'Zebra' in the Airway

Abstract

INTRODUCTION: Immigration and cultural exchanges bring global diversity to diseases we see in our practice. We present here the story of a young Burmese man with chronic cough causing multiple physicians and emergency room (ER) visits over 1.5 years, without a diagnosis or improvement. CASE PRESENTATION: A 23 y old Burmese non-smoking man was referred with chronic cough for -1.5 yrs. History included treated tuberculosis 5 yrs. ago. Expectoration had occasional blood streaks, not associated with fever, chills, chest pain, weight loss or appetite loss. In last 18 months, he visited many physicians and ERs, including a nearby university hospital for this cough. Repeatedly, he had normal chest x-rays, and was discharged with cough suppressants and antibiotics, without relief. He was eventually referred to a pulmonologist. A CT chest done after chest x-ray was again unrevealing showed a calcified granuloma in right lower lobe, and a calcified endobronchial lesion in bronchus intermedius. Flexible bronchoscopy revealed near-complete obliteration of bronchus intermedius by a zebra-skin patterned lesion surrounded by well-formed bronchial tissue. Direct pressure by tip of bronchoscope moved it distally, partly freeing it from the surrounding tissue. After failing with forceps, snare and fogarthy catheter, we extracted a 1.5 cm sharp stone-like object using the basket catheter. A closer inspection identified the object as betel-nut (Areca catechu), a mouth-freshener frequently chewed in South Asia. Patient's wife later confirmed his betel-nut addiction. Cough finally resolved after bronchoscopy. Biopsy of surrounding tissue revealed non-malignant squamous metaplasia with fibrin, and inflamed granulation tissue. DISCUSSION: Areca catechu is a WHO group-1 carcinogen, known to cause squamous cell carcinoma. Worldwide, it is the fourth most common addictive substance, and is culturally prevalent in South Asia. Areca extract (containing Arecoline) causes airway remodeling with smooth muscle hyperplasia and fibrinous subepithelial deposits, and worsens asthma. Having developed squamous metaplasia, this patient may soon have progressed to squamous carcinoma at the endobronchial site of contact. Foreign body aspiration is more common in children and elderly, unlike our young patient. A prospective study by Magsi et al. in rural Pakistan reported betel nut as the most common airway foreign body removed. This habit is unheard of in USA, and hence, was never on our differential. This case highlights a cultural aspect of medicine uncommon in our practice. CONCLUSIONS: 1. Cultural habits should be paid attention to in history-taking of immigrant population. 2. Chest x-ray is often unrevealing for foreign body aspirations.