Endoscopic Submucosal Dissection of Seborrheic Keratosis-Like Lesion of the Esophagus: A New Entity?
Nese EKINCI1, Eylül GÜN1, Fatih ASLAN2
1Department of Pathology, Izmir Katip Celebi University Ataturk Training and Research Hospital, IZMIR, TURKEY
2Department of Gastroenterology, Koc University Hospital, ISTANBUL, TURKEY
Keywords: Endoscopic submucosal dissection, Esophagus, Seborrheic keratosis
Seborrheic keratosis, one of the most common lesions of the epidermis, is rarely seen on mucosal surfaces. We report a case of a distinctive
epithelial neoplasm of the esophagus showing close resemblance to seborrheic keratosis that was resected with endoscopic submucosal
dissection. A 65-year-old patient’s previous esophageal biopsy showed suspicious low grade dysplasia and the patient was referred for endoscopic
submucosal dissection of a flat lesion in the mid-esophagus. Macroscopic examination revealed a well circumscribed, pigmented and elevated
lesion with a diameter of 20 mm. Microscopically, the lesion was well circumscribed, with plaque-like elevation, and showed hyperkeratosis,
acanthosis, and papillomatosis. Broad coalescing solid sheets and interconnecting trabeculae of basaloid cells were the consistent feature
throughout the lesion. Squamous eddies and occasional central keratinization were present. Mitotic activity and koilocytes were not identified.
Immunohistochemically, the lesion showed diffuse nuclear positivity with p63 and negativity with p16. Ki-67 index was confined to the basal
cell layer. With the help of histopathologic and immunohistochemical findings, we diagnosed this morphologically benign case as “seborrheic
keratosis-like lesion of the esophagus”. It should be kept in mind that seborrheic keratosis-like lesions might be rarely seen on mucosal surfaces
such as the esophagus. Endoscopic submucosal dissection is a new, curative, and safe endoscopic resection technique in en-bloc resection
of superficial esophageal lesions. To our knowledge, this is the first case of the aforementioned lesion in the esophagus being resected with
endoscopic submucosal dissection.
Seborrheic keratosis is one of the most common lesions
in dermatopathology but it is rarely seen on mucosal
. Curative endoscopic resection methods such
as endoscopic mucosal resection (EMR) and endoscopic
submucosal dissection (ESD) are new resection techniques
initially developed in Japan and allow en bloc resection and
treatment of superficial gastrointestinal lesions. Although
application of ESD to the esophagus is limited in early
stage esophageal neoplasia because of its greater technical
difficulty, it avoids the high morbidity and mortality rates
of surgical treatment 2
and offers a highly effective, safe
and less expensive way for the detection and treatment
of esophageal neoplastic lesions. It is considered the
cornerstone of endoscopic treatment of Barrett’s esophagus
and early squamous cell carcinoma of the esophagus
. It is therefore increasingly and successfully in use all
around the world. We report a case of distinctive epithelial
neoplasm of the esophagus, which shows close resemblance
to seborrheic keratosis, one of the most common benign
epidermal tumors, resected with ESD.
A 65-year-old man with slight dyspnea was referred to
the gastroenterology clinic of a tertiary hospital in 2013.
Gastro-esophagoscopy was done and no abnormality was
found. The follow-up endoscopic biopsy in November 2016
showed low grade dysplasia and the patient was referred to
our hospital for further examination and treatment. After
pre-procedural assessment with narrow band imaging and
chromoendoscopy with Lugol’s solution, a flat lesion of the
esophagus with a diameter of 20 mm at approximately 28
cm from the incisor teeth was seen (Figure 1
). The resection
borders were marked with dual knife and after submucosal
injection of indigo-carmine and sodium hyaluronate
solution, and en bloc resection of the lesion 2-3 mm away
from the margins was successfully performed. Intra- and
post-procedural prophylactic coagulation with hemostatic
forceps followed. Complications such as delayed bleeding
or perforation did not occur after the ESD and the patient
was discharged 2 days after the treatment. The specimen
was pinned against a plate peripherally by stainless-steel
pins and entirely immersed in formaldehyde overnight to
preserve the tissue shape and configuration (Figure 2
Click Here to Zoom
|Figure 1: Endoscopic view of the flat lesion with a diameter of
20 mm at approximately 28 cm from the incisor teeth of the
Macroscopic examination revealed a well circumscribed,
slightly pigmented and elevated plague-like lesion with a
diameter of 20 mm on a velvety mucosal surface. Alcian
blue staining was used for macroscopic delineation of
mucosal margins and the specimen was then serially
sectioned perpendicularly at 2 mm intervals. All sections
were subjected to histopathologic review. The lesion
was well circumscribed, with plaque-like elevation on
low power magnification and the base of the lesion was
rough on an imaginary axis drawn between two mucosalsubmucosal
junctions at both ends of normal esophageal tissue. On 10x and 20x magnification, hematoxylin and
eosin-stained sections revealed hyperkeratosis, acanthosis,
and papillomatosis. Broad coalescing solid sheets and
interconnecting trabeculae of basaloid cells were the
consistent feature throughout the lesion (Figure 3).
Squamous eddies and occasional central keratinization were
present (Figure 4A). Mitotic activity and koilocytes were
not identified. Immunohistochemically, the lesion showed
negativity with p16, diffuse positivity with cytokeratin 5/6,
and diffuse nuclear positivity with p63 (Figure 4B-C). The
Ki-67 labeling index was confined only to the basal cell layer
of the lesion and normal esophageal squamous epithelium
(Figure 4D). No dysplasia was identified.
Click Here to Zoom
|Figure 3: Circumscribed plaque-like lesion with acanthosis and
the transition zone between the lesion and normal esophageal
mucosa (inset) (H&E; x10).
Click Here to Zoom
|Figure 4: A) Squamous eddies, central keratinization and basaloid cells (H&E; x40). B) Cytokeratin 5/6 positivity (IHC; x20). C) Diffuse
p63 positivity (IHC; x20). D) Ki-67 confined to basal layer (IHC; x20).
Seborrheic keratosis is one of the most common lesions seen
by dermatologists and it is considered as a benign epidermal
tumor but it may be a sign of concomitant skin cancer
and internal malignancies. They are sharply demarcated,
slightly elevated, hyperpigmented patch or plaque-like
lesions and are seen commonly in areas such as the trunk,
neck, face and upper extremities. They are considered as
hyperkeratotic lesions of the epidermis and reported not
to be seen on the mucosal surfaces 1
. However, there are
several case reports in the literature presenting seborrheic
keratosis on the conjunctiva 4
and the nasal vestibule
. All of the dermatological lesions with hyperkeratosis,
acanthosis and papillomatosis might be considered in the
clinical and pathological differential diagnosis. Our patient
did not show any similar lesions on the skin and there
was no internal malignancies detected with the imaging procedures performed before the endoscopic procedure.
The Leser-Trélat syndrome, which is characterized by
the eruptive appearance of multiple seborrheic keratoses
in association with an underlying malignant disease, was
therefore not considered during the differential diagnosis.
The term “seborrheic keratosis-like lesion” as a new entity
was previously used in a case series by Talia and McCluggage
that included a total of 7 cases of the cervix and vagina and
a relationship with the human papilloma virus (HPV)
was shown in two of these cases 6. However we did not
find any similar lesions of the esophagus reported in the
ESD is an effective method for neoplastic lesions of the
esophagus and it is a safe treatment modality in the management of early esophageal squamous cell neoplasms.
However, great skill in this technique is definitely required
2. ESD is suggested to be performed rather than EMR
while dissecting lesions that are larger than 15 mm because
it ensures en bloc resections and the recurrence rates are
lower 7. A study by Chen et al. on 296 patients with
early esophageal squamous cell neoplasms and high-grade
intraepithelial neoplasms showed no cancer-related deaths
and it was concluded that ESD is a well-accomplished and
secure procedure 8.
Rare lesions of the esophagus resected with ESD or EMR
reported in the literature include Barrett’s esophageal
cancer 3, granular cell tumor 9 and leiomyomas in the
category of stromal tumors 10.
Herein, we report a case of a superficial esophageal lesion
resected with ESD. We diagnosed this morphologically
benign case as a “seborrheic keratosis-like lesion of
the esophagus” with the help of histopathologic and
immunohistochemical findings. To our knowledge, this is
the first case of the aforementioned lesion in the esophagus.
In conclusion, it should be kept in mind that seborrheic
keratosis-like lesions might be rarely seen on mucosal
surfaces such as the esophagus and that ESD is a safe
procedure in en bloc resection of superficial esophageal
CONFLICT of INTEREST
The authors declare no conflicts of interest.
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