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DOI 10.1002/ajmg.a.62375

Медицинская библиотека · American journal of medical genetics. Part A · 2021

Demographic, clinical, and ancestry characterization of a large cluster of mucopolysaccharidosis IV A in the Brazilian Northeast region.

Dos Santos-Lopes Simone Silva, de Oliveira Jessica Maria Florêncio, de Queiroga Nascimento Denise, Montenegro Yorran Hardman Araújo, Leistner-Segal Sandra, Brusius-Facchin Ana Carolina, Eufrazino Gondim Cátia, Giugliani Roberto, de Medeiros Paula Frassinetti Vasconcelos

СтатьяПоддержка исследования: вне правительства СШАПеревод пока не подготовлен

Аннотация

Mucopolysaccharidosis (MPS) IVA is a rare autosomal recessive disease with a highly variable distribution worldwide. Discrepancies in the incidence of MPS IVA among populations of different ethnicities are mostly attributed to founder effects. Demographic and clinical data from 28 MPS IVA patients, followed at a single center, and ancestry (Y chromosome and mitochondrial markers) of a subsample of 17 patients, most with the p.Ser341Arg (c.1023C>G) mutation were analyzed. Parental consanguinity was observed in 15/20 couples; a rare homozygous N-acetylgalactosamine-6-sulfatase (GALNS) mutation was found in 7/16 families with intra-familial phenotypic heterogeneity. Paternal ancestry was 94.2% (16/17) European, 5.8% (1/17) African, and 0% Amerindian. The European paternal haplogroups R1a, R1b, and R* accounted for 94.2% (16/17) of the patients. The R1b haplogroup, identified in 59% (10/17) of the patients, is frequently found in populations from the Iberian Peninsula. European, Amerindian, and African maternal ancestry was observed in 46.9% (8/17), 35.4% (6/17), and 17.7% (3/17) of the patients, respectively. Study of a cluster of MPS IVA patients from Northeastern Brazil, with high parental consanguinity and phenotypic heterogeneity showed predominantly European parental ancestry. This ancestry finding corroborates historical data on the local settlement, formed predominantly by European men.

Для цитирования: Dos Santos-Lopes Simone Silva, de Oliveira Jessica Maria Florêncio, de Queiroga Nascimento Denise, Montenegro Yorran Hardman Araújo, Leistner-Segal Sandra, Brusius-Facchin Ana Carolina, Eufrazino Gondim Cátia, Giugliani Roberto, de Medeiros Paula Frassinetti Vasconcelos. Demographic, clinical, and ancestry characterization of a large cluster of mucopolysaccharidosis IV A in the Brazilian Northeast region.. American journal of medical genetics. Part A. 2021. DOI: 10.1002/ajmg.a.62375.

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