Three Cases of Esophageal Squamous Papilloma: A Rare and Poorly Understood Lesion

Abstract

Squamous papilloma is a rare lesion of the esophagus. The estimated prevalence is between 0.07% and 0.45% on endoscopy. The pathogenesis remains uncertain, however underlying inflammation from reflux, as well as chronic mechanical/chemical irritation have been implicated. Further, human papilloma virus has been proposed as a risk factor. Although currently considered a benign lesion, the malignant potential of esophageal squamous papilloma in causing squamous cell carcinoma is controversial.Figure 1Figure 2Figure 3We present three cases of esophageal squamous papilloma. In the first, a 51 year old asymptomatic African American female with multiple sclerosis on dimethyl fumarate underwent esophagogastroduodenoscopy (EGD) for workup of iron deficiency anemia. On exam, the Z-line appeared irregular and a single 2mm white-pink wart like projection was seen in the middle esophagus. Excisional biopsy was performed. Pathology revealed squamous papilloma with biopsies of the distal esophagus showing chronic inflammation. Of note, a recent pap smear was negative for HPV. In the second case, a 28 year old Hispanic male with 2 months of epigastric abdominal pain, and hematemesis was seen for further evaluation. An EGD revealed a single 5mm nonbleeding exophytic projection 33cm from the incisors. Excisional biopsy showed squamous papilloma. In the final case, a 42 year old female underwent evaluation for abdominal pain and melena. EGD demonstrated a single 5mm exophytic nodule in the middle esophagus and diffuse inflammation in the stomach. The lesion was excised and showed a squamous papilloma. The present cases demonstrate the heterogeneity in gender, age, esophageal location, and risk factors of squamous papilloma. Prior reports from Western countries demonstrate a male predominance, average 48 years of age and location in the lower esophagus, thought to be related to reflux disease. However, in the present series two of the three patients were female and had lesions in the middle esophagus. One patient was 28 years of age without any signs of reflux. Moreover, the present series reports the first case of esophageal squamous papilloma in a patient taking dimethyl fumarate, an agent known to increase papillomas and squamous cell carcinomas of the forestomach in mice and rat models. Further studies are needed to understand pathogenesis and risk factors of esophageal papilloma to identify at-risk patients since the natural history and malignant potential remain poorly understood.