Scrotal Involvement in Gist: a Case Report of Testicular Metastasis and Literature Review

Scrotal Involvement in Gist: a Case Report of Testicular Metastasis and Literature Review | 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 Scrotal Involvement in Gist: a Case Report of Testicular Metastasis and Literature Review AYARI JIHENE, AZZA BOUAZIZ, HAYTHEM ELASKRI, HADDAOUI ABDERRAZEK This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-6058098/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 Introduction: Gastrointestinal stromal tumors (GISTs) are mesenchymal tumors of the digestive tract originating from interstitial Cajal cells. Mutations in the KIT, PDGFRA, and occasionally BRAF genes are the main drivers of oncogenesis (1). The stomach is the most common primary location, followed by the small intestine. Metastatic GISTs most frequently involve the liver and peritoneum. However, scrotal involvement is exceedingly rare. Case Presentation: A 63-year-old man was diagnosed in 2012 with ileal GIST and peritoneal carcinomatosis. He underwent ileal resection and received Imatinib (400 mg daily) until 2018. Following disease progression in 2020, Imatinib was reintroduced at the same dose, resulting in a complete response. The patient was subsequently lost to follow-up until 2022, when he presented with abdominal and scrotal swelling. A testicular ultrasound revealed a large mass in the right scrotum, and a CT scan showed extensive intraperitoneal masses and multiple liver nodules. A right orchiectomy was performed. Histological examination identified a malignant spindle cell mesenchymal tumor with sarcomatous involvement of the testicular parenchyma without direct invasion. Postoperatively, the patient resumed Imatinib (400 mg daily), later increased to 800 mg due to disease progression in the liver and peritoneal masses in July 2023. By January 2024, further liver progression led to the initiation of Sunitinib. No additional scrotal metastases were noted. Figures Figure 1 Introduction Gastrointestinal stromal tumors (GISTs) are mesenchymal neoplasms originating from the interstitial cells of Cajal in the digestive tract. Mutations in the KIT, PDGFRA, and occasionally BRAF genes are the primary drivers of oncogenesis (1). The stomach is the most common site of origin, followed by the small intestine. Metastatic GISTs predominantly affect the liver and peritoneum. Scrotal involvement, however, remains exceedingly rare. Case Presentation A 63-year-old male was diagnosed in 2012 with ileal GIST and peritoneal carcinomatosis. He underwent ileal resection and received Imatinib (400 mg daily) until 2018. Upon disease progression in 2020, Imatinib was reintroduced at the same dose, resulting in a complete response. However, the patient was lost to follow-up until 2022, when he presented with abdominal and scrotal swelling. A testicular ultrasound revealed a large mass in the right scrotum, and a CT scan showed extensive intraperitoneal masses and multiple liver nodules. A right orchiectomy was performed. Histological examination revealed a malignant spindle cell mesenchymal tumor with sarcomatous involvement of the testicular parenchyma without direct invasion. Postoperatively, the patient resumed Imatinib (400 mg daily), which was later increased to 800 mg due to disease progression in the liver and peritoneum in July 2023. By January 2024, further progression in the liver led to the initiation of Sunitinib. No additional scrotal metastases were noted. Discussion Scrotal and Testicular Metastases of GIST: The first case of testicular metastasis from a GIST was reported by Dorić et al. in 2007 (2). The patient presented with right testicular enlargement seven months after resection of a jejunal GIST. Orchiectomy revealed tumor nodules within the tunica vaginalis, with histology confirming metastatic GIST tissue. The testicular parenchyma was not involved. Similar cases were described by Perrone et al. in 2008 (3) and Thomas et al. in 2015 (4), both proposing metastasis occurred via a persistent peritoneo-vaginalis duct. The peritoneo-vaginalis duct is an embryonic structure that connects the peritoneal cavity to the inguinal region, aiding in testicular descent. It typically closes after birth; however, its persistence may result in conditions such as indirect inguinal hernia or hydrocele (5). In the context of malignancies originating from the gastrointestinal or genitourinary tract, a patent peritoneo-vaginalis duct may facilitate metastatic spread. This hypothesis is further supported by a case reported by Ahmad et al. in 2015, where metastatic GIST tissue was found in the epididymis (6). Recently, Rota et al. (2024) described an unusual case of GIST metastasizing to the scrotum, penis, and myocardium (7). E-GIST Involving the Scrotum: Extra-gastrointestinal stromal tumors (E-GISTs) account for approximately 5% of all GISTs. Primary scrotal E-GISTs are exceptionally rare. In 2004, Froehner et al. (8) reported a case of a scrotal mass extending from the abdomen along the inguinal canal, associated with large abdominal masses. A CT-guided biopsy confirmed GIST histology, but the primary site could not be determined. In 2007, Kang et al. (9) described a scrotal E-GIST with no evidence of an alternative primary site, a finding further corroborated by Ali et al. in 2020 (10). These cases highlight the need for thorough histological evaluation to distinguish E-GISTs from other differential diagnoses, such as germ cell tumors. Conclusion GISTs can metastasize to unusual locations, including the testis and scrotum. Diagnosis is confirmed using immunohistochemical markers such as CD117 and CD34. For localized primary GISTs, surgical resection remains the cornerstone of treatment, followed by adjuvant Imatinib based on recurrence risk. However, metastatic GISTs carry a poor prognosis due to resistance to tyrosine kinase inhibitors (TKIs), rendering the management of rare metastatic sites particularly challenging. Declarations Participant Consent Statement: I, the undersigned, Pr AYARI JIHENE, hereby confirm that all participants have given their informed consent to participate in this study. This consent was obtained in writing and/or verbally after the participants were informed of the study's objectives, methods, risks, and potential benefits. The participants were given the opportunity to ask any necessary questions and expressed their willingness to participate with full knowledge of the study No founding to declare. Author Contribution ALL AUTHORS PREPARED THIS PAPER AND REVIEWED IT References Miettinen M, Lasota J. Gastrointestinal stromal tumors: review on morphology, molecular pathology, prognosis, and differential diagnosis. Arch Pathol Lab Med. 2006 Oct;130(10):1466-78. doi: 10.5858/2006-130-1466-GSTROM. PMID: 17090188. Dorić M, Radović S, Babić M, et al. Testicular metastasis of gastrointestinal stromal tumor of the jejunum. Bosn J Basic Med Sci. 2007 May;7(2):176-9. doi: 10.17305/bjbms.2007.3078. PMID: 17489757. Perrone N, Serafini G, Vitali A, et al. Gastrointestinal stromal tumor metastatic to the scrotum. J Ultrasound Med. 2008 Jun;27(6):961-4. doi: 10.7863/jum.2008.27.6.961. PMID: 18499856. Thomas R, Swamy S. Rare case of gastrointestinal stromal tumor presenting with scrotal metastasis. Indian J Surg. 2015 Apr;77(Suppl 1):188-9. doi: 10.1007/s12262-015-1260-5. PMID: 25972692. Brainwood M, Beirne G, Fenech M. Persistence of the processus vaginalis and its related disorders. Australas J Ultrasound Med. 2020 Feb;23(1):22-29. doi: 10.1002/ajum.12195. PMID: 34760578. Ahmad A, Libbey NP, Somasundar P, et al. Gastrointestinal stromal tumor metastatic to the epididymis. BMJ Case Rep. 2015 Sep;2015:bcr2015211555. doi: 10.1136/bcr-2015-211555. PMID: 26400593. Rota M, Sganzerla F, Zuffante M, et al. Case report: Male genital system, soft tissue, and myocardial metastases in a patient with exon 11-mutated GIST of unknown origin. Front Oncol. 2024 Sep;14:1450889. doi: 10.3389/fonc.2024.1450889. PMID: 39290240. Froehner M, Ockert D, Aust DE, et al. Gastrointestinal stromal tumor presenting as a scrotal mass. Int J Urol. 2004 Jun;11(6):445-7. doi: 10.1111/j.1442-2042.2004.00812.x. PMID: 15157221. Kang SH, Kim MJ, Park MG, et al. Extragastrointestinal stromal tumor presenting as a scrotal mass: an unusual case. Asian J Androl. 2007 Mar;9(2):275-9. doi: 10.1111/j.1745-7262.2007.00235.x. PMID: 17334596. Ali Q, Anand K, Bansal V. Extragastrointestinal stromal tumor presenting as a scrotal mass: First report from India. Int J Surg Case Rep. 2020;72:507-510. doi: 10.1016/j.ijscr.2020.05.079. PMID: 32698276. Additional Declarations No competing interests reported. Cite Share Download PDF Status: Posted Version 1 posted 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. We do this by developing innovative software and high quality services for the global research community. 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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-6058098","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":419566633,"identity":"bb287472-8c38-47da-ae57-2e6d7d4c45e4","order_by":0,"name":"AYARI JIHENE","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAA7ElEQVRIiWNgGAWjYBACxmYeBOfABxtStRyckcYgwcDADGQm4NOEpIWZhxgtzO28Bx8XVDDIGxw/+/CwTcK2OgaJ/KMbGH/cweMwvmTjGWcYDDecSTc4nJNwW4JBIpntBkPCM3x+MZPmbWNg3HAgjeFw7g+4lsP4tJj/5v3HYL/h/DOGwxYJxGkxY+ZtYEjccANoCwORWoyleY5JJM+88YzhYE/Cbck2nsdmNxLScGsx7D9j+Jmnxsa273wa84cfCbf5+dkTn934YINHSwOYkmBQOAAVYQMRCTg1MDDIwxkNeFSNglEwCkbByAYA28dSnEY04NAAAAAASUVORK5CYII=","orcid":"","institution":"Tunis El Manar University","correspondingAuthor":true,"prefix":"","firstName":"AYARI","middleName":"","lastName":"JIHENE","suffix":""},{"id":419566634,"identity":"b3abce6d-1893-4021-a33b-c2993f44c482","order_by":1,"name":"AZZA BOUAZIZ","email":"","orcid":"","institution":"Tunis El Manar University","correspondingAuthor":false,"prefix":"","firstName":"AZZA","middleName":"","lastName":"BOUAZIZ","suffix":""},{"id":419566635,"identity":"455b3096-08c5-44bf-b870-4779a96b2449","order_by":2,"name":"HAYTHEM ELASKRI","email":"","orcid":"","institution":"Tunis El Manar University","correspondingAuthor":false,"prefix":"","firstName":"HAYTHEM","middleName":"","lastName":"ELASKRI","suffix":""},{"id":419566636,"identity":"9ea04a1b-43d3-4c0f-b463-69c86efdc88a","order_by":3,"name":"HADDAOUI ABDERRAZEK","email":"","orcid":"","institution":"Tunis El Manar University","correspondingAuthor":false,"prefix":"","firstName":"HADDAOUI","middleName":"","lastName":"ABDERRAZEK","suffix":""}],"badges":[],"createdAt":"2025-02-18 16:53:23","currentVersionCode":1,"declarations":"","doi":"10.21203/rs.3.rs-6058098/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-6058098/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":78246957,"identity":"ec22372b-bff4-4405-9cfa-a7e2a4bab2ad","added_by":"auto","created_at":"2025-03-11 09:35:46","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":461190,"visible":true,"origin":"","legend":"\u003cp\u003eFigures 1, 2: Testicular GIST metastasis showing spindle cells arranged in intersecting fascicles with paranuclear vacuoles and eosinophilic cytoplasm.\u003c/p\u003e","description":"","filename":"1.png","url":"https://assets-eu.researchsquare.com/files/rs-6058098/v1/63973f03cfd5cb588e0fdc38.png"},{"id":78249022,"identity":"1fd708c3-06ad-49c3-abdf-4344fc30b155","added_by":"auto","created_at":"2025-03-11 09:43:51","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":716759,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-6058098/v1/e8201840-b835-48b4-a900-fe985b262f53.pdf"}],"financialInterests":"No competing interests reported.","formattedTitle":"\u003cp\u003eScrotal Involvement in Gist: a Case Report of Testicular Metastasis and Literature Review\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eGastrointestinal stromal tumors (GISTs) are mesenchymal neoplasms originating from the interstitial cells of Cajal in the digestive tract. Mutations in the KIT, PDGFRA, and occasionally BRAF genes are the primary drivers of oncogenesis (1). The stomach is the most common site of origin, followed by the small intestine. Metastatic GISTs predominantly affect the liver and peritoneum. Scrotal involvement, however, remains exceedingly rare.\u003c/p\u003e"},{"header":"Case Presentation","content":"\u003cp\u003eA 63-year-old male was diagnosed in 2012 with ileal GIST and peritoneal carcinomatosis. He underwent ileal resection and received Imatinib (400 mg daily) until 2018. Upon disease progression in 2020, Imatinib was reintroduced at the same dose, resulting in a complete response. However, the patient was lost to follow-up until 2022, when he presented with abdominal and scrotal swelling.\u003c/p\u003e\n\u003cp\u003eA testicular ultrasound revealed a large mass in the right scrotum, and a CT scan showed extensive intraperitoneal masses and multiple liver nodules. A right orchiectomy was performed. Histological examination revealed a malignant spindle cell mesenchymal tumor with sarcomatous involvement of the testicular parenchyma without direct invasion. Postoperatively, the patient resumed Imatinib (400 mg daily), which was later increased to 800 mg due to disease progression in the liver and peritoneum in July 2023. By January 2024, further progression in the liver led to the initiation of Sunitinib. No additional scrotal metastases were noted.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eScrotal and Testicular Metastases of GIST:\u003cstrong\u003e\u0026nbsp;\u003c/strong\u003eThe first case of testicular metastasis from a GIST was reported by Dorić et al. in 2007 (2). The patient presented with right testicular enlargement seven months after resection of a jejunal GIST. Orchiectomy revealed tumor nodules within the tunica vaginalis, with histology confirming metastatic GIST tissue. The testicular parenchyma was not involved. Similar cases were described by Perrone et al. in 2008 (3) and Thomas et al. in 2015 (4), both proposing metastasis occurred via a persistent peritoneo-vaginalis duct.\u003c/p\u003e\n\u003cp\u003eThe peritoneo-vaginalis duct is an embryonic structure that connects the peritoneal cavity to the inguinal region, aiding in testicular descent. It typically closes after birth; however, its persistence may result in conditions such as indirect inguinal hernia or hydrocele (5). In the context of malignancies originating from the gastrointestinal or genitourinary tract, a patent peritoneo-vaginalis duct may facilitate metastatic spread. This hypothesis is further supported by a case reported by Ahmad et al. in 2015, where metastatic GIST tissue was found in the epididymis (6). Recently, Rota et al. (2024) described an unusual case of GIST metastasizing to the scrotum, penis, and myocardium (7).\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eE-GIST Involving the Scrotum:\u0026nbsp;\u003c/strong\u003eExtra-gastrointestinal stromal tumors (E-GISTs) account for approximately 5% of all GISTs. Primary scrotal E-GISTs are exceptionally rare. In 2004, Froehner et al. (8) reported a case of a scrotal mass extending from the abdomen along the inguinal canal, associated with large abdominal masses. A CT-guided biopsy confirmed GIST histology, but the primary site could not be determined. In 2007, Kang et al. (9) described a scrotal E-GIST with no evidence of an alternative primary site, a finding further corroborated by Ali et al. in 2020 (10). These cases highlight the need for thorough histological evaluation to distinguish E-GISTs from other differential diagnoses, such as germ cell tumors.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eGISTs can metastasize to unusual locations, including the testis and scrotum. Diagnosis is confirmed using immunohistochemical markers such as CD117 and CD34. For localized primary GISTs, surgical resection remains the cornerstone of treatment, followed by adjuvant Imatinib based on recurrence risk. However, metastatic GISTs carry a poor prognosis due to resistance to tyrosine kinase inhibitors (TKIs), rendering the management of rare metastatic sites particularly challenging.\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003eParticipant Consent Statement: I, the undersigned, Pr AYARI JIHENE, hereby confirm that all participants have given their informed consent to participate in this study. This consent was obtained in writing and/or verbally after the participants were informed of the study\u0026apos;s objectives, methods, risks, and potential benefits. The participants were given the opportunity to ask any necessary questions and expressed their willingness to participate with full knowledge of the study\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eNo founding to declare.\u003c/strong\u003e\u003c/p\u003e\n\u003ch2\u003eAuthor Contribution\u003c/h2\u003e\u003cp\u003eALL AUTHORS PREPARED THIS PAPER AND REVIEWED IT\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n \u003cli\u003eMiettinen M, Lasota J. Gastrointestinal stromal tumors: review on morphology, molecular pathology, prognosis, and differential diagnosis. Arch Pathol Lab Med. 2006 Oct;130(10):1466-78. doi: 10.5858/2006-130-1466-GSTROM. PMID: 17090188.\u003c/li\u003e\n \u003cli\u003eDorić M, Radović S, Babić M, et al. Testicular metastasis of gastrointestinal stromal tumor of the jejunum. Bosn J Basic Med Sci. 2007 May;7(2):176-9. doi: 10.17305/bjbms.2007.3078. PMID: 17489757.\u003c/li\u003e\n \u003cli\u003ePerrone N, Serafini G, Vitali A, et al.\u0026nbsp;Gastrointestinal stromal tumor metastatic to the scrotum. J Ultrasound Med. 2008 Jun;27(6):961-4. doi: 10.7863/jum.2008.27.6.961. PMID: 18499856.\u003c/li\u003e\n \u003cli\u003eThomas R, Swamy S. Rare case of gastrointestinal stromal tumor presenting with scrotal metastasis. Indian J Surg. 2015 Apr;77(Suppl 1):188-9. doi: 10.1007/s12262-015-1260-5. PMID: 25972692.\u003c/li\u003e\n \u003cli\u003eBrainwood M, Beirne G, Fenech M. Persistence of the processus vaginalis and its related disorders. Australas J Ultrasound Med. 2020 Feb;23(1):22-29. doi: 10.1002/ajum.12195. PMID: 34760578.\u003c/li\u003e\n \u003cli\u003eAhmad A, Libbey NP, Somasundar P, et al. Gastrointestinal stromal tumor metastatic to the epididymis. BMJ Case Rep. 2015 Sep;2015:bcr2015211555. doi: 10.1136/bcr-2015-211555. PMID: 26400593.\u003c/li\u003e\n \u003cli\u003eRota M, Sganzerla F, Zuffante M, et al.\u0026nbsp;Case report: Male genital system, soft tissue, and myocardial metastases in a patient with exon 11-mutated GIST of unknown origin. Front Oncol. 2024 Sep;14:1450889. doi: 10.3389/fonc.2024.1450889. PMID: 39290240.\u003c/li\u003e\n \u003cli\u003eFroehner M, Ockert D, Aust DE, et al.\u0026nbsp;Gastrointestinal stromal tumor presenting as a scrotal mass. Int J Urol. 2004 Jun;11(6):445-7. doi: 10.1111/j.1442-2042.2004.00812.x. PMID: 15157221.\u003c/li\u003e\n \u003cli\u003eKang SH, Kim MJ, Park MG, et al. Extragastrointestinal stromal tumor presenting as a scrotal mass: an unusual case. Asian J Androl. 2007 Mar;9(2):275-9. doi: 10.1111/j.1745-7262.2007.00235.x. PMID: 17334596.\u003c/li\u003e\n \u003cli\u003eAli Q, Anand K, Bansal V. Extragastrointestinal stromal tumor presenting as a scrotal mass: First report from India. Int J Surg Case Rep. 2020;72:507-510. doi: 10.1016/j.ijscr.2020.05.079. PMID: 32698276.\u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"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":"","lastPublishedDoi":"10.21203/rs.3.rs-6058098/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-6058098/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cstrong\u003eIntroduction:\u0026nbsp;\u003c/strong\u003eGastrointestinal stromal tumors (GISTs) are mesenchymal tumors of the digestive tract originating from interstitial Cajal cells. Mutations in the KIT, PDGFRA, and occasionally BRAF genes are the main drivers of oncogenesis (1). The stomach is the most common primary location, followed by the small intestine. Metastatic GISTs most frequently involve the liver and peritoneum. However, scrotal involvement is exceedingly rare. \u0026nbsp;\u003c/p\u003e\n\u003cp\u003e\u003cstrong\u003eCase Presentation:\u003c/strong\u003e\u0026nbsp;A 63-year-old man was diagnosed in 2012 with ileal GIST and peritoneal carcinomatosis. He underwent ileal resection and received Imatinib (400 mg daily) until 2018. Following disease progression in 2020, Imatinib was reintroduced at the same dose, resulting in a complete response. The patient was subsequently lost to follow-up until 2022, when he presented with abdominal and scrotal swelling.\u0026nbsp;A testicular ultrasound revealed a large mass in the right scrotum, and a CT scan showed extensive intraperitoneal masses and multiple liver nodules. A right orchiectomy was performed. Histological examination identified a malignant spindle cell mesenchymal tumor with sarcomatous involvement of the testicular parenchyma without direct invasion. Postoperatively, the patient resumed Imatinib (400 mg daily), later increased to 800 mg due to disease progression in the liver and peritoneal masses in July 2023. By January 2024, further liver progression led to the initiation of Sunitinib. 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