Department of Pediatric Neurology, Chelsea and Westminster Hospital NHS Foundation Trust, 369 Fulham Road, London SW10 9NH, UK ; Unit of Genetics and Paediatric Immunology, Department of Pediatrics, University of Messina, Italy.
National Center for Rare Diseases, Istituto Superiore di Sanità, Rome, Italy ; Institute of Neurological Sciences, National Research Council, Catania, Italy.
Int J Endocrinol. 2014;2014:282489. doi: 10.1155/2014/282489. Epub 2014 Sep 3.
Various neurological and psychiatric manifestations have been recorded in children with adrenal disorders. Based on literature review and on personal case-studies and case-series we focused on the pathophysiological and clinical implications of glucocorticoid-related, mineralcorticoid-related, and catecholamine-related paediatric nervous system involvement. Childhood Cushing syndrome can be associated with long-lasting cognitive deficits and abnormal behaviour, even after resolution of the hypercortisolism. Exposure to excessive replacement of exogenous glucocorticoids in the paediatric age group (e.g., during treatments for adrenal insufficiency) has been reported with neurological and magnetic resonance imaging (MRI) abnormalities (e.g., delayed myelination and brain atrophy) due to potential corticosteroid-related myelin damage in the developing brain and the possible impairment of limbic system ontogenesis. Idiopathic intracranial hypertension (IIH), a disorder of unclear pathophysiology characterised by increased cerebrospinal fluid (CSF) pressure, has been described in children with hypercortisolism, adrenal insufficiency, and hyperaldosteronism, reflecting the potential underlying involvement of the adrenal-brain axis in the regulation of CSF pressure homeostasis. Arterial hypertension caused by paediatric adenomas or tumours of the adrenal cortex or medulla has been associated with various hypertension-related neurological manifestations. The development and maturation of the central nervous system (CNS) through childhood is tightly regulated by intrinsic, paracrine, endocrine, and external modulators, and perturbations in any of these factors, including those related to adrenal hormone imbalance, could result in consequences that affect the structure and function of the paediatric brain. Animal experiments and clinical studies demonstrated that the developing (i.e., paediatric) CNS seems to be particularly vulnerable to alterations induced by adrenal disorders and/or supraphysiological doses of corticosteroids. Physicians should be aware of potential neurological manifestations in children with adrenal dysfunction to achieve better prevention and timely diagnosis and treatment of these disorders. Further studies are needed to explore the potential neurological, cognitive, and psychiatric long-term consequences of high doses of prolonged corticosteroid administration in childhood.
各种神经和精神表现已在肾上腺疾病患儿中被记录。基于文献回顾和个人病例研究和病例系列,我们重点关注了糖皮质激素相关、盐皮质激素相关和儿茶酚胺相关儿科神经系统受累的病理生理学和临床意义。儿童库欣综合征可与长期认知缺陷和异常行为相关,即使在高皮质醇血症得到解决后也是如此。在儿科年龄组中过度使用外源性糖皮质激素替代治疗(例如,在治疗肾上腺功能不全时)已被报道与神经系统和磁共振成像(MRI)异常相关(例如,髓鞘延迟和脑萎缩),这是由于潜在的皮质类固醇相关髓鞘损伤在发育中的大脑中,并可能损害边缘系统的发生。特发性颅内高压(IIH)是一种发病机制尚不清楚的疾病,其特征为脑脊液(CSF)压力升高,已在皮质醇增多症、肾上腺功能不全和醛固酮增多症患儿中被描述,反映了肾上腺-脑轴在调节 CSF 压力平衡中的潜在潜在参与。由于儿童的肾上腺腺瘤或皮质或髓质肿瘤引起的高血压与各种与高血压相关的神经系统表现有关。通过儿童期的中枢神经系统(CNS)发育和成熟是由内在、旁分泌、内分泌和外部调节剂严格调节的,其中任何因素的失调,包括与肾上腺激素失衡相关的因素,都可能导致影响儿科大脑结构和功能的后果。动物实验和临床研究表明,发育中的(即儿科)中枢神经系统似乎特别容易受到肾上腺疾病和/或皮质类固醇超生理剂量引起的改变。医生应意识到肾上腺功能障碍儿童可能出现的潜在神经表现,以更好地预防和及时诊断和治疗这些疾病。需要进一步的研究来探讨儿童期长期大剂量皮质类固醇给药的潜在神经、认知和精神长期后果。