To shunt or not to shunt a pulmonary adenomatoid cystic malformation after 33 weeks of gestation

a case report

Authors

  • Rodrigo Ruano Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo
  • Paula Beatriz Tavares Fettback Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo
  • Vinicius Lima Ribeiro Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo
  • Marcus Marques Silva Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo
  • João Gilberto Maksoud Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo
  • Marcelo Zugaib Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

Keywords:

Cystic adenomatoid malformation of lung, congenital, Ultrasonography, Prenatal diagnosis, Fetal therapy, Abnormalities

Abstract

CONTEXT: Macrocystic adenomatoid malformation of the lung can cause severe mediastinal shift, hydrops and polyhydramnios, thereby increasing the risk of perinatal deaths. After 33 weeks of gestation, repeated puncturing of the cyst is recommended. We present a case in which a cyst-amniotic shunt was placed instead of performing this procedure. CASE REPORT: A cyst-amniotic shunt was placed at 33 weeks of gestation because of a large macrocystic adenomatoid malformation of the lung associated with severe mediastinal shift and polyhydramnios. Although it was confi rmed that the catheter was in the correct place, the cyst increased in size again two weeks later, associated with repetition of polyhydramnios. It was postulated that the catheter was blocked, and we chose to place another catheter instead of performing repeated punctures. The cystic volume, polyhydramnios and mediastinal shift regressed progressively. At 38.5 weeks, a 3,310 g male infant was delivered without presenting any respiratory distress. The infant underwent thoracotomy on the 15th day of life. Thus, in the present study, we discuss the possibility of placing a cyst-amniotic shunt instead of performing repeated cystic punctures, even at a gestational age close to full term.

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Author Biographies

Rodrigo Ruano, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

MD, PhD. Professor, Fetal Medicine Unit, Department of Obstetrics, Hospital das Clinicas, Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, Brazil.

Paula Beatriz Tavares Fettback, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

MD. Resident, Fetal Medicine Unit, Department of Obstetrics, Hospital das Clinicas, Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, Brazil.

Vinicius Lima Ribeiro, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

MD. Resident, Fetal Medicine Unit, Department of Obstetrics, Hospital das Clinicas, Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, Brazil.

Marcus Marques Silva, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

MD, Attending physician, Pediatric Surgery, Department of Pediatrics, Hospital das Clinicas, Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, Brazil.

João Gilberto Maksoud, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

MD, PhD. Professor, Pediatric Surgery, Department of Pediatrics, Hospital das Clinicas, Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, Brazil.

Marcelo Zugaib, Hospital das Clínicas, Faculdade de Medicina da Universidade de São Paulo

MD, PhD. Professor, Fetal Medicine Unit, Department of Obstetrics, Hospital das Clinicas, Faculdade de Medicina da Universidade de São Paulo (HCFMUSP), São Paulo, Brazil.

References

Stocker JT, Madewell JE, Drake RM. Congenital cystic adenomatoid malformation of the lung. Classification and morphologic spectrum. Hum Pathol. 1977;8(2):155-71.

Bunduki V, Ruano R, da Silva MM, et al. Prognostic factors associated with congenital cystic adenomatoid malformation of the lung. Prenat Diagn. 2000;20(6):459-64.

Ruano R. Recent advances in sonographic imaging of fetal tho- racic structures. Expert Rev Med Devices. 2005;2(2):217-22.

Wilson RD, Baxter JK, Johnson MP, et al. Thoracoamniotic shunts: fetal treatment of pleural effusions and congenital cystic adenomatoid malformations. Fetal Diagn Ther. 2004;19(5):413- 20.

Ruano R, Benachi A, Joubin L, et al. Three-dimensional ultrasonographic assessment of fetal lung volume as prognostic factor in isolated congenital diaphragmatic hernia. BJOG. 2004;111(5):423-9.

Tanigaki S, Miyakoshi K, Tanaka M, et al. Pulmonary hypo- plasia: prediction with use of ratio of MR imaging-measured fetal lung volume to US-estimated fetal body weight. Radiology. 2004;232(3):767-72.

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Published

2008-07-07

How to Cite

1.
Ruano R, Fettback PBT, Ribeiro VL, Silva MM, Maksoud JG, Zugaib M. To shunt or not to shunt a pulmonary adenomatoid cystic malformation after 33 weeks of gestation: a case report. Sao Paulo Med J [Internet]. 2008 Jul. 7 [cited 2025 Apr. 2];126(4):239-41. Available from: https://periodicosapm.emnuvens.com.br/spmj/article/view/1983

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Section

Case Report