Internal jugular vein hemangioma, also called pyogenic granuloma, is a rare tumor. Such a neoformation was accidentally discovered and excised in a middle-aged man. Histologic and immunohistochemical investigations were performed, and this case is compared with the poor amount of similar ones described in the literatur

Adovasio, Roberto

Calvagna, Cristiano

Cera, Chiara

Pancrazio, Francesco

Sgorlon, Giada

Zamolo, Francesca

ZAMOLO, FRANCESCA

ADOVASIO, ROBERTO

Archivio istituzionale della ricerca - Università di Trieste

DepartmenUniversity of TCorrespondSurgery, Dipargiche e dellaE-mail: r.adovAnn Vasc Surghttp://dx.doi.or 2015 ElsevieND license (htManuscript re2014; publisheInternal Jugular Vein Pyogenic CapillaryHemangioma: A Case ReportChiara Cera, Cristiano Calvagna, Giada Sgorlon, Francesca Zamolo, Francesco Pancrazio,and Roberto Adovasio, Trieste, ItalyInternal jugular vein hemangioma, also called pyogenic granuloma, is a rare tumor. Such a neo-formation was accidentally discovered and excised in a middle-aged man. Histologic and immu-nohistochemical investigations were performed, and this case is compared with the poor amountof similar ones described in the literature.Intravascular primitive tumors are very rare dis-eases and few data concerning these pathologiesare described in the literature. Moreover, thefew cases reported are mostly focused on largeveins.1 Among these tumors, internal jugularvein (IJV) hemangioma is one of the rarest. Inci-dentally, it has to be mentioned that the termscapillary pyogenic hemangioma, lobular capillaryhemangioma, and pyogenic granuloma (PG) aresynonyms.2These lesions are typically visible in thesuperficial derma. They are a common vascularproliferation that can rise after a minor traumaor a cutis infection and, although rarely, theselesions can form also subcutaneously and/orintravenously.2The cited neoformations are very often localizedin the upper part of the body, and, in particular, inarms, forearms, wrists, fingers, and, above all,neck,3 whereas only 2 cases have been reported,respectively, in the iliac vein4 and ovary.5 Lesionscan be associated with palpable swellings, and thiscould suggest a malignant event. Consequently, at of Vascular Surgery, ‘‘Ospedali Riuniti di Trieste’’,rieste, Trieste, Italy.ence to: Roberto Adovasio, MD, Department of Vasculartimento Universitario Clinico di Scienze Mediche, Chirur-Salute, strada di Fiume n447, Trieste 34100, Italy;asio@fmc.units.it2015; 29: 361.e5e361.e7g/10.1016/j.avsg.2014.08.015r Inc. This is an open access article under the CC BY-NC-tp://creativecommons.org/licenses/by-nc-nd/3.0/).ceived: May 21, 2014; manuscript accepted: August 3,d online: November 4, 2014.correct differential diagnosis is very important tobe reached.6 Anyway, PGs show benign evolution,can be even totally asymptomatic and accidentallydiscovered during investigations focused on otherpathologies.7 This is what happened for the casedescribed here.CASE REPORTDuring an echo examination focused on thyroid, a neo-formation was accidentally discovered inside the leftIJV of a 51-year-old man. The patient was asymptom-atic, without palpable masses and he did not refer tohad suffered locoregional traumas. Anyway, in thepast, he was subjected to reductive mastectomy forgynecomasty. In that occasion also hypothyroidismwas diagnosed, and he was consequently treated withsubstitutive hormone therapy (sodium levothyroxine,50 mg/day).After echo scanning, he underwent an angio computedtomography scan of the supra-aortic trunks thatconfirmed the presence of an IJV nonocclusive intralu-men neoformation with an oval, moderately irregular,and unevenmorphology. The lesionwas stuck on the pos-terioremedial face of the mentioned vein, and its dimen-sions were about 10  8.7  7.6 mm.The first diagnosis was controversial between a partialthrombosis of the vein and a primitive endoluminalneoplasia, as it was reported for previous cases.4 The sub-sequent Echo-Duplex scan investigation of the supra-aortic trunks, performed before and after echographicSonoVue (Bracco, Milano) injection, revealed the pres-ence of a markedly hypoechoic, vascularized, and appar-ently pedunculated neoformation (Fig. 1). The lesionwas compressible by the transducer and displayed preco-cious artery enhancement and progressive washout. Thislet us to rule out that the mass was ascribable to a clot361.e5Fig. 1. Echo-Doppler scan investigation after echo-graphic SonoVue injection.Fig. 2. Excised PG-containing IJV segment.Fig. 3. CD 31 stain outlining vessels’ distribution in neo-formation (20 magnification).361.e6 Case reports Annals of Vascular Surgeryand suggested a most likely productive process insidethe vein.The neoformation was surgically removed. Weapproached the IJV by a left supraclavicular incisionand then we excised the corresponding vein segmentthat displayed the internal palpable mass. The proximaland distal stumps were ligated, we didn’t perform theIJV reconstruction because of the patency and thegood size of the external jugular vein (EJV; Fig. 2). His-tologic examination, immunohistochemical coloring forCD-31 (Fig. 3), CD-34, and podoplanin, and histochemi-cal coloring for Alcian-blue pas and Azan Malloryrevealed that the neoformation was made up by myxoidstroma and little vascular structures, similar tocapillaries.Themorphologic and immunohistochemical scenarios,according to the instrumental evidences mentioned previ-ously, appear to be coherent with the diagnosis of a pyo-genic capillary hemangioma (PG).Finally, 1 month after discharge, the patientunderwent an Echo scan which revealed good patencyof the EJV.DISCUSSIONLiterature data concerning PGs localized on the in-ternal walls of the veins are extremely poor, butthis fact, almost certainly, does not reflect the realincidence of this pathology. In fact, they are veryoften asymptomatic and incidentally discovered.The first documented case of PG is dated back to1967, when a neoformation in the EJV was detectedin a 32-year-old man and a thrombotic event wasruled out after a phlebography.8The improvement of the imaging techniques haslater allowed to reach more precise diagnostic pic-tures. Anyway, their confirmation always needs asurgical approach followed by histologic, histo-chemical, and immunohistochemical evaluations.The etiology of these events remains obscure.Some authors describe the existence of congenitalvenous hemangiomas,9 but these lesions are detect-able also after traumas or infectious events thattrigger vascular hyperproliferation. In addition,pressure or hormone shocks or disorders can berelated with the incoming of a PG.10Hemangiomas are generally characterized byveins that display a single-layer endothelium and anormal endothelial cell cycle.3 The peculiar histo-logic traits of PG and vascular hemangiomas in gen-eral, allow to differentiate them from malignantintravascular tumors.4,11 Thus, a multidiscipline,radiologic, surgical, histologic approach is essentialto reach a correct and detailed diagnosis.To our knowledge, against a certain amount ofEJV hemangioma cases reported in the literature,only a couple of cases involving IJV are detectable.They were both correctly characterized only aftersurgical excision and histologic or immunohisto-logic analyses, although they displayed a radiologicVol. 29, No. 2, February 2015 Case reports 361.e7aspect that was definitely close to the known casesascribable to EJV hemangioma.7,10In our case, the patient suffered from a precedinghypothyroidism and the PG presence in the IJVcould be a consequence of an associated hormonedisorder. The investigations focused onto the controland improvement of the thyroid pathology let us tohighlight by chance the intravenous pyogenicneoformation.Its dimensions, pedunculated shape, and hypoe-choic nature, suggested us to deal with a nonthrom-botic mass, and to drive the diagnosis toward aprimitive productive neoplastic process. This wasthe main reason which led us to follow a surgicalapproach.So, echo-contrast investigation seems to be thegold standard technique for a first-level differentialdiagnosis as it has also been reported for a recentcase.12 In fact, the diagnostic enhancement becomesmore relevant and intense in contrastography.Furthermore, the exposition of patients to ionizingradiation and incoming artifacts ascribable to den-ture amalgams can be reduced in respect to moreinvasive ionizing techniques. That is, the resultswould be devoid of misleading interferences and,moreover, the relative costs of the analysis couldbe reduced.Finally, the subsequent postsurgical immunohis-tochemical investigations confirmed the presenceof PG.In conclusion, although the continuous improve-ment of radiologic techniques and clinical investiga-tions that allow to perform a correct differentialdiagnosis of PG from a partial venous thrombosis,endovenous neoformations, such as intravascularleiomyosarcoma or primary lymphoma, must besurgically excised to allow a certain diagnosis.Although in most cases vein’s reconstruction is notstrictly mandatory, the venous flow continuityrestoration may be requested only for some veins,as for example is for the inferior vena cava, to avoida consequent undesired hypertension.Thus, the possibility to reach a right diagnosis ofIJV PG remains an open challenge because only af-ter surgical excision a definitive diagnosis is possible.REFERENCES1. Harris EJ Jr, Taylor LM Jr, Porter JM. Epithelioid heman-gioendothelioma of the external iliac vein: a primaryvascular tumor presenting as traumatic venous obstruction.J Vasc Surg 1989;10:693e9.2. Barr KL, Vincek V. Subcutaneous intravascular pyogenicgranuloma: a case report and review of the literature. Cutis2010;86:130e2.3. Cooper PH, Mc Allister HA, Helwig EB. Intravenous pyo-genic granuloma. Am J Surg Pathol 1979;3:221e8.4. Pradhan S, Bazan H, Salem R, et al. Intravenous lobularcapillary hemangioma originating in the iliac veins: a casereport. J Vasc Surg 2008;47:1346e9.5. McKee GT, Fletcher CD, McKee PH. Visceral intravascularcapillary hemangioma. Arch Pathol Lab Med 1987;111:390e2.6. Maher A, Civil I. Intravenous lobular capillary hemangioma.Ann Vasc Surg 2010;24:951.e13e5.7. Al-Natour M, Kenmuir C, Khuder S, et al. Internal jugularvein hemangioma. Ann Vasc Surg 2012;2012:1129.e1e4.8. Meyers MA. Hemangioma of the external jugular vein.Radiology 1967;89:483e5.9. Werner JA, D€unne AA, Folz BJ, et al. Current concepts inthe classification, diagnosis and and treatment of hemangi-omas and vascular malformations of the head and neck.Eur Arch Otorhinolaryngol 2001;258:141e9.10. Duggal P, Chaturvedi P, Pai PS, et al. Internal jugular veinvascular malformation presenting as mass at root of neck:a case report. BMC Nose Throat Disord 2009;9:5.11. Quian LH, Hui YZ. Intravenous pyogenic granuloma: immu-nohistochemical considerationda case report. Vasc Surg2001;35:315e9.12. Ahn SE, Park JS, Ryu KN, et al. Intravenous lobular capil-lary hemangioma occurring in the cephalic vein of a 39-year-old adult. J Clin Ultrasound 2014;42:375e8.

Internal Jugular Vein Pyogenic Capillary Hemangioma: A Case Report.

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Internal Jugular Vein Pyogenic Capillary Hemangioma: A Case Report.

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