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Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect
Mucolipidosis Type IV (MLIV) is caused by a deficiency of the mucolipin cation channel encoded by Mucolipin TRP Cation Channel 1 gene (MCOLN1). It is a slowly progressive neurodevelopmental and neurodegenerative disorder causing severe psychomotor developmental delay and progressive visual impairmen...
Autores principales: | , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2022
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8872508/ https://www.ncbi.nlm.nih.gov/pubmed/35205297 http://dx.doi.org/10.3390/genes13020248 |
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author | Al-Alawi, Badriya Harikrishna, Beena Al-Thihli, Khalid Al Zuhabi, Sana Ganesh, Anuradha Al Hashami, Zainab Al Dhamhmani, Zeyana Zadjali, Razan Al Riyami, Nafila B. Zadjali, Fahad |
author_facet | Al-Alawi, Badriya Harikrishna, Beena Al-Thihli, Khalid Al Zuhabi, Sana Ganesh, Anuradha Al Hashami, Zainab Al Dhamhmani, Zeyana Zadjali, Razan Al Riyami, Nafila B. Zadjali, Fahad |
author_sort | Al-Alawi, Badriya |
collection | PubMed |
description | Mucolipidosis Type IV (MLIV) is caused by a deficiency of the mucolipin cation channel encoded by Mucolipin TRP Cation Channel 1 gene (MCOLN1). It is a slowly progressive neurodevelopmental and neurodegenerative disorder causing severe psychomotor developmental delay and progressive visual impairment, which is often misdiagnosed as cerebral palsy. We describe six patients with MLIV from two Omani families with a novel c.237+5G>A mutation in the MCOLN1 gene predicted to affect mRNA splicing. Mutation screening with a high-resolution melting (HRM) assay in a large population sample did not detect this mutation in control subjects. This report highlights the importance of considering MLIV in the differential diagnosis of patients in a pediatric age group with cerebral palsy-like presentation. Although the same rare mutation was seen in two apparently unrelated families, this was not seen in the sample screened from the general population. The HRM assay provides a cost-effective assay for population screening for the c.237+5G>A mutation. |
format | Online Article Text |
id | pubmed-8872508 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2022 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
spelling | pubmed-88725082022-02-25 Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect Al-Alawi, Badriya Harikrishna, Beena Al-Thihli, Khalid Al Zuhabi, Sana Ganesh, Anuradha Al Hashami, Zainab Al Dhamhmani, Zeyana Zadjali, Razan Al Riyami, Nafila B. Zadjali, Fahad Genes (Basel) Article Mucolipidosis Type IV (MLIV) is caused by a deficiency of the mucolipin cation channel encoded by Mucolipin TRP Cation Channel 1 gene (MCOLN1). It is a slowly progressive neurodevelopmental and neurodegenerative disorder causing severe psychomotor developmental delay and progressive visual impairment, which is often misdiagnosed as cerebral palsy. We describe six patients with MLIV from two Omani families with a novel c.237+5G>A mutation in the MCOLN1 gene predicted to affect mRNA splicing. Mutation screening with a high-resolution melting (HRM) assay in a large population sample did not detect this mutation in control subjects. This report highlights the importance of considering MLIV in the differential diagnosis of patients in a pediatric age group with cerebral palsy-like presentation. Although the same rare mutation was seen in two apparently unrelated families, this was not seen in the sample screened from the general population. The HRM assay provides a cost-effective assay for population screening for the c.237+5G>A mutation. MDPI 2022-01-28 /pmc/articles/PMC8872508/ /pubmed/35205297 http://dx.doi.org/10.3390/genes13020248 Text en © 2022 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/). |
spellingShingle | Article Al-Alawi, Badriya Harikrishna, Beena Al-Thihli, Khalid Al Zuhabi, Sana Ganesh, Anuradha Al Hashami, Zainab Al Dhamhmani, Zeyana Zadjali, Razan Al Riyami, Nafila B. Zadjali, Fahad Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect |
title | Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect |
title_full | Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect |
title_fullStr | Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect |
title_full_unstemmed | Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect |
title_short | Mucolipidosis Type IV in Omani Families with a Novel MCOLN1 Mutation: Search for Evidence of Founder Effect |
title_sort | mucolipidosis type iv in omani families with a novel mcoln1 mutation: search for evidence of founder effect |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8872508/ https://www.ncbi.nlm.nih.gov/pubmed/35205297 http://dx.doi.org/10.3390/genes13020248 |
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