Parental experiences with propranolol versus oral corticosteroids for complicated infantile hemangioma, a retrospective questionnaire study.

BACKGROUND: Infantile hemangiomas (IHs) are common and mostly emerge in the head-neck area. Recently, propranolol has been replacing oral corticosteroids (OCS) as the main treatment modality. OBJECTIVES: The aim of this study was to explore the impact of treatment, contentment with treatment outcome and quality of life for families and patients with cervicofacial IHs, treated with propranolol versus OCS. MATERIALS AND METHODS: This study was performed using questionnaires administered by a phone interview. Parents of 16 patients with a cervicofacial IH treated by OCS and 16 patients with an IH of similar localization and overall severity treated with propranolol were interviewed. The questions concerned the impact of treatment at different time periods and the contentment with treatment results. Parents were also asked to give a quality of life (QoL) score (1 to 10) for different time-points. RESULTS: Parents from the OCS group seemed to feel significantly more worried during treatment. Moreover, parents from the propranolol group perceived less negative impact on normal life issues, including work and vaccination of their child. During and after treatment, the parents of propranolol-treated IH patients gave significantly higher QoL scores. CONCLUSION: Propranolol seems to change the impact of IHs, their treatment and the quality of life. Propranolol treatment interferes less with normal issues in daily life, compared to OCS. These findings underline propranolol as the first choice treatment for life- or function-threatening IHs.

Propranolol treatment in life-threatening airway hemangiomas: a case series and review of literature.

OBJECTIVES: Infantile hemangiomas (IHs) in the airway may be potentially life-threatening during the proliferative phase. Available treatments like oral corticosteroids (OCS) and chemotherapeutic agents usually showed variable responses and serious side effects. Propranolol is a new and promising treatment option. METHODS: A case series of five IH patients with airway involvement is presented, supplemented with a review of literature. Propranolol treatment (2.0-3.0mg/kg/day) was initiated between 3 weeks and 6 months of age. Three cases were treated with propranolol monotherapy, 2 cases with OCS primarily and propranolol secondarily, in which treatment with OCS could be reduced rapidly. RESULTS: In our case series a dramatic, fast response was observed in all cases, with a permanent effect after discontinuation in four cases. In one patient a relapse of airway problems occurred two months after discontinuation of propranolol at 16 months of age; this resolved after re-start of propranolol. Review of literature together with these five cases showed 81 patients with airway IHs treated with propranolol. Propranolol was effective in 90% of the cases and seven patients were classified as non-responders. Eight IHs relapsed while weaning of propranolol or after discontinuation; dose adjustment or restart was effective in most cases but one patient appeared resistant to therapy. CONCLUSIONS: Propranolol seems to be a rapidly effective and safe treatment strategy for most IHs obstructing the airway. Because of the fast and important effects of propranolol, randomized controlled trials are hardly justifiable for this specific, relatively rare but, acute treatment indication. Despite the efficacy of propranolol, close monitoring of the patients with an airway IH is required, considering the risk of relapse of symptoms during or after treatment and the reported resistance to propranolol in at least 9% of the published cases. The dose and duration of treatment should be high and long enough to prevent relapse. Further research should focus on the optimal treatment protocol; the actual percentage of non-responders and also the mechanism of resistance to propranolol is unknown and needs to be illuminated.

Kaposiform hemangioendothelioma with Kasabach-Merritt syndrome: a new indication for propranolol treatment.

Kaposiform hemangioendothelioma is a rare vascular tumor in children. Especially, in association with the Kasabach-Merritt Phenomenon it can be life threatening. The management of these patients is very difficult and an aggressive treatment regime is required. Several multimodality and chemotherapeutic regimens have been described but with variable success and many side effects. We present a 6-week-old boy with Kaposiform hemangioendothelioma and Kasabach-Merritt Phenomenon. Ongoing propranolol treatment with only 4 initial courses of vincristine resulted in a remission that lasted at least 1 year.

Propranolol, a very promising treatment for ulceration in infantile hemangiomas: a study of 20 cases with matched historical controls.

BACKGROUND: Ulceration is a common but poorly understood complication of infantile hemangiomas (IH) that is difficult to control. OBJECTIVE: To investigate the possible role of monotherapy with propranolol for ulcerating IH. METHODS: Propranolol was given to 20 patients with IH, who suffered from ulceration at the start of treatment (mean age at onset of treatment, 3.5 months; standard error of the mean: 0.4). After cardiac screening, propranolol was administered in a progressive schedule to 2 to 2.5 mg/kg per day, divided in 3 doses. Blood pressure, heart rate, and fasting glucose levels were monitored during the first 3 days in hospital and, in the absence of complications, treatment was continued at home until the age of approximately 1 year. The 20 propranolol-treated patients were matched to patients from a historical control group, seen before the 'propranolol era'. These matches were randomly made by using clinical pictures based on type, location and size of the IH, extent of ulceration, and age at the start of ulceration. RESULTS: The time to complete healing from the onset of ulceration was significantly shorter for the propranolol-treated patients, compared with the control group (8.7 vs 22.4 weeks; t test: P < .015). In the propranolol group, a tendency to shorter ulceration duration was seen in patients starting propranolol at an earlier stage of disease. LIMITATIONS: The study was limited by the partially retrospective design and the small number of patients. CONCLUSION: Propranolol reduces the duration of ulceration in IH and seems to be more effective when started in an early phase. We propose propranolol as the treatment of first choice for ulcerating IH.

Differences between ulcerated and non-ulcerated hemangiomas, a retrospective study of 465 cases.

Our purpose was to get better insight into the ulceration of hemangiomas, by comparing patient characteristics of non-ulcerated hemangiomas with hemangiomas with active or past ulceration. A retrospective analysis was performed of files of patients who visited the Radboud University Medical Centre Nijmegen (UMCN), the Netherlands, between 1997 and 2007 for one or more infantile hemangiomas. The medical records of 465 patients were reviewed. Twenty three percent of the patients were diagnosed with ulceration. The size of ulcerated hemangiomas was significantly larger (28.6 cm2 vs. 6.0 cm2, p < 0.05). Predilection areas for ulceration were the head-neck region and the anogenital region. Ulceration was significantly most frequently seen in hemangiomas with a superficial (epidermal) component (98.5%, p < 0.05) and a segmental distribution (29.3%, p < 0.05). Ulceration most frequently took place during the proliferation phase of the hemangioma (83.1%). In the whole study population the male to female ratio was 1:2 compared to a tendency to more girls (1:3) for the group with ulcerated hemangiomas (p = 0.08). We conclude that larger, more superficial hemangiomas in areas more susceptible to trauma and contamination were more likely to ulcerate. This study contributes to the possibility of assessing the likelihood of ulceration in an individual patient.