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Published in: Journal of Medical Case Reports 1/2011

Open Access 01-12-2011 | Case report

Precocious puberty in an infant with hepatoblastoma: a case report

Authors: Usama Al-Jumaily, Ibrahim Sammour, Fadi Al-Muhaisen, Fatenah Ajlouni, Iyad Sultan

Published in: Journal of Medical Case Reports | Issue 1/2011

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Abstract

Introduction

The syndrome of isosexual precocious puberty associated with primary malignant hepatic tumors is rare. All previously reported cases in the literature are old and prognosis was grim.

Case presentation

We present the case of a 15-month-old Asian male baby who presented with precocious puberty associated with hepatoblastoma. Serum concentrations of alpha-fetoprotein and free testosterone were elevated, as was beta human chorionic gonadotropin hormone. He was treated with six courses of chemotherapy and underwent surgery. His surface markers as well as free testosterone level returned to normal during therapy. The child has now been off therapy for 18 months with no evidence of tumor recurrence at follow-up.

Conclusion

Virilizing hepatoblastoma is rare and reported with poor outcome, but the development of new chemotherapeutic agents and complete surgical resection are promising.
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Literature
1.
go back to reference Braunstein GD, Bridson WE, Glass A, Hull EW, McIntire KR: In vivo and in vitro production of human chorionic gonadotropin and alpha-feteoprotein by a virilizing hepatoblastoma. J Clin Endocrinol Metab. 1972, 35 (6): 857-862. 10.1210/jcem-35-6-857.CrossRefPubMed Braunstein GD, Bridson WE, Glass A, Hull EW, McIntire KR: In vivo and in vitro production of human chorionic gonadotropin and alpha-feteoprotein by a virilizing hepatoblastoma. J Clin Endocrinol Metab. 1972, 35 (6): 857-862. 10.1210/jcem-35-6-857.CrossRefPubMed
2.
go back to reference Komura E, Matsumura T, Kato T, Tahara T, Tsunoda Y, Sawada T: Thrombopoietin in patients with hepatoblastoma. Stem Cells. 1998, 16 (5): 329-333. 10.1002/stem.160329.CrossRefPubMed Komura E, Matsumura T, Kato T, Tahara T, Tsunoda Y, Sawada T: Thrombopoietin in patients with hepatoblastoma. Stem Cells. 1998, 16 (5): 329-333. 10.1002/stem.160329.CrossRefPubMed
3.
go back to reference Arshad RR, Woo SY, Abbassi V, Hoy GR, Sinks LF: Virilizing hepatoblastoma: precocious sexual development and partial response of pulmonary metastases to cis-platinum. CA Cancer J Clin. 1982, 32 (5): 293-300. 10.3322/canjclin.32.5.293.CrossRefPubMed Arshad RR, Woo SY, Abbassi V, Hoy GR, Sinks LF: Virilizing hepatoblastoma: precocious sexual development and partial response of pulmonary metastases to cis-platinum. CA Cancer J Clin. 1982, 32 (5): 293-300. 10.3322/canjclin.32.5.293.CrossRefPubMed
4.
go back to reference Galifer RB, Sultan C, Margueritte G, Barneon G: Testosterone-producing hepatoblastoma in a 3-year-old boy with precocious puberty. J Pediatr Surg. 1985, 20 (6): 713-714. 10.1016/S0022-3468(85)80030-0.CrossRefPubMed Galifer RB, Sultan C, Margueritte G, Barneon G: Testosterone-producing hepatoblastoma in a 3-year-old boy with precocious puberty. J Pediatr Surg. 1985, 20 (6): 713-714. 10.1016/S0022-3468(85)80030-0.CrossRefPubMed
5.
go back to reference Heinrich UE, Bolkenius M, Daum R, Oppermann HC, Mehls O, Brandeis WE: Virilizing hepatoblastoma--significance of alpha-1-fetoprotein and human chorionic gonadotropin as tumor markers in diagnosis and follow-up. Eur J Pediatr. 1981, 135 (3): 313-317. 10.1007/BF00442110.CrossRefPubMed Heinrich UE, Bolkenius M, Daum R, Oppermann HC, Mehls O, Brandeis WE: Virilizing hepatoblastoma--significance of alpha-1-fetoprotein and human chorionic gonadotropin as tumor markers in diagnosis and follow-up. Eur J Pediatr. 1981, 135 (3): 313-317. 10.1007/BF00442110.CrossRefPubMed
6.
go back to reference Herlin T, Norup K, Storm K: Treatment of unresectable hepatoblastoma with cisplatin, vincristine and 5-fluorouracil. Eur J Pediatr. 1988, 147 (5): 514-515. 10.1007/BF00441977.CrossRefPubMed Herlin T, Norup K, Storm K: Treatment of unresectable hepatoblastoma with cisplatin, vincristine and 5-fluorouracil. Eur J Pediatr. 1988, 147 (5): 514-515. 10.1007/BF00441977.CrossRefPubMed
7.
go back to reference Kumar EV, Kumar L, Pathak IC, Dash RJ, Joshi VV: Clinical, hormonal and ultrastructure studies of a virilizing hepatoblastoma. Acta Paediatr Scand. 1978, 67 (3): 389-392. 10.1111/j.1651-2227.1978.tb16340.x.CrossRefPubMed Kumar EV, Kumar L, Pathak IC, Dash RJ, Joshi VV: Clinical, hormonal and ultrastructure studies of a virilizing hepatoblastoma. Acta Paediatr Scand. 1978, 67 (3): 389-392. 10.1111/j.1651-2227.1978.tb16340.x.CrossRefPubMed
8.
go back to reference Nakagawara A, Ikeda K, Tsuneyoshi M, Daimaru Y, Enjoji M, Watanabe I, Iwafuchi M, Sawada T: Hepatoblastoma producing both alpha-fetoprotein and human chorionic gonadotropin. Clinicopathologic analysis of four cases and a review of the literature. Cancer. 1985, 56 (7): 1636-1642. 10.1002/1097-0142(19851001)56:7<1636::AID-CNCR2820560729>3.0.CO;2-R.CrossRefPubMed Nakagawara A, Ikeda K, Tsuneyoshi M, Daimaru Y, Enjoji M, Watanabe I, Iwafuchi M, Sawada T: Hepatoblastoma producing both alpha-fetoprotein and human chorionic gonadotropin. Clinicopathologic analysis of four cases and a review of the literature. Cancer. 1985, 56 (7): 1636-1642. 10.1002/1097-0142(19851001)56:7<1636::AID-CNCR2820560729>3.0.CO;2-R.CrossRefPubMed
9.
go back to reference Flores F, Solano A, Rebeil R, Osuna JA, López M, Romero CA, García E: Isosexual precocious puberty in a male infant with hepatoblastoma. Rev Invest Clin. 1979, 31 (3): 251-255.PubMed Flores F, Solano A, Rebeil R, Osuna JA, López M, Romero CA, García E: Isosexual precocious puberty in a male infant with hepatoblastoma. Rev Invest Clin. 1979, 31 (3): 251-255.PubMed
10.
go back to reference Butenandt O, Knorr D, Hecker WC, Löhrs U: Precocious puberty in a boy with HcG-producing hepatoma. Case report. Helv Paediatr Acta. 1980, 35 (2): 155-163.PubMed Butenandt O, Knorr D, Hecker WC, Löhrs U: Precocious puberty in a boy with HcG-producing hepatoma. Case report. Helv Paediatr Acta. 1980, 35 (2): 155-163.PubMed
11.
go back to reference Morinaga S, Yamaguchi M, Watanabe I, Kasai M, Ojima M, Sasano N: An immunohistochemical study of hepatoblastoma producing human chorionic gonadotropin. Cancer. 1983, 51 (9): 1647-1652. 10.1002/1097-0142(19830501)51:9<1647::AID-CNCR2820510916>3.0.CO;2-0.CrossRefPubMed Morinaga S, Yamaguchi M, Watanabe I, Kasai M, Ojima M, Sasano N: An immunohistochemical study of hepatoblastoma producing human chorionic gonadotropin. Cancer. 1983, 51 (9): 1647-1652. 10.1002/1097-0142(19830501)51:9<1647::AID-CNCR2820510916>3.0.CO;2-0.CrossRefPubMed
12.
go back to reference Beach R, Betts P, Radford M, Millward-Sadler H: Production of human chorionic gonadotrophin by a hepatoblastoma resulting in precocious puberty. J Clin Pathol. 1984, 37 (7): 734-737. 10.1136/jcp.37.7.734.CrossRefPubMedPubMedCentral Beach R, Betts P, Radford M, Millward-Sadler H: Production of human chorionic gonadotrophin by a hepatoblastoma resulting in precocious puberty. J Clin Pathol. 1984, 37 (7): 734-737. 10.1136/jcp.37.7.734.CrossRefPubMedPubMedCentral
13.
go back to reference Navarro C, Corretger J, Sancho A, Rovira J, Morales L: Paraneoplasic precocious puberty report of a new case with hepatoblastoma and review of the literature. Cancer. 1985, 56 (7): 1725-1729. 10.1002/1097-0142(19851001)56:7<1725::AID-CNCR2820560743>3.0.CO;2-7.CrossRefPubMed Navarro C, Corretger J, Sancho A, Rovira J, Morales L: Paraneoplasic precocious puberty report of a new case with hepatoblastoma and review of the literature. Cancer. 1985, 56 (7): 1725-1729. 10.1002/1097-0142(19851001)56:7<1725::AID-CNCR2820560743>3.0.CO;2-7.CrossRefPubMed
14.
go back to reference Watanabe I, Yamaguchi M, Kasai M: Histologic characteristics of gonadotropin-producing hepatoblastoma: A survey of seven cases from Japan. J Pediatr Surg. 1987, 22 (5): 406-411. 10.1016/S0022-3468(87)80258-0.CrossRefPubMed Watanabe I, Yamaguchi M, Kasai M: Histologic characteristics of gonadotropin-producing hepatoblastoma: A survey of seven cases from Japan. J Pediatr Surg. 1987, 22 (5): 406-411. 10.1016/S0022-3468(87)80258-0.CrossRefPubMed
15.
go back to reference Bode HH, Schimana W, Swai B, Bode U: Sexual precocity associated with an abdominal tumor in an African boy. J Pediatr Endocrinol Metab. 2008, 21 (3): 275-277. 10.1515/JPEM.2008.21.3.275.CrossRefPubMed Bode HH, Schimana W, Swai B, Bode U: Sexual precocity associated with an abdominal tumor in an African boy. J Pediatr Endocrinol Metab. 2008, 21 (3): 275-277. 10.1515/JPEM.2008.21.3.275.CrossRefPubMed
Metadata
Title
Precocious puberty in an infant with hepatoblastoma: a case report
Authors
Usama Al-Jumaily
Ibrahim Sammour
Fadi Al-Muhaisen
Fatenah Ajlouni
Iyad Sultan
Publication date
01-12-2011
Publisher
BioMed Central
Published in
Journal of Medical Case Reports / Issue 1/2011
Electronic ISSN: 1752-1947
DOI
https://doi.org/10.1186/1752-1947-5-422

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