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Perioperative Harlequin syndrome
- Crina L. Burlacu, Donal J. Buggy (15 September 2006)
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Coexisting Harlequin and Horners Syndromes after high thoracic paravertebral block
- Dr Anjalina Majumder, Dr Paul Farquhar-Smith (11 September 2006)
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Crina L. Burlacu , Donal J. Buggy
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Editor- We would like to thank Drs. Majumder and Farquhar-Smith for contributing with a further case report to the available literature regarding the perioperative Harlequin syndrome, and also their valuable comments regarding the mechanism underlying this syndrome (1). In regards to the prevalence of Harlequin syndrome in patients exposed to regional anaesthesia in the vicinity of thoracic and cervical sympathetic system, there are few reported encounters, indeed. At the date of our report publication (2), there was only one other published case of Harlequin appearance in adults after an internal jugular central venous cannulation (3). Other three cases were recently described (1, 4, 5), all in patients where migration of local anaesthetic at the level of preganglionic sympathetic fibres originating from T2-T4 spinal segments was highly likely. Ever since we published our first case report (2), we encountered a further similar postoperative Harlequin syndrome in a 48 years old ASA I female patient after high thoracic (T3) unilateral continuous paravertebral analgesia for breast reconstruction surgery. She presented contralateral hemifacial flushing and sweating well demarcated in the midline without the distinctive ipsilateral Horner syndrome. We were also anecdotally told of at least two other similar presentations after central venous line insertion. In all cases the hemifacial flushing and sweating was transient and not complicated. The question that arises is whether the prevalence of Harlequin syndrome after high thoracic regional anaesthesia is low or the conditions that trigger the presentation, such as heat, emotion, gustatory stimuli or exercise, are not always met, especially in the immediate perioperative period. We cannot exclude that some patients are responding with disproportionate vasodilatation and sweating of the contralateral hemiface in response to stimuli such as heat or emotion. Experimental studies in rabbit models have described a closer proximity of endothelial beta- than alpha-adrenoreceptors in relation to the sympathetic nerve terminals to the face (6). In vitro research on human facial veins has demonstrated a large inter-individual variability in the relative density and sensitivity of alpha- and beta-adrenoreceptors (7). It is possible therefore that, in some patients, the adrenergic receptors abundance and disposition may facilitate excessive beta-adrenergic vasodilatation in response to environmental stimuli. As Richardson and Cheema recently affirmed (8), there is more fascinating matter to be learnt about the interaction between the paravertebral block and the afferent sympathetic supply to the face. Crina L. Burlacu, MSc, FCARCSI, DEAA Donal J. Buggy, MD, MSc, FRCPI, FCARCSI, FRCA Department of Anaesthesia, Intensive Care and Pain Medicine Mater Misericordiae University Hospital Eccles Street Dublin 7 Ireland E-mail: crina@ireland.com 1. Majumder A, Farquhar- Smith P. Coexisting Harlequin and Horners syndromes after high thoracic paravertebral block. Br J Anaesth 2006; E- letter. 2. Burlacu CL, Buggy DJ. Coexisting harlequin and Horner syndromes after high thoracic paravertebral anaesthesia. Br J Anaesth 2005; 95: 822-4 3. Coleman P, Goddard JM. Harlequin syndrome following internal jugular vein catheterisation in an adult under general anesthetic. Anesthesiology 2002; 97: 1041 4. Mashour GA, Levine W, Ortiz VE. Intraoperative Harlequin Syndrome. Anesth Analg 2006; 102: 655 5. Crawley SM. Coexisting harlequin and Horner syndromes after high thoracic paravertebral block. Br J Anaesth 2006; 96: 537 6. Winquist RJ, Bevan JA. Relative location of alpha- and beta- adrenoceptors to sites of release of sympathetic transmitter in the rabbit facial vein. Circ Res 1981; 49: 486-92 7. Mellander S, Andersson PO, Afzelius LE, Hellstrand P. Neural beta- adrenergic dilatation of the facial vein in man. Possible mechanism in emotional blushing. Acta Physiol Scand 1982; 114 (3): 393-9 8. Richardson J, Cheema S. Thoracic paravertebral nerve block. Br J Anaesth 2006; 96: 537 Conflict of Interest:None declared |
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Dr Anjalina Majumder, Anaesthetic Specialist Registrar The Royal Marsden Hospital, Dr Paul Farquhar-Smith
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Editor- We would like to correspond further with regards to the article entitled ‘Coexisting harlequin and Horner’s syndromes after high thoracic paravertebral anaesthesia’ (1,2). A combined technique of general anaesthesia and a thoracic paravertebral block was performed. In this case a well demarcated contra lateral hemi facial flushing and ipsilateral pallor developed, without the distinctive ipsilateral Horner’s syndrome. After induction of general anaesthesia, a left paravertebral block was performed at T3/T4 using a 20G spinal needle and loss of resistance to saline technique. A total of 40 ml of local anaesthetic, 0.25% bupivacaine with 1 in 200,000 adrenaline, was injected into the paravertebral space. The operation lasted approximately 4 hours and entailed patient repositioning from right lateral to supine. Cardiovascular stability was maintained throughout and the operation was completed without incident. In the recovery room the patient was noted to have a marked hemi facial flushing of the right side (contra lateral to the block) and marked pallor on the left side, in the absence of Horner’s syndrome. The colour change persisted for approximately 5 hours post-operatively and resolved without consequence. Perioperative harlequin syndrome is caused by sympathetic block of the thermal and emotional flushing response on the pale side, with normal or excessive flushing on the contra lateral side (2). This case demonstrates that harlequin syndrome can occur without Horner’s syndrome after high volume paravertebral block (denoting T2/T3 sympathetic interruption with sparing of T1 oculomotor braches). Contrary to a previous report of harlequin syndrome without oculomotor signs (1) following a T10/11 thoracic epidural, intraoperative positioning was not a factor. Given that thoracic paravertebral injection of 15 ml of 0.5% bupivicaine causes somatic and sympathetic block over 5-8 dermatomes (4), it is perhaps surprising that harlequin syndrome, is not a common occurrence. Only three peri-operative adult cases following local anaesthetic administration have been described (1,2,3). This would imply that it is not dependent on a normal or excessive contra lateral thermal and emotional flushing response (2) but that this preserved response must be excessive and rare. Peri-operative, local anaesthetic-induced harlequin syndrome therefore may be different in mechanism to others described in the literature (5). 1. Crawly SM Coexisting harlequin and Horner syndromes after high thoracic paravertebral block. Br J Anaesth. 2006 Apr;96(4):537-8. 2. Burlacu CL and Buggy DJ Coexisting harlequin and Horner syndromes after high thoracic paravertebral anaesthesia. Br J Anaesth. 2005 Dec;95(6):822- 4. Epub 2005 Oct 14. 3. Mashour GA, Levine W, Ortiz VE. Intraoperative Harlequin syndrome. Anesth Analg. 2006 Feb;102(2):655. 4. Karamakar MK. Thoracic paravertebral block. Anesthesiology. 2001 Sep;95 (3):771-80. 5. Lance JW, Drummond PD, Gandevia SC, Morris JG. Harlequin syndrome: the sudden onset of unilateral flushing and sweating. J Neurol Neurosurg Psychiatry. 1988 May;51(5):635-42. Conflict of Interest:None declared |
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