Pediatric transplantation · 2026
Incidental Hepatoblastoma in an Infant Who Underwent Liver Transplantation for Cryptogenic Liver Cirrhosis: A Case Report.
Клинический случайСтатьяНазвание и аннотация: Перевод в очереди
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Kojima Masato, Hattori Kensuke, Uchida Hajime, Komine Ryuji, Yanagi Yusuke, Fukuda Akinari, Haga Chizuko, Yoshioka Takako, Sakamoto Seisuke, Kasahara Mureo
Аннотация
BACKGROUND: Cryptogenic liver cirrhosis in infancy is rare, and its coexistence with hepatoblastoma (HB) has not been previously documented. While HB generally arises in morphologically normal livers, the incidental discovery of HB within cirrhotic livers is exceptional.
CASE PRESENTATION: We report a 6-month-old male infant with progressive cholestasis. Prenatal assessments indicated growth restriction and blood flow redistribution, and he was delivered preterm at 33 weeks and 6 days with a birth weight of 861 g (-4.1 SD). Despite extensive clinical, metabolic, infectious, and genetic evaluations, no etiology of cholestasis was identified. Liver function was deteriorated with a Child-Pugh score of 8 (Grade B) and a Pediatric End-Stage Liver Disease Score of 20. The patient underwent living donor liver transplantation at 6 months of age (body weight: 4.446 kg). Histopathological examination of the explanted liver revealed advanced fibrosis, and unexpectedly, a microscopic hepatoblastoma (approximately 1 mm, fetal type) was detected. The high level of serum AFP rapidly normalized postoperatively, and the infant was discharged in good condition.
DISCUSSION: This case highlights a unique presentation of infantile cryptogenic cirrhosis with incidental HB. Although HB typically develops in non-cirrhotic livers, hepatic injury and regenerative processes may create a permissive environment for tumorigenesis of HB in cirrhotic liver. The incidental finding of one millimeter HB emphasizes the importance of thorough histopathological evaluation of explanted livers. Moreover, careful postoperative surveillance with AFP monitoring and imaging remains essential to detect recurrence, as localized HB after complete resection carries a reported recurrence risk of approximately 6%.
CONCLUSION: This is the first reported case of incidental HB arising in infantile cryptogenic cirrhosis. It expands the clinical spectrum of HB in infantile cryptogenic cirrhosis.
CASE PRESENTATION: We report a 6-month-old male infant with progressive cholestasis. Prenatal assessments indicated growth restriction and blood flow redistribution, and he was delivered preterm at 33 weeks and 6 days with a birth weight of 861 g (-4.1 SD). Despite extensive clinical, metabolic, infectious, and genetic evaluations, no etiology of cholestasis was identified. Liver function was deteriorated with a Child-Pugh score of 8 (Grade B) and a Pediatric End-Stage Liver Disease Score of 20. The patient underwent living donor liver transplantation at 6 months of age (body weight: 4.446 kg). Histopathological examination of the explanted liver revealed advanced fibrosis, and unexpectedly, a microscopic hepatoblastoma (approximately 1 mm, fetal type) was detected. The high level of serum AFP rapidly normalized postoperatively, and the infant was discharged in good condition.
DISCUSSION: This case highlights a unique presentation of infantile cryptogenic cirrhosis with incidental HB. Although HB typically develops in non-cirrhotic livers, hepatic injury and regenerative processes may create a permissive environment for tumorigenesis of HB in cirrhotic liver. The incidental finding of one millimeter HB emphasizes the importance of thorough histopathological evaluation of explanted livers. Moreover, careful postoperative surveillance with AFP monitoring and imaging remains essential to detect recurrence, as localized HB after complete resection carries a reported recurrence risk of approximately 6%.
CONCLUSION: This is the first reported case of incidental HB arising in infantile cryptogenic cirrhosis. It expands the clinical spectrum of HB in infantile cryptogenic cirrhosis.