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Radiol Case Rep
Radiol Case Rep
Radiology Case Reports
1930-0433
Elsevier

S1930-0433(24)00701-5
10.1016/j.radcr.2024.07.116
Case Report
Case report: Dysphagia aortica
Abdelrahman Amro aaa.360@outlook.com

Elgassim Mohamed Melgassim@hamad.qa
⁎
Ahmed Amina Aahmed100@hamad.qa

Al-Hatemi Mubarak MAlhatemi@hamad.qa

Abdalla Elhassan Mohamed eabdalla4@hamad.qa

Zaki Hany A. hanyzaki1976@gmail.com

Department of Medical Education, HMC, Doha, Qatar
⁎ Corresponding author. Melgassim@hamad.qa
27 8 2024
11 2024
27 8 2024
19 11 52805283
26 4 2024
18 7 2024
21 7 2024
© 2024 The Authors. Published by Elsevier Inc. on behalf of University of Washington.
2024

https://creativecommons.org/licenses/by-nc-nd/4.0/ This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).
Dysphagia, characterized by abnormal swallowing, presents as oropharyngeal or esophageal dysphagia. Dysphagia aortica, a rare manifestation, results from external aortic compression, leading to swallowing difficulties. Limited literature exists on this condition. We report a 22-year-old male with a complex surgical history, including aortic repairs, who presented with dysphagia and chest pain. Extensive evaluations ruled out other causes. Imaging revealed esophageal compression by an aortic graft. Endoscopy confirmed extrinsic compression. A barium swallow study was unremarkable. A diagnosis of dysphagia aortica was made, and conservative treatment was initiated. Dysphagia aortica remains a rare but noteworthy cause of dysphagia with this case highlighting the importance of considering vascular compression in patients with previous history of aortic surgery. Increased clinical awareness is essential for timely diagnosis and tailored treatment strategies. Further research is needed to establish guidelines for managing this condition, given its diverse causes.

Keywords

Aortic dysphagia
Oropharyngeal dysphagia
Esophageal dysphagia
Dysphagia
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pmcIntroduction

Dysphagia is defined as difficulties in swallowing, resulting in an abnormal delay in the passage of liquids or solids through the esophagus. This condition encompasses 2 primary types: oropharyngeal dysphagia, often arise from neuromuscular pathology, and esophageal dysphagia, which can be attributed to mechanical obstruction or functional disorders [1].

Dysphagia aortica can manifest in congenital, acquired, or iatrogenic forms. It's a condition where the esophagus faces external compression from the aorta, leading to swallowing difficulties [2]. Dysphagia aortica has been rarely reported in the literature.

We are reporting the case of a 22-year-old male with a notable history, including the repair of a coarctation of the aorta and hypoplastic aortic arch, as well as previous bypass surgery and percutaneous transluminal balloon angioplasty and a recent history of TEVAR (thoracic endovascular aortic repair). He received a diagnosis of dysphagia aortica after excluding all other possible causes of dysphagia. With this report, we aim to shed light on a unique cause of dysphagia, which is dysphagia aortica, that can be overlooked in patients with a complex surgical history of the aorta.

Case presentation

A 22-year-old male, with a past surgical history involving the repair of a coarctation of the aorta and hypoplastic aortic arch since birth, underwent bypass surgery as an infant and later had percutaneous transluminal balloon angioplasty. He presented to the hospital with complaints of difficulty swallowing and chest pain for 4 days. The patient denied any history of coughing, paroxysmal nocturnal dyspnea, or orthopnea. Apart from lumbar spine scoliosis, a review of other systems was unremarkable. Additionally, he had no history of smoking, alcohol consumption, recent dietary changes, weight fluctuations, or changes in exercise habits.

Just 2 months prior to this presentation, the patient underwent TEVAR for graft placement.

Upon arrival at the emergency department, his vital signs were stable, with a blood pressure of 128/82 mmHg, temperature of 36.8°C, heart rate of 70 beats per minute, respiratory rate of 19 breaths per minute, and oxygen saturation (SpO2) of 100% on room air. Physical examination of the chest and abdomen revealed no remarkable findings. Result of chest X-ray showed spinal scoliosis with right aortic arch (Fig. 1).Fig. 1 AP view of chest XR showing: Right sided aortic arch (red arrow) levoscoliosis.

Fig 1:

Electrocardiography (ECG) assessment showed unremarkable findings. Given the patient's history of his previous cardiac issue and complaints of chest pain, the cardiology team requested to evaluate the patient for any possible cardiac chambers enlargement with echocardiogram and it was found to be unremarkable. A blood sample was taken to the lab for investigation and the results of basic metabolic panel, complete blood count and Troponin T enzyme levels were found to be within normal limits. The radiologist on call request to assess the aorta for any hidden aortic pathology or aortic leak through Gated computed tomography (CT) thorax scan. The Gated CT scan showed middle esophagus pulled towards the right side by the aorta (Fig. 2).Fig. 2 Coronal and sagittal views of CT Aorta showing: Middle oesophagus pulled towards the right side by the aorta (yellow arrows).

Fig 2:

In light of the results of gated CT thorax, consultations were obtained from the vascular team, gastroenterology team, and upper GI surgery team. Their recommendation was to admit the patient for further investigation with upper GI endoscopy. The patient was admitted and endoscopic evaluation performed by the gastroenterology team showed evidence of extrinsic compression in the mid-distal esophagus, with no apparent changes in the esophageal mucosa (Fig. 2).

To further exclude structural abnormalities, a barium swallow study was conducted and it showed evidence of focal narrowing in the middle esophagus (Fig. 3).Fig. 3 Lateral and AP views of barium swallow showing: Focal narrowing of the mid oesophagus (oragne arrows).

Fig 3:

Based on these findings, a diagnosis of dysphagia aortica was established, and the patient was treated conservatively as his symptoms weren't severe and discharged with a follow-up appointment scheduled in the upper GI surgery clinic after an uneventful hospitalization course.

Discussion

Given the structural relationship between the esophagus and the aorta, it's plausible that the aorta could exert pressure on the esophagus [1]. Back in 1939, Pape coined the term “dysphagia aortica” to describe difficulties in swallowing arising from external compression caused by an enlarged, twisted, or aneurysmal aorta, primarily as a result of age-related degeneration [3].

Dysphagia aortica has been linked with old age, women of shorter stature, individuals with hypertension, those with kyphoscoliosis, and those with left ventricular enlargement [4].

Due to the rarity of the condition, only a limited number of cases are documented.

A previous systematic review highlighted the association of dysphagia aortica and patients with a history of aortic conditions (like thoracic aortic aneurysm or aortoesophageal fistula) or a history of vascular intervention or gastrointestinal intervention [4].

A previous study described a case of dysphagia aortica which was immediately developed after a stent grafting procedure in a female patient with thoracoabdominal aneurysm [5].

Another case also described a 55 year old male with dysphagia aortica 1 month after he had a TEVAR for aortic aneurysm [6].

In this case, our patient is a 22-year-old with spinal levoscoliosis and a surgical history that includes the repair of coarctation of the aorta due to hypoplastic left heart syndrome. Additionally, there is a recent history of TEVAR graft placement.

Given the rarity of this condition, the diagnostic approach must be comprehensive, ruling out other potential causes of dysphagia before confirming dysphagia aortica. A patient's history of aortic aneurysms or prior aortic procedures can be valuable in supporting the diagnosis. The assessment process for dysphagia aortica involves several crucial steps, including radiological imaging, manometric studies to assess motility issues in the esophagus, and upper GI endoscopy. A CT scan can be very helpful in assessing the aortic lumen and the wall of the aorta [1]. Manometric studies are also valuable for identifying esophageal motility disorders, while barium swallow studies and endoscopic examinations can help identify intraluminal causes [7].

Our patient's initial evaluation included transesophageal echocardiography due to concurrent chest pain complaints. Subsequent assessment with a gated CT thorax revealed middle esophagus pulled towards the right side by the aorta. This led to a planned endoscopic evaluation, which confirmed the presence of extrinsic compression in the mid-distal esophagus. Additionally, a barium swallow study, requested by the radiology team to rule out structural causes of dysphagia, revealed focal narrowing in the middle esophagus.

Regarding treatment, there is no established standard due to the rarity of this condition and its diverse causes. The chosen approach depends largely on the severity of symptoms and the individual patient's circumstances. Both conservative and interventional treatments are options [8].

Interestingly and paradoxically, TEVAR procedures can serve as both a potential risk factor for dysphagia aortica and a treatment option, depending on the underlying etiology.

A previous study described a patient with aortic dysphagia as a result of thoracoabdominal aneurysm who was treated with the TEVAR procedure [9].

Our patient received conservative treatment during hospitalization due to the nonserious nature of his symptoms. He was subsequently discharged with plans for follow-up care at the upper GI surgery clinic.

Conclusion

This case report highlights dysphagia aortica as an uncommon cause of swallowing difficulties. It underscores the importance of considering vascular compression as a potential factor in patients with significant aortic surgeries. Beyond its rarity, this case emphasizes the necessity of a multidisciplinary approach in treating such patients.

Increasing awareness among healthcare providers about this condition is essential for prompt diagnosis and tailored treatment strategies. Further research and collaborative efforts are needed to establish standardized guidelines for managing dysphagia aortica, given its diverse etiologies.

Availability of data and materials

All data underlying the results are available as part of the article and no additional source data are required.

Patient consent

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.

Appendix Supplementary materials

Image, application 1

Competing Interests: The authors declare that they have no known competing financial interests or personal relationships that could have appeared to influence the work reported in this paper.

Acknowledgments: This research work is funded by Qatar National Library.

Supplementary material associated with this article can be found, in the online version, at doi:10.1016/j.radcr.2024.07.116.
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