Burkholderia Cepacia Causes Frontal Mucopyocele with Anterior Cranial Fossa Extension: A Novel Case Report

Document Type : Case Report


1 Department of Otolaryngology, General University Hospital of Larisa, Greece.

2 Department of Neurosciences, Otolaryngology Section, University of Padua, Italy.

3 Department of Otolaryngology, General Hospital San Dona di Piave, Venice, Italy.


Burkholderia cepacia complex (Bcc) is a group of gram-negative bacilli that have rarely been isolated in the ear, nose and throat region in immunocompetent patients. Bcc show resistance to most available antibacterial drugs.
Case Report:
We present the case of an immunocompetent 31-year-old male reporting a pulsating headache with right supraorbital swelling associated with exophthalmos. A brain CT scan showed an expansive giant cystic lesion occupying the right frontal sinus, extending to the anterior cranial fossa. Management and outcome: drainage with the resecting of the floor of the frontal sinus from the orbital plate of the ethmoid bone to the nasal septum (Draf IIb) was performed with wide marsupialization of the mucopyocele. Polymerase chain reaction-restriction fragment length polymorphism (PCR-RFLP) analysis was used to identify the isolate. MRI 1 and 12 months after surgery showed complete lesion removal. The patient was followed for 12 months with complete recovery of symptoms.
Paranasal sinuses disease with cranial expansion and orbital complications constitutes an emergency. For the first time in the literature, Bcc was isolated in the frontal sinus, extending into the anterior cranial fossa, in an immunocompetent patient. An endoscopic surgical approach with microbiological identification and management by appropriate antibacterial drug treatment seems to be the key to success.


  1. Jones AM, Dodd ME, Webb AK. Burkholderia cepacia: current clinical issues, environmental controversies and ethical dilemmas. Eur Respir J 2001; 17:295–301.
  2. Imataki O, Kita N, Nakayama-Imaohji H, Kida J.-i, Kuwahara T, Uemura M. Bronchiolitis and bacteraemia caused by Burkholderia gladioli in a non-lung transplantation patient. New Microbes New Infect 2014; 2:175–6.
  3. Zanotti C, Munari S, Brescia G, Barion U. Burkholderia gladioli sinonasal infection. European Annals of Otorhinolaryngology, Head and Neck diseases 2019; 55–56.
  4. YOU ME Lee YM, Park KH, Moon C, Kim DY, Lee MS, Kim T, et al. Management and outcomes of Burkholderia cepacia complex bacteremia in patients without cystic fibrosis: a retrospective observational study. Eur J Clin Microbiol Infect Dis. 2020 Nov;39(11):2057-2064. doi: 10.1007/s10096-020-03960-2. Epub 2020 Jun 25. PMID: 32583228.
  5.  Zou Q, Li N, Liu J, Li X, Wang Z, Ai X, et al. Investigation of an outbreak of Burkholderia cepacia infection caused by drug contamination in a tertiary hospital in China. Am J Infect Control. 2020 Feb;48(2):199-203. doi: 10.1016/j.ajic.2019.06.011. Epub 2019 Jul 23. PMID: 31345617.
  6. TAVARES Tavares M, Kozak M, Balola A, Sá-Correia I. Burkholderia cepacia Complex Bacteria: a Feared Contamination Risk in Water-Based Pharmaceutical Products. Clin Microbiol Rev. 2020 Apr 15;33(3):e00139-19. doi:10.1128/

CMR. 00139-19. PMID: 32295766; PMCID: PMC7194853.

  1. Brook I, Frazier EH. The microbiology of mucopyocele. The Laryngoscope 2001; 111(10): 1771-3.
  2. Chobillon MA, Jankowski R. Relationship between mucoceles, nasal polyposis and nasalization. Rhinology 2004; 42(4): 219-24.
  3. Obeso S, Llorente JL, Rodrigo JP, Sánchez R,  Mancebo G,  Suárez C. Paranasal sinuses mucoceles: our experience in 72 patients. Acta Otorhinolaringol Esp 2009; 60(5):332–9.
  4. Abdel-Aziz M, El-Hoshy H, Azooz K,  Naguib N,  Ahmed Hussein A. Maxillary sinus mucocele: predisposing factors, clinical presentations, and treatment. Oral Maxillofac Surg 2017; 21 (1):55-8
  5. Har-El G. Endoscopic management of 108 sinus mucoceles. The laryngoscope 2001; (111.12): 2131-4.
  6. Scangas GA, Gudis DA, Kennedy DW. The natural history and clinical characteristics of paranasal sinus mucoceles: a clinical review. International forum of allergy & rhinology 2013; 3(9):717-7.
  7. Lund VJ, Milroy CM. Fronto-ethmoidal mucocoeles: a histopathological analysis. The Journal of Laryngology & Otology 1991; 105(11): 921-23.
  8. Dandinarasaiah M, Hegde J, Srinish G, Bijiraj V V, Prajna L. S. Atrophic Rhinitis Presenting with Ethmoidal Mucocele: A Case Report. J Clin Diag Res 2014; 8(6): KD03-KD04.
  9. Nourizadeh N,Adabizadeh A,Zarrinfar H,Majidi M,Jafariane A.H,Mohammad M.J

 Fungal biofilms in sinonasal polyposis: The role of fungal agents is notable? Journal of Oral and Maxillofacial Surgery, Medicine, and Pathology .Volume 31, Issue 4, July 2019, Pages 295-298

  1. Najafzadeh MJ, Jalaeian Samani K, Houbraken J, et al. Identification of fungal causative agents of rhinosinusitis from Mashhad, Iran. Curr Med Mycol. 2017;3(3):5-9.
  2.  Bakhshizadeh M, Hashemian HR, Najafzadeh MJ, Dolatabadi S, Zarrinfar H. First report of rhinosinusitis caused by Neoscytalidium dimidi- atum in Iran. J Med Microbiol. 2014 Jul;63(Pt 7): 1017-1019.
  3. Har-el G. Transnasal endoscopic management of frontal mucoceles. Otolaryngologic Clinics of North America 2001; 34(1): 243-51
  4.  Nam Su Ku, Sang Hoon Han, Chang Oh Kim, Ji-Hyeon Baek, Su Jin Jeong,Sung Joon Jin, Jun Yong Choi, Young Goo Song & June Myung Kim (2011) Risk factors formortality in patients with Burkholderia cepacia complex bacteraemia, Scandinavian Journal ofInfectious Diseases, 43:10, 792-797
  5. Marioni G, Rinaldi R, Staffieri C, Ottaviano G, Marchese-Ragona R, Giacomelli L, Ferraro SM, Staffieri A. Burkholderia cepacia complex nasal isolation in immunocompetent patients with sinonasal polyposis not associated with cystic fibrosis. Eur J Clin Microbiol Infect Dis. 2007 Jan; 26(1):73-5.
  6. Coutinho CP, Barreto C, Pereira L, Lito L, Cristino JM, Sá-Correia I. Incidence of Burkholderia contaminans at a cystic fibrosis centre with an unusually high representation of Burkholderia cepacia during 15 years of epidemiological surveillance. Journal of medical microbiology 2015; vol. 64, no. 8, pp. 927-935.
  7. Courson AM, Stankiewicz JA, and Lal D. Contemporary management of frontal sinus mucoceles: a meta-analysis. Laryngoscope 2014; 124:378–386.
  8. Stokken J, Wali E, Woodard T, Recinos PF, Sindwani R. Considerations in the management of giant frontal mucoceles with significant intracranial extension: a systematic review. Am J Rhinol Allergy 2016; 30:301-305.