Extralobar pulmonary sequestration manifesting as hemorrhagic infarction in a five-year-old male

Abstract

a b s t r a c tExtralobar pulmonary sequestration (EPS) is frequently asymptomatic and detected incidentally. We herein report a pediatric case of EPS that manifested as hemorrhagic infarction. A five-year-old male with previously normal X-ray films was referred to our hospital due to acute onset of right flank pain and a non-productive cough. The initial diagnosis was infection of an EPS; however, the symptoms persisted despite the administration of antibiotics. A CT-angiogram revealed interruption of the blood flow to the sequestrum, and a definitive diagnosis as hemorrhagic infarction of a pulmonary sequestration was made. During surgery, an aberrant artery was revealed to derive from the intercostal artery and the sequestrum was resected safely. In cases of infarction of EPS, early surgical resection should be considered, because progressed adhesion can complicate dissection from surrounding organs and the detection of the feeding vessels. Ó 2015 The Authors. Published by Elsevier Inc. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).A pulmonary sequestration is defined as a mass of abnormal lung tissue which has no bronchial communication with the normal tracheobronchial tree. Pulmonary sequestrations are divided into two groups; extralobar and intralobar sequestrations, based on whether they have their own visceral pleura. Extralobar pulmonary sequestration (EPS) is usually asymptomatic and detected incidentally during examinations for other clinical conditions [1]. We herein report a case of symptomatic EPS with massive bloody pleural effusion, and the initial symptom was a sudden onset of right flank pain. The pathological findings were compatible with hemorrhagic infarction of an EPS. We also review other previously reported cases of this condition. Case reportA five-year-old male was referred to our hospital due to a sudden onset of right flank pain and a non-productive cough. The patient had a past history of acute bronchitis when he was twoyears-old and had undergone adenoidectomy when he was fouryears-old. The patient received X-ray examinations each time, in which pulmonary sequestration was not found. Radiologic abnormalities were not identified even though our radiologists reviewed these X-ray films. At this presentation, the patient's temperature was 38.4 C, his pulse was 140 bpm and his respirations were 60/ min. The breath sounds were diminished over the right lower lobe. He had remarkable right flank pain, but there were no signs of peritoneal irritation or ascitic fluid. A peripheral blood examination showed elevation of the white blood cell count to 13,000 and a serum C-reactive protein concentration of 8.03 mg/dl. An X-ray film of the chest showed opacification of the right hemithorax. A contrast-enhanced computed tomography (CECT) scan revealed the presence of a solitary triangle mass without enhancement, adjacent to the posterior portion of the right lower lobe (Fig. 1). In addition, massive pleural effusion was demonstrated,...