A rare case of torsion and infarction of an extralobar pulmonary sequestration with MR, CT, and surgical correlation

Abstract

Extralobar pulmonary sequestration is a rare congenital pulmonary malformation that may present early in life or remain asymptomatic. Here we present a case of torsion of an extralobar pulmonary sequestration on its vascular pedicle. Although the patient's initial symptomatology suggested intraabdominal pathology, the correct preoperative diagnosis was determined in large part by the lesion's MRI characteristics, which strongly suggested tissue infarction.

Clinical analysis of extralobar pulmonary sequestration with torsion in children: report of 6 cases

“…The left/right ELS ratio was 1:2 in our centre’s cases. The proportion of ELS on the right side was larger in our study, while previous literature found that ELS tended to occur on the left side [ 4 – 7 ], which may be one of the reasons for the low preoperative diagnosis rate. In our cases, one child was an adolescent, three were school age, and two were preschool age.…”

“…In the past 14 years, a total of 32 cases of ELS were diagnosed in our hospital, of which only 6 cases were combined with torsion of the vascular pedicle, accounting for 18.75% of cases. To date, a total of 13 paediatric cases of ELS with torsion have been reported in the English literature [ 4 – 6 , 8 – 17 ]. To the best of our knowledge, this is the largest number of cases reported in the literature.…”

“…The cause of the abdominal pain may be due to local inflammation caused by ischaemic necrosis after torsion of the ELS vascular pedicle. The children’s symptoms disappeared immediately after removing the lesion [ 4 , 12 , 13 ]; however, antibiotic treatment could not have achieved this effect [ 13 ].…”

“…The preoperative diagnosis rate of ELS presenting with torsion is extremely low in childhood and adolescence. Among the 13 reported paediatric cases of torsed ELS in the literature, only 4 cases were diagnosed before the operation [ 4 , 6 , 10 , 11 ]. In the early years, owing to a lack of awareness about the imaging characteristics of ELS with torsion among radiologists in our hospital, the first 3 cases were not identified before the procedure.…”

“…ELS comprises 25% of all pulmonary sequestrations [ 3 ]. The majority of ELS is located between the lower lobe of the lung and the diaphragm [ 4 – 6 ], and its feeding artery mainly originates from the thoracic aorta, abdominal aorta, or other vessels in the systemic circulation. Venous drainage reaches the right atrium via the azygos vein, hemiazygos vein or vena cava [ 7 ].…”