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\begin{document}

\title{Enterocutaneous Fistula Secondary to Cecal Carcinoma Masqueraded as
Gastrointestinal Tuberculosis: A Rare Case Report}



\author[1]{Syed Halim}%
\author[1]{Mirha Ali}%
\author[1]{Rabia Nasir}%
\author[2]{Hira Moosa}%
\author[1]{Azmatullah  Yousufzai}%
\author[3]{Asma Farah}%
\affil[1]{Jinnah Sindh Medical University}%
\affil[2]{Jinnah Post Graduate Medical Centre}%
\affil[3]{East Africa University}%


\vspace{-1em}



  
  \date{June 18, 2024}


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\textbf{Case report}

Halim et al

\textbf{Enterocutaneous Fistula Secondary to Cecal Carcinoma Masqueraded
as Gastrointestinal Tuberculosis: A Rare Case Report}

Syed Ahtisham Halim\textsuperscript{1}

Mirha Ali\textsuperscript{1}

Rabia Nasir \textsuperscript{1}

Hira moosa\textsuperscript{2}

Azmatullah Khan Yousufzai\textsuperscript{1}

Asma Ahmed Farah\textsuperscript{3}

\textsuperscript{1} Department of Medicine, Jinnah Sindh Medical
University, Karachi, Pakistan

\textsuperscript{2} Department of General Surgery, Jinnah Postgraduate
Medical Centre, Karachi, Pakistan

\textsuperscript{3}Department of Medicine, East Africa University,
Boosaaso, Somalia

\textbf{Corresponding Author:}

Correspondence: Asma Ahmed Farah

Postal address: 66R3+CC4, East Africa University, Boosaaso, Somalia

Tel: +252 907 814 006

Email: asmaaaxmad16@gmail.com

ORCID: 0009-0003-8095-1729

\textbf{Email Address}

Syed Ahtisham Halim\textsuperscript{1} -

\emph{syedahtisham175@gmail.com}

Mirha Ali\textsuperscript{1} - \emph{mirha.ali780@gmail.com}

Rabia Nasir\textsuperscript{1} - rabbianasir2000@gmail.com

Hira Moosa\textsuperscript{2} - hiramoosa@gmail.com

Azmatullah Khan Yousufzai\textsuperscript{1} - \emph{azmat.ukyz@gmail}

Asma Ahmed Farah\textsuperscript{3}- \emph{asmaaaxmad16@gmail.com}

\textbf{Introduction}

Enterocutaneous Fistula is an abnormal connection between the intestinal
tract and the skin that arises iatrogenically, often due to surgical
complications such as intestinal anastomotic dehiscence and enterotomies
(1)Abdominal trauma accounts for the etiology in 25-50\% of ECF cases,
while other origins encompass cancer, radiation, inflammatory bowel
disease (IBD), ischemia, and infectious diseases. Therefore, the origins
of ECF can be postoperative, traumatic, or spontaneous.(2)

Colorectal carcinoma (CRC), in contrast, is the third most prevalent
cancer globally, with 1.8 million new diagnoses and stands as the second
leading cause of cancer-related deaths, contributing to approximately
881,000 fatalities in 2018(3). The cecum, the initial segment of the
colon, can harbor a specific form of cancer termed as carcinoma of the
cecum. Despite its often subtle clinical presentation, historical data
reveals that nearly 20\% of colorectal tumors originate in the cecum.
Notably, gastrointestinal tuberculosis is often referred to as the
''great mimic'' because it exhibits symptoms similar to those of cecal
malignancy, complicating its accurate diagnosis(4). Not only this, the
convergence of enterocutaneous fistula (ECF) with colorectal cancer
(CRC) too is a rare occurrence, typically indicative of an advanced
stage of the disease. This case report aims to elucidate the exceptional
conjunction of carcinoma of the cecum with enterocutaneous fistula, an
association rarely documented in the medical literature, thereby aiming
to underscore the possibilities of misdiagnosis, the importance of early
detection, intervention and management in advanced cases of colon cancer
that carry poor prognosis.

\textbf{Case History}

A 36-year-old woman with a history of abdominal tuberculosis had
received two months of treatment with anti-tuberculosis drug. She had
also undergone an incision and drainage procedure for a psoas abscess.
The patient now presented to the emergency department experiencing
leakage of fecal matter from her right flank region. She described that
for the previous two weeks, she had been experiencing a foul-smelling,
feculent discharge from her right lower abdomen. Fever, nausea, and
reduced appetite were present along with the other symptoms.In the
preceding six months, she had struggled with intermittent constipation
and had experienced occasional per rectal bleeding. Moreover, she
mentioned significant weight loss over the last three months.
Importantly, she confirmed that there was no family history of
tuberculosis, colon cancer, or any malignancies.

Upon physical examination, she appeared to be in a weakened state,
confined to her bed, and showed signs of malnourishment. Vital signs
taken upon admission indicated that she was running a fever and
exhibited tachycardia. Closer inspection revealed spontaneous feculent
drainage through an anterolateral opening on her anterior abdominal wall
shown in \emph{Figure 1} . This area showed signs of edema. In the right
iliac fossa, a firm to hard mass measuring approximately 10x10 cm was
identified. Aside from this, her abdomen felt soft without any
tenderness. A rectal examination revealed a normal tone, but the
examining finger was stained with feces, although no palpable mass was
identified internally.

\textbf{Methods}

For a clearer diagnosis\textbf{,} laboratory tests were conducted, and
the results indicated an elevated white blood cell count at 14 \selectlanguage{ngerman}× 10\^{}3
per L, with the normal range being 4.0--10.8 × 10\^{}3 per L. This
elevation was primarily due to neutrophilia. Furthermore, her hemoglobin
level was slightly low at 11 mg/dL, compared to the typical range of
12--16 g/dL. Her albumin level was also below the normal, registering at
3.1 g/dL, where the standard range is between 3.4 and 4.8 g/dL.

Imaging examinations provided further insights into her condition. A CT
scan displayed a substantial mass originating from the caecum and
extending to the transverse colon as shown in \emph{Figure 2} . This
mass measured 13x10x13 cm and had multiple air locules noted along the
posterior lateral wall of the ascending colon mass. This mass extended
towards the hepatic flexure and opened into the subcutaneous plane. A
colonoscopy further revealed a large ulcerated growth in the transverse
colon, causing a narrowing of the lumen. This made it difficult for the
scope to be advanced proximally. A biopsy of this growth confirmed the
diagnosis of adenocarcinoma of the colon

Expert opinion was taken and the patient was planned for an exploratory
laparotomy with extended right hemi colectomy and en bloc resection as
shown in \emph{Figure 3} . Biopsy showed poorly differentiated mucinous
adenocarcinoma, tumor extended beyond muscularis propia and perforated
serosa with positive lymph node findings. Post operatively patient was
sent to oncology for chemotherapy.

\textbf{Conclusion and Results}

This case highlights one of the rarest complications of cecal carcinoma
which is enterocutaneous fistula formation. Following the biopsy report,
the patient attended follow-up appointments, was advised to undergo
chemotherapy, but unfortunately, was lost to follow-up thereafter. These
findings, therefore, highlight the importance of clinical awareness,
early diagnosis, timely intervention, and a comprehensive approach to
patient management. Such measures ultimately lead to better prognosis
and higher survival rates for patients facing similar conditions.

\textbf{Discussion}

A fistula is an abnormal connection between two epithelialized surfaces.
An enterocutaneous fistula (ECF) specifically refers to an abnormal
communication between the intestinal tract and the skin. One study found
that 51\% of fistulas were low-output, while 49\% were high-output.
Approximately 95\% of ECFs resulted from postoperative complications,
with the ileum being the most common site of ECF formation(5).

Globally, colon cancer is the second leading cause of cancer-related
deaths. A recent report by the World Health Organization (WHO)
highlighted that in 2020, there were over 1.9 million new cases of
colorectal cancer and more than 930,000 deaths from the disease(6).
According to prior research, the cecum has been identified as the site
of colorectal malignancy in 20\% of cases, making it a lesser prevalent
tumor site(7). Up to one third of patients with CRC may present to the
emergency room with complications like perforation and obstruction,
while patients with CRC frequently present to the outpatient setting
with changes in bowel habit, rectal bleeding, or iron deficiency
anemia(2).

However, perforation of right sided colon cancer may cause a localized
collection inside the abdomen that spreads superficially into the skin
to form a fistulous tract, making it a rare occurrence. Furthermore, in
areas where tuberculosis (TB) is prevalent, Mycobacterium tuberculosis
is frequently the cause of primary psoas abscess(8).

The clinical presentations of gastrointestinal tuberculosis share
similarities with those of cecal malignancy and enterocutaneous fistula,
posing diagnostic challenges. Misdiagnosis can lead to delays in
appropriate treatment, potentially allowing the condition to progress to
an advanced stage, as demonstrated in this case.

Subsequently, the emergence of enterocutaneous fistula (ECF) as a
complex complication in cancer patients further complicates management,
especially if they have previously undergone treatments such as surgery,
radiation, and chemotherapy. Beyond addressing the intricacies of
misdiagnosis, our case also explores one of the rarest complications of
cecal carcinoma: the development of an enterocutaneous fistula. Existing
literature primarily concentrates on more common complications of
colorectal carcinoma, like perforations and bowel obstructions. The
onset of ECF in cecal cancer patients complicates their treatment, often
delaying or even precluding palliative care or additional adjuvant
therapy. This has significant consequences on patients' quality of life
and overall survival.

Initial management of ECF involves resuscitation, infection control,
skin protection, providing nutritional support to the patient, and
managing ECF effluent. Surgical repair is commonly required. The
preparatory phase is crucial for the surgical treatment of these
patients. In some scenarios, non-surgical therapy might be the primary
or sole treatment, based on the tumor type(5,7,9).The primary goal of
surgery is the curative resection of the tumor, which includes excising
the fistula tract and the tumor en bloc. In our case, this approach was
paired with postoperative chemotherapy (10).

The development of ECF in colorectal carcinoma can be traced back to
localized perforation, leading to the tumor invading neighboring regions
and the skin. Often, the presentation of such cases is delayed,
resulting in unfavorable prognosis and heightened morbidity and
mortality rate(5).In conclusion, there's an urgent need for more
extensive research and more strategic interventions to deepen our
understanding and enhance the management of these severe complications
linked to colorectal carcinoma.

\textbf{List of abbreviations:}

Enterocutaneous fistula: ECF

Tuberculosis: TB

Colorectal Carcinoma: CRC

Inflammatory Bowel Disease: IBD

World Health Organization: WHO

\textbf{Declarations}

\emph{Ethics approval and consent to participate}

The Institutional Review Board of Jinnah Postgraduate Medical Centre
with reference number: NO.F.2-95/2023-GENL/211/JPMC had granted approval
for this case report.

\emph{Consent for publication}

Written informed consent was obtained from the patient for publication
of this case report and accompanying images

\emph{Availability of data and materials}

The authors confirm that the data supporting the findings of this study
are available within the article.

\emph{Conflict of interests}

The authors declare no potential conflicts of interest concerning the
research, authorship, and/or publication of this article.

\emph{Funding}

The authors received no extramural funding for the study.

\emph{Authors' contributions}

The case discovery was done by HM and SAH. The literature and drafting
of the manuscript were conducted by HM, RN, SAH, MA, AKY, and AAF. The
editing and supervision were performed by HM and MA. All authors have
read and agreed to the final version of the manuscript.

\emph{Clinical trial registration}

Not applicable

\emph{Acknowledgements} None to declare

\textbf{References:}

1. Sci-Hub \textbar{} Enterocutaneous Fistula: Proven Strategies and
Updates. Clinics in Colon and Rectal Surgery, 29(02), 130--137
\textbar{} 10.1055/s-0036-1580732.

2. Basukala S, Khand Y, Pahari S, Mainali P, Gurung N, Gurung S.
Colorectal carcinoma presenting as spontaneous colocutaneous fistula - A
rare case report and review of literature. Int J Surg Case Rep. 2022
Jul;96.

3. Rawla P, Sunkara T, Barsouk A. Epidemiology of colorectal cancer:
incidence, mortality, survival, and risk factors. Przegla\selectlanguage{english}d
Gastroenterol. 2019;14(2):89.

4. Chong VH, Lim KS. Gastrointestinal Tuberculosis. Microbiol Spectr.
2016 Jun;4(6):638--46.

5. Kumar P, Maroju N, Kate V. Enterocutaneous fistulae: Etiology,
treatment, and outcome - A study from South India. Saudi J
Gastroenterol. 2011;

6. Colorectal cancer.

7. Hermann J, Karmelita-Katulska K, Paszkowski J, Drews M, Stajgis M.
Diagnosis of a cecal tumour with virtual colonoscopy. Polish J Radiol.
2011;

8. Vasigh M, Karoobi M, Montazeri M, Moradi G, Asefi H, Gilani A, et al.
Isolated psoas abscess caused by Mycobacterium tuberculosis: A rare case
report. Clin Case Reports. 2022 May;10(5).

9. 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;

10. Chamberlain RS, Kaufman HL, Danforth DN. Enterocutaneous fistula in
cancer patients: Etiology, management, outcome, and impact on further
treatment. Am Surg. 1998;\selectlanguage{english}
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