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Neonatal Outcomes of Multiple Congenital Thoracic Lesions

Anthony di Natale1, Sabrina Flohr1, Leny Mathew1

  • 1Richard D. Wood Jr. Center for Fetal Diagnosis and Treatment, The Children's Hospital of Philadelphia, Philadelphia, Pennsylvania, USA.

Fetal Diagnosis and Therapy
|September 8, 2024
PubMed

Insights

Multiple congenital thoracic lesions (CTL) occur in 10.5% of patients, with only 12.9% recognized prenatally. Lung-sparing surgery is a viable option, and genetic testing is recommended due to associated anomalies.

Area of Science:

  • Pediatric Surgery
  • Thoracic Surgery
  • Medical Genetics

Background:

  • Congenital thoracic lesions (CTL) like CPAM, BPS, and CLE are typically solitary.
  • Multiple CTL are rare, with limited case reports available.
  • This study addresses the scarcity of data by analyzing a large cohort of multifocal CTL patients.

Purpose of the Study:

  • To analyze the largest patient cohort with multiple congenital thoracic lesions (CTL) to date.
  • To investigate the characteristics, diagnosis, management, and outcomes of patients with multiple CTL.
  • To provide insights into the prevalence, associated anomalies, and surgical considerations for multifocal CTL.

Main Methods:

  • Retrospective chart review of patients undergoing surgery for CTL between September 1, 2013, and March 31, 2023.
  • Inclusion criteria: radiological and surgical diagnosis of multifocal CTL (≥2 lesions in >1 lobe).
  • Data collected included pre-, peri-, and postnatal characteristics.

Main Results:

  • 10.5% of 701 CTL patients had multiple CTL; only 12.9% were correctly identified prenatally.
  • Most multifocal CTL were right-sided and multilobar (44%); 12.1% were bilateral.
  • Congenital Pulmonary Airway Malformation-Bronchopulmonary Sequestration (CPAM-BPS) lesions were most common (49%); 4% had genetic syndromes, 12.9% had additional congenital anomalies.

Conclusions:

  • Multiple CTL occur in approximately 1 in 10 patients, with low prenatal detection rates.
  • Lung-sparing surgical approaches can be considered for managing multilobar CTL.
  • Increased prevalence of associated congenital anomalies and genetic syndromes warrants consideration for genetic testing, with generally favorable outcomes.
Abstract

Keywords:
Congenital lung lesionsMultifocal diseaseMultilobar diseaseMultiple congenital thoracic lesions

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