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• W3205116280 abstract "TOPIC: Lung Pathology TYPE: Medical Student/Resident Case Reports INTRODUCTION: IPH is a rare disease that causes recurrent episodes of diffuse alveolar hemorrhage, most commonly presenting in children[1]. The classic presenting triad includes iron deficiency anemia, hemoptysis, and diffuse pulmonary infiltrates; however, the initial symptom is often only anemia[1-2]. This case describes an unusual complication of IPH. CASE PRESENTATION: A 20-year-old African American female with history of IPH presented with acute onset left-sided chest pain and mild shortness of breath. She had no other associated symptoms. She was diagnosed with IPH at 15-months-old. She had previously been on azathioprine and hydroxychloroquine for many years, however stopped taking them about a year prior to presentation. She had frequent episodes of cough, hemoptysis, and fevers when she was young, however over the past 10 years she has had very minimal respiratory symptoms. She is currently taking no medications. Physical exam is significant for absent lung sounds on the left.Chest X-ray showed peripherally predominant fibrosis with apical subpleural cysts, a large pneumothorax with 3.4 centimeters of pleural separation. Atelectasis in the left infrahilar region was noted without evidence of mediastinal shift.She was placed on oxygen and a chest tube was inserted. Repeat chest X-ray showed the pneumothorax significantly decreased in size. High resolution CT chest showed peripheral fibrosis and extensive upper lung predominant subpleural cysts, bilateral perihilar and peribronchovascular coarse linear opacities, and bibasilar dependent opacities. Due to the extensive subpleural cysts, VATS pleurodesis was offered due to the high risk of pneumothorax recurrence. The patient ultimately declined the procedure. The chest tube was removed and repeat chest X-ray showed resolution of the pneumothorax. The patient followed up in pulmonology clinic and was restarted on azathioprine. DISCUSSION: This case presents a unique complication of a rare disease. With recurrent intrapulmonary hemorrhage, pulmonary fibrosis can progress with the development of extensive subpleural cysts, increasing the risk of recurrent pneumonthoraces. Previous studies have found that the most common complications of IPH include severe chronic anemia, progressive lung fibrosis[1], and recurrent hemorrhagic flares[2]. To our knowledge, there are only two prior documented patients that have presented with spontaneous pneumothorax in the setting of underlying IPH[2,3]. Despite years of apparent remission, our patient continued to have progression of fibrosis without recurrent hemorrhage or anemia, until presenting over 10 years later with a spontaneous pneumothorax. CONCLUSIONS: While this case is a unique presentation, it demonstrates that IPH may appear clinically silent for years, while pulmonary disease may continue to progress causing future and sometimes rare complications. REFERENCE #1: LaFreniere K, Gupta V. Idiopathic Pulmonary Hemosiderosis. 2021 Mar 3. In: StatPearls [Internet]. Treasure Island (FL): StatPearls Publishing; 2021 Jan–. PMID: 32644388. REFERENCE #2: Gencer M, Ceylan E, Bitiren M, Koc A. Two sisters with idiopathic pulmonary hemosiderosis. Can Respir J. 2007 Nov-Dec;14(8):490-3. doi: 10.1155/2007/150926. PMID: 18060095; PMCID: PMC2677775. REFERENCE #3: NICKOL KH. Idiopathic pulmonary haemosiderosis presenting with spontaneous pneumothorax. Tubercle. 1960 Jun;41:216-8. doi: 10.1016/s0041-3879(60)80082-7. PMID: 14426939. DISCLOSURES: No relevant relationships by John Cox, source=Web Response No relevant relationships by Ashley Reluzco, source=Web Response" @default.
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• W3205116280 date "2021-10-01" @default.
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• W3205116280 title "SECONDARY SPONTANEOUS PNEUMOTHORAX IN IDIOPATHIC PULMONARY HEMOSIDEROSIS" @default.
• W3205116280 doi "https://doi.org/10.1016/j.chest.2021.07.1569" @default.
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