Evidence map›Paper›PMID 36901946›Full record

ArticleInternational journal of molecular sciences2023

Genetic Heterogeneity of X-Linked Ichthyosis in the Republic of North Ossetia-Alania, Case Series Report.

Tatyana A Vasilyeva, Andrey V Marakhonov, Inna S Tebieva, Vitaly V Kadyshev, Artem O Borovikov, Zhanna G Markova, Alyona L Chukhrova, Evgeny K Ginter, Sergey I Kutsev, Rena A Zinchenko

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Article in International journal of molecular sciences, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 2 papers.

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2citing papers in PubMed
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1 · What the graph read from it

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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

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3 · Its place in the literature

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2 citing papers in PubMed.

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4 · The record

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5 · Who and what money

Authors and funding

10 authors.

Tatyana A VasilyevaResearch Centre for Medical Genetics, 115522 Moscow, Russia.
Andrey V MarakhonovResearch Centre for Medical Genetics, 115522 Moscow, Russia.ORCID 0000-0002-0972-5118
Inna S TebievaRepublican Children's Clinical Hospital, 362003 Vladikavkaz, Russia.
Vitaly V KadyshevResearch Centre for Medical Genetics, 115522 Moscow, Russia.
Artem O BorovikovResearch Centre for Medical Genetics, 115522 Moscow, Russia.ORCID 0000-0001-5871-8005
Zhanna G MarkovaResearch Centre for Medical Genetics, 115522 Moscow, Russia.ORCID 0000-0003-2941-2861
Alyona L ChukhrovaResearch Centre for Medical Genetics, 115522 Moscow, Russia.
Evgeny K GinterResearch Centre for Medical Genetics, 115522 Moscow, Russia.
Sergey I KutsevResearch Centre for Medical Genetics, 115522 Moscow, Russia.
Rena A ZinchenkoResearch Centre for Medical Genetics, 115522 Moscow, Russia.ORCID 0000-0003-3586-3458

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

North Caucasus has always been a residence of a lot of different authentic ethnic groups speaking different languages and still living their traditional lifestyle. The diversity appeared to be reflected in the accumulation of different mutations causing common inherited disorders. X-linked ichthyosis represents the second most common form of genodermatoses after ichthyosis vulgaris. Eight patients from three unrelated families of different ethnic origin, Kumyk, Turkish Meskhetians, and Ossetian, with X-linked ichthyosis from the North Caucasian Republic of North Ossetia-Alania were examined. NGS technology was implied for searching for disease-causing variants in one of the index patients. Known pathogenic hemizygous deletion in the short arm of chromosome X encompassing the STS gene was defined in the Kumyk family. A further analysis allowed us to establish that likely the same deletion was a cause of ichthyosis in a family belonging to the Turkish Meskhetians ethnic group. In the Ossetian family, a likely pathogenic nucleotide substitution in the STS gene was defined; it segregated with the disease in the family. We molecularly confirmed XLI in eight patients from three examined families. Though in two families, Kumyk and Turkish Meskhetian, we revealed similar hemizygous deletions in the short arm of chromosome X, but their common origin was not likely. Forensic STR markers of the alleles carrying the deletion were defined to be different. However, here, common alleles haplotype is hard to track for a high local recombination rate. We supposed the deletion could arise as a de novo event in a recombination hot spot in the described and in other populations with a recurrent character. Defined here are the different molecular genetic causes of X-linked ichthyosis in families of different ethnic origins sharing the same residence place in the Republic of North Ossetia-Alania which could point to the existing reproductive barriers even inside close neighborhoods.

Indexed as

IchthyosisIchthyosis, X-LinkedGenetic HeterogeneityHumansSteryl-SulfataseX ChromosomeSteryl-Sulfatasegross chromosomal deletionsNorth Ossetia–AlaniaSTR markersSTS geneX-linked ichthyosis

Identifiers

PMID36901946
PMCPMC10003119

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