Concurrent bronchopulmonary foregut malformations: a rare case of right-sided extralobar pulmonary sequestration and bronchogenic cyst

Carolyn Hanna,Archana Shenoy,Dhanashree Rajderkar,Jennifer Reppucci,Moiz M Mustafa,Priya G Sharma

doi:10.1186/s43055-021-00440-1

Carolyn Hanna, Archana Shenoy + Show 4 more

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https://doi.org/10.1186/s43055-021-00440-1

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Abstract

BackgroundBronchopulmonary foregut malformations are rare congenital malformations. It is extremely rare to have malformations that occur simultaneously. There is literature to show that extralobar sequestration is associated with other congenital anomalies, most commonly diaphragmatic hernias, and also with other bronchopulmonary foregut malformations (e.g., extralobar sequestration and congenital pulmonary airway malformations). However, very few case reports were found that reported extralobar sequestration and foregut duplication cysts and only one report of a right-sided complex foregut malformation with pulmonary sequestration.Case presentationWe present a case of a 3-month-old male infant with a prenatal diagnosis of a cystic lung lesion who, after developing symptoms of respiratory distress, was found to have concurrent right-sided extralobar pulmonary sequestration and a mediastinal bronchogenic cyst.ConclusionsThe concurrent occurrence of these malformations in one patient could help support the theory that these malformations result from an early error in development during the time when both the lung buds and foregut are developing simultaneously.

Highlights

Bronchopulmonary foregut malformations are rare congenital malformations
We present a unique case of a 3-month-old male infant who was prenatally diagnosed with a cystic lung lesion and developed respiratory distress postnatally with the discovery of right-sided extralobar sequestration and concurrent bronchogenic cyst
Bronchopulmonary foregut malformations (BPFMs) are a wide-encompassing term referring to several types of anomalies of pulmonary development resulting from abnormal budding of the embryonic foregut and tracheobronchial tree

Summary

Conclusions

Bronchopulmonary foregut malformations are rare, with the most common malformations of the lower respiratory tract being CPAMs and bronchopulmonary sequestration [10]. The combination of two malformations concurrently is even more rare. Very few cases have been reported in the literature that describes extralobar pulmonary sequestration and bronchogenic cyst, such as

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