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Immune-mediated encephalitis and virilization in association with a mature cystic ovarian teratoma in an adolescent girl.
Horm Res. 2009; 72(4):252-6.HR

Abstract

BACKGROUND

Mature cystic teratomas are the most common form of ovarian tumor in children and adolescents. These tumors are mostly benign and non-secreting. Virilization from an ovarian teratoma is exceptionally rare in pediatrics. Equally rare is the association of ovarian teratomas with auto-immune encephalitis.

METHODS

We describe the case of a 15-year-old girl with menstrual abnormalities and virilization, who had a past medical history of encephalitis of an unknown etiology 16 months prior to presentation.

RESULTS

Endocrine evaluation revealed an elevated serum testosterone and 17-hydroxy progesterone. A large left ovarian tumor was seen on a CT scan. Surgical excision revealed a mature cystic teratoma containing 6 liters of clear fluid with high androgen levels. Antibodies to the N-methyl-D-aspartate receptor of the hippocampus were detected in pre-operatively archived serum, but undetectable 6 months postoperatively. Immunohistochemistry studies on the tumor sections revealed that the antibodies in the patient's serum reacted with areas of the tumor expressing the N-methyl-D-aspartate receptor. Postoperatively, the patient's menstrual cycles became regular and her behavioral problems resolved. Her testosterone levels fell precipitously as well.

CONCLUSION

Both virilizing mature cystic teratomas and teratoma-associated encephalitis are extremely rare in the pediatric population. We report on the first instance of these 2 rare entities occurring in the same patient.

Authors+Show Affiliations

Division of Pediatric Endocrinology, Children's Hospital at Montefiore, Bronx, New York, USA. arunapoduval@hotmail.comNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

19786797

Citation

Poduval, A, et al. "Immune-mediated Encephalitis and Virilization in Association With a Mature Cystic Ovarian Teratoma in an Adolescent Girl." Hormone Research, vol. 72, no. 4, 2009, pp. 252-6.
Poduval A, Antal Z, Lee T, et al. Immune-mediated encephalitis and virilization in association with a mature cystic ovarian teratoma in an adolescent girl. Horm Res. 2009;72(4):252-6.
Poduval, A., Antal, Z., Lee, T., Bar, A., Dalmau, J., & Muzumdar, R. (2009). Immune-mediated encephalitis and virilization in association with a mature cystic ovarian teratoma in an adolescent girl. Hormone Research, 72(4), 252-6. https://doi.org/10.1159/000236087
Poduval A, et al. Immune-mediated Encephalitis and Virilization in Association With a Mature Cystic Ovarian Teratoma in an Adolescent Girl. Horm Res. 2009;72(4):252-6. PubMed PMID: 19786797.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Immune-mediated encephalitis and virilization in association with a mature cystic ovarian teratoma in an adolescent girl. AU - Poduval,A, AU - Antal,Z, AU - Lee,T, AU - Bar,A, AU - Dalmau,J, AU - Muzumdar,R, Y1 - 2009/09/29/ PY - 2008/05/19/received PY - 2008/11/21/accepted PY - 2009/9/30/entrez PY - 2009/9/30/pubmed PY - 2009/12/16/medline SP - 252 EP - 6 JF - Hormone research JO - Horm Res VL - 72 IS - 4 N2 - BACKGROUND: Mature cystic teratomas are the most common form of ovarian tumor in children and adolescents. These tumors are mostly benign and non-secreting. Virilization from an ovarian teratoma is exceptionally rare in pediatrics. Equally rare is the association of ovarian teratomas with auto-immune encephalitis. METHODS: We describe the case of a 15-year-old girl with menstrual abnormalities and virilization, who had a past medical history of encephalitis of an unknown etiology 16 months prior to presentation. RESULTS: Endocrine evaluation revealed an elevated serum testosterone and 17-hydroxy progesterone. A large left ovarian tumor was seen on a CT scan. Surgical excision revealed a mature cystic teratoma containing 6 liters of clear fluid with high androgen levels. Antibodies to the N-methyl-D-aspartate receptor of the hippocampus were detected in pre-operatively archived serum, but undetectable 6 months postoperatively. Immunohistochemistry studies on the tumor sections revealed that the antibodies in the patient's serum reacted with areas of the tumor expressing the N-methyl-D-aspartate receptor. Postoperatively, the patient's menstrual cycles became regular and her behavioral problems resolved. Her testosterone levels fell precipitously as well. CONCLUSION: Both virilizing mature cystic teratomas and teratoma-associated encephalitis are extremely rare in the pediatric population. We report on the first instance of these 2 rare entities occurring in the same patient. SN - 1423-0046 UR - https://www.unboundmedicine.com/medline/citation/19786797/Immune_mediated_encephalitis_and_virilization_in_association_with_a_mature_cystic_ovarian_teratoma_in_an_adolescent_girl_ L2 - https://www.karger.com?DOI=10.1159/000236087 DB - PRIME DP - Unbound Medicine ER -