Palpable Archiform Migratory Erythema Preceded by B-cell Pseudolymphoma in a Japanese Man

Abstract

Sir.Palpable archiform migratory erythema (PAME) is a rare member of the family of T-cell pseudolymphomas occurring preferentially in adull males ( I -5 ). The clinical picture of PAME is distinctive from other pseudolymphomas because infiltrated annular erythema develops into elevated migrating lesions in the trunk as the predilection site. The aetiology of this condition is unknown and the disease runs a chronic course. We report here a case of PAME in which polyclonal populations of T cells proliferated surrounding a preceding B-cell pseudolymphoma. CASE REPORTA 66-year-old man h;id developed a red, small lump iO years previously and after 7 years arc-shaped eruptions began to appear surrounding this initial lesion. The initial examination revealed an indurated smooth surfaced nodule 2.5 cm in diameter surrounded hy arcbitbrm and annular, firm, palpable erythemas on the back (Fig, la). Tbere were no subjective symptoms or superficial lymphadenopathy. The central nodule gradually resolved in 3 months after the first visit to our clinic without any treatment. The archiforEii and annular erythemas waxed and waned, as a part of the lesions disappeared in 2-3 months while other parts tended to clear, without any systemic or topical treatment (Fig. Ib). While oral penicillin, topical steroids of strong potency, and intra-lesional and intravenous injections of interferon-Y(6) were ineffective, oral administration of prednisolone at a starting dose of 30 mg/day follov^-ed by dose-tapering completely cleared the lesions in 8 months.Laboratory findings including red and white blood cell counts, haemoglobin levels, serum electrolytes, liver enzyme levels.serum protein and immunoglobulin levels and C-reactive protein were normal on several occasions during our 3-ycar observation. Anti-nuclear antibodies, and antibodies to Trcponenuipalliilum, human T-lympholrople virus-1, human immunodetlciency virus, hepatitis C virus, and Borrelia hurgihrferi were always negative. Chest X-ray showed no abnormality, and there was no lyinphadenopathy or hepatosplenomegaly hy computerized tomography scanning. Bone marrow aspiration revealed normal cellular composition.In the central nodule, atypical lymphoid cells with large, irregularly shaped, pale nuclei and small lymphocytes lay either intermingled with one another or in a follicular arrangement in the entire upper dermis (Fig, 2a). Lymphoid follicular structures consisted of CD20' large atypical B and CD3' small T cells at a ratio of 9:1 (Fig, 2b). At resolution, mononuclear cell infiltrates localized to perivascular areas with less conspicuous follicular structures that consisted of almost equal numbers of CD20' and CD3' cells. Specimens obtained from arciform erythema on 3 occasions at different siles consistently revealed a dense lymphocytic infiltrate around the blood vessels and hair follicles in the upper and mid dermis, with a CD3 ' to CD20" cell ratio of 7 to 3 (Fig, 3). No follicular structure was apparent. In addition, helper T cells outnumbered cytotoxic T cells since CD4" ...

Recurrent episodes of palpable migratory arciform erythema associated with IVIg infusions

“…Dermatology Online Journal || Case Presentation stimulus [3]. Reports of PMAE in the literature are few and a common triggering factor has not been identified.…”

“…Although some clinicians have reported success in using systemic antibiotics to induce remission of PMAE, no causative infectious etiology has been identified. Other proposed associations based on case reports have included the administration of several different medications, HIV infection, and the presence of a preceding B-cell pseudolymphoma [3][4][5]. Presumably, the large majority of cases present idiopathically.…”