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Five novel genes from the cri-du-chat critical region isolated by direct selection.
Simmons, A D; Goodart, S A; Gallardo, T D; Overhauser, J; Lovett, M.
Afiliación
  • Simmons AD; Department of Biochemistry, University of Texas Southwestern Medical Center at Dallas 75235-8591, USA.
Hum Mol Genet ; 4(2): 295-302, 1995 Feb.
Article en En | MEDLINE | ID: mdl-7757083
Cri-du-chat is a well described partial aneusomy resulting from deletion of the short arm of chromosome 5. The hallmark clinical feature of cri-du-chat, a high-pitched monochromatic cry, has recently been localized to 5p15.3, separate from the remaining clinical features of the syndrome, which have been localized to 5p15.2. Five chromosome 5-specific probes from the latter region, designated the cri-du-chat critical region (CDCCR), were used to isolate 30 cosmids from the LANL chromosome 5 specific cosmid library. The 30 framework cosmids were used in a direct selection with three cDNA sources to isolate an initial set of expressed sequences. Nine unique cDNAs were found that hybridized to four discrete sets of cosmids in the CDCCR. The nine cDNAs are novel by sequence database comparisons, and conservatively represent four transcription units. More recently, we have also constructed a YAC contig of the CDCCR which spans approximately 2 Mb. As expected, ESTs derived from the nine novel cDNAs map back to the contig. Limited expression profiles of these cDNAs have been obtained. Two cDNAs that map to one discrete set of cosmids have different expression patterns, suggesting that they represent two different genes and increasing the number of putative genes to five. Further characterization of these genes and the estimated 100 additional genes deleted in cri-du-chat should lead to better diagnostic markers and an understanding of the molecular mechanisms of the disease.
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Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Cromosomas Humanos Par 5 / Síndrome del Maullido del Gato Límite: Humans Idioma: En Revista: Hum Mol Genet Asunto de la revista: BIOLOGIA MOLECULAR / GENETICA MEDICA Año: 1995 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Reino Unido
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Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Cromosomas Humanos Par 5 / Síndrome del Maullido del Gato Límite: Humans Idioma: En Revista: Hum Mol Genet Asunto de la revista: BIOLOGIA MOLECULAR / GENETICA MEDICA Año: 1995 Tipo del documento: Article País de afiliación: Estados Unidos Pais de publicación: Reino Unido