Tapia's syndrome, first described by the Spanish otorhinolaryngologist A.G. Tapia in 1904, includes two distinct conditions, differing in clinical manifestation and pathophysiological mechanism. The central Tapia's syndrome is a typical crossed brainstem syndrome caused by a lesion of nucleus ambiguus, nucleus of hypoglossal nerve and the pyramidal tract.1 The peripheral Tapia's syndrome is due to an extracranial lesion of the hypoglossal nerve and recurrent laryngeal branch of the vagal nerve that can be provoked by tumours, head and neck operations, radiation therapy, meningitis, infectious mononucleosis and carotid artery dissection. The latter has been described, in a few cases, as a complication of oropharyngeal manipulation during anaesthetic airway management. Indeed, it has been demonstrated that the anterior displacement of the tongue during laryngoscopy may cause a substantial strain on the hypoglossal nerve. Moreover, reclination of the head, as performed in the ‘sniffing position’ during laryngoscopy, may affect distension of the nerve as well.2 Up to now, only one case of peripheral Tapia's syndrome following oropharyngeal manipulation during cardiac surgery has been reported, in particular after coronary artery bypass.3
We now describe another case of peripheral Tapia's syndrome with unilateral hypoglossal and recurrent laryngeal nerve palsy occurring after uncomplicated orotracheal intubation during cardiac surgery.
A 72-year-old man, with arterial hypertension, ulcerative rectocolitis and aortic valvular disease, was scheduled for aortic valve replacement. He was on angiotensin-converting enzyme (ACE) inhibitors, β-blockers and calcium antagonists.
Anaesthesia was induced using a combination of thiopentone 375 mg intravenously (i.v.) and sufentanil 25 μg i.v., and muscle relaxation was facilitated with vecuronium 8 mg i.v. Laryngoscopy was performed by means of a stainless steel Macintosh blade size 4 (Medicon; Tuttlingen, Germany), and transoral intubation was carried out with a 9 mm endotracheal tube. No complications were encountered during laryngoscopy and intubation. The cuff of the endotracheal tube was inflated to a pressure of 20 cmH2O. The tube was positioned in the right side of the mouth. A nasogastric tube (PVC, Levin duodenal tube, size Ch.16, Rüsch) was placed through the left nostril. General anaesthesia was maintained with sevoflurane in oxygen–air and sufentanil. After midline sternotomy, before the beginning of the extracorporeal circulation, i.v. heparin (3 mg kg−1 at a total dose of 210 mg) was administered. An uncomplicated operation was performed, and the patient was then transferred to the ICU and extubated after 25 h. After 72 h in the ICU, he was transferred to the cardiac surgery ward in good general condition.
In the next few hours, the patient complained of dyspnoea and dysphagia. Neurological examination revealed paralysis of the muscles of the left part of the tongue causing left-sided deviation with protrusion, consequent dysphagia, and dysphonia and hoarseness, suggesting the occurrence of Tapia's syndrome. Methyl-prednisolone 200 mg was given i.v., and the patient was then switched to oral therapy for 10 days. Laryngoscopy showed severe paresis of the left hemilarynx, with saliva stagnancy in the piriform sinus and normal motility of the right hemilarynx. Moreover, a haematoma of the left posterior side of the rhinopharynx was observed. A computed tomography (CT) and MRI scan did not reveal any lesion involving the brain or the neck. The patient was able to breathe adequately, but he needed a restricted diet in order to reduce the risk of aspiration. One week later, on otorhinolaryngological examination, the size of the haematoma was decreased and the movement of the left vocal cord was starting to recover. A needle electromyography (EMG) performed 4 weeks after the onset of symptoms showed fibrillation and positive sharp waves in the muscles of the left side of the tongue, which appeared hypotrophic. Three months later, the patient was re-evaluated: the neurological examination showed a marked improvement of signs and symptoms, with the disappearance of dysphonia; a minimal left-sided deviation of the tongue with protrusion was still observed. The needle EMG revealed sporadic fibrillation in the muscles of the left part of the tongue, with a voluntary pattern recruitment characterized by polyphasic, low-amplitude regenerant motor unit potentials.
The peripheral Tapia's syndrome is a rare complication of surgery, the pathophysiological mechanism of which is often a consequence of orotracheal intubation. In the case reported here, unilateral paralysis of the muscles of the tongue and of the ipsilateral vocal cord was due to a lesion of the 10th and 12th cranial nerves occurring after oropharyngeal manipulation during general anaesthesia for aortic valve replacement surgery. It has been proposed that, depending on the extent of the exerted strain, the nerve function may be temporarily impaired due to stretching (i.e. neuroapraxia) or the nerve may be permanently damaged due to extraction of its fibers (i.e. axonotmesis or neurotmesis). We assume that, in this case, a lesion of the nerves was probably caused by the rhinopharyngeal haematoma due to the placement of the nasogastric tube and worsened by heparin therapy. The haematoma was located in the pharynx along the nasogastric tube just at the crossing of the vagus and the hypoglossal nerve (which run on the lateral prominence of the anterior surface of the transverse process of C1).4 Additionally, reclination of the head, as performed in the sniffing position during general anaesthesia, might have affected distension of the nerve as well. To our knowledge, there are only four reports on peripheral unilateral Tapia's syndrome after transoral intubation for general anaesthesia and only one after cardiac surgery. Yavuzer et al.5 described a case of Tapia's syndrome after septorhinoplasty as a consequence of the compression of the nerve after inflation of the cuff within the larynx. Tesei et al.4 presented a unilateral Tapia's syndrome following endotracheal intubation after rhinoplasty under general anaesthesia. They believed that compression by the endotracheal tube just at the crossing of the vagus and hypoglossal nerve and the hyperextension of the neck were able to cause a pressure neuropathy.4 Boisseau et al.6 reported a case of Tapia's syndrome after general anaesthesia following shoulder surgery. The syndrome in their case was believed to be due to a compression by the endotracheal tube, caused by the displacement of the head.6 Johnson and Moore7 showed a case of paralysis of the 10th and 12th cranial nerves' after interscalene brachial plexus block. Tapia's syndrome has also been described in cardiac surgery by Sotiriou et al.3 as a postoperative complication of coronary artery bypass grafting (CABG) surgery. They observed a 52-year-old woman who developed airway obstruction each time extubation was attempted. A neurological examination revealed bilateral hypoglossal and recurrent laryngeal nerve palsy. The patient was unable to move the tongue and had pooled secretions in the oropharynx. Electromyographic examination showed denervation of the tongue. The authors hyphothesized that inadvertent hyperextension and lateral flexion of the neck during sternotomy and endotracheal tube malposition had occurred, leading to compression at the crossing point of the vagal and hypoglossal nerve.3
Although all the above-mentioned mechanisms proposed for Tapia's syndrome cases in the literature have to be considered, in our experience nasogastric tube placement played a central role by causing the rhinopharyngeal haematoma. In our opinion, heparin therapy, as a main step in the management of patients undergoing cardiac surgery, could have reasonably exacerbated the haematoma, leading to delayed symptoms. In conclusion, we describe here another cause of Tapia's syndrome related to airway management procedures and recommend careful manipulation of nasogastric tubes in anticoagulated patients.
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