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[Clinical thinking and decision making in practice: a full-term neonate with misunderstood respiratory insufficiency].
- 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.
- Subjects :
- Diagnosis, Differential
Fatal Outcome
Humans
Infant, Newborn
Lung pathology
Male
Proteolipids metabolism
Pulmonary Alveoli pathology
Pulmonary Surfactants metabolism
Respiratory Distress Syndrome, Newborn diagnosis
Mutation
Proteolipids genetics
Pulmonary Alveolar Proteinosis diagnosis
Pulmonary Alveolar Proteinosis genetics
Pulmonary Surfactants genetics
Respiratory Insufficiency genetics
Subjects
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