Characteristics and treatment of acute myeloid neoplasms with cutaneous involvement in infants up to 6 months of age: A retrospective study.

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State: Public
Version: Final published version
License: CC BY-NC-ND 4.0
Serval ID
serval:BIB_CCB2557653E6
Type
Article: article from journal or magazin.
Collection
Publications
Institution
Title
Characteristics and treatment of acute myeloid neoplasms with cutaneous involvement in infants up to 6 months of age: A retrospective study.
Journal
Pediatric blood & cancer
Author(s)
Renaud J., Goemans B.F., Locatelli F., Pigazzi M., Redmond S., Kuehni C.E., Destaillats A., Alonzo T.A., Gerbing R.B., Gamis A., Aplenc R., Renella R., Cooper T., Ceppi F.
ISSN
1545-5017 (Electronic)
ISSN-L
1545-5009
Publication state
Published
Issued date
07/2024
Peer-reviewed
Oui
Volume
71
Number
7
Pages
e31006
Language
english
Notes
Publication types: Journal Article ; Multicenter Study
Publication Status: ppublish
Abstract
Myeloid neoplasms account for 50% of cases of pediatric leukemias in infants. Approximately 25%-50% of patients with newborn leukemia have cutaneous extramedullary disease (EMD). In less than 10% of patients, aleukemic leukemia cutis or isolated extramedullary disease with cutaneous involvement (cEMD) occurs when skin lesions appear prior to bone marrow involvement and systemic symptoms. Interestingly, in acute myeloid leukemia with cutaneous EMD (AML-cEMD) and cEMD, spontaneous remissions have been reported.
This is a multicentric retrospective cohort study aiming to describe characteristics, treatment, and outcome of infants with either cEMD or presence of cutaneous disease with involvement of the bone marrow (AML-cEMD). This study included patients born between 1990 and 2018 from Italy, the Netherlands, Switzerland, and the United States, diagnosed between 0 and 6 months of life with cEMD or AML-cEMD. Descriptive statistics, Fisher's exact test, Kaplan-Meier method, and log rank test were applied.
The cohort consisted of n = 50 patients, including 42 AML-cEMD and eight cEMD patients. The most common genetic mutation found was a KMT2A rearrangement (n = 26, 52%). Overall 5-year event-free survival (EFS) and overall survival (OS) were 66% [confidence interval (CI): 51-78] and 75% [CI: 60-85], respectively. In two patients, complete spontaneous remission occurred without any therapy. Central nervous system (CNS) involvement was found in 25% of cEMD patients. No difference in outcomes was observed between the AML-cEMD and cEMD groups, but none of the latter patients included in the study died. KMT2A rearrangements were not associated with poorer prognosis.
In the largest cohort to date, our study describes the characteristics of infants with cutaneous involvement of myeloid neoplasms including cytomolecular findings and survival rates. Further prospective biologic and clinical studies of these infants with myeloid neoplasms will be required to individualize therapy for this rare patient population.
Keywords
Humans, Retrospective Studies, Female, Leukemia, Myeloid, Acute/mortality, Leukemia, Myeloid, Acute/pathology, Leukemia, Myeloid, Acute/genetics, Leukemia, Myeloid, Acute/therapy, Infant, Male, Infant, Newborn, Skin Neoplasms/pathology, Skin Neoplasms/mortality, Skin Neoplasms/therapy, Skin Neoplasms/genetics, Follow-Up Studies, Survival Rate, Prognosis, acute myeloid leukemia, aleukemic leukemia cutis, cutaneous infiltration, extramedullary disease, infants
Pubmed
Web of science
Open Access
Yes
Create date
15/04/2024 14:30
Last modification date
14/06/2024 7:17
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