Int J Gynaecol Obstet 17: 226-228, 1979
Adenocystic Carcinoma of the Uterine Cervix: A Clinicopathologic Study Saroj Gupta, 1 1. M. Gupta 1 and B. Sanyal 2 Departments of Pathology and Radiation Medicine, Institute of Medical Sciences, Bañaras Hindu University, Varanasi, India
ABSTRACT Gupta S, Gupta IM, Sanyal B (Depts of Pathology and Radiation Medicine, Institute of Medical Sciences, Bañaras Hindu University, Varanasi, India). Adenocystic carcinoma of the uterine cervix: a clinicopathologic study. Int J Gynaecol Obstet 17: 226-228, 1979 Eight cases of adenocystic carcinoma of the uterine cervix, a rare histologic variant, are presented, with a brief review of 41 cases previously reported in the literature. They represent only 0.27% of 3254 cervical carcinomas that we encountered between 1962 and 1977. In all eight cases, there was early paramesial involvement, and the majority behaved aggressively. All eight patients were multiparas, and six of them were postmenopausal. Interestingly, including this series, 15 of 49 reported cases of this entity have been from India.
INTRODUCTION Adenocystic carcinoma of the uterine cervix is a rare tumor of controversial histogenesis. Only eight (0.27%) of 3254 cervical cancers that we encountered during a 16-year period (1962-1977) were adenocystic carcinomas. Their clinicopathologic features are presented below.
On microscopic examination, all tumors showed solid basaloid areas of varying degrees associated with typical adenocystic patterns (Figs. 1-5). There was palisading of cells at the margins of solid islands of cells; elsewhere these cells appeared cylindrical or low cuboidal and were arranged around gland-like areas. There was pinkish granular material within the lumina of some of these spaces. A typical cylindromatous pattern of both epithelial cells with hyalinized corded stroma was seen in three cases. Areas of squamous islands within adenocystic areas were found in four cases. Additional histologic features included associated squamous cell carcinoma in situ (Case 5) and grade 2 dysplasia of the overlying squamous epithelium in two cases (Cases 3 a n d 7). Case 8 showed adenocarcinoma merging with adenocystic carcinoma. There was tumor embolization in all cases, while perineural spread was noticed in only three cases. Productive inflammatory response of stroma, comprised mainly of lymphocytes and plasma cells, was seen in two cases, while two other cases were comprised predominantly of polymorphonuclear leukocytes and another case showed a fair number of eosinophils admixed with lymphocytes and plasma cells.
DISCUSSION MATERIALS A N D M E T H O D S T h e results of eight biopsies from the suspected cases of carcinoma of the cervix are reported. Clinical data and follow-up information have been compiled from the files of the Radiotherapy Department.
RESULTS T h e clinical data on eight cases of adenocystic carcinoma of the cervix are summarized in Table I.
Int J Gynaecol Obstet 17
Paalman and Counseller (12) first described adenocystic carcinoma of the cervix in 1949. T o date, only 41 cases of adenocystic carcinoma of the cervix have been described in world literature in English (1-18); with the addition of our eight cases, the total number of reported cases of this rare entity is 49. In 1978 Lahiri et al (8) reported seven cases of adenocystic carcinoma of the cervix in India. T h u s , the present study is the second large series from India; and, although this may be a fortuitous phenomenon, it may have some etiologic bearing locally. Adenocystic carcinoma of the cervix is a distinct
Adenocystic carcinoma of the cervix
227
Table I. Clinical data on eight cases of adenocystic carcinoma of the uterine cervix.9 Case No.
Age (years)
Parity
1 2
60 40
11 6
3
30
3
4
40
5
5 6 7 8
47 52 50 55
9 7 6 4
Menopause (years)
Location
Duration of Symptoms (months)
Clinical Stage
Both lips Anterior lips
5 5
III II"
2"
IV
1
Both lips with extension Both lips
6
I
2 8 4 8
Both lips Both lips Anterior lips Both lips
6 8 2 7
III III III II
10
Follow-up None Free of disease for 3 years Died within a year Free of disease for 3 years Died within a year Well up to 1 year Well up to 1 year None
a
All patients represented with a variable degree of bleeding, pain and vaginal discharge. " During pregnancy.
Fig. 1. Characteristic adenocystic carcinomatous tumor with adenomatous and cylindroid patterns (HE, X160).
histologic entity. It usually occurs in postmenopausal women, and pain, discharge and bleeding are the common presenting symptoms. T h e ages of patients of the present series are younger than those of patients reported in Western literature, but our unpublished data show that, in general, the ages of our cervical carcinoma patients were nearly 20 years younger. In all our cases of adenocystic carcinoma, there was early parametrial involvement, and they behaved aggressively, with two cases succumbing within a year of their being reported. Vascular spread was common to all. Miles and Norris (10) came to a similar conclusion. Schàrer and Zollinger (14), Genton (5) and Ramzy, Yuzpe and Hendelman (13) stated that cylindromatous patterns and more solid tumors metastasize more frequently than predominantly cribriform patterns.
Fig. 2. Another case showing both solid and cribriform pattern (HE, X160).
Fig. 3. A more or less solid area of an adenocystic carcinoma with basaloid cells having a tendency for palisading at the margins (HE, X160).
InlJ Gynaecol Obstet 17
228
Gupta et al
(16). T h e typical cylindromatous character of the tumor found in three of our cases has been an infrequently reported finding in the cervix (18). T h e histogenesis of the tumor is controversial, but a "reserve-cell" origin in the cervix remains a possibility. REFERENCES
Fig. 5. Epithelial rounded tumor cells with compact nuclei forming gland-like spaces. Strands of hyalinized stroma are seen dissecting the tumor cells (HE, X640). Interesting findings in our series included the association of carcinoma in situ with adenocystic carcinoma (Case 5). T h e initial case reported by Paalman and Counseller (12) also had a squamous cell carcinoma in situ, a n d such an association is described in seven more cases in the literature. In addition, foci of squamous metaplasia and dysplastic changes of the squamous epithelium have also been recorded. We also noticed dysplasia of overlying squamous epithelium in two of our cases, and another four showed islands of squamous metaplasia within solid areas of tumors. Moss a n d Collins (11) a n d Miles and Norris (10) observed squamous cell carcinoma and adenocystic carcinoma merging with each other. O n e of our cases showed adenocarcinoma merging with typical adenocystic areas. This association is less frequent but has been shown in the past to occur by Miles a n d Norris (10) a n d Shingleton, Lawrence and Gore
IntJ Gynaecol Obstet 17
1. Baggish M S , Woodruff J D : Adenoid basal lesions of the cervix. Obstet Gynecol 37:807, 1971. 2. Benitez E, Rodriguez HA, Rodriguez-Cuevas H, Chavez GB: Adenoid cystic carcinoma of the uterine cervix: report of a case and review of 4 cases. Obstet Gynecol 33:151, 1969. 3. de la Maza L M , T h a y e r BA, Naeim F: Cylindroma of the uterine cervix with peritoneal metastases: report of a case and review of the literature. Am J Obstet Gynecol 112:\2\, 1972. 4. Gallager H S , Simpson C B , Ayala AG: Adenoid cystic carcinoma of the uterine cervix: report of four cases. Cancer 27.1398, 1971. 5. Genton C: Adenoid cystic carcinoma of the uterine cervix. Obstet Gynecol « : 9 0 5 , 1974. 6. Gordon H W , M c M a h a n NJ, Agliozzo C M , Rogers BA: Adenoid cystic (cylindromatous) carcinoma of the uterine cervix: report of two cases. Am J Clin Pathol 5(9:51, 1972. 7. Grafton W D , Willis G W , M a r t i n J R , Mathews W R : Adenoid cystic carcinoma of the cervix: a report of two cases. J La State Med Soc 120:216, 1968. 8. Lahiri B, Agarwal B M , Agarwal S, Lahiri V L : Adenoid cystic carcinoma of the cervix. Indian J Cancer 15:6, 1978. 9. McGee J A , Flowers C E , T a t u m BS: Adenoid cystic carcin o m a of the cervix: report of a case. Obstet Gynecol 26: 356, 1965. 10. Miles PA, Norris H J : Adenoid cystic carcinoma of the cervix: an analysis of 12 cases. Obstet Gynecol 35:103, 1971. 11. Moss LD, Collins D N : Squamous and adenoid cystic basal cell carcinoma of the cervix uteri. Am J Obstet Gynecol 88: 86, 1964. 12. Paalman R J , Counseller V S : Cylindroma of the cervix with procidentia. Am J Obstet Gynecol 5
Adenocystic Carcinoma of the Uterine Cervix: A Clinicopathologic Study Saroj Gupta, 1 1. M. Gupta 1 and B. Sanyal 2 Departments of Pathology and Radiation Medicine, Institute of Medical Sciences, Bañaras Hindu University, Varanasi, India
ABSTRACT Gupta S, Gupta IM, Sanyal B (Depts of Pathology and Radiation Medicine, Institute of Medical Sciences, Bañaras Hindu University, Varanasi, India). Adenocystic carcinoma of the uterine cervix: a clinicopathologic study. Int J Gynaecol Obstet 17: 226-228, 1979 Eight cases of adenocystic carcinoma of the uterine cervix, a rare histologic variant, are presented, with a brief review of 41 cases previously reported in the literature. They represent only 0.27% of 3254 cervical carcinomas that we encountered between 1962 and 1977. In all eight cases, there was early paramesial involvement, and the majority behaved aggressively. All eight patients were multiparas, and six of them were postmenopausal. Interestingly, including this series, 15 of 49 reported cases of this entity have been from India.
INTRODUCTION Adenocystic carcinoma of the uterine cervix is a rare tumor of controversial histogenesis. Only eight (0.27%) of 3254 cervical cancers that we encountered during a 16-year period (1962-1977) were adenocystic carcinomas. Their clinicopathologic features are presented below.
On microscopic examination, all tumors showed solid basaloid areas of varying degrees associated with typical adenocystic patterns (Figs. 1-5). There was palisading of cells at the margins of solid islands of cells; elsewhere these cells appeared cylindrical or low cuboidal and were arranged around gland-like areas. There was pinkish granular material within the lumina of some of these spaces. A typical cylindromatous pattern of both epithelial cells with hyalinized corded stroma was seen in three cases. Areas of squamous islands within adenocystic areas were found in four cases. Additional histologic features included associated squamous cell carcinoma in situ (Case 5) and grade 2 dysplasia of the overlying squamous epithelium in two cases (Cases 3 a n d 7). Case 8 showed adenocarcinoma merging with adenocystic carcinoma. There was tumor embolization in all cases, while perineural spread was noticed in only three cases. Productive inflammatory response of stroma, comprised mainly of lymphocytes and plasma cells, was seen in two cases, while two other cases were comprised predominantly of polymorphonuclear leukocytes and another case showed a fair number of eosinophils admixed with lymphocytes and plasma cells.
DISCUSSION MATERIALS A N D M E T H O D S T h e results of eight biopsies from the suspected cases of carcinoma of the cervix are reported. Clinical data and follow-up information have been compiled from the files of the Radiotherapy Department.
RESULTS T h e clinical data on eight cases of adenocystic carcinoma of the cervix are summarized in Table I.
Int J Gynaecol Obstet 17
Paalman and Counseller (12) first described adenocystic carcinoma of the cervix in 1949. T o date, only 41 cases of adenocystic carcinoma of the cervix have been described in world literature in English (1-18); with the addition of our eight cases, the total number of reported cases of this rare entity is 49. In 1978 Lahiri et al (8) reported seven cases of adenocystic carcinoma of the cervix in India. T h u s , the present study is the second large series from India; and, although this may be a fortuitous phenomenon, it may have some etiologic bearing locally. Adenocystic carcinoma of the cervix is a distinct
Adenocystic carcinoma of the cervix
227
Table I. Clinical data on eight cases of adenocystic carcinoma of the uterine cervix.9 Case No.
Age (years)
Parity
1 2
60 40
11 6
3
30
3
4
40
5
5 6 7 8
47 52 50 55
9 7 6 4
Menopause (years)
Location
Duration of Symptoms (months)
Clinical Stage
Both lips Anterior lips
5 5
III II"
2"
IV
1
Both lips with extension Both lips
6
I
2 8 4 8
Both lips Both lips Anterior lips Both lips
6 8 2 7
III III III II
10
Follow-up None Free of disease for 3 years Died within a year Free of disease for 3 years Died within a year Well up to 1 year Well up to 1 year None
a
All patients represented with a variable degree of bleeding, pain and vaginal discharge. " During pregnancy.
Fig. 1. Characteristic adenocystic carcinomatous tumor with adenomatous and cylindroid patterns (HE, X160).
histologic entity. It usually occurs in postmenopausal women, and pain, discharge and bleeding are the common presenting symptoms. T h e ages of patients of the present series are younger than those of patients reported in Western literature, but our unpublished data show that, in general, the ages of our cervical carcinoma patients were nearly 20 years younger. In all our cases of adenocystic carcinoma, there was early parametrial involvement, and they behaved aggressively, with two cases succumbing within a year of their being reported. Vascular spread was common to all. Miles and Norris (10) came to a similar conclusion. Schàrer and Zollinger (14), Genton (5) and Ramzy, Yuzpe and Hendelman (13) stated that cylindromatous patterns and more solid tumors metastasize more frequently than predominantly cribriform patterns.
Fig. 2. Another case showing both solid and cribriform pattern (HE, X160).
Fig. 3. A more or less solid area of an adenocystic carcinoma with basaloid cells having a tendency for palisading at the margins (HE, X160).
InlJ Gynaecol Obstet 17
228
Gupta et al
(16). T h e typical cylindromatous character of the tumor found in three of our cases has been an infrequently reported finding in the cervix (18). T h e histogenesis of the tumor is controversial, but a "reserve-cell" origin in the cervix remains a possibility. REFERENCES
Fig. 5. Epithelial rounded tumor cells with compact nuclei forming gland-like spaces. Strands of hyalinized stroma are seen dissecting the tumor cells (HE, X640). Interesting findings in our series included the association of carcinoma in situ with adenocystic carcinoma (Case 5). T h e initial case reported by Paalman and Counseller (12) also had a squamous cell carcinoma in situ, a n d such an association is described in seven more cases in the literature. In addition, foci of squamous metaplasia and dysplastic changes of the squamous epithelium have also been recorded. We also noticed dysplasia of overlying squamous epithelium in two of our cases, and another four showed islands of squamous metaplasia within solid areas of tumors. Moss a n d Collins (11) a n d Miles and Norris (10) observed squamous cell carcinoma and adenocystic carcinoma merging with each other. O n e of our cases showed adenocarcinoma merging with typical adenocystic areas. This association is less frequent but has been shown in the past to occur by Miles a n d Norris (10) a n d Shingleton, Lawrence and Gore
IntJ Gynaecol Obstet 17
1. Baggish M S , Woodruff J D : Adenoid basal lesions of the cervix. Obstet Gynecol 37:807, 1971. 2. Benitez E, Rodriguez HA, Rodriguez-Cuevas H, Chavez GB: Adenoid cystic carcinoma of the uterine cervix: report of a case and review of 4 cases. Obstet Gynecol 33:151, 1969. 3. de la Maza L M , T h a y e r BA, Naeim F: Cylindroma of the uterine cervix with peritoneal metastases: report of a case and review of the literature. Am J Obstet Gynecol 112:\2\, 1972. 4. Gallager H S , Simpson C B , Ayala AG: Adenoid cystic carcinoma of the uterine cervix: report of four cases. Cancer 27.1398, 1971. 5. Genton C: Adenoid cystic carcinoma of the uterine cervix. Obstet Gynecol « : 9 0 5 , 1974. 6. Gordon H W , M c M a h a n NJ, Agliozzo C M , Rogers BA: Adenoid cystic (cylindromatous) carcinoma of the uterine cervix: report of two cases. Am J Clin Pathol 5(9:51, 1972. 7. Grafton W D , Willis G W , M a r t i n J R , Mathews W R : Adenoid cystic carcinoma of the cervix: a report of two cases. J La State Med Soc 120:216, 1968. 8. Lahiri B, Agarwal B M , Agarwal S, Lahiri V L : Adenoid cystic carcinoma of the cervix. Indian J Cancer 15:6, 1978. 9. McGee J A , Flowers C E , T a t u m BS: Adenoid cystic carcin o m a of the cervix: report of a case. Obstet Gynecol 26: 356, 1965. 10. Miles PA, Norris H J : Adenoid cystic carcinoma of the cervix: an analysis of 12 cases. Obstet Gynecol 35:103, 1971. 11. Moss LD, Collins D N : Squamous and adenoid cystic basal cell carcinoma of the cervix uteri. Am J Obstet Gynecol 88: 86, 1964. 12. Paalman R J , Counseller V S : Cylindroma of the cervix with procidentia. Am J Obstet Gynecol 5
