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病例漫谈99:胸部结核的罕见临床表现:更好地了解该病症的必要性 [复制链接]



Tuberculosis is one of the most common systemicinfectious disease, and a major public health problem all over the world. Ithas several forms of presentation and clinical manifestations and remains animportant cause of preventable death in the adult population. An uncommon formof pulmonary tuberculosis is endobronchial tuberculosis, which accounts forabout 10–40% of the cases of active tuberculosis. It may be complicated bytracheobronchial stenosis, and fistula formation is an unusual complication,mainly in the region of the pleura and less frequently in the oesophagus (BEF).Increased risk of acquiring these rare complications occurs largely inimmunocompromised people (e.g., HIV, malignancies, and patients undergoingimmunesuppressi-

vetherapy). In these cases, the diagnosis is often missed or delayed.



Tubercular lymphadenitis can also lead to fistulaformation through a process of caseum necrosis and opening of a fistula betweenthe bronchus and oesophagus.


The purpose of this article is to discuss a case reportof thoracic tuberculosis that showed an unusual presentation, emphasizing theimportance of an early diagnosis in order to reduce both the risk of mortalityand prevent the spread of infection to the community.


Case Report

An immunocompetent 73-year-old Caucasian man who is anex-smoker of 42 pack-years, presented in the past 6 months with a history ofaccessional nonproductive cough that appeared during meals, especially afteringestion of liquids, and a significant weight loss of 10 kg in a year. He hadno fever, dyspnea, night sweats, chest pain, or hemoptysis. There was no familyhistory of tuberculosis or previous contact with a TB patient. He had nosignificant past medical or surgical history and prior to the onset of coughingduring meals, remarkable respiratory symptoms were not reported.



During the first 6 months, on the basis of a clinicalsuspicion of gastroesophageal reflux, he was initially, treated by a generalpractitioner, with a standard dose of proton pump inhibitors (PPI) therapy. Dueto the lack of a treatment response, in June 2013, he underwent a bariumcontrast swallow test. The chest X-ray examination performed with a bariumcontrast showed the presence of a fistula extending from the lower tract of theesophagus to the right main bronchus (Figure 1), without evidence of activeparenchymal lung infiltrates or pleural involvement.


Figure 1. Fistula demonstrated by barium contrast swallowfrom

the middle third and lower third of the esophagus to theright main bronchus.

1. 钡餐造影法可见从食管中下1/3到右主支气管的瘘道。

He was then endoscopically treated by application ofmetal clips in the esophagus for fistula closure. No chest CT scan wasperformed in this first phase of the diagnostic process.


For persistence of cough, on July 2013 the patient wassent to our Clinic where, after chest CT scan, he underwent a flexiblefiberoptic bronchoscopy in an outpatient setting.


Routine blood investigation results were normal; only anincrease of PCR (3.45 mg/dl) was found. Serum QuantiFERON testing was positive(2.41 IU/mL). The remaining systemic examination revealed no abnormality.

患者血常规检测正常;仅PCR3.45毫克/升)检测结果偏高。血清 QuantiFERON结果呈阳性(2.41 IU /毫升),其他全身检查未见异常。

The chest CT scan “with contrast” showed a significantenlargement of mediastinal lymph nodes (Figure 2A) that caused trachealcompression with deflection to the right side and reduction of its lumen forpresence of an endoluminal vegetating lesion. Several other enlarged lymphnodes were seen in the right supraclavicular and axillary area, but there wasno associated parenchymal infiltrates.


Figure 2. (A) CT scan of chest (coronal view)pre-antitubercolosis treatment showing a mediastinal lesions.

(B) CT scan of chest (coronal view) during antitubercolosistreatment showing regression of mediastinal lesions.

2.(A) CT扫描(冠状面)显示为抗结核治疗前纵膈内淋巴结肿大

(B) CT扫描(冠状面)显示抗结核治疗后纵膈淋巴结缩小。

Bronchoscopy revealed a mild inflammation of thebronchial mucosa and the presence of different vegetating lesions in thecarina, on the medial wall of the main bronchi, and on the lateral wall of theintermedius bronchus (Figure 3).


Figure 3. Bronchoscopic view showing a vegetatinglesions.

3. 支气管镜下视图显示增殖性病变

Histopathology revealed moderate chronic granulomatousinflammation characterized by the presence of lymphocytes,
histiocytes, and Langhans-type giant cells. Bronchialaspirate test results for routine culture, fungal, and AFB (acid-fast bacilli)were negative.


The patient was re-evaluated after a week byesophagoscopy and bronchoscopy, mainly for worsening symptoms and for thereevaluation of the fistula. The first reconfirmed the persistence ofbronchoesophageal fistula, so further endo-clips were placed. Thebronchoscopy control showed that the vegetating lesions had increased in size,so a laser treatment was performed with lumen desobstruction. Bronchialaspirate testing confirmed the presence of a marked lymphocytic and histiocyticinflammatory infiltrate with giant cells and granulomatous component withoutcentral necrosis. Results of a microscopic examination and molecular test (PCR)for Koch’s bacillus were negative.


Based on the pathological pattern, we decided to start astandard antitubercular treatment with isoniazid 300 mg daily, rifampin 600 mgdaily, pyrazinamide 1500 daily, and ethambutol 1200 mg daily. The patient wasplaced in isolation with negative pressure inside the room.


After 40 days, the culture of the first bronchoscopicbiopsy demonstrated the growth of Mycobacterium tuberculosis (Lowenstei-Jensenmedium and Bactec Mycobacteria Growth Indicator Tube (MGIT) 960 TB System). Thedrug susceptibility test did not show any resistance to the first-line drugs.Our patient continued anti-tuberculous treatment for 2 months, followed byrifampicin and isoniazid treatment for the following 4 months.

40天后,首次支气管镜检培养证实结核分枝杆菌的生长(Lowenstei-Jensen培养基和分枝杆菌生长指示管(MGIT960 TB系统)。药敏试验未表现出对一线药物的任何耐药性。患者进行了持续2个月的抗结核治疗,而后接受了4个月的利福平和异烟肼治疗。

During antitubercular treatment, symptoms progressivelyimproved. CT and bronchoscopy performed 2 months later showed regression oflymph node enlargement (Figure 2B) and the disappearance of intraluminallesions in the tracheobronchial tree. Bronchoscopy also showed thebronchoesophageal fistula closure (Figure 4).

抗结核治疗过程中症状逐渐减轻。2个月后行CT和支气管镜检查显示淋巴结缩小(图 2B)和支气管腔内病变消失。支气管镜也显示支气管食管瘘闭合(图4)。

Figure 4.Bronchoscopic view: scar tissue of the fistula.

4. 支气管镜下视图:肛瘘的疤痕组织

At 6-month and 1-year follow-up, physical andbronchoscopy examinations did not show evidence of recurrence, a chest CT scanat 1 year demonstrated no evidence of mediastinal lymphadenopathy, and symptomsof patient were resolved. The final diagnosis was “bronchoesophageal fistula inendobronchial tuberculosis and mediastinal lymphadenopathy”.



Bronchoesophageal or tracheoesophageal fistula isextremely rare in adults. It is more frequently a congenital condition;otherwise, the acquired forms, are usually secondary to primary neoplasm(benign, malignant, or metastatic), infectious diseases (tuberculosis,histoplasmosis, actinomycosis, and syphilis), traumatic events (sequels ofsurgical procedures), and connective tissue diseases. Broncho-esophagealfistula (BEF) in adults is commonly due to malignancy, mainly oesophaguscarcinoma and, less frequently, lymphoma, carcinoma of the lungs or trachea.Benign BEF is a rare condition. The fistula has a short course and usually thecommunication is pervious, permitting the passage of air in the stomach andliquids in the airways. The clinical symptoms and signs that occur as a resultof attempted oral feeding are closely related to the size of the fistula andare characterized by chest pain, dyspnea, barking cough, cyanosis, hemoptysis,and sputum production; in some cases episodes of pneumonia and otherrespiratory infections can occur through a mechanism of “aspiration”.



Endobronchial tuberculosis (EBTB), defined as“tuberculous infection of the tracheobronchial tree with microbial andhistopathological evidence”, is a particular form of TB. Described for thefirst time by Mortem in 1698, represents about 10–40% of cases of active TB.It’s more common in young adults, with a female predominance, and only 15% inelderly patients. Often dangerous for its consequences (trachea andbronchostenosis, recurrent pneumonia, atelectasis and respiratory failure) ispotentially an important source of infection spread in the community. Itspathogenesis remains unclear; however, different mechanisms have been suggestedand the more plausible are: direct implantation of Mycobacterium tuberculosis(Mt) in the bronchus after their inhalation, or direct extension of infectionfrom parenchymal lesions or erosion and infiltration from adjacent mediastinallymph nodes into the bronchus.


The mediastinal tuberculous lymphadenitis is rare in theabsence of simultaneous lung involvement in immunocompetent adultswhile it maybe observed more frequently in HIV-positive patients and in developingcountries with high rates of TB just as it is in sub-Saharan Africa.


During primary TB, tubercle bacillus reaches themediastinal or hilar lymph nodes causing lymph node enlargement but theparenchymal infiltrate in an immunocompetent subject may resolve withoutsequels at conventional radiography. Mycobacterium tuberculosis may stayinactive (dormant) for many years inside the lymph nodes and becomes activeagain during decreased immune status, as for example in the elderly.


Our clinical case was characterized by severalcriticisms:


• The considerable delay with which the patient has beensubject to a chest CT scan and bronchoscopy. Bronchoscopy should be donepromptly in the case of a fistula suspicion. The cough during meals wouldsuggest the presence of a bronchoesophageal fistula and this suspicion involvesthe assessment of the cause.


• Usually, it is thought that microbiological examinationof bronchial aspirate and BAL are positive for Mycobacterium tuberculosis inpatients with EBTB, and that examination provides a good diagnostic yield. Astudy conducted by Ozkaya et al. highlights the difficulty of thebacteriological diagnosis of EBTB based on BAL analysis, bronchial aspirate, orsputum analysis, and showed that a high diagnostic yield was obtained throughthe histopathologic examinations of bronchial biopsies, confirmed in ourclinical case.


• Chest x-ray, performed on our patient in the firstinstance, did not pose a suspicion of TB. There were no direct signs such asparenchymal consolidation, or pneumonia and/or excessive gastric and intestinalgasification as expression of communication between the oesophagus and airway,specific lesions of active TB or inactive as scarring; complications ofendobronchial TB (recurrent pneumonia or atelectasis) or mediastinum lymph nodeenlargement. In fact, up to 20% of patients with EBTB have a normal chestradiograph.


• The presence of non-specific symptoms such as cough,only present during meals, simulated a gastro-esophageal reflux disease. Infact, despite the bronchial obstruction, the patient didn’t have any symptomsof endobronchial tuberculosis. EBTB, may sometimes present with a veryinsidious onset and, in some cases, it may simulate other pathologicalconditions such as bronchogenic carcinoma or bronchial asthma. This symptom canmimic many diseases, misleading the doctor for a proper diagnosis. Moreover,several studies showed that, in the elderly, the classic signs and symptoms ofTB, such as fever, weight loss, night sweats, and hemoptysis and sputumproduction are sometimes absent compared to young adult.


• Pathogenesis of bronchoesophageal fistula in thisspecific case report remains difficult to interpret. It may be secondary toendobronchial tuberculosis, through a mechanism of erosion of the bronchialwall, involvement of mediastinal lymph nodes and then fistula formation into theesophagus. However, the absence of parenchymal involvement, makes unlikely thismechanism. Much more probable, was a reactivation of a primary infection inperitracheal and peribronchial lymph nodes, as a consequence of animmunodepression state, subsequent lymph node erosion intooesophagus and bronchus and fistulization with a consequent implantation of Mtin the bronchial mucosa and secondary endobronchial TB onset. Similar studiesshow that both mechanisms are uncommon or rare complications of thoracic tuberculosis.As far as we know, no cases have been reported in the literature withsimultaneous combination of bronchoesophageal fistula, endobronchialtuberculosis without parenchymal involvement, mediastinal tuberculouslymphadenitis in the elderly.


Despite the delay and the mismanagement, the patient hasresponded optimally to a standard treatment with isoniazid, rifampicin,pyrazinamide and ethambutol for 2 months, followed by isoniazid and rifampicinfor a further 4 months. The 1-year follow-up showed healing of tuberculosis,without evidence of recurrence.



Bronchoesophageal fistula is a rare manifestation of acommon disease such as tuberculosis.



We know that early diagnosis and proper treatment maymodify the natural course of this disease and increase the rate of healing.Therefore, if not treated, severe complications may occur, endangering thepatient’s life.


The difficulty of this case report, associated with thelow level of knowledge of the illness and its serious complications, caused adiagnostic delay, deferring the start of an effective antitubercular treatment.


We can not forget that a normal chest radiograph and thepresence of nonspecific symptoms do not exclude the diagnosis of tuberculosis,but even the best early detection is of no use if subsequent treatment isinadequate, or downright wrong.


Medicool医库软件 王露黔 赵美凤 编译

原文来自 Am J Case Rep

最后编辑百里小溪 最后编辑于 2016-07-11 15:07:04
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