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Acquired segmental emphysema: the enlarging spectrum of Swyer-James/Macleod's syndrome
S K Ohri1, G Rutty, S W Fountain
1Thoracic Surgical Unit, Harefield Hospital, United Kingdom.
Abstract:
Three cases of Macleod's syndrome are described, all of which required surgical resections for distressing symptoms. This provided a rare opportunity to examine the pathologic features of a condition that is usually diagnosed on its radiologic features, and the etiology of which remains unestablished. Three patients (2 women and 1 man), aged 20, 23, and 24 years, were referred from respiratory physicians because of unilateral hyperlucent lungs and associated symptoms. All 3 patients had unilateral hyperlucent lungs, but only 1 patient had demonstrable mediastinal shift on expiratory and inspiratory chest computed tomographic scan. Segmentectomies were performed (n = 4) in all the patients without perioperative morbidity or mortality. Patients have been followed up between 6 and 18 months, and remain asymptomatic with a return to normal lifestyle. Histologic examination of the specimens found inflammation of the bronchus in all 3 patients, but only two specimens had evidence of bronchiolar inflammation. In only 1 patient was there a reduction in bronchiole number. All 3 patients showed presence of emphysema. These cases are notable for the segmental distribution of the disease. Pathologic examination lends support to the theory that previous respiratory tract infection may play a role in the pathogenesis of this condition.
Insights
Macleod's syndrome, characterized by unilateral hyperlucent lungs, was surgically treated in three young adults. Pathologic findings suggest respiratory tract infections may contribute to this rare condition.
Area of Science:
- Pulmonology
- Pathology
- Radiology
Background:
- Macleod's syndrome is a rare condition typically diagnosed via imaging.
- Its etiology remains largely unestablished.
- Surgical intervention is sometimes necessary for symptomatic relief.
Purpose of the Study:
- To describe the pathological features of Macleod's syndrome in three surgically treated cases.
- To explore potential etiological factors based on histological findings.
- To report outcomes following surgical resection.
Main Methods:
- Case series of three patients (2 female, 1 male; aged 20-24) with unilateral hyperlucent lungs.
- Surgical resection (segmentectomies) performed on all patients.
- Histopathological examination of resected lung tissue.
Main Results:
- All patients presented with unilateral hyperlucent lungs; one had mediastinal shift.
- Histology revealed bronchial inflammation in all cases, bronchiolar inflammation in two, and reduced bronchiole count in one.
- Emphysema was present in all three patients, with segmental distribution.
- Surgical outcomes were excellent, with no perioperative complications and sustained asymptomatic status post-follow-up (6-18 months).
Conclusions:
- Pathological findings support the hypothesis that prior respiratory tract infection may play a role in Macleod's syndrome pathogenesis.
- Surgical resection is a viable option for symptomatic Macleod's syndrome, offering good long-term outcomes.
- The segmental distribution observed highlights a localized disease process.