Outcomes for patients with congenital hepatoblastoma

Angela D. Trobaugh-Lotrario, Barbara H. Chaiyachati, Rebecka L. Meyers, Beate Häberle, Gail E. Tomlinson, Howard M. Katzenstein, Marcio Malogolowkin, Dietrich von Schweinitz, Mark Krailo, James H. Feusner

Research output: Contribution to journalArticle

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Abstract

Background: Congenital hepatoblastoma, diagnosed in the first month of life, has been reported to have a poor prognosis; however, a comprehensive evaluation of this entity is lacking. Procedure: We retrospectively reviewed two patients from the senior authors' personal series and 25 cases identified in the databases of several multicenter group studies (INT-0098, P9645, 881, P9346, HB 89, HB94, and HB 99). We compared this series with cases of congenital hepatoblastoma previously published in the literature. Results: The 3-year survival in our case series was 86% (18/21) with a follow-up of 44-230 months (median 85.5 months). Presentation and treatment were not substantially different from hepatoblastoma cohorts unselected for age. Survival was comparable to the reported disease free survival for a similar cohort of hepatoblastoma patients unselected for age between 1986 and 2002 (82.5%) [von Schweinitz et al., Eur J Cancer 1997; 33:1243-1249]. The 2-year survival of cases reported in the literature was 0% (0/9) and 42% (10/24) for patients reported before and after 1990, respectively. Conclusions: Congenital hepatoblastoma does not appear to confer a worse prognosis. The improved survival of our current series of patients, collected from the past 20 years of German and American multicenter trials and personal series, suggests that the outcome of hepatoblastoma at this young age is much better than has been historically reported. More rigorous analysis should be conducted in future multicenter trials. It is possible that congenital hepatoblastoma should be treated like all other patients with hepatoblastoma provided that the child is stable enough to proceed with surgery and chemotherapy. Pediatr Blood Cancer 2013;60:1817-1825.

Original languageEnglish (US)
Pages (from-to)1817-1825
Number of pages9
JournalPediatric Blood and Cancer
Volume60
Issue number11
DOIs
StatePublished - Nov 1 2013
Externally publishedYes

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Hepatoblastoma
Multicenter Studies
Survival
Disease-Free Survival
Neoplasms
Databases
Drug Therapy

Keywords

  • Congenital
  • Hepatoblastoma
  • Infant
  • Neonatal

ASJC Scopus subject areas

  • Oncology
  • Pediatrics, Perinatology, and Child Health
  • Hematology

Cite this

Trobaugh-Lotrario, A. D., Chaiyachati, B. H., Meyers, R. L., Häberle, B., Tomlinson, G. E., Katzenstein, H. M., ... Feusner, J. H. (2013). Outcomes for patients with congenital hepatoblastoma. Pediatric Blood and Cancer, 60(11), 1817-1825. https://doi.org/10.1002/pbc.24655

Outcomes for patients with congenital hepatoblastoma. / Trobaugh-Lotrario, Angela D.; Chaiyachati, Barbara H.; Meyers, Rebecka L.; Häberle, Beate; Tomlinson, Gail E.; Katzenstein, Howard M.; Malogolowkin, Marcio; von Schweinitz, Dietrich; Krailo, Mark; Feusner, James H.

In: Pediatric Blood and Cancer, Vol. 60, No. 11, 01.11.2013, p. 1817-1825.

Research output: Contribution to journalArticle

Trobaugh-Lotrario, AD, Chaiyachati, BH, Meyers, RL, Häberle, B, Tomlinson, GE, Katzenstein, HM, Malogolowkin, M, von Schweinitz, D, Krailo, M & Feusner, JH 2013, 'Outcomes for patients with congenital hepatoblastoma', Pediatric Blood and Cancer, vol. 60, no. 11, pp. 1817-1825. https://doi.org/10.1002/pbc.24655
Trobaugh-Lotrario AD, Chaiyachati BH, Meyers RL, Häberle B, Tomlinson GE, Katzenstein HM et al. Outcomes for patients with congenital hepatoblastoma. Pediatric Blood and Cancer. 2013 Nov 1;60(11):1817-1825. https://doi.org/10.1002/pbc.24655
Trobaugh-Lotrario, Angela D. ; Chaiyachati, Barbara H. ; Meyers, Rebecka L. ; Häberle, Beate ; Tomlinson, Gail E. ; Katzenstein, Howard M. ; Malogolowkin, Marcio ; von Schweinitz, Dietrich ; Krailo, Mark ; Feusner, James H. / Outcomes for patients with congenital hepatoblastoma. In: Pediatric Blood and Cancer. 2013 ; Vol. 60, No. 11. pp. 1817-1825.
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abstract = "Background: Congenital hepatoblastoma, diagnosed in the first month of life, has been reported to have a poor prognosis; however, a comprehensive evaluation of this entity is lacking. Procedure: We retrospectively reviewed two patients from the senior authors' personal series and 25 cases identified in the databases of several multicenter group studies (INT-0098, P9645, 881, P9346, HB 89, HB94, and HB 99). We compared this series with cases of congenital hepatoblastoma previously published in the literature. Results: The 3-year survival in our case series was 86{\%} (18/21) with a follow-up of 44-230 months (median 85.5 months). Presentation and treatment were not substantially different from hepatoblastoma cohorts unselected for age. Survival was comparable to the reported disease free survival for a similar cohort of hepatoblastoma patients unselected for age between 1986 and 2002 (82.5{\%}) [von Schweinitz et al., Eur J Cancer 1997; 33:1243-1249]. The 2-year survival of cases reported in the literature was 0{\%} (0/9) and 42{\%} (10/24) for patients reported before and after 1990, respectively. Conclusions: Congenital hepatoblastoma does not appear to confer a worse prognosis. The improved survival of our current series of patients, collected from the past 20 years of German and American multicenter trials and personal series, suggests that the outcome of hepatoblastoma at this young age is much better than has been historically reported. More rigorous analysis should be conducted in future multicenter trials. It is possible that congenital hepatoblastoma should be treated like all other patients with hepatoblastoma provided that the child is stable enough to proceed with surgery and chemotherapy. Pediatr Blood Cancer 2013;60:1817-1825.",
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AU - Tomlinson, Gail E.

AU - Katzenstein, Howard M.

AU - Malogolowkin, Marcio

AU - von Schweinitz, Dietrich

AU - Krailo, Mark

AU - Feusner, James H.

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N2 - Background: Congenital hepatoblastoma, diagnosed in the first month of life, has been reported to have a poor prognosis; however, a comprehensive evaluation of this entity is lacking. Procedure: We retrospectively reviewed two patients from the senior authors' personal series and 25 cases identified in the databases of several multicenter group studies (INT-0098, P9645, 881, P9346, HB 89, HB94, and HB 99). We compared this series with cases of congenital hepatoblastoma previously published in the literature. Results: The 3-year survival in our case series was 86% (18/21) with a follow-up of 44-230 months (median 85.5 months). Presentation and treatment were not substantially different from hepatoblastoma cohorts unselected for age. Survival was comparable to the reported disease free survival for a similar cohort of hepatoblastoma patients unselected for age between 1986 and 2002 (82.5%) [von Schweinitz et al., Eur J Cancer 1997; 33:1243-1249]. The 2-year survival of cases reported in the literature was 0% (0/9) and 42% (10/24) for patients reported before and after 1990, respectively. Conclusions: Congenital hepatoblastoma does not appear to confer a worse prognosis. The improved survival of our current series of patients, collected from the past 20 years of German and American multicenter trials and personal series, suggests that the outcome of hepatoblastoma at this young age is much better than has been historically reported. More rigorous analysis should be conducted in future multicenter trials. It is possible that congenital hepatoblastoma should be treated like all other patients with hepatoblastoma provided that the child is stable enough to proceed with surgery and chemotherapy. Pediatr Blood Cancer 2013;60:1817-1825.

AB - Background: Congenital hepatoblastoma, diagnosed in the first month of life, has been reported to have a poor prognosis; however, a comprehensive evaluation of this entity is lacking. Procedure: We retrospectively reviewed two patients from the senior authors' personal series and 25 cases identified in the databases of several multicenter group studies (INT-0098, P9645, 881, P9346, HB 89, HB94, and HB 99). We compared this series with cases of congenital hepatoblastoma previously published in the literature. Results: The 3-year survival in our case series was 86% (18/21) with a follow-up of 44-230 months (median 85.5 months). Presentation and treatment were not substantially different from hepatoblastoma cohorts unselected for age. Survival was comparable to the reported disease free survival for a similar cohort of hepatoblastoma patients unselected for age between 1986 and 2002 (82.5%) [von Schweinitz et al., Eur J Cancer 1997; 33:1243-1249]. The 2-year survival of cases reported in the literature was 0% (0/9) and 42% (10/24) for patients reported before and after 1990, respectively. Conclusions: Congenital hepatoblastoma does not appear to confer a worse prognosis. The improved survival of our current series of patients, collected from the past 20 years of German and American multicenter trials and personal series, suggests that the outcome of hepatoblastoma at this young age is much better than has been historically reported. More rigorous analysis should be conducted in future multicenter trials. It is possible that congenital hepatoblastoma should be treated like all other patients with hepatoblastoma provided that the child is stable enough to proceed with surgery and chemotherapy. Pediatr Blood Cancer 2013;60:1817-1825.

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