Bach Ádám, Ambrus Andrea, Iványi Béla, Tóbiás Zoltán, Alim Marvasti Gholam Hossein, Rovó László
Department of Otorhinolaryngology & Head- Neck Surgery, University of Szeged; Tisza Lajos krt. 111., 6725, Szeged, Hungary.
Department of Pathology, University of Szeged, Állomás u. 2, 6725, Szeged, Hungary.
Iran J Otorhinolaryngol. 2021 Mar;33(115):119-125. doi: 10.22038/ijorl.2020.47106.2547.
Immunoglobulin G4-related disease (IgG4-RD) is a systemic fibro-inflammatory disorder. Laryngotracheal manifestation is very rare; therefore, it is usually associated with complex diagnostic and therapeutic problems.
Herein, we report the case of a 35-year-old woman with idiopathic subglottic stenosis (ISGS) treated with one-step laryngotracheal reconstruction surgery. Postoperatively, the lesion was found to be a part of the IgG4-RD spectrum. Objective and subjective phoniatric tests, spirometry, and Quality of Life Questionnaire were used for the evaluation of postoperative functional results. Slide laryngotracheoplasty as a one-step surgery without stenting and tracheostomy ensured a sufficiently wide subglottic space with no adverse effect on voice quality. During a follow-up period of 22 months, endoscopy and computed tomography scan revealed no significant restenosis. The patient was able to return to premorbid activities of daily living without any further medical treatment.
The laryngeal involvement of IgG4-RD is uncommon; however, it is a manifestation that should be included in the differential diagnosis of subglottic stenoses (SGS). Furthermore, subglottic IgG4-RD might be a potential etiological factor of ISGS and acquired airway stenosis after short-term intubation. Slide laryngotracheoplasty might be a favorable solution without stenting and tracheostomy even in special cases of SGS.
免疫球蛋白G4相关性疾病(IgG4-RD)是一种全身性纤维炎症性疾病。喉气管表现非常罕见,因此通常伴有复杂的诊断和治疗问题。
在此,我们报告一例35岁患有特发性声门下狭窄(ISGS)的女性患者,接受了一期喉气管重建手术治疗。术后发现病变是IgG4-RD谱的一部分。使用客观和主观发声测试、肺活量测定和生活质量问卷对术后功能结果进行评估。滑动喉气管成形术作为一种无需支架置入和气管切开的一期手术,确保了足够宽的声门下间隙,且对嗓音质量无不良影响。在22个月的随访期内,内镜检查和计算机断层扫描显示无明显再狭窄。患者无需进一步治疗即可恢复病前的日常生活活动。
IgG4-RD的喉部受累并不常见;然而,它是声门下狭窄(SGS)鉴别诊断中应包含的一种表现。此外,声门下IgG4-RD可能是ISGS和短期插管后获得性气道狭窄的潜在病因。即使在SGS的特殊情况下,滑动喉气管成形术可能是一种无需支架置入和气管切开的有利解决方案。