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Subsequent neoplasms and late mortality in children undergoing allogeneic transplantation for nonmalignant diseases

Authors :
Kahn, Justine M.
Brazauskas, Ruta
Tecca, Heather R.
Bo-Subait, Stephanie
Buchbinder, David
Battiwala, Minoo
Flowers, Mary E. D.
Savani, Bipin N.
Phelan, Rachel
Broglie, Larisa
Abraham, Allistair A.
Keating, Amy K.
Daly, Andrew
Wirk, Baldeep
George, Biju
Alter, Blanche P.
Ustun, Celalettin
Freytes, Cesar O.
Beitinjaneh, Amer M.
Duncan, Christine
Copelan, Edward
Hildebrandt, Gerhard C.
Murthy, Hemant S.
Lazarus, Hillard M.
Auletta, Jeffery J.
Myers, Kasiani C.
Williams, Kirsten M.
Page, Kristin M.
Vrooman, Lynda M.
Norkin, Maxim
Byrne, Michael
Diaz, Miguel Angel
Kamani, Naynesh
Bhatt, Neel S.
Rezvani, Andrew
Farhadfar, Nosha
Mehta, Parinda A.
Hematti, Peiman
Shaw, Peter J.
Kamble, Rammurti T.
Schears, Raquel
Olsson, Richard
Hayashi, Robert J.
Gale, Robert Peter
Mayo, Samantha J.
Chhabra, Saurabh
Rotz, Seth J.
Badawy, Sherif M.
Ganguly, Siddhartha
Pavletic, Steven
Nishihori, Taiga
Prestidge, Tim
Agrawal, Vaibhav
Hogan, William J.
Inamoto, Yoshihiro
Shaw, Bronwen E.
Satwani, Prakash
Kahn, Justine M.
Brazauskas, Ruta
Tecca, Heather R.
Bo-Subait, Stephanie
Buchbinder, David
Battiwala, Minoo
Flowers, Mary E. D.
Savani, Bipin N.
Phelan, Rachel
Broglie, Larisa
Abraham, Allistair A.
Keating, Amy K.
Daly, Andrew
Wirk, Baldeep
George, Biju
Alter, Blanche P.
Ustun, Celalettin
Freytes, Cesar O.
Beitinjaneh, Amer M.
Duncan, Christine
Copelan, Edward
Hildebrandt, Gerhard C.
Murthy, Hemant S.
Lazarus, Hillard M.
Auletta, Jeffery J.
Myers, Kasiani C.
Williams, Kirsten M.
Page, Kristin M.
Vrooman, Lynda M.
Norkin, Maxim
Byrne, Michael
Diaz, Miguel Angel
Kamani, Naynesh
Bhatt, Neel S.
Rezvani, Andrew
Farhadfar, Nosha
Mehta, Parinda A.
Hematti, Peiman
Shaw, Peter J.
Kamble, Rammurti T.
Schears, Raquel
Olsson, Richard
Hayashi, Robert J.
Gale, Robert Peter
Mayo, Samantha J.
Chhabra, Saurabh
Rotz, Seth J.
Badawy, Sherif M.
Ganguly, Siddhartha
Pavletic, Steven
Nishihori, Taiga
Prestidge, Tim
Agrawal, Vaibhav
Hogan, William J.
Inamoto, Yoshihiro
Shaw, Bronwen E.
Satwani, Prakash
Publication Year :
2020

Abstract

We examined the risk of subsequent neoplasms (SNs) and late mortality in children and adolescents undergoing allogeneic hematopoietic cell transplantation (HCT) for nonmalignant diseases (NMDs). Weincluded 6028 patients (median age, 6 years; interquartile range, 1-11; range, <1 to 20) from the Center for International Blood and Marrow Transplant Research (1995-2012) registry. Standardized mortality ratios (SMRs) in 2-year survivors and standardized incidence ratios (SIRs) were calculated to compare mortality and SN rates with expected rates in the general population. Median follow-up of survivors was 7.8 years. Diagnoses included severe aplastic anemia (SAA; 24%), Fanconi anemia (FA; 10%), other marrow failure (6%), hemoglobinopathy (15%), immunodeficiency (23%), and metabolic/leukodystrophy syndrome (22%). Ten-year survival was 93% (95% confidence interval [95% CI], 92% to 94%; SMR, 4.2; 95% CI, 3.7-4.8). Seventy-one patients developed SNs (1.2%). Incidence was highest in FA (5.5%), SAA (1.1%), and other marrow failure syndromes (1.7%); for other NMDs, incidence was <1%. Hematologic (27%), oropharyngeal (25%), and skin cancers (13%) were most common. Leukemia risk was highest in the first 5 years posttransplantation; oropharyngeal, skin, liver, and thyroid tumors primarily occurred after 5 years. Despite a low number of SNs, patients had an 11-fold increased SN risk (SIR, 11; 95% CI, 8.9-13.9) compared with the general population. We report excellent long-term survival and low SN incidence in an international cohort of children undergoing HCT for NMDs. The risk of SN development was highest in patients with FA and marrow failure syndromes, highlighting the need for long-term posttransplantation surveillance in this population.

Details

Database :
OAIster
Notes :
English
Publication Type :
Electronic Resource
Accession number :
edsoai.on1235305649
Document Type :
Electronic Resource
Full Text :
https://doi.org/10.1182.bloodadvances.2019000839