An uncommon cause of chronic cough: a case of tracheobroncho-

pathia osteochondroplastica

Gabija Asipauskaitė

1

, Greta Miškinaitė

1

, Rusnė Glinskytė-Bimbirė

2

1

Medical Academy, Faculty of Medicine, Lithuanian University of Health Sciences, Kaunas, Lithuania

2

respiratory tract infections. Initial imaging and spirometry were inconclusive, therefore multiple differential

diagnoses were explored. Despite several computed tomography (CT) examinations, characteristic airway

abnormalities were not recognized until retrospectively reviewed. Bronchoscopy revealed firm cartilaginous

nodules throughout the trachea and main bronchi, arising from the cartilaginous rings and sparing the posterior

membranous wall. Biopsy by forceps was technically challenging. Histological examination of the specimen

revealed only mild chronic inflammatory changes without osseous or cartilaginous tissue. The combination of

endoscopic findings, clinical presentation and retrospective imaging review supported the diagnosis of TO. The

patient was managed conservatively, as no significant airway obstruction was present.

Conclusions. TO should be considered in patients with chronic cough, recurrent infections or other unexplained

respiratory symptoms. Improved awareness among clinicians may enable earlier recognition, minimise

1. Introduction

Tracheobronchopathia osteochondroplastica

(TO) is a rare, benign airway disorder involving

the development of firm submucosal

cartilaginous or ossified nodules that protrude

into the tracheobronchial passageway [1,2]. Its

incidence is estimated at only 0.01 to 4.2 per 100

000 people [2]. Although TO aetiology remains

unclear, it is most commonly identified in

middle-aged to elderly patients and often

diagnostic gold standard [3,4]. Characteristic

nodular protrusions are typically observed

during airway endoscopy, where rigid whitish

lesions arise along the cartilaginous airway walls

but do not involve the posterior membranous

segment [5]. Due to its variable and often mild

clinical manifestations, TO may be

misinterpreted as more common conditions,

including asthma, chronic bronchitis or even

bronchogenic malignancy [6]. We present a case

month history of chronic cough, recurrent

respiratory tract infections and frequent

subfebrile fever. Her past medical history

included hypertension, chest angina and

anaemia. She had never smoked, reported no

significant occupational or environmental

exposures, and had no family history of chronic

lung diseases. The patient’s family physician

At the pulmonology follow-up visit, there was

no clinical response to budesonide, and

spirometry parameters remained unchanged.

Serology for Chlamydia pneumoniae and

Mycoplasma pneumoniae were negative.

Sputum culture grew Staphylococcus aureus,

antibacterial therapy was initiated, and the

patient was scheduled for follow-up after several

months. Repeated CT demonstrated new

endobronchial changes while spirometry

Bronchoscopy performed during hospitalization

revealed firm cartilaginous nodules throughout

the trachea and bilateral bronchi, arising from

the cartilaginous rings and sparing the posterior

membranous wall (Figure 1). Although the

nodules were difficult to biopsy, the samples

were obtained. Bronchial secretions were

obtained for microbiological culture, which

grew Candida kefyr. Blood culture yielded

Streptococcus pneumoniae. Histopathological

Figure 2. Computed tomography showing irregular nodules involving the tracheal wall with preservation of the

posterior membranous wall, with calcified nodules in the left bronchus (A) and a small nodular prominence on

the right tracheal wall (B).

The patient was informed of the TO diagnosis

and was started on antibiotic and antifungal

treatment for the ongoing infection. Upon

completion of treatment, she was discharged

with a conservative management plan under

pulmonology follow-up, as no bronchial

obstruction was present and interventional

therapy was not indicated.

3. Discussion

unaffected [1]. First described in 1932, TO is

extremely rare – under 600 cases have been

reported worldwide to date, with roughly 140

cases coming from Japan and about 80 from

China [3,7]. Its detection varies considerably, as

studies based on autopsies suggest a prevalence

of 0.25-0.30%, whereas bronchoscopic

examinations have identified it in 0.01-0.80% of

cases [1,8]. Epidemiological data regarding sex

distribution are inconsistent: some studies note a

70 years of age, although a few cases have also

been identified in children [3,7].

The aetiology of TO remains unclear, but nodule

formation is thought to result from calcium

phosphate deposition, which induces aberrant

proliferation of bone and cartilage and can lead

to narrowing of the major airways [2]. Chronic

airway inflammation has also been proposed as

The diagnosis of TO remains challenging due to

its nonspecific and often mild clinical

presentation. Patients commonly undergo

repeated evaluations for chronic cough,

recurrent infections, or unexplained dyspnoea

before characteristic findings are recognized.

Published literature shows that the condition is

often confused with more common respiratory

diseases, including asthma, tuberculosis, and, on

rare occasions, bronchogenic cancer [3,6]. In our

ray typically does not reveal any abnormalities,

computed tomography (CT) scanning is

considered the most accurate imaging technique

for evaluating TO [4,10,14]. On CT, TO

typically presents as thickening of the tracheal

cartilaginous wall with irregular sessile nodules

while consistently sparing the posterior

membranous trachea. The nodules may be

calcified or non-calcified and can occur either

focally or diffusely along the trachea and even

interpreted as normal, but TO-related changes

were retrospectively recognized when the

images were reviewed again after the diagnosis

had been confirmed by bronchoscopy or autopsy

[3,4]. CT is also highly valuable for identifying

complications such as bronchiectasis,

atelectasis, or post-obstructive pneumonia, and it

additionally contributes to the differential

or demonstrate inflammatory changes, epithelial

hyperplasia or squamous metaplasia [3,5].

Although some biopsies reveal only intact

respiratory epithelium, the most characteristic

feature described is squamous metaplasia

associated with calcified cartilage or new bone

formation. In some cases, mature

osteomedullary tissue has also been identified

[3]. Nevertheless, obtaining adequate tissue

samples may be challenging because the nodules

Currently there are no standardized guidelines

for the management of TO, and treatment

remains generally symptomatic. Most patients

receive conservative therapy aimed at relieving

cough, managing infections, and improving

breathing capacity [2–4]. This may include

antibiotics, bronchodilators, oral anti-

inflammatory agents, humidified air, oxygen or

steam inhalation, mucolytics, chest

physiotherapy, avoidance of airway irritants

excision, can be considered for patients with

more advanced disease or significant airway

obstruction, but reported outcomes have been

variable [2,4]. Overall, current management

strategies focus primarily on symptom control

and infection prevention. Further large-scale

studies are required to establish standardized

management strategies.

2. Rana A, Mezughi H, Malik SA, Mansoor K,

Al-Astal A. Rare Manifestation of Idiopathic

Tracheobronchopathia Osteochondroplastica:

Misdiagnosed and Untreated Entity? Cureus.

2020;12(7):e9407. doi:10.7759/cureus.9407.

3. Devaraja K, Surendra VU.

Clinicopathological Features and Management

Principles of Tracheobronchopathia

Osteochondroplastica - A Scoping Review.

Indian J Otolaryngol Head Neck Surg.

doi:10.21037/apm-20-316.

5. Ribeiro GMRe, Natal MRC, Silva EF, Freitas

SC, Moraes WC, Maciel FC.

Tracheobronchopathia osteochondroplastica:

computed tomography, bronchoscopy and

histopathological findings. Radiol Bras.

2016;49:56–57. doi:10.1590/0100-

3984.2014.0056.

6. Devaraja K, Sagar P, Chirom AS.

Tracheobronchopathia osteochondroplastica:

Five Cases Report and Literature Review. Ear

Nose Throat J. 2020;99(10):NP111–NP118.

doi:10.1177/0145561319897982.

8. Prakash UBS. Tracheobronchopathia

osteochondroplastica. Semin Respir Crit Care

Med. 2002;23(2):167–175. doi:10.1055/s-2002-

25305.

clinical diagnosis. The Clinical Respiratory

Journal. 2017;11(4):440–447.

doi:10.1111/crj.12356.

16. Danckers M, Raad RA, Zamuco R, Pollack

A, Rickert S, Caplan-Shaw C. A Complication

of Tracheobronchopathia Osteochondroplastica

Presenting as Acute Hypercapnic Respiratory

Failure. Am J Case Rep. 2015;16:45–49.

doi:10.12659/AJCR.892427.