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| CASE REPORT |
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| Year : 2023 | Volume
: 3
| Issue : 2 | Page : 529-530 |
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Cavernous hemangioma of zygomatic bone - A rare case report
Shivayogi R Kusagur, Daneshwari S Gaddi
Department of Ophthalmology, J. J. M. Medical College and Hospital, Davanagere, Karnataka, India
| Date of Submission | 27-Aug-2022 |
| Date of Acceptance | 31-Jan-2023 |
| Date of Web Publication | 28-Apr-2023 |
Correspondence Address:
Daneshwari S Gaddi
Department of Ophthalmology, J. J. M. Medical College and Hospital, Davanagere - 577 004, Karnataka
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/ijo.IJO_2150_22
A 45-year-old woman presented with a history of unilateral fixed, painless, gradually progressive swelling in the right frontal part of the zygomatic bone for 6 years. On examination, the best-corrected visual acuity (BCVA) of the right and left eye was 20/20. On palpation, the swelling was a solitary mass of hard consistency, painless, adhering to deep planes under normal-looking skin. Orbital computed tomography (CT) scanning showed monostotic fibrous dysplasia of the lateral wall of the right bony orbit of the zygoma. The right orbital mass was managed by excisional biopsy with bone grafting. The histopathological report revealed a cavernous hemangioma of the zygomatic bone.
Keywords: Bone graft, cavernous hemangioma, zygomatic bone
How to cite this article:
Kusagur SR, Gaddi DS. Cavernous hemangioma of zygomatic bone - A rare case report. Indian J Ophthalmol Case Rep 2023;3:529-30 |
Cavernous hemangioma is the most common benign orbital neoplasm among adults.[1] Skeletal cavernous hemangiomas are rare benign tumors of blood vessels. They occur most commonly in the spinal vertebral body, followed by the skull. These lesions account for 10% of benign calvarial tumors and 0.7% of osseous neoplasms. Among the bones of the skull, the lesions occur most commonly in the parietal bone, followed by the frontal bone.[2],[3]
The first case of cranial cavernous hemangioma was described by Toynbee in 1845.[4] Although a rudimentary lesion may be present at birth, cavernous hemangioma does not usually become symptomatic until the third to fifth decade. The tumor is characteristically unilateral and solitary, it is located most often within the muscle cone. There is no predilection for race or ethnicity.[5] It usually affects females. The prognosis after the complete excision of the tumor mass is excellent. Recurrence is rare.[6]
| Case Report | |  |
A 45-year-old female patient presented to the eye hospital with painless swelling of the lateral wall of the right orbit [Figure 1]a and [Figure 1]b, which was gradually progressive in size for 6 years. She had no known past medical or ocular history. Her past medical reports revealed that she underwent digital subtraction angiography and transarterial embolization procedure for a giant hemangioma of the liver 5 years ago. She was not on any medications. | Figure 1: (a) Clinical picture of the patient with swelling over right zygomatic region. (b) Preoperative three dimensional CT showing a bulge in the zygomatic bone. (c) Coronal section showing lesion with rounded density in the lateral wall of the right bony orbit.(d) Intraoperative picture of surgical approach of lateral orbitotomy with osteotomy. (e) Gross specimen – external surface grey brown with honey comb appearance. (f) HPE picture showing sinusoidal spaces lined by flattened endothelium and thick fibrous wall and at places, separated by bone trabeculae with osteocytes
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On physical examination, the patient's vision was 20/20 in the right eye and 20/20 in the left eye. Intraocular pressure was 14 mmHg OD and 15 mmHg OS, and there was no relative afferent pupillary defect. There was no evidence of proptosis or ocular motility restriction. On slit-lamp examination, both eyes showed normal ocular surfaces. A fixed, non-tender, hard, mass was palpable in the lateral wall region of the right orbit with no retrobulbar resistance. 5-0 Vicryl sutures were in-situ on the skin due to an incisional biopsy performed 21 days ago. Fundus evaluation was unremarkable.
A computed tomography (CT) scan showed a well-defined mass in the frontal part of the zygomatic bone of the orbit, showing homogenous hyperdensity. After obtaining informed written consent, an excisional biopsy was performed via lateral orbitotomy incision approach [Figure 1]d. A defect measuring 6 × 3 × 3 cm was noted in the lateral orbital wall after the excision of the mass, which was filled with bone graft obtained from the right iliac crest region.
The gross specimen appeared gray-brown, hard in consistency measuring 3 × 2 × 2 cm. The histopathological report revealed the presence of large cavernous blood-filled spaces lined by flattened endothelium and variable fibrous wall thickening. These channels were in between lamellar bone trabeculae. The final diagnosis of cavernous hemangioma of the zygomatic bone was established [Figure 1]f.
Her postoperative course was uneventful. At 6 months follow-up, no clinical features of local recurrence were noted [Figure 1]c.
| Discussion | | |
Our case is a cavernous hemangioma of bone arising from the frontal part of the zygomatic bone with no extra orbital extensions. Cavernous hemangioma of bone can sometimes affect the orbit by the involvement of the bony wall, causing proptosis or compression of apical structures.[7]
According to Moore,[8] CT radiographic appearances are often described as honeycombs, soap bubbles, or sunbursts. Although benign tumors do not have classic radiographic features, and hence, can be misinterpreted. CT scan remains the gold standard imaging modality for the diagnosis of bony hemangiomas. Histopathology provides the confirmative diagnosis and helps in planning further management. Histologically, the lesions are encapsulated and are composed of large cavernous spaces containing red blood cells.[1]
Treatment consists of surgical excision if the lesion compromises ocular function or optic nerve compression as evidenced by visual field defects, optic nerve swelling, and pallor. Additional indications include diplopia, significant proptosis, or significant growth for cosmetic purposes.
Although they are non-high-flow tumors, significant bleeding may occur during excision because the vessels are encased in bone and do not retract or respond to cautery in a normal fashion. Preoperative embolization using endovascular techniques allows the safe removal of the lesion without significant bleeding. When these lesions involve either the roof or posterolateral wall of the orbit, it may be necessary after resection to reconstruct the bony defect to avoid postoperative orbital pulsation or masticatory oscillopsia. There is no role in radiotherapy. The prognosis is excellent for vision and life.[9]
Although radical resection with a margin of healthy bone has been shown to lower recurrence rates, a partial resection should be considered for facial hemangiomas because of the cosmesis factor and also minimize morbidity. Proposed that a trial of partial resection be undertaken because a satisfactory cosmetic result can be achieved with less morbidity.[10]
Cavernous hemangioma of bone is a rare tumor. Zygomatic bone involvement is extremely rare, and only 23 cases have been reported in the English language literature [Figure 1]e.[11]
| Conclusion | | |
Cavernous hemangioma of the bone has rarely been reported in the literature. However, it is an important differential diagnosis for bony orbital tumors. The preferred treatment is complete excision of the tumor with bone graft and confirmation the diagnosis by histopathology. Consequently, the diagnosis is most often made after surgical excision.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient (s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initial s will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
| 1. | Prajna VN. Peymans Principles and practice of Ophthalmology 2 nd edition. 2019, p.1706.  |
| 2. | Bommie FS, KyoJoon Kangaroos. Skeletal cavernous hemangiomas of the frontal bone with orbital roof and rim involvement. Arch Craniofac Surg 2018;19:214-7. |
| 3. | Pank BH, Hwang E, Kern CH. Primary Intraosseoushemangioma in frontal bone. Arch Plast Surg 2013;40:283-5. |
| 4. | Alejandra AS, Natalia Frade P, Manuel PS. Intraosseous cavernous hemangioma: Presentation of clinical case. Egypt J Neurosurg 2018;22.doi: 10.1186/s41984-018-0018-3. |
| 5. | Albert and Jakobiec Principles and Practice of Ophthalmology. Azar Gragoudas . 3 rd ed: vol 1; p. 3012 |
| 6. | Almeida JA, Gellen PVB, Hiramatsu DM, Santos MAD, Bitencourt L, Marceliano EFV, et al. Cavernous Hemangioma in the Orbital Cavity: Case Report. Eur J Dent 2022;16:230-3. |
| 7. | Jack Rootman. Orbital Surgery a conceptual approach. 2 nd ed: 2013; P. 408. |
| 8. | Moore SL, Chun JK. Intraosseoushemangioma of the Zygoma: CT and MR findings. Am J Neuroradiol 2001;22:1383-5. |
| 9. | Myron Yanoff ,Jay S Ducker. Ophthalmology. 6 th; 1327. |
| 10. | Cheng KE, Marcinow AM, Provenzano MJ, Gurgel RK. Primary intraosseous cavernous hemanioma of the Zygoma: A case report and literature review. ENT Ear Nose Throat J 2012;91:210-5. |
| 11. | Hishiyama J, Isago T, Ito H. Intraosseoushemangioma of the zygomatic bone. JPRAS Open 2015;6:5-10. |
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