蓝色橡皮痣综合征患者感染异种分枝杆菌的复杂病例导航

Muhammad Umer Riaz Gondal, Luke Rovenstine, F. Ansari, Z. Kiyani, Devi Parvathy Jyothi Ramachandran Nair, Toqeer Khan, Anthony Donato
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摘要

导言蓝橡皮痣综合征是一种罕见的静脉畸形疾病,据报道约有 200 例。我们为您介绍一例蓝橡皮痣综合征患者感染分枝杆菌的病例。病例描述:一名患有蓝色橡皮样痣综合征、哮喘和支气管扩张的 40 岁女性因呼吸急促和咳嗽来到肺科门诊就诊。她最近因支气管扩张加重而入院治疗,但有痰咳嗽和发烧症状持续恶化。最近的胸部 CT 扫描显示,右上叶纤维空洞病间隔稳定,两年来病情逐渐加重。一年前,她的复合分枝杆菌和异种分枝杆菌培养偶尔呈阳性,被认为是定植菌,没有接受治疗。最近的医院细菌培养和耐酸杆菌涂片均为阴性。她因支气管扩张加重被送往急诊科,六周后回到诊所,两次痰培养均检出克逊酵母菌。由于这很可能是导致她肺部空洞病变和频繁感染的原因,因此决定对其进行治疗。开始使用阿奇霉素、利福平和磺胺甲恶唑/三甲氧苄氨嘧啶。后来又静脉注射了阿米卡星。一年后,她终于在一家外院接受了右肺部分切除术。在大约三年的时间里,她一直在反复使用抗生素治疗M. xenopi,但非结核分枝杆菌的重复培养结果均为阴性。结论:由于M. xenopi感染的死亡率很高(可高达69%),建议至少治疗12个月。据我们所知,这是首例在蓝色橡皮痣综合征患者中感染 M. xenopi 的病例。
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Navigating a complex case of Mycobacterium xenopi in a patient with blue rubber bleb nevus syndrome
Introduction: Blue rubber bleb nevus syndrome is a rare disorder of venous malformations, with around 200 cases reported. We present a case of Mycobacterium xenopi infection in a patient with blue rubber bleb nevus syndrome. Case Description: A 40-year-old female with blue rubber bleb nevus syndrome, asthma, and bronchiectasis came to the pulmonology clinic with shortness of breath and a cough. She was recently admitted for a bronchiectasis exacerbation but continued to have a worsening productive cough and fevers. The most recent CT scan of the chest showed interval stable right upper lobe fibrocavitary disease, demonstrating gradual progression over two years. She had occasional positive cultures for Mycobacterium Avium Complex and M. xenopi one year previously, assumed to be a colonizer and not treated. Most recent hospital cultures were negative for bacteria and an acid-fast bacilli smear. She was sent to the emergency department for bronchiectasis exacerbation and returned to the clinic six weeks later with two sputum cultures growing M. xenopi. It was decided to treat M. xenopi as this was likely the cause of her cavitary lung lesion and frequent infections. Azithromycin, rifampin, and sulfamethoxazole/trimethoprim were initiated. Intravenous amikacin was added later on. She finally had a right partial lung resection done after one year at an outside hospital. She was on and off antibiotics for M. xenopi for approximately three years with negative repeat cultures for non-tuberculous mycobacteria. Conclusion: Due to the high mortality of M. xenopi infections (which can be as high as 69%), treatment of at least twelve months is recommended. To our knowledge, this is the first reported case of M. xenopi in a patient with blue rubber bleb nevus syndrome.
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