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Skull involvement in a pediatric case of chronic recurrent multifocal osteomyelitis.

Watanabe T, Ono H, Morimoto Y, Otsuki Y, Shirai M, Endoh A, Naito M, Inoue Y, Hongo T - Nagoya J Med Sci (2015)

Bottom Line: Initially, a tumor was suspected.The slight elevation in inflammatory markers such as C-reactive protein was of little clinical value.The administration of non-steroidal anti-inflammatory drugs successfully improved his clinical symptoms.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, Iwata City Hospital, Iwata, Japan.

ABSTRACT
An 11-year-old boy was diagnosed with chronic recurrent multifocal osteomyelitis (CRMO) and presented with right sacro-femoral and occipital lesions. Initially, a tumor was suspected. However, the bone biopsy showed osteomyelitis with a negative bacterial culture. Bone scintigraphy revealed inflammatory changes on multiple bone lesions. The slight elevation in inflammatory markers such as C-reactive protein was of little clinical value. He was diagnosed with CRMO by sacral biopsy, and the clinical course progressed, with the presence of a new occipital lesion observed after the 1-year follow-up. The administration of non-steroidal anti-inflammatory drugs successfully improved his clinical symptoms. The presence of a skull lesion in the occipital bone of a pediatric patient with CRMO has not been previously reported.

No MeSH data available.


Related in: MedlinePlus

Fat-suppressed T2-weighted images. Axial (left) and sagittal (right) fat-suppressed T2-weighted images demonstrate a focus of increased signal intensity on the occipital bone. The area of increased intensity extends into the epidural space and scalp.
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fig3b: Fat-suppressed T2-weighted images. Axial (left) and sagittal (right) fat-suppressed T2-weighted images demonstrate a focus of increased signal intensity on the occipital bone. The area of increased intensity extends into the epidural space and scalp.

Mentions: A course of antibiotics was administered for 14 days but did not alter the disease course. The non-steroidal anti-inflammatory drug (NSAID) ibuprofen was started at 200 mg/day and the symptoms improved, but complete relief was not attained. Three months later, a bone biopsy was performed to rule out the possibility of malignant bone tumors, such as an osteosarcoma. Upon pathologic examination, mild, mainly lymphocytic, inflammatory cell infiltration indicated chronic osteomyelitis, without suppurative infection (Fig. 2). There was no yield from the bone tissue culture. One year later, the patient developed occipital headaches, accompanied by a decrease in pelvic pain. The headaches were intermittent and sometimes woke him during the night. There was palpable edema in the occipital lesion, and the size of the swelling was 3 cm. Head CT showed osteolytic lesions in the occipital bone (Fig. 3A). Axial and sagittal fat-suppressed T2WI demonstrated a focus of increased signal intensity in the occipital bone, with the area of increased intensity extending into the epidural space and scalp (Fig. 3B). The previous osteolytic lesion improved, but another lesion appeared as an osteolytic change in the occipital bone (Fig. 4). Technetium-99m-hydroxymethylene diphosphonate (99mTc-HMDP) scintigraphy showed increased uptake in the occipital bone and the right side of the sacrum, leading to a diagnosis of CRMO, which was controlled using ibuprofen (200 mg/day). We ruled out Langerhans cell histiocytosis (LCH) after considering the sacral biopsy and radiological findings, and because the head lesion appeared recently. The bone pain and swelling of the occipital lesion gradually improved with NSAID treatment over one year. One year and 4 months later, diffusion-weighted MRI imaging showed a new, asymptomatic region on the left (opposite) side of the sacrum. The serum cytokine profiles at this time were almost normal; the values are as follows (normal values in parentheses): IL-6 was <3 pg/mL, neopterin was 3.9 nmol/L (<5), soluble tumor necrosis factor receptor (sTNF-R)1 was 940 pg/mL (484–1,407), sTNF-R2 was 2,630 pg/mL (829–2,262), and IL-18 was 300 pg/mL (<500). Over the following two years, there were no cutaneous findings. An LPIN2 mutation seen in Majeed syndrome was not detected in our patient.


Skull involvement in a pediatric case of chronic recurrent multifocal osteomyelitis.

Watanabe T, Ono H, Morimoto Y, Otsuki Y, Shirai M, Endoh A, Naito M, Inoue Y, Hongo T - Nagoya J Med Sci (2015)

Fat-suppressed T2-weighted images. Axial (left) and sagittal (right) fat-suppressed T2-weighted images demonstrate a focus of increased signal intensity on the occipital bone. The area of increased intensity extends into the epidural space and scalp.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4574337&req=5

fig3b: Fat-suppressed T2-weighted images. Axial (left) and sagittal (right) fat-suppressed T2-weighted images demonstrate a focus of increased signal intensity on the occipital bone. The area of increased intensity extends into the epidural space and scalp.
Mentions: A course of antibiotics was administered for 14 days but did not alter the disease course. The non-steroidal anti-inflammatory drug (NSAID) ibuprofen was started at 200 mg/day and the symptoms improved, but complete relief was not attained. Three months later, a bone biopsy was performed to rule out the possibility of malignant bone tumors, such as an osteosarcoma. Upon pathologic examination, mild, mainly lymphocytic, inflammatory cell infiltration indicated chronic osteomyelitis, without suppurative infection (Fig. 2). There was no yield from the bone tissue culture. One year later, the patient developed occipital headaches, accompanied by a decrease in pelvic pain. The headaches were intermittent and sometimes woke him during the night. There was palpable edema in the occipital lesion, and the size of the swelling was 3 cm. Head CT showed osteolytic lesions in the occipital bone (Fig. 3A). Axial and sagittal fat-suppressed T2WI demonstrated a focus of increased signal intensity in the occipital bone, with the area of increased intensity extending into the epidural space and scalp (Fig. 3B). The previous osteolytic lesion improved, but another lesion appeared as an osteolytic change in the occipital bone (Fig. 4). Technetium-99m-hydroxymethylene diphosphonate (99mTc-HMDP) scintigraphy showed increased uptake in the occipital bone and the right side of the sacrum, leading to a diagnosis of CRMO, which was controlled using ibuprofen (200 mg/day). We ruled out Langerhans cell histiocytosis (LCH) after considering the sacral biopsy and radiological findings, and because the head lesion appeared recently. The bone pain and swelling of the occipital lesion gradually improved with NSAID treatment over one year. One year and 4 months later, diffusion-weighted MRI imaging showed a new, asymptomatic region on the left (opposite) side of the sacrum. The serum cytokine profiles at this time were almost normal; the values are as follows (normal values in parentheses): IL-6 was <3 pg/mL, neopterin was 3.9 nmol/L (<5), soluble tumor necrosis factor receptor (sTNF-R)1 was 940 pg/mL (484–1,407), sTNF-R2 was 2,630 pg/mL (829–2,262), and IL-18 was 300 pg/mL (<500). Over the following two years, there were no cutaneous findings. An LPIN2 mutation seen in Majeed syndrome was not detected in our patient.

Bottom Line: Initially, a tumor was suspected.The slight elevation in inflammatory markers such as C-reactive protein was of little clinical value.The administration of non-steroidal anti-inflammatory drugs successfully improved his clinical symptoms.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, Iwata City Hospital, Iwata, Japan.

ABSTRACT
An 11-year-old boy was diagnosed with chronic recurrent multifocal osteomyelitis (CRMO) and presented with right sacro-femoral and occipital lesions. Initially, a tumor was suspected. However, the bone biopsy showed osteomyelitis with a negative bacterial culture. Bone scintigraphy revealed inflammatory changes on multiple bone lesions. The slight elevation in inflammatory markers such as C-reactive protein was of little clinical value. He was diagnosed with CRMO by sacral biopsy, and the clinical course progressed, with the presence of a new occipital lesion observed after the 1-year follow-up. The administration of non-steroidal anti-inflammatory drugs successfully improved his clinical symptoms. The presence of a skull lesion in the occipital bone of a pediatric patient with CRMO has not been previously reported.

No MeSH data available.


Related in: MedlinePlus