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Functional disomy of the Xq28 chromosome region.
Eur J Hum Genet ; 13(5): 579-85, 2005 May.
Article en En | MEDLINE | ID: mdl-15741994
We report on two patients, a boy and a girl, with an additional Xq28 chromosome segment translocated onto the long arm of an autosome. The karyotypes were 46,XY,der(10)t(X;10)(q28;qter) and 46,XX,der(4)t(X;4)(q28;q34), respectively. In both cases, the de novo cryptic unbalanced X-autosome translocation resulted in a Xq28 chromosome functional disomy. To our knowledge, at least 17 patients with a distal Xq chromosome functional disomy have been described in the literature. This is the third report of a girl with an unbalanced translocation yielding such a disomy. When the clinical features of both patients are compared to those observed in patients reported in the literature, a distinct phenotype emerges including severe mental retardation, facial dysmorphic features with a wide face, a small mouth and a thin pointed nose, major axial hypotonia, severe feeding problems and proneness to infections. A clinically oriented FISH study using subtelomeric probes is necessary to detect such a cryptic rearrangement.
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Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Translocación Genética / Anomalías Múltiples / Cromosomas Humanos Par 4 / Cromosomas Humanos Par 10 / Cromosomas Humanos X / Aneuploidia Límite: Female / Humans / Infant / Male Idioma: En Revista: Eur J Hum Genet Asunto de la revista: GENETICA MEDICA Año: 2005 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Reino Unido
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Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Translocación Genética / Anomalías Múltiples / Cromosomas Humanos Par 4 / Cromosomas Humanos Par 10 / Cromosomas Humanos X / Aneuploidia Límite: Female / Humans / Infant / Male Idioma: En Revista: Eur J Hum Genet Asunto de la revista: GENETICA MEDICA Año: 2005 Tipo del documento: Article País de afiliación: Francia Pais de publicación: Reino Unido