TY - JOUR
T1 - Chronic Myopathy Associated with Anti-Signal Recognition Particle Antibodies Can Be Misdiagnosed As Facioscapulohumeral Muscular Dystrophy
AU - Ikeda, Kensuke
AU - Mori-Yoshimura, Madoka
AU - Yamamoto, Toshiyuki
AU - Sonoo, Masahiro
AU - Suzuki, Shigeaki
AU - Kondo, Yoshiyuki
AU - Nakamura, Harumasa
AU - Mitsuhashi, Kana
AU - Maeda, Meiko Hashimoto
AU - Shimizu, Jun
AU - Hayashi, Yukiko K.
AU - Nishino, Ichizo
AU - Oya, Yasushi
AU - Murata, Miho
N1 - Publisher Copyright:
Copyright © 2016 Wolters Kluwer Health, Inc. All rights reserved.
PY - 2016/6/1
Y1 - 2016/6/1
N2 - Objectives: To report cases of chronic autoimmune necrotizing myopathy with anti-signal recognition particle antibodies (anti-SRP myopathy) initially misdiagnosed as muscular dystrophy, in particular, facioscapulohumeral muscular dystrophy (FSHD). Methods: Medical records of patients with anti-SRP myopathy in our institution were retrospectively reviewed. Results: All 6 patients were initially diagnosed with muscular dystrophy because of the long-term clinical course and lack of inflammation on biopsy; 5 were diagnosed with FSHD based on a winged scapula. However, the following features suggested an alternative diagnosis, leading to anti-SRP antibody measurement: (1) lack of family history, (2) lack of facial involvement and asymmetry, (3) prominent dysphagia, and (4) profuse spontaneous activities on needle electromyography. All patients showed improvement with immunomodulating therapy. Conclusions: Anti-SRP antibody measurement should be considered in patients diagnosed with FSHD if they present with diagnostic hallmarks of anti-SRP myopathy listed above, to avoid oversight of this potentially treatable disorder.
AB - Objectives: To report cases of chronic autoimmune necrotizing myopathy with anti-signal recognition particle antibodies (anti-SRP myopathy) initially misdiagnosed as muscular dystrophy, in particular, facioscapulohumeral muscular dystrophy (FSHD). Methods: Medical records of patients with anti-SRP myopathy in our institution were retrospectively reviewed. Results: All 6 patients were initially diagnosed with muscular dystrophy because of the long-term clinical course and lack of inflammation on biopsy; 5 were diagnosed with FSHD based on a winged scapula. However, the following features suggested an alternative diagnosis, leading to anti-SRP antibody measurement: (1) lack of family history, (2) lack of facial involvement and asymmetry, (3) prominent dysphagia, and (4) profuse spontaneous activities on needle electromyography. All patients showed improvement with immunomodulating therapy. Conclusions: Anti-SRP antibody measurement should be considered in patients diagnosed with FSHD if they present with diagnostic hallmarks of anti-SRP myopathy listed above, to avoid oversight of this potentially treatable disorder.
KW - anti-signal recognition particle antibody
KW - dysphagia
KW - facioscapulohumeral muscular dystrophy
KW - winged scapulae
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U2 - 10.1097/CND.0000000000000115
DO - 10.1097/CND.0000000000000115
M3 - Article
C2 - 27224434
AN - SCOPUS:84973299449
SN - 1522-0443
VL - 17
SP - 197
EP - 206
JO - Journal of Clinical Neuromuscular Disease
JF - Journal of Clinical Neuromuscular Disease
IS - 4
ER -