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- Case Report -

Tuberous Sclerosis Complex Gene Abnormalities in 2 Patients with Lymphangioleiomyomatosis and Lung Cancer

Takahiro Sawada¹, Takuo Hayashi², Toshio Kumasaka³, Kenichi Takeuchi⁴, Haruto Hirano⁴, Hiroyuki Oura⁵, Masashi Handa⁵, Nobukazu Tomichi⁶, Sadahide Ono^6
1Department of General Thoracic Surgery, Ehime Prefectural Central Hospital, Japan, ²Department of Human Pathology, Juntendo University, Graduate School of Medicine, Japan, ³Pathological Department, Japanese Red Cross Medical Center, Japan, ⁴Department of Respiratory Internal Medicine, ⁵Department of General Thoracic Surgery, ⁶Department of Pathological Diagnosis Center, Iwate Prefectural Central Hospital, Japan

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Background. Lymphangioleiomyomatosis (LAM) is associated with abnormalities of the tuberous sclerosis complex (TSC) tumor suppressor gene, which is characterized by lymph vessel neogenesis. Futhermore, LAM concomitant with lung cancer is rare. Cases. Case 1: A tumor shadow was detected in the right upper lung field of a 64-year-old woman, with multiple cysts in bilateral lungs. Resected lung specimen tests indicated lung cancer (adenocarcinoma) in the right S¹ to S³ areas and secondary regional lymph node metastasis. Furthermore, multiple cystic lesions were considered to be LAM. In addition, multifocal micronodular pneumocyte hyperplasia (MMPH) was suspected. Gene analysis confirmed the presence of TSC1 gene abnormalities in the LAM and MMPH lesion sites. Case 2: Pulmonary tumor shadows were detected in the left S³ and S⁶ areas in a 73-year-old woman, with multiple cysts in bilateral lungs. Resected lung specimen tests indicated double lung cancer (both adenocarcinoma) and secondary regional lymph node metastasis. Furthermore, the multiple cystic lesions were considered to be LAM. Gene analysis showed TSC2 gene abnormalities in 1 lung cancer and the LAM lesion sites. However, their respective gene patterns differed. Conclusion. We report 2 rare cases in which lung cancer and LAM simultaneously occurred. However, the possibility of the involvement of TSC gene abnormalities in the pathogenesis of lung cancer was considered to be low.
key words: Lymphangioleiomyomatosis, Lung cancer, Lymphangiogenesis, Lymph node metastasis, TSC gene

Received: January 11, 2011
Accepted: March 2, 2011

JJLC 51 (3): 193-201, 2011