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Published in: Digestive Diseases and Sciences 12/2017

01-12-2017 | Original Article

A Statistically Significant Association Between Esophageal Granular Cell Tumors and Eosinophilic Esophagitis: A 16-year Analysis at Two Large Hospitals of 167,434 EGDs

Authors: Borko Nojkov, Mitual Amin, Gehad Ghaith, Mitchell S. Cappell

Published in: Digestive Diseases and Sciences | Issue 12/2017

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Abstract

Background

Esophageal granular cell tumor (eGCT) is a rare, usually benign, neoplasm of neuroectodermic origin. Eosinophilic esophagitis (EoE) is a relatively uncommon, immune-mediated, chronic disease. Both diseases commonly present with dysphagia. One case has been reported of simultaneous occurrence of both diseases.

Aims

To determine the association between diseases.

Methods

The present study was an IRB-approved, retrospective review of esophagogastroduodenoscopies (EGDs) with esophageal biopsies from two large hospitals, 1999–2014.

Results

Among 29,235 EGDs with esophageal biopsies for 16 years (167,434 total EGDs), 16 patients had pathologically diagnosed eGCT, and 1225 patients had pathologically diagnosed EoE. Five (31%) of 16 patients with eGCT had concomitant EoE (p = 0.001, OR 10.43, 95% ORCI 3.16–32.44, Fisher’s exact test). Patients with simultaneous eGCT and EoE were young (mean age = 33.6 ± 12.9 years). Three were female. Dysphagia was presenting symptom in 4 (80%) of patients. Three had asthma. All five patients had > 20 eosinophils/hpf in esophageal biopsy specimens. Three patients had endoscopic esophageal abnormalities suggesting EoE. Four patients were treated with a PPI (before and after diagnosis of EoE), and 2 patients underwent six-food-elimination diet with partial symptomatic improvement. The eGCTs averaged 13.4 ± 4.2 mm in maximal diameter and were located in upper-2, middle-2, and lower esophagus-2 (1 patient had 2 eGCTs). eGCTs were endoscopically resected-3 patients, and monitored-2 patients. Surveillance endoscopies revealed no recurrence or growth of eGCTs after resection (mean follow-up = 4.6 years).

Conclusions

This novel report of 5 patients with simultaneous EoE and eGCT adds to one, previously published case and suggests these two diseases are associated, and have a common pathophysiologic link, despite apparently different pathogenesis. Large, prospective, endoscopic and pathologic studies are warranted to further investigate this association.
Literature
1.
go back to reference Lewin KJ, Applebaum HD. Tumors of the esophagus and stomach. In: Atlas of tumor pathology, Series 3, vol. 18. Washington, DC: Armed Forces Institute of Pathology (AFIP); 1996. pp. 154–155. Lewin KJ, Applebaum HD. Tumors of the esophagus and stomach. In: Atlas of tumor pathology, Series 3, vol. 18. Washington, DC: Armed Forces Institute of Pathology (AFIP); 1996. pp. 154–155.
2.
go back to reference Gershwind ME, Chiat H, Addei KA, et al. Granular cell tumors of the esophagus. Gastrointest Radiol. 1978;2:327–330.CrossRefPubMed Gershwind ME, Chiat H, Addei KA, et al. Granular cell tumors of the esophagus. Gastrointest Radiol. 1978;2:327–330.CrossRefPubMed
3.
go back to reference Orlowska J, Pachlewski J, Gugulski A, Butruk E. A conservative approach to granular cell tumor of the esophagus: four case reports and literature review. Am J Gastroenterol. 1993;88:311–314.PubMed Orlowska J, Pachlewski J, Gugulski A, Butruk E. A conservative approach to granular cell tumor of the esophagus: four case reports and literature review. Am J Gastroenterol. 1993;88:311–314.PubMed
4.
go back to reference Liacouras CA, Furuta GT, Hirano I, et al. Eosinophilic esophagitis: updated consensus recommendations for children and adults. J Allergy Clin Immunol. 2011;128:3–20.CrossRefPubMed Liacouras CA, Furuta GT, Hirano I, et al. Eosinophilic esophagitis: updated consensus recommendations for children and adults. J Allergy Clin Immunol. 2011;128:3–20.CrossRefPubMed
7.
go back to reference Stefansson K, Wollmann RL. S-100 protein in granular cell tumors (granular cell myoblastomas). Cancer. 1982;49:1834–1838.CrossRefPubMed Stefansson K, Wollmann RL. S-100 protein in granular cell tumors (granular cell myoblastomas). Cancer. 1982;49:1834–1838.CrossRefPubMed
8.
go back to reference Dellon ES, Gonsalves N, Hirano I, et al. ACG clinical guideline: evidenced based approach to the diagnosis and management of esophageal eosinophilia and eosinophilic esophagitis (EoE). Am J Gastroenterol. 2013;108:679–692.CrossRefPubMed Dellon ES, Gonsalves N, Hirano I, et al. ACG clinical guideline: evidenced based approach to the diagnosis and management of esophageal eosinophilia and eosinophilic esophagitis (EoE). Am J Gastroenterol. 2013;108:679–692.CrossRefPubMed
9.
go back to reference Kim HP, Vance RB, Shaheen NJ, et al. The prevalence and diagnostic utility of endoscopic features of eosinophilic esophagitis: a meta-analysis. Clin Gastroenterol Hepatol. 2012;10:988–996.CrossRefPubMedPubMedCentral Kim HP, Vance RB, Shaheen NJ, et al. The prevalence and diagnostic utility of endoscopic features of eosinophilic esophagitis: a meta-analysis. Clin Gastroenterol Hepatol. 2012;10:988–996.CrossRefPubMedPubMedCentral
10.
go back to reference Nojkov B, Ghaith G, Cappell MS. Simultaneous occurrence of eosinophilic esophagitis and esophageal granular cell tumor: two case reports. Am J Gastroenterology. 2014;109:S247. doi:10.1038/ajg.2014.279. (Presented as poster P57 on October 19, 2014, American College of Gastroenterology annual convention in Philadelphia).CrossRef Nojkov B, Ghaith G, Cappell MS. Simultaneous occurrence of eosinophilic esophagitis and esophageal granular cell tumor: two case reports. Am J Gastroenterology. 2014;109:S247. doi:10.​1038/​ajg.​2014.​279. (Presented as poster P57 on October 19, 2014, American College of Gastroenterology annual convention in Philadelphia).CrossRef
12.
go back to reference Murali AR, Gupta A, Attar BM, Ravi V, Koduru P. Topical steroids in eosinophilic esophagitis: systematic review and meta-analysis of placebo-controlled randomized clinical trials. J Gastroenterol Hepatol. 2016;31:1111–1119. doi:10.1111/jgh.13281.CrossRefPubMed Murali AR, Gupta A, Attar BM, Ravi V, Koduru P. Topical steroids in eosinophilic esophagitis: systematic review and meta-analysis of placebo-controlled randomized clinical trials. J Gastroenterol Hepatol. 2016;31:1111–1119. doi:10.​1111/​jgh.​13281.CrossRefPubMed
13.
go back to reference Vitellas KM, Bennett WF, Bova JG, Johnston JC, Caldwell JH, Mayle JE. Idiopathic eosinophilic esophagitis. Radiology. 1993;186:789–793.CrossRefPubMed Vitellas KM, Bennett WF, Bova JG, Johnston JC, Caldwell JH, Mayle JE. Idiopathic eosinophilic esophagitis. Radiology. 1993;186:789–793.CrossRefPubMed
14.
go back to reference Orenstein SR, Shalaby TM, Di Lorenzo C, et al. The spectrum of pediatric eosinophilic esophagitis beyond infancy: a clinical series of 30 children. Am J Gastroenterol. 2000;95:1422–1430.CrossRefPubMed Orenstein SR, Shalaby TM, Di Lorenzo C, et al. The spectrum of pediatric eosinophilic esophagitis beyond infancy: a clinical series of 30 children. Am J Gastroenterol. 2000;95:1422–1430.CrossRefPubMed
15.
go back to reference Croese J, Fairley SK, Masson JW, et al. Clinical and endoscopic features of eosinophilic esophagitis in adults. Gastrointest Endosc. 2003;58:516–522.CrossRefPubMed Croese J, Fairley SK, Masson JW, et al. Clinical and endoscopic features of eosinophilic esophagitis in adults. Gastrointest Endosc. 2003;58:516–522.CrossRefPubMed
16.
go back to reference De Rezende L, Lucendo AJ, Alvarez-Argüellez H. Granular cell tumors of the esophagus. Report of 5 cases and review of diagnostic and therapeutic techniques. Dis Esophagus. 2007;20:436–443.CrossRefPubMed De Rezende L, Lucendo AJ, Alvarez-Argüellez H. Granular cell tumors of the esophagus. Report of 5 cases and review of diagnostic and therapeutic techniques. Dis Esophagus. 2007;20:436–443.CrossRefPubMed
17.
go back to reference Rothenberg ME. Eosinophilic gastrointestinal disorders (EGID). J Allergy Clin Immunol. 2004;113:11–28.CrossRefPubMed Rothenberg ME. Eosinophilic gastrointestinal disorders (EGID). J Allergy Clin Immunol. 2004;113:11–28.CrossRefPubMed
18.
19.
go back to reference Bischoff S, Crowe SE. Gastrointestinal food allergy: new insights into pathophysiology and clinical perspectives. Gastroenterology. 2005;128:1089–1113.CrossRefPubMed Bischoff S, Crowe SE. Gastrointestinal food allergy: new insights into pathophysiology and clinical perspectives. Gastroenterology. 2005;128:1089–1113.CrossRefPubMed
20.
go back to reference Onbasi K, Sin AZ, Doganavsargil B, et al. Eosinophil infiltration of the oesophageal mucosa in patients with pollen allergy during the season. Clin Exp Allergy. 2005;35:1423–1431.CrossRefPubMed Onbasi K, Sin AZ, Doganavsargil B, et al. Eosinophil infiltration of the oesophageal mucosa in patients with pollen allergy during the season. Clin Exp Allergy. 2005;35:1423–1431.CrossRefPubMed
21.
go back to reference Blanchard C, Stucke EM, Rodriguez-Jimenez B, et al. A striking local esophageal cytokine expression profile in eosinophilic esophagitis. J Allergy Clin Immunol. 2011;127:208–217.CrossRefPubMedPubMedCentral Blanchard C, Stucke EM, Rodriguez-Jimenez B, et al. A striking local esophageal cytokine expression profile in eosinophilic esophagitis. J Allergy Clin Immunol. 2011;127:208–217.CrossRefPubMedPubMedCentral
22.
go back to reference Sherrill JD, Kiran KC, Blanchard C, et al. Analysis and expansion of the eosinophilic esophagitis transcriptome by RNA sequencing. Genes Immun. 2014;15:361–369.CrossRefPubMedPubMedCentral Sherrill JD, Kiran KC, Blanchard C, et al. Analysis and expansion of the eosinophilic esophagitis transcriptome by RNA sequencing. Genes Immun. 2014;15:361–369.CrossRefPubMedPubMedCentral
23.
go back to reference Cappell MS, Lebwohl O. Esophageal dysmotility from a small esophageal granular cell tumor. J Clin Gastroenterol. 1991;13:432–435.CrossRefPubMed Cappell MS, Lebwohl O. Esophageal dysmotility from a small esophageal granular cell tumor. J Clin Gastroenterol. 1991;13:432–435.CrossRefPubMed
24.
go back to reference Aceves SS, Newbury RO, Dohil R, et al. Esophageal remodeling in pediatric eosinophilic esophagitis. J Allergy Clin Immunol. 2007;119:206–212.CrossRefPubMed Aceves SS, Newbury RO, Dohil R, et al. Esophageal remodeling in pediatric eosinophilic esophagitis. J Allergy Clin Immunol. 2007;119:206–212.CrossRefPubMed
25.
go back to reference Philpott H, Nandurkar S, Thien F, et al. Eosinophilic esophagitis: a clinicopathological review. Pharmacol Ther. 2015;146C:12–22.CrossRef Philpott H, Nandurkar S, Thien F, et al. Eosinophilic esophagitis: a clinicopathological review. Pharmacol Ther. 2015;146C:12–22.CrossRef
Metadata
Title
A Statistically Significant Association Between Esophageal Granular Cell Tumors and Eosinophilic Esophagitis: A 16-year Analysis at Two Large Hospitals of 167,434 EGDs
Authors
Borko Nojkov
Mitual Amin
Gehad Ghaith
Mitchell S. Cappell
Publication date
01-12-2017
Publisher
Springer US
Published in
Digestive Diseases and Sciences / Issue 12/2017
Print ISSN: 0163-2116
Electronic ISSN: 1573-2568
DOI
https://doi.org/10.1007/s10620-017-4802-9

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