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Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report

Hereditary spastic paraplegias (HSP) are inherited neurodegenerative disorders characterized by progressive paraplegia and spasticity in the lower limbs. SPG48 represents a rare genotype characterized by mutations in AP5Z1, a gene playing a role in intracellular membrane trafficking. This study desc...

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Autores principales: Jin, Ping, Wang, Yu, Nian, Na, Wang, Gong-Qiang, Fu, Xiao-Ming
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10106626/
https://www.ncbi.nlm.nih.gov/pubmed/37077568
http://dx.doi.org/10.3389/fneur.2023.1156100
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author Jin, Ping
Wang, Yu
Nian, Na
Wang, Gong-Qiang
Fu, Xiao-Ming
author_facet Jin, Ping
Wang, Yu
Nian, Na
Wang, Gong-Qiang
Fu, Xiao-Ming
author_sort Jin, Ping
collection PubMed
description Hereditary spastic paraplegias (HSP) are inherited neurodegenerative disorders characterized by progressive paraplegia and spasticity in the lower limbs. SPG48 represents a rare genotype characterized by mutations in AP5Z1, a gene playing a role in intracellular membrane trafficking. This study describes a case of a 53-year-old male patient with SPG48 presenting spastic paraplegia, infertility, hearing impairment, cognitive abnormalities and peripheral neuropathy. The Sanger sequencing revealed a homozygous deletion in the chr 7:4785904-4786677 region causing a premature stop codon in exon 10. The patient's brother was heterozygous for the mutation. The brain magnetic resonance imaging found a mild brain atrophy and white matter lesions. In the analysis of the auditory thresholds, we found a significant hearing decrease in both ears.
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spelling pubmed-101066262023-04-18 Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report Jin, Ping Wang, Yu Nian, Na Wang, Gong-Qiang Fu, Xiao-Ming Front Neurol Neurology Hereditary spastic paraplegias (HSP) are inherited neurodegenerative disorders characterized by progressive paraplegia and spasticity in the lower limbs. SPG48 represents a rare genotype characterized by mutations in AP5Z1, a gene playing a role in intracellular membrane trafficking. This study describes a case of a 53-year-old male patient with SPG48 presenting spastic paraplegia, infertility, hearing impairment, cognitive abnormalities and peripheral neuropathy. The Sanger sequencing revealed a homozygous deletion in the chr 7:4785904-4786677 region causing a premature stop codon in exon 10. The patient's brother was heterozygous for the mutation. The brain magnetic resonance imaging found a mild brain atrophy and white matter lesions. In the analysis of the auditory thresholds, we found a significant hearing decrease in both ears. Frontiers Media S.A. 2023-04-03 /pmc/articles/PMC10106626/ /pubmed/37077568 http://dx.doi.org/10.3389/fneur.2023.1156100 Text en Copyright © 2023 Jin, Wang, Nian, Wang and Fu. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Neurology
Jin, Ping
Wang, Yu
Nian, Na
Wang, Gong-Qiang
Fu, Xiao-Ming
Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report
title Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report
title_full Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report
title_fullStr Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report
title_full_unstemmed Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report
title_short Hereditary spastic paraplegia (SPG 48) with deafness and azoospermia: A case report
title_sort hereditary spastic paraplegia (spg 48) with deafness and azoospermia: a case report
topic Neurology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10106626/
https://www.ncbi.nlm.nih.gov/pubmed/37077568
http://dx.doi.org/10.3389/fneur.2023.1156100
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