Ruptured infective sinus of Valsalva aneurysm complicated by peripheral mycotic pulmonary artery aneurysm: a case report

Ruptured infective sinus of Valsalva aneurysm complicated by peripheral mycotic pulmonary artery aneurysm: a case report | 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 Ruptured infective sinus of Valsalva aneurysm complicated by peripheral mycotic pulmonary artery aneurysm: a case report Yu Nosaka, Akira Murata, Yoshitaka Yamamoto, Hideyasu Ueda, Hiroki Nakabori, and 3 more This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-8611260/v1 This work is licensed under a CC BY 4.0 License Status: Under Review Version 1 posted 13 You are reading this latest preprint version Abstract Background : Ruptured sinus of Valsalva aneurysm associated with infective endocarditis is a rare but life-threatening condition. Peripheral mycotic pulmonary artery aneurysm is even rarer and carries a high risk of rupture. The coexistence of infective ruptured sinus of Valsalva aneurysm and a peripheral mycotic pulmonary artery aneurysm has not been previously reported. Case presentation : A 58-year-old woman developed pneumonia and infective endocarditis with mobile vegetation in the right ventricular outflow tract, resulting in right ventricular outflow tract stenosis and septic pulmonary embolism. During antibiotic therapy, computed tomography incidentally revealed a peripheral mycotic pulmonary artery aneurysm. Because of the high risk of rupture, urgent transcatheter coil embolization was performed. Although ruptured sinus of Valsalva aneurysm had been considered as a differential diagnosis based on transthoracic echocardiography, definitive diagnosis was not possible due to severe right ventricular outflow tract obstruction. Subsequent transesophageal echocardiography clearly demonstrated a ruptured sinus of Valsalva aneurysm originating from the right coronary sinus and draining into the right ventricle, and definitive surgical repair was successfully performed. The postoperative course was uneventful, with no recurrence of infection or aneurysm enlargement during a two-year follow-up period. Conclusions : This case highlights the importance of early recognition and multidisciplinary decision-making in patients with infective endocarditis complicated by rare vascular lesions. Prioritizing endovascular treatment of a peripheral mycotic pulmonary artery aneurysm before open heart surgery may reduce the risk of catastrophic pulmonary hemorrhage during cardiopulmonary bypass. sinus of Valsalva aneurysm mycotic pulmonary artery aneurysm peripheral pulmonary artery aneurysm infective endocarditis coil embolization Figures Figure 1 Figure 2 Background Ruptured sinus of Valsalva aneurysm (RSVA) is a rare condition with an incidence of approximately 0.09% and is associated with infective endocarditis (IE) in about 8% of cases [ 1 – 2 ]. This pathology requires surgical repair to avoid rapid expansion or heart failure. IE may cause vascular embolism from vegetations and can lead to mycotic aneurysms in systemic and pulmonary arteries. Peripheral mycotic pulmonary artery aneurysm (MPAA) is exceedingly rare but carries a high risk of rapid enlargement and rupture, requiring early intervention [ 3 – 4 ]. We describe an extremely rare case of infective RSVA complicated by a peripheral MPAA, successfully treated by combined interventional and surgical management. Case presentation The patient was a 58-year-old woman with a history of cardiac murmur in childhood but no prior cardiac evaluation. She developed fever and was admitted to a local hospital under a diagnosis of pneumonia. She had recently begun prednisolone therapy (10 mg/day) for polymyalgia rheumatica. Blood cultures isolated Streptococcus mitis and transthoracic echocardiography (TTE) revealed a vegetation in the right ventricular outflow tract (RVOT) causing stenosis. The patient was transferred to our institution under a diagnosis of IE. Laboratory data showed high inflammatory markers (white blood cell count, 12.2 ×10 3 /µL; C-reactive protein, 4.13 mg/dL) and increased brain natriuretic peptide (396 pg/mL). Computed tomography (CT) revealed pneumonia with cavitary lesions in the right upper and lower lobes, as well as embolism of the peripheral right pulmonary arteries. TTE in our hospital also showed a mobile vegetation in the RVOT causing severe stenosis (peak velocity, 3.2 m/s) with a mosaic flow pattern (Fig. 1 A). A) Findings of transthoracic echocardiography in our hospital. White arrow indicates a vegetation in the right ventricular outflow tract with mosaic flow pattern due to severe stenosis on color Doppler imaging. B) White arrow indicates a pulmonary pseudoaneurysm in the right upper lobe. C) Coil embolization of the aneurysm. D) Transesophageal echocardiography shows biphasic flow from the right coronary cusp of the sinus of Valsalva into the right ventricle. The jet flow hindered detailed visualization around the vegetation. Although differential diagnosis of ventricular septal defect (VSD) or RSVA were considered, no definitive diagnosis could be established. Intravenous penicillin G was started, resulting in defervescence, normalization of inflammatory markers, and negative conversion of blood cultures. Two weeks after admission, follow-up CT incidentally detected a peripheral pulmonary artery aneurysm (PAA) in the right upper branch (Fig. 1 B). Because the risk of rupture was considered high and potentially life-threatening even in the absence of hemoptysis, urgent coil embolization was performed using Target coils (Stryker, Tokyo, Japan) (Fig. 1 C). The procedure was completed without complications. On postoperative day 5 after embolization for PAA, transesophageal echocardiography was performed to reassess the vegetation. Regression of the vegetation relieved RVOT stenosis, and the resulting improved visualization enabled identification of a left-to-right shunt flow. Although no VSD was identified, RSVA from the right coronary cusp of the sinus of Valsalva into the RVOT was clearly demonstrated (Fig. 1 D). Urgent surgical repair was planned. Median sternotomy was performed and cardiopulmonary bypass was established. On opening the ascending aorta and main pulmonary artery under cardioplegic arrest, a vegetation was identified in the RVOT (Fig. 2A). Figure 2 A, B) Findings from pulmonary arteriotomy. A large vegetation is present in the right ventricular outflow tract. The fistula is identified by passing a metal probe from the fistula located at the right coronary cusp of the sinus of Valsalva. After debridement, the fistula is closed using the standard technique for VSD closure with a bovine pericardial patch. C, D) Findings from ascending aortotomy. The opening of the same fistula at the right coronary cusp of the sinus of Valsalva is also closed using another bovine pericardial patch. A fistula was unclear from the RVOT side because the vegetation was large. From the aortic side, the fistula was identified at the right coronary cusp. When a metal probe was passed through the fistula, the exit was identified from the center of the vegetation in the RVOT. No VSD was identified, but localized fibrotic thickening was present beneath the right coronary cusp, suggesting a possible history of spontaneous VSD closure. Debridement of the vegetation was followed by closure of the fistula from both the aortic and RVOT sides using bovine pericardial patches (Fig. 2B-D). Operative time was 4 hours 44 minutes, cardiopulmonary bypass time was 171 minutes, and aortic cross-clamp time was 134 minutes. Intraoperative cultures of the vegetation were negative, and intravenous penicillin G was continued postoperatively. Postoperative TTE showed no residual shunt and no recurrence of vegetation. CT showed regression of the pulmonary abscesses and no enlargement of the embolized MPAA. Antibiotic therapy was switched to oral amoxicillin six weeks after initiation of penicillin G. The patient was transferred to another hospital for rehabilitation on postoperative day 21. She remained in good clinical condition at the two-year outpatient follow-up. Discussion The anatomical classification of RSVA proposed by Sakakibara and Konno is widely used [ 5 ]. Type I, characterized by rupture from the right coronary sinus into the RVOT just below the pulmonary valve, accounts for approximately 80% of cases in Japan, whereas type IIIa and type IV are more common in Western countries [ 5 ]. The etiology of SVA includes congenital and acquired causes. Congenital SVA typically originates in the right coronary sinus and is frequently associated with VSD. VSD allows prolapse of the aortic valve and sinus of Valsalva, increasing the risk of RSVA and concomitant aortic regurgitation [ 1 , 6 – 7 ]. RSVA may also occur after spontaneous or surgical closure of VSD [ 6 – 7 ], suggesting a close association between RSVA and VSD. Acquired etiologies include IE, atherosclerosis, syphilis, and trauma [ 1 ]. The present case was classified as type I, and both congenital and acquired etiologies were considered possible because of the history and intraoperative findings. The standard treatment for RSVA is surgical repair [ 8 ]. A modified clinical classification to select the optimal surgical approach was proposed by Xin-jin et al. [ 2 ]. This classification suggests that type I cases are repaired from the right ventricular side alone. However, in the present case, closure was performed using double patches from both the aortic and right ventricular sides because of the fragility of the surrounding infected tissue. The feature that made the present case clinically unique was the coexistence of a peripheral MPAA. The incidence of PAA is extremely low (0.007%), with 11% classified as peripheral lesions [ 9 ]. Although several cases of peripheral MPAA secondary to septic embolism from IE have been reported, no previous report has described the coexistence of peripheral MPAA and infective RSVA. Infection is one of the major causes of peripheral PAA. Proposed mechanisms include direct invasion of the pulmonary artery by an infected focus, such as a lung abscess or cavitary lesion, and ischemic injury caused by infection of the vasa vasorum. In addition, septic emboli can directly penetrate the arterial wall, resulting in MPAA formation [ 4 , 10 ]. MPAA shows a high risk of rupture, with mortality exceeding 50% [ 4 ]. Hemoptysis is regarded as a warning sign of rupture [ 11 – 12 ], and more than half of reported cases of peripheral PAA deaths occurred due to rupture [ 13 ]. This indicates the importance of intervention before hemoptysis occurs. Because of the rarity of PAA, no standardized criteria exist for treatment indication. Surgical lung resection and interventional radiology (IVR) are available treatment options, but IVR is particularly suitable for peripheral lesions because of the minimal invasiveness [ 14 ]. In the present case, preceding IVR for the PAA reduced the overall invasiveness of treatment and prevented fatal pulmonary hemorrhage due to full heparinization during cardiac surgery. Conclusion We encountered an exceptionally rare case of infective RSVA complicated by a peripheral MPAA. Prompt coil embolization of the MPAA followed by surgical repair of the RSVA resulted in a favorable clinical outcome. Early and suitable intervention is crucial for achieving treatment success in patients with coexisting IE and mycotic aneurysms. Abbreviations CT Computed tomography IE Infective endocarditis IVR Interventional radiology MPAA Mycotic pulmonary artery aneurysm PAA Pulmonary artery aneurysm RSVA Ruptured sinus of Valsalva aneurysm RVOT Right ventricular outflow tract TEE Transesophageal echocardiography TTE Transthoracic echocardiography VSD Ventricular septal defect Declarations Ethics approval and consent to participate: Not applicable Consent for publication: A statement that formal consent was obtained Availability of data and materials: No new data were created or analyzed Competing interests: No competing interests Funding: Not applicable Authors’ contributions: Writing; N.Y, Review and editing; M.A, Final approval of the article; all authors Acknowledgements: The authors thank FORTE Science Communications (https://www.forte-science.co.jp/) for English language editing. References Zhaoqi Sun X, Xu Z, Liu. Infectious endocarditis complicated with intracranial infected aneurysm rupture and sinus of valsalva aneurysm rupture. BMC Neurol. 2024;24:372. Xuan LX-JL, Bo P, et al. Modified Sakakibara classification system for ruptured sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg. 2013;146:874–8. Liang C. Biraj Bista. Multiple pulmonary artery mycotic aneurysms and septic emboli in a patient with tricuspid valve vegetation and infective endocarditis. Radiol Case Rep. 2020;16:128–31. Benvenieste O, Bruneel F, Bedos JP, et al. Ruptured mycotic pulmonary artery aneurysm (an unusual complication of right-sided endocarditis). Scand J Infect Dis. 1998;30:626–9. Sakakibara S, Konno S. Congenital aneurysm of the sinus of Valsalva: anatomy and classification. Am Heart J. 1962;63:405–24. Jin Y, Han XM, Wang HS, et al. Coexisting ventricular septal defect affects the features of ruptured sinus of Valsalva aneurysms. Saudi Med J. 2017;38:257–61. Matsushita T, Masuda S, Inoue T, et al. Perforation of sinus Valsalva 10 years after repair of ventricular septal defect. Asian Cardiovasc Thorac Ann. 2012;20:353. Vural KM, Sener E, Taşdemir O, et al. Approach to sinus of Valsalva aneurysms: a review of 53 cases. Eur J Cardiothorac Surg. 2001;20:71–6. Maximilian Kreibich M, Siepe J, Kroll, et al. Aneurysms of the pulmonary artery. Circulation. 2015;131:310–6. Bozkurt AK, Oztunc F, Akman C, et al. Multiple pulmonary artery aneurysms due to infective endocarditis. Ann Thorac Surg. 2003;75:593–6. Shih HH, Kang PL, Lin CY, et al. Main pulmonary artery aneurysm. J Chin Med Assoc. 2007;70:453–5. Tsui EY, Cheung YK, Chow L, et al. Idiopathic pulmonary artery aneurysm: digital subtraction pulmonary angiography grossly underestimates the size of the aneurysm. Clin Imaging. 2001;25:178–80. Monchik J, Wilkins EW Jr. Solitary aneurysm of the middle lobe artery. A case report and review of solitary peripheral pulmonary artery aneurysms. Ann Thorac Surg. 1974;17:496–503. Chiaki Sato T, Kubo E, Shibata, et al. Successful transcatheter embolization of an unruptured idiopathic peripheral pulmonary artery aneurysm. Radiol Case Rep. 2022;18:8–10. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Under Review Version 1 posted Editorial decision: Revision requested 05 Apr, 2026 Reviews received at journal 21 Feb, 2026 Reviews received at journal 15 Feb, 2026 Reviewers agreed at journal 10 Feb, 2026 Reviews received at journal 06 Feb, 2026 Reviewers agreed at journal 06 Feb, 2026 Reviews received at journal 06 Feb, 2026 Reviewers agreed at journal 06 Feb, 2026 Reviewers agreed at journal 05 Feb, 2026 Reviewers invited by journal 05 Feb, 2026 Editor assigned by journal 20 Jan, 2026 Submission checks completed at journal 20 Jan, 2026 First submitted to journal 15 Jan, 2026 You are reading this latest preprint version Research Square lets you share your work early, gain feedback from the community, and start making changes to your manuscript prior to peer review in a journal. As a division of Research Square Company, we’re committed to making research communication faster, fairer, and more useful. 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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-8611260","acceptedTermsAndConditions":true,"allowDirectSubmit":false,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":587707770,"identity":"8f70a98d-0836-4cf6-9ca3-92b86dda3fe5","order_by":0,"name":"Yu Nosaka","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAABBUlEQVRIiWNgGAWjYFADHgaGAx8M/smB2AceEKmF8eGMggPGYC0JRGphNub5cCCxAcTBp8XgeI8Bw8e2e3L8PQfYpHkM7qTPDzv8EGiLnZxuAw4tZ84YMM44U2wscbaBTXKOwbPcjbfTDIBako3NDuDQciPHgJmnIiFxAz8Dm8QbA+bcjbMTQFoOJG7Dp+WPAVQLjwFzuuHs9A+EtTCAbOFtYDbkMTicIC+dg98WyTPHCg72nEkwljhzsPHhDIM0ww3SOQUHEgxw+4XvePPGBz/bEoAhlnzgwIc/NvLys9M3f/hQYSeHS4sCUBwqxdgAcSqYa4BdOQjINxAWGQWjYBSMgpEOANJUZbYS3/CKAAAAAElFTkSuQmCC","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":true,"prefix":"","firstName":"Yu","middleName":"","lastName":"Nosaka","suffix":""},{"id":587707771,"identity":"6bfd90b7-60d8-4b22-85c1-5e0a074e47e3","order_by":1,"name":"Akira Murata","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Akira","middleName":"","lastName":"Murata","suffix":""},{"id":587707772,"identity":"69aa8ca2-c988-497d-a5cb-300a572e335d","order_by":2,"name":"Yoshitaka Yamamoto","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Yoshitaka","middleName":"","lastName":"Yamamoto","suffix":""},{"id":587707773,"identity":"c287c1a9-b471-4dcd-bb13-d66e27f8f35b","order_by":3,"name":"Hideyasu Ueda","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Hideyasu","middleName":"","lastName":"Ueda","suffix":""},{"id":587707774,"identity":"a3e86082-18ca-4227-ac61-f737b8dfa131","order_by":4,"name":"Hiroki Nakabori","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Hiroki","middleName":"","lastName":"Nakabori","suffix":""},{"id":587707775,"identity":"6a23fcd9-45cb-46ed-85d9-35fe15fd95f3","order_by":5,"name":"Ai Sakai","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Ai","middleName":"","lastName":"Sakai","suffix":""},{"id":587707778,"identity":"b3a0995f-db5d-43bb-99f3-99578c9e0d36","order_by":6,"name":"Masaki Ushijima","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Masaki","middleName":"","lastName":"Ushijima","suffix":""},{"id":587707780,"identity":"242a5665-e38f-48e4-b7d7-fce1a80338da","order_by":7,"name":"Kenji Iino","email":"","orcid":"","institution":"Kanazawa University Hospital","correspondingAuthor":false,"prefix":"","firstName":"Kenji","middleName":"","lastName":"Iino","suffix":""}],"badges":[],"createdAt":"2026-01-15 14:08:21","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-8611260/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-8611260/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":102244869,"identity":"1a6eba37-c69a-41d0-9bc4-30228e987e7f","added_by":"auto","created_at":"2026-02-09 17:41:02","extension":"jpeg","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":214117,"visible":true,"origin":"","legend":"\u003cp\u003eA) Findings of transthoracic echocardiography in our hospital. White arrow indicates a vegetation in the right ventricular outflow tract with mosaic flow pattern due to severe stenosis on color Doppler imaging. B) White arrow indicates a pulmonary pseudoaneurysm in the right upper lobe. C) Coil embolization of the aneurysm. D) Transesophageal echocardiography shows biphasic flow from the right coronary cusp of the sinus of Valsalva into the right ventricle.\u003c/p\u003e","description":"","filename":"floatimage1.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8611260/v1/827f5c7df02347e8ecd803db.jpeg"},{"id":102244758,"identity":"5134311c-18f5-4c14-aea2-b9a12be7fe77","added_by":"auto","created_at":"2026-02-09 17:40:47","extension":"jpeg","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":241143,"visible":true,"origin":"","legend":"\u003cp\u003eA, B) Findings from pulmonary arteriotomy. A large vegetation is present in the right ventricular outflow tract. The fistula is identified by passing a metal probe from the fistula located at the right coronary cusp of the sinus of Valsalva. After debridement, the fistula is closed using the standard technique for VSD closure with a bovine pericardial patch. C, D) Findings from ascending aortotomy. The opening of the same fistula at the right coronary cusp of the sinus of Valsalva is also closed using another bovine pericardial patch.\u003c/p\u003e","description":"","filename":"floatimage2.jpeg","url":"https://assets-eu.researchsquare.com/files/rs-8611260/v1/192248cbc78ad024a61b405a.jpeg"},{"id":102244907,"identity":"b5a56b53-e530-46c0-965a-1325547c319c","added_by":"auto","created_at":"2026-02-09 17:41:17","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":780875,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-8611260/v1/b887e3f7-30d8-4fd2-847b-add6531632be.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"Ruptured infective sinus of Valsalva aneurysm complicated by peripheral mycotic pulmonary artery aneurysm: a case report","fulltext":[{"header":"Background","content":"\u003cp\u003eRuptured sinus of Valsalva aneurysm (RSVA) is a rare condition with an incidence of approximately 0.09% and is associated with infective endocarditis (IE) in about 8% of cases [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. This pathology requires surgical repair to avoid rapid expansion or heart failure. IE may cause vascular embolism from vegetations and can lead to mycotic aneurysms in systemic and pulmonary arteries. Peripheral mycotic pulmonary artery aneurysm (MPAA) is exceedingly rare but carries a high risk of rapid enlargement and rupture, requiring early intervention [\u003cspan citationid=\"CR3\" class=\"CitationRef\"\u003e3\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e]. We describe an extremely rare case of infective RSVA complicated by a peripheral MPAA, successfully treated by combined interventional and surgical management.\u003c/p\u003e "},{"header":"Case presentation","content":"\u003cp\u003eThe patient was a 58-year-old woman with a history of cardiac murmur in childhood but no prior cardiac evaluation. She developed fever and was admitted to a local hospital under a diagnosis of pneumonia. She had recently begun prednisolone therapy (10 mg/day) for polymyalgia rheumatica. Blood cultures isolated \u003cem\u003eStreptococcus mitis\u003c/em\u003e and transthoracic echocardiography (TTE) revealed a vegetation in the right ventricular outflow tract (RVOT) causing stenosis. The patient was transferred to our institution under a diagnosis of IE.\u003c/p\u003e \u003cp\u003eLaboratory data showed high inflammatory markers (white blood cell count, 12.2 \u0026times;10\u003csup\u003e3\u003c/sup\u003e/\u0026micro;L; C-reactive protein, 4.13 mg/dL) and increased brain natriuretic peptide (396 pg/mL). Computed tomography (CT) revealed pneumonia with cavitary lesions in the right upper and lower lobes, as well as embolism of the peripheral right pulmonary arteries. TTE in our hospital also showed a mobile vegetation in the RVOT causing severe stenosis (peak velocity, 3.2 m/s) with a mosaic flow pattern (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eA).\u003c/p\u003e \u003cp\u003e \u003c/p\u003e \u003cp\u003eA) Findings of transthoracic echocardiography in our hospital. White arrow indicates a vegetation in the right ventricular outflow tract with mosaic flow pattern due to severe stenosis on color Doppler imaging. B) White arrow indicates a pulmonary pseudoaneurysm in the right upper lobe. C) Coil embolization of the aneurysm. D) Transesophageal echocardiography shows biphasic flow from the right coronary cusp of the sinus of Valsalva into the right ventricle.\u003c/p\u003e \u003cp\u003eThe jet flow hindered detailed visualization around the vegetation. Although differential diagnosis of ventricular septal defect (VSD) or RSVA were considered, no definitive diagnosis could be established.\u003c/p\u003e \u003cp\u003eIntravenous penicillin G was started, resulting in defervescence, normalization of inflammatory markers, and negative conversion of blood cultures. Two weeks after admission, follow-up CT incidentally detected a peripheral pulmonary artery aneurysm (PAA) in the right upper branch (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eB). Because the risk of rupture was considered high and potentially life-threatening even in the absence of hemoptysis, urgent coil embolization was performed using Target coils (Stryker, Tokyo, Japan) (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eC). The procedure was completed without complications. On postoperative day 5 after embolization for PAA, transesophageal echocardiography was performed to reassess the vegetation. Regression of the vegetation relieved RVOT stenosis, and the resulting improved visualization enabled identification of a left-to-right shunt flow. Although no VSD was identified, RSVA from the right coronary cusp of the sinus of Valsalva into the RVOT was clearly demonstrated (Fig.\u0026nbsp;\u003cspan refid=\"Fig1\" class=\"InternalRef\"\u003e1\u003c/span\u003eD). Urgent surgical repair was planned.\u003c/p\u003e \u003cp\u003e Median sternotomy was performed and cardiopulmonary bypass was established. On opening the ascending aorta and main pulmonary artery under cardioplegic arrest, a vegetation was identified in the RVOT (Fig.\u0026nbsp;2A).\u003c/p\u003e \u003cp\u003eFigure\u0026nbsp;2\u003c/p\u003e \u003cp\u003eA, B) Findings from pulmonary arteriotomy. A large vegetation is present in the right ventricular outflow tract. The fistula is identified by passing a metal probe from the fistula located at the right coronary cusp of the sinus of Valsalva. After debridement, the fistula is closed using the standard technique for VSD closure with a bovine pericardial patch.\u003c/p\u003e \u003cp\u003eC, D) Findings from ascending aortotomy. The opening of the same fistula at the right coronary cusp of the sinus of Valsalva is also closed using another bovine pericardial patch.\u003c/p\u003e \u003cp\u003eA fistula was unclear from the RVOT side because the vegetation was large. From the aortic side, the fistula was identified at the right coronary cusp. When a metal probe was passed through the fistula, the exit was identified from the center of the vegetation in the RVOT. No VSD was identified, but localized fibrotic thickening was present beneath the right coronary cusp, suggesting a possible history of spontaneous VSD closure. Debridement of the vegetation was followed by closure of the fistula from both the aortic and RVOT sides using bovine pericardial patches (Fig.\u0026nbsp;2B-D). Operative time was 4 hours 44 minutes, cardiopulmonary bypass time was 171 minutes, and aortic cross-clamp time was 134 minutes.\u003c/p\u003e \u003cp\u003eIntraoperative cultures of the vegetation were negative, and intravenous penicillin G was continued postoperatively. Postoperative TTE showed no residual shunt and no recurrence of vegetation. CT showed regression of the pulmonary abscesses and no enlargement of the embolized MPAA. Antibiotic therapy was switched to oral amoxicillin six weeks after initiation of penicillin G. The patient was transferred to another hospital for rehabilitation on postoperative day 21. She remained in good clinical condition at the two-year outpatient follow-up.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eThe anatomical classification of RSVA proposed by Sakakibara and Konno is widely used [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e]. Type I, characterized by rupture from the right coronary sinus into the RVOT just below the pulmonary valve, accounts for approximately 80% of cases in Japan, whereas type IIIa and type IV are more common in Western countries [\u003cspan citationid=\"CR5\" class=\"CitationRef\"\u003e5\u003c/span\u003e]. The etiology of SVA includes congenital and acquired causes. Congenital SVA typically originates in the right coronary sinus and is frequently associated with VSD. VSD allows prolapse of the aortic valve and sinus of Valsalva, increasing the risk of RSVA and concomitant aortic regurgitation [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e, \u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e]. RSVA may also occur after spontaneous or surgical closure of VSD [\u003cspan citationid=\"CR6\" class=\"CitationRef\"\u003e6\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR7\" class=\"CitationRef\"\u003e7\u003c/span\u003e], suggesting a close association between RSVA and VSD. Acquired etiologies include IE, atherosclerosis, syphilis, and trauma [\u003cspan citationid=\"CR1\" class=\"CitationRef\"\u003e1\u003c/span\u003e]. The present case was classified as type I, and both congenital and acquired etiologies were considered possible because of the history and intraoperative findings. The standard treatment for RSVA is surgical repair [\u003cspan citationid=\"CR8\" class=\"CitationRef\"\u003e8\u003c/span\u003e]. A modified clinical classification to select the optimal surgical approach was proposed by Xin-jin et al. [\u003cspan citationid=\"CR2\" class=\"CitationRef\"\u003e2\u003c/span\u003e]. This classification suggests that type I cases are repaired from the right ventricular side alone. However, in the present case, closure was performed using double patches from both the aortic and right ventricular sides because of the fragility of the surrounding infected tissue.\u003c/p\u003e \u003cp\u003eThe feature that made the present case clinically unique was the coexistence of a peripheral MPAA. The incidence of PAA is extremely low (0.007%), with 11% classified as peripheral lesions [\u003cspan citationid=\"CR9\" class=\"CitationRef\"\u003e9\u003c/span\u003e]. Although several cases of peripheral MPAA secondary to septic embolism from IE have been reported, no previous report has described the coexistence of peripheral MPAA and infective RSVA. Infection is one of the major causes of peripheral PAA. Proposed mechanisms include direct invasion of the pulmonary artery by an infected focus, such as a lung abscess or cavitary lesion, and ischemic injury caused by infection of the vasa vasorum. In addition, septic emboli can directly penetrate the arterial wall, resulting in MPAA formation [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e, \u003cspan citationid=\"CR10\" class=\"CitationRef\"\u003e10\u003c/span\u003e]. MPAA shows a high risk of rupture, with mortality exceeding 50% [\u003cspan citationid=\"CR4\" class=\"CitationRef\"\u003e4\u003c/span\u003e]. Hemoptysis is regarded as a warning sign of rupture [\u003cspan citationid=\"CR11\" class=\"CitationRef\"\u003e11\u003c/span\u003e\u0026ndash;\u003cspan citationid=\"CR12\" class=\"CitationRef\"\u003e12\u003c/span\u003e], and more than half of reported cases of peripheral PAA deaths occurred due to rupture [\u003cspan citationid=\"CR13\" class=\"CitationRef\"\u003e13\u003c/span\u003e]. This indicates the importance of intervention before hemoptysis occurs. Because of the rarity of PAA, no standardized criteria exist for treatment indication. Surgical lung resection and interventional radiology (IVR) are available treatment options, but IVR is particularly suitable for peripheral lesions because of the minimal invasiveness [\u003cspan citationid=\"CR14\" class=\"CitationRef\"\u003e14\u003c/span\u003e]. In the present case, preceding IVR for the PAA reduced the overall invasiveness of treatment and prevented fatal pulmonary hemorrhage due to full heparinization during cardiac surgery.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eWe encountered an exceptionally rare case of infective RSVA complicated by a peripheral MPAA. Prompt coil embolization of the MPAA followed by surgical repair of the RSVA resulted in a favorable clinical outcome. Early and suitable intervention is crucial for achieving treatment success in patients with coexisting IE and mycotic aneurysms.\u003c/p\u003e"},{"header":"Abbreviations","content":"\u003cdiv class=\"DefinitionList\"\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eCT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eComputed tomography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eIE\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eInfective endocarditis\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eIVR\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eInterventional radiology\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eMPAA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eMycotic pulmonary artery aneurysm\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003ePAA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003ePulmonary artery aneurysm\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eRSVA\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eRuptured sinus of Valsalva aneurysm\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eRVOT\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eRight ventricular outflow tract\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eTEE\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eTransesophageal echocardiography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eTTE\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eTransthoracic echocardiography\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003cdiv class=\"DefinitionListEntry\"\u003e \u003cdiv class=\"Term\"\u003eVSD\u003c/div\u003e \u003cdiv class=\"Description\"\u003e \u003cp\u003eVentricular septal defect\u003c/p\u003e \u003c/div\u003e \u003c/div\u003e \u003c/div\u003e"},{"header":"Declarations","content":"\u003cp\u003eEthics approval and consent to participate: Not applicable\u003c/p\u003e\n\u003cp\u003eConsent for publication: A statement that formal consent was obtained\u003c/p\u003e\n\u003cp\u003eAvailability of data and materials: No new data were created or analyzed\u003c/p\u003e\n\u003cp\u003eCompeting interests: No competing interests\u003c/p\u003e\n\u003cp\u003eFunding: Not applicable\u003c/p\u003e\n\u003cp\u003eAuthors\u0026rsquo; contributions: Writing; N.Y, Review and editing; M.A, Final approval of the article; all authors\u003c/p\u003e\n\u003cp\u003eAcknowledgements:\u003c/p\u003e\n\u003cp\u003eThe authors thank FORTE Science Communications (https://www.forte-science.co.jp/) for English language editing.\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\u003cli\u003e\u003cspan\u003eZhaoqi Sun X, Xu Z, Liu. Infectious endocarditis complicated with intracranial infected aneurysm rupture and sinus of valsalva aneurysm rupture. BMC Neurol. 2024;24:372.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eXuan LX-JL, Bo P, et al. Modified Sakakibara classification system for ruptured sinus of Valsalva aneurysm. J Thorac Cardiovasc Surg. 2013;146:874\u0026ndash;8.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eLiang C. Biraj Bista. Multiple pulmonary artery mycotic aneurysms and septic emboli in a patient with tricuspid valve vegetation and infective endocarditis. Radiol Case Rep. 2020;16:128\u0026ndash;31.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBenvenieste O, Bruneel F, Bedos JP, et al. Ruptured mycotic pulmonary artery aneurysm (an unusual complication of right-sided endocarditis). Scand J Infect Dis. 1998;30:626\u0026ndash;9.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eSakakibara S, Konno S. Congenital aneurysm of the sinus of Valsalva: anatomy and classification. Am Heart J. 1962;63:405\u0026ndash;24.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eJin Y, Han XM, Wang HS, et al. Coexisting ventricular septal defect affects the features of ruptured sinus of Valsalva aneurysms. Saudi Med J. 2017;38:257\u0026ndash;61.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMatsushita T, Masuda S, Inoue T, et al. Perforation of sinus Valsalva 10 years after repair of ventricular septal defect. Asian Cardiovasc Thorac Ann. 2012;20:353.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eVural KM, Sener E, Taşdemir O, et al. Approach to sinus of Valsalva aneurysms: a review of 53 cases. Eur J Cardiothorac Surg. 2001;20:71\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMaximilian Kreibich M, Siepe J, Kroll, et al. Aneurysms of the pulmonary artery. Circulation. 2015;131:310\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eBozkurt AK, Oztunc F, Akman C, et al. Multiple pulmonary artery aneurysms due to infective endocarditis. Ann Thorac Surg. 2003;75:593\u0026ndash;6.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eShih HH, Kang PL, Lin CY, et al. Main pulmonary artery aneurysm. J Chin Med Assoc. 2007;70:453\u0026ndash;5.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eTsui EY, Cheung YK, Chow L, et al. Idiopathic pulmonary artery aneurysm: digital subtraction pulmonary angiography grossly underestimates the size of the aneurysm. Clin Imaging. 2001;25:178\u0026ndash;80.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eMonchik J, Wilkins EW Jr. Solitary aneurysm of the middle lobe artery. A case report and review of solitary peripheral pulmonary artery aneurysms. Ann Thorac Surg. 1974;17:496\u0026ndash;503.\u003c/span\u003e\u003c/li\u003e \u003cli\u003e\u003cspan\u003eChiaki Sato T, Kubo E, Shibata, et al. Successful transcatheter embolization of an unruptured idiopathic peripheral pulmonary artery aneurysm. Radiol Case Rep. 2022;18:8\u0026ndash;10.\u003c/span\u003e\u003c/li\u003e\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":false,"hideJournal":false,"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":"journal-of-cardiothoracic-surgery","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"jcts","sideBox":"Learn more about [Journal of Cardiothoracic Surgery](http://cardiothoracicsurgery.biomedcentral.com)","snPcode":"13019","submissionUrl":"https://submission.nature.com/new-submission/13019/3","title":"Journal of Cardiothoracic Surgery","twitterHandle":"@BioMedCentral","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true},"keywords":"sinus of Valsalva aneurysm, mycotic pulmonary artery aneurysm, peripheral pulmonary artery aneurysm, infective endocarditis, coil embolization","lastPublishedDoi":"10.21203/rs.3.rs-8611260/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-8611260/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eBackground\u003c/strong\u003e: Ruptured sinus of Valsalva aneurysm associated with infective endocarditis is a rare but life-threatening condition. Peripheral mycotic pulmonary artery aneurysm is even rarer and carries a high risk of rupture. The coexistence of infective ruptured sinus of Valsalva aneurysm and a peripheral mycotic pulmonary artery aneurysm has not been previously reported.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase presentation\u003c/strong\u003e: A 58-year-old woman developed pneumonia and infective endocarditis with mobile vegetation in the right ventricular outflow tract, resulting in right ventricular outflow tract stenosis and septic pulmonary embolism. During antibiotic therapy, computed tomography incidentally revealed a peripheral mycotic pulmonary artery aneurysm. Because of the high risk of rupture, urgent transcatheter coil embolization was performed. Although ruptured sinus of Valsalva aneurysm had been considered as a differential diagnosis based on transthoracic echocardiography, definitive diagnosis was not possible due to severe right ventricular outflow tract obstruction. Subsequent transesophageal echocardiography clearly demonstrated a ruptured sinus of Valsalva aneurysm originating from the right coronary sinus and draining into the right ventricle, and definitive surgical repair was successfully performed. The postoperative course was uneventful, with no recurrence of infection or aneurysm enlargement during a two-year follow-up period.\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eConclusions\u003c/strong\u003e: This case highlights the importance of early recognition and multidisciplinary decision-making in patients with infective endocarditis complicated by rare vascular lesions. Prioritizing endovascular treatment of a peripheral mycotic pulmonary artery aneurysm before open heart surgery may reduce the risk of catastrophic pulmonary hemorrhage during cardiopulmonary bypass.\u003c/p\u003e","manuscriptTitle":"Ruptured infective sinus of Valsalva aneurysm complicated by peripheral mycotic pulmonary artery aneurysm: a case report","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2026-02-09 17:40:10","doi":"10.21203/rs.3.rs-8611260/v1","editorialEvents":[{"type":"communityComments","content":0},{"type":"decision","content":"Revision requested","date":"2026-04-05T13:03:17+00:00","index":"","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2026-02-21T07:07:22+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2026-02-15T15:57:24+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"236653946240815554906174506084913619421","date":"2026-02-10T23:56:13+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2026-02-06T17:26:07+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"339372094022686357661504260429668727926","date":"2026-02-06T16:41:33+00:00","index":"hide","fulltext":""},{"type":"editorInvitedReview","content":"","date":"2026-02-06T07:32:00+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"304800627683722335422515104565847744675","date":"2026-02-06T06:37:01+00:00","index":"hide","fulltext":""},{"type":"reviewerAgreed","content":"138990949105972405038642017270039370176","date":"2026-02-05T12:15:53+00:00","index":"hide","fulltext":""},{"type":"reviewersInvited","content":"","date":"2026-02-05T09:34:37+00:00","index":"","fulltext":""},{"type":"editorAssigned","content":"","date":"2026-01-20T06:15:56+00:00","index":"","fulltext":""},{"type":"checksComplete","content":"","date":"2026-01-20T06:15:04+00:00","index":"","fulltext":""},{"type":"submitted","content":"Journal of Cardiothoracic Surgery","date":"2026-01-15T13:58:42+00:00","index":"","fulltext":""}],"status":"published","journal":{"display":true,"email":"[email protected]","identity":"journal-of-cardiothoracic-surgery","isNatureJournal":false,"hasQc":true,"allowDirectSubmit":false,"externalIdentity":"jcts","sideBox":"Learn more about [Journal of Cardiothoracic Surgery](http://cardiothoracicsurgery.biomedcentral.com)","snPcode":"13019","submissionUrl":"https://submission.nature.com/new-submission/13019/3","title":"Journal of Cardiothoracic Surgery","twitterHandle":"@BioMedCentral","acdcEnabled":true,"dfaEnabled":true,"editorialSystem":"em","reportingPortfolio":"BMC/SO AJ","inReviewEnabled":true,"inReviewRevisionsEnabled":true}}],"origin":"","ownerIdentity":"1c1af23c-2bd9-43ea-bdb4-54a0c3269230","owner":[],"postedDate":"February 9th, 2026","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"under-review","subjectAreas":[],"tags":[],"updatedAt":"2026-04-19T04:08:18+00:00","versionOfRecord":[],"versionCreatedAt":"2026-02-09 17:40:10","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-8611260","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-8611260","identity":"rs-8611260","version":["v1"]},"buildId":"XKTyCvWXoU3ODBz1xrDgd","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}