Chronic recurrent multifocal osteomyelitis involving the mandible: case reports and review of the literature
PAJ Monsour* and JB Dalton
The University of Queensland, School of Dentistry, Brisbane, Australia
Chronic recurrent multifocal osteomyelitis (CRMO)is an aseptic inflammatory disorder of unknown cause occurring in children and adolescents. It is characterized by multifocal bone lesions with pain and swelling recurring over months to years. Lesions usually involve the metaphyses of the long bones and involvement of the jaw is rare. The clinical presentation, radiographic appearance and histology of a case of CRMO involving the mandible in an8year-old girl are documented. The radiographic appearance of another three cases is also described. Dentomaxillofacial Radiology (2010) 39, 184–190. doi: 10.1259/dmfr/23060413 Keywords: chronic recurrent multifocal osteomyelitis; mandible; children; computed tomography
Introduction Chronic recurrent multifocal osteomyelitis (CRMO) is an uncommon aseptic inflammatory disorder. It is characterizedby bone lesions with pain and swelling with periods of exacerbation and improvement in different locations over several months to years. The aetiology of the condition is unknown.1,2 It is traditionally thought to affect children and adolescents.1 The current opinion is that CRMO is the paediatric and most severe form of SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis, osteitis).3,4 Thedisease was first described by Giedion et al in 19725 and the term chronic recurrent multifocal osteomyelitis was coined by Bjorksten et al.2 It affects females four times more than males,1 and has a median age of onset of around 9years-old.1,6,7 The median duration of disease activity is 5 years.6,8 Lesions are most commonly found in the metaphyses of the long bones and involvement of themandible is rare.1 This article describes the clinical presentation, radiographic appearance and histology of a case of CRMO involving the mandible in an 8-year-old girl. The radiographic appearances using CT of another three cases are also described. Case reports Case A An 8-year-old girl was referred to Queensland Diagnostic Imaging for radiographic investigation of a swelling in the left mandible.Clinical examination revealed swelling of the left masseter muscle and expansion of the left ramus of the mandible. Ultrasound of the region was performed and normal muscle striation and vascularity were reported. Bulging of the surface of the ramus of the mandible was noted, suggesting the presence of bony pathology. An orthopantomograph was obtained, which demonstrated expansion of the left ramus,loss of the normal trabecular pattern with patchy radiolucencies and absence of the cortical outline of the inferior dental canal (Figure 1). A CT examination using a multislice CT scanner was performed. Marked expansion, patchy sclerosis and areas of lysis were obvious in the left ramus, extending superiorly into the coronoid process and inferiorly into the left body to the first molar. Multipleareas of cortical perforation were noted in the left ramus. The thin overlying expanded cortex formed a ‘‘bone-onbone’’ appearance (Figure 2a). The inferior dental canal was visible running through the medullary sclerosis without deviation. There was swelling of the soft tissues, including the masseter, overlying the area (Figure 3). An MRI of the mandible and skull base with contrast and fatsaturation sequences were performed. Thickening of the
*Correspondence to: Professor Paul Monsour, University of Queensland, School of Dentistry, 200 Turbot Street, Brisbane, Qld 4000, Australia; E-mail: firstname.lastname@example.org Received 3 March 2009; revised 25 May 2009; accepted 11 June 2009
Chronic recurrent multifocal osteomyelitis PAJ Monsour and JB Dalton
left ascending ramus and...