Descriptive data are presented as mean with standard deviation and as percentage where appropriate. Patients were grouped based on the recovery status of their tongue deviation at time of follow-up (complete or partial recovery versus no recovery) and analyzed using a Kaplan-Meier survival technique. Using the statistical software R version 3.0.0 (R Foundation for Statistical Computing, Vienna, Austria), we conducted a log-rank test to compare the time-to-recovery curves between airway type, gender, diagnosis (isolated versus multiple cranial nerves), and treatment subgroups. Kaplan-Meier summary statistics provide the mean and median times to recovery, and the times to recovery for the 25th, 50th, and 75th patient quartiles. For patients with complete or partial recovery, a Pearson correlation coefficient was calculated to examine the relationship between age or operative duration and the reported follow-up interval.
RESULTS AND DISCUSSION
HNP: Airway-Related Mechanisms and Clinical Manifestations
We identified 59 publications reporting 69 patients with HNP after procedural airway management in the literature through 2013 (Table 1). Diagnoses include isolated unilateral or bilateral HNP (n = 46), as well as combined hypoglossal-lingual nerve neurapraxia (n = 8) or hypoglossal-recurrent laryngeal neurapraxia (Tapia syndrome) (n = 15).
Clinical symptoms of HNP are nonspecific and include dysarthria (difficulty with articulation), dysphagia, and even dyspnea. On examination, unilateral deviation and elevation of the tongue ipsilateral to the injured side are pathognomonic for hypoglossal nerve injury and can be attributed to paralysis of the superior and inferior longitudinal muscles.65 Later physical examination findings revealed unilateral atrophy and genioglossus muscle fasciculation, signifying denervation–reinnervation injury.66,67
Radiographic imaging, including computed tomography and magnetic resonance imaging, can help exclude ischemic stroke and hemorrhage, and provides confirmation of both supraglottic airway trauma and tongue atrophy68,69 (Fig. 1). Extracranial Doppler and ultrasound studies can aid in the diagnosis of vascular dissection as a cause of HNP.11,16 In persistent cases of HNP, electromyographic and nerve conduction studies demonstrated damage to the neural elements, a pathology that is not typical of transient neurapraxia.6
Proposed HNP Mechanisms
Most reported cases of HNP after airway management suggest involvement of the extracranial section of the hypoglossal nerve, which exits the skull through the hypoglossal canal and descends caudally, along with the internal carotid artery and jugular vein. At the mandibular angle, it passes anteriorly deep to the posterior belly of the digastric muscle and reaches the submandibular region to enter the tongue.67 At the undersurface of the tongue, numerous branches pass upward to supply its intrinsic muscles.11,29,67 The 4 mechanisms of injury leading to HNP proposed in the literature are described in Figure 2. Tapia syndrome (unilateral recurrent laryngeal nerve and hypoglossal nerve paralysis), a subset of hypoglossal nerve injury, is attributed to compression injury to intersecting extracranial fibers of both the hypoglossal and vagus nerves at the base of the tongue.13,18,33,37,53
Table 1 and Figure 3A present the demographics of patients with HNP after procedural airway management. The majority of patients with isolated hypoglossal neurapraxia and Tapia syndrome are male. No differences in demographics are seen between cases of solitary hypoglossal injury and combined cranial nerve neurapraxia. Although reporting bias must be considered, morphometric and forensic studies of the hyoid bone demonstrate greater absolute dimensions in males.70,71 Ito et al. show that males have a longer length of the greater cornu (33.8 vs 29.8 mm) and larger hyoid volume (4.31 vs 2.95 cm3) and exhibit earlier ossification of the connection between the hyoid body and greater cornu. Given these anatomical differences, male patients are more likely to experience hypoglossal nerve compression at the hyoid cornu level.
There does not appear to be any specific age range associated with anecdotal reports of HNP. Harnett et al.72 found a higher incidence of minor airway complications in infants receiving an LMA, and multiple authors report postoperative HNP after LMA placement in adolescents.26,28,31 On the other hand, several authors report findings of a calcified stylohyoid ligament on radiographic imaging in elderly patients.17,30,38,50,59 In addition, Nagai et al.50 report a patient with rheumatoid arthritis, which is known to cause cervical (C1-C2) joint instability and bony ligamentous abnormalities. It is conceivable that these anatomical abnormalities could stretch the nerve at the angle of the mandible and cause HNP. Additionally, a short neck may predispose to nerve stretching during laryngoscopy.1,34
Laterality of HNP
An earlier report of HNP suggests that right-sided neurapraxias are more common, a finding that was originally attributed to the fact that most anesthesiologists are right-handed.8 Nevertheless, most operators would use their right hand to introduce the ETT into the trachea regardless of their handedness. Theoretically, pressure exerted from the laryngoscope blade could predispose to unilateral hypoglossal injury when sweeping the tongue from the right to left before intubation. However, multiple subsequent case reports have demonstrated the bilateral incidence of this neurapraxia. Moreover, in those reports that mention the tube being taped to the right side, there is an equal prevalence of neurapraxic symptoms on either side.7,15,16,30,52
Airway Management Characteristics
Various case reports address airway management strategies including tube size, method of laryngoscopy, and blade type/size. All studies that provide laryngoscope information for orotracheal intubation report the use of a Macintosh blade, either size 3 or 4.2,7,8,15,27,30,45,47,48,52,54,57 Figure 3B displays the reported airway management techniques that were used in patients who subsequently developed isolated or multiple cranial nerve neurapraxia. In their review, Dziewas and Ludemann11 show that HNP occurs after direct laryngoscopy and endotracheal intubation, LMA placement, and even after bronchoscopy. Zamora and Saha54 discuss HNP after Combitube placement. We found a greater number of patients with HNP after ETT placement. During orotracheal intubation, neck hyperextension stretches the hypoglossal nerve on the anterior aspect of the C1 transverse process by as much as 1.3 cm. In addition, direct pressure exerted by the Macintosh blade at the base of the tongue causes soft-tissue compression against the hyoid bone, possibly exacerbating the neurapraxia.1,3,12,15,26,30,46,49,73,74
Some authors suggest that ETT cuff pressure and LMA cuff insufflation may be associated with HNP, suggesting injury at the hyoid bone.21,29,31,32,37,45,53,54 Seven patients were reported to have received at least 30 minutes of N2O as part of their anesthetic management, which would predispose to diffusion of N2O into the cuff resulting in increased cuff pressures.12,19,29,32,36,45,50
ETT Cuff Pressure
In 9 patients, the ETT cuff pressure was maintained <20 cm H2O,2,7,10,12,16,22,27,52 while in 1 patient, the intracuff pressure was maintained at 30 cm H2O before surgical draping.10 Al-Benna described hypoglossal nerve injury in a patient with a maximum measured ETT cuff pressure of 34 cm H2O.45 While there is an anatomical disparity between the location of the inflated ETT cuff and the hypoglossal nerve, it is possible that ETT cuff–related damage may be explained by anatomical variants, such as a low-looping hypoglossal nerve or tongue innervation from the superior root of the ansa cervicalis.
LMA Cuff Volume
Eight cases with LMA use mention cuff insufflation volume in the range of 15 to 40 mL of air19,21,28,29,31,32,37,50 but did not mention goals for cuff pressure titration or intraoperative monitoring. Lumb and Wrigley75 demonstrated that LMA cuff pressures can increase by as much as 50% during brief periods of N2O anesthesia. Similarly, Trumpelmann and Cook32 reported on an overdistended LMA cuff after removal in a patient who had received N2O during anesthesia. Although the cuff insufflation volume varies considerably by LMA size and type, these unanticipated increases in cuff volume during longer cases can compress the hypoglossal nerve against the hyoid bone and cause HNP symptoms.
Operative Duration and Reintubation
Anesthetic duration and procedural duration in the reported cases vary (Table 1). Aside from the complications associated with prolonged intubation, no studies have evaluated its relative contribution specifically to HNP. On the other hand, repeated airway management attempts,5,29 intra- and postoperative reintubations,6,27,28,40,57 and prolonged ventilatory support27,35,40 increase the risk of iatrogenic trauma to the airway mucosa and underlying nerve structures. Three patients required reintubation due to respiratory failure.6,27,40 Two additional patients required LMA replacement28 or conversion to ETT34 for preoperative supraglottic airway device dislocation. All 5 patients with bilateral isolated HNP included 1 of these factors of complex airway management.6,27–29,35
Surgical subspecialties associated with subsequent isolated HNP or combined neurapraxias are displayed in Figure 3C and listed in Appendix Table 1. However, HNP is frequently reported after otolaryngologic surgery. Dysarthria and ipsilateral tongue deviation are mentioned after rhinoplasty3,7,22,38,52 and sinus surgery,30,34 as well as after tonsillectomy1,4,49,51,58,59 and periglottic excisions.1,8,14 Throat pack placement during these surgeries can create pressure at the greater cornu of the hyoid,2 and their frequent use is linked to combined hypoglossal-recurrent (Tapia syndrome) and hypoglossal-lingual nerve palsies.12,22,52 Similarly, hematoma and other postsurgical upper airway swelling can result in delayed symptoms and dysarthria due to nerve compression.9,10,19,76 The otolaryngology team can detect tongue deviation in patients with subclinical HNP (i.e., without symptomatic dysarthria) through frequent routine neurologic examinations that are not consistently used in other specialties, contributing to an increased diagnostic rate and reporting bias.
Several authors report HNP after shoulder surgeries.10,11,16,19,21,23,25,50 Neck rotation and undetected head movement underneath the surgical drapes can lead to prolonged traction of the hypoglossal nerve throughout the case.5,16,37,46 During cardiac surgery, neck hyperextension and lateral flexion during sternotomy can compress the ETT cuff against the hypoglossal-recurrent laryngeal nerve, resulting in Tapia syndrome.13,42,43,57 Similarly, unanticipated position changes resulting in accidental extubation,36 LMA malposition,28 or change in airway management34 (e.g., switching from LMA to ETT) are associated with HNP.
Routine position changes after intubation, such as from semisupine (30 degrees) to the Fowler position (70 degrees), can cause pressure injury to the nerve throughout its superficial course anterior to the mandible.7,16,23,25,38,48,52 It is possible that even small position changes after airway securement, including during surgical preparation and draping, could predispose to hypoglossal nerve trauma. Conrardy et al.77 demonstrated that the ETT cuff can migrate from 3.8 to 6.4 cm with neck flexion or extension during intubation, potentially injuring the subglottis. This pattern of trauma would more likely result in recurrent laryngeal nerve injury and dysphonia as in Tapia syndrome.
Clinical Course and Management
HNP is typically diagnosed in a delayed fashion, with more than half of the reported cases diagnosed the day after surgery. Nevertheless, all but 3 patients exhibited tongue deviation by the end of the first postoperative day.10,12,33 Residual anesthesia may interfere with an early diagnosis of neurapraxia. Some patients after otolaryngology or general surgery do not exhibit signs or symptoms until their first postoperative day or later.11,15,22,24,32,52 Due to the delayed onset of symptoms, neurapraxia can potentially develop after discharge and remain undiagnosed.
HNP appears to be largely self-limited; of 60 patients with a reported recovery status and follow-up interval, 26 patients (43.3%) achieved resolution within 6 weeks after surgery, and an additional 24 patients (40.0%) were symptom-free within 6 months of their operative date. The 25th percentile, median, and 75th percentile are 28, 60, and 120 days, respectively. Several authors reported complete resolution 1 year after diagnosis,6,9,12,14,20,22 while others found only partial recovery at variable follow-up periods.1,15,33,49 Five patients (8.3%) had persistent tongue deviation and dysarthria at follow-up intervals. The follow-up interval was not reported for 4 additional patients with persistent symptoms.1,3,4,13,55,56 Patients with partial recovery demonstrated similar demographics and operative durations when compared to fully recovered patients. More than half of the patients with partial recovery are associated with Tapia syndrome, and remaining neurologic deficits include persistent tongue deviation15 or vocal fold immobility. Patients with isolated or combined cranial nerve neurapraxias (recurrent laryngeal, lingual, or glossopharyngeal nerves) are reported to recover at similar follow-up intervals (P = 0.34). However, patients receiving an ETT exhibited later recovery postoperatively than patients in whom an LMA was used (P = 0.003) (Fig. 4). Indeed, the invasive technique (direct laryngoscopy), neck positioning, and cuff pressures associated with ETT placement can be more traumatic to the airway mucosa and require longer healing times. In addition, the reported follow-up period for patients exhibiting complete recovery is similar between genders (P = 0.09), and age is poorly correlated with the recovery follow-up interval (r = −0.090). There is a moderate positive correlation between operative duration and follow-up interval for patients with reported recovery status (r = 0.49).
A few shortcomings must be considered when interpreting these recovery estimates and analyses. The reported data are extracted retrospectively from individual publications and several authors instead of 1 study. In addition, patients may have recovered earlier than the reported follow-up period in each publication, and patients with longer recovery times may be disproportionately represented in this sample of case reports. On the other hand, the correlations between age or operative duration and time to recovery exclude patients with persistent HNP who may have needed longer recovery times past the last recorded encounter.
Possible Preventive Measures
Potentially preventive measures are deduced by the postulated mechanisms of injury, with an emphasis on the use of less invasive methods of airway management (LMA instead of ETT) (Appendix Table 2). Indeed, some authors postulate that routine cuff pressure monitoring could decrease the incidence of HNP after surgery.31,32 Although no neurapraxias were noted in their study of 200 patients receiving an LMA for ambulatory surgery, Seet et al.78 demonstrated a decrease in dysphagia and dysphonia at 1 hour and 1 day after surgery in patients whose LMA cuff pressures were limited to <60 cm H2O. Similarly, Ratnaraj et al.79 showed that maintaining ETT cuff pressure <20 cm H2O in patients undergoing cervical spine surgery significantly decreased the incidence of sore throat 24 hours after extubation. Intermittent pressure cuff lowering during long operations or pressure-relief valves can decrease the risk of nerve compression,29,75,80 and it follows that LMA or ETT cuff deflation during surgical positioning could also prevent iatrogenic injury to the hypoglossal and recurrent laryngeal nerves, respectively. Bohner et al.81 described the first use of a nerve stimulator for the successful identification and continuous monitoring of the hypoglossal nerve during an anatomically challenging carotid endarterectomy under general anesthesia.
Supportive measures for HNP during initial evaluation in the immediate postoperative period may include supplemental oxygen and respiratory monitoring. Otolaryngology-guided rehabilitation measures include dietary modifications, logopedic treatment, and electrical stimulation therapy.82 Corticosteroid therapy has been shown to accelerate spontaneous recovery after Bell palsy,83 and multiple authors advocate a short course of high-dose steroids such as prednisone if airway edema is suspected.5,7,15,24,26,28,33,46,50,53 However, there are no controlled studies of the benefits of these treatments on neurapraxic patients after surgery. In our review, patients receiving corticosteroid treatment demonstrated complete or partial recovery at similar follow-up periods compared to nontreated patients.
Closed Claims Data
There are only 4 nonsurgical hypoglossal nerve injury claims in the Anesthesia Closed Claims database (1980–present: 10,093 claims). A difficult intubation with pharyngeal injury occurred in 1 claim, and an LMA was used in 2 claims. Three of the nonsurgical injuries were permanent, and 1 temporary. Only 1 of these 4 claims resulted in payment ($30,500 in 2012 inflation-adjusted dollars), a significantly smaller proportion when compared to other surgical anesthesia claims (58%, P = 0.012) (Domino KB, University of Washington Medical Center, personal communication, March 17, 2014).
Hypoglossal neurapraxia after airway extubation is repeatedly reported after various surgeries. Nerve compression and overstretching can occur during both unexpected and routine position changes, including neck hyperextension for laryngoscopy and surgical positioning. Male patients may be more vulnerable given their larger hyoid bone dimensions. Excessive pressure in the ETT or LMA cuff, perhaps exacerbated by the use of N2O, may produce injurious malposition of the airway devices. Early postoperative detection of tongue deviation and dysarthria, as well as consultation with neurology and otolaryngology consultants, can help exclude other serious etiologies including stroke and carotid dissection. Minimizing airway instrumentation during endotracheal intubation, along with consideration for intermittent pressure monitoring of the ETT cuff and position during long surgical procedures, may decrease the incidence of cranial nerve neurapraxias. While a short course of steroids may decrease swelling after airway removal, further studies need to be performed to ascertain their effect on the incidence of postoperative HNP and the recovery period for neurapraxic patients.
Name: Aalap C. Shah, MD.
Contribution: This author helped prepare the manuscript, conduct the literature review, and choose and execute statistical tests.
Attestations: Aalap C. Shah approves the final manuscript, attests to the integrity of the analysis reported in the manuscript, and is the archival author.
Name: Christopher Barnes, MD.
Contribution: This author helped prepare original artwork, the figure layout, and manuscript preparation.
Attestations: Christopher Barnes approves the final manuscript, and attests to the integrity of the analysis reported in the manuscript.
Name: Charles F. Spiekerman, PhD.
Contribution: This author helped prepare the manuscript and choose and execute statistical tests.
Attestation: Charles F. Spiekerman approves the final manuscript, and attests to the integrity of the analysis reported in the manuscript.
Name: Laurent A. Bollag, MD.
Contribution: This author helped prepare the manuscript and choose and execute the statistical tests.
Attestation: Laurent A. Bollag approves the final manuscript, and attests to the integrity of the analysis reported in the manuscript.
This manuscript was handled by: Sorin J. Brull, MD, FCARCSI (Hon).
We thank Dr. Karen Domino for providing data from the ASA Closed Claims Database. We also thank Dr. Allan Goldman and Paul Constanthin for their critical review of our manuscript.
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