Treatment responsive opsoclonus-ataxia associated with ovarian teratoma

Kawachi, Izumi; Saji, Etsuji; Toyoshima, Yuta; Dalmau, Josep; Nishizawa, M

doi:10.1136/jnnp.2009.177261

Cited by 8 publications

(13 citation statements)

References 5 publications

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“…In addition, Kawachi et al . suggested two types of PNS associated with ovarian teratoma: anti‐NMDAR encephalitis presenting with acute psychiatric symptoms; and brainstem encephalitis presenting with OAS, with the immune targets of the latter being currently unknown, as in the present case. Kurian et al .…”

Section: Discussionmentioning

confidence: 67%

“…Opsoclonus–ataxia syndrome (OAS) is a type of PNS of unknown etiology, with acute or subacute progression, and the predominant symptoms of opsoclonus (characterized by saccadic, multivectorial, involuntary eye movements) and cerebellar ataxia. Although very rare, OAS been reported in association with ovarian teratoma, similar to anti‐NMDAR encephalitis . We report a rare case of OAS associated with ovarian teratoma in which a good clinical course was achieved after intensive immunotherapy and early surgical treatment.…”

Section: Introductionmentioning

confidence: 84%

“…and Lou et al . described ovarian tumors that had already been diagnosed at the time of admission, and the tumor was promptly resected . This was presumably because the tumors were large and those patients complained of abdominal pain.…”

Section: Discussionmentioning

confidence: 99%

“…Although very rare, OAS been reported in association with ovarian teratoma, similar to anti-NMDAR encephalitis. [2][3][4] We report a rare case of OAS associated with ovarian teratoma in which a good clinical course was achieved after intensive immunotherapy and early surgical treatment.…”

Section: Introductionmentioning

confidence: 97%

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Opsoclonus–ataxia syndrome associated with ovarian mature teratoma

Kawakami

Kin

Hirose

et al. 2015

J of Obstet and Gynaecol

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A 16-year-old girl with no prior medical history developed vertigo and nausea following alimentary infection. Neurological examination showed limb and truncal ataxia, opsoclonus, myoclonus, and hyperreflexia. Brain magnetic resonance imaging and cerebrospinal fluid analysis showed no abnormalities. Treatment with i.v. high-dose methylprednisolone and immunoglobulin was started, but this proved ineffective. The clinical course was unusual, so whole-body computed tomography was done to evaluate other differential diagnoses. Imaging identified right ovarian mature teratoma. Paraneoplastic opsoclonus-ataxia syndrome was suspected, therefore single-incision laparoscopic ovarian cystectomy was done 10 days after admission. Two months after therapy, the patient had complete recovery and remained asymptomatic at 1 year after onset. Serum testing for anti-neuronal antibodies was negative, including for anti-N-methyl-d-aspartate-receptor antibody. Young women with ataxia and opsoclonus of unclear etiology should be examined for the presence of ovarian teratoma, then intensive immunotherapy and prompt tumor resection can lead to good clinical outcome.

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Section: Discussionmentioning

confidence: 67%

Section: Introductionmentioning

confidence: 84%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 97%

See 2 more Smart Citations

Opsoclonus–ataxia syndrome associated with ovarian mature teratoma

Kawakami

Kin

Hirose

et al. 2015

J of Obstet and Gynaecol

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“…In another patient, the presence of a subacute severe cerebellar syndrome preceded the diagnosis of a burn-out testicular germinoma for 4 years [11]. Benign teratomas, almost always of ovarian origin may cause encephalitis, associated with antibodies against the NR1 subunit of the glutamate receptor NMDA [12] and exceptionally opsoclonus-myoclonus syndrome without any associated antibody [13]. However, no cases of isolated cerebellar ataxia have been reported in association with this tumor.…”

Section: Discussionmentioning

confidence: 91%

Paraneoplastic cerebellar degeneration associated with thymic germinoma

Solà-Valls¹,

Gaba²,

Muñoz³

et al. 2012

Journal of the Neurological Sciences

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A novel treatment‐responsive encephalitis with frequent opsoclonus and teratoma

Armangué

Titulaer

Sabater

et al. 2013

Annals of Neurology

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Among 249 patients with teratoma-associated encephalitis, 211 had N-methyl-D-aspartate receptor antibodies and 38 were negative for these antibodies. Whereas antibody-positive patients rarely developed prominent brainstem–cerebellar symptoms, 22 (58%) antibody-negative patients developed a brainstem–cerebellar syndrome, which in 45% occurred with opsoclonus. The median age of these patients was 28.5 years (range = 12–41), 91% were women, and 74% had full recovery after therapy and tumor resection. These findings uncover a novel phenotype of paraneoplastic opsoclonus that until recently was likely considered idiopathic or postinfectious. The triad of young age (teenager to young adult), systemic teratoma, and high response to treatment characterize this novel brain-stem–cerebellar syndrome.

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Treatment responsive opsoclonus-ataxia associated with ovarian teratoma

Cited by 8 publications

References 5 publications

Opsoclonus–ataxia syndrome associated with ovarian mature teratoma

Opsoclonus–ataxia syndrome associated with ovarian mature teratoma

Paraneoplastic cerebellar degeneration associated with thymic germinoma

A novel treatment‐responsive encephalitis with frequent opsoclonus and teratoma

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