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CHRONIC RECURRENT MULTIFOCAL OSTEOMYELITIS STAM et al. 213

Work-up

Radiography of the pelvis and hips (Fig. 1)shows multiple mixed lesions at the apophysis ofboth trochanter major.

Tc-99m bone scintigraphy (Fig. 2) demonstratesan increased uptake in the left wrist, in the righttibia and in both trochanter major.

Radiography of the right knee (Fig. 3), AP (A) andlateral view (B) shows mixed lesions, merely oste-olytic at the center and osteoblastic at the periph-ery of the metaphysis of the right femur and tibia.

Radiography of the hands (Fig. 4) reveals anarea of osteolysis with surrounding osteosclerosisis seen at the metaphysis of the left distal radius.

Radiological diagnosis

Histology was obtained with Yamshidi biopsiesof the right hip and cultures were taken.Histopathological examination showed chronicinflammation, the cultures were negative.

Based on the clinical findings, plain radiography,

additional bone scintigraphy, histopathologicalexamination and cultures, the diagnosis of chronicrecurrent multifocal osteomyelitis (CRMO) wasmade.

Discussion

Chronic recurrent multifocal osteomyelitis(CRMO) is a rare inflammatory disease distinctfrom bacterial osteomyelitis. CRMO primarilyaffects young girls (peak age of onset, 10 years)with a female:male ratio of 5:1. CRMO is often adiagnosis of exclusion, based on the following cri-teria: (1) bone lesions with radiographic findingssuggestive for subacute or chronic osteomyelitis;(2) an atypical location of lesions compared withinfectious osteomyelitis, often multifocal and a fre-quent involvement of the clavicle; (3) no abscess,fistula or sequestra formation; (4) lack of causativeorganism; (5) nonspecific histopathological andlaboratory findings compatible with subacute orchronic osteomyelitis; (6) a characteristic pro-

longed, fluctuating course with recurrent episodesof pain occurring over several years (7) occasionalaccompanying skin disease, most frequent pustu-losis palmoplantaris, less commonly acne, psoria-sis vulgaris and pyoderma gangrenosum.

By some authors, CRMO is considered to be apediatric form of the SAPHO (synovitis, acne, pus-tulosis, hyperostosis, osteitis) syndrome, an entityof diseases whereby sterile inflammatory bonelesions and skin eruptions are associated.

The distribution of the lesions seen in patientswith CRMO is often multifocal and sometimessymme-trical, but unifocal distribution can occur.There is a predilection for the metaphysis of thelong bones usually in the lower extremity (tibia,femur, fibula) followed by the clavicula and spine.In tubular bones osteolytic and osteoproliferativechanges are seen, sometimes with periostal reac-tion. Plain radiography and additional bonescintigraphy contribute to the diagnosis CRMO.MRI is valuable in the assessment of the localextent and activity of symptomatic lesions.

When (multifocal) lytic or sclerotic bone findings

are seen in a patient with recurring episodes ofpain, CRMO should be included in the differentialdiagnosis, when bacterial infections and malignan-cy are excluded.

Bibliography

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3. Huber A.M., Lam P.Y., Duffy C.M., et al.: Chronicrecurrent multifocal osteomyelitis: clinical out-comes after more than five years of follow-up.J Pediatr, 2002, 141: 198-203.

4. Jurik A.G.: Chronic Recurrent MultifocalOsteomyelitis. Semin Musculoskelet Radiol,2004, 8: 243-253.