Hypopigmented T-cell dyscrasia evolving to hypopigmented mycosis fungoides during etanercept therapy

Hypopigmented T-cell dyscrasia evolving to hypopigmented mycosis fungoides during etanercept therapy

Letters S121 J AM ACAD DERMATOL VOLUME 59, NUMBER 5 REFERENCES 1. Langley RGB, Carey WP, Rafal ES, Tyring SK, Caro I, Wang X, et al. Incidence of in...

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Letters S121

J AM ACAD DERMATOL VOLUME 59, NUMBER 5

REFERENCES 1. Langley RGB, Carey WP, Rafal ES, Tyring SK, Caro I, Wang X, et al. Incidence of infection during efalizumab therapy for psoriasis: analysis of the clinical trial experience. Clin Ther 2005;27:1317-28. 2. Carey W, Toth DP, Bissonnette R, Langley R. No evidence for increased risk of infection during efalizumab treatment: a review of the clinical data. Presented at the 13th Annual Meeting of the European Academy of Dermatology and Venereology, Florence, Italy, November 18-21, 2004. Poster #79. 3. Papp KA, Camisa C, Stone SP, Caro I, Wang X, Compton P, et al. Safety of efalizumab in patients with moderate to severe chronic plaque psoriasis: review of clinical data. Part II. J Cutan Med Surg 2005;9:313-23. 4. Tuxen AJ, Yong MK, Street AC, Dolianitis C. Disseminated cryptococcal infection in a patient with severe psoriasis treated with efalizumab, methotrexate and ciclosporin. Br J Dermatol 2007;157:1067-8. 5. Gaylor MN, Duvic M. Generalized pustular psoriasis following withdrawal of efalizumab. J Drugs Dermatol 2004;3:77-9.

Fig 1. A hypopigmented patch without scales on the patient’s left upper back.

doi:10.1016/j.jaad.2008.06.035

Hypopigmented T-cell dyscrasia evolving to hypopigmented mycosis fungoides during etanercept therapy To the Editor: Hypopigmented mycosis fungoides (MF) is a rare variant of MF, described mostly in patients of Asian or African descent.1 The pathogenesis of hypopigmented MF remains to be elucidated. The hypopigmentation is thought to be caused by the cytotoxic effect of T-suppressor lymphocytes on the surrounding melanocytes. The condition is easily misdiagnosed.1,2 The exact incidence of hypopigmented MF is still unknown, likely because of underor misdiagnosis. Biopsy of early hypopigmented MF may initially suggest T-cell dyscrasia, later progressing into MF. Because of the risk of lymphoma, tumor necrosis factor-alfa (TNF-a) inhibitors, such as infliximab, adalimumab, and etanercept, are typically avoided in patients with history of systemic lymphoma. There are a few reports of infliximab and adalimumab treatment associated with the onset or exacerbation of cutaneous T-cell lymphoma.3,4 Here we report a case of hypopigmented T-cell dyscrasia on the skin that progressed to hypopigmented MF after etanercept treatment over a 6-month period. The patient is a 61-year-old African American female with a history of seronegative rheumatoid arthritis who presented to the dermatology department with a 20-year history of a few pruritic hypopigmented patches on her face, trunk, and extremities (Fig 1). Although T-cell dyscrasia was suspected 7 years before the initiation of etanercept, multiple skin biopsies were nondiagnostic. For the arthopathy, the patient was treated initially with methotrexate and later switched to etanercept for

Fig 2. A, Subtle alignment of lymphocytes with halos along the dermoepidermal junction. B, Focal intraepidermal aggregate of atypical lymphocytes (Pautrier microabscess). (Hematoxylineeosin stain; original magnification: A, 320; B, 340.)

6 months. After initiating etanercept, the patient reported an increasing number of hypopigmented patches with a worsening of pruritus. After 6 months of twice-weekly etanercept injections, physical examination of the chest, back, arms, and legs revealed multiple 1- to 10-cm hypopigmented patches with no scale. No ectropion, alopecia, or lymphadenopathy was noted. A punch biopsy taken from the left back revealed atypical lymphoid epidermotropism consistent with cutaneous T-cell lymphoma (Fig 2, A). The specimen was noted to exhibit mild to moderate superficial perivascular atypical lymphocyte infiltrate with alignment along the basement membrane zone with vacuolization and epidermotropism with focal Pautrier microabscess, coarse papillary dermal fibrosis, and occasional plasma cell and numerous melanophages, consistent with the diagnosis of MF (Fig 2, B). A follow-up T-cell flow cytometry panel supported the diagnosis of MF. Etanercept was

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J AM ACAD DERMATOL NOVEMBER 2008

discontinued. The patient was placed back on methotrexate and ultraviolet phototherapy with reduction of pruritus and number of lesions. In hypopigmented MF, the predominant location of the persistent hypopigmented lesions on nonphotodistributed areas of the body is a clue to diagnosis. Pruritus is a commonly associated symptom. In this case, the patient was started on etanercept, a TNF-a inhibitor, for rheumatoid arthritis, which worsened her pruritus and increased the number of hypopigmented lesions. A worsening of systemic lymphoma is a well known risk of TNF-a blockers. There have been few reports of onset or worsening MF variants associated with treatment with other TNF-a blockers including infliximab and adalimumab.3,4 The hypopigmented variant joins the list of subtypes of MF unmasked by the initiation of etanercept treatment. Gary S. Chuang, MD, Daniel I. Wasserman, MD, H. Randolph Byers, MD, PhD, and Marie-France Demierre, MD, FRCPC From the Department of Dermatology, Boston University Medical Center, Boston, Massachusetts Funding source: None. Conflicts of interest: None declared. Reprint requests: Marie-France Demierre, MD, FRCPC, Skin Oncology Program, Boston University Medical Center/Boston Medical Center, 720 Harrison Ave, DOB-801A, Boston, MA, 02118 E-mail: [email protected] REFERENCES 1. Qari MS, Li N, Demierre MF. Hypopigmented mycosis fungoides and literature review. J Cut Med Surg 2000;4:142-8. 2. Sezer E, Sezer T, Senavli A, Koseoglu D, Filiz N. Hypopigmented mycosis fungoides in a Caucasian child. Eur J Dermatol 2006;16:584. 3. Dalle S, Balme B, Berger F, Hayett S, Thomas L. Mycosis fungoideseassociated follicular mucinosis under adalimumab. Br J Dermatol 2005;153:207-8. 4. Sanli H, Ataman S, Akay BN, Yilmaz A, Yildizlar D, Gurgey E. Mycosis fungoides in a patient with ankylosing spondylitis during infliximab therapy. J Drugs Dermatol 2007;6:834-6. doi:10.1016/j.jaad.2008.06.042

Multiple eccrine hidrocystomas treated with glycopyrrolate To the Editor: Eccrine hidrocystomas are benign cysts with eccrine differentiation. They may be multiple or solitary. In 1893, Robinson1 reported multiple hidrocystomas in women who worked in hot, humid environments. Eccrine hidrocystomas tend to

Fig 1. Note multiple 2- to 2.5-mm smooth, shiny, soft papules on the nose.

enlarge with excessive sweating caused by increased ambient temperature or exercise, likely because of sweat retention. Treatment of multiple eccrine hidrocystomas is difficult because of the number and location of lesions. We report a case of multiple eccrine hidrocystomas that responded to glycopyrrolate, an oral anticholinergic agent. A 39-year-old white male reported a 15-year history of asymptomatic lesions on the face which were more noticeable during the summer months and significantly enlarged after exercise. Examination revealed multiple 2- to 2.5-mm, dome-shaped, skincolored to bluish papules located predominately over the cheeks and nose (Fig 1). Histopathology showed a cystic space within the dermis lined by a flattened layer of epithelium characteristic of eccrine hidrocystoma (Fig 2). He failed topical aluminum chloride 6.25% daily for 1 month. He was then started on an oral anticholinergic agent ( glycopyrrolate 1 mg twice daily). This significantly inhibited the enlargement of the lesions during the summer and after exercise. He tolerated treatment without side effects. He takes glycopyrrolate only during the summer months because the lesions are not prominent during the rest of the year and do not significantly enlarge after exercising in the cooler months. Topical anticholinergic medications (atropine and scopolamine) that reduce sweating have been shown to induce transitory resolution of hidrocystoma2; however, they do not work consistently, and they can be associated with anticholinergic side effects, such as nausea and blurred vision. Electrodesiccation or other local destructive methods are useful but run the risk of scarring.3 Lesional puncture and drainage may be helpful, but recurrence is common.4 Pulseddye laser produces variable success and has a slight