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A 49-year old female with history of chronic pancreatitis, 14 cm peri-pancreatic pseudocyst with cystogastrostomy stent placed 1 year earlier, and splenic vein thrombosis presented with massive hematemesis and pre-syncope requiring aggressive volume resuscitation, high dose IV pantoprazole and octreotide drip.CASE PRESENTATION: Initial CT angiography (CTA) of the abdomen revealed a 4 mm splenic artery pseudoaneurysm without evidence of rupture or extravasation, and remonstrated a previously identified splenic vein thrombosis with reduction in size of existing pancreatic pseudocyst. Upper endoscopy showed a large blood clot in the gastric body, suspected to be caused by irritation due to the cystogastrostomy stent. Her hematemesis promptly resolved but she developed frequent melena. The next day, she became severely hypotensive and repeat CTA of the abdomen showed the splenic artery pseudoaneurysm abutting the pseudocyst had enlarged from 6x4 mm to 8x6 mm. There was no evidence of active bleeding but there was a large blood clot in the stomach. She continued to be hemodynamically unstable, and developed hematochezia 4 days later. Repeat CTA showed no active extravasation of contrast and stable splenic artery pseudoaneurysm. Repeat upper endoscopy did not show active bleeding and the clot in her stomach had cleared. She developed recurrent hematemesis which prompted intubation for airway protection. A repeat CTA of the abdomen showed gastric blood, and lack of opacification of the splenic pseudoaneurysm with no active extravasation of contrast later confirmed by splenic artery angiogram. A subtle defect was noted within the expected location of the pseudoaneurysm with spasm within the mid/distal splenic artery. Coil embolization of the proximal splenic artery across the region of the pseudoaneurysm was performed. Post embolization angiogram showed no antegrade flow within the splenic artery. She continued to have melena, cystogastrostomy stent was removed, and upper endoscopy revealed only old blood. At 1 month follow-up, her melena and hematochezia had resolved, CT Abdomen did not show the splenic artery pseudoaneurysm. DISCUSSION: Our case outlines one of the less frequent causes of gastrointestinal bleeding and highlights the necessity of a multi-disciplinary team in critical care. Careful management of our persistently hemodynamically unstable patient and close coordination with the gastroenterology and interventional radiology teams led to the patients survival.CONCLUSIONS: Splenic artery pseudoaneurysms, while rare, are typically caused by pancreatitis, trauma, or peptic ulcer disease and almost always necessitate ICU stay when bleeding occurs. The multitude of precipitating factors in this case with focus on the cystogastrostomy stent outlines the necessity of considering splenic artery pseudoaneurysm as a potential cause of GI bleeding while addressing stent-related complications.

INTRODUCTION:Often identified in the first 3 years of life, congenital atresia of the pulmonary vein is a rare anomaly accompanied by congenital heart disease in approximately 30% of cases1. It is associated with significant morbidity and mortality due to recurrent lower respiratory tract infection and persistent dyspnea. Treatment for such anomalies is not clear. CASE PRESENTATION:A healthy 35-year-old male presented with complaints of left sided sharp sub-sternal chest pain radiating to his left arm and neck. He denied exertional dyspnea, hemoptysis, history of recurrent pulmonary infections, epistaxis, or telangiectasias. Computed tomography angiography (CTA) of his chest revealed a large left lower lobe pulmonary arteriovenous malformation (AVM) and a prominent right coronary artery with a left main coronary artery that is not clearly discerned. A CTA coronary revealed the entire coronary system arises from the right coronary cusp and congenital atresia of the left superior pulmonary vein with dilated collateral vessels emptying into the left inferior pulmonary vein. Pulmonary angiography revealed dilated venous structure in the left lung consistent with congenital atresia of the left superior pulmonary vein with collateralized flow of the left upper lobe to the left lower lobe. This abnormal structure ultimately drained into the left atrium via the left inferior pulmonary vein. No embolization or other treatment modalities were performed by interventional radiology. Cardiac coronary angiography revealed both the left and right coronary system arose from the right coronary cusp. The patient's chest pain was self-limited and was ultimately felt to be unrelated to his congenital venous malformation.DISCUSSION: Congenital pulmonary vein atresia is the result of failure of incorporation of the pulmonary vein into the left atrium1 typically identified at birth. Our case is unique both in the lateness of presentation and the patients lack of exertional dyspnea, hemoptysis, or recurrent pulmonary infections. Treatment approaches include pneumonectomy, corrective surgery, or conservative management. Given our patients relative asymptomatic presentation we pursued a more conservative route. Hereditary hemorrhagic telangiectasias is an important differential diagnosis but was ultimately inconsistent with our patients isolated radiographic findings and clinical history. CONCLUSIONS: Our patient's atypical and delayed presentation shows one of the many anatomical variants accompanying congenital pulmonary vein atresia. His collateralized flow from the left upper lobe likely contributed to his lack of classically associated symptoms.

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