Department of Oral and Maxillofacial Diseases, University of Helsinki, P.O. Box 41, FI-00014, Helsinki, Finland.
Research Program for Clinical and Molecular Metabolism, Faculty of Medicine, University of Helsinki, Helsinki, Finland.
BMC Musculoskelet Disord. 2021 Jan 11;22(1):61. doi: 10.1186/s12891-020-03932-9.
Obstructive sleep apnea (OSA) is prevalent in individuals with Osteogenesis imperfecta (OI). To date, no study has investigated treatment of OSA in adult individuals with OI using positive airway pressure (PAP). This observational pilot study examined the adherence of adults with OI to treatment of OSA with PAP therapy, and the evolution of self-experienced sleepiness and depression symptoms before and after treatment.
We included 20 patients, with a mean age of 51 years, who represented varying severity of OI and displayed an apnea and hypopnea index ≥ 5 /sleeping hour as recorded by an overnight polysomnography. PAP therapy was proposed to all patients. Epworth Sleepiness Scale (ESS) questionnaire to evaluate daytime sleepiness, and a validated self-rating depression questionnaire to identify possible depression, were completed prior to PAP therapy and repeated after a minimum of one year. The datasets supporting the conclusions of this article are included within the article.
From the 20 patients, 15 initiated PAP therapy, and two patients later interrupted it. The mean PAP follow-up period was 1230 days. At baseline, an abnormally high ESS score was reported by 29% of the respondents, and an abnormally high number of symptoms suggesting depression by 29%. Follow-up questionnaires were completed by 60% of the patients, of whom 83% were adherent to PAP treatment. ESS score and depression symptoms did not decrease significantly with PAP therapy.
Patients with OI accepted well PAP therapy and remained compliant. Sleepiness and depression persisted unaltered despite good PAP adherence. These unexpectedly poor improvements in symptoms by PAP therapy may be due to subjective depression symptoms and the complexity of factors underlying persisting sleepiness in OI. Further research is needed to confirm this novel finding.
阻塞性睡眠呼吸暂停（OSA）在成骨不全症（OI）患者中较为常见。迄今为止，尚无研究使用气道正压通气（PAP）治疗 OI 成人 OSA。本观察性初步研究调查了使用 PAP 治疗 OSA 时 OI 成人的依从性，以及治疗前后自我报告的嗜睡和抑郁症状的演变。
我们纳入了 20 名患者，平均年龄 51 岁，他们代表了不同严重程度的 OI，并在夜间多导睡眠图记录中显示每小时呼吸暂停和低通气指数≥5。向所有患者提出了 PAP 治疗方案。使用 Epworth 嗜睡量表（ESS）问卷评估白天嗜睡，并用经过验证的自评抑郁问卷识别可能的抑郁，在 PAP 治疗前完成并在至少一年后重复。支持本文结论的数据集包含在本文中。
20 名患者中，有 15 名开始 PAP 治疗，有 2 名患者后来中断了治疗。平均 PAP 随访时间为 1230 天。在基线时，29%的受访者报告 ESS 评分异常高，29%的受访者报告异常多的抑郁症状。完成了随访问卷的有 60%的患者，其中 83%的患者坚持 PAP 治疗。ESS 评分和抑郁症状在 PAP 治疗后并未显著下降。
OI 患者对 PAP 治疗的接受程度较好且保持依从性。尽管 PAP 治疗依从性良好，但嗜睡和抑郁症状仍未改善。PAP 治疗对症状的改善不佳可能是由于主观抑郁症状和导致 OI 持续嗜睡的因素的复杂性。需要进一步研究来证实这一新发现。
