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Esophageal Ectopic Sebaceous Glands

Bahdi, Firas MD1; Zarrin-Khameh, Neda MD, MPH2; Agarwal, Suneal Kumar MD, FACG3

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doi: 10.14309/crj.0000000000000508
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CASE REPORT

A 63-year-old White woman with gastroesophageal reflux disease, controlled with aggressive lifestyle interventions and high-dose proton pump inhibitors, underwent her first upper endoscopy to screen for Barrett's esophagus, which revealed numerous nonscrapable small yellowish plaques involving the middle and lower third of the esophagus diffusely (Figure 1). Given the broad macroscopic differential diagnosis, these lesions were biopsied for histopathological characterization. The histopathological examination of these esophageal biopsies using hematoxylin and eosin stains revealed stratified squamous epithelium with multiple lobulated sebaceous glands without hair follicles in the lamina propria consistent with esophageal ectopic sebaceous glands (EESG; Figure 2). There was no evidence of intestinal metaplasia, candida, or viral cytopathic effect. The patient was counseled about this benign finding, and no changes were made to her established management plan.

Figure 1.
Figure 1.:
Diffuse numerous nonscrapable small yellowish plaques involving the middle and distal third of the esophagus.
Figure 2.
Figure 2.:
Esophageal biopsy with hematoxylin and eosin stain: (A) low-power microscopy revealing stratified squamous epithelium with multiple subepithelial lobulated glands and (B) high-power microscopy revealing multiple lobulated sebaceous glands without hair follicles in the lamina propria.

EESG are a rare condition with an estimated incidence rate of 0.0046%.1 It is unclear whether EESG are the result of a congenital anomaly or a metaplastic change. The congenital heterotopia theory suggests abnormal separation of the embryonic layers during embryogenesis resulting in the presence of the ectodermal sebaceous glands in the endodermal esophagus.1,3,4 The metaplastic theory suggests these glands form after birth in response to unknown triggers that are likely found after puberty as there have been no reported cases found in the pediatric population.5

Epidemiologically, the mean age of diagnosis for reported cases of EESG was approximately 50 years with an approximate 1:1 male-to-female ratio.1,3,5 Approximately two-thirds of patients with EESG reported associated reflux or dyspepsia symptoms, while the remaining third of cases were incidentally diagnosed on upper endoscopy for alternative screening indications.4 The relationship between the EESG and gastroesophageal reflux disease or its severity remains unclear.

Endoscopically, the EESG usually manifest as nonscrapable small yellowish plaques involving the middle and lower third of the esophagus, ranging in size from 1 to 20 mm and in number from 1 to over a 100 lesions.3 The EESG are often macroscopically misdiagnosed for candidiasis, xanthomas, squamous papillomas, or glycogen acanthoses.1,2 Accurate diagnosis was more likely to be made by senior endoscopists who are motivated to seek a tissue biopsy as histopathological examination is key to accurate diagnosis.2 The EESG are a rare benign condition that does not necessitate specific treatment or endoscopic follow-up because it does not carry any malignant potential.1–5

DISCLOSURES

Author contributions: F. Bahdi wrote the manuscript. N. Zarrin-Khameh provided the histology images. SK Agarwal revised the manuscript for intellectual content and is the article guarantor. All authors have approved the final manuscript.

Financial disclosure: None to report.

Informed consent was obtained for this case report.

REFERENCES

1. Park A, Lee JH, Park A, et al. Prevalence rate and clinical characteristics of esophageal ectopic sebaceous glands in asymptomatic health screen examinees, Dis Esophagus. 2017;30(1):1–5.
2. Chiu KW, Wu CK, Lu LS, Eng HL, Chiou SS. Diagnostic pitfall of sebaceous gland metaplasia of the esophagus. World J Clin Cases. 2014;2(7):311–5.
3. Lee SH, Lee DJ, Kim KM, Kim KN, Kang JK. Ectopic sebaceous glands in the oesophagus: A case report and review of literature. Scott Med J. 2014;59(3):e1–5.
4. Montalvo N, Tapia V, Padilla H, Redrobán L. Heterotopic sebaceous glands in the esophagus, a very rare histopathological diagnosis: A case report and review of the literature. Clin Case Rep. 2016;5(2):89–92.
5. Nakanishi Y, Ochiai A, Shimoda T, et al. Heterotopic sebaceous glands in the esophagus: Histopathological and immunohistochemical study of a resected esophagus. Pathol Int. 1999;49:364–8.
© 2020 The Author(s). Published by Wolters Kluwer Health, Inc. on behalf of The American College of Gastroenterology.