Osteogenesis imperfecta in a child presenting with neurological features.

Discussion This case history is of interest because of the diagnostic problems presented by the onset of haematuria during the course of severe staphy-lococcal septicaemia treated with massive doses of penicillin, since it may be a complication both of the disease and of the therapy. Haematuria may arise during the course of staphylococcal septicaemia but in this patient the onset was later than the height of the infection, and haematuria resulting from infection is usually associated with pus cells in the urine. There are on record two cases of haematuria during probenecid therapy (Boger & Strickland, 1954) where it was ascribed directly to the uricosuria rather than to drug sensitivity, but in our case the dose was low and the duration of treatment short. When the probenecid was discontinued the haema-turia diminished. However, it again increased when the daily intravenous dose of methicillin was raised from 12 g to 16 g. The persistence of the haematuria coupled with the continued fever despite clinical recovery suggested a methicillin sensitivity. Following reduction of the intravenous methicillin to 8 g/day, the haematuria again temporarily decreased and gradually stopped after the drug was discontinued. It seems likely that the temporary fall in the degree of haematuria after stopping the probenecid was due to a fall in the blood levels of methicillin at that time. Baldwin et al., (1968) described the syndrome of hypersensitivity as characterized by fever, evanescent morbilliform rash, haematuria, azotaemia and eosinophilia. Our patient showed all these features. Of their seven patients four were on doses of methi-cillin ranging from 7 to 24 g/day and demonstrated some or all of these features. All recovered after discontinuing penicillin therapy. Two of their patients had renal biopsies which showed an insterstitial nethritis with tubular damage but no evidence of glomerular lesion or arteritis. This hypersensitivity only appears to develop in patients given high doses of methicillin over prolonged periods and the allergen has been postulated as a penicillin hapten/renal protein. The delayed hyper-sensitivity skin test (Redmond & Levine, 1968) appears to have little diagnostic usefulness since it is present in 21 % of patients who have no recent exposure to penicillin and who can tolerate penicillin without allergic reactions. It may be of interest to note that no difficulties or side-effects were noted while doses of fusidic acid were given in the form of a new preparation in the maximum doses (2 g/day). Acknowledgments …