Congenital diaphragmatic hernia with associated broncho-pulmonary sequestration: A report of two cases and a literature review

Article type: Case Report

Authors: Alhamad, Moath^{a; b; *} | Anand, Dhullipala^{a; b}

Affiliations: [a] Sidra Medicine, Doha, Qatar | [b] Weill Cornell Medicine, Doha, Qatar

Correspondence: [*] Address for correspondence: Moath Alhamad, MD, FAAP. P.O. Box 26999, Sidra medicine, Doha, Qatar. Tel.: +974 3100 7772; E-mails: [email protected], [email protected].

Abstract: BACKGROUND:Congenital Diaphragmatic Hernia (CDH) is a severe congenital anomaly with significant morbidity and mortality. It can be isolated or can be associated with other congenital anomalies, including broncho-pulmonary sequestration (BPS). The association of CDH with BPS (CDH+BPS) is uncommon but has been previously reported, and it can complicate the course of the disease in patients with CDH. We report two cases of CDH+BPS that were recently treated at our CDH-Qatar (CDH-Q) program. METHODS:We reviewed CDH-Q program registry to search for CDH+BPS and extracted the data for the identified cases. We also reviewed the previously published literature available on PubMed for similar cases. RESULTS:Out of 53 cases of CDH referred to CDH-Q from January 2018 to December 2022, two cases of CDH+BPS were identified, with an estimated prevalence of 3.8% of this association in our CDH population. Both cases were born at term. Case 1 was diagnosed with CDH+BPS postnatally, while case 2 was diagnosed with CDH antenatally but BPS was diagnosed after birth. Both cases underwent a surgical repair of the CDH with resection of the associated BPS, and the histopathology of the resected lung tissue confirmed the presence of BPS in both. Both cases survived to discharge. CONCLUSION:The association of CDH+BPS is uncommon; however, it can have significant consequences on the management and the prognosis of patients with CDH. Reporting these cases is important to provide a better understanding of this association and its impact on CDH patients.

Keywords: Broncho-pulmonary sequestration, congenital diaphragmatic hernia

DOI: 10.3233/NPM-230042

Journal: Journal of Neonatal-Perinatal Medicine, vol. 17, no. 1, pp. 123-131, 2024