Hipertensão pulmonar em lactente associada a pulmão em ferradura: relato de caso

Abstract

This is the case report of a young infant with early respiratory distress and pulmonary hypertension, diagnosed as a variant of horseshoe lung and we have reviewed the literature to seek information about this rare pulmonary malformation and its cardiac and hemodynamic repercussions.

Horseshoe Lung Associated with Left Lung Hypoplasia: Case Report and Systematic Review of the Literature

“…Based on the definitions of aplasia and hypoplasia of the lung mentioned above [ 14 ], it may be controversial to diagnose those cases as HL involving left lung hypoplasia rather than left lung aplasia. Lutterman et al [ 4 ] and Neves et al [ 10 ] reported that in their case the right pulmonary artery branch ran to the isthmus of the left lung, and the origins of the left pulmonary artery and vein could not be clearly depicted on angiography. Ersöz et al [ 2 ] reported that in their case the left pulmonary artery was not observed on angiography, but small collateral vessels arising from the main pulmonary artery were seen.…”

“…To the best of our knowledge, only 10 cases of HL involving left lung hypoplasia have been reported [ 2 – 11 ]. In those cases, an abnormal number of left pulmonary veins [ 8 , 11 ] and cardiovascular malformations such as ASD, VSD and DORV were observed [ 2 , 3 , 5 , 6 , 9 , 10 ]. In our case, an ASD and a VSD were detected with 128-DSCT, but the number of the pulmonary veins was normal.…”

“…Among the 10 reported cases in Table 1 , the left pulmonary artery could not be detected in 5 cases [ 2 , 4 , 5 , 9 , 10 ]. Jeewa et al [ 9 ] reported that in their case the left pulmonary artery, left pulmonary vein, and left main bronchus were not detected on CE-CT images.…”

“…Ersöz et al [ 2 ] reported that in their case the left pulmonary artery was not observed on angiography, but small collateral vessels arising from the main pulmonary artery were seen. Among the 10 reported cases of HL involving left lung hypoplasia, the pulmonary arteries of 3 cases [ 2 , 4 , 10 ] were evaluated with angiography. The absence of the pulmonary artery is considered to be a diagnostic criterion for lung agenesis, but not lung hypoplasia [ 14 ].…”

“…Abnormal numbers of left pulmonary veins were observed in 2 patients, and cardiovascular malformations such as ASD, VSD and double outlet right ventricle (DORV) were detected in 7 patients. The left pulmonary artery could not be detected in 5 cases [ 2 , 4 , 5 , 9 , 10 ]. Seven cases involved pulmonary hypertension, whereas pulmonary hypertension was not mentioned in the reports on the other 4 cases.…”