Makras Polyzois, Erickson Dana, Davidge-Pitts Caroline J, Diamond Eli L, Allen Carl E, McClain Kenneth L, Abeykoon Jithma P, Go Ronald S, Siwakoti Krishmita, Sotoudeh Houman, Ravindran Aishwarya, Gruber Lucinda M, Goyal Gaurav
Department of Endocrinology and Diabetes and Department of Medical Research, LCH Adult Clinic, 251 Hellenic Air Force & VA General Hospital, Athens 11525, Greece.
Division of Endocrinology, Diabetes, Metabolism and Nutrition, Mayo Clinic, Rochester, MN 55902, USA.
J Clin Endocrinol Metab. 2025 May 19;110(6):1756-1766. doi: 10.1210/clinem/dgae827.
Endocrinopathies are frequently the initial presentation of histiocytic neoplasms, which are rare hematologic disorders affecting multiple organ systems. Langerhans cell histiocytosis and Erdheim-Chester disease are 2 such disorders known to infiltrate the hypothalamus and/or pituitary gland, leading to arginine vasopressin deficiency (AVP-D) and anterior pituitary dysfunction (APD) in 20% to 30% of cases, often as the first manifestation. Conversely, histiocytic disorders account for a notable proportion (10-15%) of all pituitary stalk lesions. The diagnosis of histiocytoses is often delayed in such cases due to the nonspecific presentation of endocrinopathies and pituitary involvement. Consequently, endocrinologists are at the frontline and uniquely positioned to achieve early diagnosis by recognizing the varied nonendocrine features of these disorders. This article provides an overview of the endocrine manifestations of histiocytic disorders and presents a simplified algorithm to guide the diagnostic workup in cases presenting with "idiopathic" AVP-D or APD. Such cases should be evaluated for histiocytic neoplasms with additional imaging studies and biopsies of suspected disease sites. If no disease site beyond the pituitary is identified, the risks and benefits of a pituitary stalk lesion biopsy must be carefully considered. While treatments of histiocytic neoplasms are highly efficacious, endocrinopathies are considered permanent and require long-term hormone replacement. It remains unclear whether early diagnosis and novel targeted therapies can reverse these endocrine disorders. Therefore, the role of the endocrinologist role is critical in the diagnosis and management of these rare diseases.
内分泌病常为组织细胞肿瘤的首发表现,组织细胞肿瘤是一类罕见的血液系统疾病,可累及多个器官系统。朗格汉斯细胞组织细胞增多症和厄尔德海姆-切斯特病就是已知会累及下丘脑和/或垂体的两种此类疾病,在20%至30%的病例中会导致精氨酸血管加压素缺乏(AVP-D)和垂体前叶功能障碍(APD),且常为首发表现。相反,组织细胞疾病在所有垂体柄病变中占相当比例(10 - 15%)。由于内分泌病和垂体受累的表现不具特异性,此类病例中组织细胞增多症的诊断往往会延迟。因此,内分泌科医生处于前沿位置,且具备独特优势,可通过识别这些疾病多样的非内分泌特征来实现早期诊断。本文概述了组织细胞疾病的内分泌表现,并提出了一种简化的算法,以指导对出现“特发性”AVP-D或APD的病例进行诊断检查。此类病例应通过额外的影像学检查和对可疑疾病部位进行活检来评估是否存在组织细胞肿瘤。如果未发现垂体以外的疾病部位,则必须仔细权衡垂体柄病变活检的风险和益处。虽然组织细胞肿瘤的治疗效果显著,但内分泌病被认为是永久性的,需要长期激素替代治疗。早期诊断和新型靶向治疗能否逆转这些内分泌疾病仍不明确。因此,内分泌科医生在这些罕见疾病的诊断和管理中起着关键作用。