Drug-induced hypersensitivity syndrome (DIHS) is a severe multiorgan reaction related to the reactivation of human herpesvirus (HHV)-6 (1). Recently, the involvement of other types of HHV, including herpes simplex virus (HSV), Epstein-Barr virus (EBV), HHV-7, and cytomegalovirus (CMV), has been reported (2-5). We report here the first case of DIHS possibly associated with reactivation of varicella-zoster virus (VZV).
CASE REPORTA 72-year-old woman was referred to our clinic because of skin eruptions covering almost her entire body. About 4 weeks before, she had taken salazosulphapyridine, bucillamine, and prednisolone (PSL) for treatment of rheumatoid arthritis. However, she stopped taking the drugs one week before referral due to vomiting and diarrhoea. She had a slight fever and generalised skin lesions for 3 more days. She was first hospitalised in the local hospital with a primary diagnosis of drug eruption. Thirty mg/day of PSL was administrated intravenously on the first day of hospitalisation. However, there was no improvement in symptoms and she was referred to our clinic. Her vitals on admission were as follows: temperature of 37.2ºC and blood pressure of 185/82 mmHg. Physical examination revealed infiltrative erythematous macules and papules on the entire body and facial oedema (Fig. 1a). There was no lymphadenopathy. Laboratory data were as follows: white blood cell (WBC) count of 11,200 /mm 3 with 65% neutrophils, 23% lymphocytes, 4% eosinophils, 0% monocytes and C-reactive protein of 3.2 mg/dl (normal < 0.20 mg/dl). Liver and renal functions were within normal limits.A skin biopsy specimen obtained from the left thigh revealed focal spongiosis and basal vacuolar change with lymphocyte exocytosis in the epidermis and superficial perivascular lymphocytic infiltration in the dermis. We suspected drug eruption including a possibility of DIHS because of the prescription of salazosulphapyridine. We prescribed 60 mg/day of oral PSL. We consulted the department of internal medicine regarding gastrointestinal symptoms. She was diagnosed with acute colitis associated with drug eruption and was recommended treatment with antiflatulent and no fasting temporarily. Since the skin lesions and fever were improved, we gradually tapered the daily dose of PSL and terminated administration after 15 days.However, on the 17 th day after onset of the first rash, she developed erythemas and papules on the abdomen again with high fever of 38.0ºC and she showed abnormal values in liver function tests: aspartate aminotransferase (AST) level, 70 IU/l (normal 13-33 IU/l); alanine aminotransferase (ALT) level, 280 IU/l (normal 8-42 IU/l); gamma-glutamyltransferase (γ-GT) level, 66 IU/l (normal 10-47 IU/l); lactate dehydrogenase (LDH) level, 329 IU/l (normal 119-229 IU/l). Ten days later, though WBC count was normalised to 5,500/μl, eosinophilia of 22% occurred. A second skin biopsy from the patient's abdomen revealed marked spongiosis with lymphocyte exocytosis, extravasated erythrocytes in the epidermis and severe basal vacuola...