Atypical location of a lymphangioma in the thymic cavity in an adult: case report and review of the literature

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Abstract Introduction: Lymphangiomas are rare primary tumours of the anterior mediastinum in adults. They are usually asymptomatic. In other cases, there may be a cervical mass or signs of mediastinal compression such as exertional dyspnoea or dysphagia. The diagnosis is made with certainty by histology. Surgery is the main curative treatment. Case report: We report the case of a 34-year-old female patient who presented with a painless anterior cervico-mediastinal mass associated with dyspnoea. Cervico-thoracic imaging revealed a non-compressive mass in the thymic cavity, suggestive of a thymoma but not excluding a lymphoma. Biology and histology after scannoguided biopsy were negative. Further histological examination following surgical biopsy revealed the vascular nature of the mass, consistent with lymphangioma. Conclusion: Lymphangiomas are rarely seen in adults, particularly in the anterior mediastinum. Our observation of a lymphangioma found in the thymic cavity adds to the other cases found in the literature. This could lead us to include it in the nosological panel of an anterior cervico-mediastinal mass found in adults. Furthermore, as its diagnosis is essentially histological, we could also consider surgery as a means of diagnosis in addition to the therapeutic aspect, especially in cases of diagnostic doubt.
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Atypical location of a lymphangioma in the thymic cavity in an adult: case report and review of the literature | 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 Atypical location of a lymphangioma in the thymic cavity in an adult: case report and review of the literature Dr Joël Cedric Bayem This is a preprint; it has not been peer reviewed by a journal. https://doi.org/ 10.21203/rs.3.rs-6746821/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 : Lymphangiomas are rare primary tumours of the anterior mediastinum in adults. They are usually asymptomatic. In other cases, there may be a cervical mass or signs of mediastinal compression such as exertional dyspnoea or dysphagia. The diagnosis is made with certainty by histology. Surgery is the main curative treatment. Case report : We report the case of a 34-year-old female patient who presented with a painless anterior cervico-mediastinal mass associated with dyspnoea. Cervico-thoracic imaging revealed a non-compressive mass in the thymic cavity, suggestive of a thymoma but not excluding a lymphoma. Biology and histology after scannoguided biopsy were negative. Further histological examination following surgical biopsy revealed the vascular nature of the mass, consistent with lymphangioma. Conclusion : Lymphangiomas are rarely seen in adults, particularly in the anterior mediastinum. Our observation of a lymphangioma found in the thymic cavity adds to the other cases found in the literature. This could lead us to include it in the nosological panel of an anterior cervico-mediastinal mass found in adults. Furthermore, as its diagnosis is essentially histological, we could also consider surgery as a means of diagnosis in addition to the therapeutic aspect, especially in cases of diagnostic doubt. Cardiothoracic Surgery Lymphangioma Adult Mediastinum Thymic cavity Figures Figure 1 Figure 2 Introduction Lymphangiomas are rare primary tumours of the mediastinum resulting from a fusion anomaly of the embryonic lymphatic network and the venous system [1]. They account for less than 1% of primary mediastinal tumours [1]. They are more frequent and symptomatic in children, and occur in the cervical, cervicothoracic or anterior mediastinal regions. Their discovery in adults is usually fortuitous because they are asymptomatic and usually occur in the middle mediastinum [1]. The curative treatment is surgical removal [2,4]. We report a case of lymphangioma found in the thymic cavity in a patient operated on in the thoracic surgery department of the ARRAZI Hospital at the Mohammed VI University Hospital, Marrakech, and review the literature. Observation This is Patient O.F aged 34 admitted to the thoracic surgery department for management of an anterior cervical mass, slowly evolving over 02 months. The history revealed a patient with dyspnoea on exertion. Clinically, she presented with a painful anterior cervical mass, adherent to the deep plane, not indurated, not plunging on swallowing, not pulsatile, with no local inflammatory signs or collateral circulation, limiting neck movements and whose lower pole could not be palpated. Cervicothoracic scans showed a tissue mass measuring 4.6*1.7*6cm, located in the superior-anterior mediastinum, in the pre-vascular region, well limited, spontaneously hypodense, homogeneously enhancing after injection of PDC. It was connected anteriorly with the sternal manubrium without bone lysis and posteriorly with the anterior wall of the ascending aorta and the right and left brachiocephalic veins without signs of compression or invasion. The thyroid gland was normal in size and structure. (Figure 1) The hypothesised diagnosis was thymoma, without ruling out a lymphomatous origin. Biological tests for aetiology were negative. We performed 02 CT biopsies, which were non-contributory. Given this diagnostic impasse, we opted for surgical biopsy and excision. Exploration by sternotomy revealed a mass in the thymic cavity, well encapsulated with no locoregional invasion. Surgery consisted of a monobloc excision. Histological examination of the mass showed a fibrofatty tissue with numerous ectatic vascular elements of lymphatic appearance and cavernous architecture, bordered by a single layer of regular endothelial cells and containing amorphous eosinophilic material. (Figure 2) The connective tissue between these structures consisted of regular fibroblasts punctuated by lymphoplasmacytic elements. Reactive lymph nodes and thymic parenchyma of subnormal morphology were also present. No malignant tumour proliferation was visible. The morphological appearance was that of a lymphangioma with reactive lymphadenitis without signs of malignancy. Discussion Lymphangiomas are rare conditions that develop in the lymphatic vascular system and are considered to be malformative or hamartomatous rather than tumourous[5]. These malformations are most often congenital in origin and are found in around 90% of cases in children under the age of 02[6]. Lymphangiomas are found throughout the body with the exception of the brain, with 75% occurring in the cervical region, 15% in the axilla and 10% in the cervicomediastinal, retroperitoneal, splenic or colonic regions[7]. In adults, they are located in the middle mediastinum, in the peri-tracheobronchial lymph node chains, and their acquired origin cannot be ruled out [1,2]. The anterior mediastinal location is thought to result from the sequestrated lymphatic tissue evolving on its own. The reported case of thymic localisation is consistent with the hypothesis that the lymphatic elements initially sequestered in the cervical region migrated into the mediastinum following migratory organs such as the thymus, heart or pericardium [7,8]. Lymphangiomas of the mediastinum in adults are rarely symptomatic. Their clinical expression is often due to compressive phenomena such as cough, dyspnoea and chest pain, or complications such as intra-cystic haemorrhage or intra-pleural rupture [11]. In their series of 37 cases, Riquet et al. reported 59% of patients asymptomatic, 30% with revealing symptoms such as a palpable mass, dysphagia, exertional dyspnoea, chylothorax and 11% with signs of borrowing [2]. In 50% of cases, however, the condition is discovered by chance during a chest X-ray [7]. This reveals a non-specific opacity in the anterior, middle or posterior mediastinum. A CT scan of the chest usually reveals a homogeneous, multilocular, hypodense, well-limited mass in the anterior superior mediastinum, involving neighbouring structures without any signs of compression. Imaging is particularly useful for determining the exact topography of the lymphangioma and for analysing the precise extent of the lymphangioma in the mediastinum, enabling total removal to be considered [9,10]. The presence of a cervico-mediastinal mass usually points to conditions such as a plunging goitre, thymic tumour, germ cell tumour or lymphoma. In the face of this multitude of hypotheses, biology plays an important role in orientation. This includes measuring TSH, looking for anti-acetylcholine receptor antibodies, measuring alpha-fetoprotein, betaHCG and serum LDH[1]. However, in our case they were all negative, making the diagnosis even more difficult. Lymphangiomas of the thymic cavity are rare and their diagnosis is mainly confirmed histologically[1,4,7]. Histologically, they mimic the structure of lymphatic vessels, the wall of which is based on collagen fibres with a few smooth muscle fibres, and the lumen of the vessels may contain lymphocytes. They are classified into three categories according to the size of the lymphatic vessels that make them up. There are capillary lymphangiomas, cavernous lymphangiomas and cystic lymphangiomas [3,4]. The cavernous type is made up of dilated lymphatic ducts with supporting tissue that insinuates itself into adjacent structures[7], as found in our case. Surgery remains the gold standard and definitive treatment for mediastinal lymphangioma[14]. It is particularly necessary when the lymphangioma is symptomatic. The classic approach is thoracotomy[1], although we have opted for sternotomy, which gives a better view because of the thymic location and ratios of the mass. Other therapeutic alternatives have been described, such as systemic treatment with a mechanistic target of rapamycin (mTOR) inhibitor [3], puncture-aspiration followed by an injection of picibanil (OK-432) [13] or an injection of fibrin-based glue [12]. An alcohol-based solution can also be used as a sclerosing agent. Conclusion Lymphangiomas are rarely seen in adults, particularly in the anterior mediastinum. Our observation of a lymphangioma found in the thymic cavity adds to the other cases found in the literature. This could lead us to include it in the nosological panel of an anterior cervico-mediastinal mass found in adults. Furthermore, as its diagnosis is essentially histological, we could also consider surgery as a means of diagnosis in addition to the therapeutic aspect, especially in cases of diagnostic doubt. Declarations The authors confirm that they have consent for publication from the patient. 1- Ethics approval and consent to participate : Not applicable 2- Consent for publication : The authors declare that they have consent for publication 3- Availability of data and materials : Not applicable 4- Competing interests : The authors declare that they have no competing interests 5- Fundings :All authors declare that they have no financial relationships at present or within the previous three yers with any organizations that might have an interest in the submitted work. 6- Author contributions : H. Rais performed the histological examination of the Thymus , and was a major contributor in writing the manuscript. All authors read and approved the final manuscript 7- Acknowledgments : Not applicable References Rivera C, Belaroussi Y, Mazères F, Le Pimpec Barthes F. Traitement chirurgical des tumeurs du médiastin. EMC - Techniques chirurgicales - Thorax 2020;37(4):1-18 [Article 42-180]. Riquet M, Briere J, Le Pimpec-Barthes Fet al. Cystic lymphangioma of the neck and mediastinum: are there acquired forms? Report of 37 cases. Rev Mal Respir. 1999;16(1):71–9. Nasser M, Ahmad K, Cottin V. Mediastinal lymphangioma in an adult. The Journal of Thoracic and Cardiovascular Surgery . 2018 ;155(6):e195–7. Le Pimpec-Barthes F, Cazes A, Bagan P, et al. Les kystes du médiastin : approche diagnostique et traitement. Rev Pneum Clin. 2010 ;66(1):52–62. Academie Nationale de Medecine. Definition du Lymphangiome. Dictionnaire médical de l’Académie de Médecine 2020. Visite le 07 Jan 2025 a 19h10. Disponible : https://www.academie-medecine.fr/le-dictionnaire/index.php?q=lymphangiome+ , Khabbaza J, Sethi S, Raymond D, et al. Mediastinal Cyst Mimicking Malignancy in a Pipe Smoker. Chest 2013 ;144(4):21A. Rakotosamimanana J1, Raharisolo Vololonantenaina CR2, Ratovoson H1, et al.Lymphangiome kystique cervico-médiastinal : à propos d’un cas et revue de la littérature. Arch Inst Pasteur Madagascar 2000; 66 (1&2) : 61-64 Childress Me, Baker Cp, Samson Pc. Lymphangioma of the mediastinum; report of a case with review of the literature. J Thorac Surg. 1956;31(3):338–48. Adil A, Ksiyer M. Unusual mediastinal cystic lymphangioma. Apropos of a case and review of the literature. Ann Radiol. 1996;39(6):249–52. Dia, Aliou Amadou et al. Apport de la tomodensitométrie dans le diagnostic du lymphangiome kystique cervical. A propos d’un cas. Médecine d'Afrique Noire 2013 ; 6006: 294-296. Swarnakar RN, Hazarey JD, Dhoble C, et al. A 36-Year-Old Female with Recurrent Left Sided Pleural Effusion: A Rare Case of Mediastinal Lymphangioma. Am J Case Rep. 2016;17:799–804. Zhou S, Dong S, Du J. Percutaneous therapy of a mediastinal lymphangioma with fibrin glue: case report with clinical success after 4 years. BMC Surg. 2018 ;18(1):4. Golinelli G, Toso A, Borello G, et al. Percutaneous Sclerotherapy With OK-432 of a Cervicomediastinal Lymphangioma. Ann Thorac Surg. 2015;100(5):1879–81. Fokkema J, Paul M, Vrouenraets B. Mediastinal lymphangioma in an adult. Ann R Coll Surg Engl. 2014;96(5):e24–5. Additional Declarations The authors declare no competing interests. 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. Our growing team is made up of researchers and industry professionals working together to solve the most critical problems facing scientific publishing. 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-6746821","acceptedTermsAndConditions":true,"allowDirectSubmit":true,"archivedVersions":[],"articleType":"Case Report","associatedPublications":[],"authors":[{"id":461695688,"identity":"6e185014-ce6b-4137-ba77-c37630082f2d","order_by":0,"name":"Dr Joël Cedric Bayem","email":"data:image/png;base64,iVBORw0KGgoAAAANSUhEUgAAAZAAAAAyAQMAAABI0h/eAAAABlBMVEX///8AAABVwtN+AAAACXBIWXMAAA7EAAAOxAGVKw4bAAAAwElEQVRIiWNgGAWjYBACAzDJY8PAIAFi2BCvJQ2qJY1oLQyHSdBizn7G8NENmfOJ/bObDz5gSLhHWItlT46xcQ7P7cQZd44lGzAkFBPhsAM5ZtIgLQ03cswkGH8kEKHl/Bvz3zk85xLng7QwJBCjBaiSOYfnQOIGorVYznhWDHRYsvHGG2nJBgnEaDHnT974ObfHTnbejeSDDz4Qo4WBgcOAgbGHwbEBxCZKAwMD+wMGhh8M9sQpHgWjYBSMghEJAMV9O6MCDXygAAAAAElFTkSuQmCC","orcid":"https://orcid.org/0009-0003-4846-9705","institution":"Centre Hospitalier Universitaire Mohammed VI Marrakech","correspondingAuthor":true,"prefix":"Dr","firstName":"Joël","middleName":"Cedric","lastName":"Bayem","suffix":""}],"badges":[],"createdAt":"2025-05-26 04:04:22","currentVersionCode":1,"declarations":{"humanSubjects":true,"vertebrateSubjects":true,"conflictsOfInterestStatement":false,"humanSubjectEthicalGuidelines":true,"humanSubjectConsent":true,"humanSubjectClinicalTrial":false,"humanSubjectCaseReport":true,"vertebrateSubjectEthicalGuidelines":true},"doi":"10.21203/rs.3.rs-6746821/v1","doiUrl":"https://doi.org/10.21203/rs.3.rs-6746821/v1","draftVersion":[],"editorialEvents":[],"editorialNote":"","failedWorkflow":false,"files":[{"id":83836260,"identity":"260901e9-aab1-4ab9-acfe-4231b339ed8e","added_by":"auto","created_at":"2025-06-03 13:15:43","extension":"png","order_by":1,"title":"Figure 1","display":"","copyAsset":false,"role":"figure","size":5713,"visible":true,"origin":"","legend":"\u003cp\u003eThe thyroid gland was normal in size and structure.\u003c/p\u003e","description":"","filename":"placeholderimageCopy.png","url":"https://assets-eu.researchsquare.com/files/rs-6746821/v1/bf3180e5baa9075c650d70ae.png"},{"id":83836263,"identity":"7acf6702-6720-4975-8b3a-48948d27e46c","added_by":"auto","created_at":"2025-06-03 13:15:43","extension":"png","order_by":2,"title":"Figure 2","display":"","copyAsset":false,"role":"figure","size":5713,"visible":true,"origin":"","legend":"\u003cp\u003eHistological examination of the mass.\u003c/p\u003e","description":"","filename":"placeholderimage.png","url":"https://assets-eu.researchsquare.com/files/rs-6746821/v1/206eea408e32cb9dff582991.png"},{"id":83837470,"identity":"cb2e3a0e-cde6-48a0-803c-291b861be7f3","added_by":"auto","created_at":"2025-06-03 13:24:01","extension":"pdf","order_by":0,"title":"","display":"","copyAsset":false,"role":"manuscript-pdf","size":320322,"visible":true,"origin":"","legend":"","description":"","filename":"manuscript.pdf","url":"https://assets-eu.researchsquare.com/files/rs-6746821/v1/cdea9568-0271-4768-8918-4c9e194b54ce.pdf"}],"financialInterests":"The authors declare no competing interests.","formattedTitle":"\u003cp\u003e\u003cstrong\u003eAtypical location of a lymphangioma in the thymic cavity in an adult: case report and review of the literature\u003c/strong\u003e\u003c/p\u003e","fulltext":[{"header":"Introduction","content":"\u003cp\u003eLymphangiomas are rare primary tumours of the mediastinum resulting from a fusion anomaly of the embryonic lymphatic network and the venous system [1]. They account for less than 1% of primary mediastinal tumours [1].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThey are more frequent and symptomatic in children, and occur in the cervical, cervicothoracic or anterior mediastinal regions. Their discovery in adults is usually fortuitous because they are asymptomatic and usually occur in the middle mediastinum [1].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe curative treatment is surgical removal [2,4]. We report a case of lymphangioma found in the thymic cavity in a patient operated on in the thoracic surgery department of the ARRAZI Hospital at the Mohammed VI University Hospital, Marrakech, and review the literature.\u0026nbsp;\u003c/p\u003e"},{"header":"Observation","content":"\u003cp\u003eThis is Patient O.F aged 34 admitted to the thoracic surgery department for management of an anterior cervical mass, slowly evolving over 02 months.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe history revealed a patient with dyspnoea on exertion. Clinically, she presented with a painful anterior cervical mass, adherent to the deep plane, not indurated, not plunging on swallowing, not pulsatile, with no local inflammatory signs or collateral circulation, limiting neck movements and whose lower pole could not be palpated.\u003c/p\u003e\n\u003cp\u003eCervicothoracic scans showed a tissue mass measuring 4.6*1.7*6cm, located in the superior-anterior mediastinum, in the pre-vascular region, well limited, spontaneously hypodense, homogeneously enhancing after injection of PDC. It was connected anteriorly with the sternal manubrium without bone lysis and posteriorly with the anterior wall of the ascending aorta and the right and left brachiocephalic veins without signs of compression or invasion. The thyroid gland was normal in size and structure. (Figure 1)\u003c/p\u003e\n\u003cp\u003eThe hypothesised diagnosis was thymoma, without ruling out a lymphomatous origin.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eBiological tests for aetiology were negative. We performed 02 CT biopsies, which were non-contributory.\u003c/p\u003e\n\u003cp\u003eGiven this diagnostic impasse, we opted for surgical biopsy and excision.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eExploration by sternotomy revealed a mass in the thymic cavity, well encapsulated with no locoregional invasion. Surgery consisted of a monobloc excision.\u003c/p\u003e\n\u003cp\u003eHistological examination of the mass showed a fibrofatty tissue with numerous ectatic vascular elements of lymphatic appearance and cavernous architecture, bordered by a single layer of regular endothelial cells and containing amorphous eosinophilic material. (Figure 2)\u003c/p\u003e\n\u003cp\u003eThe connective tissue between these structures consisted of regular fibroblasts punctuated by lymphoplasmacytic elements. Reactive lymph nodes and thymic parenchyma of subnormal morphology were also present. No malignant tumour proliferation was visible. The morphological appearance was that of a lymphangioma with reactive lymphadenitis without signs of malignancy.\u003c/p\u003e"},{"header":"Discussion","content":"\u003cp\u003eLymphangiomas are rare conditions that develop in the lymphatic vascular system and are considered to be malformative or hamartomatous rather than tumourous[5].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThese malformations are most often congenital in origin and are found in around 90% of cases in children under the age of 02[6]. Lymphangiomas are found throughout the body with the exception of the brain, with 75% occurring in the cervical region, 15% in the axilla and 10% in the cervicomediastinal, retroperitoneal, splenic or colonic regions[7].\u003c/p\u003e\n\u003cp\u003eIn adults, they are located in the middle mediastinum, in the peri-tracheobronchial lymph node chains, and their acquired origin cannot be ruled out [1,2]. The anterior mediastinal location is thought to result from the sequestrated lymphatic tissue evolving on its own.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe reported case of thymic localisation is consistent with the hypothesis that the lymphatic elements initially sequestered in the cervical region migrated into the mediastinum following migratory organs such as the thymus, heart or pericardium [7,8].\u003c/p\u003e\n\u003cp\u003eLymphangiomas of the mediastinum in adults are rarely symptomatic. Their clinical expression is often due to compressive phenomena such as cough, dyspnoea and chest pain, or complications such as intra-cystic haemorrhage or intra-pleural rupture [11].\u003c/p\u003e\n\u003cp\u003eIn their series of \u0026nbsp;37 cases, Riquet et al. reported 59% of patients asymptomatic, 30% with revealing symptoms such as a palpable mass, dysphagia, exertional dyspnoea, chylothorax and 11% with signs of borrowing [2]. \u0026nbsp;In 50% of cases, however, the condition is discovered by chance during a chest X-ray [7]. This reveals a non-specific opacity in the anterior, middle or posterior mediastinum.\u003c/p\u003e\n\u003cp\u003e\u0026nbsp;A CT scan of the chest usually reveals a homogeneous, multilocular, hypodense, well-limited mass in the anterior superior mediastinum, involving neighbouring structures without any signs of compression. Imaging is particularly useful for determining the exact topography of the lymphangioma and for analysing the precise extent of the lymphangioma in the mediastinum, enabling total removal to be considered [9,10].\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eThe presence of a cervico-mediastinal mass usually points to conditions such as a plunging goitre, thymic tumour, germ cell tumour or lymphoma. In the face of this multitude of hypotheses, biology plays an important role in orientation. This includes measuring TSH, looking for anti-acetylcholine receptor antibodies, measuring alpha-fetoprotein, betaHCG and serum LDH[1]. However, in our case they were all negative, making the diagnosis even more difficult.\u0026nbsp;\u003c/p\u003e\n\u003cp\u003eLymphangiomas of the thymic cavity are rare and their diagnosis is mainly confirmed histologically[1,4,7]. Histologically, they mimic the structure of lymphatic vessels, the wall of which is based on collagen fibres with a few smooth muscle fibres, and the lumen of the vessels may contain lymphocytes. They are classified into three categories according to the size of the lymphatic vessels that make them up. There are capillary lymphangiomas, cavernous lymphangiomas and cystic lymphangiomas [3,4]. The cavernous type is made up of dilated lymphatic ducts with supporting tissue that insinuates itself into adjacent structures[7], as found in our case.\u003c/p\u003e\n\u003cp\u003eSurgery remains the gold standard and definitive treatment for mediastinal lymphangioma[14]. \u0026nbsp;It is particularly necessary when the lymphangioma is symptomatic. The classic approach is thoracotomy[1], although we have opted for sternotomy, which gives a better view because of the thymic location and ratios of the mass.\u003c/p\u003e\n\u003cp\u003eOther therapeutic alternatives have been described, such as systemic treatment with a mechanistic target of rapamycin (mTOR) inhibitor [3], puncture-aspiration followed by an injection of picibanil (OK-432) [13] or an injection of fibrin-based glue [12]. An alcohol-based solution can also be used as a sclerosing agent.\u003c/p\u003e"},{"header":"Conclusion","content":"\u003cp\u003eLymphangiomas are rarely seen in adults, particularly in the anterior mediastinum. Our observation of a lymphangioma found in the thymic cavity adds to the other cases found in the literature. This could lead us to include it in the nosological panel of an anterior cervico-mediastinal mass found in adults. Furthermore, as its diagnosis is essentially histological, we could also consider surgery as a means of diagnosis in addition to the therapeutic aspect, especially in cases of diagnostic doubt.\u0026nbsp;\u003c/p\u003e"},{"header":"Declarations","content":"\u003cp\u003e\u003cspan\u003eThe authors confirm that they have consent for publication from the patient.\u003c/span\u003e\u003c/p\u003e\u003cp\u003e1- \u003cstrong\u003eEthics approval and consent to participate\u003c/strong\u003e : Not applicable\u003c/p\u003e\n\u003cp\u003e2- \u003cstrong\u003eConsent for publication\u0026nbsp;\u003c/strong\u003e: The authors declare that they have consent for publication\u003c/p\u003e\n\u003cp\u003e3- \u003cstrong\u003eAvailability of data and materials\u0026nbsp;\u003c/strong\u003e: Not applicable\u003c/p\u003e\n\u003cp\u003e4- \u003cstrong\u003eCompeting interests\u0026nbsp;\u003c/strong\u003e: The authors declare that they have no competing interests\u003c/p\u003e\n\u003cp\u003e5- \u003cstrong\u003eFundings\u0026nbsp;\u003c/strong\u003e:All authors declare that they have no financial relationships at present or within the previous three yers with any organizations that might have an interest in the submitted work.\u003c/p\u003e\n\u003cp\u003e6- \u003cstrong\u003eAuthor contributions\u0026nbsp;\u003c/strong\u003e: H. Rais performed the histological examination of the Thymus , and was a major contributor in writing the manuscript. All authors read and approved the final manuscript\u0026nbsp;\u003c/p\u003e\n\u003cp\u003e7- \u003cstrong\u003eAcknowledgments\u003c/strong\u003e : Not applicable\u003c/p\u003e"},{"header":"References","content":"\u003col\u003e\n\u003cli\u003eRivera C, Belaroussi Y, Mazères F, Le Pimpec Barthes F. Traitement chirurgical des tumeurs du médiastin. EMC - Techniques chirurgicales - Thorax 2020;37(4):1-18 [Article 42-180].\u003c/li\u003e\n\u003cli\u003eRiquet M, Briere J, Le Pimpec-Barthes Fet al. Cystic lymphangioma of the neck and mediastinum: are there acquired forms? Report of 37 cases. Rev Mal Respir. 1999;16(1):71\u0026ndash;9.\u003c/li\u003e\n\u003cli\u003eNasser M, Ahmad K, Cottin V. Mediastinal lymphangioma in an adult. The Journal of Thoracic and Cardiovascular Surgery . 2018 ;155(6):e195\u0026ndash;7. \u003c/li\u003e\n\u003cli\u003eLe Pimpec-Barthes F, Cazes A, Bagan P, et al. Les kystes du m\u0026eacute;diastin : approche diagnostique et traitement. Rev Pneum Clin. 2010 ;66(1):52\u0026ndash;62.\u003c/li\u003e\n\u003cli\u003eAcademie Nationale de Medecine. Definition du Lymphangiome. Dictionnaire m\u0026eacute;dical de l\u0026rsquo;Acad\u0026eacute;mie de M\u0026eacute;decine 2020. Visite le 07 Jan 2025 a 19h10. Disponible : https://www.academie-medecine.fr/le-dictionnaire/index.php?q=lymphangiome+ , \u003c/li\u003e\n\u003cli\u003eKhabbaza J, Sethi S, Raymond D, et al. Mediastinal Cyst Mimicking Malignancy in a Pipe Smoker. Chest 2013 ;144(4):21A.\u003c/li\u003e\n\u003cli\u003eRakotosamimanana J1, Raharisolo Vololonantenaina CR2, Ratovoson H1, et al.Lymphangiome kystique cervico-médiastinal : à propos d\u0026rsquo;un cas et revue de la littérature. Arch Inst Pasteur Madagascar 2000; 66 (1\u0026amp;2) : 61-64\u003c/li\u003e\n\u003cli\u003eChildress Me, Baker Cp, Samson Pc. Lymphangioma of the mediastinum; report of a case with review of the literature. J Thorac Surg. 1956;31(3):338\u0026ndash;48.\u003c/li\u003e\n\u003cli\u003eAdil A, Ksiyer M. Unusual mediastinal cystic lymphangioma. Apropos of a case and review of the literature. Ann Radiol. 1996;39(6):249\u0026ndash;52.\u003c/li\u003e\n\u003cli\u003eDia, Aliou Amadou et al. Apport de la tomodensitom\u0026eacute;trie dans le diagnostic du lymphangiome kystique cervical. A propos d\u0026rsquo;un cas. M\u0026eacute;decine d\u0026apos;Afrique Noire 2013 ; 6006: 294-296.\u003c/li\u003e\n\u003cli\u003eSwarnakar RN, Hazarey JD, Dhoble C, et al. A 36-Year-Old Female with Recurrent Left Sided Pleural Effusion: A Rare Case of Mediastinal Lymphangioma. Am J Case Rep. 2016;17:799\u0026ndash;804.\u003c/li\u003e\n\u003cli\u003eZhou S, Dong S, Du J. Percutaneous therapy of a mediastinal lymphangioma with fibrin glue: case report with clinical success after 4 years. BMC Surg. 2018 ;18(1):4.\u003c/li\u003e\n\u003cli\u003eGolinelli G, Toso A, Borello G, et al. Percutaneous Sclerotherapy With OK-432 of a Cervicomediastinal Lymphangioma. Ann Thorac Surg. 2015;100(5):1879\u0026ndash;81.\u003c/li\u003e\n\u003cli\u003eFokkema J, Paul M, Vrouenraets B. Mediastinal lymphangioma in an adult. Ann R Coll Surg Engl. 2014;96(5):e24\u0026ndash;5. \u003c/li\u003e\n\u003c/ol\u003e"}],"fulltextSource":"","fullText":"","funders":[],"hasAdminPriorityOnWorkflow":false,"hasManuscriptDocX":true,"hasOptedInToPreprint":true,"hasPassedJournalQc":"","hasAnyPriority":true,"hideJournal":true,"highlight":"","institution":"Centre Hospitalier Universitaire Mohammed VI","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":"Lymphangioma, Adult, Mediastinum, Thymic cavity","lastPublishedDoi":"10.21203/rs.3.rs-6746821/v1","lastPublishedDoiUrl":"https://doi.org/10.21203/rs.3.rs-6746821/v1","license":{"name":"CC BY 4.0","url":"https://creativecommons.org/licenses/by/4.0/"},"manuscriptAbstract":"\u003cp\u003e\u003cu\u003e\u003cstrong\u003eIntroduction\u003c/strong\u003e\u003c/u\u003e: Lymphangiomas are rare primary tumours of the anterior mediastinum in adults. They are usually asymptomatic. In other cases, there may be a cervical mass or signs of mediastinal compression such as exertional dyspnoea or dysphagia. The diagnosis is made with certainty by histology. Surgery is the main curative treatment.\u003c/p\u003e\n\u003cp\u003e\u003cu\u003e\u003cstrong\u003eCase report\u003c/strong\u003e\u003c/u\u003e: We report the case of a 34-year-old female patient who presented with a painless anterior cervico-mediastinal mass associated with dyspnoea. Cervico-thoracic imaging revealed a non-compressive mass in the thymic cavity, suggestive of a thymoma but not excluding a lymphoma. Biology and histology after scannoguided biopsy were negative. Further histological examination following surgical biopsy revealed the vascular nature of the mass, consistent with lymphangioma.\u003c/p\u003e\n\u003cp\u003e\u003cu\u003e\u003cstrong\u003eConclusion\u003c/strong\u003e\u003c/u\u003e: Lymphangiomas are rarely seen in adults, particularly in the anterior mediastinum. Our observation of a lymphangioma found in the thymic cavity adds to the other cases found in the literature. This could lead us to include it in the nosological panel of an anterior cervico-mediastinal mass found in adults. Furthermore, as its diagnosis is essentially histological, we could also consider surgery as a means of diagnosis in addition to the therapeutic aspect, especially in cases of diagnostic doubt.\u003c/p\u003e","manuscriptTitle":"Atypical location of a lymphangioma in the thymic cavity in an adult: case report and review of the literature","msid":"","msnumber":"","nonDraftVersions":[{"code":1,"date":"2025-06-03 13:15:38","doi":"10.21203/rs.3.rs-6746821/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":"18b38bf3-3d5e-4fcc-aa12-c36556393398","owner":[],"postedDate":"June 3rd, 2025","published":true,"recentEditorialEvents":[],"rejectedJournal":[],"revision":"","amendment":"","status":"posted","subjectAreas":[{"id":49027429,"name":"Cardiothoracic Surgery"}],"tags":[],"updatedAt":"2025-06-03T13:15:38+00:00","versionOfRecord":[],"versionCreatedAt":"2025-06-03 13:15:38","video":"","vorDoi":"","vorDoiUrl":"","workflowStages":[]},"version":"v1","identity":"rs-6746821","journalConfig":"researchsquare"},"__N_SSP":true},"page":"/article/[identity]/[[...version]]","query":{"redirect":"/article/rs-6746821","identity":"rs-6746821","version":["v1"]},"buildId":"8U1c8b4HqxoKbykW_rLl7","isFallback":false,"isExperimentalCompile":false,"dynamicIds":[84888],"gssp":true,"scriptLoader":[]}

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