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Вопросы гематологии/онкологии и иммунопатологии в педиатрии. 2017; 16: 26-32

Результаты терапии сарком Юинга в Республике Беларусь

Киселёв Л. П., Алейникова О. В.

https://doi.org/10.24287/1726-1708-2017-16-2-26-32

Аннотация

Полихимиотерапия и локальный контроль - обязательные компоненты лечения пациентов с саркомой Юинга (СЮ). Цель исследования: оценка показателей долгосрочной выживаемости и анализ исходов пациентов с СЮ для установления отличий в клинико-патологических характеристиках пациентов с благоприятными и неблагоприятными результатами терапии. Использованы данные пациентов с СЮ (n = 108), полученные из детского канцер-регистра Республики Беларусь, включенного в Интернациональное агентство исследования рака (IARC). Бессобытийная выживаемость для всей когорты пациентов - 49,2%, при локализованных и метастатических формах - соответственно 64,5 и 19,5%. Такие характеристики пациентов, как пол, возраст, уровень лактатдегидрогеназы, размер и локализация опухоли, имели широкий диапазон и не позволяли однозначно определять группы благоприятного или неблагоприятного исхода заболевания. Представленные результаты свидетельствуют о том, что выживаемость пациентов детского возраста с СЮ в Республике Беларусь соответствует этому показателю в странах с развитой системой здравоохранения. Новые подходы необходимы для курации большинства случаев с метастатическими и трети случаев с локализованными формами заболевания.
Список литературы

1. Алейникова О.В., Потапнев М.П., Сыцкевич О.Н., Петрович С.В., Исмаил-заде Р.С., Стронгин Ю.С. и др. Достижения детской онкологии и гематологии в Республике Беларусь. Актуальные вопросы детской онкологии и гематологии: Материалы VIII Междунар. симпозиума. Минск, 2000; 3-8.

2. Румянцев А.Г., Варфоломеева С.Р., Грачев Н.С., Карачунский А.И., Нович-кова Г.А. Принципы и инструменты доказательной медицины в детской гематологии/онкологии. Доктор.Ру 2015; 10 (111): 6-13.

3. Schaefer K.L., Eisenacher M., Braun Y., Brachwitz K., Wai D.H., Dirksen U., et al. Microarray analysis of Ewing’s sarcoma family of tumours reveals characteristic gene expression signatures associated with metastasis and resistance to chemotherapy. Eur J Cancer 2008; 44: 699-709.

4. Padhye B., McCowage G. Chemotherapy regimens in newly diagnosed and recurrent ewing sarcoma in children and young adults. Cancer Forum 2010; 34: 3.

5. Smith M.A., Altekruse S.F., Adamson P.C., Reaman G.H., Seibel N.L. Declining childhood and adolescent cancer mortality. Cancer 2014; 120: 2497-506.

6. Cotterill S.J., Ahrens S., Paulussen M., Jurgens H.F., Voute P.A., Gadner H., et al. Prognostic factors in Ewing’s tumor of bone: analysis of 975 patients from the European Intergroup Cooperative Ewing’s Sarcoma Study Group. J Clin Oncol 2000; 18: 3108-14.

7. Rodriguez-Galindo C., Navid F., Liu T., Billups C.A., Rao B.N., Krasin M.J. Prognostic factors for local and distant control in Ewing sarcoma family of tumors. Ann Oncol 2008; 19: 814-20.

8. Arndt V., Lacour B., Steliarova-Foucher E., Spix C., Znaor A., Pastore G., et al. Up-to-date monitoring of childhood cancer long-term survival in Europe: tumours of the sympathetic nervous system, retinoblastoma, renal and bone tumours, and soft tissue sarcomas. Ann Oncol 2007; 18: 1722-33.

9. Applebaum M.A., Worch J., Matthay K.K., Goldsby R., Neuhaus J., West D.C., Dubois S.G., et al. Clinical features and outcomes in patients with extraskeletal Ewing sarcoma. Cancer 2011; 117 (13): 3027-32.

10. Petrovich S., Aleynikova O., Shumikhina T. Epidemiological aspects of childhood onco-hematological morbidity in the Republic of Belarus. Vopr Onkol 2002; 48 (3): 301-5.

11. Paulussen M., Craft A.W., Lewis I., Hackshaw A., Douglas C., Dunst J., et al. Results of the EICESS-92 Study: two randomized trials of Ewing’s sarcoma treatment-cyclophosphamide compared with ifosfamide in standard-risk patients and assessment of benefit of etoposide added to standard treatment in high-risk patients. J Clin Oncol 2008; 26: 4385-93.

12. Granowetter L., Womer R., Devidas M., Krailo M., Wang C., Bernstein M., et al. Dose-intensified compared with standard chemotherapy for nonmetastatic Ewing sarcoma family of tumours: a Children’s Oncology Group study. J Clin Oncol 2009; 27: 2536-41.

13. Oberlin O., Rey A., Desfachelles A.S., Philip T., Plantaz D., Schmitt C., et al. Impact of high-dose busulfan plus melphalan as consolidation in metastatic Ewing tumours: a study by the Societe Francaise des Cancers de l’Enfant. J Clin Oncol 2006; 24: 3997-4002.

14. Juergens C., Weston C., Lewis I., Whelan J., Paulussen M., Oberlin O., et al. Safety assessment of intensive induction with vincristine, ifosfamide, doxorubicin and etoposide (VIDE) in the treatment of Ewing tumours in the EURO-E.W.I.N.G. 99 clinical trial. Pediatr Blood Cancer 2006; 47: 22-9.

15. Neerav S., Joshua D., Schiffman D. R., Davis I.J., Womer R.B., Lessnick S.L., Lawlor E.R. The COG Ewing Sarcoma Biology Committee. Biomarkers in Ewing sarcoma: the promise and challenge of personalized medicine. A report from the Children’s Oncology Group. Frontiers in oncology. 2013; 3: 1-13.

16. Киселёв Л.П., Савицкая Т.В., Липай Н.В., Алейникова О.В. TFPI2, VEGFА165 и VEGFА189 как независимые факторы прогноза локализованных форм саркомы Юинга // Онкологический журнал (Минск, Республика Беларусь), 2016; 10: 93-100.

Pediatric Hematology/Oncology and Immunopathology. 2017; 16: 26-32

The treatment results of Ewing's sarcomas in children in Belarus Republic

Kisialeu L. , Aleinikova O.

https://doi.org/10.24287/1726-1708-2017-16-2-26-32

Abstract

Chemotherapy and local control are mandatory components of treatment for patients with Ewing's sarcoma (ES). The aim of the study where the survival rates and clinical and pathological characteristics of patients with ES in the Belarus Republic (RB) over a 15-year. Data of ES patients (n=108) obtained from the RB children's cancer-registry (included in the International Agency for Research on Cancer, IARC) were used in the work. Event-free survival was superior for localized EES compared with metastatic forms (64,5% vs 19,5%; р < 0.0001). Patient characteristics: sex, age, level of lactate dehydrogenase, tumor size and location were characterized by a wide range and did not allow distinguish groups of favorable or unfavorable outcome. Treatment results for RB patients with ES are similar to generally accepted standards. Patients with metastatic status need new treatment strategies. Also new approaches are needed for identification and curate for third of patients with localized ES forms and unfavorable outcomes.

References

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3. Schaefer K.L., Eisenacher M., Braun Y., Brachwitz K., Wai D.H., Dirksen U., et al. Microarray analysis of Ewing’s sarcoma family of tumours reveals characteristic gene expression signatures associated with metastasis and resistance to chemotherapy. Eur J Cancer 2008; 44: 699-709.

4. Padhye B., McCowage G. Chemotherapy regimens in newly diagnosed and recurrent ewing sarcoma in children and young adults. Cancer Forum 2010; 34: 3.

5. Smith M.A., Altekruse S.F., Adamson P.C., Reaman G.H., Seibel N.L. Declining childhood and adolescent cancer mortality. Cancer 2014; 120: 2497-506.

6. Cotterill S.J., Ahrens S., Paulussen M., Jurgens H.F., Voute P.A., Gadner H., et al. Prognostic factors in Ewing’s tumor of bone: analysis of 975 patients from the European Intergroup Cooperative Ewing’s Sarcoma Study Group. J Clin Oncol 2000; 18: 3108-14.

7. Rodriguez-Galindo C., Navid F., Liu T., Billups C.A., Rao B.N., Krasin M.J. Prognostic factors for local and distant control in Ewing sarcoma family of tumors. Ann Oncol 2008; 19: 814-20.

8. Arndt V., Lacour B., Steliarova-Foucher E., Spix C., Znaor A., Pastore G., et al. Up-to-date monitoring of childhood cancer long-term survival in Europe: tumours of the sympathetic nervous system, retinoblastoma, renal and bone tumours, and soft tissue sarcomas. Ann Oncol 2007; 18: 1722-33.

9. Applebaum M.A., Worch J., Matthay K.K., Goldsby R., Neuhaus J., West D.C., Dubois S.G., et al. Clinical features and outcomes in patients with extraskeletal Ewing sarcoma. Cancer 2011; 117 (13): 3027-32.

10. Petrovich S., Aleynikova O., Shumikhina T. Epidemiological aspects of childhood onco-hematological morbidity in the Republic of Belarus. Vopr Onkol 2002; 48 (3): 301-5.

11. Paulussen M., Craft A.W., Lewis I., Hackshaw A., Douglas C., Dunst J., et al. Results of the EICESS-92 Study: two randomized trials of Ewing’s sarcoma treatment-cyclophosphamide compared with ifosfamide in standard-risk patients and assessment of benefit of etoposide added to standard treatment in high-risk patients. J Clin Oncol 2008; 26: 4385-93.

12. Granowetter L., Womer R., Devidas M., Krailo M., Wang C., Bernstein M., et al. Dose-intensified compared with standard chemotherapy for nonmetastatic Ewing sarcoma family of tumours: a Children’s Oncology Group study. J Clin Oncol 2009; 27: 2536-41.

13. Oberlin O., Rey A., Desfachelles A.S., Philip T., Plantaz D., Schmitt C., et al. Impact of high-dose busulfan plus melphalan as consolidation in metastatic Ewing tumours: a study by the Societe Francaise des Cancers de l’Enfant. J Clin Oncol 2006; 24: 3997-4002.

14. Juergens C., Weston C., Lewis I., Whelan J., Paulussen M., Oberlin O., et al. Safety assessment of intensive induction with vincristine, ifosfamide, doxorubicin and etoposide (VIDE) in the treatment of Ewing tumours in the EURO-E.W.I.N.G. 99 clinical trial. Pediatr Blood Cancer 2006; 47: 22-9.

15. Neerav S., Joshua D., Schiffman D. R., Davis I.J., Womer R.B., Lessnick S.L., Lawlor E.R. The COG Ewing Sarcoma Biology Committee. Biomarkers in Ewing sarcoma: the promise and challenge of personalized medicine. A report from the Children’s Oncology Group. Frontiers in oncology. 2013; 3: 1-13.

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