Secondary cardiac involvement in anti-SRP-antibody-positive myopathy: an 87-year-old woman with heart failure symptoms as the first clinical presentation.


Journal

BMC neurology
ISSN: 1471-2377
Titre abrégé: BMC Neurol
Pays: England
ID NLM: 100968555

Informations de publication

Date de publication:
17 Jan 2020
Historique:
received: 09 10 2019
accepted: 02 01 2020
entrez: 19 1 2020
pubmed: 19 1 2020
medline: 9 4 2020
Statut: epublish

Résumé

Necrotizing myopathy (NM) is defined by the dominant pathological feature of necrosis of muscle fibers without substantial lymphocytic inflammatory infiltration. Anti-signal recognition particle (SRP)-antibody-positive myopathy is related to NM. Anti-SRP-antibody-positive myopathy can comorbid with other disorders in some patients, however, comorbidity with malignant tumor and myopericarditis has still not been reported. An 87-year-old woman with dyspnea on exertion and leg edema was referred to our hospital because of suspected heart failure and elevated serum creatine kinase level. Upon hospitalization, she developed muscle weakness predominantly in the proximal muscles. Muscle biopsy and immunological blood test led to the diagnosis of anti-SRP-antibody-positive myopathy. A colon carcinoma was also found and surgically removed. The muscle weakness remained despite the tumor resection and treatment with methylprednisolone. Cardiac screening revealed arrhythmia and diastolic dysfunction with pericardial effusion, which recovered with intravenous immunoglobulin (IVIg) treatment. We reported the first case of anti-SRP-positive myopathy comorbid with colon carcinoma and myopericarditis. This case is rare in the point that heart failure symptoms were the first clinical presentation. The underlying mechanism is still not clear, however, physicians should be carefully aware of the neoplasm and cardiac involvement in anti-SRP-antibody positive-myopathy patients and should consider farther evaluation and management.

Sections du résumé

BACKGROUND BACKGROUND
Necrotizing myopathy (NM) is defined by the dominant pathological feature of necrosis of muscle fibers without substantial lymphocytic inflammatory infiltration. Anti-signal recognition particle (SRP)-antibody-positive myopathy is related to NM. Anti-SRP-antibody-positive myopathy can comorbid with other disorders in some patients, however, comorbidity with malignant tumor and myopericarditis has still not been reported.
CASE PRESENTATION METHODS
An 87-year-old woman with dyspnea on exertion and leg edema was referred to our hospital because of suspected heart failure and elevated serum creatine kinase level. Upon hospitalization, she developed muscle weakness predominantly in the proximal muscles. Muscle biopsy and immunological blood test led to the diagnosis of anti-SRP-antibody-positive myopathy. A colon carcinoma was also found and surgically removed. The muscle weakness remained despite the tumor resection and treatment with methylprednisolone. Cardiac screening revealed arrhythmia and diastolic dysfunction with pericardial effusion, which recovered with intravenous immunoglobulin (IVIg) treatment.
CONCLUSIONS CONCLUSIONS
We reported the first case of anti-SRP-positive myopathy comorbid with colon carcinoma and myopericarditis. This case is rare in the point that heart failure symptoms were the first clinical presentation. The underlying mechanism is still not clear, however, physicians should be carefully aware of the neoplasm and cardiac involvement in anti-SRP-antibody positive-myopathy patients and should consider farther evaluation and management.

Identifiants

pubmed: 31952503
doi: 10.1186/s12883-020-1599-5
pii: 10.1186/s12883-020-1599-5
pmc: PMC6969444
doi:

Substances chimiques

Autoantibodies 0
Immunoglobulins, Intravenous 0
Signal Recognition Particle 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

29

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Auteurs

Arika Hara (A)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Ryota Amano (R)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan. d10sm003@yahoo.co.jp.

Hiroaki Yokote (H)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Masahide Ijima (M)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Satoshi Zeniya (S)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Toshiki Uchihara (T)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Sawako Yada (S)

Department of Internal Medicine, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Mayumi Masumura (M)

Department of Internal Medicine, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Hidenobu Takei (H)

Department of Internal Medicine, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

Ichizo Nishino (I)

Department of Neuromuscular Research, National Institute of Neuroscience, National Center of Neurology and Psychiatry, 4-1-1 Ogawahigashi-cho, Kodaira, Tokyo, 187-8502, Japan.

Shuta Toru (S)

Department of Neurology, Nitobe Memorial Nakano General Hospital, 4-59-16 Chuo Nakano, Tokyo, 164-8607, Japan.

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Classifications MeSH