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Chronic recurrent multifocal osteomyelitis exhibiting predominance of periosteal reaction
Rev Assoc Med B ras 2017; 63(4):303-306 303
IMAGE IN MEDICINE
Chronic recurrent multifocal osteomyelitis exhibiting
predominance of periosteal reaction
Rodolfo Mendes Queiroz 1*, Pedro Henrique P ereira Rocha1, Lara Z upelli Lauar2, Mauro José Brandão da Costa1,
Claudio Benedini Laguna1, Rafael Gouvêa Gomes de Oliveira1
1Department of Radiology and Imaging Diagnosis, Documenta, Hospital São Francisco, Ribeirão Preto, SP, Brazil
2Division of Radiology and Imaging Diagnosis, Department of Internal Medicine, Hospital das Clínicas da Faculdade de Medicina de Ribeirão Preto da Universidade de São Paulo, Ribeirão Preto, SP, Brazil
Summary
Study conducted at Documenta –
Centro Avançado de Diagnóstico
por Imagem, Hospital São Francisco,
Ribeirão Preto, SP, Brazil
Article received: 9/11/2016
Accepted for publication: 10/4/2016
*Correspondence:
Address: Rua Bernardino de
Campos, 980
Ribeirão Preto, SP – Brazil
Postal code: 14015-130
Chronic recurrent multifocal osteomyelitis is an idiopathic nonpyogenic
autoinflammatory bone disorder involving multiple sites, with clinical progression
persisting for more than 6 months and which may have episodes of remission
and exacerbation in the long term. It represents up to 2-5% of the cases of
osteomyelitis, with an approximate incidence of up to 4/1,000,000 individuals,
and average age of disease onset estimated between 8-11 years, predominantly
in females. The legs are the most affected, with a predilection for metaphyseal
regions along the growth plate. We describe the case of a female patient, aged 2
years and 5 months, with involvement of the left ulna, right jaw and left tibia,
showing a predominance of periosteal reaction as main finding.
Keywords: osteomyelitis, chronic, multifocal, recurrent, periosteal.
Case report
A female patient aged 2 years and 5 months, being inves-
tigated due to pain and swelling in the left forearm for
one month. Her parents denied episodes of fever, trauma,
and other comorbidities. On physical examination, the
child presented normal weight, normal skin color, she
was well-hydrated, acyanotic, breathing normally and in
good general conditions. Blood counts analyzed in the
previous month revealed mild leukocytosis and normo-
cytic normochromic anemia. Levels of C-reactive protein
(CRP) and erythrocyte sedimentation rate (ESR) were
slightly elevated.
Computed tomography (CT) of the left forearm with-
out intravenous contrast revealed a periosteal reaction
and a small area of loss of cortical compaction, espe-
cially in the ulna (Figure 1A).
A biopsy of the left ulna was performed with results
describing signs suggestive of chronic osteomyelitis.
Culture of the biopsy material did not show growth of
microorganisms.
Empirical antibiotic therapy was used starting one
month after diagnosis, but there was no change in clinical
status. A new biopsy was performed in the third month,
showing the same findings associated with negative blood
culture. Radiographs of the left forearm from the second
to the fifth month showed progression of the multilamel-
lar periosteal reaction to a solid type (Figure 1B).
Chest radiography, echocardiography, levels of TSH,
T4, IgG, IgA, IgM and complement components were all
normal. A hypothesis of chronic recurrent multifocal
osteomyelitis (CRMO) was suggested and thus treated
with ibuprofen combined with methotrexate, later replaced
with sulfasalazine.
Between the eighth and ninth month, pain and swell-
ing appeared in the right mandibular region and in the
left leg. A facial CT scan showed mainly periosteal reac-
tion at the right mandibular angle with mild bone scle-
rosis (Figure 2). CT scan of the left leg also characterized
a periosteal reaction in the tibial diaphysis with a small
area of cortical alteration (Figure 3), similar to that found
in the ulna.
After 6 months of therapy, there was clinical and gen-
eral laboratory improvement, with left forearm radiogra-
phy in the 11th month showing a regression of the peri-
osteal reaction, despite persistence of cortical thickening.
Discussion
CRMO is an idiopathic nonpyogenic autoinflammatory
bone disorder involving multiple sites, with clinical pro-
gression persisting for more than six months and which