Clinical, Radiologic, and Pathologic Features of ABCA3 Mutations in Children

نویسندگان

  • Minh L Doan
  • R Paul Guillerman
  • Megan K Dishop
  • Lawrence M Nogee
  • Claire Langston
  • George B Mallory
  • Marianna M Sockrider
  • Leland L Fan
چکیده

Pediatric Pulmonary Section, Department of Pediatrics, Baylor College of Medicine, Texas Children's Hospital, Houston, Texas, U.S.A. Department of Radiology, Baylor College of Medicine, Texas Children's Hospital, Houston, Texas, U.S.A. Department of Pathology, Baylor College of Medicine, Texas Children's Hospital, Houston, Texas, U.S.A. Division of Neonatology, Department of Pediatrics, Johns Hopkins University School of Medicine, Baltimore, Maryland, U.S.A.

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Clinical, radiological and pathological features of ABCA3 mutations in children.

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In this review we reported and discussed the structural features of the ATP-Binding Cassette (ABC) transporter ABCA3 and how the use of bioinformatics tools could help researchers to obtain a reliable structural model of this important transporter. In fact, a model of ABCA3 is still lacking and no crystallographic structures (of the transporter or of its orthologues) are available. With the adv...

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BACKGROUND ABCA3 transporter (ATP-binding cassette transporter of the A subfamily) is localized to the limiting membrane of lamellar bodies, organelles for assembly and storage of pulmonary surfactant in alveolar epithelial type II cells (AECII). It transports surfactant phospholipids into lamellar bodies and absence of ABCA3 function disrupts lamellar body biogenesis. Mutations of the ABCA3 ge...

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تاریخ انتشار 2007