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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...
Autores principales: | , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Hindawi Publishing Corporation
2016
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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. |
format | Online Article Text |
id | pubmed-4745298 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2016 |
publisher | Hindawi Publishing Corporation |
record_format | MEDLINE/PubMed |
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
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title_full | Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
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title_fullStr | Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
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title_full_unstemmed | Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
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title_short | Osteoporosis-Pseudoglioma in a Mauritanian Child due to a Novel Mutation in LRP5
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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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