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Вопросы гематологии/онкологии и иммунопатологии в педиатрии. 2021; 20: 66-74

Хориоидкарцинома у детей

Валиахметова Э. Ф., Папуша Л. И., Санакоева А. В., Шишкина Л. В., Быданов О. И., Новичкова Г. А., Горелышев С. К., Карачунский А. И.

https://doi.org/10.24287/1726-1708-2021-20-1-66-74

Аннотация

Хориоидкарцинома (ХК) представляeт собой редкую, агрессивную опухоль головного мозга у детей с низкими показателями выживаемости. В настоящее время не существует определенной оптимальной стратегии лечения этого новообразования. Данное исследование одобрено независимым этическим комитетом и утверждено решением ученого совета ФГБУ «НМИЦ ДГОИ им. Дмитрия Рогачева» Минздрава России. В исследование были включены больные с верифицированным диагнозом «хориоидкарцинома» в возрасте от 0 до 18 лет в период с 01.01.2009 по 31.12.2019. Всего было зарегистрировано 32 пациента. Медиана возраста составила 2,65 года, 93,5% ХК локализовались в боковых желудочках. Метастазы на момент постановки диагноза были обнаружены в 21,8% случаев, 5-летняя бессобытийная выживаемость (БСВ) у детей с метастазами была ниже, чем у пациентов без метастазов: 29 ± 17% и 49 ± 12% соответственно. В нашей когорте полное удаление опухоли было выполено 65,6% пациентов. Пятилетняя БСВ у пациентов с полным удалением ХК была выше по сравнению с детьми, которым было выполнено субтотальное и частичное удаление опухоли (63 ± 13%, 12 ± 11% и 0% соответственно). К тому же общая выживаемость (ОВ) была немного выше у тех, кому было выполнено полное удаление опухоли, по сравнению с субтотальным и частичным удалением (74 ± 12%, 67 ± 16% и 0% соответственно). Из 32 детей с ХК программную химиотерапию (ХТ) получили 15 детей, непрограммную ХТ – 17, 5-летняя БСВ у пациентов, получивших программную и непрограммную ХТ, составила 79 ± 11% и 0% соответственно (р = 0,0006), 5-летняя ОВ у пациентов, получивших программную и непрограммную ХТ, составила 93 ± 7% и 36 ± 14% соответственно (р = 0,0054). 

Список литературы

1. Sun M.Z., Oh M.C., Ivan M.E., Kaur G., Safaee M., Kim J.M., et al. Current management of choroid plexus carcinomas. Neurosurgical Review. 2014; 37 (2): 179– 92. DOI: 10.1007/s10143-013-0499-1

2. Louis D.N., Perry A., Reifenberger G., von Deimling A., Figarella-Branger D., Cavenee W.K., et al. The 2016 World Health Organization Classification of Tumors of the Central Nervous System: a summary. Acta Neuropathol 2016; 131 (6): 1–18. DOI: 10.1007/s00401-016-1545-1

3. Cannon D.M., Mohindra P., Gondi V., Kruser T.J., Kozak K.R. Choroid plexus tumor epidemiology and outcomes: implications for surgical and radiotherapeutic management. J Neurooncol 2015; 121 (1): 151–7. DOI: 10.1007/s11060-014-1616-x

4. Wolff J.E.A., Sajedi M., Brant R., Coppes M.J., Egeler R.M. Choroid plexus tumours. Br J Cancer. 2002; 87 (10): 1086–91. DOI: 10.1038/sj.bjc.6600609

5. Meyers S.P., Chuang S.H., Pollack I.F., Korones D.N., Zimmerman R.A. Choroid plexus carcinomas in children: MRI features and patient outcomes. Neuroradiology 2004; 46 (9): 770–80. DOI: 10.1007/s00234-004-1238-7

6. Wrede B., Hasselblatt M., Peters O., Thall P.F., Kutluk T., Moghrabi A., et al. Atypical choroid plexus papilloma: Clinical experience in the CPT-SIOP-2000 study. J Neurooncol 2009; 95 (3): 383– 92. DOI: 10.1007/s11060-009-9936-y

7. Wolff J.E.A., Sajedi M., Coppes M.J., Anderson R.A., Maarten Egeler R. Radiation therapy and survival in choroid plexus carcinoma. Lancet 1999; 353 (9170): 2126. DOI: 10.1016/S0140-6736(99)01744-4

8. Gozali A.E., Britt B., Shane L., Gonzalez I., Gilles F., McComb J.G., et al. Choroid Plexus Tumors; Management, Outcome, and Association With the Li–Fraumeni Syndrome: The Children’s Hospital Los Angeles (CHLA) Experience, 1991–2010. Pediatr Blood Cancer 2012; 58 (5): 905– 9. DOI: 10.1002/pbc

9. Zaky W., Dhall G., Khatua S., Brown R.J., Ginn K.F., Gardner S.L., et al. Choroid Plexus Carcinoma in Children: The Head Start Experience. Pediatr Blood Cancer 2015; 62 (5): 784–9. DOI: 10.1002/pbc.27031

10. Siegfried A., Morin S., Munzer C., Delisle M., Gambart M., Puget S., et al. A French retrospective study on clinical outcome in 102 choroid plexus tumors in children. J Neurooncol 2017; 135 (1): 151–60. DOI: 10.1007/s11060-017-2561-2

11. Mallick S., Benson R., Melgandi W., Rath GK. Effect of Surgery, Adjuvant Therapy, and Other Prognostic Factors on Choroid Plexus Carcinoma: A Systematic Review and Individual Patient Data Analysis. Int J Radiat Oncol 2017; 99 (5): 1199– 206. DOI: 10.1016/j.ijrobp.2017.08.012

12. Sun M.Z., Ivan M.E., Oh M.C., Delance A.R., Clark A.J., Safaee M., et al. Effects of adjuvant chemotherapy and radiation on overall survival in children with choroid plexus carcinoma. J Neurooncol 2014; 120 (2): 353–60. DOI: 10.1007/s11060-014-1559-2

13. Louis D.N. WHO classification of tumours of central nervous system. In: Louis D.N., Ohgaki H., Wiestler O.D., Cavenee W.K., Ellison D.W., Branger F.D., et al. (eds.). International Agency for Research on Cancer. WHO Classification of Tumours of the Central Nervous System. 4th ed. Lyon: International Agency for Research Centre; 2007. 309 p. DOI: 10.1007/s00401-007-0243-4

14. Lafay-Cousin L., Keene D., Carret A., Fryer C., Larouche V., Silva M., et al. Choroid plexus tumors in children less than 36 months: the Canadian Pediatric Brain Tumor Consortium (CPBTC) experience. Child’s Nerv Syst 2011; 27 (2): 259–64. DOI: 10.3171/2010.3.PEDS09354

15. Bettegowda C., Adogwa O., Mehta V., Chaichana K.L., Weingart J., Carson B.S., et al. Treatment of choroid plexus tumors: a 20-year single institutional experience. Neurosurg Pediatr. 2012; 14 (11): 871–82. DOI: 10.3171/2012.8.PEDS12132

16. Hosmann A., Hinker F., Dorfer C., Slavc I., Haberler C., Dieckmann K., et al. Management of choroid plexus tumors — an institutional experience. Acta Neurochir (Wien) 2019; 161 (4): 745–54. DOI: 10.1007/s00701-019-03832-5

17. Zhou J.W.L., Wang X., Peng J.Y., Ma S.C., Zhang D.N., Guan X.D., et al. Clinical Features and Prognostic Risk Factors of Choroid Plexus Tumors in Children. Chinese Med J 2018; 131 (24): 2938–46. DOI: 10.4103/0366-6999.247195

18. Packer R.J., Perilongo G., Johnson D., Sutton L.N., Vezina G., Zimmerman R.A., et al. Choroid plexus carcinoma of childhood. Cancer 1992; 69 (2): 580–5. DOI: 10.1002/1097-0142(19920115)69:2 ˂580::AIDCNCR2820690250˃ 3.0.CO;2-O

19. Wrede B., Liu P., Wolff J.E.A. Chemotherapy improves the survival of patients with choroid plexus carcinoma: A meta-analysis of individual cases with choroid plexus tumors. J Neurooncol 2007; 85 (3): 345–51. DOI: 10.1007/s11060-007-9428-x

20. Валиахметова Э.Ф., Папуша Л.И., Ясько Л.А., Друй А.Е., Горелышев С.К., Карачунский А.И. Хориоидкарциномы у детей: обзор литературы. Российский журнал детской гематологии и онкологии 2020; 7 (1): 51–61. DOI: 10.21682/2311-1267-2020-7-1-51-61

21. Bahar M., Dhir A., Kordes U., Wolff J. Intercontinental multidisciplinary data collection and treatment optimization study for patients with choroid plexus tumor. Neuro-Oncology 2015; 17 (suppl 5): v186. DOI: 10.1093/neuonc/nov229.01

Pediatric Hematology/Oncology and Immunopathology. 2021; 20: 66-74

Choroid plexus carcinoma in children

Valiakhmetova A. F., Papusha L. I., Sanakoeva A. V., Shishkina L. V., Budanov O. I., Novichkova G. A., Gorelyshev S. K., Karachunskiy A. I.

https://doi.org/10.24287/1726-1708-2021-20-1-66-74

Abstract

Choroid plexus carcinomas (CPCs) are rare pediatric tumors with a generally poor prognosis. Currently there is no definite optimal treatment strategy for this neoplasm. This study is supported by the Independent Ethics Committee and approved by the Academic Council of the Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology. This study included patients with a verified diagnosis of CPC at the age 0 to 18 years in the period from 01.01.2009 to 31.12.2019. A total of 32 patients were registered. The median age was 2.65 years; 93.5% of CPCs were localized in the lateral ventricles. Initial metastases were found in 21.8% of cases; 5-year event-free survival (EFS) in children with metastases was lower than in those who did not have metastases 29 ± 17% and 49 ± 12%. In our cohort, gross total resection (GTR) was performed in 65.6% of patients. The five-year EFS in patients with complete CPC removal was higher than in patients who underwent subtotal and partial resection (63 ± 13%, 12 ± 11%, and 0%, respectively). In addition, overall survival (OS) was slightly higher in those who underwent GTR compared with subtotal and partial resection (74 ± 12%, 67 ± 16%, and 0%, respectively). Of the 32 children with CPCs, 15 children received programm chemotherapy, 17 non-programm chemotherapy, 5-year EFS in patients who received programm and non-programm chemotherapy was 79 ± 11% and 0%, respectively (p = 0.0006), 5-year OS in patients who received programm and non-programm chemotherapy was 93 ± 7% and 36 ± 14% (p = 0.0054). 

References

1. Sun M.Z., Oh M.C., Ivan M.E., Kaur G., Safaee M., Kim J.M., et al. Current management of choroid plexus carcinomas. Neurosurgical Review. 2014; 37 (2): 179– 92. DOI: 10.1007/s10143-013-0499-1

2. Louis D.N., Perry A., Reifenberger G., von Deimling A., Figarella-Branger D., Cavenee W.K., et al. The 2016 World Health Organization Classification of Tumors of the Central Nervous System: a summary. Acta Neuropathol 2016; 131 (6): 1–18. DOI: 10.1007/s00401-016-1545-1

3. Cannon D.M., Mohindra P., Gondi V., Kruser T.J., Kozak K.R. Choroid plexus tumor epidemiology and outcomes: implications for surgical and radiotherapeutic management. J Neurooncol 2015; 121 (1): 151–7. DOI: 10.1007/s11060-014-1616-x

4. Wolff J.E.A., Sajedi M., Brant R., Coppes M.J., Egeler R.M. Choroid plexus tumours. Br J Cancer. 2002; 87 (10): 1086–91. DOI: 10.1038/sj.bjc.6600609

5. Meyers S.P., Chuang S.H., Pollack I.F., Korones D.N., Zimmerman R.A. Choroid plexus carcinomas in children: MRI features and patient outcomes. Neuroradiology 2004; 46 (9): 770–80. DOI: 10.1007/s00234-004-1238-7

6. Wrede B., Hasselblatt M., Peters O., Thall P.F., Kutluk T., Moghrabi A., et al. Atypical choroid plexus papilloma: Clinical experience in the CPT-SIOP-2000 study. J Neurooncol 2009; 95 (3): 383– 92. DOI: 10.1007/s11060-009-9936-y

7. Wolff J.E.A., Sajedi M., Coppes M.J., Anderson R.A., Maarten Egeler R. Radiation therapy and survival in choroid plexus carcinoma. Lancet 1999; 353 (9170): 2126. DOI: 10.1016/S0140-6736(99)01744-4

8. Gozali A.E., Britt B., Shane L., Gonzalez I., Gilles F., McComb J.G., et al. Choroid Plexus Tumors; Management, Outcome, and Association With the Li–Fraumeni Syndrome: The Children’s Hospital Los Angeles (CHLA) Experience, 1991–2010. Pediatr Blood Cancer 2012; 58 (5): 905– 9. DOI: 10.1002/pbc

9. Zaky W., Dhall G., Khatua S., Brown R.J., Ginn K.F., Gardner S.L., et al. Choroid Plexus Carcinoma in Children: The Head Start Experience. Pediatr Blood Cancer 2015; 62 (5): 784–9. DOI: 10.1002/pbc.27031

10. Siegfried A., Morin S., Munzer C., Delisle M., Gambart M., Puget S., et al. A French retrospective study on clinical outcome in 102 choroid plexus tumors in children. J Neurooncol 2017; 135 (1): 151–60. DOI: 10.1007/s11060-017-2561-2

11. Mallick S., Benson R., Melgandi W., Rath GK. Effect of Surgery, Adjuvant Therapy, and Other Prognostic Factors on Choroid Plexus Carcinoma: A Systematic Review and Individual Patient Data Analysis. Int J Radiat Oncol 2017; 99 (5): 1199– 206. DOI: 10.1016/j.ijrobp.2017.08.012

12. Sun M.Z., Ivan M.E., Oh M.C., Delance A.R., Clark A.J., Safaee M., et al. Effects of adjuvant chemotherapy and radiation on overall survival in children with choroid plexus carcinoma. J Neurooncol 2014; 120 (2): 353–60. DOI: 10.1007/s11060-014-1559-2

13. Louis D.N. WHO classification of tumours of central nervous system. In: Louis D.N., Ohgaki H., Wiestler O.D., Cavenee W.K., Ellison D.W., Branger F.D., et al. (eds.). International Agency for Research on Cancer. WHO Classification of Tumours of the Central Nervous System. 4th ed. Lyon: International Agency for Research Centre; 2007. 309 p. DOI: 10.1007/s00401-007-0243-4

14. Lafay-Cousin L., Keene D., Carret A., Fryer C., Larouche V., Silva M., et al. Choroid plexus tumors in children less than 36 months: the Canadian Pediatric Brain Tumor Consortium (CPBTC) experience. Child’s Nerv Syst 2011; 27 (2): 259–64. DOI: 10.3171/2010.3.PEDS09354

15. Bettegowda C., Adogwa O., Mehta V., Chaichana K.L., Weingart J., Carson B.S., et al. Treatment of choroid plexus tumors: a 20-year single institutional experience. Neurosurg Pediatr. 2012; 14 (11): 871–82. DOI: 10.3171/2012.8.PEDS12132

16. Hosmann A., Hinker F., Dorfer C., Slavc I., Haberler C., Dieckmann K., et al. Management of choroid plexus tumors — an institutional experience. Acta Neurochir (Wien) 2019; 161 (4): 745–54. DOI: 10.1007/s00701-019-03832-5

17. Zhou J.W.L., Wang X., Peng J.Y., Ma S.C., Zhang D.N., Guan X.D., et al. Clinical Features and Prognostic Risk Factors of Choroid Plexus Tumors in Children. Chinese Med J 2018; 131 (24): 2938–46. DOI: 10.4103/0366-6999.247195

18. Packer R.J., Perilongo G., Johnson D., Sutton L.N., Vezina G., Zimmerman R.A., et al. Choroid plexus carcinoma of childhood. Cancer 1992; 69 (2): 580–5. DOI: 10.1002/1097-0142(19920115)69:2 ˂580::AIDCNCR2820690250˃ 3.0.CO;2-O

19. Wrede B., Liu P., Wolff J.E.A. Chemotherapy improves the survival of patients with choroid plexus carcinoma: A meta-analysis of individual cases with choroid plexus tumors. J Neurooncol 2007; 85 (3): 345–51. DOI: 10.1007/s11060-007-9428-x

20. Valiakhmetova E.F., Papusha L.I., Yas'ko L.A., Drui A.E., Gorelyshev S.K., Karachunskii A.I. Khorioidkartsinomy u detei: obzor literatury. Rossiiskii zhurnal detskoi gematologii i onkologii 2020; 7 (1): 51–61. DOI: 10.21682/2311-1267-2020-7-1-51-61

21. Bahar M., Dhir A., Kordes U., Wolff J. Intercontinental multidisciplinary data collection and treatment optimization study for patients with choroid plexus tumor. Neuro-Oncology 2015; 17 (suppl 5): v186. DOI: 10.1093/neuonc/nov229.01