Percutaneous and Invasive Management of a Rare Congenital Pulmonary Malformation in an Active Duty Service Member LT Brennan Shutt, D.O. (Associate) LCDR.

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Percutaneous and Invasive Management of a Rare Congenital Pulmonary Malformation in an Active Duty Service Member LT Brennan Shutt, D.O. (Associate) LCDR Eric Shafer, D.O. (Member) CDR Keshav Nayak, M.D. CAPT Daniel Gramins, M.D. Departments of Internal Medicine, Cardiology, and Cardiothoracic Surgery Naval Medical Center San Diego

Disclosures Brennan J. Shutt, DO Internal Medicine Resident, PGY-3 I have no relevant financial disclosures

Case Presentation A 26-year-old Caucasian active duty male LCPL USMC was referred to the cardiology department after an incidental vascular abnormality was on imaging work up for hematuria Further clinical investigation revealed a history which was significant for recurrent pulmonary infections, intermittent dyspnea with chest discomfort, and episodic hemoptysis for 12 months prior to presentation

History PMH: Rhabdomyolysis PSH: None Meds: None ALL: No known drug allergies SOC: No tobacco or alcohol use, Marine Unit Combat Logistics Battalion-7

Computed tomography scan axial cut

3-D Reconstructions

Diagnosis Right-sided intralobar sequestration with left-to-left shunting from an anomalous 6- millimeter systemic arterial vessel

Pulmonary Sequestration Mass of non-functioning lung tissue separated from the normal bronchopulmonary tree and vascularized by an aberrant systemic artery 0.2 to 6% of all congenital pulmonary malformations Intralobar 75% vs Extralobar 25% Pryce Classification I, II, III

Clinical presentation Age at diagnosis Repeated respiratory tract infections, hemoptysis, exertional dyspnea, chest pain, hemothorax, congestive heart failure, pneumothorax, heart murmur, incidental finding on imaging Associated congenital malformations

Treatment Options Surgery vs endovascular management vs both Bleeding complications with surgical management alone Transcatheter embolization Amplatzer® vascular plug vs microcoil vs PVA

Multi-disciplinary Management Multi-disciplinary conference consensus decision to reduce intra-operative bleeding with prophylactic plug embolization of feeder artery Underwent successful catheterization procedure with Amplatzer® Vascular Plug II with acute closure of feeder artery

Amplatzer® vascular plug II Indicated for peripheral arterial and venous embolization Multi-layered, multi-lobed Nitinol mesh for rapid embolization Targeted delivery with repositioning Occludes vessel faster with single device Reduces radiation exposure

Patient Outcome Uncomplicated VATS wedge resection of right lower lobe Minimal intraoperative blood loss Discharged 2 days post-operatively At 14 months post procedure, no further episodes of recurrent pulmonary infections, hemoptysis, or functional impairments

Conclusions Rare congenital malformation, good prognosis Second case of prophylactic embolization prior to definitive surgical management Minimally invasive embolization, minimally invasive surgery Decreases length of hospitalization, complications mitigated

References Halkic N, Cuénoud PF, Corthésy ME, Ksontini R, Boumghar M. Pulmonary sequestration: a review of 26 cases. Eur J Cardiothorac Surg 1998; 14: Jiang S, Shi JY, Zhu XH, Chen C, Sun XW, Yu D, Jie B. Endovascular embolization of the complete type of anomalous systemic arterial supply to normal basal lung segments. Chest 2011; 139(6): Kim TE, Kwon JH, Kim JS. Transcatheter embolization for massive hemoptysis from an intralobar pulmonary sequestration: a case report. Clinical Key; 38(3): Cho MJ, Kim DY, Kim SC, Kim KS, Kim EA, Lee BS. Embolization versus surgical resection of pulmonary sequestration: clinical experiences with a thorascopic approach. Journal of Pediatric Surgery 2012; 47: Madhusudhan KS, Das CJ, Dutta R, Kumar A, Bhalla AS. Endovascular embolization of pulmonary sequestration in an adult. J Vasc Interv Radiol 2009; 20: Marine L, Valdes FE, Mertens RM, Bergoeing MR, Kramer A. Endovascular treatment of symptomatic pulmonary sequestration. Ann Vasc Surg 2011;25:11-15 Thurber JS, Unger JA, DeVries WC. A United States Marine presenting with hemoptysis after push-ups. Military Medicine 2014; 179: Oermann CM. Bronchopulmonary sequestration. Leoncini G, Rossi UG, Ferro C, Chessa L. Endovascular treatment of pulmonary sequestration in adults using Amplatzer® vascular plugs. Interactive Cardiovascular and Thoracic Surgery 2011; 12: Image from Image from devices/amplatzer-family-of-vascular-plugs