Giant oesophageal hamartoma.

Abstract

Benign tumours of the oesophagus are rare, representing less than 1 % of all oesophageal neoplasms.' Most of them are intramural leiomyomas and other benign tumours encountered only infrequently; among these pedunculated intraluminal hamartomas form a particularly rare group. Special difficulties are associated with their diagnosis, but early detection is important because despite their benign histological nature life threatening sequelae may ensue. We present the case history of a patient with a protracted illness due to a large oesophageal hamartoma, which had remained undiagnosed despite multiple endoscopies and biopsy.Case report A 52 year old non-smoker presented in late 1979 with a dry cough and mild but progressive dysphagia. Barium studies showed a megaoesophagus with large, lobulated filling defects. At oesophagoscopy grossly dilated mucosal folds were visualised within the dilated oesophagus, biopsy of which showed only chronic inflammatory changes. The differential diagnosis included achalasia, oesophagitis with polyp formation, and giant mucosal folds of the oesophagus. The patient was treated symptomatically until November 1982, when he presented again with rapidly progressive dysphagia. The results of the investigations were as before; the condition was thought to be inflammatory and the patient was treated with a brief course of prednisolone, which produced a transient but substantial symptomatic improvement.The patient was readmitted five months later with an episode of complete dysphagia. On admission he was malnourished, hypoalbuminaemic, and anaemic with an iron deficiency picture. Nasoenteric feeding was started and he transferred to the Brompton Hospital. A plain posteroanterior chest radiograph showed a grossly dilated oesophagus with a shelf like filling defect in the upper third. Barium studies (fig 1) showed a megaoesophagus with large, lobulated filling defects. The findings at oesophagoscopy were unchanged. The patient, requiring urgent relief of his complete dysphagia, proceeded to thoracotomy. The approach was made initially through the left chest and a long oesophagotomy was made at the level of the inferior pulmonary vein before the true nature of the lesion was appreciated. This allowed delivery of the apex of a single large polypoid lesion. We decided at this stage to proceed to formal oesophagectomy as primary closure of the compressed and dilated oesophagus was likely to be hazardous. The stomach was mobilised and the thoracotomy closed, the patient was turned, and a right thoracotomy was performed. A mobile mass could be felt

Chondromatous hamartoma of cervical esophagus: a case report and literature review

“…To the best of our knowledge, twenty studies with twenty esophageal hamartoma cases (1,(4)(5)(6)(7)(8)(9)(10)(11)(12)(13)(14)(15)(16)(17)(18)(19)(20)(21)(22) have been reported and eight of these studies described (4,6,8,10,11) were cervical or upper esophagus hamartomas [Halfhide et al (10) and Ginai et al (11) reported the same case], and five cases (5,7,9,13) were in distal or lower esophagus [one (9) was in Chinese, Table 1]. Several other studies (23-32) also reported congenital esophageal stenosis caused by tracheobronchial remnants.…”

A hamartoma presenting as an intramural upper oesophageal tumour

“…The most common tumour type is leiomyoma, while other benign tumours are rare, such as neurofibromas (schwannoma), granular cell tumours, hemangiomas, lipomas, and other types of polyps like hamartoma (13). Previous reports have described intraluminal polypomatous descriptions of oesophageal hamartoma or lower intramural oesophageal hamartoma (2,3,5,14) or lower intramural oesophageal hamartoma (4). Hamartoma in cervical oesophagus of adults is extremely rare and only five case reports on this entity have been published this far (2,3,5,8,15).…”

“…Hamartomas were introduced in 1904 by Albrecht (1) as tumour-like malformations with mixing normal tissue components in the organ in which they occur. Hamartomas of the lung and airways are relatively common tumours, but only a few reports describe a hamartoma of the oesophagus in adults (2)(3)(4)(5)(6)(7)(8). Also, hamartomas of the nasotracheal tract have been presented, and most of them in paediatric population (9,10).…”

Adult Asymptomatic Hamartoma in the Distal Esophagus: A Rare Case

“…Due to its rarity, there are no epidemiological data regarding the occurrence of this disease in the esophagus. Limited data have been published as case reports, scattered in sometimes obscure or relatively inaccessible journals (Table) (2,(4)(5)(6)(7)(8)(9)(10)(11)(12)(13).…”

“…Most lesions have been discovered as a result of the patient's clinical presentation, for example, the presence of obstruction due to intraluminal growth (4,11) or the regurgitation of a pedunculated tumor (9, 13). Other tumors have been vague or asymptomatic and discovered incidentally in late childhood or adulthood (2, 5, 10), as in the case of the current patient.…”