Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis

Background: Several results support the hypothesis that a group of pathologies falling within the Neuromyelitis Optica Spectrum Disorders (NMOSD) diagnostic criteria may coexist with Connective Tissue Diseases (CTD) in patients with a high susceptibility to autoimmune conditions. However, the relati...

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Main Authors: Jessica Elisabetta Esposito, Giovanni Annoni, Milena D’Amato, Alessandra Graziosi, Francesca Troilo, Annalisa Di Risio, Gilda Angelini, Caterina Castiglione, Pierluigi Tocco, Paola Volpe, Giancarlo Di Iorio, Giustino Parruti, Ennio Polilli
Format: Article
Language:English
Published: IMR Press 2024-02-01
Series:Journal of Integrative Neuroscience
Subjects:
Online Access:https://www.imrpress.com/journal/JIN/23/2/10.31083/j.jin2302035
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author Jessica Elisabetta Esposito
Giovanni Annoni
Milena D’Amato
Alessandra Graziosi
Francesca Troilo
Annalisa Di Risio
Gilda Angelini
Caterina Castiglione
Pierluigi Tocco
Paola Volpe
Giancarlo Di Iorio
Giustino Parruti
Ennio Polilli
author_facet Jessica Elisabetta Esposito
Giovanni Annoni
Milena D’Amato
Alessandra Graziosi
Francesca Troilo
Annalisa Di Risio
Gilda Angelini
Caterina Castiglione
Pierluigi Tocco
Paola Volpe
Giancarlo Di Iorio
Giustino Parruti
Ennio Polilli
author_sort Jessica Elisabetta Esposito
collection DOAJ
description Background: Several results support the hypothesis that a group of pathologies falling within the Neuromyelitis Optica Spectrum Disorders (NMOSD) diagnostic criteria may coexist with Connective Tissue Diseases (CTD) in patients with a high susceptibility to autoimmune conditions. However, the relationship between NMOSD and rheumatologic diseases deserves further investigations to clarify all clinical aspects of this coexistence. We designed a systematic review and a proportional meta-analysis to estimate the association between CTD and MNOSD, with the aim of helping to plan the best strategy to achieve the most significant public health benefit for these conditions. Methods: We conducted a systematic review of the literature published until February 2023, searching in four databases: PubMed, Web of Science, EmBase, and OVID. Then, we conducted a random-effects proportional meta-analysis and assessed the risk of bias of the included studies using the Joanna Briggs Institute checklist. Results: The literature search yielded an overall result of 3176 publications (272 from PubMed, 880 from Web of Science, 634 from EmBase and 1390 from OVID). Of these, 29 were included in this systematic review. Analyzing studies that recruited unselected patients with Systemic Lupus Erythematosus (SLE) and Sjogren Syndrome (SjS), the pooled percentages of NMOSD overlapping were 0.6% (95% Confidence Interval [95% CI]: 0.1%–1.4%,) and 6.5% (95% CI: 4.7–8.6), respectively. Studies enrolling rheumatologic patients with nervous system symptoms involvement reported higher percentage of NMOSD (i.e., among SjS patients, a pooled percentage of 26.5%, 95% CI: 5.5–54.6%, was found). Similarly, recruiting patients with NMOSD, we found pooled percentages of SjS or SLE respectively of 7.0% and 3.5%. Conclusions: Our research found that the coexistence of these two disorders was more frequent in female rheumatologic patients with a SjS diagnosis with neurological manifestations and in neurologic patients for whom a SjS diagnosis was suspected. Similarly, NMOSD are less frequently found in SLE and very rarely incident in Mixed Connective Tissue Disease (MCTD) patients. These considerations should be taken into account in clinical experience of rheumatologists and neurologists, since early diagnosis of both conditions may influence the timing of immunosuppressive therapy and the prevention of systemic disabilities.
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spelling doaj.art-e2d9dff0f9794b60b6c5baadae9839482024-02-29T08:00:15ZengIMR PressJournal of Integrative Neuroscience0219-63522024-02-012323510.31083/j.jin2302035S0219-6352(23)00670-8Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-AnalysisJessica Elisabetta Esposito0Giovanni Annoni1Milena D’Amato2Alessandra Graziosi3Francesca Troilo4Annalisa Di Risio5Gilda Angelini6Caterina Castiglione7Pierluigi Tocco8Paola Volpe9Giancarlo Di Iorio10Giustino Parruti11Ennio Polilli12Clinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyDepartment of Innovative Technology in Medicine and Odontoiatrics, University “G. d’Annunzio”, 66013 Chieti, ItalyClinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyDepartment of Innovative Technology in Medicine and Odontoiatrics, University “G. d’Annunzio”, 66013 Chieti, ItalyDepartment of Innovative Technology in Medicine and Odontoiatrics, University “G. d’Annunzio”, 66013 Chieti, ItalyClinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyClinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyClinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyNeurology and Stroke Unit, Pescara General Hospital, 65124 Pescara, ItalyReumathology Unit, Pescara General Hospital, 65124 Pescara, ItalyClinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyDepartment of Medicine, Pescara General Hospital, 65124 Pescara, ItalyClinical Pathology Unit, Pescara General Hospital, 65124 Pescara, ItalyBackground: Several results support the hypothesis that a group of pathologies falling within the Neuromyelitis Optica Spectrum Disorders (NMOSD) diagnostic criteria may coexist with Connective Tissue Diseases (CTD) in patients with a high susceptibility to autoimmune conditions. However, the relationship between NMOSD and rheumatologic diseases deserves further investigations to clarify all clinical aspects of this coexistence. We designed a systematic review and a proportional meta-analysis to estimate the association between CTD and MNOSD, with the aim of helping to plan the best strategy to achieve the most significant public health benefit for these conditions. Methods: We conducted a systematic review of the literature published until February 2023, searching in four databases: PubMed, Web of Science, EmBase, and OVID. Then, we conducted a random-effects proportional meta-analysis and assessed the risk of bias of the included studies using the Joanna Briggs Institute checklist. Results: The literature search yielded an overall result of 3176 publications (272 from PubMed, 880 from Web of Science, 634 from EmBase and 1390 from OVID). Of these, 29 were included in this systematic review. Analyzing studies that recruited unselected patients with Systemic Lupus Erythematosus (SLE) and Sjogren Syndrome (SjS), the pooled percentages of NMOSD overlapping were 0.6% (95% Confidence Interval [95% CI]: 0.1%–1.4%,) and 6.5% (95% CI: 4.7–8.6), respectively. Studies enrolling rheumatologic patients with nervous system symptoms involvement reported higher percentage of NMOSD (i.e., among SjS patients, a pooled percentage of 26.5%, 95% CI: 5.5–54.6%, was found). Similarly, recruiting patients with NMOSD, we found pooled percentages of SjS or SLE respectively of 7.0% and 3.5%. Conclusions: Our research found that the coexistence of these two disorders was more frequent in female rheumatologic patients with a SjS diagnosis with neurological manifestations and in neurologic patients for whom a SjS diagnosis was suspected. Similarly, NMOSD are less frequently found in SLE and very rarely incident in Mixed Connective Tissue Disease (MCTD) patients. These considerations should be taken into account in clinical experience of rheumatologists and neurologists, since early diagnosis of both conditions may influence the timing of immunosuppressive therapy and the prevention of systemic disabilities.https://www.imrpress.com/journal/JIN/23/2/10.31083/j.jin2302035connective tissue diseasecoexistence of autoimmune diseasesautoimmunityneuromyelitis optica spectrum disorders
spellingShingle Jessica Elisabetta Esposito
Giovanni Annoni
Milena D’Amato
Alessandra Graziosi
Francesca Troilo
Annalisa Di Risio
Gilda Angelini
Caterina Castiglione
Pierluigi Tocco
Paola Volpe
Giancarlo Di Iorio
Giustino Parruti
Ennio Polilli
Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis
Journal of Integrative Neuroscience
connective tissue disease
coexistence of autoimmune diseases
autoimmunity
neuromyelitis optica spectrum disorders
title Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis
title_full Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis
title_fullStr Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis
title_full_unstemmed Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis
title_short Systemic Connective Tissue Disease and Neuromyelitis Optica Spectrum Disorder Coexistence: A Systematic Review and Meta-Analysis
title_sort systemic connective tissue disease and neuromyelitis optica spectrum disorder coexistence a systematic review and meta analysis
topic connective tissue disease
coexistence of autoimmune diseases
autoimmunity
neuromyelitis optica spectrum disorders
url https://www.imrpress.com/journal/JIN/23/2/10.31083/j.jin2302035
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