A 54-year-old woman was admitted because regular gastroendoscopy incidentally revealed
an 8-mm subepithelial lesion 30 cm from the incisors ([Fig. 1]). Magnifying narrow-band imaging (NBI) showed regular intrapapillary capillary loops,
several of which showed slight dilatation (type A) ([Fig. 2]). Lugol’s iodine staining and ultrasound gastroscopy yielded negative findings.
Endoscopic biopsy revealed several scattered heterocyst clusters. Positron emission
tomography-computed tomography did not indicate metastasis.
Fig. 1 Gastroscopy revealed an 8-mm subepithelial lesion (circled) 30 cm from the incisors.
Fig. 2 Magnifying narrow-band imaging showed regular intrapapillary capillary loops (circled),
several of which showed slight dilatation.
Endoscopic submucosal dissection (ESD) was performed for further diagnosis and treatment
([Video 1]). Microscopically, the lesion was covered with normal epithelium ([Fig. 3 a]). Several poorly differentiated squamous carcinoma cells with dense lymphoplasmacytic
infiltration were distributed in the mucosal lamina propria and muscularis mucosa
(M3) ([Fig. 3 b]), consistent with lymphoepithelioma-like carcinoma (LELC). In situ hybridization
for Epstein-Barr virus showed negative findings. In addition, a dermoid cyst was identified
in the muscularis mucosa ([Fig. 4]). At 3 months post-ESD, the ESD wound had healed well without distant metastasis.
Video 1 Gastroscopy showing a subepithelial lesion. Submucosal dissection was performed.
Histological results revealed a lymphoepithelioma-like carcinoma and a dermoid cyst.
Fig. 3 Histopathological section from endoscopic submucosal dissection sample showed lymphoepithelioma-like
carcinoma (LELC). a The LELC was covered with normal epithelium. b The LELC was composed of poorly differentiated squamous carcinoma cells with dense
lymphoplasmacytic infiltration (hematoxylin and eosin stain).
Fig. 4 Histopathological section revealed a dermoid cyst lined with squamous epithelium
and containing hair follicles (hematoxylin and eosin stain).
Esophageal LELCs are rare and characterized by poorly differentiated carcinoma with
prominent lymphocytic infiltration covered with normal epithelium [1]. Epstein-Barr virus positivity is common but not necessary for diagnosis. Esophagectomy
and adjuvant chemoradiotherapy are traditional treatments [2]. Complete removal of early-stage LELC by ESD without distant metastases or recurrence
has only been reported once [1]. Dermoid cysts, benign cutaneous tumors of ectoderm origin, have not been found
in the esophagus [3]. Squamous cell carcinoma arising from a cranial dermoid cyst has only been reported
by Tsugu et al. [4], indicating malignant potential of dermoid cysts, especially of those co-existing
with squamous cell carcinoma. To our knowledge, this is the first report of co-existing
primary LELC and dermoid cyst in the esophagus, indicating that LELC and dermoid cysts
have appearances similar to benign subepithelial lesion on gastroscopy, even with
the help of Lugol’s iodine staining, magnifying NBI, and ultrasound gastroscopy. Endoscopists
are encouraged to perform regular biopsies for differential diagnosis.
Endoscopy_UCTN_Code_CCL_1AB_2AC_3AB
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