Originally published as JCO Early Release 10.1200/JCO.2008.16.0622 on June 9 2008

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Mature and Immature Extracranial Teratomas in Children: The UK Children's Cancer Study Group Experience

Jillian R. Mann, Elizabeth S. Gray, Claire Thornton, Faro Raafat, Kathleen Robinson, Gary S. Collins, Peter Gornall, Simon N. Huddart, Juliet P. Hale, Anthony Oakhill

From the Departments of Pediatric Oncology, Histopathology, Pediatric Surgery, Birmingham Children's Hospital, Birmingham; Department of Pathology, Medical School, University of Aberdeen; Department of Histopathology, Institute of Clinical Sciences, Belfast; The Children's Cancer and Leukaemia Group Data Centre and University of Leicester, Leicester; Centre for Statistics in Medicine, University of Oxford, Oxford; University Hospital of Wales, Cardiff; Department of Pediatric Oncology, Royal Victoria Infirmary, Newcastle-upon Tyne; and the Department of Pediatric Oncology, Royal Hospital for Children, Bristol, United Kingdom
Deceased

Corresponding author: Jillian R. Mann, FRCPCH, Oncology Department, Birmingham Children's Hospital, Steelhouse Lane, Birmingham, B4 6NH, United Kingdom; e-mail: jillmann{at}doctors.org.uk

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.

published online ahead of print at www.jco.org on June 9, 2008

Supported by Cancer Research UK (for data management).

Presented in part at the Germ Cell Tumours IV meeting, in Leeds, United Kingdom, November 1998.

The UK Children's Cancer Study Group is now the Children's Cancer and Leukaemia Group.

Authors’ disclosures of potential conflicts of interest and author contributions are found at the end of this article.

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