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DOI 10.1002/ajmg.a.33018

Медицинская библиотека · American journal of medical genetics. Part A · 2009

Progeroid syndrome with scleroderma-like skin changes associated with homozygous R435C LMNA mutation.

Madej-Pilarczyk Agnieszka, Rosińska-Borkowska Danuta, Rekawek Joanna, Marchel Michał, Szaluś Ewa, Jabłońska Stefania, Hausmanowa-Petrusewicz Irena

Клинический случайСтатьяПоддержка исследования: вне правительства СШАПеревод в очереди

Аннотация

Hutchinson-Gilford progeria is a rare genetic disorder resulting from mutations in the LMNA gene encoding lamin A/C. In addition to the classical phenotype usually caused by the 1824C>T mutation of LMNA, a number of atypical progeroid syndromes have been described. They have some distinct features, such as skeletal deformities or scleroderma-like skin changes. The underlying defect is usually a homozygous mutation of LMNA, or a combined defect of LMNA and another gene, for example, ZMPSTE-24. We present a 2-year-old girl born to consanguineous parents affected by progeroid syndrome with scleroderma-like skin changes. Genetic analysis revealed the homozygous LMNA mutation 1303C>T (R435C). The same heterozygous mutation was found in the patient's parents and 11 other family members. The progeroid syndrome in our patient shares the signs of two laminopathies: progeria and restrictive dermatopathy. Two other children in the family died at the age of 2 due to a disease similar to that in the proposita. On the basis of the family pedigree we presume that these children probably had the same homozygous LMNA mutation. Scleroderma-like skin changes in infants, associated with growth retardation and dysmorphic features, suggest premature aging syndrome, requiring genetic testing and counseling of asymptomatic carriers of LMNA mutations.

Для цитирования: Madej-Pilarczyk Agnieszka, Rosińska-Borkowska Danuta, Rekawek Joanna, Marchel Michał, Szaluś Ewa, Jabłońska Stefania, Hausmanowa-Petrusewicz Irena. Progeroid syndrome with scleroderma-like skin changes associated with homozygous R435C LMNA mutation.. American journal of medical genetics. Part A. 2009. DOI: 10.1002/ajmg.a.33018.

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