|
 
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| CASE REPORT |
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| Year : 2022 | Volume
: 9
| Issue : 4 | Page : 152-154 |
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Osteomyelitis of maxilla with orbital cellulitis after tooth extraction
Qiong Yao, Xihong Hu
Department of Radiology, Children's Hospital of Fudan University, Shanghai, China
| Date of Submission | 25-Feb-2022 |
| Date of Decision | 10-Nov-2022 |
| Date of Acceptance | 20-Dec-2022 |
| Date of Web Publication | 21-Mar-2023 |
Correspondence Address:
Xihong Hu
Department of Radiology, Children's Hospital of Fudan University, Shanghai 201102
China
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/RID.RID_16_22
Osteomyelitis of the maxilla with orbital cellulitis, an uncommon and life-threatening disease, can be misdiagnosed. We here report a 13-year-old boy, possibly with combined immunodeficiency disease, who presented with osteomyelitis of the maxilla and orbital cellulitis after tooth extraction. Computed tomography demonstrated thickening of the left maxillary bone. Magnetic resonance imaging showed inflammation in the left maxillary bone and retrobulbar space. Metagenomic analysis of an aspiration biopsy resulted in a diagnosis of infection with Porphyromonas endodontalis. He was successfully treated with metronidazole.
Keywords: Children, magnetic resonance imaging, orbital cellulitis, osteomyelitis
How to cite this article:
Yao Q, Hu X. Osteomyelitis of maxilla with orbital cellulitis after tooth extraction. Radiol Infect Dis 2022;9:152-4 |
| Introduction | |  |
It's known that osteomyelitis of maxilla with orbital cellulitis is an uncommon and life threatening disease. Early diagnosis and treatment can reduce complications and improve the prognosis. In this case, we reported a 13-year-old boy possibly presenting osteomyelitis of maxilla and orbital cellulitis after tooth exbraction. The CT and MRI images helped to show the infections.
| Case Report | | |
A 13-year-old boy was admitted to our hospital for facial swelling and deteriorating vision in the left eye. He had developed orbital cellulitis after a tooth extraction 3 years previously. He had previously been diagnosed with mutation of the tapasin-binding protein (TAPBP) gene and a variety of infections, including pneumonia, appendicitis, and lymphadenitis. Examination revealed edema, exophthalmos, and drooping of the left upper eyelid. Relevant laboratory data included white cell count 11.9 × 109/L, 86.1% neutrophils, and C-reactive protein 105 mg/L. Computed tomography (CT) demonstrated that the left maxillary bone was thickened and contained lytic and sclerotic lesions. The left maxillary sinus was smaller than the right and the medial orbital wall had been destroyed [Figure 1]. Magnetic resonance imaging (MRI) of the head and eyes also showed low signal intensity on T1-weighted images and high signal intensity on T2-weighted images in the left maxillary bone and inflammation in the left maxillary and ethmoid sinuses [Figure 2]. The left retrobulbar space, including the extraocular muscles and optic nerve, was also affected. Other MRI findings included proptosis, enlarged extraocular muscles, optic atrophy, and extensive swelling of retrobulbar soft tissue [Figure 3]. Taking these findings together, osteomyelitis of the maxilla with orbital cellulitis was diagnosed. Aspiration biopsy and pathogen metagenomic analysis revealed that the pathogen was Porphyromonas endodontalis (P. endodontalis). We therefore prescribed a large dose of metronidazole (0.3 g, three times per day) and levofloxacin eye drops (three times per day). The infection regressed and the patient's general condition improved during follow-up. A repeat MRI showed absorption of high-intensity signals on T2-weighted images of the left maxillary bone [Figure 4]. | Figure 1: Coronal reformatted computed tomography image demonstrating thickening of the left maxillary bone (long arrow) and destruction of the medial orbital wall (short arrow)
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 | Figure 2: T2-weighted fat-saturated coronal magnetic resonance image showing high signal intensity in the left maxillary bone (long arrow) and retrobulbar space infection (short arrow)
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 | Figure 3: Enhanced axial T1-weighted image showing extensive swelling in the retrobulbar soft tissue (arrow)
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 | Figure 4: T2-weighted fat-saturated coronal magnetic resonance image showing absorption of high-intensity signals by the left maxillary bone (long arrow)
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| Discussion | | |
Osteomyelitis of the maxilla with orbital cellulitis, an uncommon and life-threatening condition, can be misdiagnosed.[1] In most cases, osteomyelitis of the maxilla is caused by nasosinusitis or odontogenic disease.[2],[3] It can also be caused by periodontal or periapical diseases. Hematogenous spread of bacteria has also been reported.[1],[4] Our patient had previously had a tooth extraction. We believed that the associated injury to the alveolar bone had triggered the infection because P. endodontalis is specifically located in the gingival area. The previous infections listed above had occurred long before the current infection and possibly indicated impairment of our patient's immune function. He had a mutation of the TAPBP gene, which binds to MHC Class I in the endoplasmic reticulum.[5] However, the relationship between mutation of this gene and immune dysfunction is still being investigated.
Pathogens that can cause osteomyelitis of the maxilla include both Gram-positive and Gram-negative bacteria, such as Staphylococcus aureus, Staphylococcus epidermidis, and Peptostreptococcus, and mucormycetes, which can cause mucormycosis.[6] P. endodontalis, an asaccharolytic, black-pigmented, Gram-negative anaerobic bacterium, has been shown to be associated with chronic periodontitis and periapical infections.[7] Osteomyelitis caused by P. endodontalis has rarely been reported. However, this patient appeared to have an immunodeficiency disease, which may have contributed to the extensive spread of his infection. His severe infection with P. endodontalis induced acute osteomyelitis of the maxilla that penetrated through the cortical bone and progressed to orbital cellulitis. In such cases, the type of antibiotic should be tailored according to the responsible pathogen(s).
| Conclusions | | |
Orbital cellulitis caused by osteomyelitis of the maxilla is often misdiagnosed. Our case is important because of the rarity of the pathogen, the severity of the infection, and the misdiagnosis in the early stages. Early diagnosis and appropriate antibiotic therapy can reduce complications and improve the prognosis. CT and MRI scans are necessary to determine the origin and extent of infection.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Ethic statement
Not appliable.
Acknowledgment
We thank Dr. Trish Reynolds, MBBS, FRACP, from Liwen Bianji (Edanz) (www.liwenbianji.cn/), for editing the English text of a draft of this manuscript.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
| References | | |
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| 2. | Skouteris CA, Velegrakis G, Christodoulou P, Helidonis E. Infantile osteomyelitis of the maxilla with concomitant subperiosteal orbital abscess: A case report. J Oral Maxillofac Surg 1995;53:67-70. |
| 3. | Prasad KC, Prasad SC, Mouli N, Agarwal S. Osteomyelitis in the head and neck. Acta Otolaryngol 2007;127:194-205. |
| 4. | Whyte NS, Bielski RJ. Acute hematogenous osteomyelitis in children. Pediatr Ann 2016;45:e204-8. |
| 5. | Boyle LH, Hermann C, Boname JM, Porter KM, Patel PA, Burr ML, et al. Tapasin-related protein TAPBPR is an additional component of the MHC class I presentation pathway. Proc Natl Acad Sci U S A 2013;110:3465-70. |
| 6. | Sadaksharam J, Murugesan M. Osteomyelitis of maxilla: A rare finding from a radiologist point of view. Contemp Clin Dent 2019;10:394-6. [Full text] |
| 7. | Lombardo Bedran TB, Marcantonio RA, Spin Neto R, Alves Mayer MP, Grenier D, Spolidorio LC, et al. Porphyromonas endodontalis in chronic periodontitis: A clinical and microbiological cross-sectional study. J Oral Microbiol 2012;4:10-3402. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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