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Bilateral juvenile granulosa cell tumors in a 4-month-old dysmorphic infant. A clinical, histologic, and
Insights
Bilateral juvenile granulosa cell tumors in an infant presented as ovarian masses. These rare tumors in young children are treatable with conservative therapy, showing no recurrence after 16 months.
Area of Science:
- Gynecologic Oncology
- Pediatric Pathology
- Developmental Biology
Background:
- Juvenile granulosa cell tumors (JGCTs) are rare ovarian neoplasms typically affecting young girls.
- Bilateral involvement is uncommon, and presentation in infancy is exceptionally rare.
Observation:
- A 4-month-old infant presented with bilateral cystic ovarian masses and congenital anomalies including microcephaly and facial asymmetry.
- Serum alpha-fetoprotein was mildly elevated; imaging revealed well-defined masses without metastasis.
- Histopathology showed dense tumor cell proliferation with necrosis and microcyst formation, consistent with JGCTs.
Findings:
- Immunoperoxidase and ultrastructural studies confirmed the granulosa cell nature of the tumors.
- Despite initial findings, no alpha-fetoprotein was detected by immunoperoxidase, and no recurrence was observed during a 16-month follow-up.
- The infant's serum alpha-fetoprotein levels normalized post-treatment.
Implications:
- This case highlights JGCTs as a rare but treatable cause of ovarian masses in infants.
- The findings support the potential for conservative management of bilateral JGCTs in this age group.
- Early diagnosis and monitoring are crucial for successful outcomes in pediatric ovarian tumors.
Abstract:
Juvenile granulosa cell tumors were encountered within bilateral cystic ovarian masses in a 4-month-old infant. The child was the product of a consanguinous pregnancy, and manifested poor growth, relative microcephaly, facial asymmetry, and a malformed left ear. There was no history of gestational drug or hormone ingestion, and no evidence of abnormal endocrine activity after birth, however, the serum alpha-fetoprotein level was mildly elevated. The tumors were well defined by sonography and there was no evidence of metastasis. Histologically, a dense proliferation of round to oval tumor cells showed considerable individual cell necrosis and frequent microcyst formation. There was no evidence of luteinization and only mild nuclear pleomorphism. Immunoperoxidase study failed to reveal alpha-fetoprotein. Ultrastructural study supported the granulosa cell nature of the tumor, but a few cells contained bundles of intracytoplasmic filaments. There has been no evidence of recurrent disease during a 16-month follow-up period, and serial alpha-fetoprotein determinations have remained in the reference range. Comparison with two previously reported bilateral juvenile granulosa cell tumors suggests that this tumor occurs in young infants, and is amenable to conservative therapy.
