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        "resumen" => "<span class="elsevierStyleSectionTitle">Background</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">The clinical spectrum of congenital cystic adenomatoid malformation of the lung &#40;CCAML&#41; ranges from asymptomatic lesions to neonatal respiratory distress and hydrops fetalis&#46;</p> <span class="elsevierStyleSectionTitle">Aim</span><p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">To review our experience with CCAML&#44; emphasising natural history&#44; management and outcome&#46;</p> <span class="elsevierStyleSectionTitle">Material and methods</span><p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">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&#46;</p> <span class="elsevierStyleSectionTitle">Results</span><p id="spar0045" class="elsevierStyleSimplePara elsevierViewall">Fifteen neonates with CCAML were identified&#44; 9F&#47;6M&#44; birth weight 3100 g &#40;645&#8211;3975&#41;&#44; gestational age 38 weeks &#40;24&#8211;40&#41;&#46; The incidence of CCAML was 1&#58; 9300 births&#46; There were 11 &#40;73&#37;&#41; cases of cystic lung lesion diagnosed during pregnancy&#44; median age 22 weeks &#40;19&#8211;30&#41;&#46; The lesion was right sided in six &#40;40&#37;&#41; and left sided in nine &#40;60&#37;&#41; cases&#46; <span class="elsevierStyleItalic">In utero</span> spontaneous regression of the lesion was observed in two cases&#46; Antenatal intervention &#40;pleurocentesis and thoracoamniotic shunting&#41; was performed in one foetus with impending hydrops&#46; Normal lung radiographic findings at birth were present in five cases&#44; with an abnormal CT scan&#46; Three &#40;20&#37;&#41; neonates became symptomatic during the neonatal period &#40;respiratory distress&#41; and one &#40;70&#37;&#41; after the neonatal period &#40;spontaneous pneumothorax&#41;&#46; Two neonates &#40;13&#37;&#41; died&#46; Six &#40;40&#37;&#41; patients underwent thoracotomy and appropriate excisional surgery&#46; Histological examination showed definitive features of CCAML &#40;Stocker classification&#58; type I &#61; 4&#59; type II &#61; 1&#59; type III &#61; 2&#41;&#46; Eight &#40;53&#37;&#41; patients remain asymptomatic and did not undergo surgery&#46;</p> <span class="elsevierStyleSectionTitle">Conclusions</span><p id="spar0050" class="elsevierStyleSimplePara elsevierViewall">Antenatally diagnosed CCAML has a good prognosis in the absence of severe foetal distress&#59; normal radiographic findings at birth do not rule out CCAML&#59; treatment of asymptomatic CCAML is controversial&#59; surgery may be advocated because of its low morbidity and the prevention of late complications such as malignancy&#46;</p>"
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        "resumen" => "<span class="elsevierStyleSectionTitle">Introdu&#231;&#227;o</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">O espectro cl&#237;nico da malforma&#231;&#227;o adenomat&#243;ide c&#237;stica pulmonar cong&#233;nita &#40;MACPC&#41; varia desde les&#245;es assintom&#225;ticas e dificuldade respirat&#243;ria no rec&#233;m-nascido a hidr&#243;psia fetal&#46;</p> <span class="elsevierStyleSectionTitle">Objectivos</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Avalia&#231;&#227;o da casu&#237;stica de MACPC&#44; real&#231;ando a hist&#243;ria natural&#44; tratamento e evolu&#231;&#227;o&#46;</p> <span class="elsevierStyleSectionTitle">Material e m&#233;todos</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">An&#225;lise retrospectiva dos processos cl&#237;nicos de todos os rec&#233;m-nascidos com o diagn&#243;stico de MACPC&#44; admitidos nas unidades de cui-dados intensivos neonatais de cinco centros terci&#225;rios do Norte de Portugal&#44; entre 1996 e 2005&#46;</p> <span class="elsevierStyleSectionTitle">Resultados</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">Foram identificados 15 rec&#233;m-nascidos&#44; 9F&#47;6M&#44; peso ao nascimento 3100 g &#40;645&#8211;3975&#41;&#44; idade gestacional de 38 semanas &#40;24&#8211;40&#41;&#46; A incid&#234;ncia de MACPC foi de 1&#58;9300 nascimentos&#46; Onze &#40;73&#37;&#41; rec&#233;m-nascidos apresentaram diagn&#243;stico pr&#233;-natal de les&#227;o pulmonar c&#237;stica pelas 22 semanas &#40;19&#8211;30&#41;&#46; Esta ocorreu no hemit&#243;rax direito em seis &#40;40&#37;&#41; casos e no lado esquerdo em 9 &#40;60&#37;&#41;&#46; Foi documentada regress&#227;o <span class="elsevierStyleItalic">in utero</span> da les&#227;o em dois casos&#46; Foi necess&#225;ria interven&#231;&#227;o ante-natal &#40;pleurocentese e coloca&#231;&#227;o de deriva&#231;&#227;o toraco-amni&#243;tica&#41; num feto com hidr&#243;psia fetal&#46; Ao nascimento&#44; cinco rec&#233;m-nascidos apresentaram radiografia de t&#243;rax dentro da normalidade&#44; sendo a tomografia axial computorizada diagn&#243;stica da les&#227;o&#46; Tr&#234;s &#40;20&#37;&#41; rec&#233;m-nascidos tornaram-se sintom&#225;ticos durante o per&#237;odo neonatal &#40;dificuldade respirat&#243;ria&#41; e um &#40;7&#37;&#41; ap&#243;s o per&#237;odo neonatal &#40;pneumot&#243;rax espont&#226;neo&#41;&#46; Dois &#40;13&#37;&#41; faleceram&#46; Seis &#40;40&#37;&#41; doentes foram submetidos a toracotomia e ex&#233;rese da les&#227;o&#46; O estudo anatomopatol&#243;gico demonstrou achados definitivos de MACPC &#40;classifica&#231;&#227;o de Stoker&#58; tipo I &#61; 4&#59; tipo II &#61; 1&#59; tipo III &#61; 2&#41;&#46; Oito &#40;53&#37;&#41; crian&#231;as mantiveram-se assintom&#225;ticas e permanecem em vigil&#226;ncia cl&#237;nica&#46;</p> <span class="elsevierStyleSectionTitle">Conclus&#245;es</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">A MACPC com diagn&#243;stico pr&#233;-natal tem bom progn&#243;stico na aus&#234;ncia de sofrimento fetal&#59; uma radiografia de t&#243;rax sem altera&#231;&#245;es ao nascimento n&#227;o exclui MACPC&#59; o tratamento da les&#227;o assintom&#225;tica &#233; controverso&#59; a cirurgia est&#225; indicada devido &#224; baixa morbilidade e possibilidade de preven&#231;&#227;o de complica&#231;&#245;es tardias&#44; como a degeneresc&#234;ncia maligna&#46;</p>"
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Vol. 13. Issue 4.
Pages 511-523 (July - August 2007)
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Vol. 13. Issue 4.
Pages 511-523 (July - August 2007)
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Congenital cystic adenomatoid malformation of the lung – The experience of five medical centres
Malformação congénita das vias aéreas pulmonares – Experiência de cinco centros
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948
Gustavo Rocha1,
Corresponding author
gusrocha@oninet.pt

Correspondence to/Correspondência: 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.
, Paula Cristina Fernandes2, Elisa Proença3, Conceição Quintas4, Teresa Martins5, Inês Azevedo6, Hercília Guimarães1
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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Article information
Abstract
Background

The clinical spectrum of congenital cystic adenomatoid malformation of the lung (CCAML) ranges from asymptomatic lesions to neonatal respiratory distress and hydrops fetalis.

Aim

To review our experience with CCAML, emphasising natural history, management and outcome.

Material and methods

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.

Results

Fifteen neonates with CCAML were identified, 9F/6M, birth weight 3100 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. In utero 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 pneumothorax). Two neonates (13%) died. Six (40%) patients underwent thoracotomy and appropriate excisional surgery. Histological examination showed definitive features of CCAML (Stocker classification: type I = 4; type II = 1; type III = 2). Eight (53%) patients remain asymptomatic and did not undergo surgery.

Conclusions

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.

Key-words:
Congenital
cystic adenomatoid malformation
Resumo
Introdução

O espectro clínico da malformação adenomatóide cística pulmonar congénita (MACPC) varia desde lesões assintomáticas e dificuldade respiratória no recém-nascido a hidrópsia fetal.

Objectivos

Avaliação da casuística de MACPC, realçando a história natural, tratamento e evolução.

Material e métodos

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.

Resultados

Foram identificados 15 recém-nascidos, 9F/6M, peso ao nascimento 3100 g (645–3975), idade gestacional de 38 semanas (24–40). A incidência 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 in utero da lesão em dois casos. Foi necessária intervenção ante-natal (pleurocentese e colocação de derivação toraco-amniótica) 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 espontâneo). Dois (13%) faleceram. Seis (40%) doentes foram submetidos a toracotomia e exérese da lesão. O estudo anatomopatológico demonstrou achados definitivos de MACPC (classificação de Stoker: tipo I = 4; tipo II = 1; tipo III = 2). Oito (53%) crianças mantiveram-se assintomáticas e permanecem em vigilância clínica.

Conclusões

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 prevenção de complicações tardias, como a degenerescência maligna.

Palavras-chave:
Congénito
alformação adenomatóide ística
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