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\begin{document}

\title{Life-threatening gastrointestinal bleeding from splenic artery
pseudoaneurysm due to gastric ulcer penetration treated by surgical
hemostasis with resuscitative endovascular balloon occlusion of the
aorta: a case report}



\author[1]{Tomonori Nakata}%
\author[1]{Yuko Okishio}%
\author[1]{Kentaro Ueda}%
\author[1]{Toru Nasu}%
\author[1]{Shuji Kawashima}%
\author[1]{Kosei Kunitatsu}%
\author[1]{Seiya Kato}%
\affil[1]{Wakayama Medical University}%


\vspace{-1em}



  
  \date{May 25, 2024}


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\begin{abstract}
We report the case of a patient for whom surgical hemostasis of
gastrointestinal bleeding from a splenic artery pseudoaneurysm that
developed due to gastric ulcer penetration was achieved with
resuscitative endovascular balloon occlusion of the aorta.%
\end{abstract}%



\sloppy


\textbf{Introduction}

Splenic artery pseudoaneurysms (SAPs) are rare causes of upper
gastrointestinal bleeding (UGIB), with less than 250 reported cases in
the literature.\textsuperscript{1} Among these reported cases, SAPs are
most often caused by pancreatitis (52\%) or trauma
(29\%),\textsuperscript{2} and rarely by peptic ulcer disease. To the
best of our knowledge, only nine cases of SAPs caused by peptic ulcers
have been previously reported.\textsuperscript{2-10} UGIB caused by a
ruptured SAP presents a significant risk for patient survival, since a
mortality rate of up to 90\% has been reported for untreated
cases\textsuperscript{1} and the treatment strategies used can determine
whether a patient lives or dies. The use of resuscitative endovascular
balloon occlusion of the aorta (REBOA) to control non-traumatic bleeding
has increased\textsuperscript{11-13}; however, REBOA has not been used
in any case report of UGIB from a SAP that was caused by a peptic ulcer.
Herein, we report the case of a patient with UGIB from a SAP caused by a
peptic ulcer, who survived as the result of surgery using REBOA.

\textbf{Case report}

A 72-year-old female with a history of consumption of nonsteroidal
anti-inflammatory drugs for back pain was presented to our hospital for
melena and shock by the helicopter emergency medical service. The
patient had a history of atrial fibrillation and hypertension, for which
she was receiving rivaroxaban (10 mg/day) and carvedilol (10 mg/day).
During the emergency medical flight, the patient's blood pressure was
49/30 mmHg and her heart rate was 50 beats per minute (bpm). The patient
received an intravenous transfusion of crystalloid fluid and tracheal
intubation in transit to the hospital. Upon arrival at our emergency
department, the patient's vital signs and laboratory results included:
heart rate, 60 bpm; blood pressure, 95/44 mmHg; and hemoglobin, 4.0
g/dL. The patient received a transfusion of red blood cells and
fresh-frozen plasma for the treatment of anemia. Subsequent changes in
her vital signs and laboratory results, and the transfusion volume
administered, are shown in Figure 1. The patient underwent upper
gastrointestinal endoscopy; however, a lesion was not identified with
this technique due to profuse hemorrhage from the posterior wall of the
stomach. Consequently, contrast-enhanced computed tomography (CT) was
performed and showed the presence of free air in the abdominal cavity
and a large amount of extravasated fluid in the gastric lumen (Figure
2); thus, we decided to perform surgery to achieve hemostasis. Following
CT, the patient developed shock. We inserted a 7-French sheath into the
right femoral artery using the Seldinger technique. Then, a REBOA
catheter (RESCUE balloon catheter; Tokai Medical Products, Aichi,
Japan) was advanced to the distal thoracic aorta. Although the patient's
blood pressure had decreased immediately prior to surgery, we were able
to control her blood pressure and the intragastric hemorrhage by
completely occluding the aorta with balloon inflation using the REBOA
technique. During the surgery, a circular transmural ulcer with a
diameter of 30 mm was found in the posterior wall of the stomach, and a
pyloric gastrectomy was performed (Figure 3). Active hemorrhage from the
splenic artery resumed after the endovascular balloon was deflated;
therefore, the ulcer was closed with sutures to achieve hemostasis. The
patient was admitted to the intensive care unit (ICU) due to the
development of coagulopathy and required open abdominal management to
evaluate blood flow in the gastric remnant. Subsequently, the patient's
vital signs stabilized. A Roux-en-Y reconstruction procedure was
performed on the second day of hospitalization. The patient was
discharged from the ICU on the third day of hospitalization and was
discharged from the hospital on the 29th day of hospitalization without
organ dysfunction.

\textbf{Discussion}

SAP is uncommon. In a large case series from the Mayo Clinic, 10 SAPs
were found during an 18-year period.\textsuperscript{2} Among previously
reported cases, SAPs are most often caused by pancreatitis (52\%) or
trauma (29\%)\textsuperscript{2} and are rarely caused by peptic ulcer
disease. To the best of our knowledge, there are only nine reported
cases of SAPs caused by peptic ulcers.\textsuperscript{2-10} Thus, for a
patient with UGIB after pancreatitis or trauma, a ruptured SAP should be
considered as a differential diagnosis. However, the diagnosis of SAP
due to a peptic ulcer poses a significant challenge for a clinician. In
contrast to true aneurysms, which involve all three layers (intima,
media, and adventitia) of an arterial wall, pseudoaneurysms typically
involve only the intima and media. Unlike true aneurysms, SAPs carry a
much higher risk of rupture.\textsuperscript{3} Control of UGIB caused
by ruptured SAPs is critical to the survival of patients since the
mortality rate for this condition can reach 90\% in untreated
cases\textsuperscript{1} and the treatment strategies selected can
determine whether patients live or die. Recently, retrospective studies
and case reports involving multiple patients have discussed the use of
REBOA for the treatment of non-traumatic hemorrhagic
shock.\textsuperscript{11-13}

In cases of traumatic shock and cardiopulmonary arrest, aortic occlusion
with resuscitative thoracotomy (RT) is a method of temporizing distal
hemorrhage while augmenting cerebral and coronary
perfusion.\textsuperscript{14} REBOA is also widely performed in cases
of traumatic shock and is minimally invasive compared with
RT.\textsuperscript{14,15} Among patients in traumatic shock who did not
require cardiopulmonary resuscitation before REBOA or RT, those who
underwent REBOA had a significantly higher survival
rate.\textsuperscript{14} There are two types of REBOA balloon
management strategies: intermittent and partial. Intermittent REBOA
occludes the aorta completely, occlusion time is limited to 35--40 min
in the distal thoracic aorta,\textsuperscript{15} and the occlusion
process involves repeated inflation and deflation of the balloon. In
contrast, partial REBOA occludes the aorta partially to minimize
ischemic injury caudal to the balloon, while maintaining cerebral and
coronary perfusion. Although the retrospective studies included cases of
hemostasis with REBOA, little consideration was given to the influence
of balloon management in these cases. In our patient's case, management
by intermittent REBOA was useful, not only for maintaining cerebral and
coronary artery perfusion, but also for securing the surgical field and
identifying the source of the hemorrhage. Moreover, the durations of the
complete occlusion times were 20 min and 5 min, respectively. Thus, the
patient recovered without organ dysfunction. Since there are no
conclusive indications as to whether partial or intermittent REBOA is
better for balloon management, we propose that the choice of REBOA for
balloon management should be based on the hemodynamics and bleeding
characteristics of lesions. Future studies must aim to clarify the
adaptation of REBOA for non-traumatic hemorrhage and to consider the
type of REBOA balloon management used.

\textbf{Conclusion}

This case report showed that REBOA can be used to control UGIB and blood
pressure during surgery for a ruptured SAP.

\textbf{Authorship list}

TN wrote the first draft of the manuscript. YO, KK, and SK reviewed and
revised the manuscript. YO, KU, TN, and SK performed the surgery and
contributed to patient care. All authors read and approved the final
manuscript.

\textbf{Acknowledgements}

None.

\textbf{References}

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hemorrhage due to gastric cancer eroding into a splenic artery
pseudoaneurysm: two dangerously rare etiologies of upper
gastrointestinal bleeding. Cureus 12:e10685.

2. Tessier D. J., Stone W. M., Fowl R. J., et al. 2003. Clinical
features and management of splenic artery pseudoaneurysm: case series
and cumulative review of literature. Journal of Vascular Surgery
38:969-974.

3. Varshney P., Songra B., Mathur S., et al. 2014. Splenic artery
pseudoaneurysm presenting as massive hematemesis: a diagnostic dilemma.
Case Reports in Surgery 2014:501937.

4. Sawicki M., Marlicz W., Czapla N., et al. 2015. Massive upper
gastrointestinal bleeding from a splenic artery pseudoaneurysm caused by
a penetrating gastric ulcer: case report and review of literature.
Polish Journal of Radiology 80:384-387.

5. Pasumarthy L., Kumar R. R., Srour J., and D. Ahlbrandt. 2009.
Penetration of gastric ulcer into the splenic artery: a rare
complication. Gastroenterology Research 2:350-352.

6. Panzera F., Inchingolo R., Rizzi M., et al. 2020. Giant splenic
artery aneurysm presenting with massive upper gastrointestinal bleeding:
a case report and review of literature. World Journal of
Gastroenterology 26:3110-3117.

7. Menaria P., and V. Muddana. 2019. Unusual case of life-threatening
gastro intestinal bleed from a splenic artery pseudoaneurysm: case
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Medicine 2019:8528906.

8. Syed S. M., Moradian S., Ahmed M., Ahmed U., Shaheen S., and V.
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9. Cho S. B., Park S. E., Lee C. M., et al. 2018. Splenic artery
pseudoaneurysm with splenic infarction induced by a benign gastric
ulcer: a case report. Medicine (Baltimore) 97:e11589.

10. Haivas C. D., and S. Kessler. 2016. Splenic artery pseudoaneurysm
causing brisk upper-GI bleed from gastric ulcer. Gastrointestinal
Endoscopy 84:540-541

11. Hoehn M. R., Hansraj N. Z., Pasley A. M., et al. 2019. Resuscitative
endovascular balloon occlusion of the aorta for non-traumatic
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12. Matsumura Y., Matsumoto J., Idoguchi K., et al. 2018. Non-traumatic
hemorrhage is controlled with REBOA in acute phase then mortality
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13. Sano H., Tsurukiri J., Hoshiai A., Oomura T., Tanaka Y., and S.
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14. Brenner M., Inaba K., Aiolfi A., et al. 2018. Resuscitative
endovascular balloon occlusion of the aorta and resuscitative
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15. Hoareau G. L., Tibbits E. M., Beyer C. A., et al. 2019.
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\textbf{Figure legends}

\textbf{Figure 1.} Time course of blood pressure, heart rate, and
laboratory data trends in a 72-year-old woman with melena. RBC, red
blood cell; FFP, fresh-frozen plasma; CT, computed tomography; REBOA,
resuscitative endovascular balloon occlusion of the aorta; ED, emergency
department; Lac, lactate; Hb, hemoglobin; BE, base excess; Plt,
platelet; Fib, fibrinogen

\textbf{Figure 2.} Contrast-enhanced computed tomography findings for a
72-year-old woman with melena. Free air is observed in the abdominal
cavity (A). The red arrows indicate a large amount of extravasated fluid
in the gastric lumen (B, C)

\textbf{Figure 3.} Abdominal surgery findings for a 72-year-old woman
with melena. A defect with a diameter of 30 mm is seen on the posterior
wall of the stomach. The white arrows indicate the perforated ulcer
within the posterior wall of the stomach (A, B). The yellow arrow
indicates the suture site of the splenic artery aneurysm (B)\selectlanguage{english}
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