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Abstract

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Article in Japanese

Case Report

A case of diffuse pulmonary meningotheliomatosis

Yuichi Shimaokaa  Kosuke Ichikawaa  Tatsuki Oharaa  Katsuaki Asakawab  Masaki Teradac  Jouji Toyamaa 

aDepartment of Respiratory Medicine, Joetsu General Hospital
bDivision of Respiratory Medicine, Niigata University
cDepartment of Respiratory Medicine, Saiseikai Niigata Second Hospital

ABSTRACT

A 54-year-old woman was admitted because of an abnormality in the chest X-ray. At the age of 17, she had been diagnosed as Turner syndrome and received female hormone replacement therapy. Although she had no symptom, the chest CT showed multiple small nodular shadows in bilateral lung fields. Laboratory data showed mild elevation of ProGRP. Video-assisted thoracoscopic lung biopsy was performed. Histologically these nodules contained cell foci with pathognomonic whorl formation. Immunohistochemically, the cells were positive for EMA, vimentin, and progesterone receptor, whereas negative for cytokeratin marker. Ultrastructurally, complex interdigitating cell processes were connected by desmosomes. The diagnosis was "diffuse pulmonary meningotheliomatosis," which contained diffuse multiple "minute pulmonary meningothelial-like nodules." During 4 years of observation, interval changes in the size and distribution of the nodules have not been detected. It is necessary to consider this disease when we encounter multiple nodular shadows.

KEYWORDS

Minute pulmonary meningothelial-like nodule  Turner syndrome  Diffuse pulmonary meningotheliomatosis 

Received 4 Feb 2014 / Accepted 2 May 2014

AJRS, 3(5): 695-699, 2014

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