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Case Report| Volume 35, ISSUE 3, P400-403, June 2022

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Anti-N-Methyl-D-Aspartate Receptor Encephalitis Associated with Mature Ovarian Teratoma in a Young Adolescent: A Case Report

Published:December 01, 2021DOI:https://doi.org/10.1016/j.jpag.2021.11.009

      Abstract

      Background

      Ovarian teratoma has an uncommon association with anti-N-methyl-d-aspartate receptor (NMDAR) encephalitis. This is a life-threatening condition, and here, we describe a case of an adolescent girl with anti-NMDAR encephalitis caused by an ovarian teratoma.

      Case

      A 14-year-old girl presented with acute features of fever, severe headache, altered behavioral changes, delirium, autonomic instability, episodes of seizure, and involuntary movement over a period of 1 month. Upon investigation, electroencephalogram (EEG), computed tomography (CT), and magnetic resonance imaging (MRI) of her brain showed normal findings. Her serum and cerebrospinal fluid were positive for anti-NMDARs, and a diagnosis of anti-NMDAR encephalitis was made, so she received a course of intravenous methyl prednisolone and immunoglobulin and was discharged after her neurological status improved. Upon further workup, she was suspected to have a left ovarian dermoid cyst on transabdominal ultrasonogram, which was an incidental finding. Her tumor marker panel showed normal serum lactate dehydrogenase, beta human chorionic gonadotropin, inhibin, alpha-fetoprotein, and carcinoembryonic antigen and a cancer antigen 125 level of 71.5 U/L. She eventually underwent laparoscopic left ovarian cystectomy and received immunotherapy in the postoperative period. She was discharged in stable condition on postoperative day 4. On histopathological examination, the specimen revealed a mature cystic teratoma with glial component. One year after surgery, the patient has recovered completely and has no residual psychiatric or neurological symptoms.

      Conclusion

      Complete recovery after surgery in cases of anti-NMDAR encephalitis with ovarian teratoma emphasizes the need for early recognition of the entity, search for underlying tumor, and tumor removal to improve the prognosis.

      Key Words

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      References

      1. Dalmau J, Tuzun E, Wu HY, et al: Paraneoplastic anti-N-methyl-d-aspartate receptor encephalitis associated with ovarian teratoma. Ann Neurol 2007; 61:25-36.

      2. <bib id=“bib2” type=“Periodical”>2.Martin AL, Jolliffe E, Hertweck SP: Ovarian teratoma associated with coexisting anti-n-methyl-d-aspartate receptor and glial fibrillary acidic protein autoimmune meningoencephalitis in an adolescent girl: a case report. J Pediatr Adolesc Gynecol 2018; 31:321-324.

      3. <bib id=“bib3” type=“Periodical”>3.Dalmau J, Armangué T, Planagumà J, et al: An update on anti-NMDA receptor encephalitis for neurologists and psychiatrists: mechanisms and models. Lancet Neurol 2019; 18:1045-1057.

      4. <bib id=“bib4” type=“Periodical”>4.Dai Y, Zhang J, Ren H, et al: Surgical outcomes in patients with anti-N-methyl d-aspartate receptor encephalitis with ovarian teratoma. Am J Obstet Gynecol 2019; 221:485. e1-10.

      5. <bib id=“bib5” type=“Periodical”>5.Graus F, Titulaer MJ, Balu R, et al: A clinical approach to diagnosis of autoimmune encephalitis. Lancet Neurol 2016; 15:391-404.

      6. <bib id=“bib6” type=“Periodical”>6.Timmerman D, Valentin L, Bourne TH, et al: International Ovarian Tumor Analysis (IOTA) group. Terms, definitions and measurements to describe the sonographic features of adnexal tumors: a consensus opinion from the International Ovarian Tumor Analysis (IOTA) group. Ultrasound Obstet Gynecol 2000; 16:500-505.

      7. <bib id=“bib7” type=“Periodical”>7.Abe KK, Koli RL, Yamamoto LG: Emergency department presentations of anti-N-methyl-d-aspartate receptor encephalitis. Pediatr Emerg Care 2016; 32:107-112.

      8. <bib id=“bib8” type=“Periodical”>8.Dalmau J, Armangue T, Planaguma J, et al: An update on anti-NMDA receptor encephalitis for neurologists and psychiatrists: mechanisms and models. Lancet Neurol 2019; 18:1045-1057. Doi:10.1016/S1474-4422(19)30244-3.

      9. <bib id=“bib9” type=“Periodical”>9.Garg D, Mohammad SS, Sharma S: Autoimmune encephalitis in children: an update. Indian Pediatr 2020; 57:662-670. PMID: 32727942.

      10. <bib id=“bib10” type=“Periodical”>10.Nosadini M, Mohammad SS, Ramanathan S, et al: Immune therapy in autoimmune encephalitis: a systematic review. Expert Rev Neurother 2015; 15:1391-1419.

      11. <bib id=“bib11” type=“Periodical”>11.Yan B, Wang Y, Zhang Y, et al: Teratoma-associated anti-N-methyl-d-aspartate receptor encephalitis: a case report and literature review. Medicine (Baltimore) 2019; 98:e15765.

      12. <bib id=“bib12” type=“Periodical”>12.Liu H, Jian M, Liang F, et al: Anti-N-methyl-d-aspartate receptor encephalitis associated with an ovarian teratoma: two cases report and anaesthesia considerations. BMC Anesthesiol 2015; 15:150.

      13. <bib id=“bib13” type=“Periodical”>13.Dalmau J, Lancaster E, Martinez-Hernandez E, et al: Clinical experience and laboratory investigations in patients with anti-NMDAR encephalitis. Lancet Neurol 2011; 10:63-74.

      14. <bib id=“bib14” type=“Periodical”>14.Tumbi A, Gilani A, Scarff JR, et al: Anti-N-methyl-D encephalitis. Innov Clin Neurosci 2011; 8:24-25.

      15. <bib id=“bib15” type=“Periodical”>15.Titulaer MJ, McCracken L, Gabilondo I, et al: Treatment and prognosis factors for long-term outcome in patients with anti NMDA receptor encephalitis: an observational cohort study. Lancet Neurol 2013; 12:157-165.

      16. <bib id=“bib16” type=“Periodical”>16.Dalmau J, Gleichman AJ, Hughes EG, et al: Anti-NMDA-receptor encephalitis: case series and analysis of the effects of antibodies. Lancet Neurol 2008; 7:1091-1098.