| Abstract|| |
Isolation of the left brachiocephalic artery (ILBA) is an extremely rare anomaly of aortic arch with diverse manifestations in the neurologic system, heart, and left upper arm. This anomaly is defined as the absence of connection of the left brachiocephalic artery (LBA) to aortic arch and connection of LBA to pulmonary artery (PA) through a patent arterial duct (PAD). However, this definition is not inclusive of all cases. Not only are there inconsistencies in the definition and terminology of this aortic arch anomaly but also there is no classification for this anomaly despite its heterogeneous nature in terms of anatomy, clinical presentation and prognosis. We performed a 52-year comprehensive literature review in the period between 1966 and 2018. Our inclusion criteria were any manuscript that included a case report or case series, with confirmed diagnosis of ILBA. All quantitative data were analyzed using descriptive analysis by SPSS version 21 (IBM SPSS Statistics, USA). Results were presented as mean ± standard deviation and median. Based on the presence or absence of connection of LBA to PA and the number of sources of steal from the LBA, we classified ILBA into three types: single-steal type with no connection of LBA to PA and single source of blood flow steal from LBA through the left subclavian artery (LSCA), double-steal type with connection of LBA to PA through PAD and two sources of steal through LSCA and arterial duct (AD), and triple-steal type with bilateral PADs and therefore, three sources of blood flow steal from LBA including the LSCA and the double ADs. Patients with single-steal type have the best prognosis and present latest with symptoms of cerebrovascular insufficiency or left arm claudication. The oldest reported patient was 69 years of age with symptoms of dizziness and near syncope. No death was reported in these patients. Double-steal type is the most common type and is often associated with genetic syndromes and/or extracardiac anomalies. Triple-steal type is the rarest type with the earliest presentation and worst prognosis. The oldest reported patient was 60 days of age. All reported cases had cardiac symptoms, pulmonary overcirculation, pulmonary hypertension, and fatal outcome.
Keywords: Anatomic-clinical-prognostic classifi cation, isolation of left brachiocephalic artery, left subclavian steal syndrome
|How to cite this article:|
Malakan Rad E, Pouraliakbar HR. Isolation of the left brachiocephalic artery revisited: A 52-year literature review and introduction of a novel anatomic-clinical-prognostic classification. Ann Pediatr Card 2019;12:117-29
|How to cite this URL:|
Malakan Rad E, Pouraliakbar HR. Isolation of the left brachiocephalic artery revisited: A 52-year literature review and introduction of a novel anatomic-clinical-prognostic classification. Ann Pediatr Card [serial online] 2019 [cited 2019 May 22];12:117-29. Available from: http://www.annalspc.com/text.asp?2019/12/2/117/257402
| Introduction|| |
Isolation of the left brachiocephalic artery (ILBA) is an extremely rare anomaly of aortic arch with diverse manifestations in the neurologic system, heart, and left upper arm. This anomaly is defined as the absence of connection of the left brachiocephalic artery (LBA) to aortic arch and connection of LBA to pulmonary artery (PA) through an arterial duct (AD).,,,,, However, this definition is not inclusive of all cases, including our present case. We present an asymptomatic child with ILBA and without connection of LBA to PA. Since 1966 when Levine et al. reported the first case of ILBA as “atresia of left common carotid and left subclavian arteries,” there are inconsistencies in the definition and terminology of this aortic arch anomaly. There is no classification for this anomaly despite its heterogeneous nature in terms of anatomy, clinical presentation, and prognosis.
We performed a comprehensive literature review in the period between 1966 and 2018. Based on the anatomy of our case and the 52-year literature review of ILBA, from 1966 to April 2018, we propose a comprehensive, novel, and practical anatomic-clinical-prognostic classification of ILBA.
| Shortcomings of the Current Terminology and Definition and the Need of a Classification for Isolation of the Left Brachiocephalic Artery|| |
There are confusing inconsistencies not only in terminology but also in the definition of isolation of LBA in the literature. Different terminologies have been used for this anomaly including “anomalous origin of LBA from pulmonary artery,” “aplasia of left brachiocephalic trunk,” “isolation,” “anomalous origin of LBA from left pulmonary artery,” and “origin of LBA from right pulmonary artery.”,,,, The term “origin” from PA is not justified because the direction of blood flow is not from PA into the LBA. This hemodynamic point differentiates ILBA from interrupted aortic arch Type C [Figure 1] and [Table 1].
|Figure 1: Aortic arch and cerebrovascular circulation in three different conditions: (b) normal, (b) isolation of the left brachiocephalic artery without connection to pulmonary artery, and (c) Type C interruption of aortic arch. For the sake of simplicity, all annotations are shown only in Figure 1b and are not repeated in Figure 1a. The white arrow in each of the figures shows the direction of blood flow into the left brachiocephalic artery|
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|Table 1: Clarifying the confusion on the source of perfusion of the left brachiocephalic artery and direction of blood flow between left brachiocephalic artery and pulmonary artery in four conditions: Normal, isolation of the left brachiocephalic artery (without and with connection to pulmonary artery), and type C interruption of aortic arch|
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The original case reported by Levine et al. was a 33-year-old woman with symptoms of dizziness and poor perfusion of the left arm without any connection of LBA to PA through a patent AD (PAD). However, later, some authors defined ILBA as origin of LBA from left PA through an AD. Review of literature and study of our case, who had no connection of LBA to PA, indicate the shortcoming of the current definition in inclusion of all cases of ILBA. Obviously, there is significant hemodynamic difference between patients with PAD and those with obliterated AD. Left to right shunt across the AD occurs only in the former.
Patients with ILBA have widely heterogeneous characteristics in terms of anatomy, clinical presentation, and outcome. These diversities indicate the heterogeneous nature of these patients and the need for an appropriate anatomic-clinical-prognostic classification system.
| Clinical Importance of Isolation of the Left Brachiocephalic Artery|| |
ILBA results in a heterogeneous spectrum of clinical manifestations expanding over three organ systems, which can be acronymed as three “H's.” Three H's include head (i.e., cerebral symptoms due to vertebral-basilar insufficiency), heart (cardiac manifestations secondary to pulmonary overcirculation and congestive heart failure), and hand (claudication and numbness of the left arm due to decreased flow to the left subclavian artery [LSCA]) [Figure 2]. The cerebral and upper extremity symptoms are due to “under-perfusion” of these organs whereas the cardiac symptoms are secondary to pulmonary “over-circulation” resulting from the left to right shunt that occurs from LBA into PA through the PAD or ducts.
|Figure 2: Schematic diagram showing “the anatomy and the number of steal phenomenon” and “the source of symptoms” in patients with isolation of the left brachiocephalic artery. Symptoms are related to head (manifested as dizziness and transient ischemic attacks due to cerebrovascular insufficiency), heart (manifested as congestive heart failure due to left to right shunt from left brachiocephalic artery to pulmonary artery), and hand (manifested as left arm claudication due to decreased flow to left subclavian artery)|
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Patients with ILBA are also at risk of subclavian artery steal phenomenon. Connection of ILBA to PA provides the setting for an additional left to right shunt by PA steal (double steal) with consequent cardiac manifestations secondary to pulmonary overcirculation. In patients with bilateral ADs, there is a potential for triple steal to occur from LBA, including LSCA steal and pulmonary arterial steal through the left and the right ADs.
| 52-Year Literature Review on Isolation Ofthe Left Brachiocephalic Artery: 1966–2018|| |
We performed a 52-year literature review of ILBA from 1966 to April 2018.
Our inclusion criteria were any manuscript that included a case report or case series, with confirmed diagnosis of ILBA. The diagnostic criteria were lack of connection of LBA to aorta, Retrograde late filling of LBA through the collateral arteries from circle of Willis with or without connection to PA through unilateral or bilateral patent arterial duct or ducts.
The electronic database of PubMed, Embase, Cochrane, Web of Science, Scopus, Ovid MedLine, Clinical Key, and Essential Science Indicators and electronic journals of ScienceDirect, Wiley Online Library, Springer, Proquest, BMJ, and SAGE were searched respectively. We also searched Google Scholar and Google. We used the search terms of “isolation,” “isolated,” “anomalous origin,” and “atresia” in combination with LBA or brachiocephalic artery, brachiocephalic artery and LBA without any limitation for age or language. We searched the references of each paper and included the relevant articles. We excluded cases with isolation of only left common carotid artery (LCCA) or only LSCA. We extracted the following twelve information about each case: age at presentation, main clinical presentation, presence of other associated congenital heart disease, presence of pulmonary hypertension, presence of extracardiac anomaly or syndromes, presence of weak or absent pulse of LCCA, smaller size, lower blood pressure (BP) and lower oxygen saturation of the left hand, smaller left cerebral hemisphere, type of treatment, and final outcome of the patient. All quantitative data were analyzed using descriptive analysis by SPSS version 21 (IBM SPSS Statistics, New york, USA). Results were presented as mean ± standard deviation and median. We found 30 cases that had the diagnostic criteria for inclusion in our study. After studying the full text of all papers, we found that case number 15 was a case of left aortic arch with isolated right brachiocephalic artery. Neither the title of this paper nor the full text clearly stated the sidedness of the brachiocephalic artery. Sidedness was shown in the figure of this case report.
A separate literature search performed to find further cases of the left aortic arch and isolated right brachiocephalic artery proved the uniqueness of this case.
We also included the information of an asymptomatic 3.5-year-old child with ventricular septal defect (VSD) and ILBA. She was referred to our clinic with a wrong diagnosis of VSD and interrupted right aortic arch. On physical examination, she had normal growth and development. First and second heart sounds were normal and a grade 4/6 holosystolic murmur was heard at the left sternal border. A bruit was heard on the cranium. The left carotid, left brachial, and left radial pulses were weaker than the right ones. The BP in the right and left arms were 111/77 mmHg (mean = 90 mmHg) and 87/60 mmHg (mean = 70 mmHg), respectively. Oxygen saturation was 98% in both hands. Both hands were of the same size. On cardiac catheterization and angiography, there was a right aortic arch with two arch branches including right common carotid artery and right subclavian artery with ILBA as seen by late and retrograde filling of the LBA with no connection to PA [Figure 3] and [Video 1]. The pulmonary arterial pressure was normal. Imaging of the brain and the heart by 384-slice computed tomographic angiography revealed normal size of both cerebral hemispheres, normal cerebral arteries and a long distance (21 mm) between the isolated LBA and the aortic arch [Figure 4]. The child was scheduled for surgical VSD closure. Considering the long distance between the LBA and the aortic arch, absence of symptoms of subclavian artery steal at this stage of life, and lack of consent of the parents, reimplantation of the isolated LBA was not planned at this age.
|Figure 3: (a) Aortogram in anteroposterior view indicates right aortic arch with two branches of RCCA (yellow arrow) and RSCA. (b) After several beats, LCCA is visualized by retrograde flow from the cerebral circulation from above (red arrows). RSCA is shown by yellow arrow. LCCA is smaller than RCCA. (c) The left subclavian artery appears late through retrograde flow from above which is shown by the red arrows. RCCA: Right common carotid artery, LCCA: Left common carotid artery, RSCA: Right subclavian artery|
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|Figure 4: Three-dimensional, volume -rendered image of computed tomographic angiography shows the right aortic arch with isolation of the left brachiocephalic artery. LCC artery is smaller than RCC artery. The distance between the isolated left brachiocephalic artery with the aortic arch is significant. RCC: Right common carotid; LCC: Left common carotid, RSCA: Right subclavian artery, LSCA: Left subclavian artery, AO: Aorta, PA: Pulmonary artery|
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The results of the 52-year literature review on cases with ILBA, including our case, are tabulated in [Table 2].
|Table 2: A 52-year literature review of complete isolation of brachiocephalic artery* (in order of the date from April 2018 back to 1966)|
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| Novel Anatomic-Clinical-Prognostic Classification of Isolation of the Left Brachiocephalic Artery|| |
Based on this literature review, we classified all the 31 cases based on the number of sources of steal from the brachiocephalic artery into three anatomic-clinical-prognostic types [Figure 5]. The basis for classification was the presence or absence of connection of LBA to PA and the number of sources of steal of flow from LBA. Case number 15 in [Table 2] was a unique case of 2-month-old infant with left aortic arch and isolated right brachiocephalic artery.
- Single-steal type with single source of steal from LBA through the LSCA and with no connection to PA
- Double-steal type with double sources of steal from LBA through LSCA and PAD
- Triple-steal type with triple sources of steal from LBA through LSCA and bilateral PAD.
Clinical and prognostic characteristics of the three types of isolation of the left brachiocephalic artery
Comparison of clinical and prognostic characteristics of these three types is summarized in [Table 3].
|Table 3: Clinical and prognostic characteristics of the three types of isolated left brachiocephalic artery*|
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In these patients with no connection of LBA to PA, the only source of steal from LBA is subclavian artery steal. Therefore, the least amount of steal occurs from the cerebral circulation. During infancy and early childhood when both the physical activities and the linguistic ability to express symptoms are limited, we do not expect overt complaints or symptoms of cerebrovascular insufficiency. The clinical and prognostic characteristics of this type are higher age at presentation, dominance of symptoms of cerebrovascular and left arm perfusion insufficiency, lack of association with genetic syndromes and extracardiac anomalies and universally good outcomes. Although atrial septal defect (ASD), VSD, pulmonary stenosis, and double-chamber right ventricle have been reported in these patients, the vast majority of these patients do not have congenital heart disease. Smaller left cerebral hemisphere, reported in other types, has not been reported in this type. There is no report of death in this type.,,,,,,,
LBA is connected to PA in these patients, either directly or through a left-sided PAD. This type is the most common type of ILBA. These patients present much earlier than patients with single-steal type. More than half of these patients have associated genetic syndromes, including Down syndrome and particularly DiGeorge syndrome, or other extracardiac anomalies. CHARGE association and polysplenia syndrome have been reported. Cardiac manifestations are the main clinical presentation and 76.4% of these patients have at least one congenital heart disease other than PAD. Primum and secundum ASD, cleft mitral valve VSD, complete atrio ventricular septal defect (VSD), cervical aortic arch, subaortic stenosis, coarctation, and bilateral superior caval vein have been reported. More than 90% of cases had symptoms of pulmonary overcirculation. The majority of reported patients had pulmonary hypertension. Two patients died in this group, one died at the age of 9 months and the other before 2 years of age, had Prader–Willi syndrome and DiGeorge syndrome, respectively. Evaluation for DiGeorge syndrome and other genetic syndromes is recommended in this group.,,,,,,,,,,,,,,,
These patients have bilateral PADs which set the stage for triple steal to occur from the LBA. This is the rarest type with the earliest presentation, the worst prognosis, and 100% mortality. Despite the three sources of steal from LBA, the cause of death is not attributable to the steal phenomenon. The main culprit for mortality in these patients was the associated complex congenital heart disease. All reported cases had pulmonary overcirculation and pulmonary hypertension. Left to right shunt across the two PADs contributed to pulmonary congestion and heart failure. However, the smaller left cerebral hemisphere, reported in one-fourth of these patients on autopsy, can be explained by the triple-steal phenomenon. All reported cases had pulmonary overcirculation and pulmonary hypertension. A more detailed description of the cardiac and extracardiac anomalies of the four patients with this type is indicated in [Table 4].,,,
|Table 4: Details of cardiac and extracardiac anomalies and other important characteristics of the four cases with triple-steal type of isolated left brachiocephalic artery|
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Advantages of the novel classification
This classification system is comprehensive and includes all the reported cases of ILBA. It provides practical information on anatomy, expected clinical manifestations and outcome. It also provides hints on the essential diagnostic workup.
| Three Simple and Helpful Diagnostic Clues for Early Recognition of the Tricky Diagnosis of Isolation of the Left Brachiocephalic Artery|| |
Look for the triad of “right aortic arch,” “two aortic branches,” and “weak pulse of the left arm”
The triad of “right aortic arch, two aortic arch branches and weak pulse of left upper arm” was present in almost 90% of all cases [Table 5].
|Table 5: The frequency of the triad of right aortic arch, two aortic arch branches, and weak or absent pulse of left upper limb in isolation of left brachiocephalic artery (total number of 30 cases*)|
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All reviewed cases had right aortic arch. All patients except two cases had two aortic arch branches. The case reported by Pauliukas et al. had right aortic arch with mirror-imaging branches (case number 14 with single-steal type in [Table 1]). Case number 11, a double-steal type, reported by Reeves et al. also had three aortic arch branches. It is noteworthy that there is only once case with left aortic arch and isolation of the right brachiocephalic artery. All other patients had right aortic arch with two aortic arch branches.
Absent or weak pulses of the left carotid, left brachial, and left radial artery are present in the vast majority of all types when pulses were examined. However, in about half of cases, bilateral examination of pulses was either not performed or not reported.
In all reported cases, except two, left hand pulse was either absent or lower than the right side. Of those 2 cases with equal pulses, one was due to origin of the right subclavian artery in the segment with severe coarctation. The pathophysiology of equal pulses in the other case who was a 56-year-old woman with systemic hypertension and history of multiple transient ischemic attacks affecting the right side of the body is not clear.
Therefore, simple palpation of the pulse of the left hand in a child with right aortic arch with two-aortic arch branches can be extremely helpful in early detection of this tricky and extremely rare anomaly.
Compare the blood pressure and the oxygen saturation of the left hand with the right hand
The BP in the left arm was lower than the right in patients with reported BP of both hands. In the 5-year-old girl reported by Delgado and Barturen, the slightly higher BP in the left arm was because of the long-segment coarctation involving the right subclavian artery.
Mild decreased oxygen saturation of the left arm in comparison to the right arm may occur. Oxygen saturation was not reported in 73.3% of cases. In 7 cases with measured oxygen saturation, decreased saturation was seen in 4. The higher left arm oxygen saturation, reported in the case of Miyaji et al., represents the unique case of isolation of the right brachiocephalic artery and left aortic arch. Thus, higher BP of the left arm in a patient with ILBA should raise the suspicion to associated lesions. As seen in [Table 1], differential size of the left upper arm is rare and is not expected to be present at a very early age.
Auscultate the cranium for cranial bruits in isolation of the left brachiocephalic artery
Bruits in the cranium and in the neck can be detected in all types. However, as in our case, auscultation of bruits does not necessarily indicate a serious abnormality in intracranial arteries and may simply reflect the collateral flow that supplies the left vertebral artery in retrograde.
In summary, a weak radial pulse in a child with right aortic arch and two arch branches, without any history that indicates other etiologies, should very strongly raise the suspicion to the presence of ILBA. Bilateral palpation of radial pulses is an often forgotten and extremely simple physical examination that directs us to the early diagnosis of this extremely rare and tricky diagnosis, with potentially diverse neurologic, cardiac, and left upper extremity manifestations. We propose a simple, novel, and practical anatomic-clinical-prognostic classification that categorizes these patients into three types. As we go from single-steal type to triple-steal type, the prognosis worsens and the age at presentation decreases. Complex congenital heart disease or mortality is not reported in patients with single-steal type. These patients often present in adulthood with symptoms of vertebral-basilar artery insufficiency or left arm claudication. About one-fourth of patients with the double-steal type have DiGeorge syndrome. Evaluation for microdeletion of 22q11 is recommended in these patients. Triple-steal type is the rarest type and has the earliest presentation with worst prognosis and 100% mortality. Of note, in this subset of patients, the mortality does not result from the steal phenomenon but from the associated complex congenital heart disease. All patients with triple-steal had extracardiac anomaly.
The authors deeply appreciate the kind permission of the parents of the patient who allowed us to report the cardiac images of their child.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
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Dr. Elaheh Malakan Rad
Department of Pediatrics, Children's Hospital Medical Center (Pediatric Center of Excellence), No. 62, Dr. Gharib's Street, End of Keshavarz Boulevard, Tehran 1419733151
Source of Support: None, Conflict of Interest: None
[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
[Table 1], [Table 2], [Table 3], [Table 4], [Table 5]