Important components of a programme for children with medical complexity: An Australian perspective.

BACKGROUND: Children with medical complexity (CMC) have high care needs, often unmet by traditional healthcare models. In response to this need, the Complex Care Service (CCS) at The Royal Children's Hospital (RCH), Melbourne was created. Although preliminary parent satisfaction data were available, we lacked knowledge of how the various components of the expanded service were valued and contributed to overall caregiver satisfaction. AIM: The aims of this study were to (a) determine what caregivers value most about the CCS and (b) explore caregiver perceptions of care. METHODS: All caregivers of children enrolled in the RCH CCS in April 2017 were invited to participate. A purposefully designed survey explored caregiver perceptions of care, including patient quality of care; the extent to which the CCS components added value and satisfaction; and frequency of contact. Participants were also invited to answer open-ended questions and provide general comments. RESULTS: Responses were received from 53 families (51%). We found that 24-hr phone advice, coordination of appointments, a key contact, and access to timely information were the most important components of the service. More than 90% of caregivers indicated that they were satisfied with care and that the CCS improved their child's quality of care. Coordination, communication, family-centred care, quality care, and access were emergent themes within comments. CONCLUSION: This study provides important information regarding the design and operation of services for CMC throughout Australia and further afield. Our findings highlight the importance of the key contact and family-centred care. This has implications for practice, as maintaining service quality, as the CCS expands and is implemented more widely, is a major sustainability challenge. It is crucial that we have a detailed understanding of what elements are required to support effective care coordination, to achieve successful implementation on a larger scale.

Enuresis Management in Children: Retrospective Clinical Audit of 2861 Cases Treated with Practitioner-Assisted Bell-and-Pad Alarm.

OBJECTIVE: To establish the treatment efficacy of practitioner-assisted bell-and-pad alarm therapy in children with enuresis between the ages of 5 and 16 years by retrospective medical chart review of 2861 children in multiple clinical settings. STUDY DESIGN: This review was conducted across 7 Australian clinical practices. The primary outcome measure was the time taken for children with either primary, secondary, monosymptomatic, or nonmonosymptomatic enuresis to be dry for 14 consecutive nights. The secondary outcome measure was to determine relapse rates, defined as 1 symptom recurrence per month post interruption of treatment. Data were analyzed by correlation and χ2 test via IBM SPSS Statistics (version 22). RESULTS: The overall success rate of the bell and pad treatment was 76%, irrespective of age. The mean treatment time to achieve dryness was 62.1 ± 30.8 days, and the relapse rate was 23%. Concurrent bowel dysfunction was associated with a slightly lower success rate (74%). Concurrent lower urinary tract symptoms were associated with a lower success rate (73%) and greater relapse (1.75 times more likely to relapse). Children with secondary enuresis had significantly greater success than those with primary enuresis (82% vs 74%). CONCLUSION: The type of alarm therapy reported in this study is highly effective. This study will provide the basis for clinical guidelines and practice tools for clinicians, which will help to reduce variation in care pathways for alarm treatment for enuresis.

Daily transabdominal electrical stimulation at home increased defecation in children with slow-transit constipation: a pilot study.

PURPOSE: Transcutaneous electrical stimulation (TES) (3 sessions/wk) over the abdomen stimulated bowel functions in a randomized controlled trial. This pilot study assessed whether daily TES at home with a safe, portable machine would be possible and more efficacious than trial results. METHODS: Eleven patients (6 male/5 female; mean age, 14 years; range, 12-18 years) with slow-transit constipation who relapsed or responded poorly in the trial were recruited (11 +/- 5 months later). An EPM-IF-4160 (Fuji Dynamics, Hong Kong) portable machine (sine waveform, 4 kHz carrier frequency, 80-160 Hz beat frequency, intensity <33 mA) delivering interferential current (2 electrodes over epigastrium + 2 over kidneys) was applied 1 hour daily at home. Continence diaries were kept for 1 month before and 2 months during treatment. RESULTS: All children completed more than 1 month of treatment after baseline recording. Defecation increased in 9 of 11 children, and soiling decreased in 4 of 11 children. There was a significant increase in total episodes of defecation per week (mean +/- SD, 2.5 +/- 2.1 vs 6.7 +/- 4.4; P = .008) and a nonsignificant decrease in soiling (3.8 +/- 1.6 vs 1.1 +/- 0.5 episodes/wk, P = .1). Daily stimulation does not affect abdominal pain. No adverse events occurred. CONCLUSIONS: Daily TES at home is safe and significantly improved bowel function in children who did not respond to 3 times per week of TES. Home TES may be a novel treatment of intractable slow transit constipation, avoiding hospital visits.

Improvement of quality of life in children with slow transit constipation after treatment with transcutaneous electrical stimulation.

BACKGROUND: Slow transit constipation (STC) causes intractable symptoms not responsive to medical treatment. Children have irregular bowel motions, colicky abdominal pain, and frequent soiling. Transcutaneous electrical stimulation using interferential current (interferential therapy [IFT]) is a novel treatment of STC. This study assessed quality of life (QOL) in STC children before and after IFT treatment. METHODS: Eligible STC children were randomized to receive either real or placebo IFT (12 sessions for 4 weeks). Questionnaires (Pediatric Quality of Life Inventory) were administered before and 6 weeks after treatment, with parallel parent and child self-report scales. Higher scores indicate better QOL. Holschneider and Templeton scores were also obtained. The QOL scores were compared using paired t tests. RESULTS: Thirty-three children (21 male), with a mean age of 11.8 years (range, 7.4-16.5 years), were recruited; 16 received real IFT. Child-perceived QOL was improved after real IFT compared with baseline (81.1 vs 72.9, P = .005) but not after placebo IFT (78.1 vs 74.9, P = .120). The Holschneider score improved after real IFT (10 vs 8, P = .015) but not after placebo IFT (9 vs 8, P = .112). Parentally perceived QOL was similar after real IFT (70.1 vs 70.3, P = .927) and placebo IFT (70.2 vs 69.8, P = .899). There were no differences in Templeton scores. CONCLUSION: Interferential therapy is a novel therapy for children with STC that improves their self-perceived QOL.

Standard medical therapies do not alter colonic transit time in children with treatment-resistant slow-transit constipation.

BACKGROUND: Slow transit constipation (STC) is a form of chronic constipation characterised by prolonged passage of faecal matter through the colon. It is diagnosed by demonstrating delayed colonic transit on gastrointestinal transit studies. Traditionally, radio-opaque marker studies are performed. Recently, radioisotope nuclear transit studies (NTS) have been used in our centre to assess gastrointestinal transit time. This study aimed to evaluate if there are changes in colonic transit in STC children resistant to standard medical treatment over a prolonged period. METHODS: Children with STC resistant to standard medical therapy for > or =2 years who had undergone two separate NTS to assess their colonic transit (where the first study had identified slow colonic transit without anorectal retention) were identified after ethical approval. The geometric centre (GC) of radioisotope activity at 6, 24, 30 and 48 h was compared in the two transit studies to determine if changes occurred. RESULTS: Seven children (4 males) with proven STC resistant to standard medical therapy and two transit studies performed at different times were identified. Mean age was 7.0 years (5.4-10.8 years) at first study, and 11.4 years (9.7-14.2 years) at second study, with a mean of 4.4 years (1-8.5 years) between studies. There was no significant difference in colonic transit at any timepoint in the two tests (paired t test). CONCLUSIONS: We conclude that nuclear transit studies are reproducible in assessing slow colonic transit in children with treatment-resistant STC and demonstrate that conventional medical treatment over many years has no effect on underlying colonic motility.

Slow-transit constipation in children: our experience.

Constipation is a common problem in children, with childhood prevalence estimated at between 1 and 30%. It accounts for a significant percentage of referrals to paediatricians and paediatric gastroenterologists. It commonly runs in families, suggesting either an underlying genetic predisposition or common environmental factors, such as dietary exposure. The peak age for presentation of constipation is shortly after toilet training, when passage of hard stools can cause pain on defecation, which then triggers holding-on behaviour in the child. At the time of the next call to stool the toddler may try to prevent defecation by contraction of the pelvic floor muscles and anal sphincter. Unless the holding-on behaviour is quickly corrected by interventions to soften faeces and prevent further pain, the constipation can very rapidly become severe and chronic. Until recently, this mechanism was thought to be the only significant primary cause of constipation in childhood. In this review, we will summarise recent evidence to suggest that severe chronic constipation in children may also be due to slowed colonic transit.

Decreased colonic transit time after transcutaneous interferential electrical stimulation in children with slow transit constipation.

PURPOSE: Idiopathic slow transit constipation (STC) describes a clinical syndrome characterised by intractable constipation. It is diagnosed by demonstrating delayed colonic transit on nuclear transit studies (NTS). A possible new treatment is interferential therapy (IFT), which is a form of electrical stimulation that involves the transcutaneous application of electrical current. This study aimed to ascertain the effect of IFT on colonic transit time. METHODS: Children with STC diagnosed by NTS were randomised to receive either 12 real or placebo IFT sessions for a 4-week period. After a 2-month break, they all received 12 real IFT sessions-again for a 4-week period. A NTS was repeated 6 to 8 weeks after cessation of each treatment period where able. Geometric centres (GCs) of activity were calculated for all studies at 6, 24, 30, and 48 hours. Pretreatment and posttreatment GCs were compared by statistical parametric analysis (paired t test). RESULTS: Thirty-one pretreatment, 22 postreal IFT, and 8 postplacebo IFT studies were identified in 26 children (mean age, 12.7 years; 16 male). Colonic transit was significantly faster in children given real treatment when compared to their pretreatment NTS at 24 (mean CG, 2.39 vs 3.04; P < or = .0001), 30 (mean GC, 2.79 vs 3.47; P = .0039), and 48 (mean GC, 3.34 vs 4.32; P = .0001) hours. By contrast, those children who received placebo IFT had no significant change in colonic transit. CONCLUSIONS: Transcutaneous electrical stimulation with interferential therapy can significantly speed up colonic transit in children with slow transit constipation.

Connective tissue disorder--a new subgroup of boys with slow transit constipation?

PURPOSE: Slow transit constipation (STC) is a form of chronic constipation, with delayed colonic passage of stool. Possible etiologies include reduced neurotransmitter levels, reduced interstitial cells of Cajal density, or a disorder of connective tissue (CT) synthesis. A common CT disorder is generalized joint hypermobility (GJH). This study aimed to investigate whether there was a greater prevalence of GJH among patients with STC than controls. METHODS: Children (aged 7-17) diagnosed with STC by radio/nuclear transit study were recruited from outpatient clinics. Controls (no history of constipation) were recruited from outpatient clinics and a scout jamboree. Hypermobility was assessed using the Beighton score (4 or more = hypermobile). This project received ethical approval by the human research ethics committee. RESULTS: Thirty-nine STC subjects and 41 controls were measured. Of 39 STC subjects, 15 (38%) were hypermobile, compared to 8 (20%) of 41 controls (P = .06). Analyzed by gender, 10 (38%) of 26 STC males and 1 (4%) of 23 control males were hypermobile (P < .01). CONCLUSIONS: These results show that GJH is higher in STC children, particularly males, suggesting that a disorder of CT synthesis plays a role in the etiology of STC. Further research is required to ascertain the nature of any relationship and how this knowledge may aid our understanding and treatment of STC.

Quality of life in children with slow transit constipation.

BACKGROUND: Slow transit constipation (STC) causes intractable symptoms not readily responsive to laxatives, diet, or life-style changes. Children with STC have irregular bowel motions associated with colicky abdominal pain and frequent uncontrollable soiling. This study assessed the physical and psychosocial quality of life (QOL) in children with long-standing (> or =2 years) STC vs healthy controls. METHODS: Children (aged 8-18) were recruited from gastrointestinal and surgical clinics and a Scout Jamboree. After informed consent was obtained, the questionnaire (Pediatric Quality of Life Inventory) was administered. This consists of parallel child and parent self-report scales encompassing physical functioning, emotional functioning, social functioning, and school functioning. Higher scores indicate better QOL. P value less than .05 was considered statistically significant. RESULTS: In 51 children with STC (mean, 11.5 years; male/female, 2:1) and 79 controls (mean, 12.1 years; male/female, 1.9:1), Pediatric Quality of Life Inventory QOL score was significantly lower in the STC group (72.90 vs 85.99; P < .0001). In addition, parents of children with STC reported a significantly lower QOL score than their child compared with the child's own report (64.43 vs 72.90; P = .0034). Parents of controls did not (84.25 vs 85.99; P = .12). CONCLUSIONS: Slow transit constipation is a debilitating condition affecting both physical and emotional functioning in children. Parental perception of QOL is significantly worse, highlighting the considerable family impact of constipation and uncontrollable soiling.

Pilot study using transcutaneous electrical stimulation (interferential current) to treat chronic treatment-resistant constipation and soiling in children.

BACKGROUND: Chronic constipation in children may have organic or behavioral causes. The purpose of the present study was to investigate the effect of treatment with transcutaneous electrical stimulation (using interferential current) in children with chronic treatment-resistant constipation with proven organic disorders. METHODS: Eight children (7-16 years) with at least 4 years of chronic treatment-resistant constipation and soiling, who had failed diet, laxative treatment and behavioral therapy were given 1 month of transcutaneous electrical stimulation. The three most severe cases had appendicostomies with antegrade washouts every 2-3 days to prevent impaction and reduce their soiling. Children and carers kept a daily diary of bowel habits, recording number of spontaneous defecations, episodes of soiling, use of bowel washouts and medications. Transcutaneous stimulation using interferential current was applied three times per week for 3-4 weeks using four surface electrodes, two to the paraspinal area of T9-10 to L2 and one to either side of the anterior abdominal wall beneath the costal margin. Diaries were recorded for 1 month before, during, and after stimulation and for 2 weeks 3 months later. RESULTS: Transcutaneous electrical stimulation using interferential current stopped soiling in 7/8 children and increased the frequency of spontaneous defecations in 5/8. Defecations remained high and soiling low for 3 months in 3/6 children (with data). CONCLUSIONS: These results suggest that transcutaneous electrical stimulation using interferential current has a beneficial effect for children with chronic treatment-resistant constipation. Further trials using larger series of patients are needed to confirm this benefit, to determine the ideal stimulation parameters and to investigate why electrical stimulation might be effective.

Evidence against a synergistic effect of desmopressin with conditioning in the treatment of nocturnal enuresis.

OBJECTIVE: To test the hypothesis that desmopressin facilitates acquisition of continence, we aimed to establish whether, in children with nocturnal enuresis who are desmopressin nonresponders, adjunct desmopressin increases the rate of sustained continence after treatment with a conditioning alarm. Study design Patients with nocturnal enuresis (n=358; age range, 6-16 years) completed a 4-week "run-in" course of intranasal desmopressin (20-40 microg). Of these, 207 defined as nonresponders (<50% reduction in wet nights) were randomly assigned to receive either desmopressin (n=101) or placebo (n=106) nasal spray, together with conditioning alarm therapy for 8 weeks. Principal outcome measures were remission (28 continuous dry nights) and relapse (>2 wet nights in 2 weeks after having achieved remission). RESULTS: Remission rates were similar in both groups (51.5% desmopressin, 48.1% placebo; 95% CI on difference, -10%, 17%; P=.63), and relapse rates were not significantly different (13.5% vs 5.9%; 95% CI on difference, -3.7%, 19%; P=.19). Although remission rates were similar, children treated with desmopressin had significantly more dry nights during treatment than those in the placebo group. CONCLUSIONS: Desmopressin did not act synergistically with alarm treatment to achieve remission. Therefore, we infer that in partial or nonresponders, desmopressin does not enhance learning.