International Journal of Pediatric Otorhinolaryngology 131 (2020) 109885
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Case Report
Subcutaneous emphysema with pneumomediastinum after tonsillectomy: Case report and review of the literature
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Jenna H. Barengoa, Sonia N. Yuenb, Patrick Kennedyb, Sally R. Shottb,c,∗ a
University of Cincinnati College of Medicine, Cincinnati, OH, USA Department of Otolaryngology-Head & Neck Surgery, University of Cincinnati Medical Center, Cincinnati, OH, USA c Department of Pediatric Otolaryngology-Head and Neck Surgery, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA b
A R T I C LE I N FO
A B S T R A C T
Keywords: Tonsillectomy Down syndrome Subcutaneous emphysema Subcutaneous crepitus Pneumomediastinum
Subcutaneous emphysema is a rare complication of tonsillectomy.We report a case of post-tonsillectomy crepitus with radiographic extravasation of contrast from the tonsil fossa into the neck, subcutaneous emphysema, pneumomediastinum and small pneumothorax in a patient with Down Syndrome. Subsequent direct laryngoscopy showed no visible defect in the mucosal or muscle layers of the tonsil fossa. We explore common presenting symptoms, clinical course, and treatment of subcutaneous emphysema secondary to tonsillectomy.
1. Introduction Tonsillectomy is one of the most common pediatric procedures, with over 350,000 performed annually in the United States [1]. The procedure has an overall complication rate of 8–9%. Respiratory compromise, bleeding, pain, and secondary dehydration are the most common complications [2–4]. Subcutaneous emphysema is a rarely reported complication, first mentioned in literature in 1910 [5]. Published reports point to underlying tonsillar fossa muscle injury as a cause of this complication, but the mechanism is poorly understood [6,7]. 2. Case report We present a three-year-old male with a history of Down syndrome, partial atrioventricular canal repaired five months prior to tonsillectomy, gastroesophageal reflux disease, possible celiac disease, chronic otitis media with effusion, obstructive sleep apnea, and tonsillar hypertrophy. He underwent a tonsillectomy, left ear exam under anesthesia, right ear myringotomy with tympanostomy tube insertion, and esophagogastroduodenoscopy (EGD) with biopsies. EGD was performed initially by the Gastroenterology service. Tonsillectomy was performed in standard fashion with bovie electrocautery, with minimal bleeding and no pharyngeal fat exposed. The senior author's routine technique at the completion of tonsillectomy is to release the mouth gag, palpate the neck, and then re-examine the tonsillar fossae for bleeding. No crepitance was palpated during this maneuver. Emergence
from general anesthesia was without complication with no need for extended positive pressure ventilation. In the post-anesthesia care unit, family noted swelling around the jaw and left cheek, which had not been appreciated while patient was in the operating room. The decision was made to observe the swelling during his planned overnight observation due to his history of Down Syndrome and obstructive sleep apnea. Overnight, there was increased swelling and crepitus extending from the temples to the clavicles, with the left greater than the right side. There was no cardiorespiratory compromise. Chest x-ray (CXR) showed extensive subcutaneous emphysema of the neck and face, with evidence of pneumomediastinum and a small pneumothorax (Fig. 1). Nasopharyngeal flexible endoscopy showed soft palate swelling, but a patent airway in the posterior oropharynx, supraglottis and glottis. The decision was made to place the patient on supplemental oxygen, start intravenous piperacillin-tazobactam (Zosyn), and perform serial physical exams. On post-operative day (POD) one, an upper gastrointestinal fluoroscopy revealed extravasation of barium contrast into the neck at the level of the left tonsillar fossa. On POD two, he was taken to the operating room to place a nasogastric tube and examine the tonsillar fossa, oropharynx and hypopharynx. No abnormalities were seen - no unexpected mucosal trauma, no evidence of underlying muscle violation in the tonsillar fossae, and no obvious air escape from the tonsillar fossae on manipulation. Over the next 72 h, facial swelling decreased, and crepitus subsided. Daily CXRs showed improving pneumomediastinum. The apical
∗ Corresponding author. Department of Pediatric Otolaryngology-Head and Neck Surgery, Cincinnati Children's Hospital Medical Center, Ohio. 3333 Burnet Avenue, Cincinnati, OH, 45229, USA. E-mail address:
[email protected] (S.R. Shott).
https://doi.org/10.1016/j.ijporl.2020.109885 Received 26 September 2019; Received in revised form 12 January 2020; Accepted 13 January 2020 Available online 16 January 2020 0165-5876/ © 2020 Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 131 (2020) 109885
J.H. Barengo, et al.
Fig. 1. Lateral (a) and anterior-posterior (b) chest x-rays obtained 18 hours after tonsillectomy. There is extensive pneumomediastinum, small left apical pneumothorax, subcutaneous air within the neck, and patchy atelectasis. Sternotomy wires are intact from cardiac surgery five months prior.
during the procedure or during the examination under anesthesia on POD two. We suspect that an undetected injury in the tonsillar bed allowed air to track into the neck when the patient was ventilated with positive pressure at the time of extubation, or during post-operative coughing and other Valsalva maneuvers. The patient had a previous sternotomy to repair an atrioventricular septal defect and cleft atrioventricular valve five months prior to this tonsillectomy. It is unlikely that this was a contributing factor to the development of subcutaneous emphysema as there were no issues with wound healing and no residual defect. A literature review performed through PubMed identified 30 case reports, written in English and with full texts available, of subcutaneous emphysema following tonsillectomy (Table 1). The most common treatment was with antibiotics and observation. For our patient, we felt it was important to bypass the area of the injury with a feeding tube until healing was complete in order to minimize the risk of infection. We also made daily skin markings of the spread and resolution of the subcutaneous air and used serial CXRs to confirm the pneumomediastinum was resolving. Most reported cases resolved spontaneously without need for intervention. Three cases required intubation of the patient [8,14,15], and one patient required a thoracotomy [16]. Down Syndrome was a comorbidity in one other report following an uncomplicated adenotonsillectomy. In this case the child had a very serious postoperative course of bacterial mediastinitis, and cardiopulmonary arrest requiring resuscitation [8]. This literature review does not suggest that patients with Down Syndrome are more susceptible to subcutaneous emphysema following tonsillectomy.
pneumothorax resolved within 24 h. At no time did patient have any respiratory difficulty. He was discharged on POD five in stable condition with his nutrition through the nasogastric tube, and on two weeks of amoxicillin-clavulanate (Augmentin) based on Infectious Disease recommendations. Follow-up upper gastrointestinal fluoroscopy series, performed POD ten, showed no evidence of contrast extravasation into the neck, and the feeding tube was removed with no feeding difficulty. He has had no residual deficits. 3. Discussion Subcutaneous emphysema and pneumomediastinum have been described in the literature as rare complications following tonsillectomy. Though the mechanism of air entry into the subcutaneous space is not yet known, most reports describe one of two likely mechanisms. The first is rupture along the tracheobronchial tree from an alveolar bleb or traumatic intubation, leading to air extravasation into the mediastinum that tracks into the subcutaneous space [8–10]. Rupture may occur in the hours and days following the procedure with sudden increase in intrathoracic pressure such as from coughing, vomiting, or Valsalva maneuvers. The second proposed mechanism is injury to superior constrictor muscle and deep cervical fascia through the tonsillar bed, allowing air into the pharyngeal neck space [8,9,11,12]. Once air is in the pharyngeal space, it can track through to the connected mediastinum, causing pneumomediastinum [10]. This injury can occur during the removal of tonsils and likely occurs more frequently when dissection is difficult, adhesions are present, or deep dissection planes are necessary [6,7,13]. If injury occurs during the procedure, complications are most likely seen during the procedure or immediate postoperative period. Increased pressure or irritation to a healing tonsillar bed, such as from positive pressure ventilation or vomiting, is more likely to cause injury in the days following tonsillectomy [8,9]. In our case, intubation and/or EGD performed prior to tonsillectomy may have placed increased pressure on the tracheobronchial tree. However, our examination of the oropharynx and hypopharynx did not find trauma to the mucosa of the upper airway. The injury most likely occurred within the left tonsillar bed given the barium swallow indicated extravasation at that level, though no gross injury was noted
4. Conclusion Subcutaneous emphysema with pneumomediastinum is a rare complication of tonsillectomy. If subcutaneous air and crepitus develop after tonsillectomy, treatment should include antibiotics, examination to identify possible sites of injury, and other appropriate interventions. Conservative treatment is usually all that is needed with close observation and antibiotics.
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International Journal of Pediatric Otorhinolaryngology 131 (2020) 109885
J.H. Barengo, et al.
Table 1 Published case reports on post-tonsillectomy emphysema and their management. Authors
Year Published
Age (years)
Gender
Symptoms
Treatment
Parish Baker Knutson and Oulette Ferguson et al. Anderson Prupas et al. Hampton et al. Hampton et al. Braverman et al. Miman et al. Nishino et al. Marioni et al. Watanabe et al. Stewart et al. Yammine et al.
1910 1936 1954 1955 1958 1977 1997 1997 1997 2001 2003 2003 2004 2004 2004
28 27 6 4 8 22 18 33 22 11 55 34 24 22 36
M F M F F M F F F M F F M F M
observation observation antibiotics thoracotomy, transfusion antibiotics observation observation antibiotics, observation observation suture repair of tonsillar fossae, packing, antibiotics antibiotics antibiotics antibiotics antibiotics antibiotics
Lima et al. Patel et al. Shine et al. Panerari et al.
2005 2005 2005 2005
25 31 7 33
M M F F
swelling, cyanosis cough, swelling unknown swelling, tachycardia, pallor, dyspnea swelling, vomiting, retching, fever swelling swelling swelling dyspnea swelling, sore throat, chest pain, dyspnea swelling swelling swelling swelling swelling swelling, dysphagia decreased neck motion swelling swelling, dyspnea swelling Dyspnea, stridor
Hung et al. Kim et al. Koukoutsis et al. Yelnoorkar et al. Bizaki et al. Tran et al. Erol et al. Crosbie et al. Saravakos et al. Mahmood et al. Duvekot et al.
2009 2010 2013 2014 2014 2015 2016 2017 2017 2018 2019
37 36 21 18 29 30 4 12 21 40 22 mo
M F M M F F M F M F M
swelling, crepitus swelling, pain swelling, pain swelling swelling, sore throat, pressure Neck pain, cough swelling, gagging, vomiting swelling pain, dysphagia swelling swelling swelling, increased fluid intake
Financial disclosures
observation antibiotics, observation oxygen, antibiotics, intravenous fluids intubation, surgical closure of muscular planes, tracheostomy, antibiotics intubation, antibiotics antibiotics antibiotics, observation antibiotics antibiotics antibiotics, observation observation antibiotics, observation antibiotics, cough suppressants, laxatives antibiotics, intravenous steroids antibiotics, inotropes, intravenous immunoglobulin, intubation
pneumomediastinum after tonsillectomy, Am. J. Otolaryngol. 31 (2010) 212–215. [7] A.E. Stewart, D.F. Brewster, P.E. Bernstein, Subcutaneous emphysema and pneumomediastinum complicating tonsillectomy, Arch. Otolaryngol. Head Neck Surg. 130 (11) (2004) 1324–1327. [8] A. Duvekot, G. van Heesch, L. Veder, Subcutaneous and mediastinal emphysema followed by group A beta-hemolytic Streptococci mediastinitis. A complicated course after adenotonsillectomy: case report, Diagnostics 9 (1) (2019 Jan) E11. [9] P. Saravakos, M. Taxeidis, I. Kastanioudakis, O. Reichel, Subcutaneous emphysema as a complication of tonsillectomy: systematic literature review and case report, Iran. J. Otorhinolaryngol. 30 (2018) 3–10. [10] D.D. Tran, P.D. Littlefield, Late presentation of subcutaneous emphysema and pneumomediastinum following elective tonsillectomy, Am. J. Otolaryngol. 36 (2) (2015) 299–302. [11] N.P. Shine, C. Sader, H. Coates, Cervicofacial emphysema and pneumomediastinum following pediatric adenotonsillectomy: a rare complication, Int. J. Pediatr. Otorhinolaryngol. 69 (11) (2005) 1579–1582. [12] O. Ero, E. Aydin, A rare complication of tonsillectomy: subcutaneous emphysema, Turk. Arch. Otolaryngol. 54 (2016) 172–174. [13] A. Bizaki, J. Kaariainen, T. Harju, M. Rautiainen, Facial subcutaneous emphysema after tonsillectomy, Head Face Med. 10 (2014) 11. [14] A.C. Panerari, A.C. Soter, et al., Onset of subcutaneous emphysema and pneumomediastinum after tonsillectomy: a case report, Braz. J. Otorhinolaryngol. 71 (1) (2005) 94–96. [15] M.H. Hung, P.Y. Shih, et al., Cervicofacial subcutaneous emphysema following tonsillectomy: implications for anesthesiologists, Acta Anaesthesiol. Taiwanica 47 (3) (2009) 134–137. [16] C.C. Ferguson, P.M. McGarry, I.H. Beckman, M. Broder, Surgical emphysema complicating tonsillectomy and dental extraction, Can. Med. Assoc. J. 72 (11) (1955 Jun) 847–848.
None to disclose. Declaration of competing interest None to disclose. References [1] C.A. Steiner, Z. Karaca, B.J. Moore, M.C. Imshaug, G. Pickens, Surgeries in HospitalBased Ambulatory Surgery and Hospital Inpatient Settings, Agency for Healthcare Research Quality, Rockville, MD, 2014 HCUP Statistical Brief #223. May 2017, revised Feb 2018 https://hcup-us.ahrq.gov/reports/statbriefs/sb223-AmbulatoryInpatient-Surgeries-2014.jsp. [2] J.L. Paradise, C.D. Bluestone, D.K. Colborn, B.S. Bernard, H.E. Rockette, M. KursLasky, Tonsillectomy and adenotonsillectomy for recurrent throat infection in moderately affected children, Pediatrics 110 (1 Pt 1) (2002) 7. [3] J. Granell, P. Gete, M. Villafruela, C. Bolaños, Vicent, Safety of outpatient tonsillectomy in children: a review of 6 years in a tertiary hospital experience, J. Otolaryngol. Head Neck Surg. 131 (4) (2004) 383. [4] D.L. Canto, C. Pachêco-Pereira, et al., Adenotonsillectomy complications: a metaanalysis, Pediatrics 136 (4) (2015) 702–718 Oct. [5] B.D. Parish, A case of subcutaneous surgical emphysema. An unusual complication following the removal of faucial tonsils, The Laryngoscope 20 (11) (1910 Nov) 1046–1048. [6] J.P. Kim, J.J. Park, H.S. Kang, M.S. Song, Subcutaneous emphysema and
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