Literature DB >> 23123320

Homozygous stop mutation in the SNX10 gene in a consanguineous Iraqi boy with osteopetrosis and corpus callosum hypoplasia.

André Mégarbané1, Alessandra Pangrazio, Anna Villa, Eliane Chouery, Joseph Maarawi, Sandra Sabbagh, Gérard Lefranc, Cristina Sobacchi.   

Abstract

Recently a mutation in the SNX10 gene that belongs to the sorting nexin family was identified as a cause of a new subset of human autosomal recessive osteopetrosis. Here, we identified a novel homozygous mutation (c.46C > T, p.Arg16X) in SNX10, in an Iraqi boy from a consanguineous family with a history of infantile osteopetrosis. The proband exhibited macrocephaly, prominent forehead, proptosis of the eyes, strabismus, splenomegaly and joint hyperlaxity. Bone X-rays showed increased bone density, metaphyseal under-modelling, transverse alternating bands of greater and lesser density in tubular bones, anteriorly notched vertebral bodies and bone-in-bone appearance. Brain atrophy, external hydrocephalus and thin corpus callosum were noted at the brain MRI and CT scan. Blood test results revealed the presence of anaemia and leukopenia. Our findings confirm the role of SNX10 in autosomal recessive osteopetrosis and help to better define the core set of manifestations associated with this new pathological entity.
Copyright © 2012 Elsevier Masson SAS. All rights reserved.

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Year:  2012        PMID: 23123320     DOI: 10.1016/j.ejmg.2012.10.010

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  13 in total

Review 1.  Regulation of lysosome biogenesis and functions in osteoclasts.

Authors:  Julie Lacombe; Gérard Karsenty; Mathieu Ferron
Journal:  Cell Cycle       Date:  2013-08-05       Impact factor: 4.534

2.  Snx10: a newly identified locus associated with human osteopetrosis.

Authors:  Liang Ye; Leslie R Morse; Ricardo A Battaglino
Journal:  IBMS Bonekey       Date:  2013

Review 3.  Osteopetrosis: genetics, treatment and new insights into osteoclast function.

Authors:  Cristina Sobacchi; Ansgar Schulz; Fraser P Coxon; Anna Villa; Miep H Helfrich
Journal:  Nat Rev Endocrinol       Date:  2013-07-23       Impact factor: 43.330

4.  TRAFD1 (FLN29) Interacts with Plekhm1 and Regulates Osteoclast Acidification and Resorption.

Authors:  Hanna Witwicka; Hong Jia; Artem Kutikov; Pablo Reyes-Gutierrez; Xiangdong Li; Paul R Odgren
Journal:  PLoS One       Date:  2015-05-19       Impact factor: 3.240

5.  Osteopetrorickets due to Snx10 deficiency in mice results from both failed osteoclast activity and loss of gastric acid-dependent calcium absorption.

Authors:  Liang Ye; Leslie R Morse; Li Zhang; Hajime Sasaki; Jason C Mills; Paul R Odgren; Greg Sibbel; James R L Stanley; Gee Wong; Ariane Zamarioli; Ricardo A Battaglino
Journal:  PLoS Genet       Date:  2015-03-26       Impact factor: 5.917

Review 6.  Osteopetrosis and its relevance for the discovery of new functions associated with the skeleton.

Authors:  Amélie E Coudert; Marie-Christine de Vernejoul; Maurizio Muraca; Andrea Del Fattore
Journal:  Int J Endocrinol       Date:  2015-03-19       Impact factor: 3.257

7.  SNX16 negatively regulates the migration and tumorigenesis of MCF-7 cells.

Authors:  Leilei Zhang; Dajiang Qin; Chunfang Hao; Xiaodong Shu; Duanqing Pei
Journal:  Cell Regen (Lond)       Date:  2013-04-30

8.  SNX10 gene mutation leading to osteopetrosis with dysfunctional osteoclasts.

Authors:  Eva-Lena Stattin; Petra Henning; Joakim Klar; Emma McDermott; Christina Stecksen-Blicks; Per-Erik Sandström; Therese G Kellgren; Patrik Rydén; Göran Hallmans; Torsten Lönnerholm; Adam Ameur; Miep H Helfrich; Fraser P Coxon; Niklas Dahl; Johan Wikström; Ulf H Lerner
Journal:  Sci Rep       Date:  2017-06-07       Impact factor: 4.379

9.  Cervical spine fractures in osteopetrosis: a case report and review of the literature.

Authors:  Arjang Ahmadpour; Amir Goodarzi; Darrin J Lee; Ripul R Panchal; Kee D Kim
Journal:  J Biomed Res       Date:  2018-01-18

Review 10.  Membrane trafficking in osteoclasts and implications for osteoporosis.

Authors:  Pei Ying Ng; Amy Brigitte Patricia Ribet; Nathan John Pavlos
Journal:  Biochem Soc Trans       Date:  2019-03-05       Impact factor: 5.407

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