Thattaruparambil Prakash Vinayak, Suravi Mohanty, Kanishka Das
All India Institute of Medical Sciences. St. John’s Medical College.
India
Fetal and Pediatric Pathology
Fetal Pediatr Pathol 2022; 41: 505-510
DOI: 10.1080/15513815.2020.1849474
Abstract
Background: Idiopathic lobar emphysema (ILE) and bronchopulmonary sequestration (BPS) are two of the well-characterized pulmonary malformations.
Case report: An antenatally detected case of a left bronchopulmonary malformation (BPM) was clinicoradiologically diagnosed to be a left upper lobar emphysema with isolated dextrocardia in the neonatal period. Besides the emphysematous left upper lobe, an accessory lobe akin to an extra lobar BPS was an operative surprise. Histopathological examination of both excised lobes led to a revised diagnosis of a ‘hybrid’ malformation comprising lobar emphysema and extra lobar BPS. The postoperative recovery was uneventful.
Discussion/conclusion: The observations suggest that BPS and ILE may be interim entities in a continuum of abnormal embryogenesis. Such a hybrid malformation has not been hitherto reported.
Category
Abnormal Systemic to Pulmonary Arterial Collaterals or Connections
Surgical and Catheter-mediated Interventions for Pulmonary Vascular Disease
Age Focus: Pediatric Pulmonary Vascular Disease
Fresh or Filed Publication: Filed (PHiled). Greater than 1-2 years since publication
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