Literature DB >> 25692795

Novel STAMBP mutation and additional findings in an Arabic family.

Eissa A Faqeih1, Laila Bastaki, Rasim Ozgur Rosti, Emily G Spencer, AbdulAli P Zada, Mohammad A M Saleh, Kyongmi Um, Joseph G Gleeson.   

Abstract

Microcephaly-capillary malformation syndrome (MIC-CAP syndrome) is a newly recognized autosomal recessive congenital neurocutaneous central nervous system disorder characterized by severe microcephaly, early-onset seizures, profound psychomotor disability, and multiple cutaneous capillary lesions. In addition, affected patients have variable dysmorphic facial features and hypoplastic distal phalanges. It is distinctively caused by mutations in a newly characterized gene, STAMBP, encoding the deubiquitinating (DUB) isopeptidase that has a key role in cell surface receptor-mediated endocytosis and sorting. Herein, we describe an Arab family of two siblings with classic features of MIC-CAP syndrome that harbor a novel predicted splice mutation in STAMBP, which additionally display previously unreported findings of congenital hypothyroidism and alopecia areata.
© 2015 Wiley Periodicals, Inc.

Entities:  

Keywords:  MIC-CAP syndrome; STAMBP gene; alopecia areata; capillary malformation; congenital hypothyroidism

Mesh:

Substances:

Year:  2015        PMID: 25692795     DOI: 10.1002/ajmg.a.36782

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  3 in total

1.  A novel homozygous missense mutation in the SH3-binding motif of STAMBP causing microcephaly-capillary malformation syndrome.

Authors:  Ikumi Hori; Fuyuki Miya; Yutaka Negishi; Ayako Hattori; Naoki Ando; Keith A Boroevich; Nobuhiko Okamoto; Mitsuhiro Kato; Tatsuhiko Tsunoda; Mami Yamasaki; Yonehiro Kanemura; Kenjiro Kosaki; Shinji Saitoh
Journal:  J Hum Genet       Date:  2018-06-15       Impact factor: 3.172

Review 2.  Early‑onset epilepsy and microcephaly‑capillary malformation syndrome caused by a novel STAMBP mutation in a Chinese boy.

Authors:  Fangrui Wu; Ying Dai; Juan Wang; Min Cheng; Yanqin Wang; Xiujuan Li; Ping Yuan; Shuang Liao; Li Jiang; Jin Chen; Lisi Yan; Min Zhong
Journal:  Mol Med Rep       Date:  2019-10-17       Impact factor: 2.952

3.  Novel compound heterozygous mutation in STAMBP causes a neurodevelopmental disorder by disrupting cortical proliferation.

Authors:  Meixin Hu; Huiping Li; Zhuxi Huang; Dongyun Li; Ying Xu; Qiong Xu; Bo Chen; Yi Wang; Jingxin Deng; Ming Zhu; Weijun Feng; Xiu Xu
Journal:  Front Neurosci       Date:  2022-08-10       Impact factor: 5.152

  3 in total

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