- Email: [email protected]
Transition of Care to an Adult Spina Bifida Clinic: Patient Perspectives and Medical Outcomes
Transition of Care to an Adult Spina Bifida Clinic: Patient Perspectives and Medical Outcomes Ashley Cox, Lynn Breau, Liette Connor, P. Daniel McNeely, Peter A. Anderson and Dawn L. MacLellan* From the Department of Urology (AC), QEII Health Science Centre, School of Nursing (LB), Division of Neurosurgery (PDM) and Department of Urology (PAA, DLM), Dalhousie University and Department of Urology, Capital Health District Authority (LC), Halifax, Nova Scotia, Canada
Abbreviations and Acronyms ACES ⫽ Ambulatory Care Experience Survey SB ⫽ spina bifida TCS ⫽ Transition of Care Survey TCS-p ⫽ TCS-parent version Study received institutional ethics review board approval. Supplementary material for this article can be obtained at http://urology.medicine.dal.ca/ surveys.htm. * Correspondence: Department of Urology, Dalhousie University, P. O. Box 9700, IWK Health Centre, 5850 University Ave., Halifax, Nova Scotia, B3K 6R8, Canada (telephone: 1-902-470-8943; FAX: 1-902-470-8267; e-mail: Dawn.MacLellan@ iwk.nshealth.ca).
Purpose: Transitioning care of patients with spina bifida to adult centers poses a challenge. We sought patient and parent perspectives on the transition process at our center and correlated these perspectives with medical outcomes. Materials and Methods: Patients who attended the adult spina bifida clinic at our institution were invited to complete SF-36®, the Ambulatory Care Experience Survey and a Transition of Care Survey. Urological and neurosurgical medical outcomes were correlated with health status, clinic experience and perspectives on the transition process. Statistical analysis was done using SPSS®, version 16.0. Results: Of 105 patients with spina bifida 24 participated in the study. SF-36 results showed that the physical health domain correlated with lack of employment (p ⫽ 0.006). Patients whose parents completed the surveys on their behalf were more likely to have physical limitations than the patients who completed surveys (p ⫽ 0.011). Urological and neurosurgical outcomes did not significantly affect SF-36, Ambulatory Care Experience Survey or Transition of Care Survey scores in this patient population. Patients and caregivers identified similar key elements and barriers in the transition process. Satisfaction with care at the pediatric center was higher than at the adult center. Conclusions: Transitioning care of patients with spina bifida from pediatric to adult care poses challenges for the health care system. Medical outcomes do not impact the patient perspective of the transition process. To optimize the transition of care we must address the barriers identified by patients and their caregivers. Key Words: kidney; spinal dysraphism; adult; health care facilities, manpower, and services; urinary tract
1590
www.jurology.com
SPINA bifida is the most common congenital abnormality of the spinal cord. Of children born with myelomeningocele 75% now live into adulthood.1 SB results in multisystem medical problems and limitations in psychosocial function. Urological complications occur in 24% to 94% of adults with SB.1 Multidisciplinary health care teams at pediatric centers are required to
provide care to individuals with SB. At the most successful centers case managers, subspecialist physicians, access to mental health care resources and vocational training are provided.2 Multidisciplinary health care is necessary for adults with SB to prevent adverse outcomes such as renal failure, ventriculoperitoneal shunt mal-
0022-5347/11/1864-1590/0 THE JOURNAL OF UROLOGY® © 2011 by AMERICAN UROLOGICAL ASSOCIATION EDUCATION
Vol. 186, 1590-1594, October 2011 Printed in U.S.A. DOI:10.1016/j.juro.2011.04.011
AND
RESEARCH, INC.
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
function or early death.3 Transitioning care to adult centers has proved to be a challenge.4 Binks et al identified barriers and key elements necessary for a successful transition.5 Barriers include the inability of pediatric health care providers to let go of patients, patient reluctance to leave the familiarity of a pediatric center and a lack of interest by adult health care providers. Key elements include adequate preparation of the patient and caregivers, flexible timing of transition, early introduction to the adult clinic, knowledgeable adult care providers and coordinated care. Across the pediatric, urological and neurosurgical literature there is a paucity of evidence on the transition process for patients with SB. Current data are extrapolated from studies of other populations.5 We acquired patient and caregiver perspectives on the transition experience at our center. We also determined how medical outcomes impact the global wellbeing of adults with SB, their experience at the adult clinic and their satisfaction with the transition process.
1591
we calculated frequencies. MANOVA was used to explore the SF-36 subscales between patients who completed SF-36 and those whose parents completed it. An independence score was determined based on the patient living alone, being employed and ambulating independently. A series of MANCOVAs was used to determine whether patient independence was related to SF-36 subscales and TCS: patient version scores. A series of MANCOVAs was used to determine whether specific urological and neurological outcomes were related to SF-36, ACES and patient transition questionnaire scores. The urological outcomes examined were upper or lower urinary tract stones, urinary tract infection within the last year, need for another urological operation, need for clean intermittent catheterization, hospital admission for a nonurological event and urinary incontinence. The neurosurgical outcomes examined included a ventriculoperitoneal shunt, need for shunt revision in adulthood, neuropathic pain syndrome or seizures and the need for a detethering procedure. For each MANCOVA each outcome was examined independently by including the other factors as covariates. Nephrological outcomes were recorded but extensive analysis was not performed.
RESULTS MATERIALS AND METHODS Patients who attended the adult SB clinic at our institution were identified for study participation. From June 2009 to July 2010 patients were invited to complete 3 telephone surveys, including SF-36, ACES and TCS-patient version. Patients had the option to share the invitation with their parent/caregiver, who was also invited to complete TCS-p. Parents were invited to complete 3 surveys (SF-36, ACES and TCS-p) on behalf of the patient if the patient was unable to complete the surveys independently due to disability. Consent was obtained via mail, telephone contact was made and surveys were done by a research associate. SF-36 was used as a generic measurement of functional health and well-being.6 ACES was used to evaluate the patient experience with health care professionals at the adult SB clinic.7 The 2 surveys have been validated (http:// www.sf-36.org/tools/sf36.shtml and http://160.109.101. 132/icrhps/resprog/thi/aces_publist.asp, respectively). TCS was created for the purpose of this study and has not been validated. Two versions of this survey were created to investigate the patient and caregiver perspectives, respectively. Each survey consisted of 22 questions derived from the transitioning care barriers and key elements identified by Binks et al.5 Urological, nephrological and neurosurgical medical outcomes were recorded retrospectively. The institutional ethics review board provided approval for this study. Data were analyzed with SPSS, version 16.0. All tests were 2-tailed with ␣ ⫽ 0.05. For all MANOVAs and MANCOVAs Wilks’ was used to judge significance. Subscales were calculated for the SF-36 survey and ACES according to instructions in the manuals. Descriptive statistics (mean ⫾ SD) were calculated for items on all questionnaires, for questionnaire subscales, and for demographic and medical characteristics. For categorical data
A total of 105 patients were contacted, of whom 24 responded for a 22.8% response rate (see figure). Nine participants did not complete the TCS-patient version since they did not attend the pediatric SB clinic at our center before attending the adult SB clinic. One patient did not complete ACES since it had been more than 12 months since the participant attended the adult clinic. Table 1 shows patient characteristics. Table 2 lists nephrological, urological and neurosurgical outcomes. No patient was on dialysis or underwent transplantation or augmentation revision. SF-36 subscales include physical health, pain, general health, vitality, social, emotional health and mental health. We examined whether patient independence affected the SF-36 subscales. Patients who were not employed and those whose parents completed the surveys on their behalf scored lower in the physical role subscale than patients who were employed or who completed the surveys (p ⫽ 0.011 and 0.006, respectively). Decreased mobility was associated with a trend toward decreased general health and vitality (p ⫽ 0.083 and 0.045, respectively) but it did not affect the SF-36 physical role. Patients who lived alone trended toward decreased emotional well-being and mental health (p ⫽ 0.074 and 0.083, respectively). Urological outcomes did not significantly affect SF-36 scores. There was an association between hospital admission for nongenitourinary reasons and a low score on the vitality subscale (p ⫽ 0.051). Neurosurgical outcomes did not significantly affect SF-36 scores. Patients with a neuropathic pain syn-
1592
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
Survey responses
drome trended toward a decreased general health subscale score (p ⫽ 0.072). The ACES domains analyzed included quality of physician-patient interactions, shared decision making, coordination of care and access. Patients with urinary tract infections rated coordination of care higher than patients without urinary tract infections (p ⫽ 0.058). Patients who underwent detethering procedures in adulthood rated the quality of physician-patient interactions and coordination of care poorly (p ⫽ 0.073 and 0.061, respectively) compared to patients who did not undergo these procedures. The mean age of patients transitioned to the adult spina bifida clinic was 26.8 years (range 19 to 66). Table 1. Patient demographics and characteristics Independence Measures Living: Independent Parent/group home Ambulation: Wheelchair dependent Aid Independent Employment: Yes No Education: High school Community college University Lesion type: Myelomeningocele Lipomeningocele Sacral agenesis Lesion level: Thoracic Thoracolumbar Lumbar Lumbosacral Sacral Unknown
We assessed whether patient satisfaction correlated with markers of independence. Living alone, having good mobility or being employed did not statistically significantly impact satisfaction with the transition process. There were also no urological or neurosurgical outcomes that statistically significantly affected patient satisfaction with the transition of care. TCS-patient version and TCS-p were derived based on barriers and key elements of the transition process identified by Binks et al.5 Patients and parents reported similar barriers and key elements in the transition process. Each group thought that the adult clinic did not address financial/employment issues and each identified not visiting the adult clinic before transition as a barrier. Patients also wanted to make more decisions regarding care. Patients did not think that their parents were unable
No. Pts (%) 7 (29.2) 17 (70.8) 15 (62.5) 4 (16.7) 5 (20.8) 8 (33.3) 16 (67.7) 12 (50) 6 (25) 6 (25) 20 (83.3) 3 (12.5) 1 (4.2) 3 (12.5) 6 (25) 7 (29.2) 3 (12.5) 1 (4.2) 4 (16.7)
Table 2. Medical outcomes of patients attending adult SB clinic No. Pts (%) Nephrological: Increased creatinine Hypertension Nephrology referral Urological: Hydronephrosis Lower tract stone Upper tract stone Urinary tract infection Urological procedure Bladder augmentation Incontinence Clean intermittent catheterization Anticholinergics Erectile dysfunction Neurosurgical: Ventriculoperitoneal shunt Shunt revision Detethering procedure Seizure Neuropathic pain syndrome
3 (12.5) 3 (12.5) 2 (8.3) 3 (12.5) 2 (8.3) 2 (8.3) 17 (70.8) 5 (20.8) 1 (4.2) 11 (45.8) 19 (79.2) 12 (50) 2 (8.3) 17 (70.8) 13 (54.2) 2 (8.3) 2 (8.3) 2 (8.3)
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
to give up control of their care. Each group thought that pediatric caregivers appropriately let go of care, adult caregivers were knowledgeable, and personnel at the adult and pediatric centers communicated well. They thought that the transition was flexible and coordinated. Patients and parents were asked to rate the care provided at the pediatric SB clinic and at the adult SB clinic. Patient and parent perspectives were comparable. Mean patient scores for the pediatric and adult clinics was 9.31 ⫾ 1.251 and 7.73 ⫾ 2.127 (p ⫽ 0.057), and mean parent scores were 9.70 ⫾ 0.675 and 7.70 ⫾ 2.869 (p ⫽ 0.060), respectively.
DISCUSSION Based on a mean score of 7.73/10 and 7.70/10, respectively, patients and parents were satisfied with the adult SB clinic. However, each group considered it inferior to the pediatric SB clinic. Patients and parents shared the same view of the transition process at our center. Our study included 4 TCS-p questionnaires that were completed on behalf of the patient by a parent. Several reports of transition medicine show that patients and parents share similar views of the transition process.8 –10 The small proportion of surveys completed by parents may limit the applicability of our study. However, a study goal was to address the parent perspective. Our study results confirm that the transition process at our institution meets several key elements. However, certain barriers exist. Therefore, changes could be made to improve the transition process. Two important changes would be to give patients the opportunity to attend the adult clinic before the final transition and allow patients to make more decisions. Patients thought that our clinic failed to address financial and employment issues. Although this was not a principle identified by Binks et al,5 employment or the lack thereof was significantly related to decreased physical capabilities in our analysis of SF-36. Adult patients with SB seek guidance on work, education, social provision and sexual counseling.11 Services provided by the most successful transition programs include case management, subspecialist physicians, mental health care, career/ vocational training and health education.12 Our results contribute more evidence to the body of literature highlighting the importance of providing multiple resources for adult patients with SB. Many key elements were met in the transition process at our center. This may reflect the fact that at our institution the pediatric urologists and neurosurgeons who provide care in the adult setting are the same physicians who work at the pediatric SB
1593
clinic. Clinics that combine pediatric and adult health care teams lead to better communication and patient confidence, which improves the transition process.3 This appears to be an ideal situation for transitioning care but it is unlikely to be feasible at most institutions. This could contribute to the high level of satisfaction reported and, thus, our findings may not be applicable to other centers. The medical outcomes of study participants differed from previously studied adult SB populations. Between 1990 and 1998 McDonnell and McCann reviewed the records of 237 adult patients with SB and found that 48% had renal dysfunction, 15% required antihypertensive medication, ventricular shunts were present in 37%, clean intermittent catheterization was performed by 28% and 21.8% had undergone urinary diversion.13 The high rate of VP shunts (70.8%) and shunt revisions (54.2%) in our population may have contributed to protect bladder and renal function in our study.14 Notably our study population included 3 patients with lipomyelomeningocele and 1 with sacral agenesis, who may have better mobility and less neurosurgical issues than patients with myelomeningocele. This may have skewed our results. However, evidence suggests that sacral agenesis may contribute to worsening urological health compared to myelomeningocele.15 Including these patients may prevent our findings from being generalized to a truly homogenous myelomeningocele population but this study is representative of a transitioned population at an adult SB clinic. SF-36 and ACES results were not significantly impacted by urological and neurosurgical outcomes. Similar studies have shown that medical outcomes may not correlate with quality of life issues.16 Medical outcomes in our patient population may reflect selection bias with patients who participated in the study being healthier and, thus, more satisfied with the transition process. However, it may reflect an overall improvement in care during the last 1 to 2 decades. The most common cause of death in adults with SB is related to shunt malfunction or renal failure.14 The risk of neurosurgical and urological deterioration remains constant throughout adulthood, which warrants close followup of these patients. The response rate to our study was low at 22.8%. This is consistent with similar studies of adults with cystic fibrosis17 and reflects the difficulty of studying a young patient population with chronic illness. Our recruitment process contributed to the low response rate since multiple steps were required to contact patients and complete the surveys. Future studies may yield higher response rates if interviews and surveys were completed at clinic appointments.
1594
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
CONCLUSIONS Our study shows that the general health of adults with SB, their experience at the adult SB clinic and their perspective on the transition process were indepen-
dent of urological and neurosurgical medical outcomes. Patients and their caregivers preferred the care received at the pediatric center but remained satisfied with the adult SB clinic at our institution.
REFERENCES 1. Dicianno BE, Kurowski BG, Yang JM et al: Rehabilitation and medical management of the adult with spina bifida. Am J Phys Med Rehabil 2008; 87: 1027.
7. Rodriguez HP, Rogers WH, Marshall RE et al: The effects of primary care physician visit continuity on patients’ experiences with care. J Gen Intern Med 2007; 22: 787.
13. McDonnell GV and McCann JP: Issues of medical management in adults with spina bifida. Child Nerv Syst 2000; 16: 222.
2. Scal P, Evans T, Blozis S et al: Trends in transition from pediatric to adult health care services for young adults with chronic conditions. J Adolesc Health 1999; 24: 259.
8. Rutishauser C, Akré C and Surìs JC: Transition from pediatric to adult health care: expectations of adolescents with chronic disorders and their parents. Eur J Pediatr 2011; 170: 865.
14. Bowman RH, McLone DG, Grant JA et al: Spina bifida outcome: a 25-year prospective. Pediatr Neurosurg 2001; 34: 114.
3. Kennedy A, Sloman F, Douglass JA et al: Young people with chronic illness: the approach to transition. Intern Med J 2007; 37: 555.
9. Boyle MP, Farukhi Z and Nosky ML: Strategies for improving transition of adult cystic fibrosis care, based on patient and parent views. Pediatr Pulmonol 2001; 32: 428.
4. Steinbeck KS, Brodie L and Towns SJ: Transition in chronic illness: who is going where? J Paediatr Child Health 2008; 44: 478. 5. Binks JA, Barden WS, Burke TA et al: What do we really know about the transition to adultcentered health care? A focus on cerebral palsy and spina bifida. Arch Phys Med Rehabil 2007; 88: 1064. 6. Ware JE Jr: SF-36 health survey update. Spine (Phila Pa 1976) 2000; 25: 3130.
10. Wong LHL, Chan FWK, Wong FYY et al: Transition of care for adolescents and families with chronic illness. J Adolesc Health 2010; 47: 540. 11. Stellman-Ward G, Bannister CM and Lewis M: Assessing the needs of the adult with spina bifida. Eur J Pediatr Surg, suppl., 1993; 14: 14. 12. Mennito SH and Clark JK: Transition medicine: a review of current theory and practice. South Med J 2010; 103: 339.
15. Torre M, Buffa P, Jasonni V et al: Long-term urologic outcome in patients with caudal regression syndrome, compared with meningomyelocele and spinal cord lipoma. J Pediatr Surg 2008; 43: 530. 16. Bellin MH, Zabel TA, Dicianno BE et al: Correlates of depressive and anxiety symptoms in young adults with spina bifida. J Pediatr Psych 2010; 35: 778. 17. Anderson DL, Flume PA, Hardy KK et al: Transition Programs in Cystic Fibrosis Centers: Perception of Patients. Pediatr Pulmonol 2002; 33: 327.
Abbreviations and Acronyms ACES ⫽ Ambulatory Care Experience Survey SB ⫽ spina bifida TCS ⫽ Transition of Care Survey TCS-p ⫽ TCS-parent version Study received institutional ethics review board approval. Supplementary material for this article can be obtained at http://urology.medicine.dal.ca/ surveys.htm. * Correspondence: Department of Urology, Dalhousie University, P. O. Box 9700, IWK Health Centre, 5850 University Ave., Halifax, Nova Scotia, B3K 6R8, Canada (telephone: 1-902-470-8943; FAX: 1-902-470-8267; e-mail: Dawn.MacLellan@ iwk.nshealth.ca).
Purpose: Transitioning care of patients with spina bifida to adult centers poses a challenge. We sought patient and parent perspectives on the transition process at our center and correlated these perspectives with medical outcomes. Materials and Methods: Patients who attended the adult spina bifida clinic at our institution were invited to complete SF-36®, the Ambulatory Care Experience Survey and a Transition of Care Survey. Urological and neurosurgical medical outcomes were correlated with health status, clinic experience and perspectives on the transition process. Statistical analysis was done using SPSS®, version 16.0. Results: Of 105 patients with spina bifida 24 participated in the study. SF-36 results showed that the physical health domain correlated with lack of employment (p ⫽ 0.006). Patients whose parents completed the surveys on their behalf were more likely to have physical limitations than the patients who completed surveys (p ⫽ 0.011). Urological and neurosurgical outcomes did not significantly affect SF-36, Ambulatory Care Experience Survey or Transition of Care Survey scores in this patient population. Patients and caregivers identified similar key elements and barriers in the transition process. Satisfaction with care at the pediatric center was higher than at the adult center. Conclusions: Transitioning care of patients with spina bifida from pediatric to adult care poses challenges for the health care system. Medical outcomes do not impact the patient perspective of the transition process. To optimize the transition of care we must address the barriers identified by patients and their caregivers. Key Words: kidney; spinal dysraphism; adult; health care facilities, manpower, and services; urinary tract
1590
www.jurology.com
SPINA bifida is the most common congenital abnormality of the spinal cord. Of children born with myelomeningocele 75% now live into adulthood.1 SB results in multisystem medical problems and limitations in psychosocial function. Urological complications occur in 24% to 94% of adults with SB.1 Multidisciplinary health care teams at pediatric centers are required to
provide care to individuals with SB. At the most successful centers case managers, subspecialist physicians, access to mental health care resources and vocational training are provided.2 Multidisciplinary health care is necessary for adults with SB to prevent adverse outcomes such as renal failure, ventriculoperitoneal shunt mal-
0022-5347/11/1864-1590/0 THE JOURNAL OF UROLOGY® © 2011 by AMERICAN UROLOGICAL ASSOCIATION EDUCATION
Vol. 186, 1590-1594, October 2011 Printed in U.S.A. DOI:10.1016/j.juro.2011.04.011
AND
RESEARCH, INC.
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
function or early death.3 Transitioning care to adult centers has proved to be a challenge.4 Binks et al identified barriers and key elements necessary for a successful transition.5 Barriers include the inability of pediatric health care providers to let go of patients, patient reluctance to leave the familiarity of a pediatric center and a lack of interest by adult health care providers. Key elements include adequate preparation of the patient and caregivers, flexible timing of transition, early introduction to the adult clinic, knowledgeable adult care providers and coordinated care. Across the pediatric, urological and neurosurgical literature there is a paucity of evidence on the transition process for patients with SB. Current data are extrapolated from studies of other populations.5 We acquired patient and caregiver perspectives on the transition experience at our center. We also determined how medical outcomes impact the global wellbeing of adults with SB, their experience at the adult clinic and their satisfaction with the transition process.
1591
we calculated frequencies. MANOVA was used to explore the SF-36 subscales between patients who completed SF-36 and those whose parents completed it. An independence score was determined based on the patient living alone, being employed and ambulating independently. A series of MANCOVAs was used to determine whether patient independence was related to SF-36 subscales and TCS: patient version scores. A series of MANCOVAs was used to determine whether specific urological and neurological outcomes were related to SF-36, ACES and patient transition questionnaire scores. The urological outcomes examined were upper or lower urinary tract stones, urinary tract infection within the last year, need for another urological operation, need for clean intermittent catheterization, hospital admission for a nonurological event and urinary incontinence. The neurosurgical outcomes examined included a ventriculoperitoneal shunt, need for shunt revision in adulthood, neuropathic pain syndrome or seizures and the need for a detethering procedure. For each MANCOVA each outcome was examined independently by including the other factors as covariates. Nephrological outcomes were recorded but extensive analysis was not performed.
RESULTS MATERIALS AND METHODS Patients who attended the adult SB clinic at our institution were identified for study participation. From June 2009 to July 2010 patients were invited to complete 3 telephone surveys, including SF-36, ACES and TCS-patient version. Patients had the option to share the invitation with their parent/caregiver, who was also invited to complete TCS-p. Parents were invited to complete 3 surveys (SF-36, ACES and TCS-p) on behalf of the patient if the patient was unable to complete the surveys independently due to disability. Consent was obtained via mail, telephone contact was made and surveys were done by a research associate. SF-36 was used as a generic measurement of functional health and well-being.6 ACES was used to evaluate the patient experience with health care professionals at the adult SB clinic.7 The 2 surveys have been validated (http:// www.sf-36.org/tools/sf36.shtml and http://160.109.101. 132/icrhps/resprog/thi/aces_publist.asp, respectively). TCS was created for the purpose of this study and has not been validated. Two versions of this survey were created to investigate the patient and caregiver perspectives, respectively. Each survey consisted of 22 questions derived from the transitioning care barriers and key elements identified by Binks et al.5 Urological, nephrological and neurosurgical medical outcomes were recorded retrospectively. The institutional ethics review board provided approval for this study. Data were analyzed with SPSS, version 16.0. All tests were 2-tailed with ␣ ⫽ 0.05. For all MANOVAs and MANCOVAs Wilks’ was used to judge significance. Subscales were calculated for the SF-36 survey and ACES according to instructions in the manuals. Descriptive statistics (mean ⫾ SD) were calculated for items on all questionnaires, for questionnaire subscales, and for demographic and medical characteristics. For categorical data
A total of 105 patients were contacted, of whom 24 responded for a 22.8% response rate (see figure). Nine participants did not complete the TCS-patient version since they did not attend the pediatric SB clinic at our center before attending the adult SB clinic. One patient did not complete ACES since it had been more than 12 months since the participant attended the adult clinic. Table 1 shows patient characteristics. Table 2 lists nephrological, urological and neurosurgical outcomes. No patient was on dialysis or underwent transplantation or augmentation revision. SF-36 subscales include physical health, pain, general health, vitality, social, emotional health and mental health. We examined whether patient independence affected the SF-36 subscales. Patients who were not employed and those whose parents completed the surveys on their behalf scored lower in the physical role subscale than patients who were employed or who completed the surveys (p ⫽ 0.011 and 0.006, respectively). Decreased mobility was associated with a trend toward decreased general health and vitality (p ⫽ 0.083 and 0.045, respectively) but it did not affect the SF-36 physical role. Patients who lived alone trended toward decreased emotional well-being and mental health (p ⫽ 0.074 and 0.083, respectively). Urological outcomes did not significantly affect SF-36 scores. There was an association between hospital admission for nongenitourinary reasons and a low score on the vitality subscale (p ⫽ 0.051). Neurosurgical outcomes did not significantly affect SF-36 scores. Patients with a neuropathic pain syn-
1592
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
Survey responses
drome trended toward a decreased general health subscale score (p ⫽ 0.072). The ACES domains analyzed included quality of physician-patient interactions, shared decision making, coordination of care and access. Patients with urinary tract infections rated coordination of care higher than patients without urinary tract infections (p ⫽ 0.058). Patients who underwent detethering procedures in adulthood rated the quality of physician-patient interactions and coordination of care poorly (p ⫽ 0.073 and 0.061, respectively) compared to patients who did not undergo these procedures. The mean age of patients transitioned to the adult spina bifida clinic was 26.8 years (range 19 to 66). Table 1. Patient demographics and characteristics Independence Measures Living: Independent Parent/group home Ambulation: Wheelchair dependent Aid Independent Employment: Yes No Education: High school Community college University Lesion type: Myelomeningocele Lipomeningocele Sacral agenesis Lesion level: Thoracic Thoracolumbar Lumbar Lumbosacral Sacral Unknown
We assessed whether patient satisfaction correlated with markers of independence. Living alone, having good mobility or being employed did not statistically significantly impact satisfaction with the transition process. There were also no urological or neurosurgical outcomes that statistically significantly affected patient satisfaction with the transition of care. TCS-patient version and TCS-p were derived based on barriers and key elements of the transition process identified by Binks et al.5 Patients and parents reported similar barriers and key elements in the transition process. Each group thought that the adult clinic did not address financial/employment issues and each identified not visiting the adult clinic before transition as a barrier. Patients also wanted to make more decisions regarding care. Patients did not think that their parents were unable
No. Pts (%) 7 (29.2) 17 (70.8) 15 (62.5) 4 (16.7) 5 (20.8) 8 (33.3) 16 (67.7) 12 (50) 6 (25) 6 (25) 20 (83.3) 3 (12.5) 1 (4.2) 3 (12.5) 6 (25) 7 (29.2) 3 (12.5) 1 (4.2) 4 (16.7)
Table 2. Medical outcomes of patients attending adult SB clinic No. Pts (%) Nephrological: Increased creatinine Hypertension Nephrology referral Urological: Hydronephrosis Lower tract stone Upper tract stone Urinary tract infection Urological procedure Bladder augmentation Incontinence Clean intermittent catheterization Anticholinergics Erectile dysfunction Neurosurgical: Ventriculoperitoneal shunt Shunt revision Detethering procedure Seizure Neuropathic pain syndrome
3 (12.5) 3 (12.5) 2 (8.3) 3 (12.5) 2 (8.3) 2 (8.3) 17 (70.8) 5 (20.8) 1 (4.2) 11 (45.8) 19 (79.2) 12 (50) 2 (8.3) 17 (70.8) 13 (54.2) 2 (8.3) 2 (8.3) 2 (8.3)
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
to give up control of their care. Each group thought that pediatric caregivers appropriately let go of care, adult caregivers were knowledgeable, and personnel at the adult and pediatric centers communicated well. They thought that the transition was flexible and coordinated. Patients and parents were asked to rate the care provided at the pediatric SB clinic and at the adult SB clinic. Patient and parent perspectives were comparable. Mean patient scores for the pediatric and adult clinics was 9.31 ⫾ 1.251 and 7.73 ⫾ 2.127 (p ⫽ 0.057), and mean parent scores were 9.70 ⫾ 0.675 and 7.70 ⫾ 2.869 (p ⫽ 0.060), respectively.
DISCUSSION Based on a mean score of 7.73/10 and 7.70/10, respectively, patients and parents were satisfied with the adult SB clinic. However, each group considered it inferior to the pediatric SB clinic. Patients and parents shared the same view of the transition process at our center. Our study included 4 TCS-p questionnaires that were completed on behalf of the patient by a parent. Several reports of transition medicine show that patients and parents share similar views of the transition process.8 –10 The small proportion of surveys completed by parents may limit the applicability of our study. However, a study goal was to address the parent perspective. Our study results confirm that the transition process at our institution meets several key elements. However, certain barriers exist. Therefore, changes could be made to improve the transition process. Two important changes would be to give patients the opportunity to attend the adult clinic before the final transition and allow patients to make more decisions. Patients thought that our clinic failed to address financial and employment issues. Although this was not a principle identified by Binks et al,5 employment or the lack thereof was significantly related to decreased physical capabilities in our analysis of SF-36. Adult patients with SB seek guidance on work, education, social provision and sexual counseling.11 Services provided by the most successful transition programs include case management, subspecialist physicians, mental health care, career/ vocational training and health education.12 Our results contribute more evidence to the body of literature highlighting the importance of providing multiple resources for adult patients with SB. Many key elements were met in the transition process at our center. This may reflect the fact that at our institution the pediatric urologists and neurosurgeons who provide care in the adult setting are the same physicians who work at the pediatric SB
1593
clinic. Clinics that combine pediatric and adult health care teams lead to better communication and patient confidence, which improves the transition process.3 This appears to be an ideal situation for transitioning care but it is unlikely to be feasible at most institutions. This could contribute to the high level of satisfaction reported and, thus, our findings may not be applicable to other centers. The medical outcomes of study participants differed from previously studied adult SB populations. Between 1990 and 1998 McDonnell and McCann reviewed the records of 237 adult patients with SB and found that 48% had renal dysfunction, 15% required antihypertensive medication, ventricular shunts were present in 37%, clean intermittent catheterization was performed by 28% and 21.8% had undergone urinary diversion.13 The high rate of VP shunts (70.8%) and shunt revisions (54.2%) in our population may have contributed to protect bladder and renal function in our study.14 Notably our study population included 3 patients with lipomyelomeningocele and 1 with sacral agenesis, who may have better mobility and less neurosurgical issues than patients with myelomeningocele. This may have skewed our results. However, evidence suggests that sacral agenesis may contribute to worsening urological health compared to myelomeningocele.15 Including these patients may prevent our findings from being generalized to a truly homogenous myelomeningocele population but this study is representative of a transitioned population at an adult SB clinic. SF-36 and ACES results were not significantly impacted by urological and neurosurgical outcomes. Similar studies have shown that medical outcomes may not correlate with quality of life issues.16 Medical outcomes in our patient population may reflect selection bias with patients who participated in the study being healthier and, thus, more satisfied with the transition process. However, it may reflect an overall improvement in care during the last 1 to 2 decades. The most common cause of death in adults with SB is related to shunt malfunction or renal failure.14 The risk of neurosurgical and urological deterioration remains constant throughout adulthood, which warrants close followup of these patients. The response rate to our study was low at 22.8%. This is consistent with similar studies of adults with cystic fibrosis17 and reflects the difficulty of studying a young patient population with chronic illness. Our recruitment process contributed to the low response rate since multiple steps were required to contact patients and complete the surveys. Future studies may yield higher response rates if interviews and surveys were completed at clinic appointments.
1594
TRANSITION OF CARE TO ADULT SPINA BIFIDA CLINIC
CONCLUSIONS Our study shows that the general health of adults with SB, their experience at the adult SB clinic and their perspective on the transition process were indepen-
dent of urological and neurosurgical medical outcomes. Patients and their caregivers preferred the care received at the pediatric center but remained satisfied with the adult SB clinic at our institution.
REFERENCES 1. Dicianno BE, Kurowski BG, Yang JM et al: Rehabilitation and medical management of the adult with spina bifida. Am J Phys Med Rehabil 2008; 87: 1027.
7. Rodriguez HP, Rogers WH, Marshall RE et al: The effects of primary care physician visit continuity on patients’ experiences with care. J Gen Intern Med 2007; 22: 787.
13. McDonnell GV and McCann JP: Issues of medical management in adults with spina bifida. Child Nerv Syst 2000; 16: 222.
2. Scal P, Evans T, Blozis S et al: Trends in transition from pediatric to adult health care services for young adults with chronic conditions. J Adolesc Health 1999; 24: 259.
8. Rutishauser C, Akré C and Surìs JC: Transition from pediatric to adult health care: expectations of adolescents with chronic disorders and their parents. Eur J Pediatr 2011; 170: 865.
14. Bowman RH, McLone DG, Grant JA et al: Spina bifida outcome: a 25-year prospective. Pediatr Neurosurg 2001; 34: 114.
3. Kennedy A, Sloman F, Douglass JA et al: Young people with chronic illness: the approach to transition. Intern Med J 2007; 37: 555.
9. Boyle MP, Farukhi Z and Nosky ML: Strategies for improving transition of adult cystic fibrosis care, based on patient and parent views. Pediatr Pulmonol 2001; 32: 428.
4. Steinbeck KS, Brodie L and Towns SJ: Transition in chronic illness: who is going where? J Paediatr Child Health 2008; 44: 478. 5. Binks JA, Barden WS, Burke TA et al: What do we really know about the transition to adultcentered health care? A focus on cerebral palsy and spina bifida. Arch Phys Med Rehabil 2007; 88: 1064. 6. Ware JE Jr: SF-36 health survey update. Spine (Phila Pa 1976) 2000; 25: 3130.
10. Wong LHL, Chan FWK, Wong FYY et al: Transition of care for adolescents and families with chronic illness. J Adolesc Health 2010; 47: 540. 11. Stellman-Ward G, Bannister CM and Lewis M: Assessing the needs of the adult with spina bifida. Eur J Pediatr Surg, suppl., 1993; 14: 14. 12. Mennito SH and Clark JK: Transition medicine: a review of current theory and practice. South Med J 2010; 103: 339.
15. Torre M, Buffa P, Jasonni V et al: Long-term urologic outcome in patients with caudal regression syndrome, compared with meningomyelocele and spinal cord lipoma. J Pediatr Surg 2008; 43: 530. 16. Bellin MH, Zabel TA, Dicianno BE et al: Correlates of depressive and anxiety symptoms in young adults with spina bifida. J Pediatr Psych 2010; 35: 778. 17. Anderson DL, Flume PA, Hardy KK et al: Transition Programs in Cystic Fibrosis Centers: Perception of Patients. Pediatr Pulmonol 2002; 33: 327.