A Case of Periorbital Infantile Capillary Hemangioma Treated With Propranolol

PURPOSE: To report a case of periorbital infantile capillary hemangioma treated with propranolol. CASE SUMMARY: A 6-month-old girl visited our clinic for a mass on the right upper eyelid, which had been present since birth. A dome-shaped, red-purple colored mass occupying almost the entire right upper eyelid was observed, and overlying branch-shaped telangiectases were also detected. The visual axis of the right eye was partially obscured, due to the ptotic eyelid, and her ability to fix and follow was poor in the right eye. The patient was diagnosed with infantile capillary hemagioma. Because amblyopia in her right eye was suspected, treatment with propranolol was initiated. After 2.5 months of propranolol treatment, the hemangioma decreased in size and volume dramatically, changed from red-purple to light red color, and softened. The ptosis of the right upper eyelid improved, as well as the ability of the right eye to fix and follow. No significant adverse effects (e.g. hypotension, bradycardia, hypoglycemia, bronchospasm, or elevated liver enzymes) were observed, except transient QTc prolongation during cotreatment with ibuprofen, due to an upper respiratory infection. CONCLUSIONS: Propranolol should be considered as a treatment option for periorbital infantile capillary hemangioma. Further clinical studies are required to establish the optimal guidelines and long-term outcome.

Clinicopathological Observation of Hemangioma of Infancy: The Diagnostic Usefulness of GLUT-1 Immunohistochemical Stain / 대한피부과학회지

BACKGROUND: Hemangioma of infancy (HOI) is one of the most common tumors in childhood. Their clinical features can on occasion look like those of other vascular tumors and malformations, so the correct differentiation of HOI may not always be easy. OBJECTIVE: The purpose of this study was to review the clinicopathological features of HOIs and to particularlyexamine the usefulness of glucose transporter protein isoform-1 (GLUT-1) immunohistochemical stain (IHS) as a specific marker of HOI. METHODS: This study was conducted at the Vascular Anomaly Clinic of Kyungpook National University Hospital during 11 years (1998~2008). Sixty-two cases were selected in addition to selecting other tumors and malformation as a control group. We then reviewed their clinical features and the results of H&E and GLUT-1 IHS for making the differential diagnosis. RESULTS: The male to female ratio was 1 : 2.88. The head and neck were the most commonly involved sites. The complications included ulceration and/or bleeding (12.9%), scarring (9.7%), infection (4.8%), ocular complications (4.8%) like visual axis occlusion, and pain (1.6%). Three phases of proliferating, involuting and involuted of a HOI comprised 11.3%, 37.1% and 51.6% of the lesions, respectively, from the clinical view point and 11.3%, 48.4% and 40.3% of the lesions, respectively, from the pathological point of view. GLUT-1 was positively expressed on almost all of the HOIs (47/57, 82.5%), except for 10 HOIs of the involuted phase and the other controls. CONCLUSION: The clinicopathological findings of this study were similar with those of the previous reports. The expression of GLUT-1 by a HOI can be helpful to make the correct diagnosis from other vascular tumors and malformations.

Six Cases of Congenital Tufted Angioma / 대한피부과학회지

Tufted angioma is a rare slowly progressive vascular tumor, characterized by histopathologic findings of numerous angiomatous lobules of "cannonball" appearance within the dermis. It affects children but occurs infrequently at birth. It appears as a clinical imitator of hemangioma of infancy (HOI), so it needs to be differentiated from HOI. Clinically, lesions most often present as red to purple, indurated or nodular plaque, and are usually located on the neck, upper trunk or proximal limb. We report six cases of tufted angioma of congenital onset. We undertook immunohistochemical study using CD31, D2-40 and GLUT1 to rule out other vascular tumors or malformations, particularly HOI. These cases may demonstrate the keys of differential diagnosis between HOI and tufted angioma, which share common features clinically and rarely histopathologically.

Clinical trial of 5% Imiquimod Cream for Eleven Cases of Infantile Hemangioma / 대한피부과학회지

BACKGROUND: Infantile hemangioma is a common benign tumor with preponderance in females. Even though most of them have self-limiting course after growing phase, many parents seek more effective and less aggressive therapy. Motivated by a report on the experience of successful treatment with imiquimod, we planned to confirm the beneficial effect of imiquimod on the regression of infantile hemangioma. OBJECTIVE: To enhance the noninvasive treatment modality of infantile hemangioma. METHODS: Eleven patients with various ages were enrolled in this study. Eight female and three male patients applied 5% imiquimod cream every other day or daily. RESULTS: There was no statistically significant difference of treatment results between both sexes. The number of lesions and locations didn't alter the outcome. But, the growing phase versus regression phase affected the response to treatment significantly after two months of application. As a whole, most of the patients tolerated the imiquimod well. Three of them experienced mild scales and erosions. One showed shallow ulcers and crusts that needed medical treatments. But, by reducing the frequency of application and some dressings, all the complications were managed well and didn't hinder the therapy. There were no significant systemic adverse effects. CONCLUSION: Imiquimod is effective for infantile hemangioma regardless of site of occurrence, especially when used early in growing phase. It is noninvasive and relatively safe method. For a beneficial effect, the therapy must be continued for at least two months.

Clinicopathological Study of Hemangioma of Infancy and the Immunohistochemical Study of GLUT1 Expression / 대한피부과학회지

BACKGROUND: Hemangiomas of infancy are common, benign, pediatric tumors of endothelial cells. This is characterized by an initial phase of rapid proliferation, which is followed by slow involution, often leading to complete regression. There has been no large clinicopathological study in Korea. OBJECTIVE: The purpose of this study was to investigate the clinicopathological findings of hemangioma of infancy in Korean patients. METHODS: The clinical study included 256 patients of hemangioma of infancy. Histopathological examination of 18 cases was done with H&E staining. We also performed immunohistochemical staining with GLUT1 in 10 cases. RESULTS: The study results are summarized as follows: 1. The male to female ratio was 1: 3.1. Half of the lesions were noticed at birth. 2. The sites of predilection were head and neck (68.9%), upper extremities (11.7%), trunk (10.2%), and lower extremities (8.8%). 3. The results of clinical classification by depth of lesion were: superficial hemangioma (63.7%), deep hemangioma (20.9%), and combined hemangioma (15.4%). 4. The results of histopathological classification by the developmental phase were: proliferating phase (16.7%), involuting phase (83.3%). 5. In the immunohistochemical study using GLUT1, three out of ten cases of hemangioma were stained positively. CONCLUSION: The clinical findings of our study were identical with previous published reports. The characteristic expression of GLUT1 in hemangioma of infancy can be helpful in differentiating them from other vascular tumors and vascular malformations, and in the examination of developmental pathogenesis in hemangioma.