Chronic sclerosing osteomyelitis of the clavicle. A manifestation of chronic recurrent multifocal osteomyelitis

The cases of five children/adolescents and two young adults with unilateral chronic osteomyelitis of the clavicle are reported. The clinical course was prolonged and characterized by intermittent periods of exacerbation followed by improvement. Extensive investigations revealed no causative organisms and the disease seemed resistant to antibiotic therapy. The only laboratory abnormality was elevated ESR, present in five cases. Osteomyelitis of other bones was detected in four cases and pustulosis palmoplantaris in two, suggesting that the disease is a manifestation of chronic recurrent multifocal osteomyelitis.     

Treatment of chronic recurrent multifocal osteomyelitis with bisphosphonates in children

ObjectivesAssessment of bisphosphonates efficiency in the therapy of NSAIDs-refractory Chronic Recurrent Multifocal Osteomyelitis.MethodsRetrospective analysis of records of patients treated for Chronic Recurrent Multifocal Osteomyelitis between 2012 and 2018.ResultsBetween 2012 and 2018, 76 children and adolescents were diagnosed with Chronic Recurrent Multifocal Osteomyelitis in our department. All patients underwent an initial course of NSAIDs therapy that provided a remission in 46% of cases. Of 41 NSAIDs-resistant cases, 7 patients were male and 34 were female. Disease started meanly in the age of 10. Most frequently pain localised in foot, clavicle and hip. In presented group, pamidronate was administered intravenously in the dose of 1 mg/kg/day for 3 days. Patients received 6 series (1–17 series) on average with mean interval of 10 weeks (4–14 weeks). Our observations demonstrated rapid decrease of symptoms intensity after first dose of pamidronate with relapse of pain after 3–4 weeks. The frequency of pamidronate dosage was dependent of patient's symptoms. No serious adverse effects were reported. We finished the therapy after complete remission of symptoms and complete bone remodelling in imaging. Of 41 patients, 32 achieved remission and 9 continue their therapy. In remission group patients received 7 series of pamidronate on average and their treatment lasted meanly 20 months.ConclusionsPamidronate is a safe and efficient method of CRMO therapy, particularly in cases refractory to NSAIDs treatment. Treatment with pamidronate provides both symptomatic relief as well as normalisation of bone morphology.     

Chronic recurrent multifocal osteomyelitis

Chronic recurrent multifocal osteomyelitis (CRMO), also known as chronic nonbacterial osteomyelitis, is an orphan disease that manifests as recurrent flares of inflammatory bone pain with or without a fever. The pain is related to one or more foci of nonbacterial osteomyelitis. To distinguish unifocal CRMO from a tumor or an infection, a bone biopsy is required in nearly all patients and a trial of antibiotic therapy in many. CRMO is now considered the pediatric equivalent of SAPHO syndrome, and recent data suggest that CRMO should be classified among the autoinflammatory diseases. The treatment of CRMO is not standardized. Although no randomized placebo-controlled trials are available, there is general agreement that nonsteroidal antiinflammatory drugs constitute the best first-line treatment and that bisphosphonates and biotherapies such as TNFα antagonists are effective in the most severe forms. Although CRMO is considered a benign disease, recent data suggest an up to 50% rate of residual impairments despite optimal management.     

Chronic recurrent multifocal osteomyelitis

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare, inflammatory, skeletal disease of unknown origin, which mainly affects children and adolescents in terms of cleido-spondylo-metaphysal skeletal inflammation. Only 10% of the patients are older than 20 years. To date, only about 200 cases have been reported in the literature. In the course of the disease, the initial radiological signs are osteolysis followed by sclerosis and hyperostosis in the end stage. The histological investigations reveal chronic inflammatory infiltrates with lymphocytes and hyperostosis. Although the prognosis of CRMO, to our current understanding, is self limiting, serious complications have been reported such as pathological fractures and compression fractures of the spine. A recently recommended therapy scheme is based on the administration of azithromycin combined with calcitonin. We present the case of a 25 year old female patient who has suffered from CRMO for 1.5 years with the cervical spine and the manubrium sterni being affected. The current state of diagnosis, therapy, and prognostic outlook of this rare disease are discussed.     

Acute multifocal haematogenous osteomyelitis in children

Purpose  

Haematogenous multifocal osteomyelitis in children represents a dangerous form of osteomyelitis in which sepsis can develop quickly if it is not treated early. A retrospective analysis of 26 children with acute multifocal haematogenous osteomyelitis over a period of 5 years was undertaken in order to assess the clinical presentation, infective organism, laboratory investigations and risk factors involved.     

Chronic recurrent multifocal osteomyelitis causing spinal cord compression

Chronic recurrent multifocal osteomyelitis (CRMO) is a very rare condition of unknown etiology and most commonly occurs during childhood or adolescence. The purpose of this paper is to present a case of CRMO in a vertebral location with severe kyphosis, spinal cord compression, and neurological dysfunction requiring anterior decompression and fusion. After 12 weeks, the patient was physically able to return to school. At 2-year follow-up, neurological and functional outcomes are fair. Magnetic resonance imaging shows good restoration of the sagittal spine alignment despite residual mild kyphosis, and restoration of a normal sagittal diameter of the spinal canal. Received: 4 August 1997 Revised: 16 January 1998 Accepted: 22 January 1998     

Chronic recurrent multifocal osteomyelitis: a follow-up study

Thirteen children/adolescents and two young adults with unifocal or multifocal nonpyogenic inflammatory bony lesions with a prolonged, fluctuating course are reported. The lesions were most often located at the metaphyseal region of tubular bones and the clavicle, but also at the spine, ischiopubic bone, and the sacroiliac joint. Progressive sclerosis and hyperostosis occurred mostly in the clavicle and occasionally in the tibia, femur, metatarsal, and ischiopubic bone, linking the changes to those described under the name Garré osteomyelitis. Seven patients had pustulosis palmoplantaris, and the skeletal disorder may be regarded a form of pustulotic arthroosteitis.     

Chronic recurrent multifocal osteomyelitis after acute lymphoblastic leukaemia

Summary We describe an 8 year old girl who developed chronic recurrent multifocal osteomyelitis (CRMO) in the ilium and clavicle. Treatment for an acute lymphoblastic leukaemia had been finished two months before. After antibiotic therapy, the clinical symptoms improved and no fresh lesions appeared. The aetiology of CRMO is unknown, but we feel that infection may precipitate an immunological reaction.

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Résumé Les auteurs rapportent l'observation d'une fillette de 8 ans, qui présentait une ostéomyélite récurrente chronique multifocale (OCRM), atteignant l'os iliaque et la clavicule. Elle était d'abord soignée pour une leucémie lymphoblastique aigu? dont le traitement était terminé deux mois avant la survenue de l'ORCM. Un traitement antibiotique entra?na l'amélioration clinique sans qu'apparaissent d'autres localisations. L'étiologie de cette maladie est inconnue, mais nous estimons comme probable qu'une infection soit la cause d'une réaction immunologique.

     

Normophosphatemic type tumoral calcinosis associated with chronic recurrent multifocal osteomyelitis: a case report

Chronic recurrent multifocal osteomyelitis (CRMO) and tumoral calcinosis are two distinct musculoskeletal diseases with unclear etiopathogenesis. Previously, two CRMO cases with associated tumoral calcinosis were reported. We report a patient who developed tumoral calcinosis after the surgical treatment of CRMO. To our knowledge it is the third patient in whom tumoral calcinosis developed sporadically during follow-up for CRMO.     

Chronic recurrent multifocal osteomyelitis. Association with vertebra plana

In seven children who had chronic recurrent multifocal osteomyelitis, the radiographic and histological findings were consistent with those of osteomyelitis, but the results of cultures were negative. We studied the clinical, radiographic, histological, and microbiological findings in these patients, who had a total of thirty-nine lesions. The lesions occurred most frequently in the spine, tibia, and femur; three patients had vertebra plana. The natural history of chronic recurrent multifocal osteomyelitis appears to be slow, spontaneous resolution of the osseous lesions without specific treatment. The diagnosis is one of exclusion. Biopsy is recommended, and results of cultures must be negative before therapy with antibiotics can be withheld.     

Chronic recurrent multifocal osteomyelitis: a distinct clinical entity

We reviewed the cases of five children with the diagnosis of chronic recurrent multifocal osteomyelitis (CRMO) and compared and contrasted them to 11 cases of subacute osteomyelitis. Significant differences were found between these two groups in the number of cases with positive biopsy cultures, number of clinical episodes, and number of bones involved. In CRMO, cultures are negative, and recurrent clinical episodes involve different bones at different times. The data indicate that CRMO is a distinct clinical entity, different from subacute osteomyelitis; it is a benign, self-limiting inflammatory disease of bone, and no chronic problems have occurred as a result of CRMO. Restraint in antibiotic treatment and in performing repeated biopsies is indicated in CRMO.     

Superinfection of chronic multifocal osteomyelitis with Enterobacter cloacae

A case is presented of a 6-year-old boy from Nigeria who developed chronic multifocal osteomyelitis with bilateral and symmetrical proximal humeral involvement. Superimposed changes of acute osteomyelitis were noted on the histopathological evaluation of the lesions. Cultures from both sides were positive for Enterobacter cloacae, which was presumably contracted during intravenous therapy for typhoid fever in Nigeria 1 month prior to his presentation.     

Successful treatment of chronic recurrent multifocal osteomyelitis with indomethacin: a preliminary report of five cases

Chronic recurrent multifocal osteomyelitis (CRMO) is a disease of children and young adults. Clinically, the disease is characterized by the insidious onset of local pain and swelling in affected bones. Its course is one of intermittent periods of exacerbation and remission with successive bones affected. The pathogenesis of CRMO remains unknown, although an autoinflammatory disorder may be the cause, with inflammation of bone. This lesion is radiologically characterized as multiple lucencies surrounded by defined zones of patchy but dense sclerosis, cortical thickening from periosteal new bone formation, and increased bone size with different bones involved. Multiple therapeutic regimens had shown only a partial or temporary response. Because indomethacin has been successfully applied in inhibition of ossification and inflammatory processes, we initiated therapy with indomethacin in patients with CRMO. We report on the cases of 5 patients who responded dramatically to treatment with indomethacin. All underwent progressive clinical improvement (mean, 2.8 months). Radiological lesions disappeared after a mean period of 10.5 months. In 1 case where treatment was started late, small osteolytic zones persisted but with no clinical consequences. There were no additional recurrences or new bones affected during follow-up period (mean, 4 years). Our observation indicates that indomethacin may be an effective treatment for CRMO.