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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...
Autores principales: | , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
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Frontiers Media S.A.
2023
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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. |
format | Online Article Text |
id | pubmed-10106626 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | Frontiers Media S.A. |
record_format | MEDLINE/PubMed |
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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