Chronic Recurrent Multifocal Osteomyelitis: Two Cases of Sacral Disease Responsive to Corticosteroids

Holden, Wendy; David, Joel

doi:10.1086/427217

Cited by 35 publications

(23 citation statements)

References 11 publications

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“…Other possible pharmacologic treatments include bisphosphonates, sulfasalazine, methotrexate, colchicine, interferons, and gamma globulins [23]. Oral glucocorticoids are likely to be effective as well, but should be used sparingly given the adverse long-term side effects, especially in children and adolescents [24].…”

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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Section: Discussionmentioning

confidence: 99%

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

et al. 2015

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“…En el contexto de artritis o compromiso vertebral, los Aines han demostrado no ser tan efectivos y se debería considerar otra estrategia. Los esteroides también son comúnmente citados como beneficiosos en el manejo de pacientes con CRMO, pero hay pocos reportes en la literatura que respalden su uso y no se tiene un consenso respecto a la dosis apropiada y duración del tratamiento en una población particularmente susceptible a los efectos secundarios de estos fármacos por su uso continuo, tal como el cierre temprano de las placas de crecimiento 42 .…”

Section: Tratamientounclassified

Osteomielitis crónica multifocal recurrente: estado del arte

Eusse

Velásquez-Franco

Navas

2016

MedUPB

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RESUMENLa osteomielitis crónica multifocal recurrente es una enfermedad autoinflamatoria que se caracteriza por focos estériles de inflamación ósea que producen dolor recurrente. Es una entidad poco frecuente que tiene como manifestaciones cardinales la presencia de lesiones óseas inflamatorias en las regiones metaepifisiarias de los huesos largos que predomina en miembros inferiores. Esta enfermedad, con frecuencia asociada con sínto-mas sistémicos y lesiones en piel, hace parte del grupo de enfermedades que conforma el síndrome: artritis-dermatitis. Los hallazgos radiológicos más comunes son las áreas de osteolisis rodeadas de un halo esclerótico delgado, sin reacción perióstica asociada o la formación de secuestros óseos. La resonancia nuclear magnética permite hacer una detección precoz de la entidad y facilita su diagnóstico diferencial, principalmente, con infecciones y tumores óseos. El tratamiento está basado en la información obtenida mediante reportes de caso y series de pacientes y consiste en el uso de antiinflamatorios no esteroideos y en casos refractarios, bisfosfonatos y antagonistas del factor de necrosis tumoral alfa. El pronóstico de esta enfermedad, en general, es bueno, con un curso clínico benigno y autolimitado, en la mayoría de los casos. Palabras clave: osteomielitis; inflamación; hiperostosis. ABSTRACTChronic recurrent multifocal osteomyelitis is a sterile autoinflammatory disease characterized by foci of sterile inflammation in the bone that generate recurrent pain. It is a rare entity in which the cardinal clinical manifestation is the presence of inflammatory bone lesions in metaepiphyseal regions of long bones, predominating in lower limbs. The disease is often associated with systemic symptoms and skin involvement making it part of the arthritis-dermatitis syndrome. The most common radiographic finding is osteolysis surrounded by a thin sclerotic halo, without periosteal reaction associated with bone formation or bone sequestrum. Magnetic resonance imaging allows early detection and facilitates the differential diagnosis between osteomyelitis and infections or bone tumors. Treatment is supported mainly by case reports and series of patients and consists of the use of nonsteroidal anti-inflammatory drugs and the use of bisphosphonates or tumor necrosis factor alpha antagonists in refractory cases. The prognosis of this disease is generally good, with a benign and self-limited clinical course in most cases.

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“…However, the resolution of pain may be transient with symptoms recurring once steroids are stopped [9,29,44,45]. …”

Section: Steroidsmentioning

confidence: 99%

Chronic Recurrent Multifocal Osteomyelitis

Roderick

Ramanan

2012

Advances in Experimental Medicine and Biology

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Chronic recurrent multifocal osteomyelitis (CRMO) is an autoinflammatory bone disease occurring primarily in children and adolescents. Episodes of systemic inflammation occur due to immune dysregulation without autoantibodies, pathogens or antigen-specific T cells. CRMO is characterised by the insidious onset of pain with swelling and tenderness over the affected bones. Clavicular involvement was the classical description; however, the metaphyses and epiphyses of long bones are frequently affected. Lesions may occur in any bone, including vertebrae. Characteristic imaging includes bone oedema, lytic areas, periosteal reaction and soft tissue reaction. Biopsies from affected areas display polymorphonuclear leucocytes with osteoclasts and necrosis in the early stages. Subsequently, lymphocytes and plasma cells predominate followed by fibrosis and signs of reactive new bone forming around the inflammation. Diagnosis is facilitated by the use of STIR MRI scanning, potentially obviating the need for biopsy and unnecessary long-term antibiotics due to incorrect diagnosis. Treatment options include non-steroidal anti-inflammatory drugs and bisphosphonates. Biologics have been tried in resistant cases with promising initial results. Gene identification has not proved easy although research in this area continues. Early descriptions of the disease suggested a benign course; however, longer-term follow up shows that it can cause significant morbidity and longer-term disability. Although it has always been thought of as very rare, the prevalence is likely to be vastly underestimated due to poor recognition of the disease.

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Chronic Recurrent Multifocal Osteomyelitis: Two Cases of Sacral Disease Responsive to Corticosteroids

Cited by 35 publications

References 11 publications

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

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

Osteomielitis crónica multifocal recurrente: estado del arte

Chronic Recurrent Multifocal Osteomyelitis

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