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新生儿全身性淋巴管发育异常的新型临床表型:漏诊病例

Novel clinical phenotype of generalised lymphatic dysplasia in a neonate: a missed diagnosis.

作者信息

Abiramalatha Thangaraj, Johnson Thanka, Balakrishnan Umamaheswari, Amboiram Prakash

机构信息

Neonatology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India.

Pathology, Sri Ramachandra Institute of Higher Education and Research, Chennai, Tamil Nadu, India.

出版信息

BMJ Case Rep. 2019 Aug 30;12(8):e229260. doi: 10.1136/bcr-2019-229260.

Abstract

We report a preterm neonate who had a large cervical cystic hygroma and right chylothorax. She was operated on day-21 and a near-complete resection of cystic hygroma was done. She developed refractory hypoxemia and shock post surgery and died after 24 hours. During autopsy, the chest cavity was found to be filled with chyle. Histopathological examination showed dilated lymphatics in the pleura, hepatic capsule, serosa of stomach and intestines, peri-pancreatic regions, peri-renal capsule and peri-adrenal tissues suggestive of generalised lymphatic dysplasia. Clinical exome sequencing did not reveal any pathogenic mutation in the genes involved in primary lymphatic dysplasia, noonan syndrome or rasopathies.

摘要

我们报告了一名患有巨大颈部囊状水瘤和右侧乳糜胸的早产儿。她在出生后第21天接受了手术,对囊状水瘤进行了近乎完全的切除。术后她出现了难治性低氧血症和休克,并在24小时后死亡。尸检时发现胸腔充满乳糜。组织病理学检查显示,胸膜、肝包膜、胃和肠的浆膜、胰腺周围区域、肾包膜和肾上腺周围组织中的淋巴管扩张,提示全身性淋巴管发育异常。临床外显子组测序未发现原发性淋巴管发育异常、努南综合征或RAS病相关基因的任何致病突变。

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本文引用的文献

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Treatment guidelines of lymphatic malformations of the head and neck.头颈部淋巴管畸形治疗指南。
Oral Oncol. 2011 Dec;47(12):1105-9. doi: 10.1016/j.oraloncology.2011.08.001. Epub 2011 Sep 8.
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Sclerotherapy of lymphangiomas of the head and neck.头颈部淋巴管瘤的硬化疗法。
Head Neck. 2011 Nov;33(11):1649-55. doi: 10.1002/hed.21552. Epub 2010 Aug 24.

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