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J Pediatr ; 132(2): 244-8, 1998 Feb.
Artículo en Inglés | MEDLINE | ID: mdl-9506635

RESUMEN

A 38-day-old male infant with persistent pulmonary hypertension and respiratory failure since birth was found to have a complete absence of surfactant protein B (SP-B) along with an aberrant form of SP-C in his tracheal aspirate fluid, findings consistent with the diagnosis of hereditary SP-B deficiency. Surprisingly, SP-B and SP-B messenger ribonucleic acid were present in lung biopsy tissue. However, DNA sequence analysis demonstrated a point mutation in exon 5 of one of the SP-B gene alleles. The infant's mother was found to be a carrier of this mutation. The infant's other SP-B allele did not differ from the published DNA sequence for the SP-B gene. We conclude that this patient had a transient deficiency of SP-B, in contrast to that of previously described infants with irreversible respiratory failure caused by hereditary SP-B deficiency. We recommend that infants with suspected SP-B deficiency have serial analysis of tracheal fluid samples for both SP-B and SP-C before lung biopsy, along with genetic analysis for the known SP-B mutations. We speculate that the new mutation found in one of this patient's SP-B genes was in part responsible for the transient deficiency of SP-B.


Asunto(s)
Errores Innatos del Metabolismo/genética , Proteolípidos/metabolismo , Surfactantes Pulmonares/deficiencia , Surfactantes Pulmonares/metabolismo , Síndrome de Dificultad Respiratoria del Recién Nacido/metabolismo , Exudados y Transudados/química , Humanos , Immunoblotting , Lactante , Recién Nacido , Masculino , Errores Innatos del Metabolismo/complicaciones , Mutación , Reacción en Cadena de la Polimerasa , Proteolípidos/genética , Surfactantes Pulmonares/genética , Síndrome de Dificultad Respiratoria del Recién Nacido/complicaciones , Análisis de Secuencia de ADN , Factores de Tiempo , Tráquea
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