Pituitary and systemic autoimmunity in a case of intrasellar germinoma.
Pituitary and systemic autoimmunity in a case of intrasellar germinoma.
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DOI：
10.1007/s11102-009-0187-x
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发表时间：
2011-12
期刊：
影响因子：
3.8
通讯作者：
Caturegli, Patrizio
中科院分区：
文献类型：
--
作者：
Gutenberg, Angelika;Bell, Jennifer J.;Lupi, Isabella;Tzou, Shey-Cherng;Landek-Salgado, Melissa A.;Kimura, Hiroaki;Su, Jack;Karaviti, Lefkothea P.;Salvatori, Roberto;Caturegli, Patrizio
摘要蝶鞍生殖细胞瘤与自体免疫性脑下垂体炎有相似的临床及病理特徴，因此很难与两者鉴别。然而，由于这两种疾病的不同治疗方法，这种差异对于患者护理至关重要。我们报告的情况下，一个11岁的女孩谁提出了尿崩症和生长迟缓，并被发现有鞍内和鞍上肿块。垂体活检的初步检查显示腺垂体弥漫性淋巴细胞浸润和胎盘碱性磷酸酶表达缺失，导致垂体炎和糖皮质激素治疗的诊断。由于缺乏临床和放射学反应，再次检查垂体标本，发现这次存在散在的c-kit和Oct 4阳性生殖细胞瘤细胞。修改后的诊断提示开始放射治疗，这导致垂体肿块消失。免疫学研究表明，患者的血清识别由患者自身生殖细胞瘤细胞表达的抗原，以及垂体抗原（如生长激素）和系统性抗原（如干燥综合征抗原B和α-烯醇化酶）。该研究首次报道了鞍内生殖细胞瘤患者存在垂体和全身抗体，并提醒我们，垂体弥漫性淋巴细胞浸润和垂体抗体并不总是表明诊断为自身免疫性垂体炎。
Germinomas arising in the sella turcica are difficult to differentiate from autoimmune hypophysitis because of similar clinical and pathological features. This differentiation, nevertheless, is critical for patient care due to different treatments of the two diseases. We report the case of an 11-year-old girl who presented with diabetes insipidus and growth retardation, and was found to have an intra- and supra-sellar mass. Initial examination of the pituitary biopsy showed diffuse lymphocytic infiltration of the adenohypophysis and absent placental alkaline phosphatase expression, leading to a diagnosis of hypophysitis and glucocorticoid treatment. Because of the lack of clinical and radiological response, the pituitary specimen was re-examined, revealing this time the presence of scattered c-kit and Oct4 positive germinoma cells. The revised diagnosis prompted the initiation of radiotherapy, which induced disappearance of the pituitary mass. Immunological studies showed that the patient’s serum recognized antigens expressed by the patient’s own germinoma cells, as well as pituitary antigens like growth hormone and systemic antigens like the Sjögren syndrome antigen B and alpha-enolase. The study first reports the presence of pituitary and systemic antibodies in a patient with intrasellar germinoma, and reminds us that diffuse lymphocytic infiltration of the pituitary gland and pituitary antibodies does not always indicate a diagnosis of autoimmune hypophysitis.
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