丝状体肌病
张力减退
医学
肌张力过低
肌肉活检
儿科
肌病
无症状的
近端肌无力
体格检查
星云素
先天性肌病
基因检测
罕见病
活检
病理
疾病
内科学
提丁
肌节
心肌细胞
作者
Neha Dalal,Kirti Naranje,Amita Moriangthem,Anita Singh
出处
期刊:Case Reports
[BMJ]
日期:2024-06-01
卷期号:17 (6): e259303-e259303
标识
DOI:10.1136/bcr-2023-259303
摘要
Nemaline rod myopathy is an extremely rare muscle disease responsible for hypotonia and poor muscle strength in infants. The disease has variable phenotypic presentations across different ages, ranging from neonatal to the adult onset and from severe to asymptomatic varieties. Clinical features, muscle biopsy and genetic testing help in diagnosis. The histopathological examination shows the presence of rod-like structures or nemaline bodies in muscles. Management remains mainly supportive, and currently, there is no available curative treatment. This case report describes an infant presenting with gross hypotonia, poor handling of secretions and multiple extubation failures who was diagnosed by clinical exome sequencing. The patient harboured compound heterozygous variants in the NEB gene suggestive of nemaline rod myopathy. The newborn showed significant improvement in muscle strength after he was started on dietary L-tyrosine supplementation. This case highlights the emerging role of L-tyrosine in the supportive care of infants with nemaline rod myopathy.
科研通智能强力驱动
Strongly Powered by AbleSci AI