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Med Sci Monit, 2009; 15(9): CS139-142 WWW. M ED S CI M ONIT .COM

PMID: 19721403 Case Study

Received: 2008.01.16
Pneumatosis intestinalis in ulcerative colitis CS
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Accepted: 2008.03.12
Published: 2009.09.01

Authors Contribution: Ayako Matsumoto1 BF, Hajime Isomoto2 ACDEF, Saburo Shikuwa1 ABD,
A Study Design
B Data Collection
Kenta Okamoto1 B, Naoyuki Yamaguchi1 BC, Ken Ohnita2 D, Yohei Mizuta2 C,

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C Statistical Analysis Masahiro Fujii3 B, Shigeru Kohno2 DEF
D Data Interpretation
1
E Manuscript Preparation Department of Internal Medicine, National Nagasaki Medical Center, Omura, Japan
2
F Literature Search 2nd Department of Internal Medicine, Nagasaki University School of Medicine, Nagasaki, Japan
3
G Funds Collection Fujii Clinic, Omura, Japan

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Source of support: Departmental sources

LY LSummary
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Background:
N A UC patients are considered to be at risk for pneumatosis intestinalis (PI).
Case Report: A 50-year-old man who had been treated with prednisolone for left-sided ulcerative colitis (UC)
underwent follow-up colonoscopy. In addition to active colitis in the left colon, some cystic lesions
O N
were found in the unaffected ascending colon. Endoscopic ultrasonography and computed tomog-
raphy conrmed the presence of intramural air, consistent with PI. Since corticosteroid use might
contribute to the development of PI, the patient was successfully treated with leukocytapheresis.
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At the last follow-up colonoscopy the UC was still in remission and the pneumatic cysts were re-
solving.
Conclusions: It is important to determine the clinical signicance of PI in each patient to ensure appropriate
therapy.
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key words: ulcerative colitis pneumatosis intestinalis prednisolone

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Full-text PDF: http://www.medscimonit.com/fulltxt.php?ICID=878165

Word count: 1202
Tables:
Figures: 3
References: 21
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Authors address: Hajime Isomoto, 2nd Department of Internal Medicine, Nagasaki University School of Medicine, 1-7-1 Sakamoto,
Nagasaki 852-8501, Japan, e-mail: hajimei2002@yahoo.co.jp

Current Contents/Clinical Medicine IF(2008)=1.514 Index Medicus/MEDLINE EMBASE/Excerpta Medica Chemical Abstracts Index Copernicus CS139

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Case Study Med Sci Monit, 2009; 15(9): CS139-142

BACKGROUND A
Pneumatosis intestinalis (PI), also known as pneumatosis
cystoides intestinalis or pneumatosis coli, is dened as the
presence of gas within the bowel wall. Although there have
been several reported cases of idiopathic PI, most cases oc-
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cur in association with underlying or coexistent diseases

[1,2]. PI is classically found in the following clinical settings:
premature infants with necrotizing enterocolitis, children
with congenital heart defects, adults with obstructive pul-
monary disease, adults and children with a wide variety of

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gastrointestinal pathology, connective tissue diseases, trans-
plant, and drug therapy, particularly steroids and immuno-
suppressive agents [1,2]. However, PI has rarely been found
in association with inammatory bowel disease, including
ulcerative colitis (UC). In this report a case of left-sided UC
treated with daily prednisolone that developed PI in the un-

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affected ascending colon is presented. B
CASE REPORT

LY L
A 50-year-old man presented with a four-week history of rec-
tal bleeding. He had been diagnosed with left-sided UC at
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the age of 36 years and had been treated with oral prednis-
N A
olone plus mesalazine followed by oral mesalazine alone.
The patient had been in remission until the latest recur-
rence, at which time colonoscopy showed active left-sided
O N
UC. He was treated with intravenous prednisolone, starting
with 60 mg/day, which resulted in immediate disappear-
ance of the disease. Four months later he appeared to have
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a are of his symptoms despite taking 10 mg of oral pred-

nisolone daily and he was admitted to hospital. His family
history was noncontributory.

On physical examination, the patient was afebrile and had C

stable vital signs. The palpebral and bulbar conjunctivae
were normal and without anemia or jaundice, respectively.
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No wheezing was audible in lung elds. The patients ab-

domen was soft, with no distension and no evidence of re-
bound, guarding, or tenderness; the bowel sounds were nor-
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mal. Admission laboratory investigations showed a leukocyte

count of 6400/mm3 (normal: 35009100/mm3), platelet
count of 23.6104/mm3 (normal: 13.036.9104/mm3), he-
moglobin of 15.3 g/dl (normal: 11.315.2 g/dl), C-reactive
protein (CRP) of 1.29 mg/ml (normal: <0.3 mg/ml), and
serum total protein of 6.8 g/dl (normal: 6.58.2 g/dl).
Urinalysis was normal and no infectious agents were iden-
tied in stool specimens. Clostridium difcile toxin was neg-
ative in the feces. No air was visualized within the colon
wall on abdominal X-ray. Barium examination including
the small intestine and upper gastrointestinal endoscopy
was normal.
Figure 1. Colonoscopy on the next hospitalization day shows friable,
Colonoscopy performed on the next hospitalization day re- edematous mucosa with granularity, mucous exudates, and
vealed friable, edematous mucosa with granularity, mucous slight bleeding as well as diffuse erosions in the rectum (A).
exudates, and spontaneous bleeding throughout the rec- Several cystic lesions are seen on the normal-appearing fold
in the ascending colon, which was uninvolved by UC (B).
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tum and sigmoid colon, where diffuse erosions were noted

with no normal-appearing intervening mucosa (Figure 1A).
Histopathology of the colorectal biopsies showed diffuse
mucosal inltration of inammatory cells, deformed atro-
phic crypts, goblet cell depletion, and crypt abscesses. In
addition, several cystic lesions with intact overlying mucosa
were identied in the ascending colon, which was unaffect-
ed by UC (Figure 1B). Endoscopic ultrasonography showed
Endoscopic ultrasonography shows high-amplitude echoes
with acoustic shadowing within the submucosal cysts (C).
high-amplitude echoes with acoustic shadowing within the
submucosal cysts (Figure 1C), consistent with PI. Computed
tomography (CT) of the abdomen conrmed intramural

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Med Sci Monit, 2009; 15(9): CS139-142 Matsumoto A et al Pneumatosis intestinalis in ulcerative colitis

CS
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Figure 2. Computed tomography of the abdomen showed no porto
venous gas.

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B
air in the ascending colon without any porto venous gas
(Figure 2), excluding bowel ischemia.

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Based on the clinical, radiological, endoscopic, and histo-
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pathological ndings, the patient was diagnosed as having

N A
moderate active left-sided UC along with PI in the unaf-
fected ascending colon. The patient was treated with leu-
kocytapheresis (LCAP), which was performed using a com-
O N
mercially available leukocyte removal column (Cellsorba;
Asahi Medical, Tokyo, Japan) once a week for 10 succes-
sive weeks. The LCAP treatment led to improvement of
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the clinical symptoms without any adverse reactions and to

normalization of the CRP level. At the last follow-up colo-
noscopy three months after discharge, the UC was still in
remission (Figure 3A) and the pneumatic cysts were resolv-
ing (Figure 3B). Figure 3. Colonoscopy at the last follow-up shows that the UC was in
remission (A) and the pneumatic cysts were resolving (B).
DISCUSSION
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The pathogenesis of PI is not completely understood, but it of PI is unclear, it is possible that in the present case, lym-
is thought to be multifactorial [2]. Both bacterial and me- phodepletion secondary to steroid therapy may have creat-
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chanical theories have been proposed, based largely on clin-
ical entities known to be associated with PI [2]. According
to the bacterial theory, the hosts endogenous gut ora is
implicated as the principal gas producer. The gas-forming
bacterial overgrowth along with increased peristalsis and
increased intraluminal pressure may provide the mechani-
cal force that results in air entry into the bowel wall [3]. In
support of the mechanical theory [4], gas may dissect into
the bowel wall from the luminal surface through mucosal
breaks in gastrointestinal inammation, as seen in patients
with inammatory bowel disease (IBD) [5,6]. In addition,
steroids, as well as other immunosuppressants, are listed
among the clinical variables related to the development of
PI [7,8]. These risk factors place IBD patients, who are of-
ten treated with such medications, at an increased risk of PI.
However, only a handful of cases of PI have been reported
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ed functional defects in the mucosa, thereby permitting dis-
section of air into the non-inamed bowel [12,13]. See et al.
reported the case of a woman who had recently been treated
for left-sided UC with high-dose prednisone plus mercapto-
purine and then developed PI in the small intestine [10].
There is limited information regarding the management of
PI; however, the critical decision is whether to treat conser-
vatively or proceed with emergency surgery [11]. PI patients
with toxemia, those who have complications (obstruction,
intussusception, or volvulus), and those with underlying
causes associated with a high mortality rate require sur-
gery [1,2,1315]. In particular, concomitant hepatic por-
tal venous gas in a patient with PI is usually considered an
ominous prognostic sign [10,16]. This generally occurs in
acutely ill patients, most frequently in necrotizing enteroco-

in patients with IBD, either UC or Crohns disease [811].
Most patients with PI are asymptomatic [11] and PI may be
discovered incidentally on radiographic or endoscopic ex-
amination [11,12]. In our patient, PI was identied in the
uninvolved segment of the right colon when colonoscopy
was performed for relapse of UC, primarily affecting the
left colon. Although the role of steroids in the occurrence
litis. Heng et al. found that 10 of 12 patients with the com-
bination of PI and HPVG died within 48 hours [16]. Once
an abdominal emergency has been ruled out, and because
PI cysts may resolve over time, patients with no or mini-
mal symptoms only require treatment for the underlying
causes. Given that glucocorticoid use appears to be an im-
portant contributing factor [7, 8], as in the present case,
LCAP may be used as a treatment modality for active UC

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This copy is for personal use only - distrib
Case Study
complicated by PI. In our patient, LCAP led to signicant
improvement of the UC that had repeatedly ared during
treatment with prednisolone. Recent studies have demon-
strated that LCAP may be more effective than high-dose ste-
roid treatment with fewer adverse effects [17]. However, no
consensus has been reached on the signicance and man-
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agement of PI complicating IBD. It is also uncertain wheth-
er PI is a reliable indicator of severity in IBD. For patients
who are symptomatic and do not need surgery, there are
several medical treatment options available, including oxy-
gen therapy, either high-ow or hyperbaric, antibiotics, and
elementary diets [1821].
CONCLUSIONS
UC, especially when it is treated with corticosteroids, must
be considered a condition that may be associated with PI.
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As with any disease entity, the clinician must determine the
clinical signicance of PI in each patient to make appropri-
ate therapeutic decisions.
REFERENCES:
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