Redhu Rakesh, Nadkarni Trimurti, Mahesh R
Department of Neurosurgery, King Edward Memorial Hospital, Seth G. S. Medical College, Parel, Mumbai - 400012, India.
J Pediatr Neurosci. 2011 Jan;6(1):62-4. doi: 10.4103/1817-1745.84412.
Diabetes insipidus (DI) associated with a thickened pituitary stalk is a diagnostic challenge in the pediatric population. Langerhans Cell Histiocytosis (LCH) is a rare cause of this entity. A 4-year-old male child presented with central DI of 1-year duration, associated with a thickened pituitary stalk. The etiology for the same remained elusive as the patient had no other manifestation to suggest LCH. A year later, the patient developed a left frontal scalp swelling. Neuroradiology demonstrated multiple punched out osteolytic lesions in both the frontal bones. The infundibulum was thickened and showed post-contrast enhancement. Histology and immunohistochemistry (IHC) of the biopsy specimen confirmed LCH. The child was administered chemotherapy according to LCH protocol, which resulted in 33% reduction in the size of the skull lesions. The DI was controlled with medical management. The present case highlights the need for serial follow-up and magnetic resonance (MR) imaging that led to a diagnosis of LCH. The clinical presentation and management of central DI and a thickened pituitary stalk is presented and the relevant literature is discussed.
伴有垂体柄增粗的尿崩症(DI)在儿科人群中是一个诊断难题。朗格汉斯细胞组织细胞增多症(LCH)是导致该病症的罕见病因。一名4岁男童出现了持续1年的中枢性尿崩症,并伴有垂体柄增粗。由于该患者没有其他提示LCH的表现,其病因仍不明确。1年后,该患者左侧额部头皮出现肿胀。神经放射学检查显示双侧额骨有多个凿孔样溶骨性病变。漏斗部增粗,增强扫描后有强化。活检标本的组织学和免疫组织化学(IHC)检查证实为LCH。该患儿按照LCH治疗方案接受了化疗,颅骨病变大小缩小了33%。尿崩症通过药物治疗得到了控制。本病例强调了需要进行系列随访和磁共振(MR)成像以诊断LCH。本文介绍了中枢性尿崩症和垂体柄增粗的临床表现及治疗,并讨论了相关文献。