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Q J Med 1988; 68: 645-655
© 1988 Association of Physicians

other

Lymphomatoid Granulomatosis - Evidence of a Clonal T-Cell Origin and an Association with Lethal Midline Granuloma

SEAN WHITTAKER, LETIZIA FORONI, LUCIO LUZZATTO, IRWIN LAMPERT, PETER AMLOTT, ALISTAIR MUNRO and ROBIN RUSSELL JONES

Departments of Dermatology, Haematology, Oncology and Pathology, Hammersmith Hospital, Royal Postgraduate Medical School Ducane Road, London W12 ONN Department of Immunology, Royal Free Hospital Pond Street, London NW3

Address correspondence to Dr S. Whittaker, Department of Dermatology, Hammersmith Hospital.

Accepted for publication 7 April 1988.

Lymphomatoid granulomatosis and lethal midline granuloma are both characterized histologically by atypical pleomorphic angiocentric infiltrates. Whether these conditions are malignant lymphoproliferative disorders remains controversial.

Here we report the results of studies carried out in a patient with coeliac disease, who developed recurrent self-healing subcutaneous nodules with the histological changes of lymphomatoid granulomatosis and an invasive nasal tumour with the histological features of lethal midline granuloma. The patient subsequently also developed an erythrophagocytic syndrome. Immunocytochemical labelling of both cutaneous and nasal lesions demonstrated a predominant population of T cells with a CD4-negative CD8-positive phenotype. Analysis of DNA from cutaneous tissue revealed a discrete rearrangement of the ß and {gamma} T-cell receptor genes.

These findings suggest that lymphomatoid granulomatosis is a clonal T-cell lymphoproliferative disorder and its association with lethal midline granuloma indicates that both conditions may have a common histogenesis.


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