We report a case that presents a diagnostic challenge in a 22 year-old female. CT-Scan and MRI showed a soft-density mass (12 cm) causing middle arch erosion of the fifth rib. In this rapidly-growing chest wall tumor a surgical-biopsy was very hemorrhagic and frozen section was unabled to disclose a sarcoma. Angiography and embolization of the feeding arteries were done. The final histopathology pointed out hemangioma. Complete resection was performed without prosthesis interposition. We emphasize two points regarding vascular chest wall tumors: (1) its possibility to mimick a sarcoma, so the surgical planning demands preoperative diagnosis; (2) the positive role of embolization in large and fast-growing lesions

Dartevelle, Philippe G.

de Montpreville, Vincent

Fadel, Elie

Macedo, Amarilio

Paul, Jean François

English

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CHEST WALL HEMANGIOMA: A DIFFICULT PREOPERATIVE DIAGNOSIS

Archives of the Faculty of Veterinary Medicine UFRGS

Volume 23 (1/2)     •   24CHEST WALL HEMANGIOMA: A DIFFICULT PREOPERATIVE DIAGNOSISAmarilio Macedo, MD, PhD, Elie Fadel, MD, PhD, Jean François Paul, MD, Vincent de Montpreville, MD,Philippe G. Dartevelle, MDABSTRACTWe report a case that presents a diagnostic challenge in a 22 year-old female. CT-Scanand MRI showed a soft-density mass (12 cm) causing middle arch erosion of the fifth rib. Inthis rapidly-growing chest wall tumor a surgical-biopsy was very hemorrhagic and frozensection was unabled to disclose a sarcoma. Angiography and embolization of the feedingarteries were done. The final histopathology pointed out hemangioma. Complete resectionwas performed without prosthesis interposition. We emphasize two points regarding vascularchest wall tumors: (1) its possibility to mimick a sarcoma, so the surgical planning demandspreoperative diagnosis; (2) the positive role of embolization in large and fast-growing lesions.INTRODUCTIONAccordingly to the International Society for the Studyof Vascular Anomalies (ISSVA) vascular anomalies(angiomas) should be divided in two large groups : vasculartumors and vascular malformations. The former includesmainly hemangiomas and are characterized by hyperplasiadue to proliferation of endothelial cells, pericytes and smoothmuscle cells . Vascular malformations have been defined asa dysmorphogenic process made of dysplasic vessels andwithout cellular proliferation [1]. We report a case of a chestwall hemangioma pointing out the importance of definitediagnosis to establish the correct surgery.A 22-year-old woman was referred to ourDepartment for evaluation of a right chest wall tumorthat appeared recently (2 months) and rapidly becameprominent. She denied trauma. Her physicalexamination was remarkable for a firm and smooth lesionmeasuring approximately 8 x 12 cm and located in theaxillary region at the level of the third, fourth and fifthribs. No thrills or bruits were heard on auscultation andthe overlying skin was intact. A chest radiographdemonstrated erosion in the middle arch of the fifth rib.A CT-Scan and a MRI showed a huge tumor of 12 cmin the great axis promoting an irregularity in the adjacentvisceral pleura. A surgical-biopsy was followed byhemorrhage and the frozen section was unable to disclosea sarcoma. Postoperatively an angiographic studyDepartments of Thoracic and Vascular Surgery, and Heart-Lung TransplantationHôpital Marie Lannelongue, Paris-Sud University, Plessis- Robinson, FranceHospital de Clinicas de Porto Alegre - Serviço de Cirurgia CardiotoracicaAddress reprint requests to Dr. Amarilio Macedo, Department of Cardiothoracic Surgery –Hospital de Clinicas de Porto Alegre, 2350 RuaRamiro Barcelos,  Porto Alegre, RS, Brazil.Phone/fax: +55 51 2101 8276, e-mail: amariliomacedo@terra.com.brallowed a clear visualisation of 4 pedicles feeding thetumor, mainly the 3rd intercostal artery. Embolizationwas accomplished with Ivalon and micro-coils. Finalhistopathologic diagnosis pointed out a benign vasculartumor.  A chest wall resection including the third riband the musculature adjacent to the tumor was done.There was no evidence of pleural or pulmonaryinvolvement. Definitive histopathology confirmed askeletal muscle hemangioma. The patient had anuneventfull recovery and was sent home in the 7thpostoperative day. A six-month follow-up was absolutelynormal.COMMENTIt is well established that hemangiomas are anunusual group of chest wall tumors. Although 73% ofhemangiomas are present at birth [2] in the setting ofintramuscular hemangiomas, a traumatic etiology has beenimplicated [3], at least as a promoter of the tumor expansion[4]. Concerning the hemangiomas, those involving the ribcage could also be divided in intramuscular hemangiomaand bone hemangioma. Skeletal muscles hemangiomasrepresent 0.7% of all benign hemangiomas and had a wideanatomic distribution [2]. They were located mostly inthe quadriceps muscle (19%) with the intercostalmusculature been affected in only 1.4% [5]. Bonehemangiomas showed a great predilection for the spineVolume 23 (1/2)     •   25CHEST WALL HEMANGIOMA: A DIFFICULT PREOPERATIVE DIAGNOSISand skull and not more than 2.8% were located in theribs[6]. The tumor appeared early in life or has been notedfor several years. Others observed evolutions through a 12-month [4, 8] or 36-month [3]. On palpation a tender non-pulsatile mass is generally found to be fixed along the mus-cular plane. The presence of thrills or bruits may indicate ahigh-flux vascular malformation.Diagnosis was stated preoperatively in only 19% of335 cases of skeletal muscle hemangiomas [7]. Commonlymistaken diagnosis include sarcoma, lipoma, and abscess.Transthoracic needle biopsies are useless [3] and evenhazardous as massive hemothorax and emergencythoracotomy had already been reported [8]. After ahemorrhagic surgical-biopsy that demanded bloodtransfusion our strategy was to attend a definite diagnosisand perform an angiographic study. Besides, frozen sectionwas dubious about malignancy. The lesion presented somemalignant charateristics like: large size, deep location, boneerosion and heterogeneity. The MRI was not able to show“worm-like” structures that could suggest a hemangioma-type presentation. On the other hand even agressiveneoplasms do not typically reach this size in two months.Painless tumor with such a rapid growth suggestshemorrhage into the lesion. In our case-report even a frozensection was not able to disclose sarcoma giving rise to thefact that chest wall hemangiomas may mimick a much moreaggressive tumor, that would demand a a well planned andmore radical chest wall resection. With obvious cosmeticand functional consequences. Vascular lesions should bebiopsied if there is any suspicion of malignancy [9].Selective angiography has played a major role as adiagnostic as well as therapeutic tool in some cases [10]. Weperformed an angiographic study that demonstratedhypervascularization and embolization of the 3rd, 4th, 5th in-tercostal arteries and a branch of the external thoracic arterywas done as a bridge to a less problematic surgical resection.Hemangiomas in the proliferation phase expressesbFGF, vascular endothelial growth factor (VEGF), andcolagenase IV [11]. Perhaps in the near future laboratorydata may facilitate diagnosis of vascular chest wall tumors.Complete excision with or without prosthetic replacementis the treatment that should be applied, alwaysremembering that local recurrence up to 18% was observedin a review of 89 cases of skeletal hemangioma [13].We concluded that in rapid-growing chest walltumors the diagnosis of hemangioma should be kept inmind and surgical-biopsy is a main step to a correct surgicalschedule. CT-Scan, some features of MRI images andspecially angiography may be helpful to suggesthemangioma. Finally, embolization of the vascularpedicles seems to facilitate the surgical approach byshrinkage of large tumors.REFERENCES1. Enjolras O. Vascular tumors and vascularmalformations: Are we at the dawn of a betterknowledge? Pediatr Dermatol 1999;16:238-41.2. Watson WL, McCarthy WD. Blood and lymph vesseltumors. A report of 1056 cases. Surg Gynecol Obstet1940; 71:569-88.3. Winchester DJ, Victor TA, Fry WA. Intercostalhemangioma presenting as a chest wall tumor. AnnThorac Surg 1992;54 :145-6.4. Yonehara Y, Nakatsuka T, Ichioka I, Takato T,Matsumoto S, Yamada A. Intramuscularhaemangioma of the anterior chest wall. Br J PlastSurg 2000;53:257-9.5. Scott JES. Hemangiomas in skeletal muscle. Br J Surg1957;44:496-501.6. Wenger DE, Wold LE. Benign vascular lesions of bone:radiologic and pathologic features. Skeletal Radiol2000;29:63-74.7. Shallow TA, Eyer SA, Wagner FB. Primaryhemangiomatous tumors of skeletal muscle. AnnSurg 1974;119:700-40.8. Yokoi K, Igarashi S, Matsugama H, Sawafuji M.Epithelioid hemangioendothelioma presenting as achest wall tumor. Thorac Cardiovasc Surgeon1997;45:254-8.9. Mueller BU, Mulliken JB. The infant with a vasculartumor. Semin Perinatol 1999;23:332-40.10. Pillsbury HC, Jones K. Vascular tumors of the headand neck: presentation, prognosis and dilemmas.Oncology 1989;3:23-9.11. Takahashi K, Mulliken JB, Kosakewich HP, RogersRA, Folkman J, Ezekowitz RA. Cellular markersthat distinguish the phases of hemangioma duringinfancy and childhood. J Clin Invest 1994;93:2357-64.12. Allen PW, Enzinger FM. Hemangioma of skeletalmuscle. Cancer 1972;29:8-22.

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CHEST WALL HEMANGIOMA: A DIFFICULT PREOPERATIVE DIAGNOSIS

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