IMAGE OF THE MONTH An Unexpected Cause of Dysphagia Keyur Parikh,*,‡ Jonathan Umbel,*,‡ and Deepak Venkat*,‡ *Division of Gastroenterology and Liver Disease University Hospitals, Case Medical Center, Cleveland, Ohio; and ‡Division of Gastroenterology and Liver Disease, Louis Stokes Cleveland VA Medical Center, Cleveland, Ohio
39-year-old man with a history of human immunodeficiency virus infection and intermittent compliance with antiretroviral therapy with a cluster of differentiation 4 cell count of 26 cells/mm3 was admitted for dysphagia/odynophagia and diarrhea. His history was notable for multiple episodes of esophageal candidiasis and oropharyngeal lesions caused by human papillomavirus (HPV) and herpes simplex virus, respectively. For his diarrhea, he was found to be positive for Clostridium difficile infection, and this resolved after treatment with metronidazole alone. He concurrently complained of a 2-week history of recurrent dysphagia, odynophagia, and globus sensation in his throat that was worse with solids more so than liquids. He denied regurgitation or difficulty tolerating secretions. He was empirically treated with intravenous micafungin because he had previously been infected with fluconazole-resistant species of Candida; however, his symptoms did not resolve, and an esophagogastroduodenoscopy was pursued. Endoscopic findings showed evidence of hyperkeratotic and hypertrophic appearing lesions (Figure A) diffusely scattered throughout the entire length of his esophagus in addition to his hypopharynx (Figure B) without endoscopic evidence of the classically described white plaques associated with esophageal candidiasis or esophageal ulceration suggestive of cytomegalovirus or herpes simplex virus esophagitis. These lesions were biopsied and then sent for pathologic examination (Figure C), which found evidence of epithelial hyperparakeratosis and koilocytotic changes consistent with HPV infection subtype p16.
A
Although HPV-related squamous cell carcinoma is a well-characterized etiology causing esophageal odynophagia and globus sensation,1 benign HPV-related lesions causing these symptoms are less understood, and data are limited to case reports and series.2,3 This patient had lesions diffusely in his hypopharynx that likely produced his globus sensation with odynophagia, and the scattered verrucae throughout his esophagus may have contributed to his symptoms of dysphagia. The most important step in the management of this patient is to ensure close adherence to his antiretroviral therapy because improved immune system function should suppress HPV viral activity and resolve these lesions. Although there are no clear guideline recommendations regarding endoscopic surveillance of HPV-associated benign lesions, we believe that surveillance is warranted because HPV subtype p16 infection is associated with an increased risk of squamous cell carcinoma. In addition, there is an increased risk for developing the much rarer but equally significant condition of verrucous carcinoma, which can arise from hyperkeratotic plaques despite previously benign pathology.4 The patient in question was referred for a repeat esophagogastroduodenoscopy in 1 year for surveillance. On follow-up, the patient continues to remain noncompliant with his highly active antiretroviral therapy, and his cluster of differentiation 4 count remains below 10 cells/mm3 several months after his reported presentation. He has been admitted repeatedly with similar complaints. This case highlights the importance of endoscopy and consideration of atypical causes of dysphagia in the immunosuppressed patient.
Clinical Gastroenterology and Hepatology 2015;13:xxxiii–xxxiv
IMAGE OF THE MONTH, continued References 1.
McIlwain WR, Sood AJ, Nguyen SA, et al. Initial symptoms in patients with HPV-positive and HPV-negative oropharyngeal cancer. JAMA Otolaryngol Head Neck Surg 2014;140:441–447.
2.
Sandvik AK, Aase S, Kveberg KH, et al. Papillomatosis of the esophagus. J Clin Gastroenterol 1996;22:35–37. Fekete F, Chazouilleres O, Ganthier V, et al. [A case of esophageal papillomatosis in adults]. Gastroenterol Clin Biol 1988; 12:66–70.
3.
xxxiv
4.
Tonna J, et al. Esophageal verrucous carcinoma arising from hyperkeratotic plaques associated with human papilloma virus type 51. Dis Esophagus 2010;23:E17–E20.
Conflicts of interest The authors disclose no conflicts. © 2015 by the AGA Institute 1542-3565/$36.00 http://dx.doi.org/10.1016/j.cgh.2015.02.025
39-year-old man with a history of human immunodeficiency virus infection and intermittent compliance with antiretroviral therapy with a cluster of differentiation 4 cell count of 26 cells/mm3 was admitted for dysphagia/odynophagia and diarrhea. His history was notable for multiple episodes of esophageal candidiasis and oropharyngeal lesions caused by human papillomavirus (HPV) and herpes simplex virus, respectively. For his diarrhea, he was found to be positive for Clostridium difficile infection, and this resolved after treatment with metronidazole alone. He concurrently complained of a 2-week history of recurrent dysphagia, odynophagia, and globus sensation in his throat that was worse with solids more so than liquids. He denied regurgitation or difficulty tolerating secretions. He was empirically treated with intravenous micafungin because he had previously been infected with fluconazole-resistant species of Candida; however, his symptoms did not resolve, and an esophagogastroduodenoscopy was pursued. Endoscopic findings showed evidence of hyperkeratotic and hypertrophic appearing lesions (Figure A) diffusely scattered throughout the entire length of his esophagus in addition to his hypopharynx (Figure B) without endoscopic evidence of the classically described white plaques associated with esophageal candidiasis or esophageal ulceration suggestive of cytomegalovirus or herpes simplex virus esophagitis. These lesions were biopsied and then sent for pathologic examination (Figure C), which found evidence of epithelial hyperparakeratosis and koilocytotic changes consistent with HPV infection subtype p16.
A
Although HPV-related squamous cell carcinoma is a well-characterized etiology causing esophageal odynophagia and globus sensation,1 benign HPV-related lesions causing these symptoms are less understood, and data are limited to case reports and series.2,3 This patient had lesions diffusely in his hypopharynx that likely produced his globus sensation with odynophagia, and the scattered verrucae throughout his esophagus may have contributed to his symptoms of dysphagia. The most important step in the management of this patient is to ensure close adherence to his antiretroviral therapy because improved immune system function should suppress HPV viral activity and resolve these lesions. Although there are no clear guideline recommendations regarding endoscopic surveillance of HPV-associated benign lesions, we believe that surveillance is warranted because HPV subtype p16 infection is associated with an increased risk of squamous cell carcinoma. In addition, there is an increased risk for developing the much rarer but equally significant condition of verrucous carcinoma, which can arise from hyperkeratotic plaques despite previously benign pathology.4 The patient in question was referred for a repeat esophagogastroduodenoscopy in 1 year for surveillance. On follow-up, the patient continues to remain noncompliant with his highly active antiretroviral therapy, and his cluster of differentiation 4 count remains below 10 cells/mm3 several months after his reported presentation. He has been admitted repeatedly with similar complaints. This case highlights the importance of endoscopy and consideration of atypical causes of dysphagia in the immunosuppressed patient.
Clinical Gastroenterology and Hepatology 2015;13:xxxiii–xxxiv
IMAGE OF THE MONTH, continued References 1.
McIlwain WR, Sood AJ, Nguyen SA, et al. Initial symptoms in patients with HPV-positive and HPV-negative oropharyngeal cancer. JAMA Otolaryngol Head Neck Surg 2014;140:441–447.
2.
Sandvik AK, Aase S, Kveberg KH, et al. Papillomatosis of the esophagus. J Clin Gastroenterol 1996;22:35–37. Fekete F, Chazouilleres O, Ganthier V, et al. [A case of esophageal papillomatosis in adults]. Gastroenterol Clin Biol 1988; 12:66–70.
3.
xxxiv
4.
Tonna J, et al. Esophageal verrucous carcinoma arising from hyperkeratotic plaques associated with human papilloma virus type 51. Dis Esophagus 2010;23:E17–E20.
Conflicts of interest The authors disclose no conflicts. © 2015 by the AGA Institute 1542-3565/$36.00 http://dx.doi.org/10.1016/j.cgh.2015.02.025
