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OBJECTIVES: Combined oral contraceptives (COC) and spironolactone are the first and second-line treatments of mild hirsutism, since the use of cyproterone acetate was restricted to the treatment of severe hirsutism by the French guidelines for hyperandrogenism published in May 2020. Because spironolactone was until now barely used in France, the aim of this study was to evaluate the indication, efficacy and impact on quality of life of COC and spironolactone treatments on mild hirsutism in non-menopausal women. METHODS: This retrospective monocentric study was conducted between June 2020 and October 2021. It included patients with mild hirsutism who received a prescription of COC or/and spironolactone. Modified Ferriman and Gallwey score (FGm) was performed by clinicians and self-rated by patients during the follow-up. Hirsutism-related quality of life was assessed using the Dermatology Life Quality Index (DLQI) and a visual analog scale. RESULTS: A total of 44 patients were included, but only 30 patients received the treatment for 6 months. 70% of patients were free of side effects. Clinically we observed a decrease of 26% in the FGm score rated by clinicians and patients after 6 months of treatment (P<0,01). This was not correlated with an improvement in quality of life. CONCLUSIONS: The data collected showed the clinical efficacy of both COC and spironolactone in the treatment of mild hirsutism. These two treatments were well-tolerated. However, the quality of life scores did not improve after 6 months. These treatments should be evaluated after a longer period.

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OBJECTIVES: Cyproterone acetate is actually the first-line anti-androgenic treatment for severe hyperandrogenism in women. However, as this treatment is currently controversial, the objective of the study was to evaluate the safety and efficacy of spironolactone as a relay for cyproterone acetate. METHODS: This is a monocentric retrospective study conducted between December 2002 and January 2018 at the Jeanne de Flandre Hospital at the University Hospital Center of Lille. Patients with clinical hyperandrogenism who received treatment with cyproterone acetate and then spironolactone were included. A total of 37 patients were clinically and biologically evaluated before treatment, followed by cyproterone acetate and spironolactone. RESULTS: Clinically, the vast majority of patients were satisfied with the relay by spironolactone and found no difference between the two treatments. Biologically, testosterone and delta-4 androstenedione levels were significantly decreased with cyproterone acetate and spironolactone compared to no treatment. No significant differences were found when comparing levels under cyproterone acetate and under spironolactone. In addition, 87,5% of patients were free of side effects. CONCLUSIONS: The data collected show the clinical and biological efficacy of spironolactone as a relay for cyproterone acetate in the treatment of hyperandrogenism. This anti-androgen therefore appears as an effective and well-tolerated alternative, as a relay for cyproterone acetate in patients with hyperandrogenism.

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The authors are reporting here for the first time a documented case of androgen-secreting luteal cyst responsible for primary sterility in Mali. A 26-year married woman with a history of familial hyper androgenia of diabetes and high blood pressure who consulted for hyper androgen syndrome and primary infertility. Hirsutism with the presence of hairs on her chin, upper lips, thorax, forearms, arms, and her legs under waxing which made a Ferriman and Galloway scoreof10. A pelvic ultrasound coupled with a laparoscopy allowed us to retain the diagnosis of luteal cyst. CONCLUSION: Hyper androgenicity in women is rare and the luteal cystis one of its multiple etiologies.

Les auteurs rapportent le premier cas de kyste lutéal androgéno-sécrétant responsable d'une stérilité primaire documenté au Mali. Il s'agissait d'une femme mariée de 26ans, avec des antécédents d'hyper androgénie familiale de diabète et hypertension artérielle qui a consulté pour syndrome d'hyper androgénie et stérilité primaire. Hirsute avec la présence de poils au niveau du menton, des lèvres supérieurs, du thorax, des avant-bras, des bras, des jambes sous épilation ce qui faisait un score de Ferriman et Galloway à 10. Une échographie pelvienne couplée à une coelioscopie nous a permis de retenir le diagnostic de kyste lutéal. CONCLUSION: L'hyper androgénie chez la femme est rare et d'étiologie multiple parmi lesquelles le kyste lutéal.

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BACKGROUND: Non-classical congenital adrenal hyperplasia (NC-CAH) is a recessive autosomal disease caused by a deficiency of adrenal steroidogenesis enzymes. It must be distinguished from classical CAH, either simple virilising or salt-wasting, diagnosed during the neonatal period and responsible for potentially lethal disorders of sexual differentiation. NC-CAH presents a simpler and less specific clinical picture. Herein, we present two cases comprising twin girls consulting for diffuse hypertrichosis. PATIENTS AND METHODS: Two 5-year-old twin girls were seen at our consultation for increased pilosity on all four limbs, but with no facial pilosity or synophrys, as well as comedones on the chin. Their height and weight and psychomotor development was normal, with no signs of precocious puberty and no clitoral hypertrophy. Levels of 17OH-P and SDHA were high, while FSH and LH were low and IGF1 and TSH were normal. Analysis of gene CYP21 associated with NC-CAH showed mutations p.V281L and IVS2-13A/C>G. Mutation p.V281L was present in the heterozygous state in the older sister and the father, together with moderate hyperpilosity but without hirsutism or acne. No mutations were found in the mother, indicating either de novo appearance of mutation IVS2-13A/C>G in the twins or germline mosaicism in the mother. DISCUSSION: We diagnosed NC-CAH as the cause of diffuse hypertrichosis in these twins. This disease is not rare, with a prevalence of 1/1000 to 1500 among peoples of European descent. It is often diagnosed late since routine neonatal screening is not performed. In some cases, NC-CAH remains asymptomatic. The appearance of pubic hair at around 5 to 7 years is the initial reason for consultation, particularly with a dermatologist. Hyperandrogenism varies, involving hirsutism, acne, fertility disorders and premature ageing of bone. Cortisol and aldosterone levels are generally normal. The risk of acute adrenal insufficiency is extremely low. Differential diagnosis concerns ovarian or adrenal tumors and polycystic ovary syndrome. 21-OH deficiency results in defective cortisol and aldosterone synthesis, and thus in raised ACTH, leading to increased adrenal androgen secretion. The early appearance of secondary sexual characteristics is associated with a gradual accumulation of 17-OHP. Depending on severity, hydrocortisone or anti-androgens may be given, or where treatment fails, aesthetic measures such as epilation or hair discolouration may be performed. CONCLUSION: In children presenting NC-CAH, the appearance of pubic hair and hirsutism often constitute the initial reasons for consultation, including with dermatologists. However, hypertrichosis may occur in isolation. It is important that these abnormalities be clearly known to enable early diagnosis and rapid initiation of simple and readily accessible treatment.

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