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[Clinical thinking and decision making in practice: a full-term neonate with misunderstood respiratory insufficiency].

Authors :
van Heijst AF
Semmekrot BA
Kollée LA
van de Bor M
Source :
Nederlands tijdschrift voor geneeskunde [Ned Tijdschr Geneeskd] 1999 Jun 12; Vol. 143 (24), pp. 1256-60.
Publication Year :
1999

Abstract

A fullterm newborn boy developed severe respiratory insufficiency, multiple air leaks and severe pulmonary hypertension, leading to his death on the third day of life. Family history revealed that a sister of the patient had died earlier after a similar course with respiratory problems. The most common causes of respiratory insufficiency could be subsequently excluded. After extensive postmortem investigation alveolar proteinosis was found in the lung tissue. DNA investigation was then performed in the parents, and both appeared to be heterozygotic for the 121ins2 mutation. This finding suggests both children in this family to have been homozygotic for the 121ins2 mutation resulting in a lack of synthesis of surfactant protein B (SP-B). Homozygotic SP-B deficiency in the newborn is a fatal disease with no curative perspectives, except for lung transplantation and gene therapy.

Details

Language :
Dutch; Flemish
ISSN :
0028-2162
Volume :
143
Issue :
24
Database :
MEDLINE
Journal :
Nederlands tijdschrift voor geneeskunde
Publication Type :
Academic Journal
Accession number :
10396336