title: Epidural metastases from endodermal sinus tumor arising from benign sacral teratoma. Case report and review of the literature.
abstract: The recurrence of benign sacral teratomas is a small but significant possibility. Recurrence as an endodermal sinus tumor (EST) with epidural metastases, however, has not been previously reported. The authors describe a case of a mature sacrococcygeal teratoma in a 4-day-old female patient that recurred after 22 months as an EST with epidural metastases. The child presented with abdominal pain, urinary retention, and difficulty walking. On imaging, a large pelvic mass and an epidural spinal mass were revealed. The patient's alpha-fetal protein (AFP) level was 68,000 ng/ml. Her neurological examination was significant for 3/5 plantar and dorsiflexion strength bilaterally. The patient underwent L-3, L-4, and L-5 bilateral laminectomies followed by subtotal resection of the recurrent pelvic tumor. Pathological testing of samples of both the recurrent pelvic and the extradural spinal tumors led to a diagnosis of EST. The patient underwent four cycles of chemotherapy with normalization of her AFP level to 13 ng/ml. In the weeks that followed, her AFP level steadily rose again to 167 ng/ml. Follow-up imaging revealed no tumor recurrence. The patient underwent a second course of chemotherapy followed by two tandem courses of high-dose chemotherapy with autologous stem-cell rescue. Since completing this therapy the patient has been clinically stable with an AFP level of 1.3 ng/ml for 14 months. At the 1-year follow-up examination, her plantar and dorsiflexion strength had markedly improved to 4+/5 bilaterally. To the authors' knowledge, this is the first case of a mature sacrococcygeal tumor that recurred as an EST and caused spinal canal compromise.
authors.affiliation: Department of Neurological Surgery, University of Washington, Seattle, 98104-2499, USA. ericpete@u.washington.edu
authors.forename: Eric C
authors.initials: EC
authors.lastname: Peterson
authors.forename: Tord D
authors.initials: TD
authors.lastname: Alden
authors.forename: Kathy
authors.initials: K
authors.lastname: Patterson
authors.forename: Adam
authors.initials: A
authors.lastname: Lipson
authors.forename: Debra
authors.initials: D
authors.lastname: Friedman
authors.forename: Jacob
authors.initials: J
authors.lastname: Garcia
authors.forename: Anthony M
authors.initials: AM
authors.lastname: Avellino
mesh_terms: D000970:Antineoplastic Agents / Q000627:therapeutic use; D003050:Coccyx; D018240:Endodermal Sinus Tumor / Q000175:diagnosis / Q000188:drug therapy / Q000556:secondary*; D004824:Epidural Space; D005260:Female; D006801:Humans; D007231:Infant, Newborn; D008279:Magnetic Resonance Imaging; D009364:Neoplasm Recurrence, Local / Q000473:pathology* / Q000601:surgery; D019635:Neurosurgical Procedures; D010386:Pelvic Neoplasms / Q000473:pathology* / Q000601:surgery; D012447:Sacrum; D013125:Spinal Neoplasms / Q000097:blood / Q000175:diagnosis / Q000188:drug therapy / Q000556:secondary*; D013724:Teratoma / Q000473:pathology* / Q000601:surgery; D000509:alpha-Fetoproteins / Q000378:metabolism
chemical_list: D000970:Antineoplastic Agents; D000509:alpha-Fetoproteins