Giant duodenal ulcers after neurosurgery for brainstem tumors that required reoperation for gastric disconnection: a report of two cases
Nobori et al. BMC Surgery
Giant duodenal ulcers after neurosurgery for brainstem tumors that required reoperation for gastric disconnection: a report of two cases
Chihoko Nobori 0
Kenjiro Kimura 0
Go Ohira 0
Ryosuke Amano 0
Sadaaki Yamazoe 0
Hiroaki Tanaka 0
Kentaro Naito 1
Toshihiro Takami 1
Kosei Hirakawa 0
Masaichi Ohira 0
0 Department of Surgical Oncology, Osaka City University Graduate School of Medicine , 1-4-3 Asahi-machi, Abeno-ku, Osaka , Japan
1 Department of Neurosurgery, Osaka City University Graduate School of Medicine , 1-4-3 Asahi-machi, Abeno-ku, Osaka , Japan
Background: Despite the efficacy of pharmacotherapy for gastrointestinal ulcers, severe cases of bleeding or perforation due to gastrointestinal ulcers still occur. Giant duodenal ulcer perforation is an uncommon but difficultto-manage pathology with a high mortality rate. We report two cases of giant duodenal ulcer perforation after neurosurgery for brainstem tumors that needed reoperation for gastric disconnection because of postoperative leakage and bleeding. Case presentation: Both cases had undergone neurosurgery for brainstem tumors, and the patients were in a shock state for several days with peritonitis due to giant duodenal perforation. In Case 1, antrectomy with Billroth II reconstruction was performed. However, reoperation for gastric disconnection was needed because of major leakage of gastrojejunostomy and jejunojejunostomy. In Case 2, an omental patch, cholecystectomy, and insertion of a bile drainage tube from the cystic duct were performed for the giant duodenal ulcer, but leakage and bleeding from the ulcer edge required reoperation for gastric disconnection. Conclusions: Brainstem tumors in these cases might have been related to duodenal ulcer perforation with late diagnosis that progressed to severe sepsis. For giant duodenal ulcer perforation with poor general condition, simple closure including omental patch or antrectomy with reconstruction is hazardous. Antrectomy with gastric disconnection, meaning gastrostomy, duodenostomy, feeding jejunostomy and cholecystectomy, is recommended.
Giant duodenal ulcer; Gastric disconnection; Brainstem tumor
Cushing reported gastroduodenal ulcers produced by
elevated intracranial pressure caused by an intracranial
tumor, head injury, or other space-occupying lesion, which
have been called Cushing’s ulcer . The use of histamine
H2-receptor antagonists or proton pump inhibitors can
decrease the incidence of Cushing’s ulcer and its
complications, such as bleeding and perforation. However, cases
of severe bleeding or perforation from gastroduodenal
ulcers still occur. Generally, duodenal ulcer perforation is a
surgical emergency. Factors such as advanced age,
concomitant disease, preoperative shock, large size of the
perforation, and delays in presentation and operation have
been identified as risk factors for mortality from duodenal
ulcer perforation . Gapta et al. classified duodenal ulcer
perforations into three groups based on the size of the
perforations: ‘small’ perforations less than 1 cm in
diameter; ‘large’ perforations more than 1 cm but less than
3 cm in diameter; and ‘giant’ perforations exceeding 3 cm
. Small and large perforations are common and
relatively easy to manage, resulting in low mortality rates. On
the other hand, giant perforations are uncommon but
difficult to manage and associated with higher mortality
rates. Simple closure or omental patching alone have been
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reported as unsafe. Two cases of giant duodenal ulcer
perforation after neurosurgery that needed re-operation
because of postoperative leakage and bleeding are described.
Taking these cases into account, we discuss how to cope
with perforation of a giant duodenal ulcer that has
progressed to sepsis because of late diagnosis.
Case 1 involved a 25-year-old man who had undergone
surgical resection of anaplastic ependymoma extending
from the brainstem to the fourth ventricle (Fig. 1). Two
days after neurosurgery, laboratory data showed an
unexpectedly severe inflammatory response (white cell count,
18,900/μL; C-reactive protein (CRP), 12.8 mg/dl). The
patient was observed with administration of meropenem.
Two days later, he developed shock and the abdomen
appeared severely distended. Vital signs were: temperature,
39.1 °C; heart rate, 130 beats/min; blood pressure, 73/
37 mmHg under medication with dopamine 8 μg/kg/min
and noradrenaline 0.25 μg/kg/min; and oxygen saturation,
94 % in room air. Laboratory data showed: white cell
count, 23,100/μL; platelet count, 32,000/μL; CRP, 5.48 mg/
dL. Computed tomography (CT) showed free air and
massive ascites (Fig. 2), and emergency surgery was
performed under a presumptive diagnosis of gastrointestinal
perforation. On laparotomy, 3 L of muddy ascites was
removed, and a perforation 3.5 cm in diameter was found in
the second portion of the duodenal bulb (Fig. 3).
Antrectomy including the ulcerated portion using Billroth II
reconstruction with Braun anastomosis, insertion of a
duodenal drainage tube from the duodenal stump, and
cholecystectomy with insertion of a bile drainage tube from the
cystic duct were performed.
Ten days after the ulcer operation, major leakage of
the gastrojejunostomy and jejunojejunostomy required
re-operation, involving gastric disconnection,
gastrostomy, duodenostomy, and feeding jejunostomy. After
reoperation, the patient developed multiple-organ failure,
but he recovered with intensive care. Eight months after
the reoperation, digestive tract reconstruction surgery
was performed using the Roux-en-Y method. Since that
reconstruction surgery, the patient has been making
Case 2 involved a 62-year-old woman. She had
undergone surgical resection of a brainstem hemangioblastoma
that progressed acutely after stereotactic radiosurgery
(Fig. 4). Six days after neurosurgery, laboratory data
revealed an unexpectedly severe inflammatory response
(white cell count, 23,100/μL; CRP, 18.5 mg/dL). However,
she was observed with administration of cefepime. After
another 3 days, she developed shock and the abdomen
appeared distended. Vital signs were: temperature, 38.1 °C;
heart rate, 140 beats/min; blood pressure, 60/40 mmHg
under medication with dopamine 10 μg/kg/min and
noradrenaline 0.15 μg/kg/min; and oxygen saturation, 92 % in
room air. Laboratory data showed: white cell count, 18,100/
μL; platelet count, 29,000/μL; CRP, 4.1 mg/dL. CT showed
massive ascites, but no free air at that time (Fig. 5).
Aspirated ascites showed intestinal juice, so emergency
surgery was performed under a diagnosis of
On laparotomy, 4 L of biliary ascites was removed, and a
perforation 4 cm in length was found at the duodenal bulb
(Fig. 6). An omental patch over the perforation site,
insertion of a drainage tube into the duodenum from the
anterior wall of the stomach, and cholecystectomy with
insertion of a bile drainage tube from the cystic duct were
performed. Fifteen days after the ulcer operation,
continuous bleeding at the wall edge of the duodenal ulcer
required reoperation. Operative findings revealed ulcer
bleeding and dehiscence of the perforation site. Gastric
disconnection was performed, comprising antrectomy
including resection of the ulcerated portion, tube
duodenostomy, and tube gastrostomy. The patient also needed
intensive care, and her condition improved after 3 months.
However, digestive reconstruction surgery has not yet
Fig. 1 Head MRI. The MRI scan reveals an anaplastic ependymoma that extended from brainstem to forth ventricle
been performed as of the time of writing, as the brain
tumor recurred during recovery.
In these two cases, the brainstem tumors might have been
related to duodenal ulcer perforation that progressed to
septicemia. In 1841, Rokitansky suggested for the first time
that ulcerative processes of the stomach might involve
dysfunction of nervous mechanisms . In 1932, Cushing
reported gastroduodenal ulcers produced by elevated
intracranial pressure caused by an intracranial tumor, head
injury, or other space-occupying lesion. He suggested that
such ulcerative processes might be related to diencephalic
or brainstem disorders affecting the parasympathetic
Fig. 3 The intra-operative finding. The perforation 3.5-cm in diameter
was found in the second portion of the duodenal bulb
nervous system. Since then, ulcers of this type have been
called Cushing’s ulcers .
The mechanism of ulceration appears to involve three
routes from the central nervous system to the stomach: 1)
anterior hypothalamus – vagus nerve; 2) posterior
hypothalamus – sympathetic nerve; and 3) posterior
hypothalamus – anterior pituitary gland – adrenal cortex. Through
these three routes, factors that aggravate the stomach are
increased or protective factors are decreased. The
sympathetic and parasympathetic nervous systems usually
maintain a balance of the blood supply, gastric secretion, and
gastric motility. Dysfunction of the central nervous system
stimulates the hypothalamus, which then stimulates the
sympathetic and parasympathetic nervous systems.
Stimulation of sympathetic nerves decreases blood supply to the
stomach, and stimulation of parasympathetic nerves
increases gastric secretion. Moreover, adrenal cortical
hormones through the anterior pituitary gland decrease
gastric mucus secretion [4–10]. These factors then
contribute to the development of gastroduodenal ulcers.
In general, factors such as advanced age, concomitant
disease, preoperative shock, large size of the perforation,
and delays in presentation and operation have been
identified as risk factors for mortality in duodenal ulcer
perforation . Based on these factors, several scoring
systems have been used to evaluate the condition of the
patient with duodenal ulcer perforation, such as the
Boey score , Mannheim Peritonitis Index [12–14],
APACHE II score  and Jabalpur score . In
particular, the perforation-operation interval seems to
represent an important factor for mortality. Mishra et al.
reported that the mortality rate is 3 % within 24 h, 57 %
from 25 to 72 h, and 80 % over 120 h after duodenal
ulcer perforation . Many reports have stated that an
interval to operation larger than 24 h increases the
mortality rate [17–19], because heavier bacterial
contamination occurs in patients with delayed treatment . In
the present two cases, decreased level of consciousness
was the major cause of delayed diagnosis in both
patients. Although gauging the interval since ulcer
perforation is difficult, at least 48 h may have elapsed in both
cases, given the presence of septic shock. Of the above
risk factors, our two cases showed large perforations,
delayed diagnosis, concomitant disease, and preoperative
shock, as well as advanced age in Case 2. Operations in
such cases are generally difficult. Nonetheless,
antrectomy with Billroth II reconstruction was performed for
Case 1 and omental patching was performed for Case 2.
Because gastric disconnection requires a second operation
for digestive reconstruction, we hesitated to perform this
procedure, but gastric disconnection was unavoidable at
the first emergency surgery.
Most duodenal ulcer perforations are less than 1 cm in
length, and can be successfully treated with one-layer
Fig. 4 Head MRI. The MRI scan reveals an a brainstem hemangioblastoma
closure plus a pedicled omental patch (Cellan-Jones
technique) or an omental patch repair (Graham technique)
[21–23]. On the other hand, giant duodenal ulcers are
uncommon, with duodenal ulcer perforation more than
3 cm in length reportedly accounting for about 1.23 % of
cases . Giant duodenal ulcers are difficult to manage
and are associated with high rates of both morbidity (20–
70 %) and mortality (15–40 %) because of the extensive
duodenal tissue loss and surrounding tissue inflammation
. The Cellan-Jones and Graham techniques often fail
to achieve closure of the perforation, resulting in
postoperative leakage or gastric outlet obstruction.
Several reports have described surgical procedures for
giant ulcers, including partial gastrectomy, jejunal
serosal patch , free omental plug , and jejunal pedicle
graft . Lal et al. reported the efficacy of
triple-tubeostomy (tube gastrostomy, retrograde tube duodenostomy,
and feeding jejunostomy) with repair of the perforation for
large duodenal ulcer perforations . Cranford et al.
advocated gastric disconnection with truncal vagotomy,
antrectomy, and triple-tube-ostomy . This surgical
approach is considered the most appropriate procedure
for giant duodenal ulcer perforation in cases with poor
general conditions owing to late diagnosis. Because one
of the present cases showed bleeding and leakage from
the repaired duodenal ulcer, antrectomy including the
ulcerative portion was thought to be necessary for giant
duodenal ulcer. In cases with poor general conditions
owing to late diagnosis, digestive tract reconstruction is
hazardous, and gastric disconnection might be needed.
This approach necessitates a second elective operation
for digestive reconstruction, but is thought to represent
the safest procedure given the high mortality rate of
this condition. Moreover, cholecystectomy with
insertion of a bile drainage tube from the cystic duct might
Fig. 5 Abdominal CT. The CT scan reveals massive fluid accumulation
and an irregular duodenal wall
Fig. 6 The intra-operative finding. An 4-cm perforation was noted at
the anterior wall of the duodenval bulb
We have reported two cases of giant duodenal ulcer
perforation after neurosurgery that needed reoperations because
of postoperative leakage and bleeding. For giant duodenal
ulcer with poor general condition owing to late diagnosis,
simple closure including omental patching or antrectomy
with reconstruction is hazardous. Antrectomy with gastric
disconnection, which means gastrostomy, duodenostomy,
feeding jejunostomy and cholecystectomy, is recommended.
CN, KK, and GO were involved in data collection, case analysis and writing
the manuscript. RA, SY, HT, MO, and KH assisted in drafting the manuscript
and reviewed the article. TT and KN performed the first surgeries. All authors
read and approved the final manuscript.
Consent for publication
Written informed consent was obtained from the patients for publication of
this case report and the accompanying images. A copy of written consent is
available for review by the editor of this journal.
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