Spina bifida occulta in functional enuresis
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《美国医学杂志》
Department of Pediatrics, Kalawati Saran Children's Hospital, Lady Hardinge Medical College, New Delhi, India
Abstract
OBJECTIVE: To study incidence of spina bifida occulta in nocturnal enuresis cases and to compare outcome of enuresis with spina bifida occulta and enuresis without spina bifida occulta. METHODS: Patients with enuresis divided into two groups based on X-ray lumbosacral spine. Outcome of these patients were compared on behavioral therapy. RESULTS: Spina bifida occulta was detected in 18 out of total 48 patients. Levels of spina bifida were L5 vertebrae in 3,L5-S1 in 5,S1 in 8 and S1-S2 in 2 patients. Outcome was evaluated in 42 patients who were followed up for more than 6 months. There was no significant difference between both the groups. CONCLUSION: Spina bifida occulta is a common finding in enuresis. Outcome of patients with spina bifida occulta is not different than the patients without spina bifida occulta.
Keywords: Spina bifida occulta; enuresis
Enuresis is a common problem seen in children and adolescents. Functional enuresis is defined as urination in the clothing or bed-wetting beyond the age when children should be toilet trained and in the absence of neurological abnormality[1]. Approximately 15-20% of 5 years olds wet their beds.[2] Nearly 15% of these children spontaneously achieve control each year so that after the age of 15 years only 1 % of the children have enuresis[3].
Enuresis is best viewed as a symptom rather than a disease. It is a manifestation that can be caused by or affected by a variety of factors including developmental delay, abnormal sleep patterns or organic factors such as urinary tract infection (UTI).[4]
Spina bifida occulta is a congenital spinal anomaly, which occurs due to failure of fusion of the posterior arches of the lumbosacral spine. Spina bifida occulta has been linked with a wide spectrum of urodynamic abnormalities including upper and lower motor neuron types of bladder and urethral dysfunction.[5]
Few reports have suggested that enuretic children have higher incidence of spina bifida occulta[6],[7],[8] and these enuretics with spina bifida occulta may have a worse clinical outcome. Khoury et el identified a group of 31 diurnal enuretic children with spina bifida occulta who failed to respond to conservative measures, and they reported resolution of the incontinence in 70% of them after dividing the filum terminale.[6]
The modalities of treatment presently available include tricyclic antidepressant, desmopressin, behavioral therapy, alarm system, acupuncture etc. Out of all available modalities behavioral modification methods are most successful in permanently curing nocturnal enuresis.
In the present study, an attempt has been made to study incidence of spina bifida occulta in enuretic children and clinical outcome of these enuretic with spina bifida occulta were compared with enuretics without spina bifida, managed with behavioral modification therapy.
Materials and methods
This prospective observational study was conducted in the Neurology clinic of a pediatric tertiary hospital in Delhi. All children who came with the complaints of bed-wetting and were found to be monosymptomatic, constituted the study material.
DSM IV definition of enuresis was used for defining a case of enuresis[9] i.e.
Repeated involuntary or intentional voiding during the day or night in clothes or bed.
At least 2 such events per week for at least 3 consecutive months or the presence of clinically significant distress or impairment in social, academic or other important areas of functioning.
Chronological age of at least 5 years or equivalent age
Detailed history of each child fulfilling above-mentioned criteria was recorded in a preformed proforma and complete neurological examination was done. Lumbosacral spine was examined to look for nevus, dimple, gluteal folds asymmetry or tuft of hairs. Abdomen was palpated for distended bladder, kidneys and fecoliths.
All patients underwent urine analysis & culture, stool examinations for ova and cysts and X-ray lumbosacral spine-AP and lateral view for spina bifida occulta. Ultrasound abdomen was also done in each case for any renal/bladder wall changes and residual urine after voiding.
Based on X-ray lumbosacral spine patients were divided into two groups
Group-I=Enuretics with spina bifida occulta
Group-II= Enuretics with normal spine
A child psychologist counseled children, parents, and treatment options were explained to them. Parents and children who gave consent for behavioral modification trial for at least 6 months were taken for further study.
The children were encouraged to void after awakening, after every 2 hours, and then before going to bed. The children were instructed not to drink excess of water with dinner and to withhold fluids 2 hours before going to bed. Children and parents were asked to maintain voiding charts and parents were asked to give rewards for dry nights. Children were followed up monthly in the neurology clinic. At each visit, no of events of voiding, compliance were recorded. Outcome of both groups were evaluated as per the following definitions[10].
Complete cure (responder) - as a reduction in wet nights of at least 90 %
Improvement or partial responder as a reduction in wet nights of >50%but <90%
Data was analyzed by using chi-square test to compare both the groups.
Results
A total of 48 patients who came with complaints of bed-wetting (night, day or both) and gave consent for the trial constituted material for the present study. There were 22 males and 26 were females. Age ranged from five and half years to 14 years. 38 patients had only nocturnal enuresis while 10 patients had history of bed-wetting during daytime also when they fell asleep. None of the patients had urgency, urge incontinence or hesitancy of urine. Positive family history was observed in 8 patients and usual time of bed-wetting was 2-3 hrs after going to bed. 5 patients complained bed-wetting more than once in single night.
Spina bifida occulta was detected in 18 out of total 48 patients. Level of spina bifida was L5 vertebrae in 3,L5-S1 in 5,S1 in 8 and S1-S2 in 2 patients table1.
Outcome was evaluated in 42 patients who were followed up for more than 6 months. 6 patients who were lost in follow up before 6 months were excluded from outcome analysis. Outcome findings are summarized in the table2. There was no significant difference between both the groups in outcome.
Ultrasound abdomen did not reveal renal anomalies or bladder wall changes in any of the patients. Three patients had >20 ml residual urine (2 in group I and 1 in group II). Among these three one did not improve and two improved.
Discussion
Enuresis or bed-wetting remains one of the society's unsolved problems. Despite a voluminous literature, the causes are not clearly understood and treatment remains controversial. Enuresis is defined as inappropriate or involuntary voiding of urine at an age by which control should be present. Enuresis has to be differentiated from the term incontinence, which is used for leakage of urine in a child with structural or neurological disorders. Nocturnal enuresis is the involuntary and undesirable wetting that occurs at night or during sleep beyond the anticipated age while complicated enuresis encompasses a wide spectrum of functional voiding disorders in children with enuresis and daytime voiding symptoms who don't otherwise exhibit any neurological disorders[11].
Enuresis is best viewed as a symptom rather than a disease. A variety of diverse factors have been reported as a cause e.g. developmental delay, abnormal sleep patterns or organic factors like UTI. A no of studies have also reported higher incidence of spina bifida occulta in these patients. Sakakabara et al in their study of 28 patients of spina bifida cystica and occulta, out of which 12 were with occult form, reported detrusor hyperreflexia in 43% of patients[5].
Kawauchi et al in their study of urological abnormalities in 1328 patients with nocturnal enuresis reported occult spina bifida in 346(36%) of 962 who had plain X-ray of the spine.[12] Ritchey et al, in their retrospective study of 456 patients with diurnal enuresis to determine the relationship between spina bifida occulta and attainment of continence, reported spina bifida occulta in 48 patients (38%) out of 127 patients who had undergone X-ray of spine.[13] Boone et al who looked for spina bifida occulta in 653 patients attending an accident and emergency department without presenting complains of backache or enuresis reported incidence of spina bifida occulta much higher in those below the age of 40 yrs (29.2%) compared with those above 40 yr (9.8%)[14].Based on this they suggested that spina bifida occulta is a common anomaly, of no clinical significance on its own. However we could not find it very high as reported by Kalra et al (89%).[15] In the present study 18 patients (37%) out of 48 studied had spina bifida occulta which is similar to Kawauchi et al[5] and Ritchey et al.[13] This incidence is higher than reported in normal population by Boone et al who reported an overall age-adjusted incidence as 17.3%.[14]
The present study also aimed to study impact of presence of spina bifida occulta in these monosymptomatic enuretics on clinical outcome. Questionnaires and voiding charts are most important tools in reporting the outcome and results of therapeutic interventions.[10] Out of all available therapeutic interventions, behavioral modifications are most successful for long lasting improvement.
In the present study outcomes were similar in both the groups when we compared on traditional therapy and using voiding charts as a tool for outcome evaluation. table2.
Fidas et al in their review of 138 adults with varying urological complaints, who underwent plain radiographs, found spina bifida occulta in 55% of men and 43% of women. All patients underwent urodynamic evaluation but they could not find correlation between the urodynamic abnormalities and spina bifida occulta.[8] However, clinical outcome of these patients was not reported. Ritchey et al in a retrospective review reported incidence of spina bifida occulta in 38% of children with enuresis. Findings of urodynamic studies were similar in both the groups. Two third of their patients with spina bifida improved and they concluded that most of the children with spina bifida occulta can be managed conservatively.
It is said that the presence of spina bifida occulta suggests the existence of subclinical neurological bladder. In the present study it was found that although incidence of spina bifida occulta is higher (37%) than usually reported in normal population they do not have adverse effect on outcome. No significant difference could be found in clinical outcome of patients with enuresis who also had spina bifida occulta as compared to those enuretics with normal spine on X-ray. The present study finding of higher incidence of spina bifida is similar to other studies however total cured was less than as reported by Ritchey et al which may be due to shorter duration of follow-up in the present study. The present study does not agree with Horowitz et al who suggested that night wetting in spina bifida has a much lower incidence of spontaneous resolution.[16]
It may be concluded that although incidence of spina bifida occulta is higher in enuresis, they do not affect the clinical outcome.
Key Message
Spina bifida occulta is a common finding in enuresis
References
1. Doleys DM. Behavioral treatments for nocturnal enuresis: A review of the recent literature. Psychol Bull 1977; 84(1) : 30-34.
2. Rushton HG. Nocturnal enuresis: Epidemiology, evaluation and currently available treatment options. J Pediatr 1989; 114: 691-696.
3. Forsythe WI, Redmond A. Enuresis and spontaneous cure rate: Study of 1129 enuretics. Arch Dis Child 1974; 49: 259-263.
4. Dodge WF, West EF, Bridgforth EB et al. Nocturnal enuresis in 6 to 10 year old children: Correlation with bacteriuria, proteinuria, and dysuria. Am J Dis Child 1970: 120; 32-35.
5. Sakakibara R, Hattori T, Uchiyama T, Kamura K, Yamanishi T. Uroneurological assessment of spina bifida cystica and occulta. Neurourol Urodyn 2003; 22(4): 328-334.
6. Khoury AE, Hendrick EB, McLorie GA, Kulkarni A, Churchill BM. Occult spinal dysraphism: Clinical and urodynamic outcome after division of the filum terminale. J Urol 1990; 144: 426.
7. Galloway NT, Tainsh J. Minor defects of the sacrum and neurogenic bladder dysfunction. Brit J Urol 1985; 57: 154.
8. Fidas A, MacDonald HL, Elton RA, McInnes A, Wild SR, Chisholm GD. Prevalence of spina bifida occulta in patients with functional disorders of the lower urinary tract and its relation to urodynamic and neurophysiological measurements. Brit Med J 1989; 298: 357
9. American Psychiatry Association; Diagnostic and Statistical Manual of Mental Disorders. 4 edn. Washington DC; American Psychiatry Association 1994.
10. Norgaard JP, van Gool JD, Hjalmas K, Djurhuus JC, Hellstrom AI. Standardization and definitions in the lower urinary tract dysfunction in children. Brit J Urol 1998; 81(supp3): 1-6.
11. Aneja S. Nocturnal Enuresis. Indian J Pediatr 2002: 69; 707-712.
12. Kawauchi A, Kitamori T, Imada N, Tanaka Y, Watanabe H. Urological abnormalities in1, 328 patients with nocturnal enuresis. Eur Urol 1996; 29; 231-234.
13. Ritchey ML, Sinha A, Dipietro MA, Huang C, Flood H, Bloom DA. Significance of spina bifida occulta in children with diurnal enuresis. J Urol 1994; 152: 815-818.
14. Boone D, Parsons D, Lachmann SM, Sherwood T. Spina bifida occulta: lesion or anomaly Clin Radiol 1985; 36(2): 159-161.
15. Kalra V, Palaksha HK. Lumbosacral Spina Bifida Occulta in Functional enuresis observed during Laser Reflexo Therapy. J Child Neurol 1999; 14(3): 541-543.
16. Horowitz M, Combs Ajand Gerdes D. Desmopressin for nocturnal incontinence in the spina bifida population. J Urol 1997; 158 : 2267-2268.(Kumar Praveen, Aneja S, K)
Abstract
OBJECTIVE: To study incidence of spina bifida occulta in nocturnal enuresis cases and to compare outcome of enuresis with spina bifida occulta and enuresis without spina bifida occulta. METHODS: Patients with enuresis divided into two groups based on X-ray lumbosacral spine. Outcome of these patients were compared on behavioral therapy. RESULTS: Spina bifida occulta was detected in 18 out of total 48 patients. Levels of spina bifida were L5 vertebrae in 3,L5-S1 in 5,S1 in 8 and S1-S2 in 2 patients. Outcome was evaluated in 42 patients who were followed up for more than 6 months. There was no significant difference between both the groups. CONCLUSION: Spina bifida occulta is a common finding in enuresis. Outcome of patients with spina bifida occulta is not different than the patients without spina bifida occulta.
Keywords: Spina bifida occulta; enuresis
Enuresis is a common problem seen in children and adolescents. Functional enuresis is defined as urination in the clothing or bed-wetting beyond the age when children should be toilet trained and in the absence of neurological abnormality[1]. Approximately 15-20% of 5 years olds wet their beds.[2] Nearly 15% of these children spontaneously achieve control each year so that after the age of 15 years only 1 % of the children have enuresis[3].
Enuresis is best viewed as a symptom rather than a disease. It is a manifestation that can be caused by or affected by a variety of factors including developmental delay, abnormal sleep patterns or organic factors such as urinary tract infection (UTI).[4]
Spina bifida occulta is a congenital spinal anomaly, which occurs due to failure of fusion of the posterior arches of the lumbosacral spine. Spina bifida occulta has been linked with a wide spectrum of urodynamic abnormalities including upper and lower motor neuron types of bladder and urethral dysfunction.[5]
Few reports have suggested that enuretic children have higher incidence of spina bifida occulta[6],[7],[8] and these enuretics with spina bifida occulta may have a worse clinical outcome. Khoury et el identified a group of 31 diurnal enuretic children with spina bifida occulta who failed to respond to conservative measures, and they reported resolution of the incontinence in 70% of them after dividing the filum terminale.[6]
The modalities of treatment presently available include tricyclic antidepressant, desmopressin, behavioral therapy, alarm system, acupuncture etc. Out of all available modalities behavioral modification methods are most successful in permanently curing nocturnal enuresis.
In the present study, an attempt has been made to study incidence of spina bifida occulta in enuretic children and clinical outcome of these enuretic with spina bifida occulta were compared with enuretics without spina bifida, managed with behavioral modification therapy.
Materials and methods
This prospective observational study was conducted in the Neurology clinic of a pediatric tertiary hospital in Delhi. All children who came with the complaints of bed-wetting and were found to be monosymptomatic, constituted the study material.
DSM IV definition of enuresis was used for defining a case of enuresis[9] i.e.
Repeated involuntary or intentional voiding during the day or night in clothes or bed.
At least 2 such events per week for at least 3 consecutive months or the presence of clinically significant distress or impairment in social, academic or other important areas of functioning.
Chronological age of at least 5 years or equivalent age
Detailed history of each child fulfilling above-mentioned criteria was recorded in a preformed proforma and complete neurological examination was done. Lumbosacral spine was examined to look for nevus, dimple, gluteal folds asymmetry or tuft of hairs. Abdomen was palpated for distended bladder, kidneys and fecoliths.
All patients underwent urine analysis & culture, stool examinations for ova and cysts and X-ray lumbosacral spine-AP and lateral view for spina bifida occulta. Ultrasound abdomen was also done in each case for any renal/bladder wall changes and residual urine after voiding.
Based on X-ray lumbosacral spine patients were divided into two groups
Group-I=Enuretics with spina bifida occulta
Group-II= Enuretics with normal spine
A child psychologist counseled children, parents, and treatment options were explained to them. Parents and children who gave consent for behavioral modification trial for at least 6 months were taken for further study.
The children were encouraged to void after awakening, after every 2 hours, and then before going to bed. The children were instructed not to drink excess of water with dinner and to withhold fluids 2 hours before going to bed. Children and parents were asked to maintain voiding charts and parents were asked to give rewards for dry nights. Children were followed up monthly in the neurology clinic. At each visit, no of events of voiding, compliance were recorded. Outcome of both groups were evaluated as per the following definitions[10].
Complete cure (responder) - as a reduction in wet nights of at least 90 %
Improvement or partial responder as a reduction in wet nights of >50%but <90%
Data was analyzed by using chi-square test to compare both the groups.
Results
A total of 48 patients who came with complaints of bed-wetting (night, day or both) and gave consent for the trial constituted material for the present study. There were 22 males and 26 were females. Age ranged from five and half years to 14 years. 38 patients had only nocturnal enuresis while 10 patients had history of bed-wetting during daytime also when they fell asleep. None of the patients had urgency, urge incontinence or hesitancy of urine. Positive family history was observed in 8 patients and usual time of bed-wetting was 2-3 hrs after going to bed. 5 patients complained bed-wetting more than once in single night.
Spina bifida occulta was detected in 18 out of total 48 patients. Level of spina bifida was L5 vertebrae in 3,L5-S1 in 5,S1 in 8 and S1-S2 in 2 patients table1.
Outcome was evaluated in 42 patients who were followed up for more than 6 months. 6 patients who were lost in follow up before 6 months were excluded from outcome analysis. Outcome findings are summarized in the table2. There was no significant difference between both the groups in outcome.
Ultrasound abdomen did not reveal renal anomalies or bladder wall changes in any of the patients. Three patients had >20 ml residual urine (2 in group I and 1 in group II). Among these three one did not improve and two improved.
Discussion
Enuresis or bed-wetting remains one of the society's unsolved problems. Despite a voluminous literature, the causes are not clearly understood and treatment remains controversial. Enuresis is defined as inappropriate or involuntary voiding of urine at an age by which control should be present. Enuresis has to be differentiated from the term incontinence, which is used for leakage of urine in a child with structural or neurological disorders. Nocturnal enuresis is the involuntary and undesirable wetting that occurs at night or during sleep beyond the anticipated age while complicated enuresis encompasses a wide spectrum of functional voiding disorders in children with enuresis and daytime voiding symptoms who don't otherwise exhibit any neurological disorders[11].
Enuresis is best viewed as a symptom rather than a disease. A variety of diverse factors have been reported as a cause e.g. developmental delay, abnormal sleep patterns or organic factors like UTI. A no of studies have also reported higher incidence of spina bifida occulta in these patients. Sakakabara et al in their study of 28 patients of spina bifida cystica and occulta, out of which 12 were with occult form, reported detrusor hyperreflexia in 43% of patients[5].
Kawauchi et al in their study of urological abnormalities in 1328 patients with nocturnal enuresis reported occult spina bifida in 346(36%) of 962 who had plain X-ray of the spine.[12] Ritchey et al, in their retrospective study of 456 patients with diurnal enuresis to determine the relationship between spina bifida occulta and attainment of continence, reported spina bifida occulta in 48 patients (38%) out of 127 patients who had undergone X-ray of spine.[13] Boone et al who looked for spina bifida occulta in 653 patients attending an accident and emergency department without presenting complains of backache or enuresis reported incidence of spina bifida occulta much higher in those below the age of 40 yrs (29.2%) compared with those above 40 yr (9.8%)[14].Based on this they suggested that spina bifida occulta is a common anomaly, of no clinical significance on its own. However we could not find it very high as reported by Kalra et al (89%).[15] In the present study 18 patients (37%) out of 48 studied had spina bifida occulta which is similar to Kawauchi et al[5] and Ritchey et al.[13] This incidence is higher than reported in normal population by Boone et al who reported an overall age-adjusted incidence as 17.3%.[14]
The present study also aimed to study impact of presence of spina bifida occulta in these monosymptomatic enuretics on clinical outcome. Questionnaires and voiding charts are most important tools in reporting the outcome and results of therapeutic interventions.[10] Out of all available therapeutic interventions, behavioral modifications are most successful for long lasting improvement.
In the present study outcomes were similar in both the groups when we compared on traditional therapy and using voiding charts as a tool for outcome evaluation. table2.
Fidas et al in their review of 138 adults with varying urological complaints, who underwent plain radiographs, found spina bifida occulta in 55% of men and 43% of women. All patients underwent urodynamic evaluation but they could not find correlation between the urodynamic abnormalities and spina bifida occulta.[8] However, clinical outcome of these patients was not reported. Ritchey et al in a retrospective review reported incidence of spina bifida occulta in 38% of children with enuresis. Findings of urodynamic studies were similar in both the groups. Two third of their patients with spina bifida improved and they concluded that most of the children with spina bifida occulta can be managed conservatively.
It is said that the presence of spina bifida occulta suggests the existence of subclinical neurological bladder. In the present study it was found that although incidence of spina bifida occulta is higher (37%) than usually reported in normal population they do not have adverse effect on outcome. No significant difference could be found in clinical outcome of patients with enuresis who also had spina bifida occulta as compared to those enuretics with normal spine on X-ray. The present study finding of higher incidence of spina bifida is similar to other studies however total cured was less than as reported by Ritchey et al which may be due to shorter duration of follow-up in the present study. The present study does not agree with Horowitz et al who suggested that night wetting in spina bifida has a much lower incidence of spontaneous resolution.[16]
It may be concluded that although incidence of spina bifida occulta is higher in enuresis, they do not affect the clinical outcome.
Key Message
Spina bifida occulta is a common finding in enuresis
References
1. Doleys DM. Behavioral treatments for nocturnal enuresis: A review of the recent literature. Psychol Bull 1977; 84(1) : 30-34.
2. Rushton HG. Nocturnal enuresis: Epidemiology, evaluation and currently available treatment options. J Pediatr 1989; 114: 691-696.
3. Forsythe WI, Redmond A. Enuresis and spontaneous cure rate: Study of 1129 enuretics. Arch Dis Child 1974; 49: 259-263.
4. Dodge WF, West EF, Bridgforth EB et al. Nocturnal enuresis in 6 to 10 year old children: Correlation with bacteriuria, proteinuria, and dysuria. Am J Dis Child 1970: 120; 32-35.
5. Sakakibara R, Hattori T, Uchiyama T, Kamura K, Yamanishi T. Uroneurological assessment of spina bifida cystica and occulta. Neurourol Urodyn 2003; 22(4): 328-334.
6. Khoury AE, Hendrick EB, McLorie GA, Kulkarni A, Churchill BM. Occult spinal dysraphism: Clinical and urodynamic outcome after division of the filum terminale. J Urol 1990; 144: 426.
7. Galloway NT, Tainsh J. Minor defects of the sacrum and neurogenic bladder dysfunction. Brit J Urol 1985; 57: 154.
8. Fidas A, MacDonald HL, Elton RA, McInnes A, Wild SR, Chisholm GD. Prevalence of spina bifida occulta in patients with functional disorders of the lower urinary tract and its relation to urodynamic and neurophysiological measurements. Brit Med J 1989; 298: 357
9. American Psychiatry Association; Diagnostic and Statistical Manual of Mental Disorders. 4 edn. Washington DC; American Psychiatry Association 1994.
10. Norgaard JP, van Gool JD, Hjalmas K, Djurhuus JC, Hellstrom AI. Standardization and definitions in the lower urinary tract dysfunction in children. Brit J Urol 1998; 81(supp3): 1-6.
11. Aneja S. Nocturnal Enuresis. Indian J Pediatr 2002: 69; 707-712.
12. Kawauchi A, Kitamori T, Imada N, Tanaka Y, Watanabe H. Urological abnormalities in1, 328 patients with nocturnal enuresis. Eur Urol 1996; 29; 231-234.
13. Ritchey ML, Sinha A, Dipietro MA, Huang C, Flood H, Bloom DA. Significance of spina bifida occulta in children with diurnal enuresis. J Urol 1994; 152: 815-818.
14. Boone D, Parsons D, Lachmann SM, Sherwood T. Spina bifida occulta: lesion or anomaly Clin Radiol 1985; 36(2): 159-161.
15. Kalra V, Palaksha HK. Lumbosacral Spina Bifida Occulta in Functional enuresis observed during Laser Reflexo Therapy. J Child Neurol 1999; 14(3): 541-543.
16. Horowitz M, Combs Ajand Gerdes D. Desmopressin for nocturnal incontinence in the spina bifida population. J Urol 1997; 158 : 2267-2268.(Kumar Praveen, Aneja S, K)