산전진단된 선천성 낭종성선종양폐기형의 임신결과
Pregnancy outcomes of antenatally diagnosed congenital cystic adenomatoid malformation of lung
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목적: 산전진단된 선천성 낭종성선종양폐기형의 진단 정확도와 임신결과 및 예후에 영향을 미치는 인자를 확인하고자 하였다. 연구 방법: 1999년 1월부터 2008년 6월까지 서울대학교병원에서 산전초음파를 통해 선천성 낭종성선종양폐기형으로 진단된 32예를 대상으로 산모 및 신생아의 의무기록을 후향적으로 조사하였다. 결과: 산전초음파검사를 통해 선천성 낭종성선종양폐기형으로 진단된 32예 중 24예에서 추적관찰이 이루어 졌다. 출생 후 조직검사나 영상의학검사를 통해 3예는 폐분리증으로, 1예는 선천성 대엽성폐기종으로 진단되어 선천성 낭종성선종양폐기형의 산전진단의 정확도는 83.3% (20/24)였다. 선천성 낭종성선종양폐기형으로 최종 진단이 확인된 20예 중 태아수종이나 복수가 동반된 경우는 2예 (10%)였다. 태아수종이나 복수가 동반된 2예 중 한 예는 흉강-양막강단락술을 시행하였고, 다른 한 예는 산모가 원하여 임신을 종결하였다. 임신을 유지한 15예 중 흉강-양막강단락술을 시행한 1예를 제외하면 모두 만삭분만 하였으며 (평균 분만 주수: 39.6±2.2주, 평균체중: 3,200±500 gram), 15예 모두 출생 후 다른 합병증 없이 생존 퇴원하였다. 산전초음파 분류에 의해 대낭성으로 확인된 13예 중 출생 후 조직학적 분류에서 미세낭이나 고형성 병변인 III형은 없었으며, 소낭성으로 진단된 7예의 경우 III형이 5예(83%)로 산전초음파 분류와 출생 후 분류간의 연관성을 확인할 수 있었다 (P<0.01). 결론: 산전진단된 선천성 낭종성선종양폐기형에서 태아수종이나 복수와 같은 합병증의 빈도는 10%였으며, 이러한 합병증이 없이 임신을 지속한 경우 임신결과는 매우 양호하였다.
Abstract
Objective: To determine the accuracy of antenatal diagnosis, pregnancy outcome and the predictors for adverse outcome in antenatally diagnosed congenital cystic adenomatoid malformation (CCAM). Methods: A retrospective review was performed on a total of 32 patients with antenatally diagnosed CCAM between January, 1999 and June, 2008. Results: Of 32 cases with antenatally diagnosed CCAM, 8 patients were lost to follow-up. In remaining 24 patients, postnatal histologic examination and/or radiologic study showed definitive diagnostic features of CCAM (n=20), pulmonary sequestration (n=3) and congenital lobar emphysema (n=1). The accuracy of antenatal diagnosis was 83.3% (20/24). The rate of presence of hydrops and/or ascites was 10% (2/20) in those with CCAM confirmed postnatally. Of the 2 patients with ascites and/or hydrops, thoraco-amniotic (TA) shunt was performed in one case with hydrops and termination was done in the other case. All of the 15 cases with ongoing pregnancy were delivered at term, except one with TA-shunt who was born at 33+3 weeks of gestation (mean gestational age at birth±standard deviation; 39.6±2.2 weeks, mean birth weight; 3,200±500 gram). All 15 neonates were discharged alive without any complication. Antenatal sonographic microcystic lesion was associated with postnatal histologic type III lesion [5 type III lesions in 7 microcystitc cases (71.4%) vs. no type III lesion in 13 macrocystic cases (0%), P<0.01]. Conclusion: The incidence of ascites and/or hydrops was 10% (2/20) in fetuses with CCAM. Antenatally diagnosed CCAM has an excellent prognosis in the absence of signs of ascites and/or hydrops.
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