Belde DEMİR, Demet ALAYGUT, Caner ALPARSLAN, Eren SOYALTIN, Fatma MUTLUBAŞ, Seçil ARSLANSOYU ÇAMLAR, Önder YAVAŞCAN

Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab

Background. Myalgia is one of the presenting symptoms of Familial Mediterranean Fever (FMF), which is reported at a rate of 27-39.6%. Recurrent fasciitis in those cases are extremely rare. We aimed to present a case with FMF having radiologically proven fasciitis.Case. An 11-year-old male patient with a diagnosis of FMF and M694V homozygote mutation, using colchicine regularly at a dose of 2 mg/day (0.08 mg/kg/day) for 4 years; was admitted to the hospital with severe pain and tenderness on the right calf. There were no accompanying symptoms like fever or abdominal pain. He described three similar episodes with pain and tenderness in left and right calves, which were not accompanied by fever in the last 6 months. The erythrocyte sedimentation rate and C-reactive protein levels were high and serum creatinine kinase was normal. The day after initiation of non-steroidal anti-inflammatory drug (NSAID), his complaints regressed. However, after a week, he again had a severe calf pain. Lower extremity arterial and venous doppler ultrasonography was normal. Increased peripheral signal intensity and fasciitis around the soleus muscle was defined in MRI. With NSAIDs, myalgia disappeared in a few days and acute phase reactants decreased within a week. In the follow up, canakinumab was prescribed due to febrile attacks as frequent as once a month and calf pain observed almost weekly. Thereafter, both febrile attacks and recurrent debilitating calf pain were completely ceased. Conclusion. There are three cases in the literature with fasciitis related myalgia. With this case, we wanted to emphasize fasciitis as a cause of FMF associated myalgia on MRI. In such cases MRI may be helpful to demonstrate fascial involvement.

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1. Majeed HA. Differential diagnosis of fever of unknown origin in children. Curr Opin Rheumatol 2000; 12: 439-444.

2. Zemer D. Muscle pains in familial Mediterranean fever. Harefuah 1984; 106: 232-233.

3. Soylu A, Oztürk Y, Kasap B, Akman N, Türkmen M, Kavukçu S. Clinical quiz: a pediatric case presenting with fever and diffuse myalgia. Rheumatol Int 2005; 25: 314-316.

4. Fujikawa K, Migita K, Tsukada T, Kawakami A, Eguchi K. Protracted febrile myalgia syndrome in a Japanese patient with fasciitis detected on MRI. Intern Med 2014; 53: 2817-2819.

5. Hull KM, Wong K, Wood GM, Chu WS, Kastner DL. Monocytic fasciitis: a newly recognized clinical feature of tumor necrosis factor receptor dysfunction. Arthritis Rheum 2002; 46: 2189-2194.

6. Quillinan N, Mohammad A, Mannion G, et al. Imaging evidence for persistent subclinical fasciitis and arthritis in tumour necrosis factor receptorassociated periodic syndrome (TRAPS) between febrile attacks. Ann Rheum Dis 2010; 69: 1408-1409.

7. Otar Yener G, Ekici Tekin Z, Yuksel S. Psoriatic fasciitis in a pediatric patient: a case report. World J Clin Cases 2019; 7: 69-72.

8. Kaplan E, Mukamel M, Barash J, et al. Protracted febrile myalgia in children and young adults with familial mediterranean fever: analysis of 15 patients and suggested criteria for working diagnosis. Clin Exp Rheumatol 2007; 25(4 Suppl 45): S114-S117.

9. Abdel Halim MM, Al-Otaibi T, Donia F, et al. Protracted febrile myalgia syndrome in a kidney transplant recipient with familial mediterranean fever. Exp Clin Transplant 2015; 13: 188-192.

10. Schapira D, Ludatscher R, Nahir M, Lorber M, Scharf Y. Severe myalgia in familial mediterranean fever: clinical and ultrastructural aspects. Ann Rheum Dis 1988; 47: 80-83.

11. Mercan R, Turan A, Bitik B, Tufan A, Haznedaroglu S, Goker B. Rapid resolution of protracted febrile myalgia syndrome with anakinra: report of two cases. Mod Rheumatol 2016; 26: 458-459.

12. Kotevoglu N, Sahin F, Ozkiris SO, Bankaoglu M, Sakiz D, Kuran B. Protracted febrile myalgia of familial mediterranean fever. Clin Exp Rheumatol 2004; 22(4 Suppl 34): S69-S70.

13. Umeda M, Aramaki T, Fujikawa K, et al. Tocilizumab is effective in a familial mediterranean fever patient complicated with histologically proven recurrent fasciitis and myositis. Int J Rheum Dis 2017; 20: 1868- 1871.

14. Gül A. Approach to the patients with inadequate response to colchicine in familial mediterranean fever. Best Pract Res Clin Rheumatol 2016; 30: 296- 303.