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takayasu 's arteritis ( ta ) is a progressive inflammatory vasculitis , potentially affecting all large and medium calibre arteries , leading to a varying degree of narrowing , occlusion or dilatation with specific predilection for young asian women .
this disease is also referred to as pulseless disease , aortic arch syndrome , young female arteritis , idiopathic aortitis , and martorell syndrome .
the major clinical finding is loss of palpable pulses in the upper limbs and neck .
the unsuspected ischaemia in vital regional vascular beds may be associated with high anaesthetic risks for these patients .
this case was particularly challenging because of the coexisting cushing 's syndrome ( cs ) , which required the anaesthesiologist to be geared up to solve the additional problems of volume overload , hypertension , glucose intolerance and hypokalaemic metabolic alkalosis .
a 15-year - old male patient presented to our hospital 6 months before with a history of headache , swelling over face and body and episodic numbness and tingling in both upper limbs and clumsiness of the right hand while working for last 6 months .
magnetic resonance angiogram revealed narrowing of distal thoracic aorta above diaphragm , dissection above diaphragm extending below up to bifurcation , occlusion of left renal artery with distal reformation , subtle narrowing of right renal artery and occlusion of right subclavian artery with reformation of the brachial artery . computed tomography ( ct ) angiography also supported the findings , and diagnosis of ta type v with left adrenal mass ( ?
patient underwent digital subtraction angiography , and a stent was placed in proximal left renal artery 4 months back .
since then patient was on clopidogrel along with antihypertensive medications . during the present visit ,
pre - operative blood pressure ( bp ) recorded on left upper limb was 150/92 mmhg and correlated well with lower limb recordings .
bp was non - recordable on right arm as his right radial , brachial and axillary arterial pulsations were non - palpable .
both carotid pulsations were palpable , and bruit could be heard over right carotid artery .
chest radiography showed cardiomegaly and standard 12-lead electrocardiogram ( ecg ) showed sinus tachycardia and left axis deviation suggestive of left ventricular hypertrophy ( lvh ) with repolarization abnormality .
nmol / l ( normal 110 - 520 nmol / l ) and was not suppressed with dexamethasone .
serum adrenocorticotropic hormone ( acth ) was < 11.40 pg / ml ( normal 4 - 80 pg / ml ) and 24 h urinary cortisol was 461.03 mcg / day ( normal range 20 - 100 mcg / day ) .
there was no history suggestive of any focal neurological deficit or visual disturbance and no features suggesting cerebral involvement or ischaemia with head and neck extension .
his airway was graded as mallampatti class i. patient was pre - medicated with alprazolam 0.25 mg orally , and all anti - hypertensive medications were continued until the day of surgery . in the operating room ,
a 20 g epidural catheter was inserted at t10 - 11 interspace , and 12 ml of 0.25% bupivacaine with 50 mcg fentanyl was given as a bolus dose .
approximately , 10 - 12 min after the bolus dose , patient was induced with fentanyl 100 mcg , midazolam 1 mg and thiopentone 150 mg and trachea was intubated after giving vecuronium 5 mg .
the lungs were ventilated with 50% oxygen in air and sevoflurane 1 minimum alveolar concentration .
the anaesthesia was maintained with sevoflurane , propofol infusion and intermittent boluses of fentanyl and vecuronium as needed .
intravenous infusion of hydrocortisone was started pre - operatively at the rate of 5 mg / h and continued with tapering doses till second post - operative day .
the left radial artery was selected for direct arterial pressure monitoring as the pulse was strong and was easily accessible .
intraoperative monitoring included ecg , pulse oximetry , body temperature , capnography , blood sugar level , blood gases , urine output and central venous pressure ( cvp ) .
epidural infusion of bupivacaine 0.125% with fentanyl 2 mcg / ml was used for intraoperative and post - operative analgesia .
intra - operatively , cvp was maintained between 6 and 8 cm of water , and the patient remained haemodynamically stable .
a total of 1.5 l of crystalloids and 500 ml of colloids ( 6% hydroxyl ethyl starch ) were given intra - operatively .
patient was observed in high - dependency unit for 24 h and then shifted to the ward .
he was discharged on the 8 post - operative day and referred to an endocrinologist for further care .
takayasu 's arteritis is characterised by a focal stenosis process preferentially involving the aorta and the proximal segments of its main branches . as per the new angiographic classification
, it is divided into six types ( i , iia , iib , iii , iv and v ) and the involvement of coronary or pulmonary arteries is designated as c ( + ) or p ( + ) , respectively .
hypertension in ta is commonly renovascular , and ta is the most common cause of renovascular hypertension in india .
hypertension in our patient may be related to cs . besides calcium channel blocker and alpha - adrenergic blocker ,
our patient was on an angiotensin converting enzyme inhibitor , enalapril , which was started only after stenting of left renal artery .
previous reports of the anaesthetic management of ta as well as that of cs have greatly emphasized the importance of adequate cardiovascular monitoring . fortunately , our patient had good pulses in his left arm for reliable monitoring of arterial bp .
meikle and milne had suggested that monitoring of both upper and lower limb bp should be considered in order to have some measure of overall perfusion . in our case , since left upper limb pressures correlated very well with the lower limb pressures pre - operatively , monitoring of bp in the leg was not deemed necessary .
we did not insert pulmonary artery catheter as there were no signs or symptoms of pulmonary hypertension .
we used epidural blockade as part of the anaesthetic management to achieve this without recourse to potent intravenous vasodilator agents .
hypertensive episodes during general anaesthesia can lead to cerebral haemorrhage and infarction in a patient with ta so cerebral perfusion pressure should be maintained and procedures that diminish carotid blood flow including the hyperextension of the head should be avoided .
no specialised neurological monitoring was used perioperatively . however , this patient recovered without any neurological deficits .
there is only one report of post - operative cerebral infarction in a patient with ta .
apart from steroid supplementation ( given in ta patients ) other causes of cs are unilateral or bilateral adrenal hyperplasia , pituitary adenoma , cd and ectopic acth production .
after testing the serum cortisol and acth , and the dexamethasone suppression test , patient underwent ct angiography and ultrasound examinations , which further confirmed the diagnosis .
it is well - known that patients with cs tend to suffer from volume overload , hypertension , glucose intolerance and hypokalaemic metabolic alkalosis . in our patient , anaesthesia induction was unremarkable , and there was no difficulty in airway management .
special attention was paid to patient positioning , in order to avoid fractures and/or skin damage .
there were no significant changes in the electrolytes and blood glucose level , as well as in the acid - base status .
perioperative steroid supplementation is needed in patients with cs and ta who are on steroids to prevent the occurrence of addisonian hypotensive crises in such patients .
the steroid infusion was administered to the patient in the perioperative period in the present case .
have reported two patients with concomitant ta and cd , in whom ta was exacerbated after normalisation of cortisol secretion following treatment .
similarly , there can be reactivation of ta activity after adrenalectomy due to the loss of the immunosuppressive effect of endogenous glucocorticoids and hence we should follow - up and monitor these patients for this possibility .
patients with rare association of takayasu 's arteritis with the coexisting cushing 's syndrome can be encountered in daily practice .
careful pre - operative evaluation and proper intraoperative planning and management can lead to a fruitful outcome in these patients .
takayasu 's arteritis ( ta ) is a rare , chronic progressive panendarteritis involving the aorta and its main branches . 
 anaesthesia for patients with ta is complicated by their severe uncontrolled hypertension , end - organ dysfunction , stenosis of major blood vessels , and difficulties encountered in monitoring arterial blood pressure . in a patient with cushing 's syndrome ( cs ) 
 , the anaesthesiologist needs to deal with volume overload , hyperglycaemia , hypokalaemia , difficult airway and ventilation . 
 anaesthetic management of a patient with concomitant ta and cs undergoing adrenalectomy has hardly ever been reported . 
 we present the successful anaesthetic management of a 15-year - old child with coexisting ta and cs undergoing open adrenalectomy .