Department of Urology, The University of Texas Southwestern Medical Center and Children's Medical Center Dallas, TX, USA.
J Pediatr Urol. 2010 Jun;6(3):265-9. doi: 10.1016/j.jpurol.2009.08.009. Epub 2009 Oct 24.
To report a novel cause of nocturnal enuresis (NE) and highlight the literature giving insight into this novel mechanism.
A 12-year-old morbidly obese female with 2-year history of nightly secondary monosymptomatic NE.
On evaluation, a history of severe sleep disturbed breathing was elicited. Anticipating obstructive sleep apnea (OSA), polysomnography was performed, detecting severe central sleep apnea (CSA) without OSA. Brain magnetic resonance imaging revealed severe Chiari malformation Type I (CM1) with abnormal cerebrospinal fluid dynamics. She had no other classic signs or symptoms of CM1. Neurosurgical decompression halted the NE and normalized nocturnal breathing and cerebrospinal fluid dynamics, confirming that the CSA was caused by the CM1 and resulted in the NE. A thorough literature review found no prior reports of CSA-induced NE. Since CSA and OSA differ by the absence of negative intrathoracic pressures in CSA, this case suggests that such pressures are not a key mechanistic component of SA-induced NE.
This first report of secondary NE caused by CSA from CM1 emphasizes obtaining a sleep history in the enuretic child, introduces a new cause of NE, and challenges hypotheses underlying SA-induced NE.
报告一种夜间遗尿（NE）的新病因，并重点介绍有关该新机制的文献。
一名 12 岁的病态肥胖女性，有两年夜间继发性单症状 NE 病史。
评估时，出现严重睡眠呼吸障碍史。预计为阻塞性睡眠呼吸暂停（OSA），进行了多导睡眠图检查，检测到严重中枢性睡眠呼吸暂停（CSA）而无 OSA。脑磁共振成像显示严重的 Chiari 畸形 I 型（CM1），伴有异常脑脊液动力学。她没有 CM1 的其他典型体征或症状。神经外科减压停止了遗尿，并使夜间呼吸和脑脊液动力学正常化，证实 CSA 是由 CM1 引起的，并导致了 NE。彻底的文献复习未发现 CSA 引起的 NE 的先前报告。由于 CSA 缺乏 CSA 中的负胸腔内压力，而 OSA 存在这种压力，因此该病例表明，这种压力不是 SA 引起的 NE 的关键机制组成部分。
这是首例由 CM1 引起的继发性 CSA 引起的 NE，强调在遗尿儿童中获取睡眠史，介绍了 NE 的新病因，并对 SA 引起的 NE 的假设提出了挑战。
