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完美风暴:一例伴有通气不足、下丘脑自主神经调节障碍、神经内分泌肿瘤(ROHHADNET)的快速发作性肥胖病例,合并心力衰竭、发作性睡病及盆腔神经内分泌肿瘤的罕见部位。

The Perfect Storm: A Case of Rapid-Onset Obesity With Hypoventilation, Hypothalamic, Autonomic Dysregulation, Neuroendocrine Tumor (ROHHADNET) With Heart Failure, Narcolepsy, and a Rare Location of a Pelvic Neuroendocrine Tumor.

作者信息

Roby Paul, Smith Beltran Gretta, Finch Casey, Malhotra Sonal, Reiling Krista, Dayyat Ehab, Birkemeier Krista, Raju Muppala, Macmurdo Colleen, Hernandez Edwin, Sagar Malvika

机构信息

Internal Medicine, Baylor Scott & White Health, Temple, USA.

Pediatrics, Baylor Scott & White Health, Temple, USA.

出版信息

Cureus. 2023 Dec 11;15(12):e50341. doi: 10.7759/cureus.50341. eCollection 2023 Dec.

Abstract

Rapid-onset obesity with hypothalamic dysfunction, hypoventilation, and autonomic dysregulation (ROHHAD) syndrome is a rare disease of concurrent respiratory dysfunction and autonomic dysregulation with endocrine abnormalities. ROHHADNET includes ROHHAD plus coexisting neuroendocrine tumors (NETs). We describe an eight-year-old boy, who originally presented at four years of age with rapid weight gain and hyperhidrosis and who developed mild obstructive sleep apnea (OSA). His clinical course was eventually complicated by hypoxic respiratory failure requiring admission to the pediatric intensive care unit (PICU). Echocardiogram at that time demonstrated dilated cardiomyopathy left ventricular ejection fraction (LVEF) of 28% at time of admission. His respiratory failure persisted despite average volume-assured pressure support (AVAPS) around the clock leading to tracheostomy placement for cardiopulmonary support. He also demonstrated autonomic instability with multiple pituitary hormone deficiencies. Computed tomography (CT) imaging of the abdomen and pelvis demonstrated a presacral soft tissue mass consistent with a tumor of neural crest origin. Daytime somnolence and confusion progressed and a low cerebrospinal fluid hypocretin level revealed a diagnosis of narcolepsy type 1.

摘要

伴有下丘脑功能障碍、通气不足和自主神经调节异常的快速发作性肥胖(ROHHAD)综合征是一种并发呼吸功能障碍、自主神经调节异常及内分泌异常的罕见疾病。ROHHADNET包括ROHHAD以及并存的神经内分泌肿瘤(NETs)。我们描述一名8岁男孩,他4岁时最初表现为体重快速增加和多汗,并发展为轻度阻塞性睡眠呼吸暂停(OSA)。其临床病程最终因缺氧性呼吸衰竭而复杂化,需要入住儿科重症监护病房(PICU)。当时的超声心动图显示扩张型心肌病,入院时左心室射血分数(LVEF)为28%。尽管持续进行平均容量保证压力支持(AVAPS),其呼吸衰竭仍持续存在,导致为进行心肺支持而实施气管造口术。他还表现出自主神经不稳定以及多种垂体激素缺乏。腹部和骨盆的计算机断层扫描(CT)成像显示骶前软组织肿块,符合神经嵴起源的肿瘤。白天嗜睡和意识模糊加重,脑脊液下丘脑分泌素水平降低,提示诊断为1型发作性睡病。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/83bd/10777333/ebb40ca4c6a9/cureus-0015-00000050341-i01.jpg

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