Minoxidil 5% Solution for Topical Treatment

of Loose Anagen Hair Syndrome

Abstract: A 2-year-old girl with a diagnosis ofloose anagen hair syndrome was treated with atapering regime of minoxidil 5% solution over 28months, resulting in quick, significant clinicalimprovement with no adverse effects.

A2-year-old girl presented for excessive hair loss leadingto sparse hair since birth. The family historywas positivefor lichen planopilaris in the patient’s grandmother andalopecia areata in an aunt. Examination revealed ahealthy child with diffusely sparse, thin, lusterless, shortblondhairwithnoassociated scalpdermatitis (Fig. 1).Ahair pull test yielded more than 10 hairs withoutdiscomfort. Microscopic analysis demonstrated anagenhairs with misshapen hair bulbs and ruffled cuticles. Adiagnosis of loose anagen hair syndrome (LAHS) wasmade. Family history revealed no other cases.

Minoxidil 5% solution once daily was started. Sixmonths later, significant improvement in hair densityand a normal hair pull test were noted (Fig. 2A).Follow-up at 6, 12 (Fig. 2B), and 20 months showedcontinued improvement in hair density, and minoxidil5% solution was reduced to thrice, twice, and once aweek, respectively. Twenty-eight months after treat-ment initiation, her hair was visibly darker and denserand treatment was discontinued (Fig. 2C). The hair

pull test remained normal at all follow-up visits. Noadverse effects were noted.

DISCUSSION

LAHS is a sporadic or autosomal dominant hairdisorder that primarily affects children but occasion-ally manifests in adults (1). With an estimatedincidence of 2 to 2.25 cases per million per year,LAHS is more frequently reported in girls, althoughunderdiagnosis in boys is likely due to their conven-tionally shorter hairstyles (2). Patients are typicallygirls with short blond hair that is slow growing orunruly or sheds easily (3). Diagnosis depends on aconsistent clinical picture combined with a positivepull test with painless extraction of at least 10 looseanagen hairs or a trichogram showing more than 80%loose anagen hairs (1). The presence of loose anagenhairs per se is nondiagnostic (3).

Abnormal anagen hair anchorage underlies thepathogenesis, with premature and irregular keratini-zation of Henle and Huxley cells of the inner rootsheath (IRS) complex resulting in abnormal adhesionbetween the cuticle of the IRS and that of the hairshaft (4). This leads to poor support of the hair shaftand premature cessation of the anagen phase. Muta-tions in K6hf of the companion layer of the hairfollicle have been found in only some patients withLAHS, suggesting that mutations of other keratins ofthe IRS may play a role.

Although the clinical expression of LAHS improveswith age, with hair becoming longer, denser, anddarker by adolescence or adulthood, it has beensuggested that the phenomenon of loosely anchoredanagen hairs persists throughout the individual’slifetime (4). Most children are managed expectantly,but the use of topical minoxidil is promising inanecdotal reports and has been suggested as a first-line therapy for children with clinically severe LAHS(4,5).

Possible mechanisms of action of topical minoxidilon hair physiology include increased local cutaneousblood flow, prolongation of keratinocyte lifespan,enhanced cell proliferation, and DNA synthesis infollicular and perifollicular keratinocytes, with alter-ation of the hair growth pattern and phenotype or ashift from telogen to anagen (6).

Chong et al (7) followed up LAHS patients at 6-month intervals for up to 18 months and found thatthe results of the hair pull test fluctuated over this time,with no correlation to clinical severity. The hair pull testremained negative in our patient at up to 28 months offollow-up, suggesting that minoxidil’s effect on cell

Figure 1. Child with diffusely sparse, thin, short blondhair.

© 2014 Wiley Periodicals, Inc. 389

BRIEF REPORTS

Pediatric Dermatology Vol. 31 No. 3 389–409, 2014

proliferation and DNA synthesis could have positivelymodified the pathologic disordered keratinization of theIRS, leading to firmer anchorage of the hair. Similarresults have been noted in the treatment of short anagenhair syndrome (8). Further studies would clarifywhether improved anagen hair anchorage persists orfluctuations ensue after treatment cessation.

Reported adverse effects of topical minoxidil inadults include hypertrichosis, irritant and allergiccontact dermatitis, and worsening of seborrheicdermatitis (6). Georgala et al (9) reported threechildren (10–14 years old) with alopecia areata andalopecia totalis who experienced mild reversiblecardiovascular adverse effects (palpitations, dizziness,sinus tachycardia) between 20 days and 1 monthafter starting 2% minoxidil solution. Our experiencewith minoxidil 5% solution in treating children withvarious nonscarring hair conditions has beenfavorable, with no adverse effects noted. The treat-ment of LAHS described here is a short-term,tapering regime, thus we believe it to be safe inchildren.

In conclusion, this is the first description of atapering regime of topical minoxidil 5% solutionresulting in significant clinical improvement and noadverse effects in a patient with LAHS. We recom-mend this as a first-line treatment of LAHS, regardlessof severity, to hasten the resolution of clinical symp-toms in patients.

REFERENCES

1. Tosti A, Peluso A, Misciali C et al. Loose anagen hair.Arch Dermatol 1997;133:1089–1093.

2. Pham CM, Krejci-Manwaring J. Loose anagen hairsyndrome: an underdiagnosed condition in males. Pedi-atr Dermatol 2010;27:408–409.

3. Olsen EA, Bettencourt MS, Cote NL. The presence ofloose anagen hairs obtained by hair pull in the normal

population. J Investig Dermatol Symp Proc 1999;4:258–260.

4. Mirmirani P, Uno H, Price VH. Abnormal inner rootsheath of the hair follicle in the loose anagen hairsyndrome: an ultrastructural study. J Am Acad Derma-tol 2011;64:129–134.

5. Cantatore-Francis JL, Orlow SJ. Practical guidelines forevaluation of loose anagen hair syndrome. Arch Derma-tol 2009;145:1123–1128.

6. Rogers NE, Avram MR. Medical treatments for maleand female pattern hair loss. J Am Acad Dermatol2008;59:547–566; quiz 567–568.

7. Chong AH, Sinclair R. Loose anagen syndrome: aprospective study of three families. Australas J Dermatol2002;43:120–124.

8. Giacomini F, Starace M, Tosti A. Short anagensyndrome. Pediatr Dermatol 2011;28:133–134.

9. Georgala S, Befon A, Maniatopoulou E et al. Topicaluse of minoxidil in children and systemic side effects.Dermatology 2007;214:101–102.

Nisha S. Chandran, M.B.B.S., M.Med., M.R.C.P. (UK)*Arnold P. Oranje, M.D., Ph.D.†*Division of Dermatology, University Medicine Cluster,National University Hospital, Singapore†Department of Dermatology, Maasstad Ziekenhuis,Rotterdam, The Netherlands

Address correspondence to Nisha Suyien Chandran, M.B.B.S.,M.Med., M.R.C.P. (UK), Division of Dermatology, UniversityMedicine Cluster, National University Hospital, 5 Lower KentRidge Road, Singapore 119074, or e-mail: nishasuch@gmail.com.

PHACE Syndrome: A Retrospective Review

of 23 Patients

Abstract: We present 23 patients with PHACEsyndrome showing similarities in our population withdata that already exist while highlighting neurodeve-lopmental occurrences arising in a subset of thesepatients.

A B C

Figure 2. Child at (A) 6, (B) 12, and (C) 28 months after initiation of treatment with minoxidil 5% solution.

390 Pediatric Dermatology Vol. 31 No. 3 May/June 2014