Lymphangioleiomyomatosis, multifocal micronodular pneumocyte hyperplasia, and sarcoidosis: more pathological findings in the same chest CT, or a single pathological pathway?

BMC Pulm Med. 2017 Jul 28;17(1):107. doi: 10.1186/s12890-017-0447-x.

Abstract

Background: Autoimmune hepatitis/primary biliary cirrhosis overlap syndrome, lymphangioleiomyomatosis/tuberous sclerosis complex (LAM-TSC), and sarcoidosis are three rare diseases. Here we present, to the best of our knowledge, the first description of a patient with the coexistence of these three diseases.

Case presentation: A 47-year-old woman affected by LAM-TSC and primary biliary cirrosis/autoimmune hepatitis overlap syndrome. During her follow up a high resolution chest CT scan (HRTC) confirmed the presence of both multiple cysts and micronodular opacities consistent with multifocal micronodular pneumocytes hyperlasia (MMPH), and revealed multiple hilar-mediastinal symmetrical lymphadenopathies suggestive of sarcoidosis. Simultaneously, subcutaneous nodules appeared on her forearm bilaterally. Cutaneous biopsy showed granulomatous dermatitis with sarcoid-like granulomas. A diagnosis of stage I pulmonary sarcoidosis was made. No treatment for sarcoidosis was initiated since the patient had neither systemic involvement, nor respiratory impairment.

Conclusions: The presence of more than one rare disease should challenge the concept of a potential common underlying mechanism, since the a priori probability of the concomitant presence of different conditions with different pathogenic mechanisms - especially if rare diseases - is low. We speculate that the dysregulation of the pathway involving mTOR and MAPK and their interaction might play a role in the pathogenesis of other diseases, including sarcoidosis.

Keywords: LAM; Sarcoidosis; TSC; Thoracic images.

Publication types

  • Case Reports

MeSH terms

  • Alveolar Epithelial Cells / pathology
  • Female
  • Hepatitis, Autoimmune / complications*
  • Humans
  • Hyperplasia
  • Liver Cirrhosis, Biliary / complications*
  • Lung / diagnostic imaging
  • Lung / pathology
  • Lung Neoplasms / complications*
  • Lung Neoplasms / diagnostic imaging
  • Lymphangioleiomyomatosis / complications*
  • Lymphangioleiomyomatosis / diagnostic imaging
  • Middle Aged
  • Sarcoidosis, Pulmonary / complications*
  • Sarcoidosis, Pulmonary / diagnostic imaging
  • Tomography, X-Ray Computed
  • Tuberous Sclerosis / complications*
  • Tuberous Sclerosis / diagnostic imaging