Lymphangioleiomyomatosis Diagnosed by Immunocytochemical and Genetic Analysis of Lymphangioleiomyomatosis Cell Clusters Found in Chylous Pleural Effusion

Michihiro Hirama; Ryo Atsuta; Keiko Mitani; Toshio Kumasaka; Yoko Gunji; Shin-ichi Sasaki; Akihiko Iwase; Kazuhisa Takahashi; Kuniaki Seyama

doi:10.2169/internalmedicine.46.0225

CASE REPORTS

Lymphangioleiomyomatosis Diagnosed by Immunocytochemical and Genetic Analysis of Lymphangioleiomyomatosis Cell Clusters Found in Chylous Pleural Effusion

Michihiro Hirama, Ryo Atsuta, Keiko Mitani, Toshio Kumasaka, Yoko Gunji, Shin-ichi Sasaki, Akihiko Iwase, Kazuhisa Takahashi, Kuniaki Seyama

Author information

Michihiro Hirama
Department of Respiratory Medicine, Juntendo University School of Medicine
Ryo Atsuta
Department of Respiratory Medicine, Juntendo University School of Medicine
Keiko Mitani
Department of Human Pathology, Juntendo University School of Medicine
Toshio Kumasaka
Department of Human Pathology, Juntendo University School of Medicine
Yoko Gunji
Department of Respiratory Medicine, Juntendo University School of Medicine
Shin-ichi Sasaki
Department of Respiratory Medicine, Juntendo University Urayasu Hospital
Akihiko Iwase
Department of Respiratory Medicine, Tokyo Metropolitan Koto Geriatric Medical Center
Kazuhisa Takahashi
Department of Respiratory Medicine, Juntendo University School of Medicine
Kuniaki Seyama
Department of Respiratory Medicine, Juntendo University School of Medicine

Keywords: lymphangiogenesis, tumor suppressor gene, pleurodesis, chyle

JOURNAL OPEN ACCESS

2007 Volume 46 Issue 18 Pages 1593-1596

DOI https://doi.org/10.2169/internalmedicine.46.0225

[Correction] Intern. Med. Vol.47 (2008) No.3 pp.192-192

Details

Abstract

A 37-year-old woman presented with a cough and discomfort in the chest. Computed tomography revealed the right pleural effusion and a number of cysts in the lungs. Thoracentesis revealed LAM cell clusters (LCC) in chylous pleural effusion, confirmed by immunocytochemical examinations showing that the cells at the center of cluster were LAM cells positive for α-smooth muscle actin and HMB45 and the outer layer was lymphatic endothelium cells. When LCC were cultured in vitro, the loss of heterozygosity of TSC2 markers was detected. This case illustrates that LAM can be diagnosed by the identification of LCC without an invasive biopsy if complicated with chylous effusion.

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