Pancreaticoduodenal artery (PDA) aneurysms are rare, accounting for only 2% of all visceral artery aneurysms [1]. Approximately 50% of reported PDA aneurysms are associated with celiac artery stenosis or occlusion [2,3]. The overall rupture rate is around 40%, with a mortality rate of up to 50% [1,2,4].

Regarding treatment, an endovascular approach is preferred and considered more effective than surgical options [5,6]. The literature includes case reports on the endovascular treatment of PDA aneurysms associated with celiac artery stenosis or occlusion. However, this case report highlights the endovascular treatment and outcomes of a much rarer condition: multiple PDA aneurysms. Written informed consent was obtained from the patient for the publication of this case report.

A 54-year-old male presented to our general surgery clinic with long-standing abdominal pain (exacerbated after meals). Intravenous and oral contrast-enhanced abdominal computed tomography (CT) was performed, revealing critical stenosis of the celiac artery caused by median arcuate ligament syndrome (Fig. 1A), along with three sequential saccular aneurysms in the PDA (Fig. 1B-D). The diameters of the aneurysms (from proximal to distal) were 34, 15, and 21 mm. The patient had no comorbidities other than hypertension and no personal or family history of diseases potentially associated with visceral artery aneurysms. The patient was evaluated by the rheumatology department for connective tissue disorders and vasculitis; however, the results were negative. Blood test results and physical examination were unremarkable. The patient was referred to the interventional radiology department for endovascular therapy.

Figure 1. Computed tomography scan of a 54-year-old male showed celiac artery stenosis (arrow) (A) and pancreaticoduodenal artery saccular aneurysms measuring 34 mm (B), 15 mm (C), and 21 mm (D) from proximal to distal.

Under ultrasound guidance, a 5-F introducer sheath was placed in the right femoral artery under local anesthesia using the Seldinger technique. During abdominal digital subtraction angiography (DSA), the celiac artery origin was not visualized. The PDA and associated aneurysms demonstrated retrograde filling from the superior mesenteric artery (SMA), with collateral branches supplying the celiac trunk, main hepatic artery, and splenic artery. Subsequently, a 5-F Cobra catheter (Imager^TM II, Boston Scientific) was placed in the SMA (Fig. 2A). Accessing the most proximal (34-mm) aneurysm sac was achieved via a 2.7-F microcatheter (Progreat^TM, Terumo) (Fig. 2B). Sixteen detachable fiber coils (Interlock^TM, Boston Scientific) of different sizes were used for embolization, starting from the proximal aneurysm and proceeding distally (Fig. 2C, D). Post-procedural DSA confirmed the occlusion of the aneurysms and collateral filling of the celiac trunk and its branches via the SMA (Fig. 2D). Follow-up Doppler ultrasonography (DUS) performed 1 day later confirmed the aneurysm occlusion. The patient was discharged with no additional complaints and was scheduled for follow-up at 1 week and 1 month.

Figure 2. The angiogram obtained from the superior mesenteric artery showed three consecutive saccular aneurysms in the pancreaticoduodenal artery (A), the microcatheter (arrow) placed in the proximal aneurysm (B), coil embolization of the aneurysms and pathways from proximal to distal (C), and the post-procedural angiogram showing closure of the aneurysms and retrograde filling of the hepatic (arrow) and splenic artery (dashed arrow) via collateral branches (D).

At the 1-week follow-up DUS, the aneurysms remained occluded. However, at the 1-month follow-up DUS and CT, the distal aneurysm had expanded, with a 20-mm patent lumen (Fig. 3A, B). The patient then underwent a second intervention. DSA from the SMA indicated that the previous coils in the distal aneurysm had been pushed peripherally, with partial thrombosis of the lumen (Fig. 3C). The aneurysm lumen was re-embolized using six coils (Fig. 3D).

Figure 3. Follow-up ultrasonography (A) and computed tomography (B) 1 month later showed that the distal aneurysm had reopened; the angiogram from the superior mesenteric artery showed that the coils were pushed peripherally (C), and the open lumen was closed with additional coils (D).

During the 10-month follow-up period, with monthly checks for the first 3 months and bimonthly thereafter, the aneurysms remained occluded, and the patient reported no further complaints.

PDA aneurysms should be treated regardless of size [7]. The association between celiac occlusive disease and PDA aneurysms is well-known. Case reports indicate that treatment of the celiac artery can lead to auto-occlusion of PDA aneurysms [2], likely due to restored antegrade blood flow through the celiac axis, which reduces hemodynamic stress on the PDA collateral network via the retrograde SMA blood flow; however, due to the high risk of rupture, embolization is preferred approach [5,8]. Moreover, treating the celiac artery reduces the risk of aneurysm recurrence.

In this case, the initial plan was to embolize the aneurysms after placing a stent in the celiac artery; however, access to the celiac artery was not possible. Consequently, the aneurysms were embolized in a retrograde manner from the SMA. Before embolization, collateral branches supplying the hepatic artery, separate from the PDA aneurysms, were confirmed. After embolization, increased blood flow through the collateral branches was observed (Fig. 2D).

The patient’s medical history revealed hypertension resistant to medical treatment, with a systolic blood pressure ranging between 160 and 180 mmHg. This condition was anticipated to contribute to recurrence after embolization; hence, the patient was advised to adhere to close follow-up. One month later, the distal aneurysm reopened and was occluded more tightly by utilizing additional coils. During the second procedure, placement of a covered stent in the SMA to close off the PDA orifice was considered but ultimately not performed due to concerns about the collateral branches supplying the hepatic artery from the patent PDA segment and the possibility of secondary complications related to stenting.

The patient postprandial abdominal pain resolved after the initial procedure and did not recur during follow-up. Despite the untreated celiac artery stenosis, the resolution of symptoms was attributed to the strengthening of SMA flow following the closure of the PDA aneurysms, which indirectly enhanced the flow in the SMA collateral branches associated with the celiac artery branches. When endovascular approaches fail, bypass or decompression surgery of the celiac artery should be considered. These options were explained to the patient; however, the patient declined surgical intervention.

Treatment of the celiac artery is recommended for management of PDA aneurysms associated with celiac artery stenosis or occlusion. If left untreated, continued retrograde blood flow to the collateral branches of the SMA increases the risk of recurrence [7]. In this case, the celiac artery could not be treated, and together with uncontrolled hypertension, this risk was further increased, leading to reopening of the previously embolized aneurysm sac within just 1 month. In such cases, we recommend tighter coil packing for the first aneurysm sac exposed to blood flow and closer follow-up owing to the risk of recurrence.

None.

The author has nothing to disclose.