Journal of Clinical Anesthesia (2005) 17, 650 – 654
Case conference of the University of Florida Series Editors: A. Joseph Layon MD ! Michael E. Mahla MD Associate Series Editors: Lawrence Caruso MD ! Andrea Gabrielli MD
Apparent sialadenopathy of the sublingual glands after general anesthesia with placement of laryngeal mask airwayB Joshua Fuhrmeister MD (Resident)a, Jennifer Janelle MD (Assistant Professor)b, R. Victor Zhang MD, PhD (Assistant Professor)a,* a
Department of Anesthesiology, University of Florida College of Medicine, Box 100254, Gainesville, Florida 32610-0254, USA Department of Medicine, University of Florida College of Medicine, Box 100254, Gainesville, Florida 32610-0254, USA c Department of Otolaryngology, University of Florida College of Medicine, Box 100254, Gainesville, Florida 32610-0254, USA b
Received 17 May 2005; accepted 17 May 2005
Comment by Jack D. Sedwick MDc Keywords: Sialadenopathy; Salivary glands; Endoscopic procedures
Abstract Sialadenopathy, inflammation of the salivary glands, is a rare anesthetic complication. This case conference describes a patient who developed sialadenopathy of the sublingual glands after placement of a laryngeal mask airway during a brief surgical procedure. The patient’s history is detailed, the surgical and anesthetic procedures are described, and a discussion follows, which describes the different causes of this inflammatory process. In the comment, the physiology of the salivary glands is described in detail.
1. Introduction Sialadenopathy, the inflammation of salivary glands, rarely occurs in anesthesia practice. Since first described by Attas et al [1] in 1968, there have been several reports of postoperative sialadenopathy, usually associated with endotracheal intubation and after perioral endoscopic procedures under local anesthesia [1-5]. More recently, sialadenopathy
has been reported in association with the use of a laryngeal mask airway (LMA) [6,7]. These reports described swelling of bilateral parotid glands or bilateral submandibular glands after general anesthesia with the placement of an LMA. In our case report, however, we describe a case of bilateral sublingual sialadenopathy after general anesthesia with the use of an LMA.
2. Case report B
Case conference presentations are selected and edited at the Department of Anesthesiology, University of Florida College of Medicine. T Corresponding author. Tel.: +1 352 395 8012; fax: +1 352 395 8013. 0952-8180/$ – see front matter doi:10.1016/j.jclinane.2005.05.002
A 38-year-old woman with end-stage renal disease presented for removal of a nonfunctioning Tenckhoff
Sialadenopathy of the sublingual glands peritoneal dialysis catheter. She previously had a cadaver kidney transplant and multiple arteriovenous fistulas. Her medical history was also significant for recurrent deep vein thrombosis, hypertension, and peripheral neuropathy and was positive for lupus anticoagulant. The patient was not on any anticoagulants at the time of this admission due to a recent gastrointestinal bleed. One year before this admission, she had experienced an episode of postoperative tongue swelling for several days after the placement of a Tenckhoff catheter under general anesthesia with endotracheal intubation. On examination, she weighed 80 kg, was 155 cm in height, and had acceptable vital signs. She had a small mouth opening (two and a half finger breath), a Mallampati score of III, and a relatively large tongue. After induction of anesthesia with intravenous propofol and fentanyl, a lubricated size 4 Proseal LMA was placed into the hypopharynx, but could not be positioned to achieve a good seal, and was thus removed. Subsequently, a lubricated size 4 Classic LMA was placed into the hypopharynx without difficulty using the standard technique. The insertion was atraumatic, without coughing or gagging. The cuff was inflated with the recommended volume of air, and a good seal was obtained with a leak at 20 cm H2O. The placement was judged satisfactory by observing chest rise with gentle positive pressure ventilation. General anesthesia was maintained with sevoflurane and 65% nitrous oxide in oxygen, with the patient breathing spontaneously throughout the procedure. Surgery was uneventful and lasted less than 30 minutes. No signs of excessive salivation or other oral pathology were observed. At the end of surgery, the patient emerged from anesthesia smoothly, and the LMA was removed after the patient awakened. She maintained a good airway and recovered in the postanesthesia recovery room without any complaints.
Fig. 1 Picture taken about 3 hours after the surgery showing large and hyperemic sublingual glands. To reduce the pain, a tongue depressor was used by the patient to keep the tongue away from the swollen glands. The sublingual area was very painful to touch and the patient was unable to tolerate oral intake.
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Fig. 2 On postoperative day 1, the sublingual glands were less swollen, less hyperemic, and less painful. Topical application of 2% viscous lidocaine was effective in reducing the pain. The patient was able to start eating some liquid food, although the patient still had burning pain when the area was in contact with the food.
Soon after arrival to the ward, the patient began to complain of increasing pain in her sublingual area. Examination showed very engorged and hyperemic sublingual glands, which were very painful on touch. To reduce the pain at the sublingual area, the patient used a tongue depressor to hold the tongue up from the swollen sublingual glands (Fig. 1). Topical application of 2% viscous lidocaine to the sublingual area was also helpful in relieving the pain. On postoperative day 1, the sublingual glands were still swollen and painful, but the patient no longer needed to hold up the tongue (Fig. 2). The patient was able to begin taking liquid enteral nutrition, although she complained of burning pain at the sublingual area upon contact with this material. Over the next 3 to 4 days, the swelling and pain dissipated completely (Fig. 3). There were no long-term sequelae.
Fig. 3 The pain and swelling of the sublingual glands dissipated completely over 5 to 6 days after the surgery. This is a photo of the sublingual glands 10 days after the surgery, showing a complete recovery of the sublingual glands from the sialadenopathy.
652 In the following year, the patient returned to the operating room on two occasions for the placement and then removal of another Tenckhoff catheter. She was given general anesthesia with endotracheal intubation with smaller endotracheal tubes (6.0 and 6.5 mm), and there was no postoperative sialadenopathy.
3. Discussion Various local complications of LMA use have been described. They include abrasion damage to the posterior oropharyngeal wall, ulcers of the soft palate and uvula, lingual nerve injury leading to numbness of the tongue, lingual artery compression with subsequent tongue cyanosis, recurrent laryngeal nerve injury leading to hoarseness, hypoglossal nerve injury, and sialadenopathy [8]. These injuries are thought to be caused by traumatic placement, malpositioning, and overinflation of the LMA device. Sialadenopathy, in particular, is postulated to occur as a result of gland and ductal deformation from a malpositioned or overinflated LMA [8]. The large volume of the LMA displaces adjacent tissues and transmits pressure to salivary ducts, leading to obstruction of salivary drainage with resultant gland enlargement and inflammation [6-8]. Using the largest LMA size that is practical for a particular patient is thought to minimize complications, which may minimize the risk for malposition leading to compression injury [8]. However, using a larger LMA without adjustment for cuff volume and correct intracuff pressure may cause more compression to adjacent tissues, potentially leading to complications like the one our patient experienced. The submandibular triangle has been shown to be deformed by the placement of an LMA. Measuring submandibular structures with ultrasonography while changing the LMA intracuff pressure, Ogata et al [9] found that the submandibular glands deformed in a pressuredependent manner when the LMA intracuff pressure was greater than 50 cm H2O. They concluded that intracuff pressure should be readjusted to recommended values to avoid complications involving surrounding tissues. Monitoring cuff pressure, with the goal of maintaining pressure at the lowest level permitting adequate ventilation, may be an effective way of minimizing the risk for injury. This is particularly relevant when nitrous oxide is used because this gas can increase cuff volume by 9% to 38% [8]. Another possible mechanism for sialadenopathy is that an overactive pharyngeal reflex stimulates the salivary glands causing vasodilation and hyperemia [7]. The salivary glands are controlled mainly by parasympathetic nerve signals from the superior and inferior salivatory nuclei in the brain stem. The salivatory nuclei are excited both by taste and tactile stimuli from the tongue and other areas of the mouth and pharynx. The parasympathetic nerve signals induce copious salivation and at the same time dilate the blood vessels. In addition, salivation itself directly dilates
J. Fuhrmeister et al. the blood vessels, thus providing increased blood flow as it is needed. Sympathetic stimulation can also increase salivation moderately, but much less than the parasympathetic stimulation [10]. This would contribute to swelling of the salivary glands if there were salivary ductal obstruction due to the presence of an LMA. Attas et al [1] related sialadenopathy to violent straining and coughing produced by intubation or extubation causing venous engorgement due to an increase in intrathoracic pressure. This results in acute exudation of fluid out of the vascular compartment and rapid enlargement of the gland [1,6]. In our patient, a size 4 Classic LMA was placed based on the patient’s weight and inflated with the recommended air volume. However, the intracuff pressure was not monitored. This relatively large size LMA with recommended volume of air in combination with her small oropharyngeal and hypopharyngeal cavities may have caused an excessive pressure and tissue distortion. The use of nitrous oxide may also have contributed to increased intracuff pressure in the LMA. Increased risk for postoperative sialadenopathy is indicated by her history of tongue swelling after endotracheal intubation, which is consistent with submandibular duct or venous obstruction. It is interesting to note that her most recent two endotracheal intubations with smaller endotracheal tubes were without postoperative sialadenopathy. This again suggests that her small oropharyngeal and hypopharyngeal cavities are likely contributing factors for her increased risk of postoperative sialadenopathy. Although we believe the etiology of our patient’s sialadenopathy was likely mechanical compression, other causes must be considered. As stated above, parasympathetic or sympathetic input can cause salivary gland stimulation. However, the patient did not receive any autonomic drugs, which might have stimulated salivary gland function, and no excessive salivation was observed throughout the procedure. Anesthetic induction and emergence were smooth and uneventful; therefore, venous engorgement by coughing is an unlikely cause. An increase in intrathoracic pressure by straining would affect other organs in a similar manner, but in our case, only the sublingual glands were involved. Sialolithiasis, the presence of salivary calculi, can also lead to inflammation of the salivary glands, but this usually does not occur in the sublingual gland as it lacks a single dominant duct [11]. Finally, we must consider the patient’s history of hypercoagulability and whether venous stasis may have caused the sublingual sialadenopathy. However, the venous drainage of the sublingual glands is provided by the lingual and facial veins, and thrombosis of these veins would have had more widespread implications [11]. The above discussion is focused on the clinical presentation of our patient. However, a much broader range of etiologies, as listed in Table 1, should be considered as possible causes of sialadenopathy under various clinical situations [12,13].
Sialadenopathy of the sublingual glands Table 1
Differential diagnosis of sialadenopathy
Etiology
Examples
Infection
Mumps Cytomegalovirus HIV Staphylococcus aureus Anticholinergics Autonomic stimulation Venous engorgement Sialolithiasis Extrinsic mass Sjfgren’s syndrome Pleomorphic adenoma Mucoepidermoid carcinoma Radiation Surgery Diabetes
Pharmacological/autonomic Obstruction
Immune disorder Neoplasm Xerostomia
The unique feature in our patient is that only the sublingual glands were involved. Based on an extensive literature search, sialadenopathy of the sublingual glands after the use of an LMA has not been reported. Previous reports of sialadenopathy after the use of an LMA involved either submandibular or parotid glands [6,7]. All these LMA-related sialadenopathies share some common features: they are rare complications after the use of an LMA that do not cause appreciable obstruction of the airway and resolve spontaneously without the need for medical intervention.
4. Conclusion Although the LMA is a safe alternative to endotracheal intubation, its use is not without complications. To minimize the risk of local neurovascular compression, one should consider the size of the oropharyngeal cavity and select the size of the LMA appropriately. Vigilance should be maintained in checking the intracuff volume and pressure to avoid excessive pressure to the surrounding tissues. Maintaining the cuff pressure at the lowest level permitting adequate ventilation may be an effective way to minimize the risk for compression injury. The LMArelated sublingual sialadenopathy described in this case report is a painful inflammatory process that resolves on its own over a few days.
5. Comment by Jack D. Sedwick MD The authors present a very unusual case of sialadenopathy of the sublingual glands after an LMA was used to assist with anesthesia in a relatively minor procedure. The patient required two subsequent general anesthetics in which the LMA was not used without subsequent sialadenopathy. Sialadenopathy or nonneoplastic salivary
653 gland disease has many causalities including both acute and chronic inflammatory disorders, acute suppurative sialadenitis, autoimmune causes (eg, Sjfgren’s syndrome), granulomatous diseases, blunt and penetrating trauma, sialolithiasis, pneumoparotitis, radiation sialadenitis, and human immunodeficiency virus (HIV)–associated salivary gland disease. These disorders are broadly defined as inflammatory and noninflammatory. While hundreds of minor salivary glands line the entire oral cavity, the majority of sialadenopathy affects the major salivary glands consisting of the paired parotid, submandibular, and sublingual glands. The parotid and submandibular glands are much more frequently involved in both inflammatory and noninflammatory causes of salivary gland dysfunction than the sublingual glands. This is due to the unique anatomy of the sublingual gland, making this case report an infrequent and unlikely event. The sublingual glands are the smallest of the major salivary glands located in the floor of the mouth below the oral mucosa just adjacent to the submandibular duct. The gland is almost touching the mandible anteriorly and the genioglossus muscle laterally with the sling of the mylohyoid muscle lying directly inferior. Unlike the larger parotid and submandibular glands, a fascial capsule does not surround the sublingual gland. This lack of an adherent fascial capsule makes inflammation of the gland less painful and less restricted than the larger salivary glands. Although the submandibular duct passes between the sublingual gland and the genioglossus, it does not provide an egress for saliva from the sublingual gland. The ducts of Rivinus provide drainage of saliva from the sublingual gland. There are approximately 10 small ducts of Rivinus, which open intraorally along the sublingual fold in most patients. Rarely, the ducts of Rivinus will join to form two larger (one on each side) sublingual (Bartholin’s) ducts. Unlike the longer submandibular duct and Stensen’s duct of the parotid gland, which are between 4 and 6 cm in length, the ducts of the sublingual gland are very short, much less than a centimeter. Anatomically, the multiple small short ducts make obstruction and sialadenopathy a much less common occurrence in the sublingual glands. Because of the large number of short ducts in the sublingual glands, sialadenopathy must be much more severe to cause gland dysfunction and swelling. In this case, with symptoms of sudden onset of diffuse enlargement of a major salivary gland with associated tenderness and no history of sialadenopathy, the diagnosis of acute suppurative sialadenitis must be considered. Even if the cause of the gland dysfunction and swelling is felt to be blunt force trauma or, as in this case, high pressure, management that would more rapidly improve salivary flow and decreased edema of the gland should be instituted to prevent the condition from degenerating into an acute suppurative sialadenitis. Initial treatment of acute sialadenitis should include adequate hydration,
654 improved oral hygiene, repeated massage of the gland, and sialogogues. In addition to these measures, empiric treatment with a penicillinase-resistant anti-staphylococcal antibiotic therapy should be instituted to treat suppurative or developing suppurative sialadenitis cases while awaiting cultures. Inadequate hydration often leads to decreased salivary flow and acute sialadenitis. Acute suppurative disease is often seen in older hospitalized or nursing home patients who are underhydrated. Without appropriate treatment, or in severe cases, acute suppurative sialadenitis can progress to abscess formation. When an abscess is suspected due to a lack of response to therapy or by physical examination, a computed tomographic scan should be preformed. If an abscess is present, it should be drained surgically. In summary, the authors present a very rare case of pressure-induced sialadenopathy of the sublingual glands. Sialadenopathy is a very rare complication of using an LMA, which is usually mildly debilitating and self-limited. I know of no accurate way to predict who might be at risk for this type of complication in the preoperative evaluation. In patients with other risk factors for acute suppurative sialadenitis, such as advanced age and dehydration, it is important to institute massage, hydration, and sialogogues to prevent suppurative sialadenitis in patients with traumatic sialadenopathy.
J. Fuhrmeister et al.
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