TY - JOUR
T1 - A rare case of sporadic inclusion body myositis and rheumatoid arthritis exhibiting ectopic lymphoid follicle-like structures
T2 - a case report and literature review
AU - Konomatsu, Kazutoshi
AU - Izumi, Rumiko
AU - Suzuki, Naoki
AU - Takai, Yoshiki
AU - Shirota, Yuko
AU - Saito, Ryoko
AU - Kuroda, Hiroshi
AU - Aoki, Masashi
N1 - Funding Information:
This work was supported by AMED under grant number 20dk0310086 , an Intramural Research Grant ( 20FC1036 ) for Neurological and Psychiatric Disorders of NCNP, Grant-in-Aid for challenging Exploratory Research ( 20K21563 ), JSPS KAKENHI grant number JP20K16571 and Research on Measures for Intractable Diseases from the Japanese Ministry of Health Labor and Welfare ( 20FC1036 ).
Publisher Copyright:
© 2021 Elsevier B.V.
PY - 2021/9
Y1 - 2021/9
N2 - Sporadic inclusion body myositis (sIBM) is a degenerative, intractable, inflammatory myopathy with an immune pathomechanism. We report on a case of a 44-year-old Japanese man who began developing progressive muscle weakness at age 40. Rheumatoid arthritis symptoms manifested at 43 with strongly positive anti-cyclic citrullinated peptide antibodies. Along with typical sIBM pathology, a muscle biopsy revealed dramatic inflammation with prominent perivascular B-cell infiltration forming ectopic lymphoid follicle-like structures (ELFLSs). Exome sequencing identified no causative variants of hereditary myopathy or immune disorders. A combination of immunotherapy slowed the progression of the muscular symptoms. This unusual form of sIBM, including earlier age at onset, a partial response to immunotherapy, and a histopathology presenting B-cell infiltrate with ectopic lymphoid follicle-like structures, indicates a possible association of rheumatoid arthritis and heterogeneity with the autoimmune involvement of sIBM. We review the clinical and pathological features of patients with rheumatoid arthritis associated sIBM in the literature.
AB - Sporadic inclusion body myositis (sIBM) is a degenerative, intractable, inflammatory myopathy with an immune pathomechanism. We report on a case of a 44-year-old Japanese man who began developing progressive muscle weakness at age 40. Rheumatoid arthritis symptoms manifested at 43 with strongly positive anti-cyclic citrullinated peptide antibodies. Along with typical sIBM pathology, a muscle biopsy revealed dramatic inflammation with prominent perivascular B-cell infiltration forming ectopic lymphoid follicle-like structures (ELFLSs). Exome sequencing identified no causative variants of hereditary myopathy or immune disorders. A combination of immunotherapy slowed the progression of the muscular symptoms. This unusual form of sIBM, including earlier age at onset, a partial response to immunotherapy, and a histopathology presenting B-cell infiltrate with ectopic lymphoid follicle-like structures, indicates a possible association of rheumatoid arthritis and heterogeneity with the autoimmune involvement of sIBM. We review the clinical and pathological features of patients with rheumatoid arthritis associated sIBM in the literature.
KW - Ectopic lymphoid follicle-like structure
KW - Perivascular B-cell infiltration
KW - Rheumatoid arthritis
KW - Sporadic inclusion body myositis
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U2 - 10.1016/j.nmd.2021.07.002
DO - 10.1016/j.nmd.2021.07.002
M3 - Article
C2 - 34465502
AN - SCOPUS:85113831015
SN - 0960-8966
VL - 31
SP - 870
EP - 876
JO - Neuromuscular Disorders
JF - Neuromuscular Disorders
IS - 9
ER -