Diphencyprone in the treatment of Satoyoshi syndrome

P8513Azathioprine: A therapy to be considered in alopecia universalis

Sivanie Vivehanantha, MBBS, Birmingham Skin Centre, Birmingham, UnitedKingdom; Shireen Velangi, MBBS, Birmingham Skin Centre, Birmingham, UnitedKingdom

Alopecia areata (AA) is a T lymphocyteemediated autoimmune disorder of the hairfollicle. It is characterized by patchy hair loss developing in otherwise normal skin,with ‘‘exclamation mark’’ hairs around margins of expanding areas. Most cases arelimited to 1 or more patches of hair loss, but in severe cases there may be completeloss of hair on the scalp (alopecia totalis, AT) or on all hair-bearing sites (alopeciauniversalis, AU). Treatment is not mandatory because the condition is benign andspontaneous remissions and relapses are common. However, the condition oftenresults in significant psychological morbidity, thereby justifying treatment. Patientswith AT or AU usually have a poorer prognosis and pose a further therapeuticchallenge. We report a case of AU responsive to azathioprine on 2 separateoccasions. A 36-year-old woman with lifelong severe atopic eczema started todevelop AA as a teenager. From the age of 29, the AA rapidly worsened and within ayear it progressed to AU. In 2007 at the age of 29, she was commenced on oralcyclosporine (stopped after a month because of side effects) and although hereczema responded to treatment, the alopecia continued to progress. In February2008, she was commenced on oral azathioprine for her eczema which respondedwell, and within 6 weeks there was rapid hair regrowth and almost completeregrowth within 8 months. In 2010, azathioprine was stopped as it had lost itsefficacy in controlling the severity of her eczema. Oral methotrexate was started inJune 2010 and although there was variable control of her eczema, the alopeciarelapsed. Methotrexate was stopped in April 2013 as the eczema was poorlycontrolled. Azathioprine was recommenced in May 2013, again with a goodresponse of both her eczema and AU. There are numerous treatment modalitiesfor AA suggested in the literature: intralesional, topical, and systemic corticoste-roids, topical immunotherapy, topical irritants, topical minoxidil, topical prosta-glandin analogue, PUVA, oral immunosuppression, cryotherapy, and excimer lasers.However, there are just 2 reports of the use of azathioprine in the treatment of AA inthe literature; 1 case report of AT and 1 small pilot study involving patients with AAand AT. To the best of our knowledge, this is the first report of AU responding toazathioprine. The rapid regrowth on commencement of azathioprine on the 2separate occasions makes it unlikely that the response is purely related to chance.

MAY 201

cial support: None identified.

P8074Cortexolone 17a-propionate: A new antiandrogen acting on hair dermalpapilla cells for the treatment of androgenic alopecia

Giuseppe Celasco, MD, Cosmo Research and Development, SpA, Lainate-Milano,Italy; Angela Milasi, PhD, Cosmo Research and Development, SpA, CataniaLaboratory, Catania, Italy; Daniel Piacquadio, MD, Therapeutics Inc, San Diego,CA, United States; Luigi Moro, PhD, Cosmo Research and Development, SpA,Lainate-Milano, Italy; Robert Gauthier, Therapeutics Inc, San Diego, CA, UnitedStates

Background: The negative effects (miniaturization, shortening the anagen phase) ofdihydrotestosterone (DHT) on hair dermal papilla cells (DPCs) are well recognized.As a consequence, substances endowed with antiandrogenic properties areregarded as potentially useful in counteracting the hair growth effects of DHT. Inthis study we evaluated the ability of 2 antiandrogens (cortexolone 17a-propionate[CB-03-01] and cyproterone acetate [CA]) to antagonize in vitro the negative effectsof DHT on proliferation of DPC of mice expressing androgen-dependent baldness(AGA-mouse).

Methods: DPCswere obtained from the dorsal skin ofmale B6CBAF1/J mice (CharlesRiver, Italy) just sacrificed. Isolated DPCs were incubated for 48 hrs with DHT alone(0.5-5 �M) or with the associations of DHT+CB-03-01 or DHT+CA at equimolarconcentrations (2.5 �M). At the end of the incubation period the DPCswere labelledwith 5-bromodeoxyuridine, the label was detected by ELISA, and the proliferationindex was quantified using the photometry absorbance method.

Results: DHT incubated alone induced evident dose-dependent inhibition of DPCproliferation up to -32.8% (P\.001). When CB-03-01 was incubated together withDHT, the proliferation index was only minimally reduced (-2.4%), so that CB-03-01almost completely abolished the inhibitory effect of DHTon DPC proliferation (P¼.007 vs. DHT). When CA and DHTwere incubated together, the proliferation indexwas reduced by -24.2%, not significantly different (P ¼ .210) from that induced byDHT alone. The protective effect of CB-03-01 on DPC was ;103 higher than thatinduced by CA. The reason CA, in spite of its well recognized antiandrogenic activity,yielded poorly effective results is not understood. It is possible that the additionalendocrine properties of CA, such as its intrinsic progestinic activity, could interferewith the effects at DPC level.

Conclusions: CB-03-01 represents a potent antagonist of DHT at the DPC level,notably more effective than CA. Because of this mechanism of action, CB-03-01should be regarded as an antiandrogen potentially useful for use in the topicaltreatment of androgenic alopecia.

provided by Cosmo Research and Development, SpA andtics Inc.

Support IntrepidTherapeu

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P8715Differential diagnosis of trichotillomania by trichoscopy

Adriana Rakowska, MD, PhD, Department of Dermatology CSK MSW, Warsaw,Poland; Lidia Rudnicka, MD, PhD, Specjalisci Dermatolodzy, Warszawa, Poland;Malgorzata Olszewska, MD, PhD, Department of Dermatology Medical Universityof Warsaw, Warsaw, Poland

The differential diagnosis between trichotillomania and other causes of hair may bedifficult in dermatologic practice. Trichoscopy (hair and scalp dermoscopy)effectively supports differential diagnosis of various hair and scalp diseases. Theaim of this study was to analyze the usefulness of trichoscopy in differentialdiagnosis of trichotillomania. Trichoscopy was performed in 370 patients (44 withtrichotillomania, 314 with alopecia areata, and 12 with tinea capitis). Statisticalanalysis revealed that the main and most characteristic trichoscopic findings oftrichotillomania are: irregularly broken hairs (44/44; 100% of patients), v-sign(24/44; 57%), flame hairs (11/44; 25%), hair powder (7/44; 16%), and coiled hairs(17/44; 39%). Flame hairs, v-sign, and hair powder were not observed in other typesof hair loss. In conclusion, flame hairs, v-sign, and hair powder are specifictrichoscopy features, which may aid quick, noninvasive, in-office differentialdiagnosis of trichotillomania.

cial support: None identified.

P7924Diphencyprone in the treatment of Satoyoshi syndrome

Nayra Merino de Paz, MD, Hospital Quir�on Tenerife, Santa Cruz de Tenerife,Spain; Francisco Guimera Martin-Neda, MD, PhD, CHUC, La Laguna, Spain;Marina Rodriguez Martin, MD, PhD, Hospital Quir�on Tenerife, Santa Cruz deTenerife, Spain; Miguel Saez Rodriguez, MD, Centro Madre, Santa Cruz deTenerife, Spain; Monica Merino de Paz, NP, CHUC, La Laguna, Spain; PatriciaContreras Ferrer, MD, Hospital Quiron Tenerife, Santa Cruz de Tenerife, Spain;Ruth Pitti Perez, MD, HUNSC, Santa Cruz de Tenerife, Spain

Introduction: Satoyoshi syndrome is a rare syndrome that it is characterized by thepresence of alopecia, diarrhea, muscular spasms, osseous abnormalities, andendocrinopathies. About 52 cases have been described in the literature. Oralglucocorticoids, azathioprine, tacrolimus, methotrexate, and intravenous immuno-globulins have been described as useful treatments in this syndrome.

Case report: This report describes a 12-year-old girl with universal hair loss fromscalp, eyebrows, and eyelashes since 7 years ago. She also presented with painfulintermittent muscle spasms and growth retardation with metaphyseal and epiph-yseal dystrophy. Carbamazepine and otilonium bromide treatment led to thedisappearance of her muscles spasms and diarrhea, and she is currently beingtreated with growth hormone. Several systemic treatments were attempted withoutimprovement 7 years ago, with several adverse events and quality of life affectation.We decided to use safety therapies for the alopecia. Intermittent oral glucocorticoidtherapy (30 mg once a week) was attempted for a month, but a lot of adverse eventsappeared, especially very disturbing diarrhea. Topical glucocorticoids after 4months did not induce hair regrowth. UVB phototherapy induced a hard headache.We started topical diphencyprone once a week with a good response and withoutadverse events.

Discussion: Dyphencyprone is one of the options to treat alopecia areata. It is atopically administered drug that induces a local immune response. In the literature,Satoyoshi syndrome has been treated by oral glucocorticoids, with good results forspasms, alopecia, and diarrhea. Intravenous immunoglobulins have improvedspasms and decreased anti-DNA antibodies. Few patients have been treated withazathioprine, methotrexate and tacrolimus with good response. But all of thesetreatments have a lot of adverse effects and risks, as immunosuppression.

Conclusion: This is the first report of diphencyprone therapy of alopecia associatedwith Satoyoshi syndrome.

cial support: None identified.

J AM ACAD DERMATOL AB89