a case of cutaneous rosai–dorfman disease

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Page 1: A case of cutaneous Rosai–Dorfman disease

CLINICAL DERMATOLOGY AND OTHERCUTANEOUS DISORDERS

P6133Vibration plate purpura: A new lifestyle dermatosis

Allan Gordon Matthews, MBChB, Lanarkshire Centre for Dermatology, Airdrie,United Kingdom; Christopher David Evans, MBBS, Lanarkshire Centre forDermatology, Airdrie, United Kingdom; Joanna Weir, MBChB, LanarkshireCentre for Dermatology, Airdrie, United Kingdom; Valerie Dudgeon, MBChB,Lanarkshire Centre for Dermatology, Airdrie, United Kingdom

We report the case of a localized purpuric eruption on the abdomen induced bywhole body vibration training. A 61-year-old male hair stylist presented with a 4-week history of an itchy rash affecting his abdominal wall. Clinically, the rashresembled a pigmented purpuric dermatosis. Multiple tiny petechiae were super-imposed on ill-defined brownish patches. Skin biopsy revealed a superficialperivascular lymphocytic infiltrate. The patient had hired a whole body vibrationmachine (also known as a vibration plate) in a NewYear plan to tone his muscles andease stiff joints. Twice a day, he positioned himself on the device in an isometricpartial squat, with tensed abdominal muscles, whilst the platform vibrated contin-uously for 10 minutes. The frequency and amplitude of vibrations were preprog-rammed and fluctuated between 0.1-27 Hz and 1-10 mm respectively. He developedpruritus over his abdominal wall within 2 weeks of starting this regime and the rashappeared 4 weeks later. After being advised stop using the machine, the rash fadedwithin 2 weeks, with no adverse sequelae. Whole body vibration (WBV) is arelatively new exercise method, which involves a person performing variousexercises on a platformwhich applies vertical vibrations to the body.WBVmachinesare installed in many gymnasia and health clubs, and some models are marketed foruse in the home. More recently, hospitals and physiotherapy suites have startedintroducing machines for use in rehabilitation populations. During WBV training,vertical vibrations transmitted through the body cause vigorous oscillation of thesoft tissues. In this case, we hypothesize that the resonant reciprocating movementof the soft tissues overlying the patient’s tensed abdominal muscles causedsignificant shearing forces on cutaneous small vessels, resulting in the rash. Analternative explanation is that the rash represented a variant of exercise-inducedpurpura, occurring in an unusual distribution. Exercise-induced purpura typicallypresents as a purpuric rash in the lower limbs and is considered as an acute,microcirculatory deficiency and thermoregulation decompensation that occursafter episodes of exhaustive major muscular activity or unusual or excessiveexercise. With WBV machines becoming more widely used, ‘‘vibration platepurpura’’ is likely to present more frequently. The exact pathophysiology is unclearbut the condition appears benign and does not require directed therapy.

AB38

cial support: None identified.

Commer

P6263Tacrolimus ointment 0.1% in the treatment of scrotal lichen simplexchronicus: An open-label study

Eugene Sern-Ting Tan, MBBS, National Skin Centre, Singapore; Andy Soon-LeongTan, MPH, Annenberg School for Communication, University of Pennsylvania,Philadelphia, PA, United States; Hong Liang Tey, MBBS, National Skin Centre,Singapore

Background: Scrotal lichen simplex chronicus poses a therapeutic challenge asconventional treatment with topical corticosteroids and oral antihistamines is oftenunsatisfactory.

Objective: To evaluate the efficacy of topical 0.1% tacrolimus ointment in thetreatment of scrotal lichen simplex chronicus.

Methods: We performed a prospective, open-label study of adult patients withscrotal lichen simplex chronicus over a period of 6 months from December 2011 toMay 2012 in the Itch Clinic at the National Skin Centre, Singapore. All subjects wereinstructed to apply 0.1% tacrolimus ointment twice daily for 6 weeks. Beforetreatment and at 6 weeks of treatment, the subjects were assessed subjectively ontheir severity and frequency of itch, degree of itch-induced sleep impairment anddisease-related quality of life, and objectively on the clinical disease severity by asingle investigator. We performed Wilcoxon signed-rank tests to compare theseoutcome measures pre- and posttreatment.

Results: Nineteen subjects participated in this study, with a mean age of 48.0 years.Thirteen (68.4%) attended the second visit at 6 weeks. Among them, there wereimprovements in mean itch score (maximum of 10) from 6.7 to 2.9, mean itchfrequency from 14.8 to 3.4 times per week, mean sleep score (maximum of 5) from2.8 to 1.1, mean Dermatology Life Quality Index score (maximum of 30) from 9.6 to3.5, mean disease severity score (maximum of 6) from 3.5 to 1.4, and reduction inmean scrotal surface area involvement from 80% to 50% posttreatment. Theimprovements in these outcomes were all statistically significant (P \ .05). Themean onset of action of topical tacrolimus was 4.6 days. No major adverse effectswere reported apart from 2 subjects who experienced a tolerable mild burningsensation. Of the remaining 6 subjects (31.6%) who dropped out of the study, 4(21.1%) experienced intolerable burning sensation with topical tacrolimus anddiscontinued treatment; 2 subjects reported significant improvement in their itchscore with tacrolimus but defaulted because of personal reasons. We performed asensitivity analysis assuming that the participants who defaulted had no improve-ment in their outcome measures at 6 weeks and repeated the comparisons above.The overall improvements in these outcomes remained statistically significant.

Conclusion: Topical 0.1% tacrolimus ointment, when tolerated, was an effective andsafe treatment for scrotal lichen simplex chronicus in our study population.

cial support: None identified.

Commer

J AM ACAD DERMATOL

P6720A case of cutaneous metastases from multiple myeloma

Karen Connolly, MD, Henry Ford Health System Department of Dermatology,Detroit, Michigan, United States; Margaret Douglass, MD, Henry Ford HealthSystem Department of Dermatology, Detroit, MI, United States

Multiple myeloma is a hematologic cancer of plasma cells. Cutaneous metastasesfrom multiple myeloma are a rare complication seen in aggressive variants of thisdisorder. Patients with multiple myeloma actively being treated who developcutaneous metastases can present a diagnostic dilemma, as many of the newerchemotherapy agents produce novel cutaneous eruptions. We describe the case of a49-year-old white man with a known history of stage IIIA metastatic multiplemyeloma. He was actively undergoing treatment with Velcade (bortezemib), aproteasome inhibitor. Over a 1-month period he developed multiple friableerythematous papules and nodules over the chest, scalp, and lower extremities. Abiopsy of one of the lesions revealed an atypical plasmacytoid proliferationconsistent with metastatic multiple melanoma. Immunostaining revealed CD138positivity and weak focal staining with LCA. Cutaneous metastases of multiplemelanoma are uncommon; a review of the literature in 2003 reported 100 describedcases. They are an extremely poor prognostic indicator and a sign of high tumorburden. On average, death occurswithin 1 year of developing cutaneousmetastases.The IgG subtype is most commonly seen, and treatment is typically chemotherapyaimed at treating the underlying disease.

cial support: None identified.

Commer

P6358A case of cutaneous RosaieDorfman disease

Ezra Mirvish, MD, University of Pittsburgh Medical Center Department ofDermatology, Pittsburgh, PA, United States; Larisa Geskin, MD, University ofPittsburgh Medical Center Department of Dermatology, Pittsburgh, PA, UnitedStates; Rebecca Pomerantz, MD, University of Pittsburgh Medical CenterDepartment of Dermatology, Pittsburgh, PA, United States

RosaieDorfman disease is a rare histiocytic proliferative disorder often character-ized by cervical lymphadenopathy, although patients may present with exclusivelyextranodal disease. We report the case of a 69-year-old woman with ‘‘bumps on theface’’ for approximately 18 months that had temporarily resolved after a steroidinjection in her shoulder for musculoskeletal pain. She had chronic joint pain ofmany years’ duration, but otherwise denied constitutional or system-specificsymptoms, including fever, fatigue, unexplained weight loss, and night sweats.Physical examination revealed several asymptomatic erythematous translucent firmpapules present on her temples and zygomatic arches bilaterally and only 1 addi-tional lesion on the patient’s left arm. Differential diagnosis included cutaneouslymphoma, lymphomatoid papulosis, and foreign body reaction. Biopsy at anoutside institution was interpreted as cutaneous RosaieDorfman versus benignlymphoid hyperplasia. Cutaneous lesions, which are nonspecific appearing, canrarely represent the exclusive manifestation of RosaieDorfman. This case highlightscutaneous RosaieDorfman as being a clinical entity, distinct from its systemiccounterpart, which may be considered in the clinician’s differential diagnosis evenwhen cervical lymphadenopathy is not present.

cial support: None identified.

Commer

APRIL 2013