1- InfectiousDiseasesUnit, Hospital de Dona Estefânia, CHULC - EPE, Lisbon, Portugal
2- OphthalmologyUnit, Hospital São José, CHULC - EPE, Lisbon, Portugal
3- PediatricOtolaryngologyUnit, Hospital de Dona Estefânia, CHULC - EPE, Lisbon, Portugal
4- RheumatogyUnit, Hospital de Dona Estefânia, CHULC - EPE, Lisbon, Portugal
- 36th Annual Meeting of the European Society for Paediatric Infectious Diseases, Malmo - Sweden, May 28 – June 2 2018
- International meeting
- E-poster
Resumo:
Introduction: Adalimumab is a tumor necrosis factor-alpha (TNF-α) used for refractory non-infectious uveitis in children. Anti-TNF therapy has been be associated with opportunistic infections, such as atypical mycobacteriosis.
Case Report: A 12-year-old girl with idiopathic anterior and intermediate uveitis, under treatment with adalimumab and cyclosporine, presented with a 4-months nodular, erythematous vegetative lesions of the nasal septum, palate, tonsil and right foot. She denied fever, weight loss or cough and had no response to antibiotics. She had a domestic aquarium and dealt with horses, but did not recall preceding trauma.Blood tests showed leukocytosis, an erythrocyte sedimentation rate of 53mm/h and a C-reactive protein level of 0,9mg/L. The interferon-gamma release assay test and the thorax x-ray was negative.The nasal septum, palate and tonsil biopsy revealed giant cellsnon-necrotic granuloma, withDNA amplification for HSV-1 and the foot skin biopsy shown wart-like epithelium.All lesions amplified for atypical mycobacteria DNA. Anti-TNF-α was discontinued and intravenous acyclovir together withclarithromycin, rifampicin and levofloxacin. The culture turned positive for Mycobacterium marinum and she is currently on clarithromycin and rifampicin for six months, with slow but complete regression ofmuco-cutaneous lesions.
Discussion: Although rare, atypical mycobacteria infections in patients taking TNF-α inhibitors have been reported in the literature. The diagnosis of Mycobacterium marinum infection is difficult and requires a high degree of suspicion. Treatment is challenging and usually includesclarithromycin combined with rifampicin or ethambutolfor at least 2 months after disease regression. The multifocal mucousinvolvement is to our knowledge not previously reported and the authors wonder whether treatmentor prophylaxis should be maintained longer to prevent relapses.
Palavras Chave: anti-TNF-α, atypical mycobacteriosis, idiopathic uveitis, Mycobacterium marinum