Artigo Original / Original Article
Malformação congénita das vias aéreas pulmonares – Experiência de cinco centros
Congenital cystic adenomatoid malformation of the lung – The experience of five medical centres
Gustavo Rocha1,
, Paula Cristina Fernandes2, Elisa Proença3, Conceição Quintas4, Teresa Martins5, Inês Azevedo6, Hercília Guimarães1
Corresponding author
gusrocha@oninet.pt
Correspondência/Correspondence to: Gustavo Rocha, Serviço de Neonatologia / Unidade Autónoma de Gestão da Mulher e da Criança, Hospital de São João – Piso 2, Alameda Prof. Hernâni Monteiro, 4202-451 Porto, Telefone / Telephone: 225095816 Fax: 225505919.
Correspondência/Correspondence to: Gustavo Rocha, Serviço de Neonatologia / Unidade Autónoma de Gestão da Mulher e da Criança, Hospital de São João – Piso 2, Alameda Prof. Hernâni Monteiro, 4202-451 Porto, Telefone / Telephone: 225095816 Fax: 225505919.
1 Serviço de Neonatologia, Unidade Autónoma de Gestão da Mulher e da Criança (UAGMC), Hospital de São João (HSJ), Faculdade de Medicina da Universidade do Porto (FMUP) / Neonatal Unit, Unidade Autónoma de Gestão da Mulher e da Criança (UAGMC), Hospital de São João (HSJ), Faculdade de Medicina da Universidade do Porto (FMUP)
2 Serviço de Cuidados Intensivos Neonatais e Pediátricos, Hospital Geral de Santo António / Neonatal and Paediatric Intensive Care Unit, Hospital Geral de Santo António
3 Unidade de Cuidados Intensivos Neonatais, Maternidade Júlio Dinis / Neonatal Intensive Care Unit, Maternidade Júlio Dinis
4 Unidade de Neonatologia, Serviço de Pediatria, Centro Hospitalar de Vila Nova de Gaia / Neonatal Unit, Paediatic Unit, Centro Hospitalar de Vila Nova de Gaia
5 Unidade de Neonatologia, Serviço de Pediatria, Hospital Pedro Hispano, Matosinhos / Neonatal Unit, Paediatic Unit, Hospital Pedro Hispano, Matosinhos
6 Unidade de Pneumologia, UAGMC, HSJ, FMUP / Pulmonology Unit, UAGMC, HSJ, FMUP
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"af0015" ] 3 => array:3 [ "entidad" => "Unidade de Neonatologia, Serviço de Pediatria, Centro Hospitalar de Vila Nova de Gaia / Neonatal Unit, Paediatic Unit, Centro Hospitalar de Vila Nova de Gaia" "etiqueta" => "4" "identificador" => "af0020" ] 4 => array:3 [ "entidad" => "Unidade de Neonatologia, Serviço de Pediatria, Hospital Pedro Hispano, Matosinhos / Neonatal Unit, Paediatic Unit, Hospital Pedro Hispano, Matosinhos" "etiqueta" => "5" "identificador" => "af0025" ] 5 => array:3 [ "entidad" => "Unidade de Pneumologia, UAGMC, HSJ, FMUP / Pulmonology Unit, UAGMC, HSJ, FMUP" "etiqueta" => "6" "identificador" => "af0030" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cr0005" "etiqueta" => "*" "correspondencia" => "Correspondência/Correspondence to: Gustavo Rocha, Serviço de Neonatologia / Unidade Autónoma de Gestão da Mulher e da Criança, Hospital de São João – Piso 2, Alameda Prof. Hernâni Monteiro, 4202-451 Porto, Telefone / Telephone: 225095816 Fax: 225505919." ] ] ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Congenital cystic adenomatoid malformation of the lung – The experience of five medical centres" ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2006-06-09" "fechaAceptado" => "2007-02-05" "PalabrasClave" => array:2 [ "pt" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palavras-chave" "identificador" => "xpalclavsec570339" "palabras" => array:2 [ 0 => "Congénito" 1 => "malformação adenomatóide cística" ] ] ] "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Key-words" "identificador" => "xpalclavsec570340" "palabras" => array:2 [ 0 => "Congenital" 1 => "cystic adenomatoid malformation" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "pt" => array:2 [ "titulo" => "Resumo" "resumen" => "<span id="as0005" class="elsevierStyleSection elsevierViewall"><p id="sp0005" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Introdução</span>: O espectro clínico da malformação adenomatóide cística pulmonar congénita (MACPC) va-ria desde lesões assintomáticas e dificuldade respiratória no recém-nascido a hidrópsia fetal.</p><p id="sp0010" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Objectivos</span>: Avaliação da casuística de MACPC, real-çando a história natural, tratamento e evolução.</p><p id="sp0015" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Material e métodos</span>: Análise retrospectiva dos processos clínicos de todos os recém-nascidos com o diagnóstico de MACPC, admitidos nas unidades de cui-dados intensivos neonatais de cinco centros terciários do Norte de Portugal, entre 1996 e 2005.</p><p id="sp0020" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Resultados</span>: Foram identificados 15 recém-nascidos, 9<span class="elsevierStyleHsp" style=""></span>F/6<span class="elsevierStyleHsp" style=""></span>M, peso ao nascimento 3100<span class="elsevierStyleHsp" style=""></span>g (645-3975), idade gestacional de 38 semanas (24-40). A incidên-cia de MACPC foi de 1:9300 nascimentos. Onze (73%) recém-nascidos apresentaram diagnóstico pré-natal de lesão pulmonar cística pelas 22 semanas (19-30). Esta ocorreu no hemitórax direito em seis (40%) casos e no lado esquerdo em 9 (60%). Foi documentada regressão <span class="elsevierStyleItalic">in utero</span> da lesão em dois casos. Foi necessária intervenção ante-natal (pleu-rocentese e colocação de derivação toraco-amnió-tica) num feto com hidrópsia fetal. Ao nascimento, cinco recém-nascidos apresentaram radiografia de tórax dentro da normalidade, sendo a tomografia axial computorizada diagnóstica da lesão. Três (20%) recém-nascidos tornaram-se sintomáticos durante o período neonatal (dificuldade respiratória) e um (7%) após o período neonatal (pneumotórax espon-tâneo). Dois (13%) faleceram. Seis (40%) doentes foram submetidos a toracotomia e exérese da lesão. O estudo anatomopatológico demonstrou acha-dos definitivos de MACPC (classificação de Stoker: tipo I<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>4; tipo II<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>1; tipo III<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>2). Oito (53%) crianças mantiveram-se assintomáticas e permane-cem em vigilância clínica.</p><p id="sp0025" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Conclusões</span>: A MACPC com diagnóstico pré-natal tem bom prognóstico na ausência de sofrimento fetal; uma radiografia de tórax sem alterações ao nascimento não exclui MACPC; o tratamento da lesão assintomática é controverso; a cirurgia está indicada devido à baixa morbilidade e possibilidade de preven-ção de complicações tardias, como a degenerescência maligna.</p><p id="sp0030" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Rev Port Pneumol 2007; XIII (4): 511-523</span></p></span>" ] "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="as0010" class="elsevierStyleSection elsevierViewall"><p id="sp0035" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Background</span>: The clinical spectrum of congenital cystic adenomatoid malformation of the lung (CCAML) ranges from asymptomatic lesions to neonatal respiratory distress and hydrops fetalis.</p><p id="sp0040" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Aim</span>: To review our experience with CCAML, emphasising natural history, management and out-come.</p><p id="sp0045" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Material and methods</span>: A retrospective chart review of all CCAML-diagnosed neonates admitted to the neonatal intensive care units of five tertiary medical centres in the north of Portugal between 1996 and 2005.</p><p id="sp0050" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Results</span>: Fifteen neonates with CCAML were identified, 9<span class="elsevierStyleHsp" style=""></span>F/6<span class="elsevierStyleHsp" style=""></span>M, birth weight 3100<span class="elsevierStyleHsp" style=""></span>g (645-3975), gestational age 38 weeks (24-40). The incidence of CCAML was 1: 9300 births. There were 11 (73%) cases of cystic lung lesion diagnosed during pregnancy, median age 22 weeks (19-30). The lesion was right sided in six (40%) and left sided in nine (60%) cases. <span class="elsevierStyleItalic">In utero</span> spontaneous regression of the lesion was observed in two cases. Antenatal intervention (pleurocentesis and thoracoamniotic shunting) was performed in one foetus with impending hydrops. Normal lung radiographic findings at birth were present in five cases, with an abnormal CT scan. Three (20%) neonates became symptomatic during the neonatal period (respiratory distress) and one (70%) after the neonatal period (spontaneous pneu-mothorax). Two neonates (13%) died. Six (40%) patients underwent thoracotomy and appropriate excisional surgery. Histological examination showed definitive features of CCAML (Stocker classification: type I<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>4; type II<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>1; type III<span class="elsevierStyleHsp" style=""></span>=<span class="elsevierStyleHsp" style=""></span>2). Eight (53%) patients remain asymptomatic and did not undergo surgery.</p><p id="sp0055" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Conclusions</span>: Antenatally diagnosed CCAML has a good prognosis in the absence of severe foetal distress; normal radiographic findings at birth do not rule out CCAML; treatment of asymptomatic CCAML is controversial; surgery may be advocated because of its low morbidity and the prevention of late complications such as malignancy.</p><p id="sp0060" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleBold">Rev Port Pneumol 2007; XIII (4): 495-523</span></p></span>" ] ] "lecturaRecomendada" => array:1 [ 0 => array:3 [ "vista" => "all" "titulo" => "<span class="elsevierStyleSectionTitle" id="st0025">Bibliografia / Bibliography</span>" "seccion" => array:1 [ 0 => array:2 [ "vista" => "all" "bibliografiaReferencia" => array:27 [ 0 => array:3 [ "identificador" => "bb0005" "etiqueta" => "1." 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Article information
ISSN: 08732159Original language: Portuguese
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