[DRESS syndrome related to Hexaquine (quinine and thiamine)].

BACKGROUND

Drug Reaction with Eosinophilia and Systemic Symptoms (DRESS syndrome) is a severe drug eruption defined as an association of a drug reaction, mostly cutaneous, laboratory abnormalities with the presence of hypereosinophilia and/or atypical lymphocytes, and involvement of one or more organs. We report a case of DRESS syndrome associated with quinine or thiamine (which in France are combined in Hexaquine with serious hepatitis and renal failure and associated viral reactivation of Human Herpes Virus 6 (HHV6).

METHODS

A twenty-three year-old woman was hospitalized for asthenia, fever, erythroderma, facial edema and painful cervical lymph nodes. Laboratory results showed inflammatory syndrome, hypogammaglobulinemia and the presence of activated atypical lymphocytes. Broad screening for infectious serology showed HHV6 reactivation with marked increase in specific IgG levels within two weeks and detection of circulating DNA in blood by PCR (polymerase chain reaction). Two severe complications subsequently appeared: cytolytic hepatitis and interstitial nephritis. The outcome was favorable with oral corticosteroid therapy. Repeated history-taking did not initially identify any causal agent, but several weeks later, the patient remembered taking Hexaquine (quinine and thiamine) for essential muscular cramps daily for 27 days before the onset of symptoms. The patient had taken no other medication during the previous weeks. One year later, at the end of an enteroviral infection, a skin rash similar to the first episode was seen but in isolation on this occasion, suggesting minor reactivation of DRESS syndrome.

CONCLUSIONS

To our knowledge, ours is the first reported case of DRESS syndrome associated with this drug. Because of the difficulty of identifying the causative drug, even though taken alone, our case once again illustrates the importance of spending adequate time on history taking. Although the physiopathology of DRESS syndrome has not yet been fully elucidated, it is nevertheless probable that it is a multi-factor phenomenon (involving medication and viruses among others).