eng Science and Education Publishing American Journal of Medical Case Reports 2374-216X 2016-09-14 4 8 293 295 10.12691/ajmcr-4-8-9 AJMCR2016489 article Imene Boukhris imen.boukhris@laposte.net 1 Hana Zoubaidi 1 Ines kechaou 1 Eya Chérif 1 Ines mahmoud 2 Anis Hariz 2 Nédia Znaidi 3 Narjes Khalfallah 3 Internal Medicine Department, University Hospital of Charles Nicolle, Tunis, Tunisia Rheumatology Department, University Hospital of Charles Nicolle, Tunis, Tunisia Department of Diagnostic Pathology, University Hospital of Charles Nicolle, Tunis, Tunisia In Langerhans cell histiocytosis pulmonary involvement is often isolated. It is rarely a part of a multifocal form. Tenosynovitis is exceptional. Hand bones involvement is atypical. We report the case of a 75-year-old, non-smoker man, without medical history, who presented with arthritis and fever. Examination found arthritis affecting large and small joints. In biology, there was an inflammatory syndrome. Radiographs of the hands showed erosions of the carpal bones. Doppler ultrasonography of the hands showed active synovitis. Tc99 bone scintigraphy highlighted joint uptake in the hands, knees, spine and right upper jaw. Blood cultures and infectious investigations were negative. Anti-nuclear antibodies and rheumatoid factor were negatives. CT scan showed cystic lung lesions and nodular aspect. Bronchoalveolar lavage fluid revealed positivity of anti-CD1a at 6%. A surgical biopsy showed an infiltrate of eosinophilic cells, positive for CD1a in immunohistochemistry studies. The diagnosis of Langerhans cell histiocytosis was retained. http://pubs.sciepub.com/ajmcr/4/8/9/ajmcr-4-8-9.pdf langerhans cell histiocytosis lung arthritis synovitis bronchoalveolar lavage