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Intestinal heterotopia in urethral caruncle

  • Author(s): Charfi, Slim
  • Makni, Saloua
  • Amouri, Meriem
  • Ellouze, Sameh
  • Turki, Hamida
  • Boudawara, Tahya Sellami
  • et al.
Main Content

Letter: Intestinal heterotopia in urethral caruncle
Slim Charfi1, Saloua Makni1, Meriem Amouri2, Sameh Ellouze1, Hamida Turki2, Tahya Sellami Boudawara1
Dermatology Online Journal 18 (7): 13

1. Department of Pathology, Habib Bourguiba University Hospital Center, Sfax, Tunisia
2. Department of Dermatology, Hedi Chaker University Hospital Center, Sfax, Tunisia


Abstract

We report a case of urethral caruncle with intestinal heterotopia in a 26-year-old woman. This entity is rarely reported.



Case

We report an unusual vulvar lesion arising in a 26-year-old woman with a history of pemphigus vulgaris receiving long-term corticosteroid treatment. She presented for consultation for a vulvar lesion. Clinical examination found a red and polypoid 10 x 5 mm nodule of the periurethral region.


Figure 1
Figure 1. A red and polypoid nodule of the periurethral region

A complete resection was performed. At surgery, the nodule showed microcystic and hemorrhagic features. Histologically, the lesion was covered by an intestinal colonic epithelium in continuity with both a squamous and transitional epithelium. The latter was invaginated in the lamina propria demonstrating cystic nests with bloody content. At the surface, there was an exulcerated area associated with abundant inflammatory infiltrates including lymphocytes, plasma cells, and neutrophils Histochemical stain with blue alcian was strongly CD positive in “goblet” cells of the intestinal metaplasia. Immunohistochemical study showed a postivity of glandular cells with keratin 20, although, they were keratin 7 negative.


Figure 2Figure 3
Figure 2. A polypoid lesion recovered by a squamous epithelium in continuity with an intestinal colonic-type epithelium (H&E x40)

Figure 3. Glandular cells demonstrate positive immunostaining with keratin 20 whereas they were negative for keratin 7 (x40)

Discussion

Urethral caruncle is a common growth of the urethral meatus, encountered in middle-aged women [1]. Although its etiology and pathogenesis are not well understood, many theories have been proposed [1, 2]. Urethral irritation and inflammation of distal urethral mucosa have been suggested as potential etiologies. Mucosal prolapse of the urethra may also contribute to the development of caruncle [2]. Intestinal heterotopia in urethral caruncle has rarely been described [3, 4, 5]. To the best of our knowledge, only seven cases were reported with several denominations “intestinal heterotopia in urethral caruncle,” “periurethral colonic type polyp,” and “urethral caruncle with coexistence of intestinal heterotopia” [3, 4, 5, 6]. Intestinal heterotopia in these lesions is considered as a metaplastic change similar to lesions seen in transitional bladder mucosa (cystitis glandularis with intestinal metaplasia) [4]. The occurrence of two cases in the pediatric population supports an additional congenital etiology [6]. The natural history of intestinal metaplasia in transitional mucosa and its exact clinical significance remains indeterminate. Although rare case reports have described adenocarcinoma adjacent to cystitis glandularis with intestinal metaplasia, this lesion is relatively common and is not currently regarded as a precursor to urothelial carcinoma [7]. Finally, we recommend that all caruncles in young women should be submitted to histological study.

References

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2. Dmochowski RR, Ganabathi K, Zimmern PE, Leach GE. Benign female periurethral masses. J Urol. 1994 Dec;152(6 Pt 1):1943-51. [PubMed]

3. Järvi OH, Marin S. Intestinal mucosal heterotopia of an urethral caruncle. Acta Pathol Microbiol Immunol Scand A. 1982 May;90(3):213-9. [PubMed]

4. Järvi OH, Marin S, de Boer WG. Further studies of intestinal heterotopia in urethral caruncle. Acta Pathol Microbiol Immunol Scand A. 1984 Nov;92(6):469-74. [PubMed]

5. Willett GD, Lack EE. Periurethral colonic-type polyp simulating urethral caruncle. A case report. J Reprod Med. 1990 Nov;35(11):1017-8. [PubMed]

6. Urakami S, Igawa M, Shiina H, Ishibe T. Urethral caruncle with coexistence of intestinal heterotopia. A case report. Int Urol Nephrol. 1997;29(3):341-4. [PubMed]

7. Sung MT, Lopez-Beltran A, Eble JN, MacLennan GT, Tan PH, Montironi R, Jones TD, Ulbright TM, Blair JE, Cheng L. et al. Divergent pathway of intestinal metaplasia and cystitis glandularis of the urinary bladder. Mod Pathol. 2006 Nov;19(11):1395-401. [PubMed]

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