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 Table of Contents  
ORIGINAL ARTICLE
Year : 2021  |  Volume : 1  |  Issue : 1  |  Page : 43-47

Posthysterectomy malignant pelvic masses – A diagnostic dilemma


Department of Gynecologic Oncology, Dr. B. Borooah Cancer Institute, Guwahati, Assam, India

Date of Submission17-May-2021
Date of Decision20-May-2021
Date of Acceptance20-May-2021
Date of Web Publication23-Jul-2021

Correspondence Address:
Dr. Roma Jethani
A5, Doctors' Quarters, BBCI Campus, Gopinath Nagar, A K Azaad Road, Guwahati - 781 016, Assam
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/aort.aort_11_21

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  Abstract 


BACKGROUND: Pelvic masses following hysterectomy are common findings in Gynecologic Oncology centers. Incomplete preoperative evaluation, inadequate surgery (subtotal hysterectomy/only hysterectomy without salpingo-oophorectomy in postmenopausal women), and delay in histopathological diagnosis are few reasons for missing out on malignant etiologies. The diagnostic dilemma exists because of unknown primary malignancy. This study aims to highlight the various pathologies that present as pelvic masses after hysterectomy and their management based on histopathological examination and immunohistochemical (IHC) markers.
MATERIALS AND METHODS: This was a retrospective analysis of all women who presented in 1-year duration (January 2019–December 2019) to the gynecologic oncology department with pelvic mass and prior history of hysterectomy done outside. The data of these women were critically analyzed in regard to their demographic profile, preoperative and postoperative characteristics, histopathological and IHC markers of pelvic mass, management of the disease, and their outcome.
RESULTS: The total number of patients eligible for the study was 17. The median time to presentation after hysterectomy was 5 years (range: 1–20 years). The origin was female genital tract in 16 women and urothelial in 1 patient. Management options of these patients were concurrent chemoradiation/palliative radiotherapy/palliative chemotherapy/palliative care. The overall survival of these patients was dismal.
CONCLUSION: Before proceeding with hysterectomy, thorough evaluation should be done for all patients with even minimal symptoms. Early identification of malignant disease and management by a multidisciplinary team can greatly affect the overall prognosis of the patient.

Keywords: Hysterectomy, malignancy, pelvic mass


How to cite this article:
Jethani R, Patra S, Barmon D, Chhangte Z, Baruah U, Begum D, Kataki AC. Posthysterectomy malignant pelvic masses – A diagnostic dilemma. Ann Oncol Res Ther 2021;1:43-7

How to cite this URL:
Jethani R, Patra S, Barmon D, Chhangte Z, Baruah U, Begum D, Kataki AC. Posthysterectomy malignant pelvic masses – A diagnostic dilemma. Ann Oncol Res Ther [serial online] 2021 [cited 2022 May 23];1:43-7. Available from: http://www.aort.com/text.asp?2021/1/1/43/322153




  Introduction Top


Hysterectomy is one of the most common gynecological operations performed in females for benign indications such as abnormal uterine bleeding mostly due to uterine fibroids and adenomyosis. A proper evaluation of abnormal uterine bleeding to rule out gynecological malignancy is one of the major parts of workup. However, an incomplete preoperative evaluation, inadequate surgery (subtotal hysterectomy/only hysterectomy without salpingo-oophorectomy in postmenopausal women), and no histopathological diagnosis are few reasons for missing out on malignant etiologies. This leads to local relapse at the vault in the form of malignant pelvic masses which not only poses a dilemma in diagnosing the primary site of tumor but the management thereafter.

The present study is a review of malignant pelvic masses in women who had undergone hysterectomy outside for benign indications.


  Materials and Methods Top


The medical records of women who presented with isolated malignant pelvic mass posthysterectomy (done in other hospitals) in the outpatient department of Gynecologic oncology between January 2019 and December 2019 were collected. The data of these women were retrospectively analyzed with regard to their demographic profile, clinical profile, factors related to hysterectomy, biopsy and immunohistochemical (IHC) markers of pelvic mass, management of the disease, and their outcome. All women were evaluated completely as per the standard protocol of the hospital, namely complete history and physical examination. A proper evaluation of pelvic mass was done based on clinical, per abdominal, per speculum, per vaginum, and per rectal examination. After proper clinical examination, routine investigations including blood tests and tumor markers were done. Contrast-enhanced computed tomography/magnetic resonance imaging of the whole abdomen and pelvis and chest X-ray were done. All the patients underwent biopsy of pelvic mass. Based on the histology, IHC tests were done for determining the primary origin of the tumor. The IHC markers used were Paired box gene 8 (PAX8), Wilms tumor 1 (WT) 1, p53, estrogen receptor (ER), progesterone receptor (PR), p16, vimentin, Human Melanoma Black 45 (HMB45), smooth muscle actin (SMA), cytokeratin (CK), CK7, CK20, GATA-binding protein 3 (GATA3), p63, p40, carcinoembryonic antigen (CEA), alpha-fetoprotein (AFP), and placental alkaline phosphatase (PLAP).


  Results Top


Demographic profile

A total number of patients presenting with pelvic mass posthysterectomy in 1 year were 17. The mean age of patients at presentation was 47 years (range: 38–71 years), and the main symptom was bleeding per vaginum. The mean age of women at the time of hysterectomy was 45 years (range: 40–60 years). The mean duration from hysterectomy to development of pelvic mass was 5 years (range: 1–20 years). Majority of the patients presented 3–5 years after hysterectomy (47%) [Table 1].
Table 1: Interval from hysterectomy and treatment received

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Clinical profile

The most common indication of hysterectomy was abnormal uterine bleeding (35%) followed by chronic pelvic pain (23%). Seventeen percent of patients had nonspecific symptoms, 11% of patients had persistent vaginal discharge, 5% had presented with abdominal lump, and another 5% had uterovaginal prolapse. [Table 2] shows the indications of hysterectomy in the study population. The Eastern Cooperative Oncology Group performance status in majority was 1. None of the patients had preoperative cervical cancer screening and endometrial sampling. Operative procedure performed in most women was total abdominal hysterectomy (70%), followed by subtotal hysterectomy (11%), and total abdominal hysterectomy and bilateral salpingo-oophorectomy (11%). There was one woman (5%) who had undergone vaginal hysterectomy. Other operative details such as intraoperative findings and histopathological reports of the specimen were not available.
Table 2: Indications of hysterectomy

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[Table 3] shows the biopsy results of the pelvic masses. The histology of pelvic mass was squamous cell carcinoma (47%), adenocarcinoma (35%), poorly differentiated carcinoma (11%), and uterine sarcoma (5%). With the help of IHC markers (PAX-8, WT-1, vimentin, ER, PR, p53, and p16), adenocarcinoma was differentiated into endometrioid adenocarcinoma (5%), endocervical adenocarcinoma (11%), and high-grade adenocarcinoma (17%). The primary tissue of origin for patients with poorly differentiated adenocarcinoma was determined using CK7, GATA3, p63, p40, p53, PR, ER, CK20, CEA, AFP, PLAP, vimentin, SMA, and HMB45. Leiomyosarcoma in 5% of patients and urothelial carcinoma with squamous differentiation in the rest 5% were diagnosed.
Table 3: Histology of malignant pelvic masses

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[Table 1] summarizes the interval between hysterectomy and presentation based on origin of cancer. The mean time to presentation of women with vaginal vault/cervical cancer, ovarian cancer, uterine (endometrioid) cancer, uterine leiomyosarcoma, rhabdomyosarcoma and urothelial cancer was 6.1 years, 5.6years, 3 years,5 years,4 years, and 1year respectively. The various treatments given to these women based on primary are shown in [Table 4].
Table 4: Outcome at the end of 1 year

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Survival outcome

At the end of 1 year, 16/17 patients are alive and on regular follow-up. One out of 17 had died (5%) [Table 4].


  Discussion Top


The present analysis of malignant pelvic masses developed posthysterectomy is of concern as regards the dilemma of diagnosing the primary tumor whether gynecological or nongynecological and the management thereafter. Hysterectomy is the second most common surgery among women done for benign gynecological, mostly fibroids.[1] The burden of cancers in women as depicted in the GLOBOCAN 2020 reported 604,127 new cases of cervix uteri cancer, 417,367 new cases of corpus uteri cancer, 313,959 new cases of ovarian cancer, 45,240 new cases of vulvar cancer, and 17,908 new cases of vaginal cancer, worldwide.[2] Thus, before going for hysterectomy, it is very crucial to rule out malignancy. Incomplete preoperative evaluation, hysterectomy without salpingo-oophorectomy (after 45 years) or subtotal hysterectomy, and no histopathological examination of the postoperative specimen are the reasons for missing out on malignant pathology. In the present study, in 1 year, 17 women presented with malignant masses in the pelvis, vault, or vagina years after undergoing hysterectomy for benign indications. Loft et al. in their study followed women 12.5 years posthysterectomy done for benign conditions and reported the lifetime risk of developing ovarian cancer 2.1% after hysterectomy compared to 2.7% in the general population.[3] Holub et al. reported a 50.7% incidence of pelvic mass after hysterectomy and the patients requiring reoperation accounted for 2.7%–5.5%.[4] Similar to the present study, Chao et al. evaluated 247 patients posthysterectomy with pelvic masses, of which one-third (34.01%) were malignant masses and the rest 65.99% were benign masses.[5] The present study confronted malignant masses which could be due to high referral load from outside to our cancer center.

On further classifying the primary origin of the pelvic mass based on biopsy and IHC tests, it was observed that majority 10 (58%) were cervical/vaginal vault carcinoma. The presentation was from 1 year to as long as 20 years (mean = 6.1 years). Two out of these 10 women had subtotal hysterectomy in the past. Although in majority, there was vault recurrence posttotal hysterectomy (mean time to presentation = 4.6 years). The histology was of squamous (n = 8) and adenocarcinoma (n = 2). In the study by Park et al., the authors evaluated women who underwent inadvertent simple hysterectomy with undiagnosed occult cervical cancer, 34.6% (9 out 26) of patients who did not get any definitive radiation developed recurrence. The median time to recurrence was 4 years ranging from 4 months to 11 years.[6] This is in accordance with the observation of the present study. A Papanicolaou (PAP) smear screening before hysterectomy is a simple measure to rule out cervical cancer which might be the reason of missing out cancer in cervix, thus women presented with vault recurrence. The occurrence of cervical cancer in cervix postsubtotal hysterectomy has been reported by many, thus the need to screen with cytology.

The second common primary in the present study was uterus, endometrioid adenocarcinoma (n = 1, time to presentation = 3 years), leiomyosarcoma (n = 1, time to presentation = 5 years), and rhabdomyosarcoma (n = 1, time to presentation = 5 years). Various case reports in the literature have documented pelvic masses with endometrioid adenocarcinoma histology presenting after 2–17 years of hysterectomy.[7],[8],[9] All these women had received hormonal replacement therapy postoperatively. Our patient, in contrast, had no history of hormonal replacement therapy. She was managed with external beam radiotherapy to the pelvis.

One of the rarest tumors, rhabdomyosarcoma of the uterus in adults, was encountered in the present analysis. This woman presented after 4 years of hysterectomy with bleeding per vaginum and lower abdominal pain. [Figure 1] shows the contrast-enhanced computed tomography of the pelvis. Initial histology revealed poorly differentiated carcinoma; IHC tests (CK, HMB45-negative, vimentin, and SMA-positive) confirmed the diagnosis of rhabdomyosarcoma. [Figure 2] and [Figure 3] show the histology and IHC of the pelvic mass. The patient had undergone radiation and chemotherapy as the recurrent mass was inoperable. The patient is on regular follow-up with no evidence of residual disease at the end of the study. Very few cases of adult recurrent rhabdomyosarcoma of the uterus have been reported so far. Similar to our report, Alkhaledi et al. reported a case of metastatic rhabdomyosarcoma of the uterus in a 60-year-old woman presenting 2 years after hysterectomy with muscle weakness due to metastasis. The patient was managed with combination chemotherapy and had residual disease at the end of the study.[10]
Figure 1: Contrast-enhanced computed tomography (pelvis) – 10.2 cm × 5.9 cm lobulated pelvic mass of soft-tissue attenuation extending to the vaginal vault. It extends to the right pelvic sidewalls and closely abuts the right internal iliac vessels. Right ureter is compressed with Grade II Hydronephrosis

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Figure 2: Histopathological slide (H and E stain) of pelvic mass. The biopsy of the pelvic mass revealed poorly differentiated malignant tumor

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Figure 3: Immunohistochemical test – vimentin positive

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Leiomyosarcoma is the most common (25%–36%) uterine sarcomas. A rare case of posthysterectomy leiomyosarcoma was reported by Bhuyar et al. The patient had presented after 4 years of hysterectomy. Our patient presented after 5 years and was treated with palliative chemotherapy in view of poor performance status.[11]

The incidence of ovarian endometrioid carcinoma and ovarian clear cell carcinoma was higher in those with a history of hysterectomy due to endometriosis/adenomyosis.[5] However, in our study, exact indication of hysterectomy could not be ascertained due to lack of histopathology report of hysterectomy specimen. Three out of 17 patients were diagnosed with high-grade serous adenocarcinoma after IHC tests (PAX-8, WT1, and p53 positive). Yen-Chiu et al. reported a case of ovarian malignancy (serous tumor with low-grade malignant potential) 13 years after a total hysterectomy and bilateral salpingo-oophorectomy for endometriosis.[12] Our patients presented mean 5.6 years after hysterectomy (range: 5–7 years).

Urothelial cancer rarely presents as chronic pelvic pain and pelvic mass, as seen in our case. Farley et al. reported the case of a 76-year-old postmenopausal woman with low-grade ureteral carcinoma presenting with complex pelvic mass.[13] Urothelial cancer presenting as pelvic mass recurrence is not being reported so far.

The present study depicts that undiagnosed genital tract malignancy, especially uterus and cervix, if missed/untreated, can relapse years after the procedure and poses a diagnostic and therapeutic challenge to the gyne-oncologists. Moreover, the outcome also gets affected decreasing the overall survival of the women due to delay in commencement of treatment. One out of 17 patients (5%) died at the end of our study which highlights the grim outcome due to delayed treatment.


  Conclusion Top


A complete evaluation and thorough workup are crucial steps in the management of women presenting with gynecological symptoms keeping in mind the possibility of malignancy so as not to deal with such long-standing recurrent pelvic disease thereafter. A checklist comprising PAP smear and endometrial biopsy for all patients before hysterectomy should be ensured. Removal of bilateral tubes and ovaries must be done during hysterectomy in all postmenopausal women. The importance of histopathological examination of the hysterectomy specimen should be emphasized for detection of incidental malignancy. Strict follow-up of patients with abnormal histopathological reports and early referral to gyne-oncologist for an appropriate and timely intervention can greatly affect the overall prognosis of the patient.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Novetsky AP, Boyd LR, Curtin JP. Trends in bilateral oophorectomy at the time of hysterectomy for benign disease. Obstet Gynecol 2011;118:1280-6.  Back to cited text no. 1
    
2.
Bray F, Ferlay J, Soerjomataram I, Siegel RL, Torre LA, Jemal A. Global cancer statistics 2018: GLOBOCAN estimates of incidence and mortality worldwide for 36 cancers in 185 countries. CA Cancer J Clin 2018;68:394-424.  Back to cited text no. 2
    
3.
Loft A, Lidegaard O, Tabor A. Incidence of ovarian cancer after hysterectomy: A nationwide controlled follow up. Br J Obstet Gynaecol 1997;104:1296-301.  Back to cited text no. 3
    
4.
Holub Z, Jandourek M, Jabor A, Kliment L, Wágnerová M. Does hysterectomy without salpingo-oophorectomy influence the reoperation rate for adnexal pathology? A retrospective study. Clin Exp Obstet Gynecol 2000;27:109-12.  Back to cited text no. 4
    
5.
Chao X, Liu Y, Ji M, Wang S, Shi H, Fan Q, et al. Malignant risk of pelvic mass after hysterectomy for adenomyosis or endometriosis. Medicine (Baltimore) 2020;99:e19712.  Back to cited text no. 5
    
6.
Park JY, Kim DY, Kim JH, Kim YM, Kim YT, Nam JH. Management of occult invasive cervical cancer found after simple hysterectomy. Ann Oncol 2010;21:994-1000.  Back to cited text no. 6
    
7.
Abu MA, Sinha P, Totoe L, McCune G. Endometrial cancer thirteen years after total abdominal hysterectomy and bilateral salpingo-oophorectomy and hormone replacement therapy: A case report. Eur J Gynaecol Oncol 1997;18:482-3.  Back to cited text no. 7
    
8.
Debus G, Schuhmacher I. Endometrial adenocarcinoma arising during estrogenic treatment 17 years after total abdominal hysterectomy and bilateral salpingo-oophorectomy: A case report. Acta Obstet Gynecol Scand 2001;80:589-90.  Back to cited text no. 8
    
9.
Nomura S, Suganuma T, Suzuki T, Ito T, Kajiyama H, Okada M, et al. Endometrioid adenocarcinoma arising from endometriosis during 2 years of estrogen replacement therapy after total hysterectomy and bilateral salpingo-oophorectomy. Acta Obstet Gynecol Scand 2006;85:1019-21.  Back to cited text no. 9
    
10.
Alkhaledi A, Hanafi I, Alsabe H, Chatty EM. Rhabdomyosarcoma of the uterus with multiple metastases in a post-menopausal woman. Oxf Med Case Reports 2019;2019:omz017.  Back to cited text no. 10
    
11.
Bhuyar S, Sontakke B, Dharmale N. A rare case of leiomyosarcoma arising from vault post-hysterectomy. Int J Reprod Contracept Obstet Gynecol 2018;7:349-51.  Back to cited text no. 11
    
12.
Chiu YH, Chen CH, Chiu LH, Yen YK, Chang CW, Tzeng CR, et al. Occurrence of ovarian cancer 13 years after a total hysterectomy and bilateral salpingo-oophorectomy for endometriosis: A case report and literature review. Gynecol Minim Invasive Ther 2015;4:23-5.  Back to cited text no. 12
    
13.
Farley JH, Douglas TH, Mcleod DG, Harrison CR. Ureteral carcinoma presenting as a complex pelvic mass in a post menopausal patient. Gynecol Oncol 1998;70:134-6.  Back to cited text no. 13
    


    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2], [Table 3], [Table 4]



 

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