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Unilateral Darier's disease complicated by Kaposi's varicelliform eruption limited to the affected skin

Sir, Granulomatous infiltrates have been described in systemic lymphomas such as Hodgkin’s disease, but also in mycosis fungoides (MF). Indeed, granulomatous MF is a rare histological subtype of MF with about 40 cases reported since its initial description by Ackerman and Flaxman in 1970. While granuloma annulare has been described in association with classical lesions of MF, we have recently observed an unusual granulomatous MF mimicking a granuloma annulare both clinically and histologically, without any clinical lesion of classical MF. A 66-year-old man was seen in our Department in September 1995 with a 1-year history of pruriginous cutaneous eruption lesions that first developed on the dorsum of the feet and secondarily extended to the trunk, legs and arms. Lesions were annular with an erythematous centre without atrophy and a papular erythematous peripheral ring without scaling (Fig. 1). This clinical aspect was suggestive of granuloma annulare. No other skin lesions such as plaques or patches were noticed. Skin biopsies immediately showed patterns of MF and granuloma annulare. Indeed, atypical small and medium-sized lymphocytes proliferated in the upper dermis and infiltrated the epidermis, forming small intraepidermal aggregates (Fig. 2a). Simultaneously, collagenic necrosis was observed, surrounded by an abundant infiltrate consisting of histiocytes, monocyte ⁄ macrophagederived giant cells, and atypical lymphocytes (Fig. 2b,c) that have a similar cytological aspect to epidermotropic lymphocytes. No elastolysis was observed. Polymerase chain reaction (PCR) detected a monoclonal rearrangement of the T-cell receptor c chain on skin biopsy. General investigations were otherwise normal. Psoralen ultraviolet A therapy was initially performed, but was stopped after 2 months because of the efflorescence of multiple actinic keratoses. Daily topical steroids (betamethasone) were prescribed and remission was achieved in 3 months. This treatment was pursued three times weekly for 4 years. Similar annular lesions recurred twice on the legs and arms but regressed when topical steroids were applied more frequently. The patient has been in remission since December 1998 with a weekly application of topical steroids. This patient presented lesions clinically suggestive of granuloma annulare. The cutaneous biopsies showed a pattern compatible with this diagnosis, but the presence of atypical lymphocytes both around the granulomatous infiltrate and in the epidermis, forming microabscesses, led to the diagnosis of granulomatous MF. Clinical presentation of granulomatous MF is variable and the diagnosis may be a fortuitous histological discovery. Lesions may consist of patches, plaques, tumours or erythroderma as in Sézary syndrome. Lesions may be poikilodermic or erythemato-violaceous mimicking sarcoidosis. Granulomatous slack skin syndrome is also a rare variant of granulomatous MF with a particular clinical presentation with areas of pendulous lax skin in axillae and groins. To our knowledge, no case of granulomatous MF clinically mimicking granuloma annulare has been described. Histologically, granulomatous MF may consist of a sarcoidlike granuloma pattern with well-circumscribed collections of epithelioid histiocytes and multinucleated giant cells or less commonly in a granuloma annulare-like pattern with palisaded collection of epithelioid histiocytes with central necrosis. Elastolysis is observed in the granulomatous slack skin variant. Atypical lymphocytes may be hidden by an extensive granulomatous inflammation. In these cases, the pathologist must be vigilant for the atypical features of the granulomatous infiltrate, such as the presence of monocytes ⁄ macrophage-derived giant cells as individuals cells in a small-size infiltrate with little eosinophilic collagenic necrosis. The presence of epidermotropic atypical lymphocytes associated with granulomatous infiltrate is one of the clues to diagnosis and PCR detection of a T-cell monoclonal population is a further argument for granulomatous MF. Granuloma annulare has been described in association with malignant lymphoproliferations such as Hodgkin’s disease, B-cell lymphoma, Sézary syndrome and MF, with a parallel course between granuloma annulare and lymphomatous disease. The histological pattern was that of granuloma annulare without any lymphomatous cells mixed with the granulomatous infiltrate. In these patients, granuloma annulare consists in a non-specific sign of an underlying lymphoma and a sudden increase in the number of granuloma annulare may be concomitant with recurrence or progression of lymphomatous disease. Recently, granuloma annulare has been reported in association with an aggressive granulomatous MF with a parallel evolution of granuloma annulare and granulomatous MF but in this