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罗哈达综合征之谜:挑战与未来策略

The Enigma That Is ROHHAD Syndrome: Challenges and Future Strategies.

作者信息

Hawton Katherine, Giri Dinesh, Crowne Elizabeth, Greenwood Rosemary, Hamilton-Shield Julian

机构信息

Bristol Royal Hospital for Children, University Hospitals Bristol and Weston NHS Foundation Trust, Bristol BS2 8BJ, UK.

Translational Health Sciences, University of Bristol, Bristol BS8 1QU, UK.

出版信息

Brain Sci. 2024 Oct 23;14(11):1046. doi: 10.3390/brainsci14111046.

Abstract

Rapid-onset obesity with hypoventilation, hypothalamic dysfunction, and autonomic dysregulation (ROHHAD) is a rare syndrome presenting in early childhood associated with a high risk of mortality between 50 and 60%. It is characterised by rapid, early onset of obesity between 1.5-7 years, along with central hypoventilation and hypothalamic dysfunction, such as central hypothyroidism, hyperprolactinemia, disorders of sodium and water balance, growth hormone deficiency, adrenocortical insufficiency, or disorders of puberty and features of autonomic dysregulation. Up to half of cases have neural crest tumours, most commonly ganglioneuromas or ganglioneuroblastomas. The incidence of ROHHAD syndrome in any population is unknown. Currently, there is no specific diagnostic or genetic biomarker for ROHHAD, and diagnosis is based on clinical signs and symptoms, which is often challenging, and consequently may be delayed or unrecognised. Early diagnosis is important, as without intervention, ROHHAD is associated with high morbidity and mortality. Aetiology remains unclear; an autoimmune origin has been postulated, with immunosuppressive agents being used with variable benefit. With no cure, multidisciplinary management is largely supportive. Therefore, there are many unanswered questions in ROHHAD syndrome. In this review article, we outline the challenges posed by ROHHAD syndrome, including aetiology, genetics, diagnosis, screening, management, and prognosis. We present research priorities to tackle these issues to improve outcomes.

摘要

快速发作性肥胖伴通气不足、下丘脑功能障碍和自主神经调节异常(ROHHAD)是一种罕见综合征,于儿童早期发病,死亡率高达50%至60%。其特征为在1.5至7岁之间迅速、早期出现肥胖,同时伴有中枢性通气不足和下丘脑功能障碍,如中枢性甲状腺功能减退、高催乳素血症、钠和水平衡紊乱、生长激素缺乏、肾上腺皮质功能不全、青春期障碍以及自主神经调节异常的特征。多达一半的病例患有神经嵴肿瘤,最常见的是神经节瘤或神经节神经母细胞瘤。ROHHAD综合征在任何人群中的发病率均未知。目前,尚无针对ROHHAD的特异性诊断或基因生物标志物诊断基于临床体征和症状,这往往具有挑战性,因此可能会延迟诊断或无法识别。早期诊断很重要,因为未经干预的ROHHAD会导致高发病率和死亡率。病因仍不清楚;有人推测其起源于自身免疫,免疫抑制剂的使用效果不一。由于无法治愈,多学科管理主要是支持性的。因此,ROHHAD综合征存在许多未解决的问题。在这篇综述文章中,我们概述了ROHHAD综合征带来的挑战,包括病因、遗传学、诊断、筛查、管理和预后。我们提出了应对这些问题以改善治疗结果的研究重点。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/663a/11591771/aabb8ebac2f5/brainsci-14-01046-g001.jpg

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