Case Presentation : Tracheal obstruction by calcified TB gland in a child Aneesa Vanker, Pierre Goussard, Sharon Kling, JT Janson, B Barnard, M Connellan. Tygerberg Children`s Hospital.Department of Paediatrics and Cardiothoracic surgery.University of Stellenbosch

Background 9 month old baby GA referred from Worcester Hospital Problems: # Recurrent “stridor” for 3 months # Pulmonary tuberculosis on treatment for 5 months

TB diagnosis & Mx TB diagnosed at 4 months of age based on - +ve tuberculin skin test - CXR with suggestive features of TB - No gastric washings done  Commenced on TB Rx and was already on continuation phase Rx  Apparently “many” TB contacts

Stridor background History of repeated admission to Caledon Hospital from 6 months of age with stridor. Treated with nebulisations and sent home. Eventually at 9 months of age, referred to Worcester Hospital for Ix of stridor. Noted on CXR to have ?mass in right main bronchus area

TCH course Clinically: Well grown child on 10 th centile for weight. Resp exam: Monophonic wheeze Minimal stridor No differential air entry Other systems normal

Investigations CXR – calcified lesion in area of right main bronchus – most likely lymph node ENT consult – Not able to detect any abnormality The next step - Bronchoscopy

CXR showing calcified node

Bronchoscopy Large gland herniating into the trachea with >90% occlusion Right main bronchus occluded by herniating gland Areas of gland removed piecemeal at bronchoscopy (endoscopic enucleation)– still significant occlusion BAL done and cultures sent

Large gland herniating into the trachea with >90% occlusion

Further developments Intubated post bronchoscopy to protect the airway Transferred to PICU Urgent chest CT scan done  confirmed large gland of tuberculous nature herniating into trachea and right main bronchus

Calcified gland eroding into trachea

Surgery Enucleation done Large amount of caseous material removed Small tracheal defect closed

Post-surgery Repeated bronchoscopy 1 hour after returning to PICU Trachea now only 50% occluded (prev >90%), RMB still occluded Changed to MDR TB Rx (INH, Rif, Oflox, Amik, Etham) + Steroids Reason – no response to previous Rx, possible MDR TB Extubated

Before surgery Post surgery

Further course ZN stain positive for AFB on enucleated gland Culture pending Clinically wheeze improved Repeat bronchoscopy 1 week post- enucleation  Trachea patent, no gland herniation, RMB only 50% occluded by herniating gland

Transferred back to Worcester to continue TB treatment For follow-up in 1 months time

Airway involvement in TB Trachea and 2 main bronchi most affected. Upper airway involvement rare in children

Presentation Enlarged glands can cause external compression of the airways. May herniate into airways. Varying degrees of obstruction rarely complete obstruction. Partial obstruction  “ball-valve” effect – air enters lung but trapped on expiration Complete obstruction  lung or lobar collapse

Assessment CXR – 4 patterns of compression 1) airway narrowing 2) ball-valve effect 3) expansile pneumonia 4) lobar collapse  Bronchoscopy – degree of obstruction, BAL, endoscopic enucleation  CT scan – Confirmation, assist in planning further interventions

Management TB treatment – standard 3 drug regime Corticosteroid – prednisone 2mg/kg for 1 month then weaned Evaluated for enucleation – life- threatening obstruction, poor response to Rx and steroids.

Learning points Differentiating stridor from monophonic wheeze Stridor - Harsh, high-pitched inspiratory sound usually audible without a stethoscope - Extrathoracic obstruction  Monophonic wheeze – Intrathoracic obstruction

Recurrent presentations of stridor/wheezing warrant further investigation Although on CXR – gland look calcified, still needed further management TB cultures are always important especially when the diagnosis is made

Summary Unusual presentation of endobronchial TB. Potential for life-threatening complications. Thus far the outcome has been favourable.

Review of the literature Endobronchial TB with gland herniation has been described: Airway involvement in pulmonary tuberculosis. Goussard P, Gie R Paediatr Respir Rev Jun;8(2): However, very little described on gland herniation into the trachea 2 articles : Tuberculous cavitating node communicating with the trachea. Case report with radiographic and pathologic review. Palacios EJ, Tirman RM, White HJ. J Ark Med Soc May;68(12): Airway obstruction secondary to tuberculosis lymph node erosion into the trachea: drainage via bronchoscopy. Schwartz MS, Kahlstrom EJ, Hawkins DB. Otolaryngol Head Neck Surg Dec;99(6):604-6.