Literature DB >> 27108015

Reductions in pulmonary function detected in patients with lymphangioleiomyomatosis: An analysis of the Japanese National Research Project on Intractable Diseases database.

Mie Hayashida1, Masanori Yasuo2, Masayuki Hanaoka3, Kuniaki Seyama4, Yoshikazu Inoue5, Koichiro Tatsumi6, Michiaki Mishima7.   

Abstract

BACKGROUND: In lymphangioleiomyomatosis (LAM), predicting lung disease progression is essential for treatment planning. However, no previous Japanese studies have attempted to predict the reductions in pulmonary function that occur in LAM patients.
METHODS: The data for 89 LAM patients who had undergone ≥3 spirometry tests and whose data had been registered in the Japanese National Research Project on Intractable Diseases database between October 2009 and March 2014 were analyzed after excluding patients who had undergone (1) a lung transplant; (2) mTOR inhibitor treatment; or (3) thoracic drainage, pleurodesis, surgery, or thoracic duct ligation during the study period. The rates of change (slope) in pulmonary parameters were calculated, and their associations with clinical background factors were investigated.
RESULTS: Among the whole study population, the median (quartiles) slope of forced expiratory volume in one second (FEV1) was -46.7 (-95.2; -15.0)mL per year. Episodes of conservatively treated pneumothorax during the study period were found to be associated with rapid reductions in FEV1 (% predicted). Pregnancy during the study period was associated with a reduction in FEV1 (% predicted). When the patients were divided into those who exhibited initial FEV1 (% predicted) values of >70% (Group A) and ≤70% (Group B), Group B displayed significantly faster reductions in FEV1 (% predicted) than Group A.
CONCLUSIONS: LAM patients whose initial FEV1 (% predicted) values are ≤70% subsequently exhibit rapid reductions in their FEV1 values, and hence, require treatment. However, the FEV1 reduction rate varies markedly among individuals and should be monitored in all cases.
Copyright © 2015 The Japanese Respiratory Society. Published by Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Database; Disease progression; Lymphangioleiomyomatosis; Pulmonary function; Rare lung disease

Mesh:

Year:  2015        PMID: 27108015     DOI: 10.1016/j.resinv.2015.11.003

Source DB:  PubMed          Journal:  Respir Investig        ISSN: 2212-5345


  8 in total

1.  Analysis of the MILES cohort reveals determinants of disease progression and treatment response in lymphangioleiomyomatosis.

Authors:  Nishant Gupta; Hye-Seung Lee; Lisa R Young; Charlie Strange; Joel Moss; Lianne G Singer; Koh Nakata; Alan F Barker; Jeffrey T Chapman; Mark L Brantly; James M Stocks; Kevin K Brown; Joseph P Lynch; Hilary J Goldberg; Gregory P Downey; Angelo M Taveira-DaSilva; Jeffrey P Krischer; Kenneth Setchell; Bruce C Trapnell; Yoshikazu Inoue; Francis X McCormack
Journal:  Eur Respir J       Date:  2019-04-04       Impact factor: 16.671

Review 2.  Pulmonary manifestations in tuberous sclerosis complex.

Authors:  Nishant Gupta; Elizabeth P Henske
Journal:  Am J Med Genet C Semin Med Genet       Date:  2018-07-28       Impact factor: 3.908

3.  The NHLBI LAM Registry: Prognostic Physiologic and Radiologic Biomarkers Emerge From a 15-Year Prospective Longitudinal Analysis.

Authors:  Nishant Gupta; Hye-Seung Lee; Jay H Ryu; Angelo M Taveira-DaSilva; Gerald J Beck; Jar-Chi Lee; Kevin McCarthy; Geraldine A Finlay; Kevin K Brown; Stephen J Ruoss; Nilo A Avila; Joel Moss; Francis X McCormack
Journal:  Chest       Date:  2018-06-22       Impact factor: 9.410

4.  Long-term clinical course and progression of lymphangioleiomyomatosis in a single lung transplant referral centre in Korea.

Authors:  Shihwan Chang; Ji Soo Choi; Ah Young Leem; Su Hwan Lee; Sang Hoon Lee; Song Yee Kim; Kyung Soo Chung; Ji Ye Jung; Young Ae Kang; Young Sam Kim; Jin Gu Lee; Hyo Chae Paik; Hyo Sup Shim; Eun Hye Lee; Moo Suk Park
Journal:  Sci Rep       Date:  2022-05-18       Impact factor: 4.996

5.  Pregnancy after the diagnosis of lymphangioleiomyomatosis (LAM).

Authors:  Lisha Shen; Whenshuai Xu; Jinsong Gao; Jun Wang; Jiannan Huang; Yani Wang; Yudi He; Yanli Yang; Xinlun Tian; Kai-Feng Xu
Journal:  Orphanet J Rare Dis       Date:  2021-03-17       Impact factor: 4.123

Review 6.  Lymphangioleiomyomatosis: a clinical review.

Authors:  Anne M O'Mahony; Evelyn Lynn; David J Murphy; Aurelie Fabre; Cormac McCarthy
Journal:  Breathe (Sheff)       Date:  2020-06

7.  Rates of change in FEV1 and DLCO as potential indicators for mTOR inhibitor therapy in premenopausal lymphangioleiomyomatosis patients.

Authors:  Angelo M Taveira-DaSilva; Patricia Julien-Williams; Amanda M Jones; Mario Stylianou; Joel Moss
Journal:  Eur Respir J       Date:  2018-04-19       Impact factor: 16.671

Review 8.  Lung Diseases Unique to Women.

Authors:  Rachel N Criner; Abdullah Al-Abcha; Allison A Lambert; MeiLan K Han
Journal:  Clin Chest Med       Date:  2021-09       Impact factor: 4.967

  8 in total

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