healthmedicinet

Case 1

All persons have given their informed consent prior to their inclusion in the study,
and all human studies have been approved by the local Ethics Committee and performed
in accordance with the ethical standards.

A 20-year-old woman was admitted to her local hospital in June, 2010 because of cough
and expectoration for 3 months. She was diagnosed as tracheal tuberculosis according
to the positive acid-fast bacilli in her sputum and granuloma-like lesions on the
tracheal wall with caseous necrosis by bronchoscopy with biopsy. She was given anti-TB
medications consisting of Isoniazid (INH), Rifampicin (RFP), Ethambutol (EMB) and
Pyrazinamide (PZA). However, her symptoms were not relieved, but deteriorated after
1 month. The bronchoscopy was performed again, revealing tracheal scar stenosis. The
biopsy showed chronic inflammation of the upper tracheal mucosa with fibrous tissue
proliferation. She was admitted to our hospital on December 9, 2010 for further treatment.

On admission, physical examinations showed that she was clear-minded, breath was 25
times per min, pulse was 93 beats per min, and blood pressure was 126/78 mmHg. On
chest examination, her breath sound was rough with wheeze on both sides. The result
of abdominal examination was normal. Pulmonary function testing (PFT) showed that
her pulmonary function was impaired [vital capacity (VC) =3.29 L (91.9 % pred), the
forced expired volume in 1 s (FEV1) =1.11 L (35.2 % pred), the ratio between FEV1
and the forced vital capacity (FEV1/FVC) =33.65 %]. A chest computed tomography (CT;
Fig. 1) revealed tracheal stenosis at the thoracic inlet, patchy, nodule-like and streaking
lesions with uneven density in both lobes of the lung, and normal lymph node sat mediastinum
and lung hilus.

Fig. 1. A chest computed tomography scan showing tracheal stenosis at thoracic inlet before
treatment in Case 1

After admission, she continued to receive anti-TB drugs (INH, RFP, EMB and PZA), and
then changed to anti-TB drugs [INH, RFP, EMB and streptomycin(SM)] due to liver injury.
Electronic bronchoscopy was performed at 3 days after admission (December, 13) under
general anesthesia. Mechanical ventilation was used to assist her breathing. The bronchoscopy
showed a ring-like scar stenosis about 2 cm below the normal glottis. The bronchoscope
(BF BC-260, Olympus Corporation, Tokyo, Japan) with an external diameter of4.9 mmcould
pass through the stenosis, but not the bronchoscope (BF IT-260, Olympus Corporation,
Tokyo, Japan) with the outer diameter of 5.9 mm. The bronchoscopy (BF BC-260) revealed
that the airway wall was rough, the narrowed airway was about 5 cm in length, and
the lower end of the stenosis was about 5 cm away from the sharp tracheal carina.
No abnormality in the bilateral lobar bronchus was observed.

The tracheal stenosis was treated using the MK58880balloon (1 cm in diameter, Boston
Scientific Cork Company, Ireland) with the pressure increased to 303.9 Kpa (3 atm)
to dilate the trachea for 10s during which the mechanical ventilation was stopped.
Subsequently, the pressure started to decrease and the mechanical ventilation was
recovered. The balloon dilatation was repeated 3 times. The bronchial lumen expanded
only slightly following the procedure (Fig. 2a, b). On the second day of the balloon dilatation, her symptoms including coughing, shortness
of breath, were alleviated. The wheeze and stridor of lungs were reduced under physical
examination.

Fig. 2. Photographs showing bronchus stenosis before and after treatment in Case 1. a Photographs showing bronchus lumen before treatment; b Photographs showing bronchus lumen immediately after treatment; c Photographs showing bronchus lumen 50 days after treatment

Ten days later, she received bronchoscopic re-examination of the airways and the results
still indicated that the bronchoscope (BF BC-260, Olympus Corporation, Tokyo, Japan)
could pass through the stenosis, but not the bronchoscope (BF IT-260, Olympus Corporation,
Tokyo, Japan). Thus, the high-pressure balloon dilatation treatment was performed
again during which the pressure of the balloon was increased to 5 atm and then maintained
for 10 s. After 8 inflation cycles, the bronchoscope (BF IT-260, Olympus Corporation,
Tokyo, Japan) can pass through the stenosis. However, her cough and shortness of breath
symptoms were deteriorated following the treatment, which may be caused by the presence
of bronchial edema and spasm. These symptoms were resolved by intravenous and intranasal
administration of dexamethasone using atomization device. On the third day after treatment,
her cough and shortness of breath symptoms were apparently ameliorated. The physical
examinations showed that wheeze and stridor of lungs were remarkably reduced. Then,
she was discharged.

Fifty days after the treatment, a repeat PFT showed that her pulmonary function was
remarkably improved (VC?=?3.40 L (94.8 % pred), FEV1?=?2.06 L (65.5 % pred), FEV1/FVC?=?60.51 %).
A repeat bronchoscopy showed that the ring-like scar stenosis was dilated, allowing
the passage of bronchoscopy (BF IT-260, Olympus Corporation, Tokyo, Japan) (Fig. 2c). During her 1 year outpatient follow-up, no tracheal stenosis was observed. Timeline
of past history of the patient was listed in Table 1.

Table 1. Timeline of case 1

Case 2

A 39-year-old female patient was admitted to our hospital on October 13, 2010 because
of cough and expectoration for 3 years, chest tightness and shortness of breath for
approximately 1 month. On November, 2007, she was ever admitted to a local hospital
with complaints of cough, expectoration and white phlegm, but not fatigue, night sweats,
fever and chest pain. The local hospital diagnosis was bronchial asthma. She was given
anti-inflammatory drugs, but her symptoms persisted.

On admission, physical examinations revealed that she was clear-minded, breath was
26 times per min, pulse was 100 beats per min, and blood pressure was 132/80 mmHg.
Chest examination revealed wheeze in the trachea, and rough breath sound with in aspiratory
and expiratory wheeze in both lungs. Her abdominal examination was normal. No pathological
syndrome was observed. Chest CT scan revealed pulmonary TB on both sides, especially
in the middle and lower lobe. Lower tracheal stenosis, right bronchus stenosis and
right pleural thickening were also observed (Fig. 3). Sputum smear for acid-fast bacilli was positive. Subsequently she was given combined
anti-TB treatment (INH, RFP, EMB and PZA) since December 13, 2010. On December 17,
2010, bronchoscopy was performed under local anesthesia, revealing that as shown in
Fig. 4a, the glottis was normal, false diverticula was present in the middle segment of trachea,
and stenosis was present in the middle and lower trachea, barely allowing the passage
of the bronchoscopy (BF BC-260, Olympus Corporation, Tokyo, Japan). Tracheal mucosal
congestion and swelling were present, and the left main bronchus and lobar bronchus
on the left side was patent, although with the presence of viscous secretions. However,
the right main bronchial stenosis was observed with caseous necrotic material attached
to the bronchial orifice and could not permit the passage of the bronchoscopy (BF
BC-260, Olympus Corporation, Tokyo, Japan). Our final diagnosis was active secondary
pulmonary tuberculosis, tracheal and right main bronchial tuberculosis. On January
12, 2011, the patient was administrated with endotracheal intubation. After a month,
the endotracheal catheter was removed and then, the patient was discharged.

Fig. 3. A chest computed tomography scan showing tracheal stenosis and right bronchial stenosis
of the patient in Case 2

Fig. 4. Photographs showing tracheal and bronchus stenosis in Case 2. a Photographs showing the middle and lower tracheal stenosis, and right main bronchus
stenosis with caseous necrotic material attached to the nozzle exit before the treatment;
b Photographs showing the narrowed trachea and right main bronchus immediately after
the first treatment; c Photographs showing the middle and lower tracheal, and right main bronchus after
the fifth treatment

On February18, 2011, she was re-admitted to our hospital. The bronchoscopic high-pressure
(6 atm) balloon dilatation (MK58880, Boston Scientific Cork Company, Ireland) was
performed in the middle and lower trachea for the patient under general anesthesia
and electrocardiogram (ECG) monitoring. Mechanical ventilation was applied to assist
the patient’s breathing. When the oxygen saturation was below 95 %, the balloon stopped
dilatation and started empting. The balloon dilatation lasted as long as 1.5 min for
each cycle. When the oxygen saturation recovered to be above 99 %, the second balloon
dilatation was performed and repeated. The bronchoscopic high-pressure (5 atm) balloon
dilatation was also performed under ECG monitoring for the right main bronchial stenosis
twice with5 min for each cycle. After the balloon dilatation, the BC-260bronchoscope
could pass through the middle and lower trachea stenosis but not the IT-260bronchoscope.
For the right main bronchus stenosis, BC-260 bronchoscope could not even pass (Fig. 4b). Her chest tightness, shortness of breath symptoms were improved after the balloon
dilatation treatment, but still could not tolerate PFT.

She received balloon dilatation treatment for 5 more times on March 28, April 22,
May 6, June 7 and July 18, 2011, respectively. On June 28, 2012, a repeat bronchoscopy
showed that the middle and lower trachea stenosis was dilated and patent without the
presence of congestion and swelling, allowing the passage of the IT-260 bronchoscope.
The orifice of the right main bronchus was still narrowed, could not permit the passage
of the bronchoscopy (BC-260), but the caseous necrotic materials disappeared (Fig. 4c). No restenosis occurred in the patient in 1 year outpatient follow-up. Timeline
of case 2 was listed in Table 2.

Table 2. Timeline of case 2

Literature review

We searched the WANFANG and PubMed database for articles published between 1984 and
2014 in the Chinese-and English-language literature. There were several studies on
the application of balloon dilatation in management of combination of main bronchus
and lobe bronchus stenosis, yet rare reports on specific treatment of TB-associated
tracheal stenosis by BBD were retrieved. The search of PubMed database yielded 13
articles. A total of 474 patients were involved in the retrieved articles from PubMed
database and the present study. The total information retrieved was summarized in
Table 38]–20]. Of the 474 patients, the tracheal stenosis of 4 infant patients results from endotracheal
intubation 10]. The etiologies of the 474 patients include bronchial carcinoma, other malignancy,
lung carcinoma post-tracheal resection, endotracheal intubation, tracheal tuberculosis,
prolonged mechanical ventilation, lung transplantation, Wegener’s granulomatosis,
idiopathic stenosis, post-tracheostomy, sarcoidosis and amyloidosis.

Table 3. Summary of clinical characteristics of the patients in the retrieved articles and
the present study