A retrospective study of myeloid leukaemia in children with Down syndrome in Ireland.


Journal

Irish journal of medical science
ISSN: 1863-4362
Titre abrégé: Ir J Med Sci
Pays: Ireland
ID NLM: 7806864

Informations de publication

Date de publication:
Aug 2020
Historique:
received: 08 10 2019
accepted: 22 01 2020
pubmed: 2 2 2020
medline: 29 8 2020
entrez: 2 2 2020
Statut: ppublish

Résumé

Acute megakaryoblastic leukaemia (AMKL) is a subtype of myeloid leukaemia and is the most common leukaemia type in children with Down syndrome (DS) under 4 years of age. AMKL is often preceded by a transient neonatal pre-leukaemic syndrome, transient myeloproliferative disorder (TMD). Although TMD often spontaneously resolves, 20-30% of these patients subsequently develop AMKL within the first 4 years of life. To perform a retrospective consecutive national audit of all documented cases of childhood TMD and AMKL-DS from 1990 to 2018 at Our Lady's Children's Hospital, Crumlin (OLCHC), Ireland. All patients with a diagnosis of AMKL treated consecutively at (OLCHC) between 1990 and 2018 were reviewed. Kaplan-Meier survival curves were constructed. Twenty-seven patients with AMKL-DS were identified. A prior neonatal diagnosis of TMD was described in 10 patients (37%). Nineteen patients (70%) are alive and well, in complete remission, at a median follow-up of 11.4 years. Overall survival (OS) of this cohort has risen from 54% from those treated between the years 1990 and 2004 (n = 13) to 93% for those treated between the years 2005 and 2018 (n = 14). High cure rates are observed in AMKL-DS using current polychemotherapy protocols. The finding of a low platelet count at time of diagnosis is in keeping with the knowledge that AMKL-DS is a malignancy of platelet progenitor cells.

Sections du résumé

BACKGROUND BACKGROUND
Acute megakaryoblastic leukaemia (AMKL) is a subtype of myeloid leukaemia and is the most common leukaemia type in children with Down syndrome (DS) under 4 years of age. AMKL is often preceded by a transient neonatal pre-leukaemic syndrome, transient myeloproliferative disorder (TMD). Although TMD often spontaneously resolves, 20-30% of these patients subsequently develop AMKL within the first 4 years of life.
AIMS OBJECTIVE
To perform a retrospective consecutive national audit of all documented cases of childhood TMD and AMKL-DS from 1990 to 2018 at Our Lady's Children's Hospital, Crumlin (OLCHC), Ireland.
METHODS METHODS
All patients with a diagnosis of AMKL treated consecutively at (OLCHC) between 1990 and 2018 were reviewed. Kaplan-Meier survival curves were constructed.
RESULTS RESULTS
Twenty-seven patients with AMKL-DS were identified. A prior neonatal diagnosis of TMD was described in 10 patients (37%). Nineteen patients (70%) are alive and well, in complete remission, at a median follow-up of 11.4 years. Overall survival (OS) of this cohort has risen from 54% from those treated between the years 1990 and 2004 (n = 13) to 93% for those treated between the years 2005 and 2018 (n = 14).
CONCLUSION CONCLUSIONS
High cure rates are observed in AMKL-DS using current polychemotherapy protocols. The finding of a low platelet count at time of diagnosis is in keeping with the knowledge that AMKL-DS is a malignancy of platelet progenitor cells.

Identifiants

pubmed: 32006388
doi: 10.1007/s11845-020-02181-y
pii: 10.1007/s11845-020-02181-y
doi:

Types de publication

Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

979-984

Auteurs

Gavin P Dowling (GP)

Royal College of Surgeons in Ireland, Dublin, Ireland.

Andrea Piccin (A)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland. apiccin@gmail.com.
Internal Medicine V, Haematology and Oncology, University of Medicine, Innsbruck, Austria. apiccin@gmail.com.

Katherine T Gavin (KT)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.

David R Betts (DR)

Department of Clinical Genetics, Children's Health Ireland at Crumlin, Dublin, Ireland.

Andrea Malone (A)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.

Melanie Cotter (M)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.
Temple Street Children's University Hospital, Dublin, Ireland.

Aoife Sills (A)

Royal College of Surgeons in Ireland, Dublin, Ireland.

Pamela Evans (P)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.

Aengus O' Marcaigh (A)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.

Owen P Smith (OP)

Department of Haematology and Oncology, Children's Health Ireland at Crumlin, Dublin, Ireland.
University College Dublin (UCD), Dublin, Ireland.

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