Prosch Helmut, Grois Nicole, Bökkerink Jos, Prayer Daniela, Leuschner Ivo, Minkov Milen, Gadner Helmut
Children's Cancer Research Institute, Vienna, Austria.
Pediatr Blood Cancer. 2006 Mar;46(3):363-6. doi: 10.1002/pbc.20027.
Central diabetes insipidus (CDI) is a rare disorder that may be caused by a variety of diseases. In pediatric and adolescent patients the most common causes for CDI are Langerhans cell histiocytosis (LCH) and germinoma. To avoid a potentially hazardous biopsy of the hypothalamic pituitary region it is recommended to evaluate patients with CDI carefully to identify potential extracranial lesions. Since LCH is the most common systemic disease that may cause CDI, special focus is paid to the identification of LCH lesions. We report on a 9(1/2) year old girl who presented with central diabetes insipidus and a thickening of the pituitary stalk on magnetic resonance imaging. Diagnostic workup revealed a history of recurrent ear infections and a compressed 6th thoracic vertebral body on radiographs. Based on these findings LCH was anticipated. Upon growth of the pituitary stalk lesion the patient was treated with LCH standard chemotherapy. After an initial shrinkage of the lesion, a further growth of the pituitary stalk lesion was observed and the tumor was resected. Histopathology revealed germinoma. This case underscores the importance of a istopathologically proven diagnosis in patients with HPR tumors before the initiation of a specific therapy, even if the clinical findings are highly suggestive.
中枢性尿崩症(CDI)是一种罕见的疾病,可能由多种疾病引起。在儿童和青少年患者中,CDI最常见的病因是朗格汉斯细胞组织细胞增多症(LCH)和生殖细胞瘤。为避免对下丘脑 - 垂体区域进行有潜在风险的活检,建议对CDI患者进行仔细评估,以识别潜在的颅外病变。由于LCH是可能导致CDI的最常见全身性疾病,因此特别关注LCH病变的识别。我们报告了一名9岁半的女孩,她患有中枢性尿崩症,磁共振成像显示垂体柄增厚。诊断性检查发现有反复耳部感染史,X线片显示第6胸椎椎体受压。基于这些发现,推测为LCH。垂体柄病变增大后,该患者接受了LCH标准化疗。病变最初缩小后,观察到垂体柄病变进一步增大,遂将肿瘤切除。组织病理学检查显示为生殖细胞瘤。该病例强调了在开始特定治疗之前,对下丘脑 - 垂体区域(HPR)肿瘤患者进行组织病理学确诊的重要性,即使临床发现高度提示某种疾病。