Background to the study

The narrative interview data for this paper was collected between December 2012 and July 2014 as part of the ‘Understanding and Improving the Outcome of Encephalitis’ (ENCEPH-UK) applied research programme grant funded by the National Institute for Health Research (NIHR). The programme is made up of a series of inter-related cohort studies and a range of sub-studies, further details of which can be found at www.encephuk.org. The narrative data presented in this paper was collected for one of the programme’s sub-studies, called the End User Experience study. This aimed to develop a detailed understanding about HSV encephalitis and its management from the perspective of people who had been treated for the condition, as well as their significant others.

Narrative research is a form of qualitative inquiry which focuses upon the remembered account or story in order to explore how people recall, account for, and make sense of past events and actions which occur [29]. A narrative methodology largely, but not exclusively, utilises in-depth interviews which encourage people to develop an account of their experiences [30]. Additionally, narrative interviewing allows individual accounts to be situated within a wider social milieu, in this case, medical institutions or healthcare settings [31]. This enables the research to provide insight into the social structures and cultural contexts which underpin experiences, without losing the specificity of the individual narrative.

Data collection

Participants were recruited to the End User Experience study from the main ENCEPH-UK cohort studies. These consisted of two groups of participants who were recruited from 60 hospitals across the UK: 1) a retrospective cohort who had encephalitis at any time between 2005 and 2012, and 2) a prospective cohort of patients who were recruited at the point of having suspected encephalitis in the hospital sites. The prospective paediatric patients in ENCEPH-UK were recruited via the Childhood Meningitis and Encephalitis study (UK-ChiMES); a collaborative study with the University of Oxford (www.encephuk.org/studies/ukchimes). Cases of neonatal HSV encephalitis were excluded. Due to the rarity of HSV encephalitis, additional retrospective participants were recruited by advertising through The Encephalitis Society in the UK.

Participants enrolled into the main cohort studies were purposively recruited to the End User Experience study on the basis of them having received a diagnosis of HSV encephalitis. Since it was important to understand how the experience of encephalitis had changed over time, participants were sampled across those who had recently been discharged from hospital (between 3 and 6 months, from the prospective cohort), and those who had been treated for encephalitis further in the past (between 1 and 8 years, from the retrospective cohort). Additionally, cases were varied between people treated in general hospitals and those who were managed in tertiary centres (specialist infectious diseases, neurology units, or paediatric centres).

Participants were initially contacted by phone or email by JC and were sent an information sheet about the study if they were interested in taking part. In total, 30 narrative interviews were conducted by JC with 45 participants in their own homes (17 interviews with 26 participants from the retrospective cohort, and 13 interviews with 19 participants from the prospective cohort); one participant from the prospective cohort subsequently withdrew from the study, making 29 transcripts available for the final analysis. Since people with encephalitis often had little memory of the time during their acute illness, many chose to be interviewed with a relative or friend who was present during their hospitalisation. Many of the accounts were therefore co-produced by encephalitis patients and their relatives. In the case of children under the age of 16, ethical constraints meant that their parents were recruited for interview (N = 5). Additionally, in two adult cases, interviews were done solely with their relatives. This was because in one case the patient had died, and in the other the patient had acquired severe neurocognitive problems, meaning she was unable to participate (Table 1).

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Table 1. Participant characteristics and interview details of patients with HSV encephalitis.

https://doi.org/10.1371/journal.pone.0151145.t001

The study was approved by the National Research Ethics Service (NRES), East Midlands Committee (11/EM/0442). Written consent was taken from participants prior to each interview, and all interviews were digitally recorded and transcribed in full. Participants were assigned pseudonyms, and other identifying aspects of the accounts were anonymised during transcription. The interviews focused upon participants’ experiences of: diagnosis, which encompasses the experience of initial symptoms, diagnostic investigations, and admission into hospital; the care received in hospital; everyday life post-hospitalisation; and care received after discharge from hospital. Interviews followed an adapted version of Wengraf’s format for narrative interviewing and lasted between 20 minutes and three and a half hours [30]. Consideration was also given to the level of fatigue experienced by participants, for example, since people are more commonly fatigued in the first few months post-discharge, interviews tended to be shorter for participants who had recently left hospital.

Analysis

Narrative inquiry is interested in privileging the way in which people make sense of the world around them, how they reflect on what they do within this world, and the context and production of meaning within narrative accounts. The narrative interviews for this study generated rich insight into the experience of diagnosis and treatment for encephalitis, and the processes involved in accessing and shaping amorphous care systems around the condition.

While the narratives demonstrated a diversity of experiences around these processes, the analysis was principally concerned with ‘structural commonalities’ across the accounts [32, 33]. This refers to the way in which the accounts emphasised, and were similarly shaped by, particular institutional constraints or modes of organisation: for example, how the diagnosis of HSV encephalitis was experienced as a particular issue in relation to the perceived lack of recognition for the problem from health professionals, and how people made decisions and took actions in the context of these limitations. To enable insight into these processes, we adopted a dual analytic approach [29]. This incorporated narrative analysis with a more general thematic analysis. The narrative analysis used an adapted version of Labov’s structural analytic approach [34, 35]. This focused upon examining the organisation of a narrative, in relation to how events were described and interpreted by the teller. The categories generated from the narrative analysis were then used as the basis for the thematic analysis. This was concerned with interpreting patterns across accounts, in terms of the common challenges which people were faced with when being diagnosed and treated for HSV encephalitis, and the kinds of strategies people employed in response, such as what they did when encountering particular symptoms. In so doing, we were able to characterise the conditions which give rise to the particular experiences across the narrative accounts as a whole.

In order to illustrate our findings, we present three encephalitis cases. While the cases all have their own idiosyncrasies, these cases were chosen due to their typicality in experiences across the dataset. Before turning to the results, it is important to emphasise that the narrative data we present is not seen as a ‘factual’ account of the events as they occurred, but instead, is a remembered account, where peoples’ interpretations and understandings are privileged [29, 36].

Stephanie

Stephanie first realised that something was wrong while she was out driving one day and started to feel “a bit distanced from reality and cool and separate”. Over the following few days she experienced headaches, strange “feelings of unreality” and became aware that her urine was a “peculiar dark colour”. Five days later, after realising these symptoms were persisting, Stephanie made an appointment with her GP. At the doctors, Stephanie felt that the examining GP had an “extreme disinterest” in her case: she took a urine sample and told Stephanie to wait a week for the results. Stephanie remembered “sitting there feeling unreal, aware that I really was in need and was ill, and being told quite rudely [by the GP] to get up and go”. Nine days after Stephanie first experienced symptoms, her daughter, Sarah, worried about the lack of contact from her mother, tried to telephone her. When Stephanie answered the phone in an incoherent manner, Sarah was sufficiently concerned to call an ambulance.

Once in hospital, Stephanie was told by her daughter that she spoke in a “peculiarly over simplistic fashion” to the examining doctor, who saw her ability to speak as a good sign. However, her daughter made it clear that this manner of speech was totally out of character for her mother: who worked as a psychologist and was a very articulate woman. That evening, doctors told Stephanie’s children that she was probably suffering from either meningitis, septicaemia, or, the least likely, encephalitis, and she was given antiviral medication as a precautionary treatment for encephalitis. Stephanie had a lumbar puncture the same day, and the result confirmed a diagnosis of HSV encephalitis a week later.

Using her notes, Stephanie detailed how her family were a constant presence on the ward during her time in hospital. Sarah drew up a family rota, and made sure that someone was with Stephanie every day. She also supplemented the formal care of staff by making her own notes about Stephanie’s medical charts and staying overnight with her mother when the ward was understaffed. Stephanie’s brother, a GP, sought out information about encephalitis by consulting his colleagues and staff on the ward. His acquired knowledge about the condition meant he was able to dispute the decision of doctors regarding the time-frame that Stephanie should be kept on aciclovir. Despite these issues, Stephanie was keen to highlight the care given to her by staff on the ward, who she described as “wonderful”. As an example, Stephanie recounted having “horrendous” dreams one evening, and the comfort given to her by one of the nurses, who sat with her, held her hand, and told her to concentrate on the pictures of her family pinned to the wall. Reflecting on this incident, Stephanie explained how: “that was powerful treatment; it wasn’t just comforting it was actually really powerful”.

Two weeks after being admitted to hospital, Stephanie was discharged home.

Greg and Nicola

Nicola explained that the first indication that something was wrong with Greg was when he arrived home from work one evening and told her he was tired and had a headache; she also noticed he seemed wobbly on his feet. The next day, after “stumbling” about the house and slurring his speech, Greg told Nicola that he was feeling unwell. Since Greg was not someone who usually complained when he was ill, Nicola was suitably worried and made him an appointment with the GP. In the GP consultation, Nicola explained that Greg kept on “disappearing into a world of his own” and she expressed her fear to the doctor that he might be suffering from meningitis. Concerned, the GP sent Greg to the Urgent Care Unit at his local hospital. Here, Greg was assessed for a stroke, which Nicola perceived as a mistake—since he was not exhibiting the signs of stroke, which she understood to be a drooping face and weakness on one side. When Greg’s CT scan came back normal, he was sent home to wait for an “urgent” MRI scan. The next day, Nicola became increasingly alarmed at Greg’s behaviour, recalling how she: “couldn't rouse him, short of probably punching him in the face he probably wouldn't have snapped out of it”. Unhappy with the situation, she took him back to the Urgent Care unit and insisted that they do something, telling them: “he’s not right, he’s got worse. I’m not taking him home until you find out what’s going on with him”.

Greg was started on aciclovir that evening and placed in an overflow ward; it was explained to Nicola that he had suspected viral encephalitis. In reflecting on that evening, Greg described feeling distressed at the “horrendous” conditions on the ward: the bright lights, disgruntled staff, and constant noise from elderly patients compounded his splitting headache. To make matters worse, Greg and Nicola experienced an inconsistent approach to his care. Greg explained that, a few days after he was admitted, the doctors made an “incredibly stupid decision” and stopped his medication. Two days later, after he complained of feeling unwell again, Greg was told he would need to go back on aciclovir. However, by that evening he was still waiting for the medication, and Nicola complained to the ward staff about the lack of action. She reflected on this delay in terms of the possible consequences it could have caused: “You can't say it takes 10 hours to get an antiviral medication for a patient that is potentially going to be left with a more serious brain injury if you don't give it to them soon”. Greg and Nicola were also frustrated at the poor communication from staff about Greg’s diagnosis and care plan. As a result, Nicola took it upon herself to research viral encephalitis on the internet, and requested that doctors write down answers to her questions, so that Greg could process the information in his own time. She described how her request was ignored, and doctors continued to provide information orally to Greg. Less than a week into his stay in hospital the conditions on the ward meant that Greg had “had enough” and he tried to discharge himself before his treatment had finished. He was subsequently allowed home, with aciclovir continued via a home treatment team.

Ben and Janet

Ben first experienced “strange” symptoms when he was out walking his dog one day and began to fall over. He knew something was amiss since he was not generally prone to clumsiness. Ben described feeling “really odd” after his walk, and told Janet that he thought there was something wrong with his brain. Later on that day, Janet found Ben slumped in a chair; assuming that he had had a stroke she took him to the GP and told the doctor she thought Ben should go to hospital. When the GP examined Ben he was sufficiently concerned to call for an ambulance. However, when the paramedics arrived, they disputed the doctor’s suspicion of a stroke. They questioned Janet about whether Ben had suffered headaches or been sick, and told her they thought he had a norovirus infection. Unsure of this diagnosis, Janet questioned the paramedics’ judgement, asking them whether norovirus would “cause confusion and sleeping a lot, and falling down. And he [the paramedic] said ‘oh yes it can cause all that’”.

Once at the hospital, Ben was put into an isolation unit where he was told he would have to wait to be admitted because there was a lack of available beds. In the early hours of the morning, Ben called Janet to tell her he had been discharged after the doctors had said there was nothing wrong with him. Janet was incredulous at the decision, and Ben recalls how he felt as if he was “in cloud cuckoo land”, and found it very difficult trying to call a taxi to get home. In the days which followed, Ben was, according to Janet, “out of it” and spent much of the time asleep. Unhappy with his lack of improvement, Janet called the GP who said he would see Ben the next day, and assured Janet that he would refer him back into hospital. That evening, Ben tried to get up to go to the toilet; his last memory of that day is of falling over when he tried to get out of bed. Janet later discovered Ben collapsed on the floor and immediately called an ambulance.

In the hospital, doctors informed Janet that they suspected an infection on Ben’s brain. This prompted Janet’s memory, who suggested a possible link to the cold sore virus to the doctor, explaining how “years and years ago I had read in a book or a paper that a cold sore had killed a woman, and I used to get loads of cold sores so I always worried about [it], so when he [the doctor] had said that [about the brain infection] I just said to him, ‘it couldn't be to do with a cold sore could it?’ Anyway the doctor come back and he said thanks for that [suggestion] because we could have been looking [for a diagnosis] for a long time.”

1. Access to diagnosis

2. Care within acute settings

Implications of the findings for encephalitis diagnosis and care

This paper has shown that people with HSV encephalitis and their significant others play a critical role in the diagnosis and treatment of HSV encephalitis. Specifically, they are crucial to: a) identifying that there is a serious medical problem, and b) providing a route by which a diagnosis can be made. While previous social science research has emphasised the importance of patients and their significant others in shaping patient pathways, or what has been termed ‘illness trajectories’ [38, 39, 40], this work has gone further to reveal the specific contribution that patients and families can play in helping to forge a medical diagnosis.

The route to achieving a timely diagnosis of HSV encephalitis, and, ultimately, improving patient outcomes should therefore be understood as a participatory process. In other words, the intimate knowledge of individuals and their families that something is amiss, and their subsequent actions to get help can be as crucial to diagnosis as the practices and decisions of health professionals [23]. Moreover, the role of families in facilitating a medical response to HSV encephalitis is not isolated to diagnosis: it continues into acute care and the work often required of families to adjust the care around the specific needs of their relatives. Taken together, these results have obvious implications for improving the practices involved in the diagnosis, treatment, and ongoing care for people with HSV encephalitis.

Recommendations for practice could be incorporated into the existing National Clinical Guidelines, and also be used to inform the production of a care pathway for HSV encephalitis; they involve recognising the vital work of individuals and their significant others in encephalitis management in two ways. Firstly, by taking seriously their accounts when individuals and families judge behaviour that is clearly out of character or has shifted from ‘normality’. While management guidelines emphasise altered behaviour and personality as one of the clinical features that should lead to suspicion of encephalitis, they do not necessarily acknowledge the critical role played by patients and families in recognising, and drawing attention to these changes. We therefore recommend the use of a key diagnostic question when individuals/families express such concerns: what is normal for you/your relative, and how have things/has their behaviour recently differed from this? This question could be asked not only by hospital clinicians but also by GPs and paramedic staff, who act as key gatekeepers to the provision of urgent assessment in hospital. Asking this question of patients and families may go some way to ensuring more timely recognition of the problem. Secondly, the action of families in shaping the course of acute care highlights the need to formalise the role of relatives and carers in the ongoing care for people with encephalitis. We therefore recommend that multidisciplinary treatment teams (e.g. clinicians, nurses, neurologists, physiotherapists, etc) should consider families as active team members in the process of caring for patients with HSV encephalitis, for example, by taking seriously their concerns and suggestions for patients’ needs around communication and comfort, and including them as participants in the decision-making processes of care. In so doing, this would also enable the creation of more personalised care plans, and the practical implementation of care more fully attentive to the individual needs of each patient, something which was highlighted as a key aspect of ‘good’ care within the accounts.

Recognising the roles of patients and their families in these ways, and developing the existing National Clinical Guidelines for viral encephalitis to include these aspects, may enable more a more effective approach towards the diagnosis, management, and ongoing care for people with HSV encephalitis, which would ultimately aid in improving patient outcomes.

There are a number of potential limitations of this study, and avenues for further research which need to be further explored. Firstly, since the research participants all had help from friends and family, this leaves the question of what happens to people who do not have significant others to advocate on their behalf. Future research could therefore examine how the experiences and processes of care might differ for this group. Secondly, our findings incorporated the experiences of a small number of parents whose babies and children suffered from HSV encephalitis. Whilst the recommendations for practice could obviously be applied to the management of HSV encephalitis in paediatric settings, the small sample size of parents and the variety of ages of the paediatric cases (from six months to 15 years) make it difficult to draw conclusions about the mechanisms involved in the diagnosis and care for this sample. There is a need, therefore, to conduct further research into these experiences from the perspective of parents; this would include sampling cases across a range of ages, to understand how the processes of diagnosis and treatment can be characterised for paediatrics. Details of one of the paediatric cases in this study can be found in a previous publication [41]. Thirdly, since the results in this study relate specifically to HSV encephalitis, we are not able to comment on the generalisability of the experiences to other patient populations. However, since the findings highlight institutional issues in the diagnosis and treatment of HSV encephalitis, it is plausible to suggest that they may help to make sense of, and open up questions about, the processes involved in the clinical management of other acute neurological conditions [42, 43]. Finally, a further limitation relates to how the narratives did not focus in detail on the diagnostic investigations for encephalitis, such as the experience of having a lumbar puncture. The fact that these processes were not often discussed indicates that they were not well remembered by participants, perhaps because they were overshadowed by struggles to gain recognition for symptoms. This limitation also extends to the fact that the study did not directly observe the interactions between healthcare professionals, patients, and families. Future ethnographic research is therefore needed, to extend the insights of this study by observing the processes of HSV encephalitis diagnosis and management as they occur.

Expanding the research insights in these ways would further our aim to create more responsive guidelines and healthcare interventions to improve the care of, and outcomes for people with the potentially devastating condition of HSV encephalitis.