Success in the use of oral propranolol in the treatment of infantile hemangioma in nasal tip - Report of two cases,

AnBrasDermatol.2020;95(2):207---209

Anais

Brasileiros

de

Dermatologia

CASE

REPORT

Success

in

the

use

of

oral

propranolol

in

the

treatment

of

infantile

hemangioma

in

nasal

tip

---

Report

of

two

cases

夽,夽夽

Mariana

Carvalho

Costa

_,

_Odil

_Garrido

_Campos

_de

_Andrade

_,

Lethícia

de

Castro

Pereira

_,

_Izelda

_Maria

_Carvalho

_Costa

a_{ServiceofDermatology,HospitalUniversitáriodeBrasília,Brasília,DF,Brazil}

b_{FaculdadedeCiênciasdaSaúdeeEducac¸ão,CentroUniversitáriodeBrasília,Brasília,DF,Brazil}

Received15March2019;accepted19May2019

Availableonline21January2020

KEYWORDS

Hemangioma; Infant; Propranolol

Abstract Infantilehemangiomaisthemostcommonpediatricvasculartumor,withthe

fol-lowingriskfactors:lowbirthweight,prematurity,whiteskin,femalegender,multiparityand

advancedmaternalage.Theuseoforalandtopicalbeta-blockers,althoughrecent,hasemerged

asthefirstlineoftreatment,withsuperiorsafetyandefficacytopreviouslyusedtherapies,

such as corticosteroidsandsurgeries.This reportdescribes twocases ofnasal tipinfantile

hemangioma,treatedwithoralpropranolol.Bothpresentedexcellenttherapeuticresponses.

©2020SociedadeBrasileira deDermatologia.PublishedbyElsevierEspa˜na,S.L.U.Thisisan

openaccessarticleundertheCCBYlicense(http://creativecommons.org/licenses/by/4.0/).

Introduction

Infantile Hemangioma (IH) is the most common pediatric vascular tumor, affecting mainly the nasal tip and head, with incidence of 3% to 10%. Its pathogenesis has yet to beclarified.Relevantriskfactorsincludelowbirthweight,

夽 _{Howtocitethisarticle: CostaMC,AndradeOGC,PereiraLC,}

CostaIMC.Successintheuseoforalpropranololinthetreatment ofinfantilehemangiomainnasaltip---Reportoftwocases.AnBras Dermatol.2020;95:207---9.

夽夽_{Study conducted at the Hospital Universitário de Brasília,}

Brasília,DF,Brazil.

∗_{Correspondingauthor.}

E-mail:odilgca@hotmail.com(O.G.Andrade).

prematurity,whiteskin,femalegender,1---3 _{multiparityand}

advancedmaternalage.1---3

The clinical course of IH is divided into three phases. First, the rapid phase is characterized by excessive pro-liferation shortly after birth. Ulcerations, bleeding and obstruction of vital structures can also occur during this phase,especiallyin preterm infants. Second, the sponta-neousregression phase---complete or partial---involves a higherriskofdeformitiesandulceration.Finally,theplateau phase(inwhichthereisnoregressionorevolution)arisesin approximately10%ofcases,requiringtherapeutic interven-tion,asthereisariskofpermanentdeformation.1---3

This reportdescribes twocases ofnasal tipIHtreated withoral propranolol. Bothshowed excellent therapeutic responses.

https://doi.org/10.1016/j.abd.2019.05.005

0365-0596/©2020SociedadeBrasileiradeDermatologia.PublishedbyElsevierEspa˜na,S.L.U.ThisisanopenaccessarticleundertheCC BYlicense(http://creativecommons.org/licenses/by/4.0/).

208 CostaMCetal.

Figure 1 Before the treatment with oral propranolol,

6-month-oldinfant.

Figure 2 Before the treatment with oral propranolol,

2-month-oldinfant.

Case

report

Six-month-old infant,female, presented an erythematous violaceousvascularlesionwithincreasedlocalvolumeatthe nosetip,compatiblewithIH(Fig.1).The lesionappeared inthe firstdaysof life asa slightlyerythematous macule and manifested progressive enlargement. Born Appropri-ateforGestationalAge(AGA),full-terminfant,multiparous mother,withoutcomplicationsduringpregnancyor impair-mentstopsychomotordevelopment.

Two-month-old infant, female, presented a violaceous vascularlesionat thenasaltip,surpassingthe anatomical limits,compatiblewithIH(Fig.2).The lesionappearedin the first days of life as a slightly erythematous and vio-laceous macule and manifested progressive enlargement. AGA, preterm birth (36 weeks), primiparous mother, in vitro fertilization,with threatof placental abruption and interruptionduetooligodramnium,withoutchangesin neu-ropsychomotordevelopment.

Inbothcases,patientsunderwentcardiologic,pulmonary andlaboratorialtests,whichdidnotrevealanyalterations. Inrelation tothe therapy,the target dose of propranolol was 2mg/kg/day, bid during meals (first 2 weeks with 0.5mg/kg/dayandafortnightlyprogressiveincrease)over 12months. The patients werecarefully monitored before treatmentandwhenthedrugdosewasincreased.

Theinfantsdevelopednoneofthereportedsideeffects --- sleepdisturbances, hypoglycemia or hypotension--- and

Figure3 Afterthetreatmentwithoralpropranolol,

9-month-oldinfant.

Figure4 Afterthetreatmentwithoralpropranolol,

5-month-oldinfant.

achieved totalinvolution without deformities or residuals scars, demonstrating the efficacy of oral propranolol and itsadvantages in relation tocorticosteroids. The patients didnot present recurrencein the3 monthsthat followed (Figs.3and4).

Discussion

Inviewofpossiblecomplications,effectivetreatment with-out side effects is necessary. Formerly, for IH located in the nasal tip, systemic corticosteroids, laser therapy and surgical excision were the most common therapeutic options to delay or interrupt the growth of the vascular tumorandenableitsremoval.Highdosesofcorticosteroids during prolonged periods are frequently accompanied by several side effects, particularly in children, such as decreases in bone formation and increases in its absorp-tion, proximal weakness and reduction of bactericidal activity, facilitating infections. In addition, consequences can include increasedhepatic gluconeogenesis, increased insulin resistance, psychosis and mood changes. Equally, laser therapy andsurgical removalcanlead tounesthetic scars.4---6

Hence,theuseofeffectivedrugswithfewersideeffects isparamount.Beta-blockers,specificallypropranolol, there-forebecamethefirstlineoftherapyandwereapprovedby theFoodandDrugAdministrationin March2014.However theuseinBrazilisnotyetwidespread.6,7

Beta-Blockers(BB)arebeta-adrenergicantagoniststhat generate systemic actions, including reductionof cardiac

Successintheuseoforalpropranololinthetreatmentofinfantilehemangiomainnasaltip---Reportoftwocases 209 outputthroughadecreaseinsinoatrial nodeactivity.This

provokes hypotension, causing reflex peripheral vasocon-striction,whichisbelievedtoberesponsibleforinvolution ofthe hemangioma.Furthermore,BBscangenerate bron-choconstriction,increasesinsodiumretentionandplasma volume secondary to the reduction in renal perfusion, as well as decreased glycogenolysis and glucagon secretion, predisposing to hypoglycemia. Specifically concerning the treatment of IH, BBs have been linked to hypoglycemia, nightmaresandhypotension.3,6,8

Regarding therisksinvolved,itis necessarytoperform cardiologic and pulmonary evaluation before treatment. Initially, an Electrocardiogram (ECG) must be requested and BloodPressure (BP), Cardiac Frequency (CF) must be checked,combinedwithdetailedcardiologicandpulmonary physical examination. At the beginning of treatment (the first dose or upon an increase in the dose) the following shouldbecheckedevery hourduring the4h period: tem-perature,CF,respiratoryrateandpulmonaryauscultation. ManyauthorsrecommendthatECGandcapillaryglycemia mustalsobeperformedpriortoadministration,and120and 240min thereafter, thus avoidingthe risk ofhypotension, bronchospasmandhypoglycemia.3,8,9

Compared to corticosteroids, BBs induce a faster response, with a response rate of around 90%. More-over, there is a lower risk of relapse and a decreased needforsurgicalintervention.3,8,10 _{Inourexperience,}

pro-pranolol has resulted in excellent therapeutic response and safety, as exemplified by the two aforementioned cases.

Financial

support

Authors’s

contributions

Mariana Carvalho Costa: Approval of the final version of the manuscript; conception and planning of the study; elaborationandwritingofthemanuscript;obtaining, anal-ysis,andinterpretationofthedata;effectiveparticipation in research orientation; intellectual participation in the propaedeutic and/or therapeutic conduct of the studied cases; critical review of the literature; critical review of themanuscript.

Odil Garrido Campos deAndrade:Approvalof thefinal versionofthemanuscript;conceptionandplanningof the study;elaborationandwritingofthemanuscript;obtaining, analysis,andinterpretationofthedata;criticalreviewof theliterature;criticalreviewofthemanuscript.

LethíciadeCastroPereira:Approvalofthefinalversionof themanuscript;elaborationandwritingofthemanuscript; criticalreviewofthemanuscript.

Izelda MariaCarvalhoCosta: Approval of thefinal ver-sion of the manuscript; elaboration and writing of the manuscript;obtaining, analysis, andinterpretation of the data;effectiveparticipationinresearchorientation; intel-lectualparticipationinthepropaedeuticand/ortherapeutic conductof thestudiedcases;criticalreviewof the litera-ture;criticalreviewofthemanuscript.

Conflicts

of

interest

References

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