Вопросы гематологии/онкологии и иммунопатологии в педиатрии. 2019; 18: 58-65
Рецидивы гепатобластомы с нормальным уровнем альфа-фетопротеина
https://doi.org/10.24287/1726-1708-2019-18-4-58-65Аннотация
Гепатобластома (ГБ) – наиболее частая злокачественная опухоль печени у детей. Значения показателя альфа-фетопротеина (АФП) используются для мониторинга ответа на противоопухолевую терапию и диагностики рецидивов заболевания. Развитие рецидивов ГБ при нормальных значениях АФП в тех случаях, когда в дебюте заболевания у пациентов отмечали повышенные значения данного онкомаркера, – редкая ситуация. В статье описаны случаи развития АФП-негативных рецидивов ГБ. В исследование включены два пациента с ГБ, инициально стратифицированные в группы высокого и стандартного риска, получавших лечение в условиях НМИЦ детской гематологии, онкологии и иммунологии им. Дмитрия Рогачева и Российского национального центра хирургии им. академика Б.В. Петровского, у которых в момент констатации рецидива заболевания отмечены нормальные уровни АФП, подтвержденные повторными измерениями. На парных образцах опухоли в дебюте и рецидиве заболевания проведено иммуногистохимическое исследование для оценки экспрессии АФП. Представленные клинические случаи свидетельствуют о том, что нормальные значения АФП при сомнительной клинической симптоматике не исключают рецидива ГБ. Возможность развития рецидивов при нормальных значениях АФП подчеркивает важность следования текущему плану диспансерного наблюдения, включающему не только оценку уровня АФП, но и рентгенографию органов грудной клетки и УЗИ органов брюшной полости. Родители пациентов дали согласие на использование информации о детях в научных исследованиях и публикациях.
Список литературы
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Pediatric Hematology/Oncology and Immunopathology. 2019; 18: 58-65
Hepatoblastoma relapses with a normal level of alpha-fetoprotein: report of two cases
https://doi.org/10.24287/1726-1708-2019-18-4-58-65Abstract
Hepatoblastoma (HB) is the most common malignant liver tumor in children. The level of alpha-fetoprotein (AFP) is used for monitoring the response to antitumor therapy and for diagnosing relapses. The occurrence of HB relapses with normal AFP levels in patients who had elevated levels of this tumor marker at disease onset is considered to be an uncommon situation. The aim of this study was to describe cases in which AFP-negative hepatoblastoma relapses developed. The study participants included two HB patients who were initially stratified into high-risk and standard-risk groups and who received treatment at the Dmitry Rogachev National Medical Research Center of Pediatric Hematology, Oncology and Immunology and the Petrovsky National Research Center of Surgery inMoscow. At the moment of relapse acknowledgement these patients had normal AFP levels, which was confirmed by serial measurements. Immunohistochemistry (IHC) tests were performed on paired samples of the tumor at disease onset and at disease relapse respectively to evaluate AFP expression. The presented clinical cases demonstrate that normal AFP levels, when accompanied by suspicious clinical symptoms, do not allow to exclude an HB relapse. The possibility of relapse with a normal AFP level reinforces the importance of following the current plan of screening tests, which includes not only an evaluation of AFP levels, but also other tests such as a chest X-ray and an abdominal ultrasound. Parents gave their consent to use information about the child in the article.
References
1. Howlader N., Noone A.M., Krapcho M., Neyman N., Aminou R., Waldron W., et al. SEER Cancer Statistics Review 2011; 1975–2008.
2. Birch L.J. Epidemiology of National Cancer Institute pediatric liver tumors. In book Pediatric liver tumors. Zimmermann A., Perilongo G. ed. Springer-Verlag, Berlin, 2011; 15–26.
3. Fuchs J., Rydzynski J., Von Schweinitz D., Bode U., Hecker H., Weinel P., et al. Study Committee of the Cooperative Pediatric Liver Tumor Study Hb 94 for the German Society for Pediatric Oncology and Hematology.Pretreatment prognostic factors and treatment results in children with hepatoblastoma: a report from the German Cooperative Pediatric Liver Tumor Study HB 94. Cancer 2002; 95 (1): 172–82.
4. Herzog C.E., Andrassy R.J., Eftekhari F. Childhood cancers: hepatoblastoma. Oncologist 2000; 5 (6): 445–53.
5. Rojas Y., Guillerman R.P., Zhang W., Vasudevan S.A., Nuchtern J.G., Thompson P.A. Relapse surveillance in AFP-positive hepatoblastoma: re-evaluating the role of imaging. Pediatric Radiology 2014; 44 (10): 1275–80.
6. Czauderna P., Lopez-Terrada D., Hiyama E., Häberle B., Malogolowkin M., Meyers R. Hepatoblastoma state of the art. Current Opinion In Pediatrics 2014; 26 (1): 19–28.
7. Perilongo G., Maibach R., Shafford E., Brugieres L., Brock P., Morland B., et al. Cisplatin versus Cisplatin plus Doxorubicin for Standard-Risk Hepatoblastoma. New England Journal of Medicine 2009; 361 (17), 1662–70.
8. Zsiros J., Brugieres L., Brock P., Roe-buck D., Maibach R., Zimmermann A., et al. Dose-dense cisplatin-based chemotherapy and surgery for children with high-risk hepatoblastoma (SIOPEL-4): a prospective, single-arm, feasibility study. The Lancet Oncology 2013; 14 (9): 834–42.
9. Semeraro M., Branchereau S., Maibach R., Zsiros J., Casanova M., Brock P. Relapses in hepatoblastoma patients: Clinical characteristics and outcome – Experience of the International Childhood Liver Tumour Strategy Group (SIOPEL). European Journal of Cancer 2013; 49 (4): 915–22.
10. Schneider D., Calaminus G., Göbel U. Diagnostic value of alpha1-fetoprotein and beta-human chorionic gonadotropin in infancy and childhood. Pediatric Hematology and Oncology 2001; 18 (1): 11–26.
11. Roebuck D., Aronson D., Clapuyt P., Czauderna P., de Ville de Goyet J., Gauthier F., et al. 2005 PRETEXT: a revised staging system for primary malignant liver tumours of childhood developed by the SIOPEL group. Pediatric Radiology 2006; 37 (2): 123–32.
12. Kachanov D.Yu., Shamanskaya T.V., Filin A.V., Moiseenko R.A., Tereshchenko G.V., Feoktistova E.V. i soavt. Dispansernoe nablyudenie patsientov c gepatoblastomoi. Rossiiskii zhurnal detskoi gematologii i onkologii 2014; 4: 79–89.
13. Katzenstein H., Furman W., Malogolowkin M., Krailo M., McCarville M., Towbin A., et al. Upfront window vincristine/irinotecan treatment of high-risk hepatoblastoma: A report from the Children's Oncology Group AHEP0731 study committee. Cancer 2017; 123 (12); 2360–7.
14. Kung F., Desai S., Dickerman J., Goorin A., Harris M., Inoue S., et al. Ifosfamide/Carboplatin/Etoposide (ICE) for Recurrent Malignant Solid Tumors of Childhood. Journal of Pediatric Hematology/Oncology 1995; 17 (3): 265–9.
15. McCarville M., Kao S. Imaging recom-mendations for malignant liver neoplasms in children. Pediatric Blood and Cancer 2005; 46 (1): 2–7.
16. Jassam N., Jones C., Briscoe T., Horner J. The hook effect: a need for constant vigilance. Annals of Clinical Biochemistry 2006; 43 (4): 314–7.
17. De Ioris M., Brugieres L., Zimmermann A., Keeling J., Brock P., Maibach R., et al. Hepatoblastoma with a low serum alpha-fetoprotein level at diagnosis: The SIOPEL group experience. European Journal of Cancer 2008; 44 (4): 545–50.
18. Maibach R., Roebuck D., Brugieres L., Capra M., Brock P., Dall’Igna P., et al. Prognostic stratification for children with hepatoblastoma: The SIOPEL experience. European Journal of Cancer 2012; 48 (10): 1543–9.
19. Trobaugh-Lotrario A., Tomlinson G., Finegold M., Gore L., Feusner J. Small cell undifferentiated variant of hepatoblastoma: Adverse clinical and molecular features similar to rhabdoid tumors. Pediatric Blood and Cancer 2009; 52 (3); 328–34.
20. Vokuhl C., Oyen F., Häberle B., von Schweinitz D., Schneppenheim R., Leuschner I. Small cell undifferentiated (SCUD) hepatoblastomas: all malignant rhabdoid tumors? Genes, Chromosomes and Cancer 2016; 55 (12): 925–31.
21. Hoshino N., Seki M., Kato M., Yoshida K., Sato Y., Nakazawa A., et al. Clonal evolution and integral analysis of hepatoblastoma. [abstract]. In: Proceedings of the 106th Annual Meeting of the American Association for Cancer Research; 2015 Apr 18–22; Philadelphia, PA. Philadelphia (PA): AACR; Cancer Res 2015; 75 (15 Suppl): Abstract nr 3294. DOI: 10.1158/1538-7445. AM 2015–3294.
