CASE REPORT
Tubulo-villous adenoma of the female genital tract: A case report and review of literature BIRTHEBR0NDUM MORTENSEN AND
KARSTEN NIELSEN
From the Institute of Pathology, Aalborg Hospital, Denmark
Acta Obstet Gynecol Scand 1991; 70: 161-163
A case is presented of tubulo-villous adenoma in the vagina of a 43-year-old woman. The possible origin of this rare condition is discussed and the need for careful follow-up is emphasized. Key words: adenocarcinoma, genital tract, intestinal metaplasia. tubulo-villous adenoma Submitted November 5, 1990 Accepted February 11, 1991
Enteric tumors arising outside the gastrointestinal tract often lead to diagnostic problems. Tubulo-villous adenomas of the vagina are extremely rare, and to our knowledge only one case has been reported previously. We report the case of a 43-year-old woman with a tubulo-villous adenorna of the vagina and discuss the possible origin of enteric tumors in the female geni tal tract.
formed from the excision-area in the vagina. In January 1989 rebiopsy was performed and reresection again in February 1990. Proctoscopy and a barium enema revealed no further lesions. Cytologic examination of cervical smears was normal.
Case report Clinicalfindings A 43-year-old nulligravida presented in November 1987 with vaginal discharge and contact bleeding of 2 months' duration. Since 1977 she had been treated several times for condylomas in the anogenital region, once with excision and later with podophyllin. The microscopical finding demonstrated papillomata planocellularia hyperkeratotica. At gynecologic examination a polypoid tumor measuring 17x10x9 rnm was found at the posterior rim Of carunculus hymenalis and a few condylomas at the anus. A surgeon excised the tumor and in January 1988 the patient was referred to hospital for further examination. A further resection was perII
Fig. 1. Microscopic appearance of the tumor. The surface and the glandular formations are covered by stratified atypical cylindrical epithelium. The connective tissue is inflamed. Hematoxylin-eosin stain, original magnification x360. Arlo Obsrer Gynecol Scand 70 (1991)
162
B. B. Mortensen and K. Nielsen
Fig. 2. Microscopic appearance of the transition from the normal squamous epithelium (lef) to the abnormal cylindrical epithelium of the tumor (right). Hematoxylin-eosin stain. original magnification ~ 3 6 0 .
Fig. 3. Parallel section to Fig.1. The goblet cells stained positive for colon-specific mucins. PBIKOHPAS-stain. original magnification ~ 3 6 0 .
Dlscusslon Gross pathology
The tumor measured 1 7 10x9 ~ mm. The surface was brownish-greyish. The gross appearance mostly resembled a colonic polyp. Microscopic appearance
The tumor tissue in 1987,1988 and in 1989 consisted of a stalk of loose connective tissue with a polypous surface composed of glandular formations. These were covered by cylindrical epithelium with basally situated nuclei and a brush border. In some glandular formations, Paneth cells and goblet cells were present. Multifocally the epithelium was stratified, with loss of orientation and variation in the sue of nuclei and cells (Fig. 1). An abrupt transition from the normal squamous epithelium of the vagina to the cylindrical epithelium was found in the margin of the tumor (Fig. 2).Microscopy of the resected tissue in 1990 showed fibrosis. Mucin histochemical and immunohistochemical examination:
The mucin in the goblet cells stained positive with a colon-specific mucin histochemical technique (Fig. 3), which stains only 0-acylated sialic acids (perboric acid-potassium hydroxide-periodic acid-Schiff technique (PBNOWPAS)) (1). The epithelial cells contained cytokeratin and CEA in the epithelium of the colonic mucosa. The cells contained no vimentin.
Aria Obster Gynecol Scand 70 (1991)
Villous adenomas are well known neoplasms of the gastrointestinal tract; other locations are rare. A few cases of enteric-like villous adenoma of the urinary tract have been reported (2). Enteric tumors of the female genital tract are extremely rare. One case of tubulo-villous adenoma of the vagina (3), one case of tubulo-villous adenoma of the vulva (4), one of tubulo-villous adenoma in a rectovaginal tract ( 5 ) . one of enteric adenocarcinoma of the vagina (3) and 3 cases of enteric adenocarcinomas of the cervix (3,6,7) have been reported. Neither the etiology nor predisposing factors in cases of enteric tumor in the female genital tract are known, but the plausible mechanisms are: 1) “congenital” ectopic intestinal epithelium of the genital tract as a result of embryological remnants, 2) “acquired” intestinal metaplasia followed by dysplastic changes of the epithelium, 3) direct outgrowth from a gastrointestinal tumor, 4) distant metastatic lesions from a gastrointestinal tumor. Intestinal metaplasia has been described in the urinary tract (23) and in one case resulting in enteric adenocarcinoma. Intestinal metaplasia of the cervix is described in 3 cases (9) and in one case coincident with adenocarcinoma (6). It suggests an ability of intestinal metaplasia to develop in tissue of Mullerian origin. Since the vagina derives from the urogenital sinus, it is probable that enteric tumors in this location arise from cloaca1 remnants. Vaginal adenosis is retention of Miillerian-derived glandular epithelium in the vagina after vaginal embryogenesis is completed and vaginal adenocarcinoma develops from this structure. This Mullerian epithelia have no colon-specific mucins. In our patient, direct outgrowth from an intestinal tumor was deemed unlikely as proctoscopy and bari-
Tubulo-villous adenoma um enema of the colon proved normal. Furthermore, no malignant histologic feature of the vaginal tumor was identified and therefore a metastatic lesion from a gastrointestinal tumor could be excluded. According to histological, mucin-histochemical and immunohistochemical investigations, the vaginal tumor was composed of the same epithelium as found in adenomas of the colon. This is supported by a case of tubulo-villous adenoma of the uterine cervix associated with invasive adenocarcinoma (7). This indicates that long term follow-up of patients with enteric tumors outside the gastrointestinal tract is advisable.
References Culling CF, Reid PE. The histochemistry of colonic mucins. J Histochem Cytochem 1979;27: 1177-9. Jacob NH, Mau W. Metaplasia of the ureteral epithelium resulting in intestinal mucosa and adenocarcinomatous transformation: report of two cases. J Urol
1951;65:2&24. Fox, Wells M,Harris M, McWilliarn W,Anderson GS.
163
Enteric tumours of the lower genital tract: a report of three cases. Histopathology 1988;12: 167-76. 4. Tiltman AJ. Knutzen VK. Primary adenocarcinoma of the vulva originating in misplaced cloaca1 tissue. Obstet Gynecol 1978;51 (suppl 1): 30-33. 5. Ciano PS, Antonioli DA, Critchlow J. Burke L, Coldman H. Villous adenoma presenting as a vaginal polyp in a rectovaginal tract. Hum Pathol 1987; 18: 863-6. 6. Azzopardi JG. Hou LT. Intestine metaplasia with argentaffin cells in cervical adenocarcinoma. J Pathol Bacteriol 1965;90:686-90. 7. Michael H, Sutton G, Hull MT, Roth LM. Villous adenoma of the uterine cervix associated with invasive adenocarcinoma: a histologic, ultrastructural, and immunohistochemical study. Int J Gynecol Pathol 1986; 5:
163-7. 8. Gordon A. Intestinal metaplasia of the urinary tract epithelium. J Pathol Bacteriol 1963;85: 4414. 9. Tromwell JE. Intestinal metaplasia with argentaffin cells in the uterine cervix. Histopathology 1985;9: 551-9. Address for correspondence: Birthe Brcbndum Mortensen, MD Skodsborgvej 52 DK-9270Klarup Denmark
Acta Obstet Gynecol Scand 70 (1991)
Tubulo-villous adenoma of the female genital tract: A case report and review of literature BIRTHEBR0NDUM MORTENSEN AND
KARSTEN NIELSEN
From the Institute of Pathology, Aalborg Hospital, Denmark
Acta Obstet Gynecol Scand 1991; 70: 161-163
A case is presented of tubulo-villous adenoma in the vagina of a 43-year-old woman. The possible origin of this rare condition is discussed and the need for careful follow-up is emphasized. Key words: adenocarcinoma, genital tract, intestinal metaplasia. tubulo-villous adenoma Submitted November 5, 1990 Accepted February 11, 1991
Enteric tumors arising outside the gastrointestinal tract often lead to diagnostic problems. Tubulo-villous adenomas of the vagina are extremely rare, and to our knowledge only one case has been reported previously. We report the case of a 43-year-old woman with a tubulo-villous adenorna of the vagina and discuss the possible origin of enteric tumors in the female geni tal tract.
formed from the excision-area in the vagina. In January 1989 rebiopsy was performed and reresection again in February 1990. Proctoscopy and a barium enema revealed no further lesions. Cytologic examination of cervical smears was normal.
Case report Clinicalfindings A 43-year-old nulligravida presented in November 1987 with vaginal discharge and contact bleeding of 2 months' duration. Since 1977 she had been treated several times for condylomas in the anogenital region, once with excision and later with podophyllin. The microscopical finding demonstrated papillomata planocellularia hyperkeratotica. At gynecologic examination a polypoid tumor measuring 17x10x9 rnm was found at the posterior rim Of carunculus hymenalis and a few condylomas at the anus. A surgeon excised the tumor and in January 1988 the patient was referred to hospital for further examination. A further resection was perII
Fig. 1. Microscopic appearance of the tumor. The surface and the glandular formations are covered by stratified atypical cylindrical epithelium. The connective tissue is inflamed. Hematoxylin-eosin stain, original magnification x360. Arlo Obsrer Gynecol Scand 70 (1991)
162
B. B. Mortensen and K. Nielsen
Fig. 2. Microscopic appearance of the transition from the normal squamous epithelium (lef) to the abnormal cylindrical epithelium of the tumor (right). Hematoxylin-eosin stain. original magnification ~ 3 6 0 .
Fig. 3. Parallel section to Fig.1. The goblet cells stained positive for colon-specific mucins. PBIKOHPAS-stain. original magnification ~ 3 6 0 .
Dlscusslon Gross pathology
The tumor measured 1 7 10x9 ~ mm. The surface was brownish-greyish. The gross appearance mostly resembled a colonic polyp. Microscopic appearance
The tumor tissue in 1987,1988 and in 1989 consisted of a stalk of loose connective tissue with a polypous surface composed of glandular formations. These were covered by cylindrical epithelium with basally situated nuclei and a brush border. In some glandular formations, Paneth cells and goblet cells were present. Multifocally the epithelium was stratified, with loss of orientation and variation in the sue of nuclei and cells (Fig. 1). An abrupt transition from the normal squamous epithelium of the vagina to the cylindrical epithelium was found in the margin of the tumor (Fig. 2).Microscopy of the resected tissue in 1990 showed fibrosis. Mucin histochemical and immunohistochemical examination:
The mucin in the goblet cells stained positive with a colon-specific mucin histochemical technique (Fig. 3), which stains only 0-acylated sialic acids (perboric acid-potassium hydroxide-periodic acid-Schiff technique (PBNOWPAS)) (1). The epithelial cells contained cytokeratin and CEA in the epithelium of the colonic mucosa. The cells contained no vimentin.
Aria Obster Gynecol Scand 70 (1991)
Villous adenomas are well known neoplasms of the gastrointestinal tract; other locations are rare. A few cases of enteric-like villous adenoma of the urinary tract have been reported (2). Enteric tumors of the female genital tract are extremely rare. One case of tubulo-villous adenoma of the vagina (3), one case of tubulo-villous adenoma of the vulva (4), one of tubulo-villous adenoma in a rectovaginal tract ( 5 ) . one of enteric adenocarcinoma of the vagina (3) and 3 cases of enteric adenocarcinomas of the cervix (3,6,7) have been reported. Neither the etiology nor predisposing factors in cases of enteric tumor in the female genital tract are known, but the plausible mechanisms are: 1) “congenital” ectopic intestinal epithelium of the genital tract as a result of embryological remnants, 2) “acquired” intestinal metaplasia followed by dysplastic changes of the epithelium, 3) direct outgrowth from a gastrointestinal tumor, 4) distant metastatic lesions from a gastrointestinal tumor. Intestinal metaplasia has been described in the urinary tract (23) and in one case resulting in enteric adenocarcinoma. Intestinal metaplasia of the cervix is described in 3 cases (9) and in one case coincident with adenocarcinoma (6). It suggests an ability of intestinal metaplasia to develop in tissue of Mullerian origin. Since the vagina derives from the urogenital sinus, it is probable that enteric tumors in this location arise from cloaca1 remnants. Vaginal adenosis is retention of Miillerian-derived glandular epithelium in the vagina after vaginal embryogenesis is completed and vaginal adenocarcinoma develops from this structure. This Mullerian epithelia have no colon-specific mucins. In our patient, direct outgrowth from an intestinal tumor was deemed unlikely as proctoscopy and bari-
Tubulo-villous adenoma um enema of the colon proved normal. Furthermore, no malignant histologic feature of the vaginal tumor was identified and therefore a metastatic lesion from a gastrointestinal tumor could be excluded. According to histological, mucin-histochemical and immunohistochemical investigations, the vaginal tumor was composed of the same epithelium as found in adenomas of the colon. This is supported by a case of tubulo-villous adenoma of the uterine cervix associated with invasive adenocarcinoma (7). This indicates that long term follow-up of patients with enteric tumors outside the gastrointestinal tract is advisable.
References Culling CF, Reid PE. The histochemistry of colonic mucins. J Histochem Cytochem 1979;27: 1177-9. Jacob NH, Mau W. Metaplasia of the ureteral epithelium resulting in intestinal mucosa and adenocarcinomatous transformation: report of two cases. J Urol
1951;65:2&24. Fox, Wells M,Harris M, McWilliarn W,Anderson GS.
163
Enteric tumours of the lower genital tract: a report of three cases. Histopathology 1988;12: 167-76. 4. Tiltman AJ. Knutzen VK. Primary adenocarcinoma of the vulva originating in misplaced cloaca1 tissue. Obstet Gynecol 1978;51 (suppl 1): 30-33. 5. Ciano PS, Antonioli DA, Critchlow J. Burke L, Coldman H. Villous adenoma presenting as a vaginal polyp in a rectovaginal tract. Hum Pathol 1987; 18: 863-6. 6. Azzopardi JG. Hou LT. Intestine metaplasia with argentaffin cells in cervical adenocarcinoma. J Pathol Bacteriol 1965;90:686-90. 7. Michael H, Sutton G, Hull MT, Roth LM. Villous adenoma of the uterine cervix associated with invasive adenocarcinoma: a histologic, ultrastructural, and immunohistochemical study. Int J Gynecol Pathol 1986; 5:
163-7. 8. Gordon A. Intestinal metaplasia of the urinary tract epithelium. J Pathol Bacteriol 1963;85: 4414. 9. Tromwell JE. Intestinal metaplasia with argentaffin cells in the uterine cervix. Histopathology 1985;9: 551-9. Address for correspondence: Birthe Brcbndum Mortensen, MD Skodsborgvej 52 DK-9270Klarup Denmark
Acta Obstet Gynecol Scand 70 (1991)
