Yersinia enterocolitica enterocolitis in a patient with sickle ...

Clinical Medicine Insights: Pathology
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Intussusception caused by Yersinia enterocolitica
enterocolitis in a patient with sickle cell Anemia
Geetanjali Gupta1, shailesh Kumar2, reecha singh1 and Kathirvelu shanmugasamy1
1Department of Pathology, Mahatma Gandhi Medical College and research Institute, Puducherry, India 607402.
2Department of Microbiology, Mahatma Gandhi Medical College and research Institute, Puducherry, India 607402.
Corresponding author email: gng_82@y ahoo.co.in
Abstract: Yersinia enterocolitica intussusception is rarely encountered in patients without an underlying susceptibility and is most

frequently reported in iron-overloaded patients. This is thought to be related to the unusual use of iron by this microorganism. We present
a case of a 5-year old child with intussusception of the terminal ileum caused by Y. enterocolitica whose past medical history was sig-
nificant for sickle cell disease. This type of presentation is extremely rare. His monthly blood transfusions may have put him at risk for
Y. enterocolitica enterocolitis. The pathogenesis of this disease relates to the role of iron as an essential growth factor for Y. enterocolitica,
and this patient’s transfusions left him in an iron overloaded state despite treatment with Deferoxamine. Our patient’s unusual presenta-
tion of intusssuception was secondary to the mass effect caused by lymphoid hyperplasia, specifically hypertrophied Peyer’s patches in
the ileum caused by the Y. enterocolitica infection. We believe that our case demonstrates that Y. enterocolitica should be considered a
possible pathogen in patients with sickle cell disease, especially if symptoms occur shortly after blood transfusion.
Keywords: Yersinia enterocolitica, intussusception, sickle cell anemia
Clinical Medicine Insights: Pathology 2010:3 7–11
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Gupta et al
Introduction
The patient was treated with antimicrobials and had
Patients who are homozygous for sickle cell anemia
an uneventful postoperative course.
are at risk for Yersinia enterocolitica. We present a
case of a 5-year-old with intussusception of the termi-
Materials and Methods
nal ileum whose past medical history was significant
The surgical specimens were fixed in formalin and
for sickle cell anemia. His monthly blood transfu-
embedded in paraffin. The sections were stained with
sions may have put him at risk for Y. enterocolitica
routine Hematoxylin and Eosin stains. Special stains
enterocolitis. The pathogenesis of this disease relates
were perfomed including Brown and Hopps stain for
to the role of iron as an essential growth factor for
Yersinia. A culture was grown from samples taken
Y. enterocolitica, and this patient’s transfusions left
from the mesenteric lymph node.
him in an iron overloaded state, despite treatment
with Deferoxamine. Our patient’s unusual presenta-
tion of intussusception was secondary to the mass
Results
effect caused by lymphoid hyperplasia, specifically
Surgical specimens included a section of terminal
hypertrophied Peyer’s patches in the ileum caused by
ileum measuring 18 × 4 × 4 cm that telescoped into
Y. enterocolitica infection. To our knowledge, this is
the ileocecal region (Fig. 1). Other specimens
the first case of intussusception as a complication of
included a section of the cecum and ascending
Y. enterocolitica enterocolitis in a patient with sickle
colon measuring 9 × 5 × 4 cm, and another section of
cell anemia.
the cecum attached to the vermiform appendix that

measured 5 × 1 × 1 cm. All serosal surfaces were
case presentation
erythematous. Multiple discrete lymph nodes were
A 5-year-old boy was hospitalized with a 3-day
seen in the mesentery. Microscopically extensive,

history of abdominal cramps, diarrhoea, bloody
severe, transmural, acute and chronic inflammation
stools with mucus, intermittent emesis and fevers to
was present in the terminal ileum, cecum, right
40 °C. His past medical history was significant for
colon and appendix, as were ulceration, abscess for-
sickle cell disease, and he had received blood trans-
mation, lymphoid hypertrophy, focal necrosis and
fusions once every 3–4 weeks for the 3 years previ-
focal peritonitis (Figs. 2, 3, 4 and 5). Lymph nodes
ous. His last transfusion (225 ml of packed cells) had
showed necrotizing lymphadenitis and extensive
occurred 5 days prior to admission. He also received
infarction. The Brown and Hopps stain revealed
weekly Deferoxamine treatment over the previous
gram negative bacilli. The culture of the mesenteric
1.5 years.
On admission, the child appeared ill and was
dehydrated and tachypneic. His abdomen was dis-
tended, with diffuse tenderness, guarding and peri-
toneal signs in the right lower quadrant but with
positive bowel sounds. A computed tomography scan
of the abdomen revealed an inflammatory process in
the right lower quadrant, with a small amount of free
air and fluid collection that was consistent with an
abscess. The patient was taken for exploratory lapa-
rotomy, ileocecectomy and ileostomy, during which
time a 3 cm ileocolic intussusception was manually
reduced. Mesenteric adenitis was noted. The appen-
dix was erythematous and bulging. A preoperative
stool culture in Cefsoludin-Irgasan-Novobiocin
(CIN) agar grew Y. enterocolitica (serotype 0:3).
Figure 1. Photograph of the terminal ileum telescoped into the ileocecal
region.
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Yersinia enterocolitica intussusception in a patient with sickle cell anemia
Figure 2. Microscopic view (Hematoxylin and eosin stain; 200X
Figure 4. Microscopic view (Hematoxylin and eosin stain; 200X
­
magnification)­showing­severe­transmural­acute­and­chronic­inflamma-
­
magnification)­showing­mucosal­ulceration,­cryptitis­and­occasional­cryp-
tion, ulceration, abscess formation and lymphoid hypertrophy.
tic abscess.
lymph node (Fig. 6) yielded Y. enterocolitica of the
formation in the inguinal region and fatal septicaemia.1
same serotype, biotype and isotype as the organism
Bacterial infection in an intussusception has been
isolated from the stool sample.
described only sporadically. Y. enterocolitica is the
only bacterial agent responsible for infected intus-
susceptions in in children. Y. enterocolitica has been
Discussion
reported in three previous cases in North American lit-
Y. enterocolitica is a gram-negative coccobacillus with
erature and seven previous cases worldwide.2–6
worldwide distribution that commonly causes gastro-
Y. enterocolitica is spread primarily via the
enteritis and mesenteric adenitis in children and ado-

fecal—oral route, and infection most often results
lescents. Reported complications have included severe
from human consumption of contaminated meat
enterocolitis involving the ileocecal region, appendici-
products (especially pork), milk products, water
tis, small bowel gangrene, intestinal perforation, peri-
and vegetables. Less commonly, exposure to human
tonitis, hepatic and splenic abscesses, chronic abscess
and zoonotic fecal carriers such as pigs, dogs, cats
Figure 3. Microscopic view (Hematoxylin and eosin stain; 200X
­
magnification)­showing­lymphoid­hypertrophy,­and­lymphoid­hyperplasia­
Figure 5. Microscopic view (Hematoxylin and eosin stain; 200X
with prominent germinal centers in the lamina propria.
­
magnification)­showing­areas­of­necrosis­and­congestion.
Clinical Medicine Insights: Pathology 2010:3
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Gupta et al

solubilize ferric iron by competing with transferrin.
The iron-siderophore complexes enter the bacterial
cells following binding to specific receptors. Def-
eroxamine is a bacterial siderophore obtained from
Streptomyces piloce that is used as a chelating agent
in the treatment of acute iron toxicity and in chronic
iron overload. Y. enterocolitica is unusual among bac-
teria in that it requires iron for growth and has recep-
tors for siderophores but cannot produce siderophores
endogenously.1
conclusions
This is the first case, to our knowledge, of intussuscep-
tion as a complication of Y. enterocolitica enterocolitis
in a patient with sickle cell disease. Recommenda-
tions from this and previous articles indicate that in
a patient with iron overload, the clinician should be
alerted to the possibility of Y. enterocolitica infec-
Figure 6. “Bull’s-eye view” of a colony of Yersinia enterocolitica on
tion when abdominal symptoms and fever occur. The

Cefsoludin-Irgasan-Novobiocin (CIN) agar.
index of suspicion should be raised for patients with
sickle cell disease especially if the symptoms occur
and rats results in this disease.7 Our patient acquired
shortly after a blood transfusion.
the infection from contaminated water.
Intussusception is more common in infants aged
Disclosures
4 to 10 months, with a peak at 7 months. It is pro-
This manuscript has been read and approved by all
posed that hypertrophied Peyer’s patches, possibly
authors. This paper is unique and is not under con-
resulting from viral infection, act as a mechanical
sideration by any other publication and has not been
lead point for intussusceptions. The intussusception
published elsewhere. The authors and peer review-
in our case was in the terminal ileum, secondary to
ers of this paper report no conflicts of interest. The
mass effect caused by lymphoid hyperplasia. In one
authors confirm that they have permission to repro-
prospective study of 261 patients (228 children and
duce any copyrighted material.
33 adults) with intussusceptions, 88% of the cases
were idiopathic, without any definitive lead point. In
References
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