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184 Case Reports DOI: 10.1111/j.1610-0387.2009.07266.

Successful treatment of a large traces of blood on the infant’s clothing,


suggesting rapid growth and pressure.
The lesion developed central ulcerations
hemangioma with propranolol which steadily increased in size. Given
the strong tendency toward growth and
Waseem Mousa1, Kirsten Kues1, Ellen Haas1, Peter Lauerer2, the deep components of the tumor, we
Helena Pavlakovic2, Michael P. Schön1, Markus Zutt1 chose to initiate systemic therapy after
(1) Department of Dermatology, Venereology and Allergology, Georg August consultation with pediatrics.
University Göttingen, Germany Off-label systemic therapy was begun
(2) Department of Pediatric Medicine I, Georg August University Göttingen, with oral propranolol (initial dose of
Germany 1 mg /kg of body weight) after discus-
sion with the parents who gave written
consent. On the second day the dose was
increased to 2 mg/kg body weight, which
is well below that given for cardiac indi-
cations. Treatment was performed on an
inpatient basis with monitoring of heart
rate, blood pressure, electrolyte and
blood glucose levels; ECG and echocar-
JDDG; 2010 • 8:184–186 Submitted: 21. 7. 2009 | Accepted: 4. 8. 2009
diography were also performed. Therapy
was well tolerated. Two complications
Summary Introduction occurred, but were readily manageable.
Hemangiomas are the most common Hemanigiomas of infancy often have a Initial hypotension of 60/35 mmHg was
vascular tumors in children. They occur self-limiting course, but treatment may treated with i.v. volume substitution and
in 8–12 % of all infants and in 22 % of be necessary if complications arise due to did not recur. Hyperkalemia with values
premature infants (female: male = 3: 1). the affected site, growth of the lesion, or of up to 6.1 mmol/l (norm =
Hemangiomas are usually sporadic; other factors. The goal of any therapy is 3.7–5.7 mmol/l) appeared after 4 days of
their etiology is unknown [1]. to stop the growth of the lesion and therapy and was first treated with
A premature female infant, born at 28 bring about regression. furosemide 1.5 mg/kg of body weight
weeks of gestation, presented with a i.v., and later with oral furosemide
large hemangioma of the right tho- Case report 1 mg/kg of body weight as well as salbu-
racic wall. Within the first few weeks, We report on a premature infant girl tamol inhalation. The patient’s status re-
the hemangioma showed rapid hori- who was delivered in the 28th week (+2 turned to normal and accompanying
zontal and vertical growth as well as days) of pregnancy. The baby measured medication was discontinued. A superin-
ulceration, which led us to initiate 32 cm and weighed 740 g. At birth she fection of the ulcerated hemangioma was
systemic therapy. had a 3 ⫻ 4 cm large, raised, sharply- treated with intravenous antibiotics.
The effectiveness of propranolol bordered bright red tumor on the right The hemangioma responded quickly to
(non-selective ß-blocker) in the man- wall of the thorax. A clinical diagnosis therapy and stopped growing in size and
agement of severe cases of heman- was made of a cutaneous/subcutaneous thickness. After a few days the tumor be-
gioma has been shown in a recent infantile hemangioma (Figure 1). Regular came flatter and decreased in diameter.
series of cases [2]. We began oral pro- clinical examinations were performed The ulcerations also healed and there
pranolol treatment, in close interdisci- and the size of the rapidly growing tu- were areas of regression (Figure 1). Out-
plinary cooperation. After a few days of mor was documented. By the eighth patient treatment with given 8 weeks of
therapy, the tumor had stopped week of life, the hemangioma grown to oral propranolol2 mg/kg/KG 18 weeks
expanding. After 18 weeks, there has 6.6 ⫻ 8.8 cm. Imaging studies were or- ambulant on an outpatient basis was well
been marked regression but the thera- dered to assess the depth of the heman- tolerated. A follow-up duplex ultrasound
py is still being continued. We propose gioma and to rule out deeper sources after 6 weeks showed a significant de-
that propranolol may be an effective (e.g., an arteriovenous shunt) and in- crease in penetration depth and vascular-
and relatively well tolerable alternative volvement of internal organs. Color- ization (Figure 2). A follow-up MRI is
in the management of selected cases coded duplex ultrasound showed a depth planned.
of severe hemangiomas in infancy, of up to > 2 cm with extensive superficial
providing interdisciplinary coopera- and deep vascularizations (Figure 2). Discussion
tion between dermatologists and Magnetic resonance imaging (MRI) Given rapid progression and potential
pediatricians is available. showed involvement of the back muscles complications of a large hemangioma of
as well as intrathoracic components of infancy, quick and sufficient therapy is
Keywords the hemangioma (Figure 2) and a small needed. In our patient, laser (e.g.,
emangioma – propranolol – hemangioma measuring about 3 cm in ND:YAG laser along with pulsed dye
beta-blocker diameter in the liver. During the first few laser) and cryotherapy were ruled out
weeks of life, the tumor rapidly grew in due to the diameter and depth of the
size and thickness. The mother reported lesion Systemic therapy was chosen in

JDDG | 3 ˙2010 (Band 8) © The Authors • Journal compilation © Blackwell Verlag GmbH, Berlin • JDDG • 1610-0379/2010/0803
Case Reports 185

Figure 1: Clinical course. Picture above left side shows 8 week old patient before therapy, the other photos show the interval from start of therapy.

regulation of the Raf mitogen-activated


protein kinase signaling pathway with
reduced expression of vascular endothe-
lial growth factor (VEGF) and basic fi-
broblast growth factor (bFGF). VEGF
and bFGF are important angiogenic fac-
tors during the growth phase of heman-
giomas. Propranolol is also thought to
cause direct induction of apoptosis in
capillary endothelial cells [2]. Standard
recommendations on the length of ther-
apy with propranolol are still lacking.
Treatment regimens of 4–6 months have
been reported [3]. This question should
also be addressed in future randomized
studies. Presumably, therapy should be
continued until growth of the lesion has
been stopped. If after discontinuing pro-
pranolol therapy superficial heman-
gioma components remain, these can be
readily removed with combined laser
therapy. The patient should be ade-
Figure 2: MRI with intrathoracic hemangioma (a); Duplex ultrasound before therapy (b); Duplex quately monitored for any adverse effects
ultrasound after 7 weeks of therapy (c), green lines show the depth.
which must be treated promptly if they
occur, as in the patient described here.
accordance with current guideline rec- blocker) in large hemangiomas [2]. To summarize, propranolol is an
ommendations [1]. The goal of systemic There are probably various factors re- effective treatment option with rela-
therapy is to stop the expansion of the lated to the mechanism of action of pro- tively few side effects for large and
hemangioma by stopping growth and if pranolol that make it successful in the deep hemangiomas or for heman-
possible to induce regression. Pulse ther- treatment of large hemangiomas: propra- giomatosis. In infants, therapy can be
apy is generally with prednisolone nolol causes vasoconstriction and thus initiated and carried out under the co-
(2–5 mg/kg body weight daily). Vin- possible occlusion of superficial, and es- operation of the treating pediatrician
cristine or cyclophosphamide are also oc- pecially deep supplying blood vessels. and dermatologist. <<<
casionally used [1]. Within a relatively short time after start-
Recent studies have shown the effective- ing therapy, the hemangioma begins to Conflict of interest
ness of propranolol (a non-selective beta lighten. Also under discussion are down- None.

© The Authors • Journal compilation © Blackwell Verlag GmbH, Berlin • JDDG • 1610-0379/2010/0803 JDDG | 3 ˙2010 (Band 8)
186 Case Reports

References 3 Cremer H. Hämangiome Klassifizie-


Correspondence to 1 Hohenleutner U, Landthaler M, rung und Therapie-Empfehlungen.
Dr. med. Markus Zutt Hamm H, Sebastian G. Hämangiome pädiatrie hautnah 2009; 2: 133–46.
Abteilung Dermatologie, Venerologie im Säuglings- und Kindesalter. J Dtsch
und Allergologie Dermatol Ges 2007; 4: 334–9.
der Universitätsmedizin Göttingen 2 Letaute-Labreze C, Dumas de la
Von-Siebold-Straße 3 Roque E, Hubiche T, Boralive F,
D-37075 Göttingen Thambo J-B, Taieb A. Propranolol
Tel.: +49-551-39-6410 for severe hemangiomas of infancy.
Fax: +49-551-39-12811 N Engl J Med 2008; 358: 2649–
E-mail: mzutt@gwdg.de 51.

JDDG | 3 ˙2010 (Band 8) © The Authors • Journal compilation © Blackwell Verlag GmbH, Berlin • JDDG • 1610-0379/2010/0803
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