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Quality of life issues in pediatric dermatology
Quality of Life Issues in Pediatric Dermatology Sarah L. Chamlin, MD and Mary-Margaret Chren, MD
Outcomes research, or the measurement and improvem e n t of the end results of health care, is a rapidly growing area of interest in both pediatric s and dermatology. Traditionally, efficacy of treatment has been assessed by objective outcome measures such as blood pressure, clinical severity, and laboratory examinations. More recently, the measurement of quality of life as a health outcome has been recognized as an important part of patient assessment, particularly for chronic diseases that do not affect survival or the results of laboratory tests. After decades of technologyfocused advances in laboratory and imaging studies, research efforts are currently focusing on the development and use of quality-of-life measures. Quality of life is a subjective concept that includes many dimensions of health and well-being, including physical, psychological, and social aspects. A measurement of quality of life allows patients and families to describe quantitatively how a disease and its therapy affects many aspects of their lives. Quality-oflife measures can be used in both research and clinical practice to evaluate efficacy and impact of therapy on this important health outcome. Development of feasible, accurate quality-of-life measures is a complex process of instrument development, testing, and refinement. Several quality-of-life instruments have been developed to assess adults with skin disease. The development of similar outcome measures in pediatric dermatology lags far behind. Qualitative and quantitative data exist that describe the psychological and social effects of cutaneous disease on the lives of affected children. The Pediatric Symptom Checklist has been used to assess the prevalence of psychosocial dysfunction in pediatric dermatology clinics. The Pediatric Symptom Checklist is a 35 item questionnaire developed to identify psychosocial morbidity in school-aged children. 1 Children seen in dermatology clinics and a control group of healthy children demonstrated similar rates of dysfunction with this instrument (13% and 14%, respectively). Although this quantitative study did not identify an overall increased rate of dysfunction, 44% of the dermatology patients with scores indicating dysfunction Curr Probl Dermatol, May/June 2000
were reported to have skin disorders affecting their appearance. The association between psychosocial dysfunction and a condition affecting appearance was statistically significant. 2 The social and psychological impact of facial hemangiomas, the most common benign tumor of infancy, has been recently investigated.3 This descriptive study examined the effects of facial hemangiomas on parental emotion and adaptation, experiences with public reactions, parental-child interactions, and satisfaction with medical care. Parents of children with hemangiomas reported feelings of disbelief, panic, fear, loss, and mourning. Social stigmatization and a high rate of dissatisfaction with medical care were also documented. Importantly, this qualitative examination of the impact of facial hemangiomas illustrates the complex relationship between perception of disease and disease prognosis. Often a disease with a good prognosis nonetheless dramatically impacts a family. Acne vulgaris is thought to have significant adverse psychosocial effects on adolescents and adults. A study of teenagers with acne documented feelings of dissatisfaction with facial appearance in 58% of the teens examined. The dissatisfaction correlated with feelings of embarrassment and social inhibition.4 Adults with acne are found to have increased levels of anxiety, with the degree of anxiety increasing as severity increases. 5 Several other studies document depression, anxiety, and poor self-esteem in adults with acne, while a paucity o f quantitative or qualitative data exists for children with acne. 6-s Atopic dermatitis has a major impact psychologically and socially on the affected child and his/her family. Many investigators have studied the effects of childhood atopic dermatitis on sleep patterns. Children with atopic dermatitis are documented to have significant sleep abnormalities including increased difficulty falling asleep, increased restless sleep, difficulty with morning awakening, and decreased total sleep time when assessed with the Child Sleep Behavior Scale. Difficulty falling asleep and night awakening correlated with daytime discipline problems. 9 Atopic dermatitis significantly increases the number of arousals and 123
awakenings even when in remission. 1° Children with moderate and severe atopic dermatitis have been found to have twice the rate of psychological disturbance compared to control children with minor skin lesions when tested with the Rutter Questionnaire. 11 In addition to the effect on the child, parents of children with atopic dermatitis are reported to have significantly lower rates of employment, higher levels of psychosocial stress, and lower levels of social support. 12 These examples provide evidence that cutaneous disease in children likely significantly affects their quality of life, through psychosocial dysfunction, family stress, and physical dysfunction such as sleep abnormalities. In adults, several direct measures have been developed to quantify the adverse effects of skin disease on quality of life. These instruments include, among others, the Dermatology Life Quality Index t3 and SkindexJ 4 The Children's Dermatology Life Quality Index (CDLQI), has been developed for use in children with skin disease. CDLQI was developed in Britain to assess the quality of life in children with skin disease of any sort. This 10 question index With proven reliability has been used in children with inflammatory and non-inflammatory skin diseases as well as normal children. 15 Formal validity and responsiveness testing has not been reported. The CDLQI is the only skin-specific quality=of-life instrument for children and is an important first step in applying quality-of-life measurement to pediatric dermatology. However, this measure may not be broadly applicable for comprehensive assessments. The small number of questions may limit the assessment of the complexities encountered in understanding and quantifying quality of life. In addition, this measure was developed generically for children 3-16 years of age and does not account for developmental differences. Two outcome instruments have been developed to quantify the impact of childhood disease on the family: the Impact-on-Family Scale and the Dermatitis Family Impact questionnaire. The Impact-on-Family Scale is a generic measure developed to assess the effect of childhood illness on the family system. Reliability was evaluated, but formal validity and responsiveness testing has not been reported. 16 A recent study using the Impact-on-Family Scale compared the impact of both juvenile onset diabetes mellitus and atopic dermatitis on families. The Australian investigators found a significantly higher impact on families of children with moderate or severe atopic dermatitis when compared to families of children with juvenile onset diabetes, iv 124
The second instrument, the Dermatitis Family Impact questionnaire, recently developed in Britain, is a 10 question survey with proven reliability. A significantly greater impact on families of children with atopic dermatitis was reported when compared with control families of healthy children.18 Formal validity and responsiveness studies have not been reported. The American Academy of Pediatrics has identified the need to measure quality of life or functional outcomes in children. The Academy launched the Functional Outcomes Project in 1992 to develop age and disease specific instruments assessing asthma, otitis media, juvenile rheumatoid arthritis, and head injury. A core set of domains has been identified that include the many dimensions of health in children. The dimensions include activity, physical health, emotional health, social interactions, and health care utilization. Self-administered questionnaires are developed to measure these dimensions of life. An age and disease specific quality-of-life measure for atopic dermatitis would be widely useful. Atopic dermatitis is the most common skin disease in children, occurring in approximately 20-30% of children. 19 In addition to the psychosocial effects, atopic dermatitis has a great economic impact in the United States. A cost analysis of atopic dermatitis estimated the total national cost to be $364 million annually. An inappropriately high rate of emergency room use was reported, accounting for $87 million dollars of this total annually. 2° This misuse of resources suggests that current practices of care are inadequate. Some practitioners have suggested that multidisciplinary care may improve the lives and care of children with atopic dermatitis. Because quality-of-life measures can be used in clinical research to assess current care practices and determine the efficacy of alternative therapies and systems of care, a quality-of-life measure for atopic dermatitis could be used in a study to test this hypothesis. In the future, home visits, multidisciplinary clinics, and specialist nurses may be found to lessen the severity of disease, improve the quality of life, and decrease the economic burden in children with atopic dermatitis and their families.
REFERENCES 1. Jellinek MS, Murphy JM, Bums B. Brief psychosocial screening in outpatient pediatric practice. J Pediatr 1986;109:3718. 2. Rauch PK, Jellinek MS, Murphy JM, Schachner L, Hansen R, Esterly NB, et al. Screening for psychosocial dysfunction in pediatric dermatology practice. Clin Pediatr 1991 ;30:493-7. Curr Probl Dermatol, May/June 2000
3. Tanner JL, Dechert MP, Frieden IJ. Growing up with a facial hemangioma: parent and child coping and adaptation. Pediatrics 1998;101:446-52. 4. Krowchuk DP, Stancin T, Keskinen R, Walker R, Bass J, Anglin TM. The psychosocial effects of ache on adolescents. Pediatr Dermatol 1991;8:332-8. 5. Wu SF, Kinder BN, Tmnnell TN, Fulton JE. Role of anxiety and anger in acne patients: a relationship with the severity of the disorder. J Amer Acad Dermatol 1988;18:325-33. 6. Rubinow DR, Peck GL, Squillace KM, Gantt GG. Reduced anxiety and depression in cystic ache patients after successful treatment with oral isotretinoin. J Amer Acad Dermatol 1987;17:25-32. 7. Newton JN, Mallon E, Klassen A, Ryan TJ, Finlay AY. The effectiveness of acne treatment; an assessment by patients of the outcome of therapy. Br J Dermatol 1997;137:563-7. 8. Lasek RJ, Chren MM. The effects of acne vulgaris on the quality of life of adult dermatology patients. Arch Dermatol 1998;134:454-8. 9. Dahl RE, Bernhisel-Broadbent J, Scanlon-Holdford S, Sampson HA, Lupo M. Sleep disturbance in children with atopic dermatitis. Arch Pediatr Adolesc Med 1995;149:85660. 10. Reuveni HR, Chapnick G, Tal A, Tarasiuk A. Sleep fragmentation in children with atopic dermatitis. Arch Pediatr Adolesc Med 1999; 153:249-53. 11. Absolon CM, Cottrell D, Eldridge SM, Glover MT. Psychological disturbance in atopic eczema: the extent of the problem in school-aged children. Br J Dermatol 1997;137: 241-5.
Curr Probl Dermatol, May/June 2000
12. Daud LR, Garralda ME, David TJ. Psychosocial adjustment in preschool children with atopic eczema. Arch Dis Child 1993;69:670-6. 13. Finlay AY, Khan GK. Dermatology life quality index (DLQI)-a simple practical measure for routine clinical use. Clin Exp Dermatol 1994;19:210-6. 14. Chren MM, Lasek RJ, Quinn LM, Mostow EN, Zyzanski SJ. Skindex, a quality-of-life measure for patients with skin disease: reliability, validity, and responsiveness. J Invest Dermatol 1996;107:707-13. 15. Lewis-Jones MS, Finlay AY. The Children's Dermatology Life Quality Index (CDLQI): initial validation and practical use. Br J Dermatol 1995;132:942-9. 16. Stein RE, Riessman CK. The development of an impact-onfamily scale: preliminary findings. Med Care 1980;18:46572. 17. Su JC, Kemp AS, Varigos GA, Nolan TM. Atopic eczema: its impact on the family and financial cost. Arch Dis Child 1997;76:159-62. 18. Lawson V, Lewis-Jones MS, Finlay AY, Reid P, Owens RG. The family impact of childhood atopic dermatitis: the dermatitis family impact questionnaire. Br J Dermatol 1998; 138:107-13. 19. Mar A, Tam M, Jolley D, Marks R. The cumulative incidence of atopic dermatitis in the first 12 months among Chinese, Vietnamese, and Caucasian infants born in Melbourne, Australia. J Amer Acad Dermatol 1999;40:597-02. 20. Lapidus CS, Schwarz DF, Honig PJ. Atopic dermatitis in children: Who cares? Who pays? J Amer Acad Dermatol 1993;28:699-03.
125
Outcomes research, or the measurement and improvem e n t of the end results of health care, is a rapidly growing area of interest in both pediatric s and dermatology. Traditionally, efficacy of treatment has been assessed by objective outcome measures such as blood pressure, clinical severity, and laboratory examinations. More recently, the measurement of quality of life as a health outcome has been recognized as an important part of patient assessment, particularly for chronic diseases that do not affect survival or the results of laboratory tests. After decades of technologyfocused advances in laboratory and imaging studies, research efforts are currently focusing on the development and use of quality-of-life measures. Quality of life is a subjective concept that includes many dimensions of health and well-being, including physical, psychological, and social aspects. A measurement of quality of life allows patients and families to describe quantitatively how a disease and its therapy affects many aspects of their lives. Quality-oflife measures can be used in both research and clinical practice to evaluate efficacy and impact of therapy on this important health outcome. Development of feasible, accurate quality-of-life measures is a complex process of instrument development, testing, and refinement. Several quality-of-life instruments have been developed to assess adults with skin disease. The development of similar outcome measures in pediatric dermatology lags far behind. Qualitative and quantitative data exist that describe the psychological and social effects of cutaneous disease on the lives of affected children. The Pediatric Symptom Checklist has been used to assess the prevalence of psychosocial dysfunction in pediatric dermatology clinics. The Pediatric Symptom Checklist is a 35 item questionnaire developed to identify psychosocial morbidity in school-aged children. 1 Children seen in dermatology clinics and a control group of healthy children demonstrated similar rates of dysfunction with this instrument (13% and 14%, respectively). Although this quantitative study did not identify an overall increased rate of dysfunction, 44% of the dermatology patients with scores indicating dysfunction Curr Probl Dermatol, May/June 2000
were reported to have skin disorders affecting their appearance. The association between psychosocial dysfunction and a condition affecting appearance was statistically significant. 2 The social and psychological impact of facial hemangiomas, the most common benign tumor of infancy, has been recently investigated.3 This descriptive study examined the effects of facial hemangiomas on parental emotion and adaptation, experiences with public reactions, parental-child interactions, and satisfaction with medical care. Parents of children with hemangiomas reported feelings of disbelief, panic, fear, loss, and mourning. Social stigmatization and a high rate of dissatisfaction with medical care were also documented. Importantly, this qualitative examination of the impact of facial hemangiomas illustrates the complex relationship between perception of disease and disease prognosis. Often a disease with a good prognosis nonetheless dramatically impacts a family. Acne vulgaris is thought to have significant adverse psychosocial effects on adolescents and adults. A study of teenagers with acne documented feelings of dissatisfaction with facial appearance in 58% of the teens examined. The dissatisfaction correlated with feelings of embarrassment and social inhibition.4 Adults with acne are found to have increased levels of anxiety, with the degree of anxiety increasing as severity increases. 5 Several other studies document depression, anxiety, and poor self-esteem in adults with acne, while a paucity o f quantitative or qualitative data exists for children with acne. 6-s Atopic dermatitis has a major impact psychologically and socially on the affected child and his/her family. Many investigators have studied the effects of childhood atopic dermatitis on sleep patterns. Children with atopic dermatitis are documented to have significant sleep abnormalities including increased difficulty falling asleep, increased restless sleep, difficulty with morning awakening, and decreased total sleep time when assessed with the Child Sleep Behavior Scale. Difficulty falling asleep and night awakening correlated with daytime discipline problems. 9 Atopic dermatitis significantly increases the number of arousals and 123
awakenings even when in remission. 1° Children with moderate and severe atopic dermatitis have been found to have twice the rate of psychological disturbance compared to control children with minor skin lesions when tested with the Rutter Questionnaire. 11 In addition to the effect on the child, parents of children with atopic dermatitis are reported to have significantly lower rates of employment, higher levels of psychosocial stress, and lower levels of social support. 12 These examples provide evidence that cutaneous disease in children likely significantly affects their quality of life, through psychosocial dysfunction, family stress, and physical dysfunction such as sleep abnormalities. In adults, several direct measures have been developed to quantify the adverse effects of skin disease on quality of life. These instruments include, among others, the Dermatology Life Quality Index t3 and SkindexJ 4 The Children's Dermatology Life Quality Index (CDLQI), has been developed for use in children with skin disease. CDLQI was developed in Britain to assess the quality of life in children with skin disease of any sort. This 10 question index With proven reliability has been used in children with inflammatory and non-inflammatory skin diseases as well as normal children. 15 Formal validity and responsiveness testing has not been reported. The CDLQI is the only skin-specific quality=of-life instrument for children and is an important first step in applying quality-of-life measurement to pediatric dermatology. However, this measure may not be broadly applicable for comprehensive assessments. The small number of questions may limit the assessment of the complexities encountered in understanding and quantifying quality of life. In addition, this measure was developed generically for children 3-16 years of age and does not account for developmental differences. Two outcome instruments have been developed to quantify the impact of childhood disease on the family: the Impact-on-Family Scale and the Dermatitis Family Impact questionnaire. The Impact-on-Family Scale is a generic measure developed to assess the effect of childhood illness on the family system. Reliability was evaluated, but formal validity and responsiveness testing has not been reported. 16 A recent study using the Impact-on-Family Scale compared the impact of both juvenile onset diabetes mellitus and atopic dermatitis on families. The Australian investigators found a significantly higher impact on families of children with moderate or severe atopic dermatitis when compared to families of children with juvenile onset diabetes, iv 124
The second instrument, the Dermatitis Family Impact questionnaire, recently developed in Britain, is a 10 question survey with proven reliability. A significantly greater impact on families of children with atopic dermatitis was reported when compared with control families of healthy children.18 Formal validity and responsiveness studies have not been reported. The American Academy of Pediatrics has identified the need to measure quality of life or functional outcomes in children. The Academy launched the Functional Outcomes Project in 1992 to develop age and disease specific instruments assessing asthma, otitis media, juvenile rheumatoid arthritis, and head injury. A core set of domains has been identified that include the many dimensions of health in children. The dimensions include activity, physical health, emotional health, social interactions, and health care utilization. Self-administered questionnaires are developed to measure these dimensions of life. An age and disease specific quality-of-life measure for atopic dermatitis would be widely useful. Atopic dermatitis is the most common skin disease in children, occurring in approximately 20-30% of children. 19 In addition to the psychosocial effects, atopic dermatitis has a great economic impact in the United States. A cost analysis of atopic dermatitis estimated the total national cost to be $364 million annually. An inappropriately high rate of emergency room use was reported, accounting for $87 million dollars of this total annually. 2° This misuse of resources suggests that current practices of care are inadequate. Some practitioners have suggested that multidisciplinary care may improve the lives and care of children with atopic dermatitis. Because quality-of-life measures can be used in clinical research to assess current care practices and determine the efficacy of alternative therapies and systems of care, a quality-of-life measure for atopic dermatitis could be used in a study to test this hypothesis. In the future, home visits, multidisciplinary clinics, and specialist nurses may be found to lessen the severity of disease, improve the quality of life, and decrease the economic burden in children with atopic dermatitis and their families.
REFERENCES 1. Jellinek MS, Murphy JM, Bums B. Brief psychosocial screening in outpatient pediatric practice. J Pediatr 1986;109:3718. 2. Rauch PK, Jellinek MS, Murphy JM, Schachner L, Hansen R, Esterly NB, et al. Screening for psychosocial dysfunction in pediatric dermatology practice. Clin Pediatr 1991 ;30:493-7. Curr Probl Dermatol, May/June 2000
3. Tanner JL, Dechert MP, Frieden IJ. Growing up with a facial hemangioma: parent and child coping and adaptation. Pediatrics 1998;101:446-52. 4. Krowchuk DP, Stancin T, Keskinen R, Walker R, Bass J, Anglin TM. The psychosocial effects of ache on adolescents. Pediatr Dermatol 1991;8:332-8. 5. Wu SF, Kinder BN, Tmnnell TN, Fulton JE. Role of anxiety and anger in acne patients: a relationship with the severity of the disorder. J Amer Acad Dermatol 1988;18:325-33. 6. Rubinow DR, Peck GL, Squillace KM, Gantt GG. Reduced anxiety and depression in cystic ache patients after successful treatment with oral isotretinoin. J Amer Acad Dermatol 1987;17:25-32. 7. Newton JN, Mallon E, Klassen A, Ryan TJ, Finlay AY. The effectiveness of acne treatment; an assessment by patients of the outcome of therapy. Br J Dermatol 1997;137:563-7. 8. Lasek RJ, Chren MM. The effects of acne vulgaris on the quality of life of adult dermatology patients. Arch Dermatol 1998;134:454-8. 9. Dahl RE, Bernhisel-Broadbent J, Scanlon-Holdford S, Sampson HA, Lupo M. Sleep disturbance in children with atopic dermatitis. Arch Pediatr Adolesc Med 1995;149:85660. 10. Reuveni HR, Chapnick G, Tal A, Tarasiuk A. Sleep fragmentation in children with atopic dermatitis. Arch Pediatr Adolesc Med 1999; 153:249-53. 11. Absolon CM, Cottrell D, Eldridge SM, Glover MT. Psychological disturbance in atopic eczema: the extent of the problem in school-aged children. Br J Dermatol 1997;137: 241-5.
Curr Probl Dermatol, May/June 2000
12. Daud LR, Garralda ME, David TJ. Psychosocial adjustment in preschool children with atopic eczema. Arch Dis Child 1993;69:670-6. 13. Finlay AY, Khan GK. Dermatology life quality index (DLQI)-a simple practical measure for routine clinical use. Clin Exp Dermatol 1994;19:210-6. 14. Chren MM, Lasek RJ, Quinn LM, Mostow EN, Zyzanski SJ. Skindex, a quality-of-life measure for patients with skin disease: reliability, validity, and responsiveness. J Invest Dermatol 1996;107:707-13. 15. Lewis-Jones MS, Finlay AY. The Children's Dermatology Life Quality Index (CDLQI): initial validation and practical use. Br J Dermatol 1995;132:942-9. 16. Stein RE, Riessman CK. The development of an impact-onfamily scale: preliminary findings. Med Care 1980;18:46572. 17. Su JC, Kemp AS, Varigos GA, Nolan TM. Atopic eczema: its impact on the family and financial cost. Arch Dis Child 1997;76:159-62. 18. Lawson V, Lewis-Jones MS, Finlay AY, Reid P, Owens RG. The family impact of childhood atopic dermatitis: the dermatitis family impact questionnaire. Br J Dermatol 1998; 138:107-13. 19. Mar A, Tam M, Jolley D, Marks R. The cumulative incidence of atopic dermatitis in the first 12 months among Chinese, Vietnamese, and Caucasian infants born in Melbourne, Australia. J Amer Acad Dermatol 1999;40:597-02. 20. Lapidus CS, Schwarz DF, Honig PJ. Atopic dermatitis in children: Who cares? Who pays? J Amer Acad Dermatol 1993;28:699-03.
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