Assessment of pulmonary outcomes, exercise capacity, and longitudinal changes in lung function in pediatric survivors of high-risk neuroblastoma Journal Article


Authors: Stone, A.; Friedman, D. N.; Kushner, B. H.; Wolden, S.; Modak, S.; LaQuaglia, M. P.; Costello, J.; Wu, X.; Cheung, N. K.; Sklar, C. A.
Article Title: Assessment of pulmonary outcomes, exercise capacity, and longitudinal changes in lung function in pediatric survivors of high-risk neuroblastoma
Abstract: Background/objectives: Survivors of high-risk neuroblastoma (NB) are exposed to multimodality therapies early in life and confront late therapy-related toxicities. This study assessed respiratory symptoms, exercise capacity, and longitudinal changes in pulmonary function tests (PFTs) among survivors. Design/methods: Survivors of high-risk NB followed in the long-term follow-up clinic at Memorial Sloan Kettering Cancer Center were enrolled. Symptom and physical activity questionnaires were completed. Medical records were reviewed for treatments and comorbidities. Participants completed spirometry, plethysmography, diffusion capacity of the lung for carbon monoxide, 6-minute walk tests (6MWTs), and cardiopulmonary exercise testing. Questionnaires and PFTs were repeated at least one year after enrollment. Results: Sixty-two survivors participated (median age at study: 10.92 years; median age at diagnosis: 2.75 years; median time since completion of therapy: 5.29 years). Thirty-two percent had chronic respiratory symptoms. Seventy-seven percent had PFT abnormalities, mostly mild to moderate severity. Thirty-three completed 6MWTs (median, 634.3 meters); eight completed cardiopulmonary exercise tests (mean VO2 max: 63% predicted); 23 completed a second PFT revealing declines over a median 2.97 years (mean percent predicted forced vital capacity: 79.9 to 70.0; mean forced expiratory volume in 1 second: 81.6 to 69.9). Risks for abnormalities included thoracic surgery, chest radiation therapy (RT), thoracic surgery plus chest RT, and hematopoietic stem cell transplant. Conclusions: In this cohort of survivors of high-risk NB, PFT abnormalities were common but mostly mild or moderate. Maximal exercise capacity may be affected by respiratory limitations and declines in lung function may occur over time. Continued pulmonary surveillance of this at-risk population is warranted. © 2019 Wiley Periodicals, Inc.
Keywords: adolescent; adult; child; cancer surgery; major clinical study; busulfan; fludarabine; lenalidomide; thalidomide; cisplatin; doxorubicin; cancer combination chemotherapy; multimodality cancer therapy; cancer patient; cancer radiotherapy; cytarabine; temozolomide; topotecan; cancer staging; outcome assessment; follow up; carboplatin; etoposide; maintenance therapy; risk factors; exercise; cyclophosphamide; melphalan; vincristine; practice guideline; hematopoietic stem cell transplantation; prediction; ifosfamide; thiotepa; high risk patient; cancer survivor; survivors; irinotecan; coughing; dyspnea; pneumonia; disease severity; neuroblastoma; celecoxib; eflornithine; laboratory; medical record; comorbidity; long term care; asparaginase; thorax radiography; asthma; isotretinoin; lung function; beta glucan; radioimmunotherapy; forced expiratory volume; cardiopulmonary exercise test; oxygen consumption; lung fibrosis; electrocardiogram; rapamycin; environmental exposure; perifosine; oxygen supply; longitudinal study; plethysmography; spirometry; wheezing; lung diffusion capacity; forced vital capacity; crizotinib; respiratory symptoms; pulmonary function tests; exercise capacity; human; male; female; priority journal; article; total lung capacity; lung gas exchange; six minute walk test
Journal Title: Pediatric Blood and Cancer
Volume: 66
Issue: 11
ISSN: 1545-5009
Publisher: Wiley Periodicals, Inc  
Date Published: 2019-11-01
Start Page: e27960
Language: English
DOI: 10.1002/pbc.27960
PUBMED: 31407504
PROVIDER: scopus
PMCID: PMC6927011
DOI/URL:
Notes: Article -- Export Date: 1 November 2019 -- Source: Scopus
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MSK Authors
  1. Brian Kushner
    230 Kushner
  2. Nai-Kong Cheung
    531 Cheung
  3. Charles A Sklar
    298 Sklar
  4. Shakeel Modak
    185 Modak
  5. Suzanne L Wolden
    453 Wolden