Lamback Elisa, Filho Ferdinand Duenas Cabrera, Ventura Nina, Chimelli Leila, Christ-Crain Mirjam, Gadelha Mônica R
Neuroendocrinology Research Center, Endocrinology Section, Medical School and Hospital Universitário Clementino Fraga Filho, Universidade Federal do Rio de Janeiro, Rio de Janeiro 21941-617, Brazil.
Neuropathology and Molecular Genetics Laboratory, Instituto Estadual do Cérebro Paulo Niemeyer, Secretaria Estadual de Saúde, Rio de Janeiro 20231-092, Brazil.
JCEM Case Rep. 2024 Aug 16;2(9):luae099. doi: 10.1210/jcemcr/luae099. eCollection 2024 Sep.
Neurocytomas are neuronal tumors that are usually intraventricular. Rare cases can arise from extraventricular sites. To our knowledge, only 29 cases of extraventricular neurocytoma of the sellar region (EVNSR) have been reported in the literature. We describe a case of a 39-year-old woman who presented with a one-month history of refractory headache, nausea and vomiting. Magnetic resonance imaging (MRI) showed a 5.1 × 3.1 × 2.2 cm sellar and suprasellar mass, suggestive of a pituitary adenoma (PA). She had hyponatremia, obstructive hydrocephalus, and panhypopituitarism at presentation (hypogonadism, adrenal insufficiency). After glucocorticoid replacement therapy and ventriculoperitoneal shunt, the vomiting and headache resolved, but she remained with nausea and hyponatremia. She was submitted to surgery, and histopathological analysis revealed a neurocytoma with positive immunostaining for arginine vasopressin. Syndrome of inappropriate antidiuresis (SIAD) was diagnosed but did not resolve after surgery due to residual tumor, despite fluid restriction and saline replacement. SIAD later resolved with empagliflozin. In conclusion, EVNSR is extremely rare and can be misdiagnosed as PA on MRI. In the context of SIAD and extraventricular neurocytoma, a secreting arginine vasopressin tumor must be considered. SIAD can be challenging to treat, with excision of the EVNSR the treatment choice and, alternatively, empagliflozin associated with fluid restriction.
神经细胞瘤是一种通常位于脑室内的神经元肿瘤。罕见情况下可起源于脑室外部位。据我们所知,文献中仅报道了29例鞍区脑室外神经细胞瘤(EVNSR)。我们描述了一例39岁女性,她有1个月的难治性头痛、恶心和呕吐病史。磁共振成像(MRI)显示鞍区和鞍上有一个5.1×3.1×2.2 cm的肿块,提示垂体腺瘤(PA)。她就诊时存在低钠血症、梗阻性脑积水和全垂体功能减退(性腺功能减退、肾上腺功能不全)。在接受糖皮质激素替代治疗和脑室腹腔分流术后,呕吐和头痛缓解,但仍有恶心和低钠血症。她接受了手术,组织病理学分析显示为神经细胞瘤,精氨酸加压素免疫染色呈阳性。诊断为抗利尿激素分泌异常综合征(SIAD),尽管进行了液体限制和盐水补充,但术后由于残留肿瘤该综合征仍未缓解。SIAD后来使用恩格列净得以缓解。总之,EVNSR极为罕见,在MRI上可能被误诊为PA。在SIAD和脑室外神经细胞瘤的情况下,必须考虑分泌精氨酸加压素的肿瘤。SIAD的治疗可能具有挑战性,治疗选择是切除EVNSR,或者使用恩格列净并限制液体摄入。