Fernández Silvia Carolina, Bernhardt María Celina, Grondona Ezequiel, Venier Ana Clara, Bertolino María Lorena, Pautasso Mauro José, Mezzano Emilio, Damilano Roxana Analía, Sala Claudia Susana, Herrera Enrique José, Pesaola Favio Nicolás, Maldonado Cristina Alicia, Quintar Amado Alfredo, De Paul Ana Lucía
Centro de Microscopía Electrónica, Facultad de Ciencias Médicas, Universidad Nacional de Córdoba, Córdoba, Argentina.
Servicio de Patología, Clínica Universitaria Reina Fabiola, Córdoba, Argentina.
Front Neurosci. 2023 Sep 22;17:1227144. doi: 10.3389/fnins.2023.1227144. eCollection 2023.
Xanthogranulomas are considered rare tumors, with their sellar and non-sellar frequency ranging from 1.6 to 7% among intracranial lesions, and described as a separate entity by the World Health Organization in 2000. The diagnosis of sellar xanthogranulomas is challenging, given their uncertain origin and clinical course. In addition, the limited reporting of sellar xanthogranuloma cases and the absence of characteristic images make these entities difficult to distinguish from other cystic lesions of the sellar region, such as adamantinomatous craniopharyngiomas, Rathke's cleft cysts, pituitary tumors, arachnoid cysts, epidermoid cysts, and dermoid cysts. Here, we describe the clinical presentation, radiological findings, immunohistochemical/histopathological analysis, and the ultrastructural examination by transmission electron microscopy of five sellar xanthogranulomas cases reported in two care centers in Cordoba, Argentina. Two males and three females between 37 and 73 years of age (average 51.8 years) presented with persistent headaches, generalized endocrine defects, and visual problems. MRI revealed cystic formations in the sellar region, which usually projected into adjacent tissues such as the suprasellar region or cavernous sinuses, and compressed other structures such as the optic chiasm, pituitary gland, and cranial nerves. All patients underwent surgical intervention to remove the tumor tissue. The histopathological analysis of the samples showed cellular tissue with a xanthogranulomatous appearance, inflammatory cellular infiltrate (mainly lymphocytes and macrophages), fibroblasts, abundant collagen fibers, and hemorrhages. An ultrastructural analysis helped to identify cellular infiltrates and granules resulting from tumor cell activity. The data support the hypothesis that sellar xanthogranulomas could occur as an inflammatory reaction secondary to the rupture and hemorrhage of a previous cystic process, thereby generating an expansion of the tumor body toward adjacent tissues. The information obtained from these cases contributes to the current knowledge about this disease's origin and clinical and histological evolution. However, the scarcity of patients and the observed phenotypic heterogeneity make its diagnosis still challenging. Undoubtedly, more investigations are needed to provide additional information in order to be able to achieve a more accurate diagnosis and effective treatment of this rare disease.
黄色肉芽肿被认为是罕见肿瘤,在颅内病变中,其鞍区和非鞍区发生率在1.6%至7%之间,2000年世界卫生组织将其列为一个独立的实体。鞍区黄色肉芽肿的诊断具有挑战性,因为其起源和临床病程尚不确定。此外,鞍区黄色肉芽肿病例报告有限且缺乏特征性影像,使得这些实体难以与鞍区其他囊性病变区分开来,如造釉细胞瘤型颅咽管瘤、拉克氏囊肿、垂体瘤、蛛网膜囊肿、表皮样囊肿和皮样囊肿。在此,我们描述了阿根廷科尔多瓦两个医疗中心报告的5例鞍区黄色肉芽肿病例的临床表现、影像学表现、免疫组化/组织病理学分析以及透射电子显微镜超微结构检查。2名男性和3名女性,年龄在37至73岁之间(平均51.8岁),表现为持续性头痛、全身性内分泌缺陷和视觉问题。磁共振成像(MRI)显示鞍区有囊性结构,通常向鞍上区或海绵窦等相邻组织突出,并压迫视交叉、垂体和颅神经等其他结构。所有患者均接受了手术干预以切除肿瘤组织。样本的组织病理学分析显示细胞组织呈黄色肉芽肿外观,有炎性细胞浸润(主要是淋巴细胞和巨噬细胞)、成纤维细胞、丰富的胶原纤维和出血。超微结构分析有助于识别肿瘤细胞活动产生的细胞浸润和颗粒。这些数据支持这样一种假说,即鞍区黄色肉芽肿可能是先前囊性病变破裂和出血继发的炎症反应,从而使肿瘤主体向相邻组织扩展。从这些病例中获得的信息有助于增进目前对该疾病起源以及临床和组织学演变的了解。然而,患者数量稀少以及观察到的表型异质性使其诊断仍然具有挑战性。毫无疑问,需要进行更多研究以提供更多信息,从而能够对这种罕见疾病实现更准确的诊断和有效治疗。