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© 1995 British Society for Rheumatology

case-report

ARTHROPATHY, LEUCOPENIA AND RECURRENT INFECTION ASSOCIATED WITH A TcR {gamma} {delta}POPULATION

E. HODGES, C. QUIN, A. M. FARRELL*, S. CHRISTMAS{dagger}, H. F. SEWELL*, M. DOHERTY{ddagger}, R. J. POWELL{dagger} and J. L. SMITH

Molecular Immunology Group, Southampton University Hospitals NHS Trust
*Department of Immunology, University Hospital NHS Trust Nottingham
{dagger}Department of Immunology, Liverpool University City Hospital NHS Trust Nottingham
{ddagger}Department of Rheumatology, City Hospital NHS Trust Nottingham

Correspondence to: Correspondence to: E. Hodges, Molecular Immunology Group, Wessex Immunology, Southampton University Hospitals NHS Trust, Tremona Road, Southampton SO16 6YD.

This report documents the presence of a clonal {gamma}{delta} T-cell receptor (TcR) population in the blood of a patient who presented with an arthropathy of undetermined cause, leucopenia and splenomegaly. There was no evidence for lymphoid malignancy clinically or at post-mortem. The phenotype and genotype of the clonal T-cell population were not associated with the predominant TcR{delta} rearrangement found in peripheral blood {gamma}{delta} cells, but were similar to those found in {gamma}{delta} TcR cells infiltrating rheumatoid synovium. The data indicate the presence of a monoclonal population of {gamma}{delta} TcR cells which in the face of continued immunosuppression behaved benignly. The case may represent a cytomorphologically atypical example of the large granular lymphocytes, neutropenia and arthropathy syndrome/lymphoproliferative disease of granular lymphocytes and, although the patient's clinical features were not ‘classical’, rheumatoid arthritis (RA) may have been the underlying primary disorder.

KEY WORDS: Arthropathy, {gamma}{delta} TcR Lymphocytes, Genetic analysis


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