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\begin{document}

\title{Inflammatory Myofibroblastic Tumor Presenting as Ileocolic
Intussusception: A Case Report}



\author[1]{Nischal  Khanal}%
\author[2]{Rupak Subedi}%
\author[2]{NIrajan  Shrestha}%
\author[2]{Shristi Shrestha}%
\affil[1]{Tribhuvan University Institute of Medicine}%
\affil[2]{Madan Bhandari Academy of Health Sciences}%


\vspace{-1em}



  
  \date{May 29, 2024}


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\textbf{Title:}

\textbf{Inflammatory Myofibroblastic Tumor Presenting as Ileocolic
Intussusception: A Case Report}

\begin{enumerate}
\tightlist
\item
  Dr. Nischal Khanal, MBBS, MS General Surgery, Madan Bhandari Academy
  of Health Sciences (corresponding author).
\item
  Dr. Rupak Subedi, MBBS, Madan Bhandari Academy of Health Sciences.
\item
  Dr. Nirajan Shrestha, MBBS, MS General Surgery, Madan Bhandari Academy
  of Health Sciences.
\item
  Dr. Shristi Shrestha, MBBS, Madan Bhandari Academy of Health Sciences.
\end{enumerate}

\textbf{Consent} : Written informed consent was obtained from the
patient to publish this report.

\textbf{Conflict of Interest Statement} : There are no conflict of
interest regarding this manuscript.

\textbf{Abstract}

We present a case of a 36-year-old male who presented with abdominal
pain and vomiting. Inflammatory markers were elevated in routine
investigations, while other laboratory parameters were within normal
limits. Ultrasound imaging revealed a target lesion, which required
further evaluation with a CT scan and confirmed ileocolic
intussusception. An exploratory laparotomy showed a solid lesion
measuring 5 by 6 cm in the cecum, along with evidence of ileocolic
intussusception. A standard right hemicolectomy was performed, and the
postoperative course was uneventful. Histopathological examination
showed an inflammatory myofibroblastic tumor with nodular hyperplasia.
The report highlighted the importance of assessing rare neoplastic
causes in patients with intussusception.

\textbf{Keywords:} Inflammatory Myofibroblastic tumor, intussusception,
hemicolectomy, spindle cell proliferation, abdominal pain, vomiting

\textbf{Introduction} : Inflammatory Myofibroblastic tumors (IMTs) are
rare neoplasms characterized by proliferative Myofibroblastic spindle
cells accompanied by a prominent inflammatory infiltrate. {[}1{]} IMTs
predominantly affect children and young adults but can occur at any age.
{[}2{]} The most common site of occurrence is the lung, followed by the
extrapulmonary sites like liver, pancreas, intestine, and bones {[}3{]}.
Intestinal involvement is relatively rare, and ileocolic intussusception
caused by IMT is an even rarer presentation {[}4{]}. We report a case of
a 36-year-old male who presented with abdominal pain, vomiting, and
subsequent diagnosis of IMT leading to ileocolic intussusception.

\textbf{Case presentation:} A 36-year-old male presented to the
emergency department with a three-day history of diffuse abdominal pain,
followed by vomiting. The pain started from the right lower quadrant and
later generalized. On physical examination, mild tenderness was present
on deep palpation in the right lower quadrant of the abdomen. The
patient had no significant medical history and denied any recent weight
loss or changes in bowel habits.

Laboratory investigations revealed raised inflammatory marker. ~A
complete blood count revealed elevated WBCs (17,100/\selectlanguage{greek}µ\selectlanguage{english}L) with
neutrophilia (80\%). Blood urea nitrogen and creatinine level were
normal and other hematological and biochemical parameters were within
normal limits. An ultrasound examination of the abdomen showed a target
lesion in the right lower quadrant, raising the suspicion of
intussusception. A subsequent CT scan confirmed segmental,
circumferential thickening of the terminal ileocolic junction showing a
target stratification pattern \textbf{(Fig 1)} . The protrusion of
terminal ileum into ascending colon gave an intraluminal pseudomass
appearance \textbf{(Fig 2)} . The small bowel loops proximal to
ileocolic junction was dilated with outer-to-outer diameter of 3.2cm and
mottled appearance giving small bowel feces sign.\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image1/image1}

\end{center}
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\includegraphics[width=0.70\columnwidth]{figures/image2/image2}

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Fig 2: Coronal image showing intraluminal

pseudomass appearance with perlesional

fat strandings Fig 1: Axial image showing thickened ileocolic junction
with target stratification pattern\selectlanguage{english}
\begin{figure}[H]
\begin{center}
\includegraphics[width=0.70\columnwidth]{figures/image3/image3}

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The patient underwent an exploratory laparotomy. Intraoperatively there
was a solid lesion measuring approximately 5\selectlanguage{ngerman}×6 cm, well-demarcated
smooth mass involving the cecum, approximately 2 cm proximal to
ileocolic junction \textbf{(Fig: 3 and 4)} . There was no evidence of
ascites, no mesenteric lymphadenopathy and liver surface appeared
normal. Lead point of ileocolic intussusception was the mass and right
sided standard hemicolectomy performed. On cut surface, the lesion had
diffuse grey to yellowish appearance. The procedure completed without
complications and the specimen was marked and sent for histopathological
examination.

Fig 3: Tumor mass removed along with segment of ileum\selectlanguage{english}
\begin{figure}[H]
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\includegraphics[width=0.70\columnwidth]{figures/image4/image4}

\end{center}
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.

Fig 4: Intraoperative image with tumor insitu

The post-operative course was unremarkable, and after 24 hours of
observation in Intensive care Unit (ICU) the patient was transferred to
the surgery ward. He recovered well and discharged after fifth
Post-operative day.

Histopathological examination of the resected specimen showed an
Inflammatory Myofibroblastic tumor. The tumor showed nodular hyperplasia
with diffuse eosinophilic infiltrate, subserosal bland spindle to
stellate cells proliferation along with lymphoid aggregates,
lymphocytes, plasma cells, and eosinophils.

\textbf{Discussion} : Inflammatory Myofibroblastic tumors being rare
neoplasms with diverse clinical presentations can affect various organs
like lungs, liver, endolarynx, maxillary sinus, oral cavity etc.{[}5{]}
However, their involvement in the gastrointestinal tract is particularly
rare and their expression as intussusception is even rarer. {[}4{]}.
Their exact etiology remains uncertain but they can even mimic various
inflammatory conditions and their behavior ranges from benign to locally
aggressive or rarely malignant, making the diagnosis challenging.

Some common causes of adult ileocolic intussusception are adhesions,
polyp, inflammatory bowel disease, gastro intestinal tumor (GIST) etc.
{[}6{]}. Inflammatory Myofibroblastic Tumor Presenting as Ileocolic
Intussusception is a rare diagnosis, so it is even difficult for a
resources equipped centers to diagnose. Diagnosing this rare condition
in Hetauda Hospital, a district hospital located in sub-urban region
with inadequate resources is quite challenging. The diagnosis of this
condition at our center either reflects the inadequacies in studies or
not frequently reported.

In our case, the patient's presentation with abdominal pain, vomiting,
and imaging findings of intussusception prompted further investigation,
leading to the diagnosis of an IMT. Surgical resection followed by
histopathological examination of the resected specimen performed.

Immuno histochemical staining (IHC) which helps to identify specific
marker expressed by tumor cells like Smooth muscle actin (SMA),
anaplastic lymphoma kinase (ALK Protein), vimentin, and CD34. These IHC
features can help in distinguishing benign or malignant nature of the
tumor. Together in conjunction with histopathological features, they can
help to establish confirmatory diagnosis. However, in sub-urban area
like ours, patient presents with their complaints and after getting
relief from their issues, they do not come up to hospital for follow up.
Therefore, in our case we are unable to give Immunohistochemistry (IHC)
reports as he was lost to follow up.

Further studies, research and case reports are necessary to understand
the etiology, behavior, and optimal management strategies for IMTs,
particularly those involving gastrointestinal tract. Additionally,
increased awareness among clinicians is also essential to ensure early
recognition, prompt diagnosis, and appropriate surgical intervention, as
demonstrated in this case, are crucial for optimal management and
favorable patient outcomes of these rare tumors.

\textbf{Conclusion} :

This case report shares a very rare condition of inflammatory
myofibroblastic tumor (IMT), presenting with ileocolic intussusception
in a middle-aged man, which is extremely uncommon and can be confused
with other inflammatory conditions however, through diagnostic
evaluation and multidisciplinary approach can result in early diagnosis,
optimal management and favorable patient outcomes of these rare tumors.

\textbf{References}

1. Koyuncuer, A., \emph{Inflammatory myofibroblastic tumor of the
small-bowel mesentery: A case report of nonspecific clinical
presentation and a review of the literature.} Int J Surg Case Rep,
2014.\textbf{5} (12): p. 1214-7.

2. Camela, F., et al., \emph{Pulmonary Inflammatory Myofibroblastic
Tumor in Children: A Case Report and Brief Review of Literature.} Front
Pediatr, 2018. \textbf{6} : p. 35.

3. Khalil, S., T. Ghafoor, and A.K.F. Raja, \emph{Inflammatory
Myofibroblastic Tumor: A Rare Presentation and an Effective Treatment
with Crizotinib.} Case Reports in Oncological Medicine,
2020.\textbf{2020} : p. 6923103.

4. Hameed, T., et al., \emph{Acute Intestinal Obstruction Due to
Ileocolic Intussusception in an Adult; A Rare Presentation of
Inflammatory Myofibroblastic Tumor.} Am J Case Rep, 2020. \textbf{21} :
p. e920438.

5. Palaskar, S., et al., \emph{Inflammatory myofibroblastic
tumor.}Contemp Clin Dent, 2011. \textbf{2} (4): p. 274-7.

6. Tarchouli, M. and A. Ait~Ali, \emph{Adult Intussusception: An
Uncommon Condition and Challenging Management.} Visceral Medicine,
2020.\textbf{37} (2): p. 120-127.\selectlanguage{english}
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