Cervical aortic arch with aneurysm formation and an anomalous right subclavian artery and left vertebral artery:A case report

lNTRODUCTlON

A cervical aortic arch (CAA) is a rare congenital anomaly of vascular development and refers to a highriding aortic arch (AA) that often extends above the level of the clavicle[1-4]. This paper reports a case of a unique CAA with aneurysm formation and an anomalous right subclavian artery (RSCA) and left vertebral artery (LVA).

I guess our love lasts because it is comfortable. No, the sky is not bluer: it s just a familiar hue15. We don t feel particularly young: we ve experienced too much that has contributed to our growth and wisdom, taking its toll16 on our bodies, and created our memories.

CASE PRESENTATlON

Chief complaints

A 29-year-old woman presented repeated bilateral purulent nasal discharge for 3 mo.

History of present illness

The symptoms started 3 mo prior to presentation, with dizziness and headache.

History of past illness

The patient underwent a cesarean birth at a local hospital 4 years prior.

Personal and family history

The patient had no family history that was related to the present illness.

The final diagnosis of the present case was left CAA with aneurysm formation and an anomalous RSCA and LVA.

Physical examination

The total hospital stay was 30 d. The patient’s recovery was smooth, and no abnormality was found at the 2-mo follow-up after discharge.

Laboratory examinations

Imaging examinations

A computed tomography scan at another hospital showed bilateral maxillary sinusitis and ethmoiditis and bilateral inferior turbinate hypertrophy. The patient was diagnosed with chronic sinusitis and chronic rhinitis at admission. A preoperative noncontrast chest computed tomography scan showed a high-riding, tortuous AA extending to the mid-upper level of the first thoracic vertebra with local cystic dilatation, and a few calcified plaques were identified in the descending aorta (DA). A further computed tomography angiography examination showed that the ascending aorta was located to the right of the spine. The brachiocephalic trunk (BCT), left common carotid artery (LCCA), LVA (slender), and left subclavian artery (LSCA) sequentially branched off of the aorta from the proximal end to the distal end of the AA. The BCT bifurcated into the right common carotid artery and the RSCA. The proximal end of the RSCA was tortuous and dilated (approximately 1.5 cm in diameter). The AA showed tumor-like local expansion, with a maximum diameter of approximately 4.0 cm. The long and tortuous AA distal to the aneurysm was located to the left of the spine, extended upward, surpassed the level of the clavicle, and reached the mid-upper level of the first thoracic vertebra. Then, it extended downward tortuously along the left side of the spine to become the DA (Figure 1).

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FlNAL DlAGNOSlS

And as she drank thus, and leaned right over the water, the rag containing the three drops of blood fell from her bosom12 and floated down the stream, and she in her anxiety never even noticed her loss.21 But the waiting-maid had observed it with delight, as she knew it gave her power over the bride, for in losing the drops of blood the Princess had become weak and powerless.22 When she wished to get on her horse Falada again, the waiting- maid called out: I mean to ride Falada: you must mount my beast ; and this too she had to submit to. Then the waiting-maid commanded her harshly to take off her royal robes, and to put on her common ones,23 and finally she made her swear by heaven not to say a word about the matter24 when they reached the palace; and if she hadn t taken this oath she would have been killed on the spot. But Falada observed everything, and laid it all to heart.

TREATMENT

After consultation with the department of cardiac macrovascular surgery, the patient was diagnosed with left CAA plus aneurysm formation and an anomalous RSCA and LVA and was transferred to that department. The patient underwent AA aneurysm resection and artificial blood vessel replacement under general anesthesia and cardiopulmonary bypass (CPB) on July 10, 2021. The left CAA and the aneurysm (with a maximum diameter of approximately 4.0 cm) that had formed between the LCCA and the LSCA were observed during the operation. During the intraoperative period, a total of 2 units of type O suspended red blood cells, 10 units of cryoprecipitated antihemophilic factors, and 500 mL of fresh frozen plasma were transfused, and vasopressor drugs, including 8 μg deoxyepinephrine, 128 μg adrenaline, and 38478 μg dopamine, were infused with an infusion pump control to manage blood pressure. The total CPB time was 106 min, the ascending aorta was blocked for 44 min, and circulation was stopped for 15 min. After successful surgery, the patient received intensive care, anti-infection treatment, respiratory and circulatory function maintenance, and microcirculation improvement therapy for 44 h and 50 min.

OUTCOME AND FOLLOW-UP

Physical examination showed a slightly congested mucosa of the bilateral nasal cavity, a slightly enlarged inferior turbinate, a hypertrophic middle turbinate, and a small amount of purulent nasal discharge in both middle nasal passages and posterior nostrils.

DlSCUSSlON

CAA was first reported by Reid[5] in 1914. CAA is very rare, with an incidence of less than 1/10000[1,3,6], although it is relatively more common in young women[6]. In the 35 cases of CAA reported by Zhong[6], the average patient age was 34.2 years, and females accounted for 65.7% of the sample.

That night, my girlfriend cried heavily in front of me. I asked her whether it was her father’s stupid deeds that made her feel humiliated8. But she said that she felt guilty for her dad and that though the rest of the whole world betrayed her, her old father would still back her up and help her get the justice she deserved, just as when she was young and the neighboring boy grabbed her ball, her father would get it back for her. However, this changed world was no longer the stage for her old father and his deeds became funnily obsolete9（） . No one managed to see the real and ever-lasting love to his daughter behind his seemingly rude behaviors.

Most CAA cases are asymptomatic and are usually detected incidentally[4,6,12]. A few cases manifest as a pulsatile neck mass[4,6] or dyspnea and dysphagia due to compression of the trachea and esophagus[6,12,13]. Some CAA cases are combined with congenital cardiovascular anomalies, such as patent ductus arteriosus, tetralogy of Fallot, ventricular septal defect, pulmonary atresia[1,14], and various anatomical variations of the AA branches[2,3,13]. Approximately 20% of CAA cases are complicated with aneurysm formation[1,9,11], which is more common in women[8,9]. The aneurysm may be due to abnormal hemodynamics and arterial wall pressure changes caused by embryonic development, connective tissue anomalies, and a long and tortuous aorta[11,14]. Haughton[15] classified CAA into five types. Type A:The CAA and DA are contralateral, one side of the common carotid artery is missing, and the independent internal and external carotid arteries directly branch from the AA. Type B:The CAA and DA are contralateral, and two common carotid arteries branch from the AA. Type C:The CAA and DA are contralateral, and a bicarotid trunk appears. Type D:The CAA and DA are ipsilateral, and the sequence of brachiocephalic branching is normal. Type E:This type refers to a right CAA with an ipsilateral DA. Haughton[15] type D is the most common type of CAA that is combined with aneurysms[3,16], most aneurysms are located in the AA between the LCCA and the LSCA[6,8,16], but a few are located in the DA or simultaneously involve the AA and DA[16]. Shayan[11] reported eight cases of CAA with aneurysms (type D aneurysm in seven cases and type A aneurysm in one case), including six cases in which the aneurysm was located between the LCCA and the LSCA and two cases in which the aneurysm was located between the LSCA and the DA.

The patient described in this study had a left CAA with an ipsilateral DA. Her long and tortuous AA straddled the left side of the spine, extended upward above the clavicle, and then extended downward tortuously along the left side of the spine. The concurrent AA aneurysm was located between the LCCA and the ostium of the LSCA, and the origins of the two blood vessels were far apart. At the same time, the CAA was combined with the proximal dilatation and tortuosity of the RSCA and the anomalous origin of the LVA, reflecting the uniqueness of her manifestations.

CONCLUSlON

A CAA is a rare congenital anomaly of vascular development. Some CAA cases are combined with congenital cardiovascular anomalies, and approximately 20% of CAA cases are complicated with aneurysm formation. The present unique case of CAA with aneurysm formation and an anomalous RSCA and LVA enriches existing CAA data.

We thank Mr. Yang F and Ms. Ma LJ for their contributions to computed tomography image reconstruction.

There we sat, side by side, watching the sun go down behind the old Texaco service station across the busy street. A street I had been allowed to cross by myself. Grampy stretched his long legs down over two stairs. I dangled22 mine, a bit closer to the first step this time, I m sure.

Grandpa filled his hours with television. He watched the news and game shows while Grandma went about her day. They made a pact—he was not to leave his chair or his bed without her assistance.

Laboratory tests showed that the serum uric acid (428.9 μmol/L), fibrin (4.9 g/L), erythrocyte sedimentation rate (30 mm/h) and ultrasensitive thyroid stimulating hormone (5.1 μIU/mL) values were slightly elevated, the myoglobin (＜ 21 ng/mL) level was slightly decreased, and the complete blood count, electrolyte profiles and liver function were normal.

Informed written consent was obtained from the patient for publication of this report and any accompanying images.

Wu YΚ wrote the paper; Mao Q, Zhou MT, Liu N, Yu X, Peng JC, Tao YY and Gong XQ contributed to the literature search and manuscript preparation; Yang L and Zhang XM designed the study; all of the authors read and approved the final manuscript.

The etiology of CAA is unclear but is possibly related to abnormal embryonic development of the AA (persistence of the 2or 3arch or incomplete descension of the normally developed fourth arch, resulting in incomplete entry of the AA into the thoracic cavity)[7-10]. Chromosome 22q11 deletion is evident in some CAA cases[3,4,11].

The authors declare that they have no conflict of interest.

The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).

This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BYNC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is noncommercial. See:https://creativecommons.org/Licenses/by-nc/4.0/

Then the hermit looked out and saw how near they were, and he said to the youth, Do you see that? The youth shrieked20 and cried, Ah, good father, it is a fleet of ships, and they are chasing us, and in a few moments they will be upon us

China

Yao-Kun Wu 0000-0002-1959-5599; Qi Mao 0000-0003-1301-069X; Mao-Ting Zhou 0000-0003-2034-2086; Ning Liu 0000-0003-3587-5440; Xi Yu 0000-0002-5785-5144; Jin-Cheng Peng 0000-0002-9686-3913; Yun-Yun Tao 0000-0001-5081-3315; Xue-Qin Gong 0000-0001-8389-8765; Lin Yang 0000-0001-8746-9255; Xiao-Ming Zhang 0000-0001-5327-8506.

The Chinese Society of Interventional Oncology.

Guo XR

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But the clever flute-player was quite a match for the little man in cunning, and said: All right, you needn t be afraid, you shall get your beard back before we part; but you must allow my bride and me to accompany you a bit on your homeward way

Guo XR