Letter to the editor

Palmoplantar hyperkeratosis as the first symptom of mycosis fungoides

To the editor: Palmoplantar keratoderma is a not uncommon manifestation of cutaneous T-cell lym- phoma; however, it usually occurs in the advanced stages of SCzary syndrome. We report a patient who first showed hyperkeratotic lesions of the palms and soles, developing years after psoriasiform lesions on his extremities. Biopsy of both types of lesions showed typical dermatopathologic changes of myco- sis fungoides (MF), although SCzary cells were not present in the peripheral blood smear.

A 57-year-old man began showing signs of pal- moplantar hyperkeratosis three years ago. One year later, psoriasiform plaques appeared on the elbows and legs that persist to this day. Four years ago, the patient was operated for giant hyperplasia of both lingual tonsils, that histologically showed follicular lymphoid hyperplasia. Subsequently, he underwent cobalt-beam therapy because of relapse of the tonsil disease.

Physical examination disclosed a palmoplantar hyperkeratosis with an erythematous base that trans- gressed up to the back of the feet, remaining on the

palm of the hands (Fig. 1). On the elbow, knee and leg, there were red violet and infiltrated plaques with moderate hyperkeratosis and scaling. Neither lym- phadenopathy nor organomegaly was found. Biop- sies were done of the palms, soles and elbows; all confirmed the diagnosis of MF due to the existence of characteristic infiltrate of atypical lymphocytes with epidermotropism and Pautrier microabscesses. Monoclonal antibody studies showed that the cells of the infiltrate were T-helper lymphocytes (OKT4). Electron microscopy revealed large atypical cells with folded nuclei, but the peripheral blood smear did not show SCzary cells. Findings from a further search for lymphatic and systemic involvement were normal. The patient was treated with Re-PUVA for 3 months and improved initially but never reached complete remission.

Atypical manifestations of MF have been de- scribed, such as granulomatous, pustular, bullous, hypopigmented, blue polka dot macules, fixed-drug eruption and hyperkeratotic lesions. When the palms and soles are affected by MF, eczematous [l], ulcer- ous [2], sclerodermic [3], ischemic [4], pustular [51 and bullous lesions 161 have been reported. Credit for describing the association of palmoplantar hyperker-

Fig. 1. Hyperkeratotic lesions in palms and soles.

atosis and MF is attributed to Hallopeau and Bureau [7]. Since then, only a few such cases have been reported in the literature [8-lo]. All affected patients had advanced stages of cutaneous T-cell lymphoma, unlike our case who presented palmoplantar hyperk- eratosis as the first clinical feature of MF. Differen- tial diagnosis considered was Bazex syndrome and adult T-cell leukemia/lymphoma affecting tongue with cutaneous lesions of MF. However, our case did not show atypical cells in the excised tongue tonsils.

In conclusion, we report a case with palmoplantar hyperkeratosis as the first symptom of MF unlike previously published cases where this occurred only in advanced stages of cutaneous T-cell lymphoma or SCzary syndrome.

Francisco Russo *, Miguel Ortega, Jose Carlos Moreno, Francisco Camacho

Department of Medical Surgical Dermatology and Venereology, University School of Medicine, AC&. Dr. Fedriani 3, Seville

31075, Spain

[l] Voigtfinder V, Hartmann AA, Adam W, et al. Mycosis fungo]de: ttiologie innattendue d’un eczema chronique des

mains avec gigantisme digital. Ann Dermatol Venereol 1988;115:1212-4.

Letter to the editor

[2] Robert A, Mark J, Donald M, John A. Ulceration of the

palms and soles: an unusual feature of cutaneous T cell

lymphoma. Acta Derm Venereol @to&h) 1990;70:523-5. [3] Wei N, Foon KA. Sclerodactyly in a patient with mycosis

fungoides. Arch Intern Med 1985;145:139-40.

[4] Garioch JJ, Todd P, Soukop M, Thomson J. T-Cell Lym- phoma presenting with severe digital ischaemia. Clin Exp Dermatol 1991;16:202-3.

[S] Moreno JC; Ortega M, Conejo-Mir JS; Sanchez Pedreho P. Palmoplantar pustulosis as a manifestation of cutaneous T cell Lymphoma (mycosis fungoides). J Am Acad Dermatol

1990;23:758-9. [6] Aractingi S, Robert C, Reygagne P, Verola 0, Dubertret L.

Syndrome de SCzary avec lesions bulleuses palmo-plantaires.

Ann Dermatol Venereol 1992;119:894-7. [7] Hallopeau H, Bureau G. Sur une erythrodermie mycosique

avec hiperkeratose plantaire et palmaire et peut-etre neo-

plasie initiale. Bull Sot Dermatol 1896;7:222. [8] Tomsick RS: Hyperkeratosis in mycosis fungoides. Cutis

1982;29:621-3. [9] Thiers H, Moulin G, Racouchot J, Fayolle J, Perrot H. Etat

hyperkdratosique paraneoplasique au tours d’un mycosis fon- g$de ganglionnaire secondaire d un parapsoriasis en plaques.

Bull Sot Fr Dermatol Syphiliogr 1967;74:633-5. [lo] Homayoun A, Moshe 2. Palmoplantar hyperkeratosis in my-

cosis fungoides. J Am Acad Dermatol 1985;13:897-9.

* Corresponding author. Tel.: 954376474.

SSDI 0926-9959(95)00037-2

Unusual onset of Lyme borreliosis simulating ber- pes zoster

To the editor: Erythema (chronicum) migrans (EM) is a cardinal sign of early Lyme borreliosis (LB) and occurs in 60% to 80% of cases [1,2]. EM is sometimes associated with constitutional signs and symptoms, including arthralgias, myalgias, neck stiffness, lymphoadenopathy, fever, headache, chills, photophobia and disesthesias [l-3]. When the onset of LB is typical, the diagnosis is usually easy. How- ever LB sometimes starts with larvate, largely in- complete or atypical manifestations similar to those of other diseases [4].

We report the case of an unusual onset of LB characterized by metamerical radiculitis initially wrongly mistaken for herpes zoster.

In May 1994, a 63-year-old female presented with metameric, sub-continuous neuralgic pain started two weeks before, misinterpreted as herpes zoster and

treated ineffectively with specific anti-viral therapy (intravenous acyclovir, 250 mg 3 times per day).

She had a history of numerous tick bites; the last one had occurred two weeks before the onset of symptoms in her left upper thoracic paravertebral area.

The patient described a sub-continuous severe burning sensation refractory to analgesics, localized in the lumbar region with a dermatomal distribution. This area resulted hyperpathic to mild touch. No other neurological signs were observed. On general medical examination three pale erythematous patches of 2 cm diameter, surrounded by normal appearing skin, were observed in the left lumbar area lying in the direction of the skin lines.

Radiographic signs of a slight lumbar-sacral arthrosis were present. Renal ecography resulted negative.

Routine laboratory parameters were completely normal. Serum samples were examined by both im-