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128


Successful Treatment of Generalized Essential Telangiectasia With 6-Mercaptopurine

Glazer, Alex M; Sofen, Bryan D; Rigel, Darrell S; Shupack, Jerome L

Generalized essential telangiectasia (GET) is a notoriously difficult to treat disorder with no current satisfactory treatments. This case and discussion report the use of 6-mercaptopurine (6-MP) as a successful treatment for GET. Moreover, we show that GET may represent a state of increased angiogenesis, a paradigm shift from the current understanding that these telangiectasias represent dilatations of only pre-existing vessels. This new view of GET may drive others to look at novel agents for treatment

J Drugs Dermatol. 2017;16(3):280-282

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PMID: 28301625
ISSN: 1545-9616
CID: 2529272

Adiposis dolorosa

Schaffer, Panta Rouhani; Hale, Christopher S; Meehan, Shane A; Shupack, Jerome L; Ramachandran, Sarika
We report a 46-year-old woman with a nine-year history of obesity; chronic diffuse pain in the adipose tissue of her medial upper arms, lower trunk, and thighs; multiple biopsy-proven lipomas and angiolipomas; and a lipomatous pseudomass of the breast. Her systemic symptoms included generalized weakness, fatigue, memory impairment, and arthralgias. Although some of the lesions were tender, most were only appreciated with palpation. Her clinical history and histopathologic data suggested adiposis dolorosa (Dercum's disease). Owing to the chronic pain, an interdisciplinary approach with the use of analgesics and doxycycline has been initiated.
PMID: 25526345
ISSN: 1087-2108
CID: 1411612

Development of linear IgA bullous dermatosis in a patient with psoriasis taking ustekinumab

Becker, Joanna G; Mundi, Jyoti P; Newlove, Tracey; Mones, Joan; Shupack, Jerome
PMID: 22980274
ISSN: 0190-9622
CID: 178231

Familial benign chronic pemphigus (Hailey-Hailey disease)

Hunt, Raegan; O'Reilly, Kathryn; Ralston, Jonathan; Kamino, Hideko; Shupack, Jerome L
We present an atypical case of familial benign chronic pemphigus (Hailey-Hailey disease) that manifested with relapsing, flaccid vesicles and erosions, which were limited to the upper chest, anterior aspect of the neck, and anterior aspects of the upper arms without intertriginous involvement. Although individual eruptions in this patient demonstrated asymmetry, relapses did not obey a segmental distribution. To the best of our knowledge, no other patient has been described with symmetric lesions that were localized solely to the anterior upper body without a prior history of lesions at commonly affected disease sites, which include skin folds, the back, and the posterior and lateral aspects of the neck. This unusual presentation of Hailey-Hailey disease highlights the variable nature of the disease
PMID: 21163165
ISSN: 1087-2108
CID: 115722

Generalized discoid lupus erythematosus [Case Report]

Farley-Loftus, Rachel; Mahlberg, Matthew; Merola, Joseph F; Votava, Henry J; Meehan, Shane; Stein, Jennifer; Shupack, Jerome L
A 79-year-old woman presented with a four-year history of generalized, erythematous, indurated plaques on the malar areas, back, and extremities. The lesions had been recalcitrant in the past to topical glucocorticoid therapy. A skin biopsy specimen was diagnostic of discoid lupus erythematosus (DLE). A minority of patients with DLE progress to develop systemic lupus erythematosus although generalized DLE is more frequently associated with systemic involvement than is limited disease. Standard therapy of cutaneous lupus includes broad spectrum sunscreens, topical and intralesional glucocorticoids, and antimalarial agents
PMID: 19891926
ISSN: 1087-2108
CID: 108419

Porokeratosis palmaris et plantaris disseminata or a disseminated late-onset variant of porokeratotic eccrine ostial and dermal ductal nevus (PEODDN) with follicular involvement [Case Report]

Hartman, Rachael; Rizzo, Carina; Patel, Rishi; Kamino, Hideko; Shupack, Jerome L
A 48-year-old woman presented with a four-year history of pruritic, hyperkeratotic, spiny papules that began on her chest and spread to her extremities, groin, palms, face, and scalp where it caused non-scarring alopecia. Histopathologic features included cornoid lamella, which is the hallmark of porokeratosis. However, the patient's constellation of findings does not meet diagnostic criteria for any of the five clinical variants of porokeratosis. Her presentation is most compatible with either porokeratosis palmaris et plantaris disseminata (PPPD), which is a rare variant of punctate porokeratosis that can involve any area of the body or late-onset porokeratotic eccrine ostial and dermal ductal nevus (PEODDN), which is a rare, benign hamartoma of the eccrine sweat glands with porokeratotic histopathologic features that has been reported to occasionally have systemic involvement. Treatment of either condition is difficult although there have been reported successes with cryotherapy, surgical excision, and CO2 laser. Since porokeratosis is a disorder of keratinization and our patient has prominent follicular involvement, isotretinoin may be another reasonable therapeutic option
PMID: 19891916
ISSN: 1087-2108
CID: 105294

Acquired smooth-muscle hamartoma [Case Report]

Yancovitz, Molly; Gonzalez, Mercedes E; Votava, Henry J; Walters, Ruth; Kundu, Roopal; Shupack, Jerome L
A 52-year-old woman presented with an occasionally pruritic, hyperpigmented and hypertrichotic, indurated plaque on the left upper arm that initially developed during childhood. Histopathologic examination showed changes that were consistent with a smooth-muscle hamartoma. Cutaneous smooth-muscle hamartomas are uncommon benign neoplasms. Most lesions are congenital, but there have been a few reports of acquired lesions. These lesions have been described as part of a spectrum of neoplasms that include Becker nevi since they share many clinical and histopathologic features
PMID: 19891920
ISSN: 1087-2108
CID: 108420

Generalized essential telangiectasia [Case Report]

Karen, Julie K; Mengden, Stephanie J; Kamino, Hideko; Shupack, Jerome L
A 56-year-old woman presented with multiple, erythematous cutaneous vessels that blanched with diascopy and were symmetrically distributed over both lower and upper extremities. The lesions had been present for approximately 6 years and were asymptomatic. Generalized essential telangiectasia (GET) is an idiopathic syndrome of widespread, asymptomatic telangiectases of unknown cause. In our patient, an extensive laboratory evaluation showed low levels of circulating vitamin C as well as the presence of urinary matrix metalloproteinases. The relevance of these abnormal findings is unclear. No uniformly effective treatment exists for GET. Our patient was started on oral doxycycline and was referred for laser therapy
PMID: 18627745
ISSN: 1087-2108
CID: 94719

Failure of etanercept therapy in disseminated granuloma annulare - Reply [Letter]

Shupack, JL; Siu, K
ISI:000240593100027
ISSN: 0003-987x
CID: 68681

Comparison of tazarotene and minocycline maintenance therapies in acne vulgaris: a multicenter, double-blind, randomized, parallel-group study

Leyden, James; Thiboutot, Diane M; Shalita, Alan R; Webster, Guy; Washenik, Kenneth; Strober, Bruce E; Shupack, Jerome
OBJECTIVE: To evaluate the efficacy of 3 maintenance regimens (topical tazarotene, oral minocycline hydrochloride, or both) in sustaining improvement in acne. DESIGN: Multicenter, open-label treatment phase followed by double-blind, randomized, parallel-group maintenance phase. SETTING: Ambulatory patients in research or referral centers. PATIENTS: Volunteer sample of 189 patients with moderately severe to severe acne vulgaris (110 entered maintenance phase, 90 completed, and 2 discontinued because of adverse events). INTERVENTIONS: All patients were treated with 0.1% tazarotene gel (each evening) and a 100-mg capsule (twice daily) of minocycline hydrochloride for up to 12 weeks. Patients with 75% or greater global improvement at week 12 were randomly assigned to 12 weeks of maintenance therapy with tazarotene gel plus placebo capsules, vehicle gel plus minocycline capsules, or tazarotene gel plus minocycline capsules. MAIN OUTCOME MEASURES: Overall disease severity, global improvement, and lesion counts. RESULTS: All regimens were effective in sustaining improvements in acne. After 12 weeks of maintenance therapy, the mean reductions from baseline in noninflammatory and inflammatory lesion count, respectively, were 60% and 54% with tazarotene, 52% and 66% with minocycline, and 64% and 66% with tazarotene plus minocycline. At week 24, more than 80% of patients in each group had maintained a 50% or greater global improvement from baseline, and more than 50% had maintained a 75% or greater global improvement. CONCLUSIONS: A high percentage of patients with moderately severe to severe acne can maintain improvement in their condition with topical retinoid monotherapy. Maintenance with combination tazarotene and minocycline therapy showed a trend for greater efficacy but no statistical significance vs tazarotene alone. Topical retinoid monotherapy should be considered for maintenance to help minimize antibiotic exposure
PMID: 16702498
ISSN: 0003-987x
CID: 94895