Clinical Case Summary
This case presents a 64-year old man with verrucous carcinoma of the esophagus (VCE), a rare variant of squamous cell carcinoma. The patient has a history of type 2 diabetes and tobacco abuse, and presented in September 2019 with complaints of dysphagia. Upper endoscopy revealed reflux-, and candida esophagitis, and a proximal stenosis. Treatment was initiated with proton pump inhibitors, antifungal therapy, and dilatations. Over three years, multiple endoscopies were conducted, almost all revealing extensive white hyperkeratotic and exophytic lesions. Histopathology reports described fungal threads and no dysplasia, leading to a diagnosis of therapy-resistant candida esophagitis due to poorly regulated diabetes. After two times failure of intravenous antifungal therapy, in February 2023, the patient was referred to our center, a tertiary hospital. Targeted bite on bite biopsies revealed candida aggregates, parakeratosis, and squamous cell proliferation, highly suggestive of well-differentiated VCE. A PET-CT showed no signs of metastatic disease; therefore, considering the early stage and endoscopic extent of the lesion, a laparoscopic esophagostomy was performed.
The prolonged interval from clinical presentation to VCE diagnosis is thought to result from the hyperkeratotic growth pattern, which causes inadequate material for histologic confirmation of underlying neoplastic tissue. Additionally, the presence of fungal threads and white lesions results in diagnosis of candida esophagitis, misleading physicians and causing diagnostic delay. In patients with therapy-resistant candida esophagitis and atypical hyperkeratotic esophageal lesions VCE should be considered, enabling early diagnosis and treatment.
References
Figures that will be presented when accepted:
Figure 1: upper GI endoscopy during initial presentation showing white/yellow keratotic plaques and a proximal stenosis.
Figure 2: upper GI endoscopy at time of VCE diagnosis at our center 4-years after the initial presentation, showing a circumferentially narrowed esophagus, with white, granular and keratotic plaques and lesions at white light endoscopy
Figure 3: upper GI endoscopy at time of VCE diagnosis at our center 4-years after the initial presentation, with NBI showing a circumferentially narrowed esophagus, and diffusely irregular mucosa with white plaques and keratotic lesions.
References:
1. Sweetser S, Jacobs NL, Wong Kee Song LM. Endoscopic diagnosis and treatment of esophageal verrucous squamous cell cancer. Dis Esophagus. 2014;27(5):452-6.
2. Egeland C, Achiam MP, Federspiel B, Svendsen LB. Verrucous Squamous Cell Cancer in the Esophagus: An Obscure Diagnosis. Case Rep Gastroenterol. 2016;10(2):466-71.
3. Tabuchi S, Koyanagi K, Nagata K, Ozawa S, Kawachi S. Verrucous carcinoma of the esophagus: a case report and literature review. Surg Case Rep. 2020;6(1):35.