Tapias Luis F, Rogan Thomas J, Wright Cameron D, Mathisen Douglas J
Division of Thoracic Surgery, Massachusetts General Hospital, Boston, MA, USA.
Eur J Cardiothorac Surg. 2021 Jan 4;59(1):122-129. doi: 10.1093/ejcts/ezaa296.
Idiopathic laryngotracheal stenosis (ILTS) is an uncommon problem arising mostly in women. In some, it arises during or is exacerbated by pregnancy. Experience with management of patients with this rare association is limited. This study seeks to evaluate the management of patients with pregnancy-associated ILTS and compare outcomes to cases not associated with pregnancy.
Retrospective review of 15 patients undergoing surgical treatment of pregnancy-associated ILTS from 1971 to 2013. Variables of interest and airway outcomes were compared to patients with non-pregnancy-associated ILTS.
Pregnancy-associated ILTS was observed in 15/263 (5.7%) patients. Symptoms developed during their first pregnancy. When compared to non-pregnancy patients, these patients were younger (37 vs 47 years; P = 0.0003), had more prior tracheostomies (26.7% vs 10.9%; P = 0.085) and had more preoperative airway dilatations (86.7% vs 57.7%; P = 0.030). All patients completed pregnancy without complications and ultimately underwent laryngotracheal resection. The expression of hormonal receptors in the surgical specimens was similar in both groups (oestrogen receptors: 100% vs 75% and progesterone receptors: 71.4% vs 72.1%, in pregnancy and non-pregnancy patients, respectively). Airway outcomes were good/excellent in 13 (86.7%) patients with pregnancy-associated ILTS and 225 (90.7%) patients without pregnancy association (P = 0.642), and did not change when adjusting for other risk factors.
Pregnancy-associated ILTS is rare. The pathophysiology is unclear, but appearance of symptoms during pregnancy may suggest hormonal factors. To minimize foetal risk, dilatation during pregnancy followed by laryngotracheal resection after delivery is the preferred treatment. Pregnancy association does not seem to affect outcomes with expected satisfactory results in most patients.
特发性喉气管狭窄(ILTS)是一种罕见的疾病,多见于女性。部分患者在孕期发病或病情加重。对于这种罕见关联疾病患者的治疗经验有限。本研究旨在评估妊娠相关ILTS患者的治疗情况,并将其结果与非妊娠相关病例进行比较。
回顾性分析1971年至2013年间接受手术治疗的15例妊娠相关ILTS患者。将感兴趣的变量和气道结局与非妊娠相关ILTS患者进行比较。
在263例患者中有15例(5.7%)出现妊娠相关ILTS。症状在首次妊娠期间出现。与非妊娠患者相比,这些患者更年轻(37岁对47岁;P = 0.0003),既往气管切开术更多(26.7%对10.9%;P = 0.085),术前气道扩张更多(86.7%对57.7%;P = 0.030)。所有患者均顺利完成妊娠且无并发症,最终接受了喉气管切除术。两组手术标本中激素受体的表达相似(雌激素受体:妊娠患者和非妊娠患者分别为100%对75%,孕激素受体:71.4%对72.1%)。13例(86.7%)妊娠相关ILTS患者和225例(90.7%)非妊娠相关患者的气道结局为良好/优秀(P = 0.642),在调整其他危险因素后结果未改变。
妊娠相关ILTS较为罕见。其病理生理机制尚不清楚,但孕期症状的出现可能提示激素因素。为使胎儿风险最小化,孕期扩张治疗,产后行喉气管切除术是首选治疗方法。妊娠似乎不影响治疗结果,大多数患者可获得预期的满意结果。