Unusual Esophageal Mass as a Cause of Dysphagia

Abstract

Question: A 73-year-old man with underlying alcoholic liver cirrhosis presented with a 1-month history of dysphagia. Upper endoscopy performed by his primary physician showed a polypoid lesion with a stricture in the middle esophagus; thus, he was referred for management. The patient has a 25 pack-year smoking history. Physical examination and laboratory testing revealed no remarkable abnormalities. Repeat endoscopy showed a small polypoid lesion but the endoscope could not be inserted due to severe stricture. Ultrathin endoscopy (GIF-XP260N; Olympus Optical Co., Ltd, Tokyo, Japan) revealed a 1.0-cm polypoid lesion with normal appearing mucosa in the esophagus, 27 cm from the incisors (Figure A). A biopsy from the lesion revealed proliferated and dysregulated epithelia throughout the mucosal layer, which was consistent with squamous intraepithelial neoplasm. Computed tomography scan showed only short segmental thickening of the middle esophageal wall. Because the patient declined surgery, we performed endoscopic resection (Figure B); however, remnant tumor under the small polypoid lesion could not be approached endoscopically (Figure C). Pathologically, the lobulating mass had proliferation of the capillaries with inflammatory cell infiltration and edematous stroma below the normal stratified squamous epithelium (Figure D). There was also disorganized squamous epithelium in the area surrounding spindle-shaped lymphovascular endothelia (Figure E). What is the diagnosis? See the Gastroenterology web site (www.gastrojournal.org) for more information on submitting your favorite image to Clinical Challenges and Images in GI. Pyogenic granuloma (PG), a polypoid capillary hemangioma, is a benign lesion arising from blood vessels of the skin or the oral and nasal mucosa.1Kusakabe A. Kato H. Hayashi K. et al.Pyogenic granuloma of the stomach successfully treated by endoscopic resection after transarterial embolization of the feeding artery.J Gastroenterol. 2005; 40: 530-535Crossref PubMed Scopus (25) Google Scholar In contrast with classic capillary hemangiomas, PG is composed of numerous newly formed capillaries of variable sizes arranged in a distinctive lobular pattern with or without ulceration on the surface. PGs are usually diagnosed in children and young adults and are rarely found in the alimentary tract. To date, only 42 cases of PGs in the alimentary tract have been reported, with 23 cases located in the esophagus.2Seoung H.G. Kim G.H. Song G.A. et al.Esophageal pyogenic granuloma: endosonographic findings and endoscopic treatments.Clin Endosc. 2013; 46: 81-84Crossref PubMed Scopus (6) Google Scholar No case of malignant transformation has been reported. To our knowledge, this is the first described case of PG associated with esophageal squamous cell carcinoma. Although the precise etiology is unknown, suggested etiologic factors include mechanical trauma, infection, chemical irritation, pregnancy, and hormonal mechanisms that induce reactive inflammatory and abnormal vascular responses.2Seoung H.G. Kim G.H. Song G.A. et al.Esophageal pyogenic granuloma: endosonographic findings and endoscopic treatments.Clin Endosc. 2013; 46: 81-84Crossref PubMed Scopus (6) Google Scholar Previously, a case of PG was reported originating in Barrett’s epithelium.3Craig R.M. Carlson S. Nordbrock H.A. et al.Pyogenic granuloma in Barrett's esophagus mimicking esophageal carcinoma.Gastroenterology. 1995; 108: 1894-1896Abstract Full Text PDF PubMed Scopus (26) Google Scholar In this case, the patient was treated with repeated dilations because of stricture formation caused by severe esophagitis, suggesting that it had been caused by trauma. However, the pathogenesis of co-occurring PG and squamous cell carcinoma is unclear in our case. Endoscopically, PG is seen as a protruding tumor or, sometimes, a pedunculated mass, with its color ranging from dark red to friable sanguine, reflecting the blood-rich nature of the lesion.1Kusakabe A. Kato H. Hayashi K. et al.Pyogenic granuloma of the stomach successfully treated by endoscopic resection after transarterial embolization of the feeding artery.J Gastroenterol. 2005; 40: 530-535Crossref PubMed Scopus (25) Google Scholar Although small polypoid lesions can be removed endoscopically, surgical excision is required in patients with large or deep lesions, or lesions that mimic malignant transformation.1Kusakabe A. Kato H. Hayashi K. et al.Pyogenic granuloma of the stomach successfully treated by endoscopic resection after transarterial embolization of the feeding artery.J Gastroenterol. 2005; 40: 530-535Crossref PubMed Scopus (25) Google Scholar Symptoms of esophageal PG include dysphagia, retrosternal pain, weight loss, and bleeding. PG should be differentially diagnosed from inflammatory polyps, benign and malignant vascular malformations, and angiomatous variants of Kaposi sarcoma.1Kusakabe A. Kato H. Hayashi K. et al.Pyogenic granuloma of the stomach successfully treated by endoscopic resection after transarterial embolization of the feeding artery.J Gastroenterol. 2005; 40: 530-535Crossref PubMed Scopus (25) Google Scholar Surgical resection would have been the best treatment choice in our case; however, the patient declined operative intervention. Instead, he received regional radiation of 55.8 Gy; an alternative treatment would have been photodynamic therapy. After radiation treatment, complete remission was achieved and the stricture improved (Figure F). Follow-up esophagogastroduodenoscopy at 3 and 6 months showed no evidence of recurrence.