ESP is classified as a benign epithelial tumor of the esophagus, with an endoscopic incidence of 0.01%–0.45%.1 Although the etiology of ESP is unknown, it is suspected to have causes or contributing factors that include chronic irritation due to gastric acid reflux, smoking, alcohol, and human papillomavirus.2 In the present case, there were no findings such as esophagitis or esophageal hiatal hernia, and the involvement of human papillomavirus was negative in the pathological findings. ESP usually shows an incidental finding as a small (<0.5 cm) single lesion, most commonly located in the distal esophagus.1,3 In this rare case, multiple ESPs were present predominantly in the distal esophagus.
ESP is typically reported as fleshy pink in color, with a soft to warty texture; it usually exists in either a sessile or pedunculated form.3 ESP is difficult to diagnose based on a biopsy, and a sufficient amount of specimen is necessary for diagnosis. In the past, surgery was performed for the diagnosis, but now, less-invasive endoscopic mucosal resection is often recommended. We performed an endoscopic esophageal mucosal resection using a cap-fitted esophagogastroduodenoscope to obtain sufficient volume of samples. There have been no reports of ESP that could be diagnosed by the biopsy method to date.
Three main histologic patterns of ESP have been described. It is generally classified as the exophytic type (fingerlike squamous papillae overlying fibrovascular cores of lamina propria), the endophytic type (a round, smooth surface contour and an inverted papillomatous appearance), and the spiked type (a verrucoid appearance, a corrugated surface, hyperkeratosis, and a prominent granular cell layer).4 The exophytic type is more common although the present case led to its classification as the endophytic type. Prominent eosinophilic infiltrate to the epithelium was observed in this case, which may be also diagnosed as an eosinophilic esophagitis. A case of ESP associated with Goltz syndrome with eosinophilic esophagitis has been reported, but according to that report, eosinophilic esophagitis is considered as a disorder that related to connective tissue disease, and its association with ESP is unknown.5
ESP is benign but may be associated with cancer although extremely rare.2 However, whether these lesions represent de novo carcinomas from the onset or true malignant degeneration of a squamous papilloma is unknown.6 In addition, Cowden disease causing gastrointestinal polyposis could be an alternative diagnosis for the present case. However, it was denied by the physical findings, family history, and pathological findings (glycogenic acanthosis seen with Cowden disease). ESP often merges with Goltz syndrome, but the present case was totally different in terms of the absence of birth defects or skin symptoms.7,8
Small isolated lesions have been commonly and successfully treated by endoscopic resection. Because of the paucity of reported cases, however, the best clinical management of extensive ESP has remained unclear.2 ESPs should be considered as a differential diagnosis for esophageal polyps that even manifest as multiple polyps such as polyposis (eg, Cowden disease) in the esophagus.
Author contributions: S. Makise and K. Hiraka wrote the manuscript. H. Watanabe, K. Atsumi, K. Inoue, K. Miyajima, K. Makisumi, A. Mizushima, T. Sasaguri, D. Tsurumaru, H. Honda edited the manuscript. S. Makise is the article guarantor.
Financial disclosure: None to report.
Informed consent was obtained for this case report.
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© 2019 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of The American College of Gastroenterology.
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