Perspectives on informed assent and bodily integrity in prospective deep brain stimulation for youth with refractory obsessive-compulsive disorder

Abstract

Background

Deep brain stimulation is approved for treating refractory obsessive-compulsive disorder in adults under the US Food and Drug Administration Humanitarian Device Exemption, and studies have shown its efficacy in reducing symptom severity and improving quality of life. While similar deep brain stimulation treatment is available for pediatric patients with dystonia, it is not yet available for pediatric patients with obsessive-compulsive disorder, although soon could be. The prospect of growing indications for pediatric deep brain stimulation raises several ethical concerns relating to bodily integrity, the ability to offer informed assent, and the role pediatric patients play in the decision-making process.

Objective

The aim of this study is to solicit and assess the views of stakeholders (children, parents, clinicians) on pediatric assent, autonomy, and bodily integrity in the context of potential pediatric deep brain stimulation for obsessive-compulsive disorder.

Methods

Semi-structured interviews were conducted with pediatric obsessive-compulsive disorder patients (n = 21), caregivers of pediatric obsessive-compulsive disorder patients aged 14–18 (n = 19), and clinicians with experience treating refractory obsessive-compulsive disorder (n = 25). Interviews were transcribed and coded in MAXQDA 2018 and 2020 software and processed for thematic content analysis to isolate and compare specific themes.

Results

A majority of respondents (74%, 48/65) across all three stakeholder groups voiced that the decision-making process should be collaborative and involve everyone (clinicians: 84% or 21/25, caregivers 71% or 15/21, and patients 63% or 12/19). We identified a split between respondents’ views on who should have the final say in the event of disagreement (38% or 25/65 favored the patient versus 35% or 23/65 favoring caregivers). A split between respondents also emerged concerning the maturity relevant for deep brain stimulation decision-making, with 45% (29/65) favoring developmental maturity (age/physiological development) and 45% (29/65) favoring decisional maturity (capacity to understand and weigh information). A majority of clinicians indicated that they would not move forward with deep brain stimulation without securing patient assent (80% or 20/25), with some stating the only exception is if patient quality of life was very poor and/or they lacked insight. Both caregivers and patients expressed a significant respect for the patient's right to bodily integrity, with 67% of caregivers (14/21) and 68% of patients (13/19) justifying patient involvement in decision-making specifically with reference to infringements of bodily integrity.

Conclusion

Our findings demonstrate that despite broad agreement across stakeholders that the decision-making process for pediatric deep brain stimulation for obsessive-compulsive disorder should be collaborative and somehow involve pediatric patients, there is disagreement about what this process entails and what factors determine patient involvement in the process. However, there is agreement that children have a right to bodily and brain integrity, which should only be infringed upon in rare circumstances.

Keywords

Bioethics and medical ethics clinical ethics minors informed consent mental health therapies and neurosciences psychosurgery deep brain stimulation

Introduction

Deep brain stimulation (DBS) is a form of neuromodulation where leads are surgically inserted into the brain to provide targeted electrical stimulation for symptom relief. DBS was first used in the US for treating essential tremor and the tremors resulting from Parkinson's disease (PD) after clinicians found that electrical stimulation to particular areas of the brain could produce effects similar to lesion/ablation therapy but which could be modulated.¹ Following developments since the 1980s in stimulation, devices, and targeting, DBS has been used to treat a variety of both movement and psychiatric conditions,² including essential tremor and PD,^3,4 epilepsy, adult and pediatric (≥18) dystonia,^5–7 Tourette syndrome (TS),^8,9 and adult obsessive-compulsive disorder (OCD).^10–13

Researchers continue to investigate the viability of DBS for treating other conditions, such as major depressive disorder and Alzheimer's disorder.^1,2 Additionally, recent studies have begun to research whether pediatric patients with conditions treated with DBS in adults (e.g. pediatric TS or pediatric OCD) may safely benefit from early DBS intervention. This research is complicated by distinct state and federal regulations pertaining to when a minor may legally consent to a given medical treatment; for example, age for medical consent ranges 14–18 in the European Union depending on the country, and some US states recognize the common-law doctrine of the “mature minor” (typically ≥14).^14–16 Where pediatric patients are not legally permitted to provide informed consent for medical treatment, their involvement in medical decision-making raises ethical and logistical questions about the relationship between autonomy and consent as well as the extent (and role of) of a child's right to bodily integrity.

In 2009, the US FDA approved DBS for adult OCD under the Humanitarian Device Exemption (#HO50003 09M-0090) (HDE), making OCD the first psychiatric disorder to receive some form of FDA approval for DBS treatment.¹⁷ In the US, HDE approval does not require evidence that DBS for OCD is effective. While DBS is offered to pediatric patients with refractory dystonia under the FDA's HDE (#H020007 03M-0157), it is not yet approved for refractory OCD in pediatric patients.¹⁸ Presently, it is estimated that there are over 100,000 cases of pediatric refractory OCD in the US alone.^19,20 DBS for refractory OCD in adult patients has demonstrated efficacy of long term reduction in symptoms.^10–12,21 These results, paired with the efficacy of DBS for refractory dystonia in pediatric patients, suggest that DBS may be effective in treating refractory OCD in pediatric patients.^5,6

This paper shares and analyzes the views of US stakeholders (pediatric patients, caregivers, and clinicians) on pediatric assent, autonomy, and bodily integrity in the context of prospective pediatric DBS (pDBS) for OCD. Previous studies have investigated the ways in which these concepts (assent, autonomy, and bodily integrity) impact medical decision-making regarding pDBS for non-OCD conditions; this is the first to focus on a primarily psychiatric condition.²² Existing work on pDBS for OCD has been limited to the perspectives of caregivers of children with OCD and clinicians who treat pediatric OCD on the use of pDBS; this is the first to analyze the views of youth diagnosed with OCD and to compare them with the perspectives of these other stakeholders.^23,24

Salient ethical dimensions of pDBS decision-making

Certain ethical dimensions of medical decision-making become more salient when treating pediatric patients or are not present when treating adult patients with DBS. Below we briefly discuss how some of these dimensions have been addressed in previous research.

The first such dimension made salient by pDBS for OCD concerns the pediatric patient's right to bodily integrity (respecting the physical boundaries of a person), whether this right may be violated or infringed and under what conditions. In the case of autonomous agents, having one's right to bodily integrity violated concerns “any penetration into a bodily orifice, breaking of the skin, or alteration of a person's physical form” where they do not consent.²⁵ Agents who are not autonomous (or perhaps not fully autonomous, like young children) can still have their right to bodily integrity violated even though they are unable to provide informed consent. For example, in the US, children enjoy a constitutional right to bodily integrity, which is legally recognized in many contexts.²⁶ Theorists disagree about the conditions under which this right is violated and when an intervention should be considered a justified infringement (e.g. in the patient's best interest), or no infringement at all.²⁷

Directing our attention to the pediatric patient's right to bodily integrity leads to the second dimension made salient by pDBS for OCD, which concerns the core practice of securing informed consent or assent from pediatric patients prior to treatment. Receiving informed consent from a patient is both ethically and legally important; it requires disclosing relevant medical information, assessing patient understanding and decisional capacity, and securing legal permission for treatment.^28,29 Not only does the legal age of consent differ across states and countries, a person may exhibit appropriate decision-making capacity before reaching the age of consent. For this reason, there are two senses of informed consent to distinguish which will become relevant later in this paper: autonomous authorization and institutional/effective consent. Faden, Beauchamp, and King (who propose the distinction) define autonomous authorization as being given when “a patient or subject with (1) substantial understanding and (2) in substantial absence of control by others (3) intentionally (4) authorizes a professional (to do [X]).”³⁰^(p278) This sense of informed consent focuses on the autonomous act of the patient to authorize (rather than merely agree or comply with) some plan or procedure. By contrast, effective consent refers to authorization from a patient, which “has been obtained through procedures that satisfy the rules and requirements defining a specific institutional practice in health care or in research.”³⁰^(p280) This distinction allows us to characterize situations where a patient may provide autonomous authorization for care even if they are unable to provide effective consent (e.g. due to laws governing age of consent). In such cases, typically the patient must (or ideally) provide what is termed assent, in addition to caregiver or guardian consent for treatment.^31–33

The confluence of these two ethical dimensions (bodily integrity and the importance of consent/assent) lead directly to a third, viz. the proper role of the guardian or caregiver in instances where treatment like DBS is indicated, given that pediatric patients (1) have a right to bodily integrity, which must be respected, and (2) can potentially give autonomous authorization absent effective consent. This necessitates both patient and caregiver involvement in some form, and researchers on pDBS have proposed some form of dynamic shared decision-making. For example, Farah Focquaert argues in favor of a model of decision-making that avoids the risks resulting from either extreme of the decision solely residing with either the caregiver or the child patient. Instead, she favors mutual medical decision-making where there is a dynamic back-and-forth between the medical expert, the patient, and the caregiver negotiating the patient's understanding of their situation and the options available.³⁴ This necessitates ongoing conversations as well as private meetings with the patient to ensure both that there is no inappropriate influence being exerted by family but also that there is proper psychosocial support from family.^35,36

Finally, we must note that pediatric decision-making is highly heterogenous and depends significantly on both a pediatric patient's age as well as their relative maturity and the nature of the decision being made. The level of involvement for a 7-year-old is much different from that of an adolescent or teenager, and this is especially true given the seriousness of neuromodulation as a treatment. While the patient respondents for our study fall within the adolescent category by age, all the respondents were asked about DBS for patients ≤18 generally. In the discussion, we consider how the relative age (e.g. young child versus late adolescent) impacts our conclusions.

Methods

Participants and procedures

Semi-structured interviews were conducted with US-based clinicians (n = 25) with experience treating pediatric OCD, as well as pediatric patients aged 14–18 (n = 19) and caregivers of patients (n = 21) with OCD to understand stakeholder perspectives on informed consent and decision-making about pDBS. An interview guide was developed based on issues raised in the clinical and ethics literature and through discussions with DBS and movement disorders experts. A draft of the guide was reviewed by an advisory team and piloted with a psychologist specializing in refractory OCD in children and adults, resulting in minor changes for improved clarity. The study was approved by the Institutional Review Board at Baylor College of Medicine (approval number: H-45996). Interviews were conducted via phone or Zoom, lasting an average of 50 min. Table 1 includes demographic information for the clinician respondents; table 2 contains demographic information for caregiver respondents; table 3 includes demographic information for patient respondents. Note that one patient, one caregiver, and one clinician were excluded from analysis, but their demographic information has been included below.

Table 1.

Demographic information for the clinician respondents.

Clinicians	n = 26	%
Gender
Male	12	46
Female	8	31
Prefer not to answer	6	23
Race/Ethnicity
Non-Hispanic/Latinx, White	16	61.5
Hispanic/Latinx	2	8
Asian	1	4
Prefer not to answer	7	27
Clinical Background/Training
Psychologist	14	54
Psychiatrist	8	31
Licensed professional counselor or marriage and family therapist	3	11.5
Prefer not to answer	1	4
Years treating children and adolescents with OCD
5–10	7	27
10–15	5	19
15–20	3	11.5
20–25	2	8
25+	3	11.5
Prefer not to answer	6	23

Table 2.

Demographic information for the caregiver respondents.

Caregivers	n = 22	%
Gender
Male	3	13.6
Female	16	72.3
No response	3	13.6
Race/ethnicity
Hispanic/Latinx	1	4.5
White or European American	18	82
Asian	1	4.5
No response	2	9.1
Age
41–45	4	18.2
46–50	7	32
51–55	5	23
56–60	3	13.6
No response	3	13.6

Table 3.

Demographic information for the patient respondents.

Patients	n = 20	%
Gender
Male	3	15
Female	12	60
Trans male	1	5
Nonbinary	1	5
Prefer not to answer	0	0
No response	3	15
Race/ethnicity
Hispanic/Latinx	5	25
White or European American	12	60
Prefer not to answer	0	0
No response	3	15
Age
14	4	20
15	0	0
16	3	15
17	2	10
18	0	0
Prefer not to answer	8	40
No response	3	15

Data analysis

Interviews were audio-recorded, transcribed verbatim, and analyzed using MAXQDA 2018 and 2020 qualitative data analysis software. Team members (led by a qualitative methods expert and medical anthropologist) developed a shared codebook to identify thematic patterns in caregivers’, clinicians’, and patients’ responses to questions about their views on potential pDBS treatment. Team members independently coded transcripts to ensure inter-coder reliability in application of the codebook. We then iteratively abstracted the coded information and identified thematic patterns across interviews utilizing thematic content analysis, calculated final theme and sub-theme frequencies, and completed primary data analysis.

Ethics approval obtained

This study was approved by the Institutional Review Board for Human Subject Research for Baylor College of Medicine and Affiliated Hospitals (approval #H-45996).

Results

Collaboration in decision-making

A majority of all respondents (74% or 48/65) indicated that the decision-making process for DBS should be collaborative and involve patient, caregiver, and clinician(s). This sentiment was most prominently voiced by clinicians (84% or 21/25), followed by caregivers (71% or 15/21), and then patients (63% or 12/19). On the topic of collaboration in decision-making, one clinician said: “I think it's definitely something I would discuss with them and with their parents. I would want both parties - the adolescents and the parents - to be informed and to be able to talk through the decision together” (C09).

This focus on shared decision-making was frequently echoed. An example of this included a clinician raising the need for a diverse group involved in the decision-making process: “Multiple people are very involved in the decision. And I think what's important is having people who have the background, the medical knowledge, the OCD knowledge, and then balancing that with what the parents understand, who's educating the parents, who's educating the child, and having everyone sort of come to the table and really having quite a frank discussion as a group about it,” (C25).

Some clinician respondents (44% or 11/25) expressed that one goal of collaborating with a pediatric patient on treatment decision is to ensure that the patient primarily was informed about the treatment, trajectory, and risks of DBS. As one clinician said, “But I think the biggest thing is trying to educate and really […] being able to communicate ideas in a way that whoever you're speaking to can understand and make sense to them, and that's going to be different for adolescents and kids than it's going to be for adults, and I think finding a way to make sure that you're explaining in a way that they actually understand what you're talking about” (C21).

Several clinicians gave more detail and described a tiered collaboration approach, which begins with one-on-one meetings with patients and caregivers separately before integrating them into a group discussion: “Okay. I think I'd start with the parents. I'd at least sort of put out the feelers with the parents before broaching it with the teen, because I think it can be overwhelming, or it can seem a little frightening that, “Oh my gosh, is something so wrong with me I need to have brain surgery?” Correct? So, I would use the parents, assuming they're supportive, motivated and on board parents. I would use them as the first screen to see how they think about it, and then to gauge how they think their teen would feel about it. Then if they think it's okay, then I would engage the teen in the process,” (C18).

Final decider for DBS treatment

Favoring a collaborative decision-making process does not necessarily mean that no one party has more of a say, or has the final say, in whether to engage with DBS treatment. The respondents differed in their perspectives of who should have the final decisional say in pursuing DBS treatment. One-third of the respondents (35% or 23/65) expressed that, when patient and caregiver conflict about trying DBS, the caregiver should have the final say: “Listen, as a parent, you make a lot of decisions for your kids that they don't like and they don't want. Ultimately, I would have to weigh his feelings in on the decision, but ultimately I would make the decision,” (CG02).

Among caregivers in particular, 62% voiced that the caregivers should have the ultimate decision about DBS treatment (13/21). This contrasts the sentiment expressed by over a third of the respondents (38% or 25/65), who said the patient should have the final say in conflicts. On this, one patient said: “I wouldn't want my parents to make decisions for me […] because I'm the one who is going to have to live with it. So I guess, just being present in what the doctor has to say and being educated on what it's going to do. I would want to know everything about it if it's going to be in my brain,” (PT15). Among patients in particular, 47% (9/19) expressed that patients should have the ultimate decision about DBS.

Bodily integrity of pediatric patients

A recurring theme across the respondents was the consideration of the pediatric patient's right to bodily integrity as a reason to secure their assent and involve them in the decision-making process (31/65 respondents or 48%). This value was most strongly represented among caregivers (67% or 14/21) followed by patients (68% or 13/19). One caregiver said: “If I just took her to the hospital to get brain surgery, she would never trust me again, or she would probably run away. […] It's her body. She's almost 15. I don't sneak vegetables into her food. I wouldn't sneak an electrode or whatever into her brain,” (CG13).

Compare this to what one patient had to say about their involvement and their bodily integrity: “Oh, I'd be so involved. Because I get, parental consent and stuff. Say if, I don't know, if I wanted plastic surgery for something I would have to say yes and my parents would have to say yes too. But for brain surgery, I would be the one saying yes. My parents don't get to make that decision for me. That is my choice,” (PT16).

Generally, with respect to bodily integrity, the respondents did not distinguish between interference with the body or the brain and would often use them interchangeably.

Securing patient assent

Clinicians in particular (88% or 22/25) voiced that pediatric patient assent would be necessary for them to suggest moving forward with DBS. One clinician said: “You have to make sure they have a good informed assent. They're adolescents, so they legally can't consent, but a good informed assent where they understand all of the potential risks and the potential benefits, and that those are not glossed over. I know in a lot of medical settings, I've had family who are in hospitals, and procedures and things like that, a lot of times, the risks are a little bit glossed over, especially in front of kids,” (C03).

Several clinician respondents indicated there was only a narrow set of disparate circumstances under which they would consider pDBS without patient assent, nearly all of which concern either:

the patient lacking insight into their OCD (“Okay, it would depend on … clinically, it would depend on the amount of insight the child has. If I truly believed that DBS would help the kid, because they can't see it because of the OCD, yes. I would have no problem with that. No problem whatsoever. I wouldn't like it. But I wouldn't have a problem with it,” (C26));

having extremely low quality of life due to symptoms (“So, I think only in the case where it was determined perhaps that the person wasn't competent to make their own decisions in terms of caring for themselves, so maybe if someone were on an inpatient unit and it was determined that they didn't have the capacity to take care of themselves, like in terms of because their OCD was so impairing, maybe in that case, I could see a case being made for moving forward, even if the individual didn't, if the kid didn't assent,” (C16));

having symptoms that are prohibitive of securing assent (“So if they have health-related OCD symptoms and it's like, no, if you touch me I'm going to die of brain crabs or something, then yeah, that weighs very differently in my mind than my OCD sucks and it's making my life miserable but I know who I am and I have some real objections to what this is going to do, what this is going to be like,” (C08)).

Maturity of patient—developmental versus decisional

A split was seen in stakeholder conceptualization of patient preparedness to be involved in the decision-making process for DBS. The views were chiefly divided between the respondents who prioritize the patient's developmental maturity (being fully developed physiologically) as opposed to those prioritizing the patient's decisional maturity (having the capacity to weigh and consider information, regardless of biological age).

Developmental maturity—the patient's level of biological or physiological development—was viewed by 45% of the respondents (29/65) as being crucial for moving forward with pDBS for OCD. A meaningful number of respondents (18/65 or 28%) articulated a loose continuum of development and participation, where pediatric patient involvement in the decision-making process is minimal for young children but more involved for adolescents and teens. Similarly, a meaningful number of respondents (17/65 or 26%) specifically indicated that they would want to wait until the patient was at least 18 years old before receiving DBS due to brain development. Consider what one clinician said about brain development and maturity: “I think the strongest [consideration] of all is the developing brain and the degree that DBS may have consequences for brain development that are hard to predict before getting to either age of adult level consent or to a point where we're confident that all the brain regions involved are fully developed. I think that's probably the most compelling argument that I would have for waiting until 18 or over,” (C13).

Decisional maturity—the patient's preparedness to understand and weigh various considerations for and against treatment—was viewed by 45% (29/65) of the respondents as being more important for decision-making than biological age, including half of clinicians and over half of caregivers (13/25 and 12/21, respectively). One clinician underscored that understanding and decisional maturity are what truly matter for assent: “It's actually about the true meaning of consent, it's about everybody understanding what can happen if we do. What can happen if we don’t? The kid could be 15 and get it or somebody can be 26 and not get it. It's a lot more complicated than that. I really hate the 18-year-old cut off thing because it's so black and white,” (C26).

Comparing this similar sentiment echoed by a caregiver that decisional maturity is highly variable and particular to the individual: “Oh, I don't worry about the legal consent. To me, it's the mental capacity. So I don't think age matters […] I feel like just like some kids are ready to drive, or vote, or drink at certain ages, not just because they're state mandated, some are ready before others. Some never are,” (CG15). On this view, it is more important to assess patient maturity on a case-by-case basis rather than use of age-cutoffs, since some patients may be more “decisionally mature” at a younger age than others.

As that caregiver noted, this view of decisional maturity can cut both ways; consider that a minority of the respondents favoring decisional maturity (24% or 7/29) voiced that, because decisional maturity is decoupled from age, there is no guarantee that 18-year-old patients (who are legally able to decide on DBS for themselves) have the requisite maturity. As one caregiver said, “Even if they're the legal age of consent they're still not mature enough to know. I mean, just because you're 18 or whatever does not mean that you have the information you need to make the right decision,” (CG12).

Discussion

Our findings highlight conceptual and ethical issues raised by the prospect of pDBS for refractory OCD and provide empirical support for some existing analyses involving securing informed assent, respecting bodily integrity, and collaborative participation in decision-making.

One aspect of pDBS decision-making generally that must be addressed first is: why not wait until the patient is older if we are concerned about either interfering with brain development and/or guaranteeing informed consent is obtained from the patient? The answer to this question depends on the complex interplay of several factors; different conditions have different ages of onset, available treatment alternatives (with variable efficacy), and progressions such that delaying DBS for one condition may be more harmful than for another. For example, the mean age of onset for Gilles Tourette Syndrome (GTS) is 7 years,^22,37 whereas the average age of onset for OCD is 19.5 years with 50% experiencing adolescent onset of symptoms (around age 10).^38,39 Further complicating this, pDBS is predominantly appropriate for refractory conditions, which are resistant to standard treatment. For an adult to be eligible for DBS for OCD, they must have tried cognitive behavioral therapy and multiple medications without relief.^11,40,41 What is worse is that OCD often takes comparatively longer to diagnose than other conditions treatable with DBS (the average time to treatment after onset of OCD symptoms is 11 years).³⁹

As Farah Focquaert notes, some conditions may remit on their own (e.g. approximately 75% of children diagnosed with GTS remit by the time of brain maturation)⁸ while others, such as dystonia,⁶ remain the same or worsen over time. If a patient receives pDBS for a condition the symptoms of which would have otherwise resolved (or become subclinical) without intervention, resulting harm or disadvantage to the patient from pDBS would represent a wronging of the patient.³⁵ Conversely, the costs of delaying pDBS until the age of legal consent differ dramatically from one condition to another. As Munoz et al. note, the severity of pediatric dystonia can cause pain and physical distortions, which at best interfere significantly with activities of daily living and at worst are life-threatening.⁴² Comparatively, while severe pediatric OCD can drastically interfere with daily life and is extremely distressing, it is not life-threatening. Furthermore, a recent analysis of clinician perspectives on pDBS candidacy for dystonia indicated that young age/early intervention is a primary candidacy consideration.⁴³ This dovetails with research suggesting that pediatric and adult DBS interventions early in the onset of symptoms are more effective and have better outcomes than treatment that occurs later.^5,7,44–46 And so, while the costs of delaying DBS for OCD are less dire than for conditions like dystonia, early treatment can have a significant impact on the trajectory of the illness.

A central theme emerging from the data concerned the need to secure patient assent in the autonomous authorization sense of informed consent. As discussed, this contrasts with the legal or institutional sense of effective consent. As a foundational ethical and legal feature of medicine, patients have the legal right to be properly informed about their condition and possible treatment options, including side effects and risks of each option.²⁸ Crucially, pediatric patients are unable to provide effective consent because they are thought to still be developing the capacity for autonomy.³¹ Generally, in pediatric medical decision-making in the US, clinicians must secure consent of the guardian as well as assent of the patient.^47–49 Pediatric assent is obtained to move forward with treatment or research except in limited, exceptional circumstances, but there are (1) not clear federal guidelines about how to obtain assent and (2) disagreements about the ethical or theoretical grounds for pediatric assent.^29,31 When our respondents spoke about assent, they implicitly spoke in ways consistent with the distinction between autonomous authorization and effective consent outlined earlier, as when CG015 indicates that a patient's biological age (i.e. ability to give effective consent) is not what is most relevant but instead something like what we have called their decisional maturity (ability to give autonomous authorization).^a

There is an asymmetry built into the decision-making relationship privileging the caregiver; while it is possible to move forward with DBS with only caregiver consent, it is not possible to move forward with DBS with only the patient's autonomous authorization (assent). Guardians have the legal authority to give effective consent on behalf of pediatric patients and the ethical obligation to act as fiduciaries, choosing treatment courses that are in the best interest of the patient who cannot give effective consent.⁴⁹ Challenges arise when the caregiver and patient disagree about DBS, including the degree to which caregiver interests and values influence the patient's interest in, or commitment to, DBS therapy. For example, caregivers may be motivated to press for DBS treatment without having thoroughly tried less invasive treatment options if they are overwhelmed with patient symptoms and their impact on the family. Developing decisional aids geared toward shared decision-making in pediatric contexts should help standardize these conversations and provide a blueprint for comparable care across institutions.^b

A second central issue raised by pDBS and our findings concerns the right to bodily integrity for children and the invasive nature of DBS treatment. In its simplest iteration, the right to bodily integrity is “the right not to have your body touched or your body interfered with without your consent”—broadly, a right against physical interference.⁵⁰^(p568) Bodily integrity is distinct from the right to autonomy, although they are intimately related. This distinction is important, since pediatric patients are thought to lack a legal right to autonomy (which is why they cannot give institutional consent) while possessing a constitutional and moral right to bodily integrity.²⁶ Our finding that caregivers (67% or 14/21 CGs) and patients (68% or 13/19 PTs) cite bodily integrity as a reason to involve patients in the decision-making process demonstrates that both of these stakeholder groups consider DBS to be a serious medical procedure that warrants increased participation on the part of the pediatric patient. This signals a shared value between caregivers and patients about the importance of maintaining the patient's right to bodily integrity.

There are different conceptions of the right to bodily integrity that could inform whether specific actions constitute an impermissible infringement of this right. One conception of this right casts infringements as resulting from any “serious” bodily intervention while others, for example, Mazer's recent view, emphasize that interventions may not even infringe upon the patient's bodily integrity if they are in the patient's best interest.²⁷ On this first conception, DBS invades bodily integrity and infringes on that right because it requires the implantation of both a device and leads to deep brain regions and is thus a “serious” bodily intervention in the strict sense, especially when compared to the current leading treatments for OCD: pharmacotherapy and cognitive behavioral therapy. On Mazor's view of the right to bodily integrity, decisions made by a fiduciary (even those that are “serious” bodily interventions) are not even infringements when they are in the pediatric patient's best interest, meaning in some cases pDBS without patient assent would not infringe their rights. Patients and caregiver responses in our study align closer with the “serious encroachment” view than Mazor's view given their general hesitancy to treat patients without their assent qua autonomous authorization.

Interestingly, clinicians and caregivers said they would only move forward with DBS without patient assent if either the patient's quality of life was abysmal and/or the patient lacks insight into the severity of their OCD. This may signal conditions under which caregivers and clinicians view DBS absent assent as sometimes being a justified infringement, although, these conditions have disparate justifications.^c

Questions about the boundaries of bodily integrity are fundamental to our current investigation because pediatric patients are still in the process of psychological, neurological, and physiological development, unlike adult DBS patients.^51,52 As lead site mapping in the brain for OCD becomes better understood and more reliable, the risks of adverse effect on pediatric patients will hopefully abate. It is currently unclear when DBS treatment will be in the best interests of pediatric patients given the above concerns, so we cannot say definitively that the caregiver's choice to pursue DBS absent assent would be justified.

These two issues—that of assent and the right to bodily integrity—coalesce into a third: the role of the guardian. We believe that our findings provide some empirical support for Focquaert's analysis of the role of the guardian in pDBS decision-making. For example, Focquaert offers two “defining elements” of ideal collaborative decision-making in pDBS: (1) an exchange of information between clinicians, caregivers, and patients (at age-appropriate levels) as well as (2) involvement from all stakeholders in the decision-making process in reaching a mutual decision.³⁵^{(pp452–454)} Nearly three-quarters of the total respondents voiced that collaborative discussion should be used to ensure that patients partake in the decision-making process and come to an age-appropriate understanding of their condition, the treatment, and have the opportunity to ask questions, in line with (1). Some clinicians described a tiered process where they act as a kind of intermediary, meeting with the patient and caregiver separately before having a group meeting, with the goal of evaluating each of their attitudes and reasons for seeking treatment, and their understanding of its expected efficacy and maintenance. Concerning (2), caregivers and clinicians were acutely aware of the risks from excluding the patient from discussions (e.g. “If I just took her to the hospital to get brain surgery, she would never trust me again”) and the importance of reaching a mutual decision between all stakeholders.

Forming a set of best practices for decision-making in these contexts, perhaps involving a decision aid, will go a long way toward helping standardize these treatment conversations, providing suggested guidelines for DBS centers around the world to rely upon and further refine over time as the volume of pDBS increases and new treatment targets are identified. These questions become even more pressing when we compare increased interest in DBS for pediatric patients for a variety of conditions alongside the relative lack of pediatric neuroethics research on pDBS: there are presently 82 clinical trials in the US for DBS where pediatric patients are eligible for enrollment, and an increasing number of studies focusing on pDBS for both movement and psychiatric disorders.^4,6,8,53,54

Conclusion

Refractory OCD is a debilitating illness that impacts tens of thousands of people across the world, and presently it is the only psychiatric illness to receive an FDA HDE in the US for DBS treatment in adults. Current data shows adult DBS for OCD to be effective at reducing symptom severity, but novel ethical issues arise with the use of DBS in pediatric OCD patients. Our study has pinpointed that these issues surround when and how to secure patient assent and to balance this against caregiver informed consent, the potential for DBS to disrupt ordinary brain development in these patients given its nature, and the question of how to organize a collaborative decision-making framework that respects not just the authority of parents to decide for their children but also the role children play in agreeing to (and understanding) their own care and their right to bodily integrity. Since DBS is continually being researched for new treatment applications, and as DBS use across the world increases, setting shared guidelines surrounding collaborative decision-making for DBS helps provide blueprints for DBS centers not currently treating pediatric patients but will soon may, and may also inform future discussions about DBS for adults who lack capacity.^55,56

Footnotes

Declaration of conflicting interests

Dr. Storch reports receiving research funding to his institution from the Ream Foundation, International OCD Foundation, and NIH. He is a consultant for Brainsway and Biohaven Pharmaceuticals. He owns stock less than $5000 in NView. He receives book royalties from Elsevier, Wiley, Oxford, American Psychological Association, Guildford, Springer, Routledge, and Jessica Kingsley.

Funding

The authors disclosed receipt of the following financial support for the research, authorship, and/or publication of this article: This work was supported by the National Institute of Mental Health (grant number 1RF1MH121371-01).

ORCID iD

Jared N Smith

Notes

References

Frey

Cagle

Johnson

, et al. Past, present, and future of deep brain stimulation: Hardware, software, imaging, physiology and novel approaches. Front Neurol 2022; 13: 1–20. https://www.frontiersin.org/articles/10.3389/fneur.2022.825178. Accessed May 24, 2023.

Barrett

. Psychiatric neurosurgery in the 21st century: overview and the growth of deep brain stimulation. BJPsych Bull 2017; 41: 281–286.

Hariz

Blomstedt

. Deep brain stimulation for Parkinson’s disease. J Intern Med 2022; n/a. doi:https://doi.org/10.1111/joim.13541

Flora

Perera

Cameron

, et al. Deep brain stimulation for essential tremor: a systematic review. Mov Disord 2010; 25: 1550–1559.

Elkaim

Alotaibi

Sigal

, et al. Deep brain stimulation for pediatric dystonia: a meta-analysis with individual participant data. Dev Med Child Neurol 2019; 61: 49–56.

Hale

Monsour

Rolston

, et al. Deep brain stimulation in pediatric dystonia: a systematic review. Neurosurg Rev 2020; 43: 873–880.

Mehrkens

Borggraefe

Feddersen

, et al. Early globus pallidus internus stimulation in pediatric patients with generalized primary dystonia: long-term efficacy and safety. J Child Neurol 2010; 25: 1355–1361.

Coulombe

Elkaim

Alotaibi

, et al. Deep brain stimulation for Gilles de la Tourette syndrome in children and youth: a meta-analysis with individual participant data. J Neurosurg: Pediatrics 2019; 23: 236–246.

Schrock

Mink

Woods

, et al. Tourette syndrome deep brain stimulation: a review and updated recommendations. Mov Disord 2015; 30: 448–471.

10.

Mar-Barrutia

Real

Segalás

, et al. Deep brain stimulation for obsessive-compulsive disorder: a systematic review of worldwide experience after 20 years. World J Psychiatry 2021; 11: 659–680.

11.

Cruz

Gutiérrez-Rojas

González-Domenech

, et al. Deep brain stimulation in obsessive-compulsive disorder: results from meta-analysis. Psychiatry Res 2022; 317: 114869.

12.

Kahn

Sutton

Winston

, et al. Deep brain stimulation for obsessive-compulsive disorder: real world experience post-FDA-humanitarian use device approval. Front Psychiatry 2021; 12: 1–13. https://www.frontiersin.org/articles/10.3389/fpsyt.2021.568932. Accessed November 7, 2022.

13.

Haber

Yendiki

Jbabdi

. Four deep brain stimulation targets for obsessive-compulsive disorder: are they different? Biol Psychiatry 2021; 90: 667–677.

14.

Consenting to medical treatment without parental consent. European Union Agency for Fundamental Rights. Published November 14, 2017. Accessed June 27, 2023. http://fra.europa.eu/en/publication/2017/mapping-minimum-age-requirements-concerning-rights-child-eu/consenting-medical-treatment-without-parental-consent

15.

Boceta

Martínez-Casares

Albert

. The informed consent in the mature minor: understanding and decision-making capacity. Anales de Pediatría (English Edition) 2021; 95: 413–422.

16.

Coleman

Rosoff

. The legal authority of mature minors to consent to general medical treatment. Pediatrics 2013; 131: 786–793.

17.

Humanitarian Device Exemption (HDE) OCD. Accessed November 7, 2022. https://www.accessdata.fda.gov/scripts/cdrh/cfdocs/cfhde/hde.cfm?id=H050003

18.

Humanitarian Device Exemption (HDE) DYSTONIA. Accessed November 7, 2022. https://www.accessdata.fda.gov/scripts/cdrh/cfdocs/cfhde/hde.cfm?id=H020007

19.

Denys

. Pharmacotherapy of obsessive-compulsive disorder and obsessive-compulsive Spectrum disorders. Psychiatric Clin North Am 2006; 29: 553–584.

20.

Bloch

Storch

. Assessment and management of treatment-refractory obsessive-compulsive disorder in children. J Am Acad Child Adolesc Psychiatry 2015; 54: 251–262.

21.

Kohl

Schönherr

Luigjes

, et al. Deep brain stimulation for treatment-refractory obsessive compulsive disorder: a systematic review. BMC Psychiatry 2014; 14: 214.

22.

Behmer Hansen

Dubey

Smith

, et al. Paediatric deep brain stimulation: ethical considerations in malignant Tourette syndrome. J Med Ethics 2020; 46: 668–673.

23.

Weinzimmer

Schneider

Cepeda

, et al. Perceptions of deep brain stimulation for adolescents with obsessive-compulsive disorder. J Child Adolesc Psychopharmacol 2021; 31: 109–117.

24.

Muñoz

Kostick

Torgerson

, et al. Pressing ethical issues in considering pediatric deep brain stimulation for obsessive-compulsive disorder. Brain Stimul 2021; 14: 1566–1572.

25.

Earp

. The child’s right to bodily integrity. In: Ethics and the contemporary world. London UK: Routledge, 2019, pp.217–235.

26.

Hill

. Constituting children’s bodily integrity. Duke Law J 64 (2015): 1295–1362.

27.

Mazor

. On the child’s right to bodily integrity: when is the right infringed? J Med Philos: Forum Bioethics Philos Med 2021; 46: 451–465.

28.

Informed Consent. American Medical Association. Accessed November 7, 2022. https://www.ama-assn.org/delivering-care/ethics/informed-consent

29.

Katz

Webb

, COMMITTEE ON BIOETHICS , et al. Informed consent in decision-making in pediatric practice. Pediatrics 2016; 138: e20161485.

30.

Faden

Beauchamp

King

NMP

. A history and theory of informed consent. Oxford University Press, Incorporated, 1986. Accessed June 27, 2023. http://ebookcentral.proquest.com/lib/tmclibrary/detail.action?docID=3052042

31.

Navin

Wasserman

. Capacity for preferences and pediatric assent: implications for pediatric practice. Hastings Center Report 2019; 49: 43–51.

32.

Wasserman

Navin

Vercler

. Pediatric assent and treating children over objection. Pediatrics 2019; 144: e20190382.

33.

Cherry

. Adolescents lack sufficient maturity to consent to medical research. J Law Med Ethics 2017; 45: 307–317.

34.

Austin

Lin

Selway

, et al. What parents think and feel about deep brain stimulation in paediatric secondary dystonia including cerebral palsy: a qualitative study of parental decision-making. Eur J Paediatr Neurol 2017; 21: 185–192.

35.

Focquaert

. Deep brain stimulation in children: parental authority versus shared decision-making. Neuroethics 2013; 6: 447–455.

36.

Boulicault

Goering

Klein

, et al. The role of family members in psychiatric deep brain stimulation trials: more than psychosocial support. Neuroethics 2023; 16: 14.

37.

Silvestri

Chiarotti

Baglioni

, et al. A preliminary examination of self-concept in older adolescents and young adults with Gilles de la Tourette syndrome. Eur J Paediatr Neurol 2017; 21: 468–474.

38.

Goodman

Grice

Lapidus

KAB

, et al. Obsessive-compulsive disorder. Psychiatric Clin North Am 2014; 37: 257–267.

39.

Fenske

Petersen

. Obsessive-compulsive disorder: Diagnosis and management. Am Fam Physician 2015; 92: 896–903.

40.

Garnaat

Greenberg

Sibrava

, et al. Who qualifies for deep brain stimulation for OCD? Data from a naturalistic clinical sample. J Neuropsychiatry Clin Neurosci 2014; 26: 81–86.

41.

Medtronic. Deep Brain Stimulation - Obsessive Compulsive Disorder. Accessed June 28, 2023. https://www.medtronic.com/us-en/healthcare-professionals/therapies-procedures/neurological/deep-brain-stimulation/indications/obsessive-compulsive-disorder.html

42.

Muñoz

Blumenthal-Barby

Storch

, et al. Pediatric deep brain stimulation for dystonia: current state and ethical considerations. Camb Q Healthc Ethics 2020; 29: 557–573.

43.

Torgerson

Munoz

Kostick

, et al. Clinical and psychosocial factors considered when deciding whether to offer deep brain stimulation for childhood dystonia. Neuromodulation: Technol Neural Interface. Published online January 3, 2022. doi:https://doi.org/10.1016/j.neurom.2021.10.018

44.

Aravamuthan

Waugh

Stone

. Deep brain stimulation for monogenic dystonia. Curr Opin Pediatr 2017; 29: 691–696.

45.

Lumsden

Kaminska

Gimeno

, et al. Proportion of life lived with dystonia inversely correlates with response to pallidal deep brain stimulation in both primary and secondary childhood dystonia. Dev Med Child Neurol 2013; 55: 567–574.

46.

Isaias

Alterman

Tagliati

. Outcome predictors of pallidal stimulation in patients with primary dystonia: the role of disease duration. Brain 2008; 131: 1895–1902.

47.

De Lourdes Levy

Larcher

Kurz

. Informed consent/assent in children. Statement of the ethics working group of the confederation of European specialists in paediatrics (CESP). Eur J Pediatr 2003; 162: 629–633.

48.

Diekema

. Taking children seriously: what’s so important about assent? Am J Bioeth 2003; 3: 25–26.

49.

Sen

Lee

Browd

, et al. Issues of consent and assent in pediatric neurosurgery. Childs Nerv Syst 2021; 37: 33–37.

50.

Herring

Wall

. The nature and significance of the right to bodily integrity. Camb Law J 2017; 76: 566–588.

51.

Becht

Nelemans

Branje

SJT

, et al. Identity uncertainty and commitment making across adolescence: five-year within-person associations using daily identity reports. Dev Psychol 2017; 53: 2103–2112.

52.

Erikson

. Identity, youth, and crisis. 1st ed. W. W. Norton, 1968.

53.

Van Dyck-Lippens

Santegoeds

, et al. Deep-brain stimulation for Anorexia Nervosa. World Neurosurg 2013; 80: S29.e1–S29.e10.

54.

Search of: deep brain stimulation | Recruiting, Not yet recruiting, Active, not recruiting, Completed, Unknown status Studies | Child - List Results - ClinicalTrials.gov. Accessed November 9, 2022. https://clinicaltrials.gov/ct2/results?term=deep+brain+stimulation&Search=Apply&recrs=b&recrs=a&recr%20s=f&recrs=d&recrs=e&recrs=m&age_v=&age=0&gndr=&type=&rslt=.

55.

Smajdor

. Reification and assent in research involving those who lack capacity. J Med Ethics. Published online January 23, 2023:jme-2022-108710. doi:https://doi.org/10.1136/jme-2022-108710

56.

Smith

Blumenthal-Barby

. Call for moral recognition as part of paediatric assent. J Med Ethics 2023; 49: 481–482.