Yamamoto Akiko, Komatsu Nagisa, Iwata Naoko, Fujisawa Haruki, Suzuki Atsushi, Sugimura Yoshihisa
Division of Pediatrics, Kumamoto Chuo Hospital, Kumamoto 862-0965, Japan.
Department of Endocrinology and Diabetes, Daido Hospital, Nagoya, Aichi 457-8511, Japan.
JCEM Case Rep. 2024 Dec 26;3(1):luae214. doi: 10.1210/jcemcr/luae214. eCollection 2025 Jan.
Lymphocytic infundibuloneurohypophysitis (LINH) is a disease with an etiology involving an autoimmune mechanism, characterized by lymphocytic inflammation of the posterior pituitary and infundibular stalk, resulting in arginine vasopressin deficiency. It is difficult to distinguish from pituitary neoplasm or infiltrative diseases, and biopsy is necessary for a definitive diagnosis, but this is highly invasive. In children, it is especially important to distinguish LINH from tumors such as germ cell tumors. Recently, the usefulness of anti-rabphilin-3A antibody as a serum marker for LINH has been reported. To date, only a limited number of pediatric cases have been reported. We present a 4-year-old boy with arginine vasopressin deficiency. Magnetic resonance imaging of the head showed thickening of the pituitary stalk without a posterior pituitary bright spot, and anti-rabphilin-3A antibody was positive. Consequently, pituitary biopsy was not performed because of the strong suspicion of LINH. Five months after symptom onset, the pituitary stalk thickening had resolved. This case represents the first report of probable or definitive LINH with anti-rabphilin-3A antibody positivity in a 4-year-old child, making it the youngest positive case reported to date. Our case highlights the importance of noninvasive approaches and careful follow-up to avoid invasive interventions for children with LINH.
淋巴细胞性漏斗神经垂体炎(LINH)是一种病因涉及自身免疫机制的疾病,其特征是垂体后叶和漏斗柄的淋巴细胞炎症,导致精氨酸加压素缺乏。它很难与垂体肿瘤或浸润性疾病区分开来,活检是明确诊断所必需的,但这具有高度侵入性。在儿童中,将LINH与生殖细胞肿瘤等肿瘤区分开来尤为重要。最近,有报道称抗rabphilin - 3A抗体作为LINH的血清标志物具有实用性。迄今为止,仅报道了有限数量的儿科病例。我们报告一名患有精氨酸加压素缺乏的4岁男孩。头部磁共振成像显示垂体柄增厚且垂体后叶无亮点,抗rabphilin - 3A抗体呈阳性。因此,由于强烈怀疑LINH,未进行垂体活检。症状出现五个月后,垂体柄增厚已消退。本病例是4岁儿童中可能或确诊的LINH且抗rabphilin - 3A抗体阳性的首例报告,使其成为迄今为止报道的最年轻的阳性病例。我们的病例强调了非侵入性方法和仔细随访对于避免对LINH儿童进行侵入性干预的重要性。