Ann Thorac Surg 2009;87:1258-1260. doi:10.1016/j.athoracsur.2008.08.053
© 2009 The Society of Thoracic Surgeons
Case Reports
Diffuse Esophageal Polyposis: An Uncommon Occurrence
Erica M. Giblin, MDa,*,
Carolyn E. Reed, MDb
a Department of Cardiothoracic Surgery, Medical University of South Carolina, Charleston, South Carolina
b Department of General Thoracic Surgery, Department of Cardiothoracic Surgery, Medical University of South Carolina, Charleston, South Carolina
Accepted for publication August 20, 2008.
* Address correspondence to Dr Giblin, Department of Cardiothoracic Surgery, Medical University of South Carolina, Ashley River Tower, 25 Courtenay Dr, Suite 7018, Charleston, SC 29425 (Email: gibline{at}hotmail.com).
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Abstract
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The presence of multiple esophageal polyps on endoscopy is a rare entity. Most of the literature cited on this phenomenon is based on case reports and small series. A large proportion of the literature describes one or two polyps, with the majority of polyps occurring in the area of the gastroesophageal junction. We present a case of a 66-year-old woman with a history of gastroesophageal reflux disease that was found to have extensive esophageal polyposis of the mid-esophagus on upper endoscopy. The patient underwent a transhiatal esophagectomy. Final pathology was consistent with extensive polypoid dsyplasia in the presence of Barrett's esophagus.
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Introduction
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Diffuse esophageal polyposis is an exceptionally uncommon finding on endoscopy. Esophageal polyposis in conjunction with Barrett's esophagus is even less common [1]. The natural history of diffuse esophageal polyposis is relatively unknown. Also unknown is the role of diffuse esophageal polyps as a marker for neoplastic progression. We report a rare case of a patient with diffuse esophageal polyposis and Barrett's esophagus who underwent definitive treatment with transhiatal esophagectomy.
The patient is a 66-year-old woman with a longstanding history of gastroesophageal reflux disease and hiatal hernia. She had been diagnosed with Barrett's esophagus in 1994 and had undergone surveillance every 2 to 3 years. Multiple biopsies were significant for low-grade dysplasia. Approximately 1 year ago, an exacerbation of her reflux symptoms developed. Upper endoscopy at the time was significant for multiple polypoid lesions in her esophagus extending from 20 to 29 cm, and Barrett's esophagus extending from 20 cm to the gastroesophageal junction at 34 cm from the incisors (Fig 1). Biopsy of the lesions revealed polypoid low-grade dysplasia arising in a goblet cell, containing columnar mucosa of the type seen in Barrett's mucosa. The patient underwent endoscopic ultrasound evaluation that demonstrated the polypoid process was confined to the mucosa. A computed tomographic scan showed a markedly dilated esophagus starting from the esophageal inlet and extending to the gastroesophageal junction where a large hiatal hernia was present (Fig 2). She was seen in consultation with thoracic surgery and surgical resection was recommended. Her pulmonary function tests were significant for a forced vital capacity of 2.77 (95%) and forced expiratory volume in 1 second of 2.20 (94%). The patient underwent a transhiatal esophagectomy with cervical esophagogastric anastamosis, pyloroplasty, and feeding jejunostomy tube placement. Final pathology was relevant for extensive polypoid dysplasia with low-grade Barrett's esophagus (Fig 3). A focus of high-grade dysplasia was identified, but no frank invasive carcinoma was demonstrated. No evidence of Barrett's or dysplasia was noted at the proximal resection margin. Immunohistochemistry was relevant for Ki-67 focally positive and p53 focally positive. On postoperative day 7, there was noted to be cloudy fluid in the patient's cervical drain.
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Fig 1. Endoscopic evaluation of the esophagus notable for diffuse polyposis extending from 20 to 29 cm from the incisors.
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Fig 2. Chest computed tomographic scan with intravenous contrast demonstrating a dilated esophagus and hiatal hernia.
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A gastrografin swallow was significant for a small anastamotic leak. The patient's neck incision was opened and an endoscopy revealed a small opening at the level of the gastrotomy. The wound was packed and the patient was discharged on a clear liquid diet and jejunostomy tube feeds. A repeat swallow 1 week later showed resolution of the anastamotic leak and the patient's diet was progressively advanced. At 6 months postoperatively, the patient is eating without difficulty.
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Comment
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Polypoid lesions of the esophagus are a distinctly unusual finding on endoscopic examination. The most common presentation is that of a solitary esophageal polyp. Less commonly seen are multiple polyps, and diffuse polyposis is a very rare finding. Most reports of polypoid lesions are in the form of case reports and small series. In one of the largest series, Abraham and colleagues [1] discussed 27 patients with hyperplastic esophageal polyps. The most common location of the polyps was in the region of the gastroesophageal junction (67%) followed by the distal esophagus (30%) and the mid-esophagus (3%). Multiple (two) polyps were seen in only 3 patients in this particular series.
The mucosa comprising mostly of the hyperplastic polyps included cardiac-type (80%). Squamous mucosa was seen in 17%, and intestinal metaplasia was seen in two polyps (6.7%). Barrett's esophagus was present in only 15% of patients in Abraham and colleagues' [1] study. There was also a strong association between hyperplastic polyps of the gastroesophageal junction and the presence of active or recent esophageal ulceration or erosion (67%) noted.
The cause of esophageal polyps is still yet undetermined. Gastrointestinal reflux disease appears to play a large role, but other causes may also play a role in a select subset of patients. Factors such as medication-associated esophagitis, infectious esophagitis, severe vomiting, anatomic considerations (ie, Calcinosis, Raynaud's phenomenon, Esophageal dysmotility disorders, sclerodactyly, and telangiectasia [CREST], dysmotility syndromes), and inflammatory bowel diseases may also play a role in the development of esophageal polyps [1–3].
Some authors have suggested that patients with polyps tend to be symptomatic at the time of diagnosis. Brock and colleagues [4] in a series of 14 patients with adenocarcinoma in distal esophageal polyps undergoing esophagectomies noted that all but one of the 14 patients (93%) were symptomatic at diagnosis. The majority of patients in this series presented with gastrointestinal bleeding or gastroesophageal reflux disease (79%). Patients with polyps had early-stage esophageal adenocarcinoma, and Barrett's was present in 71% of these patients. High-grade dysplasia was seen in 80% of the cases and most of the polyps contained poorly-differentiated adenocarcinomas.
Whether or not polyps of the esophagus are one marker in the progression of the dysplasia-to-carcinoma sequence is unknown. Thurberg and colleagues' [5] report on five cases with sessile or pedunculated polypoid lesions of the esophagus associated with Barrett's esophagus revealed adenocarcinoma arising within the polyp in four of the cases, and one case in the adjacent Barrett's esophagus [5].
Given the uncertainty of the degree of association of esophageal polyposis with progression to adenocarcinoma, resection is recommended, particularly in patients with associated Barrett's esophagus. Endoscopic mucosal resection of large hyperplastic polyps in patients with Barrett's esophagus has been described in the literature [6]. In a small series of 3 patients with polyps and low-grade dysplasia, metaplastic tissue was completely excised in 2 patients. Esophageal recurrence occurred in 1 patient that was successfully treated with endoscopic mucosal resection [6]. In the rare case of diffuse polypoid esophagus, esophagectomy is the standard of care. As in the case presented, there are often diffuse areas of dysplasia (low-grade and high-grade) and early cancer can not be identified without complete resection. In all cases, the presence of esophageal polyps demands that investigation of the associated esophageal mucosa be undertaken to assess the underlying contributing conditions (ie, gastrointestinal reflux disease, Barrett's, adenocarcinoma, infection, Crohn's, and so forth) that appropriately directed therapy can be undertaken.
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References
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- Abraham SC, Singh VK, Yardley JH, Wu TT. Hyperplastic polyps of the esophagus and esophagogastric junction: histologic and clinicopathologic findings Am J Surg Pathol 2001;25:1180-1187.[Medline]
- Murney Jr RG, Huston JD. Endoscopic evaluation of the esophagogastric polyp and fold Gastrointest Endosc 1983;29:294-296.[Medline]
- Cockey BM, Jones B, Bayless TM, et al. Filiform polyps of the esophagus with inflammatory bowel disease AJR Am J Roentgenol 1985;144:1207-1208.[Free Full Text]
- Brock MV, Are C, Wu TT, Yang SC, Heitmiller RF. Polypoid esophageal adenocarcinoma-a clinicopathological variant of esophageal cancer? Curr Surg 2002;59:336-341.[Medline]
- Thurberg BL, Duray PH, Odze RD. Polypoid dysplasia in Barrett's esophagus: a clinicopathologic, immunohistochemical, and molecular study of five cases Hum Pathol 1999;30:745-752.[Medline]
- De Ceglie A, Lapertossa G, Blanchi S, et al. Endoscopic mucosal resection of large hyperplastic polyps in 3 patients with Barrett's esophagus World J Gastroenterol 2006;12:5699-5704.[Medline]
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Abstract
Introduction
