International Journal of Surgery Case Reports 106 (2023) 108189

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International Journal of Surgery Case Reports

journal homepage: www.elsevier.com/locate/ijscr

Case report

An unusual presentation of ovarian carcinoma with supraclavicular lymph

node and colorectal metastases leading to spontaneous rectovaginal fistula
Ikram Kharmach a, b, *, Samia Malki b, c, Ouissam Al Jarroudi a, b, Tijani El Harroudi d, b,
Badr Serji d, b, Said Afqir a, b
a
Department of Medical Oncology, Mohammed VI University Hospital, Oujda, Morocco
b
Faculty of Medicine and Pharmacy, Mohammed Ist University, Oujda, Morocco
c
Department of Anatomic Pathology, Mohammed VI University Hospital, Oujda, Morocco
d
Department of Surgical Oncology, Mohammed VI University Hospital, Oujda, Morocco

A R T I C L E I N F O A B S T R A C T

Keywords: Introduction and importance: Secondary metastases to the rectum from primary ovarian cancer are a rare entity
Metastatic ovarian cancer and their diagnosis and management are challenging. In this report, we discuss the findings of the case of
Rectal metastasis metastatic ovarian cancer to supraclavicular lymph nodes and the rectum complicated with rectovaginal fistula.
Rectovaginal fistula
Case presentation: A 68-year-old woman was admitted for abdominal pain with rectal bleeding. Pelvic exami­
Supraclavicular lymph node
nation revealed a left latero-uterine mass. Abdominal-pelvic CT scan showed a tumor mass on the left ovary. A
cytoreductive surgery and resection of a non-imaged rectal nodule identified during surgery were performed. The
tumor specimens including the rectal metastasis were immunohistochemically confirming a metastatic ovarian
cancer using CK7, WT1 and CK20. The patient received chemotherapy and had complete remission. However,
she had a recto-vaginal fistula confirmed by imaging and had developed right supraclavicular lymphadenopathy
from ovarian cancer later.
Clinical discussion: The dissemination of ovarian cancer in the digestive tract can be frequently, through direct
invasion, abdominal implantation and lymphatic system. Unusually, ovarian cancer cells may spread to supra-
clavicular nodes, because of the connection of the two diaphragmatic stages allowing the lymph flows
through the lymphatic vessels. Moreover, rectovaginal fistula is an uncommon complication which can be seen
spontaneously or due to certain patient's features.
Conclusion: In advanced ovarian carcinoma, it is required to properly assess the digestive tract during surgery
because imaging can miss metastatic lesions such as our case. The use of immunohistochemistry is recommended
to differentiate between primary ovarian carcinoma and secondary metastasis.

1. Introduction
Ovarian cancer (OC) is one of the major causes of death among
women diagnosed with reproductive tract malignancies and it mostly
affects women after menopause and rarely before the age of 40 [1].
Patients with OC are often diagnosed at advanced stage particularly
distant sites which is associated with high mortality rates [2]. The
peritoneal cavity is the most frequent site for OC seeding. Although,
other unusual and rare sites may be involved such as the Colo rectum
and distant lymph nodes [2]. Several complications can be developed
related to either treatment or surgery for OC such as rectovaginal fistula
which can be seen spontaneously or associated with patient's features
[3].
In this case report based on SCARE 2020 guidelines for better
reporting [4], we discuss the findings of a patient who underwent sur­
gery for an epithelial ovarian cancer and developed rectal metastasis
and recto-vaginal fistula with particular attention to the impact of his­
topathological examination and periodic follow-up of patients with
unusual metastatic sites.
2. Presentation of case
A 68-year-old Moroccan Amazigh housewife living in a rural area
with normal body mass index presented hypogastric pain which was

* Corresponding author at: Mohammed VI University Hospital, BP 60050 Oujda, Morocco.

E-mail address: khikram2@gmail.com (I. Kharmach).

https://doi.org/10.1016/j.ijscr.2023.108189
Received 23 February 2023; Received in revised form 3 April 2023; Accepted 4 April 2023
Available online 13 April 2023
2210-2612/© 2023 The Authors. Published by Elsevier Ltd on behalf of IJS Publishing Group Ltd. This is an open access article under the CC BY license
(http://creativecommons.org/licenses/by/4.0/).
I. Kharmach et al.
more accentuated on the left, associated with rectal bleeding of 2 weeks
duration. The patient denied alcohol abuse or any history of smoking.
She had also no previous social, gynecologic or family history of cancer.
She took oral medication for diabetes (metformin at the dose of 1 g per
day). She had a slim figure and her abdominal examination shows no
abnormalities. On pelvic examination, there is a firm, irregular mass in
the left latero-uterine area. There were no signs of perianal sepsis, or any
palpable mass.
A thoraco-abdominopelvic computed tomography (CT) scan showed
a large heterogeneous polylobed mass measuring 80 mm suggesting a
tumor of the left ovary, with no ascites or deep adenopathy (Fig. 1A).
Cancer antigen 125 (CA-125) was prescribed but not done due to the
low-income status of our patient. The patient was healthy enough to
undergo anesthesia, no particular measures was taken before surgery.
An exploratory laparotomy was performed under general anesthesia in
the supine position by an experienced senior surgical oncologist sur­
geon. A left ovarian mass of malignant appearance, adherent to the
sigmoid colon at the top of the wall and a nodule of peritoneal carci­
nomatosis invading the upper rectum were noticed. The surgical treat­
ment included a total hysterectomy, a bilateral annexectomy,
omentectomy and appendectomy involving a resection of the rectal
nodule. Pelvic and lumbo-aortic lymph node dissection was not per­
formed as per the latest recommendations. After debulking surgery, an
abdominal-pelvic CT revealed an enlargement of the uterine cavity with
a posterior invasion to the rectum (Fig. 1B). The initial anatomopatho­
logical study showed a tumor lesion measuring 8 × 3 × 2 cm, pre­
dominantly of solid cystic consistency (90 %). The cystic component was
multilocular with the presence of endocytic vegetations. The capsule
was intact with no invasion or exocytic vegetations. The patient un­
derwent colonoscopy that revealed extended ulcerations of 1 to 9 cm
from the anal margin. Immunohistochemistry (IHC) was performed and
Fig. 1. (A) CT scan image of the abdomen showed a left ovary mass, (B) CT scan image showing an irregular circumferential thickening of the rectum, (C) CT scan
image of the thoracic-abdominal-pelvic level showing a right supra-clavicular adenopathy, and (D) CT scan image showing complete remission of the primary tumor.
2
International Journal of Surgery Case Reports 106 (2023) 108189
showed tumor cells expressing cytokeratin 7 (CK7) but negative for
cytokeratin 20 (CK20), synaptophysin, progesterone receptors and
cluster of differentiation 30 (CD30). The rectal resection was infiltrated
with carcinoma (Fig. 2A and B).
The IHC analysis confirmed the ovarian origin of this infiltration and
it was positive for CK7 and Wilms tumor 1 (WT1) and negative for CK20
(Fig. 2C and D). Based on these findings, high-grade serous adenocar­
cinoma of the ovary according to the world health organization classi­
fication (2020) was retained and classified as stage IVB according to the
International Federation of Gynecology and Obstetrics (FIGO) staging
system for ovarian cancer (8th edition,2017). Within a week, the patient
was discharged from the hospital and continued treatment with antibi­
otics and preventive anticoagulation at home. The patient was lost to
follow-up for 4 months and during her visit, a cervical thoraco-
abdominopelvic CT scan was performed and revealed a right cervical
tissue mass measuring 29 mm × 31 mm suggesting an adenopathy
(Fig. 1C) which was confirmed on physical examination. A biopsy was
performed and the histopathological examination of the supraclavicular
nodule confirmed the secondary lymph node location of a known high-
grade serous carcinoma in our patient. No homologous recombination
deficiency testing was performed due to its unavailability at the hospital.
The patient received six cycles of combination chemotherapy con­
sisting of paclitaxel at a dose of 175 mg/m2 and carboplatin AUC 6.
Bevacizumab was not available and not affordable to our patient. The
most common adverse event was neutropenia grade II with no fever
managed successfully with treatment abstinence.
Nine months after initial chemotherapy, the patient reported leakage
of stool from the vagina. A CT scan revealed a rectovaginal fistula
(Fig. 3) and almost complete remission of the primary tumor (Fig. 1D).
An ostomy was proposed as a therapeutic option but rejected by the
patient. She has been monitored regularly with clinical examination and
I. Kharmach et al.
Fig. 2. (A) microphotograph showing a colonic wall with carcinomatous proliferation in its submucosa arranged in clusters and micropapillary (HE, ×100), (B)
tumor cells are atypical with hyperchromatic nuclei and abundant eosinophilic cytoplasm (HE, ×200), (C) the tumor cells express the anti-CK7 antibody and (D)
tumor cells express anti-WT1 antibody.
CT scans every 3 months. No recurrent disease was detected at 15
months of follow up. Our patient is still alive at the time of writing this
case report. On the other hand, our patient was satisfied with our
management. However, she wished to have been treated with targeted
agents such as bevacizumab and poly (ADP-ribose) polymerases (PARP)
inhibitors which were not available in our under-resourced setting.
3. Discussion
OC accounts for approximately 5 % of all female cancers and it in­
cludes several histological types [1]. The majority of OCs are of
epithelial origin. 90 % of cases fall into this category including
mucinous, clear cell, endometrioid and serous carcinomas which
represent the most common type [5]. In fact, unlike ovarian metastases
of digestive origin such as the classic Krukenberg tumor characterized by
the presence of signet ring cells in the ovaries which are more common
[5]. According to Lindner et al. ovarian metastases account for about 7
% of all OCs and present a colorectal origin in about 45 % of cases [6]. To
determine the etiological origin of the metastasis, not only the
morphology of the tumor is required, but also the IHC study based on the
expression of cytokeratins the tumor cells is essential because of the
indistinguishable histological appearance of OCs from the primary
gastrointestinal tumor.
In order to determine the cut-point between high- and low-grade OC,
Manu et al. [7] showed in his study that tumor protein P53, P16 and
WT1 had significantly higher staining scores in the group of ovarian
serous carcinoma comparing to the benign group, furthermore, the as­
sociation of moderate to high P53 and WT1 scores provides a solid
3
International Journal of Surgery Case Reports 106 (2023) 108189
argument to confirm high grade serous ovarian tumors. According to
Chu et al. CK7+, CK20- represents 98 % of OCs versus 0 % of colorectal
cancers. Beyond that, two markers were included in the study of
Lagendijk et al. [8] who reached the same conclusions suggesting that
CK7+, CA125+ /CK20-, carcino-embryonic antigen (CEA)- are bio­
markers for diagnosing an ovarian origin, and CK7 - /CA-125 - /CK20 +
/CEA+ in case of a colon origin. However, CA125 remains normal in 15
% of OCs. In our case, the combination of the morphological study and
the results of the IHC analysis confirmed the primary ovarian origin.
The dissemination of OC in the digestive tract can take various routes
frequently by a peritoneal route, which is the most common form. OC
cells disseminate through the exfoliation of tumor cells in the cavity and
are transported then by the ascitic fluid to the organs of the peritoneal
cavity [9]. Subsequently, the invasion can occur directly by invading
adjacent structures such as the colon. Then lymphatic route can also be
involved, initially through the para-aortic nodes, followed by the broad
ligament nodes which then drain into the hypogastric nodes. Finally,
hematological dissemination, which is less frequent, usually occurs in
the presence of advanced peritoneal disease and leads to the invasion of
the pleura, the lung, the liver and rarely the kidneys [5].
The interval time between diagnosis of OC and identification of
metastases is usually delayed, it ranged from 1 to 139 months and the
survival time after diagnosis of dissemination ranged from 1 to 51
months [10]. Trastour et al. [5] described a case of rectal recurrence 20
years after the resection of a stage III papillary clear cell OC. IHC study
confirmed CK7 positivity and CK20 negativity, with a positive CA-125.
The patient received a total proctectomy with protective colostomy.
Two years later, abdominal MRI showed a hepatic metastasis. The
I. Kharmach et al. International Journal of Surgery Case Reports 106 (2023) 108189

to rectal metastasis, a right supra clavicular adenopathy resulting from a

histologically confirmed OC. Only few cases have been described in the
literature with this trait. It is observed that lymph node metastases are
present in 70 % of the patients, however supra clavicular metastases are
reported in only 4 % of them, this is partly explained by the connection
of the two supra and sub diaphragmatic stages and the flow of lymphatic
fluid through the lymph nodes until the supra-clavicular nodes [13].
In OC, the rate of complications by fistula is rare, estimated between
0.8 % and 8.2 % [14]. It is affected by many factors including the
development of ascites greater than 500 mL [15], hypoalbuminemia less
than 3 g/dL [14] or the use of targeted therapy [16]. It has been reported
in the literature that a recto-vaginal fistula can appear when introducing
an antiangiogenic treatment [17,18]. Bevacizumab has confirmed its
efficacy especially in terms of progression-free survival [19]. However,
the use of this molecule seems to enhance certain complications. Cher­
eau et al. [20] discussed the first case of recto-vaginal fistula in a 45-
year-old woman treated for OC and who complained of having stools
through the vagina after 2 cycles of bevacizumab. Therefore, they rec­
ommended some precautions; first, bevacizumab should be introduced
in the second cycle of chemotherapy and secondly, an ileostomy may be
suggested for selected individuals. Presumably, this is the first study
suggesting that recto-vaginal fistula may be developed spontaneously
regardless of any established risk factors.

4. Conclusion

When an OC is associated with colorectal cancer, pathologic diag­

nosis is key to identify which one is the primary tumor, using immu­
nohistochemical staining with cytokeratin 7 and 20. The IHC based
finding can provide an optimal therapeutic approach. It is essential that
clinicians bear in mind that a proper peri-operative assessment of the
gastrointestinal tract should be performed for ovarian tumors to exclude
synchronous lesions. The occurrence of a spontaneous rectovaginal fis­
tula is an uncommon complication that should be considered while
treating advanced ovarian neoplasia; this situation highlights the
importance of periodic follow-up of patients with unusual metastatic
sites.

Consent to participate

Written informed consent was obtained from the patient for publi­
cation of this case report and accompanying images. A copy of the
Fig. 3. (A and B). CT scan images after opacification by vaginal route showing written consent is available for review by the Editor-in-Chief of this
the passage of the contrast product at the vaginal level revealing a rec­ journal on request.
tovaginal fistula.
Provenance and peer review
histopathological exam pointed to a gynecological origin (CK7 +, CK 20
-, CA-125 +). Zighelboim et al. [11] observed the development of a Not commissioned, externally peer-reviewed.
sigmoid tumor six months after left adnexectomy of an ovarian carci­
noma with low malignant potential, and no further treatment was rec­ Ethical approval
ommended. The histology of the sigmoid tumor was suggestive of an
ovarian origin; however, it was a high-grade papillary carcinoma with Ethical approval has been exempted by our institution.
areas of endometrioid carcinoma, the tumor was immunohistochemi­
cally positive for CK7+, CEA+, CA-125+, and negative for transcription Funding
termination factor 1 (TTF1), and CK20-.
Other site of metastasis, besides colorectal, is the gastric parenchyma None.
which is an extremely rare location, few cases have been reported, Zhou
and Miao [12] discussed a case report of 61-year-old women with no Guarantor
clinical manifestation but a high CA-125 and a history of ovarian
adenocarcinoma treated, 12 years ago, by surgery + adjuvant chemo­ Kharmach Ikram.
therapy. The diagnosis of metastatic OC was confirmed after the per­
formance of gastrectomy. The immunohistochemical staining was Research registration number
positive for WT1, CA-125, CK7 and negative for caudal-related ho­
meobox transcription factor 2 (CDX2). In our case, the rectal metastasis Not applicable.
occurred earlier and discovered at the operation of the primary tumor.
The particular feature of our case is that the patient has, in addition

4
I. Kharmach et al.
CRediT authorship contribution statement
Kharmach Ikram: Writing, review and editing of the manuscript.
Malki Samia: interpretation of histological data.
Al Jarroudi Ouissam: supervised the writing of manuscript.
El Harroudi Tijani: contributor.
Serji Badr: supervision and surgeon of the patient.
Afqir Said: supervision and data validation.
Declaration of competing interest
The authors have no conflict of interest to declare.
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