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Successful Sirolimus Treatment of Lymphangioleiomyomatosis in a Hepatitis B Virus Carrier.

Authors
  • Sonobe, Shoko1
  • Arai, Toru2
  • Tanimoto, Yasushi3
  • Sugimoto, Chikatoshi2
  • Kitaichi, Masanori4
  • Akira, Masanori5
  • Kasai, Takahiko6
  • Hirose, Masaki2
  • Inoue, Yoshikazu2
  • 1 Department of Internal Medicine, National Hospital Organization Kinki-Chuo Chest Medical Center, Japan. , (Japan)
  • 2 Clinical Research Center, National Hospital Organization Kinki-Chuo Chest Medical Center, Japan. , (Japan)
  • 3 Department of Allergy and Respiratory Medicine, National Hospital Organization Minami-Okayama Medical Center, Japan. , (Japan)
  • 4 Department of Pathology, National Hospital Organization Minami Wakayama Medical Center, Japan. , (Japan)
  • 5 Department of Radiology, National Hospital Organization Kinki-Chuo Chest Medical Center, Japan. , (Japan)
  • 6 Department of Pathology, National Hospital Organization Kinki-Chuo Chest Medical Center, Japan. , (Japan)
Type
Published Article
Journal
Internal medicine (Tokyo, Japan)
Publication Date
Feb 15, 2019
Volume
58
Issue
4
Pages
569–574
Identifiers
DOI: 10.2169/internalmedicine.1329-18
PMID: 30333386
Source
Medline
Keywords
Language
English
License
Unknown

Abstract

A 34-year-old woman experiencing shortness of breath was referred to our hospital. The patient was diagnosed with sporadic lymphangioleiomyomatosis based on the observation of bilateral diffuse multiple thin-walled cysts on computed tomography of the chest, chylous effusion, elevated serum vascular endothelial growth factor-D levels and transbronchial biopsy findings. This patient was a hepatitis B virus (HBV) carrier. Treatment with 1 mg daily of sirolimus was started after HBV DNA was brought below the cut-off level using entecavir. Sirolimus was effective, as the chylous effusion resolved completely and the dyspnea improved. The sirolimus dosage was increased to 2 mg daily without causing HBV reactivation.

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