Servicio de Pediatría, Hospital de Puerto Montt, 5507798, Puerto Montt, Chile.
Department of Human Genetics, Yokohama City University Graduate School of Medicine, Yokohama, 236-0004, Japan.
J Hum Genet. 2018 May;63(5):677-681. doi: 10.1038/s10038-018-0426-y. Epub 2018 Feb 26.
Prolyl endopeptidase-like (PREPL) deficiency (MIM# 616224) is a rare autosomal recessive inherited congenital myasthenic syndrome characterized by neonatal hypotonia, feeding problems, mild dysmorphism, and neuromuscular symptoms, followed by hyperphagia and obesity in later childhood. Some patients also exhibit growth deficits, sexual hormone deficiency, and cognitive impairments. This syndrome is caused by biallelic mutations in PREPL. To date, only one nucleotide deletion and seven small microdeletions in PREPL have been reported. Here we report a female patient with a novel homozygous frameshift mutation in PREPL (NM_006036.4, c.342delA:p.Val115Leufs*39). Her clinical features are similar to those of previously reported cases. The mutation is the first homozygous point mutation reported in humans.
脯氨酰内肽酶样(PREPL)缺乏症(MIM# 616224)是一种罕见的常染色体隐性遗传性先天性肌无力综合征,其特征为新生儿低张力、喂养问题、轻度畸形和神经肌肉症状,随后在儿童后期出现多食和肥胖。一些患者还表现出生长缺陷、性激素缺乏和认知障碍。该综合征由 PREPL 的双等位基因突变引起。迄今为止,仅报道了 PREPL 中的一个核苷酸缺失和七个小微缺失。在这里,我们报告了一例 PREPL 中新型纯合移码突变的女性患者(NM_006036.4,c.342delA:p.Val115Leufs*39)。她的临床特征与先前报道的病例相似。该突变是人类中首次报道的纯合点突变。