International Journal of Pediatric Otorhinolaryngology (2006) 70, 941—946 www.elsevier.com/locate/ijporl CASE REPORT Three extraordinary complications of adenotonsillectomy Michael J. Reilly a,b,*, Gregory Milmoe a,b, Maria Pena a,b a Georgetown University Hospital, Otolaryngology — Head & Neck Surgery, 3800 Reservoir Rd., Washington, DC 20007, USA b Children’s National Medical Center, 111 Michigan Ave NW, Washington, DC 20010, USA Received 25 June 2005; received in revised form 25 September 2005; accepted 26 September 2005 KEYWORDS Adenotonsillectomy; Complication; Atlanto-axial subluxation; Cerebral venous thrombosis; Electrocautery malfunction; Oropharyngeal burn; Oral cavity burn Summary In a 6-month period, three patients aged 5—11 years were transferred to our tertiary care children’s hospital for management of severe complications following adenotonsillectomy. The first patient presented with headaches and lethargy and was found to have a sagittal sinus thrombosis from severe dehydration. The second patient was admitted immediately following an intra-operative oral cavity fire due to electrocautery malfunction. She suffered partial-thickness burns to the buccal mucosa, palate, and lips. The third patient was admitted with torticollis. Grisel’s syndrome was initially suspected, but a thorough work up resulted in the diagnosis of a conversion disorder. These cases comprise an interesting cohort of three little-known complications of adenotonsillectomy. # 2005 Elsevier Ireland Ltd. All rights reserved. 1. Introduction Each year in the United States, adenotonsillectomy is performed in nearly 300,000 children age 15 years and under, making it one of the most common procedures practiced in American medicine [1]. Adenotonsillectomy is a relatively simple procedure and has been thoroughly studied. Known complications have been reported throughout the literature. In one study, Colclasure and Graham studied over 3000 patients undergoing adenotonsillectomy and published a major complication rate of 1.4% [2]. This included hemorrhage, anesthetic * Corresponding author. Tel.: +1 202 641 3260. E-mail address: mikereillydc@gmail.com (M.J. Reilly). complications, severe nausea, and dehydration. Further well-established complications of adenotonsillectomy include infection, velopharyngeal insufficiency, nasopharyngeal stenosis, Eagle’s Syndrome, taste disturbance, and Grisel’s syndrome [3,4]. Goins and Pitovski have suggested that adenotonsillectomy complications be characterized depending on the timing of their occurrence in relation to surgery: intra-operative, immediate postoperative (<24 h), delayed (<2 weeks), and long-term [5]. The cases discussed in this presentation identify three additional complications to be considered in pre-operative counseling, intra-operative decisionmaking, and postoperative management of adenotonsillectomy patients. 0165-5876/$ — see front matter # 2005 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2005.09.023 942 M.J. Reilly et al. 2. Case reports 2.1. Case one A 7-year-old girl underwent uncomplicated adenotonsillectomy for chronic adenotonsillitis. Two days later, she was re-admitted for a week-long hospital stay for decreased oral intake and dehydration. She was discharged home on postoperative day 9, but continued to have poor oral intake. On postoperative day 11, the patient presented to her local Emergency Department with sudden-onset drooling, tonic head-tilting, and unresponsiveness. She had also undergone a full-body tonic-clonic episode en route to the hospital. Upon arrival, the patient required intubation for airway protection. Head CT scan was performed, which was negative. The patient was stabilized on the ventilator and transferred to a tertiary care children’s hospital for further management. Neurological work-up was initiated. EEG revealed an abnormal focus of activity in the left frontal lobe. The patient was started on anti-epileptics, and MRI/MRV was done because of continued headaches. It revealed cortical infarction involving the bilateral frontal lobes and a sinus venous thrombosis of the anterior superior sagittal sinus. Hematological work-up for hypercoaguability was negative, and the patient was not placed on thrombolytics. This patient continued to refuse oral hydration, and a nasogastric tube was placed. She was evaluated by speech therapy, who documented a delayed oropharyngeal swallow and continued supplementation with liquid food alternatives via Fig. 1 nasogastric tube. On postoperative day 25, the patient was discharged home in stable condition with no evidence of residual neurological impairment (Figs. 1 and 2). 2.2. Case two A 5-year-old girl underwent adenotonsillectomy for sleep-disordered breathing. The procedure was done under general anesthesia with a 4.0 uncuffed endotracheal tube. Intra-operatively, mucosal burns were noted on the superior lip, posterior buccal mucosa, soft palate, and hard palate after a reported ‘‘spark’’ from the electrocautery instrument. The patient was awakened and extubated without difficulty. In the ensuing hours, there was an increase in oral edema, specifically involving the soft and hard palates. The patient was transferred to our tertiary care children’s hospital for further management. Upon arrival in the Emergency Department, the patient was nasotracheally intubated for airway protection. Examination of the oral cavity revealed partial-thickness burns to the soft palate, hard palate, and superior lip, but not involving the oral commisures. There were also partial-thickness burns to the posterior buccal mucosa bilaterally. No other abnormalities were noted. The patient was managed conservatively with artificial ventilation, sedation, and hydration. On postoperative day 6, the patient was taken to the operating room for examination and extubation. The soft palate and buccal mucosa had remucosalized. The hard palate was improving, but had Sagittal MRV depicting thrombosis of the anterior half of the superior sagittal sinus. Three extraordinary complications of adenotonsillectomy Fig. 2 943 Noncontrast axial T1 MRI demonstrating cortical infarctions of the right and left frontal lobes. continued evidence of partial-thickness burn with white exudate. The superior lip had small areas of partial-thickness burns with the majority demonstrating epithelialization. The pharynx, nasopharynx and hypopharynx were completely normal. On the basis of these findings, the patient was awakened from anesthesia and extubated. Serial debridements and applications of bacitracin were utilized for wound care. The patient showed continued progress and her diet was advanced slowly to regular. She was discharged home on postoperative day 17 in stable condition. Six-month follow-up revealed complete healing of all burns other than persistence of a small scar of her right superior lip. No operative intervention has been chosen at this time. to identify an etiology of the patient’s torticollis. Laboratory studies were normal and radiographic imaging of the neck revealed no atlanto-axial or C2—C3 subluxation during rotational movement. Upon further observation of the patient and discussion with the family, it was found that the torticollis was intermittent, often associated with the anticipation of medical intervention, and absent during sleep. As the sporadic nature of the torticollis was in clear violation of known physiology, a psychiatric etiology was considered. The patient was ultimately diagnosed with a conversion disorder based on DSM-IV criteria. He was discharged home on postoperative day 28 (hospital day 4). At the time of his 6-week follow-up, the patient’s pain and torticollis had resolved completely (Figs. 3 and 4). 2.3. Case three An 11-year-old boy presented to the Emergency Department at his local hospital with torticollis and trismus. Computed tomography revealed a peritonsillar abscess, which resolved with intravenous followed by oral antibiotic therapy. Four weeks later, the patient underwent an uneventful tonsillectomy and was discharged home in stable condition the same day. By report, the patient did well until postoperative day 25 when he presented to our tertiary care children’s hospital for evaluation of neck pain and torticollis. Grisel’s syndrome was considered, and consultations were placed with the orthopedic, neurology, and neurosurgical services. None of the consulting experts were able 3. Discussion 3.1. Case one The suspected etiology of this patient’s superior sagittal sinus thrombosis was severe dehydration in the first 2 weeks after adenotonsillectomy. Cerebral venous thrombosis (CVT) is associated with a known risk factor in 77% of cases, mostly related to an alteration of cerebral hemodynamics. Dehydration, shock, congestive heart failure, systemic sepsis, and local infection (perioral, mastoid, periorbital) are all associated with an increased risk of CVT [6,7]. Inherited hypercoagulable disorders are 944 Fig. 3 M.J. Reilly et al. CT scan of a different patient, revealing the classic atlanto-axial subluxation of Grisel’s syndrome [4]. only present in one-third of children with CVTs. In this patient, hypercoagulability work-up was negative. CVT can present with a broad spectrum of symptoms, including headache, vomiting, seizures, or coma. The mainstay of treatment is anticoagulant therapy with a target INR of 2.5 for 3—6 months, even in the presence of hemorrhagic infarction [8]. Fig. 4 CT scan of our patient revealing appropriate movement of C1 on C2. Normal craniocervical and C2—C3 junctions were demonstrated as well. The architecture of the vertebral bodies was normal. Soft tissues were unremarkable and the intervertebral disc spaces maintained their height. Three extraordinary complications of adenotonsillectomy Of interest is the notion that the patient’s difficulty with the oropharyngeal phase of swallowing may have been exacerbated by neurologic deficits from the cerebral venous thrombosis. However, the absence of any long-term sequelae in this patient is consistent with previous reports of successful outcomes in children with CVT. 3.2. Case two Malfunction of the electrocautery instrument is the most likely mechanism of injury causing the partialthickness burns on this child’s posterior buccal mucosa, hard palate, soft palate, and superior lip. Ignition of the anesthetic agent was also considered as a possible etiology as there have been published reports of airway fire during the use of electrocautery [9]. However, these incidents occurred during tracheostomy procedures where there was direct exposure of concentrated inhalational anesthetic agent upon incising the trachea. On the contrary, there is minimal leak of anesthetic agent into the oral cavity with an uncuffed pediatric tracheostomy tube, making this an unlikely explanation. Still, the anesthesiologist and surgeon should be cognizant of the combustibility of an inhalational anesthetic with a high fraction of inspired oxygen. This is especially important when electrocautery will be used in any procedure involving the airway. Electrical burns of the mouth generally result in significant edema and progressive enlargement in the hours following the initial insult. While early excision of the wound with primary closure may be possible in some instances, this approach carries increased risk of wound dehiscence and is dependent on the size of the involved area [10]. The conservative approach was selected for this patient, which consists of cleansing the area, keeping it moist, and assessing the extent of healing at 7—14 days. If necessary, surgical reconstruction may be considered at that time. Whenever the oral commisure is involved in an oral cavity burn, the use of a microstomia prevention appliance is indicated. The rapid wound healing and the absence of oral commisure involvement obviated the need for surgery or microstomia appliance. 3.3. Case three Initially, the suspected etiology of this patient’s neck pain and torticollis 4 weeks after tonsillectomy and 8 weeks after peritonsillar abscess was atlantoaxial subluxation (AAS), also known as Grisel’s syndrome. Upon further evaluation, however, the torticollis was noted to be intermittent and absent 945 during sleep. CT scan of the neck demonstrated no atlanto-axial or C2—C3 subluxation during rotational movement. The diagnosis of a conversion reaction then became the most likely etiology for this child’s torticollis. The diagnosis of conversion disorder cannot be made without demonstrating that symptomatology clearly violates the laws of anatomy and physiology [11]. In this case, the absence of AAS was not enough to assert this diagnosis. A retrospective review by Mezue et al. of 13 children with torticollis and fever demonstrated that only 3/13 had radiographic findings consistent with AAS [12]. They theorized that inflammatory torticollis may be a precursor to AAS. However, the intermittent nature of this patient’s torticollis, its absence during sleep, and its association with impending medical intervention are clear violations of the known pathophysiology of inflammatory torticollis. The patient’s prior history of true inflammatory torticollis is notable in that conversion disorder is usually characterized by symptoms that corresponds with the patient’s particular conception of how an illness might manifest itself, either from personal experience or observation of another. It is important to remember that unlike malingerers, patients with conversion disorder do not intentionally feign their symptoms. 4. Conclusion There is limited information in the English literature about unusual complications of adenotonsillectomy. Three such cases are presented. All complications occurred at different local facilities and were transferred to our tertiary care children’s hospital for further management. 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