3MC综合征:先前报道和较轻患者的分子发现扩大了自然史和表型谱。

IF 0.4 4区 医学 Q4 GENETICS & HEREDITY
Chloe Jade Ashton, Rahat Perveen, Glenda Beaman, Giangiorgio Crisponi, Ariadna González-Del Angel, Gilda Garza-Mayén, Miguel Angel Alcántara-Ortigoza, James O'Sullivan, Jill Clayton-Smith
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引用次数: 0

摘要

3MC综合征1-3型(MIM#257920、265050和248340)是一种罕见的常染色体隐性遗传病,由编码凝集素补体途径的基因的致病变异引起。3MC综合征患者具有独特的面部表型,包括远视、眉毛高度弓形和上睑下垂。相当数量的患者有双侧唇腭裂,他们经常表现出泌尿生殖系统和骨骼异常。3MC综合征的临床线索是存在特征性的尾侧附属物。MASP1, COLEC11和COLEC10基因的遗传变异已被确定为导致该综合征的原因,但迄今为止描述的患者相对较少。我们通过描述5例患者的临床和分子发现,巩固和扩展了目前对3MC综合征的表型特征和分子诊断的知识。这包括对两位兄弟的随访,他们的临床表型于1999年由Crisponi等人首次报道。我们的研究有助于3MC综合征的临床和分子谱的发展。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
3MC syndrome: molecular findings in previously reported and milder patients expand the natural history and phenotypic spectrum.

The 3MC syndromes types 1-3 (MIM#257920, 265050 and 248340, respectively) are rare autosomal recessive genetic disorders caused by pathogenic variants in genes encoding the lectin complement pathway. Patients with 3MC syndrome have a distinctive facial phenotype including hypertelorism, highly arched eyebrows and ptosis. A significant number of patients have bilateral cleft lip and palate and they often exhibit genitourinary and skeletal anomalies. A clinical clue to 3MC syndrome is the presence of a characteristic caudal appendage. Genetic variants in MASP1, COLEC11 and COLEC10 genes have been identified as the causation of this syndrome, yet relatively few patients have been described so far. We consolidate and expand current knowledge of phenotypic features and molecular diagnosis of 3MC syndrome by describing the clinical and molecular findings in five patients. This includes follow-up of two brothers whose clinical phenotypes were first reported by Crisponi et al in 1999. Our study contributes to the evolving clinical and molecular spectrum of 3MC syndrome.

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来源期刊
Clinical Dysmorphology
Clinical Dysmorphology 医学-遗传学
CiteScore
1.20
自引率
0.00%
发文量
64
审稿时长
6-12 weeks
期刊介绍: Clinical Dysmorphology publishes succinct case reports on the etiology, clinical delineation, genetic mapping, and molecular embryology of birth defects. This journal covers such topics as multiple congenital anomaly syndromes - with particular emphasis on previously undescribed conditions, rare findings, ethnic differences in existing syndromes, fetal abnormalities, and cytogenetic aberrations that might give clues to the localization of developmental genes. Regular features include original, peer-reviewed articles, conference reports, book and software reviews, abstracts and summaries from the UK Dysmorphology Club, and literature summaries. Submitted articles undergo a preliminary review by the editor. Some articles may be returned to authors wihtout further consideration. Those being considered for publication will undergo further assessment and peer-review by the editors and those invited to do so from a reviewer pool.
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