Literature DB >> 22325252

3M syndrome: an easily recognizable yet underdiagnosed cause of proportionate short stature.

Mohammed S Al-Dosari1, Muneera Al-Shammari, Ranad Shaheen, Eissa Faqeih, Mohammed A Alghofely, Ahmad Boukai, Fowzan S Alkuraya.   

Abstract

OBJECTIVE: To characterize, via clinical and molecul criteria, a cohort of patients with 3M syndrome and thereby increase awareness of this syndrome as a recognizable cause of proportionate short stature. STUDY
DESIGN: We conducted a case series of patients referred to clinical genetics for proportionate short stature. CUL7, OBSL1, and CCDC8 genes were clinically phenotyped and sequenced.
RESULTS: In 6 Saudi families with 3M syndrome, we identified three CUL7, one OBSL1, and one CCDC8 novel mutations, which we show result in a remarkably similar clinical phenotype. Despite their typical and easily discernible clinical phenotype, all these patients have been extensively investigated for alternative causes of their short stature and received erroneous diagnoses.
CONCLUSION: Increased awareness about this syndrome among pediatricians and endocrinologists is needed to avoid a costly and unnecessary diagnostic odyssey.
Copyright © 2012 Mosby, Inc. All rights reserved.

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Year:  2012        PMID: 22325252     DOI: 10.1016/j.jpeds.2011.12.051

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  12 in total

Review 1.  Genetic evaluation of short stature.

Authors:  Andrew Dauber; Ron G Rosenfeld; Joel N Hirschhorn
Journal:  J Clin Endocrinol Metab       Date:  2014-06-10       Impact factor: 5.958

2.  Expanding the clinical and genetic heterogeneity of hereditary disorders of connective tissue.

Authors:  Anas M Alazami; Sarah M Al-Qattan; Eissa Faqeih; Amal Alhashem; Muneera Alshammari; Fatema Alzahrani; Mohammed S Al-Dosari; Nisha Patel; Afaf Alsagheir; Bassam Binabbas; Hamad Alzaidan; Abdulmonem Alsiddiky; Nasser Alharbi; Majid Alfadhel; Amal Kentab; Riza M Daza; Martin Kircher; Jay Shendure; Mais Hashem; Saif Alshahrani; Zuhair Rahbeeni; Ola Khalifa; Ranad Shaheen; Fowzan S Alkuraya
Journal:  Hum Genet       Date:  2016-03-29       Impact factor: 4.132

3.  Whole exome sequencing reveals a novel mutation in CUL7 in a patient with an undiagnosed growth disorder.

Authors:  Andrew Dauber; Joan Stoler; Eliana Hechter; Jason Safer; Joel N Hirschhorn
Journal:  J Pediatr       Date:  2012-09-10       Impact factor: 4.406

4.  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

5.  Two Siblings with a Mutation in CCDC8 Presenting with Mild Short Stature: A Case of 3-M Syndrome.

Authors:  Lihong Liao; Hoong-Wei Gan; Vivian Hwa; Mehul Dattani; Andrew Dauber
Journal:  Horm Res Paediatr       Date:  2017-07-04       Impact factor: 2.852

Review 6.  Cullin-RING E3 Ubiquitin Ligase 7 in Growth Control and Cancer.

Authors:  Zhen-Qiang Pan
Journal:  Adv Exp Med Biol       Date:  2020       Impact factor: 2.622

7.  3-M syndrome: a novel CUL7 mutation associated with respiratory distress and a good response to GH therapy.

Authors:  A Deeb; O Afandi; S Attia; A El Fatih
Journal:  Endocrinol Diabetes Metab Case Rep       Date:  2015-04-01

8.  Genomic analysis of primordial dwarfism reveals novel disease genes.

Authors:  Ranad Shaheen; Eissa Faqeih; Shinu Ansari; Ghada Abdel-Salam; Zuhair N Al-Hassnan; Tarfa Al-Shidi; Rana Alomar; Sameera Sogaty; Fowzan S Alkuraya
Journal:  Genome Res       Date:  2014-01-03       Impact factor: 9.043

9.  A novel CUL7 mutation in a Japanese patient with 3M syndrome.

Authors:  Tomozumi Takatani; Tadashi Shiohama; Rieko Takatani; Naoki Shimojo
Journal:  Hum Genome Var       Date:  2018-10-23

10.  Molecular basis of a new ovine model for human 3M syndrome-2.

Authors:  S A Woolley; S E Hayes; M R Shariflou; F W Nicholas; C E Willet; B A O'Rourke; I Tammen
Journal:  BMC Genet       Date:  2020-09-15       Impact factor: 2.797

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