Isotretinoin
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SAPHO syndrome: case report A 15-year-old boy developed SAPHO syndrome during treatment with isotretinoin for acne fulminans. The boy complained of severe acne and low back pain with inability to walk. Assuming acne fulminans, a course of systemic isotretinoin was administered at the dose of 0.5 mg/kg/day without any benefit [route not stated]. Concomitantly, sertraline was also started due to depressive symptoms associated with insomnia as well as irritability. Ten weeks following initiation of isotretinoin, he experienced increasing low-back pain associated with progressive inability to walk and restriction of daily activities. On admission to the emergency department, he was suffering and unable to walk. Physical examination showed sacroiliac pain, a decreased axial range of movement and severe nodulocystic acne with abscesses on face, neck and thorax. Laboratory investigations was as follows: systemic inflammation, CPR 7.15 mg/dL, ESR 84 mm/h, WBC 14,950 /mmc and 70% neutrophils. Pustular lesions culture was positive for Staphylococcus aureus. Treatment with isotretinoin was interrupted and antibiotic therapy with clindamycin was initiated. MRI demonstrated symmetrical sacroiliitis, moderate bone marrow oedema and osteitis of transverse process of fifth lumbar vertebra. The association of acne fulminans and osteitis was consistent with the diagnosis of SAPHO syndrome. Whole body MRI confirmed this hypothesis. In particular coronal views showed sternoclavicular and costoclavicular osteitis, and axial involvement, including sacroiliac joint and spine. Subsequently, IV antibiotics were held. NSAIDs were started in order to control the inflammatory status and oral rifampicin for the cutaneous lesions. Despite treatment, the depressive disorder was persisting along with the other complaints. In the following weeks, osteoarticular involvement and cutaneous manifestations worsened and he developed gluteal bilateral hidradenitis suppurativa (HS). Therefore, adalimumab was administered. Following 4 weeks of treatment, there was a progressive improvement of cutaneous lesions and osteoarticular symptoms. Adalimumab administration was extended to 4 weeks. After 6 months of favourable adalimumab treatment, there was a relapse of osteoarticular symptoms. Subsequently, adalimumab was administered again every 2 weeks resulting in a long-lasting remission. Twelve months later, an MRI showed no evidence of abnormal vertebral bone marrow signal, sterno-clavear or sacroiliac effusions. After 24 months of treatment with adalimumab, the disease maintained a complete remission. Luzzati M, et al. SAPHO syndrome: the supposed trigger by isotretinoin, the efficacy of adalimumab and the specter of depressive disorder: a case report. Italian Journal of 803519500 Pediatrics 46: 169, No. 1, Dec 2020. Available from: URL: http://doi.org/10.1186/s13052-020-00933-1
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Reactions 5 Dec 2020 No. 1833
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