Literature DB >> 26791357

SOFT syndrome caused by compound heterozygous mutations of POC1A and its skeletal manifestation.

Jung Min Ko1,2, Soyoon Jung1, Jieun Seo3, Choong Ho Shin1, Hae Il Cheong1,2, Murim Choi3, Ok-Hwa Kim4, Tae-Joon Cho5.   

Abstract

SOFT syndrome (MIM614813) is an extremely rare primordial dwarfism characterized by short stature, onychodysplasia, facial dysmorphism and hypotrichosis, which is caused by biallelic mutations in the POC1A gene. Only 19 patients with mutation-confirmed SOFT syndrome have been reported to date, all of whom carried homozygous variants that were strongly associated with consanguineous marriages. We report an 8.5-year-old boy with SOFT syndrome showing primordial dwarfism, no effect of growth-hormone therapy and skeletal dysplasia. This is the first report of compound heterozygous variants in POC1A, one previously reported and the other novel. A characteristic skeletal manifestation is reported.

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Year:  2016        PMID: 26791357     DOI: 10.1038/jhg.2015.174

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  14 in total

Review 1.  Novel POC1A mutation in primordial dwarfism reveals new insights for centriole biogenesis.

Authors:  Asuman Koparir; Omer F Karatas; Betul Yuceturk; Bayram Yuksel; Ali O Bayrak; Omer F Gerdan; Mahmut S Sagiroglu; Alper Gezdirici; Koray Kirimtay; Ece Selcuk; Arzu Karabay; Chad J Creighton; Adnan Yuksel; Mustafa Ozen
Journal:  Hum Mol Genet       Date:  2015-07-10       Impact factor: 6.150

2.  A distinctive autosomal recessive syndrome of severe disproportionate short stature with short long bones, brachydactyly, and hypotrichosis in two consanguineous Arab families.

Authors:  Stavit A Shalev; Ronen Spiegel; Zvi U Borochowitz
Journal:  Eur J Med Genet       Date:  2012-03-03       Impact factor: 2.708

3.  POC1A truncation mutation causes a ciliopathy in humans characterized by primordial dwarfism.

Authors:  Ranad Shaheen; Eissa Faqeih; Hanan E Shamseldin; Ramil R Noche; Asma Sunker; Muneera J Alshammari; Tarfa Al-Sheddi; Nouran Adly; Mohammed S Al-Dosari; Sean G Megason; Muneera Al-Husain; Futwan Al-Mohanna; Fowzan S Alkuraya
Journal:  Am J Hum Genet       Date:  2012-07-26       Impact factor: 11.025

4.  Short stature, onychodysplasia, facial dysmorphism, and hypotrichosis syndrome is caused by a POC1A mutation.

Authors:  Ofer Sarig; Sagi Nahum; Debora Rapaport; Akemi Ishida-Yamamoto; Dana Fuchs-Telem; Li Qiaoli; Ksenya Cohen-Katsenelson; Ronen Spiegel; Janna Nousbeck; Shirli Israeli; Zvi-Uri Borochowitz; Gilly Padalon-Brauch; Jouni Uitto; Mia Horowitz; Stavit Shalev; Eli Sprecher
Journal:  Am J Hum Genet       Date:  2012-07-26       Impact factor: 11.025

5.  Application of whole exome sequencing to identify disease-causing variants in inherited human diseases.

Authors:  Gerald Goh; Murim Choi
Journal:  Genomics Inform       Date:  2012-12-31

6.  Growth hormone treatment in growth hormone-sufficient and -insufficient children with intrauterine growth retardation/Russell-Silver syndrome.

Authors:  C Azcona; A Albanese; P Bareille; R Stanhope
Journal:  Horm Res       Date:  1998

7.  Immunologic and clinical features of 25 Amish patients with RMRP 70 A-->G cartilage hair hypoplasia.

Authors:  Nicholas L Rider; D Holmes Morton; Erik Puffenberger; Christine L Hendrickson; Donna L Robinson; Kevin A Strauss
Journal:  Clin Immunol       Date:  2009-01-16       Impact factor: 3.969

Review 8.  The nonmotile ciliopathies.

Authors:  Jonathan L Tobin; Philip L Beales
Journal:  Genet Med       Date:  2009-06       Impact factor: 8.822

9.  Basal body stability and ciliogenesis requires the conserved component Poc1.

Authors:  Chad G Pearson; Daniel P S Osborn; Thomas H Giddings; Philip L Beales; Mark Winey
Journal:  J Cell Biol       Date:  2009-12-14       Impact factor: 10.539

10.  Truncation of POC1A associated with short stature and extreme insulin resistance.

Authors:  Jian-Hua Chen; Maria Segni; Felicity Payne; Isabel Huang-Doran; Alison Sleigh; Claire Adams; David B Savage; Stephen O'Rahilly; Robert K Semple; Inês Barroso
Journal:  J Mol Endocrinol       Date:  2015-10       Impact factor: 5.098

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  5 in total

1.  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

2.  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

Review 3.  Microcephalic Osteodysplastic Primordial Dwarfism, Type II: a Clinical Review.

Authors:  Michael B Bober; Andrew P Jackson
Journal:  Curr Osteoporos Rep       Date:  2017-04       Impact factor: 5.096

4.  SOFT Syndrome: The First Case in Iran.

Authors:  Neda Mostofizadeh; Mahshid Gheidarloo; Mahin Hashemipour; Elham Hashemi Dehkordi
Journal:  Adv Biomed Res       Date:  2018-09-21

5.  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

  5 in total

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