To evaluate the safety and efficacy of our institutional beta-blocker protocol for treatment of complicated infantile hemangiomas IH. Based on preliminary data showing hemangioma recrudescence off-treatment, we reviewed 9 additional patients with recrudescence between August and December Propranolol appears to be associated with minor, not severe symptomatic adverse events. Propranolol appears to be effective in treating complicated IH.
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While ulceration is a common, predominantly benign complication in infantile hemangioma, little is known about the prognosis of ulcerated CH. However, it has been observed that ulcerated CH may be complicated by life-threatening bleeding episodes.
In both cases, the CHs were fed by high-flow vessels and the ensuing massive bleeding was due to superficial vessel wall erosion induced by the ulceration. Both patients were successfully treated with intravascular embolization; one patient underwent additional hemostatic surgery. Embolization is the treatment of choice in the case of severe bleeding, as the natural history of RICH is to spontaneously regress. Infantile hemangiomas IHs are the most common benign vascular tumors found in children; they appear around 2 weeks after birth, proliferate rapidly for up to 1 year, and then slowly spontaneously regress by the age of 3 to 5 years.
Congenital hemangiomas fall into 2 clinical subtypes: rapidly involuting congenital hemangiomas RICH , which tend to rapidly involute in the postnatal period so that the lesion is fully resolved by age 8 to 14 months, and noninvoluting CHs, which do not regress 3 but instead grow in proportion to the child and often require surgical resection.
Recently, a third clinical subtype based on partially involuting clinical behavior has been described. Ulceration is the most common complication of IH, 8 , 9 which can result in pain and infection. Ulceration of IH can also induce bleeding episodes that are mainly mild and benign.
In contrast, little is known about the prognosis of ulcerated CH. To our knowledge, only 3 well-documented cases of severe bleeding have been reported in patients with CH. Institutional review board approval was not required for this study. Parents of the patients provided written consent. A newborn girl was referred to our institution for a large cutaneous congenital vascular tumor. She was born at term by vaginal delivery after an unremarkable pregnancy.
At birth, she had a telangiectatic violaceous protuberant tumor 5 cm in diameter on her right knee Figure 1 A. A small central ulceration with a crust was observed. She also had a superficial veinous ectasia at the inner edge of the thigh with a vibratory sensation felt on the skin at palpation, suggesting a fast-flow tumor.
A, Exophytic violaceous tumor with telangiectasias and central ulceration, at birth. B, Evolution at age 4 months. Findings from color Doppler ultrasonography revealed a moderately fast-flow vascular tumor with a large dilatation of superficial veins, in particular the right saphenous vein with another dilated branch. There was no visible shunt or abnormal vessels. Findings from magnetic resonance imaging showed a mass with T2 high-intensity signals associated with arteriovenous shunts, tortuous veins, and flow void.
Results of echocardiography were normal, and the patient did not have thrombocytopenia or coagulopathy. Results of cutaneous biopsy were consistent with a benign vascular tumor with lobular proliferation and large ectatic veins Figure 2. Active treatment was not initially advised. Two weeks later, the patient suddenly developed a massive hemorrhage from the crust area of the CH that led to hemorrhagic shock and necessitated transfer to the intensive care unit.
The bleeding was partially controlled using a compression dressing. At 3 weeks of life, given the persistence of the hemorrhage, we gave the patient several blood transfusions. A selective vascular embolization was then performed with coils and N -butyl cyanoacrylate with metacryloxisulfolane.
A series of arteriograms of the common femoral artery showed that voluminous pedicles were supplying the CH Figure 3. However, when the patient was almost 2 months old, the persistence of severe hemorrhagic episodes required several more transfusions.
Therefore, we decided to perform selective hemostatic surgery, which finally stopped the bleeding. At this point, the CH rapidly regressed, and by 4 months of age, only a telangiectatic scar with redundant skin remained Figure 1 B.
Femoral arteriogram performed at 20 days of life, with enlarged right common femoral artery and vein red arrowheads and intralesional arteriovenous shunts yellow arrowhead. A newborn boy was hospitalized in the pediatric ward for a hemorrhage resulting from an ulcerated CH. The boy was born at term by vaginal delivery after an unremarkable pregnancy.
He had a 3. The lesion was surrounded by a halo-like rim of pallor with a large central ulceration Figure 4. Telangiectatic purplish tumor on the upper left thigh, halo-like rim of pallor, and large central ulceration.
Simple occlusive dressings were initially used for treatment. At 10 days of life, massive pulsatile bleeding occurred during the replacement of the dressing.
Findings from the computed tomographic angiography revealed a 3. Drainage occurred through the large veins into the left external iliac vein by the saphenous vein. However, because of the persistent episodes of massive bleeding that required several transfusions, a selective vascular embolization was performed when the patient was 3 weeks old, first using alcohol and then using microbeads. After this treatment, the bleeding quickly became less abundant, and the evolution of the tumor was favorable.
The CH regressed, and the bleeding episodes stopped. The ulceration healed completely within 2 months. By 1 year of age, although the CH continued to regress, a purplish swelling still persisted, with central lipoatrophy and a vascular mass seen on magnetic resonance imaging. The parents refused surgical excision of the remaining lesion. We report 2 neonatal cases of ulcerated CH that were complicated by life-threatening hemorrhagic episodes. While IHs are frequent vascular benign tumors in children, CHs are rare.
RICH are violaceous tumors, firm to hard on palpation with telangiectasias on the surface, surrounded by a halo-like rim of pallor Figure 4. The most frequently involved sites are the head and the limbs near a joint.
Infantile fibrosarcomas may display similar clinical and radiologic features; therefore, a biopsy is recommended to confirm the diagnosis if there is any doubt. However, diagnosis is more frequently made in the second trimester of pregnancy, 12 as in our second case. In our first case, even though the tumor at birth was very large, it was not diagnosed during antenatal ultrasonography. This could be because these tumors grow extremely rapidly during the later stages of pregnancy. Although very large CHs may be complicated by cardiac failure, thrombocytopenia, or coagulopathy, 13 these lesions are usually benign, and no treatment is recommended.
Ulceration is a common, predominantly benign complication in IH and bleeding seems to occur only rarely and most often is moderate. Some case reports have suggested that ulcerated CH can lead to massive hemorrhages, 1 , 10 , 14 , 15 but the risk factors for this severe complication are unknown. Although some cases of life-threatening hemorrhages have also been described in ulcerated IH, especially in segmental IH, the data reported in the literature are sometimes too unclear to differentiate between true IH cases and misdiagnosed CH cases.
Massive bleeding in ulcerated CH may be because of the erosion of the large superficial vessel that is associated with this tumor. As with IH, CHs are vascular tumors with a high blood flow.
However, CH differs from IH based on the presence of the larger vessels that are seen in imaging test results. In our first case, as in previous reports, 10 , 14 , 15 a large superficial vessel was eroded and resulted in high-flow hemorrhaging. It is unknown whether ulcerated CH localization or subtype could be a risk factor for severe hemorrhaging, but it is remarkable that our 2 cases and 2 of 3 other well-documented cases in the literature 1 , 10 were of the RICH subtype, located on the thigh, and vascularized by the femoral fast-flow vessels.
Therefore, Doppler ultrasonography, magnetic resonance imaging, or computed tomographic angiography should be discussed early in cases of ulcerated RICH to evaluate the size and depth of the vessels involved, as well as their distance to the ulcerated area. Powell et al 1 also propose to treat severe bleeding episodes caused by CH with tranexamic acid, an antifibrinolytic agent that can be used topically and helps to stabilize the clot. In contrast to cases of IH, 9 propranolol treatment is now known to be ineffective in reducing CH volume, as our 2 cases clearly illustrated.
Furthermore, because propranolol can be harmful for hemodynamically unstable children, this treatment should be avoided in children with severe bleeding that can occur in ulcerated CH.
Although it is a rare complication, neonates with ulcerated CH are at risk for severe hemorrhaging. Close clinical monitoring is recommended for cases of CH with ulceration, even when the ulceration is small.
Early Doppler ultrasonography, magnetic resonance imaging, or computed tomographic angiography should be used to visualize the subcutaneous vessels and assess the possible damage. Embolization should be the treatment of choice in case of severe bleeding as the natural history of RICH is to spontaneously regress. Published Online: January 7, Author Contributions: Dr Barbarot had full access to all the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Mss Vildy and Macher contributed equally to the present work. Conflict of Interest Disclosures: None reported. She was compensated for her contribution.
View Large Download. Figure 2. Biopsy Stain, Case 1. Figure 3.
Analysis of Beta-Blocker Instituted for Treatment of Hemangiomas (RABBIT Study)
We performed a retrospective case series of IHs with prolonged growth to further characterize these lesions and their treatment. Recognition of this subset of hemangiomas is important for clinicians, and further study of IHs may provide clues to their pathogenesis. Infantile hemangiomas are the most common tumor of infancy and have a well-described natural history of rapid growth during early infancy followed by gradual involution. Although there are other types of vascular tumors sometimes referred to under the umbrella term hemangiomas eg, lobular capillary hemangioma [pyogenic granuloma] , hereafter in this article the term hemangioma will refer to infantile hemangiomas specifically.
Detection of p53 and Bcl-2 expression in cutaneous hemangioma through the quantum dot technique
While ulceration is a common, predominantly benign complication in infantile hemangioma, little is known about the prognosis of ulcerated CH. However, it has been observed that ulcerated CH may be complicated by life-threatening bleeding episodes. In both cases, the CHs were fed by high-flow vessels and the ensuing massive bleeding was due to superficial vessel wall erosion induced by the ulceration. Both patients were successfully treated with intravascular embolization; one patient underwent additional hemostatic surgery. Embolization is the treatment of choice in the case of severe bleeding, as the natural history of RICH is to spontaneously regress.
Hemangioma is one of the most common types of infantile vascular benign tumor. The aim of the present study was to investigate the role of B-cell lymphoma 2 Bcl-2 and tumor protein p53 p53 in the proliferation and apoptosis of hemangioma cells. A total of 38 paraffin-embedded hemangioma specimens 16 males and 22 females and another 5 paraffin-embedded healthy surrounding tissue samples, collected between January and December , were obtained from the Department of Pathology at Renmin Hospital of Wuhan University Wuhan, China. Immunohistochemistry, hematoxylin and eosin staining, and quantum dot double staining were used to detect the expression of proliferating cell nuclear antigen PCNA , Bcl-2 and p53 in hemangioma and healthy surrounding skin tissue samples.
Read more about the Hemangioma Investigator Group here. Am J Cardiol. Mol Syndromol. Propranolol treatment of infantile hemangiomas: anticipatory guidance for parents and caretakers.