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Bi-insular cortical involvement in anti-NMDA-receptor encephalitis - a case report.
BMC Neurol. 2016 Aug 08; 16:130.BN

Abstract

BACKGROUND

Anti-NMDA-receptor encephalitis is an immune-mediated inflammatory disorder of the central nervous system. Brain MRI is unremarkable in at least 50% of patients and highly variable in the remaining patients with signal abnormalities in different brain regions. Only scarce reports exist on other imaging modalities.

CASE PRESENTATION

A 31-year-old woman sub-acutely developed psychosis, behavioural changes, amnesia, alternating states of agitation and mutism, fever and epileptic seizures. Clinically suspected diagnosis of anti-NMDA-receptor encephalitis was confirmed by the detection of anti-NMDA receptor antibodies in CSF and serum. During the acute phase, brain MRI abnormalities were found in both insular cortices and hippocampi, whereas F(18)-FDG-PET showed hypermetabolism bilaterally in insular and prefrontal cortex. After resection of the underlying ovarian teratoma and with multimodal immunotherapy the patient substantially improved reaching a modified Rankin Scale score of 2 after 3 months. At follow-up, both hippocampi were still affected on MRI, whereas insular cortex appeared normal; however, both regions showed prominent glucose hypometabolism.

CONCLUSIONS

Here, we report bi-insular cortical abnormalities on MRI and F(18)-FDG-PET in a patient with anti-NMDA-receptor encephalitis during the acute phase and after clinical improvement.

Authors+Show Affiliations

Department of Neurology, Medical University of Innsbruck, Anichstrasse 35, A-6020, Innsbruck, Austria. harald.hegen@i-med.ac.at.Department of Nuclear Medicine, Medical University of Innsbruck, Innsbruck, Austria.Department of Neuroradiology, Medical University of Innsbruck, Innsbruck, Austria.Department of Nuclear Medicine, Medical University of Innsbruck, Innsbruck, Austria.Neuroimmunology Laboratory, Department of Neurology, Medical University of Innsbruck, Innsbruck, Austria.Department of Neurology, Medical University of Innsbruck, Anichstrasse 35, A-6020, Innsbruck, Austria.Department of Neurology, Medical University of Innsbruck, Anichstrasse 35, A-6020, Innsbruck, Austria.Department of Neurology, Medical University of Innsbruck, Anichstrasse 35, A-6020, Innsbruck, Austria.Department of Neurology, Medical University of Innsbruck, Anichstrasse 35, A-6020, Innsbruck, Austria.

Pub Type(s)

Case Reports
Journal Article

Language

eng

PubMed ID

27502387

Citation

Hegen, Harald, et al. "Bi-insular Cortical Involvement in anti-NMDA-receptor Encephalitis - a Case Report." BMC Neurology, vol. 16, 2016, p. 130.
Hegen H, Uprimny C, Grams A, et al. Bi-insular cortical involvement in anti-NMDA-receptor encephalitis - a case report. BMC Neurol. 2016;16:130.
Hegen, H., Uprimny, C., Grams, A., Virgolini, I., Ramberger, M., Beer, R., Helbok, R., Pfausler, B., & Schmutzhard, E. (2016). Bi-insular cortical involvement in anti-NMDA-receptor encephalitis - a case report. BMC Neurology, 16, 130. https://doi.org/10.1186/s12883-016-0653-9
Hegen H, et al. Bi-insular Cortical Involvement in anti-NMDA-receptor Encephalitis - a Case Report. BMC Neurol. 2016 Aug 8;16:130. PubMed PMID: 27502387.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Bi-insular cortical involvement in anti-NMDA-receptor encephalitis - a case report. AU - Hegen,Harald, AU - Uprimny,Christian, AU - Grams,Astrid, AU - Virgolini,Irene, AU - Ramberger,Melanie, AU - Beer,Ronny, AU - Helbok,Raimund, AU - Pfausler,Bettina, AU - Schmutzhard,Erich, Y1 - 2016/08/08/ PY - 2016/04/28/received PY - 2016/07/29/accepted PY - 2016/8/10/entrez PY - 2016/8/10/pubmed PY - 2016/12/15/medline KW - Autoimmune encephalitis KW - Case report KW - Frontal lobe KW - Hippocampus KW - Insular cortex KW - MRI KW - Medial temporal lobe KW - NMDA receptor KW - PET SP - 130 EP - 130 JF - BMC neurology JO - BMC Neurol VL - 16 N2 - BACKGROUND: Anti-NMDA-receptor encephalitis is an immune-mediated inflammatory disorder of the central nervous system. Brain MRI is unremarkable in at least 50% of patients and highly variable in the remaining patients with signal abnormalities in different brain regions. Only scarce reports exist on other imaging modalities. CASE PRESENTATION: A 31-year-old woman sub-acutely developed psychosis, behavioural changes, amnesia, alternating states of agitation and mutism, fever and epileptic seizures. Clinically suspected diagnosis of anti-NMDA-receptor encephalitis was confirmed by the detection of anti-NMDA receptor antibodies in CSF and serum. During the acute phase, brain MRI abnormalities were found in both insular cortices and hippocampi, whereas F(18)-FDG-PET showed hypermetabolism bilaterally in insular and prefrontal cortex. After resection of the underlying ovarian teratoma and with multimodal immunotherapy the patient substantially improved reaching a modified Rankin Scale score of 2 after 3 months. At follow-up, both hippocampi were still affected on MRI, whereas insular cortex appeared normal; however, both regions showed prominent glucose hypometabolism. CONCLUSIONS: Here, we report bi-insular cortical abnormalities on MRI and F(18)-FDG-PET in a patient with anti-NMDA-receptor encephalitis during the acute phase and after clinical improvement. SN - 1471-2377 UR - https://www.unboundmedicine.com/medline/citation/27502387/Bi_insular_cortical_involvement_in_anti_NMDA_receptor_encephalitis___a_case_report_ L2 - https://bmcneurol.biomedcentral.com/articles/10.1186/s12883-016-0653-9 DB - PRIME DP - Unbound Medicine ER -