1. Aspergillus Temporomandibular Joint Osteomyelitis as a Complication of Otitis Externa
Lalee Varghese, Rabin Chacko, George M Varghese, Anand Job
Christian Medical College, Vellore
ABSTRACT
DISCUSSION
Osteomyelitis of the temporomandibular joint (TMJ) is a rare complication of otitis externa which can lead to ankylosis and destruction of the joint. We report a 74 year old diabetic who presented with persistent earache in spite of multiple courses of antibiotic therapy. TMJ involvement was associated with tender preauricular swelling and limitation in mouth opening. CT scan showed erosion of the right mandibular condyle with soft tissue swelling around the TMJ and external ear along with thrombosis of right internal jugular vein. Surgical findings included erosion of the mandibular condyle with granulation tissue. Peroperative culture grew Aspergillus flavus. To our knowledge, this is the first case of temporomandibular osteomyelitis due to Aspergillus flavus. The patient was successfully treated with right condylectomy and debridement followed by oral voriconazole.
Septic arthritis of the TM joint is an uncommon disease. Direct spread of local sepsis in adults has been reported from otitis externa, infection of maxillary molars, an intra-articular injection, extraction of third molars, and a facial burn. Adult patients with rheumatoid disease, diabetes, on immunosuppression or taking steroids are often predisposed to infection[1]. On reviewing the literature, there are only few reports of septic arthritis of the temporomandibular joint following otitis externa and in almost all cases in both adults and children the causative organism usually is Staphylococcus aureus[2].
Otitis externa is a common condition especially in patients with diabetes mellitus. Invasive otitis externa (IOE) is a particular entity where main feature is its spreading from the external auditory canal to adjacent anatomical structures including soft tissues, cartilage, and bone. The most common pathogen in this condition is Pseudomonas aeruginosa. The invasive process can lead to skull base osteomyelitis, progressive cranial nerve palsies, and fatal outcome if not recognized and treated early. Yet spread of infection to the temporomandibular joint is extremely rare. Anatomically the anterior bony canal wall is related to the joint; the joint capsule is attached to the squamotympanic suture. Congenital dehiscences of the cartilaginous canal, the inconstant fissures of Santorini, have been described, and these or a dehiscent squamotympanic suture may account for spread of infection to the joint[3]. Diagnosis of the condition may be difficult and delayed, as painful jaw movements, preauricular swelling and tenderness are not uncommon in otitis externa.
Invasive external otitis typically occurs in elderly diabetic patients, and Pseudomonas aeruginosa is the most common causative microbial pathogen[4], [5]. Fungal pathogens, mostly Aspergillus species can rarely cause IOE [6] in patients with uncontrolled diabetes mellitus, immunocompromised patients or in those under long-term steroid therapy.
Radiographs of the temporomandibular joint in the early stages may be normal although later signs of bony destruction may become apparent. Aspiration of the joint and appropriate antibiotic therapy may be sufficient to treat septic arthritis if instigated early. Once abscess formation is established and signs of bony destruction are apparent then exploration of the joint is required. The treatment of Aspergillus sp. IOE classically includes extensive surgical debridement and long-term antifungal therapy. The joint is approached by a parotidectomy incision, keeping close to the cartilage of the external meatus to avoid damage to the facial nerve. Bony defects and large cavities may need obliteration with muscle flaps.
Voriconazole is currently considered the first-line therapeutic option for invasive aspergillosis [7], based on its high intrinsic anti-Aspergillus activity and its superiority against intravenous amphotericin B in a large randomized trial [8]. In addition, this broad-spectrum azole is distributed throughout the body, including soft tissues and bone, where its good diffusion has been recently documented. Furthermore, long-term voriconazole therapy has been demonstrated to be effective in several patients with Aspergillus bone infections[9]. This antifungal agent is well tolerated despite prolonged treatment and available intravenously and orally.
To our knowledge, our patient is the first case of temporomandibular joint osteomyelitis due to Aspergillus flavus. Based on its favorable bone penetration, its tolerance, and its efficacy as demonstrated in this case, voriconazole may be considered an attractive first-line therapeutic option for Aspergillus IOE.
CASE REPORT
A 74-year-old man, a known diabetic for past four years on oral hypoglycemic agents, presented with a 5-month history of right-sided otalgia and otorrhea. He also had pain around right ear on opening the mouth for four months. The ear discharge was minimal and watery initially but later developed severe ear ache with mucopurulent discharge. He was also treated with several courses of oral antibiotics and topical ear drops, with minimal improvement. Four months after the initial diagnosis, he was admitted to our hospital with worsening pain. On clinical examination, the right external ear canal was markedly painful and filled with whitish otomycotic debris. Tympanic membrane showed a pinpoint perforation with pulsatile discharge. There was an ill-defined tender swelling in the right preauricular area. Physical examination also revealed weakness of 9, 10 & 11 cranial nerves. A diagnosis of otitis externa with skull base involvement was made and computed tomography of base of skull showed erosion of the right condylar process and enhancing soft tissue within the temporomandibular (TM) joint. There was focal erosion of the posterosuperior wall of the TM joint. Right external auditory canal showed concentric mild soft tissue swelling. Soft tissue with ill-defined margins, likely inflammatory changes, obliterated the parapharyngeal fat and fat planes in the masticator space & infratemporal fossa, and extended into nasopharyx with obliteration of the right Fossa of Rosenmuller. There was loss of fat plane around carotid sheath with thrombosis of right internal jugular vein. Findings were suggestive of septic arthritis of right TM joint with extension into the parapharyngeal space and nasopharynx with right IJV thrombosis.
After optimal control of blood sugars, right condylectomy and debridement was done under GA and granulation tissue filling joint space was sent for histopathological and microbiological examination. Histopathological examination showed fragments of fibrocollagenous tissue and inflammatory granulation tissue with patchy moderate infiltrates of lymphocytes admixed with plasma cells. Entrapped spicules of necrotic bone were present in foci. Gram staining showed pus cells with no bacteria and the bacterial culture was negative. Fungal culture yielded Aspergillus flavus.
The patient was initiated on treatment with oral voriconazole (200mg orally twice a day). On follow-up after 10 weeks the patient is asymptomatic. He has shown good clinical and biological tolerance with out any adverse effects to voriconazole.
CT scan showing enhancing soft tissue within the right TM joint with erosion of the condylar process & articular margins
REFERENCES
1] Amos MJ, Patterson AR, Worrall SF. Septic arthritis of the temporomandibular joint in a 6-year-old child. Br J Oral Maxillofac Surg Apr;46(3):242-3.
[2] Thomson HG. Septic arthritis of the temporomandibular joint complicating otitis externa. J Laryngol Otol Mar;103(3):
[3] Smith, P. G. and Lucente, F. E. (1986) In Otolaryngology—Head and Neck Surgery (Cummings, C,. W., Harker, L. A., Krause, C. J., Shuller, D. E. (eds) Vol IV p.2899, The C. V. Mosby Company, St Louis, Toronto
[4] Doroghazi, R. M., J. B. Nadol, Jr., N. E. Hyslop, Jr., A. S. Baker, and L. Axelrod Invasive external otitis. Report of 21 cases and review of the literature. Am. J. Med. 71:
[5] Rubin Grandis, J., B. F. Branstetter IV, and V. L. Yu The changing face of malignant (necrotising) external otitis: clinical, radiological, and anatomic correlations. Lancet Infect. Dis. 4:34-39.
[6] Carfrae, M. J., and B. W. Kesser Malignant otitis externa. Otolaryngol. Clin. N. Am. 41:
[7] Walsh, T. J., E. J. Anaissie, D. W. Denning, R. Herbrecht, D. P. Kontoyiannis, K. A. Marr, V. A. Morrison, B. H. Segal, W. J. Steinbach, D. A. Stevens, J. A. van Burik, J. R. Wingard, and T. F. Patterson Treatment of aspergillosis: clinical practice guidelines of the Infectious Diseases Society of America. Clin. Infect. Dis. 46:
[8] Herbrecht, R., D. W. Denning, T. F. Patterson, J. E. Bennett, R. E. Greene, J. W. Oestmann, W. V. Kern, K. A. Marr, P. Ribaud, O. Lortholary, R. Sylvester, R. H. Rubin, J. R. Wingard, P. Stark, C. Durand, D. Caillot, E. Thiel, P. H. Chandrasekar, M. R. Hodges, H. T. Schlamm, P. F. Troke, and B. de Pauw Voriconazole versus amphotericin B for primary therapy of invasive aspergillosis. N. Engl. J. Med. 347:
[9] Mouas, H., I. Lutsar, B. Dupont, O. Fain, R. Herbrecht, F. X. Lescure, and O. Lortholary Voriconazole for invasive bone aspergillosis: a worldwide experience of 20 cases. Clin. Infect. Dis. 40:
Lalee Varghese