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A Recurrent Mutation in Growth Hormone Receptor (GHR) Gene Underlying Laron-type Dwarfism in a Pakistani Family
Laron syndrome (LS) is a rare autosomal recessively segregating disorder of severe short stature. The condition is characterized by short limbs, delayed puberty, hypoglycemia in infancy, and obesity. Mutations in growth hormone receptor (GHR) have been implicated in LS; hence, it is also known as gr...
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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YJBM
2023
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10524814/ https://www.ncbi.nlm.nih.gov/pubmed/37780997 http://dx.doi.org/10.59249/TCAA2040 |
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author | Shabbir, Rana Muhammad Kamran Nalbant, Gökhan Zaman, Qamar Tolun, Aslıhan Malik, Sajid Mumtaz, Sara |
author_facet | Shabbir, Rana Muhammad Kamran Nalbant, Gökhan Zaman, Qamar Tolun, Aslıhan Malik, Sajid Mumtaz, Sara |
author_sort | Shabbir, Rana Muhammad Kamran |
collection | PubMed |
description | Laron syndrome (LS) is a rare autosomal recessively segregating disorder of severe short stature. The condition is characterized by short limbs, delayed puberty, hypoglycemia in infancy, and obesity. Mutations in growth hormone receptor (GHR) have been implicated in LS; hence, it is also known as growth hormone insensitivity syndrome (MIM-262500). Here we represent a consanguineous Pakistani family in which three siblings were afflicted with LS. Patients had rather similar phenotypic presentations marked with short stature, delayed bone age, limited extension of elbows, truncal obesity, delayed puberty, childish appearance, and frontal bossing. They also had additional features such as hypo-muscularity, early fatigue, large ears, widely-spaced breasts, and attention deficit behavior, which are rarely reported in LS. The unusual combination of the features hindered a straightforward diagnosis and prompted us to first detect the regions of shared homozygosity and subsequently the disease-causing variant by next generation technologies, like SNP genotyping and exome sequencing. A homozygous pathogenic variant c.508G>C (p.(Asp170His)) in GHR was detected. The variant is known to be implicated in LS, supporting the molecular diagnosis of LS. Also, we present detailed clinical, hematological, and hormonal profiling of the siblings. |
format | Online Article Text |
id | pubmed-10524814 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | YJBM |
record_format | MEDLINE/PubMed |
spelling | pubmed-105248142023-09-29 A Recurrent Mutation in Growth Hormone Receptor (GHR) Gene Underlying Laron-type Dwarfism in a Pakistani Family Shabbir, Rana Muhammad Kamran Nalbant, Gökhan Zaman, Qamar Tolun, Aslıhan Malik, Sajid Mumtaz, Sara Yale J Biol Med Original Contribution Laron syndrome (LS) is a rare autosomal recessively segregating disorder of severe short stature. The condition is characterized by short limbs, delayed puberty, hypoglycemia in infancy, and obesity. Mutations in growth hormone receptor (GHR) have been implicated in LS; hence, it is also known as growth hormone insensitivity syndrome (MIM-262500). Here we represent a consanguineous Pakistani family in which three siblings were afflicted with LS. Patients had rather similar phenotypic presentations marked with short stature, delayed bone age, limited extension of elbows, truncal obesity, delayed puberty, childish appearance, and frontal bossing. They also had additional features such as hypo-muscularity, early fatigue, large ears, widely-spaced breasts, and attention deficit behavior, which are rarely reported in LS. The unusual combination of the features hindered a straightforward diagnosis and prompted us to first detect the regions of shared homozygosity and subsequently the disease-causing variant by next generation technologies, like SNP genotyping and exome sequencing. A homozygous pathogenic variant c.508G>C (p.(Asp170His)) in GHR was detected. The variant is known to be implicated in LS, supporting the molecular diagnosis of LS. Also, we present detailed clinical, hematological, and hormonal profiling of the siblings. YJBM 2023-09-29 /pmc/articles/PMC10524814/ /pubmed/37780997 http://dx.doi.org/10.59249/TCAA2040 Text en Copyright ©2023, Yale Journal of Biology and Medicine https://creativecommons.org/licenses/by-nc/4.0/This is an open access article distributed under the terms of the Creative Commons CC BY-NC license, which permits use, distribution, and reproduction in any medium, provided the original work is properly cited. You may not use the material for commercial purposes. |
spellingShingle | Original Contribution Shabbir, Rana Muhammad Kamran Nalbant, Gökhan Zaman, Qamar Tolun, Aslıhan Malik, Sajid Mumtaz, Sara A Recurrent Mutation in Growth Hormone Receptor (GHR) Gene Underlying Laron-type Dwarfism in a Pakistani Family |
title | A Recurrent Mutation in Growth Hormone Receptor
(GHR) Gene Underlying Laron-type Dwarfism in a Pakistani
Family |
title_full | A Recurrent Mutation in Growth Hormone Receptor
(GHR) Gene Underlying Laron-type Dwarfism in a Pakistani
Family |
title_fullStr | A Recurrent Mutation in Growth Hormone Receptor
(GHR) Gene Underlying Laron-type Dwarfism in a Pakistani
Family |
title_full_unstemmed | A Recurrent Mutation in Growth Hormone Receptor
(GHR) Gene Underlying Laron-type Dwarfism in a Pakistani
Family |
title_short | A Recurrent Mutation in Growth Hormone Receptor
(GHR) Gene Underlying Laron-type Dwarfism in a Pakistani
Family |
title_sort | recurrent mutation in growth hormone receptor
(ghr) gene underlying laron-type dwarfism in a pakistani
family |
topic | Original Contribution |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10524814/ https://www.ncbi.nlm.nih.gov/pubmed/37780997 http://dx.doi.org/10.59249/TCAA2040 |
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