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Hagit Peleg, Eldad Ben-Chetrit
PURPOSE OF REVIEW: This article addresses the prevalence and relationship between autoinflammatory diseases and vasculitis. RECENT FINDINGS: Autoimmune diseases (AIDs) are a group of syndromes characterized by episodes of unprovoked inflammation due to dysregulation of the innate immune system. Despite the common occurrence of rashes and other skin lesions in these diseases, vasculitis is reported in only a few. On the other hand, neutrophilic dermatoses are more prevalent...
October 13, 2016: Current Opinion in Rheumatology
A Schnabel, U Range, G Hahn, T Siepmann, R Berner, C M Hedrich
Historically, osteomyelitis was considered an infectious disorder. More recently, inflammatory mechanisms were recognized causing a significant proportion of pediatric osteomyelitis. This study was to compare characteristics of children with chronic non-bacterial (CNO) and bacterial osteomyelitis (BOM). A chart review of osteomyelitis patients from the departments of pediatrics, pediatric surgery, orthopedic surgery, and oral and maxillofacial surgery was conducted in a tertiary referral center, covering the years 2004-2014...
October 11, 2016: Rheumatology International
Noa Safra, Peta L Hitchens, Emanual Maverakis, Anupam Mitra, Courtney Korff, Eric Johnson, Amir Kol, Michael J Bannasch, Niels C Pedersen, Danika L Bannasch
Metaphyseal osteopathy (MO) (hypertrophic osteodystrophy) is a developmental disorder of unexplained etiology affecting dogs during rapid growth. Affected dogs experience relapsing episodes of lytic/sclerotic metaphyseal lesions and systemic inflammation. MO is rare in the general dog population; however, some breeds (Weimaraner, Great Dane and Irish Setter) have a much higher incidence, supporting a hereditary etiology. Autoinflammatory childhood disorders of parallel presentation such as chronic recurrent multifocal osteomyelitis (CRMO), and deficiency of interleukin-1 receptor antagonist (DIRA), involve impaired innate immunity pathways and aberrant cytokine production...
October 15, 2016: Veterinary Immunology and Immunopathology
Matthias Christian Wurm, Ines Brecht, Michael Lell, Kathrin Brunner, Konstantinos Theodorou Mitsimponas, Martin Chada, Julia Jahn, Friedrich-Wilhelm Neukam, Cornelius von Wilmowsky
BACKGROUND: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare acquired inflammatory skeletal disorder of unknown origin. CRMO was first described by Gideon in 1972 and mainly affects children and young adults of female gender. The CRMO is part of the clinical picture of non-bacterial Osteomyelitis (NBO) and typically presents a relapsing recurring course with both remission and spontaneous exacerbation. CRMO is typically encountered in the limbs and the metaphysis of long bones in particular...
2016: BMC Oral Health
Sigrun R Hofmann, Anja Schnabel, Angela Rösen-Wolff, Henner Morbach, Hermann J Girschick, Christian M Hedrich
Chronic nonbacterial osteomyelitis (CNO) is an autoinflammatory bone disorder, covering a clinical spectrum with asymptomatic inflammation of single bones at the one end, and chronic recurrent multifocal osteomyelitis (CRMO) at the other end. The exact molecular pathophysiology of CNO remains largely unknown. Provided familial clusters and the association with inflammatory disorders of the skin and intestine suggest a genetic predisposition. Recently, profound dysregulation of cytokine responses was demonstrated in CRMO...
September 1, 2016: Journal of Rheumatology
M R Roderick, R Shah, V Rogers, A Finn, A V Ramanan
BACKGROUND: Chronic recurrent multifocal osteomyelitis (CRMO) is a little known inflammatory bone disease occurring primarily in children and adolescents. Delays in referral and diagnosis may lead to prolonged courses of antibiotics with in-patient care, unnecessary radiation exposure from multiple plain radiographs or bone scans and repeated surgery including bone biopsies. Children (aged < 18 years) diagnosed with CRMO between January 2005 and December 2012, reviewed at Bristol Royal Hospital for Children were included and all available data collected...
2016: Pediatric Rheumatology Online Journal
Bonnie L Padwa, Kelley Dentino, Caroline D Robson, Sook Bin Woo, Kyle Kurek, Cory M Resnick
PURPOSE: Chronic nonbacterial osteomyelitis (CNO) is a focal sterile inflammatory osteitis in children that most commonly develops in the long bones, but can occur in any bone. The disease course is variable, ranging from acute and self-resolving isolated lesions to chronic recurrent multifocal osteomyelitis (CRMO), which is frequently associated with extraosseous inflammatory disease. The purpose of this study was to present our clinical experience with CNO of the mandible in children...
May 26, 2016: Journal of Oral and Maxillofacial Surgery
Daniel J Matthew, Sang Hoon Oh, Andrew Sevy, Michael D Morse
Supersonically cooled CrW was studied using resonant two-photon ionization spectroscopy. The vibronically resolved spectrum was recorded over the region 21 100 to 23 400 cm(-1), showing a very large number of bands. Seventeen of these bands, across three different isotopologues, were rotationally resolved and analyzed. All were found to arise from the ground (1)Σ(+) state of the molecule and to terminate on states with Ω' = 0. The average r0 bond length across the three isotopic forms was determined to be 1...
June 7, 2016: Journal of Chemical Physics
N H von der Höh, A Völker, D Jeszenszky, C-E Heyde
Chronic non-bacterial osteomyelitis (CNO) in childhood and adolescence is a non-infectious autoinflammatory disease of the bone with partial involvement of adjacent joints and soft tissue. The etiology is unknown. The disease can occur singular or recurrent. Individual bones can be affected and multiple lesions can occur. Chronic recurrent multifocal osteomyelitis (CRMO) shows the whole picture of CNO. Accompanying but temporally independent of the bouts of osteomyelitis, some patients show manifestations in the skin, eyes, lungs and the gastrointestinal tract...
June 2016: Der Orthopäde
N Hendel, M K Akmatov, J Hamel, C Vogelberg, F Pessler
We aimed to evaluate the fraction of exhaled nitric oxide (FENO50) and deaerated exhaled breath condensate pH (dEBCpH) as non-invasive markers of subclinical airway inflammation in pediatric patients with rheumatologic disorders. We determined FENO50 and dEBCpH in a prospective study spanning at least 12 months, comprising 85 pediatric patients with rheumatologic disorders, including juvenile idiopathic arthritis (JIA, n  =  63), chronic recurrent multifocal osteomyelitis (CRMO, n  =  6), systemic lupus erythematosus (SLE, n  =  3), juvenile dermatomyositis (JDM, n  =  1) and other rheumatic disorders (n  =  12)...
June 2016: Journal of Breath Research
Sigrun Renate Hofmann, Anne Sophie Kubasch, Ursula Range, Martin Walther Laass, Henner Morbach, Hermann Joseph Girschick, Christian Michael Hedrich
Chronic recurrent multifocal osteomyelitis (CRMO), the most severe form of chronic nonbacterial osteomyelitis, is an autoinflammatory bone disorder. A timely diagnosis and treatment initiation is complicated by the absence of widely accepted diagnostic criteria and an incomplete pathophysiological understanding. The aim of this study was to determine biomarkers for the diagnosis and follow-up of CRMO. Serum of 56 CRMO patients was collected at the time of diagnosis. As controls, sera from treatment-naïve age-matched patients with Crohn's disease (N = 62) or JIA (N = 28) as well as healthy individuals (N = 62) were collected...
June 2016: Rheumatology International
Nadine Leclair, Gregor Thörmer, Ina Sorge, Lutz Ritter, Volker Schuster, Franz Wolfgang Hirsch
OBJECTIVE: Chronic recurrent multifocal osteomyelitis/ chronic non-bacterial osteomyelitis (CRMO/ CNO) is a rare auto-inflammatory disease and typically manifests in terms of musculoskeletal pain. Because of a high frequency of musculoskeletal disorders in children/ adolescents, it can be quite challenging to distinguish CRMO/ CNO from nonspecific musculoskeletal pain or from malignancies. The purpose of this study was to evaluate the visibility of CRMO lesions in a whole-body diffusion-weighted imaging (WB-DWI) technique and its potential clinical value to better characterize MR-visible lesions...
2016: PloS One
M Barrani, F Massei, M Scaglione, A Paolicchi, S Vitali, E M Ciancia, S Crovella, M C Caparello, R Consolini
BACKGROUND: Chronic recurrent multifocal osteomyelitis (CRMO) is a rare condition that commonly affects the clavicle and pelvis. CASE PRESENTATION: We report here a case a 12 years old girl with CRMO arising with recurrent episodes of left supraorbital headache, followed by the appearance of a periorbital dyschromia. Magnetic resonance imaging (MRI) of the skull and orbits revealed an important subacute inflammatory process. Few months after, the child presented a painful swelling of the left clavicle; the histological examination of the related biopsy allowed to establish the diagnosis of CRMO...
2015: Pediatric Rheumatology Online Journal
S R Hofmann, A S Kubasch, C Ioannidis, A Rösen-Wolff, H J Girschick, H Morbach, C M Hedrich
Chronic recurrent multifocal osteomyelitis (CRMO) is characterized by reduced activation of protein kinases ERK1 and 2 in monocytes resulting in impaired IL-10 expression. IL10 and its homologs IL19 and IL20 are organized in the IL10 cluster on chromosome 1q32. IL-10 and IL-19 are immune-regulatory cytokines, while IL-20 acts in a pro-inflammatory manner. The NLRP3 inflammasome, a multi-protein complex forming in response to innate stimuli, mediates IL-1β cleavage and release. Here, we investigated IL-10-related cytokine expression in CRMO monocytes, underlying molecular events, and effects on inflammatory responses...
December 2015: Clinical Immunology: the Official Journal of the Clinical Immunology Society
Deniz Aygun, Kenan Barut, Yildiz Camcioglu, Ozgur Kasapcopur
Chronic recurrent multifocal osteomyelitis (CRMO) is a rare non-infectious inflammatory bone disease of unknown aetiology. CRMO mainly affects the metaphyses of long bones and spine in children and young adolescents. It presents with recurrent episodes of bone pain and fever, resembling bacterial osteomyelitis, but cultures of lesions are sterile and it is unresponsive to antibiotic therapy. We report a case of a 3-year-old boy diagnosed with CRMO, who was initially treated for bacterial osteomyelitis, and received prolonged antibiotic therapy for chronic pain, and swelling of mandible and ulna...
August 25, 2015: BMJ Case Reports
C van Ommen, J Dehoorne, Fr De Baets, S Vande Velde, M Van Winckel, S Van Biervliet
Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disease of unknown etiology, most commonly affecting the metaphysis of long bones, especially the tibia, femur and clavicle. The clinical spectrum varies from self-limited uni-or multi-focal lesions to chronic recurrent courses. Diagnosis is based on clinical, radiologic and pathological findings, is probably underdiagnosed due to poor recognition of the disease. A dysregulated innate immunity causes immune cell infiltration of the bones with subsequent osteoclast activation leading to sterile bone lesions...
June 2015: Acta Gastro-enterologica Belgica
Michael J Ombrello
Monogenic diseases usually demonstrate Mendelian inheritance and are caused by highly penetrant genetic variants of a single gene. In contrast, genetically complex diseases arise from a combination of multiple genetic and environmental factors. The concept of autoinflammation originally emerged from the identification of individual, activating lesions of the innate immune system as the molecular basis of the hereditary periodic fever syndromes. In addition to these rare, monogenic forms of autoinflammation, genetically complex autoinflammatory diseases like the periodic fever, aphthous stomatitis, pharyngitis, and cervical adenitis (PFAPA) syndrome, chronic recurrent multifocal osteomyelitis (CRMO), Behçet's disease, and systemic arthritis also fulfill the definition of autoinflammatory diseases-namely, the development of apparently unprovoked episodes of inflammation without identifiable exogenous triggers and in the absence of autoimmunity...
July 2015: Seminars in Immunopathology
Agnes M Voit, Andreas P Arnoldi, Hassan Douis, Felicitas Bleisteiner, Moritz K Jansson, Maximilian F Reiser, Sabine Weckbach, Annette F Jansson
OBJECTIVE: (1) To examine how many patients have clinically and/or radiologically active chronic recurrent multifocal osteomyelitis (CRMO) ≥ 10 years after first onset of symptoms, and (2) to compare clinical and whole-body magnetic resonance imaging (WB-MRI) findings. METHODS: Seventeen patients (82% women) who were diagnosed with childhood-onset CRMO at least 10 years (average 12) before reexamination were reevaluated. Patients completed a standardized questionnaire, and underwent clinical and laboratory investigation and WB-MRI...
August 2015: Journal of Rheumatology
Pavla Walsh, Prudence J Manners, Jack Vercoe, David Burgner, Kevin J Murray
OBJECTIVE: To describe the clinical features, management and outcome of 34 children with chronic recurrent multifocal osteomyelitis (CRMO) diagnosed at a single centre over 9 years. METHODS: All children identified with CRMO for the period 2005-13 were identified from a prospectively collected database, with additional data from hospital records. RESULTS: Thirty-four patients, 21 female and 13 male, were identified. The average age at symptom onset was 9...
September 2015: Rheumatology
Cheng William Hong, Edward C Hsiao, Andrew E Horvai, Thomas M Link
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...
September 2015: Skeletal Radiology
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