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Large Pulmonary Artery Aneurysm Rupture in Hughes-Stovin Syndrome

2006; Lippincott Williams & Wilkins; Volume: 114; Issue: 10 Linguagem: Inglês

10.1161/circulationaha.106.614636

1524-4539

Antoine Khalil, Antoine Parrot, Muriel Fartoukh, C. Marsault, Marie‐France Carette,

Histiocytic Disorders and Treatments

HomeCirculationVol. 114, No. 10Large Pulmonary Artery Aneurysm Rupture in Hughes-Stovin Syndrome Free AccessReview ArticlePDF/EPUBAboutView PDFView EPUBSections ToolsAdd to favoritesDownload citationsTrack citationsPermissions ShareShare onFacebookTwitterLinked InMendeleyReddit Jump toFree AccessReview ArticlePDF/EPUBLarge Pulmonary Artery Aneurysm Rupture in Hughes-Stovin SyndromeMultidetector Computed Tomography Pattern and Endovascular Treatment Antoine Khalil, Antoine Parrot, Muriel Fartoukh, Claude Marsault and Marie-France Carette Antoine KhalilAntoine Khalil From the Radiology Department (A.K., C.M., M.-F.C.) and Pneumology and Respiratory Intensive Care Unit (A.P., M.F.), AP-HP, Tenon Hospital, and Pierre et Marie Curie University (C.M., M.-F.C.), Paris, France. , Antoine ParrotAntoine Parrot From the Radiology Department (A.K., C.M., M.-F.C.) and Pneumology and Respiratory Intensive Care Unit (A.P., M.F.), AP-HP, Tenon Hospital, and Pierre et Marie Curie University (C.M., M.-F.C.), Paris, France. , Muriel FartoukhMuriel Fartoukh From the Radiology Department (A.K., C.M., M.-F.C.) and Pneumology and Respiratory Intensive Care Unit (A.P., M.F.), AP-HP, Tenon Hospital, and Pierre et Marie Curie University (C.M., M.-F.C.), Paris, France. , Claude MarsaultClaude Marsault From the Radiology Department (A.K., C.M., M.-F.C.) and Pneumology and Respiratory Intensive Care Unit (A.P., M.F.), AP-HP, Tenon Hospital, and Pierre et Marie Curie University (C.M., M.-F.C.), Paris, France. and Marie-France CaretteMarie-France Carette From the Radiology Department (A.K., C.M., M.-F.C.) and Pneumology and Respiratory Intensive Care Unit (A.P., M.F.), AP-HP, Tenon Hospital, and Pierre et Marie Curie University (C.M., M.-F.C.), Paris, France. Originally published5 Sep 2006https://doi.org/10.1161/CIRCULATIONAHA.106.614636Circulation. 2006;114:e380–e381A 42-year-old man with a history of recurrent pulmonary embolism was admitted to the respiratory intensive care unit of our hospital for massive hemoptysis (600 mL in one time). The multidetector computed tomography angiography (MDCTA) showed a large aneurysm originating from the middle lobe pulmonary artery with an air bubble in the aneurysm sac (Figure, A) and enlargement of systemic bronchial and nonbronchial arteries (Figure, B). The MDCTA showed 5 aneurysms of the pulmonary arteries at the right lower lobe (n=2), left upper lobe (n=1), and left lower lobe (n=2). Percutaneous vaso-occlusion of the aneurysm in the pulmonary artery of the middle lobe was performed (Figure, C). Cardiac MDCT indicated a thrombus in the right atrium (Figure, D) and an anomalous origin of the right coronary artery. Hughes-Stovin syndrome (HSS) was diagnosed on the basis of the association of multiple pulmonary arteries aneurysms, pulmonary embolism, and thrombus into the right atrium. Immunosuppressive therapy was started. The clinical condition of the patient improved at the 4-month follow-up. Download figureDownload PowerPointA, Axial thin-slab maximum-intensity projection (3 mm) shows a large aneurysm originating from the right middle lobe (*), the wall thrombus (white arrowheads), and an air bubble in the aneurysm sac (white arrow). In addition, presence of an aneurysm of the anterior segment of the left upper lobe and a widening of the left lower pulmonary artery can be seen. B, Three-dimensional posteroanterior view of the intrathoracic vessels using a volume-rendering technique shows a large aneurysm originating from the right middle lobe (*) and a smaller one originating from the right lower lobe segmental artery (black arrow). Note the enlargement of the systemic bronchial arteries in the right bronchointercostal trunk (white arrow) and an aberrant left bronchial artery originating from the left subclavian artery (white arrowheads). Note also the enlargement of the right and left inferior phrenic arteries (black arrowheads). C, Right-sided pulmonary angiography. Occlusion of the large aneurysm originating from the middle lobe pulmonary artery was achieved using a total of 2.07 meters of coils. D, Coronal oblique thin-slab maximum-intensity projection (3 mm) shows the right atrial thrombus (*).The typical presentation of HSS involves in 3 phases: a first stage involving symptoms of thrombophlebitis, a second stage consisting of formation and enlargement of pulmonary aneurysms, and a third stage of aneurysmal rupture that triggers massive hemoptysis, leading to death. This case illustrates the endovascular treatment option for large pulmonary artery aneurysm rupture in HSS.DisclosuresNone.FootnotesReprint requests to A. Khalil, Service de Radiologie Hôpital Tenon, 4 rue de la Chine, 75020 Paris, France. E-mail [email protected] Previous Back to top Next FiguresReferencesRelatedDetailsCited By Kechida M, Daadaa S and Jomaa W (2022) Clinical presentation, radiological findings and treatment options in Hughes-Stovin syndrome, Rheumatology, 10.5114/reum.2022.115666, 60:2, (148-152), Online publication date: 30-Apr-2022. Emad Y, Ragab Y, Robinson C, Pankl S, Young P, Fabi M, Bawaskar P, Ibrahim O, Erkan D, Barman B, Tekavec-Trkanjec J, Jayakrishnan B, Kindermann M, Kechida M, Guffroy A, Silva R, Frikha F, Abou-Zeid A, Hassan M, Farber H, Abdelbary M, Tornes L, Margolesky J, El-Shaarawy N, Bennji S, Agarwala M, Saad A, Amezyane T, Ghirardo S, Cruz V, Niemeyer B, Al-Zeedy K, Al-Jahdali H, Jaramillo N, Demirkan S, Kably I, Kim J and Rasker J (2021) Pulmonary vasculitis in Hughes-Stovin syndrome (HSS): a reference atlas and computed tomography pulmonary angiography guide—a report by the HSS International Study Group, Clinical Rheumatology, 10.1007/s10067-021-05912-3, 40:12, (4993-5008), Online publication date: 1-Dec-2021. Hamdy F, Tharwat S, Abdelwahab H, Okasha H and Eltoraby E (2020) Hughes-Stovin syndrome without venous involvement – Unusual presentation of a rare disease: Case report and review of the literature, The Egyptian Rheumatologist, 10.1016/j.ejr.2019.09.001, 42:2, (159-163), Online publication date: 1-Apr-2020. El Jammal T, Gavand P, Martin M, Korganow A and Guffroy A (2019) Syndrome de Hughes-Stovin : à propos d'un cas chez un jeune patient avec thromboses récurrentes et anévrysme de l'artère pulmonaire et revue de la littérature, La Revue de Médecine Interne, 10.1016/j.revmed.2018.07.009, 40:2, (120-125), Online publication date: 1-Feb-2019. Voiriot G, Parrot A, Antoine M, Gibelin A, Haddad S, Carette M, Fartoukh M and Khalil A (2018) Transcatheter embolotherapy of pulmonary artery aneurysms as emergency treatment of hemoptysis in Behcet patients: experience of a referral center and a review of the literature, Internal and Emergency Medicine, 10.1007/s11739-018-1817-y, 13:4, (491-500), Online publication date: 1-Jun-2018. Cabot R, Rosenberg E, Pierce V, Dudzinski D, Baggett M, Sgroi D, Shepard J, Bond A, McDonald E, Ebeling S, Farber H, McDermott S, Witkin A, Kelly N, Miloslavsky E and Stone J (2018) Case 11-2018: A 48-Year-Old Woman with Recurrent Venous Thromboembolism and Pulmonary Artery Aneurysm, New England Journal of Medicine, 10.1056/NEJMcpc1800323, 378:15, (1430-1438), Online publication date: 12-Apr-2018. 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Khalil A, Parrot A, Fartoukh M, Djibre M, Tassart M and Carette M (2012) Pulmonary Artery Occlusion With Ethylene Vinyl Alcohol Copolymer in Patients With Hemoptysis: Initial Experience in 12 Cases, American Journal of Roentgenology, 10.2214/AJR.10.5370, 198:1, (207-212), Online publication date: 1-Jan-2012. Khalid U and Saleem T (2011) Hughes-Stovin Syndrome, Orphanet Journal of Rare Diseases, 10.1186/1750-1172-6-15, 6:1, (15), . Hacking C and Weerakkody Y (2010) Hughes-Stovin syndrome Radiopaedia.org, 10.53347/rID-12482 Chalazonitis A, Lachanis S, Mitseas P, Argyriou P, Tzovara J, Porfyrides P, Sotiropoulou E and Ptohis N (2009) Hughes-Stovin Syndrome: a case report and review of the literature, Cases Journal, 10.1186/1757-1626-2-98, 2:1, Online publication date: 1-Dec-2009. 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September 5, 2006Vol 114, Issue 10 Advertisement Article InformationMetrics https://doi.org/10.1161/CIRCULATIONAHA.106.614636PMID: 16952990 Originally publishedSeptember 5, 2006 PDF download Advertisement SubjectsComputerized Tomography (CT)Pulmonary HypertensionTreatment