Mohammad Hamad, Tala Mohammad, Mira Bishtawi, Randa Shahwan, Jaber H Jaradat, Wafa' A Hazaymeh, Omar F Jbarah, Ethar Hazaimeh
{"title":"Prevalence of neurological manifestation in familial mediterranean fever patients: systematic review & meta-analysis.","authors":"Mohammad Hamad, Tala Mohammad, Mira Bishtawi, Randa Shahwan, Jaber H Jaradat, Wafa' A Hazaymeh, Omar F Jbarah, Ethar Hazaimeh","doi":"10.1007/s10072-025-08351-z","DOIUrl":null,"url":null,"abstract":"<p><strong>Background: </strong>Familial Mediterranean Fever (FMF) is the most common autoinflammatory disease worldwide, characterized by recurrent attacks of fever and polyserositis. Several studies have reported FMF cases with neurological conditions. As well as multiple neurological diseases have been reported in multiple studies in patients with FMF.</p><p><strong>Aims: </strong>to summarize the frequency of neurologic manifestations reported among FMF patients.</p><p><strong>Methods: </strong>PubMed, Web of Science, and SCOPUS were searched for studies that reported any neurological manifestation in FMF patients. Studies were screened first by abstract and then by full text, any conflict was resolved by a discussion. The JBI tool was used for assessing the risk of bias in studies. A meta-analysis of the proportions was performed for neurological manifestations in patients with FMF. A random effects model was indicated when I<sup>2</sup> was greater than 50%, and a fixed model was used otherwise. R version 4.3.2 for performing the analysis.</p><p><strong>Results: </strong>Out of 787 citations, 36 studies (14 case reports, 22 others) were included with a total of 906 FMF patients (14 case reports, 892 others) with neurological manifestations. For case-report studies, the common manifestation was headache (42.9%) followed by Muscle weakness, diplopia (35.7%), Sensory disorders (35.7%) and facial nerve palsy (14.3%), The most common comorbid neurological condition was Multiple sclerosis (28.6%). Regarding Other study types, the Pooled prevalence of the most common manifestation was Neuropathy (0.45 [0.00; 0.99]), then Myalgia (0.27, 95%CI: [0.11; 0.47]), Headache (0.23, 95%CI: [0.12; 0.36]), and visual disorders (0.13 [0.01; 0.37]). The most common comorbid was ADHD (0.31, 95%CI: [0.25; 0.37]). Most studies were evaluated at a low risk of bias.</p><p><strong>Conclusion: </strong>Nearly half of the patients in our review had Neuropathy in this review. ADHD was co-comorbid in 31% of patients. These findings underscore the importance of increased awareness and surveillance for these manifestations in patients with FMF. Further research is needed to better understand the pathogenesis of this manifestation.</p>","PeriodicalId":19191,"journal":{"name":"Neurological Sciences","volume":" ","pages":"4943-4953"},"PeriodicalIF":2.4000,"publicationDate":"2025-10-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":"0","resultStr":null,"platform":"Semanticscholar","paperid":null,"PeriodicalName":"Neurological Sciences","FirstCategoryId":"3","ListUrlMain":"https://doi.org/10.1007/s10072-025-08351-z","RegionNum":4,"RegionCategory":"医学","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":null,"EPubDate":"2025/7/11 0:00:00","PubModel":"Epub","JCR":"Q2","JCRName":"CLINICAL NEUROLOGY","Score":null,"Total":0}
引用次数: 0
Abstract
Background: Familial Mediterranean Fever (FMF) is the most common autoinflammatory disease worldwide, characterized by recurrent attacks of fever and polyserositis. Several studies have reported FMF cases with neurological conditions. As well as multiple neurological diseases have been reported in multiple studies in patients with FMF.
Aims: to summarize the frequency of neurologic manifestations reported among FMF patients.
Methods: PubMed, Web of Science, and SCOPUS were searched for studies that reported any neurological manifestation in FMF patients. Studies were screened first by abstract and then by full text, any conflict was resolved by a discussion. The JBI tool was used for assessing the risk of bias in studies. A meta-analysis of the proportions was performed for neurological manifestations in patients with FMF. A random effects model was indicated when I2 was greater than 50%, and a fixed model was used otherwise. R version 4.3.2 for performing the analysis.
Results: Out of 787 citations, 36 studies (14 case reports, 22 others) were included with a total of 906 FMF patients (14 case reports, 892 others) with neurological manifestations. For case-report studies, the common manifestation was headache (42.9%) followed by Muscle weakness, diplopia (35.7%), Sensory disorders (35.7%) and facial nerve palsy (14.3%), The most common comorbid neurological condition was Multiple sclerosis (28.6%). Regarding Other study types, the Pooled prevalence of the most common manifestation was Neuropathy (0.45 [0.00; 0.99]), then Myalgia (0.27, 95%CI: [0.11; 0.47]), Headache (0.23, 95%CI: [0.12; 0.36]), and visual disorders (0.13 [0.01; 0.37]). The most common comorbid was ADHD (0.31, 95%CI: [0.25; 0.37]). Most studies were evaluated at a low risk of bias.
Conclusion: Nearly half of the patients in our review had Neuropathy in this review. ADHD was co-comorbid in 31% of patients. These findings underscore the importance of increased awareness and surveillance for these manifestations in patients with FMF. Further research is needed to better understand the pathogenesis of this manifestation.
期刊介绍:
Neurological Sciences is intended to provide a medium for the communication of results and ideas in the field of neuroscience. The journal welcomes contributions in both the basic and clinical aspects of the neurosciences. The official language of the journal is English. Reports are published in the form of original articles, short communications, editorials, reviews and letters to the editor. Original articles present the results of experimental or clinical studies in the neurosciences, while short communications are succinct reports permitting the rapid publication of novel results. Original contributions may be submitted for the special sections History of Neurology, Health Care and Neurological Digressions - a forum for cultural topics related to the neurosciences. The journal also publishes correspondence book reviews, meeting reports and announcements.