title: [Orbital myositis associated with ipilimumab].
abstract: BACKGROUND
  Ipilimumab is a monoclonal antibody targeting cytotoxic T-lymphocyte antigen 4 (CTLA-4) that allows increased survival and, occasionally, complete remission, in the treatment of metastatic melanoma. The most frequent adverse effects are attributed to dysimmunity. We report the case of a female patient who developed orbital myositis during treatment with ipilimumab.
  
  
  METHODS
  A woman on ipilimumab for a heel melanoma with mediastinal metastases was referred for evaluation of painful diplopia and proptosis that began three days after the fourth infusion of ipilimumab. The clinical examination disclosed a left abductiondeficit associated with conjunctival hyperaemia and palpebral oedema. Orbital MRI disclosed enlargement of the left lateral rectus, enhancing after contrast. An extensive work-up did not find any evidence for thyroid-related eye disease, as well as other orbital inflammatory processes, orbital cellulitis or orbital metastases. Treatment with high-dose oral steroids resulted in complete clinical recovery within a few days.
  
  
  CONCLUSIONS
  To our knowledge, this is the first clinical report of orbital myositis as an adverse event related to anti-CTLA-4 antibody treatment. Both timing and usual profile of adverse events support the hypothesis that orbital myositis has to be attributed there to ipilimumab. Several dysimmune toxicities were observed with ipilimumab. Ophtalmic toxicity has unusually been described. Most cases were uveitis. Whether immune-related adverse events correlate with clinical response to ipilimumab treatment remains to be determined.
authors.affiliation: Service de dermatologie, université L'UNAM, CHU d'Angers, 4, rue Larrey, 49933 Angers cedex 9, France. marie.lecouflet@gmail.com
authors.forename: M
authors.initials: M
authors.lastname: Lecouflet
authors.forename: M
authors.initials: M
authors.lastname: Verschoore
authors.forename: C
authors.initials: C
authors.lastname: Giard
authors.forename: P
authors.initials: P
authors.lastname: Gohier
authors.forename: Y
authors.initials: Y
authors.lastname: Le Corre
authors.forename: D
authors.initials: D
authors.lastname: Milea
authors.forename: L
authors.initials: L
authors.lastname: Martin
mesh_terms: D000368:Aged; D000911:Antibodies, Monoclonal / Q000009:adverse effects* / Q000276:immunology / Q000627:therapeutic use; D000970:Antineoplastic Agents / Q000009:adverse effects* / Q000276:immunology / Q000627:therapeutic use; D015496:CD4-Positive T-Lymphocytes / Q000187:drug effects; D060908:CTLA-4 Antigen / Q000276:immunology; D020226:Cavernous Sinus Thrombosis / Q000175:diagnosis; D002481:Cellulitis / Q000175:diagnosis; D003131:Combined Modality Therapy; D003937:Diagnosis, Differential; D004172:Diplopia / Q000209:etiology; D005094:Exophthalmos / Q000209:etiology; D005260:Female; D005534:Foot Diseases / Q000601:surgery / Q000628:therapy; D006801:Humans; D007167:Immunotherapy; D000074324:Ipilimumab; D008207:Lymphatic Metastasis; D008545:Melanoma / Q000556:secondary / Q000601:surgery / Q000628:therapy; D055622:Orbital Myositis / Q000139:chemically induced* / Q000150:complications / Q000175:diagnosis / Q000188:drug therapy; D012878:Skin Neoplasms / Q000601:surgery / Q000628:therapy
chemical_list: D000911:Antibodies, Monoclonal; D000970:Antineoplastic Agents; D060908:CTLA-4 Antigen; C556706:CTLA4 protein, human; D000074324:Ipilimumab