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一名12岁男孩在接受高剂量生长激素(GH)治疗后出现与中枢性睡眠呼吸暂停相关的Chiari I型畸形:病例报告。

Chiari type 1 malformation associated with central sleep apnea after high dose growth hormone (GH) therapy in a 12-year-old boy: A case report.

作者信息

Mori Toshihiko, Nishino Eri, Jitsukawa Tomomi, Hoshino Emiko, Hirakawa Satoshi, Kuroiwa Yuki, Fuse Shigeto, Yoto Yuko, Tsutsumi Hiroyuki

机构信息

Department of Pediatrics, NTT East Sapporo Hospital, Hokkaido, Japan.

Department of Pediatrics, Sapporo Medical University School of Medicine, Hokkaido, Japan.

出版信息

Clin Pediatr Endocrinol. 2018;27(1):45-51. doi: 10.1297/cpe.27.45. Epub 2018 Jan 30.

Abstract

We describe the case of a short-statured 12-yr-old boy who developed a Chiari type 1 malformation associated with central sleep apnea after administration of high-dose GH therapy, which he had been receiving since the age of 10 yr and 4 mo. He responded well to GH therapy, and his height increased by 18.8 cm in 2 yr. At 12 yr and 4 mo of age, his mother reported that he had developed sleep apnea during the previous year and it had worsened over a month prior to presentation at our hospital. Otolaryngological examination did not reveal tonsillar or adenoidal hypertrophy. Polysomnography demonstrated severe central sleep apnea with an apnea-hypopnea index of 46.5/h. Sagittal T1-weighted magnetic resonance imaging (MRI) demonstrated herniation of the cerebellar tonsils 15 mm below the foramen magnum into the cervical spinal cord. Continuous positive airway pressure therapy initiated prior to performing neurosurgery was ineffective. Following uncomplicated foramen magnum decompression, his breathing pattern during sleep returned to normal. Sagittal MRI examination should be considered in patients who develop sleep apnea during/following administration of GH therapy.

摘要

我们描述了一名身材矮小的12岁男孩的病例,该男孩在接受高剂量生长激素(GH)治疗后出现了与中枢性睡眠呼吸暂停相关的Chiari I型畸形,他从10岁4个月起就开始接受这种治疗。他对GH治疗反应良好,2年内身高增加了18.8厘米。在12岁4个月时,他的母亲报告说他在前一年出现了睡眠呼吸暂停,并且在到我院就诊前一个月病情恶化。耳鼻喉科检查未发现扁桃体或腺样体肥大。多导睡眠图显示严重的中枢性睡眠呼吸暂停,呼吸暂停低通气指数为46.5次/小时。矢状位T1加权磁共振成像(MRI)显示小脑扁桃体疝入枕骨大孔下方15毫米处的颈脊髓。在进行神经外科手术之前开始的持续气道正压通气治疗无效。在进行简单的枕骨大孔减压术后,他睡眠期间的呼吸模式恢复正常。对于在GH治疗期间/之后出现睡眠呼吸暂停的患者,应考虑进行矢状位MRI检查。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/3c03/5792821/88ca9a42fc4d/cpe-27-045-g001.jpg

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