Congenital misalignment of pulmonary veins with alveolar capillary dysplasia causing persistent neonatal pulmonary hypertension: Report of two affected siblings

被引：34

作者：

Gutierrez, C

Rodriguez, A

Palenzuela, S

Forteza, C

Rossello, JLD

机构：

[1] Hosp Pereira Rossell, Pediat Pathol Lab, Montevideo 16059, Uruguay

[2] Hosp Clin Montevideo, Latin Amer Ctr Perinatol & Human Dev, Montevideo 11600, Uruguay

来源：

PEDIATRIC AND DEVELOPMENTAL PATHOLOGY | 2000年 / 3卷 / 03期

关键词：

congenital alveolar capillary dysplasia; family occurrence; misalignment of the pulmonary vessels; persistent neonatal pulmonary hypertension;

D O I：

10.1007/s100249910035

中图分类号：

R36 [病理学];

学科分类号：

100104 ;

摘要：

Misalignment of pulmonary vessels with alveolar capillary dysplasia is a rare cause of persistent pulmonary hypertension of the newborn. Most of the reported cases have been sporadic. We present two consecutive affected siblings with this disorder. This is the fifth reported family occurrence of this condition. In addition to the pulmonary abnormality, one of our cases had duodenal atresia.

引用

收藏

页码：271 / 276

页数：6

相关论文

共 28 条

[21] PRIMARY ALVEOLAR CAPILLARY DYSPLASIA [J].

NEWMAN, B ;

YUNIS, E .

PEDIATRIC RADIOLOGY, 1990, 21 (01) :20-22

[22] CONGENITAL MISALIGNMENT OF PULMONARY VESSELS - AN UNUSUAL SYNDROME-ASSOCIATED WITH PPHN [J].

NG, PC ;

LEWINDON, PJ ;

SIU, YK ;

TO, KF ;

WONG, W .

ACTA PAEDIATRICA, 1995, 84 (03) :349-353

[23] MISALIGNMENT OF LUNG VESSELS AND ALVEOLAR-CAPILLARY DYSPLASIA [J].

OLDENBURG, J ;

VANDERPAL, HJH ;

SCHREVEL, LS ;

BLOK, APR ;

WAGENVOORT, CA .

HISTOPATHOLOGY, 1995, 27 (02) :192-194

[24] Pathological case of the month - Congenital alveolar capillary dysplasia and misalignment of lung vessels [J].

Rehan, V ;

Phillips, S ;

Fajardo, C ;

Seshia, MMK .

ARCHIVES OF PEDIATRICS & ADOLESCENT MEDICINE, 1997, 151 (11) :1163-1164

[25] Alveolar capillary dysplasia: Lung biopsy diagnosis, nitric oxide responsiveness, and bronchial generation count [J].

Sirkin, W ;

OHare, BP ;

Cox, PN ;

Perrin, D ;

Cutz, E ;

Silver, MM .

PEDIATRIC PATHOLOGY & LABORATORY MEDICINE, 1997, 17 (01) :125-132

[26] Inhaled nitric oxide enhances oxygenation but not survival in infants with alveolar capillary dysplasia [J].

Steinhorn, RH ;

Cox, PN ;

Fineman, JR ;

Finer, NN ;

Rosenberg, EM ;

Silver, MM ;

Tyebkhan, J ;

Zwass, MS ;

Morin, FC .

JOURNAL OF PEDIATRICS, 1997, 130 (03) :417-422

[27] Familial persistent pulmonary hypertension of the newborn resulting from misalignment of the pulmonary vessels (congenital alveolar capillary dysplasia) [J].

Vassal, HB ;

Malone, M ;

Petros, AJ ;

Winter, RM .

JOURNAL OF MEDICAL GENETICS, 1998, 35 (01) :58-60

[28] MISALIGNMENT OF LUNG VESSELS - A SYNDROME CAUSING PERSISTENT NEONATAL PULMONARY-HYPERTENSION [J].

WAGENVOORT, CA .

HUMAN PATHOLOGY, 1986, 17 (07) :727-730