Loose anagen syndrome: A little response to minoxidil

Autosomal recessive hypotrichosis with loose anagen hairs associated with TKFC mutations*

“…4 Minoxidil 5% lotion has been suggested as a possible treatment for loose anagen hair but most reports are anecdotal and separating therapeutic response from natural resolution of the hair anchorage is difficult. 5,6 The aim of our study was to search for an underlying genetic defect in a patient presenting with hypotrichosis and loose anagen hair using next-generation sequencing and functional characterization of potentially pathogenic variants. We identified biallelic mutations in TKFC, whereas recently, different biallelic mutations in TKFC have also been reported to cause cataracts and multisystem disease without any hair abnormalities.…”

“…Most cases occur sporadically and are self‐limiting, but in other cases a genetic basis has been proposed, 2,3 and variants in the hair follicle keratin K6HF (now known as keratin 75 within the companion layer of the hair follicle) have only rarely been identified 4 . Minoxidil 5% lotion has been suggested as a possible treatment for loose anagen hair but most reports are anecdotal and separating therapeutic response from natural resolution of the hair anchorage is difficult 5,6 . The aim of our study was to search for an underlying genetic defect in a patient presenting with hypotrichosis and loose anagen hair using next‐generation sequencing and functional characterization of potentially pathogenic variants.…”

Low‐dose oral minoxidil improves global hair density and length in children with loose anagen hair syndrome

“…Premature and defective keratinization of the inner root sheath (IRS) and impaired anchorage of anagen hairs underlie the pathogenesis 1 . A favourable response was demonstrated in three patients with LAHS treated with topical minoxidil 1–3 . Low‐dose oral minoxidil (LDOM) has been shown to be effective in adult androgenetic alopecia, alopecia areata and telogen effluvium 4 .…”

“…1 A favourable response was demonstrated in three patients with LAHS treated with topical minoxidil. [1][2][3] Low-dose oral minoxidil (LDOM) has been shown to be effective in adult androgenetic alopecia, alopecia areata and telogen effluvium. 4 Herein, we describe the efficacy and safety of LDOM in LAHS.…”

Loose anagen syndrome in one identical twin girl

“…Loose anagen syndrome is attributed to an autosomal dominant or sporadic mutation in the gene encoding keratin 6 [2,3]. One mutation in K6HF, the companion layer keratin which represents the innermost layer of the outer root sheath, prematurely keratinizes the inner root sheath which impairs adhesion between the inner root sheath cuticle and the hair shaft cuticle [1,5,6]. An additional mutation in K6IRS, which is specific to the inner root sheath, impairs adhesion between the inner and outer root sheaths and prematurely terminates anagen [6].…”