- Email: [email protected]
CLEAN INTERMITTENT CATHETERIZATION IN GENITALLY SENSATE CHILDREN: PATIENT EXPERIENCE AND HEALTH RELATED QUALITY OF LIFE
0022-5347/05/1744-1616/0 THE JOURNAL OF UROLOGY® Copyright © 2005 by AMERICAN UROLOGICAL ASSOCIATION
Vol. 174, 1616 –1619, October 2005 Printed in U.S.A.
DOI: 10.1097/01.ju.0000176620.02031.67
CLEAN INTERMITTENT CATHETERIZATION IN GENITALLY SENSATE CHILDREN: PATIENT EXPERIENCE AND HEALTH RELATED QUALITY OF LIFE SETH A. ALPERT,* EARL Y. CHENG, KERRY F. ZEBOLD
AND
WILLIAM E. KAPLAN
From the Division of Urology, Children’s Memorial Hospital, Chicago, Illinois
ABSTRACT
Purpose: Clean intermittent catheterization (CIC) has proven successful for bladder emptying in children without genital sensation with success rates of 94% to 100% in select groups. A subset of the pediatric population requires CIC for bladder dysfunction, yet has normal genital sensation. This study was designed to assess our experience with CIC in genitally sensate children and the health related (HR) quality of life (QOL) for them and their families. Materials and Methods: A multimodality questionnaire on the usage, learning curve and degree of difficulty of CIC was developed. The PedsQL™ 4.0 Generic Core Scales, a reliable, validated HRQOL survey developed for pediatric patients with chronic disease, was used to assess patient and parental QOL. Children in our practice with normal genital sensation and bladder dysfunction requiring CIC were contacted to complete these questionnaires. When possible, QOL data were collected from the patient (self-report) and one or both of the parents (parent-proxy report). Results: Data for 12 males and 8 females were collected, including 30 QOL questionnaires (12 self, 18 proxy). Of the patients 80% were able to learn CIC technique in 1 clinic visit with the help of a nurse. On a 10-point scale (1—not difficult, 10 —very difficult) the mean degree of difficulty for learning the CIC technique was 3.25 for males and 4.00 for females (3.55 overall). On a 10-point scale (1— uncomfortable, 10 —very comfortable) mean comfort level with the CIC technique was 9.12 for females and 9.46 for males (9.33 overall). On the PedsQL 100-point scale mean QOL was 83.24 for the self-report compared to 81.78 for the parent proxy report. The QOL for normal children has previously been shown as 83 (self) to 87.61 (proxy). Conclusions: CIC was an easy technique for most sensate children to learn in 1 visit and master in a short time. Overall comfort with the technique was excellent and few problems were encountered. Their HRQOL was comparable to that of normal children. KEY WORDS: urinary catheterization, quality of life, bladder
Clean (nonsterile) intermittent catheterization (CIC) was introduced by Lapides et al in 1972 to assist those in whom bladder emptying was incomplete for a variety of reasons.1 CIC revolutionized the care of the patient with dysfunctional voiding and/or neurogenic bladder. Several previous studies have shown that the success of CIC in the pediatric population ranges from 94% to 100%, primarily in children with neurogenic bladder from myelomeningocele or spinal cord injury.2, 3 General perception regarding CIC in genitally sensate children with neurogenic bladder dysfunction is that success rates are lower since those with normal genital sensation will not tolerate catheterization as easily as those without sensation. A limited number of studies have investigated CIC in children with normal genital sensation and have reported success rates of 65% to 70% overall.4, 5 Van Savage et al found that patients with normal sensation on CIC had normal self-esteem5 but no previous studies have specifically examined the health related (HR) quality of life (QOL) of these patients while on CIC. We evaluate sensate children on CIC to assess their experience with the technique and determine the HRQOL for these patients and their parents. * Correspondence: Division of Pediatric Urology, Children’s Memorial Hospital, 2300 Children’s Plaza, Box 24, Chicago, Illinois 60614 (telephone: 773-880-4428; FAX: 773-880-3339; e-mail: [email protected]).
MATERIALS AND METHODS
Upon study approval from the institutional review board, patients seen in our practice during the last 17 years with normal genital sensation and who have been started on CIC for bladder dysfunction were identified and approached for inclusion in this study. We developed a multimodality questionnaire on the use, learning curve and degree of difficulty of CIC. The quality of life study was performed using the PedsQL™ 4.0, developed by Dr. James W. Varni. The PedsQL™ 4.0 is a reliable, validated HRQOL survey developed for pediatric patients with chronic disease, which has separate modules that can be used to assess patient and parental QOL.6, 7 Each QOL questionnaire consists of 21 or 23 age-appropriate questions encompassing the 4 areas of Physical Functioning, Emotional Functioning, Social Functioning and School Functioning. A 5-point response scale is used for patients 8 to 18 years old, whereas a simplified 3-point scale is used for children younger than 8 years to increase ease of use for this age group. Items on these questionnaires are reverse-scored and linearly transformed to a 0 to 100 scale such that higher scores indicate better HRQOL. Questionnaires are available for patients who are old enough to respond themselves (self-report form) and for parents of subjects to respond for them (parent-proxy form). Each patient and/or parents were contacted in person, by mail and/or by telephone and completed these question-
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CATHETERIZATION IN GENITALLY SENSATE CHILDREN
naires. When possible, QOL data were collected from the patient (self-report) and 1 or both parents (parent-proxy report). If the child was too young to complete the PedsQL form, then data were collected for that patient from the parentproxy report. Statistical comparisons were done using a 2-sided t test with p ⱕ0.05 considered significant. Data are reported as mean ⫾ standard deviation. When the concept of the CIC technique as a necessary medical treatment is first introduced, it is beneficial if the family already has a relationship with the nurse, nurse practitioner or other health care professional who will be providing instruction. If this is not the case, then a short period is allowed between the recommendation for CIC and formal instruction in the technique to allow the family and the CIC teacher to build a trusting and therapeutic relationship. This also allows time for the patient and family to adjust emotionally to the decision and be part of the decision-making process. Teaching sessions are scheduled for a time and place that permit uninterrupted instruction in a quiet setting. In addition to the participation of parents, other family members who can provide support or may be asked to perform CIC are encouraged to attend and learn the technique. The patient and family are told that the CIC technique itself is simple, while acknowledging that the emotional impact it carries is difficult. The family is assured that they will soon become the experts in catheterizing their own child. From the beginning of the session, the family understands that catheterizing the child will be accomplished before they leave. First, the instructor gives a verbal review of the technique, using appropriate analogies and/or props to help demonstrate. Concerns about the technique and/or fears of discomfort are discussed and the family is told that instruction will proceed at whatever pace is necessary. Next, the instructor performs CIC on the child after assuring him/her that it will be done slowly and that any discomfort during the procedure will be addressed. Once this has been accomplished, either one or both of the parents perform CIC, followed by any other family members who may wish or need to learn. If the child is old enough to catheterize independently, then he/she will also perform CIC during the appointment. Once all are comfortable with the technique, supplies are either provided to the family at that time or arranged to be delivered to the home. The instructor gives the family multiple contact numbers (ie office, pager, e-mail) so that they have the knowledge and comfort that help and support are always available. A followup telephone call from the instructor within 24 to 48 hours of the appointment is mandatory to answer any questions that may have arisen and to ensure that the family is comfortable with performing CIC in the home setting. For the vast majority of sensate patients additional instruction or appointments have not been necessary. RESULTS
We identified 33 patients with bladder dysfunction but normal genital sensation, of whom 20 were able to be contacted for this study and completed the study instruments. The reasons for starting CIC in these patients are listed in the table. Data for the 12 males and 8 females were collected, including 30 QOL questionnaires (12 self-report, 18 parentproxy). All patients or their parents completed the multimodality questionnaire, which included inquiries about patient demographic information, the level of difficulty to learn CIC and the level of comfort with performance of CIC. Mean patient age at study enrollment was 12.3 ⫾ 6.9 years (range 2.4 to 26.3), and mean age at initiation of CIC was 6.9 ⫾ 4.5 years (range 15 months to 17 years). Mean length of followup on CIC was 5.45 ⫾ 5.37 years (range 6.5 months to 16.5 years), and mean number of catheterizations a day was 4.80 ⫾ 1.52 (range 2 to 8). There were no failures in learning or continuing CIC, although 3 patients stopped CIC
Diagnosis of sensate patients on clean intermittent catheterization Diagnosis Imperforate anus/tethered cord Neurogenic bladder after multiple bladder surgeries Spinal tumor Hinman’s syndrome Neurogenic bladder of unknown etiology Posterior urethral valves Sacral lipoma Lipomeningocele Atonic bladder Ochoa syndrome Total
No. Pts 4 3 3 2 2 2 1 1 1 1 20
either temporarily or permanently on the instructions of their treating physician. Of the patients and/or their parents 80% were able to learn the CIC technique in 1 outpatient clinic visit of an hour or less with the help of a nurse who is experienced in teaching CIC. The remaining 20% required 2 to 3 sessions to learn the technique. On a 10-point scale of difficulty of learning the CIC technique (1—very easy, 10 —very difficult) the mean level of difficulty was 3.55 ⫾ 2.71 for all patients, 3.25 ⫾ 2.57 for boys and 4.00 ⫾ 3.02 for girls. According to the responses on the questionnaire, factors that made learning CIC more challenging included apprehensiveness or uncooperativeness of the patient, discomfort during the procedure or unwillingness of the patient to learn. The main factor that aided in learning the technique was the helpfulness and explanations of the nurse teaching CIC. In regard to patient and/or parent present comfort level with the CIC technique on a 10-point scale of increasing comfort level (1—not comfortable, 10 — very comfortable), the mean comfort level was 9.33 ⫾ 1.10 for all, 9.46 ⫾ 0.84 for boys and 9.12 ⫾ 1.46 for girls. Results from the PedsQL™ 4.0 HRQOL questionnaire showed that the mean QOL score was 83.24 ⫾ 10.32 on the self-report form and 81.78 ⫾ 12.90 on the parent-proxy form. The mean QOL in our study by gender (using self-report and parent-proxy data) was 85.28 ⫾ 9.76 for males and 76.66 ⫾ 13.59 for females. When parent-proxy data were excluded, the QOL by gender for the self-report data was 82.74 ⫾ 10.38 for males and 84.24 ⫾ 11.72 for females. DISCUSSION
When Lapides et al first reported the concept of selfcatheterization, 3 of the 14 patients described had no neurological deficit but had atonic bladders from infrequent voiding.1 The remaining patients had neurogenic bladder due to multiple sclerosis, myelomeningocele, lipoma of the spinal cord or transverse myelitis. Since then, the technique of CIC has become an integral component of the care of patients with the inability to partially or completely empty the bladder. When the clinical situation dictates, the decision to initiate CIC in a patient without genital sensation is usually straightforward. Conversely, the perceived sentiment regarding instituting CIC in genitally sensate individuals, especially children, is that genital discomfort limits its practical use. In 1 study of lower urinary tract dysfunction in children with anorectal malformation Boemers et al found that it was much more difficult for these children and their parents to accept CIC because normal genital sensation caused them pain and/or distress during catheterization attempts.8 They found that those who were started on CIC in the neonatal period or in infancy accepted and were compliant with CIC, but it was nearly impossible to introduce CIC in older children. Of 19 children in whom the use of CIC was deemed desirable for urological care only 7 and their parents (37%) were amenable to it. Our study showed that CIC could
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CATHETERIZATION IN GENITALLY SENSATE CHILDREN
be successfully started in older children, since the mean age in our series was 6.9 years and the oldest patient started CIC at age 17 years. More recent studies have shown better success with instituting CIC in populations of genitally sensate children but the success rates are much lower than those seen with diseases without genital sensation (eg myelodysplasia, spinal cord injury or sacral agenesis). While the success rates for CIC in patients with myelodysplasia range from 94% to100%, Pohl et al showed a 70% success rate in 23 pediatric patients with high post-void residuals but no identifiable anatomic or neurological abnormality.4 Similarly, Van Savage et al demonstrated a 65% success rate in a group of children with a variety of diagnoses who required CIC for bladder management.5 All failures occurred within 6 months in both studies. Of our 20 patients 80% were able to learn CIC within a 1-hour outpatient clinic visit and at a mean of almost 5.5 years of followup, no patient had stopped catheterizing unless directed to do so by a physician. We believe that our high success rate is due to the heavy involvement of an experienced nurse practitioner who works closely with the patients and their families, and is directly available to assist in the teaching and maintenance of the CIC technique. Establishing a trusting relationship between the nurse practitioner and the family from the start is crucial for reinforcing the importance of maintaining CIC as well as for emotional and technical support. Measuring quality of life in patients has become increasingly important in medicine in general and in pediatrics in particular. As Eiser and Morse state: “Where it is possible to manage but not cure a disease, it is important to determine how far treatment and disease compromise the child’s QOL. In this way, informed judgments can be made about whether or not treatment is appropriate, and, where there is a choice, which choice might be the best option for the child.”9 In urological diseases there is a complex interplay between QOL and the control of evacuation.10 Private matters for children such as incontinence may become public when it limits ability to participate in normal childhood activities such as swimming or playing with other children, which may have a profound effect on QOL. Current and future studies of medical treatments in pediatrics have the responsibility to not only assess therapeutic success, but also the impact on the patient as a whole. Previous large studies using the PedsQL to assess QOL of normal, healthy control children showed a self-report QOL score of 83.00 ⫾ 14.79 and a parent-proxy report score of 87.61 ⫾ 12.33.11 Comparison of the QOL scores of these normal children to our CIC patient self-reported QOL scores showed no significant difference (p ⬎0.99). Parent-proxy results between CIC patients and normals approached statistical difference (p ⫽ 0.055). The mean self-report QOL by gender for normal, healthy children in previous studies was 83.16 ⫾ 13.06 for males and 82.54 ⫾ 13.27 for females.12 Our male and female CIC patient QOL scores were not statistically different from those of healthy controls (males p ⫽ 0.93, females p ⫽ 0.80). We also compared our data to those of other studies of children with chronic conditions such as diabetes and pediatric cancer. In a study of children 5 to 18 years old with type 1 or 2 diabetes and their parents the self-reported QOL was 80.37 ⫾ 12.90 and parent-proxy QOL was 76.56 ⫾ 14.10.13 Although significant differences were found in this study between those with diabetes and normal controls, when the diabetes QOL data were compared to those of genitally sensate children on CIC, there was no significant difference in (self-reported QOL p ⫽ 0.43 or parent-proxy QOL p ⫽ 0.10). When our CIC study group was compared with pediatric oncology patients,14 there were statistically significant differences between self-reported QOL (p ⫽ 0.016) and parentproxy QOL (p ⫽ 0.006).
Previous studies on CIC in genitally sensate children have not specifically focused on QOL. Using a proven HRQOL instrument, we were able to determine that the QOL for these children is not significantly different compared to that previously documented in normal, healthy controls. CIC may prevent incontinence and allow these children to better regulate a voiding schedule such that they perceive themselves as no different from their normal peers who do not have to catheterize. Also, since these children are generally more functional and ambulatory than the majority of genitally insensate children who may also have other neurological impairments, they may be able to participate in a wider range of activities with fewer difficulties so that QOL is better overall than those with more serious neurological conditions. One potential bias in our study is the selection bias inherent in those responding to our questionnaires. Of 33 patients we attempted to enroll 20 (60%) responded and completed both study instruments. It is possible that those who responded were more positive in their attitude toward CIC than those who did not respond to the questionnaires. Also, the questionnaire used to evaluate HRQOL is a general instrument evaluating overall QOL for children with chronic diseases and does not specifically address issues regarding urological conditions or catheterization. There is no such questionnaire available at present but efforts to design one specific to this patient group are under way. Work for the future should include comparison of QOL before and after the institution of CIC in genitally sensate children to longitudinally demonstrate that CIC provides a positive benefit to these children, or at least does not have a substantially negative affect on their QOL. The overall number of patients in this study was smaller than that from previous studies of QOL of normal controls, which makes statistical comparison of these values more difficult to validate. CONCLUSIONS
Clean intermittent catheterization can be taught to genitally sensate children and their parents in a short period with high long-term success rates. The assistance of an individual experienced with the technique, such as a nurse or nurse practitioner, is integral to teach the procedure and reinforce the need to continue. Over time, patients and their families become comfortable with CIC and report quality of life similar to that of normal children. REFERENCES
1. Lapides, J., Diokno, A. C., Silber, S. J. and Lowe, B. S.: Clean, intermittent self-catheterization in the treatment of urinary tract disease. J Urol, 107: 458, 1972 2. Kass, E. J., McHugh, T. and Diokno, A. C.: Intermittent catheterization in children less than 6 years old. J Urol, 121: 792, 1979 3. Plunkett, J. M. and Braren, V.: Clean intermittent catheterization in children. J Urol, 121: 469, 1979 4. Pohl, H. G., Bauer, S. B., Borer, J. G., Diamond, D. A., Kelly, M. D., Grant, R. et al: The outcome of voiding dysfunction managed with clean intermittent catheterization in neurologically and anatomically normal children. BJU Int, 89: 923, 2002 5. Van Savage, J. G., Sackett, C. K., Wilhelm, C. L., Sessions, R. P. and Mesrobian, H. G.: Indications for and outcomes of clean intermittent catheterization in children with normal genital sensation. J Urol, 157: 1866, 1997 6. Varni, J. W., Seid, M. and Rode, C. A.: The PedsQL: measurement model for the pediatric quality of life inventory. Med Care, 37: 126, 1999 7. Varni, J. W., Seid, M. and Kurtin, P. S.: PedsQL 4.0: reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Med Care, 39: 800, 2001 8. Boemers, T. M., de Jong, T. P., van Gool, J. D. and Bax, K. M.: Urologic problems in anorectal malformations. Part 2: func-
CATHETERIZATION IN GENITALLY SENSATE CHILDREN tional urologic sequelae. J Pediatr Surg, 31: 634, 1996 9. Eiser, C. and Morse, R.: The measurement of quality of life in children: past and future perspectives. J Dev Behav Pediatr, 22: 248, 2001 10. Gerharz, E. W., Eiser, C. and Woodhouse, C. R.: Current approaches to assessing the quality of life in children and adolescents. BJU Int, 91: 150, 2003 11. Varni, J. W., Seid, M., Knight, T. S., Uzark, K. and Szer, I. S.: The PedsQL 4.0 Generic Core Scales: sensitivity, responsiveness, and impact on clinical decision-making. J Behav Med, 25: 175, 2002 12. Varni, J. W., Burwinkle, T. M., Seid, M. and Skarr, D.: The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr, 3: 329, 2003 13. Varni, J. W., Burwinkle, T. M., Jacobs, J. R., Gottschalk, M., Kaufman, F. and Jones, K. L.: The PedsQL in type 1 and type 2 diabetes: reliability and validity of the Pediatric Quality of Life Inventory Generic Core Scales and type 1 Diabetes Module. Diabetes Care, 26: 631, 2003 14. Varni, J. W., Burwinkle, T. M., Katz, E. R., Meeske, K. and Dickinson, P.: The PedsQL in pediatric cancer: reliability and validity of the Pediatric Quality of Life Inventory Generic Core Scales, Multidimensional Fatigue Scale, and Cancer Module. Cancer, 94: 2090, 2002 EDITORIAL COMMENTS Clean intermittent catheterization was first described in the early 1970s, and since that time it has become a mainstay of management for patients unable to empty the bladder spontaneously. In the pediatric population the majority of these are children with spina bifida, who generally have an insensate bladder and urethra. However, there is a subset of patients, including boys with posterior urethral valves, who have sensation but still need CIC. The common teaching has been that such children are poor candidates for CIC,
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especially if it is begun at a later age. However, as Alpert et al note, there has been a paucity of literature to support this finding. They evaluated quality of life in patients on CIC with a validated instrument, and found it to be comparable to controls. In addition, they evaluated patient discomfort with CIC, and found it to be relatively mild. The authors point out that a key part of the success is the participation of a dedicated clinician, often a nurse practitioner. I think that is one of the most important messages of this article: patients can learn to accept seemingly onerous treatments, provided they receive the help and support of a caring team of clinicians. Barry Duel Urology and Pediatrics University of California, Irvine Orange, California As pediatric urologists, we all have sensate patients in our practice with nonneurogenic bladder dysfunction who require clean intermittent catheterization. Although initially apprehensive, a majority of patients adjust well enough to be compliant with a CIC program. Historical success rates have varied from 37% to 65% (references 4, 5 and 8 in article). Earlier work has reported no change in selfesteem or body image after implementation of a CIC program. However, to date the well-being and ease with which children become proficient in CIC have yet to be evaluated. The authors present a well designed protocol that uses a validated survey instrument to demonstrate no adverse impact on the quality of life for these patients after institution of CIC. Moreover, they highlight the fundamental component, patient education, to achieving durable success with their program. The authors should be applauded for their efforts. C. D. Anthony Herndon Department of Surgery/Urology University of Alabama at Birmingham Birmingham, Alabama
DISCUSSION Dr. William Reiner. When you are designing scales of 1 to 10 for children, you find 4, 7 and 8 are virtually never used. One to 10 is too big a spread for kids, and it is really too big for adults as well. It is much better to use a 1 to 5 scale, which is more reproducible and more understandable for us. Unless you divide it into sectors, such as quartiles, it is difficult for kids to use. However, this was a good study—I liked it. Mr. David Thomas. In your abstract you say that these are patients with normal genital sensation. When I looked at the patient group, you had children with anorectal malformations, who presumably had sacral agenesis, and spinal tumors. Some of these children presumably did have a neuropathic bladder and had reduced sensation. Did you have any children with posterior urethral valves who would have unequivocally normal sensation? Dr. Seth Alpert. We did. If you look at that table again, we had 2 or 3 patients with posterior urethral valves. We also had I think 1 or 2 children with Hinman’s syndrome, and a girl with Ochoa syndrome with no other abnormalities who had unequivocally normal sensation. It is possible that some of our patients had reduced sensation but none had lack of sensation such as those in the spina bifida population.
Vol. 174, 1616 –1619, October 2005 Printed in U.S.A.
DOI: 10.1097/01.ju.0000176620.02031.67
CLEAN INTERMITTENT CATHETERIZATION IN GENITALLY SENSATE CHILDREN: PATIENT EXPERIENCE AND HEALTH RELATED QUALITY OF LIFE SETH A. ALPERT,* EARL Y. CHENG, KERRY F. ZEBOLD
AND
WILLIAM E. KAPLAN
From the Division of Urology, Children’s Memorial Hospital, Chicago, Illinois
ABSTRACT
Purpose: Clean intermittent catheterization (CIC) has proven successful for bladder emptying in children without genital sensation with success rates of 94% to 100% in select groups. A subset of the pediatric population requires CIC for bladder dysfunction, yet has normal genital sensation. This study was designed to assess our experience with CIC in genitally sensate children and the health related (HR) quality of life (QOL) for them and their families. Materials and Methods: A multimodality questionnaire on the usage, learning curve and degree of difficulty of CIC was developed. The PedsQL™ 4.0 Generic Core Scales, a reliable, validated HRQOL survey developed for pediatric patients with chronic disease, was used to assess patient and parental QOL. Children in our practice with normal genital sensation and bladder dysfunction requiring CIC were contacted to complete these questionnaires. When possible, QOL data were collected from the patient (self-report) and one or both of the parents (parent-proxy report). Results: Data for 12 males and 8 females were collected, including 30 QOL questionnaires (12 self, 18 proxy). Of the patients 80% were able to learn CIC technique in 1 clinic visit with the help of a nurse. On a 10-point scale (1—not difficult, 10 —very difficult) the mean degree of difficulty for learning the CIC technique was 3.25 for males and 4.00 for females (3.55 overall). On a 10-point scale (1— uncomfortable, 10 —very comfortable) mean comfort level with the CIC technique was 9.12 for females and 9.46 for males (9.33 overall). On the PedsQL 100-point scale mean QOL was 83.24 for the self-report compared to 81.78 for the parent proxy report. The QOL for normal children has previously been shown as 83 (self) to 87.61 (proxy). Conclusions: CIC was an easy technique for most sensate children to learn in 1 visit and master in a short time. Overall comfort with the technique was excellent and few problems were encountered. Their HRQOL was comparable to that of normal children. KEY WORDS: urinary catheterization, quality of life, bladder
Clean (nonsterile) intermittent catheterization (CIC) was introduced by Lapides et al in 1972 to assist those in whom bladder emptying was incomplete for a variety of reasons.1 CIC revolutionized the care of the patient with dysfunctional voiding and/or neurogenic bladder. Several previous studies have shown that the success of CIC in the pediatric population ranges from 94% to 100%, primarily in children with neurogenic bladder from myelomeningocele or spinal cord injury.2, 3 General perception regarding CIC in genitally sensate children with neurogenic bladder dysfunction is that success rates are lower since those with normal genital sensation will not tolerate catheterization as easily as those without sensation. A limited number of studies have investigated CIC in children with normal genital sensation and have reported success rates of 65% to 70% overall.4, 5 Van Savage et al found that patients with normal sensation on CIC had normal self-esteem5 but no previous studies have specifically examined the health related (HR) quality of life (QOL) of these patients while on CIC. We evaluate sensate children on CIC to assess their experience with the technique and determine the HRQOL for these patients and their parents. * Correspondence: Division of Pediatric Urology, Children’s Memorial Hospital, 2300 Children’s Plaza, Box 24, Chicago, Illinois 60614 (telephone: 773-880-4428; FAX: 773-880-3339; e-mail: [email protected]).
MATERIALS AND METHODS
Upon study approval from the institutional review board, patients seen in our practice during the last 17 years with normal genital sensation and who have been started on CIC for bladder dysfunction were identified and approached for inclusion in this study. We developed a multimodality questionnaire on the use, learning curve and degree of difficulty of CIC. The quality of life study was performed using the PedsQL™ 4.0, developed by Dr. James W. Varni. The PedsQL™ 4.0 is a reliable, validated HRQOL survey developed for pediatric patients with chronic disease, which has separate modules that can be used to assess patient and parental QOL.6, 7 Each QOL questionnaire consists of 21 or 23 age-appropriate questions encompassing the 4 areas of Physical Functioning, Emotional Functioning, Social Functioning and School Functioning. A 5-point response scale is used for patients 8 to 18 years old, whereas a simplified 3-point scale is used for children younger than 8 years to increase ease of use for this age group. Items on these questionnaires are reverse-scored and linearly transformed to a 0 to 100 scale such that higher scores indicate better HRQOL. Questionnaires are available for patients who are old enough to respond themselves (self-report form) and for parents of subjects to respond for them (parent-proxy form). Each patient and/or parents were contacted in person, by mail and/or by telephone and completed these question-
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CATHETERIZATION IN GENITALLY SENSATE CHILDREN
naires. When possible, QOL data were collected from the patient (self-report) and 1 or both parents (parent-proxy report). If the child was too young to complete the PedsQL form, then data were collected for that patient from the parentproxy report. Statistical comparisons were done using a 2-sided t test with p ⱕ0.05 considered significant. Data are reported as mean ⫾ standard deviation. When the concept of the CIC technique as a necessary medical treatment is first introduced, it is beneficial if the family already has a relationship with the nurse, nurse practitioner or other health care professional who will be providing instruction. If this is not the case, then a short period is allowed between the recommendation for CIC and formal instruction in the technique to allow the family and the CIC teacher to build a trusting and therapeutic relationship. This also allows time for the patient and family to adjust emotionally to the decision and be part of the decision-making process. Teaching sessions are scheduled for a time and place that permit uninterrupted instruction in a quiet setting. In addition to the participation of parents, other family members who can provide support or may be asked to perform CIC are encouraged to attend and learn the technique. The patient and family are told that the CIC technique itself is simple, while acknowledging that the emotional impact it carries is difficult. The family is assured that they will soon become the experts in catheterizing their own child. From the beginning of the session, the family understands that catheterizing the child will be accomplished before they leave. First, the instructor gives a verbal review of the technique, using appropriate analogies and/or props to help demonstrate. Concerns about the technique and/or fears of discomfort are discussed and the family is told that instruction will proceed at whatever pace is necessary. Next, the instructor performs CIC on the child after assuring him/her that it will be done slowly and that any discomfort during the procedure will be addressed. Once this has been accomplished, either one or both of the parents perform CIC, followed by any other family members who may wish or need to learn. If the child is old enough to catheterize independently, then he/she will also perform CIC during the appointment. Once all are comfortable with the technique, supplies are either provided to the family at that time or arranged to be delivered to the home. The instructor gives the family multiple contact numbers (ie office, pager, e-mail) so that they have the knowledge and comfort that help and support are always available. A followup telephone call from the instructor within 24 to 48 hours of the appointment is mandatory to answer any questions that may have arisen and to ensure that the family is comfortable with performing CIC in the home setting. For the vast majority of sensate patients additional instruction or appointments have not been necessary. RESULTS
We identified 33 patients with bladder dysfunction but normal genital sensation, of whom 20 were able to be contacted for this study and completed the study instruments. The reasons for starting CIC in these patients are listed in the table. Data for the 12 males and 8 females were collected, including 30 QOL questionnaires (12 self-report, 18 parentproxy). All patients or their parents completed the multimodality questionnaire, which included inquiries about patient demographic information, the level of difficulty to learn CIC and the level of comfort with performance of CIC. Mean patient age at study enrollment was 12.3 ⫾ 6.9 years (range 2.4 to 26.3), and mean age at initiation of CIC was 6.9 ⫾ 4.5 years (range 15 months to 17 years). Mean length of followup on CIC was 5.45 ⫾ 5.37 years (range 6.5 months to 16.5 years), and mean number of catheterizations a day was 4.80 ⫾ 1.52 (range 2 to 8). There were no failures in learning or continuing CIC, although 3 patients stopped CIC
Diagnosis of sensate patients on clean intermittent catheterization Diagnosis Imperforate anus/tethered cord Neurogenic bladder after multiple bladder surgeries Spinal tumor Hinman’s syndrome Neurogenic bladder of unknown etiology Posterior urethral valves Sacral lipoma Lipomeningocele Atonic bladder Ochoa syndrome Total
No. Pts 4 3 3 2 2 2 1 1 1 1 20
either temporarily or permanently on the instructions of their treating physician. Of the patients and/or their parents 80% were able to learn the CIC technique in 1 outpatient clinic visit of an hour or less with the help of a nurse who is experienced in teaching CIC. The remaining 20% required 2 to 3 sessions to learn the technique. On a 10-point scale of difficulty of learning the CIC technique (1—very easy, 10 —very difficult) the mean level of difficulty was 3.55 ⫾ 2.71 for all patients, 3.25 ⫾ 2.57 for boys and 4.00 ⫾ 3.02 for girls. According to the responses on the questionnaire, factors that made learning CIC more challenging included apprehensiveness or uncooperativeness of the patient, discomfort during the procedure or unwillingness of the patient to learn. The main factor that aided in learning the technique was the helpfulness and explanations of the nurse teaching CIC. In regard to patient and/or parent present comfort level with the CIC technique on a 10-point scale of increasing comfort level (1—not comfortable, 10 — very comfortable), the mean comfort level was 9.33 ⫾ 1.10 for all, 9.46 ⫾ 0.84 for boys and 9.12 ⫾ 1.46 for girls. Results from the PedsQL™ 4.0 HRQOL questionnaire showed that the mean QOL score was 83.24 ⫾ 10.32 on the self-report form and 81.78 ⫾ 12.90 on the parent-proxy form. The mean QOL in our study by gender (using self-report and parent-proxy data) was 85.28 ⫾ 9.76 for males and 76.66 ⫾ 13.59 for females. When parent-proxy data were excluded, the QOL by gender for the self-report data was 82.74 ⫾ 10.38 for males and 84.24 ⫾ 11.72 for females. DISCUSSION
When Lapides et al first reported the concept of selfcatheterization, 3 of the 14 patients described had no neurological deficit but had atonic bladders from infrequent voiding.1 The remaining patients had neurogenic bladder due to multiple sclerosis, myelomeningocele, lipoma of the spinal cord or transverse myelitis. Since then, the technique of CIC has become an integral component of the care of patients with the inability to partially or completely empty the bladder. When the clinical situation dictates, the decision to initiate CIC in a patient without genital sensation is usually straightforward. Conversely, the perceived sentiment regarding instituting CIC in genitally sensate individuals, especially children, is that genital discomfort limits its practical use. In 1 study of lower urinary tract dysfunction in children with anorectal malformation Boemers et al found that it was much more difficult for these children and their parents to accept CIC because normal genital sensation caused them pain and/or distress during catheterization attempts.8 They found that those who were started on CIC in the neonatal period or in infancy accepted and were compliant with CIC, but it was nearly impossible to introduce CIC in older children. Of 19 children in whom the use of CIC was deemed desirable for urological care only 7 and their parents (37%) were amenable to it. Our study showed that CIC could
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be successfully started in older children, since the mean age in our series was 6.9 years and the oldest patient started CIC at age 17 years. More recent studies have shown better success with instituting CIC in populations of genitally sensate children but the success rates are much lower than those seen with diseases without genital sensation (eg myelodysplasia, spinal cord injury or sacral agenesis). While the success rates for CIC in patients with myelodysplasia range from 94% to100%, Pohl et al showed a 70% success rate in 23 pediatric patients with high post-void residuals but no identifiable anatomic or neurological abnormality.4 Similarly, Van Savage et al demonstrated a 65% success rate in a group of children with a variety of diagnoses who required CIC for bladder management.5 All failures occurred within 6 months in both studies. Of our 20 patients 80% were able to learn CIC within a 1-hour outpatient clinic visit and at a mean of almost 5.5 years of followup, no patient had stopped catheterizing unless directed to do so by a physician. We believe that our high success rate is due to the heavy involvement of an experienced nurse practitioner who works closely with the patients and their families, and is directly available to assist in the teaching and maintenance of the CIC technique. Establishing a trusting relationship between the nurse practitioner and the family from the start is crucial for reinforcing the importance of maintaining CIC as well as for emotional and technical support. Measuring quality of life in patients has become increasingly important in medicine in general and in pediatrics in particular. As Eiser and Morse state: “Where it is possible to manage but not cure a disease, it is important to determine how far treatment and disease compromise the child’s QOL. In this way, informed judgments can be made about whether or not treatment is appropriate, and, where there is a choice, which choice might be the best option for the child.”9 In urological diseases there is a complex interplay between QOL and the control of evacuation.10 Private matters for children such as incontinence may become public when it limits ability to participate in normal childhood activities such as swimming or playing with other children, which may have a profound effect on QOL. Current and future studies of medical treatments in pediatrics have the responsibility to not only assess therapeutic success, but also the impact on the patient as a whole. Previous large studies using the PedsQL to assess QOL of normal, healthy control children showed a self-report QOL score of 83.00 ⫾ 14.79 and a parent-proxy report score of 87.61 ⫾ 12.33.11 Comparison of the QOL scores of these normal children to our CIC patient self-reported QOL scores showed no significant difference (p ⬎0.99). Parent-proxy results between CIC patients and normals approached statistical difference (p ⫽ 0.055). The mean self-report QOL by gender for normal, healthy children in previous studies was 83.16 ⫾ 13.06 for males and 82.54 ⫾ 13.27 for females.12 Our male and female CIC patient QOL scores were not statistically different from those of healthy controls (males p ⫽ 0.93, females p ⫽ 0.80). We also compared our data to those of other studies of children with chronic conditions such as diabetes and pediatric cancer. In a study of children 5 to 18 years old with type 1 or 2 diabetes and their parents the self-reported QOL was 80.37 ⫾ 12.90 and parent-proxy QOL was 76.56 ⫾ 14.10.13 Although significant differences were found in this study between those with diabetes and normal controls, when the diabetes QOL data were compared to those of genitally sensate children on CIC, there was no significant difference in (self-reported QOL p ⫽ 0.43 or parent-proxy QOL p ⫽ 0.10). When our CIC study group was compared with pediatric oncology patients,14 there were statistically significant differences between self-reported QOL (p ⫽ 0.016) and parentproxy QOL (p ⫽ 0.006).
Previous studies on CIC in genitally sensate children have not specifically focused on QOL. Using a proven HRQOL instrument, we were able to determine that the QOL for these children is not significantly different compared to that previously documented in normal, healthy controls. CIC may prevent incontinence and allow these children to better regulate a voiding schedule such that they perceive themselves as no different from their normal peers who do not have to catheterize. Also, since these children are generally more functional and ambulatory than the majority of genitally insensate children who may also have other neurological impairments, they may be able to participate in a wider range of activities with fewer difficulties so that QOL is better overall than those with more serious neurological conditions. One potential bias in our study is the selection bias inherent in those responding to our questionnaires. Of 33 patients we attempted to enroll 20 (60%) responded and completed both study instruments. It is possible that those who responded were more positive in their attitude toward CIC than those who did not respond to the questionnaires. Also, the questionnaire used to evaluate HRQOL is a general instrument evaluating overall QOL for children with chronic diseases and does not specifically address issues regarding urological conditions or catheterization. There is no such questionnaire available at present but efforts to design one specific to this patient group are under way. Work for the future should include comparison of QOL before and after the institution of CIC in genitally sensate children to longitudinally demonstrate that CIC provides a positive benefit to these children, or at least does not have a substantially negative affect on their QOL. The overall number of patients in this study was smaller than that from previous studies of QOL of normal controls, which makes statistical comparison of these values more difficult to validate. CONCLUSIONS
Clean intermittent catheterization can be taught to genitally sensate children and their parents in a short period with high long-term success rates. The assistance of an individual experienced with the technique, such as a nurse or nurse practitioner, is integral to teach the procedure and reinforce the need to continue. Over time, patients and their families become comfortable with CIC and report quality of life similar to that of normal children. REFERENCES
1. Lapides, J., Diokno, A. C., Silber, S. J. and Lowe, B. S.: Clean, intermittent self-catheterization in the treatment of urinary tract disease. J Urol, 107: 458, 1972 2. Kass, E. J., McHugh, T. and Diokno, A. C.: Intermittent catheterization in children less than 6 years old. J Urol, 121: 792, 1979 3. Plunkett, J. M. and Braren, V.: Clean intermittent catheterization in children. J Urol, 121: 469, 1979 4. Pohl, H. G., Bauer, S. B., Borer, J. G., Diamond, D. A., Kelly, M. D., Grant, R. et al: The outcome of voiding dysfunction managed with clean intermittent catheterization in neurologically and anatomically normal children. BJU Int, 89: 923, 2002 5. Van Savage, J. G., Sackett, C. K., Wilhelm, C. L., Sessions, R. P. and Mesrobian, H. G.: Indications for and outcomes of clean intermittent catheterization in children with normal genital sensation. J Urol, 157: 1866, 1997 6. Varni, J. W., Seid, M. and Rode, C. A.: The PedsQL: measurement model for the pediatric quality of life inventory. Med Care, 37: 126, 1999 7. Varni, J. W., Seid, M. and Kurtin, P. S.: PedsQL 4.0: reliability and validity of the Pediatric Quality of Life Inventory version 4.0 generic core scales in healthy and patient populations. Med Care, 39: 800, 2001 8. Boemers, T. M., de Jong, T. P., van Gool, J. D. and Bax, K. M.: Urologic problems in anorectal malformations. Part 2: func-
CATHETERIZATION IN GENITALLY SENSATE CHILDREN tional urologic sequelae. J Pediatr Surg, 31: 634, 1996 9. Eiser, C. and Morse, R.: The measurement of quality of life in children: past and future perspectives. J Dev Behav Pediatr, 22: 248, 2001 10. Gerharz, E. W., Eiser, C. and Woodhouse, C. R.: Current approaches to assessing the quality of life in children and adolescents. BJU Int, 91: 150, 2003 11. Varni, J. W., Seid, M., Knight, T. S., Uzark, K. and Szer, I. S.: The PedsQL 4.0 Generic Core Scales: sensitivity, responsiveness, and impact on clinical decision-making. J Behav Med, 25: 175, 2002 12. Varni, J. W., Burwinkle, T. M., Seid, M. and Skarr, D.: The PedsQL 4.0 as a pediatric population health measure: feasibility, reliability, and validity. Ambul Pediatr, 3: 329, 2003 13. Varni, J. W., Burwinkle, T. M., Jacobs, J. R., Gottschalk, M., Kaufman, F. and Jones, K. L.: The PedsQL in type 1 and type 2 diabetes: reliability and validity of the Pediatric Quality of Life Inventory Generic Core Scales and type 1 Diabetes Module. Diabetes Care, 26: 631, 2003 14. Varni, J. W., Burwinkle, T. M., Katz, E. R., Meeske, K. and Dickinson, P.: The PedsQL in pediatric cancer: reliability and validity of the Pediatric Quality of Life Inventory Generic Core Scales, Multidimensional Fatigue Scale, and Cancer Module. Cancer, 94: 2090, 2002 EDITORIAL COMMENTS Clean intermittent catheterization was first described in the early 1970s, and since that time it has become a mainstay of management for patients unable to empty the bladder spontaneously. In the pediatric population the majority of these are children with spina bifida, who generally have an insensate bladder and urethra. However, there is a subset of patients, including boys with posterior urethral valves, who have sensation but still need CIC. The common teaching has been that such children are poor candidates for CIC,
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especially if it is begun at a later age. However, as Alpert et al note, there has been a paucity of literature to support this finding. They evaluated quality of life in patients on CIC with a validated instrument, and found it to be comparable to controls. In addition, they evaluated patient discomfort with CIC, and found it to be relatively mild. The authors point out that a key part of the success is the participation of a dedicated clinician, often a nurse practitioner. I think that is one of the most important messages of this article: patients can learn to accept seemingly onerous treatments, provided they receive the help and support of a caring team of clinicians. Barry Duel Urology and Pediatrics University of California, Irvine Orange, California As pediatric urologists, we all have sensate patients in our practice with nonneurogenic bladder dysfunction who require clean intermittent catheterization. Although initially apprehensive, a majority of patients adjust well enough to be compliant with a CIC program. Historical success rates have varied from 37% to 65% (references 4, 5 and 8 in article). Earlier work has reported no change in selfesteem or body image after implementation of a CIC program. However, to date the well-being and ease with which children become proficient in CIC have yet to be evaluated. The authors present a well designed protocol that uses a validated survey instrument to demonstrate no adverse impact on the quality of life for these patients after institution of CIC. Moreover, they highlight the fundamental component, patient education, to achieving durable success with their program. The authors should be applauded for their efforts. C. D. Anthony Herndon Department of Surgery/Urology University of Alabama at Birmingham Birmingham, Alabama
DISCUSSION Dr. William Reiner. When you are designing scales of 1 to 10 for children, you find 4, 7 and 8 are virtually never used. One to 10 is too big a spread for kids, and it is really too big for adults as well. It is much better to use a 1 to 5 scale, which is more reproducible and more understandable for us. Unless you divide it into sectors, such as quartiles, it is difficult for kids to use. However, this was a good study—I liked it. Mr. David Thomas. In your abstract you say that these are patients with normal genital sensation. When I looked at the patient group, you had children with anorectal malformations, who presumably had sacral agenesis, and spinal tumors. Some of these children presumably did have a neuropathic bladder and had reduced sensation. Did you have any children with posterior urethral valves who would have unequivocally normal sensation? Dr. Seth Alpert. We did. If you look at that table again, we had 2 or 3 patients with posterior urethral valves. We also had I think 1 or 2 children with Hinman’s syndrome, and a girl with Ochoa syndrome with no other abnormalities who had unequivocally normal sensation. It is possible that some of our patients had reduced sensation but none had lack of sensation such as those in the spina bifida population.