Clinical analysis of paraneoplastic encephalitis associated with ovarian teratoma

Tonomura, Yasuyo; Kataoka, Hiroshi; Hara, Yasuo; Takamure, Miwa; Naba, Itirou; Kitauti, Takanori; Saito, Kozue; Ueno, S.

doi:10.1007/s11060-007-9372-9

Cited by 34 publications

(35 citation statements)

References 21 publications

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“…Immunotherapy, including corticosteroids, intravenous immunoglobulin or plasma exchange, is often effective, and it has been suggested that prompt resection of the teratoma expedites recovery 1. In most previously reported cases of anti-NMDAR encephalitis, the ovarian teratoma was removed a few months (median: 9 weeks) after neurological symptom presentation, sometimes when symptoms had already partially responded to immunotherapy 1-5. We report the clinical outcome and follow-up of antibody titres in a Japanese woman whose ovarian tumour was removed early (20 days) after neurological symptom onset.…”

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confidence: 99%

Neurological response to early removal of ovarian teratoma in anti-NMDAR encephalitis

Seki

Suzuki

Iizuka

et al. 2008

Journal of Neurology, Neurosurgery & Psychiatry

131

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We report an 18-year-old woman with anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis, who developed psychiatric symptoms, progressive unresponsiveness, dyskinesias, hypoventilation, hypersalivation and seizures. Early removal of an ovarian teratoma followed by plasma exchange and corticosteroids resulted in a prompt neurological response and eventual full recovery. Serial analysis of antibodies to NR1/NR2B heteromers of the NMDAR showed an early decrease of serum titres, although the cerebrospinal fluid titres correlated better with clinical outcome. The patients' antibodies reacted with areas of the tumour that contained NMDAR-expressing tissue. Search for and removal of a teratoma should be promptly considered after the diagnosis of anti-NMDAR encephalitis.Paraneoplastic encephalitis associated with ovarian teratoma has recently been related to the development of antibodies to the NR1/NR2B heteromers of the N-methyl-D-asparate receptor (NMDAR). 1 The disorder, called anti-NMDAR encephalitis, results in a characteristic syndrome that presents with prominent psychiatric symptoms or, less frequently, memory deficits, followed by a rapid decline of the level of consciousness, central hypoventilation, seizures, involuntary movements and dysautonomia. Despite the severity of symptoms and prolonged clinical course, most patients recover if the disorder is recognised and treated. 1 Immunotherapy, including corticosteroids, intravenous immunoglobulin or plasma exchange, is often effective, and it has been suggested that prompt resection of the teratoma expedites recovery. 1 In most previously reported cases of anti-NMDAR encephalitis, the ovarian teratoma was removed a few months (median: 9 weeks) after neurological symptom presentation, sometimes when symptoms had already partially responded to immunotherapy. 1-5 We report the clinical outcome and follow-up of antibody titres in a Japanese woman whose ovarian tumour was removed early (20 days) after neurological symptom onset. CASE REPORTAn 18-year-old woman without a past medical history of interest developed headache and fever for a few days. She also complained of the sensation that the left half of her body was twisting. She was admitted to our hospital with progressive psychosis, and emotional and behavioural changes. Her temperature was 37.1°C; no meningeal signs were noted. Sustained involuntary movements were observed around her mouth, resembling orofacial dyskinesias. She was disorientated to place and person, and had schizophrenia-like symptoms, such as disorganized 1). She constantly repeated "I'm in the world of Harry Potter!" and "I have no idea who I am!" and knocked her head against the wall. Intravenous administration of acyclovir (1500 mg/day) and dexamethasone (6 mg/day) resulted in no improvement and she became mute and unresponsive to verbal commands. The orofacial dyskinesias gradually worsened, showing sustained bizarre movements such as widely opening and tightly closing of the eyes and mouth, sticking out the tongue and grimacin...

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confidence: 99%

Neurological response to early removal of ovarian teratoma in anti-NMDAR encephalitis

Seki

Suzuki

Iizuka

et al. 2008

Journal of Neurology, Neurosurgery & Psychiatry

131

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“…Mature teratomas are benign and usually composed of 3 germ-cell layers (ectoderm, mesoderm, and endoderm). 7 Benign teratomas have been associated with a variety of paraneoplastic syndromes including limbic encephalitis, 8,9 opsoclonus-myoclonus syndrome, 10 seronegative polyarthritis, tenosynovitis, 11 and autoimmune hemolytic anemia, 12 but the association with JDM reported here is, to our knowledge, a novel observation. The MRI with fat suppression of the patient's proximal lower extremities, which revealed an increased T2-weighted signal in many of the muscles of the thighs bilaterally.…”

Section: Discussionmentioning

confidence: 66%

Ovarian Teratoma Mimicking Features of Juvenile Dermatomyositis in a Child

2011

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An 8-year-old girl complained for 4 months of right arm pain, weakness in both legs, difficulty in arising from a seated or squatting position, and 1 month of pain in her hips, ankles, and knees. On physical examination, she had weak neck flexors, weak proximal and abdominal muscles, and an assisted Gower maneuver; both knees and ankles were painful. Erythematous macules on her elbows, knees, and medial ankles were present without heliotrope rash or dilated eyelid capillaries. She had nail-fold erythema and decreased numbers of nail-fold capillary end-row loops (ERLs) . MRI of her thigh muscles revealed serpiginous increased T-2 signals consistent with inflammation and a complex round mass in the left pelvis. A muscle biopsy did not indicate juvenile dermatomyositis. Pelvic ultrasound confirmed a solid mass of the left ovary consistent with a mature teratoma. After surgical removal of the teratoma, the myositis, synovitis, and cutaneous findings resolved over 4 months without further therapy.

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“…2 It is not stated whether any of the arrhythmias were related to seizures. Tachycardia, bradycardia, cardiac pauses and asystole have also been reported in other case series of anti-NMDAR encephalitis [8][9][10] and are presumed to be related to central autonomic dysfunction.…”

Section: Discussionmentioning

confidence: 78%

Anti-NMDA receptor encephalitis associated with ictal asystole

Lee

Lawn

Prentice

et al. 2011

Journal of Clinical Neuroscience

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Clinical analysis of paraneoplastic encephalitis associated with ovarian teratoma

Abstract: OTE presents with cardiac conduction problems and hypersalivation in addition to psychiatric symptoms, seizures, and central hypoventilation.

Cited by 34 publications

References 21 publications

Neurological response to early removal of ovarian teratoma in anti-NMDAR encephalitis

Neurological response to early removal of ovarian teratoma in anti-NMDAR encephalitis

Ovarian Teratoma Mimicking Features of Juvenile Dermatomyositis in a Child

Anti-NMDA receptor encephalitis associated with ictal asystole

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