Chronic recurrent multifocal osteomyelitis with MR correlation: A case report

Chronic recurrent multifocal osteomyelitis (CRMO) is a rare disorder of unknown etiology. The main characteristics on plain X-rays are a lytic destruction in the metaphyseal region of the long bones followed by progressive sclerosis. The symmetrical pattern and the frequent involvement of the sternoclavicular joints and vertebrae are typical. An association with palmoplantar pustulosis has also been described. Laboratory findings are nonspecific. Although MRI is not necessary to make the diagnosis of chronic osteomyelitis, it is useful in assessing the extent and the evolution of the lesions. CRMO of the tibial and fibular bones is described in a 14-year-old girl, who suffered from pain and soft tissue swelling in both ankles. Initial plain X-rays and laboratory findings were normal. After relapsing clinically, progressive sclerosis of both fibular bones occurred. Lytic lesions in the left tibia and both fibular bones were visible. Scintigraphic examination showed pathologic tracer accumulation in both legs. The diagnosis of CRMO was suggested based on CT and MRI findings. CRMO was confirmed after curettage and bone biopsy. Histopathological findings were consistent with active chronic osteomyelitis.     

Chronic recurrent multifocal osteomyelitis – report of eight patients

The authors undertook a retrospective review of the clinical records and radiologic findings of eight children with chronic, recurrent multifocal osteomyelitis (CRMO). This disease is a recognized clinicopathologic entity with typical radiographic findings, mostly in the metaphyses of the long bones. The diagnosis is one of exclusion without pathognomonic findings. The patients were treated with non-steroidal anti-inflammatory drugs. There was no evidence of altered bone growth or abnormal joint development. It is concluded that CRMO is a distinct clinical entity that is different from acute or subacute bacterial osteomyelitis. Recognition of this condition is important to avoid treatment with antibiotics and repeated operations. Accepted: 8 April 1998     

Rib osteomyelitis in children. Early radiologic and ultrasonic findings

The earliest radiographic changes of osteomyelitis in the long bones is deep-seated edema manifesting as soft tissue swelling and obliteration of the intermuscular planes adjacent to the affected bone. Similarly, the early change of rib osteomyelitis is pericostal edema demonstrated by soft tissue swelling of the thoracic wall accompanied by an adjacent inward pleural displacement. In both osteomyelitis of the rib and the long bones, the bony changes will appear 1–2 weeks later. Pericostal edema can be readily diagnosed by ultrasound scan. Pericostal edema, although non specific and can occur in other conditions, yet it is a strong warning sign, set within the overall clinical picture of osteomyelitis.     

Osteomyelitis: early scintigraphic detection in children.

The value of scintigraphy in the early detection of osteomyelitis is demonstrated in children who had signs and symptoms suggestive of bone infection. Nine patients between 2 and 13 years of age were evaluated with technetium Tc 99m diphosphonate bone scintigrams and roentgenograms. Blood cultures were performed in all patients, and bone biopsy or drainage was obtained in six patients. The diagnosis of osteomyelitis was made in seven patients; one patient had a subperiosteal abscess surrounded by osteomyelitis, and one patient had cellulitis. The seven children with osteomyelitis had focal increase of radiopharmaceutical uptake in the bone. The child with the subperiosteal abscess had an area of decreased uptake in the center of the abscess surrounded by a zone of increased uptake of the radioactive bone-seeker. The patient with cellulitis had soft tissue changes by X-ray and a normal bone scintigram. In the seven patients with osteomyelitis, the bone scintigram was performed during the early phase of the disease and no bony changes were present on the roentgenogram. In one patient with subacute osteomyelitis, soft tissue changes were seen radiologically. Only three of the seven children with osteomyelitis developed radiological bony changes. Since bone scintigraphy can detect early local bone derangement, it is recommended in the initial evaluation of children in whom osteomyelitis is suspected.     

Pyogenic arthritis associated with adjacent osteomyelitis: identification of the sequela-prone child.

We treated 96 cases of pyogenic arthritis from January 1, 1980, to December 31, 1990, 16 of whom had adjacent osteomyelitis. Presenting symptoms in the latter were indistinguishable from those in 80 cases of primary pyogenic arthritis with regard to involved joints, aspirate findings and pathogens; however, adjacent osteomyelitis patients tended to be younger and were more likely to be symptomatic more than 7 days and to have received prior antibiotics. Prompt joint drainage was done in 15 of 16 cases but adjacent osteomyelitis was recognized in only 5. Bone scan was misleading in 3 of the 4 cases where it was performed as part of the initial evaluation. Persistent pain, swelling and/or fever occurred in 9 patients, 6 of whom underwent further joint drainage. Radiographs were diagnostic in 1 patient at admission, 10 during hospitalization and in 5 at follow-up. Sequelae were found in 8 of 13 patients with bone and joint infections vs. 8 of 41 patients with primary joint infection. Patients with a positive culture from the hip or shoulder who had been pretreated with antibiotics had the worst prognosis. Osteomyelitis should be considered in patients who present with symptoms and signs of pyogenic arthritis for longer than 1 week, especially if they have received prior antibiotics. Earlier recognition and bone debridement may improve outcome.     

The syndrome of chronic recurrent multifocal osteomyelitis and congenital dyserythropoietic anaemia. Report of a new family and a review

A new autosomal recessive syndrome of chronic recurrent multifocal osteomyelitis (CRMO) and congenital dyserythropoietic anaemia (CDA) with microcytosis has recently been described in four children (two sibships) of one consangineous Arab family. In this report, we describe the clinical features and course of the syndrome of CRMO and CDA in two additional patients (one sibship) from another consanguineous Arab family and review the literature. The two patients (brother and sister), the products of a consanguineous marriage, developed the syndrome at an early age of 3 weeks and 2 months respectively. The diagnosis of CRMO was confirmed by radiological and technetium isotope bone scans. Bone marrow studies confirmed the diagnosis of CDA. Peripheral blood films showed hypochromia and microcytosis. The sites involved by CRMO were periarticular, mainly around the elbow, knee, wrist and small joints of the hand. The brother is now 21 years old and the sister 3.5 years old and CRMO is still active with frequent relapses. The brother developed flexion deformities at the age of 13 years. Both patients failed to thrive; weight and height were below the 5th percentile. CONCLUSION: This is the second report of the syndrome of chronic recurrent multifocal osteomyelitis and microcytic congenital dyserythropoietic anaemia, confirming it as a clinical entity, inherited as an autosomal recessive trait. The disease is characterised by an early onset, long clinical course of remissions and relapses, and seems to be different from the sporadic form of chronic recurrent multifocal osteomyelitis.     

Chronic recurrent multifocal osteomyelitis of the mandible. A case report]

Chronic recurrent multifocal osteomyelitis (CRMO) is a disorder rarely localized to the lower jaw. CASE REPORT: A fourteen-year-old boy complained of a swollen of his lower jaw. After a CT Scan, a bone biopsy was performed and yielded S. oralis against which an adapted intravenous antibiotherapy was administered without efficacy. The absence of malignant process and the revelation of an other focus of fixation at the Tc bone scan localized on humerus called to mind the diagnosis of CRMO. CONCLUSION: The diagnosis of this disease is difficult and based on a number of concording arguments:clinical and radiological signs of osteomyelitis, multifocal presentation, recurrent relapses and remissions, inaction of antibiotics, elimination of the other differential diagnosis, in particular the infectious osteitis.     

Spinal involvement in chronic recurrent multifocal osteomyelitis (CRMO) in childhood and effect of pamidronate

There are only a few studies that address the frequency and type of spinal involvement in patients with chronic recurrent multifocal osteomyelitis (CRMO) as well as the outcome of these patients treated with pamidronate (PAM). We performed a retrospective study on patients with CRMO and analyzed clinical and pain assessments as well as regional and whole body MRI findings and compared with posttreatment findings. Of 102 children and adolescents with CRMO, 27 (26%) had involvement of the spine. Vertebral deformities were seen in 14 of these 27 patients, scoliosis or kyphosis in 6. After routine whole body MRI, 19 complained of back pain, whereas eight were asymptomatic with spinal lesions detected incidentally. A total of 72 spinal lesions were detected, thoracic vertebrae being the most commonly affected. Seven patients were treated with PAM; all of whom had vertebral deformities and ongoing back pain. Pain resolution was achieved within 3 months of PAM treatment in every case. One patient subsequently developed a pain amplification syndrome. Repeat MRI performed at a mean interval of 13 months revealed partial or complete resolution of vertebral hyperintensities in every patient. Improvement of vertebral height was seen in a total of three vertebrae in two patients. Severe side effects were not observed. In conclusion, we demonstrated that spinal involvement and associated vertebral deformities with or without kyphoscoliosis are not rare in CRMO, and PAM appears to be an effective and safe treatment for this condition. Although controlled studies are urgently needed, the use of PAM for refractory CRMO with extended spinal involvement (vertebral deformities, kyphosis, and scoliosis) should be considered, especially after failing of conventional therapy.     

Dramatic pain relief and resolution of bone inflammation following pamidronate in 9 pediatric patients with persistent chronic recurrent multifocal osteomyelitis (CRMO)

Background  

Chronic recurrent multifocal osteomyelitis (CRMO) is an inflammatory, non-infectious osteopathy that affects predominantly patients ≤ 18 years of age. There is no uniformly effective treatment. Our objective is to describe clinical, magnetic resonance imaging (MRI), and bone resorption response to intravenous pamidronate in pediatric CRMO.     

Ultrasonic signs of pelvic osteomyelitis in children

The ultrasonic findings were reviewed in 13 children in whom pelvic osteomyelitis was diagnosed by a positive^99mtechnetium methylene diphosphonate (MDP) bone scan in conjunction with clinical and laboratory features of osteomyelitis. All patients presented with pain in the region of the hip joint. In six patients the ultrasound study was confined to the hip joint, and all six had normal findings. In seven patients the ultrasound study was extended to include the pelvis. Deep soft tissue swelling was demonstrated in six of these, including a periosseous abscess in one case. Ultrasonography was negative in one patient with a 5-week history, whose pelvic osteomyelitis was resolving at the time of the ultrasound study. Oedema of the obturator internus and externus muscles was observed in osteomyelitis affecting the pubis and ischium, and of the iliacus and/or the gluteus medius muscle in osteomyelitis of the ilium. In children presenting with hip pain who have a normal hip ultrasound study, extension of the ultrasound examination to include these four pelvic muscles may help to identify and document the progression of acute pelvic osteomyelitis.     

Magnetic resonance imaging in children with acute hip pain

45 children presenting with acute hip pain were prospectively evaluated with conventional radiography, radioisotope bone scan, and magnetic resonance imaging (MRI). The final diagnoses were transient synovitis (n = 17), septic arthritis (n = 2), Legg-Calve-Perthes disease (LCPD, n = 13), epiphyseal dysplasia (n = 2), other conditions (n = 4), and normal findings (n = 7). In the work-up MRI provided more morphologic information than other techniques and enlarged the diagnostic possibilities. It was the only imaging technique able to give an early indication of bone marrow involvement in systemic diseases. For the early diagnosis of LCPD, MRI was as sensitive as isotope bone scan and more precise than conventional radiography. In the follow-up of LCPD patients MRI was not able to indicate the start of revascularisation of the necrotic area, which bone scans showed reliably in six patients: but MRI provided excellent evaluation of the position, form and size of the femoral head and the surrounding soft tissues.     

Osteomyelitis in patient with sickle cell disease

From 1977 to 1989, 23 children with sickle cell disease were identified as having 21 episodes of acute and 3 episodes of chronic osteomyelitis, respectively. The responsible organisms were found in 17 cases: Salmonella (12 cases), coagulase-negative Staphylococcus (3 cases). Haemophilus influenzae (1 case), Escherichia coli (1 case). The mean age was 7 7/12 years. In 15 patients, osteomyelitis occurred in 1 bone; osteomyelitis of more than one bone was recorded in 9 cases. The most commonly affected bone was the femur (7 episodes); 5 episodes of hand-foot syndrome with osteomyelitis occurred in children in the first 2 years of life (mean age 16 months). Two patients had a Salmonella vertebral osteomyelitis. Incision and drainage were performed in 5 cases and bone aspiration in 9 cases. Etiologic agents were obtained with these two procedures in respectively 5 and 3 cases. Radionuclide scans were used in 7 episodes: uptake on bone scan was increased in 5 cases and normal in 2. In all cases, the outcome was satisfactory. Differentiation from acute bone infarcts in difficult. An extensive workup is required to confirm the diagnosis of infection: early scintigraphy, bone aspiration or surgical biopsy in patients with negative blood cultures should be performed. Until the results of cultures, the antimicrobial regimen chosen for initial therapy should be broad enough to treat the likely etiologic agents including Salmonella.     

Oral alendronate in pediatric chronic recurrent multifocal osteomyelitis

Chronic recurrent multifocal osteomyelitis (CRMO) primarily affects children and adolescents, and is characterized by episodic sterile osteomyelitis over several years. No definitive treatment is available. Non‐steroidal anti‐inflammatory drugs (NSAID) are common first‐line agents, but provide limited improvement in bone pain and do not affect disease duration. Several agents are utilized in the case of non‐response to NSAID, including corticosteroids, methotrexate, and tumor necrosis factor‐blocking agents. Bisphosphonates are increasingly being used. Most case series involve cyclic i.v. pamidronate, but this restricts the social lives of children and their families. Although oral medication has advantages over cyclic i.v. infusion because it does not require repeated hospital admissions, there have been no reports on treatment with oral bisphosphonates, such as alendronate, in pediatric CRMO patients. This case report describes the use of oral bisphosphonate as an alternative treatment in CRMO patients in whom standard therapy has failed.     

Ostéomyélite chronique multifocale récurrente. À propos d'un cas de la mandibuleChronic recurrent multifocal osteomyelitis of the mandible. A case report

Chronic recurrent multifocal osteomyelitis (CRMO) is a disorder rarely localized to the lower jaw.Case report. – A fourteen-year-old boy complained of a swollen of his lower jaw. After a CT Scan, a bone biopsy was performed and yielded S. oralis against which an adapted intravenous antibiotherapy was administered without efficacy. The absence of malignant process and the revelation of an other focus of fixation at the Tc bone scan localized on humerus called to mind the diagnosis of CRMO.Conclusion. – The diagnosis of this disease is difficult and based on a number of concording arguments:clinical and radiological signs of osteomyelitis, multifocal presentation, recurrent relapses and remissions, inaction of antibiotics, elimination of the other differential diagnosis, in particular the infectious osteitis.     

MR imaging evaluation of subacute and chronic bone abscesses in children

Objective. The aim of the present study was to assess the value of magnetic resonance (MR) imaging in subacute and chronic bone abscesses in children. ¶Materials and methods. Seventy-four patients underwent MR imaging because of suspected musculoskeletal infections between January 1996 and January 1999 in Montreal Children's Hospital. The clinical, radiographic, scintigraphic and MR imaging features of patients with a bone abscess were studied. ¶Results. Eleven patients had osteomyelitis with no bone abscess and six had osteomyelitis with a subacute or chronic bone abscess. Although the lucency was eventually seen on plain radiographs in all cases, MR imaging made a significant contribution, as it helped narrow the differential diagnosis and showed better delineated medullary involvement and extension into the epiphysis. ¶Conclusion. MR imaging is valuable in the diagnostic evaluation of children with bone infection and abscess. It reveals the extent of subperiosteal and epiphyseal involvement not seen on plain radiographs. The extent of the medullary involvement around the abscess is best visualized with MR imaging, which can also distinguish between isolated soft tissue infection adjacent to bone and true bone infection.     

Chronic recurrent multifocal osteomyelitis-- I. Review

Juvenile and adolescent "Chronic Recurrent Multifocal Osteomyelitis" (CRMO) is described on the basis of literature and analysis of 43 own cases (23 cases in children or adolescents).This systemic, non-purulent inflammatory disease occurs mainly metaphyseal in long bones, in pelvic bones or as spondylitis and is not as rare as it seemed. Basis of the disease is a primarily chronic, sterile, in phase of onset often monotopic (e.g. clavicle) and later frequently polytopic osteomyelitis, possibly triggered by an immuno-pathological process (e.g. Proprionibacterium acnes), and showing histologically plasmacellular invasion and a sclerosing process in different stages. Association with pustulous dermatosis (psoriasis, acne, palmo-plantar pustulosis) is found in about 25 % of children and adolescents and in more than 50 % of the adult patients. 5 differents types of distribution of osteomyelitic lesions can be found by using Te99m-bone scan primarily, of which the "pelvic type" is the most common. Because of the close neighbourhood of meta-/epiphyseal osteomyelitic focuses, "sympathetic arthritis" with synovitis is seen frequently. A therapeutic approach with azithromycine and calcitonine is presented.     

Scintigraphic differentiation of bone infarction from osteomyelitis in children with sickle cell disease

Bone scans or bone marrow scans or both were obtained during 42 episodes of bone pain in 40 children with sickle cell disease, and the usefulness of these procedures was compared. On the basis of the subsequent clinical course, a diagnosis of bone infarction was made in 34 episodes, and osteomyelitis in eight. Among 22 patients with bone infarction, uptake on bone scan was increased in 14, decreased in three, and normal in five. Seven of eight patients with osteomyelitis had increased uptake on bone scan; one had normal uptake. In contrast, marrow scan uptake was markedly decreased in 15 of 16 patients with bone infarction, and was normal in five of five patients with osteomyelitis. Thus, decreased uptake on bone marrow scan in a patient with sickle cell disease and bone pain almost invariably indicates infarction, whereas normal uptake strongly suggests the diagnosis of osteomyelitis. We found marrow scans more useful than bone scans for this differential diagnosis.     

Changing pattern of osteomyelitis in infants and children

A retrospective analysis of 332 children with osteomyelitis (OM), managed from 1966 to 1996, was undertaken to evaluate etiology, clinical course and treatment results. In 64% of all patients positive bacterial cultures were obtained, Staphylococcus aureus, streptococci, pneumococci, and Haemophilus influenzae were the most frequently cultured pathogens. In two-thirds of the cases long bones (femur, tibia, humerus) were affected. Osteoarthritis or suppurative arthritis was evident in 27%; 32 of 170 (19%) re-evaluated patients had moderate or severe sequelae. Risk factors for an unfavorable course were the onset of disease in early infancy, suppurative arthritis, and an affected epiphysis. Suppurative arthritis, in particular, needs early evacuation to prevent sequelae. In recent years we observed an increasing number of patients presenting with atypical forms of OM. Since 1989 10 patients were considered to have chronic recurrent multifocal OM (CRMO). In 6 of them the clavicle was involved; their ages ranged from 3 to 14 years. The erythrocyte sedimentation rate was elevated (median 48, range 9–110 mm), while other inflammatory parameters like C-reactive protein (median 9, range <5–85 mg/l) or leucocyte count were slightly elevated or normal. Histopathology was stage-dependent, with a predominance of lymphoplasmacellular infiltration. A nonbacterial origin of CRMO is probable but not proven. Histopathology is not suitable for differentiation between bacterial and nonbacterial forms of bone inflammation. Accepted: 5 January 1999     

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??Objective To discuss the value of combination of high frequency ultrasound and MRI in assessment of changes of joints involvement in children with haemophiliac arthropathy. To compare the consistency of the two methods in detecting the disease. Methods Totally 17 cases of children with hemophilia were included??and 6 knee joints??6 ankle joints and 5 elbow joints were performed by ultrasonic and MRI examination. According to IPSG scores??assesement of effusion/hemothrosis??hemosidaring??cartilage loss and bone cyst was performed to get the comparison of two kinds of examination method in the detection consistency of soft tissue and osteochondral lesions. Results The two kinds of examination methods for detection of joint disease has a high consistency in synovial thickening?? hemosiderin deposition??cartilage loss and bone destruction??Kappa??1.000??0.850??0.761 and 0.876??P??0.05????in joint effusion they showed medium consistency??and kappa value was 0.605??P??0.01??. Detection consistency in joint cyst was poor??and Kappa value was 0.261??P??0.110??. Conclusion??On the detection of soft tissues and osteochondral joint changes of hemophilia in children?? ultrasound and magnetic resonance imaging have poor consistency in the bone cyst??while in the detection of other pathological changes??two kinds of examination have high consistency in detection rate.