Skip to main content
Top
Published in: BMC Pulmonary Medicine 1/2021

Open Access 01-12-2021 | Computed Tomography | Research article

The value of transbronchial lung biopsy in the diagnosis of lymphangioleiomyomatosis

Authors: Wenshuai Xu, Han Cui, Hongrui Liu, Ruie Feng, Xinlun Tian, Yanli Yang, Kai-Feng Xu

Published in: BMC Pulmonary Medicine | Issue 1/2021

Login to get access

Abstract

Background

Transbronchial lung biopsy (TBLB) in the diagnosis of lymphangioleiomyomatosis (LAM) is not a common approach, although TBLB is often performed in diffuse lung diseases. We aimed to examine the diagnostic value and safety of TBLB in LAM patients based on the data collected in our center.

Methods

We reviewed LAM patients registered in our LAM Clinic from December 8, 2006, to December 31, 2019. All patients with definite or probable diagnosis of LAM who had been examined using TBLB were included. All available pathology slides were reviewed by an experienced LAM pathologist. All complications were reviewed by the medical records and confirmed using telephone interviews.

Results

The pathology results of 86 patients (including 74 definite LAM and 12 probable LAM) were available. The positive rate of TBLB in LAM patients was 49/86 (57.0%). The positive rates of SMA, HMB-45, ER, and PR in LAM patients were 97.6%, 93%, 84.6%, and 78.4% respectively. The positive rate of TBLB was 40%, 60% and 60.8% in patients with CT Grade I, Grade II, and Grade III respectively, and the difference was not significant. Patients who had 3–4 or 5–6 biopsied specimens had a higher rate of diagnosis than those with 1–2 biopsied specimens. Four patients (5.6%) reported pneumothorax. No major hemoptysis was reported.

Conclusions

TBLB is a feasible and safe procedure for obtaining a pathological diagnosis of LAM. Taking more than 2 samples during the biopsy procedure increased the rate of diagnosis.
Literature
1.
go back to reference Johnson SR. Lymphangioleiomyomatosis. Eur Respir J. 2006;27(5):1056–65. Johnson SR. Lymphangioleiomyomatosis. Eur Respir J. 2006;27(5):1056–65.
2.
go back to reference Johnson SR, Cordier JF, Lazor R, Cottin V, Costabel U, Harari S, et al. European Respiratory Society guidelines for the diagnosis and management of lymphangioleiomyomatosis. Eur Respir J. 2010;35(1):14–26. Johnson SR, Cordier JF, Lazor R, Cottin V, Costabel U, Harari S, et al. European Respiratory Society guidelines for the diagnosis and management of lymphangioleiomyomatosis. Eur Respir J. 2010;35(1):14–26.
3.
go back to reference Gupta N. Lymphangioleiomyomatosis diagnosis and management: high-resolution chest computed tomography, transbronchial lung biopsy, and pleural disease management. Offi Am Thorac Soc. 2017;196(10):1337–48. Gupta N. Lymphangioleiomyomatosis diagnosis and management: high-resolution chest computed tomography, transbronchial lung biopsy, and pleural disease management. Offi Am Thorac Soc. 2017;196(10):1337–48.
4.
go back to reference Koo HK, Yoo CG. Multiple cystic lung disease. Tubercu Respir Dis. 2013;74(3):97–103. Koo HK, Yoo CG. Multiple cystic lung disease. Tubercu Respir Dis. 2013;74(3):97–103.
5.
go back to reference Koba T, Arai T, Kitaichi M, Kasai T, Hirose M, Tachibana K, Sugimoto C, Akira M, Hayashi S, Inoue Y. Efficacy and safety of transbronchial lung biopsy for the diagnosis of lymphangioleiomyomatosis: a report of 24 consecutive patients. Respirology. 2018;23(3):331–8. Koba T, Arai T, Kitaichi M, Kasai T, Hirose M, Tachibana K, Sugimoto C, Akira M, Hayashi S, Inoue Y. Efficacy and safety of transbronchial lung biopsy for the diagnosis of lymphangioleiomyomatosis: a report of 24 consecutive patients. Respirology. 2018;23(3):331–8.
6.
go back to reference Okamoto S, Suzuki K, Hayashi T, Muraki K, Nagaoka T, Nishino K, et al. Transbronchial lung biopsy for the diagnosis of lymphangioleiomyomatosis: the severity of cystic lung destruction assessed by the modified Goddard scoring system as a predictor for establishing the diagnosis. Orphanet J Rare Dis. 2020;15(1):125. Okamoto S, Suzuki K, Hayashi T, Muraki K, Nagaoka T, Nishino K, et al. Transbronchial lung biopsy for the diagnosis of lymphangioleiomyomatosis: the severity of cystic lung destruction assessed by the modified Goddard scoring system as a predictor for establishing the diagnosis. Orphanet J Rare Dis. 2020;15(1):125.
7.
go back to reference Avila NA, Chen CC, Chu SC, Wu M, Jones EC, Neumann RD, et al. Pulmonary lymphangioleiomyomatosis: correlation of ventilation-perfusion scintigraphy, chest radiography, and CT with pulmonary function tests. Radiology. 2000;214(2):441–6. Avila NA, Chen CC, Chu SC, Wu M, Jones EC, Neumann RD, et al. Pulmonary lymphangioleiomyomatosis: correlation of ventilation-perfusion scintigraphy, chest radiography, and CT with pulmonary function tests. Radiology. 2000;214(2):441–6.
8.
go back to reference Díaz Pedreira JA, González-Carreró J, Cameselle Veloso R, Barros Tizón JC, Bandrés Gimeno R. [Pulmonary lymphangioleiomyomatosis. Presentation of a new case]. Anales de medicina interna (Madrid, Spain: 1984). 1991;8 (12):614–6. Díaz Pedreira JA, González-Carreró J, Cameselle Veloso R, Barros Tizón JC, Bandrés Gimeno R. [Pulmonary lymphangioleiomyomatosis. Presentation of a new case]. Anales de medicina interna (Madrid, Spain: 1984). 1991;8 (12):614–6.
9.
go back to reference Bonetti F, Chiodera PL, Pea M, Martignoni G, Bosi F, Zamboni G, et al. Transbronchial biopsy in lymphangiomyomatosis of the lung. HMB45 for diagnosis. Am J Surg Pathol. 1993;17(11):1092–102. Bonetti F, Chiodera PL, Pea M, Martignoni G, Bosi F, Zamboni G, et al. Transbronchial biopsy in lymphangiomyomatosis of the lung. HMB45 for diagnosis. Am J Surg Pathol. 1993;17(11):1092–102.
10.
go back to reference Harari S, Torre O, Cassandro R, Taveira-DaSilva AM, Moss J. Bronchoscopic diagnosis of Langerhans cell histiocytosis and lymphangioleiomyomatosis. Respir Med. 2012;106(9):1286–92. Harari S, Torre O, Cassandro R, Taveira-DaSilva AM, Moss J. Bronchoscopic diagnosis of Langerhans cell histiocytosis and lymphangioleiomyomatosis. Respir Med. 2012;106(9):1286–92.
11.
go back to reference Meraj R, Wikenheiser-Brokamp KA, Young LR, Byrnes S, McCormack FX. Utility of transbronchial biopsy in the diagnosis of lymphangioleiomyomatosis. Front Med. 2012;6(4):395–405. Meraj R, Wikenheiser-Brokamp KA, Young LR, Byrnes S, McCormack FX. Utility of transbronchial biopsy in the diagnosis of lymphangioleiomyomatosis. Front Med. 2012;6(4):395–405.
12.
go back to reference Meraj R, Wikenheiser-Brokamp KA, Young LR, Byrnes S, McCormack FX. Utility of transbronchial biopsy in the diagnosis of lymphangioleiomyomatosis. Respirology (Carlton, Vic). 2012;6(4):395–405. Meraj R, Wikenheiser-Brokamp KA, Young LR, Byrnes S, McCormack FX. Utility of transbronchial biopsy in the diagnosis of lymphangioleiomyomatosis. Respirology (Carlton, Vic). 2012;6(4):395–405.
13.
go back to reference Ferrans VJ, Yu ZX, Nelson WK, Valencia JC, Tatsuguchi A, Avila NA, et al. Lymphangioleiomyomatosis (LAM): a review of clinical and morphological features. J Nippon Med Sch. 2000;67(5):311–29. Ferrans VJ, Yu ZX, Nelson WK, Valencia JC, Tatsuguchi A, Avila NA, et al. Lymphangioleiomyomatosis (LAM): a review of clinical and morphological features. J Nippon Med Sch. 2000;67(5):311–29.
14.
go back to reference Gao L, Yue MM, Davis J, Hyjek E, Schuger L. In pulmonary lymphangioleiomyomatosis expression of progesterone receptor is frequently higher than that of estrogen receptor. Virchows Arch. 2014;464(4):495–503. Gao L, Yue MM, Davis J, Hyjek E, Schuger L. In pulmonary lymphangioleiomyomatosis expression of progesterone receptor is frequently higher than that of estrogen receptor. Virchows Arch. 2014;464(4):495–503.
15.
go back to reference de Fenoyl O, Capron F, Lebeau B, Rochemaure J. Transbronchial biopsy without fluoroscopy: a five year experience in outpatients. Thorax. 1989;44(11):956–9. de Fenoyl O, Capron F, Lebeau B, Rochemaure J. Transbronchial biopsy without fluoroscopy: a five year experience in outpatients. Thorax. 1989;44(11):956–9.
16.
go back to reference Babiak A, Hetzel J, Krishna G, Fritz P, Moeller P, Balli T, et al. Transbronchial cryobiopsy: a new tool for lung biopsies. Respiration. 2009;78(2):203–8. Babiak A, Hetzel J, Krishna G, Fritz P, Moeller P, Balli T, et al. Transbronchial cryobiopsy: a new tool for lung biopsies. Respiration. 2009;78(2):203–8.
17.
go back to reference Ravaglia C, Bonifazi M, Wells AU, Tomassetti S, Gurioli C, Piciucchi S, et al. Safety and diagnostic yield of transbronchial lung cryobiopsy in diffuse parenchymal lung diseases: a comparative study versus video-assisted thoracoscopic lung biopsy and a systematic review of the literature. Respiration. 2016;91(3):215–27. Ravaglia C, Bonifazi M, Wells AU, Tomassetti S, Gurioli C, Piciucchi S, et al. Safety and diagnostic yield of transbronchial lung cryobiopsy in diffuse parenchymal lung diseases: a comparative study versus video-assisted thoracoscopic lung biopsy and a systematic review of the literature. Respiration. 2016;91(3):215–27.
18.
go back to reference Johannson KA, Marcoux VS, Ronksley PE, Ryerson CJ. Diagnostic yield and complications of transbronchial lung cryobiopsy for interstitial lung disease. A systematic review and metaanalysis. Ann Am Thorac Soc. 2016;13(10):1828–38. Johannson KA, Marcoux VS, Ronksley PE, Ryerson CJ. Diagnostic yield and complications of transbronchial lung cryobiopsy for interstitial lung disease. A systematic review and metaanalysis. Ann Am Thorac Soc. 2016;13(10):1828–38.
19.
go back to reference Fruchter O, Fridel L, El Raouf BA, Abdel-Rahman N, Rosengarten D, Kramer MR. Histological diagnosis of interstitial lung diseases by cryo-transbronchial biopsy. Respirology (Carlton, Vic). 2014;19(5):683–8. Fruchter O, Fridel L, El Raouf BA, Abdel-Rahman N, Rosengarten D, Kramer MR. Histological diagnosis of interstitial lung diseases by cryo-transbronchial biopsy. Respirology (Carlton, Vic). 2014;19(5):683–8.
20.
go back to reference Casoni GL, Tomassetti S, Cavazza A, Colby TV, Dubini A, Ryu JH, et al. Transbronchial lung cryobiopsy in the diagnosis of fibrotic interstitial lung diseases. PLoS ONE. 2014;9(2):e86716. Casoni GL, Tomassetti S, Cavazza A, Colby TV, Dubini A, Ryu JH, et al. Transbronchial lung cryobiopsy in the diagnosis of fibrotic interstitial lung diseases. PLoS ONE. 2014;9(2):e86716.
Metadata
Title
The value of transbronchial lung biopsy in the diagnosis of lymphangioleiomyomatosis
Authors
Wenshuai Xu
Han Cui
Hongrui Liu
Ruie Feng
Xinlun Tian
Yanli Yang
Kai-Feng Xu
Publication date
01-12-2021
Publisher
BioMed Central
Published in
BMC Pulmonary Medicine / Issue 1/2021
Electronic ISSN: 1471-2466
DOI
https://doi.org/10.1186/s12890-021-01518-2

Other articles of this Issue 1/2021

BMC Pulmonary Medicine 1/2021 Go to the issue
Live Webinar | 27-06-2024 | 18:00 (CEST)

Keynote webinar | Spotlight on medication adherence

Live: Thursday 27th June 2024, 18:00-19:30 (CEST)

WHO estimates that half of all patients worldwide are non-adherent to their prescribed medication. The consequences of poor adherence can be catastrophic, on both the individual and population level.

Join our expert panel to discover why you need to understand the drivers of non-adherence in your patients, and how you can optimize medication adherence in your clinics to drastically improve patient outcomes.

Prof. Kevin Dolgin
Prof. Florian Limbourg
Prof. Anoop Chauhan
Developed by: Springer Medicine
Obesity Clinical Trial Summary

At a glance: The STEP trials

A round-up of the STEP phase 3 clinical trials evaluating semaglutide for weight loss in people with overweight or obesity.

Developed by: Springer Medicine

Highlights from the ACC 2024 Congress

Year in Review: Pediatric cardiology

Watch Dr. Anne Marie Valente present the last year's highlights in pediatric and congenital heart disease in the official ACC.24 Year in Review session.

Year in Review: Pulmonary vascular disease

The last year's highlights in pulmonary vascular disease are presented by Dr. Jane Leopold in this official video from ACC.24.

Year in Review: Valvular heart disease

Watch Prof. William Zoghbi present the last year's highlights in valvular heart disease from the official ACC.24 Year in Review session.

Year in Review: Heart failure and cardiomyopathies

Watch this official video from ACC.24. Dr. Biykem Bozkurt discusses last year's major advances in heart failure and cardiomyopathies.