Swyer-James-MacLeod Syndrome (SJMS) occurs as a result of childhood bronchiolitis obliterans. Typically, this disorder is diagnosed in childhood after evaluations for recurrent respiratory infections. One of the reasons to explain the difficulty in diagnosis is that when patients develop little bronchiectasis, and therefore, few symptoms, then this syndrome may not be recognized until adulthood. Here, we are presenting a 22-year-old female patient who was diagnosed with SJMS who was initially misdiagnosed with a pneumothorax and treated by multiple chest tube drainages. This case highlights the significance of taking a careful history, the application of computed tomography and scintigraphy in confirming the diagnosis of SJMS and in eliminating other diseases.