Discussion
Retroperitoneal bleeding is a rare, but potentially life-threatening
cause of abdominal pain. It is caused by a complication of femoral
artery catheterization or other imaging procedures, pelvic/lumber
trauma, oral coagulants, and aortic dissection.5Visceral aneurysm and dissection are the rare causes of retroperitoneal
bleeding. The IPDA contributes to only 2% of visceral
aneurysms.2 The mortality rate for ruptured IPDA
aneurysms is approximately 30%, requiring early identification and
treatment.4 Bleeding from the IPDA dissection has also
been reported.2 The IPDA aneurysms are associated with
celiac artery stenosis, atherosclerosis, infection, trauma, and
pancreatitis. They were related to celiac artery stenosis in 60%-75%
of cases.6 Celiac artery stenosis can be detected by
contrast-enhanced CT or Doppler ultrasonography. Since celiac artery
stenosis is found in approximately 7% of asymptomatic patients, the
presence of typical symptoms, such as chronic abdominal pain (especially
postprandial), nausea/vomiting, and mild weight loss, is needed for the
diagnosis of CACS.7,8 The mechanism by which the
celiac artery stenosis leads to the development of IPDA aneurysms is
associated with the formation of arterial pancreatic
arcade.9,10. The superior and inferior portions of the
anterior and posterior pancreaticoduodenal arteries form a pancreatic
arcade between the celiac artery and SMA. Celiac artery stenosis reduces
blood flow to the pancreatic arcades, which increases blood flow and
pressure in the IPDA from SMA, promoting the IPDA aneurysm or dissection
formation.
In this case, the patient presented with severe abdominal pain, which
led to the diagnosis of retroperitoneal hemorrhage. The patient had a
celiac artery stenosis detected in CT scan, which we believe, was the
cause of the PIPDA dissection. In addition, the patient had chronic,
non-specific abdominal pain, which may be a symptom of CACS. Typical
symptoms of CACS are chronic abdominal pain (especially postprandial),
nausea/vomiting, and mild weight loss. In the present case, nausea,
vomiting, and weight loss, which are typical symptoms of CACS, were not
observed. The other possibility is that atherosclerosis may have caused
the PIPDA dissection, not related to CACS, since dyslipidemia is an
underlying disease in this case.
The patient had elevated liver enzyme levels, which resolved with
treatment; however, the relationship between elevated liver enzyme
levels and CACS or IPDA aneurysm/dissection has not been reported in
previous literature. Our hypothesis is that blood flow to the liver
originating from the celiac artery was reduced due to celiac artery
stenosis prior to this episode, and as a result, blood flow from the SMA
region to the liver was supplemented via the pancreatic cascade. In this
patient, we believe that the PIPDA dissection reduced the total blood
flow to the liver from the SMA region, causing transient liver ischemia.
The first-line treatment for ruptured IPDA aneurysms and dissections is
endovascular embolization.11 Metallic coils are
increasingly used to occlude the ruptured aneurysm or dissection.
Regarding embolization, coils are placed at both the afferent and
efferent arteries close to the bleeding site because of the presence of
collateral arteries around the pancreas.12 In some
cases, surgery may be an option due to the complexity of the pancreatic
arcade and the difficulty of catheterization due to celiac artery
stenosis. Surgical treatment is also indicated when embolization is
unsuccessful. Performing surgical ligament release in CACS patients with
ruptured aneurysms is not yet established.3,13Abdominal artery release surgery is expected to improve the symptoms of
CACS and prevent subsequent aneurysms, but its long-term effects are
unknown. In this case, after discussing the benefits and risks of
surgery with the patient, we chose to observe the patient without
performing surgery.
In conclusion, we report the successful endovascular treatment of
ruptured PIPDA dissection associated with CACS. Celiac artery stenosis
can cause retroperitoneal hemorrhage secondary to visceral aneurysm and
dissection. Clinicians may consider CACS as the cause of visceral
aneurysm and dissection in patients with chronic abdominal symptoms.
Conflicts of Interest: The authors declare no conflicts of
interest.
Acknowledgments: None.
Funding: None.
Author contributions : KN: drafted the manuscript. HA, TI, JK,
and HK: critically revised the manuscript. All authors read and approved
the final manuscript.
Ethical approval: Applicable.
Informed consent: Informed consent was obtained from the
patient.