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Ovarian teratoma associated with anti-N-methyl D-aspartate receptor encephalitis: a report of 5 cases documenting prominent intratumoral lymphoid infiltrates.

Abstract Anti-N-methyl D-aspartate receptor (NMDAR) encephalitis is a recently described severe neurological disorder predominantly affecting young women, which presents with psychosis, memory deficits, seizures, and encephalopathy, often requiring prolonged hospitalization. The condition is frequently associated with an underlying neoplasm, most often an ovarian teratoma, and in such cases appears to be a para-neoplastic, immune-mediated encephalopathy. The histologic features of the teratomas associated with anti-NMDAR encephalitis have seldom been described in detail. Therefore, in this report, we have compared ovarian teratomas (4 mature and 1 immature) from 5 patients with anti-NMDAR encephalitis with 22 sporadic control teratomas (14 mature and 8 immature) that included neuroglial elements. The encephalitis-associated tumors ranged from 0.7 to 9.5 cm diameter, and 1 case was bilateral; the second teratoma was discovered 13 mo after the first when symptoms recurred. In comparison with control teratomas, the anti-NMDAR-associated tumors showed a more marked intratumoral lymphoid infiltrate that colocalized to the mature neuroglial elements. Reactive germinal centers (3 cases) and diffuse lymphoplasmacytic infiltrates within the neuroglial matrix (4 cases), and degenerative neuronal changes (2 cases), were seen only in the anti-NMDAR-positive cases. Pathologists encountering ovarian teratomas with these distinctive reactive lymphoid elements should consider the possibility of anti-NMDAR encephalitis, particularly because the neurological symptoms may develop after tumor resection. Careful histopathologic examination may be required to identify small, radiologically occult teratomas, and to demonstrate the presence of subtle neoplastic neuroglial components in teratomas associated with anti-NMDAR encephalitis.
PMID
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Authors

Mayor MeshTerms
Keywords
Journal Title international journal of gynecological pathology : official journal of the international society of gynecological pathologists
Publication Year Start




PMID- 22833082
OWN - NLM
STAT- MEDLINE
DA  - 20120808
DCOM- 20121217
LR  - 20120808
IS  - 1538-7151 (Electronic)
IS  - 0277-1691 (Linking)
VI  - 31
IP  - 5
DP  - 2012 Sep
TI  - Ovarian teratoma associated with anti-N-methyl D-aspartate receptor encephalitis:
      a report of 5 cases documenting prominent intratumoral lymphoid infiltrates.
PG  - 429-37
AB  - Anti-N-methyl D-aspartate receptor (NMDAR) encephalitis is a recently described
      severe neurological disorder predominantly affecting young women, which presents 
      with psychosis, memory deficits, seizures, and encephalopathy, often requiring
      prolonged hospitalization. The condition is frequently associated with an
      underlying neoplasm, most often an ovarian teratoma, and in such cases appears to
      be a para-neoplastic, immune-mediated encephalopathy. The histologic features of 
      the teratomas associated with anti-NMDAR encephalitis have seldom been described 
      in detail. Therefore, in this report, we have compared ovarian teratomas (4
      mature and 1 immature) from 5 patients with anti-NMDAR encephalitis with 22
      sporadic control teratomas (14 mature and 8 immature) that included neuroglial
      elements. The encephalitis-associated tumors ranged from 0.7 to 9.5 cm diameter, 
      and 1 case was bilateral; the second teratoma was discovered 13 mo after the
      first when symptoms recurred. In comparison with control teratomas, the
      anti-NMDAR-associated tumors showed a more marked intratumoral lymphoid
      infiltrate that colocalized to the mature neuroglial elements. Reactive germinal 
      centers (3 cases) and diffuse lymphoplasmacytic infiltrates within the neuroglial
      matrix (4 cases), and degenerative neuronal changes (2 cases), were seen only in 
      the anti-NMDAR-positive cases. Pathologists encountering ovarian teratomas with
      these distinctive reactive lymphoid elements should consider the possibility of
      anti-NMDAR encephalitis, particularly because the neurological symptoms may
      develop after tumor resection. Careful histopathologic examination may be
      required to identify small, radiologically occult teratomas, and to demonstrate
      the presence of subtle neoplastic neuroglial components in teratomas associated
      with anti-NMDAR encephalitis.
FAU - Dabner, Marcus
AU  - Dabner M
AD  - Department of Histopathology, King Edward Memorial Hospital, Perth, Western
      Australia.
FAU - McCluggage, W Glenn
AU  - McCluggage WG
FAU - Bundell, Chris
AU  - Bundell C
FAU - Carr, Aisling
AU  - Carr A
FAU - Leung, Yee
AU  - Leung Y
FAU - Sharma, Raghwa
AU  - Sharma R
FAU - Stewart, Colin J R
AU  - Stewart CJ
LA  - eng
PT  - Case Reports
PT  - Journal Article
PL  - United States
TA  - Int J Gynecol Pathol
JT  - International journal of gynecological pathology : official journal of the
      International Society of Gynecological Pathologists
JID - 8214845
SB  - IM
MH  - Adolescent
MH  - Adult
MH  - Anti-N-Methyl-D-Aspartate Receptor Encephalitis/*etiology
MH  - Cell Aggregation
MH  - Female
MH  - Humans
MH  - Lymphocytes, Tumor-Infiltrating/*pathology
MH  - Ovarian Neoplasms/complications/*pathology
MH  - Teratoma/complications/*pathology
EDAT- 2012/07/27 06:00
MHDA- 2012/12/18 06:00
CRDT- 2012/07/27 06:00
AID - 10.1097/PGP.0b013e31824a1de2 [doi]
PST - ppublish
SO  - Int J Gynecol Pathol. 2012 Sep;31(5):429-37.