Congenital capillary hemangiomas are usually seen in females and typically appear at birth or within the first 2 months of life. They generally locate to the superior orbit and lids. But acquired capillary hemangioma (ACH) of the periocular region is very rare.
Till date, only four cases of acquired capillary hemangioma of the eyelid have been reported. Three of these (two were male, and one was a pregnant woman) were reported in adults. One was reported in a child. To the best of our knowledge, this is the first case of a woman who was not pregnant.
A 49-year-old woman visited our clinic with a complaint of a left upper-eyelid mass. The lesion first appeared 2 months earlier and had gradually increased in size. The patient did not have any history of other cutaneous lesions or antecedent trauma.
On examination, the unaided visual acuities were 20/20 for each eye. Appearance of the mass was with a smooth surface and dark brown, violet color. The mass was sessile and well demarcated. A vein had drained the mass from the right side. There were telangiectatic vessels around the periorbital region. Under the eyelid skin, vessels were appeared sharp-cut and at dark purple color. The rest of the ophthalmic and orbital examination was normal. We clinically diagnosed the lesion as pyogenic granuloma and planned an excisional biopsy. Unfortunately, we didn’t have a photo-documentation facility at that time in the hospital so we could not present preoperative photo.
Excisional biopsy of the mass was performed and hemostasis maintained. The wound was repaired primer suturing with 6-0 Vicryl. On the macroscobic examination, size and characteristic of the mass was 0.6 × 0.4 × 0.2 cm and at soft temper nodular structure. Histopathological examination of the mass showed with numerous capillary lumina lined by endothelial cells, without nuclear atypism [Fig. 1].
The patient was assessed at postoperative 1st week. The wound had healed and the sutures were removed. The previously noted under-skin vessels were still clinically evident at postoperative 1st week. These vessels became less obvious and got light violet color at postoperative 6th month control, and mass had not recurred again. Postoperative cosmetic result was very good. After histopathological diagnoses, we realized that it was a rare case, and we sent the patient to the Education and Research Hospital for photo-documentation and advanced systemic examination [Figs. 2 and 3].
The most common congenital vascular tumors of the periorbital region are capillary hemangiomas; and involution of this entity usually occur in the first decade. Acquired capillary hemangioma of the eyelid and periocular region is a very rare conditions. Six cases have been reported in the literature. Four of these six were acquired capillary hemangioma of the eyelid. Two cases were in the lacrimal sac region. Both genders have been affected by ACH of the eyelid. Two of four cases were in the female gender. One was in a pregnant woman, and the other was in a 12-year-old girl. Hormonal changes during puberty and pregnancy can cause the development of vascular lesions and also trauma, and irritative agents may cause inciting factors for capillary hemangiomas. But these were absent in our case.
The acquired form of capillary hemangioma looks like a mature type of congenital capillary hemangioma. In immature stages of the lesion, there are numerous newly formed capillaries with narrow lumina. When immature lesion getting mature, the vascular lumina becomes dilated the endothelial cells get more flattened, stroma becomes edematous and hyalinized, and blood flow establishes.
The main differential diagnoses for ACH are, pyogenic granuloma, angiosarcoma, and acquired tufted angioma of the eyelids. Acquired tufted angioma of the eyelid is also a rare entity. A recent report about tufted angioma of the eyelid was published in two patients. Clinically, tenderness, hypertrichosis, and induration are useful signs in differentiating tufted angioma from hemangioma. Histopathologically, angiomatous lobules of tufted angioma are composed of relatively bloodless and poorly canalized capillaries which are lined by plump endothelial cells. Tufted angioma in childhood needs differentiation from juvenil hemangioma. Angiomatous lobules of juvenile hemangiomas are more massive, and lesions tend to have involvement of deeper periocular structures at later stages.
Congenital capillary hemangiomas usually regress spontaneously, especially immunohistochemically bcl-2 negative lesions more prone to spontaneous involution. Bcl-2 positivity is inversely correlated with the regression in infantile hemangiomas. However, in ACH, results of surgical excision were excellent. In our case, we performed total excision. After resection, a venule which had drained the capillary hemangioma became indistinct. The vessels around the mass which were obvious became less manifest at postoperative 6th month [Figs. 2 and 3].
To the best of our knowledge, this is the first ACH of the eyelid case whose age was over 40, and the first case of a woman who was not pregnant.
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Conflict of Interest: None declared.