Liraglutide administration improves hormonal/metabolic profile and reproductive features in women with HAIR-AN syndrome

S Livadas; I Androulakis; N Angelopoulos; A Lytras; F Papagiannopoulos; G Kassi

doi:10.1530/EDM-19-0150

Liraglutide administration improves hormonal/metabolic profile and reproductive features in women with HAIR-AN syndrome

Endocrinol Diabetes Metab Case Rep. 2020 Jun 4:2020:19-0150. doi: 10.1530/EDM-19-0150. Online ahead of print.

Authors

S Livadas¹, I Androulakis¹, N Angelopoulos¹, A Lytras¹, F Papagiannopoulos², G Kassi³

Affiliations

¹ Endocrine Unit, Metropolitan Hospital, Athens, Greece.
² Novo-Nordisk, Athens, Greece.
³ Endocrine Unit, Alexandra Hospital, Athens, Greece.

Abstract

Summary: HAIR-AN syndrome, the coexistence of Hirsutism, Insulin Resistance (IR) and Acanthosis Nigricans, constitutes a rare nosologic entity. It is characterized from clinical and biochemical hyperandrogenism accompanied with severe insulin resistance, chronic anovulation and metabolic abnormalities. Literally, HAIR-AN represents an extreme case of polycystic ovary syndrome (PCOS). In everyday practice, the management of HAIR-AN constitutes a therapeutic challenge with the available pharmaceutical agents. Specifically, the degree of IR cannot be significantly ameliorated with metformin administration, whereas oral contraceptives chronic administration is associated with worsening of metabolic profile. Liraglutide and exenatide, in combination with metformin, have been introduced in the management of significantly obese women with PCOS with satisfactory results. Based on this notion, we prescribed liraglutide in five women with HAIR-AN. In all participants a significant improvement regarding the degree of IR, fat depositions, androgen levels and the pattern of menstrual cycle was observed, with minimal weight loss. Furthermore, one woman became pregnant during liraglutide treatment giving birth to a healthy child. Accordingly, we conclude that liraglutide constitutes an effective alternative in the management of women with HAIR-AN.

Learning points: HAIR-AN management is challenging and classic therapeutic regimens are ineffective. Literally HAIR-AN syndrome, the coexistence of Hirsutism, Insulin Resistance and Acanthosis Nigricans, represents an extreme case of polycystic ovary syndrome. In cases of HAIR-AN, liraglutide constitutes an effective and safe choice.

Keywords: 2020; Acanthosis nigricans; Acne; Adult; Amenorrhoea; Androgens; Androstenedione; Anovulation; Female; GLP1 agonists; Glucose tolerance (oral); Greece; Gynaecological endocrinology; Gynaecology; HAIR-AN syndrome*; HOMA; Hirsutism; Hyperandrogenism; Hyperinsulinaemia; Insulin; Insulin resistance; June; Liraglutide; Novel treatment; Ovaries; PCOS; Testosterone; White.