Androgen therapy in inherited bone marrow failure syndromes: analysis from the Canadian Inherited Marrow Failure Registry
Summary Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to improve blood counts in different bone marrow failure conditions. Little is known about efficacy and toxicity with new androgens (i.e.,...
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| Published in | British journal of haematology Vol. 189; no. 5; pp. 976 - 981 |
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| Main Authors | , , , , , , , , , , , , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
England
Blackwell Publishing Ltd
01.06.2020
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0007-1048 1365-2141 1365-2141 |
| DOI | 10.1111/bjh.16445 |
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| Abstract | Summary
Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to improve blood counts in different bone marrow failure conditions. Little is known about efficacy and toxicity with new androgens (i.e., danazol) in different types of IBMFS. We identified 29 patients from the Canadian Inherited Marrow Failure Registry, who received oxymetholone or danazol. Sixteen (55%) had haematological response including patients with unclassified IBMFS (45%). Danazol showed a better toxicity profile and similar efficacy compared to oxymetholone. Androgens are an effective and safe option to ameliorate bone marrow failure in IBMFS. |
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| AbstractList | Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to improve blood counts in different bone marrow failure conditions. Little is known about efficacy and toxicity with new androgens (i.e., danazol) in different types of IBMFS. We identified 29 patients from the Canadian Inherited Marrow Failure Registry, who received oxymetholone or danazol. Sixteen (55%) had haematological response including patients with unclassified IBMFS (45%). Danazol showed a better toxicity profile and similar efficacy compared to oxymetholone. Androgens are an effective and safe option to ameliorate bone marrow failure in IBMFS. Summary Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to improve blood counts in different bone marrow failure conditions. Little is known about efficacy and toxicity with new androgens (i.e., danazol) in different types of IBMFS. We identified 29 patients from the Canadian Inherited Marrow Failure Registry, who received oxymetholone or danazol. Sixteen (55%) had haematological response including patients with unclassified IBMFS (45%). Danazol showed a better toxicity profile and similar efficacy compared to oxymetholone. Androgens are an effective and safe option to ameliorate bone marrow failure in IBMFS. Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to improve blood counts in different bone marrow failure conditions. Little is known about efficacy and toxicity with new androgens (i.e., danazol) in different types of IBMFS. We identified 29 patients from the Canadian Inherited Marrow Failure Registry, who received oxymetholone or danazol. Sixteen (55%) had haematological response including patients with unclassified IBMFS (45%). Danazol showed a better toxicity profile and similar efficacy compared to oxymetholone. Androgens are an effective and safe option to ameliorate bone marrow failure in IBMFS.Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to improve blood counts in different bone marrow failure conditions. Little is known about efficacy and toxicity with new androgens (i.e., danazol) in different types of IBMFS. We identified 29 patients from the Canadian Inherited Marrow Failure Registry, who received oxymetholone or danazol. Sixteen (55%) had haematological response including patients with unclassified IBMFS (45%). Danazol showed a better toxicity profile and similar efficacy compared to oxymetholone. Androgens are an effective and safe option to ameliorate bone marrow failure in IBMFS. |
| Author | Zlateska, Bozana Brossard, Josee Goodyear, Lisa Cada, Michaela Lipton, Jeffrey H. Jardine, Lawrence Silva, Mariana Ali, Salah S. Cuvelier, Geoffrey D. E. Michon, Bruno Lauhasurayotin, Supanun Steele, MacGregor Abish, Sharon Klaassen, Robert J. Dror, Yigal Sinha, Roona Català, Albert Fernandez, Conrad V. Sung, Lillian Corriveau‐Bourque, Catherine Breakey, Vicky R. Pastore, Yves D. Rayar, Meera |
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| Cites_doi | 10.3109/08880018.2015.1129567 10.1016/S0149-2918(01)80114-4 10.1002/pbc.24617 10.1182/blood-2008-09-178871 10.1007/s12185-016-1951-0 10.1111/bjh.12748 10.1002/ajh.20183 10.1007/s00228-009-0771-z 10.1016/j.bcmd.2011.11.006 10.1007/s12185-012-1249-9 10.1182/asheducation-2017.1.96 10.1111/bjh.12432 10.1056/NEJMoa1515319 |
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Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been... Progressive cytopenia is a serious complication among paediatric patients with inherited bone marrow failure syndromes (IBMFS). Androgens have been used to... |
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| SubjectTerms | Adolescent Adult Androgens Androgens - adverse effects Androgens - therapeutic use Bone marrow bone marrow failure Bone Marrow Failure Disorders - blood Bone Marrow Failure Disorders - drug therapy Bone Marrow Failure Disorders - genetics Bone Marrow Failure Disorders - therapy Canada - epidemiology Cell Lineage Child Child, Preschool Combined Modality Therapy danazol Danazol - adverse effects Danazol - therapeutic use Disease Progression Drug Substitution dyskeratosis congenita Fanconi anaemia Female Hematology Hematopoietic Stem Cell Transplantation Humans Infant Male Middle Aged Oxymetholone - adverse effects Oxymetholone - therapeutic use Pancytopenia - drug therapy Pancytopenia - etiology Registries Thrombocytopenia - drug therapy Thrombocytopenia - etiology Toxicity Treatment Outcome Virilism - chemically induced |
| Title | Androgen therapy in inherited bone marrow failure syndromes: analysis from the Canadian Inherited Marrow Failure Registry |
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