Missed bronchial web in a 4-years old boy with foreign
body aspiration: a case report
Abstract:Congenital bronchial webs are extremely rare in incidence and usually
remained undiagnosed due to non-specific symptoms. Herein, we reported a
4-years-old case of bronchial web who was initially undiagnosed upon
bronchoscopy following foreign-body aspiration and afterward
misdiagnosed as childhood-asthma through his consistent cough and
exertional dyspnea for several months.
Keywords: congenital tracheal stenosis, bronchial
stenosis, chest 3d-ct; virtual bronchoscopy, bronchoscopy.
Introduction: Bronchial stenosis could be either
acquired following trauma, infection, inflammation, and autoimmune
condition or congenital due to bronchial webs though scarce in incidence
[1]. Congenital bronchial webs are often undiagnosed during infancy
and misdiagnosed later as they are not presented with specific
manifestations. Bronchial webs could sometimes mimic asthma or
foreign-body aspiration presentations. Patients mainly complain
shortness of breath, especially during exertion, persistent cough,
wheezing, or even infection[2]. The true incidence of the bronchial
web is not precisely known as they may go unrecognized throughout the
life. It is estimated that 1 in 10,000 births could be afflicted with
bronchial webs[3]. Lung computed tomography (CT) scan could be
helpful for screening of airway anomalies. Moreover, bronchoscopy is
both diagnostic and a therapeutic option. Herein, we described a
4-year-old boy with a right bronchial web who had been primary diagnosed
as case of foreign body aspiration and afterward treated approaching
childhood-asthma.
Case-report : A 4-year-old boy with anamneses of seasonal
allergy which was predominant in the spring and occasional cough without
other comorbidities was referred to emergency department in Shiraz
following sudden cough and dyspnea. The patient underwent bronchoscopy
due to foreign body aspiration suspicious. A little plastic toy was
extracted along through the bronchoscopy. Although Bronchoscopy revealed
that the trachea and bronchi of the left lung was normal, the severe
inflammation was reported in the right bronchus possibly due to foreign
body irritation. In order not to healing the cough and other symptom,
the patient referred several doctors suggested to be treated as
childhood-asthma, but the symptoms did not have a remarkably improved.
After five months of persistent in symptoms, especially cough and
exertional dyspnea, the patient was referred to our institution for more
evaluation. His physical examination was unremarkable without audible
wheezing or stridor. Results of a routine hematological panel and the
chest x-ray upon-admission was interpreted as normal(Figure.1) . The chest CT-scan showed a partial obstruction in
the main right bronchus (Figure.2) . To more evaluation,
pulmonary 3-dimensional constructed CT-scan was conducted which revealed
a narrowing structure at the third-thoracic (T3) vertebrae bone level
without any mass, adenopathy, or vascular sling formation in mediastinum(Figure.3) . The patient underwent bronchoscopy and a
string-like, pallor structure stretching across the main right bronchial
lumen obstructed the mainstream of lumen with just a 2 mm opening was
revealed (Figure.4) . No additional anomalies of the trachea,
carina, vocal cords, or other organs were found. The lesion was
partially removed using knife during the bronchoscopy procedure. Removal
of the lesion by knife, which was easily broken, revealed a fibrous
string without bronchial glands or cartilage diagnosed as a “bronchial
web” (Figure.4) . The patients’ symptoms improved remarkably
following the procedure. The second and third follow up bronchoscopy was
performed for accomplishment of both bronchial web removal and to
confirm that no additional microscopic changes was constructed. Of note,
during the second bronchoscopy the granulation tissue was taken. On
follow=up in the clinic, the patient’s dyspnea had resolved; he had no
limitations in his daily activity or exercise.
Discussion: A bronchial web is formed by a thin layer of
membranous tissue containing small holes that cause the bronchial lumen
to narrow leading to partial airway obstruction. The etiology of the
lesion is unknown. If such webs result in complete obstruction, they
will be fatal unless treated at birth. The literature about which
published this topic revealed that most of bronchial cases were
congenital by nature and similar to our finding, anatomical occurrence
was mostly in the right bronchial site [2-4]. Most of these cases
went initially unrecognized due to the nonspecific symptoms. Similarly,
our patient who initially had an episode of foreign body aspiration, had
been treated as asthma for frequent months due to persistent cough and
dyspnea. We believe that appearance of clinical symptoms just after the
foreign body aspiration was due to inflammation reaction to foreign body
at the bronchial web site, which leaded to exacerbate the partial lumen
narrowing. In other meaning, the worsening of his symptoms, probably was
associated with the progression of the size of the stenosis. Our case
demonstrated the need for bronchoscopy evaluation due to persistent
symptom not ameliorating by previous treatment. Diagnostic bronchoscopy
is essential for diagnosis of congenital bronchial web and also for
acquired cases. Meanwhile, in recent years, tendencies to spiral
pulmonary 3D-CT scan and reconstructed virtual bronchoscopy navigation
as diagnostic option has been increased[5]. By this imaging, the
internal layer of the airway’s lumen and also neighboring structures of
external lumen such as fistula could be inspected[6]. By evaluation
of the primary chest x-ray in our patient, it was not plausible
suspecting bronchial narrowing (Figure.1) ; however, the
findings of bronchial stenosis were remarkably evident in the pulmonary
3D-CT-scan. Moreover, as the internal aspect of bronchial lumen images
were constructed by virtual bronchoscopy using the pulmonary CT-scan
images, the findings suggested a web shaped stenosis in right bronchus.
The virtual bronchoscopy is less effective for the dynamic imaging
inspection or evaluation of mucosal formations by the colorful tonnage
compared to bronchoscopy; meanwhile the virtual bronchoscopy advantage
is that bronchial lumen does not get invaded directly by the
bronchoscope instrument; hence the possible injury to high-risk lesions
is prevented. Therefore, if a fixed and predominant stenotic structure
is doubted at a level where bronchoscopy is hard to utilized, primary
assessment using 3D pulmonary CT-scan and virtual bronchoscopy could be
helpful[5, 7]. Finally, the interventional lesion removal using
knife during two stages of flexible bronchoscopy with one week interval
has not been previously reported for improvement of these lesions. Due
to completely remission of clinical symptoms and no procedure related
side-effects, this technique is found to be a safe and effective way to
treat the airway obstruction following bronchial web.
Conclusion : Herein, we described a mis-diagnosed case of
child with prolonged cough and dyspnea who was ultimately diagnosed as
bronchial web after evaluation via pulmonary 3D-CT scan and treated by
interventional bronchoscopy. Bronchial stenosis due to web structures
should be considered in prolonged asthma-like symptoms, and assessment
based on using 3D pulmonary CT-scan, virtual bronchoscopy, and following
bronchoscopy as ultimate diagnostic and treatment option could be
advantageous.
Conflicts of interest: no conflict of interest is
declared by the authors.
Funding/Support: There is no funding to present essay.
Ethics approval statement: The authors all declare that
this manuscript is not published or under consideration in other
journals.
Patient consent statement: Written informed consent has
been acquired from patient parents to publish this study according to
the journal’s patient consent policy. Moreover, the authors all declare
that patients’ confidentiality has been respected.
Data Availability Statement: The data supporting the
findings of the study such as electronical medical reports and full
video of bronchoscopy procedures are available on request from the
corresponding author.