RESUMEN
El síndrome SAPHO es una enfermedad autoinflamatoria crónica que engloba una serie de manifestaciones cutáneas y osteoarticulares que incluyen sinovitis, acné, pustulosis, hiperostosis y osteítis. Se presenta el caso de un paciente masculino de 18 años de edad, con antecedente de esclerodermia localizada diagnosticada por biopsia hace 3 años, quien debutó con lesiones cutáneas y osteoarticulares 6 meses después de suspender tratamiento con metotrexate y colchicina, para dicho antecedente. Se reportó una resonancia magnética sugestiva de una sacroileítis con presencia de edema óseo; y la gammagrafía ósea reveló zonas de captación con imagen en cabeza de toro en articulación esternoclavicular, sugiriendo síndrome de SAPHO. Se decidió reincorporar el metotrexate y colchicina, evolucionando satisfactoriamente. No se encontraron reportes similares concomitando estas dos entidades que tienen como punto en común el tratamiento con los mismos fármacos.
SAPHO syndrome is a chronic autoinflammatory disease that encompasses a series of cutaneous and osteoarticular manifestations that include synovitis, acne, pustulosis, hyperostosis, and osteitis. We present the case of an 18-year-old male patient, with a history of localized scleroderma diagnosed by biopsy 3 years ago, who debuted with skin and osteoarticular lesions 6 months after discontinuing treatment with methotrexate and colchicine, for said history. An MRI suggestive of sacroiliitis with the presence of bone edema was reported, and bone scintigraphy revealed uptake areas with a bull's head image in the sternoclavicular joint, suggesting SAPHO syndrome. It was decided to reincorporate methotrexate and colchicine, evolving satisfactorily. No similar reports were found coexisting with these two entities that have treatment with the same drugs as a point in common.
RESUMEN
El síndrome SAPHO es una enfermedad inflamatoria de origen multifactorial que incluye una asociación entre manifestaciones dermatológicas y osteoarticulares. La presencia de osteítis e hiperostosis definen la enfermedad. No existe un tratamiento estándar, pero se usan AINES como primera línea. Se presenta el caso de un varón de 18 años con acné severo en dorso, pecho, barbilla y zona retroauricular, asociado a fiebre alta y placas eritematosas en ambas piernas. La gammagrafía presenta hipercaptación en tibia derecha, con relación a osteítis no infecciosa. Tuvo evolución favorable con antibióticos y corticoide oral.
SAPHO syndrome (synovitis, acne, pustulosis, hyperostosis and osteitis) is a multifactorial inflammatory condition that includes an association between dermatological and bone and joint manifestations. The presence of osteitis and hyperostosis define this disease. There is no standard therapy for SAPHO syndrome, but NSAIDs are used as first line. We present the case of an 18-year-old male subject with severe acne on his back, chest, chin, and the retroauricular area, being this associated to high fever and erythematous plaques in both legs. The bone scan showed right leg hyper-uptake, related to non-infectious osteitis. The patient did well with antibiotics and oral steroids.
RESUMEN
INTRODUCTION: SAPHO syndrome is defined as the association of a group of rare sterile osteoarticular disorders and inflammatory skin diseases whose etiology, although not yet determined, probably involves genetic, immunological and infectious mechanisms. The recurrent multifocal osteomyelitis, an inflammatory disease, can be associated with this syndrome even as a single event. CASE REPORT: A case of a young female patient, with a definite diagnosis of SAPHO and an inflammatory mandibular atypical disease for which therapeutic options with immunosuppressants were being used, is reported. The adverse evolution of the clinical conditions led to the hypothesis that the patient suffered from associated mandibular odontogenic bacterial osteomyelitis. The extraction of all teeth was recommended. After our evaluation, we recommended a conservative treatment, and after 2 months of treatment with an endovenous antibiotic, the patient showed improvement of clinical and laboratory results. CONCLUSION: Early diagnosis is necessary to avoid successive and unnecessary tooth loss in the treatment of chronic osteomyelitis mandibular.