Nd Perera Gamage, C Jayasinghe Anusha, D Dias Lalindra, Kulatunga Aruna
National Hospital of Sri Lanka, Colombo, Sri Lanka.
BMC Res Notes. 2012 Aug 20;5:447. doi: 10.1186/1756-0500-5-447.
Takayasu arteritis is a large vessel vasculitis occurring in young females. We report a rare presentation of Takayasu arteritis in a Sri Lankan woman. She presented with bronchiectasis and left recurrent laryngeal nerve palsy prior to the onset of vascular symptoms. This case illustrates an atypical presentation of this disease and the diagnostic dilemma that the physician may be faced with.
A 39-year-old woman presented with chronic cough, haemoptysis and hoarseness of voice. She had left recurrent laryngeal nerve palsy and high inflammatory markers on investigation. CT thorax revealed aortic wall thickening and traction bronchiectasis. 2 D echocardiogram revealed grade 1 aortic regurgitation compatible with aortitis. She did not have weak peripheral pulses or a blood pressure discrepancy and did not meet American College of Rheumatology (ACR) criteria for diagnosis of Takayasu arteritis at this stage. Tuberculosis, syphilis and sarcoidosis was excluded. While awaiting angiography, she developed left arm claudication and a pericardial effusion. Angiography revealed evidence of Takayasu arteritis and absence of flow in the left subclavian artery. Takayasu arteritis was diagnosed at this stage after a period of eight months from the onset of initial symptoms. She is currently on prednisolone, azathioprine and aspirin.
Bronchiectasis and recurrent laryngeal nerve palsy is a rare presentation of Takayasu arteritis. Atypical presentations can occur in Takayasu arteritis prior to the onset of vascular symptoms. Elevation of inflammatory markers are an early finding. A high degree of suspicion is needed to identify these patients in the early course of the disease.
大动脉炎是一种发生于年轻女性的大血管血管炎。我们报告了一名斯里兰卡女性大动脉炎的罕见表现。她在血管症状出现之前就表现为支气管扩张和左侧喉返神经麻痹。该病例说明了这种疾病的非典型表现以及医生可能面临的诊断困境。
一名39岁女性因慢性咳嗽、咯血和声音嘶哑就诊。检查发现她有左侧喉返神经麻痹且炎症指标升高。胸部CT显示主动脉壁增厚和牵拉性支气管扩张。二维超声心动图显示1级主动脉瓣反流,符合主动脉炎表现。她没有外周脉搏减弱或血压差异,且此时不符合美国风湿病学会(ACR)大动脉炎的诊断标准。已排除结核病、梅毒和结节病。在等待血管造影期间,她出现了左臂间歇性跛行和心包积液。血管造影显示有大动脉炎的证据且左锁骨下动脉无血流。从最初症状出现起8个月后,此时诊断为大动脉炎。她目前正在服用泼尼松龙、硫唑嘌呤和阿司匹林。
支气管扩张和喉返神经麻痹是大动脉炎的罕见表现。大动脉炎在血管症状出现之前可出现非典型表现。炎症指标升高是早期表现。在疾病早期需要高度怀疑以识别这些患者。