a case of anesthesia mumps that required postoperative re ... · congestion, and excess saliva...

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CASE REPORT Open Access A case of anesthesia mumps that required postoperative re-intubation Takayuki Hamaguchi * , Naho Suzuki and Ichiro Kondo Abstract We encountered a 59-year-old man who first underwent left internal carotid endarterectomy for left internal carotid artery stenosis and then presented with postoperative swelling of the bilateral salivary glands. He then developed upper airway obstruction that required emergency tracheal intubation. The most likely cause was thought to be anesthesia mumps, which involves a complex interaction of multiple factors including pneumoparotitis, venous congestion, and excess saliva secretion. Many cases of salivary gland swelling recover after follow-up observation alone if there are no inflammatory findings; however, severe complications may sometimes occur. If upper airway obstruction develops as in the present case, then emergency airway management must also be considered and conscientious observation is necessary. Keywords: Anesthesia mumps, Salivary gland swelling, Parotid gland swelling, Upper airway obstruction Background Anesthesia mumps consists of acute and transient salivary gland swelling caused by general anesthesia. It is an extremely rare postoperative complication and occurs following various surgical procedures. It is usually a self-limiting disease and requires only follow-up observation [1, 2], and in few case reports, evaluation with imaging studies have been performed. We encountered a patient who required emergency tracheal intubation due to upper airway obstruction after anesthesia mumps following an internal carotid artery (ICA) endarterectomy (CEA). We include discussion on imaging evaluationsince preoperative and postoperative neck computed tomography (CT) images were available for this patient partly because neck surgery was performedand a brief literature review. Case presentation A 59-year-old man (170 cm, 78 kg) had cerebral infarction of the right precentral gyrus 2 years prior presentation and was diagnosed with bilateral ICA stenosis. He was treated conservatively (oral cilostazol and clopidogrel) and followed up; however, the stenosis of the left ICA pro- gressed. Therefore, he was scheduled for CEA. There were no apparent complications related to his previous cerebral infarction. He was receiving an oral treatment for hyperten- sion and dyslipidemia and had a long history of smoking (40 cigarettes per day for 40 years). There were no other notable findings in preoperative examinations. On the day of surgery, he was admitted to the operating room without premedication. Pre-oxygenation was performed using a mask without headband. Anesthesia was induced using 200 μg fentanyl and 5 mg midazolam, and 60 mg rocur- onium was given to facilitate tracheal intubation. A Macintosh laryngoscope was used to expose the larynx. The view was classified as Cormack III. We attempted a tracheal intubation with a endotracheal tube (ShileyEndotracheal Tube with TaperGuardCuff 7.5 mm), but the esophagus was inadvertently intubated, so it was removed. The second tracheal intubation by using a Macintosh laryngoscope was successful. Wheals appeared on the upper limbs, neck, and precordium without any vital sign changes after infusion of cefazolin sodium as a pre- operative antibiotic, so administration was discontinued. The skin signs were believed to be an allergic reaction to cefazolin, which was changed to fosfomycin, and the patient also received an intravenous infusion containing 200 mg of hydrocortisone sodium phosphate. During surgery, he was placed in the supine position, but the neck was slightly rotated and lateroflexed to the right to secure the surgical field. Anesthesia was maintained * Correspondence: [email protected] Department of Anesthesiology, Jikei University School of Medicine, Nishi-shinbashi 3-19-18, Minato-ku, Tokyo, Japan © The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. Hamaguchi et al. JA Clinical Reports (2018) 4:22 https://doi.org/10.1186/s40981-018-0159-0

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Page 1: A case of anesthesia mumps that required postoperative re ... · congestion, and excess saliva secretion. Many cases of salivary gland swelling recover after follow-up observation

CASE REPORT Open Access

A case of anesthesia mumps that requiredpostoperative re-intubationTakayuki Hamaguchi* , Naho Suzuki and Ichiro Kondo

Abstract

We encountered a 59-year-old man who first underwent left internal carotid endarterectomy for left internal carotidartery stenosis and then presented with postoperative swelling of the bilateral salivary glands. He then developedupper airway obstruction that required emergency tracheal intubation. The most likely cause was thought to beanesthesia mumps, which involves a complex interaction of multiple factors including pneumoparotitis, venouscongestion, and excess saliva secretion. Many cases of salivary gland swelling recover after follow-up observationalone if there are no inflammatory findings; however, severe complications may sometimes occur. If upper airwayobstruction develops as in the present case, then emergency airway management must also be considered andconscientious observation is necessary.

Keywords: Anesthesia mumps, Salivary gland swelling, Parotid gland swelling, Upper airway obstruction

BackgroundAnesthesia mumps consists of acute and transient salivarygland swelling caused by general anesthesia. It is anextremely rare postoperative complication and occursfollowing various surgical procedures. It is usually aself-limiting disease and requires only follow-up observation[1, 2], and in few case reports, evaluation with imagingstudies have been performed.We encountered a patient who required emergency

tracheal intubation due to upper airway obstruction afteranesthesia mumps following an internal carotid artery (ICA)endarterectomy (CEA). We include discussion on imagingevaluation—since preoperative and postoperative neckcomputed tomography (CT) images were available for thispatient partly because neck surgery was performed—and abrief literature review.

Case presentationA 59-year-old man (170 cm, 78 kg) had cerebral infarctionof the right precentral gyrus 2 years prior presentationand was diagnosed with bilateral ICA stenosis. He wastreated conservatively (oral cilostazol and clopidogrel) andfollowed up; however, the stenosis of the left ICA pro-gressed. Therefore, he was scheduled for CEA. There were

no apparent complications related to his previous cerebralinfarction. He was receiving an oral treatment for hyperten-sion and dyslipidemia and had a long history of smoking(40 cigarettes per day for 40 years). There were no othernotable findings in preoperative examinations. On the dayof surgery, he was admitted to the operating room withoutpremedication. Pre-oxygenation was performed using amask without headband. Anesthesia was induced using200 μg fentanyl and 5 mg midazolam, and 60 mg rocur-onium was given to facilitate tracheal intubation. AMacintosh laryngoscope was used to expose the larynx.The view was classified as Cormack III. We attempteda tracheal intubation with a endotracheal tube (Shiley™Endotracheal Tube with TaperGuard™ Cuff 7.5 mm),but the esophagus was inadvertently intubated, so itwas removed. The second tracheal intubation by using aMacintosh laryngoscope was successful. Wheals appearedon the upper limbs, neck, and precordium without any vitalsign changes after infusion of cefazolin sodium as a pre-operative antibiotic, so administration was discontinued.The skin signs were believed to be an allergic reaction tocefazolin, which was changed to fosfomycin, and thepatient also received an intravenous infusion containing200 mg of hydrocortisone sodium phosphate. Duringsurgery, he was placed in the supine position, but theneck was slightly rotated and lateroflexed to the rightto secure the surgical field. Anesthesia was maintained

* Correspondence: [email protected] of Anesthesiology, Jikei University School of Medicine,Nishi-shinbashi 3-19-18, Minato-ku, Tokyo, Japan

© The Author(s). 2018 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0International License (http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, andreproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link tothe Creative Commons license, and indicate if changes were made.

Hamaguchi et al. JA Clinical Reports (2018) 4:22 https://doi.org/10.1186/s40981-018-0159-0

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with 1.4% sevoflurane and remifentanil at a dose of0.2 μg/kg/min. Mild hypotension was observed intraop-eratively. This was managed with fluid infusions andcontinuous administration of an appropriate dose ofnoradrenaline. Surgery was concluded without problems,and the preoperatively observed wheals resolved. Endo-tracheal aspiration was performed after waking up thepatient, but it triggered a strong cough reflex. The patientwas then extubated. The duration of surgery was 4 h and57 min, the duration of anesthesia was 6 h and 29 min,the volume of blood loss was negligible, the in-out balancewas + 3210 mL (crystalloid fluid 4175 mL, urine volume965 mL), and mild swelling of the face and both upperlimbs was noted. The patient was admitted to the inten-sive care unit (ICU) in a lucid state with a blood pressureof 120/70 mmHg, a heart rate of 110 beats/min, oxygensaturation of 100% on 3 L of nasal oxygen and respiratoryrate of 12 breaths/min. His arterial blood gas analysis wasnormal, and mild hoarseness was noted without swellingof the neck or stridor. Six hours after admission to theICU mild bilateral neck swelling appeared. The hoarsenessrapidly worsened, and tachypnea and stridor appeared 1 hlater without oxygen desaturation. Emergency intubationwith a tube (Shiley™ Evac Endotracheal Tube withTaperGuard™ Cuff 7.5 mm) was immediately performedwith mild sedation with propofol under spontaneousbreathing on suspicion of upper airway obstruction. Weused a video laryngoscope (HOYA Co. Ltd., airwayscope) with a gum elastic bougie tube introducer.Laryngopharyngeal findings at this time included noepiglottic edema. However, the edema of the lateral andposterior pharyngeal walls was present, and it narrowedthe oral and pharyngeal cavities. The neck swelling waspronounced, and the neck circumference was 63 cm(Fig. 1). A CT scan was performed to investigate thecause and to facilitate differential diagnosis of possiblepostoperative hemorrhage, but no bleeding was ob-served. The bilateral parotid and salivary glands weremarkedly swollen (Fig. 2), and the edema of the poster-ior pharyngeal wall was developed (Fig. 3) when com-pared to the preoperative state. Blood test resultsrevealed a leukocyte count of 8000/μL (eosinophils 0%),amylase at 1790 U/L, mumps immunoglobulin G (IgG)at 16.6 (+), mumps IgM at 0.27 (−), and C-reactiveprotein at 0.18. Based on these findings, we concludedthat the patient had previously suffered from a mumpsinfection, but had no active disease. Moreover, he onlypresented with the clinical features of sialadenitis. Wecommenced administration of steroids to reduce theedema. On postoperative day (POD) 2, the serum amylasedecreased to 457 U/L, gradual improvement of the bilateralparotid gland swelling was observed, and the patient wasextubated. The subsequent clinical course was favorable,and the neck swelling disappeared. The general condition,

airways, and ability to swallow were all normal when thepatient was discharged on POD 11.

ConclusionsIn this case, emergency intubation was performedbecause upper airway obstruction strongly suspected dueto expiratory stridor and rapidly worsen dyspnea. His neckwas bilaterally swelling and CT showed the bilateral parotidglands swelling and the edema of the posterior pharyngealwall without hematoma around surgical site. Blood testindicated serum amylase was abnormally elevated. Thus, wediagnosed upper airway obstruction due to the anesthesiamumps. Furthermore, he developed an allergic reaction,which might have caused or intensified the edematouschanges in the pharyngeal wall. This might be one ofthe etiologies that caused upper airway obstruction.Anesthesia mumps is considered to be an extremely

rare complication. According to reports in the literature,its occurrence is highly variable (0.2–17%) [3–5], andthe exact incidence remains unknown. Although patientscan develop postoperative salivary gland swelling, it istypically mild, with few subjective symptoms. Therefore,many cases resolve spontaneously without being noticed.Thus, we believe that the discrepancies in the incidencerate are largely due to variable amount of attention thatis paid to this condition.The cause and detailed pathophysiology of anesthesia

mumps remains unknown, but salivary duct occlusion,sialorrhea, venous congestion and venostasis, involvementof the autonomic nerves, and side effects of medicationshave been suggested [5, 6].Causes of salivary gland obstruction include (1) physical

compression by lateral position, positions that rotate and

Fig. 1 Photograph showing the swelling of his neck 7 h afterthe operation

Hamaguchi et al. JA Clinical Reports (2018) 4:22 Page 2 of 4

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flex the neck, compression by endotracheal tubes, mucosallesions and edema; (2) pneumoparotitis induced by thepenetration of air as a result of retrograde flow into thesalivary duct due to positive pressure in the oral cavityby mask ventilation, pharyngeal reflex, and cough reflex;and (3) dehydration, administration of atropine, and sym-pathetic nervous system activation due to invasiveness ofsurgery causing increased salivary viscosity, which may inturn itself cause an occlusion.Salivation is mediated by the autonomic nerves, and the

main secretory innervation is parasympathetic. Intratra-cheal manipulation stimulates parasympathetic nerves thatmediate the pharyngeal reflex, which promotes salivationand leads to vasodilation and hyperemia in the salivarygland [2]. Stimulation of the sympathetic nerves alsoevokes salivation, but it is relatively short-lasting, the saliva

is usually thick and mucinous, and vasoconstriction occurs[7]. Actually, noradrenaline infusion increases salivaryalpha-amylase, a digestive enzyme secreted from thesalivary glands that has been proposed as a sensitivesurrogate maker for activity stress [8].In the present case, we observed bilateral swelling of

the parotid and submandibular glands. Therefore, weconsidered it unlikely that there was only a mechanicalregional occlusion related to patient positioning andpneumoparotitis. We believe that variety of causes ledto development of anesthesia mumps. One of them is avenous congestion due to surgical procedure or stimuliof CEA, and the others are excess secretion of saliva dueto reflex salivation, the pharyngeal reflex or continuousadministration of noradrenaline. However, the exactcauses remain unclear.

Fig. 2 Post-operative image of computed tomography scans at the parotid glands level shows the bilateral parotid glands were swollencompared to the pre-operative state

Fig. 3 Post-operative image of computed tomography scans shows the edema of the posterior pharyngeal wall was developed compared to thepre-operative state

Hamaguchi et al. JA Clinical Reports (2018) 4:22 Page 3 of 4

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Several reports state that most cases of anesthesiamumps resolve spontaneously with follow-up observationalone, it has been considered that rehydration therapy andanti-inflammatory drugs if needed are sufficient to treat it[1, 2]. However, there may be cases, such as the presentcase, in which patients suffer from severe complications,including upper airway obstruction [9, 10]. In our patient,the combined approach of a video laryngoscope and agum elastic bougie tube introducer resulted in a successfulre-intubation without the need for tracheostomy.Emergency tracheostomy might be often chosen because ofdifficult mask ventilation and the possibility of intubationdifficulty for postoperative dyspnea and acute airwayobstruction [10]. However, in cases involving massiveedema and swelling of the neck, it is difficult to identify thelocation of the tracheostomy. Video laryngoscopes couldwell become the standard procedure for these types ofpatients who need emergency tracheal intubation. Hence,additional measures to treat these conditions are required,and conscientious follow-up observation is necessary.

AbbreviationsCEA: Carotid endarterectomy; CT: Computed tomography; ICA: Internalcarotid artery; ICU: Intensive care unit; POD: Postoperative day

FundingNo funding

Authors’ contributionsTH wrote this manuscript. NS helped to revise this manuscript. IK supervised toshape this manuscript. All authors read and approved the final manuscript.

Ethics approval and consent to participateNot applicable

Consent for publicationWritten informed consent was obtained from the patient for publication ofthis case report and accompanying images.

Competing interestsThe authors declare that they have no competing interests.

Publisher’s NoteSpringer Nature remains neutral with regard to jurisdictional claims inpublished maps and institutional affiliations.

Received: 19 December 2017 Accepted: 20 February 2018

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5. Matsuki A, Wakayama S, Oyama T. Acute transient swelling of the salivaryglands during and following endotracheal anaesthesia. Anaesthesist. 1975;24:125–8.

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9. Franco C, Giorgio C, Maria GA, Angelo G, Alessandro G, Rodolfo P. Massivefacial edema and airway obstruction secondary to acute postoperativesialadenitis or “anesthesia mumps”: case report. J Med Case Rep. 2009;3:7073.

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