Rare presentation of a rare disease: Bilateral congenital lobar overinflation

Abstract

Congenital lobar overinflation is a rare but well-recognized congenital cause of neonatal and infantile respiratory distress. At times, the condition can mimic other congenital or acquired diseases and have atypical distribution and imaging patterns. Lobectomy of the involved lobe(s) is curative. We present our experience with 3 surgically confirmed cases of congenital lobar overinflation. Referral papers, patient's charts, including operation notes, and radiographic records were reviewed. All of them were initially misdiagnosed or underdiagnosed based on the initial radiographic examination alone. All 3 were referred to our center with respiratory distress, and the first 2 were treated with antibiotics prior to the settlement of their diagnosis. Chest computed tomography was key in diagnosing all 3 cases. The first patient was a 10-day-old neonate diagnosed with bilateral congenital lobar overinflation. The second patient was a 2-month-old infant diagnosed with right middle lobe disease. In these 2 cases, the initial assessment of the vascularity was atypically excessive in the affected lobe(s). Eventually, correlation with typical concurrent imaging features and the clinical condition of the patients led to the correct diagnosis. The third case was a 4-month-old infant with left upper lobe congenital lobar overinflation. All cases underwent successful surgical treatment. Congenital lobar overinflation is a rare anomaly, and multiple-lobe involvement is even rarer. Vascularity within the affected lobes is a subjective assessment that can be overestimated, leading to confusion, and a feature that needs correlation with other common imaging features and the clinical course of patients.

Keywords

• Congenital lobar overinflation
• Pneumothorax
• Pediatric chest
• Chest CT