Misdiagnosed childhood sarcoidosis as non-Langerhans' cell histiocytosis treated with tumor necrosis factors-α antagonists

• Published in: Anales de pediatría (Barcelona, Spain : 2003) Spain : 2003), 2012-10, Vol.77 (4), p.267-271
• Main Authors: Petiti Martin, G, Castellanos González, M, Sanz Bueno, J, Burgués Calderón, M, Villar Buil, M, Vanaclocha, F, Peralto, J L
• Format: Article
• Language: Spanish
• Subjects: Adalimumab; Adrenal Cortex Hormones - therapeutic use; Antibodies, Monoclonal - therapeutic use; Antibodies, Monoclonal, Humanized - therapeutic use; Child; Diagnostic Errors; Female; Histiocytosis, Non-Langerhans-Cell - diagnosis; Humans; Infliximab; Methotrexate - therapeutic use; Sarcoidosis - diagnosis; Sarcoidosis - drug therapy; Sarcoidosis - pathology; Tumor Necrosis Factor-alpha - antagonists & inhibitors; Uveitis - drug therapy
• ISSN: 1695-9531
• DOI: 10.1016/j.anpedi.2012.01.024

Sarcoidosis is a chronic multisystemic granulomatous disease of unknown origin. Generalised eruptive histiocytosis is a rare, benign, self-healing, non-Langerhans' cell histiocytosis (non-LCH). We report the case of an 8-year-old girl with sarcoidosis who was misdiagnosed as non-LCH. She was treated with oral corticosteroids, methotrexate and adalimumab, but there was insufficient control of ocular disease. The introduction of infliximab achieved a control of the uveitis and enabled the corticosteroid dose to be tapered. In some cases of sarcoidosis the lack of well-organised granuloma formation at the beginning of the disease, and the presence of prominent giant cells may suggest alternative diagnoses, such as non-LCH. Although the experience of tumour necrosis factor-α antagonists use in children with sarcoidosis is limited, these drugs may be helpful for those patients experiencing a severe and refractory disease.