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Cited by 26Transplantation: Methods and OutcomesTuberous Sclerosis Complex ResearchPulmonary Hypertension Research and Treatments

The DOI matches its Crossref record; 24 of the 24 references it cites were found in a registry or an index.

Mayo clinic experience of lung transplantation in pulmonary lymphangioleiomyomatosis

Open access
Ussavarungsi, Kamonpun, Hu, Xiaowen, Scott, J.P., Erasmus, David B., Mallea, Jorge M., Alvarez, Francisco, Lee, Augustine S., Keller, Cesar A., Ryu, Jay H., Burger, Charles D.·2015·Respiratory Medicine
DOI 10.1016/j.rmed.2015.08.014
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Abstract

Objectives Lymphangioleiomyomatosis (LAM) is a rare, cystic lung disease that generally results in progressive decline in lung function. Despite advancement of pharmacological therapy for LAM, lung transplantation remains an important option for women with end-stage LAM.Methods Patients with LAM undergoing lung transplantation at the Mayo Clinic campuses in Rochester, Minnesota and Jacksonville, Florida since 1995 were retrospectively reviewed.Results Overall, 12 women underwent lung transplantation. Nine of 12 (75%) underwent double lung transplant. The mean age was 42 ± 8 years at the time of transplant. One patient (8%) had a chylothorax and 7 (58%) had recurrent pneumothoraces, 4 (33%) of which required pleurodesis. All had diffuse, cystic lung disease on chest CT consistent with LAM which was confirmed in the explant of all patients. The average length of ICU and hospital stays were 5 ± 4 and 19 ± 19 days, respectively. Mild to moderate anastomotic ischemia was evident in all patients but resolved with time. No patient was treated with sirolimus pre-transplant. Seven patients received sirolimus post-transplant; however, clinical benefit was documented in only 2 patients, 1 of which was treated for large retroperitoneal cysts with ureteral obstruction and another with persistent chylothorax and retroperitoneal lymphangioleimyomas. Five patients are deceased. The median survival by Kaplan-Meier analysis was 119 months with a median follow-up of 68 months (range 2-225 months).Conclusions Lung transplant remains a viable treatment for patients with end-stage LAM. The role of sirolimus peri-transplantation remains ill-defined.

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Crossref · 10.1016/j.rmed.2015.08.014

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  • Lung transplant for lymphangioleiomyomatosis may be surgically challenging because of a high prevalence of adhesions from prior management of pleural complications.

    “Lung transplant for LAM may be challenging surgically due to the high prevalence of adhesions resulting from the management of pleural complications prior to transplant.(309, 312–314, 319)”

    From Consensus document for the selection of lung transplant candidates: An update from the International Society for Heart and Lung Transplantation. (2021) · cites this paper as [313]

    The Discussion (page unavailable) says prior pleurodesis can create challenges for lung transplantation in LAM, and the Results describe significant intraoperative bleeding in one patient on the side of prior pleurodesis. Our copy does not state that adhesions are highly prevalent or identify them as the cause of those surgical challenges. (LLM, conf=0.84)

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  • Fewer patients with lymphangioleiomyomatosis have required lung transplant since standardized use of mTOR inhibitors.

    “Fewer patients with LAM have required lung transplant since the standardized use of mTOR inhibitors as treatment, but it remains an indication for those with severely abnormal lung function (FEV1 < 30%), exertional dyspnea (NYHA class III or IV), or hypoxemia at rest.(303–306, 308–315)”

    From Consensus document for the selection of lung transplant candidates: An update from the International Society for Heart and Lung Transplantation. (2021) · cites this paper as [313]

    The Discussion (page unavailable) reports that sirolimus can slow lung-function decline and that transplantation remains an option when medical treatment does not work. It does not report that fewer patients have required transplantation since mTOR inhibitors became standard treatment. Nor does it give the cited FEV1, NYHA, or resting-hypoxemia thresholds as transplant indications. (LLM, conf=0.84)

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Research works printed but not linked: 1

  1. Erasmus, 2014, Treatment of intra-abdominal lymphangioleiomyomas with sirolimus in a lung transplant recipient with lymphangioleiomyomatosis (LAM), South Afr. Respir. J., 18, 17

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Metadata

Authors
Ussavarungsi, K., Hu, X., Scott, J. P., Erasmus, D. B., Mallea, J. M., Alvarez, F., Lee, A. S., Keller, C. A., Ryu, J. H., & Burger, C. D.
Affiliations
Mayo Clinic (US)
Corresponding author
  • Ussavarungsi, Kamonpun
  • Hu, Xiaowen
  • Scott, J.P.
  • Erasmus, David B.
  • Mallea, Jorge M.
  • Alvarez, Francisco
  • Lee, Augustine S.
  • Keller, Cesar A.
  • Ryu, Jay H.
  • Burger, Charles D.
ORCID
Venue
Respiratory Medicine
Publisher
Elsevier BV
Published
2015-10
Volume
109
Issue
10
Pages
1354-1359
ISSN
0954-6111 (ISSN-L)
DOI
10.1016/j.rmed.2015.08.014
OpenAlex
W2142923206
Semantic Scholar
cd6b761fb02d63424ea1e4aefc1f9258216df97f
Cited by
26
References
24
Citations by year
2026: 1 · 2025: 1 · 2024: 1 · 2023: 1 · 2022: 4 · 2021: 3 · 2020: 2 · 2019: 7 · 2018: 2 · 2017: 3 · 2015: 1
FWCI
2.01
Type
journal article
Language
en
Open access
Licence
Locations
  • Respiratory Medicine (publishedVersion)
  • PubMed (publishedVersion)
Topics
  • Health Sciences › Medicine › Surgery › Transplantation: Methods and Outcomes
  • Health Sciences › Medicine › Genetics (clinical) › Tuberous Sclerosis Complex Research
  • Health Sciences › Medicine › Pulmonary and Respiratory Medicine › Pulmonary Hypertension Research and Treatments
Keywords
pulmonary lymphangioleiomyomatosis, lung transplantation, end-stage lung disease, rapamycin, chylothorax, recurrent pneumothorax, pleurodesis, anastomotic ischemia, survival, women
MeSH terms
Adult, Echocardiography, Female, Humans, Immunosuppressive Agents, Lung Neoplasms, Middle Aged, Retrospective Studies, Severity of Illness Index, Tomography, X-Ray Computed, Lung Transplantation, Treatment Outcome, Lymphangioleiomyomatosis, Pleurodesis, Sirolimus

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