Omphalocele and alveolar capillary dysplasia: A new association

被引：14

作者：

Gerrits, Luella C. ^{[1
]}

De Mol, Amerik C. ^{[2
]}

Bulten, Johan ^{[3
]}

Van der Staak, Frans H. ^{[4
]}

Van Heijst, Arno F. J. ^{[1
]}

机构：

[1] Radboud Univ Nijmegen, Med Ctr, Dept Neonatol, NL-6525 ED Nijmegen, Netherlands

[2] Albert Schweitzer Hosp, Dept Pediat, Dordrecht, Netherlands

[3] Radboud Univ Nijmegen, Med Ctr, Dept Pathol, NL-6525 ED Nijmegen, Netherlands

[4] Radboud Univ Nijmegen, Med Ctr, Dept Pediat Surg, NL-6525 ED Nijmegen, Netherlands

来源：

PEDIATRIC CRITICAL CARE MEDICINE | 2010年 / 11卷 / 03期

关键词：

omphalocele; neonate; alveolar capillary dysplasia; extracorporeal membrane oxygenation; PERSISTENT PULMONARY-HYPERTENSION; ABDOMINAL-WALL DEFECTS; GIANT OMPHALOCELE; MISALIGNMENT; VESSELS;

D O I：

10.1097/PCC.0b013e3181b80a75

中图分类号：

R4 [临床医学];

学科分类号：

1002 ; 100602 ;

摘要：

Objective: First report of an infant with coexistent omphalocele and alveolar capillary dysplasia. Design: Descriptive case report. Setting: Neonatal intensive care unit of a tertiary care children's hospital. Patient: We describe a term infant with omphalocele and respiratory insufficiency attributable to pulmonary hypertension. The patient was placed on extracorporeal membrane oxygenation, but the pulmonary hypertension persisted. After 10 days on extracorporeal membrane oxygenation, a lung biopsy was per-formed. It showed alveolar capillary dysplasia. Because of the lethal prognosis, extracorporeal membrane oxygenation was withdrawn and the patient expired. Conclusions: This is the first description of an association between omphalocele and alveolar capillary dysplasia. In new-borns with omphalocele who have severe respiratory insufficiency and pulmonary hypertension, alveolar capillary dysplasia should be considered. (Pediatr Crit Care Med 2010; 11:e36-e37)

引用

页码：E36 / E37

页数：2

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