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Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5

Osteoporosis-pseudoglioma (OPPG) syndrome is a very rare autosomal recessive disorder, caused by mutations in the low-density lipoprotein receptor-related protein 5 (LRP5) gene. It manifests by severe juvenile osteoporosis with congenital or infancy-onset visual loss. We describe a case of OPPG due...

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Autores principales: Biha, Noura, Ghaber, S. M., Hacen, M. M., Collet, Corinne
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi Publishing Corporation 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4745298/
https://www.ncbi.nlm.nih.gov/pubmed/26904320
http://dx.doi.org/10.1155/2016/9814928
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author Biha, Noura
Ghaber, S. M.
Hacen, M. M.
Collet, Corinne
author_facet Biha, Noura
Ghaber, S. M.
Hacen, M. M.
Collet, Corinne
author_sort Biha, Noura
collection PubMed
description Osteoporosis-pseudoglioma (OPPG) syndrome is a very rare autosomal recessive disorder, caused by mutations in the low-density lipoprotein receptor-related protein 5 (LRP5) gene. It manifests by severe juvenile osteoporosis with congenital or infancy-onset visual loss. We describe a case of OPPG due to novel mutation in LRP5 gene, occurring in a female Mauritanian child. This 10-year-old female child was born blind, and after then multiple fragility fractures appeared. PCR amplification and sequencing revealed a novel homozygous nonsense mutation in exon 10 of the LRP5 gene (c.2270G>A; pTrP757(⁎)); this mutation leads to the production of a truncated protein containing 757 amino acids instead of 1615, located in the third β-propeller domain of the LRP5 protein. Both parents were heterozygous for the mutation. This is the first case of the OPPG described in black Africans, which broadens the spectrum of LRP5 gene mutations in OPPG.
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spelling pubmed-47452982016-02-22 Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5 Biha, Noura Ghaber, S. M. Hacen, M. M. Collet, Corinne Case Rep Genet Case Report Osteoporosis-pseudoglioma (OPPG) syndrome is a very rare autosomal recessive disorder, caused by mutations in the low-density lipoprotein receptor-related protein 5 (LRP5) gene. It manifests by severe juvenile osteoporosis with congenital or infancy-onset visual loss. We describe a case of OPPG due to novel mutation in LRP5 gene, occurring in a female Mauritanian child. This 10-year-old female child was born blind, and after then multiple fragility fractures appeared. PCR amplification and sequencing revealed a novel homozygous nonsense mutation in exon 10 of the LRP5 gene (c.2270G>A; pTrP757(⁎)); this mutation leads to the production of a truncated protein containing 757 amino acids instead of 1615, located in the third β-propeller domain of the LRP5 protein. Both parents were heterozygous for the mutation. This is the first case of the OPPG described in black Africans, which broadens the spectrum of LRP5 gene mutations in OPPG. Hindawi Publishing Corporation 2016 2016-01-19 /pmc/articles/PMC4745298/ /pubmed/26904320 http://dx.doi.org/10.1155/2016/9814928 Text en Copyright © 2016 Noura Biha et al. https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Case Report
Biha, Noura
Ghaber, S. M.
Hacen, M. M.
Collet, Corinne
Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
title Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
title_full Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
title_fullStr Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
title_full_unstemmed Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
title_short Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
title_sort osteoporosis-pseudoglioma in a mauritanian child due to a novel mutation in lrp5
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4745298/
https://www.ncbi.nlm.nih.gov/pubmed/26904320
http://dx.doi.org/10.1155/2016/9814928
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