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True PDA aneurysms may be associated with coeliac axis stenosis or occlusion.
Ruptured true PDA aneurysms may result in fatal haemorrhage.
Concomitant median arcuate ligament compression is a reversible cause of coeliac axis stenosis.
Ligament release can be performed as a staged elective procedure following emergency treatment.
Pancreaticoduodenal artery (PDA) aneurysms represent a small portion of rare visceral artery aneurysms. Rupture of these aneurysms results in fatal haemorrhage in up to 50% of cases, necessitating prompt endovascular or open intervention. As highlighted by a recent retrospective review, median arcuate ligament (MAL) release is an important part of management when these aneurysms are diagnosed in conjunction with median arcuate ligament compression (MALC). Two cases of successful urgent management of a ruptured inferior pancreatoduodenal artery aneurysm with staged MAL release are reported.
A 65 year old male presented with a ruptured PDA aneurysm in the context of MALC. The patient was treated by emergency transcatheter arterial embolisation (TAE). Staged laparoscopic MAL release required open conversion and stenting one month after rupture. A 73 year old male presented to the same institution with a ruptured PDA aneurysm, again in the context of MALC. This patient was similarly managed by emergency TAE and later had an uncomplicated laparoscopic MAL release. On table mesenteric angiography confirmed successful release. Both patients have since recovered without any recurrence of bleeding or new aneurysm formation.
Ruptured true PDA aneurysms, while uncommon, may be managed successfully using urgent endovascular techniques. Concomitant coeliac axis stenosis due to MALC requires secondary treatment and can be managed effectively using a staged approach following the urgent presentation.
Pancreaticoduodenal artery (PDA) aneurysms represent a rare subtype of visceral arterial aneurysms. True PDA aneurysms arise from altered blood flow and pressure within pancreaticoduodenal arcades secondary to a proximal coeliac axis (CA) stenosis or occlusion.
The management of two patients with ruptured inferior pancreaticoduodenal artery aneurysms and MALC using a two staged approach is presented.
A 65 year old male presented to the emergency department of a tertiary institution with acute onset epigastric pain. His history was significant for paroxysmal atrial fibrillation (AF) on apixaban, a pacemaker, and hypercholesterolaemia. On examination, he was haemodynamically stable with tenderness maximal over his epigastrium and right upper quadrant. His pathology demonstrated a haemoglobin level of 12.9 g/dL, with normal leucocyte count, liver function, and lipase. An abdominal computed tomography angiography (CTA) scan demonstrated an aneurysm in the anterior inferior pancreaticoduodenal artery (IPDA) measuring 9 mm craniocaudally with associated peri-pancreatic haemorrhage at the uncinate process.
Urgent endovascular treatment using a retrograde right femoral 6F (Brite-tip) sheath was performed. A diagnostic mesenteric digital subtraction angiogram (DSA) via a C2 catheter confirmed the presence of the aneurysm, but without active bleeding. Also noted was hypertrophy of the gastroduodenal artery, a CA stenosis, and reversal of flow through the pancreaticoduodenal arcade via the superior mesenteric artery (SMA) (Fig. 1A). Selective anterior IPDA coil embolisation from the SMA approach was performed via a 2.4 French (Progreat-Terumo) microcatheter within a C2 catheter using two 3 × 15 mm ruby coils (Penumbra). Despite some coil prolapse into the origin of the posterior IPDA, there was no residual filling of the aneurysm and all other arcades remained patent (Fig. 1B).
Post-operatively, an autoimmune screen, vasculitis studies, and infectious serology were normal. At one month, a staged laparoscopic MAL release was performed to allow time for the retroperitoneal haematoma to resolve. Despite adequate laparoscopic visibility and multidisciplinary consensus of visual release, post-operative CTA showed a residual 75% CA stenosis (Fig. 1C). Coeliac trunk stenting was initially attempted, but due to poor stent expansion from the residual MAL fibre an open release was necessary. Once the remaining band was released the stent was re-angioplastied (Fig. 1D). Anticoagulation was restarted on discharge at day 7. Follow up imaging at 12 months revealed a widely patent CA stent and no further PDA aneurysm formation.
A 73 year old male presented with a two hour history of acute upper abdominal pain on a background of AF with apixaban use. His history was significant for previous coronary stenting and dyslipidaemia. On examination, the patient was hypotensive to 97/60 mmHg and had a pulse rate of 75 beats/min. He had mild tenderness in the epigastrium without peritonism. His pathology demonstrated a haemoglobin level of 9.8 g/dL and normal leucocyte count, liver function, and lipase. Abdominal CTA identified a ruptured aneurysm arising from a branch of the anterior IPDA close to its origin, as well as a proximal CA stenosis (Fig. 2C).
Urgent coil embolisation of the bleeding branch of the IPDA was performed, accessed by a 6 French sheath in the right femoral artery (Fig. 2A). A C2 catheter was used for SMA cannulation, and this platform allowed five 2 × 4 cm ruby coils to be deployed via the 2.4F (Progreat-Terumo) microcatheter (Fig. 2B). The whole IPDA remained patent following selective branch coiling. The patient had an uncomplicated recovery and was discharged on day 6.
Three months after discharge, laparoscopic MAL release in a hybrid operating theatre with completion mesenteric angiography was undertaken. A second preparation and draping was required following laparoscopic port closure under the same anaesthetic. Owing to the experience with the first patient, intra-operative assessment of release was performed simultaneously to facilitate immediate open conversion if required. An on table mesenteric angiogram via a right femoral 6 French sheath and C2 catheter demonstrated restored antegrade CA flow with less than 30% residual CA stenosis. Therefore, open surgery was not required. At the patient's request, a post-operative mesenteric duplex ultrasound was performed instead of CTA, which confirmed antegrade flow and a widely patent CA at three months (Fig. 2D).
PDA aneurysms characterise 2% of reported visceral arterial aneurysms, with diagnosis occurring at a mean age of 60 years.
The first stage of management should focus on the PDA aneurysm. Treatment approaches for ruptured PDA aneurysms with MALC vary from transcatheter arterial embolisation (TAE) with or without coeliac artery stenting to open aneurysmorrhaphy with or without concomitant MAL release.
Illuminati et al. reviewed 57 patients with an unruptured PDA aneurysm with MALC. There was no PDA recurrence or recanalisation in those managed by open MALC treatment. Recanalisation however did occur in 11% following endovascular treatment and was correlated with CA re-stenosis.
Stenting should be reserved for intrinsic causes of CA stenosis (atherosclerosis) or following extrinsic MAL release when antegrade flow is not achieved in the arcade. Antegrade flow can be confirmed via either palpation, intra-operative ultrasound, intra-operative angiography, or on post-operative duplex.
The first of the patients had noticeable improvement in the palpable pulse at open release, while the second demonstrated restoration of antegrade flow on DSA compared with the emergency presentation images.
Staged MALC release was performed one to three months following the PDA aneurysm rupture to facilitate resolution of the retroperitoneal haematoma, which could impair operative MAL techniques.
True PDA aneurysms are rare, frequently fatal if ruptured, and can be associated with MALC as a cause of CA stenosis. In this case report, the two patients with ruptured PDA aneurysm and MALC were successfully managed with TAE as first line treatment to arrest haemorrhage, followed by a staged extrinsic release of the MALC. Treatment of underlying MALC is required for the effective correction of altered haemodynamics in the PDA arcade. When adequate collateral circulation allows, a staged MALC release offers a period of recovery from acute rupture and can improve operative success.
Conflict of interest
Inferior pancreaticoduodenal artery aneurysms in association with celiac stenosis/occlusion.
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