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Ann Dermatol Venereol. 2002 Oct;129(10 Pt 1):1161-3.
[Unilateral focal dermal hypoplasia]

[Article in French]

Denis-Thely L, Cordier MP, Cambazard F, Misery L.

Service de Dermatologie, CHU Saint-Etienne, France.

INTRODUCTION: Goltz's syndrome or focal dermal hypoplasia is an X-linked dominant disease. Patients suffer from cutaneous, bone, dental and ocular disorders. The mutated gene has not been identified. CASE-REPORT: A 16 month-old girl presented, since her birth, numerous malformations and cutaneous lesions. She had only three toes and three metatarsians on the right foot. The right hand possessed only four fingers but five metacarpiens. No bone abnormality was observed on the left side. Cutaneous lesions were also limited to the right side: linear para-median epidermal hamartoma on the face, several areas of alopecia, longitudinal atrophic and hyperpigmented striations on the lower limbs. Both median incisors were dysplasic. Nails of right hand and foot were absent or striated. Radiographies did not show infra-clinical bone lesions. At birth, trans-fontanellar sonography and sonographies of thorax and abdomen were normal, as well as ophthalmological examination. Psycho-motor development was normal. The diagnosis of Goltz syndrome was proposed. There was no other case in the family. DISCUSSION: This case is very unusual because lesions were restricted to the right side. To our knowledge, only one other case has been reported. Mutation de novo is probable and may be post-zygotic. In the future, there is no reason to fear the occurrence of another case in this family.

online pharmacy ref. source: www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=PubMed&list_uids=12442131&dopt=Abstract

Proc Natl Acad Sci U S A. 2002 May 14;99(10):7148-53.
Corticotropin-releasing hormone: an autocrine hormone that promotes lipogenesis in human sebocytes.

Zouboulis CC, Seltmann H, Hiroi N, Chen W, Young M, Oeff M, Scherbaum WA, Orfanos CE, McCann SM, Bornstein SR.

Department of Dermatology, University Medical Center Benjamin Franklin, The Free University of Berlin, 14195 Berlin, Germany. zoubouliedizin.fu-berlin.de

Sebaceous glands may be involved in a pathway conceptually similar to that of the hypothalamic-pituitary-adrenal (HPA) axis. Such a pathway has been described and may occur in human skin and lately in the sebaceous glands because they express neuropeptide receptors. Corticotropin-releasing hormone (CRH) is the most proximal element of the HPA axis, and it acts as central coordinator for neuroendocrine and behavioral responses to stress. To further examine the probability of an HPA equivalent pathway, we investigated the expression of CRH, CRH-binding protein (CRH-BP), and CRH receptors (CRH-R) in SZ95 sebocytes in vitro and their regulation by CRH and several other hormones. CRH, CRH-BP, CRH-R1, and CRH-R2 were detectable in SZ95 sebocytes at the mRNA and protein levels: CRH-R1 was the predominant type (CRH-R1/CRH-R2 = 2). CRH was biologically active on human sebocytes: it induced biphasic increase in synthesis of sebaceous lipids with a maximum stimulation at 10(-7) M and up-regulated mRNA levels of 3 beta- hydroxysteroid dehydrogenase/Delta(5-4) isomerase, although it did not affect cell viability, cell proliferation, or IL-1 beta-induced IL-8 release. CRH, dehydroepiandrosterone, and 17 beta-estradiol did not modulate CRH-R expression, whereas testosterone at 10(-7) M down-regulated CRH-R1 and CRH-R2 mRNA expression at 6 to 24 h, and growth hormone (GH) switched CRH-R1 mRNA expression to CRH-R2 at 24 h. Based on these findings, CRH may be an autocrine hormone for human sebocytes that exerts homeostatic lipogenic activity, whereas testosterone and growth hormone induce CRH negative feedback. The findings implicate CRH in the clinical development of acne, seborrhea, androgenetic alopecia, skin aging, xerosis, and other skin disorders associated with alterations in lipid formation of sebaceous origin.

online pharmacy ref. source: www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=PubMed&list_uids=12011471&dopt=Abstract

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Latanoprost, a selective FP prostanoid receptor agonist used in the treatment of glaucoma, has a hypertrichotic side effect. Using the macaque model of androgenetic alopecia, we examined the effect of latanoprost on hair growth. Eight monkeys were divided into 2 groups; one group received a daily topical application of 50 microg/ml of latanoprost for 5 months; a control group had a daily application of vehicle. For an additional 3 months, 2 monkeys from each group were given 500 microg/ml latanoprost, while the remaining monkeys continued with the previous treatment. Hair growth was evaluated by monthly photographs and phototricho-graphic analysis. Fifty microg/ml of latanoprost caused minimal hair growth. Latanoprost at 500 microg/ml induced moderate to marked hair regrowth with 5-10% conversion of vellus hairs to intermediary or terminal hairs. The vehicle group showed no effect. Further evaluation of latanoprost as an agent for treatment of human androgenetic alopecia is indicated.

online pharmacy ref. source: www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=Retrieve&db=PubMed&list_uids=12013211&dopt=Abstract

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