Is diffuse sclerosing osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome?: Analysis of seven cases

doi:10.1016/0030-4220(94)90170-8

Oral Surgery, Oral Medicine, Oral Pathology

Volume 78, Issue 5, November 1994, Pages 594-598

Oral Surgery, Oral M...

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Abstract

Diffuse sclerosing osteomyelitis of the mandible has been described as a localized disease of unknown origin. We offer evidence that mandibular involvement in the recently described synovitis acne pustulosis hyperostosis osteitis syndrome exactly fits the accepted description for diffuse sclerosing osteomyelitis of the mandible. The clinical, radiologic, and pathologic findings in seven such cases are presented and discussed.

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Cited by (108)

Collaborative approach to paediatric chronic non-bacterial osteomyelitis of the mandible: Great Ormond Street Hospital case series
2024, British Journal of Oral and Maxillofacial Surgery
This paper outlines a 10-patient case series of chronic non-bacterial osteomyelitis (CNO) of the mandible at a tertiary paediatric hospital in the UK. Our findings highlight the homogeneous presenting signs and symptoms of an intermittently painful, swollen angle and ramus of the mandible. We present the typical laboratory investigative findings (normal inflammatory markers) and imaging appearances (sclerosis and periosteal oedema). Our paper outlines an investigation protocol, including recommendations for extraoral bone biopsies and systemic magnetic resonance imaging (MRI). We explain the importance of multidisciplinary care, with combined care by rheumatologists and infectious disease specialists. Finally we demonstrate the efficacy of our treatment algorithm for oral non-steroidal anti-inflammatory drugs (NSAIDs), and in those cases refractory to NSAIDS, intravenous pamidronate. This paper provides a useful addition to the literature by informing OMF surgeons of this rare condition and given the clinical equipoise in treatments, it can hopefully guide clinicians in an investigation pathway and management protocol.
Bisphosphonate therapy in chronic diffuse sclerosing osteomyelitis/tendoperiostitis of the mandible: Retrospective case series
2022, Journal of Cranio-Maxillofacial Surgery
Citation Excerpt :
It is hypothesised that bisphosphonates inhibit osteoclast-mediated bone remodelling, which decreases the inflammatory response, resulting in a reduction of pain and swelling of the cheek (Montonen et al., 2001; Soubrier et al., 2001; Montonen and Lindqvist, 2003; Hino et al., 2005; Yamazaki et al., 2007; Urade et al., 2012; Berglund et al., 2015; Otto et al., 2015; van de Meent et al., 2017). Some authors suggest that DSO/TP of the mandible is part of a systemic spectrum with syndromes such as SCCH (sternocostoclavicular hyperostosis), CRMO (chronic recurrent multifocal osteomyelitis) or SAPHO-syndrome (synovitis, acne, pustulosis, hyperostosis, osteitis) (Kahn et al., 1994; Suei et al., 1995; Mari et al., 2014). Therapy with bisphosphonates is already a more common additional treatment in these syndromes, when refractory to anti-inflammatory medication (Aljuhani et al., 2015; Cianci et al., 2017; Hofmann et al., 2017; Yachoui et al., 2017; Li et al., 2020).
This study aims to evaluate short-term and long-term results of bisphosphonate therapy in patients with diffuse sclerosing osteomyelitis/tendoperiostitis (DSO/TP) of the mandible.
Eighteen patients (12 female, 6 male) aged 34.8 ± 22.2 years with DSO/TP of the mandible that were treated with bisphosphonates were included. In 16 patients, the bisphosphonate treatment led to remission with decrease of symptoms (pain, swelling of the cheek, trismus, tenderness of masticatory muscles) with a follow-up period of 4.5 (0.8–11.9) years between start of bisphosphonate treatment and latest follow-up consult. Of these, three patients were still in need of regular bisphosphonate therapy. Two patients were lost to follow-up.
Bisphosphonate therapy is a treatment option for DSO/TP of the mandible that is associated with a high chance of remission of symptoms. Within the limitations of the study it seems that this treatment might be an effective second step in DSO/TP refractory to conservative treatment.
Efficacy of diffusion-weighted magnetic resonance imaging in the diagnosis of osteomyelitis of the mandible
2022, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
This retrospective cohort study aimed to determine the diagnostic utility of apparent diffusion coefficient (ADC) values in the quantitative evaluation of mandibular osteomyelitis.
We analyzed the records of 70 patients aged 30 to 90 years, with and without osteomyelitis, who underwent magnetic resonance imaging at the Nihon University School of Dentistry between April 2017 and March 2019. The mean ADC of bone marrow of patients without osteomyelitis and those with acute and chronic osteomyelitis of the mandible were calculated. Differences in ADC overall and in pair-wise comparisons were analyzed. Correlations with patient age were also calculated.
The mean ADC values in the nonosteomyelitis, acute osteomyelitis, and chronic osteomyelitis groups were 0.87 ± 0.15 × 10⁻³, 1.24 ± 0.11 × 10⁻³, and 1.07 ± 0.13 × 10⁻³ mm²/s, respectively (P < .001 for all pair-wise comparisons). Receiver operating characteristic curve analysis revealed an ADC cut-off of 0.98 for osteomyelitis. Diagnostic values for predicting osteomyelitis were ≥ .81. No correlations of osteomyelitis with age were found.
The ADC was significantly higher in the osteomyelitis groups than in the nonosteomyelitis group. This suggests that ADC may be a useful parameter for quantitative evaluation of mandibular osteomyelitis.
Comparing Clinical and Radiographic Characteristics of Chronic Diffuse Sclerosing Osteomyelitis and Craniofacial Fibrous Dysplasia in the Mandible
2021, Journal of Oral and Maxillofacial Surgery
Differential diagnosis of chronic diffuse sclerosing osteomyelitis of the mandible (DSOM) and craniofacial fibrous dysplasia (CFD) involving the mandible is challenging. The purpose of this study was to explore the differences of the clinical and radiographic characteristics between these 2 conditions.
In this retrospective cross-sectional, blinded, comparative study, clinical and imaging data of patients with DSOM and CFD at the Peking University Hospital of Stomatology from 2012 to 2018 were retrieved. Clinical characteristics, mainly pain, swelling, and trismus, and radiographic findings, including sclerosis, lysis, and subperiosteal bone formation, were evaluated. The t test, χ² test, and Fisher-Freeman-Halton test were used to determine differences.
Thirty-seven patients with DSOM and 32 patients with CFD were included (mean ages, 24.2 and 28.4 years, respectively); both groups showed a female predilection. DSOM (91.9%) and CFD (84.4%) were mainly unilateral. Patients with DSOM mainly presented with pain (94.6%), soft-tissue swelling (100.0%), and trismus (54.1%), whereas those with CFD did not experience pain (90.6%) and showed bone enlargement (87.5%) without trismus (6.3%). Panoramic radiographs and computed tomography scans of patients with DSOM showed subperiosteal bone formation, cortex lysis, and poorly demarcated cortex, whereas those patients with CFD mainly showed moderate-to-severe bone expansion, well-demarcated cortex, and tooth and mandibular canal displacement.
These findings emphasize the importance of clinical and radiographic features in differentiating between DSOM and CFD. Pain, soft-tissue or bone-tissue swelling, subperiosteal bone formation, clarity of the boundary of the cortex and medulla, and continuity of the cortical bone are key points facilitating differentiation.
Application of pamidronate disodium for the treatment of diffuse sclerosing osteomyelitis of the mandible: A clinical study
2020, Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology
Citation Excerpt :
The etiologies and pathogenesis of DSOM are still unclear. Previous studies have suggested that DSOM is a type of SAPHO syndrome, and thus, its treatment is similar to the treatment for SAHPO syndrome.4,5 Currently, the most commonly used methods to treat DSOM include surgery, occlusal splint therapy, hyperbaric oxygen therapy, and drug therapy.2,12,14-16
This study aimed to assess the clinical effectiveness of using pamidronate disodium to treat patients with diffuse sclerosing osteomyelitis of the mandible (DSOM ).
Patients with DSOM who were hospitalized in the Department of Oral and Maxillofacial Surgery, Peking University Hospital of Stomatology (Beijing, China), between March 2018 and March 2019 were included. All patients were treated with intravenous administration of pamidronate disodium for 3 days. Pain intensity, mouth opening, blood workup findings, imaging examination findings, and incidence of complications before and after treatment and during the follow-up period, were recorded and compared.
Forty-three patients were treated with pamidronate disodium. Adverse reactions, including fever, hypocalcemia, hypokalemia, and diarrhea, were observed during treatment. Fourteen patients had moderate to severe pain before treatment, and the pain was markedly alleviated after days 2 and 3 of treatment. Pain and swelling were found to have disappeared at the 6-month follow-up in 39 patients (90.7%). Mean mouth opening increased from 28.5 mm to 38.1 mm. Imaging examinations showed that the bone mass in the affected area had markedly improved in 32 patients (74.4%).
Pamidronate disodium alleviated pain and swelling and improved mouth opening in patients with DSOM. Follow-up after 6 to 18 months demonstrated stable treatment effectiveness.
Outcome of different treatments for chronic diffuse sclerosing osteomyelitis of the mandible: a systematic review of published papers
2020, British Journal of Oral and Maxillofacial Surgery
Treating chronic diffuse sclerosing osteomyelitis (DSO) is challenging and many treatments have been reported. However, we know of no standard protocol or guidelines. In this systematic review of relevant publications we provide an overview of the different treatments used. We made an electronic search of PubMed, Medline, Embase, Web of Science, and the Cochrane Library databases, for papers that described the treatment of DSO of the mandible. The search yielded 48 papers that applied to all inclusion criteria, resulting in 16 case reports, 13 case series, 18 retrospective clinical cohort studies, and one randomised controlled trial. Reported treatment options included different operations; the use of antibiotics, anti-inflammatories, and antiresorptive medication; conservative treatment; and hyperbaric oxygen. Surgical treatment resulted in a low success rate and was associated with higher morbidity than other treatments. Conservative treatment, and that of bisphosphonates, yielded more promising results, so conservative treatment and bisphosphonates seem to be the most promising therapeutic options. However, because of the high risk of bias, no firm conclusions can be drawn, and larger studies with clear inclusion criteria and specified endpoints are needed.

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Is diffuse sclerosing osteomyelitis of the mandible part of the synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome?: Analysis of seven cases

Abstract

Bailliere's Clin Rheum

Rheum Dis Clin North Am

Oral Surg Oral Med Oral Pathol

Diffuse sclerosing osteomyelitis of the mandible

Int J Oral Surg

Diffuse sclerosing osteomyelitis of the mandible: clinical radiographic and histologic findings in twenty seven patients

J Oral Maxillofac Surg

Le syndrome acne´pustulose hyperostose ostei¨te (SAPHO)

Re´sultats d'une enqueˆte nationale: 85 observations

Rev Rhum Mal Osteoartic

Reiter's syndrome and reactive arthritis

Clinical features of 33 cases with pustulotic arthroosteitis

Ann Rheum Dis