Pediatric Dermatology Vol. 31 No. 3 309–318, 2014
Quality of Life Impairment in Children and Adolescents with Vitiligo Jonathan I. Silverberg, M.D., Ph.D., M.P.H., and Nanette B. Silverberg, M.D. Department of Dermatology, St. Luke’s-Roosevelt Hospital and Beth Israel Medical Centers, New York, New York
Abstract: Vitiligo significantly affects quality of life (QOL) in adults, but little is known about the effect on QOL of pediatric vitiligo and whether the extent, distribution, and duration of vitiligo are associated with QOL. We performed an online parental questionnaire-based study (N = 350) regarding children ages 0 to 17 years with vitiligo, including validated questions about body surface area (BSA), distribution, and age of onset of vitiligo, associated symptoms, and QOL using the Children’s Dermatology Life Quality Index (CDLQI). Vitiligo negatively affected numerous aspects of and total CDLQI score (median 3.0, interquartile range 5.0). Their vitiligo lesions did not bother only 4.1% of teenagers ages 15 to 17 years, versus 45.6% of children ages 0 to 6 years and 50.0% of those ages 7 to 14 years (p < 0.001). There was no association between the child’s age and whether the child’s vitiligo bothered the parents (p = 0.27). The most bothersome sites of vitiligo lesions for children and parents were the face (25.6% and 37.4%, respectively) and legs (26.2% and 26.2%, respectively). Eighty-two patients (30.1%) reported itching and painful skin within the past week. Using multivariate ordinal logistic regression models, it was found that an affected BSA of more than 25% was associated with self-consciousness, difficulty with friendships and schoolwork, and teasing and bullying. Lesions on the face and arms were associated with teasing and bullying. The extent of vitiligo is associated with QOL impairment in children and adolescents, especially self-consciousness, but also bullying and teasing. Different distributions of vitiligo lesions are associated with different aspects of QOL impairment. Teenagers ages 15 to 17 years seem to experience the most self-consciousness of all pediatric age groups.
Although vitiligo has been reported to cause significant impairment of quality of life (QOL) (1–4), specific areas of QOL impairment (e.g., functional limitations and impaired activities of daily living) have not been well characterized in large
studies. A few studies have supported the theory that skin disease in general and vitiligo in particular create an emotional burden and interference with psychosocial development. Weber et al demonstrated higher Children’s Dermatology Life Quality Index (CDLQI)
Address correspondence to Nanette B. Silverberg, M.D., Department of Dermatology Suite, St. Luke’s-Roosevelt Hospital and Beth Israel Medical Centers, 11D 1090 Amsterdam Ave. New York, NY 10025, or e-mail: [email protected]. DOI: 10.1111/pde.12226
© 2013 Wiley Periodicals, Inc.
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310 Pediatric Dermatology Vol. 31 No. 3 May/June 2014
scores for chronic skin disease (5). One study found that QOL is poorer in children with vitiligo than in those with atopic dermatitis (6). Other authors have suggested that vitiligo correlates with childhood depression, especially in the preteen years, and that disease in teenagers, especially in visible areas, may affect emotional development (7). We hypothesized further that a greater extent of vitiligo would be associated with worse QOL and that different aspects of QOL would be affected at different ages and with different distributions of vitiligo lesions. We also hypothesized that different aspects of the illness would bother parents than those that bothered their children. We conducted an online survey of parents of children with vitiligo to identify leading factors in the development of emotional disturbance for children with vitiligo and their parents.
METHODS Study Population and Questionnaire Distribution The Institutional Review Board at St. Luke’s-Roosevelt Hospital Center (www.clinicaltrials.gov registration NCT01401374) approved this parental questionnaire– based study of children with vitiligo younger than 18 years of age. Our previously validated survey showed self-reports of physician-diagnosed vitiligo, body surface area (BSA) affected by vitiligo, number of affected body parts, and disease duration were all strongly correlated with clinical assessment by a dermatologist (8). Exclusion criteria were not being diagnosed with vitiligo by a physician, vitiligo onset reported at birth (to exclude possible cases of nevus depigmentosus or pigmentary mosaicism), and incomplete survey responses. The questionnaire was uploaded to http://www. surveymonkey.com and parents of children enrolled in nonprofit support groups for vitiligo vulgaris were invited to participate by responding to the survey. Responses from initiation of the study in June 2010 through October 2012 were reviewed. The questionnaire took an average of 18 minutes (range 11–29 minutes) to complete. Data were deidentified and confidential and posed no risk to participants; the Institutional Review Board granted a waiver of informed consent.
of vitiligo, and the 11-question CDLQI (9). There were 78 questions overall (56 closed and 22 open questions). The questions related to this study; response rates are listed in supplemental Table S1. Responses were verified by screening for noninteger or implausible values (e.g., age 100). Data and Statistical Analysis Data were processed and statistics were analyzed using SAS version 9.2 (SAS Institute, Inc., Cary, NC). The duration of vitiligo and number of body parts affected were divided into tertiles for statistical analysis because of wide skewing. Analyses were performed for all children ages 0 to 17 years and specifically for those ages 4 to 16 years because the CDLQI was initially designed for the latter age group (9). Total CDLQI scores were categorized as no or small (0–5), moderate (5–9), and very or extremely large (>10) QOL effect [as previously reported (10)], owing to a highly skewed distribution of results. Ordinal logistic regression models were constructed to examine the effect of vitiligo extent on QOL, with categorized CDLQI and individual aspects of the CDLQI divided into four responses (not at all, only a little, quite a lot, very much) as the dependent variables. The independent variables were body surface area (BSA), duration of vitiligo, and number of body parts affected by vitiligo (tertiles). Adjusted odds ratios (aORs) were calculated by including sex and current age (continuous) in the full models. Linear interaction terms were tested and were included in the final models if significant (p < 0.05). Binary logistic regression models were constructed to determine whether the site of vitiligo lesions (independent variable) was a predictor of impaired aspects of QOL on the CDLQI (dependent variable). Multivariate regression analyses were used for all analyses in the study to address potential confounding from other variables, such as age and sex. Missing values were encountered from nonresponse of subjects to various questions (Table S1). Complete case analysis was performed (i.e., missing values were ignored). Correction for multiple dependent tests (k = 61) according to the approach of Benjamini and Hochberg (11) yielded a critical p-value of 0.04.
Questionnaire Questionnaire items were developed to determine novel risk and prognostic factors for vitiligo. The aim of the questionnaire was to identify QOL impairment with vitiligo. The questionnaire consisted of questions pertaining to demographic characteristics, phenotype
RESULTS Survey Population Characteristics Three hundred fifty-one parents started the survey and 350 completed it (99.7% completion rate); 342
Silverberg and Silverberg: Childhood Vitiligo and Quality of Life
reported that a physician had diagnosed their child with vitiligo. Six patients were excluded for being older than 17 years of age; 87.2% of the children were ages 4 to 16 years. Sex, age, duration of disease, BSA, face or body involvement, and presence of bilateral lesions are listed in Table 1. Most Bothersome Sites for Vitiligo Their vitiligo reportedly did not bother 23% of the children (Table 2). Upon age stratification, their vitiligo lesions did not bother 45.6% of children ages 0 to 6 years, 50.0% of those ages 7 to 14 years and 4.1% of adolescents aged 15 to 17 years (p < 0.001). In contrast, there was no significant association between the child’s age and whether their child’s vitiligo bothered the parents (p = 0.27). In subjects whose vitiligo reportedly bothered them, the most bothersome sites for children and parents were the face (25.6% and 37.4%, respectively) and legs (26.2% and 26.2%, respectively). There were no significant associations between a child’s age and which affected sites were most bothersome (p ≥ 0.03). Eighty-four percent of respondents who reported that the most bothersome site was somewhere other than the face attributed this to difficulty covering the site with clothing or makeup.
Sex, n (%) Male Female Age, mean SD Age, years, n (%) 0–3 4–16 17 Duration of vitiligo, years, mean SD Age at vitiligo onset, years, mean SD Body surface area affected,%, n (%)* 1–25 26–50 51–75 76–100 Number of body parts affected, mean SD Distribution, n (%) Face or body Face and body Laterality, n (%) Unilateral or at midline Bilateral
Effect of Vitiligo on CDLQI Scores The median total CDLQI score was 3.0 [interquartile range (IQR) 5.0] in all subjects with vitiligo and 2.5 (IQR 4.0) in children ages 4 to 16 years. CDLQI scores were significantly higher in teenagers ages 15 to 17 years (mean 10.0, IQR 14.0) than in those ages 0 to 6 years (mean 2.0, IQR 3.5) and 7 to 14 years (mean 3.0, IQR 4.0) (Kruskal–Wallis test, p = 0.0004). There was no significant difference in CDLQI scores between boys (mean 3.0, IQR 6.0) and girls (mean 2.0, IQR 4.0, p = 0.83). The total CDLQI score was categorized into no to small (0–5), moderate (5–9), and extremely large (>10) effect and modeled using ordinal logistic regression. Adolescents who developed vitiligo at age 10 years and older had significantly higher CDLQI scores than those who developed it between the ages of 0 and 9 years (odds ratio [OR] = 3.02, 95% confidence interval (CI) 1.09, 8.78, p = 0.03), although the CDLQI score was not associated with BSA, number of body parts affected, bilaterality, or duration of vitiligo lesions (Table 3). Effect of Vitiligo Extent and Duration on Individual Aspects of Lifestyle and QOL
128 (40.0) 192 (60.0)
Vitiligo negatively affected all aspects of the CDLQI (Table 4); 82 subjects (30.1%) reported itching, soreness, or painful skin within the past week. An affected BSA greater than 25% was associated with selfconsciousness, difficulty with friendships and schoolwork, and teasing and bullying (Table 5). In subset analysis of children ages 10 to 17 years, greater affected BSA was also associated with QOL impairment related to clothing decisions, playing, and hobbies (p < 0.03) (data not shown). In subset analysis of children ages 4 to 16 years, there was significantly greater self-consciousness (adjusted OR [aOR] = 2.89, 95% CI 1.30, 6.46, p = 0.009) and sleep impairment associated with a greater BSA (aOR = 9.79, 95% CI 1.75, 54.89, p = 0.009). Similarly, the highest tertile for the number of body parts affected by vitiligo lesions was associated with self-consciousness (aOR = 1.97, 95% CI 1.11, 3.51, p = 0.02) and teasing and bullying (aOR = 2.02, 95% CI 1.17, 4.22, p = 0.01). Individual aspects of the CDLQI were not associated with bilaterality of lesions (p ≥ 0.08) (data not shown).
64 (19.1) 271 (80.9)
Effect of Vitiligo Distribution on QOL
TABLE 1. Phenotype of Disease in Children and Adolescents with Physician Diagnosed Vitiligo (N = 336) Variable
311
Value 146 (43.7) 188 (56.3) 10.4 4.1 22 (6.5) 293 (87.2) 21 (6.3) 4.7 3.6 5.7 3.3 237 (71.0) 57 (17.1) 30 (9.0) 10 (3.0) 8.9 5.7
*Rows do not sum to 336 because of missing values attributed to lack of response to a particular question. Percentages of completed responses per question are reported. SD, standard deviation.
The distribution of vitiligo lesions was associated with impairment of different aspects of the CDLQI. Lesions on the hands, arms, legs, and abdomen were associated
312 Pediatric Dermatology Vol. 31 No. 3 May/June 2014
TABLE 2. Distribution of Most Bothersome Sites of Vitiligo Lesions for Children and Parents Site of vitiligo lesions
Most bothersome
All (0–17) years (n = 336)
0–6 years (n = 54)
7–14 years (n = 184)
15–17 years (n = 36)
p*
(1.9) (25.2) (1.9) (2.6) (0.7) (0.7) (3.3) (4.4) (28.2) (5.2) (22.6)
0 19 2 1 0 1 1 2 14 0 34
(0.0) (23.2) (33.3) (12.5) (0.0) (50.0) (7.7) (12.5) (16.7) (0.0) (45.6)
4 (66.7) 48 (58.5) 3 (50.0) 6 (75.0) 2 (50.0) 1 (50.0) 7 (53.9) 8 (50.0) 58 (69.1) 13 (76.5) 37 (50.0)
2 (33.3) 15 (18.3) 1 (16.7) 1 (12.5) 2 (50.0) 0 (0.0) 5 (38.5) 6 (37.5) 12 (14.3) 4 (23.5) 3 (4.1)
0.28† 0.77 0.84† 0.88† 0.16† 0.63† 0.06† 0.06† 0.18 0.03† 25 42 (79.3) Number of affected body parts 1–4 54 (90.0) (reference) 5–10 48 (87.3) 11–22 41 (78.9) Laterality Unilateral or at 27 (93.1) the midline (reference) Bilateral 124 (85.5) Duration of vitiligo, years 0.1–2.17 47 (87.0) (reference) 2.3–6.0 55 (91.7) 6.0–15.1 46 (79.3) Age of vitiligo onset, years 25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25
Not at all
Only a little
Quite a lot
Very much
Q1. Scratchy, sore, or painful skin 131 (69.7) 46 (24.5) 9 (4.8) 2 (1.1) 59 (70.2) 18 (21.4) 6 (7.1) 1 (1.2) Q2. Self-conscious about lesions 87 (46.5) 63 (33.7) 23 (12.3) 14 (7.5) 29 (34.5) 31 (36.9) 13 (15.5) 11 (13.1) Q3. Friendships 153 (82.7) 26 (14.1) 5 (2.7) 1 (0.5) 59 (71.1) 15 (18.1) 5 (6.0) 4 (4.8) Q4. Clothing decisions 125 (67.6) 32 (17.3) 17 (9.2) 11 (6.0) 48 (57.8) 16 (19.3) 5 (6.0) 14 (16.9) Q5. Going out, playing, or hobbies 133 (72.3) 32 (17.4) 13 (7.1) 6 (3.3) 56 (66.7) 18 (21.4) 3 (3.6) 7 (8.3) Q6. Swimming or other sports 144 (77.8) 23 (12.4) 5 (2.7) 13 (7.0) 62 (73.8) 9 (10.7) 5 (6.0) 8 (9.5) Q7–1. Schoolwork in past week 131 (87.3) 15 (10.0) 3 (2.0) 1 (0.7) 51 (71.8) 11 (15.5) 6 (8.5) 3 (4.2) Q7–2. Holiday in past week 126 (79.3) 21 (13.2) 9 (5.7) 3 (1.9) 59 (84.3) 6 (8.6) 2 (2.9) 3 (4.3) Q8. Teasing/bullying 124 (67.4) 49 (26.6) 11 (6.0) 0 (0.0) 46 (55.4) 20 (24.1) 11 (13.3) 6 (7.3) Q9. Sleep 159 (86.4) 19 (10.3) 4 (2.2) 2 (1.1) 68 (81.0) 10 (11.9) 5 (6.0) 1 (1.2) Q10. Affect of treatment on quality of life 121 (66.5) 44 (24.2) 12 (6.6) 5 (2.8) 52 (65.8) 16 (20.3) 9 (11.4) 2 (2.5)
OR (95% CI)
p
aOR (95% CI)
p
aOR (95% CI)
p
1.00 1.01 (0.58, 1.76)
— 0.97
1.00 0.98 (0.56, 1.72)
— 0.94
1.00 1.57 (0.61, 4.10)
— 0.35
1.00 1.66 (1.03, 2.67)
— 0.03
1.00 1.67 (1.03, 2.69)
— 0.04
1.00 2.89 (1.30, 6.46)
— 0.009
1.00 2.06 (1.13, 3.76)
— 0.02
1.00 1.90 (1.02, 3.54)
— 0.04
1.00 2.11 (0.73, 6.11)
— 0.17
1.00 1.64 (0.98, 2.73)
— 0.06
1.00 1.58 (0.94, 2.67)
— 0.08
1.00 1.95 (0.86, 4.45)
— 0.11
1.00 1.31 (0.76, 2.27)
— 0.33
1.00 1.22 (0.70, 2.13)
— 0.48
1.00 1.53 (0.63, 3.75)
— 0.35
1.00 1.29 (0.72, 2.33)
— 0.39
1.00 1.26 (0.69, 2.30)
— 0.44
1.00 2.23 (0.80, 6.17)
— 0.12
1.00 2.85 (1.42, 5.75)
— 0.003
1.00 2.52 (1.23, 5.16)
— 0.01
1.00 2.32 (0.73, 7.43)
— 0.15
1.00 0.74 (0.35, 1.55)
— 0.42
1.00 0.69 (0.32, 1.46)
— 0.33
1.00 0.93 (0.24, 3.55)
— 0.91
1.00 1.99 (1.19, 3.32)
— 0.009
1.00 2.04 (1.21, 3.44)
— 0.008
1.00 2.70 (1.11, 6.58)
— .02
1.00 1.53 (0.77, 3.03)
— 0.22
1.00 1.50 (0.75, 3.00)
— 0.26
1.00 9.79 (1.75, 54.89)
— 0.009
1.00 1.09 (0.63, 1.88)
— 0.76
1.00 1.12 (0.65, 1.94)
— 0.68
1.00 1.67 (0.65, 4.25)
— 0.28
Ordinal logistic regression models were constructed with effect of vitiligo divided into four responses (not at all, only a little, quite a lot, very much) as the dependent (outcome) variable. The independent (explanatory) variable was percentage of BSA affected by vitiligo lesions. The ORs and 95% CIs were determined across all levels of the dependent variable simultaneously rather than at an arbitrarily set cut-point and analyzed using binary logistic regression. The aORs were determined by including current age and sex. N = 221–272 subjects with complete information. Subset analyses were also performed for children ages 4–16 years.
suggesting that there may be more of an emotional impairment than a functional impairment of QOL in children. Specific sites affected with vitiligo may play a greater role in children rather than the overall extent of vitiligo lesions. That is, clothing or make-up may hide some sites, limiting functional impairment, but self-awareness of the disease and fear of being ostracized by family and peers might still cause an emotional burden. There was a significant difference between the sites of vitiligo that patients and parents found bothersome. This was particularly true in younger children. That is, younger children may not be old enough to have a sense of self-consciousness or to understand their cosmetic deformity.
Children are also more likely to face teasing and bullying and have functional impairment when they attend school. One might expect that the face would be the most bothersome site of vitiligo lesions in the vast majority of patients. A previous study of 41 children ages 8 to 18 years with vitiligo showed that vitiligo severity correlated negatively with psychosocial scores for adolescents and that involvement of specific areas, including the face in boys and genitalia in girls, was associated with greater QOL impairment (7). Another study of Korean adolescents found that facial lesions were most likely to cause QOL impairment (17). In the present study, lesions other than on the face bothered many patients because some felt that lesions on the face were more amenable to
Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present
44 (50.6) 72 (39.1) 67 (51.2) 49 (35.0) 71 (46.7) 38 (35.5) 69 (49.6) 40 (33.3) 60 (42.9) 49 (41.2) 46 (53.5) 70 (37.8) 36 (57.1) 80 (38.5) 49 (50.0) 67 (38.7) 70 (44.0) 39 (39.0)
Not at all 26 (29.9) 68 (37.0) 37 (28.2) 57 (40.7) 51 (33.6) 42 (39.3) 47 (33.8) 46 (38.3) 51 (36.4) 42 (35.3) 23 (26.7) 71 (38.4) 15 (23.8) 79 (38.0) 26 (26.5) 68 (39.3) 57 (35.9) 36 (36.0)
Only a little 11 (12.6) 25 (13.6) 14 (10.7) 22 (15.7) 18 (11.8) 15 (14.0) 14 (10.1) 19 (15.8) 16 (11.4) 17 (14.3) 9 (10.5) 27 (14.6) 7 (11.1) 29 (13.9) 12 (12.2) 24 (13.9) 18 (11.3) 15 (15.0)
Quite a lot 6 (6.9) 19 (10.3) 13 (9.9) 12 (8.6) 12 (7.9) 12 (11.2) 9 (6.5) 15 (12.5) 13 (9.3) 11 (9.2) 8 (9.3) 17 (9.2) 5 (7.9) 20 (9.6) 11 (11.2) 14 (8.1) 14 (8.8) 10 (10.0)
Very much 1.00 1.51 (0.93, 1.00 1.62 (1.04, 1.00 1.51 (0.96, 1.00 1.98 (1.25, 1.00 1.10 (0.70, 1.00 1.65 (1.02, 1.00 1.87 (1.09, 1.00 1.30 (0.82, 1.00 1.25 (0.79,
OR (95% CI)
1.99)
2.06)
3.21)
2.68)
1.72)
3.14)
2.40)
2.53)
2.43)
aOR (95% CI) 1.00 1.32 (0.81, 1.00 1.38 (0.87, 1.00 1.57 (0.99, 1.00 2.02 (1.27, 1.00 1.15 (0.73, 1.00 1.38 (0.84, 1.00 1.57 (0.89, 1.00 1.17 (0.73, 1.00 1.31 (0.82,
p — 0.09 — 0.03 — 0.34 — 0.003 — 0.69 — 0.04 — 0.02 — 0.27 — 0.34 2.10)
1.88)
2.75)
2.27)
1.82)
3.22)
2.50)
2.18)
2.16)
— 0.26 — 0.18 — 0.06 — 0.003 — 0.54 — 0.21 — 0.12 — 0.52 — 0.25
p
1.00 1.48 (0.87, 2.50) 1.00 1.39 (0.85, 2.29) 1.00 1.35 (0.82, 2.24) 1.00 1.85 (1.12, 3.07) 1.00 0.92 (0.56, 1.52) 1.00 1.20 (0.70, 2.07) 1.00 1.18 (0.65, 2.14) 1.00 1.17 (0.70, 1.97) 1.00 1.04 (0.62, 1.74)
aOR (95% CI)
Ages 4–16 years
— 0.15 — 0.19 — 0.24 — 0.01 — 0.76 — 0.50 — 0.60 — 0.56 — 0.88
p
Ordinal logistic regression models were constructed with self-consciousness divided into four responses (not at all, only a little, quite a lot, very much) as the dependent (outcome) variable. The independent (explanatory) variable was the percentage of BSA affected by vitiligo lesions. The ORs and 95% CIs were determined across all levels of the dependent variable simultaneously rather than at an arbitrarily set cut-point and analyzed using binary logistic regression. The aORs were determined by including current age and sex. N = 221–272 subjects with complete information. Subset analyses were also performed for children ages 4–16 years.
Genitals
Feet
Legs
Arms
Back
Abdomen
Chest
Hands
Face
Site
Self-consciousness, n (%)
Ages 0–17 years
TABLE 6. Ordinal Logistic Regression Analysis of Effect of Site of Vitiligo Lesions on Self-Consciousness
Silverberg and Silverberg: Childhood Vitiligo and Quality of Life 315
Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present
69 (79.3) 101 (56.1) 89 (69.5) 81 (58.3) 100 (67.1) 61 (57.6) 89 (65.4) 72 (60.5) 93 (67.4) 68 (58.1) 64 (75.3) 106 (58.2) 40 (66.7) 130 (62.8) 63 (65.6) 107 (62.6) 100 (64.1) 61 (61.6)
Not at all 15 (17.2) 54 (30.0) 30 (23.4) 39 (28.1) 35 (23.5) 32 (30.2) 37 (27.2) 30 (25.2) 34 (24.6) 33 (28.2) 14 (16.5) 55 (30.2) 15 (25.0) 54 (26.1) 25 (26.0) 44 (25.7) 44 (28.2) 23 (23.2)
Only a little 2 (2.3) 20 (11.1) 8 (6.3) 14 (10.1) 13 (8.7) 9 (8.5) 10 (7.4) 12 (10.1) 10 (7.3) 12 (10.3) 6 (7.1) 16 (8.8) 4 (6.7) 18 (8.7) 7 (7.3) 15 (8.8) 11 (7.1) 11 (11.1)
Quite a lot 1 (1.2) 5 (2.8) 1 (0.8) 5 (3.6) 1 (0.7) 4 (3.8) 0 (0.0) 5 (4.2) 1 (0.7) 4 (3.4) 1 (1.2) 5 (2.8) 1 (1.7) 5 (2.4) 1 (1.0) 5 (2.9) 1 (0.6) 4 (4.0)
Very much 1.00 3.08 (1.70, 1.00 1.70 (1.03, 1.00 1.50 (0.91, 1.00 1.35 (0.82, 1.00 1.55 (0.94, 1.00 2.10 (1.19, 1.00 1.21 (0.67, 1.00 1.18 (0.71, 1.00 1.24 (0.75,
OR (95% CI)
2.06)
1.98)
2.19)
3.69)
2.55)
2.22)
2.48)
2.79)
5.57)
aOR (95% CI) 1.00 3.00 (1.62, 5.56) 1.00 1.54 (0.92, 2.58) 1.00 1.56 (0.93, 2.60) 1.00 1.40 (0.84, 2.33) 1.00 1.65 (0.99, 2.75) 1.00 1.93 (1.07, 3.46) 1.00 1.05 (0.56, 1.96) 1.00 1.13 (0.67, 1.92) 1.00 1.30 (0.78, 2.18)
p — 0.0002 — 0.04 — 0.12 — 0.24 — 0.09 — 0.01 — 0.54 — 0.52 — 0.41
— 0.0005 — 0.09 — 0.09 — 0.19 — 0.05 — 0.03 — 0.88 — 0.65 — 0.32
p
1.00 2.68 (1.40, 5.12) 1.00 1.26 (0.72, 2.20) 1.00 1.21 (0.69, 2.12) 1.00 1.17 (0.67, 2.05) 1.00 1.21 (0.69, 2.12) 1.00 1.61 (0.86, 3.01) 1.00 0.95 (0.49, 1.86) 1.00 1.05 (0.58, 1.88) 1.00 1.17 (0.66, 2.08)
aOR (95% CI)
Ages 4–16 years
— 0.001 — 0.42 — 0.51 — 0.58 — 0.50 — 0.14 — 0.89 — 0.88 — 0.59
p
Ordinal logistic regression models were constructed with teasing and bullying divided into four responses (not at all, only a little, quite a lot, very much) as the dependent (outcome) variable. The independent (explanatory) variable was the percentage of BSA affected by vitiligo lesions. The ORs and 95% CIs were determined across all levels of the dependent variable simultaneously rather than at an arbitrarily set cut-point and analyzed using binary logistic regression. The aORs were determined by including current age and sex. N = 221–272 subjects with complete information. Subset analyses were also performed for children ages 4–16 years.
Genitals
Feet
Legs
Arms
Back
Abdomen
Chest
Hands
Face
Site
Teasing and bullying, n (%)
Ages 0–17 years
TABLE 7. Ordinal Logistic Regression Analysis of Impact of Site of Vitiligo Lesions on Teasing or Bullying
316 Pediatric Dermatology Vol. 31 No. 3 May/June 2014
Silverberg and Silverberg: Childhood Vitiligo and Quality of Life
cover-up with make-up than lesions on the extremities. This highlights that lesions on the trunk and extremities are also a significant cause of morbidity in childhood vitiligo. Vitiligo had a very large effect on QOL in 13% of patients in this study (CDLQI > 10), which highlights the importance of screening for emotional and functional impairment in the clinical assessment of childhood vitiligo. Children and adolescents with emotional impairment from vitiligo may be at risk for long-term psychological and developmental problems. Such patients may benefit from referrals for psychological intervention and systemic therapeutic interventions, including photo- and oral therapy. A previous study of 51 children with generalized vitiligo found that narrowband ultraviolet B therapy resulted in considerable repigmentation and improved CDLQI scores (18). Further studies are necessary to identify other therapeutic approaches that can improve QOL with vitiligo in adults and children. Itching and burning skin occurs in one-third of patients. We previously reported that 35.5% of adults with vitiligo had itching and burning of their skin and discussed potential mechanisms for such skin symptoms, including higher-grade inflammation, heightened sensitivity, Koebnerization of lesions due to manipulation, and symptoms secondary to comorbid autoimmune disease or skin pathology (8). Consideration of itching and burning skin is warranted in the assessment of children with vitiligo. This study has several strengths, including being prospective with a validated questionnaire for QOL (CDLQI) (9), although it has potential limitations. Subjects were recruited from vitiligo support groups and may have more psychosocial disturbance than patients who do not go online regarding their disease. Nevertheless, the study included a large number of subjects with limited vitiligo extent and mild to no reported QOL impairment. Given our hypothesis of age-related QOL changes, we included all children ages 0 to 17 years, which extends beyond the initial CDLQI design for children ages 4 to 16 years (9). To address this we performed subset analyses in children ages 4 to 16 years and achieved largely similar results. Measures of vitiligo extent are self-reported and are not assessed clinically, but our previously validated questionnaire showed that self-reports of vitiligo diagnosis and extent are accurate (8). Parents completed surveys, which may have resulted in discordance with the child’s viewpoint and overreporting of QOL impairment, although parents were apparently aware of this, as indicated by their responses about
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the most bothersome site of lesions for them versus their child. Finally, Fitzpatrick skin type was not assessed. In conclusion, vitiligo is not just a cosmetic disorder, it causes emotional and functional alterations in childhood. Greater vitiligo extent is associated with greater impairment in QOL, including skin symptoms, self-consciousness, teasing and bullying, and functional impairment. Teenagers ages 15 to 17 years appear to have the greatest QOL burden from vitiligo. Screening for QOL impairment should be included in the evaluation of childhood vitiligo and incorporated into therapeutic decision making. Further studies are needed to determine the emotional benefit of treatment of childhood vitiligo. SUPPORTING INFORMATION
Additional supporting information may be found in the online version of this article: Table S1. Questions used in the study. REFERENCES 1. Mechri A, Amri M, Douarika AA et al. Psychiatric morbidity and quality of life in vitiligo: a case controlled study. Tunis Med 2006;84:632–635. 2. Ongenae K, Dierckxsens L, Brochez L et al. Quality of life and stigmatization profile in a cohort of vitiligo patients and effect of the use of camouflage. Dermatology 2005;210:279–285. 3. Ongenae K, Van Geel N, De Schepper S et al. Effect of vitiligo on self-reported health-related quality of life. Br J Dermatol 2005;152:1165–1172. 4. Prcic S, Durovic D, Duran V et al. Some psychological characteristics of children and adolescents with vitiligo— our results. Med Pregl 2006;59:265–269. 5. Weber MB, Lorenzini D, Reinehr CP et al. Assessment of the quality of life of pediatric patients at a center of excellence in dermatology in southern Brazil. An Bras Dermatol 2012;87:697–702. 6. Dertlioglu SB, Cicek D, Balci DD et al. Dermatology life quality index scores in children with vitiligo: comparison with atopic dermatitis and healthy control subjects. Int J Dermatol 2013;52:96–101. 7. Bilgic O, Bilgic A, Akis HK et al. Depression, anxiety and health-related quality of life in children and adolescents with vitiligo. Clin Exp Dermatol 2011;36:360–365. 8. Silverberg JI, Silverberg NB. Association between vitiligo extent and distribution and quality-of-life impairment. JAMA Dermatol 2013;149:159–164. 9. Lewis-Jones MS, Finlay AY. The Children’s Dermatology Life Quality Index (CDLQI): initial validation and practical use. Br J Dermatol 1995;132:942–949. 10. Hongbo Y, Thomas CL, Harrison MA et al. Translating the science of quality of life into practice: What do
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11.
12. 13. 14.
dermatology life quality index scores mean? J Invest Dermatol 2005;125:659–664. Benjamini Y, Hochberg Y. Controlling the false discovery rate—a practical and powerful approach to multiple testing. J R Stat Soc Series B Stat Methodol 1995;57:289–300. Eagle M. Contributions of Erik Erikson. Psychoanal Rev 1997;84:337–347. Finlay AY, Basra MK. DLQI and CDLQI scores should not be combined. Br J Dermatol 2012;167: 453–454. Oostveen AM, de Jager ME, van de Kerkhof PC et al. The influence of treatments in daily clinical practice on the Children’s Dermatology Life Quality Index in juvenile psoriasis: a longitudinal study from the ChildCAPTURE patient registry. Br J Dermatol 2012;167: 145–149.
15. de Jager ME, van de Kerkhof PC, de Jong EM et al. A cross-sectional study using the Children’s Dermatology Life Quality Index (CDLQI) in childhood psoriasis: negative effect on quality of life and moderate correlation of CDLQI with severity scores. Br J Dermatol 2010;163:1099–1101. 16. Maksimovic N, Jankovic S, Marinkovic J et al. Healthrelated quality of life in patients with atopic dermatitis. J Dermatol 2012;39:42–47. 17. Choi S, Kim DY, Whang SH et al. Quality of life and psychological adaptation of Korean adolescents with vitiligo. J Eur Acad Dermatol Venereol 2010;24: 524–529. 18. Njoo MD, Bos JD, Westerhof W. Treatment of generalized vitiligo in children with narrow-band (TL01) UVB radiation therapy. J Am Acad Dermatol 2000;42:245–253.
Quality of Life Impairment in Children and Adolescents with Vitiligo Jonathan I. Silverberg, M.D., Ph.D., M.P.H., and Nanette B. Silverberg, M.D. Department of Dermatology, St. Luke’s-Roosevelt Hospital and Beth Israel Medical Centers, New York, New York
Abstract: Vitiligo significantly affects quality of life (QOL) in adults, but little is known about the effect on QOL of pediatric vitiligo and whether the extent, distribution, and duration of vitiligo are associated with QOL. We performed an online parental questionnaire-based study (N = 350) regarding children ages 0 to 17 years with vitiligo, including validated questions about body surface area (BSA), distribution, and age of onset of vitiligo, associated symptoms, and QOL using the Children’s Dermatology Life Quality Index (CDLQI). Vitiligo negatively affected numerous aspects of and total CDLQI score (median 3.0, interquartile range 5.0). Their vitiligo lesions did not bother only 4.1% of teenagers ages 15 to 17 years, versus 45.6% of children ages 0 to 6 years and 50.0% of those ages 7 to 14 years (p < 0.001). There was no association between the child’s age and whether the child’s vitiligo bothered the parents (p = 0.27). The most bothersome sites of vitiligo lesions for children and parents were the face (25.6% and 37.4%, respectively) and legs (26.2% and 26.2%, respectively). Eighty-two patients (30.1%) reported itching and painful skin within the past week. Using multivariate ordinal logistic regression models, it was found that an affected BSA of more than 25% was associated with self-consciousness, difficulty with friendships and schoolwork, and teasing and bullying. Lesions on the face and arms were associated with teasing and bullying. The extent of vitiligo is associated with QOL impairment in children and adolescents, especially self-consciousness, but also bullying and teasing. Different distributions of vitiligo lesions are associated with different aspects of QOL impairment. Teenagers ages 15 to 17 years seem to experience the most self-consciousness of all pediatric age groups.
Although vitiligo has been reported to cause significant impairment of quality of life (QOL) (1–4), specific areas of QOL impairment (e.g., functional limitations and impaired activities of daily living) have not been well characterized in large
studies. A few studies have supported the theory that skin disease in general and vitiligo in particular create an emotional burden and interference with psychosocial development. Weber et al demonstrated higher Children’s Dermatology Life Quality Index (CDLQI)
Address correspondence to Nanette B. Silverberg, M.D., Department of Dermatology Suite, St. Luke’s-Roosevelt Hospital and Beth Israel Medical Centers, 11D 1090 Amsterdam Ave. New York, NY 10025, or e-mail: [email protected]. DOI: 10.1111/pde.12226
© 2013 Wiley Periodicals, Inc.
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scores for chronic skin disease (5). One study found that QOL is poorer in children with vitiligo than in those with atopic dermatitis (6). Other authors have suggested that vitiligo correlates with childhood depression, especially in the preteen years, and that disease in teenagers, especially in visible areas, may affect emotional development (7). We hypothesized further that a greater extent of vitiligo would be associated with worse QOL and that different aspects of QOL would be affected at different ages and with different distributions of vitiligo lesions. We also hypothesized that different aspects of the illness would bother parents than those that bothered their children. We conducted an online survey of parents of children with vitiligo to identify leading factors in the development of emotional disturbance for children with vitiligo and their parents.
METHODS Study Population and Questionnaire Distribution The Institutional Review Board at St. Luke’s-Roosevelt Hospital Center (www.clinicaltrials.gov registration NCT01401374) approved this parental questionnaire– based study of children with vitiligo younger than 18 years of age. Our previously validated survey showed self-reports of physician-diagnosed vitiligo, body surface area (BSA) affected by vitiligo, number of affected body parts, and disease duration were all strongly correlated with clinical assessment by a dermatologist (8). Exclusion criteria were not being diagnosed with vitiligo by a physician, vitiligo onset reported at birth (to exclude possible cases of nevus depigmentosus or pigmentary mosaicism), and incomplete survey responses. The questionnaire was uploaded to http://www. surveymonkey.com and parents of children enrolled in nonprofit support groups for vitiligo vulgaris were invited to participate by responding to the survey. Responses from initiation of the study in June 2010 through October 2012 were reviewed. The questionnaire took an average of 18 minutes (range 11–29 minutes) to complete. Data were deidentified and confidential and posed no risk to participants; the Institutional Review Board granted a waiver of informed consent.
of vitiligo, and the 11-question CDLQI (9). There were 78 questions overall (56 closed and 22 open questions). The questions related to this study; response rates are listed in supplemental Table S1. Responses were verified by screening for noninteger or implausible values (e.g., age 100). Data and Statistical Analysis Data were processed and statistics were analyzed using SAS version 9.2 (SAS Institute, Inc., Cary, NC). The duration of vitiligo and number of body parts affected were divided into tertiles for statistical analysis because of wide skewing. Analyses were performed for all children ages 0 to 17 years and specifically for those ages 4 to 16 years because the CDLQI was initially designed for the latter age group (9). Total CDLQI scores were categorized as no or small (0–5), moderate (5–9), and very or extremely large (>10) QOL effect [as previously reported (10)], owing to a highly skewed distribution of results. Ordinal logistic regression models were constructed to examine the effect of vitiligo extent on QOL, with categorized CDLQI and individual aspects of the CDLQI divided into four responses (not at all, only a little, quite a lot, very much) as the dependent variables. The independent variables were body surface area (BSA), duration of vitiligo, and number of body parts affected by vitiligo (tertiles). Adjusted odds ratios (aORs) were calculated by including sex and current age (continuous) in the full models. Linear interaction terms were tested and were included in the final models if significant (p < 0.05). Binary logistic regression models were constructed to determine whether the site of vitiligo lesions (independent variable) was a predictor of impaired aspects of QOL on the CDLQI (dependent variable). Multivariate regression analyses were used for all analyses in the study to address potential confounding from other variables, such as age and sex. Missing values were encountered from nonresponse of subjects to various questions (Table S1). Complete case analysis was performed (i.e., missing values were ignored). Correction for multiple dependent tests (k = 61) according to the approach of Benjamini and Hochberg (11) yielded a critical p-value of 0.04.
Questionnaire Questionnaire items were developed to determine novel risk and prognostic factors for vitiligo. The aim of the questionnaire was to identify QOL impairment with vitiligo. The questionnaire consisted of questions pertaining to demographic characteristics, phenotype
RESULTS Survey Population Characteristics Three hundred fifty-one parents started the survey and 350 completed it (99.7% completion rate); 342
Silverberg and Silverberg: Childhood Vitiligo and Quality of Life
reported that a physician had diagnosed their child with vitiligo. Six patients were excluded for being older than 17 years of age; 87.2% of the children were ages 4 to 16 years. Sex, age, duration of disease, BSA, face or body involvement, and presence of bilateral lesions are listed in Table 1. Most Bothersome Sites for Vitiligo Their vitiligo reportedly did not bother 23% of the children (Table 2). Upon age stratification, their vitiligo lesions did not bother 45.6% of children ages 0 to 6 years, 50.0% of those ages 7 to 14 years and 4.1% of adolescents aged 15 to 17 years (p < 0.001). In contrast, there was no significant association between the child’s age and whether their child’s vitiligo bothered the parents (p = 0.27). In subjects whose vitiligo reportedly bothered them, the most bothersome sites for children and parents were the face (25.6% and 37.4%, respectively) and legs (26.2% and 26.2%, respectively). There were no significant associations between a child’s age and which affected sites were most bothersome (p ≥ 0.03). Eighty-four percent of respondents who reported that the most bothersome site was somewhere other than the face attributed this to difficulty covering the site with clothing or makeup.
Sex, n (%) Male Female Age, mean SD Age, years, n (%) 0–3 4–16 17 Duration of vitiligo, years, mean SD Age at vitiligo onset, years, mean SD Body surface area affected,%, n (%)* 1–25 26–50 51–75 76–100 Number of body parts affected, mean SD Distribution, n (%) Face or body Face and body Laterality, n (%) Unilateral or at midline Bilateral
Effect of Vitiligo on CDLQI Scores The median total CDLQI score was 3.0 [interquartile range (IQR) 5.0] in all subjects with vitiligo and 2.5 (IQR 4.0) in children ages 4 to 16 years. CDLQI scores were significantly higher in teenagers ages 15 to 17 years (mean 10.0, IQR 14.0) than in those ages 0 to 6 years (mean 2.0, IQR 3.5) and 7 to 14 years (mean 3.0, IQR 4.0) (Kruskal–Wallis test, p = 0.0004). There was no significant difference in CDLQI scores between boys (mean 3.0, IQR 6.0) and girls (mean 2.0, IQR 4.0, p = 0.83). The total CDLQI score was categorized into no to small (0–5), moderate (5–9), and extremely large (>10) effect and modeled using ordinal logistic regression. Adolescents who developed vitiligo at age 10 years and older had significantly higher CDLQI scores than those who developed it between the ages of 0 and 9 years (odds ratio [OR] = 3.02, 95% confidence interval (CI) 1.09, 8.78, p = 0.03), although the CDLQI score was not associated with BSA, number of body parts affected, bilaterality, or duration of vitiligo lesions (Table 3). Effect of Vitiligo Extent and Duration on Individual Aspects of Lifestyle and QOL
128 (40.0) 192 (60.0)
Vitiligo negatively affected all aspects of the CDLQI (Table 4); 82 subjects (30.1%) reported itching, soreness, or painful skin within the past week. An affected BSA greater than 25% was associated with selfconsciousness, difficulty with friendships and schoolwork, and teasing and bullying (Table 5). In subset analysis of children ages 10 to 17 years, greater affected BSA was also associated with QOL impairment related to clothing decisions, playing, and hobbies (p < 0.03) (data not shown). In subset analysis of children ages 4 to 16 years, there was significantly greater self-consciousness (adjusted OR [aOR] = 2.89, 95% CI 1.30, 6.46, p = 0.009) and sleep impairment associated with a greater BSA (aOR = 9.79, 95% CI 1.75, 54.89, p = 0.009). Similarly, the highest tertile for the number of body parts affected by vitiligo lesions was associated with self-consciousness (aOR = 1.97, 95% CI 1.11, 3.51, p = 0.02) and teasing and bullying (aOR = 2.02, 95% CI 1.17, 4.22, p = 0.01). Individual aspects of the CDLQI were not associated with bilaterality of lesions (p ≥ 0.08) (data not shown).
64 (19.1) 271 (80.9)
Effect of Vitiligo Distribution on QOL
TABLE 1. Phenotype of Disease in Children and Adolescents with Physician Diagnosed Vitiligo (N = 336) Variable
311
Value 146 (43.7) 188 (56.3) 10.4 4.1 22 (6.5) 293 (87.2) 21 (6.3) 4.7 3.6 5.7 3.3 237 (71.0) 57 (17.1) 30 (9.0) 10 (3.0) 8.9 5.7
*Rows do not sum to 336 because of missing values attributed to lack of response to a particular question. Percentages of completed responses per question are reported. SD, standard deviation.
The distribution of vitiligo lesions was associated with impairment of different aspects of the CDLQI. Lesions on the hands, arms, legs, and abdomen were associated
312 Pediatric Dermatology Vol. 31 No. 3 May/June 2014
TABLE 2. Distribution of Most Bothersome Sites of Vitiligo Lesions for Children and Parents Site of vitiligo lesions
Most bothersome
All (0–17) years (n = 336)
0–6 years (n = 54)
7–14 years (n = 184)
15–17 years (n = 36)
p*
(1.9) (25.2) (1.9) (2.6) (0.7) (0.7) (3.3) (4.4) (28.2) (5.2) (22.6)
0 19 2 1 0 1 1 2 14 0 34
(0.0) (23.2) (33.3) (12.5) (0.0) (50.0) (7.7) (12.5) (16.7) (0.0) (45.6)
4 (66.7) 48 (58.5) 3 (50.0) 6 (75.0) 2 (50.0) 1 (50.0) 7 (53.9) 8 (50.0) 58 (69.1) 13 (76.5) 37 (50.0)
2 (33.3) 15 (18.3) 1 (16.7) 1 (12.5) 2 (50.0) 0 (0.0) 5 (38.5) 6 (37.5) 12 (14.3) 4 (23.5) 3 (4.1)
0.28† 0.77 0.84† 0.88† 0.16† 0.63† 0.06† 0.06† 0.18 0.03† 25 42 (79.3) Number of affected body parts 1–4 54 (90.0) (reference) 5–10 48 (87.3) 11–22 41 (78.9) Laterality Unilateral or at 27 (93.1) the midline (reference) Bilateral 124 (85.5) Duration of vitiligo, years 0.1–2.17 47 (87.0) (reference) 2.3–6.0 55 (91.7) 6.0–15.1 46 (79.3) Age of vitiligo onset, years 25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25 1–25 >25
Not at all
Only a little
Quite a lot
Very much
Q1. Scratchy, sore, or painful skin 131 (69.7) 46 (24.5) 9 (4.8) 2 (1.1) 59 (70.2) 18 (21.4) 6 (7.1) 1 (1.2) Q2. Self-conscious about lesions 87 (46.5) 63 (33.7) 23 (12.3) 14 (7.5) 29 (34.5) 31 (36.9) 13 (15.5) 11 (13.1) Q3. Friendships 153 (82.7) 26 (14.1) 5 (2.7) 1 (0.5) 59 (71.1) 15 (18.1) 5 (6.0) 4 (4.8) Q4. Clothing decisions 125 (67.6) 32 (17.3) 17 (9.2) 11 (6.0) 48 (57.8) 16 (19.3) 5 (6.0) 14 (16.9) Q5. Going out, playing, or hobbies 133 (72.3) 32 (17.4) 13 (7.1) 6 (3.3) 56 (66.7) 18 (21.4) 3 (3.6) 7 (8.3) Q6. Swimming or other sports 144 (77.8) 23 (12.4) 5 (2.7) 13 (7.0) 62 (73.8) 9 (10.7) 5 (6.0) 8 (9.5) Q7–1. Schoolwork in past week 131 (87.3) 15 (10.0) 3 (2.0) 1 (0.7) 51 (71.8) 11 (15.5) 6 (8.5) 3 (4.2) Q7–2. Holiday in past week 126 (79.3) 21 (13.2) 9 (5.7) 3 (1.9) 59 (84.3) 6 (8.6) 2 (2.9) 3 (4.3) Q8. Teasing/bullying 124 (67.4) 49 (26.6) 11 (6.0) 0 (0.0) 46 (55.4) 20 (24.1) 11 (13.3) 6 (7.3) Q9. Sleep 159 (86.4) 19 (10.3) 4 (2.2) 2 (1.1) 68 (81.0) 10 (11.9) 5 (6.0) 1 (1.2) Q10. Affect of treatment on quality of life 121 (66.5) 44 (24.2) 12 (6.6) 5 (2.8) 52 (65.8) 16 (20.3) 9 (11.4) 2 (2.5)
OR (95% CI)
p
aOR (95% CI)
p
aOR (95% CI)
p
1.00 1.01 (0.58, 1.76)
— 0.97
1.00 0.98 (0.56, 1.72)
— 0.94
1.00 1.57 (0.61, 4.10)
— 0.35
1.00 1.66 (1.03, 2.67)
— 0.03
1.00 1.67 (1.03, 2.69)
— 0.04
1.00 2.89 (1.30, 6.46)
— 0.009
1.00 2.06 (1.13, 3.76)
— 0.02
1.00 1.90 (1.02, 3.54)
— 0.04
1.00 2.11 (0.73, 6.11)
— 0.17
1.00 1.64 (0.98, 2.73)
— 0.06
1.00 1.58 (0.94, 2.67)
— 0.08
1.00 1.95 (0.86, 4.45)
— 0.11
1.00 1.31 (0.76, 2.27)
— 0.33
1.00 1.22 (0.70, 2.13)
— 0.48
1.00 1.53 (0.63, 3.75)
— 0.35
1.00 1.29 (0.72, 2.33)
— 0.39
1.00 1.26 (0.69, 2.30)
— 0.44
1.00 2.23 (0.80, 6.17)
— 0.12
1.00 2.85 (1.42, 5.75)
— 0.003
1.00 2.52 (1.23, 5.16)
— 0.01
1.00 2.32 (0.73, 7.43)
— 0.15
1.00 0.74 (0.35, 1.55)
— 0.42
1.00 0.69 (0.32, 1.46)
— 0.33
1.00 0.93 (0.24, 3.55)
— 0.91
1.00 1.99 (1.19, 3.32)
— 0.009
1.00 2.04 (1.21, 3.44)
— 0.008
1.00 2.70 (1.11, 6.58)
— .02
1.00 1.53 (0.77, 3.03)
— 0.22
1.00 1.50 (0.75, 3.00)
— 0.26
1.00 9.79 (1.75, 54.89)
— 0.009
1.00 1.09 (0.63, 1.88)
— 0.76
1.00 1.12 (0.65, 1.94)
— 0.68
1.00 1.67 (0.65, 4.25)
— 0.28
Ordinal logistic regression models were constructed with effect of vitiligo divided into four responses (not at all, only a little, quite a lot, very much) as the dependent (outcome) variable. The independent (explanatory) variable was percentage of BSA affected by vitiligo lesions. The ORs and 95% CIs were determined across all levels of the dependent variable simultaneously rather than at an arbitrarily set cut-point and analyzed using binary logistic regression. The aORs were determined by including current age and sex. N = 221–272 subjects with complete information. Subset analyses were also performed for children ages 4–16 years.
suggesting that there may be more of an emotional impairment than a functional impairment of QOL in children. Specific sites affected with vitiligo may play a greater role in children rather than the overall extent of vitiligo lesions. That is, clothing or make-up may hide some sites, limiting functional impairment, but self-awareness of the disease and fear of being ostracized by family and peers might still cause an emotional burden. There was a significant difference between the sites of vitiligo that patients and parents found bothersome. This was particularly true in younger children. That is, younger children may not be old enough to have a sense of self-consciousness or to understand their cosmetic deformity.
Children are also more likely to face teasing and bullying and have functional impairment when they attend school. One might expect that the face would be the most bothersome site of vitiligo lesions in the vast majority of patients. A previous study of 41 children ages 8 to 18 years with vitiligo showed that vitiligo severity correlated negatively with psychosocial scores for adolescents and that involvement of specific areas, including the face in boys and genitalia in girls, was associated with greater QOL impairment (7). Another study of Korean adolescents found that facial lesions were most likely to cause QOL impairment (17). In the present study, lesions other than on the face bothered many patients because some felt that lesions on the face were more amenable to
Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present
44 (50.6) 72 (39.1) 67 (51.2) 49 (35.0) 71 (46.7) 38 (35.5) 69 (49.6) 40 (33.3) 60 (42.9) 49 (41.2) 46 (53.5) 70 (37.8) 36 (57.1) 80 (38.5) 49 (50.0) 67 (38.7) 70 (44.0) 39 (39.0)
Not at all 26 (29.9) 68 (37.0) 37 (28.2) 57 (40.7) 51 (33.6) 42 (39.3) 47 (33.8) 46 (38.3) 51 (36.4) 42 (35.3) 23 (26.7) 71 (38.4) 15 (23.8) 79 (38.0) 26 (26.5) 68 (39.3) 57 (35.9) 36 (36.0)
Only a little 11 (12.6) 25 (13.6) 14 (10.7) 22 (15.7) 18 (11.8) 15 (14.0) 14 (10.1) 19 (15.8) 16 (11.4) 17 (14.3) 9 (10.5) 27 (14.6) 7 (11.1) 29 (13.9) 12 (12.2) 24 (13.9) 18 (11.3) 15 (15.0)
Quite a lot 6 (6.9) 19 (10.3) 13 (9.9) 12 (8.6) 12 (7.9) 12 (11.2) 9 (6.5) 15 (12.5) 13 (9.3) 11 (9.2) 8 (9.3) 17 (9.2) 5 (7.9) 20 (9.6) 11 (11.2) 14 (8.1) 14 (8.8) 10 (10.0)
Very much 1.00 1.51 (0.93, 1.00 1.62 (1.04, 1.00 1.51 (0.96, 1.00 1.98 (1.25, 1.00 1.10 (0.70, 1.00 1.65 (1.02, 1.00 1.87 (1.09, 1.00 1.30 (0.82, 1.00 1.25 (0.79,
OR (95% CI)
1.99)
2.06)
3.21)
2.68)
1.72)
3.14)
2.40)
2.53)
2.43)
aOR (95% CI) 1.00 1.32 (0.81, 1.00 1.38 (0.87, 1.00 1.57 (0.99, 1.00 2.02 (1.27, 1.00 1.15 (0.73, 1.00 1.38 (0.84, 1.00 1.57 (0.89, 1.00 1.17 (0.73, 1.00 1.31 (0.82,
p — 0.09 — 0.03 — 0.34 — 0.003 — 0.69 — 0.04 — 0.02 — 0.27 — 0.34 2.10)
1.88)
2.75)
2.27)
1.82)
3.22)
2.50)
2.18)
2.16)
— 0.26 — 0.18 — 0.06 — 0.003 — 0.54 — 0.21 — 0.12 — 0.52 — 0.25
p
1.00 1.48 (0.87, 2.50) 1.00 1.39 (0.85, 2.29) 1.00 1.35 (0.82, 2.24) 1.00 1.85 (1.12, 3.07) 1.00 0.92 (0.56, 1.52) 1.00 1.20 (0.70, 2.07) 1.00 1.18 (0.65, 2.14) 1.00 1.17 (0.70, 1.97) 1.00 1.04 (0.62, 1.74)
aOR (95% CI)
Ages 4–16 years
— 0.15 — 0.19 — 0.24 — 0.01 — 0.76 — 0.50 — 0.60 — 0.56 — 0.88
p
Ordinal logistic regression models were constructed with self-consciousness divided into four responses (not at all, only a little, quite a lot, very much) as the dependent (outcome) variable. The independent (explanatory) variable was the percentage of BSA affected by vitiligo lesions. The ORs and 95% CIs were determined across all levels of the dependent variable simultaneously rather than at an arbitrarily set cut-point and analyzed using binary logistic regression. The aORs were determined by including current age and sex. N = 221–272 subjects with complete information. Subset analyses were also performed for children ages 4–16 years.
Genitals
Feet
Legs
Arms
Back
Abdomen
Chest
Hands
Face
Site
Self-consciousness, n (%)
Ages 0–17 years
TABLE 6. Ordinal Logistic Regression Analysis of Effect of Site of Vitiligo Lesions on Self-Consciousness
Silverberg and Silverberg: Childhood Vitiligo and Quality of Life 315
Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present Not present Present
69 (79.3) 101 (56.1) 89 (69.5) 81 (58.3) 100 (67.1) 61 (57.6) 89 (65.4) 72 (60.5) 93 (67.4) 68 (58.1) 64 (75.3) 106 (58.2) 40 (66.7) 130 (62.8) 63 (65.6) 107 (62.6) 100 (64.1) 61 (61.6)
Not at all 15 (17.2) 54 (30.0) 30 (23.4) 39 (28.1) 35 (23.5) 32 (30.2) 37 (27.2) 30 (25.2) 34 (24.6) 33 (28.2) 14 (16.5) 55 (30.2) 15 (25.0) 54 (26.1) 25 (26.0) 44 (25.7) 44 (28.2) 23 (23.2)
Only a little 2 (2.3) 20 (11.1) 8 (6.3) 14 (10.1) 13 (8.7) 9 (8.5) 10 (7.4) 12 (10.1) 10 (7.3) 12 (10.3) 6 (7.1) 16 (8.8) 4 (6.7) 18 (8.7) 7 (7.3) 15 (8.8) 11 (7.1) 11 (11.1)
Quite a lot 1 (1.2) 5 (2.8) 1 (0.8) 5 (3.6) 1 (0.7) 4 (3.8) 0 (0.0) 5 (4.2) 1 (0.7) 4 (3.4) 1 (1.2) 5 (2.8) 1 (1.7) 5 (2.4) 1 (1.0) 5 (2.9) 1 (0.6) 4 (4.0)
Very much 1.00 3.08 (1.70, 1.00 1.70 (1.03, 1.00 1.50 (0.91, 1.00 1.35 (0.82, 1.00 1.55 (0.94, 1.00 2.10 (1.19, 1.00 1.21 (0.67, 1.00 1.18 (0.71, 1.00 1.24 (0.75,
OR (95% CI)
2.06)
1.98)
2.19)
3.69)
2.55)
2.22)
2.48)
2.79)
5.57)
aOR (95% CI) 1.00 3.00 (1.62, 5.56) 1.00 1.54 (0.92, 2.58) 1.00 1.56 (0.93, 2.60) 1.00 1.40 (0.84, 2.33) 1.00 1.65 (0.99, 2.75) 1.00 1.93 (1.07, 3.46) 1.00 1.05 (0.56, 1.96) 1.00 1.13 (0.67, 1.92) 1.00 1.30 (0.78, 2.18)
p — 0.0002 — 0.04 — 0.12 — 0.24 — 0.09 — 0.01 — 0.54 — 0.52 — 0.41
— 0.0005 — 0.09 — 0.09 — 0.19 — 0.05 — 0.03 — 0.88 — 0.65 — 0.32
p
1.00 2.68 (1.40, 5.12) 1.00 1.26 (0.72, 2.20) 1.00 1.21 (0.69, 2.12) 1.00 1.17 (0.67, 2.05) 1.00 1.21 (0.69, 2.12) 1.00 1.61 (0.86, 3.01) 1.00 0.95 (0.49, 1.86) 1.00 1.05 (0.58, 1.88) 1.00 1.17 (0.66, 2.08)
aOR (95% CI)
Ages 4–16 years
— 0.001 — 0.42 — 0.51 — 0.58 — 0.50 — 0.14 — 0.89 — 0.88 — 0.59
p
Ordinal logistic regression models were constructed with teasing and bullying divided into four responses (not at all, only a little, quite a lot, very much) as the dependent (outcome) variable. The independent (explanatory) variable was the percentage of BSA affected by vitiligo lesions. The ORs and 95% CIs were determined across all levels of the dependent variable simultaneously rather than at an arbitrarily set cut-point and analyzed using binary logistic regression. The aORs were determined by including current age and sex. N = 221–272 subjects with complete information. Subset analyses were also performed for children ages 4–16 years.
Genitals
Feet
Legs
Arms
Back
Abdomen
Chest
Hands
Face
Site
Teasing and bullying, n (%)
Ages 0–17 years
TABLE 7. Ordinal Logistic Regression Analysis of Impact of Site of Vitiligo Lesions on Teasing or Bullying
316 Pediatric Dermatology Vol. 31 No. 3 May/June 2014
Silverberg and Silverberg: Childhood Vitiligo and Quality of Life
cover-up with make-up than lesions on the extremities. This highlights that lesions on the trunk and extremities are also a significant cause of morbidity in childhood vitiligo. Vitiligo had a very large effect on QOL in 13% of patients in this study (CDLQI > 10), which highlights the importance of screening for emotional and functional impairment in the clinical assessment of childhood vitiligo. Children and adolescents with emotional impairment from vitiligo may be at risk for long-term psychological and developmental problems. Such patients may benefit from referrals for psychological intervention and systemic therapeutic interventions, including photo- and oral therapy. A previous study of 51 children with generalized vitiligo found that narrowband ultraviolet B therapy resulted in considerable repigmentation and improved CDLQI scores (18). Further studies are necessary to identify other therapeutic approaches that can improve QOL with vitiligo in adults and children. Itching and burning skin occurs in one-third of patients. We previously reported that 35.5% of adults with vitiligo had itching and burning of their skin and discussed potential mechanisms for such skin symptoms, including higher-grade inflammation, heightened sensitivity, Koebnerization of lesions due to manipulation, and symptoms secondary to comorbid autoimmune disease or skin pathology (8). Consideration of itching and burning skin is warranted in the assessment of children with vitiligo. This study has several strengths, including being prospective with a validated questionnaire for QOL (CDLQI) (9), although it has potential limitations. Subjects were recruited from vitiligo support groups and may have more psychosocial disturbance than patients who do not go online regarding their disease. Nevertheless, the study included a large number of subjects with limited vitiligo extent and mild to no reported QOL impairment. Given our hypothesis of age-related QOL changes, we included all children ages 0 to 17 years, which extends beyond the initial CDLQI design for children ages 4 to 16 years (9). To address this we performed subset analyses in children ages 4 to 16 years and achieved largely similar results. Measures of vitiligo extent are self-reported and are not assessed clinically, but our previously validated questionnaire showed that self-reports of vitiligo diagnosis and extent are accurate (8). Parents completed surveys, which may have resulted in discordance with the child’s viewpoint and overreporting of QOL impairment, although parents were apparently aware of this, as indicated by their responses about
317
the most bothersome site of lesions for them versus their child. Finally, Fitzpatrick skin type was not assessed. In conclusion, vitiligo is not just a cosmetic disorder, it causes emotional and functional alterations in childhood. Greater vitiligo extent is associated with greater impairment in QOL, including skin symptoms, self-consciousness, teasing and bullying, and functional impairment. Teenagers ages 15 to 17 years appear to have the greatest QOL burden from vitiligo. Screening for QOL impairment should be included in the evaluation of childhood vitiligo and incorporated into therapeutic decision making. Further studies are needed to determine the emotional benefit of treatment of childhood vitiligo. SUPPORTING INFORMATION
Additional supporting information may be found in the online version of this article: Table S1. Questions used in the study. REFERENCES 1. Mechri A, Amri M, Douarika AA et al. Psychiatric morbidity and quality of life in vitiligo: a case controlled study. Tunis Med 2006;84:632–635. 2. Ongenae K, Dierckxsens L, Brochez L et al. Quality of life and stigmatization profile in a cohort of vitiligo patients and effect of the use of camouflage. Dermatology 2005;210:279–285. 3. Ongenae K, Van Geel N, De Schepper S et al. Effect of vitiligo on self-reported health-related quality of life. Br J Dermatol 2005;152:1165–1172. 4. Prcic S, Durovic D, Duran V et al. Some psychological characteristics of children and adolescents with vitiligo— our results. Med Pregl 2006;59:265–269. 5. Weber MB, Lorenzini D, Reinehr CP et al. Assessment of the quality of life of pediatric patients at a center of excellence in dermatology in southern Brazil. An Bras Dermatol 2012;87:697–702. 6. Dertlioglu SB, Cicek D, Balci DD et al. Dermatology life quality index scores in children with vitiligo: comparison with atopic dermatitis and healthy control subjects. Int J Dermatol 2013;52:96–101. 7. Bilgic O, Bilgic A, Akis HK et al. Depression, anxiety and health-related quality of life in children and adolescents with vitiligo. Clin Exp Dermatol 2011;36:360–365. 8. Silverberg JI, Silverberg NB. Association between vitiligo extent and distribution and quality-of-life impairment. JAMA Dermatol 2013;149:159–164. 9. Lewis-Jones MS, Finlay AY. The Children’s Dermatology Life Quality Index (CDLQI): initial validation and practical use. Br J Dermatol 1995;132:942–949. 10. Hongbo Y, Thomas CL, Harrison MA et al. Translating the science of quality of life into practice: What do
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