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Wen GJ, Chen J, Zhang SF, Zhou ZS, Jiao GL. Multiple sparganosis spinal infections mainly in the thoracic region: A case report. World J Clin Cases 2023; 11:8507-8511. [PMID: 38188209 PMCID: PMC10768500 DOI: 10.12998/wjcc.v11.i36.8507] [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: 08/20/2023] [Revised: 11/14/2023] [Accepted: 12/12/2023] [Indexed: 12/22/2023] Open
Abstract
BACKGROUND Spinal infection with sparganosis is rarely seen, and multiple spinal infections with sparganosis in the thoracic spine have not been reported. CASE SUMMARY In this case report, a 56-year old male patient suffered from back pain for 3 mo. Computed tomography examination of the thoracic spine showed bone destruction of the T4-5 vertebral body, as well as the right pedicle and lamina of T5. Magnetic resonance imaging showed high signals on T2W1 images and fat-suppressed images in the right vertebral body of T4-5 and the right pedicle and lamina of T5, a high signal in the vertebral canal, and similar high signals in the paravertebral and subcutaneous regions of the whole spine. Puncture biopsy showed sparganosis. Following definite diagnosis, the patient was treated with debridement of T4-5 infected lesions under a microscope, bone grafting and internal fixation. Postoperatively, the patient's back pain symptoms were significantly relieved; the incision healed after one-stage treatment, and albendazole antiparasitic treatment was administered. CONCLUSION Puncture biopsy is the most reliable method to diagnose infection by sparganum. Removal of infected lesions under the microscope and albendazole for antiparasitic treatment are safe and effective.
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Affiliation(s)
- Gan-Jun Wen
- Department of Orthopaedics, The Sixth Affiliated Hospital of Jinan University, Dongguan 523573, Guangdong Province, China
| | - Jian Chen
- Department of Orthopaedics, The Sixth Affiliated Hospital of Jinan University, Dongguan 523573, Guangdong Province, China
| | - Shi-Fei Zhang
- Department of Orthopaedics, The Sixth Affiliated Hospital of Jinan University, Dongguan 523573, Guangdong Province, China
| | - Zhi-Sen Zhou
- Department of Orthopaedics, The Sixth Affiliated Hospital of Jinan University, Dongguan 523573, Guangdong Province, China
| | - Gen-Long Jiao
- Department of Orthopaedics, The Sixth Affiliated Hospital of Jinan University, Dongguan 523573, Guangdong Province, China
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Palackdkharry CS, Wottrich S, Dienes E, Bydon M, Steinmetz MP, Traynelis VC. The leptomeninges as a critical organ for normal CNS development and function: First patient and public involved systematic review of arachnoiditis (chronic meningitis). PLoS One 2022; 17:e0274634. [PMID: 36178925 PMCID: PMC9524710 DOI: 10.1371/journal.pone.0274634] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 02/02/2022] [Accepted: 08/31/2022] [Indexed: 11/28/2022] Open
Abstract
BACKGROUND & IMPORTANCE This patient and public-involved systematic review originally focused on arachnoiditis, a supposedly rare "iatrogenic chronic meningitis" causing permanent neurologic damage and intractable pain. We sought to prove disease existence, causation, symptoms, and inform future directions. After 63 terms for the same pathology were found, the study was renamed Diseases of the Leptomeninges (DLMs). We present results that nullify traditional clinical thinking about DLMs, answer study questions, and create a unified path forward. METHODS The prospective PRISMA protocol is published at Arcsology.org. We used four platforms, 10 sources, extraction software, and critical review with ≥2 researchers at each phase. All human sources to 12/6/2020 were eligible for qualitative synthesis utilizing R. Weekly updates since cutoff strengthen conclusions. RESULTS Included were 887/14286 sources containing 12721 DLMs patients. Pathology involves the subarachnoid space (SAS) and pia. DLMs occurred in all countries as a contributor to the top 10 causes of disability-adjusted life years lost, with communicable diseases (CDs) predominating. In the USA, the ratio of CDs to iatrogenic causes is 2.4:1, contradicting arachnoiditis literature. Spinal fusion surgery comprised 54.7% of the iatrogenic category, with rhBMP-2 resulting in 2.4x more DLMs than no use (p<0.0001). Spinal injections and neuraxial anesthesia procedures cause 1.1%, and 0.2% permanent DLMs, respectively. Syringomyelia, hydrocephalus, and arachnoid cysts are complications caused by blocked CSF flow. CNS neuron death occurs due to insufficient arterial supply from compromised vasculature and nerves traversing the SAS. Contrast MRI is currently the diagnostic test of choice. Lack of radiologist recognition is problematic. DISCUSSION & CONCLUSION DLMs are common. The LM clinically functions as an organ with critical CNS-sustaining roles involving the SAS-pia structure, enclosed cells, lymphatics, and biologic pathways. Cases involve all specialties. Causes are numerous, symptoms predictable, and outcomes dependent on time to treatment and extent of residual SAS damage. An international disease classification and possible treatment trials are proposed.
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Affiliation(s)
| | - Stephanie Wottrich
- Case Western Reserve School of Medicine, Cleveland, Ohio, United States of America
| | - Erin Dienes
- Arcsology®, Mead, Colorado, United States of America
| | - Mohamad Bydon
- Department of Neurologic Surgery, Orthopedic Surgery, and Health Services Research, Mayo Clinic School of Medicine, Rochester, Minnesota, United States of America
| | - Michael P. Steinmetz
- Department of Neurological Surgery, Cleveland Clinic Lerner College of Medicine Neurologic Institute, Cleveland, Ohio, United States of America
| | - Vincent C. Traynelis
- Department of Neurosurgery, Rush University School of Medicine, Chicago, Illinois, United States of America
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Chung TT, Hsu TH, Chen YA, Chung AH. Cauda equina syndrome secondary to extramedullary spinal sparganosis. FORMOSAN JOURNAL OF SURGERY 2020. [DOI: 10.4103/fjs.fjs_53_20] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022] Open
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Noiphithak R, Doungprasert G. A case of disseminated central nervous system sparganosis. Surg Neurol Int 2016; 7:S958-S961. [PMID: 28031991 PMCID: PMC5180432 DOI: 10.4103/2152-7806.195236] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/01/2016] [Accepted: 10/07/2016] [Indexed: 11/17/2022] Open
Abstract
Background: Sparganosis is a very rare parasitic infection in various organs caused by the larvae of tapeworms called spargana. The larva usually lodges in the central nervous system (CNS) and the orbit. However, lumbar spinal canal involvement, as noted in the present case, is extremely rare. We report a rare case of disseminated CNS sparganosis involving the brain and spinal canal and review the literature. Case Description: A 54-year-old man presented with progressive low back pain and neurological deficit at the lumbosacral level for 2 months. Imaging indicated arachnoiditis and an abnormal lesion at the L4-5 vertebral level. The patient underwent laminectomy of the L4-5 with lesionectomy and lysis of adhesions between the nerve roots. Microscopic examination indicated sparganum infection. Further brain imaging revealed evidence of chronic inflammation in the left parieto-occipital area without evidence of live parasites. In addition, an ophthalmologist reported a nonactive lesion in the right conjunctiva. The patient recovered well after surgery, although he had residual back pain and bladder dysfunction probably due to severe adhesion of the lumbosacral nerve roots. Conclusion: CNS sparganosis can cause various neurological symptoms similar to those of other CNS infections. A preoperative enzyme-linked immunosorbent assay is helpful for diagnosis, especially in endemic areas. Surgical removal of the worm remains the treatment of choice.
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Affiliation(s)
- Raywat Noiphithak
- Division of Neurosurgery, Department of Surgery, Thammasat University Hospital, Faculty of Medicine, Thammasat University, Pathumthani, Thailand
| | - Gahn Doungprasert
- Division of Neurosurgery, Department of Surgery, Thammasat University Hospital, Faculty of Medicine, Thammasat University, Pathumthani, Thailand
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Rahman SMM, Kim JH, Hong ST, Choi MH. Diagnostic efficacy of a recombinant cysteine protease of Spirometra erinacei larvae for serodiagnosis of sparganosis. THE KOREAN JOURNAL OF PARASITOLOGY 2014; 52:41-6. [PMID: 24623880 PMCID: PMC3948992 DOI: 10.3347/kjp.2014.52.1.41] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 09/02/2013] [Revised: 11/21/2013] [Accepted: 12/11/2013] [Indexed: 11/23/2022]
Abstract
The mature domain of a cysteine protease of Spirometra erinacei plerocercoid larva (i.e., sparganum) was expressed in Escherichia coli, and its value as an antigen for the serodiagnosis of sparganosis was investigated. The recombinant protein (rSepCp-1) has the molecular weight of 23.4 kDa, and strongly reacted with the sparganum positive human or mice sera but not with negative sera by immunoblotting. ELISA with rSepCp-1 protein or sparganum crude antigen (SeC) was evaluated for the serodiagnosis of sparganosis using patient's sera. The sensitivity and specificity of ELISA using rSepCp-1 protein were 95.0% (19/20) and 99.1% (111/112), respectively. In contrast, the sensitivity and specificity of ELISA with SeC were 100% (20/20) and 96.4% (108/112), respectively. Moreover, in experimentally infected mice, the sensitivity and specificity of both ELISA assays were 100% for the detection of anti-sparganum IgG. It is suggested that the rSepCp-1 protein-based ELISA could provide a highly sensitive and specific assay for the diagnosis of sparganosis.
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Affiliation(s)
- S M Mazidur Rahman
- Department of Parasitology and Tropical Medicine, Seoul National University College of Medicine, and Institute of Endemic Diseases, Seoul National University Medical Research Center, Seoul 110-799, Korea
| | - Jae-Hwan Kim
- Department of Parasitology and Tropical Medicine, Seoul National University College of Medicine, and Institute of Endemic Diseases, Seoul National University Medical Research Center, Seoul 110-799, Korea
| | - Sung-Tae Hong
- Department of Parasitology and Tropical Medicine, Seoul National University College of Medicine, and Institute of Endemic Diseases, Seoul National University Medical Research Center, Seoul 110-799, Korea
| | - Min-Ho Choi
- Department of Parasitology and Tropical Medicine, Seoul National University College of Medicine, and Institute of Endemic Diseases, Seoul National University Medical Research Center, Seoul 110-799, Korea
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Abstract
Sparganosis is a zoonotic cestodiasis of human beings and animals caused by plerocercoid or second-stage larvae (sparganum) of pseudophyllidean tapeworms in host tissues. Cats are among definitive hosts in which the larva develops to adult stage in the intestines. Reports on larval infection involving various tissues and organs in cats are scarce. Rare single case reports of visceral sparganosis in cats are previously documented. The present report documents an unusual subcutaneous sparganosis in 2 Domestic Shorthair cats from southern Georgia. Veterinary clinicians should consider sparganosis as differential diagnosis for subcutaneous cyst-like masses in cats. As infected animals and animal tissues are sources of human infection, sparganosis warrants public awareness and due precaution to avoid human infection.
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Affiliation(s)
- Moges Woldemeskel
- 1Moges Woldemeskel, Tifton Veterinary Diagnostic and Investigational Laboratory, 43 Brighton Road, Tifton, GA 31793.
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Boonyasiri A, Cheunsuchon P, Srirabheebhat P, Yamasaki H, Maleewong W, Intapan PM. Sparganosis presenting as cauda equina syndrome with molecular identification of the parasite in tissue sections. THE KOREAN JOURNAL OF PARASITOLOGY 2013; 51:739-42. [PMID: 24516282 PMCID: PMC3916466 DOI: 10.3347/kjp.2013.51.6.739] [Citation(s) in RCA: 10] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Subscribe] [Scholar Register] [Received: 05/30/2013] [Revised: 07/25/2013] [Accepted: 10/11/2013] [Indexed: 11/23/2022]
Abstract
A 52-year-old woman presented with lower back pain, progressive symmetrical paraparesis with sensory impairment, and sphincter disturbance. Magnetic resonance imaging (MRI) of the whole spine revealed multiple intradural extramedullary serpiginous-mass lesions in the subarachnoid space continuously from the prepontine to the anterior part of the medulla oblongata levels, C7, T2-T8, and T12 vertebral levels distally until the end of the theca sac and filling-in the right S1 neural foramen. Sparganosis was diagnosed by demonstration of the sparganum in histopathological sections of surgically resected tissues and also by the presence of serum IgG antibodies by ELISA. DNA was extracted from unstained tissue sections, and a partial fragment of mitochondrial cytochrome c oxidase subunit 1 (cox1) gene was amplified using a primer set specific for Spirometra spp. cox1. After sequencing of the PCR-amplicon and alignment of the nucleotide sequence data, the causative agent was identified as the larva of Spirometra erinaceieuropaei.
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Affiliation(s)
- Adhiratha Boonyasiri
- Division of Infectious Diseases and Tropical Medicine, Department of Medicine, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok 10700, Thailand
| | - Pornsuk Cheunsuchon
- Department of Pathology, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok 10700, Thailand
| | - Prajak Srirabheebhat
- Division of Neurosurgery, Department of Surgery, Faculty of Medicine Siriraj Hospital, Mahidol University, Bangkok 10700, Thailand
| | - Hiroshi Yamasaki
- Department of Parasitology, National Institute of Infectious Diseases, Tokyo, Japan
| | - Wanchai Maleewong
- Department of Parasitology, Faculty of Medicine, Khon Kaen University, Khon Kaen 40002, Thailand. ; Research and Diagnostic Center for Emerging Infectious Diseases, Faculty of Medicine, Khon Kaen University, Khon Kaen, 40002, Thailand
| | - Pewpan M Intapan
- Department of Parasitology, Faculty of Medicine, Khon Kaen University, Khon Kaen 40002, Thailand. ; Research and Diagnostic Center for Emerging Infectious Diseases, Faculty of Medicine, Khon Kaen University, Khon Kaen, 40002, Thailand
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Bauchet AL, Joubert C, Helies JM, Lacour S, Bosquet N, Le Grand R, Guillot J, Lachapelle F. Disseminated Sparganosis in a Cynomolgus Macaque (Macaca fascicularis). J Comp Pathol 2013; 148:294-7. [DOI: 10.1016/j.jcpa.2012.08.003] [Citation(s) in RCA: 6] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/21/2012] [Revised: 07/20/2012] [Accepted: 08/16/2012] [Indexed: 11/24/2022]
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Jang SY, Kim CH. Migration of sparganosis from the brain to the cervical spinal cord. J Korean Neurosurg Soc 2012; 51:170-2. [PMID: 22639717 PMCID: PMC3358607 DOI: 10.3340/jkns.2012.51.3.170] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 08/17/2011] [Revised: 09/26/2011] [Accepted: 03/12/2012] [Indexed: 11/27/2022] Open
Abstract
Central nervous system (CNS) sparganosis is a rare parasitic infestation caused by ingestion of the raw or inadequately cooked snakes or frogs. Sparganum is well known for its ability of migrating though the tissue, therefore, it can cause various neurological symptoms if it involves neurological systems. A 51-year-old male patient visited our department of neurosurgery complaining of the motor weakness and radiating pain on both upper extremities over 4 months. He had a history of ingesting raw snakes untill his late twenties. The magnetic resonance (MR) images of cervical spine revealed an intramedullary ill-defined enhancing lesion with the aggregated cysts in the upper cervical spinal cord. Under presumptive diagnosis of sparganosis, we took brain MR image. The brain MR images revealed the signal change in right fronto-temporal lobe suggesting the trajectory of parasitic migration via ventricular systems. He underwent a midline myelotomy and granuloma removal followed by the posterior laminoplasty. Pathologic findings showed inflammatory changes and necrosis with keratinized tissue suggesting the CNS sparganosis. We report an uncommon case of CNS sparganosis migrated from the brain to the spinal cord with literature review.
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Affiliation(s)
- Se Youn Jang
- Department of Neurosurgery, Hanyang University Guri Hospital, Hanyang University College of Medicine, Guri, Korea
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