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Swyer-James-Macleod syndrome as a rare cause of unilateral hyperlucent lung: Three case reports
Daniela Machado1, Filipa Lima, Catarina Marques
1Pulmonology Department, Gaia/Espinho Hospital Center, Vila Nova de Gaia, Portugal.
Rationale:
Swyer-James-Macleod syndrome (SJMS) is a rare lung disorder characterized by unilateral hyperlucent lungs which arises as a complication of bronchiolitis obliterans. It is typically diagnosed during childhood, although some patients may only be diagnosed in adulthood, often as an incidental finding, but others due to recurrent chest infections.
Patient Concerns:
Three patients were referred to our institution with complaints of dyspnea on exertion, chronic productive cough, and recurrent pulmonary infections. Two of them had a history of lower respiratory tract infections during childhood.
Diagnosis:
A computed tomography scan was performed and showed unilateral reduced density of the lung and bronchiectasis in the 3 patients. Based on the clinical presentation and radiologic features, the diagnosis of SJMS was established.
Interventions:
Patients started inhaled corticosteroids and long acting beta agonist, vaccines to prevent respiratory infections were administered and airway clearance techniques were taught.
Outcomes:
Two patients became asymptomatic with inhaled therapy and no recurrent chest infections were observed over a 3-year follow-up period, being discharged from our institution to the general practitioner. The 3rd patient had some improvement in the frequency of pulmonary infections and dyspnea, without improvement in respiratory function tests, maintaining vigilance at our center.
Lessons:
These cases highlight the importance of being aware of this condition and its frequent association with bronchiectasis, which may adversely affect the prognosis, to manage patients appropriately and prevent recurrent pulmonary infections.
Insights
Swyer-James-Macleod syndrome (SJMS), a rare lung condition, can cause unilateral hyperlucent lungs. Early diagnosis and management, including inhaled therapies and airway clearance, can significantly improve patient outcomes and prevent recurrent infections.
Area of Science:
- Pulmonology
- Radiology
- Pediatric Respiratory Medicine
Background:
- Swyer-James-Macleod syndrome (SJMS) is a rare sequela of bronchiolitis obliterans, presenting as unilateral hyperlucent lungs.
- Diagnosis can occur in childhood or adulthood, often prompted by recurrent respiratory infections or incidental findings.
Observation:
- Three patients presented with dyspnea, chronic cough, and recurrent pulmonary infections, with two having a history of childhood lower respiratory tract infections.
- Computed tomography revealed unilateral lung density reduction and bronchiectasis, confirming SJMS diagnosis.
Findings:
- Inhaled corticosteroids and long-acting beta-agonists, alongside preventative measures like vaccines and airway clearance techniques, were initiated.
- Two patients achieved asymptomatic status with reduced infection frequency over a 3-year follow-up, while the third showed partial improvement.
Implications:
- SJMS management requires awareness of its association with bronchiectasis, which impacts prognosis.
- Appropriate interventions can prevent recurrent pulmonary infections and improve quality of life in SJMS patients.
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