Successful Repair of Adult Type II Aortopulmonary Window using Dual Patch Technique

Successful Repair of Adult Type II Aortopulmonary Window using Dual Patch Technique | Research Square window.SnipcartSettings = { analytics: { enabled: false } }; (function() { var accessVector = localStorage.getItem('access_vector') || ''; window.dataLayer = window.dataLayer || []; if (accessVector) { window.dataLayer.push({ user: { profile: { profileInfo: { snid: accessVector } } } }); } })(); (function(w,d,s,l,i){w[l]=w[l]||[];w[l].push({'gtm.start':new Date().getTime(),event:'gtm.js'});var f=d.getElementsByTagName(s)[0],j=d.createElement(s),dl=l!='dataLayer'?'&l='+l:'';j.async=true;j.src='https://www.googletagmanager.com/gtm.js?id='+i+dl;f.parentNode.insertBefore(j,f);})(window,document,'script','dataLayer','GTM-K279D39R'); Browse Preprints In Review Journals COVID-19 Preprints AJE Video Bytes Research Tools Research Promotion AJE Professional Editing AJE Rubriq About Preprint Platform In Review Editorial Policies Our Team Advisory Board Help Center Sign In Submit a Preprint Cite Share Download PDF Case Report Successful Repair of Adult Type II Aortopulmonary Window using Dual Patch Technique Reuben Lamiaki Kynta, Tabang Nyitan, Neelamjingbha Sun, Mrinal Mandal This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-4972015/v1 This work is licensed under a CC BY 4.0 License Status: Posted Version 1 posted You are reading this latest preprint version Abstract Background Aortopulmonary window (APW) is one of the rarest congenital cardiac defects with an incidence of 0.2–0.3% of all cardiac malformations. Patients who present in adulthood with this condition are usually in Eisenmenger syndrome and are inoperable. We present the rare case of an adult with APW with severe pulmonary hypertension who was operable on investigation and successfully treated. Case Presentation A 24 year old female diagnosed with an aortopulmonary window in childhood presented with progressive shortness of breath over the past 6 years. Her clinical examination and investigation revealed a large aortopulmonary window. Cardiac catheterization was done to evaluate the operability of this case and revealed a reversible shunt. The patient underwent successful repair using a double autologous pericardial patch technique. Conclusions Operable aortopulmonary window in an adult is a rare presentation. This report presents the successful repair of aortopulmonary window in an adult. Aortopulmonary window Paediatric Cardiac Surgery Adult Paediatric Cardiac Surgery Figures Figure 1 Figure 2 Figure 3 Background Unoperated patients with aortopulmonary window (APW) most commonly present with pulmonary vascular occlusive disease and eventually Eisenmenger syndrome. Such patients are inoperable( 1 ). We describe the case of a patient with a unrestrictive APW with bidirectional shunt with severe pulmonary artery hypertension who after evaluation was operated successfully using the double patch technique. Case Report A 24 year old female reported to the CTVS Department with progressive shortness of breath and easy fatiguability over 6 years.She was diagnosed with APW in childhood but was lost to follow up. She had no complaints of dysphagia.Clinical examination revealed a systolic murmur in the 3rd left lateral sternal border. Her room air saturation was90%. Chest X ray revealed cardiomegaly and ECG revealed left ventricle (LV)dilation. A 2D transthoracic echo revealed a 23mm APW defect with bidirectional shunt, dilated LV, with a pulmonary artery systolic pressure (PASP) of 83mm Hgand pulmonary artery pressure (PAP) of 53mm Hg with normal Biventricular function. A CT aortogram was done and revealed a large 26mm high defectbetween the great arteries with aberrant right subclavian artery (Fig. 1 ). A catheterization study was done and revealed a mean PAP of 70mmHg, pulmonary artery resistance of 5 Wood units andpulmonary hypertension with pulmonary vascular resistance index (PVRI)of 4 Wood units.m 2 . The shunt showed significantreversibility with oxygen administration, with PVRI on oxygen falling to 0.1 Wood units.m 2 . The patient was counselled and after consent was taken for surgery. AMedian sternotomy was performed. The patient was heparinized. The Aorta and branch pulmonary arteries were dissected, mobilized and looped. A patent ductus arteriosus was found and doubly ligated. In view of the distal APW,cardiopulmonary bypass (CPB) was established using the right femoral artery and dual stage venous cannulation established; the right and left pulmonary arteries were snared. The aorta was cross clamped and antegrade root del Nido cardioplegia was perfused and heart arrested. The LV was vented via the right superior pulmonary vein. An incision was made over the APW and orientation of the ostia of the coronary arteries examined. The defects in the great arteries were measured and two untreated autologous pericardial patches were harvested and sutured to each defect using continuous 5 − 0 polypropylene sutures (Fig. 2 ). The pulmonary artery snares were released and allowed to fill. Deairing manoeuvres were executed and aortic cross clamp was released. The heart resumed sinus rhythm and patient was weaned off CPB.Pre and post repair manometry was conducted and showed a decrease in PAP by 50% (Table 1 ). The cross clamp time was 56 minutes and bypass time was 78 minutes.Postoperatively, she was managed on milrinone infusion and sildenafil injections. She was weaned off ventilator on post operative day 1 and discharged on postoperative day 11.Postoperative CT scan showed no defects between the great arteries (Fig. 3 ) Table 1 Pre and post operative manometry. Pressure (mm Hg) Preoperative Postoperative Left ventricular Systolic 70 95 Diastolic 33 53 Mean 45 37 Main pulmonary artery Systolic 57 36 Diastolic 38 20 Mean 45 26 Discussion APW is one of the rarest of septal defects, accounting for 0.2–0.3% of all congenital heart malformations( 2 ).At least 45% of all APW cases are associated with complex cardiac lesions like interrupted aortic arch or ventricular septal defect( 3 ). Cardiac catheterization is recommended to evaluate the degree of pulmonary vascular disease inpatients older than 6 months of age( 4 ).However there is a recent report of an adult with isolated APW being operated with echocardiographic assessment only( 5 ). Successful treatment of adult APW has been rarely reported. In a 10 year retrospective study, Aggarwal et al reported 6 cases of adult APW who were successfully treated at a mean age of 21.5 years( 6 ). In their 37 year study, Jansen et al encountered only a single case out of their 18 case series( 7 ). Reports of adults with APWwith pregnancy have also been reported albeit with a high peripartum morbidity and mortality( 8 , 9 ) The correction of APW can be done surgically via a ligation, single patch or double patch techniques( 7 ) or by occluder devices( 10 , 11 ) in select cases. A comment on the technique of the single patch or double patch repair is difficult to make in view of the rarity of this condition as well as the rarer complications of surgery like laterrecurrenceof APW( 12 , 13 )and development of aortic pseudoaneurysm( 14 ). If left untreated, 40% of patients will eventually succumb to the sequelae of pulmonary hypertension and Eisenmenger syndrome. Most adults surviving with APW have very high pulmonary vascular pressures, making them inoperable( 1 ). This case is one of the very few reports of successfully operated APW in adulthood. Abbreviations Aortopulmonary window (APW) Left ventricle (LV) Pulmonary artery systolic pressure (PASP) Pulmonary artery pressure (PAP) Pulmonary vascular resistance index (PVRI) Cardiopulmonary bypass (CPB) Declarations Ethics approval and consent to participate This study was conducted in accordance with the Helsinki Declaration. All methods were performed in compliance with relevant guidelines and regulations. Approval from Ethics Board was waived off as it was a retrospective case. The consent to participate in this study was taken from the patient and is available on request from the Authors and Hospital Administration. Consent for publication Consent for publication has been taken from the patient and is available on request from the Hospital Administration Availability of data and materials Available on request from the Authors andHospital Administration Competing interests None Funding None Authors' contributions Investigation: Reuben Lamiaki Kynta, TabangNyitan Methodology: TabangNyitan, Neelamjingbha Sun Writing-original draft: Reuben Lamiaki Kynta, Mrinal Mandal Writing-review &editing: Reuben Lamiaki Kynta, Neelamjingbha Sun All authors read and approved the final manuscript. Acknowledgements None Authors' information Not applicable References Backer CL, Mavroudis C. Surgical management of aortopulmonary window: A 40-year experience. Eur J Cardiothorac Surg. 2002;21(5):773–9. Kutsche LM, Van Mierop LHS. Anatomy and pathogenesis of aorticopulmonary septal defect. American Journal of Cardiology [Internet]. 1987;59(5):443–7. http://dx.doi.org/10.1016/0002-9149(87)90953-2 Bagtharia R, Trivedi KR, Burkhart HM, Williams WG, Freedom RM, Van Arsdell GS, et al. Outcomes for patients with an aortopulmonary window, and the impact of associated Cardiovascular lesions. Cardiol Young. 2004;14(5):473–80. Naimo PS, Konstantinov IE. Aortopulmonary Window Repair in Children. Operative Techniques in Thoracic and Cardiovascular Surgery [Internet]. 2015;20(4):370–83. http://dx.doi.org/10.1053/j.optechstcvs.2016.04.001 Agwar FD, Ganjula SG, Waktola TG. Aorto-pulmonary window in adult patient operated on with echocardiography assessment. Annals of Thoracic Surgery Short Reports [Internet]. 2024;4–6. https://doi.org/10.1016/j.atssr.2024.04.023 Aggarwal SK, Mishra J, Sai V, Iyer VR, Panicker BKP. Aortopulmonary window in adults: Diagnosis and treatment of late-presenting patients. Congenit Heart Dis. 2008;3(5):341–6. Jansen C, Hruda J, Rammeloo L, Ottenkamp J, Hazekamp MG. Surgical repair of aortopulmonary window: Thirty-seven years of experience. PediatrCardiol. 2006;27(5):552–6. Sefiyeva GG, Malishevskaya DS, Chynybekova AN, Bautin AE, Shadrina UM, Alekseeva DV et al. Clinical features and pregnancy outcomes in women with aortopulmonary window defect: case series. Eur Heart J Case Rep [Internet]. 2023;7(10):1–5. https://doi.org/10.1093/ehjcr/ytad501 Thirunavukkarasu B, Kithan LS, Kumar N, Jain A, Bal A. Fatal outcome of congenital aortopulmonary window with patent ductus arteriosus complicating pregnancy. Autops Case Rep. 2021;11:1–7. Giordano M, Butera G. Percutaneous closure of an aortopulmonary window in a young adult patient: A case report of transcatheter closure with an occluder device. Eur Heart J Case Rep. 2020;4(3):1–4. Jureidini SB, Spadaro JJ, Rao PS. Successful transcatheter closure with the buttoned device of aortopulmonary window in an adult. Am J Cardiol. 1998;81(3):371–2. Fukumoto Y, Hosoba S, Takagi S, Goto Y. Relapse of aortopulmonary window 41 years after patch closure. Eur J Cardiothorac Surg. 2020;57(6):1218–20. Hamdy AI, Cooper GR, Shifrin R, Hamilton K, Chandran A. Aorto-Pulmonary Window in an Adult: Utility of Multimodality Imaging. J Am Coll Cardiol [Internet]. 2011;58(4):e7. http://dx.doi.org/10.1016/j.jacc.2010.11.076 Nagashima T, Taira M, Hasegawa M, Kugo Y, Watanabe T, Yoshioka D, et al. Pseudoaneurysm after aortopulmonary window repair and bilateral lung transplantation for eisenmenger syndrome: a case report. J Cardiothorac Surg. 2023;18(1):1–5. Additional Declarations No competing interests reported. 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Also discoverable on Platform About Our Team In Review Editorial Policies Advisory Board Help Center Resources Author Services Accessibility API Access RSS feed Manage Cookie Preferences © Research Square 2026 | ISSN 2693-5015 (online) Privacy Policy Terms of Service Do Not Sell My Personal Information {"props":{"pageProps":{"initialData":{"identity":"rs-4972015","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":357438763,"identity":"20f5afd4-c90f-44c6-9694-6fbb8e593736","order_by":0,"name":"Reuben Lamiaki Kynta","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA70lEQVRIiWNgGAWjYHACNhBhAGZ+AHHZidfCzMA4A8RlJkULMw+ISUiLeXuP2YOPOxiMDW73H/xs82ubPB/Qtg8fc3BrkTlzxtxw5hkGM4M7h5mlc/tuG7YBbZOcuQ23FgmJHDNp3jYGG4MbyQzSuT23GYFa2Jh58WmRf2Mm/Reihfm3Zc9te8JaJHjMpBnbgA67kcwmzfDjdiJhLTxp5Ya9bRLGkjeSzSx7G24ntzEzNuP3C/vhbQ9+ttkY9t1IfHzjx5/btvPbmw9++IhHC0wng8IBIAV0IYhsIKgeDOTB6v4Qp3gUjIJRMApGFgAAHtxKKaUx97AAAAAASUVORK5CYII=","orcid":"","institution":"North Eastern Indira Gandhi Regional Institute of Health and Medical Sciences","correspondingAuthor":true,"prefix":"","firstName":"Reuben","middleName":"Lamiaki","lastName":"Kynta","suffix":""},{"id":357438764,"identity":"3c429b2d-a632-48f0-a0ea-c3f95ef94686","order_by":1,"name":"Tabang Nyitan","email":"","orcid":"","institution":"North Eastern Indira Gandhi Regional Institute of Health and Medical Sciences","correspondingAuthor":false,"prefix":"","firstName":"Tabang","middleName":"","lastName":"Nyitan","suffix":""},{"id":357438765,"identity":"9e90de78-af5b-409f-8b12-33cc52d2104c","order_by":2,"name":"Neelamjingbha Sun","email":"","orcid":"","institution":"North Eastern Indira Gandhi Regional Institute of Health and Medical Sciences","correspondingAuthor":false,"prefix":"","firstName":"Neelamjingbha","middleName":"","lastName":"Sun","suffix":""},{"id":357438766,"identity":"5dac4d23-0079-46cf-9e87-2d4001194e2d","order_by":3,"name":"Mrinal Mandal","email":"","orcid":"","institution":"North Eastern Indira Gandhi Regional Institute of Health and Medical Sciences","correspondingAuthor":false,"prefix":"","firstName":"Mrinal","middleName":"","lastName":"Mandal","suffix":""}],"badges":[],"createdAt":"2024-08-25 09:16:34","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-4972015/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-4972015/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":66847459,"identity":"dde1cb76-5c51-4939-a383-820e866a173a","added_by":"auto","created_at":"2024-10-17 06:21:54","extension":"jpg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":106334,"visible":true,"origin":"","legend":"\u003cp\u003ePreoperative CT Aortogram of APW showing a large defect between the great arteries (*)\u003c/p\u003e","description":"","filename":"FIGURE1.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/6d4a1a0f44e41039bb874ac9.jpg"},{"id":66847461,"identity":"b2473aa7-230e-40d0-b8b2-4666faa811e9","added_by":"auto","created_at":"2024-10-17 06:21:54","extension":"jpg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":226754,"visible":true,"origin":"","legend":"\u003cp\u003eA,B: Intraoperative findings of the APW (*) C. Double patch closure of defects in the great arteries\u003c/p\u003e","description":"","filename":"FIGURE2.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/72784609bac9933def2c1258.jpg"},{"id":66847458,"identity":"71a6160b-26c3-4517-8c08-143cc400243f","added_by":"auto","created_at":"2024-10-17 06:21:54","extension":"jpg","order_by":3,"title":"Figure 3","display":"","copyAsset":false,"role":"figure","size":109790,"visible":true,"origin":"","legend":"\u003cp\u003ePostoperative CT Aortogram showing absence of defect between the great arteries\u003c/p\u003e","description":"","filename":"FIGURE3.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/c990c874692ce00deb3fc1db.jpg"},{"id":74993729,"identity":"219f328e-1781-4c93-b916-be05bf25b2f8","added_by":"auto","created_at":"2025-01-29 08:08:46","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":847445,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/5c3aef41-a3bf-4777-8093-2f43e9920932.pdf"},{"id":66848688,"identity":"6393ecaa-c7e4-47a7-9f27-9804ec6295ae","added_by":"auto","created_at":"2024-10-17 06:29:54","extension":"jpg","order_by":1,"title":"","display":"","copyAsset":false,"role":"supplement","size":141778,"visible":true,"origin":"","legend":"","description":"","filename":"POSTOPPRESSURES.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/cb832e11d1d5fed237ed019c.jpg"},{"id":66847462,"identity":"87848169-a5b6-4fe9-8d42-a022b6f3727f","added_by":"auto","created_at":"2024-10-17 06:21:54","extension":"jpg","order_by":2,"title":"","display":"","copyAsset":false,"role":"supplement","size":135894,"visible":true,"origin":"","legend":"","description":"","filename":"PREOPPRESSURES.jpg","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/8cd8e4ebd70576d1ec58a841.jpg"},{"id":66847463,"identity":"9b86f878-896c-4d27-9dba-24d70b879197","added_by":"auto","created_at":"2024-10-17 06:21:56","extension":"mp4","order_by":3,"title":"","display":"","copyAsset":false,"role":"supplement","size":80791513,"visible":true,"origin":"","legend":"","description":"","filename":"VIDEODESCRIPTION.mp4","url":"https://assets-eu.researchsquare.com/files/rs-4972015/v1/63c87557120c7ccb6f67cf62.mp4"}],"financialInterests":"No competing interests reported.","formattedTitle":"Successful Repair of Adult Type II Aortopulmonary Window using Dual Patch Technique","fulltext":[{"header":"Background","content":"\u003cp\u003eUnoperated patients with aortopulmonary window (APW) most commonly present with pulmonary vascular occlusive disease and eventually Eisenmenger syndrome. Such patients are inoperable(\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e). We describe the case of a patient with a unrestrictive APW with bidirectional shunt with severe pulmonary artery hypertension who after evaluation was operated successfully using the double patch technique.\u003c/p\u003e"},{"header":"Case Report","content":"\u003cp\u003eA 24 year old female reported to the CTVS Department with progressive shortness of breath and easy fatiguability over 6 years.She was diagnosed with APW in childhood but was lost to follow up. She had no complaints of dysphagia.Clinical examination revealed a systolic murmur in the 3rd left lateral sternal border. Her room air saturation was90%.\u003c/p\u003e \u003cp\u003eChest X ray revealed cardiomegaly and ECG revealed left ventricle (LV)dilation. A 2D transthoracic echo revealed a 23mm APW defect with bidirectional shunt, dilated LV, with a pulmonary artery systolic pressure (PASP) of 83mm Hgand pulmonary artery pressure (PAP) of 53mm Hg with normal Biventricular function.\u003c/p\u003e \u003cp\u003eA CT aortogram was done and revealed a large 26mm high defectbetween the great arteries with aberrant right subclavian artery (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eA catheterization study was done and revealed a mean PAP of 70mmHg, pulmonary artery resistance of 5 Wood units andpulmonary hypertension with pulmonary vascular resistance index (PVRI)of 4 Wood units.m\u003csup\u003e2\u003c/sup\u003e. The shunt showed significantreversibility with oxygen administration, with PVRI on oxygen falling to 0.1 Wood units.m\u003csup\u003e2\u003c/sup\u003e.\u003c/p\u003e \u003cp\u003eThe patient was counselled and after consent was taken for surgery.\u003c/p\u003e \u003cp\u003eAMedian sternotomy was performed. The patient was heparinized. The Aorta and branch pulmonary arteries were dissected, mobilized and looped. A patent ductus arteriosus was found and doubly ligated. In view of the distal APW,cardiopulmonary bypass (CPB) was established using the right femoral artery and dual stage venous cannulation established; the right and left pulmonary arteries were snared. The aorta was cross clamped and antegrade root del Nido cardioplegia was perfused and heart arrested. The LV was vented via the right superior pulmonary vein.\u003c/p\u003e \u003cp\u003eAn incision was made over the APW and orientation of the ostia of the coronary arteries examined. The defects in the great arteries were measured and two untreated autologous pericardial patches were harvested and sutured to each defect using continuous 5\u0026thinsp;\u0026minus;\u0026thinsp;0 polypropylene sutures (Fig.\u0026nbsp;\u003cspan refid=\"Fig2\" class=\"InternalRef\"\u003e2\u003c/span\u003e). The pulmonary artery snares were released and allowed to fill. Deairing manoeuvres were executed and aortic cross clamp was released.\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eThe heart resumed sinus rhythm and patient was weaned off CPB.Pre and post repair manometry was conducted and showed a decrease in PAP by 50% (Table\u0026nbsp;\u003cspan refid=\"Tab1\" class=\"InternalRef\"\u003e1\u003c/span\u003e). The cross clamp time was 56 minutes and bypass time was 78 minutes.Postoperatively, she was managed on milrinone infusion and sildenafil injections. She was weaned off ventilator on post operative day 1 and discharged on postoperative day 11.Postoperative CT scan showed no defects between the great arteries (Fig.\u0026nbsp;\u003cspan refid=\"Fig3\" class=\"InternalRef\"\u003e3\u003c/span\u003e)\u003c/p\u003e \u003cp\u003e \u003cdiv class=\"gridtable\"\u003e\u003ctable float=\"Yes\" id=\"Tab1\" border=\"1\"\u003e \u003ccaption language=\"En\"\u003e \u003cdiv class=\"CaptionNumber\"\u003eTable 1\u003c/div\u003e \u003cdiv class=\"CaptionContent\"\u003e \u003cp\u003ePre and post operative manometry.\u003c/p\u003e \u003c/div\u003e \u003c/caption\u003e \u003ccolgroup cols=\"3\"\u003e \u003cdiv align=\"left\" class=\"colspec\" colname=\"c1\" colnum=\"1\"\u003e\u003c/div\u003e \u003cdiv align=\"char\" char=\".\" class=\"colspec\" colname=\"c2\" colnum=\"2\"\u003e\u003c/div\u003e \u003cdiv align=\"char\" char=\".\" class=\"colspec\" colname=\"c3\" colnum=\"3\"\u003e\u003c/div\u003e \u003cthead\u003e \u003ctr\u003e \u003cth align=\"left\" colname=\"c1\"\u003e \u003cp\u003ePressure (mm Hg)\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c2\"\u003e \u003cp\u003ePreoperative\u003c/p\u003e \u003c/th\u003e \u003cth align=\"left\" colname=\"c3\"\u003e \u003cp\u003ePostoperative\u003c/p\u003e \u003c/th\u003e \u003c/tr\u003e \u003c/thead\u003e \u003ctbody\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eLeft ventricular\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSystolic\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e70\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e95\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDiastolic\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e33\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e53\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMean\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e45\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e37\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMain pulmonary artery\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"left\" colname=\"c2\"\u003e\u0026nbsp;\u003c/td\u003e \u003ctd align=\"left\" colname=\"c3\"\u003e\u0026nbsp;\u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eSystolic\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e57\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e36\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eDiastolic\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e38\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e20\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003ctr\u003e \u003ctd align=\"left\" colname=\"c1\"\u003e \u003cp\u003eMean\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c2\"\u003e \u003cp\u003e45\u003c/p\u003e \u003c/td\u003e \u003ctd align=\"char\" char=\".\" colname=\"c3\"\u003e \u003cp\u003e26\u003c/p\u003e \u003c/td\u003e \u003c/tr\u003e \u003c/tbody\u003e \u003c/colgroup\u003e \u003c/table\u003e\u003c/div\u003e \u003c/p\u003e \u003cp\u003e \u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eAPW is one of the rarest of septal defects, accounting for 0.2\u0026ndash;0.3% of all congenital heart malformations(\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e).At least 45% of all APW cases are associated with complex cardiac lesions like interrupted aortic arch or ventricular septal defect(\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eCardiac catheterization is recommended to evaluate the degree of pulmonary vascular disease inpatients older than 6 months of age(\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e).However there is a recent report of an adult with isolated APW being operated with echocardiographic assessment only(\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eSuccessful treatment of adult APW has been rarely reported. In a 10 year retrospective study, Aggarwal et al reported 6 cases of adult APW who were successfully treated at a mean age of 21.5 years(\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e). In their 37 year study, Jansen et al encountered only a single case out of their 18 case series(\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e). Reports of adults with APWwith pregnancy have also been reported albeit with a high peripartum morbidity and mortality(\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e, \u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e)\u003c/p\u003e \u003cp\u003eThe correction of APW can be done surgically via a ligation, single patch or double patch techniques(\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e) or by occluder devices(\u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e, \u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e) in select cases.\u003c/p\u003e \u003cp\u003eA comment on the technique of the single patch or double patch repair is difficult to make in view of the rarity of this condition as well as the rarer complications of surgery like laterrecurrenceof APW(\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e, \u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e)and development of aortic pseudoaneurysm(\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eIf left untreated, 40% of patients will eventually succumb to the sequelae of pulmonary hypertension and Eisenmenger syndrome. Most adults surviving with APW have very high pulmonary vascular pressures, making them inoperable(\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e).\u003c/p\u003e \u003cp\u003eThis case is one of the very few reports of successfully operated APW in adulthood.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cp\u003eAortopulmonary window (APW)\u003c/p\u003e \u003cp\u003eLeft ventricle (LV)\u003c/p\u003e \u003cp\u003ePulmonary artery systolic pressure (PASP)\u003c/p\u003e \u003cp\u003ePulmonary artery pressure (PAP)\u003c/p\u003e \u003cp\u003ePulmonary vascular resistance index (PVRI)\u003c/p\u003e \u003cp\u003eCardiopulmonary bypass (CPB)\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cstrong\u003eEthics approval and consent to participate\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eThis study was conducted in accordance with the Helsinki Declaration. All methods were performed in compliance with relevant guidelines and regulations. Approval from Ethics Board was waived off as it was a retrospective case. The consent to participate in this study was taken from the patient and is available on request from the Authors and Hospital Administration.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConsent for publication\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eConsent for publication has been taken from the patient and is available on request from the Hospital Administration\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAvailability of data and materials\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eAvailable on request from the Authors andHospital Administration\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCompeting interests\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNone\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eFunding\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNone\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026apos; contributions\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eInvestigation: Reuben Lamiaki Kynta, TabangNyitan\u003c/p\u003e\n\u003cp\u003eMethodology: TabangNyitan, Neelamjingbha Sun\u003c/p\u003e\n\u003cp\u003eWriting-original draft: Reuben Lamiaki Kynta, Mrinal Mandal\u003c/p\u003e\n\u003cp\u003eWriting-review \u0026amp;editing: Reuben Lamiaki Kynta, Neelamjingbha Sun\u003c/p\u003e\n\u003cp\u003eAll authors read and approved the final manuscript.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAcknowledgements\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNone\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eAuthors\u0026apos; information\u003c/strong\u003e\u003c/p\u003e\n\u003cp\u003eNot applicable\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eBacker CL, Mavroudis C. Surgical management of aortopulmonary window: A 40-year experience. Eur J Cardiothorac Surg. 2002;21(5):773\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eKutsche LM, Van Mierop LHS. Anatomy and pathogenesis of aorticopulmonary septal defect. American Journal of Cardiology [Internet]. 1987;59(5):443\u0026ndash;7. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttp://dx.doi.org/10.1016/0002-9149(87)90953-2\u003c/span\u003e\u003cspan address=\"10.1016/0002-9149(87)90953-2\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBagtharia R, Trivedi KR, Burkhart HM, Williams WG, Freedom RM, Van Arsdell GS, et al. Outcomes for patients with an aortopulmonary window, and the impact of associated Cardiovascular lesions. Cardiol Young. 2004;14(5):473\u0026ndash;80.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eNaimo PS, Konstantinov IE. Aortopulmonary Window Repair in Children. Operative Techniques in Thoracic and Cardiovascular Surgery [Internet]. 2015;20(4):370\u0026ndash;83. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttp://dx.doi.org/10.1053/j.optechstcvs.2016.04.001\u003c/span\u003e\u003cspan address=\"10.1053/j.optechstcvs.2016.04.001\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAgwar FD, Ganjula SG, Waktola TG. Aorto-pulmonary window in adult patient operated on with echocardiography assessment. Annals of Thoracic Surgery Short Reports [Internet]. 2024;4\u0026ndash;6. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1016/j.atssr.2024.04.023\u003c/span\u003e\u003cspan address=\"10.1016/j.atssr.2024.04.023\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eAggarwal SK, Mishra J, Sai V, Iyer VR, Panicker BKP. Aortopulmonary window in adults: Diagnosis and treatment of late-presenting patients. Congenit Heart Dis. 2008;3(5):341\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eJansen C, Hruda J, Rammeloo L, Ottenkamp J, Hazekamp MG. Surgical repair of aortopulmonary window: Thirty-seven years of experience. PediatrCardiol. 2006;27(5):552\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSefiyeva GG, Malishevskaya DS, Chynybekova AN, Bautin AE, Shadrina UM, Alekseeva DV et al. Clinical features and pregnancy outcomes in women with aortopulmonary window defect: case series. Eur Heart J Case Rep [Internet]. 2023;7(10):1\u0026ndash;5. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttps://doi.org/10.1093/ehjcr/ytad501\u003c/span\u003e\u003cspan address=\"10.1093/ehjcr/ytad501\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eThirunavukkarasu B, Kithan LS, Kumar N, Jain A, Bal A. Fatal outcome of congenital aortopulmonary window with patent ductus arteriosus complicating pregnancy. Autops Case Rep. 2021;11:1\u0026ndash;7.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eGiordano M, Butera G. Percutaneous closure of an aortopulmonary window in a young adult patient: A case report of transcatheter closure with an occluder device. Eur Heart J Case Rep. 2020;4(3):1\u0026ndash;4.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eJureidini SB, Spadaro JJ, Rao PS. Successful transcatheter closure with the buttoned device of aortopulmonary window in an adult. Am J Cardiol. 1998;81(3):371\u0026ndash;2.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eFukumoto Y, Hosoba S, Takagi S, Goto Y. Relapse of aortopulmonary window 41 years after patch closure. Eur J Cardiothorac Surg. 2020;57(6):1218\u0026ndash;20.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eHamdy AI, Cooper GR, Shifrin R, Hamilton K, Chandran A. Aorto-Pulmonary Window in an Adult: Utility of Multimodality Imaging. J Am Coll Cardiol [Internet]. 2011;58(4):e7. \u003cspan class=\"ExternalRef\"\u003e\u003cspan class=\"RefSource\"\u003ehttp://dx.doi.org/10.1016/j.jacc.2010.11.076\u003c/span\u003e\u003cspan address=\"10.1016/j.jacc.2010.11.076\" targettype=\"DOI\" class=\"RefTarget\"\u003e\u003c/span\u003e\u003c/span\u003e\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eNagashima T, Taira M, Hasegawa M, Kugo Y, Watanabe T, Yoshioka D, et al. Pseudoaneurysm after aortopulmonary window repair and bilateral lung transplantation for eisenmenger syndrome: a case report. J Cardiothorac Surg. 2023;18(1):1\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":true,"highlight":"","institution":"","isAcceptedByJournal":false,"isAuthorSuppliedPdf":false,"isDeskRejected":"","isHiddenFromSearch":false,"isInQc":false,"isInWorkflow":false,"isPdf":false,"isPdfUpToDate":true,"isWithdrawnOrRetracted":false,"journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true},"keywords":"Aortopulmonary window, Paediatric Cardiac Surgery, Adult Paediatric Cardiac Surgery","lastPublishedDoi":"10.21203/rs.3.rs-4972015/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-4972015/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003ch2\u003eBackground\u003c/h2\u003e \u003cp\u003eAortopulmonary window (APW) is one of the rarest congenital cardiac defects with an incidence of 0.2\u0026ndash;0.3% of all cardiac malformations. Patients who present in adulthood with this condition are usually in Eisenmenger syndrome and are inoperable. We present the rare case of an adult with APW with severe pulmonary hypertension who was operable on investigation and successfully treated.\u003c/p\u003e\u003ch2\u003eCase Presentation\u003c/h2\u003e \u003cp\u003eA 24 year old female diagnosed with an aortopulmonary window in childhood presented with progressive shortness of breath over the past 6 years. Her clinical examination and investigation revealed a large aortopulmonary window. Cardiac catheterization was done to evaluate the operability of this case and revealed a reversible shunt. The patient underwent successful repair using a double autologous pericardial patch technique.\u003c/p\u003e\u003ch2\u003eConclusions\u003c/h2\u003e \u003cp\u003eOperable aortopulmonary window in an adult is a rare presentation. This report presents the successful repair of aortopulmonary window in an adult.\u003c/p\u003e","manuscriptTitle":"Successful Repair of Adult Type II Aortopulmonary Window using Dual Patch Technique","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2024-10-17 06:21:49","doi":"10.21203/rs.3.rs-4972015/v1","editorialEvents":[{"type":"communityComments","content":0}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"researchsquare","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":true,"externalIdentity":"","sideBox":"","snPcode":"","submissionUrl":"/submission","title":"Research Square","twitterHandle":"researchsquare","acdcEnabled":true,"dfaEnabled":false,"editorialSystem":"","reportingPortfolio":"","inReviewEnabled":false,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"f7c47eb4-3cac-4b3c-a306-d37ab40c0e35","owner":[],"postedDate":"October 17th, 2024","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[],"tags":[],"updatedAt":"2025-01-29T08:08:14+00:00","versionOfRecord":[],"versionCreatedAt":"2024-10-17 06:21:49","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-4972015","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-4972015","identity":"rs-4972015","version":["v1"]},"buildId":"qtupq5eGEP_6zYnWcrvyt","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}