What is ethically required to adapt to intellectual disability in cancer care? A case study of testicular cancer management

ABSTRACT

Background

People with intellectual disability who require cancer services face several barriers and inequities, including delayed or unrecognised cancer diagnosis and inadequate attention paid to their specific concerns, needs and preferences about care.

Methods

In this case report, we present a case report of a 33-year-old male with severe intellectual disability requiring treatment for metastatic testicular cancer. The patient’s intellectual disability required major adjustments to the delivery of chemotherapy. We also describe the involvement of a clinical ethics committee to analyse the ethical pros and cons of different treatment options for this patient, and to facilitate discussions about collaboration and communication required, between different clinical teams, and with the patient’s family and carers.

Results and conclusions

We suggest this level of clinical and ethical deliberation sets the bar for what is required for healthcare services to be inclusive of and accessible for all people, especially those with intellectual disability.

KEYWORDS:

• Cancer care
• intellectual disability
• clinical ethics

People with disabilities experience poorer health outcomes, increased barriers to accessing healthcare, and reduced utilisation of preventive services, compared to the general population (Edwards et al., 2020; Willis et al., 2018). As a subgroup, cancer patients with intellectual disability, often have a delayed or even unrecognised cancer diagnosis (Cuypers et al., 2020; Heslop et al., 2014; Willis et al., 2018), and are less likely to have their concerns and preferences properly addressed (Havercamp & Scott, 2015). People with intellectual disability represent heterogeneous groups who have deficits in intellectual functioning limiting social, practical or conceptual domains of daily living (Boonman et al., 2022). Symptom assessment is often hampered by communication difficulties (Willis et al., 2018), which may result in poor pain and symptom management (Friedman et al., 2012). Physical barriers associated with accessing cancer services include location (Cuypers et al., 2020), time pressures or a lack of easy-read patient information. Patients with intellectual disability may underuse palliative care (Cross et al., 2012; Friedman et al., 2012) and cancer screening services (Iezzoni, 2022).

These barriers and inequities represent direct and indirect discrimination, both of which lead to compromised access to healthcare systems and poor cancer-related health outcomes, inferior survival rates and less intensive treatment compared to patients who do not have disabilities (Boonman et al., 2022; Cuypers et al., 2020).

They also raise important ethical considerations and questions. For example, from a justice perspective; how should available health resources be distributed to ensure cancer patients with disabilities can access and obtain the treatment they specifically require? From the principle of respect for autonomy; what is required to ensure that cancer patients with a disability are able to express their preferences about proposed treatments, and that an appropriate balance is achieved, between promoting a person’s autonomy and protecting them when their autonomy is diminished (Peterson et al., 2020)? From a risk/benefit analysis; how can health practitioners ensure the functional and cognitive effects of a patient’s disability are considered and balanced when identifying likely burdens, risks and benefits of cancer treatment.

To expand on these ethical considerations, we present the case of David (pseudonym), a 33-year male with severe intellectual disability, requiring treatment for metastatic testicular cancer. David has limited verbal capacity and a functional mental age of 2–3 years old. David’s disability restricted the available treatment options. We document how these barriers were addressed, and the ethical deliberation which underpinned treatment decisions for David.

Case study

David has lived for the last 8 years in a community house supported by a full-time carer shared amongst the residents of the facility. Although at times, ambulant, David is predominantly wheelchair bound, largely non-verbal and does not have the easy ability to express pain. He can do basic signs and gestures. David is unable to cooperate with simple medical interventions such as blood pressure monitoring, or blood test without general anaesthetic as these interventions had led to high levels of distress and agitation.

David’s carer noticed a testicular mass and referred him to an external hospital for investigation and management. He was admitted to hospital for removal of the testicle, blood work and scans whilst under general anaesthetic. Based on this a diagnosis of metastatic testicular cancer was made with evidence of spread to his lymph nodes. From an oncologic perspective, his diagnosis is associated with approximately a 95% chance of cure using standard treatment algorithms (Beyer et al. 2021), which involves outpatient chemotherapy requiring intravenous access for approximately 5 h (Monday through Friday every 3 weeks).

Both the external clinicians and David’s mother agreed that David would become highly agitated and he would not comply with remaining still during this treatment. Concerns were also expressed that supporting David in both outpatient or inpatient settings may require restraint and would likely compromise staff safety. Although David was not usually aggressive, he had the potential for becoming overwhelmed. He had previously required behavioural interventions and management for prior medical interventions. The clinical advice conveyed to David’s surrogate decision maker (his mother) was that it would not be possible to proceed with curative standard of care treatment. Instead, comfort care within a palliative approach was deemed to be the clinically and ethically appropriate option.

David’s mother and the clinical team then sought a second opinion as they were not comfortable for supportive care alone given his imminently curable malignancy. They requested a more active management route because, despite his disabilities, David had a good quality of life, enjoying his time at the local community centre and at the beach.

Second opinion and clinical ethics involvement

In addition to a second medical opinion, the second hospital’s Clinical Ethics Advisory Group was consulted and asked to respond to two questions:

1. Is it ethically reasonable to take away the option for curative standard-of-care treatment for this patient?

2. Is it ethically reasonable to offer comfort care only?

The clinical ethics group discussed the risks and burdens of delivering standard-of-care treatment for testicular cancer, including treatment burdens likely to be experienced by David and those caring for him. Based on David’s mother’s knowledge of David’s capacity to understand, express pain, anxiety and enjoyment, the clinical ethics discussion concluded, as had the first oncology opinion, that David would not understand or be able to tolerate repeated hospital visits requiring him to sit still in a chair, undergo blood tests and intravenous lines, and vital sign monitoring. Despite a high chance of cure, the burdens of standard treatment for David, the resources required to restrain him and the safety risks to staff even if restraint was considered ethically reasonable, outweighed the benefits. The ethics committee concluded, in response to the first question, that it was therefore ethically reasonable to remove the option of standard-of-care chemotherapy.

In response to the second ethics question, the group considered alternative treatments which may slow the disease process or offer a chance of cure but with less burdens than the standard treatment. In the context of a highly curable cancer and David’s mother’s request, on behalf of her son, to pursue active treatment, it was considered to be ethically inappropriate to restrict treatment to comfort care only.

From an oncologic perspective, four possible treatment options were identified. Ethical evaluation of each of the options involved referring to the foundational ethical values (beneficience, non-malificence, autonomy and justice) (Beauchamp & Childress, 2019) to consider and weigh up the benefits and burdens of each treatment option on David and his carers, and the resources required from health staff.

Option 1 was standard outpatient chemotherapy with a high chance of cure. As highlighted above, David’s family and clinicians agreed that David would not tolerate this treatment.

Option 2 was chemotherapy administration utilising chemotherapy infused over 2 h every 3 weeks for 3 to 4 cycles with international data suggesting over a 90% chance of care however it would come with the risk of substantial side effects including the need for readmission if those toxicities occurred and the potential requirement for blood and platelet transfusions (Alifrangis et al., 2022). Potential benefits of this approach include single infusion treatment that could be administered as part of a general anaesthetic.

Option 3 was daily radiotherapy for 1 month requiring general anaesthetics each day with an approximate success rate of 50%. Although no randomised data, benefits of radiotherapy are considered to be lower compared to chemotherapy, and historically not used in this stage of testicular cancer given failure rates, with the additional burden of daily general anaesthetic requirements.

Option 4 was an operation to remove the abdominal lymph nodes and historically not used given a recurrence rate similar to radiotherapy. In addition, an operation would have posed significant challenges for David, in the postoperative management including higher risk of infection, wound disruption, complications associated with poor mobility and potential difficulties if anticoagulant therapy injections were required.

The clinical ethics group achieved broad consensus about the relative burdens and benefits of each option. Option 2 was considered the best one for David compared to each of the three other alternatives. It was considered to offer the most beneficial clinical outcome whilst respecting and supporting what was known about David’s preferences and the fact that he would not cope with treatment requiring visits as an outpatient. The benefits of general anaesthetic-based chemotherapy outweighed the risks and burdens of anaesthesia for David. Option 2 also aligned with David’s mother’s request for the best available active treatment given that standard of care was not an option. The ethics committee also noted that for option 2 to work a clear plan was required to map how the treatment would be delivered, the possible effects on the patient and setbacks which may occur and processes for reviewing treatment goals. This included:

1. A detailed discussion about the proposed treatment including its risks, benefits, alternatives and evidence base as part of the informed consent process.

2. Discussions with the family and carers before treatment commences so that they are ready for possible outcomes, time points for monitoring treatment effects.

3. Consultation with David’s carer to ensure a thorough discussion of the best way to monitor David’s distress.

4. Strategies for monitoring and evaluating effects of the treatment after each cycle to ensure David is tolerating the treatment, and that family and carers are aware of signs of deterioration or side effects.

5. The role of post-chemotherapy ward based admission if required.

Treatment implementation

An expansive array of departments and services were brought together to ensure the delivery of care was safe and coordinated. This included Genitourinary Clinical Nursing Consultant support and coordination, cooperation from the National Disability Insurance Scheme (NDIS) provider, hospital access coordinators for seamless hospital attendances and admissions, theatre and anaesthetics for developmentally appropriate care provision, Day Therapy Unit managers and staff for chemotherapy preparation and treatment delivery, Hospital-In-The-Home (HITH) for ambulatory toxicity monitoring, and allied health particularly Occupational Therapy, for minimising his falls risk.

One of the early challenges was blood count monitoring. Finger prick blood draws were not tolerated by David. Instead, with consent, oral sedatives prior to any blood draw were used. If blood tests were required prior to a scheduled general anaesthetic, the tests were done after David had been put to sleep.

During his treatment, David’s carer noticed tiredness and easy bruising requiring a quick response from theatre to urgently accommodate blood and platelet transfusions whilst David was under anaesthetic. David’s falls risk was highlighted as a concern by his mother and allied health was involved in assisting with non-verbal communication tools, mobility aids, bed management and recommendations for toileting and feeding.

Clinical outcome

Despite the initial concerns around developing a non-standard treatment pathway, David safely received chemotherapy whilst under anaesthetic without significant complications and is currently disease free and back to his normal self and usual activities.

Discussion

David’s case illuminates a number of ethically important questions that were considered as part of the planning process and delivery of his cancer treatment. How to balance and prioritise the beneficial and potentially curative cancer treatments against the unique burdens experienced by a patient who was not able to understand, who was likely to be fearful or who might physically resist or be unable to express their experiences or preferences? How to adapt standard treatment to ensure the most effective but less burdensome method? How to practically adjust treatment pathways and to consider which adjustments were ethically justifiable in terms of use of resources both within and outside of the hospital? What level of restraint, if any, was ethically justified and how to balance staff safety with patient needs?

The initial decision in David’s case to move to comfort care reflects the evidence that people with disabilities experience differential treatment and higher cancer mortality than people without disabilities (Iezzoni, 2022). Seeking a second opinion and clinical ethics consultation opened up a space to consider how to tailor cancer treatment to meet David’s special needs. Clinical ethics services aim to assist clinicians and families to process and thoroughly analyse ethical challenges by applying moral reasoning, taking into account specific clinical circumstances, health ethics principles and bioethics concepts to identify and balance relevant ethical values (Crico et al., 2021; Haltaufderheide et al., 2020). The health ethics principle of respect for autonomy and the role of surrogate and supported decision-making was particularly important to ethical the deliberations. In this case, the contribution of a clinical ethics committee was integral to support the deliberation required to consider and weigh up the ethical pros and cons of available options and to ensure that decisions about what was in David’s interests also represented a supported decision based on what was known about David’s will and preferences (Cantor, 2005).

From a justice perspective, David’s case highlights that administrators and clinicians must be prepared to adapt standard treatment pathways with strong clinical evidence when using health resources to meet an individual patient’s needs (Van Spall et al., 2007). Such an approach accords with social models of disability (Davy, 2019; Ho, 2008), and accords with article 12 of the Convention on the Rights of People with Disability (CRPD), which emphasises the right of persons with disabilities to have access to the support they require (United Nations, 2006).

From the perspective of considering and weighing up benefits and burdens of treatment, David had little ability to express his needs and interests, and this has been the case since his birth. This meant clinicians had to spend extra time in learning about David via his family and carers.

David’s case highlights the value of thinking about a patient’s autonomy in a relational way (Mackenzie, 2014), where people are recognised in a condition of interdependence with others, rather than as fully autonomous and independent. Standard conceptions of what is meant to respect a patient’s autonomy presume individual capacity to understand and think about treatment options (Ho, 2008). For patients with intellectual disability, this conception of autonomy is too thin (Childress & Quante, 2022). David is profoundly disabled and has never had capacity for autonomous decisions. However, he is able to express preferences about what he enjoys and what he has trouble coping with. Facilitated ethical analysis aimed to determine what David finds meaningful in his life (Cantor, 2005), and to enable a proportional response (Peterson et al., 2020) to the protection that David required in the form of treatment for his cancer and respect for his autonomous preferences. This required asking detailed questions of David’s mother as his surrogate decisionmaker, to evaluate how David’s interests and preferences were being respected (Devi, 2013).

Conclusion

In their review of barriers to, and experiences of cancer services for adults with a physical disability, Edwards et al. (2020, p. 1) state that “cancer services, like all healthcare services, need to be inclusive of and accessible by everybody, including people with disabilities.” David’s case shows that to achieve this ethical ideal, it may not be enough to follow evidence-based standards (derived from studies which exclude participants who have disabilities) (Van Spall et al., 2007). David’s intellectual disability required major adjustments to the delivery of chemotherapy, engagement with an ethics committee to ensure in-depth examination of ethical pros and cons of treatment options, and collaboration and communication between clinical teams, family and carers (Nieuwenhuijse et al., 2019). This level of clinical and ethical deliberation sets the bar for what is required for healthcare services to be “inclusive of and accessible by everybody” (Edwards et al., 2020, p. 1).

Disclosure statement

No potential conflict of interest was reported by the author(s).