Pathology
Intraosseous hemangioma of the zygomatic bone
C u e s t a Gil, M a t i a s and Navarro-Vila, C a r l o s Complutense University of Madrid, Department of Maxillofacial Surgery and Head and Neck Surgery, Gregorio MarafiSn General Hospital (Madrid)
A case report M. Cuesta Gil, C. Navarro-Vila." Intraosseous hemangioma o f the zygomatic bone. A case report. Int. J. Oral Maxillofac. Surg. 1992; 21: 287-291. Abstract. Intraosseous hemangiomas of the facial skeleton are infrequently seen, with most cases occurring in the mandible and maxilla. Hemangioma of the zygomatic bone, however, is extremely rare. A case is presented in which selective embolization, resection of the tumor, and immediate reconstruction were carried out. Abstrat. Los hemangiomas intraoseos del esqueleto facial son raros tumores que se presentan mils frecuentemente en la mandibula y el maxilar superior. E1 hemangioma del hueso malar es extremadamente raro. Presentamos un nuevo caso de hemangioma intra6seo malar que fu6 tratado mediante embolizaci6n selectiva, resecci6n tumoral e inmediata reconstrucci6n con injerto de calota.
Introduction
Intraosseous hemangiomas are benign, slowly growing tumors arising from newly formed blood vessels. The incidence of intraosseous hemangiomas varies from 0.5 to 1% of all osseous neoplasms 4'33'34. The sites most frequently involved are the vertebral column and the calvarium 4'6'1~,~3,3°. The most commonly affected facial bones are the mandible, maxilla6'26, and nasal bones 35. Hemangioma of the zygomatic bone is extremely rare. Intraosseous hemangioma is three times more frequent in women than in men. It is found most commonly in the fourth decade, but the age ranges from infancy to the eighth decade 34. The clinical diagnosis of intraosseous hemangioma may be difficult to make, but radiographs typically show a "honeycomb" or a "sunburst" appearance. The treatment of choice is surgical excision. In 1950 SCHOF~ELD2° made the first report of a case of hemangioma in the zygomatic bone. A total of 16 cases have been published in the English literature. An additional case is presented and the treatment, including the reconstruction by calvarium bone grafts, is discussed.
Key words: hemangioma; zygomatic bone; intraosseous tumor; embolization. Accepted for publication 8 May 1992
Two and a half years previously a hard mass had been detected which had grown slowly over the left malar eminence. She was referred to our department after having been seen by other specialists who had not been able to diagnose this lesion. She denied any previous trauma. Physical examination revealed a 4- by 5-cm tumor in the left malar eminence, causing cosmetic deformity, asymmetry of the palpebral fissure, and superior vertical displacement
of the eyeball (Fig. 1). Radiographs showed an oval, radiopaque lesion in the left zygomatico-maxillary complex (Fig. 2). Computed tomography (CT) revealed a mixed density mass (bone and soft tissue) that completely occupied the left zygomatico-maxillary complex (Fig. 3). A left carotid arteriogram revealed a tumor "blush" with prominent blood supply from the facial and internal maxillary arteries (Fig. 4). A selective embolization with particulate
Fig. 1. Preoperative frontal view showing the swelling in the maxillo-zygomatic area.
Fig. 2. Waters view, showing radiopaque mass with a "sunburst" effect.
Case report
A 12-year-old girl presented with a 4year history of pain in the left maxilla.
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Fig. 3. CT scan showing dense tumor mass
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Gelfoan ® and Ivalon® (polyvinyl alcohol) was performed 24 h before the operation. A coronal and infraorbital approach was used for the tumor resection. Bleeding appeared to be minimal. The reconstruction of the defect was carried out with the inner table of the parietal bone. The outer table was used to cover the donor site. The inner table was shaped into a tripod form, to reconstruct the left zygomatic bone, infraorbital rim, and part of the zygomatic arch (Fig. 5). The postoperative course was uneventful. Histopathological examination revealed the typical features of a cavernous hemangioma (Fig. 6). At present, 3 years postoperatively, there is no evidence of recurrent disease, and the graft has survived. A satisfactory cosmetic appearance has been achieved. The patient has normal vision without diplopia, and the ocular movements are normal (Fig. 7).
Fig. 5. Inner table of parietal bone shaped in
the desired contour.
A hemangioma is a proliferation of capillaries or vascular channels lined by endothelium22. Histologically, hemangiomas are classified as cavernous, capillary, mixed, cellular, and sclerosiug25. In relation to clinical behavior, however, hemangiomas are subdivided into only two types: capillary and cavernous H. Both types have been found in the zygoma. The case presented ,appeared to be cavernous. The diagnosis of hemangiomas of the facial bones often poses difficulties. They usually present as an asymptomatic, gradually enlarging swelling. Tenderness may be present, but pain is not commonly encountered. Intraosseous hemangiomas of the zygoma may cause deformity of the cheek and orbit. Characteristically, hemangiomas show a reticulated or honeycomb pattern that is well demarcated from the normal bone. A "sunburst" effect is often present; it is created by the osseous specules radiating from the center of the lesion. Some authors claim that the diagnosis can be made from radiographs 1,23. Others consider the radiographic presentation of intraosseous hemangiomas to be nonspecific21. Computed tomography often shows an expansile process with a high-density, amorphous mass that may be suggestive of fibrous dysplasia29. Magnetic resonance imaging (MRI) appears to be extremely reliable. Because of its low flow state, intraosseous hemangioma ap-
Discussion
There are 16 well-documented cases of intraosseous hemangiomas of the zygoma presented in the English literature 3'5'8'9'12'14'19'~°'23'31'32. The clinical, histopathological, and therapeutic as-
Fig. 4. Arteriogram of the carotid artery
showing abundant blood vessels in the area of the tumor.
Fig. 6. Proliferation of blood vessels, lined by
regular endothelium and lying in a fibrous stroma with bone trabeculae. HE x 200. pects of these cases and our case are summarized in Table 1. Hemangiomas were originally classified as true neoplasms of the blood vessels24'27, but now many authors consider them to be hamartomas or hamartomatous malformations2'15'33. Trauma is sometimes mentioned as a possible cause 14, and 50% of nasal bone hemangiomas have been associated with trauma TM. In the case presented, there was no history of trauma.
Fig. 7. Postoperative frontal view. Acceptable
cosmetic appearance with only slight depression of the zygomatic area.
Zygomatic hemangioma pears as a " b l u s h " . N o b o n e windows are necessary, since M R I does n o t enh a n c e b o n e 32. A n g i o g r a p h y shows a tum o r blush, offering a d d i t i o n a l inform a t i o n , particularly if the lesion is large. A n g i o g r a p h y was carried o u t in four o t h e r patients 3,5,12. In o u r patient, angiog r a p h y was the principal diagnostic test, a n d it a p p e a r e d to b e very useful in p l a n n i n g treatment. I n t r a o s s e o u s h e m a n g i o m a s are benign lesions, m a k i n g radical excision unnecessary, b u t the skeletal d e f o r m i t y often requires t r e a t m e n t , as in the case presented. R a d i a t i o n , curettage, a n d surgical excision h a v e been p r o p o s e d for this tumor. A l t h o u g h curettage h a s been used successfully 12'14'19,p o t e n t i a l morbility because of u n c o n t r o l l e d h e m o r r h a g e is high. R a d i a t i o n t h e r a p y has b e e n s h o w n to stop the p r o g r e s s i o n of the disease, b u t does n o t alter the cosmetic deformity left b y the lesion 5'6. DAVIS & MORGAN 5 believe t h a t i r r a d i a t i o n should be the p r i m a r y m o d e o f treatm e n t for calvarial a n d skeletal hemangiomas. There are, however, reports of malignant degeneration of benign osseous n e o p l a s m s following r a d i a t i o n t h e r a p y 7. R a d i a t i o n t h e r a p y m a y cause tissue necrosis a n d tends to affect the g r o w t h potential o f vital tissues such as b o n e a n d teeth; it also m a y give rise to telangiectasis o f the skin overlying the lesion. Some a u t h o r s 27'28 describe angiograp h y a n d e m b o l i z a t i o n before surgery. Others l° have p r o p o s e d e m b o l i z a t i o n as the only therapy. DAVIS & MORGAN 5 a d v o c a t e ligating the external carotid artery o f the involved side. SMITH27 h a s p o i n t e d o u t t h a t ligation o f feeding vessels has n o t altered s u b s e q u e n t b l o o d flow. In the case presented, selective emb o l i z a t i o n 24 h before the o p e r a t i o n app e a r e d to be very effective, as m i n i m a l bleeding was e n c o u n t e r e d . Surgical t r e a t m e n t is the m o s t frequently used therapy. Resection of the zygoma, however, requires i m m e d i a t e reconstruction. T h e free calvarial b o n e graft has a n u m b e r o f advantages, in c o m p a r i s o n to free iliac b o n e a n d costal grafts, for r e c o n s t r u c t i o n of some defects in the maxillofacial area. T h e same surgical e n t r a n c e can be used to harvest the bone. T h e utilization o f the i n n e r table permits large b o n e grafts to be t a k e n so that, w h e n the outer table is replaced in its original site, there is n o area o f depression. T h e cranial b o n e is easier to m o l d t h a n iliac bone, a n d it
appears to be less resorbable, as comp a r e d with other free b o n e grafts 16'17.
Acknowledgments. The authors wish to thank Dr ALVAREZFERN/kNDEZfor the pathological studies and Mr FLORENC10 PASCUALCUESTA for typing the manuscript.
References 1. BucY PC, CArp CS. Primary hemangioma of bone with special reference to roentgenologic diagnosis. Am J Roentgenol Radium Ther Nucl Med 1930: 23: 1. 2. CHOUKAS NC, TOTO PD, VALAITIS J. Sclerosing cavernous hemangioma of the maxilla. Oral Surg 1963: 16: 17. 3. CLAUSERL, MENEGHINIF, RIGA M, RIGO L. Haemangioma of the zygoma. Report of two cases and review of the literature. J Craniomaxillofac Surg 1991: 19: 353-8. 4. DAHLIN DC. Bone tumors. 3rd edn. Springfield: Charles C. Thomas, 1978: 137 48. 5. DAWS E, MORGEN R. Hemangioma of bone. Arch Otolaryngol 1974: 99: 443. 6. DORFMAN HD, STEINER GC, JAFFE HL. Vascular tumors of bone. Hum Pathol 1971: 2:349 76. 7. GROSS CW, MONTGOMERY WW. Fibrous dysplasia and malignant degeneration. Arch Otolaryngol 1967: 85: 653-7. 8. HAR-EL G, HADAR T, ZIRKIN HY, SIDI J. Hemangioma of the zygoma. Ann Plast Surg 1987: 18: 534-40. 9. HAR-EL G, LEVY R, AVIDOR I, SEGALK, SIDI J. Hemangioma of the zygoma presenting as a tumor in the maxillary sinus. J Oral Maxillofac Surg 1986: 14:161 4. 10. HOEY MF, COURAGE GR, NEWTON TH. Management of vascular malformations of the mandible and maxilla: review and report of two cases treated by embolization and surgical obliteration. J Oral Surg 1970: 28: 696. 11. JAFFE HL. Tumors and tumorous conditions of bones and joints. Philadelphia: Lea & Febiger, 1958: 224-39. 12. JETER TS, HACKNEY FL, AUFDEMORTE TB. Cavernous hemangioma of the zygoma: report of cases. J Oral Maxillofac Surg 1990: 48: 512-15. 13. LICHTENSTEIN L. Bone tumors. 4th edn. St Louis: Mosby, 1972: 166-84. 14. MARSHAK G. Hemangioma of the zygomatic bone. Arch Otolaryngol 1980: 106: 581. 15. MATTrIEWS DN. Hemangiomata. Plast Reconst Surg 1968: 41: 528. 16. MAVES MD, MATT BH. Calvarian bone grafting of facial defects. Otolaryngol Head Neck Surg 1986: 95: 464-70. 17. POPE TL, FECHNER RE, KEATS TE. Case Report 367. Skeletal Radiol 1986: 15: 327-9. 18. POWELL NB, RILEY RW. Facial contouring with outer-table calvarial bone.
291
Arch Otolaryngol Head Neck Surg 1989: 115: 1454-9. 19. SCHMIDT GH. Hemangioma in the zygoma. Ann Plast Surg 1982: 9: 330-2. 20. SCHOFIELD AL. Primary hemangioma of the malar bone. Br J Plast Surg 1950: 3: 136-40. 21. SELEE RW, SHERMAN M, MILLER TF. Arteriovenous malformations of the mandible. ORL J Otorhinolaryngol Relat Spec 1978: 86: 659. 22. SHAFERWS, HINE MK, LEVYBM. A textbook of oral pathology. 3rd edn. Philadelphia: Saunders, 1974: 104. 23. SHERMAN RS, WILNER D. The roentgen diagnosis of hemangioma of bone. Am J Roentgenol 1961: 86:1146 59. 24. SHIRA RB, GUERNSEY LH. Central cavernous hemangioma of the mandible. J Oral Surg 1965: 23: 636. 25. SHKLAR G, MEYER I. Vascular tumors of the mouth and jaws. Oral Surg 1965: 19: 335. 26. SMITH HW. Hemangioma of the jaws. Arch Otolaryngol 1959: 70: 579. 27. SMITH RA. Central hemangioma of the maxilla: case report. Aust Dent J 1972: 17:117 19. 28. STASSI J, RAO VM, LOWRY L. Hemangioma of bone arising in the maxilla. Skeletal Radiol 1984: 12:187 91. 29. Suss RA, KUMAR A J, DOREMAN HD, MILLER NR, ROSENBAUMAE. Capillary hemangioma of the sphenoid bone. Skeletal Radiol 1984: 11: 10~7. 30. UNNI KK. Hemangioma, hemangiopericytoma and hemangioendothelioma (angiosarcoma) of bone. Cancer 1971: 27: 1403. 31. WALKER EA, MCHENRY LC. Primary hemangioma of the zygoma. Arch Otolaryngol 1965: 81: 199-203. 32. WARMAN S, MYSSIOREK D. Imagin case study of the mouth: hemangioma of the zygomatic bone. Ann Otol Rhinol Laryngol 1989: 98: 655. 33. WATSON WL, MCCARTHY WD. Blood and lymph vessel tumors: report of 1056 cases. Surg Gynecol Obstet 1940: 71: 569. 34. WYrCEBD. Primary hemangioma of skull: rare cranial tumor. Am J Roentgenol 1949: 61:302 16. 35. ZIZMORE J, ROBI~ETT WF, SPIRO RH, et al. Hemangioma of the nasal bones: radiographic appearance. Ann Otol Rhinol Laryngol 1978: 87:360 3.
Address: Dr Matlas Cuesta Gil, MD Maxillofacial Surgery Department of Head and Neck Surgery Gregorio Mara~6n General Hospital C/O Dr Esquerdo, no. 46 28007 Madrid Spain
Intraosseous hemangioma of the zygomatic bone
C u e s t a Gil, M a t i a s and Navarro-Vila, C a r l o s Complutense University of Madrid, Department of Maxillofacial Surgery and Head and Neck Surgery, Gregorio MarafiSn General Hospital (Madrid)
A case report M. Cuesta Gil, C. Navarro-Vila." Intraosseous hemangioma o f the zygomatic bone. A case report. Int. J. Oral Maxillofac. Surg. 1992; 21: 287-291. Abstract. Intraosseous hemangiomas of the facial skeleton are infrequently seen, with most cases occurring in the mandible and maxilla. Hemangioma of the zygomatic bone, however, is extremely rare. A case is presented in which selective embolization, resection of the tumor, and immediate reconstruction were carried out. Abstrat. Los hemangiomas intraoseos del esqueleto facial son raros tumores que se presentan mils frecuentemente en la mandibula y el maxilar superior. E1 hemangioma del hueso malar es extremadamente raro. Presentamos un nuevo caso de hemangioma intra6seo malar que fu6 tratado mediante embolizaci6n selectiva, resecci6n tumoral e inmediata reconstrucci6n con injerto de calota.
Introduction
Intraosseous hemangiomas are benign, slowly growing tumors arising from newly formed blood vessels. The incidence of intraosseous hemangiomas varies from 0.5 to 1% of all osseous neoplasms 4'33'34. The sites most frequently involved are the vertebral column and the calvarium 4'6'1~,~3,3°. The most commonly affected facial bones are the mandible, maxilla6'26, and nasal bones 35. Hemangioma of the zygomatic bone is extremely rare. Intraosseous hemangioma is three times more frequent in women than in men. It is found most commonly in the fourth decade, but the age ranges from infancy to the eighth decade 34. The clinical diagnosis of intraosseous hemangioma may be difficult to make, but radiographs typically show a "honeycomb" or a "sunburst" appearance. The treatment of choice is surgical excision. In 1950 SCHOF~ELD2° made the first report of a case of hemangioma in the zygomatic bone. A total of 16 cases have been published in the English literature. An additional case is presented and the treatment, including the reconstruction by calvarium bone grafts, is discussed.
Key words: hemangioma; zygomatic bone; intraosseous tumor; embolization. Accepted for publication 8 May 1992
Two and a half years previously a hard mass had been detected which had grown slowly over the left malar eminence. She was referred to our department after having been seen by other specialists who had not been able to diagnose this lesion. She denied any previous trauma. Physical examination revealed a 4- by 5-cm tumor in the left malar eminence, causing cosmetic deformity, asymmetry of the palpebral fissure, and superior vertical displacement
of the eyeball (Fig. 1). Radiographs showed an oval, radiopaque lesion in the left zygomatico-maxillary complex (Fig. 2). Computed tomography (CT) revealed a mixed density mass (bone and soft tissue) that completely occupied the left zygomatico-maxillary complex (Fig. 3). A left carotid arteriogram revealed a tumor "blush" with prominent blood supply from the facial and internal maxillary arteries (Fig. 4). A selective embolization with particulate
Fig. 1. Preoperative frontal view showing the swelling in the maxillo-zygomatic area.
Fig. 2. Waters view, showing radiopaque mass with a "sunburst" effect.
Case report
A 12-year-old girl presented with a 4year history of pain in the left maxilla.
288
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Fig. 3. CT scan showing dense tumor mass
in zygomatic bone.
Gelfoan ® and Ivalon® (polyvinyl alcohol) was performed 24 h before the operation. A coronal and infraorbital approach was used for the tumor resection. Bleeding appeared to be minimal. The reconstruction of the defect was carried out with the inner table of the parietal bone. The outer table was used to cover the donor site. The inner table was shaped into a tripod form, to reconstruct the left zygomatic bone, infraorbital rim, and part of the zygomatic arch (Fig. 5). The postoperative course was uneventful. Histopathological examination revealed the typical features of a cavernous hemangioma (Fig. 6). At present, 3 years postoperatively, there is no evidence of recurrent disease, and the graft has survived. A satisfactory cosmetic appearance has been achieved. The patient has normal vision without diplopia, and the ocular movements are normal (Fig. 7).
Fig. 5. Inner table of parietal bone shaped in
the desired contour.
A hemangioma is a proliferation of capillaries or vascular channels lined by endothelium22. Histologically, hemangiomas are classified as cavernous, capillary, mixed, cellular, and sclerosiug25. In relation to clinical behavior, however, hemangiomas are subdivided into only two types: capillary and cavernous H. Both types have been found in the zygoma. The case presented ,appeared to be cavernous. The diagnosis of hemangiomas of the facial bones often poses difficulties. They usually present as an asymptomatic, gradually enlarging swelling. Tenderness may be present, but pain is not commonly encountered. Intraosseous hemangiomas of the zygoma may cause deformity of the cheek and orbit. Characteristically, hemangiomas show a reticulated or honeycomb pattern that is well demarcated from the normal bone. A "sunburst" effect is often present; it is created by the osseous specules radiating from the center of the lesion. Some authors claim that the diagnosis can be made from radiographs 1,23. Others consider the radiographic presentation of intraosseous hemangiomas to be nonspecific21. Computed tomography often shows an expansile process with a high-density, amorphous mass that may be suggestive of fibrous dysplasia29. Magnetic resonance imaging (MRI) appears to be extremely reliable. Because of its low flow state, intraosseous hemangioma ap-
Discussion
There are 16 well-documented cases of intraosseous hemangiomas of the zygoma presented in the English literature 3'5'8'9'12'14'19'~°'23'31'32. The clinical, histopathological, and therapeutic as-
Fig. 4. Arteriogram of the carotid artery
showing abundant blood vessels in the area of the tumor.
Fig. 6. Proliferation of blood vessels, lined by
regular endothelium and lying in a fibrous stroma with bone trabeculae. HE x 200. pects of these cases and our case are summarized in Table 1. Hemangiomas were originally classified as true neoplasms of the blood vessels24'27, but now many authors consider them to be hamartomas or hamartomatous malformations2'15'33. Trauma is sometimes mentioned as a possible cause 14, and 50% of nasal bone hemangiomas have been associated with trauma TM. In the case presented, there was no history of trauma.
Fig. 7. Postoperative frontal view. Acceptable
cosmetic appearance with only slight depression of the zygomatic area.
Zygomatic hemangioma pears as a " b l u s h " . N o b o n e windows are necessary, since M R I does n o t enh a n c e b o n e 32. A n g i o g r a p h y shows a tum o r blush, offering a d d i t i o n a l inform a t i o n , particularly if the lesion is large. A n g i o g r a p h y was carried o u t in four o t h e r patients 3,5,12. In o u r patient, angiog r a p h y was the principal diagnostic test, a n d it a p p e a r e d to b e very useful in p l a n n i n g treatment. I n t r a o s s e o u s h e m a n g i o m a s are benign lesions, m a k i n g radical excision unnecessary, b u t the skeletal d e f o r m i t y often requires t r e a t m e n t , as in the case presented. R a d i a t i o n , curettage, a n d surgical excision h a v e been p r o p o s e d for this tumor. A l t h o u g h curettage h a s been used successfully 12'14'19,p o t e n t i a l morbility because of u n c o n t r o l l e d h e m o r r h a g e is high. R a d i a t i o n t h e r a p y has b e e n s h o w n to stop the p r o g r e s s i o n of the disease, b u t does n o t alter the cosmetic deformity left b y the lesion 5'6. DAVIS & MORGAN 5 believe t h a t i r r a d i a t i o n should be the p r i m a r y m o d e o f treatm e n t for calvarial a n d skeletal hemangiomas. There are, however, reports of malignant degeneration of benign osseous n e o p l a s m s following r a d i a t i o n t h e r a p y 7. R a d i a t i o n t h e r a p y m a y cause tissue necrosis a n d tends to affect the g r o w t h potential o f vital tissues such as b o n e a n d teeth; it also m a y give rise to telangiectasis o f the skin overlying the lesion. Some a u t h o r s 27'28 describe angiograp h y a n d e m b o l i z a t i o n before surgery. Others l° have p r o p o s e d e m b o l i z a t i o n as the only therapy. DAVIS & MORGAN 5 a d v o c a t e ligating the external carotid artery o f the involved side. SMITH27 h a s p o i n t e d o u t t h a t ligation o f feeding vessels has n o t altered s u b s e q u e n t b l o o d flow. In the case presented, selective emb o l i z a t i o n 24 h before the o p e r a t i o n app e a r e d to be very effective, as m i n i m a l bleeding was e n c o u n t e r e d . Surgical t r e a t m e n t is the m o s t frequently used therapy. Resection of the zygoma, however, requires i m m e d i a t e reconstruction. T h e free calvarial b o n e graft has a n u m b e r o f advantages, in c o m p a r i s o n to free iliac b o n e a n d costal grafts, for r e c o n s t r u c t i o n of some defects in the maxillofacial area. T h e same surgical e n t r a n c e can be used to harvest the bone. T h e utilization o f the i n n e r table permits large b o n e grafts to be t a k e n so that, w h e n the outer table is replaced in its original site, there is n o area o f depression. T h e cranial b o n e is easier to m o l d t h a n iliac bone, a n d it
appears to be less resorbable, as comp a r e d with other free b o n e grafts 16'17.
Acknowledgments. The authors wish to thank Dr ALVAREZFERN/kNDEZfor the pathological studies and Mr FLORENC10 PASCUALCUESTA for typing the manuscript.
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Address: Dr Matlas Cuesta Gil, MD Maxillofacial Surgery Department of Head and Neck Surgery Gregorio Mara~6n General Hospital C/O Dr Esquerdo, no. 46 28007 Madrid Spain
