Subcutaneous emphysema and pneumomediastinum after endoscopic transnasal removal of nasopharyngeal angiofibroma

Introduction. Juvenile nasopharyngeal angiofibroma is a rare, benign, well-vascularized tumor of the skull base characterized by destructive growth. The development of endoscopic techniques and experience of surgeons have enabled the removal of this tumor both at early stages and late stages (advanced disease). Patients may develop various complications in the intraoperative and postoperative periods, including massive bleeding, nasal liquorrhea, facial paresthesia, lacrimal hyposecretion, etc. Air penetration into the subcutaneous fat and mediastinum during endoscopic surgery on the paranasal sinuses is rare. Such complication as subcutaneous emphysema and pneumomediastinum after endoscopic endonasal removal of juvenile nasopharyngeal angiofibroma has not been reported in the literature.

Case report. A 19-year-old male patient has undergone endoscopic endonasal removal of juvenile nasopharyngeal angiofibroma. The tamponade was removed within the first 24 h postoperatively. Ten hours after it, the patient developed subcutaneous emphysema and pneumomediastinum triggered by sneezing. The diagnosis was confirmed by computed tomography of the neck and chest. The patient was transferred to the intensive care unit and received conservative treatment (including infusion, antibacterial, and antiinflammatory therapy). The symptoms of subcutaneous emphysema and pneumomediastinum subsided in response to treatment. Follow-up examinations (computed tomography and magnetic resonance imaging) confirmed that the tumor had been completely removed. The patient was discharged in a satisfactory condition.

Conclusion. Subcutaneous emphysema and pneumomediastinum are exceedingly rare complications of endoscopic endonasal removal of juvenile nasopharyngeal angiofibroma and are caused by anatomical connection between the parapharyngeal / retropharyngeal spaces and mediastinum. To prevent such complications, it is necessary to keep tampons in the nasal cavity for at least 2 days, as well as to instruct patients after surgery (avoid sneezing with their mouth closed, lifting weights, coughing, and vomiting). The nasoseptal flap used to repair the nasopharyngeal defect after tumor removal also ensures its sealing. Patients with complications should undergo computed tomography of the neck and chest (in case of emergency) and should be transferred to an intensive care unit. Conservative treatment (antibacterial and antiinflammatory therapy) will ensure good results in most patients.

1. Dubey S.P., Schick B. Juvenile angiofibroma. Switzerland: Springer, 2017. DOI: 10.1007/978-3-319-45343-9.

2. Kim J.S., Kim D.H., Jeon E.J. et al. A case of nasopharyngeal angiofibroma removed using a minimally invasive endoscopic endonasal technique. Wideochir Inne Tech Maloinwazyjne 2018;13(4):551–5. DOI: 10.5114/wiitm.2018.75862.

3. Sciarretta V., Pasquini E., Farneti G. et al. Endoscopic sinus surgery for the treatment of vascular tumors. Am J Rhinol 2006;20(4):426–31. DOI: 10.2500/ajr.2006.20.2888.

4. Shah S.R., Keshri A., Patadia S. et al. Stage III nasopharyngeal angiofibroma: Improving results with endoscopic-assisted midfacial degloving and modification to the Fisch staging system. J Craniomaxillofac Surg 2015;43(8):1678–83. DOI: 10.1016/j.jcms.2015.07.025.

5. Nicolai P., Villaret A.B., Farina D. et al. Endoscopic surgery for juvenile angiofibroma: a critical review of indications after 46 cases. Am J Rhinol Allergy 2010;24(2):67–72. DOI: 10.2500/ajra.2010.24.3443.

6. Huang Y., Liu Z., Want J. et al. Surgical management of juvenile nasopharyngeal angiofibroma: analysis of 162 cases from 1995 to 2012. Laryngoscope 2014;124(8):1942–6. DOI: 10.1002/lary.24522.

7. Godoy M.D., Bezerra T.F., Pinna F. de R., Voegels R.L. Complications in the endoscopic and endoscopic-assisted treatment of juvenile nasopharyngeal angiofibroma with intracranial extension. Braz J Otorhinolaryngol 2014;80(2):120–5. DOI: 10.5935/1808-8694.201.

8. Ardehali M.M., Samimi Ardestani S.H., Yazdani N. et al. Endoscopic approach for excision of juvenile nasopharyngeal angiofibroma: complications and outcomes. Am J Otolaryngol 2010;31(5):343–9. DOI: 10.1016/j.amjoto.2009.04.007.

9. Nersesyan M., Kapitanov D., Lopatin A. Our experience in endoscopic management of relapsed juvenile nasal angiofibroma. Eur Arch Otolaryngol 2009;266:1076–77. DOI: 10.1007/s00405-009-0987-7.

10. Leong S.C. A systematic review of surgical outcomes for advanced juvenile nasopharyngeal angiofibroma with intracranial involvement. Laryngoscope 2013;123(5):1125–31. DOI: 10.1002/lary.23760.

11. Liu K., Husain Q., Kanumuri V. et al. Endoscopic graduated multiangle, multicorridor resection of juvenile nasopharyngeal angiofibroma: an individualized, tailored, multicorridor, skull base approach. J Neurosurg 2016;124(5):1328–38. DOI: 10.3171/2014.1.

12. Liu Z., Hua W., Zhang H. et al. The risk factors for residual juvenile nasopharyngeal angiofibroma and the usual residual sites. Am J Otolaryngol 2019;40(3):343–6. DOI: 10.1016/j.amjoto.2018.11.010.

13. Rong Z., Zixiang Y., Chang L. et al. Lacrimal hyposecretion: a surgical complication of juvenile nasopharyngeal angiofibroma. Am J Otolaryngol 2008;29(6):367–71. DOI: 10.1016/j.amjoto.2007.10.006.

14. Hillel A.T., Metzinger R.C., Nemechek A.J., Nuss D.W. Loss of reflex tearing: an expected consequence of juvenile nasopharyngeal angiofibroma. Otolaryngol Head Neck Surg 2005;133(4):605–10. DOI: 10.1016/j.otohns.2005.05.014.

15. Eloy P., Bachy V., Grulois V., Bertrand B. Pyocele of the lachrymal sac: A late and unusual complication after surgery for a juvenile nasopharyngeal angiofibroma. Clin Ophthalmol 2008;2(1):211–15. DOI: 10.2147/opth.s2280.

16. Sharma S.B., Janakiram T.N., Baxi H., Chinnasamy B. Trigeminocardiac reflex during endoscopic juvenile nasopharyngeal angiofibroma surgery: an appraisal. Eur Arch Otorhinolaryngol 2017;274(7):2779–84. DOI: 10.1007/s00405-017-4521-z.

17. Alomari A.I., Alzoubi F.Q., Khatatbeh A. A case report of unusual pneumomediastinum after endoscopic sinus surgery. Int J Surg Case Rep 2016;29:249–53. DOI: 10.1016/j.ijscr.2016.11.028.

18. Aswani B., Anand S., Amarnath M., Lailu M. Bilateral pneumothorax, pneumomediastinum and subcutaneous emphysema following functional endoscopic sinus surgery under general anaesthesia: A case report. JMSCR 2020;8(2):933–7.

19. Respiratory medicine: Manual: in 3 vol. Ed. by A.G. Chuchalin. 2 nd edn. Moscow: Litterra, 2017. Vol. 2. 544 p. (In Russ.).

20. Saravakos P., Taxeidis M., Kastanioudakis I., Reichel O. Subcutaneous emphysema as a complication of tonsillectomy: a systematic literature review and case report. Iran J Otorhinolaryngol 2018;30(96):3–10.

21. Sohail M.A., Kishore K., Stammberger H. et al. Mediastinal emphysema associated with functional endoscopic sinus surgery. A case report. Rhinology 1995;33(2):111–2.

22. Park J.M., Park Y.C., Lee J.N. et al. Pneumomediastinum after functional endoscopic sinus surgery under general anesthesia: A case report. Korean J Anesthesiol 2013;64(4):367–72. DOI: 10.4097/kjae.2013.64.4.367.

23. Karpishchenko S.A., Beldovskaya N.Yu., Baranskaya S.V., Karpov A.A. Ophthalmic comlications of functional endoscopic sinus surgery. Oftal’mologicheskie vedomosti = Ophthalmological reports 2017;10(1): 87–92. (In Russ.). DOI: 10.17816/OV1087-92.

24. Rubinstein A., Riddell C.E., Akram I. et al. Orbital emphysema leading to blindness following routine functional endoscopic sinus surgery. Arch Ophthalmol 2005;123(10):1452. DOI: 10.1001/archopht.123.10.1452.

25. Sanu A., Jayanthi N.V., Mohan A.R. Pre-vertebral surgical emphysema following functional endoscopic sinus surgery. J Laryngol Otol 2006;120(11):e38. DOI: 10.1017/S0022215106003288.

26. Bellamy M.C., Berridge J.C., Hussain S.S. Surgical emphysema and upper airway obstruction complicating recovery from anaesthesia. Br J Anaesth 1993;71(4):592–3. DOI: 10.1093/bja/71.4.592.

27. Shah R.R., Thomas W.W., Naples J.G., Ruckenstein M.J. Subcutaneous emphysema and pneumomediastinum after eustachian tube balloon dilation. Otolaryngol Head Neck Surg 2018;159(1):203–5. DOI: 10.1177/0194599818768519.

28. Alsaleh S., Alabidi A., Mohammed Gamal A. Subcutaneous emphysema and pneumomediastinum after adenoidectomy; A rare complications. Glob J Oto 2019;18(5):97–101. DOI: 10.19080/GJO.2019.18.556000.

29. Boiko N.V., Tyukin Yu.V., Fldjyan L.Yu. Podkojnaya emfizema i pnevmomediastinum – redkoe oslojnenie tonzillektomii. Vestnik otorinolaringologii = Bulletin of Otorhinolaryngology 2019;84(5): 81–4. (In Russ.). DOI: 10.17116/otorino20198405181.

30. De Coninck L., Goderis J., Herregods N. et al. Massive pneumomediastinum with subcutaneous emphysema after elective adenotonsillectomy in children: Involvement of the Boyle-Davis mouth gag. Int J Pediatr Otorhinolaryngol 2019;122:152–4. DOI: 10.1016/j.ijporl.2019.04.023.

31. Naha A., Akhtar N., Datta P.G., Datta A. Subcutaneous emphysema and pneumomediastinum following adenotonsillectomy: A case report. Bangladesh Med Res Counc Bull 2019;42:62–5. DOI: 10.3329/bmrcb.v45i1.41810.

32. Ryu G., So Y.K., Seo M.Y. et al. Using the nasoseptal flap for reconstruction after endoscopic debridement of radionecrosis in nasopharyngeal carcinoma. Am J Rhinol Allergy 2018;32(1):61–5. DOI: 10.2500/ajra.2018.32.4486.

33. Rivera-Serrano C.M., Lentz A.K., Pinheiro-Neto C., Snyderman C.H. Cadaveric study of the posterior pedicle nasoseptal flap: a novel flap for reconstruction of pharyngeal defects and velopharyngeal insufficiency. Plast Reconstr Surg 2013;132(5):1269–75. DOI: 10.1097/PRS.0b013e3182a4c37b.