Exacerbation of recalcitrant cutaneous sarcoidosis with adalimumab - a\nparadoxical effect? A case report
A 50-year-old female was observed due to erythematous, infiltrated, sometimes ulcerated\nplaques, on the frontal and left preauricular areas lasting for 2 years. (). The patient had high blood pressure and\nwas treated with bisoprolol 5mg/day.\nThe plaque on the left preauricular area was biopsied. Biopsy was performed showing\ndermal granuloma, without central caseous necrosis and numerous Langhans multinucleated\ngiant cells. () Acid-fast bacteria stains\nwere negative as well as tissue cultures for mycobacteria, bacteria and fungi.\nInfectious etiology was excluded and the diagnosis of cutaneous sarcoidosis was made.\nExcept for cutaneous involvement, the patient was in good health with no systemic\nsymptoms. Further evaluations included a complete blood cell count and complete\nmetabolic panel, both of which revealed no significant abnormal findings.\nC-reactive-protein, angiotensin-converting-enzyme (ACE) and calcium were in the normal\nrange. Further investigation for systemic involvement was negative. Local therapy with\ntopical and intralesional corticosteroids (momethasone furoate and clobetasol propionate\ncreams; betamethasone dipropionate and betamethasone phosphate sodium aqueous\nsuspension, successively) and topical tacrolimus failed. Hydroxychloroquine sulfate (400\nmg daily), pentoxifylline (400 mg daily), methylprednisolone (up to 25 mg daily),\nazathioprine (100 mg daily) and methotrexate (up to 27,5 mg /week until cumulative dosis\nof 733 mg) produced no significant effect.\nLong-term treatment with systemic corticosteroids, azathioprine or methotrexate was felt\nto be unwarranted because of the risk of serious long-term sequelae. Treatment with\nadalimumab was proposed as an alternative to treatment with methotrexate.\nTuberculin skin test was performed before initiating the biological agent and revealed a\n1.1 millimeter papule. Therefore isoniazid (300mg/day) was started.\nMethotrexate was gradually tapered to 7.5 mg/week and methylprednisolone (4 mg/day) and\nthen stopped. Adalimumab (40 mg subcutaneously at week 1, 3 and 5) was started two\nmonths after the start of isoniazid. Adalimumab was then suspended (after the third\ninjection) because the lesions became more erythematous, infiltrated, ulcerated and\nassociated to retroauricular adenopathies (). Shortly after suspension, the lesions improved.