Pulmonary lymphangioleiomyomatosis with concomitant tuberous sclerosis complex diagnosed by video-assisted thoracoscopic surgery

Gen Thorac Cardiovasc Surg. 2008 Feb;56(2):81-4. doi: 10.1007/s11748-007-0194-7. Epub 2008 Feb 24.

Abstract

We herein describe the case of a 20-year-old woman who was diagnosed as having tuberous sclerosis complex (TSC) at the age of 10 years. The patient had a history of right pneumothorax at the age of 19. This time, a right pneumothorax recurrence was detected, and video-assisted thoracoscopic surgery (VATS) was performed. In the intraoperative findings, an infinitesimal lung cyst was detected on the lung lobe, and partial resection was performed. Pathologically, antibody-positive smooth muscle cells of the human melanoma block (HMB)-45 had grown and been diagnosed as pulmonary lymphangioleiomyomatosis (LAM). About half a year later, left pneumothorax occurred, and VATS was performed again. Pathologically, antibody-positive smooth muscle cells of HMB-45 were not detected. Occasionally, TSC is known to cause LAM complications, but in some cases it is difficult to make an accurate pathological diagnosis. Making a pathological diagnosis based on the VATS findings and taking a sufficient amount of tissue specimen are considered useful for making the diagnosis.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Antigens, Neoplasm
  • Antineoplastic Agents / therapeutic use
  • Female
  • Humans
  • Incidental Findings*
  • Lung Neoplasms / complications
  • Lung Neoplasms / diagnosis*
  • Lung Neoplasms / drug therapy
  • Lung Neoplasms / pathology
  • Lung Neoplasms / surgery
  • Lymphangioleiomyomatosis / complications
  • Lymphangioleiomyomatosis / diagnosis*
  • Lymphangioleiomyomatosis / drug therapy
  • Lymphangioleiomyomatosis / pathology
  • Lymphangioleiomyomatosis / surgery
  • Melanoma-Specific Antigens
  • Myocytes, Smooth Muscle / chemistry
  • Myocytes, Smooth Muscle / immunology
  • Myocytes, Smooth Muscle / pathology*
  • Neoplasm Proteins / analysis
  • Picibanil / therapeutic use
  • Pneumothorax / etiology
  • Pneumothorax / surgery*
  • Receptors, Estrogen / analysis
  • Receptors, Progesterone / analysis
  • Thoracic Surgery, Video-Assisted*
  • Treatment Outcome
  • Tuberous Sclerosis / complications*
  • Tuberous Sclerosis / surgery

Substances

  • Antigens, Neoplasm
  • Antineoplastic Agents
  • Melanoma-Specific Antigens
  • Neoplasm Proteins
  • Receptors, Estrogen
  • Receptors, Progesterone
  • Picibanil