Squamous papilloma is a rare fleshy pink-colored epithelial tumor first described by Adler et al in 1959 as a wart-like solitary lesion located mostly in the mid to distal esophagus.24 Analysis of the previous endoscopy series showed a prevalence ranging from 0.01% to 0.45%.1 It is more common among middle-aged men with a reported higher prevalence in Europe, accounting for approximately 70% of cases.17 Near-circumferential involvement of the esophagus, as in our patient, is extremely rare.
The etiology of ESP remains unclear, although roles of chronic mucosal irritation and HPV infection have been proposed. Prolonged chemical and mechanical mucosal irritation may generate a cellular damage-repair response followed by hyperregeneration. This hypothesis seems supported by the higher prevalence of gastroesophageal reflux disease, previous esophageal dilation, prolonged nasogastric intubation, and nitrosamine exposure among patients with ESP.17,18,25,26 Infection with HPV may also be associated with an increased risk of having esophageal squamous cell papillomas based on some observational studies.20,21 Nonetheless, it remains controversial because of extreme prevalence variability among the reported cases.22 Others suggested that tobacco and alcohol abuse are associated with an increased lifetime risk of having esophageal squamous papillomas.2,17
Patients are typically asymptomatic but can rarely present with heartburn or dysphagia.3,4,9,13,15,27 Diagnosis is usually made during upper endoscopy performed for a different indication as they appear as single or multiple round lesions that rarely exceed 1 cm in diameter. Histologically, they appear as finger-like projections of squamous epithelial cells with a connective tissue core containing small blood vessels.21 Diffuse esophageal involvement with multiple lesions is exceedingly rare.
Examination of the existing literature has shown a variable clinical course, ranging from spontaneous regression to the development of squamous cell carcinoma.23 Although it is unknown whether solitary esophageal papillomas are premalignant lesions because of the highly variable clinical course, ESP carries a significant potential of malignant transformation, as squamous cell carcinomas were identified in almost half of the reported cases.2,4–8,11,28 Thus, early diagnosis and treatment remain imperative.
Considering the benign nature of solitary lesions, treatment has been reserved only for symptomatic cases. Endoscopic resection with biopsy forceps, snare polypectomy, and cautery have been used for small single lesions. Treating multiple lesions has been challenging, and the optimal management and surveillance guidelines remain unclear because of the paucity of reported cases. Some patients with extensive ESP underwent esophagectomy because of associated dysplasia and progression to carcinoma.4,8 Nevertheless, previous methods used include endoscopic resection, laser removal, radiofrequency ablation, and cryotherapy, with variable rates of failure and recurrence.3,4,10,11,15,16,27
SCY delivers cold nitrogen gas via a catheter introduced into the esophagus through an accessory channel of an upper GI endoscope. Esophagoscopy is performed under monitored anesthesia care, and cold nitrogen gas is sprayed over the affected portion of the esophagus under direct visualization. Freezing of the esophageal mucosa is evidenced as white cryoburn with sharply demarcated margins.
It should be noted that the truFreeze SCY system is currently approved for treatment of Barrett's esophagus with low-grade dysplasia and high-grade dysplasia and malignancies. Therefore, SCY use in ESP management is considered off-label.
Previous studies suggested SCY as an effective option for treating mucosal cancer, which was not amenable to conventional treatment. There has been only 1 case report of ESP treated with SCY.15 Our patient underwent 3 sessions of SCY, and fourth surveillance endoscopy showed near-complete resolution. We believe that this case report has an important clinical implication regarding effective management of ESP, as cryotherapy is considered easy to use, minimally invasive, and has been associated with a low rate of complications and no hospital stay.
Author contributions: M. Alomari acquired and analyzed the data and drafted the manuscript. V. Wadhwa reviewed the literature and drafted the manuscript. T. Erim critically revised the manuscript for important intellectual content, and is the article guarantor. P. Bejarano obtained histopathology slides. P. Amar performed follow-up endoscopies.
Financial disclosure: None to report.
Informed consent was obtained for this case report.
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