Reversible Hypothalamic Obesity in a Girl with Suprasellar Tuberculoma.

IF 2.6 3区 医学 Q3 ENDOCRINOLOGY & METABOLISM Hormone Research in Paediatrics Pub Date : 2024-01-01 Epub Date: 2023-03-28 DOI:10.1159/000530384
Anne Rochtus, Lieven Lagae, Katrien Jansen, Lien De Somer, François Vermeulen, Francis de Zegher
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Abstract

Introduction: Suprasellar tuberculoma are extremely rare in children and most of those patients present with headache, vomiting, visual disturbances, and hypofunction of the pituitary gland. In this case report, we present a girl with tuberculosis, who developed significant weight gain in combination with pituitary dysfunction, which recovered after antituberculosis treatment.

Case presentation: An 11-year old girl presented with headache, fever and anorexia that progressively evolved into an encephalopathic status with cranial nerves III and VI paresis. Brain MRI showed meningeal contrast capture along cranial nerves II (including optic chiasm), III, V and VI bilaterally and multiple contrast enhancing brain parenchyma lesions. Tuberculin skin test was negative but interferon-gamma release assay was positive. The clinical and radiological working diagnosis was consistent with tuberculous meningoencephalitis. Pulse corticosteroids for 3 days and quadruple antituberculosis therapy were started and the girl demonstrated obvious improvement of her neurological symptoms. However, after a few months of therapy she developed remarkable weight gain (+20 kg in 1 year) and growth arrest. Her hormone profile revealed insulin resistance (homeostasis model assessment-estimated insulin resistance [HOMA-IR] 6.8) despite putative growth hormone deficiency (circulating insulin-like growth factor-I [IGF-I] 104 μg/L [-2.4 SD]). Follow-up brain MRI showed a decrease in basal meningitis, but increased parenchymal lesions in the suprasellar region extending medially into the nucleus lentiformis, with now a voluminous tuberculoma at this site. Antituberculosis treatment was continued for a total of 18 months. The patient improved clinically, she regained her pre-illness Body Mass Index (BMI) SDS and her growth rate increased slightly. On the hormonal side, disappearance of insulin resistance (HOMA-IR 2.5) and an increase in IGF-I (175 μg/L, -1.4 SD) was noted, and her last brain MRI showed a remarkable volume reduction of the suprasellar tuberculoma.

Conclusion: Suprasellar tuberculoma can have a very dynamic presentation during the active stage of the disease, which can be reversed by prolonged antituberculosis treatment. Previous studies showed that the tuberculous process can also cause long term and irreversible changes in the hypothalamic-pituitary axis. Prospective studies are however needed in the pediatric population to know the exact incidence and type of pituitary dysfunction.

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一名患有髌上结核瘤的女孩的可逆性下丘脑肥胖症
简介蝶鞍上结核瘤在儿童中极为罕见,大多数患者表现为头痛、呕吐、视力障碍和垂体功能低下。在本病例报告中,我们介绍了一名患有肺结核的女孩,她的体重明显增加,并伴有垂体功能障碍,经过抗结核治疗后痊愈:病例介绍:一名 11 岁女孩因头痛、发热和厌食而就诊,随后逐渐发展为脑病状态,并伴有颅神经 III 和 VI 麻痹。脑部核磁共振成像显示,双侧颅神经II(包括视交叉)、III、V和VI沿脑膜造影剂捕获,多处造影剂增强的脑实质病变。结核菌素皮肤试验呈阴性,但干扰素-γ释放试验呈阳性。临床和放射学诊断与结核性脑膜脑炎一致。患者开始接受为期 3 天的脉冲皮质类固醇激素治疗和四联抗结核疗法,女孩的神经症状明显好转。然而,治疗几个月后,她的体重明显增加(1 年内增加了 20 千克),生长停滞。尽管她可能缺乏生长激素(循环中的胰岛素样生长因子-I [IGF-I] 104 μg/L [-2.4 SD]),但她的激素谱却显示出胰岛素抵抗(稳态模型评估-估计胰岛素抵抗 [HOMA-IR] 6.8)。随访的脑部磁共振成像显示,基底脑膜炎有所减轻,但星状上区的实质病变增加,并向内侧延伸至扁桃体核,现在该部位出现了一个巨大的结核瘤。抗结核治疗持续了 18 个月。患者的临床症状有所改善,体重指数(BMI)恢复到患病前的水平,生长速度也略有提高。在激素方面,胰岛素抵抗消失(HOMA-IR 2.5),IGF-I 增加(175 μg/L,-1.4 SD),最后一次脑部核磁共振成像显示髌上结核瘤体积明显缩小:结论:髌上结核瘤在疾病活动期的表现非常活跃,长期抗结核治疗可使其逆转。以往的研究表明,结核过程也会导致下丘脑-垂体轴发生长期、不可逆的变化。然而,要了解垂体功能障碍的确切发病率和类型,还需要对儿童群体进行前瞻性研究。
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来源期刊
Hormone Research in Paediatrics
Hormone Research in Paediatrics ENDOCRINOLOGY & METABOLISM-PEDIATRICS
CiteScore
4.90
自引率
6.20%
发文量
88
审稿时长
4-8 weeks
期刊介绍: The mission of ''Hormone Research in Paediatrics'' is to improve the care of children with endocrine disorders by promoting basic and clinical knowledge. The journal facilitates the dissemination of information through original papers, mini reviews, clinical guidelines and papers on novel insights from clinical practice. Periodic editorials from outstanding paediatric endocrinologists address the main published novelties by critically reviewing the major strengths and weaknesses of the studies.
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