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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...

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Autores principales: Shabbir, Rana Muhammad Kamran, Nalbant, Gökhan, Zaman, Qamar, Tolun, Aslıhan, Malik, Sajid, Mumtaz, Sara
Formato: Online Artículo Texto
Lenguaje:English
Publicado: YJBM 2023
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.
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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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