Chronic Osteomyelitislike Disease With Negative Bacterial Cultures

Pelkonen, P.; Ryöppy, Soini; Jääskeläinen, J; Rapola, Juhani; Repo, Heikki; Kaitila, Ilkka

doi:10.1001/archpedi.1988.02150110045017

Cited by 43 publications

(57 citation statements)

References 20 publications

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“…12 Additional supportive evidence includes similar skin findings associated with either DSO or CRMO. Palmoplantar pustulosis has been described with both CRMO 2,6,7,10,13 and DSO. 14 Psoriasis has also been described with both CRMO 2,13 and DSO.…”

Section: Discussionmentioning

confidence: 99%

Chronic Recurrent Multifocal Osteomyelitis of the Spine and Mandible: Case Report and Review of the Literature

Chun

2004

Pediatrics

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ABSTRACT. At initial presentation, chronic recurrent multifocal osteomyelitis may mimic acute hematogenous osteomyelitis; however, cultures of affected bone are sterile. Nuclear scintigraphy identifies additional foci of involvement that present concurrently or sequentially. Unlike acute bacterial osteomyelitis, chronic recurrent multifocal osteomyelitis seems unaffected by antibiotic therapy and typically responds to treatment with antiinflammatory drugs. Surgical decortication has been reported for refractory cases. The case presented here illustrates the rare involvement of the mandible after initial presentation in the spine of a 4-year-old girl and the refractory nature of the disease over 6 years despite treatment with various medical and surgical therapies. Pediatrics 2004;113:e380 -e384. URL: http://www.pediatrics. org/cgi/content/full/113/4/e380; bone diseases, osteitis, hyperostosis.ABBREVIATIONS. CRMO, chronic recurrent multifocal osteomyelitis; MRI, magnetic resonance imaging; ESR, erythrocyte sedimentation rate; CT, computed tomography; AFB, acid-fast bacilli; Ig, immunoglobulin; PCR, polymerase chain reaction; DSO, diffuse sclerosing osteomyelitis; SAPHO, synovitis, acne, palmoplantar pustulosis, hyperostosis, osteitis syndrome. G iedion et al 1 first described acquired, culturenegative, multifocal osteomyelitis in children in 1972 and named the syndrome "subacute and chronic symmetrical osteomyelitis." In the original report, the disorder involved the metaphyses of long bones either simultaneously or successively. Subsequent publications reported involvement of additional bone sites and referred to the disorder as chronic multifocal osteomyelitis 2 and chronic recurrent multifocal osteomyelitis (CRMO). 3 Pediatric cases with mandibular involvement have been reported in the oral surgery, radiology, orthopedics, and dermatology literature.Unlike the previously reported 4 cases of CRMO with mandibular involvement in the English-language literature, 4 -7 this patient developed CRMO initially in the spine and thereafter developed mandibular involvement. She did not have prior dental infection or skin manifestations associated with CRMO. Additional aspects of interest include the progressive nature of the mandibular involvement and the disease refractoriness to different therapies over 6 years. CASE REPORT Year 1In February 1997, a previously healthy, 4-year, 2-month-old white female was seen in the clinic for left-sided lumbar pain and a fever to 104°F. There was no history of trauma, and the examination was normal. She was treated for muscle strain with ibuprofen. Over the next 3 months, the pain persisted and woke her at night, and she refused to run or jump. A bone scan revealed increased uptake at the left side of the S1 vertebra on delayed images. Magnetic resonance imaging (MRI) revealed a well-circumscribed, irregularly shaped lytic lesion of S1 extending into the sacral ala (Fig 1). Her erythrocyte sedimentation rate (ESR) was 27 mm/hour, and she underwent a computed tomography (CT)-guided perc...

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Section: Discussionmentioning

confidence: 99%

Chronic Recurrent Multifocal Osteomyelitis of the Spine and Mandible: Case Report and Review of the Literature

Chun

2004

Pediatrics

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“…17,68,92,110 Overall, approximately 30% continue to have persistent disease. 48,89 Some are left with permanent skeletal problems, including vertebra plana, vertebral fractures, vertebral fusion, secondary scoliosis, limb length discrepancy, thoracic outlet syndrome, abnormal jaw function, or persistently enlarged bone due to sclerosis. Over time, individuals can show an evolution into classic spondyloarthropathy, requiring longterm rheumatologic care.…”

Section: Prognosismentioning

confidence: 99%

Autoinflammatory Diseases Predominantly Affecting Bone and Joints

Ferguson

Goldbach‐Mansky

2014

Stiehm's Immune Deficiencies

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“…An Australian review documented patients with late physical deformities including leg length disparity [33]. In another small series, complications of thoracic outlet syndrome and progression with Wegener's disease were cited [58].…”

Section: Clinical Characteristicsmentioning

confidence: 99%

“…Most studies find elevated erythrocyte sedimentation rate [18,58,85], but one review found largely normal values for the same [31]. Immunoglobulin studies tend to show no or only non-specific elevation, e.g., up to 10% may have elevated IgM [18,31,58,86]. Most assessments for HLA-B27, antinuclear antibody, and rheumatoid factor are negative, although one report indicated a higher frequency of positive HLA-B27 compared to the general population (21% vs. 9%) [31].…”

Section: Systemic Chemistry and Immunologymentioning

confidence: 99%

Chronic multifocal osteomyelitis: Is infectious causation a moot point?

Cimolai

2011

Journal of Infection and Public Health

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Chronic multifocal osteomyelitis (CMO) is an uncommon disease entity with descriptions possibly emanating from the medical literature over one century ago, and there are numerous disease entities which have been historically detailed and which are probably synonymous. The illness is one of chronicity with exacerbating and remitting focal bony lesions. The differential diagnosis for a bony lesion which ultimately proves to be CMO is initially quite broad. There is no absolute pathognomonic clinical finding, and the diagnosis is highly dependent on clinical course, histopathology, and an absence of microbial infection. Recent studies have focused on immune dysfunction or dysregulation, and there are now many other diseases which are inflammatory in nature and which have been diagnosed among patients with CMO. Despite the aforementioned, the potential for direct infectious causation or indirect causation by infectious stimulation of immunity cannot be entirely excluded. Infection as a mechanism for pathogenesis must continue to be entertained. Multi-centre studies are key to future research.

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Chronic Osteomyelitislike Disease With Negative Bacterial Cultures

Cited by 43 publications

References 20 publications

Chronic Recurrent Multifocal Osteomyelitis of the Spine and Mandible: Case Report and Review of the Literature

Chronic Recurrent Multifocal Osteomyelitis of the Spine and Mandible: Case Report and Review of the Literature

Autoinflammatory Diseases Predominantly Affecting Bone and Joints

Chronic multifocal osteomyelitis: Is infectious causation a moot point?

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