Literature DB >> 30569574

SOFT syndrome in a patient from Chile.

Ken Saida1, Sebastian Silva2, Benjamin Solar2, Atsushi Fujita1, Kohei Hamanaka1, Satomi Mitsuhashi1, Eriko Koshimizu1, Takeshi Mizuguchi1, Satoko Miyatake1, Atsushi Takata1, Noriko Miyake1, Naomichi Matsumoto1.   

Abstract

SOFT syndrome (MIM614813) is an extremely rare primordial dwarfism caused by biallelic mutations in the POC1A gene. It is characterized by prenatal short stature, onychodysplasia, facial dysmorphism, hypotrichosis, and variable skeletal abnormalities including hypoplastic pelvis and sacrum, small hands, and cone-shaped epiphyses, as well as delayed bone age. To the best of our knowledge, only eight POC1A mutations have been reported in humans to date. We report a 7-year-old Chilean girl with SOFT syndrome arising from a novel POC1A mutation c. 649C>T, p.Arg217Trp. Although her clinical features were largely compatible with SOFT syndrome, hand X-ray examinations at 3.5 and 6 years unexpectedly showed normal bone age. Automated bone age determination was performed using image analysis software, BoneXpert. This case highlights the importance of the accumulation of patients with POC1A mutations to further elucidate the detailed clinical features of SOFT syndrome.
© 2018 Wiley Periodicals, Inc.

Entities:  

Keywords:  zzm321990POC1A; SOFT syndrome; bone age; patent ductus arteriosus

Mesh:

Substances:

Year:  2018        PMID: 30569574     DOI: 10.1002/ajmg.a.61015

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


  4 in total

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Authors:  Tomer Avidor-Reiss; Alexa Carr; Emily Lillian Fishman
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2.  Identification of SOFT syndrome caused by a pathogenic homozygous splicing variant of POC1A: a case report.

Authors:  Guoqiang Li; Guoying Chang; Chen Wang; Tingting Yu; Niu Li; Xiaodong Huang; Xiumin Wang; Jian Wang; Jiwen Wang; Ruen Yao
Journal:  BMC Med Genomics       Date:  2021-08-21       Impact factor: 3.063

3.  Further phenotypic features and two novel POC1A variants in a patient with SOFT syndrome: A case report.

Authors:  Songting Li; Yan Zhong; Yongjia Yang; Siping He; Wenjuan He
Journal:  Mol Med Rep       Date:  2021-05-06       Impact factor: 2.952

4.  Biallelic POC1A variants cause syndromic severe insulin resistance with muscle cramps.

Authors:  Veronica Mericq; Isabel Huang-Doran; Dhekra Al-Naqeb; Javiera Basaure; Claudia Castiglioni; Christiaan de Bruin; Yvonne Hendriks; Enrico Bertini; Fowzan S Alkuraya; Monique Losekoot; Khalid Al-Rubeaan; Robert K Semple; Jan M Wit
Journal:  Eur J Endocrinol       Date:  2022-03-23       Impact factor: 6.558

  4 in total

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