Protracted febrile myalgia in two children with ... - Wiley Online Library

1 downloads 0 Views 394KB Size Report
Protracted febrile myalgia in two children with familial. Mediterranean fever. Nilgun Selcuk Duru,1 Mahmut Civilibal,2 Miray Karakoyun,1 Muge Payasli,1 Murat ...
Pediatrics International (2010) 52, e137–e140

Patient Report

ped_3058

doi: 10.1111/j.1442-200X.2010.03058.x

137..140

Protracted febrile myalgia in two children with familial Mediterranean fever Nilgun Selcuk Duru,1 Mahmut Civilibal,2 Miray Karakoyun,1 Muge Payasli,1 Murat Elevli1 Departments of 1Pediatrics and 2Pediatric Nephrology, Haseki Educational and Research Hospital, Istanbul, Turkey Key words

familial Mediterranean fever, protracted febrile myalgia, streptococcus.

Familial Mediterranean fever (FMF) is an autosomal recessive disease characterized by recurrent self-limited attacks of fever and inflammatory serositis. It occurs most often in the Mediterranean region, affecting mainly people of Jewish, Turkish, Arabic and Armenian ancestry.1 FMF is associated with several types of vasculitis, including polyarteritis nodosa (PAN), Henoch– Schönlein purpura (HSP) and Behçet’s disease. The clinical spectrum of FMF has recently been expanded and protracted febrile myalgia (PFM) is now a frequently recognized component in these patients.2–4 PFM is characterized by severe paralyzing myalgia, high fever, abdominal pain, arthritis/arthralgia, and transient vasculitic rashes mimicking HSP. PFM develops usually in patients with known FMF, even under colchicine prophylaxis. Sometimes, FMF can be diagnosed with PFM as a first manifestation.2–4 We describe here two children with FMF-associated protracted febrile myalgia. The first patient presented with PFM and was subsequently diagnosed as having FMF based on clinical features and genetic analysis. The second was a patient with known FMF.

Case Reports Case 1

An 8-year-old boy was admitted with abdominal pain, high fever, arthralgia and myalgia for two weeks. He was unable to walk without help due to the myalgia. His medical history revealed abdominal pain attacks with fever and/or recurrent febrile episodes every three to four months for the last two years. His parents were not relatives and there was no family history of autoimmune or rheumatic diseases. Physical examination showed normal anthropometric development, normal blood pressure (100/60 mmHg), fever (38.7°C), tachycardia, diffuse abdominal tenderness and severe muscular tenderness in all extremities. The rest of the physical examination was normal. Correspondence: Mahmut Civilibal, MD, Ataköy 7-8 Kısım, Mimar Sinan Sitesi, L5E Blok D: 49 34156 Ataköy, Istanbul, Turkey. Email: [email protected] Received 25 October 2008; revised 20 March 2009; accepted 8 April 2009.

© 2010 Japan Pediatric Society

Laboratory investigations revealed normal urine analysis, leukocytosis (17 600/mm3) with neutrophil predominance (76%), thrombocytosis (598 000/mm3), hyperglobulinemia (2.76 g/dL), normal serum glucose, urea, creatinine, electrolytes, transaminases and creatine phosphokinase (CPK). Erythrocyte sedimentation rate (ESR), C-reactive protein (CRP) and antistreptolysin-O (ASO) were elevated to 98 mm/h, 260 mg/L (normal,