Infectious profiles in pediatric anti-N-methyl-d-aspartate receptor encephalitis
Anti-N-methyl-d-aspartate receptor autoimmune encephalitis (NMDAR AE) is an antibody-mediated neurological disorder that may be caused by post-herpes simplex virus-1 meningoencephalitis (HSV ME) and ovarian teratomas, although most pediatric cases are idiopathic. We sought to evaluate if other infec...
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Published in | Journal of neuroimmunology Vol. 381; p. 578139 |
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Main Authors | , , , , , , , , , , |
Format | Journal Article |
Language | English |
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Elsevier B.V
15.08.2023
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ISSN | 0165-5728 1872-8421 1872-8421 |
DOI | 10.1016/j.jneuroim.2023.578139 |
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Abstract | Anti-N-methyl-d-aspartate receptor autoimmune encephalitis (NMDAR AE) is an antibody-mediated neurological disorder that may be caused by post-herpes simplex virus-1 meningoencephalitis (HSV ME) and ovarian teratomas, although most pediatric cases are idiopathic. We sought to evaluate if other infections precede NMDAR AE by conducting a single-center, retrospective, case-control study of 86 pediatric cases presenting to Texas Children's Hospital between 2006 and 2022. HSV ME (HSV-1 and HSV-2) was a significantly more common preceding infection in the experimental group compared to control patients with idiopathic intracranial hypertension, while there was no difference in remote HSV infection between the two groups. Recent Epstein-Barr virus infection was evident in 8/42 (19%) tested experimental patients in comparison to 1/25 (4%) tested control patients which provided evidence for a genuine measure of effect but was not statistically significant due to small sample size (p = 0.07). The other 25 infectious etiologies were not different among the two groups and not all variables were clinically indicated or obtained in every subject, highlighting the need for future standardized, multi-institutional studies on underlying infectious precursors of autoimmune encephalitis.
•In addition to HSV- 1, HSV- 2 meningoencephalitis is a likely precipitant of anti-NMDAR encephalitis.•Other Herpesviridae like EBV may predispose to NMDAR AE greater than previously appreciated, requiring further study.•Frequently detected concomitant infections with NMDAR AE like common respiratory viruses are less likely triggers of NMDAR AE. |
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AbstractList | Anti-N-methyl-d-aspartate receptor autoimmune encephalitis (NMDAR AE) is an antibody-mediated neurological disorder that may be caused by post-herpes simplex virus-1 meningoencephalitis (HSV ME) and ovarian teratomas, although most pediatric cases are idiopathic. We sought to evaluate if other infections precede NMDAR AE by conducting a single-center, retrospective, case-control study of 86 pediatric cases presenting to Texas Children's Hospital between 2006 and 2022. HSV ME (HSV-1 and HSV-2) was a significantly more common preceding infection in the experimental group compared to control patients with idiopathic intracranial hypertension, while there was no difference in remote HSV infection between the two groups. Recent Epstein-Barr virus infection was evident in 8/42 (19%) tested experimental patients in comparison to 1/25 (4%) tested control patients which provided evidence for a genuine measure of effect but was not statistically significant due to small sample size (p = 0.07). The other 25 infectious etiologies were not different among the two groups and not all variables were clinically indicated or obtained in every subject, highlighting the need for future standardized, multi-institutional studies on underlying infectious precursors of autoimmune encephalitis.
•In addition to HSV- 1, HSV- 2 meningoencephalitis is a likely precipitant of anti-NMDAR encephalitis.•Other Herpesviridae like EBV may predispose to NMDAR AE greater than previously appreciated, requiring further study.•Frequently detected concomitant infections with NMDAR AE like common respiratory viruses are less likely triggers of NMDAR AE. Anti-N-methyl-d-aspartate receptor autoimmune encephalitis (NMDAR AE) is an antibody-mediated neurological disorder that may be caused by post-herpes simplex virus-1 meningoencephalitis (HSV ME) and ovarian teratomas, although most pediatric cases are idiopathic. We sought to evaluate if other infections precede NMDAR AE by conducting a single-center, retrospective, case-control study of 86 pediatric cases presenting to Texas Children's Hospital between 2006 and 2022. HSV ME (HSV-1 and HSV-2) was a significantly more common preceding infection in the experimental group compared to control patients with idiopathic intracranial hypertension, while there was no difference in remote HSV infection between the two groups. Recent Epstein-Barr virus infection was evident in 8/42 (19%) tested experimental patients in comparison to 1/25 (4%) tested control patients which provided evidence for a genuine measure of effect but was not statistically significant due to small sample size (p = 0.07). The other 25 infectious etiologies were not different among the two groups and not all variables were clinically indicated or obtained in every subject, highlighting the need for future standardized, multi-institutional studies on underlying infectious precursors of autoimmune encephalitis. Anti-N-methyl-d-aspartate receptor autoimmune encephalitis (NMDAR AE) is an antibody-mediated neurological disorder that may be caused by post-herpes simplex virus-1 meningoencephalitis (HSV ME) and ovarian teratomas, although most pediatric cases are idiopathic. We sought to evaluate if other infections precede NMDAR AE by conducting a single-center, retrospective, case-control study of 86 pediatric cases presenting to Texas Children's Hospital between 2006 and 2022. HSV ME (HSV-1 and HSV-2) was a significantly more common preceding infection in the experimental group compared to control patients with idiopathic intracranial hypertension, while there was no difference in remote HSV infection between the two groups. Recent Epstein-Barr virus infection was evident in 8/42 (19%) tested experimental patients in comparison to 1/25 (4%) tested control patients which provided evidence for a genuine measure of effect but was not statistically significant due to small sample size (p = 0.07). The other 25 infectious etiologies were not different among the two groups and not all variables were clinically indicated or obtained in every subject, highlighting the need for future standardized, multi-institutional studies on underlying infectious precursors of autoimmune encephalitis.Anti-N-methyl-d-aspartate receptor autoimmune encephalitis (NMDAR AE) is an antibody-mediated neurological disorder that may be caused by post-herpes simplex virus-1 meningoencephalitis (HSV ME) and ovarian teratomas, although most pediatric cases are idiopathic. We sought to evaluate if other infections precede NMDAR AE by conducting a single-center, retrospective, case-control study of 86 pediatric cases presenting to Texas Children's Hospital between 2006 and 2022. HSV ME (HSV-1 and HSV-2) was a significantly more common preceding infection in the experimental group compared to control patients with idiopathic intracranial hypertension, while there was no difference in remote HSV infection between the two groups. Recent Epstein-Barr virus infection was evident in 8/42 (19%) tested experimental patients in comparison to 1/25 (4%) tested control patients which provided evidence for a genuine measure of effect but was not statistically significant due to small sample size (p = 0.07). The other 25 infectious etiologies were not different among the two groups and not all variables were clinically indicated or obtained in every subject, highlighting the need for future standardized, multi-institutional studies on underlying infectious precursors of autoimmune encephalitis. |
ArticleNumber | 578139 |
Author | Demmler-Harrison, Gail Yarimi, Jonathan M. Muscal, Eyal Erickson, Timothy A. Kannan, Varun Jiang, Yike Sandweiss, Alexander J. Murray, Kristy O. Ronca, Shannon E. Fisher, Kristen Levine, Jesse M. |
Author_xml | – sequence: 1 givenname: Alexander J. surname: Sandweiss fullname: Sandweiss, Alexander J. organization: Department of Pediatrics, Division of Pediatric Neurology and Developmental Neuroscience, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 2 givenname: Timothy A. surname: Erickson fullname: Erickson, Timothy A. organization: Department of Pediatrics, Section of Pediatric Tropical Medicine, Center for Human Immunobiology, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 3 givenname: Yike surname: Jiang fullname: Jiang, Yike organization: Department of Pediatrics, Division of Pediatric Rheumatology, Duke University School of Medicine, United States of America – sequence: 4 givenname: Varun surname: Kannan fullname: Kannan, Varun organization: Department of Pediatrics, Division of Pediatric Neurology and Developmental Neuroscience, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 5 givenname: Jonathan M. surname: Yarimi fullname: Yarimi, Jonathan M. organization: Department of Pediatrics, Division of Pediatric Neurology and Developmental Neuroscience, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 6 givenname: Jesse M. surname: Levine fullname: Levine, Jesse M. organization: Department of Pediatrics, Division of Pediatric Neurology and Developmental Neuroscience, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 7 givenname: Kristen surname: Fisher fullname: Fisher, Kristen organization: Department of Pediatrics, Division of Pediatric Neurology and Developmental Neuroscience, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 8 givenname: Eyal surname: Muscal fullname: Muscal, Eyal organization: Department of Pediatrics, Section of Rheumatology, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 9 givenname: Gail surname: Demmler-Harrison fullname: Demmler-Harrison, Gail organization: Department of Pediatrics, Division of Infectious Disease, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 10 givenname: Kristy O. surname: Murray fullname: Murray, Kristy O. organization: Department of Pediatrics, Section of Pediatric Tropical Medicine, Center for Human Immunobiology, Baylor College of Medicine and Texas Children's Hospital, United States of America – sequence: 11 givenname: Shannon E. surname: Ronca fullname: Ronca, Shannon E. email: ronca@bcm.edu organization: Department of Pediatrics, Section of Pediatric Tropical Medicine, Center for Human Immunobiology, Baylor College of Medicine and Texas Children's Hospital, United States of America |
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Keywords | Post-infectious encephalitis NMDAR Epstein-Barr virus Seroconversion Herpes simplex virus Autoimmune encephalitis |
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SubjectTerms | Autoimmune encephalitis Epstein-Barr virus Herpes simplex virus NMDAR Post-infectious encephalitis Seroconversion |
Title | Infectious profiles in pediatric anti-N-methyl-d-aspartate receptor encephalitis |
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