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CASE SNIPPET
Year : 2023  |  Volume : 69  |  Issue : 1  |  Page : 56-58

Tracheobronchial adenoid cystic carcinoma mimicking bronchial asthma


Division of Respiratory Medicine, Department of Internal Medicine, University of Tsukuba Hospital, Ibaraki, Japan

Date of Submission25-Feb-2022
Date of Decision13-Apr-2022
Date of Acceptance26-May-2022
Date of Web Publication26-Dec-2022

Correspondence Address:
Prof. H Satoh

Japan
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jpgm.jpgm_201_22

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How to cite this article:
Ishikawa H, Satoh H, Hizawa N. Tracheobronchial adenoid cystic carcinoma mimicking bronchial asthma. J Postgrad Med 2023;69:56-8

How to cite this URL:
Ishikawa H, Satoh H, Hizawa N. Tracheobronchial adenoid cystic carcinoma mimicking bronchial asthma. J Postgrad Med [serial online] 2023 [cited 2023 Feb 1];69:56-8. Available from: https://www.jpgmonline.com/text.asp?2023/69/1/56/365563




Tracheobronchial adenoid cystic carcinomas (TACCs) arise from the submucosal glands and have indolent courses.[1],[2],[3] As a result of the submucosal location of the tumors, the clinical manifestations of TACCs are late and unspecific.[1],[2],[3] With wheezing, some of TACC patients may be misdiagnosed as having bronchial asthma (BA).[3],[4],[5],[6] Inspiratory stridor in tracheobronchial stenosis is different from expiratory wheezing due to prolonged exhalation in BA, but it is not always easy to make an accurate diagnosis. Some TACCs can grow mainly in the central airspace, even though they infiltrate the tracheobronchial wall without destroying surrounding tissue or forming a mass. Therefore, plain chest radiographs might not give significant findings even in patients with advanced TACC. These patients may be misdiagnosed as BA and, as a result, might not be able to receive appropriate treatment.[3],[4],[5],[6] We report herein two patients with TACC who had been treated as BA for years prior to the correct diagnosis.

Patient 1

A 40-year-old woman was referred to our hospital with a tracheobronchial mass on chest computed tomography (CT) scan. She had a 2-year history of wheezes and shortness of breath. She had been diagnosed as having BA and had received conventional BA therapy. Her symptoms seemed to improve slightly after use of bronchodilators but gradually deteriorated. The imaging tests were performed again. Although her plain chest radiograph was not remarkable [Figure 1]a, a chest CT scan showed a tracheobronchial mass, which almost occluded the lower part of the trachea and both main bronchi [Figure 1]b and [Figure 1]c. Bronchoscopy revealed a tracheobronchial tumor extending from lower part of the trachea to the both main bronchi, with a rich vascularization [Figure 1]d. The tumor was almost obstructing the airways. A biopsy of the tumor was obtained. The histological examination of the biopsy tissue confirmed the diagnosis of TACC [Figure 1]e. Soon after the pathological diagnosis, cisplatin-based concurrent chemoradiotherapy was initiated. Partial response was achieved and patient's symptoms improved. She has been alive without recurrence for 31 months since the final course of chemotherapy.
Figure 1: (a) normal chest radiograph; (b, c) chest CT scan (lung window, mediastional window) showing a tracheobronchial mass (arrows); (d) bronchoscopy showing a tracheobronchial tumor at lower part of the trachea (arrow); (e) Microscopic finding of the tumor (Hematoxylin and eosin staining, x200). White arrows depict cribriform island containing biphasic tissue architecture consisting of atypical glandular epithelium and epithelial/basal cells

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Patient 2

A 39-year-old woman was referred to our hospital with a tracheobronchial mass on chest CT scan. The patient had wheezes, repeated cough, and shortness of breath for 3 years. She had been diagnosed as having BA and bronchodilators had initially improved her symptoms slightly. Subsequently, her symptoms gradually worsened and her chest imaging studies were repeated. Chest radiography was unremarkable [Figure 2]a. But on a chest CT scan, tracheobronchial tumor obstructing lower part of the trachea was observed [Figure 2]b and [Figure 2]c.
Figure 2: At time of admission - (a) normal chest radiograph; (b, c) chest CT scan (lung window, mediastional window) showing a tumor obstructing lower part of the trachea (arrows); (d) bronchoscopy showing a tumor at lower part of the trachea (arrows); (e) Microscopic finding of the tumor (Hematoxylin and eosin staining, x200). White arrows depict cribriform island containing biphasic tissue architecture consisting of atypical glandular epithelium and epithelial/basal cells

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Idiopathic pneumomediastinum was also found, but disappeared after about 2 weeks of rest, and did not affect the subsequent treatment of TACC. A tumor that originated from the membranous part of the lower trachea and almost occluded the airway was revealed on bronchoscopy [Figure 2]d. A biopsy of the tumor was obtained. The histological examination of the biopsy tissue confirmed the diagnosis of TACC [Figure 2]c.

Cisplatin-based concurrent chemoradiotherapy was initiated. Partial response was achieved and symptoms subsided. She remains healthy without evidence of disease after 30 months of follow-up.

Patients with TACC often complain of nonspecific respiratory symptoms such as wheezing and shortness of breath. This may be related to airway narrowing of the tracheobronchi,[3],[4],[5],[6] whereas BA is one of the most common diseases associated with wheezing, but no patient should be labeled as “asthmatic” without appropriate evaluation. If a patient with TACC is misdiagnosed as having BA, there will be a significant delay in starting the correct treatment and even loss of life. Some previous cases reports[3],[4],[5],[6] and our 'cases indicate the fact that not all patients with wheezes are BA patients. Airway obstruction can cause asthma-like symptoms. It should be recognized that the correct diagnosis needs to be established. Before diagnosing BA, it is important to check for airway lesions. Furthermore, a diagnosis of airway lesions should always be considered in patients with BA who achieve poor control of their symptoms in spite of proper treatment and compliance. Chest CT and bronchoscopy will provide useful diagnostic information. We emphasize the following two points. First, as observed in these two patients, even if the airways are nearly obstructed, some patients with TACC may still have the symptoms observed in BA patients. Second, if appropriate treatment of BA is inadequate, chest CT scan rather than chest radiograph should be performed to assess the presence of airway lesions.

Declaration of patient consent

The authors certify that appropriate patient consent was obtained.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
 :: References Top

1.
Hsu AA, Tan EH, Takano AM. Lower respiratory tract adenoid cystic carcinoma: Its management in the past decades. Clin Oncol (R Coll Radiol) 2015;27:732-40.  Back to cited text no. 1
    
2.
Djaković Ž, Janevski Z, Cesarec V, Slobodnjak Z, Stančić-Rokotov D. Adenoid cystic carcinoma of distal trachea: A case report. Acta Clin Croat 2019;58:777-9.  Back to cited text no. 2
    
3.
Gupta D, Singh I, Sakthivel P. Adenoid cystic carcinoma of trachea: A diagnostic and therapeutic challenge. Indian J Otolaryngol Head Neck Surg 2016;68:94-6.  Back to cited text no. 3
    
4.
Amr SS, Shihabi NK. Adenoid cystic carcinoma of the trachea mimicking asthma. Saudi Med J 1999;20:640-1.  Back to cited text no. 4
    
5.
Koul PA, Khan UH, Shah TH, Dar AM. All that wheezes is not asthma. BMJ Case Rep 2014;2014:bcr2013202369.  Back to cited text no. 5
    
6.
Kokturk N, Demircan S, Kurul C, Turktas H. Tracheal adenoid cystic carcinoma masquerading asthma: A case report. BMC Pulm Med 2004;4:10.  Back to cited text no. 6
    


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