Vomiting-induced bilateral pneumothorax in a patient with MEN1-associated Zollinger-Ellison Syndrome

Abstract

Abstract Background Multiple endocrine neoplasia type 1 (MEN1) is an autosomal dominant tumor syndrome characterized by involvement of the parathyroid glands, pancreas, and pituitary gland. Pulmonary manifestations of MEN1 are uncommon. We report a patient with MEN1 who developed bilateral pneumothorax following severe vomiting associated with Zollinger-Ellison syndrome. Case presentation A 45-year-old Iranian woman with a history of pituitary macroadenoma presented with refractory vomiting and gastrointestinal bleeding associated with Zollinger-Ellison syndrome. Further evaluation demonstrated multiglandular primary hyperparathyroidism, pancreatic neuroendocrine tumor, left adrenal adenoma, and bilateral carotid body paragangliomas, fulfilling the clinical diagnostic criteria for MEN1. During hospitalization, she developed acute dyspnea, and thoracic imaging revealed bilateral pneumothorax requiring chest tube placement. Chest CT excluded esophageal perforation, and bronchoscopy, video-assisted thoracoscopic surgery (VATS), and pleural fluid analysis showed no evidence of bronchial carcinoid, thymic tumor, or primary lung malignancy. The patient subsequently underwent subtotal parathyroidectomy, carotid body tumor excision, distal pancreatectomy, splenectomy, and left adrenalectomy, with a favorable postoperative outcome. Conclusions This case highlights bilateral pneumothorax as a rare secondary mechanical complication of forceful vomiting associated with Zollinger–Ellison syndrome in a patient with MEN1. Forceful vomiting may have caused alveolar rupture through the Macklin effect. Recognition of this uncommon secondary complication may facilitate timely diagnosis and management of patients with MEN1 presenting with severe vomiting.