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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

3 months later: coronal fat suppressed T2 weighted image shows edema signal at bilateral acetabulum, major trochanter and left minor trochanter
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Figure 2: 3 months later: coronal fat suppressed T2 weighted image shows edema signal at bilateral acetabulum, major trochanter and left minor trochanter

Mentions: In a month’s time, regression of complaints occurred while occasional emergence of fever and pain on the left hip was identified. Repeated pelvic MRI (Fig. 2) revealed involvement of head of femur on the left side, L5 and S1 vertebrates characterized with hyper-density and edema.


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)

3 months later: coronal fat suppressed T2 weighted image shows edema signal at bilateral acetabulum, major trochanter and left minor trochanter
© Copyright Policy
Related In: Results  -  Collection

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

Figure 2: 3 months later: coronal fat suppressed T2 weighted image shows edema signal at bilateral acetabulum, major trochanter and left minor trochanter
Mentions: In a month’s time, regression of complaints occurred while occasional emergence of fever and pain on the left hip was identified. Repeated pelvic MRI (Fig. 2) revealed involvement of head of femur on the left side, L5 and S1 vertebrates characterized with hyper-density and edema.

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