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Sleep Quality, Dysfunctional Sleep Beliefs, and Health-Related Quality of Life and Functioning in People With Multiple Sclerosis

Multiple sclerosis (MS) is a common debilitating neurologic disease that affects mostly young women. This review provides an overview of research on the psychosocial impact of parental MS on children to inform clinicians and support people with MS considering parenthood.

A systematic review of the literature was performed by searching the MEDLINE, PsycINFO, and PSYNDEX databases. We included quantitative and mixed-method studies assessing psychosocial outcomes of children with a parent with MS. Studies were screened for eligibility and evaluated for risk of bias.

We screened 608 references, assessed 72 studies in full-text, and included 28 studies in this review. Most of the studies reported on psychosocial adjustment processes, with most results suggesting negative consequences, including difficulties with mood, behavior, or social interaction. Several studies also described associations between children with a parent with MS and increased incidences of psychiatric disorders. Nevertheless, some studies claimed that children with a parent with MS were not more likely to have psychosocial problems compared with children without a parent with MS. A few studies indicated probable positive effects of parental MS, eg potentially increased social competence. Other investigated outcomes were children’s coping skills, early childhood development, body image, and effects on education, and these were unaffected or only slightly affected by having a parent with MS.

Having a parent with MS has a relevant effect on children. However, the heterogeneous nature and varying quality of the included studies limit the interpretability of these findings. Further research is needed to provide robust evidence.

From the Institute of Neuroimmunology and Multiple Sclerosis (MH, JP, CH, ACR) and Department of Neurology (CH), University Medical Center Hamburg-Eppendorf, Hamburg, Germany; Nursing Research Unit, Institute of Social Medicine and Epidemiology, University of Lübeck, Lübeck, Germany (ACR); and private practice, Hamburg, Germany (MH). 

 Michel Haker, MD, Institute of Neuroimmunology and Multiple Sclerosis, University Medical Center Hamburg-Eppendorf, Martinistr. 52, D-20246 Hamburg, Germany; 

 The authors declare no conflicts of interest.

 This project was funded by the German Research Foundation (grant RA 3296/1-1). The funding body had no influence on the content or the implementation of the study.

It is estimated that approximately 10% of children worldwide have a parent with a chronic medical condition.

 Multiple studies have found evidence of increased risks of emotional and behavioral problems in children of parents with chronic somatic illnesses.

 It is suspected that chronic illnesses interfere with parenting through changes in parental availability, household routines, and responsibilities, as well as by contributing to parental depression and family conflict.

Affecting approximately 2.3 million people worldwide,

 multiple sclerosis (MS) is the most prevalent chronic inflammatory disease of the central nervous system.

 The demyelinating disease manifests 70% of the time in women aged 20 to 40 years.

 Thus, people with MS are commonly diagnosed during their reproductive years. People with MS can experience various physical and cognitive symptoms ranging from mild paresthesia and impaired vision to fatigue, limited mobility, and severe disability.

 Consequently, MS can affect the ability to work and to care for children or the household.

 The impact of life-threatening parental illnesses, such as cancer, on children’s development has been shown in the past.

 Having a parent with a debilitating but not life-threatening illness, such as MS, may also affect a child’s psychosocial status.

There have been 3 systematic reviews on this subject,

 providing the latest review in 2014 (literature search in 2012). All 3 reviews found evidence of predominantly negative consequences, but a few of the included studies indicated positive psychosocial effects or no significant effects.

 Considering the time frame of the last reviews, an updated, critical, systematic review is needed. By reviewing the current knowledge on this subject, we aimed to highlight further research needs and implications for clinical care. In this review, we do not specifically address the impact of children taking care of an ill parent.

In August 2020, we searched the following databases via Ovid: MEDLINE, PsycINFO, and PSYNDEX. The research question “What is the psychosocial impact of parental multiple sclerosis on children?” meant that the search consisted of 4 elements: 

. Based on these main search terms, we built search strings using all associated terms and available synonyms and spellings using the operator “OR.” Subject headings were used if available for the searched term. The detailed search strings can be accessed in 

, which is available in a PDF at the bottom of this article.

A few restrictions maximized the number of relevant search results. We included all empirical studies published in English or German and excluded data from case reports, comments, and conference abstracts. We also checked the references of previous reviews for eligible publications.

We did not establish age restrictions for the children included in the study because many studies include children of a wide age range and often do not differentiate between different age groups. We included studies that had children with a parent with MS as participants or studies that referenced them by proxy.

To be considered for inclusion, families had to have at least 1 parent with a confirmed diagnosis of MS of any clinical course. We excluded studies that had children with an MS diagnosis.

All outcomes that could generally be allocated to psychosocial effects, including quality of life issues and adjustment issues, were considered psychiatric disorders. “Good” adjustment in children and adolescents refers to healthy, normative, and age-appropriate behavior that aims toward positive adult functioning.

 Psychosocial adjustment encompasses a child’s emotions, behaviors, and social functioning. Behavior difficulties, often categorized into internalizing and externalizing symptoms, were included as well. Internalizing refers to negative behaviors that focus on inner emotions and are experienced as anxiety, depression, and somatization.

 Children’s externalizing expresses through negative behaviors focusing on the outside, such as hyperactivity, aggression, and disruptive conduct.

 We excluded studies investigating other outcomes, such as predominantly somatic or genetic effects.

We exported all search results to a reference management program (Citavi, version 6.3.0.35; Swiss Academic Software GmbH), with which we detected and deleted possible duplicates. The remaining search results were then exported to the screening tool Rayyan (Qatar Computing Research Institute – Data Analytics Group). One of us (M. Haker) reviewed all results; 2 others (J.P. and A.C.R.) each reviewed one-half of the results. The reviewers were screen blinded from each other. Conflicts were resolved by discussion. The potentially eligible studies agreed on were then subjected to blinded full-text screening in which M. Haker screened all papers and J.P. and A.C.R. each screened one-half according to the eligibility criteria using a structured screening sheet; see 

. Screening conflicts were resolved by discussion.

We created a data extraction sheet to ascertain the most relevant aspects of each included study; for details, see 

. We then extracted the data of all included studies available in full-text. The heterogeneity of the study types, outcomes, and participants did not allow a meta-analysis of the data. We performed a descriptive analysis, combining studies with similar outcomes and differentiating negative, neutral, and positive findings.

To assess the quality of the included studies, we created a quality rating form based on the quality appraisal in a previous systematic review,

 an adapted version of the Graphic Appraisal Tool for Epidemiology.

In assessing the risk of bias for multiple categories, we assigned an overall quality rating of low, medium, or high to each of the included studies. Because 12 of the included studies were already appraised by a systematic review,

 we assessed the risk of bias for the remaining 16 studies only. One of us (M. Haker) rated all included studies; 2 others (J.P. and A.C.R.) each rated one-half. We then compared, discussed, and combined the 2 separate ratings into a final rating. For the detailed criteria used, see 

The database search yielded 719 references, of which 111 were identifieds as duplicates and subsequently deleted. We screened the titles and abstracts of the remaining 608 references for eligibility. During this process, we excluded 536 references. We analyzed the remaining 72 references and 2 additional studies, identified in reference lists of previous reviews, in full-text. After the full-text screening we excluded 46 studies, leaving 28 studies for inclusion. For an overview of the inclusion process, see 

We extracted data from 28 references: 19 cross-sectional studies, 5 cohort studies, and 4 case-control studies. In 2 studies, the same participant sample was examined. Seventeen studies used comparison groups with children of healthy parents; 5 studies collected longitudinal data.

Eight studies originated in the United States, 4 in Canada, 3 in Switzerland, 3 in Australia, 3 in Israel, 2 in Greece, 2 in Denmark, 1 in the United Kingdom, and 1 in Norway; 1 study reported data from the trio of Switzerland, Greece, and Germany. Overall, the sample size varied widely and ranged from 30 to 4177 children with a parent with MS. The ages of the children studied ranged from 1.5 to 26 years, with most studies focusing on children aged 4 to 18 years.

The types of outcomes ascertained and instruments used in the studies are quite heterogeneous. The most frequently assessed outcomes were child adjustment, including emotional and behavioral problems, children’s mental health, and coping with the parent’s illness. Accordingly, the most frequently used instruments were the Strengths and Difficulties Questionnaire,

 In 8 studies, the respondent was a third party (a schoolteacher or an external rater); 6 studies had at least 1 parent and the child as respondents; 7 studies had only children as respondents; 3 studies had only 1 parent as the respondent; and 4 studies had findings based on databases or registries. For an overview of the included studies, see 

In the quality appraisal, we rated 5 studies as high quality, 6 as medium quality, and 5 as low quality. Combined with the 12 studies previously appraised in the quality assessment by Razaz et al,

 we report data from 5 high-quality studies, 11 medium-quality studies, and 12 low-quality studies in this review.

Regarding the risk of bias, the most common issue was missing data on participating children (exact age, eligibility criteria, and recruiting), as well as the children’s exposure to the parental illness, eg, exposure duration, MS course, and MS severity. Another frequent concern was the small sample size of many of the studies.

To summarize the findings, we grouped the studies based on their outcomes and reported the direction of their results.

 investigated the impact of parental MS on children’s psychosocial adjustment. Eight studies

 found that children with a parent with MS (N = 700) experienced negative emotions (eg, sadness, anxiety) and had behavior difficulties. Five studies

 compared children with a parent with MS with children with parents without MS (N = 668). Six studies,

 found that depression, fatigue, physical disability, and marital discord for parents with MS correlated with negative outcomes for their children (N = 491).

 compared how much the children of people with MS (N = 56) knew about their parent’s disease. Children with partial information internalized more (eg, felt lonely, sad) and had more social problems compared with children with full or no information about their parent’s MS. One caveat: the psychosocial status of children with no information was reported by their parents because these children were not allowed to complete their own questionnaires.

 found that the children of people with MS (N = 548) had normal psychosocial development and reported no deviation from expected norms. Whereas Rintell

 did not use a control group, Andersen et al

 compared their findings with data from 42,016 children of mothers without MS

 found that children of people with MS (N = 48), especially girls, expressed prosocial behavior more often than children of parents without MS. One case-control study

 reported that children of a parent with MS (N= 126) were less likely to report peer problems than children of parents without MS (N = 126).

 assessed the incidence of psychiatric disorders, including mood and anxiety disorders as well as attention-deficit/hyperactivity disorder (ADHD). All 4 of these studies indicated a possible negative impact for children with a parent with MS.

 children with a parent with MS (N = 1388) had higher rates of psychiatric symptoms, indicating a higher risk of psychiatric disorders, compared with children with parents without MS (N = 5217). One study

 reported that children of parents with peripartum depression were more likely to have higher rates of psychiatric disorders and that parents with MS were more likely to be affected by peripartum depression than parents without MS. One cross-sectional study

 assessed children of people with MS (N = 41) regarding their indication for psychotherapy. According to this study, 22 children of people with MS (54%) needed psychotherapy. This study did not draw a comparison with children of parents without MS.

 investigated the influence of maternal chronic illnesses on the prevalence of ADHD in their children, comparing 47,944 children with ADHD with 2,274,713 children without ADHD. Maternal MS was related to an 80% higher chance of the children having ADHD (31 children of mothers with MS out of 47,944 children with ADHD [0.06%] vs 880 children of mothers with MS out of 2,274,713 children without ADHD [0.04%]).

 (N = 274) reported on the coping skills of children with a parent with MS. Three studies

 found that these children (N = 342) likely coped better as they got older and that their coping correlated with their parent’s coping ability, especially that of the parent without MS. One of the studies

 compared data with those from children of a parent with other severe illnesses (n = 116). One cross-sectional study

 reported that children of a parent with MS (N = 48) were less likely to use coping strategies that were considered beneficial than children of parents without MS (n = 145) and tended to use coping mechanisms that correlated with negative outcomes.

 with the same study population (N = 56) ivestigated children’s feelings toward their parents. Yahav et al

 found that children of a parent with MS (expressed more feelings of obligation, concern, fear, and anger toward their parent than children of parents without MS (N = 156). Duvdevany et al

 found a high prevalence of ambivalence in children of people with MS.

The vulnerability in the early childhood development of children with a parent with MS was examined in 2 studies

 found no general difference in the early childhood development of children of parents with MS (n = 153) and children of parents without MS (n = 876), but parental mental comorbidities, as well as longer exposure to the parental illness, were associated with increased developmental vulnerability in children. A 2016 study

 indicated only 1 difference from children of healthy parents (n = 2988): children of people with MS (n = 783) appeared less vulnerable to problems with social competence. Mental and physical comorbidities in mothers with MS seemed to be associated with increased developmental vulnerability in their children.

 examined the body image of children with a parent with MS (N = 154) and found no significant impact. One of the studies

 compared the findings with those from children of parents without MS (n = 60).

 found that in the social interactions of mother-daughter dyads, daughters and their mothers with MS (n = 31) were similar to daughters and mothers without MS (n = 34).

 found that children of people with MS (n = 4,177) earned significantly higher school grades, but did not differ in highest degree attained from children of parents without chronic illness (n = 33,416). Furthermore, children of a parent with MS, especially daughters, tended to attain a health-related degree more frequently than children of parents without MS.

We conducted a systematic literature review of the psychosocial impact of having a parent with MS on children and adolescents. Most of the 28 studies reported a negative impact, but there is also evidence for positive associations as well as studies claiming no association between having a parent with MS and children’s psychosocial status. The results are largely in accordance with previous systematic reviews on the subject.

 in the present review were not described in previous work. These studies were of mixed quality, and most of them propose negative effects on children of a parent with MS, especially if the parent experiences depression. In contrast to these findings, 1 high-quality study

 reported no impact of maternal MS on children’s mental health.

 described associations between parental MS and negative outcomes in children’s psychosocial adjustment, mainly difficulties in children’s moods, behavior, and social interactions. This is also corroborated in the broader literature. A parent’s physical illness is assumed to be a risk factor for psychosocial maladjustment in children, mainly in their internalizing and externalizing problems.

 reported increased risks for children with a parent with MS of developing mental health conditions, including mood and anxiety disorders and ADHD. Nonetheless, the described effects were usually small, and study limitations have to be considered.

It remains an open question which aspects of parental illness act as the main stressors for children. It is thought that secondary illness-related factors regarding family life and parental functioning contribute to children’s psychosocial risk.

 Further work needs to clarify to what extent an evolving disease such as MS triggers an outbreak of premorbid conflicts within a couple and affects the family structure. Communication is key in managing chronic disease in families. Several researchers emphasize the value of communication and education for children affected by parental illness, thereby promoting integrated psychosocial support in the medical care of people with MS and their families,

 yet few studies have addressed the communication of parents affected by MS with their children and how and when to address the parental disease. Paliokosta et al

 highlighted the importance of communication about parental illness, possible changes to the family structure, and that parents may need support to decide on the “how, what, and how much.”

The present findings imply that communication can be an important factor in a family’s well-being and adjustment, and families may be helped by seeking professional support when they approach the difficult task of disclosure. Parents may need help to consider “how, what, and how much”

 they might disclose to their children about a parental illness and forthcoming changes in family life.

Interestingly, 6 studies in this review found that the effects on children were associated with or mediated at least in part by depression in parents with MS, especially if the mother is affected.

 This is especially relevant considering that the prevalence of depression is high in people with MS,

 but even without an accompanying MS diagnosis, maternal depression has a substantial effect on the psychological well-being of children.

 reached different conclusions about mental health, body image, childhood development, social interaction, and/or school performance, presenting evidence suggesting no difference between children with parents with MS and children with parents without MS. Positive effects of having a parent with MS were also described, such as more prosocial behavior, fewer peer problems, less vulnerability regarding social development, and higher school grades.

 mentioned the seemingly higher social competence of children of people with MS. They assumed that the greater involvement in household duties due to the parent’s illness might be one of the reasons for the positive effect on children’s social interaction.

 It is also possible that the children develop a better reflective ability to regulate their emotions. Further research could test the role of processing emotions in families of people with MS.

Although these findings indicate that a parent’s MS has at least some psychosocial impact, results must be considered with reservation. This systematic review found several methodological issues that limit the validity of the evidence from these studies.

Approximately one-third of the studies did not investigate a comparison group of children with parents without MS, which reduces the validity of the findings. A minority of the studies collected longitudinal data that would allow for some conclusions to be drawn on the continuity of the identified effects, especially considering the psychosocial dynamics of developing children and of MS as an illness. Furthermore, numerous studies failed to describe eligibility criteria, recruitment processes, outcome definitions, adjusted analyses for confounders, or the children’s exposure to their parent’s MS. For future research, children’s data should be analyzed separately for different age groups, and only 1 child per family should be included to avoid clustering effects. The use of instruments that assess data on children by proxy (eg, parents, teachers) should be evaluated carefully. Evidence indicates discrepancies between children’s self-reported outcomes and parental proxy responses.

 Therefore, the by-proxy approach should be applied only in study settings where children are too young to respond themselves or should be used only in addition to speaking with the children themselves.

This systematic review is also not without limitations. Although we searched 3 medical databases and hand searched the references of previous reviews, some relevent data may have been missed. We also included only quantitative and mixed-method studies published in English or German.

Considering the heterogeneous and partially contradictory evidence found in these 28 studies, we emphasize the need for further research. However, the findings in the present review imply that health care providers should ask people with MS about their children’s response to their illness and that parents with MS should talk to their children about it as well. In addition, neurologists need to consider a family perspective when counseling parents with MS. Because this is not a part of medical education, physicians need support and training in how to address these issues. Furthermore, health systems need to develop integrated structures to organize and finance family counseling for people with chronic illnesses. Not every child with a parent with MS will need psychosocial support, but it could be beneficial for parents and physicians to keep in mind that adjustment problems may occur. As a first step, a decision aid for people with MS considering parenthood could raise awareness and thereby help people with MS talk about this topic with their physicians and family members.

Overall, the psychosocial impact of parental MS on children is not yet clearly understood. Evidence suggests negative consequences on children’s psychosocial well-being, but a few studies also indicated positive effects or no difference from children with parents without MS. The variable study quality and contradictory findings emphasize the need for further research to allow definite conclusions and recommendations for clinical practice.

The Psychosocial Impact of Parental Multiple Sclerosis on Children and Adolescents: A Systematic Review

Urinary incontinence (UI) and fecal incontinence (FI) are challenging manifestations of multiple sclerosis (MS) that have historically been treated with limited success. Sacral neuromodulation (SNM) has provided successful resolution of UI and FI in the general population and in patients with neurologic conditions, including MS. We report on 6 patients with MS-related incontinence treated successfully with SNM and review the literature.

 Medical records were reviewed retrospectively to identify patients with MS seeking treatment for incontinence. Six patients were identified, and each is presented with follow-up assessment of the severity of UI or FI.

All 6 individuals with MS had severe incontinence that had been refractory to therapies that included medications and pelvic floor physical therapy. Five patients reported severe UI and 2 patients reported severe FI. Each patient was successfully treated with SNM, with large reductions of incontinence scores and improved quality of life.

 In this case series, SNM was effective as a treatment for UI and FI among patients with MS. These findings confirm other published series that have reported the success of SNM in patients with MS with incontinence. Sacral neuromodulation should be considered as a potential treatment option for patients with MS and UI and/or FI.

From the Reno School of Medicine, University of Nevada, Reno, NV, USA (ET, KS), and Nevada Surgical Associates, Reno, NV, USA (KS). 

Erika Thys, BA, Reno School of Medicine, University of Nevada, 1664 N Virginia St, Reno, NV 89557, USA; 

Multiple sclerosis (MS) is an autoimmune disease primarily targeting the myelin-producing oligodendrocytes of the central nervous system and can cause demyelination and axonal loss in the brain, brainstem, cerebellum, and/or spinal cord.

 These demyelinated lesions give rise to bladder problems in 50% to 90% of patients with MS, including a high prevalence of overactive bladder (OAB) and urinary incontinence (UI).

 The latter was even more frequent in patients with MS if they were disabled and female.

 Fecal incontinence (FI) is found to affect more than 50% of patients with MS, which is far greater than the 2% to 20% prevalence in the general population.

 These symptoms impact the quality of life and social interactions of patients with MS and create additional burdens for caregivers.

The current management of UI and FI in patients with MS begins with first-line behavioral modifications such as bladder training, pelvic floor physical therapy, fluid management, fiber, antidiarrheals, and dietary changes. Historically, second-line therapies for UI are pharmacotherapies, including anticholinergics and ß

-adrenergic agonists, and/or intermittent self-catheterization. Traditionally, third-line therapy options are intradetrusor botulinum toxin, consisting of repeated treatments every 6 to 12 months, or posterior tibial nerve stimulation, consisting of a weekly office-based therapy for 3 months. Advanced therapy is sacral neuromodulation (SNM), involving placement of a neuromodulation lead and a subcutaneous pulse generator.

Sacral neuromodulation has been an effective treatment for UI and FI since its approval by the US Food and Drug Administration in 1997.

 The mechanisms of action for SNM include inhibition of afferent sensory nerves of the bladder and rectum in the spinal cord, modulation of efferent voluntary somatic input to the bladder, and stimulus of autonomic motor nerves.

 Recent studies report 90% effectiveness in clinical trials for treatment of UI and FI.

 However, SNM was originally incompatible with MRI, and SNM is less accepted by insurance carriers for patients with neurologic disorders, including MS. The Centers for Medicare & Medicaid Services (CMS) and many insurance companies continue to exclude those who experience UI due to a neurologic condition, ie, cerebrovascular accident, spinal cord injury, MS, detrusor hyperref lexia, and other types of chronic neurogenic voiding dysfunctions.

In this study, we retrospectively evaluated the efficacy and safety of a newer, fully MRI-compatible SNM treatment for 6 patients with MS who were experiencing refractory UI, FI, or both before treatment.

Under an institutional review board–approved protocol (WCG IRB), a retrospective medical record review was undertaken to identify all patients with MS who sought treatment for UI and FI at the Continence Center of Nevada Surgical in Reno, Nevada. Since 2017, all patients have provided detailed responses to questionnaires regarding bowel and bladder symptoms and the presence of neurologic conditions. Informed consent was obtained from all individual patients regarding responses to questionnaires being used for clinical assessments or publication in any medium. In 2022, 6 patients with MS were identified in the database. All were ambulatory, 2 with the use of assist devices (cane or walker); no one used a wheelchair. Disease severity was not quantified, and to our knowledge, no patient referred to the center with MS declined or was refused SNM therapy. The patient characteristics are described in 

. After SNM implant (Axonics Inc), patients reported, in person or by phone, updated responses to the bladder and bowel questionnaires. The Michigan Incontinence Symptom Index (M-ISI)

 and the Fecal Incontinence Severity Index (FISI)

 were used to assess the severity of symptoms before and, on average, 9 months after therapy.

In coordination with their neurologists, each patient had a percutaneous nerve evaluation. During the procedure, a localization needle was placed into each S3 foramen, and motor response to stimulation was carefully observed, with assessment of anal bellows, perineal lift response, and great toe flexion. In subsequent days, each patient reported that the trial was successful, and SNM implant via the S3 foramen under fluoroscopic guidance was subsequently performed in the operating room using local anesthetic with sedation, with a median implant procedure time of 28 minutes. There were no adverse events during a minimum of 12 months of follow-up.

In this study, 6 patients with MS who sought treatment for UI, FI, or both were identified in the database. Each patient had failed medication therapy and pelvic floor therapy with exercises and biofeedback. One had previous botulinum injections and 1 had a failed previous bladder sling. All 6 patients experienced relief after SNM, as measured by their M-ISI and FISI scores. As depicted in Table 1, the median M-ISI score before SNM was 28 and after SNM was 6. The 2 FISI scores were 41 and 42 prior to SNM and both were 0 after SNM.

All the patients reported that SNM was successful. In addition to the improvements in UI and FI, all the patients reported significant improvements in quality of life. Patients described feeling relieved and having peace of mind going about their daily lives. Patient 1 reported being able to “take a vacation for the first time in years” after noticing that she could go longer periods of time without needing to use the bathroom. Patient 4 reported experiencing “peace of mind” and that the treatment “gave back [his] life.” This has led to an improvement in his sex life and intimacy as he is “no longer worried about having any accidents.” He reported being able to return to work and resume teaching because of SNM therapy. These anecdotes are consistent with the clinical findings that SNM allows patients to reach the toilet in time and enables complete emptying of the bladder, significantly improving quality of life, volume voided, urgency, and urge UI.

Similar to other reports in the literature on older devices (

), this small case series, using newer, MRI-compatible devices, found that SNM was a very effective therapy, resulting in significant improvement in UI and FI for patients with MS.

According to a 10-year retrospective cohort study with 4461 patients with and without MS, more patients with MS were diagnosed as having UI and FI than those without MS.

 Also, FI and UI occur at a younger age in patients with MS than in the general population. Sacral neuromodulation acts via neurologic mechanisms, offering logical plausibility to treat neurogenic incontinence. Even a single previous episode of FI can cause anxiety and lead to isolation and self-imposed restrictions to avoid the risk of embarrassment, shame, or humiliation associated with that incident.

Although not completely understood, SNM’s stimulation of the S2-S3-S4 nerve roots is thought to provide relief through a combination of efferent motor stimulus, blocking afferent inputs, and blunting spinal cord reflex responses and brain responses to detrusor and pelvic floor musculature.

 Published studies support the conclusion that SNM provides relief of UI and FI in patients with neurogenic incontinence, as does this series.

 In practice, however, patients with MS experiencing UI or FI are often considered ineligible for SNM or denied insurance coverage based on preliminary SNM studies, despite more recently published studies demonstrating its efficacy.

 In a prospective cohort study, 17 patients with MS were treated with SNM, and 16 of them (94%) had greater than 70% improvement in urinary dysfunction.

 In a 2018 study, SNM was found to improve FI in 59% to 82% of patients with MS.

 A 4-year prospective study found a statistically significant decrease in involuntary stool loss and urge defecation numbers.

 In a retrospective case series study, 60% of patients (15 of 25) reported clinically significant improvement in symptoms of frequency and incontinence, a decrease in the number of catheterizations, and an increase in void volume.

 A multicenter study analyzed therapeutic success and quality of life over 36 months and reported statistically significant improvements in urinary symptoms.

The present review of the literature found generally favorable results for SNM as a treatment for FI and UI in patients with MS. Engeler et al

 reported that SNM treatment had a high success rate after the failure of conservative treatment options. It improves the volume voided, urgency, and urge UI and reduces postvoid residual. These effects were achieved early in treatment and remained stable thereafter.

 found that SNM was a valid therapeutic option in patients with lower urinary tract symptoms due to MS. Their study confirmed a significant decrease in urgency and frequency among SNM patients and an overall improvement in quality of life.

 also determined that SNM is an effective and safe therapeutic option for treating lower urinary tract symptoms in patients with MS. In addition, Worsøe et al

 also documented SNM and its improvement in FI symptoms.

Limitations of this case series include its small size, limited generalizability, limited duration of follow-up, and that MS disability severity was not formally assessed as an independent variable. These would be important factors to include in larger-scale assessments of SNM in this patient population because SNM may not offer the same effectiveness in more advanced cases of MS. Strengths of this case series include its echoing previous findings in the literature and its raising awareness of MRI-compatible SNM as a therapy deserving of more study.

This experience with SNM in patients with MS revealed no adverse events. None of the patients in this series reported difficulty charging the device, but challenges with charging among older patients and increased, approximately 15-year, battery longevity have led us to a default recommendation of the newer, nonrechargeable device. None of the studies before the present series demonstrated adverse events or raised concern of risks that might warrant restricting SNM therapy for patients with MS. Newer SNM devices are fully compatible with 3-T MRI technology, with the recommendation that the SNM device be temporarily switched off during the scan using the accompanying remote control. Given the neurologic mechanism of pelvic floor dysfunction and incontinence in MS, effective therapy may work through a neuromodulatory mechanism.

The published literature reveals no rationale for the CMS and insurers to exclude patients with MS from effective pelvic floor therapy. The severe negative impact of incontinence and the high success at restoring continence and quality of life found in this and other studies of SNM are additional arguments that patients with MS should no longer be excluded from receiving SNM therapy by insurers. Larger-scale clinical assessment is warranted to determine the best candidates and to confirm the thesis that SNM should be considered when counseling patients with MS on treatment modalities and quality of life related to refractory UI and FI.

Sacral Neuromodulation Therapy for Urinary and Fecal Incontinence in Patients With Multiple Sclerosis: Report of 6 Cases and Literature Review

Interdisciplinary therapies for the management of people with multiple sclerosis (MS) are underappreciated. There is an urgent need to introduce music therapy (MT), either alone or in combination with physical therapy (PT), into clinical practice to achieve synergy with disease-modifying therapies. A holistic approach to rehabilitation for people with MS may mitigate symptoms and reduce polypharmacy, potentially lowering health care costs.

As MS progresses, patients experience a range of worsening symptoms, and many develop psychosocial comorbidities. As disease-modifying therapies delay disability progression, nonpharmacologic treatments become increasingly important. The main aim of PT is to improve or maintain patients’ functional mobility, strength, and flexibility. Because it targets multiple functions, MT can help improve functional and psychosocial domains and may be a valuable intervention to help patients achieve the physical, cognitive, and emotional goals of PT. Exploratory studies showed that MT, alone or in combination with PT, can lead to functional improvements in mobility, balance, gait, and fatigue. Similar to PT, MT also has benefits in improving fine motor skills, cognition, learning, and memory and in providing emotional support.

Both MT and PT have the potential to improve overall well-being and health-related quality of life in physically active patients with MS, and MT can provide added emotional support for those who are less able to engage in physical activity. However, MT is not typically a part of standard of care, and PT visits are limited. Nevertheless, interdisciplinary therapies should be incorporated into clinical practice.

From First Coast Integrative Medicine, Jacksonville Beach, FL, USA (MW); MS Achievement Center, Sacramento, CA, USA (BH); Boyer College of Music and Dance, Temple University, Philadelphia, PA, USA (WLM); A Place To Be, Middleburg, VA, USA (KL, TS); Novartis Pharmaceuticals Corporation, East Hanover, NJ, USA (JLW, WS); and John F. Kennedy Center for the Performing Arts, Washington, DC, USA (RF). 

 Megan Weigel, DNP, ARNP-C, MSCN, 3948 3rd St South, #521, Jacksonville Beach, FL 32082, USA; e-mail: megan.weigel@firstcoastintegrativemedicine.com.

 We thank Chason Goldschmitz, Office of Renée Fleming, for his help coordinating Ms Fleming’s reviews of and input on this manuscript. Medical writing support was provided by Jenny Thorp, BSc, and Claudia Brockmeyer, PhD, of Oxford PharmaGenesis and was funded by Novartis Pharmaceuticals Corporation. This manuscript was developed in accordance with Good Publication Practice guidelines. Authors had full control of the content and made the final decision on all aspects of this article.

 Dr Weigel is a consultant for Biogen, Inc, Bristol Myers Squibb, Celgene Corporation, Inc, EMD Serono, Greenwich Biosciences, Inc, Horizon Therapeutics, Novartis Pharmaceuticals Corporation (Novartis) (independent of the submitted work), Sanofi Genzyme, and TG Therapeutics, Inc and has received speakers’ fees from Bristol Myers Squibb, Horizon Therapeutics, Novartis (independent of the submitted work), and Sanofi Genzyme. Mr Hutchinson has received consultancy fees from Novartis (independent of the submitted work) and speakers’ fees from Biogen, Inc. Dr Magee has received consultancy fees from Novartis (independent of the submitted work). Dr Weiss was an employee of Novartis Pharmaceuticals Corporation, East Hanover, NJ, USA, during the development of this article. Dr Su is an employee of Novartis. Ms Fleming has received consultancy fees from Novartis (independent of the submitted work). Messrs Leong and Sweitzer declare no conflicts of interest.

 Publication of this research was supported by Novartis Pharmaceuticals Corporation.

Multiple sclerosis (MS) is a progressive disease involving demyelination of the central nervous system. It is estimated to affect more than 2 million people worldwide

 and may affect up to 1 million people in the United States.

 As the disease progresses, people with MS may experience worsening of symptoms, including, but not limited to, problems with mobility, balance, spasticity, fatigue, and cognition.

 Many also develop psychosocial comorbidities such as depression, anxiety, social isolation, and suicidal ideation; the lifetime prevalence of developing depression is 50% for individuals with MS.

This article reviews the role of music therapy (MT), a valuable but underappreciated nonpharmacologic rehabilitation therapy for MS, and how MT can support the treatment goals of physical therapy (PT), a more widely used and better reimbursed rehabilitation therapy, leading to improvements in most of the physical and psychosocial domains that are negatively affected in individuals with MS.

 Reimbursement for supportive therapies and the value of telehealth solutions for people with MS are covered as well.

Unmet Need for Interdisciplinary Therapies in MS   

Phenotypes of MS are categorized by patterns of progression and remission as evidenced by disability progression or inflammatory activities such as magnetic resonance imaging changes or clinical relapses.

 Symptoms can manifest in a relapsing-remitting manner or can occur and get progressively worse with no periods of remission.

 Approximately 85% of people with MS are diagnosed as having relapsing-remitting MS (RRMS),

 characterized by intermittent relapses with complete or near-complete recovery.

 Over time, patients are likely to transition to secondary progressive MS (SPMS), characterized by a progressive increase in disability with or without relapses and other inflammatory activities.

 This progression also corresponds with increased brain atrophy.

 If untreated, most patients with RRMS will transition to SPMS within 25 years of disease onset.

Approximately 15% of people with MS have primary progressive MS, where MS symptoms progressively worsen from onset, with no periods of remission.

 Secondary or primary progressive disease is further defined as progression with or without disease activity, which is characterized by progression of symptoms with or without worsening of disease activity as seen by magnetic resonance imaging or clinical activities, such as relapses.

 A higher proportion of patients with SPMS than with RRMS report impairments and difficulties in physical domains (eg, walking, gait and balance, muscle spasm, bladder dysfunction), cognitive domains (eg, attention and processing speed, executive function, memory), and other domains (eg, fatigue, numbness).

 Also, SPMS has a greater negative effect on patients’ working and everyday activities than does RRMS.

Disease progression is associated with increasing disability and is historically quantified in MS by the Expanded Disability Status Scale (EDSS) (

). The EDSS score ranges from 0 (no disability) to 10 (death due to MS) and is based on 8 functional systems with an emphasis on ambulation.

Although disease-modifying therapies (DMTs) are available for MS to delay disability progression and reduce relapses (

), as the disease progresses, symptomatic treatment becomes increasingly important, particularly for SPMS and primary progressive MS. An interdisciplinary approach is required to ensure that people with MS receive supportive therapy for the entire range of symptoms they experience. Indeed, 33% to 80% of patients with MS seek complementary and alternative therapies as part of their care.

 Supportive therapies may include PT, occupational therapy, social services, psychological therapies, speech and language therapy, dietetics, MT, and other creative arts therapies.

Alaina G. Tiani, PhD, DBSM

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