Journal of Child

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DOI :10.26650/jchild.2024.1477676   IUP :10.26650/jchild.2024.1477676    Full Text (PDF)

Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre

Esra Arslantaş, Ali Ayçiçek, Sibel Akpınar Tekgündüz, Saide Ertürk, Nazlı Gülsüm Akyel, Banu Yılmaz Özgüven, Erdem Kınacı, İlgin Özden

Objective: The aim of this study was to report the short-term outcomes of paediatric hepatoblastoma managed by hepatobiliary surgery and paediatric oncology from a single centre.

Methods: Children with hepatoblastoma diagnosed between May 2020 and February 2023 and treated comprehensively in a single centre, were retrospectively reviewed. Management was multidisciplinary and followed the SIOPEL protocols (SIOPEL III-IV).

Results: Eight paediatric patients with a median (range) age at diagnosis of 24 (5-68) months were included. The most common complaint was abdominal distension. There were no patients with PRETEXT stage I. Patients were graded as stage II (n=4), stage III (n=2), and stage IV (n=2). Half of the patients were classified in the standard risk group and the other half in the high-risk group. Chemotherapy was initiated in seven patients, and one was transferred directly to surgery for overt rupture. After neoadjuvant chemotherapy, complete response (CR) was not achieved in any patient, partial response (PR) was achieved in half (n=4), progressive disease and stable disease were present in one patient each, and one patient died. Four patients underwent radical hepatectomy with negative surgical margins after chemotherapy. The median (range) follow-up period was 18 (2-47) months. No recurrence was observed in any patient during follow-up, and the overall and event-free survival rates were 88% and 75%, respectively.

Conclusions: The collective work of surgery and paediatric oncology in management is essential to achieve optimal results.

Keywords: Hepatoblastoma, children, hepatectomy, chemotherapy

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References

  • Olson KSSaTA. Lanzkowsky’s manual of pediatric hematology and oncology. In: FISH J.D., ed., Hepatic Tumors. London, United Kingdom: Stacy Masucci: Elsevier Academic Press Publications; 2021:613-622. google scholar
  • Stocker JT. Hepatic tumors in children. Clin Liver Dis. 2001 Feb;5(1):259-81, viii-ix. doi: 10.1016/s1089-3261(05)70163-x. google scholar
  • Herzog CE, Andrassy RJ, Eftekhari F. Childhood cancers: hepatoblastoma. Oncologist. 2000;5(6):445-53. doi: 10.1634/ theoncologist.5-6-445. google scholar
  • Ranganathan S, Lopez-Terrada D, Alaggio R. Hepatoblastoma and Pediatric Hepatocellular Carcinoma: An Update. Pediatr Dev Pathol. 2020 Mar-Apr;23(2):79-95. doi: 10.1177/1093526619875228. google scholar
  • Pateva IB, Egler RA, Stearns DS. Hepatoblastoma in an 11-year-old: Case report and a review of the literature. Medicine (Baltimore). 2017 Jan;96(2):e5858. doi: 10.1097/MD.0000000000005858. google scholar
  • Venkatramani R, Spector LG, Georgieff M, Tomlinson G, Krailo M, Malogolowkin M, et al. Congenital abnormalities and hepatoblastoma: a report from the Children’s Oncology Group (COG) and the Utah Population Database (UPDB). Am J Med Genet A. 2014 Sep;164A(9):2250-5. doi: 10.1002/ajmg.a.36638. google scholar
  • Clericuzio CL, Chen E, McNeil DE, O’Connor T, Zackai EH, Medne L, et al. Serum alpha-fetoprotein screening for hepatoblastoma in children with Beckwith-Wiedemann syndrome or isolated hemihyperplasia. J Pediatr. 2003 Aug;143(2):270-2. doi: 10.1067/ S0022-3476(03)00306-8. google scholar
  • Harvey J, Clark S, Hyer W, Hadzic N, Tomlinson I, Hinds R. Germline APC mutations are not commonly seen in children with sporadic hepatoblastoma. J Pediatr Gastroenterol Nutr. 2008 Nov;47(5):675-7. doi: 10.1097/MPG.0b013e318174e808. google scholar
  • Brown J, Perilongo G, Shafford E, Keeling J, Pritchard J, Brock P, et al. Pretreatment prognostic factors for children with hepatoblastoma-- results from the International Society of Paediatric Oncology (SIOP) study SIOPEL 1. Eur J Cancer. 2000 Jul;36(11):1418-25. doi: 10.1016/s0959-8049(00)00074-5. google scholar
  • Horton JD, Lee S, Brown SR, Bader J, Meier DE. Survival trends in children with hepatoblastoma. Pediatr Surg Int. 2009 May;25(5):407-12. doi: 10.1007/s00383-009-2349-3. Epub 2009 Mar 24. PMID: 19308432. google scholar
  • Feng TC, Zai HY, Jiang W, Zhu Q, Jiang B, Yao L, et al. Survival and analysis of prognostic factors for hepatoblastoma: based on SEER database. Ann Transl Med. 2019 Oct;7(20):555. doi: 10.21037/ atm.2019.09.76. google scholar
  • Black CT, Cangir A, Choroszy M, Andrassy RJ. Marked response to preoperative high-dose cis-platinum in children with unresectable hepatoblastoma. J Pediatr Surg. 1991 Sep;26(9):1070-3. doi: 10.1016/0022-3468(91)90676-k. google scholar
  • Kutluk T, Yeşilipek A. Pediatric Cancer Registry in Turkey 20092021 (TPOG & TPHD). Journal of Clinical Oncology 2022 40:16_ suppl, e22020-e22020. google scholar
  • Perilongo G, Shafford E, Plaschkes J. Liver Tumour Study Group of the International Society of Paediatric Oncology. SIOPEL trials using preoperative chemotherapy in hepatoblastoma. Lancet Oncol. 2000 Oct;1:94-100. doi: 10.1016/s1470-2045(00)00018-8. google scholar
  • Malogolowkin MH, Katzenstein H, Krailo MD, Chen Z, Bowman L, Reynolds M, et al. Intensified platinum therapy is an ineffective strategy for improving outcome in pediatric patients with advanced hepatoblastoma. J Clin Oncol. 2006;24(18):2879-2884. doi: 10.1200/JCO.2005.02.6013. google scholar
  • Tayfun Küpesiz F, Akınel AN, Akbaş H, Sivrice Ç, Tüysüz Kintrup G, Karagüzel G, et al. E. Multidisciplinary Management of Pediatric Hepatoblastoma: A 20-Year Single-Center Experience. Turk J Gastroenterol. 2022 Dec;33(12):1069-1078. doi: 10.5152/ tjg.2022.21827. google scholar
  • Roebuck DJ, 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. Pediatr Radiol 2007;37(2):123-132; quiz 249-250. doi: 10.1007/s00247-006-0361-5. Epub 2006 Dec 21. google scholar
  • Schnater JM, Aronson DC, Plaschkes J, Perilongo G, Brown J, Otte JB, et al. Surgical view of the treatment of patients with hepatoblastoma: results from the first prospective trial of the International Society of Pediatric Oncology Liver Tumor Study Group. Cancer 2002 Feb 15;94(4):1111-20. google scholar
  • Meyers RL, Rowland JR, Krailo M, Chen Z, Katzenstein HM, Malogolowkin MH. Predictive power of pretreatment prognostic factors in children with hepatoblastoma: a report from the Children’s Oncology Group. Pediatr Blood Cancer. 2009;53(6):1016-1022. doi: 10.1002/pbc.22088. google scholar
  • Agarwala S, Gupta A, Bansal D, Vora T, Prasad M, Arora B, et al. Management of hepatoblastoma: ICMR consensus document. Indian J Pediatr. 2017;84(6): 456-464. doi: 10.1007/s12098-017-2301-9. google scholar
  • Fuchs J, Rydzynski J, Von Schweinitz D, Bode U, Hecker H, Weinel P, et al. 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):172-182. doi: 10.1002/cncr.10632. google scholar
  • Hishiki T. Current therapeutic strategies for childhood hepatic tumors: surgical and interventional treatments for hepatoblastoma. Int J Clin Oncol 2013;18:962-8. doi: 10.1007/ s10147-013-0625-7. google scholar
  • Yu W, Liu X, Li J, Xi Z, Jin J, Huang H, et al. A single-center retrospective analysis of childhood hepatoblastoma in China. Gland Surg. 2020 Oct;9(5):1502-1512. doi: 10.21037/gs-20-710. google scholar
  • Shanmugam N, Scott JX, Kumar V, Vij M, Ramachandran P, Narasimhan G, et al. Multidisciplinary management of hepatoblastoma in children: Experience from a developing country. Pediatr Blood Cancer. 2017 Mar;64(3). doi: 10.1002/pbc.26249. google scholar
  • Kutluk T, Yalçın B, Ekinci S, Kale G, Akyüz C, Aydın B, et al. Primary liver tumors in children: Hacettepe experience. Turk J Pediatr. 2014 Jan-Feb;56(1):1-10. google scholar
  • Zsiros J, Brugieres L, Brock P, Roebuck D, Maibach R, Zimmermann A, et al; International Childhood Liver Tumours Strategy Group (SIOPEL). Dose-dense cisplatin-based chemotherapy and surgery for children with high-risk hepatoblastoma (SIOPEL-4): a prospective, single-arm, feasibility study. Lancet Oncol. 2013 Aug;14(9):834-42. doi: 10.1016/S1470-2045(13)70272-9. google scholar
  • Rammohan A, Rela M, Kumar GV, Scott JX, Shanmugam N, Reddy MS, et al. Outcomes for high-risk hepatoblastoma in a resource-challenged setting. BJS Open. 2020 Aug;4(4):630-636. doi: 10.1002/bjs5.50297. google scholar
  • Okur MH, Yankol Y, Cimşit B, Mecit N, Ertuğrul G, Kanmaz T, et al. Liver Transplant in Children with Hepatoblastoma. Exp Clin Transplant. 2019 Oct;17(5):644-647. doi: 10.6002/ect.2016.0110. google scholar
  • Lovvorn HN 3rd, Ayers D, Zhao Z, Hilmes M, Prasad P, Shinall MC Jr, et al. Defining hepatoblastoma responsiveness to induction therapy as measured by tumor volume and serum alpha-fetoprotein kinetics. J Pediatr Surg. 2010 Jan;45(1):121-8; discussion 129. doi: 10.1016/j.jpedsurg.2009.10.023. google scholar
  • Srinivasan S, Prasad M, Parambil BC, Shrimal A, Gollamudi VRM, Subramani V, et al. Treatment outcomes and prognostic factors in children with hepatoblastoma using a risk-stratified approach. Pediatr Blood Cancer. 2023 Jul;70(7):e30302. doi: 10.1002/ pbc.30302. google scholar
  • Meyers RL, Rowland JR, Krailo M, Chen Z, Katzenstein HM, Malogolowkin MH. Predictive power of pretreatment prognostic factors in children with hepatoblastoma: a report from the Children’s Oncology Group. Pediatric Blood Cancer 2009; 53:1016-22. doi: 10.1002/pbc.22088. google scholar
  • Brown J, Perilongo G, Shafford E, Keeling J, Pritchard J, Brock Pet al. Pretreatment prognostic factors for children with hepatoblastoma e results from the International Society of Paediatric Oncology (SIOP) study SIOPEL 1. Eur J Cancer (Oxford, England 1990) 2000;36:1418-25. doi: 10.1016/s0959-8049(00)00074-5. google scholar

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APA

Arslantaş, E., Ayçiçek, A., Akpınar Tekgündüz, S., Ertürk, S., Akyel, N.G., Yılmaz Özgüven, B., Kınacı, E., & Özden, İ. (2024). Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre. Journal of Child, 24(2), 70-77. https://doi.org/10.26650/jchild.2024.1477676


AMA

Arslantaş E, Ayçiçek A, Akpınar Tekgündüz S, Ertürk S, Akyel N G, Yılmaz Özgüven B, Kınacı E, Özden İ. Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre. Journal of Child. 2024;24(2):70-77. https://doi.org/10.26650/jchild.2024.1477676


ABNT

Arslantaş, E.; Ayçiçek, A.; Akpınar Tekgündüz, S.; Ertürk, S.; Akyel, N.G.; Yılmaz Özgüven, B.; Kınacı, E.; Özden, İ. Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre. Journal of Child, [Publisher Location], v. 24, n. 2, p. 70-77, 2024.


Chicago: Author-Date Style

Arslantaş, Esra, and Ali Ayçiçek and Sibel Akpınar Tekgündüz and Saide Ertürk and Nazlı Gülsüm Akyel and Banu Yılmaz Özgüven and Erdem Kınacı and İlgin Özden. 2024. “Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre.” Journal of Child 24, no. 2: 70-77. https://doi.org/10.26650/jchild.2024.1477676


Chicago: Humanities Style

Arslantaş, Esra, and Ali Ayçiçek and Sibel Akpınar Tekgündüz and Saide Ertürk and Nazlı Gülsüm Akyel and Banu Yılmaz Özgüven and Erdem Kınacı and İlgin Özden. “Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre.” Journal of Child 24, no. 2 (Nov. 2024): 70-77. https://doi.org/10.26650/jchild.2024.1477676


Harvard: Australian Style

Arslantaş, E & Ayçiçek, A & Akpınar Tekgündüz, S & Ertürk, S & Akyel, NG & Yılmaz Özgüven, B & Kınacı, E & Özden, İ 2024, 'Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre', Journal of Child, vol. 24, no. 2, pp. 70-77, viewed 15 Nov. 2024, https://doi.org/10.26650/jchild.2024.1477676


Harvard: Author-Date Style

Arslantaş, E. and Ayçiçek, A. and Akpınar Tekgündüz, S. and Ertürk, S. and Akyel, N.G. and Yılmaz Özgüven, B. and Kınacı, E. and Özden, İ. (2024) ‘Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre’, Journal of Child, 24(2), pp. 70-77. https://doi.org/10.26650/jchild.2024.1477676 (15 Nov. 2024).


MLA

Arslantaş, Esra, and Ali Ayçiçek and Sibel Akpınar Tekgündüz and Saide Ertürk and Nazlı Gülsüm Akyel and Banu Yılmaz Özgüven and Erdem Kınacı and İlgin Özden. “Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre.” Journal of Child, vol. 24, no. 2, 2024, pp. 70-77. [Database Container], https://doi.org/10.26650/jchild.2024.1477676


Vancouver

Arslantaş E, Ayçiçek A, Akpınar Tekgündüz S, Ertürk S, Akyel NG, Yılmaz Özgüven B, Kınacı E, Özden İ. Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre. Journal of Child [Internet]. 15 Nov. 2024 [cited 15 Nov. 2024];24(2):70-77. Available from: https://doi.org/10.26650/jchild.2024.1477676 doi: 10.26650/jchild.2024.1477676


ISNAD

Arslantaş, Esra - Ayçiçek, Ali - Akpınar Tekgündüz, Sibel - Ertürk, Saide - Akyel, NazlıGülsüm - Yılmaz Özgüven, Banu - Kınacı, Erdem - Özden, İlgin. “Multidisciplinary Management of Hepatoblastoma: Three Years of Experience at a Single Centre”. Journal of Child 24/2 (Nov. 2024): 70-77. https://doi.org/10.26650/jchild.2024.1477676



TIMELINE


Submitted03.05.2024
Accepted06.06.2024
Published Online15.08.2024

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