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Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome in Childhood; A Rare Clinical Entity.

Sayılı A, Tosun O, Cobanoglu N, Bahceciler Onder N, Baba F, Kavukcu S - Iran J Pediatr (2014)

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics.

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Dear Editor, Referring to synovitis, acne, pustulosis, hyperostosis and osteitis; SAPHO syndrome is defined as a chronic, relapsing rheumatologic disease of uncertain etiology characterized by distinct osteoarticular and cutaneous manifestations... Serological tests for Salmonella and Brucella were negative... Having a BCG vaccination scar, PPD was negative and no abnormalities were found in chest radiography of the patient... Bone marrow aspiration and bone scintigraphy revealed no signs of malignancy... She stopped having this treatment on her own accord, and then applied to our hospital with vesiculopustular skin lesions on the palmar, plantar and retroauricular surfaces... Histopathologic findings revealed subcorneal intraepidermal vesicles as well as intracorneal plasma insudation... Inflammation manifests in the form of sterile osteomyelitis and hyperostosis in the bone, acne or pustulosis in the skin and synovitis in the joints... In cases with involvement of atypical sites or single skeletal lesions that lack signs of hyperostosis corresponding with radiologic findings and skin manifestations, the diagnosis becomes much more challenging for SAPHO... In CRMO, like SAPHO syndrome, skin manifestations just as described in our patient, are rare findings... Since the skin findings defined pathologically in our patient are characteristic for SAPHO syndrome together with the absence of neutrophil infiltration, we strongly believe that our case has the diagnosis of SAPHO syndrome rather than CRMO... The disease course in children just like in adults, is characterized by periods of exacerbations and remissions with an increasing number of lesions over time and considered to be a relatively benign and self-limiting disease without major sequelae... Recognition of SAPHO syndrome in our case despite the atypical presentation both in terms of age and skeletal involvement seems notable, since early diagnosis is important to avoid unnecessary invasive procedures and prolonged antibiotic treatment of osteoarticular lesions... It should be considered in the differential diagnosis of hip and lower back pain.

No MeSH data available.


Related in: MedlinePlus

Coronal fat suppressed T2 weighted image shows edema in right acetabulum and surrounding soft tissues
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Figure 1: Coronal fat suppressed T2 weighted image shows edema in right acetabulum and surrounding soft tissues

Mentions: A 12-year-old female patient was admitted to our hospital with the complaint of right hip and low back pain. She had tenderness on the right iliac crest. She had normal hemogram with an erythrocyte sedimentation rate (ESR) of 51 mm/hour. Serological tests for Salmonella and Brucella were negative. Having a BCG vaccination scar, PPD was negative and no abnormalities were found in chest radiography of the patient. Bone marrow aspiration and bone scintigraphy revealed no signs of malignancy. Anti-streptolysin O, C-reactive protein, anti-nuclear anitobody, anti double stranded DNA and HLA B27 were negative. Serum immunoglobulin levels were within the normal range. Magnetic resonance imaging (MRI) (Fig. 1), showed increased density of right acetabular area and surrounding soft tissue besides bone marrow edema. She received 15 mg/kg/day naproxen sodium administered.


Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome in Childhood; A Rare Clinical Entity.

Sayılı A, Tosun O, Cobanoglu N, Bahceciler Onder N, Baba F, Kavukcu S - Iran J Pediatr (2014)

Coronal fat suppressed T2 weighted image shows edema in right acetabulum and surrounding soft tissues
© Copyright Policy
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4359425&req=5

Figure 1: Coronal fat suppressed T2 weighted image shows edema in right acetabulum and surrounding soft tissues
Mentions: A 12-year-old female patient was admitted to our hospital with the complaint of right hip and low back pain. She had tenderness on the right iliac crest. She had normal hemogram with an erythrocyte sedimentation rate (ESR) of 51 mm/hour. Serological tests for Salmonella and Brucella were negative. Having a BCG vaccination scar, PPD was negative and no abnormalities were found in chest radiography of the patient. Bone marrow aspiration and bone scintigraphy revealed no signs of malignancy. Anti-streptolysin O, C-reactive protein, anti-nuclear anitobody, anti double stranded DNA and HLA B27 were negative. Serum immunoglobulin levels were within the normal range. Magnetic resonance imaging (MRI) (Fig. 1), showed increased density of right acetabular area and surrounding soft tissue besides bone marrow edema. She received 15 mg/kg/day naproxen sodium administered.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics.

AUTOMATICALLY GENERATED EXCERPT
Please rate it.

Dear Editor, Referring to synovitis, acne, pustulosis, hyperostosis and osteitis; SAPHO syndrome is defined as a chronic, relapsing rheumatologic disease of uncertain etiology characterized by distinct osteoarticular and cutaneous manifestations... Serological tests for Salmonella and Brucella were negative... Having a BCG vaccination scar, PPD was negative and no abnormalities were found in chest radiography of the patient... Bone marrow aspiration and bone scintigraphy revealed no signs of malignancy... She stopped having this treatment on her own accord, and then applied to our hospital with vesiculopustular skin lesions on the palmar, plantar and retroauricular surfaces... Histopathologic findings revealed subcorneal intraepidermal vesicles as well as intracorneal plasma insudation... Inflammation manifests in the form of sterile osteomyelitis and hyperostosis in the bone, acne or pustulosis in the skin and synovitis in the joints... In cases with involvement of atypical sites or single skeletal lesions that lack signs of hyperostosis corresponding with radiologic findings and skin manifestations, the diagnosis becomes much more challenging for SAPHO... In CRMO, like SAPHO syndrome, skin manifestations just as described in our patient, are rare findings... Since the skin findings defined pathologically in our patient are characteristic for SAPHO syndrome together with the absence of neutrophil infiltration, we strongly believe that our case has the diagnosis of SAPHO syndrome rather than CRMO... The disease course in children just like in adults, is characterized by periods of exacerbations and remissions with an increasing number of lesions over time and considered to be a relatively benign and self-limiting disease without major sequelae... Recognition of SAPHO syndrome in our case despite the atypical presentation both in terms of age and skeletal involvement seems notable, since early diagnosis is important to avoid unnecessary invasive procedures and prolonged antibiotic treatment of osteoarticular lesions... It should be considered in the differential diagnosis of hip and lower back pain.

No MeSH data available.


Related in: MedlinePlus