Sterile Bronchopleural Fistula Following Surgical Removal of Primary Lung Leiomyoma Inducing Secondary Hypertrophic Osteoarthropathy

Abstract

BACKGROUND Leiomyomas are benign neoplasms of the smooth muscle. When found in the pulmonary system, a rare occurrence, leiomyomas can result in hypertrophic osteoarthropathy, or significant clubbing, associated with proliferation of long bone periosteum. Bronchopulmonary fistulas, or communications between the bronchial tree and pleural space, are an uncommon postoperative complication of pneumonectomies. Even more infrequent is the presence of a bronchopulmonary fistula that is determined to be sterile. CASE REPORT The patient presented in the current case report is a 40-year-old previously healthy woman who presented with a 5-year history of chronic cough, right-sided chest discomfort, and dyspnea associated with back pain, and lower leg pain. The CT scan performed on the patient revealed a mass originating from the right lower lobe. Activity at the site of the lesion, in the long bones of the upper and lower limbs, rib cage, and vertebral bones was demonstrated by a bone scan. A CT-guided biopsy was performed, and the pathology report confirmed the presence of a leiomyoma. Following a right-sided lobectomy, the resected tumor was sent for histopathology, with the results confirming the biopsy. The patient subsequently presented with a history of persistent cough associated with increased watery secretions. The CT scan revealed the presence of a bronchopleural fistula, after which the patient underwent surgical correction. All symptoms resolved, and the patient was discharged in stable condition. CONCLUSIONS Here, we report on a patient who presented with 3 rare clinical findings: pulmonary leiomyoma, hypertrophic osteoarthropathy, and sterile bronchopulmonary fistula.

Figure 1.

(A) Chest X-ray of lung mass showing leiomyoma in right lower lung field. (B) CT scan shows a mass originating from the right lower lobe extending to the mediastinum, without lymphadenopathy. (C) PET scan showed mild activity in the lung.

Figure 2.

(A) CT chest showing the site of the bronchopleural fistula. (B) Pre-operative bone scan showing diffuse radiotracer uptake involving the sternum, upper and mid thoracic spine, as well as both femurs and tibiae, corresponding to sclerotic changes seen on CT images. A postoperative bone scan showed interval improvement of previously noted diffuse uptake in both femora, tibia, and sternum.