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Does MAP2 have a role in predicting the development of anti-NMDAR encephalitis associated with benign ovarian teratoma? A report of six new pediatric cases.

Abstract Anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis is a potentially fatal neurologic syndrome in which patients present with a spectrum of central nervous system deficits. Sixty percent of the cases can be attributed to the presence of tumors, most often ovarian teratomas. This report examines 6 pediatric patients who presented with neurologic deficits associated with the presence of such tumors. These cases illustrate a perplexing phenomenon, where benign teratomas could have a possible association with anti-NMDAR encephalitis. The purpose of this study was to compare the histology and immunohistochemistry of tumors associated with this syndrome to ovarian teratomas found in patients presenting with no neurologic symptoms. After obtaining institutional review board approval, 57 cases of ovarian teratomas were identified at our institution over 12 years. Six patients were identified with anti-NMDAR encephalitis. A panel of immunostains, including S100, GFAP, MAP2, and NeuN was applied to patients' tumor sections as well as the 6 controls from age-matched patients. No qualitative histologic or immunohistochemical differences were seen between the study cases and control group. Because no qualitative differences were identified between the study cases and the control group, testing of paired serum and cerebrospinal fluid remains the best method for diagnosis of anti-NMDAR encephalitis. Tumor banking with molecular analysis of ovarian teratomas, including whole-genome sequencing and comparative genomic hybridization between ovarian tissue saved from patients with and without anti-NMDAR encephalitis, is necessary to fully understand the etiopathogenesis of anti-NMDAR encephalitis.
PMID
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Authors

Mayor MeshTerms

Autoantibodies

Keywords

MAP2

NeuN

anti-NMDAR

encephalitis

neuronal markers

ovarian teratoma

Journal Title pediatric and developmental pathology : the official journal of the society for pediatric pathology and the paediatric pathology society
Publication Year Start




PMID- 25569473
OWN - NLM
STAT- MEDLINE
DA  - 20150415
DCOM- 20150519
LR  - 20151119
IS  - 1093-5266 (Print)
IS  - 1093-5266 (Linking)
VI  - 18
IP  - 2
DP  - 2015 Mar-Apr
TI  - Does MAP2 have a role in predicting the development of anti-NMDAR encephalitis
      associated with benign ovarian teratoma? A report of six new pediatric cases.
PG  - 122-6
LID - 10.2350/14-09-1554-OA.1 [doi]
AB  - Anti-N-methyl-d-aspartate receptor (anti-NMDAR) encephalitis is a potentially
      fatal neurologic syndrome in which patients present with a spectrum of central
      nervous system deficits. Sixty percent of the cases can be attributed to the
      presence of tumors, most often ovarian teratomas. This report examines 6
      pediatric patients who presented with neurologic deficits associated with the
      presence of such tumors. These cases illustrate a perplexing phenomenon, where
      benign teratomas could have a possible association with anti-NMDAR encephalitis. 
      The purpose of this study was to compare the histology and immunohistochemistry
      of tumors associated with this syndrome to ovarian teratomas found in patients
      presenting with no neurologic symptoms. After obtaining institutional review
      board approval, 57 cases of ovarian teratomas were identified at our institution 
      over 12 years. Six patients were identified with anti-NMDAR encephalitis. A panel
      of immunostains, including S100, GFAP, MAP2, and NeuN was applied to patients'
      tumor sections as well as the 6 controls from age-matched patients. No
      qualitative histologic or immunohistochemical differences were seen between the
      study cases and control group. Because no qualitative differences were identified
      between the study cases and the control group, testing of paired serum and
      cerebrospinal fluid remains the best method for diagnosis of anti-NMDAR
      encephalitis. Tumor banking with molecular analysis of ovarian teratomas,
      including whole-genome sequencing and comparative genomic hybridization between
      ovarian tissue saved from patients with and without anti-NMDAR encephalitis, is
      necessary to fully understand the etiopathogenesis of anti-NMDAR encephalitis.
FAU - Cundiff, Caitlin A
AU  - Cundiff CA
AD  - 1 Department of Pathology, Emory University School of Medicine and Children's
      Healthcare of Atlanta, Atlanta, GA, USA.
FAU - Elawabdeh, Nancy
AU  - Elawabdeh N
FAU - Naguib, Mina M
AU  - Naguib MM
FAU - Jactel, Samuel N
AU  - Jactel SN
FAU - Demellawy, Dina El
AU  - Demellawy DE
FAU - Abramowsky, Carlos R
AU  - Abramowsky CR
FAU - Durham, Megan M
AU  - Durham MM
FAU - Youssef, Lara
AU  - Youssef L
FAU - Wittkamp, Michael L
AU  - Wittkamp ML
FAU - Shehata, Bahig M
AU  - Shehata BM
LA  - eng
PT  - Journal Article
DEP - 20150108
PL  - United States
TA  - Pediatr Dev Pathol
JT  - Pediatric and developmental pathology : the official journal of the Society for
      Pediatric Pathology and the Paediatric Pathology Society
JID - 9809673
RN  - 0 (Autoantibodies)
RN  - 0 (Biomarkers, Tumor)
RN  - 0 (MAP2 protein, human)
RN  - 0 (Microtubule-Associated Proteins)
RN  - 0 (Receptors, N-Methyl-D-Aspartate)
SB  - IM
MH  - Adolescent
MH  - Anti-N-Methyl-D-Aspartate Receptor Encephalitis/blood/cerebrospinal
      fluid/*immunology/pathology/therapy
MH  - *Autoantibodies/blood/cerebrospinal fluid
MH  - Biomarkers, Tumor/*analysis
MH  - Case-Control Studies
MH  - Child
MH  - Child, Preschool
MH  - Female
MH  - Humans
MH  - Immunohistochemistry
MH  - Microtubule-Associated Proteins/*analysis
MH  - Ovarian Neoplasms/*chemistry/complications/pathology/therapy
MH  - Predictive Value of Tests
MH  - Prognosis
MH  - Receptors, N-Methyl-D-Aspartate/*immunology
MH  - Risk Factors
MH  - Teratoma/*chemistry/complications/pathology/therapy
OTO - NOTNLM
OT  - MAP2
OT  - NeuN
OT  - anti-NMDAR
OT  - encephalitis
OT  - neuronal markers
OT  - ovarian teratoma
EDAT- 2015/01/09 06:00
MHDA- 2015/05/20 06:00
CRDT- 2015/01/09 06:00
AID - 10.2350/14-09-1554-OA.1 [doi]
PST - ppublish
SO  - Pediatr Dev Pathol. 2015 Mar-Apr;18(2):122-6. doi: 10.2350/14-09-1554-OA.1. Epub 
      2015 Jan 8.