دورية أكاديمية
أعرض في EDS

Protracted febrile myalgia syndrome in children with familial Mediterranean fever - systematic review and a case report.

التفاصيل البيبلوغرافية
العنوان: Protracted febrile myalgia syndrome in children with familial Mediterranean fever - systematic review and a case report.
المؤلفون: Hospach T; Centre for Pediatric Rheumatology, Olgahospital Klinikum Stuttgart, Kriegsbergstr. 60, Stuttgart, 70174, Germany., Blankenburg F; Centre for Pediatric Rheumatology, Olgahospital Klinikum Stuttgart, Kriegsbergstr. 60, Stuttgart, 70174, Germany., Heinkele A; Centre for Pediatric Rheumatology, Olgahospital Klinikum Stuttgart, Kriegsbergstr. 60, Stuttgart, 70174, Germany., von Kalle T; Radiologic Institute, Olgahospital Klinikum Stuttgart, Stuttgart, Germany., Uziel Y; Faculty of Medicine, Tel Aviv University, Tel Aviv, Israel., Kallinich T; Charité, University Medicine, Berlin, Germany., Rücklová K; Centre for Pediatric Rheumatology, Olgahospital Klinikum Stuttgart, Kriegsbergstr. 60, Stuttgart, 70174, Germany. k.ruecklova@klinikum-stuttgart.de.; Charles University, Prague, Czech Republic. k.ruecklova@klinikum-stuttgart.de.
المصدر: Pediatric rheumatology online journal [Pediatr Rheumatol Online J] 2024 Sep 10; Vol. 22 (1), pp. 85. Date of Electronic Publication: 2024 Sep 10.
نوع المنشور: Case Reports; Journal Article; Systematic Review
اللغة: English
بيانات الدورية: Publisher: BioMed Central Country of Publication: England NLM ID: 101248897 Publication Model: Electronic Cited Medium: Internet ISSN: 1546-0096 (Electronic) Linking ISSN: 15460096 NLM ISO Abbreviation: Pediatr Rheumatol Online J Subsets: MEDLINE
أسماء مطبوعة: Publication: 2007- : [London] : BioMed Central
Original Publication: [Chicago, Ill. : University of Chicago, 2003]-
مواضيع طبية MeSH: Familial Mediterranean Fever*/complications , Familial Mediterranean Fever*/diagnosis , Fever*/diagnosis , Fever*/etiology , Myalgia*/diagnosis , Myalgia*/etiology, Child ; Humans ; Magnetic Resonance Imaging/methods ; Syndrome
مستخلص: Introduction: Protracted febrile myalgia syndrome (PFMS) is a rare manifestation of familial Mediterranean fever (FMF), characterized by myalgia, fever and elevated inflammatory markers lasting several weeks. As the hallmark of FMF are short episodes of disease symptoms, the long duration of PFMS may lead to a delayed diagnosis and treatment.
Objectives: 1. To perform a review of literature and rheumatology textbooks focused on clinical features and treatment of PFMS in children. 2. To present our own case.
Methods: All articles in Pub Med generated using the keywords "protracted febrile myalgia" and information on PFMS in seven rheumatology textbooks were collected. The systematic review was supplemented with our own case presentation.
Results: In total, 18 articles with 78 pediatric patients (including our own) were retrieved. More than half of the patients presented with PFMS as the first manifestation of FMF. All complained of myalgia, 65% of abdominal pain and 26% had a rash. Corticosteroids (CS) were effective in 77%. In all CS-refractory cases, anakinra was shown efficient. MRI was used in 5 patients and showed myositis in all of them. The scrutiny of seven rheumatology textbooks showed that PFMS presenting with myalgia was mentioned in six. Possible accompanying symptoms were described only once, the long duration of symptoms twice, the efficacy of corticosteroids three times and anakinra only once. The presented 6 year old patient manifested with fever, myalgia, abdominal pain and petechial rash lasting 6 weeks. She had undergone multiple diagnostic procedures before her parents mentioned a positive family history for FMF. The subsequent genetic testing confirmed a homozygosity for M694V pathogenic variant in the MEFV gene.
Conclusion: The long duration of PFMS may be misleading to clinicians especially if PFMS occurs at manifestation of FMF. The fact that more than half of the reported patients experienced PFMS as the presenting symptom of FMF is one of the key findings of our study. Our case presentation demonstrates the importance of genetic testing early in suspected autoinflammatory diseases. Furthermore, MRI may be an important diagnostic tool showing myositis in PFMS.
(© 2024. The Author(s).)
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فهرسة مساهمة: Keywords: Clinical features; Diagnostic role of MRI; Protracted febrile myalgia syndrome; Treatment
تواريخ الأحداث: Date Created: 20240910 Date Completed: 20240911 Latest Revision: 20240924
رمز التحديث: 20240925
مُعرف محوري في PubMed: PMC11389256
DOI: 10.1186/s12969-024-01019-6
PMID: 39256780
قاعدة البيانات: MEDLINE
الوصف
تدمد:1546-0096
DOI:10.1186/s12969-024-01019-6