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Case report
Dexmedetomidine in difficult airway management with a fibre-optic bronchoscope in the awake patient with Klippel–Feil Syndrome
Dexmedetomidina en el manejo de la vía aérea difícil con fibrobroncoscopio en paciente despierto afecto de síndrome de Klippel-Feil
S. Pacreua,
Corresponding author
94397@parcdesalutmar.cat

Corresponding author.
, S. Martínezb, E. Vilàa, L. Moltóa, J. Fernández-Candila
a Servicio de Anestesiología, Reanimación y Terapia del dolor, Parc de Salut Mar, Barcelona, Spain
b Servicio de Anestesiología, Reanimación y Terapia del Dolor, Hospital de la Santa Creu i Sant Pau, Barcelona, Spain
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and is one of the leading congenital causes of difficult airway&#46;<a class="elsevierStyleCrossRefs" href="#bib0070"><span class="elsevierStyleSup">2&#44;3</span></a> Short neck with limited movement and cervical instability can lead to neurological damage during laryngoscopy&#44; intubation and position during surgery&#46; Because of their limited neck mobility&#44; anaesthesiologists must consider awake fibreoptic intubation &#40;FOI&#41; in these patients&#46; Ideally&#44; the patient should be sufficiently sedated to tolerate the procedure&#44; but awake enough to cooperate&#46; Dexmedetomidine &#40;DEX&#41;&#44; an imidazoline compound&#44; is an &#945;2 adrenoceptor agonists which works by binding to the &#945;2 adrenergic receptor&#46; It exerts its hypnotic-sedative action at the level of the locus coeruleus<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">4</span></a> by binding to the &#945;2A receptors of this cell group&#44; causing a dose-dependent decrease in noradrenaline levels&#46; This reduces activity in the noradrenergic ascending pathway which&#44; together with a decrease in serotonergic neurotransmission&#44; is associated with the transition from the waking state to sleep&#44; and facilitates easy arousal&#44; thus allowing the patient to cooperate and respond&#44; despite the sedation&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">We present a case of difficult airway in a patient with KFS scheduled for extension of a previous craniectomy under general anaesthesia&#46; The patient was sedated with DEX infusion to facilitate awake FOI&#46; We obtained the patient&#39;s consent to publish this case report&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 43-year-old man&#44; ASA II&#44; with known KFS&#44; who presented congenital fusion of cervical vertebral&#44; low occipital hairline&#44; short neck and facial dysmorphism&#44; associated with syringomyelia &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; He also presented paresthesia of the left side of the tongue and soft palate&#44; right rotational nystagmus&#44; facial hemianaesthesia&#44; dysphonia due to paralysis of the left recurrent laryngeal nerve that shifted the left vocal cord to the paramedial position&#44; with normal glottic passage&#44; and dysphagia&#46; His surgical history included suboccipital decompression&#44; C1 laminectomy and resection of the atlantooccipital membrane due to Chiari&#39;s disease&#46; In the foregoing surgery&#44; awake FOI was performed&#46; Due to clinical progression and enlargement of syringomyelia &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#44; the patient was admitted to our unit for extension of craniectomy and duraplasty&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Physical examination of the airway showed mouth opening of less than 5<span class="elsevierStyleHsp" style=""></span>cm&#44; thyromental distance of less than 6&#46;5<span class="elsevierStyleHsp" style=""></span>cm&#44; severely limited neck flexion and extension&#44; limited mandible subluxation &#40;lower incisors behind the upper&#41;&#44; and Mallampati IV&#46; Given the likelihood of difficult intubation&#44; and it was decided to perform awake FOI&#46; In the operating room&#44; monitoring consisted of non-invasive blood pressure&#44; 5-lead electrocardiogram&#44; and oxygen saturation measured by pulse oximetry&#46; After placing an intravenous line&#44; the patient was premedicated with midazolam &#40;2<span class="elsevierStyleHsp" style=""></span>mg&#41; and DEX infusion&#44; starting at 0&#46;6<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;h&#46; Following this&#44; the posterior pharynx was coated with 10<span class="elsevierStyleHsp" style=""></span>ml of 2&#37; lidocaine gel and sprayed with 2 doses of 10&#37; Xylocaine&#44; and 4<span class="elsevierStyleHsp" style=""></span>l&#47;min of oxygen was delivered via nasal prongs&#46; After 10<span class="elsevierStyleHsp" style=""></span>min&#44; the fibreoptic bronchoscope &#40;FOB&#41; was inserted once the vocal cords were visualised&#44; local anaesthetic &#40;5<span class="elsevierStyleHsp" style=""></span>ml 2&#37; lidocaine&#41; was injected via the FOB and the patient was intubated using a no&#46; 7 tracheal tube&#44; without incident&#46; The patient did not cough or move his neck&#44; and oxygen saturation was maintained at between 96&#37; and 97&#37;&#46; After intubation&#44; EtCO<span class="elsevierStyleInf">2</span> was confirmed by capnography and vesicular murmur was heard in both hemithorax&#46; General anaesthesia was induced with propofol &#40;50<span class="elsevierStyleHsp" style=""></span>mg&#41;&#44; fentanyl &#40;100<span class="elsevierStyleHsp" style=""></span>&#956;g&#41; and rocuronium &#40;30<span class="elsevierStyleHsp" style=""></span>mg&#41;&#44; and maintained with sevoflurane 0&#46;5&#37; MAC to maintain BIS values between 45 and 60&#44; with a mixture of 50&#37; oxygen&#47;air and perfusion of DEX 0&#46;6<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;h to spare anaesthetic drugs&#46; Neuromuscular blockade was monitored intraoperatively by kinemiography&#44; using a Datex-Ohmeda Neuromuscular Transmission monitor &#40;GE Healthcare&#44; Helsinki&#44; Finland&#41;&#46; Surgery lasted for around 4<span class="elsevierStyleHsp" style=""></span>hours&#44; and a further bolus of rocuronium was administered to maintain a TOF of 0&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">At the end of surgery&#44; sevoflurane and DEX infusion was discontinued&#44; and sugammadex &#40;2<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#41; was administered to reverse the residual neuromuscular blockade&#46; The patient returned to spontaneous breathing and started to obey orders&#46; He was extubated 5<span class="elsevierStyleHsp" style=""></span>min after the end of surgery&#44; transferred to the postanaesthesia care unit for postoperative monitoring&#44; and from there to the hospital ward 24<span class="elsevierStyleHsp" style=""></span>h later&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0035" class="elsevierStylePara elsevierViewall">KFS is a rare entity that was first described in 1912 by Maurice Klippel and Andr&#233; Feil&#46; The syndrome is characterised by the congenital fusion of the 2nd to the 7th cervical vertebra&#46; There are several degrees of fusion&#58; &#40;a&#41; type <span class="elsevierStyleSmallCaps">I</span>&#44; massive fusion of the cervical spine to the upper dorsal level&#59; &#40;b&#41; type <span class="elsevierStyleSmallCaps">II&#44;</span> fusion of 1 or 2 vertebrae&#44; generally accompanied by occipital&#8211;cervical fusion and hemivertibrae&#46; This is the most common&#44; although it is largely asymptomatic&#59; &#40;c&#41; type III&#44; in which cervical fusion is associated with a similar disorder at the dorsal or lumbar level&#59; and &#40;d&#41; type IV&#44; which involves cervical&#44; upper thoracic&#44; lower dorsal or lumbar fusion&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">1</span></a> The syndrome&#44; particularly types I and IV&#44; can be associated with other alterations&#46; Our patient presented type <span class="elsevierStyleSmallCaps">II</span> KFS&#44; with no associated clinical manifestations&#46;</p><p id="par0040" class="elsevierStylePara elsevierViewall">Airway management in patients with this syndrome is challenging for the anaesthesiologist&#46; Different sedative drugs have been used&#44; such as benzodiazepines&#44; opioids &#40;such as iv remifentanil&#41;&#44; ketamine&#44; and propofol&#44;<a class="elsevierStyleCrossRef" href="#bib0085"><span class="elsevierStyleSup">5</span></a> but almost all cause respiratory depression&#46; We suggest DEX as an adequate and effective option in these patients with difficult airway&#44; provided it is not contraindicated due to associated cardiac dysfunction&#44; and also as a coadjuvant&#44; since it provides adequate sedation without causing respiratory depression&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">4</span></a> Local anaesthesia of the upper airway is also important to ensure successful completion of the procedure&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Other authors have reported the use of DEX for sedation in awake FOI in patients with difficult airway&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">6&#8211;8</span></a> These included 2 cases of parturients&#44; one with KFS and another with spinal muscular atrophy type <span class="elsevierStyleSmallCaps">III</span> &#40;Kugelberg-Welander disease&#41; in whom neuraxial block was ruled out due to Arnold Chiari malformation and multiple spinal surgeries&#44; and in which DEX was used for sedation in FOI during caesarean section&#46;<a class="elsevierStyleCrossRefs" href="#bib0100"><span class="elsevierStyleSup">8&#44;9</span></a> In these cases&#44; DEX was chosen for its sedative&#44; anxiolytic&#44; hypnotic and analgesic properties&#44; and also as an anaesthetic-sparing strategy&#44; Furthermore&#44; DEX is an antisialogogue that attenuates the neuroendocrine response and protects cardiovascular function without affecting breathing&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">4</span></a> All these properties&#44; together reports of it use as an analgesic during FOI&#44;<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">5&#44;7</span></a> prompted us to use 0&#46;3<span class="elsevierStyleHsp" style=""></span>g&#47;kg&#47;h iv DEX with midazolam for awake FOI&#46; The recommended loading dose is 1<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg for 10<span class="elsevierStyleHsp" style=""></span>min &#40;rapid intravenous bolus is not indicated due to the risk of hypertension&#41; followed by a maintenance infusion of 0&#46;2&#8211;0&#46;7<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;h&#46; In our case&#44; we did not administer a loading dose&#44; since the half-life of DEX is 4&#8211;10&#44; and during this time the local anaesthetic was instilled in the airway&#46;<a class="elsevierStyleCrossRefs" href="#bib0110"><span class="elsevierStyleSup">10&#44;11</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Although DEX is associated with few adverse effects&#44; the most frequent are hypotension&#44; bradycardia&#44; nausea and dry mouth typically caused by &#945;2 agonists&#46;<a class="elsevierStyleCrossRefs" href="#bib0090"><span class="elsevierStyleSup">6&#44;12</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The DEX also facilitates extubation by reducing the need for hypnotic drugs and opioids&#44; which could seriously compromise the airway&#46; In our case&#44; we decided to continue iv DEX intraoperatively to spare anaesthetic drugs and facilitate rapid awakening&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">We believe it best to plan the best possible airway management strategy in patients with KFS and known difficult airway&#46; In our case DEX&#44; with its excellent safety profile&#44; provided adequate sedation during intubation&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">The authors have no conflict of interest to declare&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Klippel&#8211;Feil syndrome is a disease characterised by congenital fusion of cervical vertebra&#44; which leads to cervical limitation and instability&#46; In these cases&#44; the best option is the orotracheal intubation with the fibre-optic bronchoscope with the patient awake&#46; The advantage is that cervical movements that could lead to neurological damage are minimised&#46; In these patients&#44; adequate sedation&#44; together with instillation of local anaesthetic in the pharynx and hypopharynx&#44; is the key to reducing patient discomfort and achieving successful orotracheal intubation&#46; Dexmedetomidine is a selective &#945;2-adrenergic receptor agonist that produces sedation and analgesia at the locus coeruleus without producing respiratory depression&#44; as well as maintaining patient collaboration&#46; The case is presented of a patient with Klippel&#8211;Feil syndrome and difficult airway management&#44; who was given a dexmedetomidine infusion at 0&#46;6<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;h as sedation for an awake fibre-optic endotracheal intubation&#46;</p></span>"
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        "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">El s&#237;ndrome de Klippel-Feil es una enfermedad que se caracteriza por la fusi&#243;n cong&#233;nita de v&#233;rtebras cervicales&#44; que condiciona una limitaci&#243;n e inestabilidad cervical&#46; En estos casos la mejor opci&#243;n es la intubaci&#243;n orotraqueal con fibrobroncoscopio con el paciente despierto&#46; La ventaja es que se minimizan los movimientos cervicales que podr&#237;an conllevar un da&#241;o neurol&#243;gico&#46; En estos pacientes una sedaci&#243;n adecuada&#44; junto con la instilaci&#243;n de anest&#233;sico local en la faringe y la hipofaringe es clave para reducir las molestias del paciente y conseguir la intubaci&#243;n orotraqueal con &#233;xito&#46; La dexmedetomidina es un agonista selectivo de los receptores &#945;-2 adren&#233;rgicos que produce sedaci&#243;n y ansiolisis al nivel del locus coeruleus&#44; sin provocar depresi&#243;n respiratoria&#44; y preservando la colaboraci&#243;n del paciente&#46; Presentamos el caso de un paciente con s&#237;ndrome de Klipple-Feil y v&#237;a a&#233;rea dif&#237;cil en el que utilizamos una perfusi&#243;n de dexmedetomidina a dosis de 0&#44;6<span class="elsevierStyleHsp" style=""></span>&#956;g&#47;kg&#47;h como sedaci&#243;n para la intubaci&#243;n orotraqueal con fibrobroncoscopio con el paciente despierto&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Pacreu S&#44; Mart&#237;nez S&#44; Vil&#224; E&#44; Molt&#243; L&#44; Fern&#225;ndez-Candil J&#46; Dexmedetomidina en el manejo de la v&#237;a a&#233;rea dif&#237;cil con fibrobroncoscopio en paciente despierto afecto de s&#237;ndrome de Klippel-Feil&#46; Rev Esp Anestesiol Reanim&#46; 2018&#59;65&#58;537&#8211;540&#46;</p>"
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