|LETTER TO THE EDITOR
|Year : 2015 | Volume
| Issue : 3 | Page : 312-313
Late recurrence in a case of vaginal clear cell adenocarcinoma unrelated to transplacental diethylstilbestrol exposure
K Khan, S Chakraborty, A Bandyopadhyay
Department of Pathology, North Bengal Medical College, Sushrutanagar, Darjeeling, West Bengal, India
|Date of Web Publication||18-Feb-2016|
Department of Pathology, North Bengal Medical College, Sushrutanagar, Darjeeling, West Bengal
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Khan K, Chakraborty S, Bandyopadhyay A. Late recurrence in a case of vaginal clear cell adenocarcinoma unrelated to transplacental diethylstilbestrol exposure. Indian J Cancer 2015;52:312-3
|How to cite this URL:|
Khan K, Chakraborty S, Bandyopadhyay A. Late recurrence in a case of vaginal clear cell adenocarcinoma unrelated to transplacental diethylstilbestrol exposure. Indian J Cancer [serial online] 2015 [cited 2019 Sep 22];52:312-3. Available from: http://www.indianjcancer.com/text.asp?2015/52/3/312/176707
Only 5-10% of all vaginal cancers are clear cell adenocarcinoma. In 87% cases of vaginal clear cell adenocarcinoma (VCCA), history of Maternal diethylstilbestrol (DES) intake by their mothers is reported. Hence in only 13% of patients with diagnosed VCCA no history of maternal intake of DES is reported i.e. in 13% of VCCA cases no transplacental or in-utero exposure to DES is reported. The fact highlighted is that, VCCA without history of maternal DES intake i.e. transplacental exposure to DES is rare. Five-years survival of patients with stage II tumors is 85% and recurrences beyond three years are reported extremely infrequently in the literature.,, Although the late recurrences occurred most commonly as local pelvic and abdominal disease those in distant extra pelvic sites like the lung has also been reported., We encountered a case of local recurrence in stage II primary VCCA unrelated to transplacental diethylstilbestrol exposure six years after adequate primary therapy.
A 37-year-old multigravida (P3+2) presented with lower abdomen pain and abnormal vaginal bleeding during late August, 2009. She was a follow-up case of VCCA diagnosed in 2003. Clinical examination revealed a 2.0 × 1.5 × 1.0 cm hemorrhagic mass at the apex of the vagina. No inguinal lymph node was palpable. Ultrasonography was corroborative and showed no other abnormalities. Chest X-ray detected no abnormality and clinical investigation revealed iron deficiency anemia. Histopathological examination revealed features of clear cell adenocarcinoma with predominantly solid pattern of growth.
In 2003, the patient had presented with pain in the lower abdomen for three months, irregular menstruation and leucorrhoea. History of last child birth was 7 years back, had no significant family history and denied any history of DES exposure in utero. Bimanual and per speculum examination showed an irregular reddish mass on the upper part of left lateral vaginal wall with left paravaginal tissue involvement. The cervix was found to be free. Per abdominal and transvaginal sonography detected a left vaginal wall mass measuring 3.6 × 3.8 × 3.3 cm which extended into the left paravaginal tissue [Figure 1]. No lymph nodes were detected and liver, spleen or bilateral kidneys showed no abnormality. Chest X-ray and X-ray pelvis were also normal. Incisional biopsy revealed histopathological features of clear cell adenocarcinoma and cervical biopsy revealed only chronic non-specific cervicitis. Examination under anesthesia was performed and the case was finally diagnosed as primary VCCA, FIGO stage II. Radical hysterectomy, bilateral pelvic lymphadenectomy along with partial vaginectomy was performed. Radical dose (7000 cGy over five weeks) of External Beam Radiotherapy (EBRT) followed by Interstitial Brachytherapy (ITB) with temporary perineal implants was instituted during late 2003. Local control of disease was achieved and complete response noted at the end of therapy with only minor transient small bowel obstruction. The patient was routinely followed up quarterly for the first year and every six months thereafter till 2009 when she presented with recurrence. She underwent exploratory laparotomy, partial vaginectomy followed by repeat intracavitary vaginal and interstitial brachytherapy with a total tumor dose of 8000 cGy and the patient is doing well without any major complications eleven months after detection of recurrence.
The histopathological features of the specimens received in 2009 and 2003 were almost similar except that in the primary specimen (2003) the predominant growth pattern was tubulocystic in contrast to a predominantly solid growth pattern in the later specimen (2009) [Figure 1] and [Figure 2]. Clear cells with hyperchromatic, moderately pleomorphic nuclei having inconspicuous nucleoli and mitotic figures <10/10 high power fields were noted [Figure 2]a and [Figure 2]b. Positive PAS stain with diastase digestion and negative mucicarmine stain confirmed intracellular glycogen deposits and no mucin. Paravaginal extension was only noted and all margins of resection were tumor-free. None of the lymph nodes resected showed evidence of metastasis.
|Figure 2: (a) Predominantly solid pattern of growth in the tumor on recurrence (2009) (H and E, ×100). (b) Clear cells with hyperchromatic, moderately pleomorphic nuclei having inconspicuous nucleoli (H and E, ×400)|
Click here to view
Most recurrences of VCCA occur within three years of initial therapy. However, recurrences have been rarely reported to occur as late as 10 to 20 years after treatment.,, Specific suggested mechanisms of carcinogenesis focus on the retention of nests of abnormal cells of mullerian duct origin, which, after stimulation by endogenous hormones during puberty, are promoted to adenocarcinoma.
The present case is extremely rare as per published literature. Even after adequate primary therapy, local recurrence occurred more than three years following that which again has been rarely reported.
| » Conclusion|| |
It can be commented that more frequent and intensive follow-up should be undertaken even after 3 years following therapy in cases of primary VCCA.
| » Acknowledgement|| |
Department of Gynecology and Obstetrics and Department of Radiotherapy, North Bengal Medical College and Hospital.
| » References|| |
Guzin K, Eser SK, Yigit A, Zemheri E. Primary clear cell carcinoma of the vagina that is not related to in utero
diethylstilbestrol use. GynecolSurg 2006;3:281-4.
Herbst AL, Robboy SJ, Scully RE, Poskanzer DC. Clear cell adenocarcinoma of the vagina and cervix in girls; analysis of 170 registry cases. Am J ObstetGynecol 1974;119:713-24.
Perez CA, Arneson AN, Galakatos A, Samanth HK. Malignant tumors of the vagina. Cancer 1973;31:36-44.
Tavassoli FA, Devilee P, editors. World Health Organization Classification of Tumours. Pathology and Genetics of Tumours of the Breast and Female Genital Organs. Lyon: IARC Press; 2003. p. 293-9.
Fishman DA, Williams S, Small W Jr, Keh P, Gerbie MV, Schwartz PE, et al
. Late recurrences of vaginal clear cell adenocarcinoma. GynecolOncol 1996;62:128-32.
Jones WB, Tan LK, Lewis JL Jr. Late recurrence of clear cell adenocarcinoma of the vagina and cervix: A report of three cases. GynecolOncol 1993;51:266-71.
[Figure 1], [Figure 2]