Unique Case of Neurogenic Thoracic Outlet Syndrome with Arterial Compression in Patient with Bilateral Cervical Ribs and Osteochondroma of the Ribs
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Background: Neurogenic thoracic outlet syndrome typically presents with paresthesia, pain, and impaired strength in the neck, shoulder, and arm, and is typically a diagnosis of exclusion. This condition is caused by compression of the brachial plexus, typically by a bony or soft tissue anomaly present congenitally and influenced by repetitive motion or significant trauma. Treatment typically involves removal of the first rib and anterior scalene to decompress the thoracic outlet and relieve stress to the brachial plexus if the patient has failed conservative treatment with physical therapy and lifestyle modifications. Case Presentation: We present a case of neurogenic thoracic outlet syndrome with arterial compression treated surgically via a transaxillary first rib and cervical rib resection in a patient with bilateral cervical ribs and osteochondromas of the ribs.Keywords:
Cervical rib
Thoracic outlet
Thoracic outlet syndrome (TOS) is the constellations of symptoms produced when the neurovascular structures that traverse the thoracic outlet are compressed. The symptoms are generally due to the compression of the subclavian artery and vein, lower trunk of the brachial plexus as they pass through the thoracic outlet. Diagnosing thoracic outlet syndrome can be difficult because the symptoms and their severity can vary greatly among people. We present a case of 30 years female presenting with arm and scapular pain radiating to hand along with restricted muscle movements; subsequently diagnosed with bilateral thoracic outlet syndrome on CT angiography and confirmed on multiphasic scan.
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Cervical rib
Neurovascular bundle
Subclavian vein
Brachial artery
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Neurovascular bundle
Subclavian vein
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Axillary artery
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Narrowing of the thoracic outlet may give rises to symptoms in the arm from compression of the nerves, the subclavian artery, and the subclavian vein. Anatomic studies show that nerve compression may affect fibers from T-1, C-8, and not infrequently C-7, providing an explanation for the diffuse nature of the nerve symptoms in the arm. Relief from this compression at the thoracic outlet is readily accomplished by transaxillary resection of the first rib together with any associated cervical rib, fibrous cervical rib analogues, or other fibrous bands encroaching on the neurovascular structures. A study of 40 patients with the syndrome who were treated in this way and followed up after an interval of three to ten years shows that those with nerve and vein symptoms can expect a good result but the results of treatment in those with arterial symptoms is less satisfactory.
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Abstract: Surgical treatment for thoracic outlet syndrome involves resection of the first rib (or a cervical rib) to resolve compression in the costoclavicular space. Depending on the structure involved (i.e. brachial plexus, subclavian artery, subclavian vein) it may be necessary to perform adjunctive vascular reconstruction. Three approaches for first rib resection are described. Transaxillary first rib resection is the most commonly performed approach. The supraclavicular approach may be preferred for arterial thoracic outlet syndrome. For vein reconstruction in venous thoracic outlet syndrome the infraclavicular approach is often more appropriate. Clinical success is obtained in the majority of patients, with slightly more variable results for the neurogenic thoracic outlet syndrome. Severe complications are rare.
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The costoclavicular compression syndrome and the cervical rib syndrome can cause arterial complications of the subclavian artery: mural thrombosis, occlusion and aneurysma. These complications can result in peripheral emboli or cerebrovascular insufficiency. Such sequelae can be avoided by timely surgical treatment of these compression syndromes. The therapy of choice is transaxillary rib resection. The same axillary approach can also be used for directly reconstructing the subclavian artery.
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Cervical rib is a well-documented congenital anomaly, and thoracic outlet syndrome (TOS) with vascular compression is extremely rare in childhood. The subclavian artery, subclavian vein, brachial plexus, or a combination of these can be affected in this syndrome. We present a case of TOS in an eight-year-old boy and describe the rare finding of subclavian artery compression with post-stenotic dilatation and severe brachial plexus compression symptoms. The patient underwent right transaxillary resection of the first thoracic and cervical rib. The presentation is unique and may be elusive.
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INTRODUCTION :
Thoracic outlet syndrome (TOS) describes a spectrum of symptoms and signs related to the passage of key anatomical structures through a narrow aperture on their way to the distal upper extremity. TOS results from compression of the neurovascular bundle in thoracic outlet area and the three components of the bundle in the thoracic outlet area are the brachial plexus, subclavian vein, and subclavian artery (SCA). Thus, there are three types of TOS, depending on which structure is compressed: neurogenic (nTOS), venous (vTOS), and arterial (aTOS).
The commonest form of TOS is neurogenic type. The arterial TOS forms less than 1% of cases, but the morbidity and mortality is severe that warrants immediate attention and management. The common etiology for aTOS is the presence of bony abnormalities. Cervical rib is found in approximately 0.5% of the general population and it contributes to the compressive neurovascular symptoms of the thoracic outlet. The symptoms of aTOS are caused by emboli arising from subclavian artery pathology, like intimal damage, stenosis, poststenotic dilatation or aneurysm.
The commonest etiology is presence of cervical rib. Other causes include anomalous first rib, post-traumatic callous formation in the clavicle, bony prominence or tumours in the clavicle, and rarely congenital fibrous band. The syndrome is caused by the anatomical narrowing caused by the cervical rib or anomalous first rib eliminating the space under the subclavian artery. This external compression results in repeated trauma in the intima of subclavian
artery, resulting in subclavian stenosis, thrombosis, aneurysm formation with mural thrombus and distal embolisation.
AIMS AND OBJECTIVES :
The aim of this study was to review our operative experience and to assess the symptomatic outcome of patients with arterial thoracic outlet syndrome who underwent decompression of the thoracic outlet.
1. To study the patient factors and their symptoms,
2. To analyze the type and site(s) of artery lesion,
3. To analyze the causative compressive agents,
4. To analyze the methods of surgical exposure and methods of decompression,
5. To study the techniques for repairing arterial lesions at the thoracic outlet,
6. To analyze the post operative outcome.
CONCLUSIONS :
Arterial complications due to compression at thoracic outlet are uncommon, comprising only less than one percent. But, it can result in significant morbidity and long term disability. Early recognition and surgical decompression provides favourable outcome in majority of patients.
Two unanticipated results in this study are (1) higher incidence in males and (2) left side cervical rib predominance. Arterial thoracic outlet syndrome is usually associated with bony component, and as cervical ribs are more common in females, the assumption is that females are more commonly associated with aTOS. This may be a wrong notion and in aTOS it may be that it is equal in both sexes. And also, this syndrome is the result of two factors added together: one is the anatomical narrowing and another is continuous trauma, causing changes in the muscle type. So, males being prone to repeated trauma, they are at higher risk in developing aTOS. Study at a larger scale is needed to confirm this.
Left sided predominance in this study cannot be considered significant, considering the size of the study.
In most patients, presence of cervical rib does not mean aTOS. In early stages of arterial compression, patient is usually asymptomatic, and the disease progresses silently. Only when, there is a distal embolism causing ischemic changes, this condition is diagnosed. If patients could be diagnosed before arterial changes occur, even at the stage of minimal post stenotic dilatation, just by decompressing thoracic outlet without any arterial repair, changes can be reversed. Mild symptoms, especially unilateral Reynaud syndrome needs further investigation to rule out thoracic outlet arterial compression.
Supraclavicular approach to tackle both bony component and arterial lesion is the best option in aTOS. Removal of the compressing bony component only without first rib removal is not affected the subjective and objective improvements in our patients. Resection of first rib is not necessary in our view. The results in our patients confirm the effectiveness of only removing the agent of compression.
The choice of arterial repair depends on the condition of the artery. No arterial intervention in simple dilatation and resection and anastomosis when there is aneurismal dilatation. This approach has minimal complications and maximum benefit. Distal embolectomy should be considered in all cases of acute limb ischemia, depending on the duration of occlusion and condition of the distal run-off vessels and collaterals.
In conclusion, this study confirms that when thromboembolic complications are present, surgery is indicated in all aTOS cases. However earlier diagnosis before thromboembolism should be the goal. We also conclude that supraclavicular approach and cervical rib excision without first rib excision is an effective procedure.
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Abstract The surgical approach to vascular complications of the thoracic outlet syndrome remains controversial. When present, removal of a cervical rib alone has produced disappointing results. Our experience of 29 consecutive first rib excisions over a 5-year period is presented. Of 20 cases with uncomplicated subclavian artery compression 19 were cured, and of six cases with aneurysm or thrombosis five were improved. Of 12 cases with neurological symptoms nine were cured and two were improved. It is suggested that first rib excision is the essential primary treatment for patients with arterial symptoms due to thoracic outlet syndrome.
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