Skip to main content
Top
Published in: Langenbeck's Archives of Surgery 8/2010

01-11-2010 | Case Management & Clinical Consequences

Adult hepatoblastoma successfully treated with multimodal treatment

Authors: Shinji Nakamura, Masayuki Sho, Hiromichi Kanehiro, Toshihiro Tanaka, Kimihiko Kichikawa, Yoshiyuki Nakajima

Published in: Langenbeck's Archives of Surgery | Issue 8/2010

Login to get access

Abstract

Purpose

Adult hepatoblastoma is rare and the prognosis is extremely poor. Although surgery offers the only chance for cure, it often recurs even after complete resection. To date, no therapeutic strategy has been established. We report here a rare case of adult hepatoblastoma successfully treated with multimodal treatment.

Methods

A 25-year-old female who had been suffering from right hypochondriac pain was referred to our hospital. Preoperative diagnosis was adult hepatoblastoma. Huge liver tumor was initially deemed unresectable. An intensive chemotherapy including arterial chemoinfusion using interventional–radiological technique resulted in a remarkable tumor reduction. Furthermore, lung metastasis that had later appeared was completely treated with additional chemotherapy. Then she underwent curative surgery at 8 months after initial treatment.

Results

The pathological diagnosis was well-differentiated hepatoblastoma. The postoperative course was uneventful and she received short-course adjuvant chemotherapy. She survived for over 4 years after diagnosis.

Conclusions

To our knowledge, this is the first report demonstrating long-term survival of a patient with initially unresectable adult hepatoblastoma. Our new therapeutic strategy may provide new insights into the treatment of this fatal disease.
Literature
2.
go back to reference Sasaki F, Matsunaga T, Iwafuchi M, Hayashi Y, Ohkawa H, Ohira M, Okamatsu T, Sugito T, Tsuchida Y, Toyosaka A, Nagahara N, Nishihira H, Hata Y, Uchino J, Misugi K, Ohnuma N (2002) Outcome of hepatoblastoma treated with the JPLT-1 (Japanese Study Group for Pediatric Liver Tumor) Protocol-1: a report from the Japanese Study Group for Pediatric Liver Tumor. J Pediatr Surg 37:851–856CrossRefPubMed Sasaki F, Matsunaga T, Iwafuchi M, Hayashi Y, Ohkawa H, Ohira M, Okamatsu T, Sugito T, Tsuchida Y, Toyosaka A, Nagahara N, Nishihira H, Hata Y, Uchino J, Misugi K, Ohnuma N (2002) Outcome of hepatoblastoma treated with the JPLT-1 (Japanese Study Group for Pediatric Liver Tumor) Protocol-1: a report from the Japanese Study Group for Pediatric Liver Tumor. J Pediatr Surg 37:851–856CrossRefPubMed
3.
go back to reference Remes-Troche JM, Montano-Loza A, Meza-Junco J, Garcia-Leiva J, Torre-Delgadillo A (2006) Hepatoblastoma in adult age. A case report and literature review. Ann Hepatol 5:179–181PubMed Remes-Troche JM, Montano-Loza A, Meza-Junco J, Garcia-Leiva J, Torre-Delgadillo A (2006) Hepatoblastoma in adult age. A case report and literature review. Ann Hepatol 5:179–181PubMed
4.
go back to reference Yamazaki M, Ryu M, Okazumi S, Kondo F, Cho A, Okada T, Takayama W, Kawashima T, Furuki A, Hirata T (2004) Hepatoblastoma in an adult A case report and clinical review of literatures. Hepatol Res 30:182–188CrossRefPubMed Yamazaki M, Ryu M, Okazumi S, Kondo F, Cho A, Okada T, Takayama W, Kawashima T, Furuki A, Hirata T (2004) Hepatoblastoma in an adult A case report and clinical review of literatures. Hepatol Res 30:182–188CrossRefPubMed
5.
go back to reference Ahn HJ, Kwon KW, Choi YJ, Kim HJ, Hong SP, Oh D, Chung JS (1997) Mixed hepatoblastoma in an adult—a case report and literature review. J Korean Med Sci 12:369–373PubMed Ahn HJ, Kwon KW, Choi YJ, Kim HJ, Hong SP, Oh D, Chung JS (1997) Mixed hepatoblastoma in an adult—a case report and literature review. J Korean Med Sci 12:369–373PubMed
6.
go back to reference Ke HY, Chen JH, Jen YM, Yu JC, Hsieh CB, Chen CJ, Liu YC, Chen TW, Chan DC (2005) Ruptured hepatoblastoma with massive internal bleeding in an adult. World J Gastroenterol 11:6235–6237PubMed Ke HY, Chen JH, Jen YM, Yu JC, Hsieh CB, Chen CJ, Liu YC, Chen TW, Chan DC (2005) Ruptured hepatoblastoma with massive internal bleeding in an adult. World J Gastroenterol 11:6235–6237PubMed
7.
go back to reference Bortolasi L, Marchiori L, Dal Dosso I, Colombari R, Nicoli N (1996) Hepatoblastoma in adult age: a report of two cases. Hepatogastroenterology 43:1073–1078PubMed Bortolasi L, Marchiori L, Dal Dosso I, Colombari R, Nicoli N (1996) Hepatoblastoma in adult age: a report of two cases. Hepatogastroenterology 43:1073–1078PubMed
8.
go back to reference Harada T, Matsuo K, Kodama S, Higashihara H, Nakayama Y, Ikeda S (1995) Adult hepatoblastoma: case report and review of the literature. Aust NZ J Surg 65:686–688CrossRef Harada T, Matsuo K, Kodama S, Higashihara H, Nakayama Y, Ikeda S (1995) Adult hepatoblastoma: case report and review of the literature. Aust NZ J Surg 65:686–688CrossRef
9.
go back to reference Mukhopadhyay P, Kundu SS, Banerjee A, Mukherjee A (2007) Adult hepatoblastoma in a female Down’s. J Assoc Physicians India 55:242–243PubMed Mukhopadhyay P, Kundu SS, Banerjee A, Mukherjee A (2007) Adult hepatoblastoma in a female Down’s. J Assoc Physicians India 55:242–243PubMed
10.
go back to reference Kasper HU, Longerich T, Stippel DL, Kern MA, Drebber U, Schirmacher P (2005) Mixed hepatoblastoma in an adult. Arch Pathol Lab Med 129:234–237PubMed Kasper HU, Longerich T, Stippel DL, Kern MA, Drebber U, Schirmacher P (2005) Mixed hepatoblastoma in an adult. Arch Pathol Lab Med 129:234–237PubMed
11.
go back to reference Inagaki M, Yagi T, Urushihara N, Shima Y, Sadamori H, Takakura N, Tanaka N, Oda M (2001) Successfully resected hepatoblastoma in a young adult with chronic hepatitis B: report of a case. Eur J Gastroenterol Hepatol 13:981–984CrossRefPubMed Inagaki M, Yagi T, Urushihara N, Shima Y, Sadamori H, Takakura N, Tanaka N, Oda M (2001) Successfully resected hepatoblastoma in a young adult with chronic hepatitis B: report of a case. Eur J Gastroenterol Hepatol 13:981–984CrossRefPubMed
12.
go back to reference Reddy KS, Karak PK, Sharma S, Rao RC, Vijayaraghavan M, Sahni P (1997) Epithelial hepatoblastoma in a middle aged woman. Trop Gastroenterol 18:163–164PubMed Reddy KS, Karak PK, Sharma S, Rao RC, Vijayaraghavan M, Sahni P (1997) Epithelial hepatoblastoma in a middle aged woman. Trop Gastroenterol 18:163–164PubMed
13.
go back to reference Parada LA, Bardi G, Hallen M, Hagerstrand I, Tranberg KG, Mitelman F, Johansson B (1997) Cytogenetic abnormalities and clonal evolution in an adult hepatoblastoma. Am J Surg Pathol 21:1381–1386CrossRefPubMed Parada LA, Bardi G, Hallen M, Hagerstrand I, Tranberg KG, Mitelman F, Johansson B (1997) Cytogenetic abnormalities and clonal evolution in an adult hepatoblastoma. Am J Surg Pathol 21:1381–1386CrossRefPubMed
14.
go back to reference Kuniyasu H, Yasui W, Shimamoto F, Fujii K, Nakahara M, Asahara T, Dohi K, Tahara E (1996) Hepatoblastoma in an adult associated with c-met proto-oncogene imbalance. Pathol Int 46:1005–1010CrossRefPubMed Kuniyasu H, Yasui W, Shimamoto F, Fujii K, Nakahara M, Asahara T, Dohi K, Tahara E (1996) Hepatoblastoma in an adult associated with c-met proto-oncogene imbalance. Pathol Int 46:1005–1010CrossRefPubMed
15.
go back to reference Kacker LK, Khan EM, Gupta R, Kapoor VK, Pandey R, Gupta RK, Saraswat VA (1995) Hepatoblastoma in an adult with biliary obstruction and associated portal venous thrombosis. HPB Surg 9:47–49CrossRefPubMed Kacker LK, Khan EM, Gupta R, Kapoor VK, Pandey R, Gupta RK, Saraswat VA (1995) Hepatoblastoma in an adult with biliary obstruction and associated portal venous thrombosis. HPB Surg 9:47–49CrossRefPubMed
16.
go back to reference Misra GC, Mansharamani GG, Deewan R, Jayaraman G (1988) Mixed hepatoblastoma in a young adult. J Assoc Physicians India 36:169–170PubMed Misra GC, Mansharamani GG, Deewan R, Jayaraman G (1988) Mixed hepatoblastoma in a young adult. J Assoc Physicians India 36:169–170PubMed
17.
go back to reference Bhatnagar A, Pathania OP, Champakam NS (1987) Hepatoblastoma in an adult female. Indian J Gastroenterol 6:125–126PubMed Bhatnagar A, Pathania OP, Champakam NS (1987) Hepatoblastoma in an adult female. Indian J Gastroenterol 6:125–126PubMed
18.
go back to reference Schnater JM, Aronson DC, Plaschkes J, Perilongo G, Brown J, Otte JB, Brugieres L, Czauderna P, MacKinlay G, Vos A (2002) 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 94:1111–1120CrossRefPubMed Schnater JM, Aronson DC, Plaschkes J, Perilongo G, Brown J, Otte JB, Brugieres L, Czauderna P, MacKinlay G, Vos A (2002) 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 94:1111–1120CrossRefPubMed
19.
go back to reference Taguchi T (1994) Chemo-occlusion for the treatment of liver cancer. A new technique using degradable starch microspheres. Clin Pharmacokinet 26:275–291CrossRefPubMed Taguchi T (1994) Chemo-occlusion for the treatment of liver cancer. A new technique using degradable starch microspheres. Clin Pharmacokinet 26:275–291CrossRefPubMed
20.
go back to reference Furuse J, Ishii H, Satake M, Onaya H, Nose H, Mikami S, Sakai H, Mera K, Maru Y, Yoshino M (2003) Pilot study of transcatheter arterial chemoembolization with degradable starch microspheres in patients with hepatocellular carcinoma. Am J Clin Oncol 26:159–164CrossRefPubMed Furuse J, Ishii H, Satake M, Onaya H, Nose H, Mikami S, Sakai H, Mera K, Maru Y, Yoshino M (2003) Pilot study of transcatheter arterial chemoembolization with degradable starch microspheres in patients with hepatocellular carcinoma. Am J Clin Oncol 26:159–164CrossRefPubMed
21.
go back to reference Ninane J, Perilongo G, Stalens JP, Guglielmi M, Otte JB, Mancini A (1991) Effectiveness and toxicity of cisplatin and doxorubicin (PLADO) in childhood hepatoblastoma and hepatocellular carcinoma: a SIOP pilot study. Med Pediatr Oncol 19:199–203CrossRefPubMed Ninane J, Perilongo G, Stalens JP, Guglielmi M, Otte JB, Mancini A (1991) Effectiveness and toxicity of cisplatin and doxorubicin (PLADO) in childhood hepatoblastoma and hepatocellular carcinoma: a SIOP pilot study. Med Pediatr Oncol 19:199–203CrossRefPubMed
22.
go back to reference Ortega JA, Krailo MD, Haas JE, King DR, Ablin AR, Quinn JJ, Feusner J, Campbell JR, Lloyd DA, Cherlow J et al (1991) Effective treatment of unresectable or metastatic hepatoblastoma with cisplatin and continuous infusion doxorubicin chemotherapy: a report from the Childrens Cancer Study Group. J Clin Oncol 9:2167–2176PubMed Ortega JA, Krailo MD, Haas JE, King DR, Ablin AR, Quinn JJ, Feusner J, Campbell JR, Lloyd DA, Cherlow J et al (1991) Effective treatment of unresectable or metastatic hepatoblastoma with cisplatin and continuous infusion doxorubicin chemotherapy: a report from the Childrens Cancer Study Group. J Clin Oncol 9:2167–2176PubMed
23.
go back to reference Okuda K, Tanaka M, Shibata J, Ando E, Ogata T, Kinoshita H, Eriguchi N, Aoyagi S, Tanikawa K (1999) Hepatic arterial infusion chemotherapy with continuous low dose administration of cisplatin and 5-fluorouracil for multiple recurrence of hepatocellular carcinoma after surgical treatment. Oncol Rep 6:587–591PubMed Okuda K, Tanaka M, Shibata J, Ando E, Ogata T, Kinoshita H, Eriguchi N, Aoyagi S, Tanikawa K (1999) Hepatic arterial infusion chemotherapy with continuous low dose administration of cisplatin and 5-fluorouracil for multiple recurrence of hepatocellular carcinoma after surgical treatment. Oncol Rep 6:587–591PubMed
24.
go back to reference Ijichi O, Ishikawa S, Shinkoda Y, Tanabe T, Okamoto Y, Takamatsu H, Inomata Y, Kawano Y (2006) Response of heavily treated and relapsed hepatoblastoma in the transplanted liver to single-agent therapy with irinotecan. Pediatr Transplant 10:635–638CrossRefPubMed Ijichi O, Ishikawa S, Shinkoda Y, Tanabe T, Okamoto Y, Takamatsu H, Inomata Y, Kawano Y (2006) Response of heavily treated and relapsed hepatoblastoma in the transplanted liver to single-agent therapy with irinotecan. Pediatr Transplant 10:635–638CrossRefPubMed
Metadata
Title
Adult hepatoblastoma successfully treated with multimodal treatment
Authors
Shinji Nakamura
Masayuki Sho
Hiromichi Kanehiro
Toshihiro Tanaka
Kimihiko Kichikawa
Yoshiyuki Nakajima
Publication date
01-11-2010
Publisher
Springer-Verlag
Published in
Langenbeck's Archives of Surgery / Issue 8/2010
Print ISSN: 1435-2443
Electronic ISSN: 1435-2451
DOI
https://doi.org/10.1007/s00423-010-0630-5

Other articles of this Issue 8/2010

Langenbeck's Archives of Surgery 8/2010 Go to the issue