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König F, Davidoff C, Ibanez K, Hwang S, Goldstein I, Feldman DR, Gyurkocza B, Giralt SA, Politikos I, Ponce DM, Scordo M, Syrkin G, Custodio CM, Shah GL. Post-Transplant Pain and Paralysis: Neurologic Amyotrophy as an Atypical Cause of Shoulder Dysfunction Following Hematopoietic Stem Cell Transplant. Cancers (Basel) 2025; 17:1816. [PMID: 40507297 PMCID: PMC12153850 DOI: 10.3390/cancers17111816] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/21/2025] [Revised: 05/24/2025] [Accepted: 05/26/2025] [Indexed: 06/16/2025] Open
Abstract
BACKGROUND Neuralgic amyotrophy (NA), also known as Parsonage-Turner syndrome, brachial neuritis, and idiopathic brachial plexopathy, is a rare and potentially debilitating peripheral nerve disorder characterized by acute-onset shoulder pain followed by progressive motor deficits. It is often under-recognized, with an estimated incidence of 1 to 3 per 100,000 annually, though some studies suggest the actual prevalence may be significantly higher. The condition typically progresses through three phases, an acute painful phase, a phase of weakness, and a recovery phase, with sensory disturbances common in addition to motor weakness. The exact pathogenesis of NA remains unclear, though it is thought to involve a combination of genetic, environmental, and immunological factors. While neurologic complications following hematopoietic stem cell transplantation (HSCT), such as neuropathies and myopathies, have been documented, NA remains exceedingly rare in this context, with only a few reported cases. The pathophysiology in HSCT patients is hypothesized to involve immune dysregulation, graft-versus-host disease (GvHD), infection, and the effects of immunosuppressive therapy. Diagnosis is primarily clinical, supported by electrodiagnostic studies and MRI, though no laboratory markers exist. The management of NA is largely supportive and multimodal, focusing on pain control and rehabilitation. OBJECTIVES The objective of this study was to describe the characteristics, clinical course, and outcomes of patients admitted for HSCT who were subsequently diagnosed with NA. STUDY DESIGN This retrospective case series from a single institution examined nine (N = 9) patients who developed acute shoulder pain following HSCT. We collected data on demographics, transplant details, clinical features, MRI findings, and electrodiagnostic studies, summarized using descriptive statistics. The diagnosis of neurologic amyotrophy was based on clinical presentation and corroborated by imaging and electrodiagnostic results. Long-term follow-up was assessed to evaluate symptom recovery. RESULTS Between August 2020 and July 2022, nine patients (44% male, median age 60) were diagnosed with NA following autologous (n = 4) or allogeneic (n = 5) HSCT. The onset of severe shoulder pain occurred at a median of 9 days post-transplant (range 1-21 days), with the majority of patients experiencing unilateral pain, predominantly affecting the right shoulder (55%). Neurologic weakness developed on average 5.1 days after pain onset, and sensory deficits were observed in all but one patient. MRI findings revealed muscle edema, atrophy, and enhancement in six patients, while electromyography confirmed NA in five. Due to the small sample size, statistical analyses, including p-values, confidence intervals, and trend comparisons, were not performed, and thus no conclusions can be drawn regarding associations between variables such as early onset and worse outcomes. Shoulder pain resolved after a median of 23 days (range 8-40 days). Long-term follow-up (>1 year) showed that three patients achieved full or near-full recovery, four partially recovered, and two showed minimal improvement. Conclusions: NA should be highly suspected in patients with acute shoulder pain and neurologic symptoms post-HSCT. To improve diagnostic accuracy and clinical outcomes, we recommend enhanced clinician awareness, the implementation of targeted diagnostic protocols (such as MRI and electrodiagnostic studies), and the establishment of standardized long-term follow-up protocols.
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Affiliation(s)
- Franchesca König
- Department of Physical Medicine & Rehabilitation, University of Colorado Anschutz Medical Campus, Aurora, CO 80045, USA;
| | - Chanel Davidoff
- Department of Physical Medicine and Rehabilitation, Lenox Hill Hospital—Northwell Health, New York, NY 10075, USA;
| | - Katarzyna Ibanez
- Rehabilitation Medicine Service, Department of Neurology, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (K.I.); (G.S.); (C.M.C.)
| | - Sinchun Hwang
- Department of Radiology, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA;
| | - Ilan Goldstein
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
| | - Darren R. Feldman
- Genitourinary Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA;
| | - Boglarka Gyurkocza
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
| | - Sergio A. Giralt
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
| | - Ioannis Politikos
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
| | - Doris M. Ponce
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
| | - Michael Scordo
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
| | - Grigory Syrkin
- Rehabilitation Medicine Service, Department of Neurology, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (K.I.); (G.S.); (C.M.C.)
| | - Christian M. Custodio
- Rehabilitation Medicine Service, Department of Neurology, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (K.I.); (G.S.); (C.M.C.)
| | - Gunjan L. Shah
- Adult Bone Marrow Transplant Service, Department of Medicine, Memorial Sloan Kettering Cancer Center, New York, NY 10065, USA; (I.G.); (B.G.); (S.A.G.); (I.P.); (D.M.P.)
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Druzhinina ES, Druzhinin DS, Tikhonova OA, Zavadenko NN. [Therapeutic options in patients with neuralgic amyotrophy]. Zh Nevrol Psikhiatr Im S S Korsakova 2025; 125:39-45. [PMID: 40195099 DOI: 10.17116/jnevro202512503139] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 04/09/2025]
Abstract
OBJECTIVE To evaluate the outcomes of patients with neuralgic amyotrophy (NA) depending on the type of treatment received - surgical or glucocorticosteroid administration. MATERIAL AND METHODS The outcome of 101 patients with verified NA was analyzed. The mean age of NA patients was 38.21±16.09 years (8 to 76 years), of which 69.3% were males (n=70) and 30.7% were females (n=31). The diagnosis was confirmed by electromyography, cervical spine MRI, and clinical data evaluation: pain according to VAS, duration of pain, timing of diagnosis from the disease onset, and severity of muscle weakness using the MRC scale. Most patients had a classic form (50.5%, n=51), followed by a distal form (39.6%, n=40), and nerve damage above the elbow joint or cranial nerves (9.9%, n=10). Most patients were examined during the phase of pain and muscle weakness (59.4%, n=60) and in the recovery phase (40.6%, n=41). 48.5% (n=49) of patients received steroids. Surgical treatment was performed in 15.8% of cases (n=16). The median follow-up of all patients was 12 months (Q1-Q3 6.00-20.00). RESULTS Patients who received GCS recovered better - 67.7% had a complete recovery compared to patients who did not receive pathogenetic therapy. Regardless of the GCS use, the outcome also depended on the NA form and the initial severity of muscle weakness (p<0.001 and p=0.001, respectively). Comparing the outcomes in the groups with and without surgery showed no statistically significant differences. However, in the non-surgery group, a higher percentage of non-recovery was found (71.4%) compared to the group receiving surgical treatment (28.6%). In 97.4% (n=38) of cases of distal NA, ultrasound revealed focal changes in the nerves, which initially indicated a poor prognosis for recovery. CONCLUSION Steroid therapy affects muscle strength recovery in patients with NA. The NA form and the initial severity of motor deficit also affect the outcome. Surgical treatment is indicated for patients with distal NA.
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Affiliation(s)
- E S Druzhinina
- Pirogov Russian National Research Medical University (Pirogov University), Moscow, Russia
| | - D S Druzhinin
- Yaroslavl State Medical University, Yaroslavl, Russia
| | - O A Tikhonova
- Immanuel Kant Baltic Federal University, Kaliningrad, Russia
| | - N N Zavadenko
- Pirogov Russian National Research Medical University (Pirogov University), Moscow, Russia
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Bang MH, Song HL, Hahn S, Kim W, Do HK. Neuralgic amyotrophy with hourglass-like constrictions: A case report. World J Clin Cases 2024; 12:6728-6735. [PMID: 39650818 PMCID: PMC11514359 DOI: 10.12998/wjcc.v12.i34.6728] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/25/2024] [Revised: 09/11/2024] [Accepted: 09/25/2024] [Indexed: 10/12/2024] Open
Abstract
BACKGROUND Neuralgic amyotrophy (NA) is a rare disease with sudden upper limb pain followed by affected muscle weakness. The most commonly affected area in NA is the upper part of the brachial plexus, and the paraspinal muscles are rarely affected (1.5%), making these cases difficult to distinguish from cervical radiculopathy. CASE SUMMARY A 76-year-old male presented to the emergency department with left hip pain post-fall. After undergoing left femoral neck fracture surgery, he experienced sudden left shoulder pain for 10 days with subsequent left arm weakness. Cervical spine computed tomography revealed mild right asymmetric intervertebral disc bulging with a decreased C5-6disc space. Three weeks later, an electrodiagnostic study confirmed brachial plexopathy findings involving the cervical root. Magnetic resonance neurography was performed for a differential diagnosis. Contrast enhancement was identified at the upper trunk of the brachial plexus, including the C5 nerve root. A suprascapular nerve hourglass-like focal constriction (HLFC) was also identified, confirming NA. After being diagnosed with NA, the patient received 15 mg prednisolone, twice daily, for 3 weeks. Physical therapy was initiated, including left arm strengthening exercises and electrical stimulation therapy. Left shoulder muscle strength significantly improved one month after comprehensive rehabilitation. CONCLUSION NA's unique features like HLFC and paraspinal involvement are crucial for accurate diagnosis, avoiding confusion with cervical radiculopathy.
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Affiliation(s)
- Mi-Hyeon Bang
- Department of Physical Medicine and Rehabilitation, Haeundae Paik Hospital, Inje University College of Medicine, Busan 48108, South Korea
| | - Ha-Lim Song
- Department of Physical Medicine and Rehabilitation, Haeundae Paik Hospital, Inje University College of Medicine, Busan 48108, South Korea
| | - Seok Hahn
- Department of Radiology, Haeundae Paik Hospital, Inje University College of Medicine, Busan 48108, South Korea
| | - Wanil Kim
- Division of Biochemistry, Department of Convergence Medical Science, Institute of Medical Science, Gyeongsang National University College of Medicine, Jinju 52727, South Korea
| | - Hwan-Kwon Do
- Department of Physical Medicine and Rehabilitation, Dongnam Institute of Radiological and Medical Sciences, Busan 46033, South Korea
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Sparasci D, Schilg‐Hafer L, Schreiner B, Scheidegger O, Peyer A, Lascano AM, Vicino A, Décard BF, Tsouni P, Humm AM, Pianezzi E, Zezza G, Hundsberger T, Dietmann A, Jung HH, Kuntzer T, Wilder‐Smith E, Martinetti‐Lucchini G, Petrini O, Fontana S, Gowland P, Niederhauser C, Gobbi C, Ripellino P. Immune triggers preceding neuralgic amyotrophy. Eur J Neurol 2024; 31:e16462. [PMID: 39364568 PMCID: PMC11554871 DOI: 10.1111/ene.16462] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/16/2024] [Revised: 06/25/2024] [Accepted: 08/18/2024] [Indexed: 10/05/2024]
Abstract
BACKGROUND AND PURPOSE Infections and vaccinations have been identified as potential immunological triggers of neuralgic amyotrophy (NA), but the exact type and frequency of the preceding agents is unknown. METHODS This was a multicentre, prospective, observational, matched case-control study. NA was diagnosed by neuromuscular experts according to validated clinical criteria and electrodiagnostic studies. Clinical data and biological samples of NA patients were collected within 90 days from disease onset between June 2018 and December 2023. All NA patients were asked about prior infection and vaccination in the month before disease onset. Serological tests for hepatitis E virus, human immunodeficiency virus, severe acute respiratory syndrome coronavirus 2, Epstein-Barr virus, cytomegalovirus, parvovirus B19, varicella-zoster virus, Borrelia burgdorferi, Mycoplasma pneumoniae and Bartonella henselae were performed in a central laboratory. Each case was matched with a healthy control for age, sex, place of residence and time of blood collection. RESULTS Fifty-seven patients and corresponding controls were included. The mean age was 45 years for both groups. NA onset was preceded by a symptomatic infectious trigger confirmed by microbiological tests in 15/57 (26.3%) patients. Coronavirus disease 2019 vaccination was considered a potential trigger in 7/57 (12.3%) subjects. An acute viral infection was associated with a bilateral involvement of the brachial plexus (p = 0.003, Cramèr's V = 0.43). CONCLUSIONS Confirmed immune triggers (infection or vaccination) preceded disease onset in 22/57 (38.6%) NA cases. We suggest to test NA patients in the acute phase for intracellular antigens, especially in the case of concomitant bilateral involvement and hepatitis.
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Affiliation(s)
- Davide Sparasci
- Department of NeurologyNeurocenter of Southern Switzerland EOCLuganoSwitzerland
| | | | - Bettina Schreiner
- Department of NeurologyUniversity and Hospital ZurichZurichSwitzerland
| | - Olivier Scheidegger
- Department of Neurology, InselspitalBern University Hospital and University of BernBernSwitzerland
| | - Anne‐Kathrin Peyer
- Department of NeurologyUniversity Hospital and University of BaselBaselSwitzerland
| | - Agustina Maria Lascano
- Neurology Division, Department of Clinical Neuroscience, University Hospitals of Geneva and Faculty of MedicineUniversity of GenevaGenevaSwitzerland
| | - Alex Vicino
- Nerve‐Muscle Unit, Neurology Service, Department of Clinical NeurosciencesLausanne University Hospital and University of LausanneLausanneSwitzerland
| | | | | | - Andrea Monika Humm
- Neurology Unit, Department of MedicineHFR Fribourg Cantonal HospitalFribourgSwitzerland
| | | | - Giulia Zezza
- Laboratory of Microbiology EOCBellinzonaSwitzerland
| | | | - Anelia Dietmann
- Department of Neurology, InselspitalBern University Hospital and University of BernBernSwitzerland
| | - Hans H. Jung
- Department of NeurologyUniversity and Hospital ZurichZurichSwitzerland
| | - Thierry Kuntzer
- Nerve‐Muscle Unit, Neurology Service, Department of Clinical NeurosciencesLausanne University Hospital and University of LausanneLausanneSwitzerland
| | - Einar Wilder‐Smith
- Department of Neurology, InselspitalBern University Hospital and University of BernBernSwitzerland
- Cantonal HospitalLucerneSwitzerland
| | | | - Orlando Petrini
- University of Applied Sciences and Arts of Southern SwitzerlandBellinzonaSwitzerland
| | - Stefano Fontana
- Blood Transfusion Service SRC Southern SwitzerlandLuganoSwitzerland
- Interregional Blood Transfusion SRCBernSwitzerland
| | | | - Christoph Niederhauser
- Interregional Blood Transfusion SRCBernSwitzerland
- Institute for Infectious DiseasesUniversity of BernBernSwitzerland
| | - Claudio Gobbi
- Department of NeurologyNeurocenter of Southern Switzerland EOCLuganoSwitzerland
- Faculty of Biomedical SciencesUniversità della Svizzera ItalianaLuganoSwitzerland
| | - Paolo Ripellino
- Department of NeurologyNeurocenter of Southern Switzerland EOCLuganoSwitzerland
- Faculty of Biomedical SciencesUniversità della Svizzera ItalianaLuganoSwitzerland
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Yoon AP, Lee EY, Tagliero LE, Pulos N, Spinner RJ, Bishop AT, Shin A. Predisposing factors for incomplete spontaneous recovery after Parsonage-Turner Syndrome. Acta Neurochir (Wien) 2024; 166:451. [PMID: 39532742 DOI: 10.1007/s00701-024-06350-1] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/20/2024] [Accepted: 11/04/2024] [Indexed: 11/16/2024]
Abstract
INTRODUCTION Parsonage-Turner Syndrome (PTS) is a rare idiopathic brachial neuritis that classically presents with sudden onset upper extremity pain followed by upper limb weakness, sensory disruption, and muscle atrophy. We hypothesized that the type of antecedent event before symptom onset would determine the degree of spontaneous recovery after PTS. METHODS We retrospectively reviewed all patients who presented with PTS to a single tertiary referral center from January 2002 to December 2021. Each medical record was reviewed to exclude patients with any other identifiable neurologic etiology. The primary outcome was improved muscle strength measured by a modified British Medical Research Council (mBMRC) grading system. The spontaneous recovery cohort included patients with appreciable or full recovery (mBMRC 4 and 5). In contrast, patients with little to no recovery (mBMRC 0 to 3) were included in the incomplete recovery group. Antecedent events, demographic variables, affected upper extremity region, time between symptom onset and final visit, and presence of nerve surgery were analyzed. Two-tailed T-test and Fisher exact tests were used to compare continuous and categorical variables. Multivariable logistic regression was modeled to identify variables associated with spontaneous recovery. RESULTS 100 patients with 144 extremities met the inclusion criteria. The average time from symptom onset to the final clinic visit was 3.5 years. 65 extremities (57.0%) spontaneously recovered without surgical intervention, whereas 49 extremities (43.0%) incompletely recovered. The spontaneous recovery cohort was younger (mean [SD] age, 47.4 [18.1] vs. 56.5 [18.7]; p = 0.02) and had fewer bilateral cases (count[%], 4 [6.5%] vs. 10 [26.3%]; p = 0.008). Multivariable logistic regression revealed that each year of increased age was associated with a 3% decreased chance of spontaneous recovery (OR[95%CI] 0.97[0.95, 0.99], p = 0.046). Bilateral involvement was associated with 93% decreased chance of recovery (OR[95%CI] 0.07[0.01, 0.39], p = 0.002), each additional muscle affected was associated with 10% decreased chance of recovery (OR[95%CI] 0.90[0.83, 0.97], p = 0.01), and traumatic antecedent events were associated with increased chance of recovery (OR[95%CI] 27.3[2.59, 287.5], p = 0.006). CONCLUSIONS Younger patients with unilateral symptoms and fewer muscle involvement have a greater chance of spontaneous recovery after PTS. PTS precipitated by trauma may have better spontaneous recovery than those precipitated by other causes. LEVEL OF EVIDENCE III
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Affiliation(s)
- Alfred P Yoon
- Division of Plastic and Reconstructive Surgery, University of California Davis School of Medicine, 2335 Stockton Blvd, Sacramento, CA, 95817, USA
| | - Ellen Y Lee
- Department of Hand and Reconstructive Microsurgery, National University Health System Singapore, 1E Kent Ridge Rd, Singapore, 119228, Singapore
| | - Lauren E Tagliero
- Department of Orthopedic Surgery, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
| | - Nicholas Pulos
- Department of Orthopedic Surgery, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
| | - Robert J Spinner
- Department of Orthopedic Surgery, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
| | - Allen T Bishop
- Department of Orthopedic Surgery, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA
| | - Alexander Shin
- Department of Orthopedic Surgery, Mayo Clinic, 200 First St. SW, Rochester, MN, 55905, USA.
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Couch B, Hayward D, Baum G, Sakthiyendran NA, Harder J, Hernandez EJ, MacKay B. A systematic review of steroid use in peripheral nerve pathologies and treatment. Front Neurol 2024; 15:1434429. [PMID: 39286807 PMCID: PMC11402678 DOI: 10.3389/fneur.2024.1434429] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/20/2024] [Accepted: 08/21/2024] [Indexed: 09/19/2024] Open
Abstract
Background The use of corticosteroids has become a part of the standard of care in various pathologies but their use in peripheral nerve injury treatment is limited. Given corticosteroids' anti-inflammatory properties and their regulatory role in neuronal protein production and myelination, corticosteroids could serve as an adjunct therapy for peripheral nerve injuries. This review aims to systematically investigate the current use of corticosteroid treatment in peripheral nerve pathologies. Methods The systematic search was performed on PubMed, MEDLINE, EMBASE, Scopus, Cochrane, and Web of Science using keywords such as "corticosteroid treatment," "peripheral nerve damage," "peripheral neuropathy," and "complications." The PRISMA guidelines were used to conduct the systematic review and all articles were reviewed by the corresponding author. After the initial search, individual study titles and abstracts were further screened and categorized using an inclusion and exclusion criteria followed by a final full-text review. Results Out of the total 27,922 identified records, 203 studies were included based on the selection criteria. These studies focused on the use and efficacy of steroids across a spectrum of compression and non-compression peripheral neuropathies such as cubital tunnel syndrome and chronic inflammatory demyelinating polyradiculoneuropathy. Various studies noted the promising role of steroids in offering pain relief, nerve block, and nerve regeneration effects. Additionally, safety considerations and potential complications regarding steroid use in peripheral nerve injuries were analyzed. Conclusion While there is currently limited clinical utilization of corticosteroids in peripheral nerve pathologies, the anti-inflammatory and regenerative effects that steroids provide may be a beneficial tool in managing various peripheral neuropathies and their associated pain. Additional clinical trials and investigation into the mechanism of action could improve the reputation of steroid use as peripheral nerve injury treatment.
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Affiliation(s)
- Brandon Couch
- Texas Tech University Health Sciences Center, Lubbock, TX, United States
| | - Dan Hayward
- Texas Tech University Health Sciences Center, Lubbock, TX, United States
| | - Gracie Baum
- Department of Orthopaedic Hand Surgery, Texas Tech University Health Sciences Center, Lubbock, TX, United States
| | | | - Justin Harder
- Department of Orthopaedic Hand Surgery, Texas Tech University Health Sciences Center, Lubbock, TX, United States
| | - Evan J Hernandez
- Department of Orthopaedic Hand Surgery, Texas Tech University Health Sciences Center, Lubbock, TX, United States
| | - Brendan MacKay
- Department of Orthopaedic Hand Surgery, Texas Tech University Health Sciences Center, Lubbock, TX, United States
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Holle JF, Limmroth V, Windisch W, Zimmerman M. Neuralgic Amyotrophy. DEUTSCHES ARZTEBLATT INTERNATIONAL 2024; 121:483-489. [PMID: 38835178 PMCID: PMC11526358 DOI: 10.3238/arztebl.m2024.0077] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 01/15/2024] [Revised: 04/16/2024] [Accepted: 04/16/2024] [Indexed: 06/06/2024]
Abstract
BACKGROUND Neuralgic amyotrophy (NA) is a multifactorial, monophasic neuritis that mainly affects the nerves of the shoulder girdle. It is characterized by very severe pain and by weakness that arises some time after the pain. Its reported incidence is high (100 cases per 100 000 persons per year), but our data suggest that many or most cases are diagnosed late or not at all. METHODS This review of the epidemiology, pathophysiology, diagnosis, and treatment of NA is based on pertinent publications retrieved by a selective literature search, and on data provided by the scientific institute of AOK, a German statutory health-insurance carrier. RESULTS It is currently thought that the combination of a genetic predisposition, an immunological trigger factor, and mechanical stress on the affected nerve segment(s) is pathophysiologically determinative. The prognosis of untreated NA is poor, with 25% of patients remaining unable to work at three years. The main form of treatment is with corticosteroids that are administered as early as possible. If there is evidence of nerve constriction or torsion, surgery may also help. There have only been six controlled cohort studies on the treatment of NA, and no randomized trials. It is not uncommon for the acute phase to develop into a chronic pain syndrome requiring multidimensional treatment. CONCLUSION Particularly in view of the high incidence and improved therapeutic options, NA should be included in the differential diagnosis of all patients with suggestive symptoms.
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Affiliation(s)
- Johannes Fabian Holle
- Department of Neurology, Cologne-Merheim, Hospitals of the City of Cologne, Cologne
- Health Faculty/Department for Human Medicine, University of Witten/Herdecke
| | - Volker Limmroth
- Department of Neurology, Cologne-Merheim, Hospitals of the City of Cologne, Cologne
| | - Wolfram Windisch
- Cologne-Merheim Lung Clinic, Hospitals of the City of Cologne, Cologne
- Health Faculty/Department for Human Medicine, University of Witten/Herdecke
| | - Maximilian Zimmerman
- Cologne-Merheim Lung Clinic, Hospitals of the City of Cologne, Cologne
- Health Faculty/Department for Human Medicine, University of Witten/Herdecke
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Ganjeh S, Aslani H, Kalantari KK, Roostayi MM. Parsonage-Turner syndrome, affecting suprascapular nerve and especially to infraspinatus muscles after COVID-19 vaccination in a professional wrestler, a case report and literature review of causes and treatments. BMC Neurol 2024; 24:187. [PMID: 38840070 PMCID: PMC11151581 DOI: 10.1186/s12883-024-03694-0] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/05/2024] [Accepted: 05/29/2024] [Indexed: 06/07/2024] Open
Abstract
BACKGROUND Acute peripheral neuropathy, also known as Parsonage-Turner syndrome or neuralgic amyotrophy, mostly affects the upper brachial plexus trunks, which include the shoulder girdle. It is typically accompanied by abrupt, intense pain, weakness, and sensory disruption. The etiology and causes of this disease are still unknown because of its low prevalence, however viral reactions-induced inflammation is one of its frequent causes. CASE PRESENTATION Here, we introduce a professional wrestler patient who was diagnosed with PTS after vaccination and was treated, and we review some articles in this field. CONCLUSION When it comes to shoulder-girdle complaints and pain, Parsonage-Turner syndrome can be a differential diagnosis. Corticosteroids during the acute period, followed by physical therapy, appear to be an efficient way to manage pain, inflammation, muscular atrophy, and the process of recovering to full nerve regeneration.
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Affiliation(s)
- Soheila Ganjeh
- Student Research Committee, Department of Physical Therapy, Faculty of Rehabilitation, Shahid Beheshti University of Medical Sciences, Tehran, Iran
| | - Hamidreza Aslani
- Department of Orthopedics, Knee and Sport Medicine Education and Research Center, School of Medicine, Shahid Beheshti University of Medical Sciences, Tehran, Iran.
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Sun J, Mao J, Zhang F, Gui X, Liao J. A case report of neuralgic amyotrophy. Front Neurol 2024; 15:1392766. [PMID: 38846034 PMCID: PMC11153766 DOI: 10.3389/fneur.2024.1392766] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/28/2024] [Accepted: 05/14/2024] [Indexed: 06/09/2024] Open
Abstract
Neuralgic muscular atrophy is not uncommon in clinical practice. Due to the different branches of brachial plexus involved in the lesion, the clinical symptoms are different, and there is a lack of clear imaging diagnostic criteria, so the diagnosis of this disease brings great challenges to clinicians. We have certain experience in the diagnosis and treatment of this disease, and hereby select a representative case of neuralgic muscular atrophy to share its diagnosis and treatment process, focusing on analyzing the characteristic symptoms of this disease, valuable imaging data and targeted treatment, so as to enable clinicians to better understand this disease.
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Affiliation(s)
- Jianfeng Sun
- Department of Rehabilitation Medicine, Southern Theater Command General Hospital of the People's Liberation Army, Guangzhou, China
| | - Ji Mao
- Department of Ultrasonography, Southern Theater Command General Hospital of the People's Liberation Army, Guangzhou, China
| | - Fengfei Zhang
- Department of Rehabilitation Medicine, Southern Theater Command General Hospital of the People's Liberation Army, Guangzhou, China
| | - Xi Gui
- Department of Rehabilitation Medicine, Southern Theater Command General Hospital of the People's Liberation Army, Guangzhou, China
| | - Junfeng Liao
- Department of Rehabilitation Medicine, Southern Theater Command General Hospital of the People's Liberation Army, Guangzhou, China
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Yeoh ZY, Ramdzan SN. Reply letter to: If SARS-CoV-2 vaccination is blamed for Parsonage-Turner syndrome, neurosurgical neurolysis is not indicated. MALAYSIAN FAMILY PHYSICIAN : THE OFFICIAL JOURNAL OF THE ACADEMY OF FAMILY PHYSICIANS OF MALAYSIA 2024; 19:34. [PMID: 38855402 PMCID: PMC11162529 DOI: 10.51866/lte.46lr] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Key Words] [Subscribe] [Scholar Register] [Indexed: 06/11/2024]
Affiliation(s)
- Zi-Yi Yeoh
- MB ChB, Department of Primary Care Medicine, Universiti Malaya Medical Centre, Kuala Lumpur, Malaysia.
| | - Siti Nurkamilla Ramdzan
- MBBS, MFamMed, PhD, Department of Primary Care Medicine, Faculty of Medicine, Universiti Malaya, Kuala Lumpur, Malaysia
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11
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Dickey Z, Sharma N. The Utility of an Epidural Steroid Injection for the Treatment of Idiopathic Brachial Neuritis. Cureus 2024; 16:e57211. [PMID: 38681293 PMCID: PMC11056218 DOI: 10.7759/cureus.57211] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/07/2024] [Accepted: 03/29/2024] [Indexed: 05/01/2024] Open
Abstract
Idiopathic brachial neuritis is an uncommon disorder that predominately affects the superior and middle trunks of the brachial plexus. Severe throbbing and aching shoulder pain is initially present for a period of days to weeks, followed by severe weakness and atrophy that can develop for an extended period of months to years. There are currently no known treatments for brachial neuritis, with the standard of care consisting of analgesics and corticosteroids, which typically provide minimal to no benefit in most cases. In this case, we will present a case of a patient who was diagnosed with idiopathic brachial neuritis and underwent an interlaminar epidural steroid injection (ESI) for treatment. Following treatment with the ESI, the patient had a subsequent resolution of symptoms. This case underscores the value of early recognition for the diagnosis of brachial neuritis and the utility of an ESI as a treatment option, thus preventing long-term pathological sequalae. To our knowledge, this is the first known reported case to have successfully cured brachial neuritis.
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Affiliation(s)
- Zachary Dickey
- Physical Medicine and Rehabilitation, Edward Via College of Osteopathic Medicine, Monroe, USA
| | - Navneet Sharma
- Physical Medicine and Rehabilitation, Green Clinic, Ruston, USA
- Rehabilitation, Ruston Regional Specialty Hospital, Ruston, USA
- Physical Medicine and Rehabilitation, Edward Via College of Osteopathic Medicine, Monroe, USA
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12
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Hagemann C, Antoniadis G, Pham M, Bischoff C, Ghosh T, Grieser T, Naumann M, Holzapfel K. [Diagnostics and treatment of hourglass-like nerve constrictions and torsions in neuralgic amyotrophy]. DER NERVENARZT 2023; 94:1157-1165. [PMID: 37943327 DOI: 10.1007/s00115-023-01562-9] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Accepted: 09/29/2023] [Indexed: 11/10/2023]
Abstract
Neuralgic amyotrophy is a disease of the peripheral nervous system characterized by severe neuropathic pain followed by peripheral paralysis. A distinction is made between a hereditary and an idiopathic form, which is assumed to have an autoimmunological origin. Conservative medicinal treatment mainly consists of nonsteroidal anti-inflammatory drugs (NSAID), opioids and glucocorticoids; however, despite treatment, symptoms in the form of pain or paralysis persist in over 50% of cases. Inflammation can lead to strictures and torsions of peripheral nerves, which can be visualized by imaging using nerve sonography or magnetic resonance (MR) neurography and confirmed intraoperatively during surgical exploration. Based on the currently available data, patients with strictures and torsions of peripheral nerves can benefit from neurosurgical treatment.
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Affiliation(s)
- Christian Hagemann
- Klinik für Neurologie und klinische Neurophysiologie, Universitätsklinikum Augsburg, Stenglinstr. 2, 86156, Augsburg, Deutschland.
| | - Gregor Antoniadis
- Sektion "Periphere Nervenchirurgie", Neurochirurgische Klinik der Universität Ulm am Bezirkskrankenhaus Günzburg, Günzburg, Deutschland
| | - Mirko Pham
- Diagnostische und interventionelle Neuroradiologie, Universitätsklinikum Würzburg, Würzburg, Deutschland
| | - Christian Bischoff
- Neurologische Gemeinschaftspraxis am Marienplatz, Burgstraße 7, 80331, München, Deutschland
| | - Tanupriya Ghosh
- Klinik für Neurologie und klinische Neurophysiologie, Universitätsklinikum Augsburg, Stenglinstr. 2, 86156, Augsburg, Deutschland
| | - Thomas Grieser
- Diagnostische und interventionelle Radiologie, Universitätsklinikum Augsburg, Augsburg, Deutschland
| | - Markus Naumann
- Klinik für Neurologie und klinische Neurophysiologie, Universitätsklinikum Augsburg, Stenglinstr. 2, 86156, Augsburg, Deutschland
| | - Korbinian Holzapfel
- Klinik für Neurologie und klinische Neurophysiologie, Universitätsklinikum Augsburg, Stenglinstr. 2, 86156, Augsburg, Deutschland
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13
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Zimmermann M, Wollsching-Strobel M, Majorski DS, Kroppen D, Schwarz SB, Berger M, Windisch W, Holle JF. [Neuralgic amyotrophy: a common cause of unilateral and bilateral diaphragmatic pareses]. Pneumologie 2023; 77:814-824. [PMID: 37647918 DOI: 10.1055/a-2113-0385] [Citation(s) in RCA: 2] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 09/01/2023]
Abstract
There are several causes for unilateral or bilateral diaphragmatic paresis. The most common cause is an (intraoperative) injury to the phrenic nerve.However, in up to 20% of cases, no explanation can be found despite extensive workup. Neuralgic amyotrophy (NA, also known as Parsonage-Turner syndrome) is a common underdiagnosed multifocal autoimmune-inflammatory disease that predominantly affects proximal nerve segments of the upper extremities. Classic symptoms include acute onset of severe pain in the shoulder girdle with delayed onset of paresis of the shoulder and arm muscles. In at least 7% of cases, the phrenic nerve is also affected. Based on the annual incidence of NA of 1:1000, the entity as a cause of diaphragmatic dysfunction is probably not as uncommon as previously thought. However, clinical experience shows that this diagnosis is often not considered, and diaphragmatic paresis gets wrongly classified as idiopathic.This is particularly disastrous because in the early stage of NA, medical therapy with corticosteroids is mostly not considered and the possibility that surgical repair of the diaphragm may be performed prematurely, given that the condition may resolve spontaneously many months after symptom onset.The aim of the present article is to raise awareness of the entity of NA as a cause of diaphragmatic paresis and to establish a standardized approach to diagnosis and treatment.
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Affiliation(s)
- Maximilian Zimmermann
- Pneumologie, Kliniken der Stadt Köln gGmbH, Köln, Deutschland
- Lehrstuhl für Pneumologie, Universität Witten/Herdecke Fakultät für Gesundheit, Köln, Deutschland
| | - Maximilian Wollsching-Strobel
- Pneumologie, Kliniken der Stadt Köln gGmbH, Köln, Deutschland
- Humanmedizin, Universität Witten/Herdecke Fakultät für Gesundheit, Witten, Deutschland
| | | | - Doreen Kroppen
- Pneumologie, Kliniken der Stadt Köln gGmbH, Universität Witten/Herdecke, Köln, Deutschland
| | - Sarah Bettina Schwarz
- Pneumologie, Kliniken der Stadt Köln gGmbH, Universität Witten/Herdecke, Köln, Deutschland
| | - Melanie Berger
- Pneumologie, Kliniken der Stadt Köln gGmbH, Universität Witten/Herdecke, Köln, Deutschland
| | - Wolfram Windisch
- Pneumologie, Kliniken der Stadt Köln gGmbH, Universität Witten/Herdecke, Köln, Deutschland
| | - Johannes Fabian Holle
- Neurologie, Kliniken der Stadt Köln gGmbH, Köln, Deutschland
- Lehrstuhl für Pneumologie, Universität Witten/Herdecke Fakultät für Gesundheit, Köln, Deutschland
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14
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Tracy JA. Autoimmune Axonal Neuropathies. Continuum (Minneap Minn) 2023; 29:1378-1400. [PMID: 37851035 DOI: 10.1212/con.0000000000001344] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 10/19/2023]
Abstract
OBJECTIVE This article reviews autoimmune axonal neuropathies, their characteristic clinical features, disease and antibody associations, appropriate ancillary testing, treatment, and prognosis. LATEST DEVELOPMENTS In 2021, the American College of Rheumatology and the Vasculitis Foundation released new summary guidelines for the treatment of antineutrophil cytoplasmic autoantibody-associated vasculitides. In addition, novel autoantibodies have been recently identified; they are often paraneoplastic and associated with axonal neuropathies. ESSENTIAL POINTS Recognition of autoimmune axonal neuropathies is important because of the potential for effective treatment to either reverse deficits or slow the progression of disease. It is necessary to properly assess for associations with other systemic disorders (eg, systemic vasculitis, connective tissue disease, neoplasm) so that adequate treatment for both neurologic and non-neurologic aspects of the disease can be initiated.
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15
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Loganathan D, Counsell N, Kesavanarayanan V, Badge R. Brachial Neuritis After a COVID-19 Booster Vaccination: A Case Report and Literature Review. Cureus 2023; 15:e45040. [PMID: 37829970 PMCID: PMC10566533 DOI: 10.7759/cureus.45040] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 09/07/2023] [Indexed: 10/14/2023] Open
Abstract
Idiopathic brachial neuritis also known as Parsonage-Turner syndrome is a rare neurological disorder characterised by pain and paraesthesia involving the shoulder girdle and upper limb, followed by weakness of the affected area. The cause is not very well understood and is often misdiagnosed leading to delayed treatment and long-term disability. There are many risk factors for the condition, with immunisations being accountable for as many as 15% of cases. In response to the coronavirus disease 2019 (COVID-19) pandemic, many companies have been producing and distributing COVID-19 vaccinations. To our knowledge, there have been 42 cases of brachial neuritis reported following COVID-19 vaccination to date. Here, we report a case of brachial neuritis following a patient's fourth COVID-19 vaccination and present a review of the literature.
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Affiliation(s)
- Deeraj Loganathan
- Trauma and Orthopaedics, Warrington and Halton Hospitals NHS Foundation Trust, Warrington, GBR
| | - Nina Counsell
- Orthopaedics and Trauma, University of Liverpool School of Medicine, Liverpool, GBR
| | | | - Ravi Badge
- Trauma and Orthopaedics, Warrington and Halton Hospitals NHS Foundation Trust, Warrington, GBR
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16
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MacRae FA, Boissonnault E, Winston P. A retrospective review of the management and outcomes of patients diagnosed with complex regional pain syndrome type II using electrodiagnostic findings. Can J Pain 2023; 7:2242892. [PMID: 38229666 PMCID: PMC10791151 DOI: 10.1080/24740527.2023.2242892] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/03/2023] [Accepted: 07/27/2023] [Indexed: 01/18/2024]
Abstract
Objectives The objective of this study was to assess the outcomes of the use of electrodiagnosis in the diagnosis and management of discrete nerve injuries in patients with complex regional pain syndrome (CRPS). Design This study is a secondary retrospective cohort analysis of patients diagnosed with CRPS from a single outpatient physical medicine and rehabilitation clinic and included all patients who had abnormal electrodiagnostic findings, in addition to CRPS. Results Sixty patients of 248 diagnosed with CRPS underwent electrodiagnosis, 41 of whom had abnormal electrodiagnostic findings indicating a discrete nerve injury. Only 51% of the 41 referrals had indicated the suspicion of a nerve injury. Nearly all patients had undergone physiotherapy. Forty-one percent responded to treatment with oral prednisone alone, 54% had a functional improvement after a combination of treatments including corticosteroids, and 5% improved with treatments that did not involve corticosteroids. Surgical interventions for nerve injuries were required for 34% of patients in the cohort. All surgeries involved the median or ulnar nerve, with the exception of one fibular nerve. After treatment, 39 of 41 patients had functional recoveries or better. Conclusions Electrodiagnosis can inform diagnosis of nerve injury and direct intervention including the need for surgical intervention. Electrodiagnosis should be considered for patients with initial signs of concomitant discrete nerve injury or with CRPS who are not responding to treatments because a nerve injury may be underlying. What is Known Complex Regional Pain Syndrome (CRPS) is a poorly understood pain condition. CRPS has been divided into two subtypes, the second subtype involves a discrete nerve injury with pain that extends beyond the territory of the nerve injury. What is New We observed that nerve injuries that may require surgical intervention are diagnosed just over half of the time upon initial assessment in patients with suspected CRPS. We observed that nerve injuries frequently required specifically directed interventions in place of or in conjunction with CRPS treatments. We suggest that electrodiagnosis is an important part of the triage protocol for CRPS II to reveal discrete nerve injuries that may be hidden. We recommend that electrodiagnosis be considered for patients with initial signs of concomitant discrete nerve injury or for CRPS patients who do not improve with medical therapies.
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Affiliation(s)
- Fraser Alexander MacRae
- Health Sciences, Western University, London, Canada
- Vancouver Island Health Authority, Victoria, Canada
| | - Eve Boissonnault
- Physical Medicine and Rehabilitation, University of Montreal, Montreal, Canada
| | - Paul Winston
- Faculty of Medicine, University of British Columbia, Victoria, Canada
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17
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Malik K, Giberson C, Ballard M, Camp N. Multimodal Pain Management for Parsonage-Turner Syndrome in the Acute Rehabilitation Setting: A Case Report. Cureus 2023; 15:e43216. [PMID: 37692705 PMCID: PMC10487277 DOI: 10.7759/cureus.43216] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 08/08/2023] [Indexed: 09/12/2023] Open
Abstract
Parsonage-Turner syndrome (PTS) is a rare neurological condition characterized by acute shoulder pain and novel neuromuscular weakness in the distribution of the brachial plexus. We present a case report highlighting the difficulties in the diagnosis and management of this condition. A multidisciplinary approach is often required to control this unique type of pain, consisting of neuropathic medications, non-steroidal anti-inflammatory drugs (NSAIDs) to control neuromuscular pain, and modalities provided by experienced physical therapists. Finally, a comprehensive, structured rehabilitation program focusing on the range of motion, neuromuscular re-education, and strengthening is imperative to regain function, reduce symptoms, and improve recovery.
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Affiliation(s)
- Kashif Malik
- Physical Medicine and Rehabilitation, Casa Colina Hospital and Centers for Healthcare, Pomona, USA
| | - Curren Giberson
- Physical Medicine and Rehabilitation, Casa Colina Hospital and Centers for Healthcare, Pomona, USA
| | - Matthew Ballard
- Physical Medicine and Rehabilitation, Casa Colina Hospital and Centers for Healthcare, Pomona, USA
| | - Nathan Camp
- Physical Medicine and Rehabilitation, Casa Colina Hospital and Centers for Healthcare, Pomona, USA
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18
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Chiang PI, Chen CM. Increased 18 F-FDG uptake in denervated muscles in a case of Parsonage-Turner syndrome. BMC Neurol 2023; 23:280. [PMID: 37495981 PMCID: PMC10369682 DOI: 10.1186/s12883-023-03328-x] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/10/2023] [Accepted: 07/12/2023] [Indexed: 07/28/2023] Open
Abstract
BACKGROUND Parsonage-Turner Syndrome (PTS) is a rare brachial plexopathy characterized by the sudden onset of pain in the shoulder girdle followed by upper limb weakness. PTS is frequently under-recognized or misdiagnosed as other more common neurological disorders presenting in a similar fashion, such as cervical radiculopathy which may require surgical intervention. Accurate diagnosis and prompt management implicate a good prognosis. Although electrophysiological studies are considered the most important for evaluating peripheral nerve injuries, it usually takes time, up to 3 weeks after the initial insult of the nerve for electromyogram (EMG) and nerve conduction studies (NCS) to display abnormalities. In the cases of PTS, especially when initial EMG/NCS and magnetic resonance neurography (MRN) results are inconclusive, 18 F-FDG positron emission tomography and computed tomography (18 F-FDG PET-CT) may be useful in helping the early detection of muscle denervation. CASE PRESENTATION A 60-year-old right-handed Taiwanese woman presented with sudden onset of intense and sharp left shoulder girdle pain without radiating to the arm, followed by muscle weakness of her left arm in abduction and elevation 3 days after the onset of pain. A detailed neurological examination and EMG and NCS suggested the clinical diagnosis of left brachial plexopathy. MRN imaging revealed no significant abnormality. 18 F-FDG PET-CT showed increased uptake in denervated muscles (supraspinatus, deltoid, and biceps muscles). Treatment with oral prednisolone and physiotherapy significantly improved pain and muscle weakness. CONCLUSIONS We present increased 18 F-FDG uptake in denervated muscles detected by 18 F-FDG PET-CT. 18 F-FDG PET-CT may serve as an adjunct examination to evaluate PTS, which has been suggested previously but rarely reported.
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Affiliation(s)
- Ping-I Chiang
- Center for Medical Education in English, Poznan University of Medical Sciences, Poznan, 60-512, Poland
| | - Chiung-Mei Chen
- Linkou Medical Center, Department of Neurology, Chang Gung Memorial Hospital, Taoyuan, Taiwan.
- Department of Neurology, College of Medicine, Chang Gung University, Taoyuan County, 33305, Taiwan.
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19
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Restoring the function in neuralgic shoulder amyotrophy by modified Somsak nerve transfer. INTERDISCIPLINARY NEUROSURGERY 2023. [DOI: 10.1016/j.inat.2023.101745] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/22/2023] Open
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20
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Salomon M, Marruganti S, Cucinotta A, Lorusso M, Bortolotti P, Brindisino F. Parsonage-Turner Syndrome mimicking musculoskeletal shoulder pain: A case report during the SARS-CoV-2 pandemic era. J Telemed Telecare 2023; 29:133-146. [PMID: 35678699 PMCID: PMC9184833 DOI: 10.1177/1357633x221100059] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/02/2023]
Abstract
Parsonage-Turner Syndrome or neuralgic amyotrophy is a peripheral neuropathy typically characterized by an abrupt onset of pain, followed by progressive neurological deficits (e.g. weakness, atrophy, occasionally sensory abnormalities) that involve the upper limb, mainly the shoulder, encompassing an extensive spectrum of clinical manifestations, somehow difficult to recognize. This case report describes the proper management of a 35-year-old, bank employee and sports amateur who reported subtle and progressive upper limb disorder with previous history of neck pain. SARS-CoV-2 pandemic era made patient's access to the healthcare system more complicated. Nevertheless, proper management of knowledge, relevant aspects of telerehabilitation-based consultation for musculoskeletal pain, advanced skills, tools and technologies led the physiotherapist to suspect an atypical presentation of Parsonage-Turner Syndrome. Further, neurologist consultation and electromyography suggested signs of denervation in the serratus anterior and supraspinatus muscle. Therefore, an appropriate physiotherapist's screening for referral is conducted to correct diagnosis and thorough treatment.
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Affiliation(s)
- Mattia Salomon
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy,Mattia Salomon, Department of Clinical
Sciences and Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, 00133, Italy.
| | - Sharon Marruganti
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy
| | - Andrea Cucinotta
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy
| | - Mariangela Lorusso
- Department of Clinical Sciences and
Translational Medicine, University of Roma “Tor Vergata” c/o
Medicine and Surgery School, Rome, Italy
| | - Paolo Bortolotti
- Department of Medicine and Health
Science “Vincenzo Tiberio”, University of Molise C/da Tappino c/o
Cardarelli Hospital, Campobasso, Italy
| | - Fabrizio Brindisino
- Department of Medicine and Health
Science “Vincenzo Tiberio”, University of Molise C/da Tappino c/o
Cardarelli Hospital, Campobasso, Italy
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21
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Janssen RMJ, Lustenhouwer R, Cup EHC, van Alfen N, Ijspeert J, Helmich RC, Cameron IGM, Geurts ACH, van Engelen BGM, Graff MJL, Groothuis JT. Effectiveness of an outpatient rehabilitation programme in patients with neuralgic amyotrophy and scapular dyskinesia: a randomised controlled trial. J Neurol Neurosurg Psychiatry 2023; 94:474-481. [PMID: 36697215 DOI: 10.1136/jnnp-2022-330296] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/29/2022] [Accepted: 01/10/2023] [Indexed: 01/27/2023]
Abstract
BACKGROUND Neuralgic amyotrophy (NA) is an acute inflammation of nerves within the brachial plexus territory leading to severe pain and multifocal paresis resulting in >60% of patients having residual complaints and functional limitations correlated with scapular dyskinesia. Our primary aim was to compare the effects of multidisciplinary rehabilitation (MR), focused on motor relearning to improve scapular dyskinesia and self-management strategies for reducing pain and fatigue, with usual care (UC) on shoulder, arm and hand functional capability in patients with NA. METHODS In a non-blinded randomised controlled trial (RCT), patients with NA (aged≥18 years, scapular dyskinesia, >8 weeks after onset) were randomised to either an MR or an UC group. MR consisted of a diagnostic multidisciplinary consultation and eight sessions of physical and occupational therapy. Primary outcome was functional capability of the shoulder, arm and hand assessed with the Shoulder Rating Questionnaire-Dutch Language Version (SRQ-DLV). RESULTS We included 47 patients with NA; due to drop-out, there were 22 participants in MR and 15 in UC for primary analysis. The mean group difference adjusted for sex, age and SRQ-DLV baseline score was 8.60 (95%CI: 0.26 to 16.94, p=0.044). The proportion attaining a minimal clinically relevant SRQ-DLV improvement (≥12) was larger for the MR group (59%) than the UC group (33%) with a number needed to treat of 4. CONCLUSION This RCT shows that an MR programme focused on motor relearning to improve scapular dyskinesia, combined with self-management strategies for reducing pain and fatigue, shows more beneficial effects on shoulder, arm and hand functional capability than UC in patients with NA. TRIAL REGISTRATION NUMBER NCT03441347.
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Affiliation(s)
- Renske M J Janssen
- Rehabilitation, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, The Netherlands
| | - Renee Lustenhouwer
- Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands.,Department of Functional Genomics, Faculty of Science, Center for Neurogenomics and Cognitive Research, Vrije Universiteit, Amsterdam, The Netherlands
| | - Edith H C Cup
- Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Nens van Alfen
- Neurology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Jos Ijspeert
- Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Rick C Helmich
- Neurology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands.,Donders Centre for Cognitive Neuroimaging, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Ian G M Cameron
- Donders Centre for Cognitive Neuroimaging, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Alexander C H Geurts
- Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Baziel G M van Engelen
- Neurology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
| | - Maud J L Graff
- Rehabilitation, Radboud Institute for Health Sciences, Radboud University Medical Center, Nijmegen, The Netherlands
| | - Jan T Groothuis
- Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Nijmegen Medical Centre, Nijmegen, The Netherlands
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22
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Nakajima A, Fukushima T, Mori H, Nozaki H, Makino K. [A case of neuralgic amyotrophy with extension disturbance of fingers after Cushing's syndrome remission]. Rinsho Shinkeigaku 2022; 62:632-636. [PMID: 35871567 DOI: 10.5692/clinicalneurol.cn-001759] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 06/15/2023]
Abstract
We describe a 57-year-old female patient who experienced hypercortisolemia caused by adrenal Cushing's syndrome. Two months post-adrenalectomy, she developed acute severe bilateral pain starting in her fingers and spreading up her arms. In the subsequent two weeks, the patient presented upper extremity patchy paralysis with extension disturbance of fingers. In the following two months, she experienced atrophy of the muscles in the hands and joint contracture. Consequently, we diagnosed her with neuralgic amyotrophy. Nerve conduction studies showed low compound muscle action potential of all the peripheral nerves in the forearms, suggesting motor neuron axonopathy. Gadolinium-enhanced MRI and ultrasound studies did not reveal any abnormalities in the brachial plexus and peripheral nerves of the forearms. The patient tested positive for anti-GalNAc-GD1a-IgM antibodies and received intravenous immunoglobulin 6 months after the onset of symptoms, which resulted in reduction of pain, muscle weakness, and contractures. This rare case of potentially immune-mediated bilateral patchy paralysis may have important implications in the understanding of clinical and pathological heterogenicity of neuralgic amyotrophy.
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Affiliation(s)
| | - Takao Fukushima
- Department of Neurology, Niigata Prefectural Shibata Hospital
| | - Hideki Mori
- Department of Neurology, Niigata Prefectural Shibata Hospital
| | - Hiroaki Nozaki
- Department of Neurology, Niigata Prefectural Shibata Hospital
| | - Kunihiko Makino
- Department of Neurology, Niigata Prefectural Shibata Hospital
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23
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Amjad MA, Hamid Z, Patel Y, Husain M, Saddique A, Liaqat A, Ochieng P. COVID-19 Vaccine-Induced Parsonage-Turner Syndrome: A Case Report and Literature Review. Cureus 2022; 14:e25493. [PMID: 35783889 PMCID: PMC9242527 DOI: 10.7759/cureus.25493] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 05/30/2022] [Indexed: 11/05/2022] Open
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24
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Miller NJ, Hooper DR, Sharma A. Bilateral neuralgic amyotrophy in a patient with livestock-associated hepatitis E virus infection. CMAJ 2022; 194:E495-E499. [PMID: 35379662 PMCID: PMC8985903 DOI: 10.1503/cmaj.211679] [Citation(s) in RCA: 3] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/22/2022] Open
Affiliation(s)
- Nicholas J Miller
- Departments of Physical Medicine and Rehabilitation (Miller, Hooper) and Internal Medicine (Sharma), Faculty of Medicine, University of Manitoba, Winnipeg, Man.
| | - Davyd R Hooper
- Departments of Physical Medicine and Rehabilitation (Miller, Hooper) and Internal Medicine (Sharma), Faculty of Medicine, University of Manitoba, Winnipeg, Man
| | - Aditya Sharma
- Departments of Physical Medicine and Rehabilitation (Miller, Hooper) and Internal Medicine (Sharma), Faculty of Medicine, University of Manitoba, Winnipeg, Man
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25
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Vitturi BK, Grandis M, Beltramini S, Orsi A, Schenone A, Icardi G, Durando P. Parsonage-Turner syndrome following coronavirus disease 2019 immunization with ChAdOx1-S vaccine: a case report and review of the literature. J Med Case Rep 2021; 15:589. [PMID: 34903275 PMCID: PMC8667016 DOI: 10.1186/s13256-021-03176-8] [Citation(s) in RCA: 17] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/25/2021] [Accepted: 11/01/2021] [Indexed: 01/01/2023] Open
Abstract
BACKGROUND Parsonage-Turner syndrome is an acute peripheral neuropathy that affects the upper brachial plexus region. Previously published reports demonstrate that the condition can be triggered by surgery, infection, autoimmune diseases, strenuous exercise, trauma, radiation, and vaccination. Parsonage-Turner syndrome has already been reported in three other patients who were vaccinated against coronavirus disease 2019. CASE PRESENTATION We report the case of a 51-year-old Caucasian man without comorbidities who received the first dose of the ChAdOx1-S recombinant vaccine (Vaxzevria, AstraZeneca, Oxford, UK) against coronavirus disease 2019 and was diagnosed with Parsonage-Turner syndrome. A few days after getting vaccinated, the patient reported a progressive increase in pain in the region of vaccine administration. One month later, the shoulder pain was followed by symptoms of hypoesthesia and muscle weakness on abduction and elevation of the left upper limb. Neurological examination revealed an atrophy of the proximal muscles of the left upper limb, accompanied by paresis of the left deltoid, biceps brachii, triceps brachii, and infraspinatus muscles. Electroneuromyography carried out 3 months after the onset of symptoms showed signs consistent with brachial plexus neuritis. The adverse reaction has been properly reported to the Italian Pharmacovigilance System (Italian Medicines Agency-Agenzia Italiana del Farmaco. CONCLUSION The increased awareness of such association is essential for early identification and diagnosis and, thus, better clinical outcomes.
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Affiliation(s)
- Bruno Kusznir Vitturi
- Department of Health Sciences, University of Genoa, L.go R. Benzi, 10 (Building 3), 16122, Genoa, Italy.
| | - Marina Grandis
- DINOGMI, University of Genoa, Genoa, Italy.,Neurology Unit, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
| | | | - Andrea Orsi
- Hygiene Unit, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
| | - Angelo Schenone
- DINOGMI, University of Genoa, Genoa, Italy.,Neurology Unit, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
| | - Giancarlo Icardi
- Department of Health Sciences, University of Genoa, L.go R. Benzi, 10 (Building 3), 16122, Genoa, Italy.,Hygiene Unit, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
| | - Paolo Durando
- Department of Health Sciences, University of Genoa, L.go R. Benzi, 10 (Building 3), 16122, Genoa, Italy.,Occupational Medicine Unit, IRCCS Ospedale Policlinico San Martino, Genoa, Italy
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26
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Kim JG, Kim SY, Oh HS, Jo DH. Parsonage-Turner Syndrome Following Typhoid Vaccination. Yonsei Med J 2021; 62:868-871. [PMID: 34427074 PMCID: PMC8382727 DOI: 10.3349/ymj.2021.62.9.868] [Citation(s) in RCA: 8] [Impact Index Per Article: 2.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 08/12/2020] [Revised: 06/15/2021] [Accepted: 07/13/2021] [Indexed: 11/27/2022] Open
Abstract
Parsonage-Turner syndrome is a rare neurological disease of varying etiology characterized by severe shoulder pain, muscle weakness, and atrophy. Mechanisms are unclear, but are thought to be genetic and immune-mediated reactions. Rarely, Parsonage-Turner syndrome occurs as a side effect of vaccination. A 20-year-old male who worked as a soldier visited the military hospital because of shoulder pain after vaccination against typhoid and was diagnosed with Parsonage-Turner syndrome based on electromyography and joint magnetic resonance imaging. Pain was controlled with a nerve block. Intravenous immunoglobulin was administered for improvement of neurologic symptoms. This case suggests that Parsonage-Turner syndrome should be considered as a side effect of vaccination. To the best of our knowledge, this is the first report of Parsonage-Turner syndrome following vaccination in Korea.
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Affiliation(s)
- Jeong Gil Kim
- Department of Rehabilitation Medicine, Armed Forces Yangju Hospital, Yangju, Korea
| | - Se Yong Kim
- Division of Respiratory and Critical Care Medicine, Department of Internal Medicine, Armed Forces Daejeon Hospital, Daejeon, Korea
| | - Hong Sang Oh
- Division of Infectious Disease, Department of Internal Medicine, Armed Forces Capital Hospital, Seongnam, Korea
| | - Dong Ho Jo
- Division of Infectious Disease, Department of Internal Medicine, Myongji Hospital, Goyang, Korea.
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27
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Garofoli R, Zauderer J, Seror P, Roren A, Guerini H, Rannou F, Drapé JL, Nguyen C, Lefèvre-Colau MM. Neuralgic amyotrophy and hepatitis E infection: 6 prospective case reports. RMD Open 2021; 6:rmdopen-2020-001401. [PMID: 33219125 PMCID: PMC8011528 DOI: 10.1136/rmdopen-2020-001401] [Citation(s) in RCA: 5] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/17/2020] [Revised: 09/30/2020] [Accepted: 11/01/2020] [Indexed: 12/23/2022] Open
Abstract
Introduction Hepatitis E virus (HEV) represents the main cause of enterically transmitted hepatitis
worldwide. It is known that neuralgic amyotrophy (NA) is one of the most frequent
neurological manifestations of HEV. However, clinical, electrodiagnostic (EDX) and MRI
characteristics, as well as long-term follow-up of HEV-related NA have not been fully
described yet. Case reports We describe longitudinally clinical, EDX, biological and MRI results of six cases of
HEV-associated NA, diagnosed from 2012 to 2017. Patients were between the ages of 33 and
57 years old and had a positive HEV serology. Clinical patterns showed the whole
spectrum of NA, varying from extensive multiple mononeuropathy damage to single
mononeuropathy. EDX results showed that the patients totalised 26 inflammatory
mononeuropathies (1 to 8 per patient). These involved classical nerves such as
suprascapular (6/6 cases), long thoracic (5/6 cases) and accessory spinal nerves (2/6
cases) and, some less frequent more distal nerves like anterior interosseous nerve (3/6
cases), as well as some unusual ones such as the lateral antebrachial cutaneous nerve
(1/6 case), sensory fibres of median nerve (1/6 case) and phrenic nerves (1/6 case).
After 2 to 8 years, all nerves had clinically recovered (muscle examination above
3/5 on MRC scale for all muscles except in one patient). Discussion HEV should be systematically screened when NA is suspected, whatever the severity, if
the onset is less than 4 months (before IgM HEV-antibodies disappear) and appears
to be frequently associated with severe clinical and EDX pattern, without increasing the
usual recovery time.
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Affiliation(s)
- Romain Garofoli
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
| | - Jennifer Zauderer
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
| | - Paul Seror
- Département De Neurophysiologie, Univ. Paris Pierre Et Marie Curie, Hôpital Pitié-Salpêtrière, Assistance Publique-Hôpitaux de Paris, Paris, France
| | - Alexandra Roren
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
| | - Henri Guerini
- Service de Radiologie ostéo-articulaire, Hôpital Cochin, Paris, France
| | - François Rannou
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France.,Université Paris Descartes, PRES Sorbonne Paris Cité, Paris, France
| | - Jean-Luc Drapé
- Service de Radiologie ostéo-articulaire, Hôpital Cochin, Paris, France
| | - Christelle Nguyen
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France.,Université de Paris, Faculté de Santé, UFR de Médecine, Paris, France.,INSERM UMR-S 1124, Toxicité Environnementale, Cibles Thérapeutiques, Signalisation Cellulaire et Biomarqueurs (T3S), Campus Saint-Germain-des-Prés, Paris, France
| | - Marie-Martine Lefèvre-Colau
- Service de Rééducation et de Réadaptation de l'Appareil Locomoteur et des Pathologies du Rachis, Hôpital Cochin, AP-HP.Centre-Université de Paris, Paris, France
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28
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Global Research on Neuropathic Pain Rehabilitation over the Last 20 Years. Neural Plast 2021; 2021:5594512. [PMID: 34306062 PMCID: PMC8282394 DOI: 10.1155/2021/5594512] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/25/2021] [Revised: 04/25/2021] [Accepted: 06/25/2021] [Indexed: 01/19/2023] Open
Abstract
Background Neuropathic pain has long been a very popular and productive field of clinical research. Neuropathic pain is difficult to cure radically because of its complicated etiology and uncertain pathogenesis. As pain worsens and persists, pain recovery techniques become more important, and medication alone is insufficient. No summary of bibliometric studies on neuropathic pain rehabilitation is yet available. The purpose of the present study is to analyze in a systematic manner the trends of neuropathic pain rehabilitation research over the period of 2000–2019. Methods Studies related to neuropathic pain rehabilitation and published between January 2000 and December 2019 were obtained from the Science Citation Index-Expanded of Web of Science. No restrictions on language, literature type, or species were established. CiteSpace V and Microsoft Excel were used to capture basic information and highlights in the field. Results Linear regression analysis showed that the number of publications on neuropathic pain rehabilitation significantly increased over time (P < 0.001). The United States showed absolute strength in terms of number of papers published, influence, and cooperation with other countries. Based on the subject categories of the Web of Science, “Rehabilitation” had the highest number of published papers (446), the highest number of citations (10,954), and the highest number of open-access papers (151); moreover, this category and “Clinical Neurology” had the same H-index (i.e., 52). “Randomized Controlled Trials” revealed the largest cluster in the cocitation map of references. The latest burst keywords included “Exercise” (2014–2019), “Functional Recovery” (2015–2019), and “Questionnaire” (2015–2019). Conclusion This study provides valuable information for neuropathic pain rehabilitation researchers seeking fresh viewpoints related to collaborators, cooperative institutions, and popular topics in this field. Some new research trends are also highlighted.
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29
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Nagao R, Ishikawa T, Mizutani Y, Niimi Y, Shima S, Ito M, Murayama K, Toyama H, Ueda A, Watanabe H. Magnetic Resonance Neurography in a Patient with Distal Neuralgic Amyotrophy. Intern Med 2021; 60:1759-1761. [PMID: 33361681 PMCID: PMC8222128 DOI: 10.2169/internalmedicine.6440-20] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/29/2022] Open
Abstract
The pathophysiology of neuralgic amyotrophy (NA) remains to be elucidated. However, high-resolution magnetic resonance imaging and ultrasound sonography have provided new insights into the mechanism underlying the development of NA and its diagnosis. We report a case of idiopathic distal NA with hyperintensity and thickening in the inferior trunk extending to the posterior and medial fasciculus of the left brachial plexus, which was detected by magnetic resonance neurography (MRN) with diffusion-weighted whole-body imaging with background body signal suppression (DWIBS). The abnormal signal intensity diminished after the improvement of symptoms following corticosteroid treatment. MRN with DWI can help diagnose distal NA and evaluate the post-therapeutic response.
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Affiliation(s)
- Ryunosuke Nagao
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Tomomasa Ishikawa
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Yasuaki Mizutani
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Yoshiki Niimi
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Sayuri Shima
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Mizuki Ito
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Kazuhiro Murayama
- Department of Radiology, Fujita Health University School of Medicine, Japan
| | - Hiroshi Toyama
- Department of Radiology, Fujita Health University School of Medicine, Japan
| | - Akihiro Ueda
- Department of Neurology, Fujita Health University School of Medicine, Japan
| | - Hirohisa Watanabe
- Department of Neurology, Fujita Health University School of Medicine, Japan
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30
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Kim TU, Chang MC. Neuralgic amyotrophy: an underrecognized entity. J Int Med Res 2021; 49:3000605211006542. [PMID: 33823638 PMCID: PMC8033465 DOI: 10.1177/03000605211006542] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/02/2021] [Accepted: 03/08/2021] [Indexed: 12/02/2022] Open
Abstract
Neuralgic amyotrophy (NA) is markedly underdiagnosed in clinical practice, and its actual incidence rate is about 1 per 1000 per year. In the current article, we provide an overview of essential information about NA, including the etiology, clinical manifestations, diagnostic investigations, differential diagnosis, treatment, and prognosis. The causes of NA are multifactorial and include immunological, mechanical, or genetic factors. Typical clinical findings are a sudden onset of pain in the shoulder region, followed by patchy flaccid paralysis of muscles in the shoulder and/or arm. A diagnosis of NA is based on a patient's clinical history and physical examination. Gadolinium-enhanced magnetic resonance imaging and high-resolution magnetic resonance neurography are useful for confirming the diagnosis and choosing the appropriate treatment. However, before a diagnosis of NA is confirmed, other disorders with similar symptoms, such as cervical radiculopathy or rotator cuff tear, need to be ruled out. The prognosis of NA depends on the degree of axonal damage. In conclusion, many patients with motor weakness and pain are encountered in clinical practice, and some of these patients will exhibit NA. It is important that clinicians understand the key features of this disorder to avoid misdiagnosis.
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Affiliation(s)
- Tae Uk Kim
- Department of Physical Medicine and Rehabilitation, College of
Medicine, Dankook University, Cheonan, Republic of Korea
| | - Min Cheol Chang
- Department of Physical Medicine and Rehabilitation, College of
Medicine, Yeungnam University, Daegu, Republic of Korea
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31
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Ismail II, Abdelnabi EA, Al-Hashel JY, Alroughani R, Ahmed SF. Neuralgic amyotrophy associated with COVID-19 infection: a case report and review of the literature. Neurol Sci 2021; 42:2161-2165. [PMID: 33745038 PMCID: PMC7980790 DOI: 10.1007/s10072-021-05197-z] [Citation(s) in RCA: 17] [Impact Index Per Article: 4.3] [Reference Citation Analysis] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/31/2021] [Accepted: 03/16/2021] [Indexed: 12/31/2022]
Affiliation(s)
- Ismail Ibrahim Ismail
- Department of Neurology, Ibn Sina Hospital, BOX 25427, Gamal Abdel Nasser Street, Sabah Medical Area, 13115, Kuwait, Kuwait.
| | | | - Jasem Y Al-Hashel
- Department of Neurology, Ibn Sina Hospital, BOX 25427, Gamal Abdel Nasser Street, Sabah Medical Area, 13115, Kuwait, Kuwait.,Health Sciences Centre, Department of Medicine, Kuwait University, Kuwait, Kuwait
| | - Raed Alroughani
- Division of Neurology, Department of Medicine, Amiri Hospital, Sharq, Kuwait, Kuwait
| | - Samar Farouk Ahmed
- Department of Neurology, Ibn Sina Hospital, BOX 25427, Gamal Abdel Nasser Street, Sabah Medical Area, 13115, Kuwait, Kuwait.,Department of Neurology and Psychiatry, Minia University, Minya, Egypt
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32
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Hu X, Jing M, Feng J, Tang J. Four cases of pediatric neuralgic amyotrophy treated with immunotherapy: one-year follow-up and literature review. J Int Med Res 2021; 48:300060520912082. [PMID: 32228355 PMCID: PMC7132571 DOI: 10.1177/0300060520912082] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/17/2022] Open
Abstract
Neuralgic amyotrophy (NA) is a neurological disease that occurs across all age
groups, but its prognosis in children is controversial. The present report adds
to the knowledge about its prognosis by describing four cases of pediatric NA in
which the patients were treated with immunotherapy and followed up for 1 year.
We also present a summary of relevant cases of pediatric NA treated with
immunotherapy. The clinical features of the four present cases were similar to
those of previously reported cases, and their symptoms improved after
immunotherapy. At the 1-year follow-up, three of the children gained near
complete recovery, and their improvement was significantly better than that
observed at the 2-month follow-up. A review of the literature showed that most
previously reported children with NA showed improvement after immunotherapy, but
no more than half of the patients recovered fully. These findings indicate that
in children with NA, immunotherapy is fairly effective and its benefits improve
with time. Thus, long-term follow-up is needed in these patients to determine
their prognosis.
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Affiliation(s)
- Xiaoyue Hu
- Department of Neurology, Children's Hospital of Soochow University, Suzhou, Jiangsu Province, China.,Department of Neurology, Wuxi Children's Hospital, Wuxi, Jiangsu Province, China
| | - Miao Jing
- Department of Neurology, Wuxi Children's Hospital, Wuxi, Jiangsu Province, China
| | - Jun Feng
- Department of Neurology, Children's Hospital of Soochow University, Suzhou, Jiangsu Province, China
| | - Jihong Tang
- Department of Neurology, Children's Hospital of Soochow University, Suzhou, Jiangsu Province, China
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33
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Gstoettner C, Mayer JA, Rassam S, Hruby LA, Salminger S, Sturma A, Aman M, Harhaus L, Platzgummer H, Aszmann OC. Neuralgic amyotrophy: a paradigm shift in diagnosis and treatment. J Neurol Neurosurg Psychiatry 2020; 91:879-888. [PMID: 32487526 DOI: 10.1136/jnnp-2020-323164] [Citation(s) in RCA: 70] [Impact Index Per Article: 14.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/07/2020] [Revised: 05/07/2020] [Accepted: 05/10/2020] [Indexed: 01/22/2023]
Abstract
Neuralgic amyotrophy (NA), also known as Parsonage-Turner syndrome, is characterised by sudden pain attacks, followed by patchy muscle paresis in the upper extremity. Recent reports have shown that incidence is much higher than previously assumed and that the majority of patients never achieve full recovery. Traditionally, the diagnosis was mainly based on clinical observations and treatment options were confined to application of corticosteroids and symptomatic management, without proven positive effects on long-term outcomes. These views, however, have been challenged in the last years. Improved imaging methods in MRI and high-resolution ultrasound have led to the identification of structural peripheral nerve pathologies in NA, most notably hourglass-like constrictions. These pathognomonic findings have paved the way for more accurate diagnosis through high-resolution imaging. Furthermore, surgery has shown to improve clinical outcomes in such cases, indicating the viability of peripheral nerve surgery as a valuable treatment option in NA. In this review, we present an update on the current knowledge on this disease, including pathophysiology and clinical presentation, moving on to diagnostic and treatment paradigms with a focus on recent radiological findings and surgical reports. Finally, we present a surgical treatment algorithm to support clinical decision making, with the aim to encourage translation into day-to-day practice.
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Affiliation(s)
- Clemens Gstoettner
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria
| | - Johannes A Mayer
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department for Hand, Plastic, Reconstructive and Burn Surgery, BG Trauma Center Tuebingen at the Eberhard Karls University Tuebingen, Tuebingen, Germany
| | - Stephanie Rassam
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of General, Visceral, Endocrine and Transplantation Surgery, Cantonal Hospital St. Gallen, St. Gallen, Switzerland
| | - Laura A Hruby
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of Orthopaedics and Trauma Surgery, Medical University of Vienna, Vienna, Austria
| | - Stefan Salminger
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Division of Plastic and Reconstructive Surgery, Department of Surgery, Medical University of Vienna, Vienna, Austria
| | - Agnes Sturma
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of Bioengineering, Imperial College London, London, UK
| | - Martin Aman
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria.,Department of Hand, Plastic and Reconstructive Surgery, Burn Center-Hand and Plastic Surgery, University of Heidelberg, BG Trauma Center Ludwigshafen, Ludwigshafen, Germany
| | - Leila Harhaus
- Department of Hand, Plastic and Reconstructive Surgery, Burn Center-Hand and Plastic Surgery, University of Heidelberg, BG Trauma Center Ludwigshafen, Ludwigshafen, Germany
| | - Hannes Platzgummer
- Department of Biomedical Imaging and Image-guided Therapy, Medical University of Vienna, Vienna, Austria
| | - Oskar C Aszmann
- Clinical Laboratory for Bionic Extremity Reconstruction, Department of Surgery, Medical University of Vienna, Vienna, Austria .,Division of Plastic and Reconstructive Surgery, Department of Surgery, Medical University of Vienna, Vienna, Austria
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Goetsch MR, Shen J, Jones JA, Memon A, Chatham W. Neuralgic Amyotrophy Presenting with Multifocal Myonecrosis and Rhabdomyolysis. Cureus 2020; 12:e7382. [PMID: 32337110 PMCID: PMC7179979 DOI: 10.7759/cureus.7382] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/29/2022] Open
Abstract
Neuralgic amyotrophy (NA), also known as Parsonage-Turner syndrome, is an idiopathic disorder characterized by rapid-onset unilateral upper extremity pain, paralysis, and sensory disturbance in the distribution of the brachial plexus. The etiology is unknown, and there is a multitude of alternative clinical presentations as well as secondary triggers, which make the diagnosis challenging. To date, there has been no report of NA presenting with frank myonecrosis. In this report, we document the first case of NA presenting with multifocal myonecrosis of the shoulder girdle muscles and rhabdomyolysis. This case posed a unique challenge in the diagnostic workup and management as many causes of myonecrosis present similarly to NA, and NA is a diagnosis of exclusion. Our patient underwent exhaustive testing and several trials of therapy before diagnosis could be made. Such evaluations are expensive and carry risks for patients. As such, it is important that physicians recognize this unique presentation of NA.
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Affiliation(s)
| | - Jeffrey Shen
- Internal Medicine, University of Alabama at Birmingham, Birmingham, USA
| | - Jeffrey A Jones
- Pathology, University of Alabama at Birmingham, Birmingham, USA
| | - Adeel Memon
- Neurology, University of Alabama at Birmingham, Birmingham, USA
| | - Walter Chatham
- Medicine, University of Alabama at Birmingham, Birmingham, USA
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35
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Manfredi M, Gismondi P, Iuliano S, Maffini V, Scivales S, Gargano G. A rare presentation of neuralgic amyotrophy in a child and a review of recent literature. J Int Med Res 2019; 47:5817-5823. [PMID: 31526173 PMCID: PMC6862908 DOI: 10.1177/0300060519868632] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/02/2022] Open
Abstract
Neuralgic amyotrophy (NA), also known as brachial neuritis and previously known as Parsonage–Turner Syndrome, has an unknown etiology. Patients with NA have a clinical pattern characterized by sudden and acute pain across the shoulder followed by flaccid paralysis. NA has an incidence of one new case per 1000 people per year with an onset of age ranging between 20 and 60 years. We describe a rare presentation of NA in a Caucasian boy who was 11 years old and did not have any other family members affected by NA. All diagnostic studies were normal and he had full recovery 5 months from the onset of symptoms. We revised the recent literature of NA. No specific diagnostic studies can confirm the diagnosis of NA, although magnetic resonance imaging or electrophysiological studies can highlight some special features. Treatment of NA is symptomatic and it is based on analgesic drugs and physical therapy, although early administration of steroids appears to improve the outcome. Prognosis of NA is generally favorable with full recovery usually within 2 years. This disease is typically an adult syndrome, but pediatricians should also be aware of this entity to avoid delays in diagnosis.
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Affiliation(s)
- Marco Manfredi
- Pediatric Unit, Maternal and Child Department, Azienda USL-IRCCS of Reggio Emilia, Sant'Anna Hospital, Castelnovo ne' Monti, Italy
| | - Pierpacifico Gismondi
- Department of Pediatrics, "Pietro Barilla" Children's Hospital, University Hospital, Parma, Italy
| | - Silvia Iuliano
- Department of Pediatrics, "Pietro Barilla" Children's Hospital, University Hospital, Parma, Italy
| | - Valentina Maffini
- Department of Pediatrics, "Pietro Barilla" Children's Hospital, University Hospital, Parma, Italy
| | - Sonya Scivales
- Pediatric Unit, Maternal and Child Department, Azienda USL-IRCCS of Reggio Emilia, Sant'Anna Hospital, Castelnovo ne' Monti, Italy
| | - Giancarlo Gargano
- Head of Maternal and Child Department, Azienda USL-IRCCS of Reggio Emilia, Reggio Emilia, Italy
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36
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Lustenhouwer R, van Alfen N, Cameron IGM, Toni I, Geurts ACH, Helmich RC, van Engelen BGM, Groothuis JT. NA-CONTROL: a study protocol for a randomised controlled trial to compare specific outpatient rehabilitation that targets cerebral mechanisms through relearning motor control and uses self-management strategies to improve functional capability of the upper extremity, to usual care in patients with neuralgic amyotrophy. Trials 2019; 20:482. [PMID: 31391076 PMCID: PMC6686223 DOI: 10.1186/s13063-019-3556-4] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/11/2019] [Accepted: 07/04/2019] [Indexed: 11/12/2022] Open
Abstract
Background Neuralgic amyotrophy (NA) is a distinct peripheral neurological disorder of the brachial plexus with a yearly incidence of 1/1000, which is characterised by acute severe upper extremity pain. Weakness of the stabilising shoulder muscles in the acute phase leads to compensatory strategies and abnormal motor control of the shoulder - scapular dyskinesia. Despite peripheral nerve recovery, scapular dyskinesia often persists, leading to debilitating residual complaints including pain and fatigue. Evidence suggests that persistent scapular dyskinesia in NA may result from maladaptive cerebral neuroplasticity, altering motor planning. Currently there is no proven effective causative treatment for the residual symptoms in NA. Moreover, the role of cerebral mechanisms in persistent scapular dyskinesia remains unclear. Methods NA-CONTROL is a single-centre, randomised controlled trial comparing specific rehabilitation to usual care in NA. The rehabilitation programme combines relearning of motor control, targeting cerebral mechanisms, with self-management strategies. Fifty patients will be included. Patients are recruited through the Radboud university medical center Nijmegen, the Netherlands. Patients with a (suspected) diagnosis of NA, with lateralized symptoms and scapular dyskinesia in the right upper extremity, who are 18 years or older and not in the acute phase can be included. The primary outcome is the Shoulder Rating Questionnaire score, which measures functional capability of the upper extremity. Secondary clinical outcomes include measures of pain, fatigue, participation, reachable workspace, muscle strength and quality of life. In addition, motor planning is assessed with first-person motor imagery and functional magnetic resonance imaging. In a sub-study the patients are compared to 25 healthy participants, to determine the involvement of cerebral mechanisms. This will enable interpretation of cerebral changes associated with the rehabilitation programme and functional impairments in NA. Discussion NA-CONTROL is the first randomised trial to investigate the effect of specific rehabilitation on residual complaints in NA. It also is the first study into the cerebral mechanisms that might underlie persistent scapular dyskinesia in NA. It thus may aid the further development of mechanism-based interventions for disturbed motor control in NA and in other peripheral neurological disorders. Trial registration ClinicalTrials.gov, NCT03441347. Registered on 20 February 2018. Electronic supplementary material The online version of this article (10.1186/s13063-019-3556-4) contains supplementary material, which is available to authorized users.
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Affiliation(s)
- Renee Lustenhouwer
- Department of Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands.,Donders Centre for Cognitive Neuroimaging, Donders Institute for Brain, Cognition and Behaviour, Radboud University, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Nens van Alfen
- Department of Neurology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Ian G M Cameron
- Donders Centre for Cognitive Neuroimaging, Donders Institute for Brain, Cognition and Behaviour, Radboud University, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Ivan Toni
- Donders Centre for Cognitive Neuroimaging, Donders Institute for Brain, Cognition and Behaviour, Radboud University, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Alexander C H Geurts
- Department of Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Rick C Helmich
- Donders Centre for Cognitive Neuroimaging, Donders Institute for Brain, Cognition and Behaviour, Radboud University, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands.,Department of Neurology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Baziel G M van Engelen
- Department of Neurology, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands
| | - Jan T Groothuis
- Department of Rehabilitation, Donders Institute for Brain, Cognition and Behaviour, Radboud University Medical Center, P.O. Box 9101, 6500 HB, Nijmegen, the Netherlands.
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Neubauer K, Boeckelmann D, Koehler U, Kracht J, Kirschner J, Pendziwiat M, Zieger B. Hereditary neuralgic amyotrophy in childhood caused by duplication within the SEPT9 gene: A family study. Cytoskeleton (Hoboken) 2018; 76:131-136. [PMID: 30019529 PMCID: PMC6585727 DOI: 10.1002/cm.21479] [Citation(s) in RCA: 9] [Impact Index Per Article: 1.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/22/2018] [Revised: 06/28/2018] [Accepted: 07/10/2018] [Indexed: 01/24/2023]
Abstract
Hereditary neuralgic amyotrophy (HNA) is an autosomal dominant disorder associated with episodic, recurrent, and painful neuropathies affecting the nerves of the brachial plexus. In this study, we report on a family of Lebanese descent with HNA onset in early childhood. The affected family members presented with platelet dysfunction. Platelet aggregation was reduced after stimulation with the agonists ADP and epinephrine in all affected family members. Flow cytometric analyses revealed impaired platelet δ‐secretion. The index patient and one brother suffered from kidney cysts. Molecular genetic analysis revealed a heterozygous duplication of exon 2 within the septin 9 (SEPT9) gene in all the affected family members. Such a young child with HNA (aged 2 years) caused by SEPT9 duplication has not been described so far.
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Affiliation(s)
- Katharina Neubauer
- Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, University Medical Center, Medical Faculty, University of Freiburg, Freiburg, Germany
| | - Doris Boeckelmann
- Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, University Medical Center, Medical Faculty, University of Freiburg, Freiburg, Germany
| | - Udo Koehler
- Department of Medical Genetics, MGZ - Medical Genetics Center, Munich, Germany
| | - Julia Kracht
- Division of General Pediatrics, Department of Pediatrics and Adolescent Medicine, University Medical Center, Medical Faculty, University of Freiburg, Freiburg, Germany
| | - Janbernd Kirschner
- Department of Neuropediatrics and Muscle Disorders, University Medical Center, Faculty of Medicine, University of Freiburg, Germany
| | - Manuela Pendziwiat
- Department of Neuropediatrics, Christian-Albrechts-University of Kiel and University Medical Center Schleswig-Holstein, Kiel, Germany
| | - Barbara Zieger
- Division of Pediatric Hematology and Oncology, Department of Pediatrics and Adolescent Medicine, University Medical Center, Medical Faculty, University of Freiburg, Freiburg, Germany
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38
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Morishima R, Nagaoka U, Nagao M, Isozaki E. Chronic Brachial Plexus Neuritis That Developed into Typical Neuralgic Amyotrophy and Positively Responded to Immunotherapy. Intern Med 2018; 57:1021-1026. [PMID: 29269655 PMCID: PMC5919865 DOI: 10.2169/internalmedicine.9482-17] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022] Open
Abstract
Based on the hypothesis that autoimmunity plays a role in the pathogenesis of neuralgic amyotrophy (NA), immunotherapy is sometimes administered. Early intervention is recommended for a good prognosis. We herein report the case of a 55-year-old man who presented with neuralgia, weakness, and muscle atrophy in his right shoulder girdle and upper arm, which progressed for ten months following a marine sports accident. The patient was diagnosed with NA. His neurological deficits gradually improved after several courses of immunotherapy, suggesting that in addition to being effective for treating early-stage disease, immunotherapy may be effective for treating chronic cases.
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Affiliation(s)
- Ryo Morishima
- Department of Neurology, Tokyo Metropolitan Neurological Hospital, Japan
| | - Utako Nagaoka
- Department of Neurology, Tokyo Metropolitan Neurological Hospital, Japan
| | - Masahiro Nagao
- Department of Neurology, Tokyo Metropolitan Neurological Hospital, Japan
| | - Eiji Isozaki
- Department of Neurology, Tokyo Metropolitan Neurological Hospital, Japan
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39
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Acute Shoulder Pain Followed by Shoulder Weakness and Atrophy: A Characteristic Presentation of Parsonage-Turner Syndrome. J Gen Intern Med 2018; 33:231. [PMID: 28956267 PMCID: PMC5789099 DOI: 10.1007/s11606-017-4187-7] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/06/2017] [Revised: 07/12/2017] [Accepted: 09/06/2017] [Indexed: 10/18/2022]
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40
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Phrenic Nerve Palsy Secondary to Parsonage–Turner Syndrome: A Diagnosis Commonly Overlooked. Lung 2017; 195:173-177. [DOI: 10.1007/s00408-016-9972-2] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/04/2016] [Accepted: 12/16/2016] [Indexed: 10/20/2022]
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41
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Cabezón I, Barreiro G, Egurbide MV. Parsonage-Turner syndrome: A case report. REUMATOLOGIA CLINICA 2017; 13:60-61. [PMID: 27079958 DOI: 10.1016/j.reuma.2016.03.001] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Received: 02/06/2016] [Revised: 02/27/2016] [Accepted: 03/04/2016] [Indexed: 06/05/2023]
Affiliation(s)
- Itxasne Cabezón
- Servicio de Medicina Interna, Hospital Universitario de Cruces, Baracaldo, Vizcaya, España.
| | - Guillermo Barreiro
- Servicio de Medicina Interna, Hospital Universitario de Cruces, Baracaldo, Vizcaya, España
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42
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Clarke CJ, Torrance E, McIntosh J, Funk L. Neuralgic amyotrophy is not the most common neurologic disorder of the shoulder: a 78-month prospective study of 60 neurologic shoulder patients in a specialist shoulder clinic. J Shoulder Elbow Surg 2016; 25:1997-2004. [PMID: 27282731 DOI: 10.1016/j.jse.2016.04.005] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 01/28/2016] [Revised: 03/31/2016] [Accepted: 04/05/2016] [Indexed: 02/01/2023]
Abstract
BACKGROUND Neuralgic amyotrophy (NA) was first described in 1948. Traditional literature describes a painful attack with sudden onset, followed by paresis, with varied outcomes. Recent studies have suggested NA is currently underdiagnosed. However, a large number of studies detailing NA originate from a small group of sources. Our study compared the onset, diagnosis, investigation, and treatment of all neurologic shoulder conditions to provide comparable data for these studies. METHODS Data were collected from 60 patients (81.6% male; median age, 41.5 years) during a 78-month period. Patients with a diagnosis of a neurologic disorder of the shoulder with confirmatory electromyogram (EMG) studies were included. RESULTS NA was diagnosed in 18 patients before the EMG investigation. Of the clinically diagnosed NA patients, only 5 (27.8%) had EMG findings supportive of NA. A further 5 patients with a clinical diagnosis other than NA were diagnosed with NA after EMG findings. Overall, 10 patients (16.6%) in our study were diagnosed with NA after EMG studies. Only 4 (40.0%) reported a sudden onset attack associated with NA. Supraspinatus and infraspinatus were involved in 9 patients (90.0%), suggesting a predictable distribution of muscle involvement. CONCLUSIONS These results suggest that NA is overdiagnosed and does not warrant the increased attention suggested by recent articles. The current study also highlights a necessity to perform EMG investigations in all cases of suspected NA because the accuracy of the clinical assessment is poor.
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Affiliation(s)
| | | | - Jerome McIntosh
- Manchester Medical School, University of Manchester, Manchester, UK
| | - Lennard Funk
- Wrightington Hospital, Appley Bridge, Lancashire, UK.
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43
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Chuk R, Sheppard M, Wallace G, Coman D. Pediatric Hereditary Neuralgic Amyotrophy: Successful Treatment With Intravenous Immunoglobulin and Insights Into SEPT9 Pathogenesis. Child Neurol Open 2016; 3:2329048X16668970. [PMID: 28503616 PMCID: PMC5417342 DOI: 10.1177/2329048x16668970] [Citation(s) in RCA: 5] [Impact Index Per Article: 0.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/13/2016] [Revised: 07/20/2016] [Accepted: 08/18/2016] [Indexed: 01/15/2023] Open
Abstract
Hereditary neuralgic amyotrophy is a rare disorder characterized by the sudden onset of recurrent episodes of painful brachial plexus neuropathies, followed by atrophy within a few weeks. The authors present the case of a 5-year-old boy who developed hereditary neuralgic amyotrophy in the right upper limb after a gastroenteritis illness. He made a full and rapid recovery with the use of intravenous immunoglobulin. A subsequent episode in the left upper limb during the course of intravenous immunoglobulin was significantly attenuated. A de novo c.262C>T mutation in exon 2 of the SEPT9 gene was identified. To our knowledge, he is the first pediatric patient with SEPT9 hereditary neuralgic amyotrophy to be treated with intravenous immunoglobulin. The authors hypothesize that the c.262C>T mutation in exon 2 of the SEPT9 gene generates pathology via the numerous isoforms under specific conditions and that intravenous immunoglobulin can play a role at the epigenetic level of improving dysfunctional SEPT9 expression.
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Affiliation(s)
- Raymond Chuk
- Discipline of Paediatrics, UnitingCare Health Clinical School, Wesley Hospital, Brisbane, Queensland, Australia.,Department of Paediatrics, Wesley Hospital, Brisbane, Queensland, Australia.,School of Medicine, University of Queensland, Queensland, Australia
| | - Megan Sheppard
- Department of Neurosciences, Lady Cilento Children's Hospital, Brisbane, Queensland, Australia
| | - Geoff Wallace
- School of Medicine, University of Queensland, Queensland, Australia.,Department of Neurosciences, Lady Cilento Children's Hospital, Brisbane, Queensland, Australia
| | - David Coman
- Discipline of Paediatrics, UnitingCare Health Clinical School, Wesley Hospital, Brisbane, Queensland, Australia.,Department of Paediatrics, Wesley Hospital, Brisbane, Queensland, Australia.,School of Medicine, University of Queensland, Queensland, Australia.,Department of Neurosciences, Lady Cilento Children's Hospital, Brisbane, Queensland, Australia.,School of Medicine, Griffith University, Gold Coast, Queensland, Australia
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44
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Milner CS, Kannan K, Iyer VG, Thirkannad SM. Parsonage-Turner Syndrome: Clinical and Epidemiological Features From a Hand Surgeon's Perspective. Hand (N Y) 2016; 11:227-31. [PMID: 27390568 PMCID: PMC4920531 DOI: 10.1177/1558944715627246] [Citation(s) in RCA: 14] [Impact Index Per Article: 1.6] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 11/15/2022]
Abstract
BACKGROUND Parsonage-Turner Syndrome (PTS) is a rare but serious condition characterized by spontaneous paresis of the upper extremity, typically lasting several months with variable recovery. With little literature on the behavior of PTS from a hand surgeon's perspective, accurate diagnosis and subsequent counseling of patients with PTS can be challenging. METHODS This study is a retrospective evaluation of the clinical features of all PTS patients seen over a 9-year period. Data was collected for gender, side affected, handedness, inciting event, clinical presentation, nerve involvement, time taken for recovery, and extent of recovery. RESULTS Thirty-eight adult cases of PTS were identified, representing an incidence of 0.34 per 1000. Fifty-five percent were female, with predomination of right handedness and dominant upper extremity involvement (60% and 58%, respectively). There was an inciting incident identified in 42% of cases, and 37% of these involved surgery. Twenty-nine percent of cases presented without experiencing typical neuropathic pain. There was a predomination of anterior interosseous nerve (AIN) or posterior interosseous nerve (PIN) involvement (42% overall). Only 44% of patients achieved a complete recovery, taking a mean duration of 10 months. CONCLUSIONS This study highlights both the rarity and atypical spectrum of clinical presentation of PTS, especially considering the more common involvement found for AIN and PIN. This highlights the likelihood that patients presenting with PTS to a hand surgical practice may differ from those typically described in the literature. The poor rate of recovery is in line with other recent reports and contrasts with the more positive outcomes found in earlier studies.
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Affiliation(s)
| | - Kumar Kannan
- Christine M. Kleinert Institute, Louisville, KY, USA
| | | | - Sunil M. Thirkannad
- Christine M. Kleinert Institute, Louisville, KY, USA
- Sunil M. Thirkannad, Christine M. Kleinert Institute, Suite 850, 225 Abraham Flexner Way, Louisville, KY 40202, USA.
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45
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Dalton HR, Kamar N, van Eijk JJJ, Mclean BN, Cintas P, Bendall RP, Jacobs BC. Hepatitis E virus and neurological injury. Nat Rev Neurol 2015; 12:77-85. [PMID: 26711839 DOI: 10.1038/nrneurol.2015.234] [Citation(s) in RCA: 166] [Impact Index Per Article: 16.6] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 12/20/2022]
Abstract
Hepatitis E is hyperendemic in many developing countries in Asia and Africa, and is caused by hepatitis E virus (HEV) genotypes 1 and 2, which are spread via the faecal-oral route by contaminated water. Recent data show that HEV infection is also endemic in developed countries. In such geographical settings, hepatitis E is caused by HEV genotypes 3 and 4, and is mainly a porcine zoonosis. In a minority of cases, HEV causes acute and chronic hepatitis, but infection is commonly asymptomatic or unrecognized. HEV infection is associated with a number of extrahepatic manifestations, including a range of neurological injuries. To date, 91 cases of HEV-associated neurological injury--most commonly, Guillain-Barré syndrome, neuralgic amyotrophy, and encephalitis/myelitis--have been reported. Here, we review the reported cases, discuss possible pathogenic mechanisms, and present our perspectives on future directions and research questions.
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Affiliation(s)
- Harry R Dalton
- Royal Cornwall Hospital, University of Exeter, Penventinnie Lane, Truro TR1 3LJ, UK.,European Centre for Environment &Human Health, University of Exeter, Penventinnie Lane, Truro TR1 3LJ, UK
| | - Nassim Kamar
- Departments of Nephrology and Organ Transplantation, CHU Rangueil, INSERM U1043, IFR-BMT, Université Paul Sabatier, 1 Avenue J. Poulhès, Toulouse Cedex 9, France
| | - Jeroen J J van Eijk
- Department of Neurology, Jeroen Bosch Ziekenhuis (JBZ), Henri Dunantstraat 1, 5223 GZ, 's-Hertogenbosch, Netherlands
| | - Brendan N Mclean
- Royal Cornwall Hospital, University of Exeter, Penventinnie Lane, Truro TR1 3LJ, UK
| | - Pascal Cintas
- Department of Neurology, Pierre Paul Riquet Hospital, CHU Purpan, Place du Dr Baylac, 31059 Toulouse, France
| | - Richard P Bendall
- Royal Cornwall Hospital, University of Exeter, Penventinnie Lane, Truro TR1 3LJ, UK.,European Centre for Environment &Human Health, University of Exeter, Penventinnie Lane, Truro TR1 3LJ, UK
| | - Bart C Jacobs
- Department of Neurology/Neuro-Immunology Erasmus MC, Erasmus MC, Afdeling Neurologie, Kamer EE 2287, Postbus 2040, 3000 CA Rotterdam, Netherlands
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46
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Chrestian N, McMillan H, Poulin C, Campbell C, Vajsar J. Hereditary neuropathy with liability to pressure palsies in childhood: Case series and literature update. Neuromuscul Disord 2015; 25:693-8. [PMID: 26189194 DOI: 10.1016/j.nmd.2015.06.463] [Citation(s) in RCA: 22] [Impact Index Per Article: 2.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/28/2015] [Revised: 06/08/2015] [Accepted: 06/26/2015] [Indexed: 11/25/2022]
Abstract
Hereditary Neuropathy with Liability to Pressure Palsy (HNPP) is a rare condition in childhood with a diverse range of clinical presentations. We analyzed the clinical presentation and electrophysiological data of 12 children with a confirmed PMP22 gene deletion and reviewed the published reports of HNPP in children and compared our data with the reports from the literature review. Peroneal palsy was the most common presentation (42%) followed by brachial plexus palsy in 25% of our cases. Nerve conduction studies were always suggestive of the diagnosis demonstrating 3 major patterns: multifocal demyelination at the area of entrapment, generalized sensory-motor polyneuropathy and a combination of the two first patterns in a vast majority (60%). Surprisingly, there was bilateral or unilateral electrophysiological entrapment of the median nerve at the carpal tunnel in all our patients. The clinical presentation of HNPP in childhood is heterogeneous and electrophysiological findings are helpful in establishing the diagnosis. Any unexplained mononeuropathy or multifocal neuropathy should lead to PMP22 gene testing to look for the deletion. Early diagnosis is important in order to facilitate appropriate genetic counseling and also for the appropriate care for these patients.
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Affiliation(s)
- Nicolas Chrestian
- The Hospital for Sick Children, University of Toronto, 555 University Avenue, Toronto, Ontario M5G 1X8, Canada.
| | - Hugh McMillan
- Children's Hospital of Eastern Ontario, University of Ottawa, 401 Smyth Road, Ottawa, Ontario K1H 8L1, Canada
| | - Chantal Poulin
- The Montreal Children's Hospital, McGill University, 1001 Boulevard Décarie, Montreal, Quebec H4A 3J1, Canada
| | - Craig Campbell
- Children's Health Research Institute Western University, 800 Commissioners Road East, London, Ontario N6C 2V5, Canada
| | - Jiri Vajsar
- The Hospital for Sick Children, University of Toronto, 555 University Avenue, Toronto, Ontario M5G 1X8, Canada
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Incidence of neuralgic amyotrophy (Parsonage Turner syndrome) in a primary care setting--a prospective cohort study. PLoS One 2015; 10:e0128361. [PMID: 26016482 PMCID: PMC4445915 DOI: 10.1371/journal.pone.0128361] [Citation(s) in RCA: 101] [Impact Index Per Article: 10.1] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 01/07/2015] [Accepted: 04/26/2015] [Indexed: 11/19/2022] Open
Abstract
OBJECTIVE Neuralgic amyotrophy is considered a rare peripheral nervous system disorder but in practice seems grossly under recognized, which negatively affects care for these patients. In this study we prospectively counted the one-year incidence rate of classic neuralgic amyotrophy in a primary care setting. METHODS In a prospective cohort study during the year 2012 we registered all new cases of neck, shoulder or arm complaints from two large primary care centers serving a population of 14,118. Prior to study, general practitioners received a short training on how to diagnose classic neuralgic amyotrophy. Neuralgic amyotrophy was defined according to published criteria irrespective of family history. Only patients with a classic phenotype were counted as definite cases. After inclusion, patients with suspected neuralgic amyotrophy who had not yet seen a neurologist were offered neurologic evaluation for diagnostic confirmation. RESULTS Of the 492 patients identified with new onset neck, shoulder or arm complaints, 34 were suspected of having neuralgic amyotrophy. After neurologic evaluation the diagnosis was confirmed in 14 patients. This amounts to a one-year incidence rate for classic neuralgic amyotrophy of 1 per 1000. CONCLUSIONS Our findings suggest that neuralgic amyotrophy is 30-50 times more common than previously thought. Unawareness of the disorder and its clinical presentation seems the most likely explanation for this difference. An incidence rate of 1 per 1000 and the long-term sequelae many patients suffer warrant more vigilance in diagnosing the disorder, to pave the way for timely treatment and prevent complications.
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48
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Jain S, Bhatt GC, Rai N, Bhan BD. Idiopathic brachial neuritis in a child: A case report and review of the literature. J Pediatr Neurosci 2015; 9:276-7. [PMID: 25624937 PMCID: PMC4302554 DOI: 10.4103/1817-1745.147593] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/10/2022] Open
Abstract
Brachial neuritis is a rare disease in children, affecting mainly the lower motor neurons of the brachial plexus and/or individual nerves or nerve branches. We report a case of idiopathic brachial plexus neuritis in a 2½-year-old female child admitted with acute respiratory distress and given antibiotic therapy following which she developed weakness of the left hand. She was diagnosed as a case of idiopathic brachial plexus neuritis and was given supportive care. Although, the association with antibiotic therapy in this case could be incidental, indeed it is intriguing and requires further studies.
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Affiliation(s)
- Shikha Jain
- Department of Pediatrics, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India
| | - Girish Chandra Bhatt
- Department of Pediatrics, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India
| | - Nirendra Rai
- Department of Neurology, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India
| | - Bhavna Dhingra Bhan
- Department of Pediatrics, All India Institute of Medical Sciences, Bhopal, Madhya Pradesh, India
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49
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Mulla SM, Buckley DN, Moulin DE, Couban R, Izhar Z, Agarwal A, Panju A, Wang L, Kallyth SM, Turan A, Montori VM, Sessler DI, Thabane L, Guyatt GH, Busse JW. Management of chronic neuropathic pain: a protocol for a multiple treatment comparison meta-analysis of randomised controlled trials. BMJ Open 2014; 4:e006112. [PMID: 25412864 PMCID: PMC4244486 DOI: 10.1136/bmjopen-2014-006112] [Citation(s) in RCA: 13] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 07/15/2014] [Revised: 10/14/2014] [Accepted: 10/31/2014] [Indexed: 11/04/2022] Open
Abstract
INTRODUCTION Chronic neuropathic pain is associated with reduced health-related quality of life and substantial socioeconomic costs. Current research addressing management of chronic neuropathic pain is limited. No review has evaluated all interventional studies for chronic neuropathic pain, which limits attempts to make inferences regarding the relative effectiveness of treatments. METHODS AND ANALYSIS We will conduct a systematic review of all randomised controlled trials evaluating therapies for chronic neuropathic pain. We will identify eligible trials, in any language, by a systematic search of CINAHL, EMBASE, MEDLINE, AMED, HealthSTAR, DARE, PsychINFO and the Cochrane Central Registry of Controlled Trials. Eligible trials will be: (1) enrol patients presenting with chronic neuropathic pain, and (2) randomise patients to alternative interventions (pharmacological or non-pharmacological) or an intervention and a control arm. Pairs of reviewers will, independently and in duplicate, screen titles and abstracts of identified citations, review the full texts of potentially eligible trials and extract information from eligible trials. We will use a modified Cochrane instrument to evaluate risk of bias of eligible studies, recommendations from the Initiative on Methods, Measurement, and Pain Assessment in Clinical Trials (IMMPACT) to inform the outcomes we will collect, and the Grading of Recommendations Assessment, Development and Evaluation (GRADE) system to evaluate our confidence in treatment effects. When possible, we will conduct: (1) in direct comparisons, a random-effects meta-analysis to establish the effect of reported therapies on patient-important outcomes; and (2) a multiple treatment comparison meta-analysis within a Bayesian framework to assess the relative effects of treatments. We will define a priori hypotheses to explain heterogeneity between studies, and conduct meta-regression and subgroup analyses consistent with the current best practices. ETHICS AND DISSEMINATION We do not require ethics approval for our proposed review. We will disseminate our findings through peer-reviewed publications and conference presentations. TRIAL REGISTRATION NUMBER PROSPERO (CRD42014009212).
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Affiliation(s)
- Sohail M Mulla
- Department of Clinical Epidemiology & Biostatistics, McMaster University, Hamilton, Ontario, Canada
- Outcomes Research Consortium, Cleveland Clinic, Cleveland, Ohio, USA
| | - D Norman Buckley
- Department of Anesthesia, McMaster University, Hamilton, Ontario, Canada
- Michael G. DeGroote Institute for Pain Research and Care, McMaster University, Hamilton, Ontario, Canada
| | - Dwight E Moulin
- Departments of Clinical Neurological Sciences and Oncology, Western University, London, Ontario, Canada
| | - Rachel Couban
- Michael G. DeGroote Institute for Pain Research and Care, McMaster University, Hamilton, Ontario, Canada
| | - Zain Izhar
- Department of Clinical Epidemiology & Biostatistics, McMaster University, Hamilton, Ontario, Canada
| | - Arnav Agarwal
- Department of Clinical Epidemiology & Biostatistics, McMaster University, Hamilton, Ontario, Canada
| | - Akbar Panju
- Michael G. DeGroote Institute for Pain Research and Care, McMaster University, Hamilton, Ontario, Canada
- Department of Medicine, McMaster University, Hamilton, Ontario, Canada
| | - Li Wang
- Department of Anesthesia, McMaster University, Hamilton, Ontario, Canada
| | - Sun Makosso Kallyth
- Michael G. DeGroote Institute for Pain Research and Care, McMaster University, Hamilton, Ontario, Canada
| | - Alparslan Turan
- Department of Outcomes Research, Cleveland Clinic, Cleveland, Ohio, USA
| | - Victor M Montori
- Knowledge and Evaluation Research Unit, Divisions of Endocrinology and Diabetes, and Health Care & Policy Research, Mayo Clinic, Rochester, Minnesota, USA
| | - Daniel I Sessler
- Department of Outcomes Research, Cleveland Clinic, Cleveland, Ohio, USA
| | - Lehana Thabane
- Department of Clinical Epidemiology & Biostatistics, McMaster University, Hamilton, Ontario, Canada
- Department of Anesthesia, McMaster University, Hamilton, Ontario, Canada
- Department of Pediatrics, McMaster University, Hamilton, Ontario, Canada
| | - Gordon H Guyatt
- Department of Clinical Epidemiology & Biostatistics, McMaster University, Hamilton, Ontario, Canada
- Department of Medicine, McMaster University, Hamilton, Ontario, Canada
| | - Jason W Busse
- Department of Clinical Epidemiology & Biostatistics, McMaster University, Hamilton, Ontario, Canada
- Department of Anesthesia, McMaster University, Hamilton, Ontario, Canada
- Michael G. DeGroote Institute for Pain Research and Care, McMaster University, Hamilton, Ontario, Canada
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Fransz DP, Schönhuth CP, Postma TJ, van Royen BJ. Parsonage-Turner syndrome following post-exposure prophylaxis. BMC Musculoskelet Disord 2014; 15:265. [PMID: 25098693 PMCID: PMC4126347 DOI: 10.1186/1471-2474-15-265] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/12/2013] [Accepted: 07/14/2014] [Indexed: 11/10/2022] Open
Abstract
BACKGROUND The 'Parsonage-Turner syndrome' (PTS) is a rare but distinct disorder with an abrupt onset of shoulder pain, followed by weakness and atrophy of the upper extremity musculature, and a slow recovery requiring months to years. To our best knowledge, this is the first case describing symptoms and signs of PTS following the administration of a post-exposure prophylaxis (PEP) regimen against possible human immunodeficiency virus (HIV) and hepatitis B virus (HBV) infection. CASE PRESENTATION A 25-year-old Caucasian man presented with pain and unilateral scapular winging following PEP against possible HIV and HBV infection. Although atrophy and weakness were observed for the right supraspinatus muscle, a full range of motion was achievable. Neurological examination, plain radiography of the right shoulder and electromyography showed no additional abnormalities. The patient was diagnosed with post-vaccination PTS and treated non-operatively. During the following 15 months the scapular winging receded and full muscle strength was regained. CONCLUSION Parsonage-Turner syndrome is a rare clinical diagnosis. The precise pathophysiological mechanism of PTS remains unclear, but it seems to involve an interaction between genetic predisposition, mechanical vulnerability and an autoimmune trigger. An immunological event, such as - in this case - a vaccination as part of PEP treatment, can trigger the onset of PTS. The clinical presentation is distinctive with acute severe pain followed by patchy paresis, atrophy and sensory symptoms that persist for months to years. No currently available tests can provide a definite confirmation or exclusion of PTS. Routine blood examination, electromyography (EMG), and computed tomography (CT) or magnetic resonance imaging (MRI) serve mainly to exclude other disorders. The recovery can be quite lengthy, non-operative treatment is the accepted practice. Supplementary administration of oral prednisolone could shorten the duration of pain. Although the outcome is typically preferable, a substantial amount of patients are left with some residual paresis and functional impairment.
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Affiliation(s)
| | - Casper P Schönhuth
- Department of Orthopaedic Surgery, VU University Medical Center, PO Box 7057, 1007 MB Amsterdam, The Netherlands.
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