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Case Reports Indian Pediatrics 2003; 40:423-425 |
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Infected Esophageal Duplication Cyst simulating Empyema |
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Devendra Kumar* R. Samujh K.L.N. Rao
Duplications of the alimentary tract are rare congenital malformations. The duplica-tions are commonly classified into two types, tubular and cystic. Thoracic duplications are usually found in the posterior mediastinum with protrusion into either the right or left thoracic cavity beneath the parietal pleura, with majority of lesions in the lower half of esophagus(1). Patients with thoracic duplica-tions usually present either with respiratory distress because of airway compression due to enlarging mass, or as asymptomatic patients with a thoracic mass found incidentally on chest X-ray. We report a thoracic duplication cyst in a 2-year-old child simulating empyema on right side. Case Report A 2-year-old child was admitted with the complaints of high grade, intermittent fever and spasmodic paroxysmal cough of 1-week duration. The child also had an attack of pneumonia around one year back lasting for about a week, but no records or X-rays were available. He had no difficulty in swallowing, cyanosis, hemoptysis, chest pain or rash. Physical examination revealed tachypnea (respiratory rate 44/min), and subcostal retractions. The percussion note was dull and air entry was decreased on the right side. Rest of the physical and systemic examination was normal. Investigations revealed total leucocyte count of 14,500/dL with 80% polymorphs. The chest X-ray showed homogeneous opacity in the right side of chest which was interpreted as empyema and an intercostal chest tube was inserted. There was no vertebral or rib anomaly. The intercostal tube drained little amount of pus followed by straw coloured thick fluid, and the culture of this fluid did not show any microorganisms. The ultrasonography of the chest showed right pleural space with multiple internal echoes and fluid debris, suggestive of empyema. The child continued to be febrile and intercostal drain was draining some fluid. On fifth day of intercostal drain placement, it stopped draining. The intercostal tube was repositioned and this time the drained fluid was blood stained. The repeat ultrasono-graphy of the chest showed loculated right pleural effusion with internal echoes and septations. The CT scan of the chest was suggestive of right lung abscess with extensive consolidation in the upper lobe. As the child was continuing to be febrile and the right lung was not expanding, right thoracotomy was planned. Operative findings revealed a 10 × 10 cm sized cyst in the right middle and lower thorax extending posteriorly into the mediastinum. The cyst was about 3-mm thick and contained hemorrhagic fluid. The lower end of the cyst was attached to the esophagus with a thin fibrous cord, without any lumen in this cord. The cyst was excised in its entirety, and chest was drained with intercostal chest tube. The child was ventilated for 2 days in the postoperative period. After the lung had expanded completely the intercostal chest tube was removed on the 5th postoperative day. The child was afebrile, and was discharged on the 7th postoperative day. The histopathological study of the cyst showed a gastric type of epithelium along with muscle wall in some areas, compatible with esophageal duplication cyst. The child is doing well at follow up of 6 months. Discussion Esophageal duplication cysts result from the failure of the originally solid esophagus to completely vacuolated to produce a hollow tube during the fifth to eighth week of embryonic life(2,3). The incidence of congenital esophagal cyst is estimated to be 1/8200, with male sex predominance (2:1)(4). In only 10% of the individuals does the cyst communicate with the adjacent esophageal lumen(5). Esophageal duplication can be associated with other congenital anomalies, such as small intestinal duplication, eso-phageal atresias distal to the duplication; tracheoesophageal fistulas, and spinal abnor-malities, including scoliosis, hemivertebrae, and fusion(6). It is important to exclude associated anomalies in patients with this congenital abnormality and special attention must be paid to the spine, diaphragm, and gastrointestinal tract. Our patient had no associated anomalies. Usually, these cysts are found incidentally on skiagram of chest in asymptomatic patients(3), however, they become sympto-matic with respiratory features when complications such as infections, displace-ment of adjacent organs, hemorrhage, rupture and neoplasia develop(7-9). In our patient the fever and cough were caused by infection of the cyst. He has been free of symptoms after the surgery. Diagnosis can be established with a barium esophagogram showing external compression of the esophagus. Computed tomography (CT) is a noninvasive radio-logical investigation, and has the advantage over conventional diagnostic procedures because it demonstrates the exact anatomic location of cyst, cystic nature of the mass and its relationship to adjacent structures. It permits simultaneous imaging and evaluation of the spine, pulmonary parenchyma, airway, and adjacent structures. The extent of involvement of vital structures also aids in determining the resectability(7). The diagnosis of esophageal duplication cyst is complicated by their rare occurrence and by the variety of locations they exhibit within the mediastinum. The diagnostic procedures employed in our case were consistent with empyema. To the best of our knowledge there have been only three case reports of enterogenic cysts in mediastinum in children from India(10-12). Of these, one presented as paraplegia and second one was diagnosed incidentally. The third presented with high fever due to infected cyst as in our case. Contributors: DK and RS designed the study. KLNR helped in the preparation of the manuscript. RS shall act as guarantor of the study. All authors were involved in drafting of manuscript and final approval. Funding: None Competing interests: None stated.
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