QJM: An International Journal of Medicine, 2016, 411–412 doi: 10.1093/qjmed/hcv209 Advance Access Publication Date: 11 November 2015 Case report
CASE REPORT
Severe cutaneous vasculitis: an unusual complication of equine antithymocyte globulin treatment K. V. Vinod1, D. Charles2, B. Hitha2, P. Radhakrishna1 and T. K. Dutta2 From the 1Department of General Medicine, Jawaharlal Institute of Postgraduate Medical Education and Research [JIPMER], Dhanvantri Nagar, Pondicherry, India and 2Department of Clinical Haematology, Jawaharlal Institute of Postgraduate Medical Education and Research [JIPMER], Dhanvantri Nagar, Pondicherry, India Address correspondence to Dr K. V. Vinod, Department of General Medicine, JIPMER, Dhanvantri Nagar 605006, Pondicherry, India. email:
[email protected]
Learning point for clinicians Equine antithymocyte globulin treatment for severe aplastic anemia, apart from causing acute severe hypersensitivity reactions such as anaphylaxis and infusion reactions, can rarely be complicated by development of severe cutaneous vasculitis, as a part of serum sickness. Treatment with systemic steroids results in its successful resolution.
Case report Antithymocyte globulin (ATG) preparations derived from hyperimmune serum of horse or rabbit immunized with human thymocytes and lymphocytes are used in the treatment of aplastic anemia. Apart from infusion reactions and anaphylaxis, serum sickness is a well-known complication developing within one to two weeks of ATG treatment initiation.1 Severe cutaneous adverse effects following equine ATG treatment are rare. A 48-year-old man presented in August 2013 with easy fatiguability, tiredness and gum bleeds for 3 months. He was found to have pancytopenia (Hb: 62 g/l, reticulocyte count: 0.5%, leucocyte count: 1.6 109/l, with a differential count of neutrophils 38% and lymphocytes 62% and platelets: 9 109/l). Bone marrow biopsy revealed severe aplastic anaemia. Paroxysmal nocturnal haemoglobinuria (PNH) work-up was negative. He was started on cyclosporine, without significant improvement. In March 2015, because of financial constraints, he received a total of 5 g (25 mg/kg/day 4 days) of equine ATG. Cyclosporine (5 mg/kg/day orally) was continued and serum sickness
prophylaxis with prednisolone (60 mg/day) were started. He received packed red cell and platelet transfusions during ATG treatment. He developed fever, arthralgia of knees, elbows and wrists, abdominal pain and purpuric rash over both legs, 11 days after ATG initiation. Examination revealed cervical lymphadenopathy and extensive palpable purpuric rash and ecchymoses over legs and feet (Figure 1a). There was no obvious focus of infection and no other sites of bleeding were evident. Blood counts revealed Hb: 69 g/l, leucocyte count: 2 109/l (differential count: neutrophils 35%, lymphocytes 64%, monocytes 1%], platelets: 12 109/l, reticulocyte count: 0.1% and erythrocyte sedimentation rate was 50 mm/h [normal
