# Renal artery reimplantation for secondary hypertension due to diaphragmatic crus compression: a case report and literature review.

Youcef Lounes, Lucien Chassin-Trubert, Kheira Hireche, Léonore Freycon-Tardy, Mariama Akodad, Ludovic Canaud, Pierre Alric

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Youcef Lounes, Lucien Chassin-Trubert, Kheira Hireche, Léonore Freycon-Tardy, Mariama Akodad, et al.. Renal artery reimplantation for secondary hypertension due to diaphragmatic crus compression: a case report and literature review.. Annals of Vascular Surgery - Brief Reports and Innovations, 2022, pp.100056. 10.1016/j.avsurg.2022.100056. hal-03605991

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# Renal artery reimplantation for secondary hypertension due to diaphragmatic crus compression: A case report and literature review.

<figure><img src="image_2.png" /></figure>

Youcef Lounes<sup>a,b,*</sup>, Lucien Chassin-Trubert<sup>a</sup>, Kheira Hireche<sup>a</sup>, Léonore Freycon-Tardy<sup>a</sup>,
Mariama Akodad<sup>b,c</sup>, Ludovic Canaud<sup>a,b</sup>, Pierre Alric<sup>a,b</sup>

## Abbreviations

<table><tr><td>CT</td><td>Computed Tomography</td></tr><tr><td>LRA</td><td>Left Renal Artery</td></tr><tr><td>RA</td><td>renal artery</td></tr><tr><td>RRA</td><td>Right Renal Artery</td></tr><tr><td>SMA</td><td>superior mesenteric artery</td></tr></table>

## Introduction

Renal artery compression due to a diaphragmatic crus entrapment is the second cause of renal stenosis after fibrodysplasia in the young population. This rare condition represents only 1.4% of secondary hypertension in that population (1). High origin and prominent diaphragmatic crus are often retrieved, leading to a compression of the renal artery (RA) at its origin defined as the median arcuate syndrome. The origin of this disease may be explained by abnormalities of cephalad migration of the kidneys during the fetus's life (2). Although rare, this compression leads to a focal stenosis causing severe hypertension. Typical clinical presentation is incidental finding of severe and resistant hypertension in a young patient with no other cardiovascular risk factors. Different treatment modalities have been described in the literature as open, laparoscopic liberation or endovascular stenting of the RA, but no recommendations exist for this rare pathology. Taking all these considerations together, we report a case of direct re-implantation of the renal artery and discuss the review of the literature.

## Case report

A 31-year-old male was admitted for dysphagia and shortness of breath. Asthma and eosinophilic esophagitis were diagnosed. During clinical examination, his systolic blood pressure was 210 mmHg without related symptoms. Consequently, he underwent several explorations including doppler ultrasound showing a flow acceleration over 300 m/sec in the proximal part of the left RA without direct visualization of the stenosis. Resistance index was 0.5 for the left side and 0.65 for the right; the left kidney measured 10.5 cm and the right 12 cm. The computed

tomography (CT)-scan confirmed the diagnosis of critical stenosis with the objectivation of a diaphragmatic crus entrapment and a high origin of the left RA, between the coeliac trunk and the superior mesenteric artery (SMA) (Fig. 1). Despite a progressive escalation of antihypertensive medications over a period of 3 years, the patient remained hypertensive with a four drug combination including Ramipril, Metoprolol, Hydrochlorothiazide and Amlodipine. The renal function was normal, and he had no other cardiovascular risk factor. After a multidisciplinary meeting, an angiogram and potential left RA angioplasty was decided. The angiogram did not allow to highlight clear stenosis, despite respiratory maneuvers. Invasive measurement of arterial pressure difference between the aorta and the renal artery was not deemed necessary as the significant stenosis was previously confirmed on CT and doppler. An angioplasty with a 6 mm non-compliant balloon in the proximal part of the LRA discovered the stenosis with rapid recoils precluding a stent placement (video 1). To ensure long-term permeability and avoid a stent placement, we decided, after evaluation of the benefits and risks of all different treatments, to perform a surgical decompression of the LRA. A median supra-umbilical laparotomy was performed and the aorta, the left renal vein, as well as the superior mesenteric were dissected. The section of the diaphragmatic crus that compressed the left RA was realized. The stenosis of the LRA was critical despite the application of local papaverine (Fig. 2A). Due to the excess length of the artery, we performed a resection of the stenosis and direct reimplantation of the left RA in the aorta in the same site of its origin with a 6-0 prolene (Fig. 2B). A 12 min supra-mesenteric aortic clamping was performed to realize the anastomosis with recovery of a normal pulsatile left RA. The outcomes were uneventful with rapid ambulation of the patient and hospital discharge at day 5. The blood pressure normalized immediately after the surgery with the discontinuation of all antihypertensive medications. The post-operative doppler ultrasound showed a perfect permeability of the left RA with no residual stenosis and a resistance index of 0.6. Renal function was normal, we observed an increase of the left kidney size (11.5 cm) at 3-month follow-up, and treatment by aspirin was prescribed for a 6-month duration. Clinical, US-doppler and CT scan follow-up showed no restenosis or hypertension recurrence up to 3-year follow-up (Fig. 3).

<footnote><sup>a</sup>Dept of Vascular and Thoracic Surgery, Univ Montpellier, CHU Montpellier, Montpellier, France</footnote>

<footnote><sup>b</sup>PhyMedExp, Univ Montpellier, CNRS, INSERM, CHU Montpellier, Montpellier, France. Montpellier, France</footnote>

<footnote><sup>c</sup>Department of Cardiology, Arnaud de Villeneuve Hospital, Montpellier, France</footnote>

<footnote>* Corresponding author.
E-mail address: youceflounes@gmail.com (Y. Lounes).</footnote>
<table><caption>**Table 1**

Literature review of all renal artery entrapments treated surgically. This table describes demographics, type of treatments, success, and follow-up.</caption><thead><tr><th>Author</th><th>Year</th><th>age</th><th>sex</th><th>Laterality</th><th>First Treatment</th><th>Second reconstruction</th><th>resolution</th><th>Follow-up (month)</th></tr></thead><tbody><tr><td>D'Abreu<sup>3</sup></td><td>1962</td><td>50</td><td>M</td><td>Bilateral</td><td>Open liberation</td><td>—</td><td>Yes</td><td>9</td></tr><tr><td>D'Abreu<sup>3</sup></td><td>1962</td><td>32</td><td>M</td><td>Left</td><td>Open liberation</td><td>—</td><td>Yes</td><td>4</td></tr><tr><td>Lampe<sup>5</sup></td><td>1965</td><td>28</td><td>M</td><td>Right</td><td>Graft Bypass</td><td>—</td><td>Yes</td><td>2</td></tr><tr><td>Villanueva<sup>6</sup></td><td>1972</td><td>—</td><td>—</td><td>—</td><td>Open liberation</td><td>—</td><td>—</td><td>—</td></tr><tr><td>Silver<sup>2</sup></td><td>1976</td><td>24</td><td>M</td><td>Left</td><td>Open liberation</td><td>—</td><td>Yes</td><td>—</td></tr><tr><td>Silver<sup>2</sup></td><td>1976</td><td>4</td><td>M</td><td>Left</td><td>Vein patch</td><td>—</td><td>Yes</td><td>—</td></tr><tr><td>Silver<sup>2</sup></td><td>1976</td><td>33</td><td>F</td><td>Right</td><td>Open liberation</td><td>—</td><td>No</td><td>—</td></tr><tr><td>Spies<sup>7</sup></td><td>1987</td><td>20</td><td>M</td><td>Left</td><td>Vein Patch</td><td>—</td><td>Yes</td><td>11</td></tr><tr><td>Clement<sup>8</sup></td><td>1990</td><td>26</td><td>F</td><td>Left</td><td>Open liberation</td><td>—</td><td>Yes</td><td>—</td></tr><tr><td>Vahdat<sup>9</sup></td><td>1991</td><td>23</td><td>—</td><td>Right</td><td>Saphenous bypass</td><td>Open Liberation</td><td>Yes</td><td>—</td></tr><tr><td>Bacourt<sup>10</sup></td><td>1992</td><td>21</td><td>M</td><td>Left</td><td>Open liberation</td><td>—</td><td>Yes</td><td>48</td></tr><tr><td>Kopecky<sup>11</sup></td><td>1997</td><td>50</td><td>F</td><td>Left</td><td>Open liberation</td><td>—</td><td>Yes</td><td>6</td></tr><tr><td>Baguet<sup>12</sup></td><td>2003</td><td>74</td><td>M</td><td>Left</td><td>RA Stenting</td><td>RA Reimplantation</td><td>Yes</td><td>2</td></tr><tr><td>Thony<sup>4</sup></td><td>2005</td><td>68</td><td>F</td><td>Left</td><td>RA stenting</td><td>—</td><td>No</td><td>6</td></tr><tr><td>Thony<sup>4</sup></td><td>2005</td><td>—</td><td>—</td><td>—</td><td>RA stenting</td><td>—</td><td>No</td><td>6</td></tr><tr><td>Deglise<sup>13</sup></td><td>2007</td><td>39</td><td>M</td><td>Bilateral</td><td>RA stenting (Left)</td><td>Graft bypass (Left)<br>RA reimplantation (Right)</td><td>Yes<br>Yes</td><td>6</td></tr><tr><td>Gaebel<sup>14</sup></td><td>2009</td><td>19</td><td>F</td><td>Left</td><td>RA angioplasty</td><td>Open liberation</td><td>Yes</td><td>—</td></tr><tr><td>Boufi<sup>15</sup></td><td>2010</td><td>56</td><td>F</td><td>Left</td><td>RA stenting</td><td>Graft Bypass</td><td>Yes</td><td>18</td></tr><tr><td>Bünger<sup>16</sup></td><td>2010</td><td>26</td><td>M</td><td>Left</td><td>Laparoscopic liberation</td><td>—</td><td>Yes</td><td>—</td></tr><tr><td>Song<sup>17</sup></td><td>2011</td><td>23</td><td>M</td><td>Bilateral</td><td>Auto transplantation (Left)<br>Open liberation (Right)</td><td>—</td><td>Yes</td><td>18</td></tr><tr><td>Rafiq<sup>18</sup></td><td>2018</td><td>21</td><td>M</td><td>Right</td><td>RA stenting</td><td>—</td><td>Yes</td><td>0,5</td></tr><tr><td>Mirza<sup>19</sup></td><td>2020</td><td>20</td><td>M</td><td>Left</td><td>Open liberation</td><td>—</td><td>Yes</td><td>17</td></tr><tr><td>Mirza<sup>19</sup></td><td>2020</td><td>65</td><td>F</td><td>Bilateral</td><td>Laparoscopic liberation</td><td>—</td><td>Yes</td><td>—</td></tr></tbody></table>

<figure><img src="image_3.png" /><figcaption>**Fig. 1.** focal stenosis due to a compression of the diaphragmatic crus.</figcaption></figure>

<figure><img src="image_4.png" /><figcaption>**Fig. 2.** intra operative photo showing the persistent stenosis and the post stenosis dilatation of the left renal artery (A). Direct reimplantation of the left renal artery (B). Ao: aorte, LRV: left renal Vein, LRA: left renal artery, St: stenosis, An: anastomosis.</figcaption></figure>

<figure><img src="image_5.png" /><figcaption>**Fig. 3.** Post-operative CT showing the resolution of the left renal stenosis.</figcaption></figure>

Discussion

In 5–10% of cases, hypertension is related to a renal stenosis (1). However, depending on the age, the large majority of those stenosis are

due to atherosclerosis or fibrodysplasia. Noilan et al. found that 1.4% of secondary hypertension may be related to an external compression of the RA by the diaphragmatic crus. This pathology has been described for the first time in 1962 by d'Abreu and Strickland (3) in London, United Kingdom. Since then, 24 cases of surgical treatments have been published in the literature. Thony et al. described a series of 15 cases with a mean age of 65 yo and a majority of female (n = 9) (4). The clinical presentation was mainly isolated hypertension (n = 7) but hypertension associated with renal insufficiency or abdominal aortic aneurysm or superior mesenteric artery aneurysm without hypertension were also observed. The right RA was involved in most cases (n = 11) with no bilateral RA involvement cases. CT-scan or Magnetic Resonance Imaging appear as the key for the diagnosis but may overestimate the stenosis as the images are taken during blocked inspiration (4). Conversely, angiogram may underestimate the stenosis regarding its proximity to the aortic wall and the potential difficulty to achieve a favorable angulation. Overall, doppler or pressure measurements are required to confirm the stenosis and the functional impact.

A literature review of surgical management is summarized in Table 1 (5–19).

Over the treated and collected cases, the mean age was younger than in the series published by Thony et al. with a male predominance (14 males and 7 females) (Table 1). Moreover, in this series, the left RA was involved in the large majority of cases (n = 18/22), and bilateral stenoses in 4 cases (3,13,17,19). Multiple visceral arteries stenosis were associated in 7 patients (9-13,16,17), with median accurate syndrome with celiac trunk stenosis, a compression of the SMA or all visceral arteries. The clinical presentation was systematically a resistant hypertension, in the majority without related symptoms.

Several surgical modalities have been described in the literature (Table 1). Endovascular treatments may be a seducing option but may not provide favorable long-term results. Complications as persistent stenosis (4), secondary restenosis and stent fracture have been described (4,13,15). Balloon-expandable stent may be associated with better results (4,18), although the resolution of the hypertension was observed in only one case with only 2 weeks of follow-up.

Liberation of the external compression is, in the authors opinion, a fundamental requirement for a suitable outcome. Indeed, in the majority of cases (n = 14/20; 70%), a surgical liberation of the diaphragmatic crus
was sufficient to obtain a complete resolution of the symptomatology. In rare cases (2), the resolution of high blood pressure was incomplete, despite the absence of residual stenosis. The authors explained that the failure to normalize the blood pressure was due to another etiology of hypertension. A stenosis can still be present after the liberation of the RA (30%). In those cases, different surgical reconstruction techniques have been described including vein patch (n = 2) (2,7) or auto-transplantation (17). In this case, direct re-implantation was successfully performed with no complications and satisfying midterm follow-up. Only few cases of direct RA re-implantation for this etiology were reported; Baguet *et al.* had to resect partially the origin of the RA to retrieve a stent priorly placed (12), and Deglise *et al.* realized a re-implantation for a persistent stenosis of the right RA (13). A supra-mesenteric aortic clamping was necessary to realize the anastomosis but for a very short time without the need of nephroplegia. We believe that doing one anastomosis with a short clamping time may offer the best outcomes for the kidney. Veinous patch have also been described in the literature with favorable outcomes but extended access to the renal artery is required.

In our case, laparotomy was preferred as re-implantation or bypass to the RA was considered less challenging thought a laparotomy than a retroperitoneal access. Indeed, the prediction of length and kinking of the bypass is difficult to appreciate after releasing the retractors. Laparoscopic liberation can offer a minimally invasive solution with safe and promising results (19). However, we observe a decreasing number of vascular surgeons that are trained for laparoscopic procedures. Moreover, this procedure would need to be associated to another surgical procedure in case of persistent renal stenosis. A RA stenting might offer a complementary minimally invasive solution, but no description of these procedures association was made in the literature. Besides, a surgical repair may offer a more durable patency than stenting in this young population (10,15) without the need of a prolonged anti-platelet treatment.

Sparse alternative treatment modalities as botulinum toxin injection into the diaphragmatic crus have been described with temporary effective results (20). This treatment may be a solution to delay the surgical treatment in pregnancy or other conditions.

## Conclusion

Renal stenosis caused by a diaphragmatic entrapment is a rare cause of renovascular hypertension. Surgical liberation with or without arterial reconstruction seems to offer the better results. We present a case of such pathology successfully treated by surgical decompression and direct re-implantation of the renal artery with satisfying short and long-term outcomes.

## Supplementary Material

**Video 1 :** Attempt of angioplasty with a 5 mm balloon, it is clearly seen that the stenosis is still present with a visible print during deflation of the balloon.

## Declaration of Competing Interest

Canaud reports a relationship with Medtronic Inc that includes: consulting or advisory.

## Supplementary materials

Supplementary material associated with this article can be found, in the online version, at doi:10.1016/j.avsurg.2022.100056.

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