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Vol. 41. Núm. 1.
Páginas 31-32 (Enero 2018)
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Vol. 41. Núm. 1.
Páginas 31-32 (Enero 2018)
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Lymphocytic esophagitis: A rare finding in adult patients with dysphagia and food impaction
Esofagitis linfocítica: una observación poco frecuente en pacientes adultos con disfagia e impactación alimentaria
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Pedro L. Gonzalez-Corderoa,
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pelu7_8@hotmail.com

Corresponding author.
, Alejandro Rubio-Fernándezb, Nuria Fernandez-Gonzalezb, Carmen Dueñas-Sadornila, Javier Molina-Infantea
a Department of Gastroenterology, Hospital Universitario San Pedro de Alcantara, Caceres, Spain
b Department of Pathology, Hospital Universitario San Pedro de Alcantara, Caceres, Spain
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Table 1. Updated prevalence rates of LyE in literature, including the present report.
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Lymphocytic esophagitis (LyE) is a novel clinicopathological condition, first described in 2006 by Rubio et al. in Sweden,1 in a series of 20 patients as a histological subset of chronic esophagitis characterized by >20 intraepithelial lymphocytes (IELs) per high-power field (HPF) with no more than rare granulocytes. Since then, a number of case reports from all over the world have confirmed the identity of this new condition.2

We report two recent patients from our center meeting clinical and histopathologic criteria compatible with LyE.

Case #1: A 52 yr-old male with smoking habit and a history of kidney stones presented with heartburn and non-progressive dysphagia over the past year. Upper endoscopy revealed no caliber or mucosal abnormalities and esophageal biopsies were taken to rule out eosinophilic esophagitis (EoE). Histopathologic analysis exhibited increased IELs increased (≥25/HPF) with marked basal–parabasal cell hyperplasia and intercellular edema (spongiosis). No eosinophils or neutrophils were detected (Fig. 1).

Figure 1.

Left panel. Esophageal biopsy revealing heavy lymphocytic infiltration with papillar and peripapillar localization (white arrows) and intercellular edema (spongiosis), in the absence of either neutrophils or eosinophils. Right panel. A more detailed picture of lymphocytic infiltration, showing >25 CD4+ intraepithelial lymphocytes per 100 epithelial cells.

(0,3MB).

Case #2: A 63-year-old male with a history of hypertension, type 2 diabetes mellitus, ischemic cardiopathy and dyslipemia presented with recurrent esophageal food impaction requiring three emergency endoscopies. The patient denied previous heartburn and/or regurgitation. Esophageal endoscopic appearance was normal and esophageal biopsies revealed no eosinophilic but dense intraepithelial lymphocytosis (≥40/HPF) with papillar and peripapillar localization, and intercellular edema.

To the best of our knowledge, these LyE cases are the first reported in Spain. The prevalence of LyE has been reported to be 0.1% in patients with oesophageal biopsies,2 albeit a recent study from Sweden has reported that LyE might be present in up to 9% of patients with food impaction3 (Table 1). In our experience as a referral center for EoE over the past decade, we have only found two cases in 967 patients (0.2%) evaluated with esophageal biopsies due to dysphagia or food impaction (Table 1).

Table 1.

Updated prevalence rates of LyE in literature, including the present report.

First author, country, year of publication,  Population  Number of cases  Prevalence 
Rokkainen J, Finland, 20055  1000 randomly selected community subjects with esophageal biopsies  0% 
Haque S, US, 20122  129,252 patients with esophageal biopsies  119  0.1% 
Pasricha S, US, 20164  Patients with esophageal biopsies (2000–2012)  27  Not reported 
Truskaite K, Sweden, 20163  185 patients with food bolus impaction and esophageal biopsies  16  9% 
Present report, Spain, 2016  972 patients with esophageal biopsies due to dysphagia/food impaction (2007–2016)  0.2% 

LyE is more common in older women without atopic conditions referring dysphagia, food impaction or gastro-esophageal reflux disease (GERD) symptoms.3,4 Association with Crohn's disease in children has shown discrepant results,6,7 whereas in adults it has been consistently associated with (GERD) and non-achalasia primary motility abnormalities.3,4,8 LyE has been proposed as an inflammatory non-specific response to different sources of esophageal injury, including radiofrequency ablation for Barrett's esophagus.9 Undoubtedly, LyE currently remains a histologic pattern in search of a disease.10 In our experience, consistent with most previous reports,2,5 it is still a rare finding (0.2%) in patients evaluated because of dysphagia or food impaction. Although this condition clinically and endoscopically may resemble EoE, the underlying etiology, its clinical significance and course, and best treatment remain to be ascertained in further studies.

References
[1]
C.A. Rubio, K. Sjödahl, J. Lagergren.
Lymphocytic esophagitis: a histologic subset of chronic esophagitis.
Am J Clin Pathol, 125 (2006), pp. 432-437
[2]
S. Haque, R.M. Genta.
Lymphocytic oesophagitis: clinicopathological aspects of an emerging condition.
[3]
K. Truskaite, A. Dlugosz.
Prevalence of eosinophilic esophagitis and lymphocytic esophagitis in adults with esophageal food bolus impaction.
Gastroenterol Res Pract, (2016), pp. 9303858
[4]
Pasricha S, Gupta A, Reed CC, Speck O, Woosley JT, Dellon ES. Lymphocytic esophagitis an emerging clinicopathologic disease associated with dysphagia. Dig Dis Sci. 2016 Jun 24. [Epub ahead of print].
[5]
J. Ronkainen, N.J. Talley, P. Aro, T. Storskrubb, S.E. Johansson, T. Lind, et al.
High prevalence of gastroesophageal reflux symptoms and esophagitis with or without symptoms in the general adult Swedish population: a Kalixanda study report.
Scand J Gastroenterol, 40 (2005), pp. 275-285
[6]
Putra J, Muller KE, Hussain ZH, Parker S, Gabbard S, Brickley, et al. Lymphocytic esophagitis in nonachalasia primary esophageal motility disorders: improved criteria, prevalence, strength of association, and natural history. Am J Surg Pathol. 2016 Aug 11. [Epub ahead of print].
[7]
J.K. Purdy, H.D. Appelman, C.P. Golembeski, B.J. McKenna.
Lymphocytic esophagitis: a chronic or recurring pattern of esophagitis resembling allergic contact dermatitis.
Am J Clin Pathol, 130 (2008), pp. 508-513
[8]
L.M. Sutton, D.D. Heintz, A.S. Patel, A.G. Weinberg.
Lymphocytic esophagitis in children.
Inflamm Bowel Dis, 20 (2014), pp. 1324-1328
[9]
J. Kissiedu, P.N. Thota, T. Gohel, R. Lopez, I.O. Gordon.
Post-ablation lymphocytic esophagitis in Barrett esophagus with high grade dysplasia or intramucosal carcinoma.
Mod Pathol, 29 (2016), pp. 599-606
[10]
J. Ronkainen, M.M. Walker, P. Aro, T. Storskrubb, N.J. Talley, Z.B. Ahmed, et al.
Lymphocytic oesophagitis, a condition in search of a disease?.
Gut, 61 (2012), pp. 1776
Copyright © 2016. Elsevier España, S.L.U., AEEH y AEG
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