Am J Perinatol 2019; 36(03): 225-232
DOI: 10.1055/s-0038-1669909
SMFM Fellowship Series Article
Thieme Medical Publishers 333 Seventh Avenue, New York, NY 10001, USA.

Fetal Congenital Pulmonary Airway Malformation: The Role of an Objective Measurement of Cardiomediastinal Shift

Rachel Shulman
1   Division of Maternal Fetal Medicine, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California, San Francisco
,
Teresa N. Sparks
1   Division of Maternal Fetal Medicine, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California, San Francisco
,
Kristen Gosnell
2   Department of Obstetrics, Gynecology, and Reproductive Sciences, Fetal Treatment Center, University of California, San Francisco
,
Cinthia Blat
1   Division of Maternal Fetal Medicine, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California, San Francisco
,
Mary E. Norton
1   Division of Maternal Fetal Medicine, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California, San Francisco
,
Hanmin Lee
3   Department of Surgery, Fetal Treatment Center, University of California, San Francisco
,
Juan Gonzalez-Velez
1   Division of Maternal Fetal Medicine, Department of Obstetrics, Gynecology, and Reproductive Sciences, University of California, San Francisco
,
Ruth B. Goldstein
2   Department of Obstetrics, Gynecology, and Reproductive Sciences, Fetal Treatment Center, University of California, San Francisco
› Author Affiliations
Further Information

Publication History

27 September 2017

24 July 2018

Publication Date:
10 September 2018 (online)

Abstract

Objective To examine the relationship between cardiomediastinal shift angle (CMSA) and adverse perinatal outcomes and hydrops in cases of congenital pulmonary airway malformation (CPAM).

Study Design This retrospective study evaluated CPAM cases referred to our institution from 2008 to 2015. The primary outcome was a composite score for adverse perinatal outcome. CMSA was measured for each case and evaluated for its association with the primary outcome. The prediction accuracy of CMSA for adverse perinatal outcome was assessed using receiver operator characteristic (ROC) curves.

Results Eighteen (21.2%) of the 85 cases experienced an adverse perinatal outcome. Increases in CMSA were associated with adverse perinatal outcomes and hydrops in bivariate analyses. Adjusted analyses found each 10-degree increase in CMSA to be associated with increased odds of an adverse perinatal outcome (adjusted odds ratio [aOR] 2.2, 95% confidence interval [CI]: 1.4–3.3) and hydrops (aOR 3.0, 95% CI: 1.5–6.1). CMSA performed well and was comparable to CPAM volume ratio in predicting adverse perinatal outcomes (area under the curve 0.81 and 0.84, respectively).

Conclusion We describe a novel measurement of mediastinal shift in cases of CPAM and its relationship with adverse perinatal outcomes and hydrops. These findings may shape the evaluation and management of CPAMs, improve our understanding of their prognosis, and influence patient counseling.

Note

The findings reported here were presented in part as posters at the Society for Maternal Fetal Medicine 37th Annual Meeting January 23–28, 2017 in Las Vegas, NV, and the American Institute of Ultrasound in Medicine Convention March 25–29, 2017 in Lake Buena Vista, FL.


 
  • References

  • 1 David M, Lamas-Pinheiro R, Henriques-Coelho T. Prenatal and postnatal management of congenital pulmonary airway malformation. Neonatology 2016; 110 (02) 101-115
  • 2 Ehrenberg-Buchner S, Stapf AM, Berman DR. , et al. Fetal lung lesions: can we start to breathe easier?. Am J Obstet Gynecol 2013; 208 (02) 151.e1-151.e7
  • 3 Lau CT, Kan A, Shek N, Tam P, Wong KK. Is congenital pulmonary airway malformation really a rare disease? Result of a prospective registry with universal antenatal screening program. Pediatr Surg Int 2017; 33 (01) 105-108
  • 4 Adzick NS, Harrison MR, Crombleholme TM, Flake AW, Howell LJ. Fetal lung lesions: management and outcome. Am J Obstet Gynecol 1998; 179 (04) 884-889
  • 5 Kunisaki SM, Ehrenberg-Buchner S, Dillman JR, Smith EA, Mychaliska GB, Treadwell MC. Vanishing fetal lung malformations: Prenatal sonographic characteristics and postnatal outcomes. J Pediatr Surg 2015; 50 (06) 978
  • 6 Hellmund A, Berg C, Geipel A. , et al. Prenatal diagnosis and evaluation of sonographic predictors for intervention and adverse outcome in congenital pulmonary airway malformation. PLoS One 2016; 11 (03) e0150474
  • 7 Yong PJ, Von Dadelszen P, Carpara D. , et al. Prediction of pediatric outcome after prenatal diagnosis and expectant antenatal management of congenital cystic adenomatoid malformation. Fetal Diagn Ther 2012; 31 (02) 94-102
  • 8 Baird R, Puligandla PS, Laberge J-M. Congenital lung malformations: informing best practice. Semin Pediatr Surg 2014; 23 (05) 270-277
  • 9 Curran PF, Jelin EB, Rand L. , et al. Prenatal steroids for microcystic congenital cystic adenomatoid malformations. J Pediatr Surg 2010; 45 (01) 145-150
  • 10 Morris LM, Lim F-Y, Livingston JC, Polzin WJ, Crombleholme TM. High-risk fetal congenital pulmonary airway malformations have a variable response to steroids. J Pediatr Surg 2009; 44 (01) 60-65
  • 11 Crombleholme TM, Coleman B, Hedrick H. , et al. Cystic adenomatoid malformation volume ratio predicts outcome in prenatally diagnosed cystic adenomatoid malformation of the lung. J Pediatr Surg 2002; 37 (03) 331-338
  • 12 Cass DL, Olutoye OO, Cassady CI. , et al. Prenatal diagnosis and outcome of fetal lung masses. J Pediatr Surg 2011; 46 (02) 292-298
  • 13 Bunduki V, Ruano R, da Silva MM. , et al. Prognostic factors associated with congenital cystic adenomatoid malformation of the lung. Prenat Diagn 2000; 20 (06) 459-464
  • 14 Vu L, Tsao K, Lee H. , et al. Characteristics of congenital cystic adenomatoid malformations associated with nonimmune hydrops and outcome. J Pediatr Surg 2007; 42 (08) 1351-1356
  • 15 Miller JA, Corteville JE, Langer JC. Congenital cystic adenomatoid malformation in the fetus: natural history and predictors of outcome. J Pediatr Surg 1996; 31 (06) 805-808
  • 16 Mallmann MR, Graham V, Rösing B. , et al. Thoracoamniotic shunting for fetal hydrothorax: predictors of intrauterine course and postnatal outcome. Fetal Diagn Ther 2017; 41 (01) 58-65
  • 17 Rice HE, Estes JM, Hedrick MH, Bealer JF, Harrison MR, Adzick NS. Congenital cystic adenomatoid malformation: a sheep model of fetal hydrops. J Pediatr Surg 1994; 29 (05) 692-696
  • 18 Thorpe-Beeston JG, Nicolaides KH. Cystic adenomatoid malformation of the lung: prenatal diagnosis and outcome. Prenat Diagn 1994; 14 (08) 677-688
  • 19 Committee on Obstetric Practice, the American Institute of Ultrasound in Medicine, and the Society for Maternal-Fetal Medicine. Committee opinion no 700: methods for estimating the due date. Obstet Gynecol 2017; 129 (05) e150-e154
  • 20 Costanzo S, Filisetti C, Vella C. , et al. Pulmonary malformations: predictors of neonatal respiratory distress and early surgery. J Neonatal Surg 2016; 5 (03) 27