Chronic recurrent multifocal osteomyelitis: an evolving clinical and radiological spectrum

Sundaram, Murali; McDonald, Douglas J.; Engel, Elizabeth; Rotman, Mitchell B.; Siegfried, Elaine C.

doi:10.1007/s002560050091

Cited by 72 publications

(39 citation statements)

References 2 publications

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“…Rheumatologists and orthopedic surgeons increasingly recognize bone lesions as nonbacterial, even if well-known associated symptoms such as palmoplantar pustolosis or hyperostosis are lacking (18,19). Particularly in immunocompetent and otherwise-healthy children and adults, NBO might be much more frequent than bacterial osteomyelitis.…”

Section: Discussionmentioning

confidence: 99%

Clinical score for nonbacterial osteitis in children and adults

Jansson¹,

Müller²,

Gliera³

et al. 2009

Arthritis & Rheumatism

137

111

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Objective. To accurately differentiate nonbacterial osteitis (NBO) from other bone lesions by applying a clinical score through the use of validated diagnostic criteria.Methods. A retrospective study was conducted to assess data on patients from a pediatric clinic and an orthopedic tertiary care clinic, using administrative International Classification of Diseases codes as well as laboratory and department records from 1996 to 2006. Two hundred twenty-four patients older than age 3 years who had either NBO (n ‫؍‬ 102), proven bacterial osteomyelitis (n ‫؍‬ 22), malignant bone tumors (n ‫؍‬ 48), or benign bone tumors (n ‫؍‬ 52) were identified by chart review. Univariate logistic regression was used to determine associations of single risk factors with a diagnosis of NBO, and multivariable logistic regression was used to assess simultaneous risk factor associations with NBO. Conclusion. The proposed scoring system helps to facilitate the diagnostic process in patients with suspected NBO. Use of this system might spare unnecessary invasive diagnostic and therapeutic procedures. Results. NBO was best predicted by a normal blood cell count (odds ratio

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Section: Discussionmentioning

confidence: 99%

Clinical score for nonbacterial osteitis in children and adults

Jansson¹,

Müller²,

Gliera³

et al. 2009

Arthritis & Rheumatism

137

111

View full text Add to dashboard Cite

show abstract

“…This is very similar to human CRMO, in which affected individuals develop histologically similar bone inflammation, with many individuals developing an associated inflammatory disorder, usually involving the skin. Palmoplantar pustulosis is the most common inflammatory dermatosis associated with CRMO but psoriasis [3,4,24,25], Sweet syndrome [16,[26][27][28], pyoderma gangrenosum [6,7,9,[28][29][30] and generalized pustulosis may also occur [31]. Gastrointestinal inflammation manifesting as Crohn's disease or ulcerative colitis has also been reported in association with CRMO [6][7][8][9] but we have yet to detect inflammation in the small or large bowel in cmo mice.…”

Section: Discussionmentioning

confidence: 99%

A missense mutation in pstpip2 is associated with the murine autoinflammatory disorder chronic multifocal osteomyelitis

Ferguson

Bing

Vasef

et al. 2006

Bone

195

154

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Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory disorder that primarily affects bone but is often accompanied by inflammation of the skin and/or gastrointestinal tract. The etiology is unknown but evidence suggests a genetic component to disease susceptibility. Although most cases of CRMO are sporadic, there is an autosomal recessive syndromic form of the disease, called Majeed syndrome, which is due to homozygous mutations in LPIN2. In addition, there is a phenotypically similar mouse, called cmo (chronic multifocal osteomyelitis) in which the disease is inherited as an autosomal recessive disorder. The cmo locus has been mapped to murine chromosome 18. In this report, we describe phenotypic abnormalities in the cmo mouse that include bone, cartilage and skin inflammation. Utilizing a backcross breeding strategy, we refined the cmo locus to a 1.3 Mb region on murine chromosome 18. Within the refined region was the gene pstpip2, which shares significant sequence homology to the PSTPIP1. Mutations in PSTPIP1 have been shown to cause the autoinflammatory disorder PAPA syndrome (pyogenic arthritis, pyoderma gangrenosum and acne). Mutation analysis, utilizing direct sequencing, revealed a single base pair change c.293T → C in the pstpip2 gene resulting in a highly conserved leucine at amino acid 98 being replaced by a proline (L98P). No other mutations were found in the coding sequence of the remaining genes in the refined interval, although a 50 kb gap remains unexplored. These data suggest that mutations in pstpip2 may be the genetic explanation for the autoinflammatory phenotype seen in the cmo mouse.

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“…Some consider CRMO to be the pediatric presentation of SAPHO syndrome, which generally affects adults, although as in this case, CRMO-like presentations can occur in adults. Presentations of CRMO in adults, as with children, occur more commonly in women [14,15]. These patients are often febrile, and while their complaints are usually Fig.…”

Section: Discussionmentioning

confidence: 99%

“…5 Axial PET/CT image at the level of the sacroiliac joints. There is widening of the sacroiliac joint and mild uptake (solid arrow) seen on the left correlating with the patient's symptoms focused on a specific site, imaging reveals other sites of disease involvement that are typically asymptomatic [14][15][16]. In one other case of CRMO in an adult man, he subsequently developed adult-onset Still's disease and was successfully treated with anakinra [16].…”

Section: Discussionmentioning

confidence: 99%

Chronic recurrent multifocal osteomyelitis with an atypical presentation in an adult man

et al. 2015

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We present the case of a 33-year-old man with no significant medical history who developed right scapular pain, left-sided sacroiliac joint pain, and lower back pain, and was eventually diagnosed with chronic recurrent multifocal osteomyelitis (CRMO). Imaging demonstrated multiple scattered T2-hyperintense lesions on MRI at the spine and the left SI joint, some of which progressed and one regressed in size on follow-up. Histopathology demonstrated only non-specific chronic inflammation compatible with CRMO. No evidence of infectious organisms or neoplastic processes was found. The pain was relapsing and remitting in nature. Laboratory investigations were notable for no evidence of hematologic malignancy or infection, but only a mild increase in alkaline phosphatase. This case highlights that CRMO, despite being thought of as a childhood-onset disease, can present in adults as well, and also provides illustrative examples of imaging and histological findings.

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Chronic recurrent multifocal osteomyelitis: an evolving clinical and radiological spectrum

Abstract: The pattern and distribution of skeletal lesions in CRMO are well recognized in the pediatric age group. The unusual clinical and/or radiological features discussed herein suggests that this is a disease that continues to evolve with a broader spectrum of features than recognized.

Cited by 72 publications

References 2 publications

Clinical score for nonbacterial osteitis in children and adults

Clinical score for nonbacterial osteitis in children and adults

A missense mutation in pstpip2 is associated with the murine autoinflammatory disorder chronic multifocal osteomyelitis

Chronic recurrent multifocal osteomyelitis with an atypical presentation in an adult man

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