CASE REPORT
Propranolol for extensive hemangiomas of infancy: two case
reports
Hemangiomas extensos da infância tratados com propranolol: relato de dois casos
Luíza Helena dos Santos Cavaleiro1, Fernanda de Oliveira Viana1, Deborah Aben Athar Unger2, Maraya de Jesus Semblano
Bittencourt3
Abstract
Hemangiomas are the most common benign tumors of childhood. They show rapid growth, followed by a regression phase that culminates in the
partial or total disappearance of the lesion. Therapeutic options should be evaluated for extensive cases. Systemic glucocorticoids are the therapy
of choice; however, there are reports that propranolol offers better and faster results. We report two cases of large volume infantile hemangioma
associated with functional limitation and aesthetic disfigurement, treated successfully with propranolol, a drug that comes as a therapeutic option
providing satisfactory and maintained results, with few side effects.
Keywords: Hemangioma; propranolol; therapeutics; adrenal cortex hormones.
Resumo
Hemangiomas são os tumores benignos mais frequentes da infância, apresentando como história natural crescimento rápido, seguido de uma
fase de regressão que culmina com o desaparecimento parcial ou total da lesão. Opções terapêuticas devem ser avaliadas para casos extensos. Os
glicocorticoides sistêmicos são a terapia de escolha; contudo, há relatos de que o propranolol oferece resultados melhores e mais rápidos. Este trabalho
descreve dois casos de hemangioma infantil de grande volume associados à limitação funcional e desfiguração estética com significativa resposta ao
propranolol, droga esta que surge como uma proposta terapêutica oferecendo resultados satisfatórios e mantidos, com poucos efeitos colaterais.
Palavras-chave: Hemangioma; propranolol; terapêutica; corticosteroides.
Introduction
Hemangiomas are the most common benign tumors of
infancy, with an incidence varying from 1% in newborns to
12% in children in the first year of life. They develop by the
uncontrolled proliferation of vessels, grow fast during the
first months of life and have a regression phase which ends
in partial or total disappearance1,2. Although spontaneous
regression makes expectant management an option, other
therapies must be considered in cases of large infantile hemangiomas that cause compression of vital structures, present visceral involvement, disfigurement, or are located at
a body orifice3,4. In such cases, systemic glycocorticoids are
the treatment of choice: oral prednisone or prednisolone
2 to 4 mg/kg/day or pulse methylprednisolone1. However,
some reports state that propanolol may provide better and
faster results in the treatment of extensive hemangiomas3-5.
In this paper, we describe two cases of infantile hemangioma that responded significantly to propanolol treatment.
Case report
Case 1
A three-month-old female infant presenting cleft lip
and cleft palate. Dermatological examination showed extensive erythematous scaly lesions extending over the right hemiface, bilateral mandibular and cervical regions.
Resident at the Service of Dermatology of Universidade Federal do Pará (UFPA), Belém (PA), Brazil.
Master’s degree in Tropical diseases at UFPA; Adjunct Professor at Departament of Dermatology of UFPA, Belém (PA), Brazil.
Master’s degree in Tropical diseases at UFPA; Physician at Fundação Santa Casa de Misericórdia do Pará, Belém (PA), Brazil.
Conflit of interest: nothing to declare
Submitted on: 09.01.2011. Accepted on: 28.04.2011
J Vasc Bras. 2011;10(2):173-176.
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Due to the involvement of the right periorbital region, the
patient was unable to open her eye, and the lesions on the
cervical region were obstructing the upper airways (Figure
1). She was treated with prednisone 1 mg/kg/day for one
month, but without improvement. After cardiology assessment, propanolol 2 mg/kg every 8 hours was introduced
with rigorous monitoring of arterial blood pressure, heart
rate and glycemic levels. The patient presented significant
improvement after one week of treatment; The lesion showed regression and became more plane, which enabled the
patient to open her right eye normally (Figure 2). Despite
clinical improvement of hemangioma, the patient died of
septic complications.
Case 2
Four-month-old female infant presenting with erythematous papules on the right preauricular regions, that had
appeared at 20 days of life. The lesions had progressive
growth and similar ones appeared. Dermatological examination showed erythematous scaly lesions of smooth consistency on the right frontal, right periocular, preauricular,
mandibular and inferior lip regions (Figures 3A-B). After
cardiologic assessment, she was given propanolol 2 mg/
kg every 8 hours, with dosage increase one month later, to
3 mg/kg every 8 hours. The lesions disappeared after four
months of treatment (Figures 4A-B). Laboratory exams did
not show alterations.
Discussion
Figure 1 - Scaly lesion on the inferior third of the face obstructing upper
airways and eye opening at the right side.
Propranolol for infantile hemangiomas - Cavaleiro LHS et al.
It is believed that only 10 to 20% of hemangiomas require treatment. Indication of treatment is made in case
of eye, upper airways and auditory canal obstruction, as
well as those that cause heart failure and hemorrhage, and
those that may turn into ulcers and disfigure the patient.
Systemic corticoids are the treatment of choice for extensive hemangiomas1,2. They are usually given to patients
in high dosages and for long periods, which makes the
patient more prone to adverse effects such as hirsutism,
cushingoid facies, hypertrophic cardiomyopathy, gastrointestinal, sleep and growth disorders2,6. Propanolol, a
nonselective beta-blocker, has been pointed out as an alternative in more aggressive cases3,4,5,7, for its effects include: vasoconstriction immediately perceptible as a color alteration associated with palpable softening of the lesions,
decrease in vascular endothelial growth factor expression
(VEGF) and basic fibroblast growth factor (bFGF) due to
protein kinase inhibition, which explains the progressive
improvement of hemangioma; and capillary endothelial
cell apoptosis deflagration. This drug should be used in
the proliferative phase, in which pro-angiogenic factors,
VEGF and bFGF are involved3. There is no consensus
in literature about the right dosage. It is recommended
to initiate the treatment with 0.16 mg/kg every 8 hours,
which may be increased to 2 mg/kg/day3. The use of this
medicine is not risk-free, and the patient may hypotension, bradycardia and asymptomatic hypoglycemia, so
the patient must be previously assessed as to cardiologic
features and have their arterial blood pressure, heart rate
and glucose levels closely monitored4. The patients of the
Figure 2 - Aspect of the lesion after propanolol (fourth day).
Propranolol for infantile hemangiomas - Cavaleiro LHS et al.
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B
Figure 3 - Erythematous scaly papules of smooth consistency on the right frontal, right periocular, preauricular, mandibular and inferior lip regions (A
and B).
A
Figure 4 - Lesion aspect after propanolol (A and B).
B
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cases described had no adverse effects to the drug, hence
it may be considered a promising alternative for extensive
hemangioma treatment with fast response.
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Correspondence:
Maraya de Jesus Semblano Bittencourt
Travessa Vileta, 1.289, apto 302
CEP: 66087-422 – Belém (PA), Brazil
E-mail: [email protected]
Author’s contribution:
Study conception and design: LHSC, FOV, DAAU, MJSB
Data analysis and interpretation: LHSC, FOV, DAAU, MJSB
Data collection: LHSC, FOV, DAAU, MJSB
Writing: LHSC, FOV, DAAU, MJSB
Critical analysis: LHSC, FOV, DAAU, MJSB
Final approval*: LHSC, FOV, DAAU, MJSB
Statistical analysis: N/A
Overall responsibility: MJSB
*All authors have read and approved the final version submitted to J Vasc Bras.