
Osteomyelitis of the oral and maxillofacial area is an uncommon inflammatory disease that occurs due to odontogenic causes such as endodontic infection, facial trauma, insufficient blood supply, and iatrogenic postoperative infections [1]. With the development of modern medicine and various antibiotics, the incidence of osteomyelitis, except for medication-related osteonecrosis (MRONJ), has decreased. However, osteomyelitis of the mandible is still considered a difficult disease to treat due to the complexity of its pathophysiology and the need for long-term broad-spectrum antibiotic administration and multiple surgeries [2].
Candida infections were previously reported in immunocompromised patient groups, such as newborns, drug addicts, and patients with catheters; however, candida osteomyelitis was rarely observed [3]. Candida osteomyelitis mainly occurs in the spine, sternum, femur, hip, facial bones, feet, ankles, and tibia. Therefore, it must be diagnosed promptly to avoid its progression to significant mortality [4]. Candida osteomyelitis of the jaw has a low incidence and no clearly established criteria for its diagnosis, treatment, and prognosis. Fungi and yeast are originally normal commensal microorganisms of the oral cavity; however, in the presence of conducive local or systemic factors, they could cause infections, including osteomyelitis. With an aging society, the number of immunocompromised patients is increasing, and more cases of candida osteomyelitis of the jaw will occur [5]. Herein, we present the case of a patient with osteomyelitis of the maxilla and mandible, including the maxillary and ethmoidal sinuses due to candida infection. The study protocol was approved by the Ethics Committee of Chosun University Dental Hospital (approval no. CUDHIRB 2206 002) and the need for written informed consent was waived by the committee.
An 81-year-old male patient visited Chosun University Hospital with a chief complaint of nasal pyorrhea and intermittent epistaxis that had occurred one month earlier. He was diagnosed with osteomyelitis based on a paranasal sinus computed tomography (CT) scan performed by an otorhinolaryngologist. At the time of admission, all his vital signs were stable, and the white blood cell count was 6,480 cells/mm3, showing no signs of an acute infection in the blood test. The patient was taking atenolol and had no underlying disease other than arrhythmia. In the department of otorhinolaryngology, endoscopic sinus surgery was planned under general anesthesia. On the CT image, necrotic tissue in the right maxillary alveolar bone was observed (Fig. 1), and a combined operation was performed after consultation with the department of oral and maxillofacial surgery. During sequestrectomy, the right maxillary sinus and oral cavity were perforated due to bone destruction, and much foreign material looking like a fungus ball was discharged from the inside along with sequestrum (Fig. 2). The patient showed good healing after the operation and was discharged from the hospital; however, two weeks later, during follow-up, the patient complained of decreased sensation in the lower lip and swelling of the right jaw. In the oral cavity, some foreign material looking like the previous fungus ball was drained from the lingual alveolar ridge of the right mandibular molar (Fig. 3). At the time of his discharge, the erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) level approached normal values (close to zero); however, when he was later re-hospitalized and retested, the CRP level had increased to 19.7 mg/dL and the ESR to 34 mm/hr. Testing was performed by collecting the drainage material and some tissue samples. As a result of the microbiological culture,
Osteomyelitis, which could be defined as an inflammatory, progressive lesion in the bone and bone marrow, can occur in any bony part of the body; however, it is relatively common in the jaw bones, including the teeth. This is because causative microorganisms can provide a route for spreading into these bones through the root canal or periodontal ligament of the contaminated tooth. In addition, the prevalence of MRONJ related to drugs such as antiresorptive agents and antiangiogenic agents, is increasing [6]. Since the etiology is so diverse, in the case of patients with osteomyelitis of the jaw, clinicians often monotonically prescribe antibiotics and implement curettage without recording the patient’s medical history in detail and approaching the patient carefully. In most cases, osteomyelitis responds to empirical antibiotics and is effectively treated via surgery; however, as in the above cases, osteomyelitis caused by candida or pathogens resistant to empirical antibiotics does not respond well to antibiotics and continues to recur even after surgery, resulting in a poor prognosis [7].
Candida, one of the commensal oral microorganisms in healthy individuals, often integrates the oropharyngeal, gastrointestinal, and vaginal microflora. Although the incidence of candida osteomyelitis of the jaw is minimal, several cases of
Fifteen candida species that cause illness in mankind are known; however, only five species cause more than 90% of invasive infections:
For successful treatment of candida osteomyelitis, speedy diagnosis and the proper use of antifungal drugs are essential. The diagnosis can be made via microbiological culture, histology, or serology; however, it is hard to distinguish between invasive infection and simple colonization in oral candida [8]. Ideally, in these infections, it is recommended to perform both microbiological culture and biopsy for diagnosis [9]. In this case,
The mechanisms by which candida induces bone infection can be classified into three categories: hematogenous spread, direct contact, and contiguous infection [10]. Among these, hematogenous spread occurs most commonly; however, as mentioned above, there are many cases that cannot be diagnosed even with blood culture, such as extremely low levels of candidemia, intermittent candidemia, or sterilization by bloodstream [11]. In this case, since the main complaint was nasal pyorrhea and intermittent epistaxis, it can be speculated that candida infection from the right maxillary sinus spread to the surrounding bone and destroyed the maxillary alveolar bone, and then continued to spread osteomyelitis through the oral cavity to the adjacent ipsilateral mandibular posterior bone. As such, osteomyelitis caused by candida is often associated with two or more sites; so, it is necessary to closely observe other bones when a patient is diagnosed with this infection [12].
In addition,
According to the 2015 Infectious Diseases Society of America guideline updates for candidiasis, for the treatment of candida osteomyelitis, it is recommended to administer fluconazole (Diflucan; Pfizer-Roerig, New York, NY, USA) 400 mg/day for 2–6 months while performing surgical debridement if necessary [14]. In this patient, micafungin was administered for two weeks, and fluconazole was used for about two months, resulting in good healing progress.
In conclusion, candida osteomyelitis has been considered a rare infectious disease with high mortality and fatality rates; however, it is occurring more and more frequently nowadays due to the change in the population structure and the increase in the number of immunosuppressors [15]. When candida osteomyelitis of the jaw is clinically suspected, prompt diagnosis (through culture tests, biopsies, and multiple blood cultures) and surgical intervention, followed by the use of appropriate antifungal agents and the prevention of opportunistic infections should be performed.
No potential conflict of interest relevant to this article was reported.
Conceptualization: HJP. Data curation: HIC. Funding acquisition: HIC. Methodology: JSO, SYM. Project administration: HJP. Visualization: JYC. Writing original draft: HIC. Writing review & editing: HJP, JSY.
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