| Abstract|| |
Epithelial inclusion cyst of the cecum (EICC) or epidermoid cyst of the cecum is extremely rare. Only seven cases of EICC have been reported in the English literature until now. A case of EICC is being reported in a 25-year-old female who presented with a pelvic mass associated with lower abdominal pain radiating to back. Abdominal contrast-enhanced computed tomography scan showed a well-defined mass juxtaposed to cecum. During operation, a subserosal cystic mass was found adherent to the anterior wall of the cecum and confirmed to be an epithelial inclusion cyst histopathologically. It is being reported due to its extreme rarity.
Keywords: Cecum, epithelial inclusion cyst, epidermoid cyst
|How to cite this article:|
Mittal S, Makkar M, Suri V. Epithelial inclusion cyst of the cecum: A rare entity. Indian J Pathol Microbiol 2015;58:506-8
| Introduction|| |
Epithelial inclusion cyst of the cecum (EICC) is an extremely rare entity with only seven cases reported in the literature till now.,,,,,, However, similar cysts of the internal organs have been reported involving the testis, epididymis, spleen, accessory spleen, appendix and liver., It is lined by well-defined stratified squamous epithelium and enlarges by cellular proliferation and by desquamation of keratinized debris into the cavity of the cyst. Histologically, it is differentiated from the dermoid cyst by the absence of dermal structures and from the benign cystic teratoma or true dermoid cyst, which is a neoplasm, by the presence of structural elements derived from the three embryonic germ layers. Its histogenesis is controversial.,,,,,, An instance of EICC presenting clinically as a pelvic mass with pain lower abdomen radiating to back is being reported due to its extreme rarity.
| Case Report|| |
A 25-year-old female presented to surgery outpatient department with complaints of pelvic mass associated with lower abdominal pain radiating to back for 2-3 years. There was no other systemic complaint or history of prior surgical operation. Physical examination of the abdomen demonstrated right lower quadrant mass. There was no tenderness. Laboratory findings were not contributory. Abdominal contrast-enhanced computed tomography scan revealed a heterogenous nonenhancing well-defined mass in pelvis superior to urinary bladder measuring 8.3 cm × 7.5 cm × 6.8 cm, juxtaposed to the cecum with an undistended appendix separate from both ovaries [Figure 1]. The uterus and both ovaries were normal. All other abdominal organs were within normal limits.
|Figure 1: Abdominal contrast enhanced computed tomography scan showing a nonenhancing heterogenous mass (arrow) juxtaposed to cecum|
Click here to view
A clinical possibility of the intestinal tumor was given.
Ultrasound-guided fine needle aspiration cytology was performed, which yielded greyish yellow material. Smears were adequate in cellularity showing many anucleate and nucleate squames [Figure 2].
|Figure 2: Gross photograph of resected right hemicolectomy specimen showing cut open cyst having pultaceous material|
Click here to view
The patient underwent right hemicolectomy. Firm growth at ileocaecal junction involving terminal ileum and cecum was seen intraoperatively with enlarged mesenteric lymph nodes. Specimen was sent for histopathological examination.
Right hemicolectomy specimen measuring 21 cm in length was received for histopathological examination. About 6.0 cm from ileal resection end, a greyish brown cecal globular mass is seen measuring 6.0 cm in diameter, adherent to the anterior wall of the cecum [Figure 3]. Cut section showed a unilocular cyst with a wall thickness of 0.1-0.2 cm with smooth inner lining filled with pultaceous material and appeared to be subserosal in location, having no communication with the intact intestinal mucosa [Figure 4]. Eight lymph nodes were resected.
|Figure 3: Gross photograph of resected right hemicolectomy specimen showing a globular mass adherent to anterior wall of cecum|
Click here to view
|Figure 4: Sections from the cyst wall showing stratified squamous epithelial lining with underlying fibrous tissue (H and E, ×400)|
Click here to view
On microscopic examination, sections from the cyst wall showed mature keratinizing stratified squamous cell epithelial lining resting on a layer of fibrous tissue. There was mild chronic inflammatory cell infiltrate in the wall of the cyst. Contents of the cyst showed anucleate squames and keratin. Sections from the gut showed normal histology. Eight lymph nodes resected showed non-specific reactive hyperplasia.
The patient had an uneventful postoperative recovery.
| Discussion|| |
The histogenesis of an epidermoid cyst is unknown. It is generally considered to be sequestration or implantation cyst and not as neoplastic growth, separating it from the benign cystic teratomas predominantly observed in the ovary. It could be acquired following implantation of epidermal fragments after trauma (e.g., Postsurgical, spinal puncture); or can arise from congenital heterotopic cutaneous tissue either at the time of closure of the neural groove (pericranial, intradiploic, intracranial and intraspinal) or of coalescence of other epithelial fusion lines (head and neck and anorectal areas). Literature review and PubMed search yielded only seven cases of EICC to date [Table 1].,,,,,,
In three cases of EICC, there was a history of previous operations abutting the cecum; therefore, these are probably acquired. Cutaneous tissue elements may be inadvertently implanted during the surgical procedure, as Pear and Wolff have suggested. The other reported cases, including the present case, have no preceding iatrogenic cause and, therefore, probably represent congenital sequestration cyst, which is unusual, considering the location of the embryonic cecum relative to the closing midline neural groove and other epithelial fusion lines. It may have taken origin where the cecum re-enters the abdominal cavity as the last part of the gut in the process of intrauterine rotation. Furthermore, the fact that most occur in a subserosal location in the cecum also supports this concept.
The theories of metaplasia of the serosa caused by chronic inflammatory stimuli and heterotopic inclusion within the organ have not been enough to explain the occurrences of these cysts in the cecum yet., In the current case, there was only mild chronic inflammatory cell infiltrate, and the absence of chronic inflammatory stimuli makes this assumption impossible.
Cecal duplication could be another possibility but was highly unlikely because of the clear external location of the cyst in relation to the cecal muscular wall and its gross appearance.
Intestinal dermoid cysts can also occur in the ileocecal region.,,,
Some possible complications of EICC that can occur include rupture, hemorrhage, necrosis, torsion, volvulus, intussusception, obstruction, peritonitis, and malignant change.
| Conclusion|| |
The possibility of EICC although very rare should be kept in mind as differential diagnosis while dealing with patients presenting with an abdominal mass.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Pan A, Rogers AG, Klass AA. Epidermoid cyst of the cecum. Can Med Assoc J 1961;84:1075.
Candreviotis N. Epidermoid cyst of cecum. J Am Med Assoc 1965; 192:425-6.
Pear BL, Wolff JN. Epidermoid cyst of the cecum. JAMA 1969;207: 1516-7.
Andiran F, Dayi S, Caydere M, Dilmen G, Dilmen U. Epidermoid cyst of the cecum. J Pediatr Surg 1999;34:1567-9.
Mady HH, Melhem MF. Epidermoid cyst of the cecum of an elderly man with no previous history of surgery: A case report and review of literature. Int J Colorectal Dis 2002;17:280-3.
Reyes CV, Paner GP, Ahn M. Epithelial inclusion cyst of the cecum. Pract Gastroenterol 2003;27:52-8.
Uzunlar A, Bukte Y. Epidermoid cyst of the cecum in an elderly man. Ann Saudi Med 2006;26:477-9.
Piserchia NE, Davey RB. Epidermoid cyst of the appendix. J Pediatr Surg 1980;15:674-5.
Pear BL. Epidermoid and dermoid sequestration cysts. Am J Roentgenol Radium Ther Nucl Med 1970;110:148-55.
Williams PL, Warwick R. Gray's Anatomy. Edinburgh: Churchill Livingstone; 1980. p. 201.
King ES. Metaplasia in the tunica vaginalis testis. Aust N
Z J Surg 1954;24:131-6.
Mossey JF, Rivers L, Patterson P. Dermoid cyst of the cecum. Can Med Assoc J 1977;117:1372.
Cotton MH, Blake JR. Dermoid cyst: A rare tumour of the appendix. Gut 1986;27:334-6.
Hirota S, Ito A. Dermoid cyst of the ileum. Am J Gastroenterol 1993;88:975.
Wilkinson N, Cairns A, Benbow EW, Donnai P, Buckley CH. Dermoid cyst of the caecum. Histopathology 1996;29:186-8.
Dr. Manisha Makkar
231, Sector 4, Panchkula, Haryana
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4]