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Mature and immature extracranial teratomas in children: the UK Children's Cancer Study Group Experience
Jillian R Mann1, Elizabeth S Gray, Claire Thornton
1Department of Pediatric Oncology, Birmingham Children's Hospital, UK. jillmann@doctors.org.uk
Insights
Surgical resection is the primary treatment for pediatric mature teratoma (MT) and immature teratoma (IT). Chemotherapy (JEB) is effective for yolk sac tumor (YST) relapse but not for pure MT or IT.
Area of Science:
- Pediatric Oncology
- Surgical Oncology
- Pediatric Pathology
Background:
- Teratomas are germ cell tumors with diverse histology, including mature teratoma (MT) and immature teratoma (IT).
- Extracranial teratomas in children require specific management strategies based on histology and location.
- Understanding risk factors for relapse is crucial for optimizing treatment and improving outcomes.
Purpose of the Study:
- To delineate features, treatment approaches, and relapse risk factors for pediatric MT and IT.
- To inform future treatment strategies for these pediatric germ cell tumors.
- To analyze outcomes based on various prognostic indicators.
Main Methods:
- Retrospective analysis of 351 children (<16 years) with biopsy-proven extracranial MT/IT.
- Evaluation of surgical resection completeness, chemotherapy use (JEB for YST relapse), and follow-up data.
- Pathology review and assessment of prognostic features and outcomes.
Main Results:
- 351 patients: 227 MT, 124 IT. Common sites: ovary, sacrococcygeal, testis.
- 5-year event-free survival: 92.2% (MT), 85.9% (IT). 5-year overall survival: 99% (MT), 95.1% (IT).
- Poorer outcomes linked to incomplete resection, tumor rupture, nongonadal sites, young age, higher stage/grade, and gliomatosis peritonei. JEB effective for YST, not MT/IT.
Conclusions:
- Primary treatment for pediatric MT and IT is surgical excision.
- JEB chemotherapy is effective for yolk sac tumor (YST) recurrence but not for pure MT or IT.
- Adjuvant chemotherapy post-surgery for sacrococcygeal teratomas is not recommended.
Purpose:
The purpose of this article is to describe the features, treatment, and risk factors for relapse of children with mature teratoma (MT) and immature teratoma (IT) to assist future treatment plans.
Patients And Methods:
Patients were younger than 16 years of age and referred to the UK Children's Cancer Study Group centers with biopsy-proven extracranial MT and IT and no prior chemotherapy. Complete excision, with the coccyx in sacrococcygeal patients, and follow-up, including serum alpha-fetoprotein monitoring for early detection of malignant yolk sac tumor (YST) recurrence, were recommended. Carboplatin, etoposide, and bleomycin (JEB) were given for YST relapse, whereas relapsed MT and IT were treated at clinicians' discretion, usually surgically. Pathology was reviewed and treatments, outcome, and prognostic features assessed.
Results:
There were 351 patients, 227 with MT, 124 with IT. Tumor sites were: testis (n = 53), ovary (n = 130), sacrococcygeal region (n = 98), thorax (n = 23), and other (n = 47). Surgical resection was incomplete in 26% of MT and 40% of IT patients; 5-year event-free survival was 92.2% and 85.9%, respectively, and 5-year overall survival was 99% and 95.1%. Poorer outcome occurred with incomplete resection, tumor rupture, nongonadal site (particularly sacrococcygeal), young age, higher stage and grade, and gliomatosis peritonei, but not with cyst fluid aspiration/spillage, tumor enucleation, nodal gliomatosis, or microfoci of YST in the tumor (Heifetz lesions). JEB was effective for YST recurrence, but not for MT or IT.
Conclusion:
Treatment remains primarily surgical, with JEB chemotherapy for YST relapse. No definite response followed JEB for pure MT and IT. Adjuvant chemotherapy after surgery for sacrococcygeal patients is not advocated.
