Giant bronchogenic cyst with pericardial defect: a case report & literature review in Japan

Abstract

Congenital pericardial defects are a rare anomaly, found during autopsy and cardiothoracic surgery. We describe a case of a 69-year-old female, with a right-sided congenital pericardial defect associated with a giant bronchogenic cyst (BC) found during surgery. The cyst was resected and the patient developed arrhythmia following surgery. A review of the literature in Japan was performed, focusing on congenital anomalies associated with pericardial defects and its pathogenesis. We paid particular attention to complications following thoracic surgery in patients with pericardial defects and indications of pericardial reconstruction in such patients. Figure 2B,C). The tumor was 72 mm in length and contained thick yellowish fluid ( Figure 2D). Pathological diagnosis was BC. Keywords: Bronchogenic cyst (BC);The patient developed a paroxysmal atrial fibrillation on the third post-operative day and medication was started. She was discharged on the 13th post-operative day, but atrial fibrillation persisted three months after surgery. DiscussionCongenital pericardial defect is a rare anomaly consisting of a partial or total defect in the pericardium. It was found in one case among 14,000 autopsies performed in a single institution (3). Most cases are found by chance at autopsies and thoracic surgeries. In a research by Ellis et al., 73% were males, and were left sided in most cases. Only three among 99 cases had right sided defects (4).During the 6th week of gestation, the pericardio-pleural canal fuses and the communication between the pleural cavity and the pericardium closes. The patency of the canal is said to be the cause of defects in the pericardium. According to a theory by Perna, premature atrophy of the duct of Cuvier results in deficient blood supply and incomplete development of the pleuro-pericardial membrane (5,6).In 1945, Rusby et al. (5) reported a case of BC associated with left sided partial pericardial defect. The association between these two anomalies may also be due to the atrophy of the duct of Cuvier. The lung buds develop from the foregut in the 5th-week embryo. As the lung develops, structures such as the duct of Cuvier, prevents its expansion to the mesial side. Premature atrophy of the duct is theorized to enable the development of abnormal lung buds and additional lobes. In a search of the English literature, we found 25 cases of pericardial defects associated with developmental abnormalities arising from the foregut, such as BCs and pulmonary sequestrations (5,7-21). There were ten cases of mediastinal BCs among which one had a partial right-sided defect in the pericardium as seen in our case (9). Many of the cases had other malformations in the lung and vessels.Though pericardial defects are usually asymptomatic, it is sometimes associated with increased mobility and stress on anchoring structures of the heart, resulting in local inflammation and chest pain. There are also occasional reports of sudden death due to herniation of the heart (4,22).We performed a review of the Japan...

Mature mediastinal bronchogenic cyst with left pericardial defect: A case report

“…Imperatori et al [ 19 ] has reported 18 cases of bronchogenic cyst with congenital pericardial defect. A number of articles[ 13 , 22 ] suggest that congenital pericardial defects may be related to bronchial cysts. At the fifth week of embryonic development, the bronchial bud develops at the tail end of the larynx and grows into the pericardioperitoneal canal, which is the future pleural cavity; during this period, the primitive heart and lungs share a common chamber and are closely linked to each other[ 23 ].…”

Bronchogenic Cyst: A Rare Cause of Respiratory Distress in Neonate - A Case Report

“…BC is also associated with various congenital anomalies of heart, chest wall and diaphragm. Imperatori A et al [9] and Kamata T et al [10] observed bronchogenic cyst with pericardial defect in adult. There is a small risk of malignant transformation within the cyst; bronchoalveolar cell carcinoma and neuroblastoma are reported with bronchogenic cyst [2].…”

Partial Left Pericardial Agenesis in a Neonate

“…[ 1 ] Smaller defects with the potential to cause strangulation should be carefully evaluated for pericardial reconstruction. [ 2 ]…”