Biomedical Genomics Unit, National Institute of Biomedical Genomics, Kolkata, India.
Am J Med Genet A. 2019 Apr;179(4):522-524. doi: 10.1002/ajmg.a.61058. Epub 2019 Jan 28.
Schuurs-Hoeijmakers syndrome (SHMS), or Autosomal Dominant Mental Retardation Syndrome type 17 (MRD17) is a rare form of intellectual disability with distinct facial features. A recurrent de novo heterozygous c.607C>T, p.Arg203Trp mutation in the PACS1 gene accounts for all reported cases except for one patient with a de novo heterozygous c.608G>A, p.Arg203Trp mutation. Ethnic background is known to affect the clinical manifestation of dysmorphic syndromes. Here we describe the first Indian patient with Schuurs-Hoeijmakers syndrome (SHMS) with a de novo heterozygous NM_018026.3 (PACS1):c.607C>T (p.Arg203Trp) variant. He is the only child with SHMS with a cleft lip. Thus our report expands the phenotypic spectrum of SHMS and establishes its occurrence across populations.
舒尔斯-霍伊杰马克斯综合征(SHMS),又称常染色体显性智力障碍综合征 17 型(MRD17),是一种罕见的智力障碍疾病,具有独特的面部特征。除了一位患者存在新发生的杂合子 c.608G>A,p.Arg203Trp 突变外,所有报道的病例均归因于 PACS1 基因中反复出现的新发生的杂合子 c.607C>T,p.Arg203Trp 突变。种族背景已知会影响发育异常综合征的临床表现。本文描述了首例印度舒尔斯-霍伊杰马克斯综合征(SHMS)患者,其存在新发生的杂合子 NM_018026.3(PACS1):c.607C>T(p.Arg203Trp)变异。他是唯一患有 SHMS 合并唇裂的患儿。因此,本报告扩展了 SHMS 的表型谱,并证实了其在不同人群中的发生。