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Chronic recurrent multifocal osteomyelitis (CRMO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome with associated neutrophilic dermatoses: a report of seven cases and review of the literature.
Pediatr Dermatol 2009 Sep-Oct; 26(5):497-505PD

Abstract

A growing body of literature has identified the association between neutrophilic dermatoses and multifocal, aseptic bone lesions in children, termed chronic recurrent multifocal osteomyelitis (CRMO). Classically, patients present with swelling, pain, and impaired mobility of the affected area, with skin lesions developing concurrently or in the future. Bone biopsy reveals inflammatory changes consistent with infectious osteomyelitis, but cultures and histologic staining invariably fail to identify an infectious source. Patients are refractory to antibiotic therapy, but dramatically respond to systemic steroids and may need to be maintained on low-dose steroids to prevent relapse. Numerous authors have suggested that CRMO and synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome lie along the same clinical spectrum. In fact some believe that CRMO is the pediatric presentation of SAPHO. The two syndromes share numerous characteristics, including osteitis, a unifocal or multifocal presentation, hyperostosis, and pustulosis, which all occur in a generally healthy individual. Our seven patients, five of whom were diagnosed with CRMO, and two of whom were diagnosed with SAPHO syndrome further strengthen the idea that CRMO and SAPHO syndrome do indeed lie along the same clinical spectrum. In addition, we include two rare cases of pediatric Sweet's syndrome with evidence of pathergy.

Authors+Show Affiliations

Department of Dermatology, New York University Langone Medical Center, New York, New York 10016, USA. brook.tlougan@gmail.comNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info availableNo affiliation info available

Pub Type(s)

Case Reports
Journal Article
Review

Language

eng

PubMed ID

19840301

Citation

Tlougan, Brook E., et al. "Chronic Recurrent Multifocal Osteomyelitis (CRMO) and Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome With Associated Neutrophilic Dermatoses: a Report of Seven Cases and Review of the Literature." Pediatric Dermatology, vol. 26, no. 5, 2009, pp. 497-505.
Tlougan BE, Podjasek JO, O'Haver J, et al. Chronic recurrent multifocal osteomyelitis (CRMO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome with associated neutrophilic dermatoses: a report of seven cases and review of the literature. Pediatr Dermatol. 2009;26(5):497-505.
Tlougan, B. E., Podjasek, J. O., O'Haver, J., Cordova, K. B., Nguyen, X. H., Tee, R., ... Hansen, R. C. (2009). Chronic recurrent multifocal osteomyelitis (CRMO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome with associated neutrophilic dermatoses: a report of seven cases and review of the literature. Pediatric Dermatology, 26(5), pp. 497-505. doi:10.1111/j.1525-1470.2009.00984.x.
Tlougan BE, et al. Chronic Recurrent Multifocal Osteomyelitis (CRMO) and Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) Syndrome With Associated Neutrophilic Dermatoses: a Report of Seven Cases and Review of the Literature. Pediatr Dermatol. 2009;26(5):497-505. PubMed PMID: 19840301.
* Article titles in AMA citation format should be in sentence-case
TY - JOUR T1 - Chronic recurrent multifocal osteomyelitis (CRMO) and synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome with associated neutrophilic dermatoses: a report of seven cases and review of the literature. AU - Tlougan,Brook E, AU - Podjasek,Joshua O, AU - O'Haver,Judith, AU - Cordova,Katherine B, AU - Nguyen,Xuan H, AU - Tee,Ronald, AU - Pinckard-Hansen,Kay C, AU - Hansen,Ronald C, PY - 2009/10/21/entrez PY - 2009/10/21/pubmed PY - 2010/1/15/medline SP - 497 EP - 505 JF - Pediatric dermatology JO - Pediatr Dermatol VL - 26 IS - 5 N2 - A growing body of literature has identified the association between neutrophilic dermatoses and multifocal, aseptic bone lesions in children, termed chronic recurrent multifocal osteomyelitis (CRMO). Classically, patients present with swelling, pain, and impaired mobility of the affected area, with skin lesions developing concurrently or in the future. Bone biopsy reveals inflammatory changes consistent with infectious osteomyelitis, but cultures and histologic staining invariably fail to identify an infectious source. Patients are refractory to antibiotic therapy, but dramatically respond to systemic steroids and may need to be maintained on low-dose steroids to prevent relapse. Numerous authors have suggested that CRMO and synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome lie along the same clinical spectrum. In fact some believe that CRMO is the pediatric presentation of SAPHO. The two syndromes share numerous characteristics, including osteitis, a unifocal or multifocal presentation, hyperostosis, and pustulosis, which all occur in a generally healthy individual. Our seven patients, five of whom were diagnosed with CRMO, and two of whom were diagnosed with SAPHO syndrome further strengthen the idea that CRMO and SAPHO syndrome do indeed lie along the same clinical spectrum. In addition, we include two rare cases of pediatric Sweet's syndrome with evidence of pathergy. SN - 1525-1470 UR - https://www.unboundmedicine.com/medline/citation/19840301/Chronic_recurrent_multifocal_osteomyelitis__CRMO__and_synovitis_acne_pustulosis_hyperostosis_and_osteitis__SAPHO__syndrome_with_associated_neutrophilic_dermatoses:_a_report_of_seven_cases_and_review_of_the_literature_ L2 - https://doi.org/10.1111/j.1525-1470.2009.00984.x DB - PRIME DP - Unbound Medicine ER -