Mathur Deepan, Somashekar Santhosh, Navarrete Cristina, Rodriguez Maria M
1Pediatric Pathology, Department of Pathology, and.
Fetal Pediatr Pathol. 2014 Aug;33(4):253-7. doi: 10.3109/15513815.2014.904026. Epub 2014 Apr 22.
Noonan Syndrome is an autosomal dominant disorder characterized by short stature, congenital heart defects, developmental delay, dysmorphic facial features and occasional lymphatic dysplasias. The features of Noonan Syndrome change with age and have variable expression. The diagnosis has historically been based on clinical grounds. We describe a child that was born with congenital refractory chylothorax and subcutaneous edema suspected to be secondary to pulmonary lymphangiectasis. The infant died of respiratory failure and anasarca at 80 days. The autopsy confirmed lymphatic dysplasia in lungs and mesentery. The baby had no dysmorphic facial features and was diagnosed postmortem with Noonan syndrome by genomic DNA sequence analysis as he had a heterozygous mutation for G503R in the PTPN11 gene.
努南综合征是一种常染色体显性疾病,其特征为身材矮小、先天性心脏缺陷、发育迟缓、面部畸形以及偶尔出现的淋巴系统发育异常。努南综合征的特征会随年龄变化且表现各异。历史上,该病症的诊断基于临床症状。我们描述了一名出生时患有先天性难治性乳糜胸和皮下水肿的患儿,怀疑这是由肺淋巴管扩张继发引起的。该婴儿在80天时死于呼吸衰竭和全身水肿。尸检证实肺部和肠系膜存在淋巴系统发育异常。这名婴儿没有面部畸形特征,在死后通过基因组DNA序列分析被诊断为努南综合征,因为他在PTPN11基因中存在G503R杂合突变。