Oligodendroglioma arising within a mature cystic ovarian teratoma: case report and review of the literature

Zannoni, Gian Franco; Fadda, Guido; Scambia, Giovanni; Capelli, Arnaldo; Carbone, Arnaldo

doi:10.1034/j.1600-0412.2002.810918.x

Cited by 13 publications

(8 citation statements)

References 9 publications

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“…However, NMDAR antibodies were negative in our patient. While, to the best of our knowledge, the presence of MOG in teratomas has not been investigated so far, expression of CNPase, an oligodendrocyte marker, has been described in mature teratomas [59], and several reports on oligodendrogliomas arising in mature teratomas exist [60–63]. Ectopic expression of MOG by the patient’s tumor therefore cannot be completely ruled out.…”

Section: Discussionmentioning

confidence: 99%

MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 3: Brainstem involvement - frequency, presentation and outcome

Jarius

Kleiter

Ruprecht

et al. 2016

J Neuroinflammation

225

199

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BackgroundMyelin oligodendrocyte glycoprotein antibodies (MOG-IgG) are present in a subset of aquaporin-4 (AQP4)-IgG-negative patients with optic neuritis (ON) and/or myelitis. Little is known so far about brainstem involvement in MOG-IgG-positive patients.ObjectiveTo investigate the frequency, clinical and paraclinical features, course, outcome, and prognostic implications of brainstem involvement in MOG-IgG-positive ON and/or myelitis.MethodsRetrospective case study.ResultsAmong 50 patients with MOG-IgG-positive ON and/or myelitis, 15 (30 %) with a history of brainstem encephalitis were identified. All were negative for AQP4-IgG. Symptoms included respiratory insufficiency, intractable nausea and vomiting (INV), dysarthria, dysphagia, impaired cough reflex, oculomotor nerve palsy and diplopia, nystagmus, internuclear ophthalmoplegia (INO), facial nerve paresis, trigeminal hypesthesia/dysesthesia, vertigo, hearing loss, balance difficulties, and gait and limb ataxia; brainstem involvement was asymptomatic in three cases. Brainstem inflammation was already present at or very shortly after disease onset in 7/15 (47 %) patients. 16/21 (76.2 %) brainstem attacks were accompanied by acute myelitis and/or ON. Lesions were located in the pons (11/13), medulla oblongata (8/14), mesencephalon (cerebral peduncles; 2/14), and cerebellar peduncles (5/14), were adjacent to the fourth ventricle in 2/12, and periaqueductal in 1/12; some had concomitant diencephalic (2/13) or cerebellar lesions (1/14). MRI or laboratory signs of blood-brain barrier damage were present in 5/12. Cerebrospinal fluid pleocytosis was found in 11/14 cases, with neutrophils in 7/11 (3-34 % of all CSF white blood cells), and oligoclonal bands in 4/14. Attacks were preceded by acute infection or vaccination in 5/15 (33.3 %). A history of teratoma was noted in one case. The disease followed a relapsing course in 13/15 (87 %); the brainstem was involved more than once in 6. Immunosuppression was not always effective in preventing relapses. Interferon-beta was followed by new attacks in two patients. While one patient died from central hypoventilation, partial or complete recovery was achieved in the remainder following treatment with high-dose steroids and/or plasma exchange. Brainstem involvement was associated with a more aggressive general disease course (higher relapse rate, more myelitis attacks, more frequently supratentorial brain lesions, worse EDSS at last follow-up).ConclusionsBrainstem involvement is present in around one third of MOG-IgG-positive patients with ON and/or myelitis. Clinical manifestations are diverse and may include symptoms typically seen in AQP4-IgG-positive neuromyelitis optica, such as INV and respiratory insufficiency, or in multiple sclerosis, such as INO. As MOG-IgG-positive brainstem encephalitis may take a serious or even fatal course, particular attention should be paid to signs or symptoms of additional brainstem involvement in patients presenting with MOG-IgG-positive ON and/or myelitis.

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Section: Discussionmentioning

confidence: 99%

MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 3: Brainstem involvement - frequency, presentation and outcome

Jarius

Kleiter

Ruprecht

et al. 2016

J Neuroinflammation

225

199

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“…Furthermore, expression of the NMDAR GluN1 subunit by the teratomatous nervous tissue was significantly more often glial in NMDAR-E teratomas than in control teratomas. Another striking particularity was that among these 27 mature teratomas, 3 contained neuroglial tissue exhibiting histopathological features of CNS neuroglial tumor, while it was exceptionally reported in the literature on sporadic ovarian teratomas ([1, 3, 10, 15, 24, 25, 31–34] the histological classification of these 20 cases is summarized in Additional file 1: Table S2 in Online Resource). We also confirmed the particular immune environment of the nervous tissue of these teratomas that exhibited massive inflammatory infiltrates.…”

Section: Discussionmentioning

confidence: 99%

Immunopathological characterization of ovarian teratomas associated with anti-N-methyl-D-aspartate receptor encephalitis

Chefdeville

Treilleux

Mayeur

et al. 2019

acta neuropathol commun

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Encephalitis with anti-NMDAR antibodies (NMDAR-E) is a severe autoimmune neurological disorder, defined by a clinical presentation of encephalitis and the presence of IgG targeting the GluN1 subunit of NMDA receptors in the CSF. An underlying ovarian teratoma is commonly associated with this autoimmune disease suggesting a role of the tumor in immunopathogenesis. In this study, we characterized the salient histopathological features of 27 ovarian teratomas associated with NMDAR-E (3 immature and 24 mature teratomas) and 40 controls without associated encephalitis. All but one NMDAR-E-associated teratomas contained a nervous tissue component, while less than 40% of control teratomas did (p < 0.001). GluN1 expression by teratomatous nervous tissue seemed to be more often glial in NMDAR-E teratomas than in control teratomas (73% vs. 29%, p < 0.05). Strikingly, 3 out of 24 NMDAR-E-associated mature teratomas contained neuroglial tissue exhibiting histopathological features of central nervous system neuroglial tumor, while such glioma-like features are exceptionally described in the literature on ovarian teratomas. Moreover, NMDAR-E associated teratomas differed from sporadic ovarian teratomas by consistent and prominent infiltration of the nervous tissue component by immune cells, comprised of T- and B-cells and mature dendritic cells organized in tertiary lymphoid structures, with IgG and IgA deposits and plasma cells in close contact to the neuroglial tissue.These data demonstrate an association between massive infiltration of NMDAR-E-associated teratomas by immune cells and particular glial features of its neuroglial component, suggesting that this glial tissue might be involved in triggering or sustaining the anti-tumor response associated with the auto-immune neurological disease.Electronic supplementary materialThe online version of this article (10.1186/s40478-019-0693-7) contains supplementary material, which is available to authorized users.

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“…Apart from this, tumors originating from neural tissue are rare, and the cooccurrence of central neurocytomas, ependymomas, glioblastomas, neuroblastomas, neuroectodermal tumors, and multiple neuroectodermal tumors together with mature cystic teratomas has been reported in the literature [4–7]. Oligodendrogliomas arising in teratomas are extremely rare, and, to our knowledge, three cases in the adulthood [3, 8, 9] and one case in the childhood [10] have been reported in the literature. Furthermore, Din et al reviewed six cases and reported that oligodendrogliomas can arise in mature and immature teratomas and those arising in immature teratomas have the worst prognosis [11].…”

Section: Discussionmentioning

confidence: 99%

“…The most common malignancy originating from mature cystic teratoma is squamous cell carcinoma [2]. Additionally, adenocarcinoma, undifferentiated carcinoma, sarcoma, papillary carcinoma, and malignant melanoma can also develop [3]. But the development of neuroepithelial tumors is very rare.…”

Section: Introductionmentioning

confidence: 99%

Oligodendroglioma Arising in Mature Cystic Teratoma

Ünal

Güleç

Sedele

2014

Case Reports in Oncological Medicine

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Background. Development of neuroepithelial tumors from mature cystic teratoma is very rare. We present a case of oligodendroglioma developing inside mature cystic teratoma. Case. Eighteen-years-old female, right adnexal mass with solid and cystic areas was detected. Sections showed all three germ layers. Also, a tumoral lesion was observed in a glial fibrillary matrix. Tumor was composed of monotonous, uniform cells which have oval-round nucleus, perinuclear halo, and indistinct cytoplasm. GFAP, EGFR, P53 were positive. Conclusions. We diagnosed oligodendroglioma arising from mature cystic teratoma. There was no recurrence at the end of 13 months followup. The number of cases which have been reported in the literature is only a few.

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Oligodendroglioma arising within a mature cystic ovarian teratoma: case report and review of the literature

Cited by 13 publications

References 9 publications

MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 3: Brainstem involvement - frequency, presentation and outcome

MOG-IgG in NMO and related disorders: a multicenter study of 50 patients. Part 3: Brainstem involvement - frequency, presentation and outcome

Immunopathological characterization of ovarian teratomas associated with anti-N-methyl-D-aspartate receptor encephalitis

Oligodendroglioma Arising in Mature Cystic Teratoma

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