A 39-year-old man was admitted to our hospital for further evaluation of a consolidated shadow and clarification of the cause of serum tumor marker elevation (CA19-9 496.2 U/ml, CA125 160.6 U/ml). Chest computed tomography revealed a well-defined homogeneous nodule in the left S(10). Angiography showed one aberrant artery, branching from the ascending aorta. Intralobar pulmonary sequestration was diagnosed and the sequestrated lung was resected. Microscopic findings of the sequestrated lung showed a mucus-containing cystically dilated bronchus, which was covered with ciliated cylindrical epithelium. Immunohistochemical staining showed positive staining for CA19-9 and CA125 in both the ciliated cylindrical epithelium and mucus. Serum values of tumor markers returned to their normal range after surgery.
"Although comparable cases have been previously reported in Japan and Korea (10–15), the exact mechanism of the condition remains a controversial subject. Previously, Yagyu et al (16) inferred that CA19-9 may be synthesized and secreted by normal bronchial epithelial cells, and gradually accumulates in the sequestrated lung with no congestion in the normal bronchial tree. In the current case, immunohistochemistry demonstrated weak staining of CA19-9 in the normal bronchial epithelia, however, marked staining was identified in the sequestrated lung tissue, particularly in the mucus of the cysts. "
[Show abstract][Hide abstract] ABSTRACT: Carbohydrate antigen 19-9 (CA19-9) is widely accepted as a tumor marker for cancers of the biliary, pancreatic and gastrointestinal tracts. Occasionally, CA19-9 is markedly elevated in the serum of patients with benign diseases. Pulmonary sequestration is a rare malformation that is characterized by the presence of lung tissue with abnormal or absent communication with the bronchi, to which blood is supplied by the systemic arteries. The current study presents a 48-year-old male who presented with upper left abdominal bloating and marked elevation of serum CA19-9 levels. The patient was referred to the Tongji Hospital (Wuhan, China) with suspected hepato-biliary-pancreatic disease and, following surgery, was diagnosed with intralobar pulmonary sequestration. Immunohistochemistry showed marked positive staining for CA19-9 in the sequestrated lung tissue. The patient's symptoms improved and the CA19-9 levels returned to the normal range following surgery. Therefore, the symptoms of upper left abdominal bloating and marked elevation of serum CA19-9 levels, in this case, may have resulted from the intralobar pulmonary sequestration.
[Show abstract][Hide abstract] ABSTRACT: Congenital lung malformations are often discovered incidentally on routine prenatal sonography or postnatal imaging. Lesions such as congenital cystic adenomatoid malformations (CCAM), sequestrations, bronchogenic cysts and congenital lobar emphysema may be asymptomatic at birth or at the time of discovery later in life. Some authors advocate simple observation because of the lack of data on the incidence of long-term complications. However, there are very few described cases where CCAM and intralobar sequestration have remained asymptomatic throughout life; complications eventually develop in virtually all patients. The most common complication is pneumonia, which may respond poorly to medical treatment. Other complications include the development of malignancies (carcinomas and pleuropulmonary blastomas), pneumothorax and hemoptysis or hemothorax. Since lung resection will be required sooner or later for CCAM, intralobar sequestration and intrapulmonary bronchogenic cysts it is best not to wait for complications to occur. For patients diagnosed prenatally, we recommend surgery at 3 to 6 months of life at the latest, so that compensatory lung growth can occur. At this age the postoperative course is usually smooth and long-term follow-up has shown normal respiratory function. Mediastinal bronchogenic cysts also tend to become symptomatic and elective resection is recommended. On the other hand, asymptomatic congenital lobar emphysema may regress spontaneously and observation is warranted. The management of small noncommunicating extralobar sequestrations is more controversial; it is known that these lesions can remain asymptomatic throughout life but complications may develop and they are sometimes difficult to differentiate from neuroblastoma.
Seminars in Pediatric Surgery 03/2005; 14(1):16-33. DOI:10.1053/j.sempedsurg.2004.10.022 · 2.22 Impact Factor
[Show abstract][Hide abstract] ABSTRACT: A 26-year-old male was admitted to our hospital with a complaint of mild dysphagia. Preoperative examinations revealed a cystic lesion at the lower esophagus, thought to be a bronchogenic cyst. The patient underwent successful thoracoscopic tumor extirpation, assisted by endoscopy, without undergoing major thoracotomy. Thoracoscopic surgery is recommended for this entity, but the muscle layer is often injured. Endoscopic ultrasonography was very useful, as it could detect damage to the proper muscle layer of the esophagus near the tumor. Thus, we could predict preoperatively that the defect in the muscle layer, which must be closed, would be caused by the surgery. Additionally, endoscopy easily facilitated the detection of the regional defect and the border of the proper muscle layer, and also could be used as a stent during wall suturing. This technique enabled us to avoid postoperative stenosis of the esophagus. Furthermore, our case presented a high level of CA125. The preoperative serum CA125 value was 93.8 U/ml and intratumoral CA125 concentration was 1,690,000 U/ml. Serum CA125 decreased to 16.5 U/ml after surgery. In this paper, we present a new strategy of the treatment for bronchogenic cyst and a review the literature.
Digestive Surgery 02/2006; 23(4):209-14. DOI:10.1159/000094751 · 2.16 Impact Factor
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