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C1 抑制剂缺乏症患者行牙科手术的短期预防:一项观察性研究。

Short-term prophylaxis in patients with angioedema due to C1-inhibitor deficiency undergoing dental procedures: An observational study.

机构信息

Department of Biomedical and Clinical Sciences Luigi Sacco, University of Milan, ASST Fatebenefratelli Sacco, Milan, Italy.

Odontoiatric Unit, ASST Fatebenefratelli Sacco, Milan, Italy.

出版信息

PLoS One. 2020 Mar 12;15(3):e0230128. doi: 10.1371/journal.pone.0230128. eCollection 2020.

Abstract

BACKGROUND

Patients affected by angioedema due to hereditary and acquired C1-inhibitor (C1-INH) deficiency (HAE and AAE, respectively) report trouble accessing dental care, due to the risk of a life-threatening oropharyngeal and laryngeal attack triggered by dental procedures. The aim of this study was to assess the identification of hurdles in receiving dental care, and the effectiveness of short-term prophylaxis (STP) in preventing angioedema attacks. In addition, the study evaluated the impact of dental care in angioedema disease. All patients affected by angioedema due to C1-INH deficiency who were treated in the dentistry outpatient department of ASST Fatebenefratelli Sacco hospital (Milan, Italy) between 2009 and 2017 were considered for the analysis. Data were collected from patients' records.

RESULTS

Twenty-nine patients were analyzed (27 with HAE and 2 with AAE). Of these, 63.0% reported that they had previously experienced hurdles in accessing dental care. Among patients with pathological oral status, at the first visit, 59.26% patients had moderate-to-severe oral disease. Seventy-five dental procedures were performed in 20 patients. Sixty procedures were preceded by STP (58 with plasma-derived C1-INH and 2 with danazol) in patients with/without long-term prophylaxis (LTP). Post-procedural attacks occurred in two patients. One HAE patient undergoing a tooth extraction without STP/LTP experienced a laryngeal attack. The other post-procedural attack occurred in an AAE patient with anti-C1-INH antibodies with STP with pdC1-INH. The angioedema disease did not worsen in any patient after dental care, but improved in four of them.

CONCLUSIONS

Most C1-INH-HAE patients reported hurdles in receiving dental care. STP protects against attacks after dental procedures. Treating oral diseases results in improvement in the frequency of attacks.

摘要

背景

遗传性和获得性 C1 抑制剂(C1-INH)缺乏症(HAE 和 AAE)患者报告在接受牙科护理时存在困难,因为牙科手术可能引发危及生命的口咽和喉水肿发作。本研究旨在评估在接受牙科护理方面遇到的障碍,并评估短期预防(STP)在预防血管性水肿发作方面的效果。此外,本研究还评估了牙科护理对血管性水肿疾病的影响。所有在 2009 年至 2017 年间在意大利米兰 ASST Fatebenefratelli Sacco 医院牙科门诊就诊的 C1-INH 缺乏症引起的血管性水肿患者均被纳入本研究。研究数据来自患者的病历记录。

结果

共分析了 29 名患者(27 名 HAE 患者和 2 名 AAE 患者)。其中,63.0%的患者报告在接受牙科护理方面存在障碍。在口腔状况异常的患者中,59.26%的患者在首次就诊时口腔疾病处于中重度。在 20 名患者中进行了 75 次牙科手术。60 次手术前,20 名患者(无长期预防或有长期预防)接受了 STP(58 名患者使用血浆衍生 C1-INH,2 名患者使用丹那唑)。2 名患者在手术后出现了发作。1 名未进行 STP/LTP 预防性治疗的 HAE 患者在拔牙后发生了喉水肿发作。另一次术后发作发生在接受 pdC1-INH 进行 STP 的 AAE 患者。在任何患者中,血管性水肿疾病在牙科护理后均未恶化,但有 4 名患者的疾病得到了改善。

结论

大多数 C1-INH-HAE 患者报告在接受牙科护理方面存在障碍。STP 可预防牙科手术后的发作。治疗口腔疾病可减少发作频率。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/925f/7067439/70c972f21b02/pone.0230128.g001.jpg

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