“Straddling two worlds”: complexities of spousal caregiving in amyotrophic lateral sclerosis during younger middle age

Abstract

Background

Family support is central to amyotrophic lateral sclerosis (ALS) care. Spouses often assume the role of primary caregiver, facing daily challenges as their partner’s needs progressively increase. Younger ALS caregivers appear to have distinct experiences and needs, yet age- or life-stage variations and processes of caregiving are seldom considered in studies of ALS family caregiver experience.

Methods

We undertook a qualitative study, guided by constructivist grounded theory methodology, to explore the dynamics of family caregiving following an ALS diagnosis in a younger middle-aged family member (≤55 years). Data were coded to identify psychosocial processes, including how family caregivers engage in caregiving.

Results

In-depth interviews were conducted with ten spousal caregivers between August 2023 and August 2025. Overall, our theoretical understanding of spousal caregiving for younger middle-aged adults with ALS (YMAs) was captured by the core category ‘straddling two worlds,’ reflecting how caregivers navigated multiple interconnected dichotomies: present and future, familiar and new norms, current and anticipated losses, and the worlds of the living and the dying. To contend with an uncertain future, caregivers anchored themselves in the present as they navigated a shifting sense of normalcy. Middle age expectations and social norms shaped how caregivers engaged in caregiving and experienced losses that were often unending and ambiguous.

Conclusions

Caregiving for a spouse with ALS in younger middle age involves temporal, practical, emotional, and existential processes. Caregivers of YMAs may benefit from interventions that help them tolerate uncertainty, stay grounded in the present, maintain normalcy, and grieve losses throughout the caregiving trajectory.

Supplementary Information

The online version contains supplementary material available at https://doi.org/10.1186/s12904-026-02230-w.

Background

Amyotrophic lateral sclerosis (ALS) is a debilitating disease resulting in progressive muscle weakness, bulbar and respiratory impairment, and cognitive and behavioural decline [1–3]. Onset is typically between 50 and 75 years of age [1], with approximately 10% of cases diagnosed in individuals under 50 [4, 5]. Although death from respiratory failure typically occurs within three to five years after onset, survival and disease progression are highly heterogeneous [1, 2]. For example, younger age at onset, among other factors, has generally been associated with longer survival and slower disease progression [4, 6]. People living with ALS (plwALS) develop complex biopsychosocial needs due to physical and psychological challenges including loss of independence and ability to communicate [7]. With no cure and limited treatment options that marginally slow disease progression, management is focused on symptomatic and supportive care [1].

Family support is central to ALS care [8–15], as formal services often address only a fraction of overall care needs [14]. Coverage for homecare services, paid support workers, and adaptive equipment varies considerably across countries and impacts access to formal home-based support for plwALS and their caregivers [16]. Even in healthcare systems with publicly funded care, inadequate subsidization; prolonged wait-times; complex approval processes; insufficient, unreliable, or inflexible allocated care hours with poor oversight by external agencies; inadequate staff training and expertise; high staff turnover; caregiver fear and mistrust of outside help; and caregiver guilt or shame about accepting help and leaving their partner in the care of others can reduce access to or utilization of formal support services, shifting care responsibilities and associated costs onto family caregivers [15–18].

With disease progression, plwALS increasingly rely on family caregivers for self-care, emotional support, and planning for future needs [15, 16, 19–21]. Caregivers often face daily challenges related to their family member’s decline [22], alongside social and emotional consequences of terminal illness, including loss of opportunities, relationship security, sense of identity, and expectations of life [23]. The progressive nature of ALS places new and evolving demands on the family unit, altering dynamics, roles, and functioning [8–10, 23]. Spouses often become the primary caregiver [10] and, when children are present, assume the other parent’s responsibilities while guiding their children through the decline and eventual loss of a parent [8, 18, 24]. As plwALS become more dependent on their spouse, the ways in which they relate to, interact with, and support each other often change as their relationship becomes less intimate [8, 21–23, 25]. The stress and demands of spousal caregiving can lead to the loss of a previously reciprocal relationship for some couples [23], though aspects of reciprocity sometimes persist as plwALS support their partner in decision-making and adaptation [14, 19, 26].

Life stage, such as middle age [27–29], and one’s sense of ‘place’ within their lifespan (contextual age) [30, 31] influence how illness is experienced [30, 32]. Middle age has boundaries that vary depending on social and cultural expectations; however, typically begins around 30 to 40 and ends at 60 or 65 years of age [29]. Role transitions outside expected age-related norms, like a spouse becoming a caregiver for their partner during middle age when independence is emphasized, are widely perceived as non-normative [27, 32]. Illness during middle age prompts couples to reevaluate future plans and their anticipated life trajectory [32]. Moreover, middle-aged spousal caregivers often face competing demands, including raising children, supporting aging parents, and managing financial and work responsibilities [33].

Younger ALS caregiver age has been associated with poorer well-being [34], greater depressive symptoms and caregiver burden [35], and less stable support networks [36], suggesting that age is an important factor shaping caregiver experiences and needs in ALS. Recent systematic reviews have highlighted the need for studies that differentiate personal characteristics of informal ALS caregivers [37], including age-related variations in experience, particularly in younger middle age [38]. Yet age- or life stage-specific nuances have rarely been accounted for in studies of ALS caregiver experience. Furthermore, the psychosocial processes of family caregiving in ALS, that is how caregivers’ actions and interactions unfold and interconnect over time to shape their experiences [39, 40], remain underexplored [21]. Understanding these processes in younger middle age could elucidate why certain patterns arise [40] and guide age-appropriate supports and interventions. Accordingly, the aim of this study was to explore the psychosocial processes underlying family caregivers’ experiences following an ALS diagnosis in a family member at 55 years or younger (younger middle-aged adult with ALS, subsequently referred to as YMA).

Methods

This study was guided by constructivist grounded theory methodology, which emphasizes participants’ lived experiences [39, 41, 42]. We adopted a constructivist worldview, recognizing that individuals construct and interpret their own experiences, resulting in multiple possible realities, and that knowledge is socially co-constructed by researchers and participants [43–45].

Setting and recruitment

Family caregivers were recruited from the Sunnybrook Health Sciences Centre ALS Clinic in Toronto, Ontario, which is the largest ALS clinic in Canada providing care for about 700 plwALS, with approximately twenty-five new diagnoses made monthly. Participants for this study were recruited as part of a dyad with a YMA, who was enrolled in a separate study, although participation of the other dyad member was optional. To be eligible, individuals had to be identified by a YMA as a family member central to their care and be able to communicate in English and provide informed consent. The first author (ASEP), a neuromuscular fellow and PhD graduate student at the time, provided study details to all eligible participants and informed consent from those who chose to participate was obtained.

Sample

The first two participants were purposively sampled as they were anticipated to be information-rich cases [46]. After analyzing their interviews, we shifted to theoretical sampling, refining our sampling strategy based on our emerging ideas [39, 47]. During coding of the first two interviews, we identified a tension between living in the moment and planning for the future, which was explored with all subsequent participants. When we identified that parenting children was central to a tentative category ‘maintaining normalcy,’ we sought caregivers with children living at home. We also theorized that time since the YMAs’ symptom onset and level of physical dependence shaped how caregivers adapted. To explore this, we sampled caregivers of YMAs living with ALS for four or more years from symptom onset and with varying levels of functional impairment based on the ALS Milano-Torino (MiToS) Staging system [48].

Data collection

Basic demographic information (participant age, sex, gender, relationship to the care recipient, employment status, and presence of dependents) as well as information on each care recipient (age at ALS diagnosis, time since symptom onset and diagnosis, functional impairment used to calculate the corresponding MiToS stage [48]), was collected prior to each interview using a questionnaire developed by the research team. Sex was categorized based on biological attributes (male, female, other) [49] and gender was defined as an individual’s self-identified personal and social identity (man, woman, gender-diverse person) [50].

The interview guide (Additional File 1) was developed by the first author in collaboration with the research team and focused on processes of caring for a family member with ALS. In line with a constructivist approach, we drew on our prior knowledge and relevant literature about family caregiving and ALS [8–10, 15, 37]. Arthur Kleinman’s work [51, 52], which conceptualizes caregiving as a learned and embodied practice that unfolds through relational and reciprocal processes, also served as a sensitizing concept [39]. The preliminary interview guide was piloted with a family caregiver of a YMA and their feedback was incorporated into the final guide. The first author conducted all interviews in English by videoconference and wrote field notes after each to capture her immediate thoughts. Recorded interviews were transcribed by the first author with the assistance of Zoom for Healthcare’s automated transcription [53].

Data analysis

Each transcript was deidentified and imported into NVivo14 [54], which was used to manage codes and memos and facilitate making linkages amongst them. Our analysis was primarily inductive, focused on identifying patterns in participants’ accounts, and reflected our interpretation [39, 55]. We used constant comparison [56] and memo-writing throughout the analysis to explore codes and then core concepts and their connections [39, 55]. The analysis was conducted by a single coder (ASEP) in alignment with constructivist frameworks [57], with ongoing input from the research team who provided critical feedback and interrogated the developing analysis.

The first phase involved line by line coding, with attention to processes or actions [39, 42]. During the second phase, initial codes that captured larger segments of data or had greater conceptual significance were sorted, synthesized, and elevated to focused codes [39], clarifying key processes and the relationships between them [55]. During the comparison of codes within and across interviews, the first author recorded ideas about possible categories, their connections, and related questions in memos to track and refine her ideas [55]. As the second phase progressed, focused codes were organized into provisional substantive categories by asking what larger analytic story they suggested, and a preliminary analytic framework was constructed [39]. All transcripts were re-examined to expand underdeveloped categories and eliminate categories that were redundant or not integral to the evolving analytic framework [39, 55]. The first author used conceptual mapping (diagramming) to visually examine key relationships and to clarify connections between categories, subcategories, and codes [39] (Supplementary Figs. 1 and 2; Additional File 2). These maps were reviewed by the research team, and their input was integrated into the final figure outlining the process of spousal caregiving in ALS during younger middle age (Fig. 1). The final level of coding involved selecting a central category, which was collaboratively developed with the senior author (JEMS) incorporating input from the co-authors, and systematically relating it to the other categories [58]. We determined that theoretical sufficiency had been reached after ten interviews, when our categories accounted for new data without requiring continual extensions or modifications [59].

Fig. 1.

Process for spousal caregiving in ALS during younger middle age

Trustworthiness and rigour

Our work was guided by four main quality criteria in constructivist grounded theory: credibility, resonance, originality, and usefulness [60]. Credibility was strengthened by collecting relevant data and interrogating it throughout collection and analysis. Reflexive examination of our positions and priorities further reinforced credibility [61]. The first author considered how her clinical background, early-adulthood experiences, and assumptions about caregiving could influence data collection and analysis, documenting insights in reflexive memos. The iterative analysis ensured codes fit tightly with the data and categories captured participants’ experience, enhancing the resonance of our findings [60]. By exploring the social processes of an under-studied group, this study contributes original insights that can inform future research and clinical practice ensuring its usefulness.

Ethical aspects

Approval from the Sunnybrook Health Sciences Centre (#5599) and University of Toronto (#43666) research ethics boards was obtained prior to commencement of the study. The first author was unknown to all but one participant, who she had encountered once during a clinic appointment three years prior to the current study, and in this instance a research assistant obtained consent to ensure consent was given voluntarily.

Results

The sample included ten caregivers of YMAs (four men and six women, aged 30–61 years), recruited between August 2023 and August 2025 (Table 1). An additional eleven caregivers consented to participate; however, ten did not respond when contacted to schedule an interview, and one did not attend a scheduled interview, with subsequent efforts to reschedule being unsuccessful. All participants lived with, and were in a committed relationship with, a YMA whose age at ALS diagnosis ranged from 30 to 55 years. Most participants were caring for a partner who had been diagnosed with ALS within the preceding three years and six partners had functional loss in two or more domains according to the ALS MiToS Staging system [48]. One partner had been experiencing symptoms for less than one year and had minimal functional impairment, while three partners had been living with symptoms for four or more years. One participant was caring for a partner with familial ALS and none reported cognitive impairment in their partner. All but one participant was employed full time and seven reported dependents, including three with children under eighteen. Five of the seven participants with dependents reported that at least one dependent was living at home with them and their partner with ALS at the time of the interview, while one non-dependent child had returned home to assist with their parent’s care. Interviews averaged 80 min (range 53–100).

Table 1.

Description of study sample (n = 10)

Characteristic	n/10
Gender
Man	4
Woman	6
Relationship type
Married	8
Engaged	1
Common-law	1
Employment status
Full-time	9
Unemployed	1
Dependents
None	3
≤18 years	3
> 18 years	4
Dependents living at home (n/7)
Yes	5
No	2
Time since partner’s symptom onset
< 1 year	1
≥1 to < 2 years	1
≥2 to < 3 years	3
≥3 to < 4 years	2
≥4 years	3
Time since partner’s ALS diagnosis
< 1 year	2
≥1 to < 2 years	3
≥2 to < 3 years	2
≥3 to < 4 years	3
Functional impairment of partner at time of interview(ALS MiToS Staging system*)
Stage 0 (functional involvement)	1
Stage 1 (loss of independence in 1 domain)	3
Stage 2 (loss of independence in 2 domains)	5
Stage 3 (loss of independence in 3 domains)	0
Stage 4 (loss of independence in 4 domains)	1

^*Domains: swallowing, walking or self-care, communicating, breathing

MiToS Milano-Torino

Through our analysis we developed a theoretical understanding of spousal caregiving for YMAs captured by the core process ‘straddling two worlds,’ which encompasses distinct yet interconnected categories (Fig. 1). Our framework positions caregivers as straddling a series of overlapping dichotomies: present and future; familiar and new norms; current and anticipated losses; and the worlds of the living and the dying. Statements from participants are provided to illustrate the categories we developed across the dataset.

Present and future

Participants grappled with making sense of and planning for a new, uncertain, and unpredictable future, processes made particularly difficult by the unpredictability and terminality of ALS. The future, and all it might entail, was often difficult for caregivers to contemplate:

“Thinking about the future is for sure like terrifying. You’re going into the unknown because on one end you can think of sort of the worst thing.” (CG12)

Early on, caregivers struggled to envision a future that no longer aligned with their present:

“When we were first diagnosed… people would come and talk to us… but it wasn’t really resonating with us exactly. …It’s like telling a child about how they’re gonna retire.” (CG17)

The uncertainty associated with an ALS diagnosis, including the unpredictability about its course or the effectiveness of treatments, was difficult for caregivers to bear:

“It may be one of the hardest things about this disease… you can never have everything organized because it is so unpredictable.” (CG13)

Caregivers compared ALS to cancer highlighting how uncertainty about the future was intensified by the absence of clearly defined disease stages or the possibility of a cure:

“Like with cancer… they know, if we don’t do this, this is gonna happen. But this disease, you have no clue, right?” (CG01)

Many caregivers described ambivalence when considering the future and this generated inner conflict for some. One participant illustrated this tension, describing being simultaneously compelled to think about the future and resistant to do so:

“At my age you start to think about, like the future, obviously, with how your kids are gonna grow up, retirement, and… there’s, like, an internal conflict. …In a normal circumstance the future would be something to look forward to. …To a certain extent, what is there to look forward to, right? What we’re looking forward to is a life that’s incomplete.” (CG17)

Uncertainty about a future with ALS created a disconnect between caregivers and healthcare providers, who sometimes conveyed urgency about planning ahead:

“And so there’s a sense among some healthcare providers of urgency, because we just don’t know, to make some big decisions. …And I think that’s where I felt a bit overwhelmed.” (CG19)

Uncertainty about the future also led to divergent perspectives on hope:

“Like we do try to be optimistic, but it’s like the wind’s just knocked out of your sails. …Don’t blow up with so much optimism and hope.” (CG21)

As caregivers tried to make sense of a life with a spouse with ALS, they struggled to prepare for the future. One caregiver explained:

“I kind of refuse to go too far. That’s too much. It’s like you’re closing your windows completely. It’s all black and I don’t want that. I know that I can get things probably set up but I don’t want to. …I don’t want to do things that are not relevant.” (CG23)

Some caregivers struggled to plan for the future out of fear that doing so might hasten the outcomes they feared:

“We always talked about, like, doing a will, planning for a funeral… so that when things do progress, that’s not something that we’re scrambling to do. We haven’t yet done that. And I think part of that is, again, this rational irrational fear of when you talk about it, because you’re preparing for it, it’s gonna happen sooner rather than later.” (CG17)

Caregivers also described how discussing the future with their partner was especially challenging, often avoiding such conversations to prevent further distress:

“We don’t really have conversations about the future… like that’s what he’s emotional over.” (CG03)

Anticipated expenses and potential loss of income further complicated future planning as participants struggled to balance preparing for their spouse’s care with maintaining financial security:

“I can’t stop working. We’re going to need an income. …And we’re going to need help. So how do you make both things happen?” (CG19)

Financial concerns were also intertwined with the broader challenge of planning for a transient and constantly evolving future. One caregiver explained:

“So, it’s a dance in terms of cost and how long he [spouse with ALS] may use a device. The amount of people when we bought our wheelchair van who made a comment about the resale value… that is insinuating that one day soon, we won’t require it. And even undercoating the van… [spouse with ALS] said, ‘let’s not bother because who knows how many winters I will be here.’ …So the logistics are a horrid thing and live in a sort of grey space of this is our life, but perhaps temporary.” (CG13)

To contend with an unpredictable and temporary future, caregivers anchored themselves in the present:

“We went to living basically in the moment. …We don’t plan too far in advance at all. Your mind completely shifted. And so it’s really about day-to-day living.” (CG19)

By anchoring themselves in the present, caregivers avoided catastrophizing about the future or ruminating about the past:

“So it’s like trying to stay focused on the present moment… don’t worry about tomorrow or think about yesterday, because if I do that, I’m ruining today. I’m ruining my chance of being present.” (CG21)

For some caregivers, living in the present was challenging because it required acknowledging their current reality, yet it also served as an act of self-preservation. One caregiver described striving to stay present, as dwelling on the past (similar to dwelling on the future) was emotionally distressing:

“I kinda try not to think of the past ‘cause I think it’s hard to think of comparing how she was before and how she is now. And I guess that’s another challenge of me trying to be as present as I can and sort of accept the way things are. …And not wish about the past because I know that only leads to more pain and depression.” (CG12)

Attending to the present also enabled caregivers to continue functioning in their everyday life:

“If I stayed like that [contemplating the future] all the time, I would never be able to work. How would I get anything done? So breathe and do my centering and just be here in the present moment.” (CG01)

The present often demanded caregivers’ full attention, leaving little room for future planning, which they usually limited to the immediate term:

“When I plan ahead, it’s for the near future. I’m planning ahead so there’s no surprises tomorrow… I have a hundred other things on my plate so I’m not going to try to plan for how do you work a feeding tube?” (CG10)

Caregivers described how living in the moment helped them manage constant change and effectively care for their spouse:

“I just try to support what he can’t do… and then allow him to do what he can do which is always changing. …Another reason why it’s really much easier to just live day-to-day ‘cause you’re just figuring it out day-to-day. It’s constantly changing.” (CG19)

However, caring for their spouse in the present was complex since planning ahead in certain instances shaped how caregivers were able to provide support in the present. They planned ahead when it provided something to look forward to or eased anxiety about the future, helping them stay grounded in the present by providing purpose or reducing uncertainty:

“And that’s why I’m really kind of hoping that we can get up to see my daughter… I think that’s what you need. You need the memories. You need something to look forward to.” (CG10)

“The doctors are in place now. So for me now, we can just take things day by day.” (CG02)

Nevertheless, caregivers also pointed out that the availability of resources did not equate with readiness to access them:

“Everything has been set up. I know we have the resources… so when we need them, we will be using them. …So when you need things, you can accept them.” (CG23)

Familiar and new norms

While caregivers anchored themselves in the present as a means to continue with daily life and support their spouse effectively, over time, caregivers continuously adapted to an evolving normal. Repeated adaptation led to ‘living a new normal,’ as they navigated the tension between their former familiar life and a new present defined by their partner’s growing dependence.

Maintaining a sense of normalcy was a key strategy used by caregivers to ground themselves in the present. When their spouse had mild functional impairment, caregivers adapted their routines and activities to preserve a sense of normalcy:

“It’s just I think being mindful of how to do things the same in a different way. …It’s just keeping your life the same… so that you don’t feel like this disease is just taking over your life.” (CG01)

For those who were also parents, their children were motivation to maintain familiar family routines:

“It was also trying to keep our lives as normal as possible for our kids.” (CG17)

Maintaining normalcy gradually transformed into redefining it, as caregivers adjusted to their spouse’s evolving needs and declining independence. Caregivers also wrestled with preserving familiar routines and making the most of limited time, sometimes prioritizing meaningful activities while their partner could still participate.

“It is an ever-shifting ground of normalcy. What was normal two months ago is no longer the case. …And that’s the messed up part of this. You want to make the most of those good times, but it’s early days and you feel like you need to keep up with our normal life as long as possible.” (CG13)

As their spouse’s needs and abilities changed, caregivers continuously adjusted to an evolving normal. Caregivers adapted their living environment, social activities, and how they communicated with their spouse or expressed intimacy:

“So intimacy, as in love, not necessarily sex, like massaging her feet, caring for her. Going there with her with those conversations as a partner. That is love and intimacy.” (CG21)

The pace of progression largely determined how quickly caregivers had to adapt. Yet even when their partner’s decline was gradual, each functional loss felt sudden:

“And then it’s strange because it feels slow but then when there’s a change, it almost feels like it’s fast.” (CG12)

Caregivers learned how to adapt to a new normal through trial and error or from others with lived experience. Early in the disease course, they supported their spouse by promoting their independence and offering emotional support:

“Caring for him physically, I don’t, he can do everything. There’s nothing that he needs me for right now. Emotionally, I would just say a support system.” (CG03)

Caregivers occasionally struggled to provide emotional support, particularly when their spouse was unreceptive to it:

“When he did fall a few times, he would start crying… and he just shuts you out. He doesn’t wanna talk about it… he doesn’t want you to be that type of support.” (CG02)

As their partner’s ALS progressed, caregivers provided physical care as well:

“Give her a shower and brushing her teeth, all that stuff that she can’t do herself anymore. I try to help her with all the physical health things.” (CG10)

Throughout the adaptation process, caregivers assumed new responsibilities and renegotiated existing age-related roles, such as parent, employee, and spouse:

“I am his spouse and caregiver but moving more and more into caregiver. …I feel that I don’t have a partner often. …And there are more times when I am parenting the kids… his input as a parent is changing… so the sense of him being a partner and a spouse is declining. …I’m not working. And I’m okay with that. But, if I had been more career minded, that would have been a very difficult thing to give up to become a caregiver.” (CG13)

Current and anticipated losses

Navigating a constantly shifting ‘normal’ required caregivers to adjust to lost routines, roles, and the future they had imagined. They grieved losses across time while witnessing the parallel losses of their partners and children. They grieved the person their spouse had been in the past:

“There’s a part of me I guess that misses her the way she was before.” (CG12)

They reflected on the loss of their partner in the present, with current losses intensified by the anticipation of those yet to come:

“I am losing my partner. We can’t go for a run anymore, we can’t do the things we like to do. …When you hear a terminal diagnosis… the grieving process might begin… because you’re now anticipating I’m gonna lose this person sooner than normal. …Like watching the person change as a person, watching them deteriorate or fade away, watching them get weaker… losing the person they once were.” (CG21)

Caregivers also described how the loss of the future they had envisioned with their spouse was particularly distressing:

“We had envisioned the next phase of our life. …And it was like all of that was gone. …All that dream, your future, what you had been thinking about completely wiped out and you were living this other life that you didn’t really want. …What you envision in your mind just wasn’t going to be what was going to happen.” (CG19)

Caregivers grieved their own losses while witnessing their spouse’s recurring losses and cycles of grief. For parents, this was especially complex, as they observed both their spouse’s diminishing capacity to engage with their children and their children’s corresponding loss:

“I think it’s sad for him [son]… seeing his dad cannot do those things around the house… maybe it’s sad for me to see that. …It’s like he doesn’t have that father figure.” (CG23)

Worlds of the living and the dying

Caregivers ultimately came to live in two overlapping realities: one in which their spouse was dying, and another in which they and their children (when present) continued to live. Caregivers often compared themselves to their friends, acutely aware of how drastically their lives had changed. One caregiver with school-aged children reflected:

“We talk about whether or not he wants the do not resuscitate form. And then [daughter] comes home and says there’s a boy that she likes and [son] is super excited about the Christmas concert they’re putting on. And [spouse with ALS] choked on something and I had to give him the Heimlich maneuver. So you’re in this sort of grey area where you’re with the living and the dying all at once. …[Spouse with ALS]’s buddies coming over and they’re talking about their trips and their work… and sometimes you realize how far away you are from everybody else. …Straddling two different worlds because that world, during those times, it feels very far away.” (CG13)

Participating in life while caring for a dying spouse required caregivers to simultaneously hold their own experience as the healthy partner while remaining attuned to their spouse’s experience of ongoing loss and deterioration:

“He’ll ask me, ‘how was your run today?’ …If I had a really good run or I felt great… I’m not gonna necessarily overly share because I know he couldn’t participate in it.” (CG02)

Caregivers also held their children’s experiences as they engaged with the world of the living, while guiding and preparing them for the reality of a parent’s terminal illness:

“Like they [children] can go for months without seeing him because they’re both away at school [university]. And so I’ll say you might see some changes. …Like when [spouse with ALS] got his walker and wheelchair… I made sure to tell them before they came home just to give them some time to get used to that idea.” (CG19)

Navigating life with a dying spouse required caregivers to reckon with coexisting, often paradoxical truths underscoring the profound complexity of caring for a spouse with ALS during younger middle age. One participant described how, despite both of their professional and financial accomplishments, his spouse’s ALS left them reliant on significant support, which was an abrupt and painful shift:

“We were at a stage in our lives where… we had accomplished a lot. …Understanding we’re gonna need a lot of support from everybody, it felt like we had failed at some point. Like, we had done something wrong… that we’re being punished.” (CG17)

Another caregiver reflected on being both strong and capable while simultaneously in need of support:

“And I think I’m a pretty independent, outspoken, stubborn, strong-willed person, but this has humbled me and made me realize that I can’t just power through it.” (CG13)

A caregiver also described a range of conflicting affective experiences:

“I feel like I’m experiencing two extremes at once. Like on one side there is this extreme pain and or suffering but on the other hand, there’s so much love being expressed through my family, between me and her.” (CG12)

Discussion

Our study presents an understanding of how spousal caregivers of YMAs respond and adapt to their unfolding reality, which may inform strategies to better support spousal caregivers of YMAs. The overarching core process, ‘straddling two worlds,’ situates caregivers as navigating multiple dichotomies: the present and an uncertain future, the familiar and newly established norms, current and anticipated losses, and the worlds of the living and the dying. This extends previous theory of informal ALS caregiving [19, 20], which emphasized how caregivers ‘secure a balance’ among practical and anticipatory tasks [20] within relationships characterized by reciprocal love, affection, and adaptability when the caregiver is a family member [19]. Our findings show that spousal caregivers of YMAs also engage in adaptive and emotional processes that extend beyond the caregiver-care recipient relationship, alongside existential processes, as they negotiate multiple and interrelated dialectical experiences shaped by expectations of middle age.

Across interviews, participants described grappling with an uncertain future following their spouse’s ALS diagnosis. They struggled to reconcile with and plan for a future that no longer aligned with their present, a process made especially difficult by ALS’s unpredictable and terminal course. Prior studies have shown that both plwALS [12, 18, 62] and their family caregivers [10, 18, 23, 25, 36] avoid thinking about the future; however, we found spousal caregivers of YMAs did contemplate it, although reluctantly, as they struggled to make emotional and cognitive sense of their future lives with and without their partner. In the face of uncertainty, plwALS can feel that it is meaningless to plan ahead [12, 18, 24], whereas caregivers in our study planned ahead when it provided a sense of purpose or reduced uncertainty. Middle age expectations of substantial life remaining [29] and, for parents, concern for their children’s future, may have intensified our participants’ more future-oriented thinking and planning, even in the face of its painful realities.

An uncertain future sometimes created tension between caregivers and healthcare providers over expectations and urgency of planning. Some caregivers chose to access support from paid homecare workers, which in some circumstances were fully or partially funded by the provincial healthcare system. Caregivers arranged external support services ahead of time when they could control when to use them or when knowing future support would be available helped them to stay grounded in the present. For ALS caregivers, external support may create security, but shame may limit its acceptance, especially if their own feelings of independence improve wellbeing or provide hope [18]. A sudden surge in support may also undermine caregivers’ sense of control [12]. Both factors may be particularly relevant in middle age—a time when individuals are expected to be independent, active, self-focused, and not dependent or engaged in intensive spousal caregiving [32]. PlwALS often pace their use of services in line with their partner’s level of acceptance [26]; therefore, understanding the factors that shape that acceptance is essential. Building on previous findings [17], we showed that age-related norms of independence and autonomy shape when spousal caregivers of YMAs engage external support and how they experience it.

Caregivers anchored themselves in the present by deliberately limiting planning to the short term. Focusing on the present can ease the emotional burden of an uncertain future, but neglecting to plan for it may undermine manageability in ALS [18]. Conversely, our findings demonstrated that living in the moment is a key adaptive process supporting manageability of an illness marked by constant change and unpredictability. This is consistent with Charmaz [63], who found that focusing on each day helped individuals manage overwhelming illness and uncertainty by fostering a sense of agency over one’s actions, thereby reinforcing a greater sense of control over one’s circumstances. For caregivers in middle age, preserving autonomy and control may be particularly crucial.

Caregivers grounded themselves in the present by maintaining normalcy, a strategy not unique to middle age [8, 13, 20, 25, 36, 64] or ALS [65–67]. Although caregivers aimed to maintain normalcy, they gradually redefined ‘normal,’ as their partner’s dependency grew. This aligns with Ray and Street [23], who found family caregivers of plwALS continually renegotiated reality as their assumptions about life were repeatedly challenged, and Penrod and colleagues [66, 67], who described ‘seeking normal’ as a unifying process in end-of-life caregiving. Penrod and colleagues [66, 67] observed caregivers ‘building a new normal’ as the care-recipient neared end-of-life and then ‘reinventing normal’ after death. While we observed similar processes, they began well before YMAs approached end-of-life and were often aimed at maintaining normalcy for the whole family, particularly children, which may explain why they began earlier. We also observed caregivers repeatedly ‘adapting to an evolving normal’ as their spouse declined, incorporating changes in management throughout the caregiving trajectory, unlike Penrod and colleagues [66, 67], who described only a brief phase of treatment integration into everyday life in terminal illnesses like ALS. This difference may reflect slower ALS progression and longer survival amongst those with younger onset [4, 6] as well as earlier efforts to maintain normalcy in our cohort.

As they redefined and adapted to a ‘new normal,’ caregivers repeatedly confronted and grieved losses. Our participants described losses across the caregiving trajectory whereas Williams and colleagues [64] identified loss only in the final stage of caregiving. Consistent with previous findings [23], our participants described losses that were often continuous, insidious, and marked by a discrepancy between their present reality and previous hopes for the future, characteristic of nonfinite loss [68, 69]. Common following life threatening or degenerative illness, nonfinite loss is characterized by the gradual loss of a world that should have been, driven by a mismatch between an emerging ‘new’ reality and a previously anticipated future [68, 69]. As their partner remained physically present but profoundly changed, caregivers in our study experienced conflicting emotions, such as sadness intertwined with joy, complicating their sense of loss, also consistent with ambiguous loss [70–72].

Often socially unacknowledged and disenfranchised, nonfinite and ambiguous loss can generate cycles of despair, temporary adaptation, and renewed uncertainty [70, 73], which resembles our findings. These types of loss may be especially important for spousal caregivers of YMAs, as a partner’s terminal illness disrupts expectations for middle age [32] and evokes anticipatory grief over future loss in later life stages [74], both amplifying uncertainty about what has been lost. Ambiguous and nonfinite loss may also contribute to burnout and other grief-related emotions [68–70], potentially explaining higher depressive symptoms and caregiver burden among younger ALS caregivers [34–36].

Implications for practice and research

Attention to the needs and emotional wellbeing of ALS caregivers is an essential component of comprehensive ALS management [75]. Support services for family members of plwALS are needed [8]; however, psychosocial resources within ALS clinics are often limited and, when available, are primarily prioritized for plwALS. Greater awareness and acknowledgement of the often paradoxical experiences associated with spousal caregiving for YMAs may better equip providers to more effectively support caregivers and, in turn, enhance care for plwALS. Models of care that support spousal caregivers of YMAs to live and adapt in the present and maintain normalcy and personal agency are likely to be beneficial, particularly when facing nonfinite losses [69].

Family members may experience tension between an obligation to provide care and a desire to preserve other aspects of their lives [13], particularly when barriers limit access to external support. Sensitive discussions by clinicians regarding family caregivers’ capacity and willingness to provide care may help navigate this ambivalence and facilitate more sustainable caregiving arrangements. Assistance in accessing available financial supports and homecare services that meet the needs of plwALS, for those who wish to supplement family-provided care, could reduce caregiving burden and enable caregivers to maintain other valued personal, familial, and occupational roles. However, careful consideration should be given to whether family caregivers desire external support, as some informal caregivers may prefer to forgo outside services or postpone their use [13, 17]. For family members providing extensive care, the development of peer support networks may facilitate adaptation to new caregiving roles, while connecting with others with similar experiences may help provide a sense of normalcy.

Clinical approaches that promote present-focused thinking and allow for multiple perspectives of the present, such as mindfulness-based practices [76], as well as those that normalize emotional ambivalence and encourage dialectical thinking, may benefit caregivers experiencing ambiguous loss [70, 71]. Although the literature is limited, there is growing interest in mindfulness-based interventions for caregivers of plwALS [76–78]. Existing studies suggest that such interventions offer benefit [76, 77]; however, caregivers have also expressed ambivalence about certain mindfulness practices [77, 78] indicating a need for approaches that are specifically tailored to the ALS caregiver population. To our knowledge, dialectical approaches remain unexplored in ALS caregivers [79, 80] and require further research. Providers are also encouraged to expand their own tolerance of ambiguity, an essential skill for supporting others in managing uncertainty [70], and its impact on care delivery warrants further study. Table 2 presents a list of specific complementary practice and research recommendations.

Table 2.

Recommendations for clinical practice and future research

Clinical practice	Future research
Recognizing and understanding loss across the caregiving trajectory
1. Acknowledge and inquire about potential nonfinite and ambiguous losses that caregivers may be facing.	1. Explore nonfinite and ambiguous loss in ALS caregivers, considering caregiver relationship-type, chronologic age, and life stage.
Supporting family caregiver adaptation and living with loss
2. Empower family caregivers to maintain a sense of normalcy while envisioning an acceptable new normal. 3. Facilitate discussions regarding family caregiver capacity and willingness to provide care, and where aligned with their preferences, provide assistance in accessing homecare services and financial supports. 4. Develop family caregiver peer support networks that enhance knowledge sharing on adjustment and enable connections with others with similar experiences. 5. Promote present-focused coping. 6. Foster autonomy and self-efficacy. 7. Cultivate tolerance for uncertainty and normalize emotional ambivalence amid it. 8. Encourage dialectical thinking (e.g., being independent and needing support).	2. Design and assess programs that support caregivers in coping with nonfinite and ambiguous loss throughout the caregiving trajectory. 3. Assess whether family caregiver peer support networks enhance knowledge sharing, facilitate connection, and support caregiver adaptation. 4. Develop and evaluate therapeutic approaches drawing on dialectical principles and mindfulness-based interventions with attention to caregiver preference. 5. Explore alternative and more flexible strategies for data collection to help improve participant retention in family caregiving studies in ALS.
Recognizing healthcare providers’ own capacity for uncertainty
9. Encourage healthcare providers to expand their own tolerance of ambiguity.	6. Examine healthcare providers’ tolerance of ambiguity including its impact on care delivery

Strengths and limitations

Our findings may not be representative of all spousal caregivers of YMAs; however, they offer insights into processes potentially transferable to similar groups, such as middle-aged caregivers of individuals with other degenerative illnesses, or spousal caregivers of plwALS in later life who identify with a younger contextual age. Although we initially aimed to include various family caregiver types, our sample consisted only of spousal or committed partners. Future research should prioritize child and parent caregivers and those supporting YMAs with familial ALS or cognitive impairment, which were also underrepresented in our sample. Our sample included fathers, employed caregivers, and those caring for plwALS with advanced disease, with data collected while their spouses were alive, addressing prior gaps [8, 10, 15, 17].

Despite extensive recruitment efforts over two years and a flexible interview schedule, our sample size was limited due to significant loss to follow-up, a challenge reflected in other ALS caregiving studies [17, 81]. Nevertheless, we collected rich data and reached theoretical sufficiency [55], with adequate data to construct robust categories [82], and a sample size consistent with other grounded theory studies of family caregiving [65, 83, 84]. The extent of loss to follow-up may reflect the time-consuming and emotionally demanding nature of caregiving [17, 81], which may be especially significant for spousal caregivers in younger middle age who are more likely to face competing demands including employment and childcare responsibilities [33]. As a result, our data may underrepresent the most overwhelmed caregivers who may have been more likely to be lost to follow-up. Future research should explore more flexible data collection strategies, which could include participant-initiated interviews within set time windows, reminder texts or calls, asynchronous email or text-based interviews, home visits, and clinic-based interviews during natural waiting periods between care provider appointments in multidisciplinary ALS clinics.

Conclusions

This study contributes to ALS literature by presenting an understanding of how spousal caregivers of YMAs respond and adapt to their unfolding reality. The core process, ‘straddling two worlds,’ captures caregivers navigating multiple interconnected dichotomies: present and future, familiar and new norms, current and anticipated losses, and the worlds of the living and the dying. Interventions should support caregivers to stay grounded in the present, maintain normalcy and personal agency, and tolerate uncertainty and ambiguity while grieving both the loss of their partner in the present and the future they had envisioned together.

Abbreviations

ALS

Amyotrophic lateral sclerosis

MiToS

Milano-Torino

plwALS

People living with amyotrophic lateral sclerosis

YMA

Younger middle-aged adult with amyotrophic lateral sclerosis

Authors’ contributions

ASEP, LGC, LZ, and JEMS conceptualized the study. ASEP carried out data collection, data analysis, and wrote the original draft of the manuscript. LGC, LZ, and JEMS contributed to data analysis. LGC, AA, LZ, and JEMS supervised the project. All authors contributed to reviewing and editing the manuscript and secured funding.

Funding

This project was made possible by the Canada Brain Research Fund (CBRF), an innovative arrangement between the Government of Canada (through Health Canada) and Brain Canada Foundation, and by ALS Canada. The views expressed herein do not necessarily represent the views of the Minister of Health or the Government of Canada.

Data availability

Data supporting study findings are available from the corresponding author upon reasonable request.

Declarations

Ethics approval and consent to participate

Approval from the Sunnybrook Health Sciences Centre (#5599) and University of Toronto (#43666) research ethics boards was obtained prior to commencement of the study. Informed consent was obtained from all individual participants included in the study. This study was performed in accordance with the Declaration of Helsinki and relevant regulations set by the Sunnybrook Health Sciences Centre and University of Toronto Ethics Boards.

Consent for publication

Not applicable.

Competing interests

The authors declare no competing interests.