Virilizing hepatoblastomas: A systematic review and meta-analysis of case reports

Objectives: Hepatoblastoma (HB) has been rarely reported to express human choriogonadotropins (hCG), causing virilization and isosexual precocious puberty in boys. The aim of this systematic review is to identify and summarize the published reports on virilizing HBs. Material and Methods: A Google scholar and PubMed search was conducted on January 9, 2024, using the terms “hepatoblastoma” AND “precocious puberty” OR “virilizing hepatoblastoma.” After back-referencing (snowballing), publications on virilizing HBs in English language were included in the study. The year of publication, study design, nationality, age, presentation, diagnostic modality, treatment, and outcomes were tabulated and the data summarized. Categorical variables were presented as percentages/proportions while continuous variables were summarized as mean and standard deviation. No risk of bias assessment was carried out as the published articles are all case reports. Results: Thirty-four case reports (43 children) on virilizing HB available were summarized. All cases reported were boys with mean age of 2.29 ± 1.61 years at presentation and average symptom duration of 4.1 ± 3.54 months. Serum β-hCG was elevated in 32 patients (103.97; range: 42–562 mIU/mL) and serum alpha-fetoprotein was elevated in 22 patients (40500; range: 2503.9–182700 ng/mL). Nine children (20.93%) had metastatic tumor at presentation. The most common subtype was mixed epithelial-mesenchymal (12/23; 52.17%). Four (9.76%) children developed local recurrences and 11 (26.83%) developed metastases postoperatively. After a median follow-up of 10 (3.75–16.5) months, 19/39 children (48.72%) survived. Conclusion: Although hCG-secreting HB is rare, it can be treated the same way as other HB patients as they have comparable outcomes.