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Girolami I, Kluge RW, Gallmetzer L, Albi C, Sorgi P, Dejaco C, Tauber M, Hanspeter E, Matzneller P. Multicentric reticulohistiocytosis with oral and laryngeal involvement in association with autoimmune/inflammatory syndrome induced by adjuvants: Expanding the spectrum of two uncommon entities. Mod Rheumatol Case Rep 2025; 9:200-206. [PMID: 39096518 DOI: 10.1093/mrcr/rxae040] [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: 03/05/2024] [Revised: 05/11/2024] [Accepted: 07/29/2024] [Indexed: 08/05/2024]
Abstract
Multicentric reticulohistiocytosis (MRH) is the most frequent entity in the group of reticulohistiocytoses. It is usually accompanied by symmetrical erosive polyarthritis and is frequently associated with cancer and autoimmune disorders. Autoimmune/inflammatory syndrome induced by adjuvants (ASIA) is an inflammatory syndrome triggered by adjuvants such as those contained in vaccines or by silicone implants. Here, we report the case of a 71-year-old woman with a history of breast cancer treated with surgery and subsequent prosthesis who developed a systemic hyperinflammatory syndrome including seronegative symmetric polyarthritis, multiple skin lesions, and two large nodular lesions in the oral cavity and larynx. The clinical picture was consistent with a clinical diagnosis of ASIA, with breast implant rupture and/or vaccination against severe acute respiratory syndrome Coronavirus 2 (SARS-CoV-2) as possible triggers. Histopathology of skin, oral nodules, and laryngeal nodules revealed cutaneous/mucous and submucosal infiltration of large epithelioid mononuclear or binucleated cells with fine granular ground glass-like cytoplasm and round to kidney-shaped nuclei with prominent nucleoli, without atypical features or relevant pleomorphism, accompanied by sparse giant cells and lymphocytes. These cells stained positive for CD68 and CD45 and negative for S100, CD1a, and markers of epithelial or neural/melanocytic differentiation, altogether consistent with a diagnosis of reticulohistiocytosis. Clinicopathological correlation allowed the final diagnosis of MRH. To our knowledge, this is the first report of a co-occurrence of MRH with ASIA, and this is relevant to broaden the spectrum of both these rare diseases.
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Affiliation(s)
- Ilaria Girolami
- Department of Pathology, Provincial Hospital of Bolzano (SABES-ASDAA), Bolzano-Bozen, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
| | - Reinhard Walter Kluge
- Department of Pathology, Provincial Hospital of Bolzano (SABES-ASDAA), Bolzano-Bozen, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
| | - Lorenz Gallmetzer
- Department of Otololaryngology, Provincial Hospital of Bolzano (SABES-ASDAA), Bolzano-Bozen, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
| | - Cecilia Albi
- Department of Otololaryngology, Provincial Hospital of Bolzano (SABES-ASDAA), Bolzano-Bozen, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
- Department of Otolaryngology, University Hospital of Ferrara, Ferrara, Italy
| | - Paola Sorgi
- Department of Dermathology, Provincial Hospital of Merano (SABES-ASDAA), Merano-Meran, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
| | - Christian Dejaco
- Service of Rheumatology, Provincial Hospital of Brunico (SABES-ASDAA), Brunico-Bruneck, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
- Department of Rheumatology, Medical University of Graz, Graz, Steiermark, Austria
| | - Martina Tauber
- Department of Pathology, Provincial Hospital of Bolzano (SABES-ASDAA), Bolzano-Bozen, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
| | - Esther Hanspeter
- Department of Pathology, Provincial Hospital of Bolzano (SABES-ASDAA), Bolzano-Bozen, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
| | - Peter Matzneller
- Service of Rheumatology, Provincial Hospital of Merano (SABES-ASDAA), Merano-Meran, Italy; Lehrkrankenhaus der Paracelsus Medizinischen Privatuniversität
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2
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Jain H, Vasdev V, Sivasami K, Kumar A, Mishra PS, Jayaprakash S, Chandwani A. Multicentric reticulohistiocytosis. QJM 2024; 117:522-523. [PMID: 38430002 DOI: 10.1093/qjmed/hcae037] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 02/23/2024] [Indexed: 03/03/2024] Open
Affiliation(s)
- Harsh Jain
- Department of Clinical Immunology and Rheumatology, Army Hospital Research and Referral, New Delhi 110010, India
| | - V Vasdev
- Department of Clinical Immunology and Rheumatology, Army Hospital Research and Referral, New Delhi 110010, India
| | - Kartik Sivasami
- Department of Clinical Immunology and Rheumatology, Army Hospital Research and Referral, New Delhi 110010, India
| | - Abhishek Kumar
- Department of Clinical Immunology and Rheumatology, Army Hospital Research and Referral, New Delhi 110010, India
| | - P S Mishra
- Department of Pathology, Army Hospital Research and Referral, New Delhi 110010, India
| | - Sankar Jayaprakash
- Department of Clinical Immunology and Rheumatology, Army Hospital Research and Referral, New Delhi 110010, India
| | - A Chandwani
- Department of Clinical Immunology and Rheumatology, Army Hospital Research and Referral, New Delhi 110010, India
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3
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Li Y, Yin H, Zhang L, Lu L. Janus kinase inhibitor for multicentric reticulohistiocytosis, a new potential treatment. Int J Rheum Dis 2023; 26:1656-1659. [PMID: 37664958 DOI: 10.1111/1756-185x.14793] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/15/2023] [Revised: 06/04/2023] [Accepted: 06/08/2023] [Indexed: 09/05/2023]
Affiliation(s)
- Yuan Li
- Department of Rheumatology, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Hanlin Yin
- Department of Rheumatology, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Le Zhang
- Department of Rheumatology, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
- Department of Pharmacy, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
| | - Liangjing Lu
- Department of Rheumatology, Renji Hospital, Shanghai Jiao Tong University School of Medicine, Shanghai, China
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Mandyam S, Onteddu J, Ray R, Basso R, Bader F, Onteddu NK. Multicentric Reticulohistiocytosis Presenting As Acute Pericarditis With Moderate-Sized Pericardial Effusion: A Case Report and Review of Multicentric Reticulohistiocytosis Treatment. Cureus 2023; 15:e39953. [PMID: 37416020 PMCID: PMC10319645 DOI: 10.7759/cureus.39953] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Accepted: 06/04/2023] [Indexed: 07/08/2023] Open
Abstract
Multicentric reticulohistiocytosis (MRH) is a rare, class IIb non-Langerhans cell histiocytosis associated with skin and joint involvement. It is more prevalent (80%) in Caucasian females in their fifth to sixth decade of life. Patients usually demonstrate symptoms and signs of symmetric polyarthritis and papulonodular cutaneous lesions. In addition to skin and joints, multiple organs can be involved, such as the lung (pleural effusion, interstitial fibrosis, hilar lymphadenopathy), heart (pericardial effusion, myocarditis), gastrointestinal system, and urogenital system (genital tract and kidney). Pericardial involvement is a rare manifestation, and around three cases have been reported in the literature so far. Our case report is a valuable contribution to the literature, which aids clinicians in contemplating MRH as one of the differentials among patients presenting with pericardial effusion. We described the characteristics of MRH along with its differentiating features from other autoimmune conditions and management.
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Affiliation(s)
| | | | - Rubela Ray
- Internal Medicine, Bankura Sammilani Medical College, Bankura, IND
| | - Rafaela Basso
- Internal Medicine, Southeast Health Medical Center, Dothan, USA
| | - Fadi Bader
- Internal Medicine, Southeast Health Medical Center, Dothan, USA
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5
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Fan J, Jiang T, Qin Y, Ding Q, Cheng X, He D. Multicentric reticulohistiocytosis with eosinophilic gastroenteritis: An unusual relationship. Int J Rheum Dis 2023. [PMID: 36823772 DOI: 10.1111/1756-185x.14625] [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: 12/18/2022] [Revised: 01/31/2023] [Accepted: 02/06/2023] [Indexed: 02/25/2023]
Abstract
Multicentric reticulohistiocytosis (MRH) is a rare disease with poorly defined therapeutic strategies. Here, we report the case of a patient with eosinophilic gastroenteritis who developed MRH after 5 years. The patient presented with disabling and rapidly progressive polyarthralgia, eosinophilic gastroenteritis, and resistance to first-line therapies. However, there was a marked improvement in the arthritis symptoms following treatment with tofacitinib. This presentation provides a useful perspective for the therapeutic management of complex scenarios involving MRH.
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Affiliation(s)
- Junyu Fan
- Department of Rheumatology, Guanghua Hospital Affiliated to Shanghai University of Traditional Chinese Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai, China.,Department of Rheumatology, Shanghai Guanghua Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai, China
| | - Ting Jiang
- Department of Rheumatology, Guanghua Hospital Affiliated to Shanghai University of Traditional Chinese Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai, China.,Department of Rheumatology, Shanghai Guanghua Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai, China
| | - Yingying Qin
- Department of Rheumatology, Guanghua Hospital Affiliated to Shanghai University of Traditional Chinese Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai, China.,Department of Rheumatology, Shanghai Guanghua Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai, China
| | - Qin Ding
- Department of Rheumatology, Guanghua Hospital Affiliated to Shanghai University of Traditional Chinese Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai, China.,Department of Rheumatology, Shanghai Guanghua Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai, China
| | - Xia Cheng
- Department of Rheumatology, Guanghua Hospital Affiliated to Shanghai University of Traditional Chinese Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai, China.,Department of Rheumatology, Shanghai Guanghua Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai, China
| | - Dongyi He
- Department of Rheumatology, Guanghua Hospital Affiliated to Shanghai University of Traditional Chinese Medicine, Shanghai University of Traditional Chinese Medicine, Shanghai, China.,Department of Rheumatology, Shanghai Guanghua Hospital of Integrated Traditional Chinese and Western Medicine, Shanghai, China.,Institute of Arthritis Research in Integrative Medicine, Shanghai Academy of Traditional Chinese Medicine, Shanghai, China
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6
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Liu PP, Shuai ZW, Lian L, Wang K. Systemic lupus erythematosus with multicentric reticulohistiocytosis: A case report. World J Clin Cases 2023; 11:456-463. [PMID: 36686350 PMCID: PMC9850977 DOI: 10.12998/wjcc.v11.i2.456] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/17/2022] [Revised: 12/08/2022] [Accepted: 12/23/2022] [Indexed: 01/12/2023] Open
Abstract
BACKGROUND Multicentric reticulohistiocytosis (MRH)/systemic lupus erythematosus (SLE) overlap syndrome is an uncommon disease in the clinic and is diagnosed through characteristic clinical manifestations, histopathology, and immunopathology. Here, we report the case of a 30-year-old woman with SLE who developed MRH.
CASE SUMMARY A 30-year-old woman with a history of polyarthritis for the past 12 years had multiple skin nodules on her body for 10 years, including the sacrococcygeal area, dorsum of the hands, interphalangeal joint of the feet and sternoclavicular joint. The histopathology of a biopsy of the distal interphalangeal joint of the hands revealed granulomatous inflammation, fibrous hyperplasia with ground-glass degeneration, inflammatory cell exudation and focal necrosis. The immunohistochemical stains showed positive staining for CD68 and negative staining for S100 and acid-fast staining. The patient was diagnosed with SLE with MRH. Her symptoms were improved after a combined treatment of prednisone, hydroxychloroquine and cyclophosphamide.
CONCLUSION MRH/SLE overlap syndrome is difficult to diagnose and treat. Cyclophosphamide may be an alternative choice for the treatment of MRH.
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Affiliation(s)
- Ping-Ping Liu
- Department of Rheumatology and Immunology, Arthritis Research Institute, The First Affiliated Hospital of Anhui Medical University, Hefei 230000, Anhui Province, China
| | - Zong-Wen Shuai
- Department of Rheumatology and Immunology, Arthritis Research Institute, The First Affiliated Hospital of Anhui Medical University, Hefei 230000, Anhui Province, China
| | - Li Lian
- Department of Rheumatology and Immunology, Arthritis Research Institute, The First Affiliated Hospital of Anhui Medical University, Hefei 230000, Anhui Province, China
| | - Kang Wang
- Department of Rheumatology and Immunology, Arthritis Research Institute, The First Affiliated Hospital of Anhui Medical University, Hefei 230000, Anhui Province, China
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Mariotti EB, Corrà A, Lemmi E, Laschi L, Aimo C, Quintarelli L, Volpi W, Nacci F, Verdelli A, Ruffo di Calabria V, Guiducci S, Caproni M. Multicentric Reticulohistiocytosis Associated with an Early Form of Systemic Lupus Erythematosus: A Case Report of a Rare Disease, with Mini Review of the Literature. J Clin Med 2022; 11:6529. [PMID: 36362761 PMCID: PMC9658768 DOI: 10.3390/jcm11216529] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/19/2022] [Revised: 10/16/2022] [Accepted: 11/01/2022] [Indexed: 08/10/2023] Open
Abstract
Multicentric reticulohistiocytosis (MRH) is the most frequently described form of reticulohistiocytosis (RH), and it is classified as a class IIb non-Langerhans cell histiocytosis. It has been designated as multicentric, being characterized by multisystemic involvement. In fact, although mainly involving the skin, along with the joints, it is a systemic inflammatory condition potentially involving every internal organ. As MRH-related skin findings can mimic rheumatoid nodules or Gottron papules, the histopathology of the cutaneous lesions is often necessary for the correct diagnosis. Approximately one-third of MRH patients have confirmed concomitant autoimmune disorders. A wide variety of autoimmune disorders associated with the disease have been reported in the literature, suggesting immune dysfunction as a factor in the pathophysiology of MRH. A case of MRH associated with autoimmune manifestation is reported in the context of a mini review of the literature, with a focus on clinical presentation, treatments, and treatment outcomes. Moreover, eight cases of MRH associated with autoimmune diseases are briefly discussed.
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Affiliation(s)
| | - Alberto Corrà
- Section of Dermatology, Department of Health Sciences, University of Florence, 50125 Florence, Italy
| | - Elisa Lemmi
- Section of Anatomical Pathology, Department of Health Sciences, University of Florence, 50125 Florence, Italy
| | - Lucrezia Laschi
- Section of Anatomical Pathology, Department of Health Sciences, University of Florence, 50125 Florence, Italy
| | - Cristina Aimo
- Section of Dermatology, Department of Health Sciences, University of Florence, 50125 Florence, Italy
| | - Lavinia Quintarelli
- Rare Disease Skin Unit, Section of Dermatology, Azienda USL Toscana Centro, University of Florence, 50125 Florence, Italy
| | - Walter Volpi
- Section of Dermatology, Azienda USL Toscana Centro, 50125 Florence, Italy
| | - Francesca Nacci
- Section of Rheumatology, Department of Clinical and Experimental Medicine, University of Florence, 50125 Florence, Italy
| | - Alice Verdelli
- Section of Dermatology, Azienda USL Toscana Centro, 50125 Florence, Italy
| | | | - Serena Guiducci
- Section of Rheumatology, Department of Clinical and Experimental Medicine, University of Florence, 50125 Florence, Italy
| | - Marzia Caproni
- Rare Disease Skin Unit, Section of Dermatology, Azienda USL Toscana Centro, University of Florence, 50125 Florence, Italy
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8
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Xu XL, Liang XH, Liu J, Deng X, Zhang L, Wang ZG. Multicentric reticulohistiocytosis with prominent skin lesions and arthritis: A case report. World J Clin Cases 2022; 10:7913-7923. [PMID: 36158473 PMCID: PMC9372849 DOI: 10.12998/wjcc.v10.i22.7913] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 09/03/2021] [Revised: 12/14/2021] [Accepted: 06/17/2022] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Multicentric reticulohistiocytosis (MRH) is a rare non-Langerhans histiocytosis of unknown etiology characterized by papulonodular skin lesions and progressive, erosive arthritis. To date, there have been approximately 300 cases of MRH reported worldwide. The majority of patients are Caucasian from western countries, and Asian patients are rare. Here, we report a case of MRH in a Chinese patient.
CASE SUMMARY A 38-year-old male was admitted to the hospital with a rash that had persisted for over 2 years and bilateral knee pain for over 1 year. The patient’s symptoms had previously been misdiagnosed as eczema when there were only skin symptoms and was finally diagnosed as MRH after a skin biopsy of the left upper back. The patient was treated with glucocorticoids combined with an immunosuppressive regimen. While the skin lesions on both arms, abdomen, and back subsided, the skin lesions on the rest of the body did not increase. The interphalangeal joints of both thumbs and bilateral knee joints remained swollen and painful.
CONCLUSION The case will help clinicians better identify and treat this disease in the absence of epidemiological studies or randomized controlled data.
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Affiliation(s)
- Xiao-Li Xu
- Department of Geriatrics, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
| | - Xiao-Hong Liang
- Department of Geriatrics, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
| | - Juan Liu
- Jiangsu Engineering Research Center for Tumor Immunotherapy, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
| | - Xu Deng
- Department of Pathology, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
| | - Lu Zhang
- Department of Rheumatology and Immunology, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
| | - Zhi-Gang Wang
- Department of Geriatrics, The Third Affiliated Hospital of Soochow University, Changzhou 213003, Jiangsu Province, China
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翟 莉, 邱 楠, 宋 惠. [Multicentric reticulohistiocytosis: A case report]. BEIJING DA XUE XUE BAO. YI XUE BAN = JOURNAL OF PEKING UNIVERSITY. HEALTH SCIENCES 2021; 53:1183-1187. [PMID: 34916702 PMCID: PMC8695165 DOI: 10.19723/j.issn.1671-167x.2021.06.029] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Subscribe] [Scholar Register] [Received: 08/16/2021] [Indexed: 06/14/2023]
Abstract
A 65-year-old woman developed erythema, papules and nodules over the body. Some nodules of her auricles and hands like string beads. Besides, she suffered from symmetrical swelling and pain of multiple joints, morning stiffness with deformity of joints; She had elevated erythrocyte sedimentation rate and C reactive protein levels; Her rheumatoid factor and antinuclear antibody were positive; Joints destruction was found with X-ray imaging; Skin pathology showed Dermal infiltrate of abundant histiocytes, part of them with a ground-glass appearance; A CD68 immunohistochemical stain was positive and the cells were negative for S100, CD1a. These findings were diagnostic evidences of multicentric reticulohistiocytosis (MRH). The patient received high-dose of glucocorticoids combinated with immunosuppressive agents, and achieved a satisfactory effect. MRH was a rare multisystem disease characterized by papulonodular mucocutaneous and destructive arthritis, and its pathogeny was not yet completely understood. The typical lesions of MRH were hard papules or nodules that usually occured on the hands, face and arms. Classic coral bead appearance from periungual cutaneous nodules that were characteristic of MRH. MRH was an inflammatory joint disease, affecting almost all the appendicular joints and characterized by joint multiple, symmetrical, destructive, progressive disability. Joints destruction of the distal interphalangeal joints was a unique feature of MRH. In addition to skin and joints, it could also involve other systems. There were no diagnostic laboratory markers for MRH. Laboratory examinations had often been found to be non-specific. Imageological examination mainly showed bone and joint destruction. Skin biopsy was the best test to diagnose MRH, the typical histopathological findings included an infiltrate with histiocytes and multinucleated giant cells with a ground-glass appearing in eosinophilic cytoplasm, and the immunohistochemical stain was positive for CD68. The diagnosis was typically made based on the clinical presentation, supportive radiographic findings and skin biopsy. MRH was easily possible to mistake for other more common autoimmune conditions, such as rheumatoid arthritis, psoriatic arthritis, osteoarthritis, and dermatomyositis, but the distinctive clinical, radiographic, and histologic features could aid in differentiating these diseases. MRH could mimic other rheumatic diseases, besides, it could also coexist with cancer or other autoimmune disorders. There was no standardized treatment for MRH. However, Nonsteroidal anti-inflammatory drugs, glucocorticoid, Immunosuppressant, biologic medications, and bisphosphonates had been used with varying degrees of curative effect. Treatment with glucocorticoid combined with immunosuppressants were effective for rash and arthritis, early use of them should be strongly considered, and refractory cases could be treated with biological agents. By reporting a MRH case and reviewing literature, this paper aims to help the clinicians improve the understanding of this rare disease, and suggests that when one diagnosis cannot explain the whole picture of the disease, and further evidence should be sought to confirm the diagnosis.
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Affiliation(s)
- 莉 翟
- 中国人民解放军联勤保障部队第960医院，山东泰安 271000Department of Rheumatology and Immunology, The 960th Hospital of the PLA Joint Logistics Support Force, Tai'an 271000, Shandong, China
| | - 楠 邱
- 中国人民解放军联勤保障部队第960医院，山东泰安 271000Department of Rheumatology and Immunology, The 960th Hospital of the PLA Joint Logistics Support Force, Tai'an 271000, Shandong, China
| | - 惠 宋
- 泰安市中心医院中医科，山东泰安 271000Department of Traditional Chinese Medicine, Taian City Central Hospital, Tai'an 271000, Shandong, China
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10
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Niaki OZ, Penn E, Scott DA, Cobos G, Vleugels RA, Weinblatt ME. Treatment of Severe Multicentric Reticulohistiocytosis With Upadacitinib. JAMA Dermatol 2021; 157:735-737. [PMID: 33950153 DOI: 10.1001/jamadermatol.2021.0996] [Citation(s) in RCA: 4] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/14/2022]
Affiliation(s)
- Omid Zahedi Niaki
- Division of Rheumatology, Department of Medicine, University of Alberta Hospital, Edmonton, Alberta, Canada
| | - Erin Penn
- Department of Rheumatologic and Immunologic Disease, Cleveland Clinic, Cleveland, Ohio
| | - Deborah A Scott
- Department of Dermatology, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Gabriela Cobos
- Department of Dermatology, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Ruth Ann Vleugels
- Department of Dermatology, Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Michael E Weinblatt
- Division of Rheumatology, Department of Medicine, Inflammation and Immunity Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
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11
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Mastorino L, Avallone G, Dapavo P, Merli M, Agostini A, Grandinetti D, Fierro MT, Quaglino P, Ribero S. Tocilizumab and its usage for skin diseases. Ital J Dermatol Venerol 2020; 157:13-22. [PMID: 33314888 DOI: 10.23736/s2784-8671.20.06772-3] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.4] [Reference Citation Analysis] [Abstract] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/08/2022]
Abstract
The monoclonal anti-IL6 receptor antibody called Tocilizumab is widely used by rheumatologists for joint diseases. Its application in dermatology has mainly concerned scleroderma and Systemic Sclerosis in the last years. The most varied skin diseases treated with tocilizumab, such as psoriasis, psoriatic arthritis, Behcet's Syndrome, Lupus, and the already mentioned scleroderma up to multi-organ syndromes with skin involvement will be discussed. At the same time, there have been several side reactions to the drug involving the skin forcing careful skin monitoring during treatment. Despite the evidence currently available in the appropriate literature, there is no formal recommendation for any of these diseases to use Tocilizumab for therapeutic purposes. The aim of this review was to collect all the main evidence on the use and involvement of the drug in dermatological practice in order to stimulate further research or hypothesize on possible therapeutic options.
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Affiliation(s)
- Luca Mastorino
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Gianluca Avallone
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Paolo Dapavo
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Martina Merli
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Andrea Agostini
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Damiano Grandinetti
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Maria T Fierro
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Pietro Quaglino
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy
| | - Simone Ribero
- Section of Dermatology, Department of Medical Sciences, University of Turin, Turin, Italy -
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12
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Bruscas Izu C, Hörndler Argarate C, García Latasa de Araníbar FJ. Multicentric reticulohistiocytosis: A case report treated with tofacitinib. Med Clin (Barc) 2020; 156:310-311. [PMID: 32173071 DOI: 10.1016/j.medcli.2019.12.011] [Citation(s) in RCA: 4] [Impact Index Per Article: 0.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/05/2019] [Revised: 12/04/2019] [Accepted: 12/19/2019] [Indexed: 11/26/2022]
Affiliation(s)
- Carlos Bruscas Izu
- Servicio de Reumatología, Hospital General de la Defensa, Zaragoza, España.
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13
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Singer SB, Thomas C, Lezcano C, Robbins M, Gordian A, Nori S, Granter SR, Merola JF, O'Malley JT. Polyarthralgias and Papulonodules in a 56-Year-Old Woman. Arthritis Care Res (Hoboken) 2018; 70:925-930. [PMID: 29342504 DOI: 10.1002/acr.23516] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/12/2017] [Accepted: 01/09/2018] [Indexed: 11/07/2022]
Affiliation(s)
- Sean B Singer
- Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Cristina Thomas
- Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Cecilia Lezcano
- Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Mark Robbins
- Harvard Vanguard Medical Associates/Atrius Health, Boston, Massachusetts
| | - Amparo Gordian
- Harvard Vanguard Medical Associates/Atrius Health, Boston, Massachusetts
| | - Sarita Nori
- Harvard Vanguard Medical Associates/Atrius Health, Boston, Massachusetts
| | - Scott R Granter
- Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - Joseph F Merola
- Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
| | - John T O'Malley
- Brigham and Women's Hospital, Harvard Medical School, Boston, Massachusetts
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15
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Lim K, D'Souza J, Vasquez JB, Vuyyuru S. Looks Can Be Deceiving: A Case Report on Multicentric Reticulohistiocytosis Successfully Treated with Rituximab. Cureus 2017; 9:e1220. [PMID: 28589069 PMCID: PMC5453733 DOI: 10.7759/cureus.1220] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/05/2022] Open
Abstract
Multicentric reticulohistiocytosis (MRH) is an idiopathic multisystemic inflammatory disease characterized by symmetric erosive polyarthritis and typical papulonodular skin lesions. MRH can be associated with autoimmune diseases, malignancy, mycobacterial infections, and hyperlipidemia, and it is important to consider appropriate screening in this population. There is no specific diagnostic laboratory test for MRH. The gold standard for diagnosis is skin or synovial biopsy, which shows characteristic multinucleated non-Langerhans giant cells and ground glass eosinophilic cytoplasm. Although the disease spontaneously remits in approximately 10 years, MRH can rapidly progress to arthritis mutilans in the majority of cases. The diagnosis and treatment are challenging due to its low prevalence and lack of robust guidelines from major rheumatological societies. Corticosteroids and methotrexate are generally first-line treatment options. However, more recently, biologic agents have been increasingly used in refractory cases with some success. Early diagnosis is crucial in preventing disease progression. We report a case of MRH in a patient whose clinical presentation mimicked rheumatoid arthritis and was subsequently treated successfully with rituximab.
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Affiliation(s)
- Khin Lim
- Internal Medicine, Florida Hospital-Orlando
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Toz B, Büyükbabani N, İnanç M. Multicentric reticulohistiocytosis: Rheumatology perspective. Best Pract Res Clin Rheumatol 2016; 30:250-260. [DOI: 10.1016/j.berh.2016.07.002] [Citation(s) in RCA: 16] [Impact Index Per Article: 1.8] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/11/2016] [Accepted: 07/13/2016] [Indexed: 12/22/2022]
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Abstract
The skin is often the herald of an underlying systemic illness, and gastrointestinal malignancies can present in numerous ways in the skin. Paraneoplastic phenomenon, such as acanthosis nigricans and tripe palm, may be the first indicator of a gastrointestinal malignancy. In addition, gastrointestinal cancers can metastasize to the skin, as described in the well-known Sister Mary Joseph's nodule. Inflammatory systemic conditions such as dermatomyositis and multicentric reticulohistiocytosis can be associated with underlying malignancy. Finally, in numerous genetic syndromes with underlying malignancies, such as Muir-Torre, recognition of the skin signs leads to early diagnosis and screening.
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[Symmetrical polyarthritis in a patient with multicentric reticulohistiocytosis]. Med Clin (Barc) 2015; 145:508-9. [PMID: 25851911 DOI: 10.1016/j.medcli.2015.02.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/25/2014] [Revised: 02/18/2015] [Accepted: 02/26/2015] [Indexed: 11/21/2022]
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Assessment and effective targeting of Interleukin-1 in multicentric reticulohistyocytosis. Joint Bone Spine 2015; 82:280-3. [DOI: 10.1016/j.jbspin.2015.02.003] [Citation(s) in RCA: 17] [Impact Index Per Article: 1.7] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 11/18/2014] [Accepted: 02/02/2015] [Indexed: 11/23/2022]
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