先天性耐药结核病合并支气管肉芽肿1例并文献复习

doi:10.3969/j.issn.1004-583X.2024.08.011

摘要/Abstract

摘要：

目的报道1例先天性耐药结核病(congenital tuberculosis,CTB)合并支气管肉芽肿患儿诊断及治疗过程,旨在为此类疾病的诊断及治疗积累经验,为临床应用提供参考。方法分析1例于2022年4月22日收住我科治疗的先天性耐药结核病患儿临床资料,分析其临床特点、诊疗情况。结果患儿系胎龄34周+4天早产,顺产,试管婴儿,生后40 d出现咳嗽、喘息伴发热等症状,反复以肺炎入院治疗。患儿母亲因输卵管堵塞行体外人工受孕,筛查发现陈旧性肺结核。入院查体示呼吸促,三凹征阳性,右肺呼吸音稍低,双肺可闻及喘鸣音,肺CT示双肺多发炎性实变,双肺多发结节影,行纤维支气管镜可见双侧支气管大量肉芽组织增生,肺泡灌洗液示TB-PCR阳性, 二代测序示结核分枝杆菌,予口服利福平、异烟肼以及利奈唑胺抗结核治疗, 出院1个月复查肺CT及纤维支气管镜检查示病情进展,结合利福平耐药实时荧光定量核酸扩增检测技术(X-pert MTB/RIF)提示利福平耐药,遂改用左氧氟沙星、利奈唑胺、吡嗪酰胺、异烟肼四联抗结核治疗,并多次行纤维支气管镜下肉芽肿电凝电切术、冷冻术以及异物钳钳取术,解除气道堵塞。患儿定期复诊并行纤维支气管镜下介入治疗近7月余,现患儿呼吸平稳,双肺通气体良好,且未发生药物不良反应,继续随访中。结论先天性结核病症状不典型,容易漏诊,需加强纤维支气管镜在结核病诊疗中的应用。输卵管堵塞导致不孕症妇女经体外受精胚胎移植术受孕可能是CTB的主要原因。此外,对于结核耐药性问题,痰结核分枝杆菌及耐药基因检查可指导临床用药,但目前对于婴幼儿耐药结核的治疗方案及指导较少,仍需要进一步的研究。

关键词: 结核, 耐药, 支气管肉芽肿

Abstract:

Objective To report the diagnosis and treatment of a case of congenital drug-resistant tuberculosis combined with bronchial granuloma, thus summarizing the clinical experience and providing references for clinical management. Methods Clinical data of a child with congenital drug-resistant tuberculosis combined with bronchial granuloma admitted to our department on April 22, 2022 were analyzed, including the clinical characteristics, diagnosis and treatment. Results The child was prematurely, vaginally delivered at 34 weeks and 4 days of gestation, in an in vitro fertilization. At 40 days of age, the child developed symptoms of cough, wheezing, and fever, and repeatedly hospitalized for pneumonia. The mother of the child underwent in vitro artificial insemination due to tubal obstruction, and presented a history of obsolete pulmonary tuberculosis. Physical examination on admission showed rapid breathing, positivity for the Hoover's sign, slightly low respiratory sounds in the right lung, and wheezing sounds in both lungs. Lung CT showed multiple inflammatory consolidations and multiple nodules in both lungs, and extensive granulation tissue proliferation in bilateral bronchi on fiberoptic bronchoscopy. The tissue polymerase chain reaction for tuberculosis (TB-PCR) test showed positive Mycobacterium tuberculosis in the alveolar lavage fluid, and second-generation sequencing consistently showed Mycobacterium tuberculosis. The child was treated by oral rifampicin, isoniazid, and linezolid for antituberculosis therapy. One month after discharge, a follow-up lung CT and fiberoptic bronchoscopy showed disease progression. The rifampicin fluorescence quantitative nucleic acid amplification detection(X-pert MTB/RIF) assay indicated rifampicin resistance, and the child was then treated with a quadruple anti-tuberculosis therapy of levofloxacin, linezolid, pyrazinamide, and isoniazid. Fiberoptic bronchoscopy procedures, including granuloma electrocoagulation and resection, cryotherapy, and foreign body forceps extraction were performed for multiple times to relieve airway obstruction. The child had regular follow-up visits and underwent interventional therapy under fiberoptic bronchoscopy for nearly 7 months. At present, the child had a stable breathing, well-ventilated in both lungs, and no adverse drug reactions. The follow-up is ongoing. Conclusion Symptoms of congenital tuberculosis are atypical, and prone to a missed diagnosis. It is necessary to strengthen the application of fiberoptic bronchoscopy in the diagnosis and treatment of tuberculosis. Tubal blockage leading to infertility in women undergoing in vitro fertilization and embryo transfer may be the main cause of congenital tuberculosis. In addition, sputum Mycobacterium tuberculosis and drug resistance gene testing can guide clinical medication in patients with drug-resistant tuberculosis. However, there is currently limited guidance and treatment plans for drug-resistant tuberculosis in infants and young children, and the further research is needed.

Key words: tuberculosis, drug resistance, bronchial granuloma

中图分类号:

穆世茵, 邹映雪, 郭永盛, 翟嘉, 黄冰. 先天性耐药结核病合并支气管肉芽肿1例并文献复习[J]. 临床荟萃, 2024, 39(8): 747-751.

Mu Shiyin, Zou Yingxue, Guo Yongsheng, Zhai Jia, Huang Bing. Congenital drug-resistant tuberculosis combined with bronchial granuloma: A case report and literature review[J]. Clinical Focus, 2024, 39(8): 747-751.

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链接本文: https://huicui.hebmu.edu.cn/CN/10.3969/j.issn.1004-583X.2024.08.011

https://huicui.hebmu.edu.cn/CN/Y2024/V39/I8/747

图/表 5

图1 肺CT(2022年3月22日) 双肺多发炎性实变及结节影,双肺透过度不均匀,上纵隔饱满,双侧肺门增大,右主支气管分支欠通畅

Fig. 1 Chest CT (March 22,2022) revealed multiple inflammatory consolidation and nodular shadow in both lungs, with non-uniform permeability, enlarged upper mediastinal lymph nodes, enlarged bilateral hilum, and inferior patency of the right main bronchus

图2 肺CT(2022年4月23日) 双肺多发结节影,炎性实变较前进展,右肺上叶气肿

Fig. 2 Chest CT (23 April 2022) showd multiple nodules in both lungs, inflammatory consolidation progressed, upper lobe emphysema of the right lung

图3 复查肺CT(2022年6月6日) 利福平+异烟肼抗结核治疗1个月,双肺多发结节较前变化不著,炎性实变较前好转,右肺上叶气肿较前无著变

Fig.3 Chest CT (June 6,2022) presented after 1 month of anti-tuberculosis treatment with rifampicin,isoniazid and linezolid, multiple nodules in both lungs were not changed, inflammatory consolidation was better than before, and emphysema in the upper lobe of the right lung was not changed than before

图4 纤维支气管镜下可见左右支气管开口大量支气管肉芽肿及干酪样坏死堵塞气道,导致通气不畅

Fig.4 Bronchoscopy showing luminal obstruction of the left and right main bronchus caused by bronchial granuloma and caseous, leading to poor ventilation

图5 纤维支气管镜下行支气管肉芽肿电凝电切术及冷冻术后,气道较前通畅

Fig. 5 Therapeutic bronchoscopy,including bronchial granuloma electrocoagulation and cryoablation, is available to relieve tracheobronchial stenosis caused by endobronchial TB

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