avium, E faecium (identified with API20 Strep, API, Biomerieux), and S epidermidis (also isolated from intravascular catheter). The E avium was resistant to vancomycin (by Stokes’ E

method with 30 Jlg discs), but was sensitive to ampicillin. Both the E faecium and S epidermidis were sensitive to vancomycin. The patient was treated with amoxycillin 500 mg three times daily and vancomycin 1 g twice a day for 10 days and recovered. Faeces cultured 24 h after the start of treatment yielded vancomycinresistant E avium and vancomycin-sensitive E faecium. E avium from blood was sent to the Antibiotic Reference Laboratory, Central Public Health Laboratory, UK, where resistance to both vancomycin and teicoplanin (minimum inhibitory concentrations > 32 mg/1) was confirmed. The strain hybridised with a DNA probe specific for the VanA glycopeptideresistance gene and could transfer vancomycin resistance in vitro to a recipient strain of Efaecalis. The origin of this vancomycin-resistant E avium strain is unclear. Neither the patient from whom it was isolated nor any of the other patients or staff on the ward had been transferred from any hospital in which vancomycin-resistant enterococci have been reported. The VanA gene has been reported in other members of ilie gut flora that would normally be regarded as non-pathogenic.5 Undisclosed transfer of this gene may be a possible explanation for the otherwise unexplained appearance of VanA in the E avium reported here. Microbiologists and clinicians should be aware that transferable vancomycin resistance in enterococci is not restricted to specialised units with large vancomycin usage but can also arise in general hospitals where vancomycin is used infrequently. We thank Mr R. Pollard for permission


report this

Third Department of Internal Medicine and Departments of Dermatology and Parasitology, Asahikwa Medical College, Asahikawa 078, Japan


1. Shulman LE. Diffuse fasciitis with hypergammaglobulinemia and eosinophilia: a new syndrome?J Rheumatol 1974; 1 (suppl): 46. 2. Stanek G, Konrad K, Jung M, Ehringer H. Shulman syndrome, a scleroderma subtype caused by Borrelia burgdorferi? Lancet 1987; i: 1490. 3. Sepp N, Schmutzhard E, Fritsch P. Shulman syndrome associated with Borrelia burgdorferi and complicated by carpal tunnel syndrome. J Am Acad Dermatol 1988, 18: 1361-62. 4. Abele DC, Anders KH. The many faces and phases of borreliosis II. J Am Acad Dermatol 1990; 23: 401-10. 5. Miyamoto K, Nakao M, Uchikawa K, Fujita H. Prevalence of Lyme borreliosis spirochetes in ixodid ticks of Japan, with special reference to a new potential vector, Ixodes ovatus (Acari: Ixodidae).J Med Entomal 1992; 29: 216-20.



Pathology Department, North Tyneside General Hospital, North Shields, Tyne & Wear NE29 8NH, UK; and Laboratory of Hospital Infection, Central Public Health Laboratory, London NW9

disappearance of eosinophilia. He is currently on 10 mg prednisolone every second day. Abele and Anders4 suggested that B burgdorferi is closely associated with diverse skin diseases including Shulman syndrome. Various tick species are candidate vectors for B burgdorferi. In Japan Ixodes persulcatus Schulze and I ovatus Neumann are the vectors of B burgdorferi5 and Hokkaido is a focus for both. The patient had a history of a tick bite and mountain excursions in Hokkaido, and he was seropositive for B burgdorferi. This case supports the view that Shulman syndrome may be another clinical expression of B burgdorferi infection.


Vancomycin-resistant enterococci. Communicable Dis Rep 1992; 2: 27. Uttley AHC, George RC, Naidoo J, et al. High-level vancomycin resistant enterococci causing hospital infections. Epidemiol Infect 1989; 103: 173-81. 3. Leclercq R, Derlot E, Duval J, Courvalin P. Plasmid-mediated resistance to vancomycin and teicoplanin in Enterococcus faecium. N Engl J Med 1988; 319: 1. 2.

157-61. 4. Noble WC, Virani

Z, Cree RGA. Co-transfer of vancomycin and other resistance genes from enterococcus faecalis NCTC 12201 to staphylococcus aureus. FEMS Microbiol Lett 1992; 93: 195-98. 5. French G, Abdulla Y, Heathcock R, et al. Vancomycin resistance in south London. Lancet 1992; 339: 818-19.

Borrelia burgdorferi and Shulman syndrome SIR,-Shulman syndrome (eosinophilic fasciitis)l is a scleroderma-like illness with firm taut skin, flexion contractures of extremities, and limitation of joint movement. Histopathology reveals striking thickening of the fascia infiltrated by lymphocytes and plasma cells. Some investigators have implicated Borrelia burgdorferi as a cause, on the basis of a history of tick bites and positive B burgdorferi serology.2,3 A 49-year-old man noticed swelling of the extremities in February, 1992. This gradually worsened, resulting in tightening of the skin and flexion contractures of the fingers. He had a history of a tick bite at age 17 when on a mountain excursion in Hokkaido, and until recently he had made other trips to mountains occasionally. He was admitted to our hospital on July 14. He had eosinophilia (white blood cells 8300/nl, 33% eosinophils) and hypergammaglobulinaemia. Rheumatoid factor, scleroderma antibodies, and serological tests for syphilis were negative. Right forearm biopsy revealed increased collagen bundles in subcutaneous fat and remarkable thickening of the fascia with infiltration of lymphocytes and plasma cells. Visceral manifestations were absent. Shulman syndrome was diagnosed. Tests for B burgdorferi (IgG/IgM FASTLYME kit; 3M Diagnostic System) were done four times while he was in hospital and were abnormal (14-15%, normal < 10%). Isolation of B burgdorferi from the skin lesion was unsuccessful. He did not respond to doxycycline and amoxycillin. Prednisolone 40 mg per day was started on Aug 20 and proved effective; his symptoms rapidly improved with the

lysosomal enzyme abnormalities in galactosaemia

reported serum glycoprotein abnormalities in galactosaemia (decreased thyroxine binding globulin [TBG] and abnormal isoelectric focusing of sialotransferrins with a shift towards the cathode).l These features are also found in the carbohydrate-deficient glycoprotein (CDG) syndromes, a newly recognised group of genetic diseases with major SiR,—We have untreated classic

system involvement.2,3 Since there





lysosomal enzyme abnormalities in CDG syndromes,2,4 we studied serum p-N-acetylhexosaminidase and ot-fucosidase in two newborn babies with galactose-1-phosphate uridyltransferase deficiency before and during treatment with a galactose-free diet. Isoelectric focusing was done with 12 ul serum on agarose gels in a pH gradient 5’0-8-0 with a Pharmacia Phast apparatus. Enzyme activities were measured with fluorogenic substrates. The isoelectric focusing pattern of p-hexosaminidase was abnormal in both patients before treatment (figure). There were several additional cathodal bands as well as an abnormal anodal

A B C D E F *





Lane A=control (newborn baby), B = patient with CDG syndrome, C and D infants with classic galactosaemia before treatment, E = D after 1 week of galactose-free diet, F=control (infant). Normally in newborn babies (A), band pattern is heavier than in infants (F). =

Borrelia burgdorferi and Shulman syndrome.

1472 avium, E faecium (identified with API20 Strep, API, Biomerieux), and S epidermidis (also isolated from intravascular catheter). The E avium was...
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