A Clinical Study of Childhood AlopeciaAreata in Singapore

Eileen Tan, M.D., Yong-Kwang Tay, M.D., and Yoke-Chin Giam, M.D.

National Skin Center, Singapore

Abstract: Alopecia areata (AA) is a common cause of nonscarringalopecia. The aim of this epidemiologic study is to review the clinical char-acteristics and treatment of childhood alopecia areata in a mixed ethnicpopulation. The study population consisted of a total of 392 children seenover a 4-year period with AA diagnosed before the age of 16 years. Thefemale:male ratio was 1:1.4. There were 309 Chinese (78.8%), 51 Malays(13.0%), and 32 Indians (8.2%). The mean age at the time of diagnosis was11.2 years. The majority of patients (71.7%) had alopecia of less than6-months duration and 6% had previous episodes of AA. Females appearedto have more severe involvement. A familial history of AA was observed in 33patients (8.4%). Associated atopy was found in 26.6% of patients and in32.3% of their first-degree relatives. Other associations such as vitiligo orDown syndrome were rare. For limited AA, topical and/or intralesionalcorticosteroid was the first-line treatment used and squaric acid dibutyl esterwas the choice of treatment for patients with extensive involvement. Theprofile of the poor respondents to therapy included young age of onset, pasthistory of AA, Down syndrome, and extensive involvement.

Alopecia areata (AA) is the most common cause oflocalized, nonscarring alopecia. This disease affects bothadults and children. There are limited data on childhoodAA. The objective of our study was to review the clinicalcharacteristics, disease associations, and treatment out-comes of young AA patients in Singapore.

MATERIALS AND METHODS

Case records of patients with AA that began before theage of 16 years were studied from 1996 to 2000 at theNational Skin Center, Singapore. The data concerninggender, ethnic group, age of onset, clinical pattern, extentof involvement, family history, disease associations, andtreatment given were recorded. The diagnosis of AAwas

made on clinical grounds. AA was classified into twogroups based on severity: limited disease (less than 50%involvement) and extensive disease (more than 50%involvement). Fisher’s exact probability test was used forstatistical evaluation. A significance level of p < 0.05was used.

RESULTS

There were a total of 392 patients with AA diagnosedbefore the age of 16 years. This represents 11.1% of thetotal number of AA cases seen during the same periodand 1.2% of the total number of cases seen in our out-patient clinic. There were 309 Chinese (78.8%), 51Malays (13.0%), and 32 Indians (8.2%). The

Address correspondence to Yong-Kwang Tay, M.D., Head,Dermatology Service, Dept. of Medicine, Changi General Hospital,2 Simei Street 3, Singapore 529889, or e-mail: yong_kwang_tay@cgh.com.sg

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female:male ratio was 1:1.4 (161 females and 231 males).The mean age of presentation was 11.2 years. Theyoungest patient was 2 years old. AA was present for ameandurationof 5.6months before the first consultation(range 1 week to 15 months). Two hundred eighty-onepatients (71.7%) had AA for less than 6 months beforetheir first visit to the doctor. Twenty-four patients (6.1%)had experienced previous episodes of AA. In this lattergroup, 15 (62.5%) had extensive involvement. Table 1summarizes the extent of alopecia with age and gender.

Three hundred ninety patients had scalp involvementwith or without other body site involvement. Mostpatients presented with patchy alopecia in oval, round,lancet, or reticular patterns (Fig. 1). Ophiasis wasreported in 9 patients (2.3%) and 5 had diffuse involve-ment. Twopatients had involvement of their eyebrows asthe sole clinical presentation. In 75 patients (19.1%),more than one body site, such as the eyebrows or beardarea, was concomitantly affected. Alopecia totalis/universalis was observed in 10 patients (2.6%). Nailchangeswere documented in 33 patients (8.4%). The nailchanges described were pitting, trachyonychia (Fig. 2),and longitudinal ridging.

One hundred four patients (26.6%) and familymembers of 130 patients (33.2%) were reported to have

an atopic history (namely atopic dermatitis, asthma, andallergic rhinitis). Atopy was present in 96 patients(29.7%) with limited AA compared to 25 patients(36.2%) with severe alopecia (p > 0.05). A 12-year-oldIndian boy had concomitant vitiligo and a 9-year-oldChinese girl had raised antimicrosomal antibody titersbut was clinically euthyroid. Three patients (0.8%) and65 patients (16.6%) had a first-degree relative with thy-roid disorder and diabetes mellitus, respectively. Fivepatients (1.3%)hadDownsyndrome.Of these, fourweremales. All presented with extensive AA. The familymembers of 33 patients (8.4%) were reported to have apast history of AA.

There are a variety of treatment modalities for AAincluding topical and intralesional corticosteroids, oralcorticosteroids, short contact dithranol, and immuno-therapy (squaric acid dibutyl ester). Evaluating treat-ment outcome can be difficult because AAmay undergospontaneous resolution. In our series, 58 patients werelost to follow-up after a few months.

Out of 392 patients, 323 had limited AA and 69 hadextensive involvement. For both limited and extensiveAA, topical therapy (topical corticosteroids such ashydrocortisone scalp lotion, 0.1% betamethasone val-erate scalp lotion, 0.025% flucinolone scalp gel) was thefirst-line treatment used. Two hundred eighty childrenwith limited AA received intralesional triamcinoloneacetonide. In 32 patients, intralesional triamcinoloneacetonide was stopped because of pain, lack of response,or progression of disease. Of the remaining 248 patients,160 (64.5%) and 211 (85.1%) experienced more than50% improvement after 12 and 24 weeks, respectively.Nine patients with extensive AA and onset less than ayear previously were started on oral prednisolone. Fivehadmore than 50% clinical improvement at the end of 6months. Prednisolone was started at a dose of 0.5 mg/kgand the dose was tapered over a period of 3–5 months.Three patients had progression of the disease while on

TABLE 1. The Extent of Alopecia Areata in Relation to Ageand Gender at the Time of Presentation

Age group(years)

Limited alopeciaareata (<50%)

Extensive alopeciaareata (>50%)

1–5Male 12 0Female 8 0

6–10Male 63 8Female 41 13

11–15Male 129 19Female 70 29

Figure 1. A 6-year-old boy with Down syndrome andextensive alopecia areata.

Figure 2. The patient from Figure 1 with trachyonychia.

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oral prednisolone and were switched to immunotherapyafter 4–6 weeks.

Forty-three young children (38 with limited AA and5 with extensive AA) were prescribed dithranol (0.5%or1%). Of these, only 10 with limited AA showed morethan 50% clinical improvement within 6 months. In therest, dithranol was discontinued after 4–6 monthsbecause of a lack of clinical improvement and side effectssuch as skin irritation.

Squaric acid dibutyl ester (SADBE) was the topicalimmunotherapy used in our center. The patient was firstsensitized with 2% SADBE in acetone applied to a 2 cmalopecicpatchonthescalp.Twoweeksaftersensitization,patientswerestartedonaweekly treatmentwithSADBE.For a majority of the cases, the starting dose of SADBEwas 0.0001%and itwas adjusted according to the clinicalresponse. Fifty-eight patients received SADBE, ofwhomfour had limited involvement (Table 2). Among 54patients with extensive AA, 40 (74.1%) achieved morethan 50% regrowth at 6 months. Side effects of SADBEinclude itchy dermatitis, blisters, mild cervical lympha-denopathy, and hyperpigmentation. The youngestpatient treated was 5 years old. The characteristics of the14 poor respondents were as follows: young age of onset,previous episodes of AA in the last 2 years, Downsyndrome, and alopecia totalis or universalis.

DISCUSSION

There is a paucity of data on childhood AA worldwide(1–3). Our study aims to review the epidemiologic profileof young AA patients in a mixed ethnic community inEast Asia. In our series, AA accounted for 11.1% of thetotal number of AA cases seen compared to 20–50% inprevious series (1–3).

In agreement with most studies (2,4,5), we observeda slight male preponderance. However, a higher pro-portion of females presented with more severe alopecia.This was in contrast to another study done in India (4)which showed a higher proportion of males with severeinvolvement.Most of the patients were Chinese, and thiswas consistent with the ethnic distribution in Singapore.The onset of AA was rare before the age of 6 years, andthis is consistent with findings in other studies (3,5).

The scalp was the predominant site of involvementand themost common clinical patternwasmultiple areasof patchy alopecia. Alopecia totalis or universalis wasreported in 10 patients, 9 of whom had a poor responseto treatment. Nail changes such as pitting, Beau lines,trachyonychia, and onychorrhexis occurred morefrequently in those with severe AA, similar to otherstudies (4,5). Overall, onychodystrophy was only recor-ded in 7.3% of patients, and this is likely an underesti-mation of the true frequency, as nail changes are usuallyasymptomatic.

Earlier studies have shown an increased incidence ofatopy among patients with alopecia areata (1,2). Thefrequency of concomitant atopy in our studywas 26.6%,which was similar to earlier reports in which it variesbetween 10 and50%(1,2,4,6–8). In accordancewith datafrom India (1,4), there was no significant correlationbetween atopy and the severity of disease, unlike in theWest (2,3).

There are few reports of an association between AAand autoimmune diseases such as vitiligo and thyroiddisorders (1,3,5–9). The incidence of AA with thyroiddisease and vitiligo varies between 1 and 20% (1–4,8,9).Both associations were rare in our cohort.

Diabetes mellitus is said to occur more frequently inrelatives of patients with AA rather than AA patientsthemselves (8). A family history of diabetes mellitusexisted in 16.6% of our study population. This figure istwo to three times higher than in the Western literature(3,8). In termsof a family historyofAA,our frequencyof8.4% was lower compared to previous reports of10–18% (1–3). Thismay possibly be due to differences ingenetic background.DuVivier andMunro (10) observedan increased incidence of AA in Down syndromepatients. In our series, the frequency of Down syndromein association with AA was 1.3% and this subgroupof patients presented with extensive involvement andhad poor response to treatment. Carter and Jegasothy(11) suggested that immunologic abnormalities inDown syndrome may contribute to the developmentof AA.

The treatment options in AA are varied. In children,the choice of therapy is determined by both the severityof disease and the tolerance of the child to treatment.Assessing treatment outcome is difficult as the diseasemay have spontaneous resolution. The majority of lim-ited AA cases have a good prognosis. Of 248 patientswho received intralesional triamcinolone acetonide,approximately two-thirds improved by more than 50%after 12 weeks. Fifty-four of 69 patients with extensiveAA received immunotherapy. Most of these children(74.1%) responded favorably to SADBE and achievedmore than 50% regrowth at the end of 6 months.

TABLE 2. SADBE Treatment in Relation to Age and Extentof Alopecia Areata

Age group(years)

Limited alopeciaareata (<50%)

Extensive alopeciaareata (>50%)

1–5 0 06–10 1 1411–15 3 40

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CONCLUSION

Certain distinctive characteristics seemed to occur in ourpatients with childhood AA. Childhood AA appears tobe less common in our mixed ethnic community. Therewas a male preponderance, although more females pre-sented with more extensive disease. A significant per-centage of our children had associated atopy, althoughthere was no significant association between atopy andextent of AA. The main mode of therapy for thetreatment of limited AA is topical and/or intralesionalcorticosteroids and immunotherapy for extensiveinvolvement. Poor respondents to therapy are those withearly onset disease, extensive involvement, Down syn-drome, and a past episode of AA.

REFERENCES

1. Sharma VK, Kumar B, Dawn G. A clinical study ofchildhood alopecia areata in Chandigarh, India. PediatrDermatol 1996;13:372–377.

2. Muller SA, Winkelmann RK. Alopecia areata: an evalu-ation of 736 patients. Arch Dermatol 1963;88:290–297.

3. DeWaard-VanDer Spek FB, Oranje AP, De RaeymaeckeDMJ, Wynia RP. Juvenile versus maturity onset alopeciaareata—a comparative retrospective clinical study. ClinExp Dermatol 1989;14:429–433.

4. SharmaVK,DawnG,KumarB. Profile of alopecia areatain northern India. Int J Dermatol 1996;35:22–27.

5. Madani S, Shapiro J. Alopecia areata update. J Am AcadDermatol 2000;42:549–566.

6. Penders AJM. Alopecia areata and atopy. Dermatologica1968;136:395–399.

7. Young E, Bruns HM, Berrens L. Alopecia areata andatopy. Dermatologica 1978;156:306–308.

8. SnellowWVR, Edwards JE,Koo JYM. Profile of alopeciaareata: a questionnaire analysis of patient and family. Int JDermatol 1992;31:186–189.

9. Cunliffe WJ, Hall R, Stevenson CJ, Weightman D.Alopecia areata, thyroid disease and autoimmunity. Br JDermatol 1969;81:877–881.

10. Du Vivier A, Munro DD. Alopecia areata, autoimmunityand Down’s syndrome. Br Med J 1975;1:191–192.

11. Carter DM, Jegasothy BV. Alopecia areata and Downsyndrome. Arch Dermatol 1976;112:1397–1399.

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