Materials and Methods:
Case presentation: Hospital database was searched for adult patients who had a discharge diagnosis of IAA at Wuhan Union Hospital over the past 10 years based on the international classification of diseases codes. The clinical notes of each potential case were reviewed, and patients younger than 14 years old or with other cardiac malformations were excluded. We also conducted a comprehensive literature search regarding IAA. Wuhan Union Hospital institutional review board approved this study.
3 patients were identified who were diagnosed with IAA at our institution. They were admitted to our department with the chief complaint of hypertension and later diagnosed with IAA using computated tomography angiography(CTA) or transcatheter angiography. Besides hypertension, they have no other symptoms. Two patients declined surgical treatment and received conservative therapy including anti-hypertensive treatment. The third patient received an extra-anatomic bypass surgery from ascending aorta to descending aorta. His post-surgical blood pressure remained high and was treated with irbesartan and hydrochlorothiazide.
Case 1:A 43-year-old man was admitted to our department with the complaint of high blood pressure in 2020. Physical examination showed regular heart beat with blood pressure of 179/102 mmHg in the right arm, 170/100 mmHg in the left arm, 135/80 mmHg in the right limb and 125/70 mmHg in the left limb, and there was a systolic murmur around the sternum. Electrocardiogram showed sinus rhythm. Echocardiography showed possible coarctation of aorta without other cardiac malformations. Then he was referred to the radiology department to further evaluate a possible aortic abnormality. CTA showed an interruption of the aortic arch distal to the origin of the left subclavian artery(Type A, Fig1 A and B) and profuse collateral circulation. The blood biochemistry laboratory results showed normal renal and liver function. There were no signs or symptoms of ischemia of splanchnic organs. Since the patient had no symptoms other than hypertension, we recommended anti-hypertensive management. After taking valsartan amlodipine and felodipine, his daily blood pressure in the left arm was stabilized at about 140/90 mmHg.
Case 2:Another 43-year-old man was admitted to our department for further treatment after he was diagnosed with IAA in another hospital in 2017. His physical examination showed regular heart beat with blood pressure of 132/96 mmHg in the right arm, 130/92 mmHg in the left arm, 110/80 mmHg in the right limb and 108/70 mmHg in the left limb, and there was a systolic murmur around the sternum. Before his referral, he had had high blood pressure for one year with the highest systolic pressure in the left arm up to 180 mmHg. Before he came to our department, he had already been diagnosed with IAA by another hospital and had been taking anti-hypertensive drugs for several weeks. His electrocardiogram showed in sinus rhythm with incomplete right bundle branch block. His echocardiography indicated possible coarctation of aorta with widened STJ (sinotubular junction). CTA revealed a coarctation or interruption of descending part of aortic arch with ascending aortic aneurysm and abundant collateral circulation. To determine whether it was a coarctation or an interruption, we performed an aortic angiography via right femoral artery and right radial artery, confirming that it was an interruption of the aortic arch distal to the origin of the left subclavian artery (Type A) with profuse collateral circulation(Fig1 C and D). His blood biochemistry laboratory results showed normal renal and liver function, without signs of ischemia of splanchnic organs. Similar to case 1, we recommended anti-hypertensives with perindopril, and the patient’s daily blood pressure in the left arm has stabilized at about 130/80 mmHg.
Case 3:A 28-year-old man was admitted to our department with the complaint of high blood pressure in 2017. Physical examination showed irregular heart beat with blood pressure of 174/110 mmHg in the right arm, 180/108 mmHg in the left arm, 130/82 mmHg in the right limb and 135/84 mmHg in the left limb, and there was a systolic murmur around the sternum. Electrocardiogram showed in sinus arrhythmia. Before he came to our hospital he had had echocardiography and CTA. His echocardiography showed coarctation of the descending aorta without other cardiac malformations, and CTA showed coarctation of the descending aorta with abundant collateral circulation. Initially, we attempted to place a stent via aortography. However, the aortography via right femoral artery and right radial artery showed an interruption of the aortic arch distal to the origin of the left subclavian artery (Type A) with abundant collateral circulation(Fig1 E and F ). The blood biochemistry results showed normal renal and liver function without signs of ischemia of splanchnic organs. Since the patient had no symptoms other than high blood pressure, as case 1 and 2, we recommended anti-hypertensives. However,given this patient’s young age and concerning regarding possible future IAA-related complications such as heart failure, renal and liver damage, etc., the patient and his family requested a surgery to correct the aortic deformities. We performed a surgical reconstruction to restore the continuity of aortic arch using extra-anatomic bypass from ascending aorta to descending aorta with 16mm artificial blood vessel. The surgery went well, but the patient’s systolic pressure in the right arm remained at approximately 170 mmHg. He received anti-hypertensives and accomplished satisfactory blood pressure (about 140/90 mmHg in the left arm), which remained adequate, 2 years after the surgery. And the patient was comatose for 2 weeks after the surgery. Since then he had been with right limb weakness and suffered from epilepsy. Significant diminished CPET performance and decreased communication ability accompanied. He could only walk slowly for about 50 meters and have a simple chat with others.