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...

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Main Authors: Belde Kasap Demir, Demet Alaygut, Caner Alparslan, Eren Soyaltın, Fatma Mutlubaş, Seçil Arslansoyu Çamlar, Önder Yavaşcan
Format: Article
Language:English
Published: Hacettepe University Institute of Child Health 2020-06-01
Series:The Turkish Journal of Pediatrics
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Online Access:https://turkjpediatr.org/article/view/470
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author Belde Kasap Demir
Demet Alaygut
Caner Alparslan
Eren Soyaltın
Fatma Mutlubaş
Seçil Arslansoyu Çamlar
Önder Yavaşcan
author_facet Belde Kasap Demir
Demet Alaygut
Caner Alparslan
Eren Soyaltın
Fatma Mutlubaş
Seçil Arslansoyu Çamlar
Önder Yavaşcan
author_sort Belde Kasap Demir
collection DOAJ
description 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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institution OA Journals
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language English
publishDate 2020-06-01
publisher Hacettepe University Institute of Child Health
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spelling doaj-art-540d349088944bafa023eb49bdcd75232025-08-20T02:01:47ZengHacettepe University Institute of Child HealthThe Turkish Journal of Pediatrics0041-43012791-64212020-06-0162310.24953/turkjped.2020.03.017Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumabBelde Kasap DemirDemet Alaygut0Caner Alparslan1Eren Soyaltın2Fatma Mutlubaş3Seçil Arslansoyu Çamlar4Önder Yavaşcan5Division of Nephrology, Department of Pediatrics, Health Sciences University, İzmir Tepecik Training and Research Hospital, İzmir, Turkey.Division of Nephrology, Department of Pediatrics, Health Sciences University, İzmir Tepecik Training and Research Hospital, İzmir, Turkey.Division of Nephrology, Department of Pediatrics, Health Sciences University, İzmir Tepecik Training and Research Hospital, İzmir, Turkey.Division of Nephrology, Department of Pediatrics, Health Sciences University, İzmir Tepecik Training and Research Hospital, İzmir, Turkey.Division of Nephrology, Department of Pediatrics, Health Sciences University, İzmir Tepecik Training and Research Hospital, İzmir, Turkey.Division of Nephrology, Department of Pediatrics, Health Sciences University, İzmir Tepecik Training and Research Hospital, İzmir, Turkey. 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. https://turkjpediatr.org/article/view/470Familial Mediaterranean Feverfasciitis
spellingShingle Belde Kasap Demir
Demet Alaygut
Caner Alparslan
Eren Soyaltın
Fatma Mutlubaş
Seçil Arslansoyu Çamlar
Önder Yavaşcan
Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab
The Turkish Journal of Pediatrics
Familial Mediaterranean Fever
fasciitis
title Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab
title_full Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab
title_fullStr Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab
title_full_unstemmed Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab
title_short Recurrent debilitating calf pain associated with fasciitis in Familial Mediterranean fever and response to canacinumab
title_sort recurrent debilitating calf pain associated with fasciitis in familial mediterranean fever and response to canacinumab
topic Familial Mediaterranean Fever
fasciitis
url https://turkjpediatr.org/article/view/470
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