The prognostic value of the morhological types of ALK-positive anaplastic large cell lymphoma in children
- Authors: Abramov D.S.1, Fedorova A.S.1, Voronin K.A.1, Levin P.A.1, Fominykh V.V.1, Volchkov E.V.1, Myakova N.V.1, Konovalov D.M.1
-
Affiliations:
- The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
- Issue: Vol 23, No 3 (2024)
- Pages: 80-90
- Section: ORIGINAL ARTICLES
- Submitted: 14.10.2024
- Accepted: 14.10.2024
- Published: 08.07.2025
- URL: https://hemoncim.com/jour/article/view/900
- DOI: https://doi.org/10.24287/1726-1708-2024-23-3-80-90
- ID: 900
Cite item
Full Text
Abstract
The aim of our study is to evaluate the prognostic value of the morphological types of ALK-positive anaplastic large cell lymphoma (ALK+ ALCL) in children. We performed a retrospective analysis of data on 81 cases of pediatric ALK+ ALCL which had been diagnosed in 2011–2022. All patients and/or their legal representatives signed voluntary informed consent for participation in the study, as well as for biological material testing. The analysis of medical records was carried out in accordance with the internal rules of the Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthсare of the Russian Federation, developed and approved by the Independent Ethics Committee of the Center. The patients received treatment according to the standard protocol. We assessed overall (OS) and event-free (EFS) survival depending on the morphological type of the tumor. The median follow-up was 55.6 months. Three-year and 5-year OS rates were 81.9% and 79.8%, respectively. Three-year and 5-year EFS rates were 59.6% and 56.0%, respectively. There were no statistically significant differences in OS and EFS between the common and non-common morphological types of ALK+ ALCL. Better survival rates were observed in the patients with a lymphohistiocytic variant. We found statistically significant differences in OS (p = 0.031) and EFS (p = 0.002) between the cases with a small cell component and without it. The results suggest that ALK+ ALCL with small cell morphology has a more aggressive course in children. Validation in larger patient cohorts and further study of the biology of different morphological types are needed to develop stratified treatment approaches.
About the authors
D. S. Abramov
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: abramovd_s@bk.ru
ORCID iD: 0000-0003-3664-2876
Dmitry S. Abramov - MD, a pathologist at the Pathology Department.
1 Samory Mashela St., Moscow 117997
Russian FederationA. S. Fedorova
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: info@eco-vector.com
Moscow
Russian FederationK. A. Voronin
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: abramovd_s@bk.ru
Moscow
Russian FederationP. A. Levin
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: abramovd_s@bk.ru
Moscow
Russian FederationV. V. Fominykh
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: abramovd_s@bk.ru
Moscow
Russian FederationE. V. Volchkov
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: abramovd_s@bk.ru
ORCID iD: 0000-0002-2574-1636
Moscow
Russian FederationN. V. Myakova
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Email: abramovd_s@bk.ru
ORCID iD: 0000-0002-4779-1896
Moscow
Russian FederationD. M. Konovalov
The Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology of Ministry of Healthcare of the Russian Federation
Author for correspondence.
Email: abramovd_s@bk.ru
ORCID iD: 0000-0001-7732-8184
Moscow
Russian FederationReferences
- Stein H., Mason D.Y., Gerdes J., O'Connor N., Wainscoat J., Pallesen G., et al. The expression of the Hodgkin’s disease associated antigen Ki-1 in reactive and neoplastic lymphoid tissue: evidence that Reed-Sternberg cells and histiocytic malignancies are derived from activated lymphoid cells. Blood 1985; 66: 848–58. doi: 10.1182/BLOOD.V66.4.848.848
- Morris S.W., Kirstein M.N., Valentine M.B., Dittmer K.G., Shapiro D.N., Saltman D.L., Look A.T. Fusion of a Kinase Gene, ALK, to a Nucleolar Protein Gene, NPM, in Non-Hodgkin’s Lymphoma. Science 1994; 263: 1281–4. doi: 10.1126/SCIENCE.8122112
- Leventaki V., Bhattacharyya S., Lim M.S. Pathology and genetics of anaplastic large cell lymphoma. Semin Diagn Pathol 2019; 37: 57–71. doi: 10.1053/J.SEMDP.2019.12.002
- Falini B., Lamant L., Campo E., Jaffe E.S., Gascoyne R.D., Stein H., et al. Anaplastic large cell lymphoma, alk positive. In: Swerdlow S.H., Campo E., Harris N.L., Jaffe E.S., Pileri S.A., Stein H. (eds.). Who Classification of Tumors of Hematopoietic and Lymphoid Tissues. IARC: Lyon (France); 2017. Рp. 413–418.
- Tole S., Wheaton L., Alexander S. Pediatric Anaplastic Large Cell Lymphoma – A Review. Oncol Hematol Rev 2018; 14 (1): 21–7.
- Lamant L., McCarthy K., D’Amore E., Klapper W., Nakagawa A., Fraga M., et al. Prognostic impact of morphologic and phenotypic features of childhood ALK-positive anaplastic large-cell lymphoma: results of the ALCL99 study. J Clin Oncol 2011; 29 (35): 4669–76. doi: 10.1200/JCO.2011.36.5411
- Mussolin L., Le Deley M.C., Carraro E., Damm-Welk C., Attarbaschi A. Williams D., et al. Prognostic Factors in Childhood Anaplastic Large Cell Lymphoma: Long Term Results of the International ALCL99 Trial. Cancers (Basel) 2020; 12: 2747. doi: 10.3390/CANCERS12102747
- Valiev T.T., Levashov A.S., Popa A.V., Mentkevich G.L. The results of differentiated therapy of anaplastic large cell lymphoma in children. Oncopediatrics 2016; 3 (2): 142. (In Russ.).
- Valiev T.T., Morozova O.V., Kovrigina A.M., Makhonova L.A., Sholokhova E.N., Serebryakova I.N., et al. Diagnosis and treatment of anaplastic large cell lymphomas in children. Hematology and transfusiology 2012; 57: 3–9. (In Russ.).
- Mussolin L., Damm-Welk C., Pillon M., Zimmermann M., Franceschetto G., Pulford K., et al. Use of minimal disseminated disease and immunity to NPM-ALK antigen to stratify ALK-positive ALCL patients with different prognosis. Leukemia 2009; 27: 416–22. doi: 10.1038/LEU.2012.205
- Pillon M., Aricò M., Mussolin L., Carraro E., Conter V., Sala A., et al. Longterm results of the AIEOP LNH-97 protocol for childhood lymphoblastic lymphoma. Pediatr Blood Cancer 2015; 62: 1388–94. doi: 10.1002/PBC.25469
- Lowe E.J., Sposto R., Perkins S.L., Gross T.G., Finlay J., Zwick D., et al. Intensive chemotherapy for systemic anaplastic large cell lymphoma in children and adolescents: final results of Children’s Cancer Group Study 5941. Pediatr Blood Cancer 2009; 52: 335–9. doi: 10.1002/PBC.21817
- Seidemann K., Tiemann M., Schrappe M., Yakisan E., Simonitsch I., Janka-Schaub G., et al. Short-pulse B-non-Hodgkin lymphoma-type chemotherapy is efficacious treatment for pediatric anaplastic large cell lymphoma: a report of the Berlin-Frankfurt-Münster Group Trial NHL-BFM 90. Blood 2001; 97: 3699–706. doi: 10.1182/blood.v97.12.3699
- Le Deley M.C., Rosolen A., Williams D.M., Horibe K., Wrobel G., Attarbaschi A., et al. Vinblastine in children and adolescents with high-risk anaplastic large-cell lymphoma: results of the randomized ALCL99-vinblastine trial. J Clin Oncol 2010; 28: 3987–93. DOI: 10.1200/ JCO.2010.28.5999
- Ait-Tahar K., Damm-Welk C., Burkhardt B., Zimmermann M., Klapper W., Reiter A., et al. Correlation of the autoantibody response to the ALK oncoantigen in pediatric anaplastic lymphoma kinase-positive anaplastic large cell lymphoma with tumor dissemination and relapse risk. Blood 2010; 115: 3314–9. doi: 10.1182/blood-2009-11-251892
Supplementary files
