The Perspective of Pituitary Patients on Work According to the Expanded ICF Model: a Qualitative Study

Purpose: As the majority of patients with pituitary disease are of working age, their health situation may negatively impact their functioning at work. However, work participation can also be inuenced by contextual (environmental and personal) factors. The aim of this qualitative study was to investigate the pituitary patients’ perspective on their functioning at work and on contextual factors contributing to work-related problems using the expanded International Classication of Functioning, Disability and Health (ICF). Methods: Semi-structured interviews, focusing on contextual factors inuencing work ability and experience, were conducted with eight patients with different types of pituitary adenomas (Cushing’s disease, prolactinoma, acromegaly, non-functioning adenoma). Following the steps of an experiential thematic analysis, forty categories were identied, which were organized into eleven themes according to the expanded ICF. Results: Patients reported various problems in work-related activities and work participation. Inuencing environmental factors included type of employment, perceived job security, nancial security, relationships with colleagues/managers, collaboration with others, physical vs. mental work, managing position, exibility at work, corporate culture, and physical work environment (work-related), and traveling distance to work, nancial savings, and (non)professional support (other environmental factors). Inuencing personal factors included professional ambition, sense of duty, motivation, job satisfaction, feeling of fullment (work-related), and personality, acceptance, coping styles, and lifestyle (general personal factors). Conclusion: Patients with pituitary disease report several contextual factors that inuence their functioning at work. Healthcare and occupational health professionals should be aware of these factors to improve patients’ well-being and functioning at work, ultimately aiming to improve quality of life.


Introduction
Pituitary adenomas are rare benign tumors of the pituitary gland, which can cause physical, but also cognitive and psychological symptoms [1]. As a result, many patients report a decrease in health-related Methods Participants Patients were recruited from the outpatient clinic of the Center for Endocrine Tumors Leiden of the Leiden University Medical Center (LUMC) and were selected by their consulting endocrinologist and/or neurosurgeon. All patients were of working age and were currently employed or had had a paid job at the time of diagnosis. Patient selection aimed at forming a representative, mixed group of patients regarding sex, employment status and tumor type. Two patients (one male, one female; one employed, one partially employed or unemployed at the time of the interview) of each tumor type (Cushing's disease, prolactinoma, acromegaly, non-functioning adenoma (NFA)) were asked to participate. Since it has been recommended to include six to ten participants for the interactive data collection method using interviews [28] in order to reach data saturation, we aimed to include a total of eight patients in this study. Data saturation was considered when after eight patients no new topics were raised. All patients gave written informed consent prior to participation and the research protocol was approved by the institutional Medical Ethical Committee before the interviews started (P17.142).

Study design and data collection
The consolidated criteria for reporting qualitative research (COREQ) were used for the design and reporting of this study [29]. With each patient, an individual semi-structured interview was conducted by CDA (MD, psychologist, female) and DJL (MD, male), who were both not involved in the patients' clinical care. These interviews took place between September 2017 and March 2018 in person (n = 6) or by phone (n = 2) and were performed based on a prede ned topic list (Supplement 1) focusing on problems at work and potential barriers and facilitators for functioning at work. Conversations were recorded and typed out verbatim.

Data analysis
Transcripts were analyzed following the steps of an experiential thematic analysis as described by Braun and Clarke (2013) [28]. Two independent researchers (MM, BS) carefully read and reread the transcripts in order to get familiar with the data and independently performed open coding (bottom-up) of the transcripts using ATLAS.ti version 8.4 (ATLAS.ti Scienti c Software Development GmbH, Berlin). The open coding was discussed in the research team (MM, BS, DJL, CDA), after which complete coding was performed (MM, BS). Discrepancies were discussed until consensus was reached. Based on the theory of the expanded ICF model, categories and subcategories were formulated (top-down) and reviewed. Categories and subcategories were de ned and renamed after which the analysis was nalized. Since this study focused on the patients' current and perioperative situation, statements relating to the situation prior to their diagnosis were not considered.

Results
Eight patients participated in individual semi-structured interviews (Table 1). Their median age was 56.8 years (range 40.7 to 63.8) and four patients were female (50%) (for privacy purposes, all individual patients will be referred to with male pronouns). For further clinical, sociodemographic-and work characteristics, see Table 1. After eight interviews, no new issues were raised and data saturation was reached. Forty categories were identi ed from the transcripts, which could be organized into eleven themes following the theory of the expanded ICF model (Fig. 1, Table 2). Under each ICF theme, the categories and subcategories pertaining to that theme are printed in bold.

Impairments in physical functions
All patients indicated they experienced physical fatigue and some patients reported malaise, which they described as feeling unwell. Patients associated this with being less alert, having sleep problems (cognitive impairments), a lower work pace, making more mistakes (activities), and di culty maintaining a healthy work-life balance (participation). Some patients also experienced joint problems, which did not clearly affect work in this small selection of patients that performed mainly mental work.

Cognitive impairments
Patients mentioned being bothered at work by various cognitive impairments. Several patients experienced mental fatigue, and concentration problems were experienced by all but one patient, resulting in problems with reading and the need to take more time to perform certain tasks (activities). These concentration problems were often associated with a lack of alertness, which some patients attributed to their medication. Sleep problems, such as daytime sleepiness, sleeping more hours per night, and less and/or fragmented sleep at night, were also reported. Furthermore, several patients experienced shortterm memory problems, which led to problems with reading, learning new information, and remembering to do certain work-related tasks (activities). Especially patients with Cushing's disease experienced a lower speed of information processing, most pronounced in the morning, resulting in a lack of time to do all their work. Finally, some patients reported a feeling of being overstimulated at work, particularly in relation to a busy work environment (working conditions).

Emotional impairments
Several patients reported problems with a changed personality, in the sense of experiencing changes in different aspects of their personality or having a different self-image. A change of self-image could be caused by physical or psychological changes and was often related to the inability to do certain workrelated tasks.
Furthermore, patients experienced a wide range of mood issues and negative feelings that affected their work. Patients reported indifference (being less assertive and caring less about things) towards work, which decreased their job satisfaction, work motivation (work-related personal factors), perseverance (general personal factor), and ability to assess risks (activity). They experienced that concentration problems (cognitive complaint) and the resulting lower work performance contributed to this indifference. Moreover, the inability to do things due to concentration problems (cognitive complaint) led to frustration and a feeling of shame. Several patients felt insecure about whether they still liked their job and about their functioning at work, which was related to (a fear of) making mistakes (activities). Furthermore, some patients experienced fear that they would not be able to nd a job anymore if they opened up about their disease and worried about their health and the resulting problems at work. Especially self-employed patients worried about their nancial situation (personal factor) if they could not work anymore. Finally, several patients reported a greater degree of irritability and impatience at work compared to before the diagnosis, which they related to making different risk assessments and to feelings of overstimulation.
Other emotions that were reported included anger, agitation, depressive feelings, and increased emotional instability, but patients did not perceive a direct relation between these complaints and their work.

Activities (limitations)
Due to fatigue (physical complaint) and cognitive complaints, several patients experienced that activities, speci cally reading, writing, driving to work, memorizing speeches, networking, working in a busy physical work environment, and starting to work early in the morning, cost greater effort. As patients were not able to work at the same pace as they used to, they perceived a need to take more time to perform tasks, and also reported the need to take more rest during the workday. Moreover, patients reported having to redo things due to memory and concentration problems (cognitive complaints), particularly while reading texts. Patients also experienced an inability to assess risks and to oversee the consequences of their actions at work, which they associated with a lack of alertness (cognitive complaints). Finally, several patients reported making (more) mistakes, due to memory and concentration problems, lack of alertness (cognitive complaints), fatigue (physical complaints), and the fact that activities cost more effort. This resulted in a feeling of guilt and insecurity (emotional complaints).

Participation (restrictions)
Several patients reported that the frequency of sick leave (absenteeism) had not changed due to their disease. Factors positively contributing to this were exibility of work times (task contents), nancial pressure due to freelance work (terms of employment), and a sense of duty (work-related personal factor).
However, several patients were working less hours than before their disease. Some patients had worked less temporarily after surgery during their return to work, slowly increasing their work hours. Some patients permanently decreased their working hours in order to cope with their health-related problems at work and to maintain a healthy work-life balance (participation). Due to nancial pressure, particularly self-employed patients experienced di culty working less. In order to be able to keep working, some patients made modi cations to their function or considered changing jobs.
Several patients reported being dismissed by their employer. Some of them saw a relation between losing their job and their disease. They thought their disease had affected their chances of keeping their job and related this to concentration problems (cognitive complaints) and mistakes they had made (activities).
As a result of fatigue (physical complaint) and a lack of alertness (cognitive complaint), some patients perceived a decreased work productivity (presenteeism). Others did not notice a decline, although some perceived that regaining their old level had taken a long time. Finally, patients experienced problems in maintaining a healthy work-life balance, mainly because they were so tired after a workday that they were unable to do anything at home.

Environmental factors
Work-related environmental factors

Terms of employment
Being employed by a company or self-employed affected patients' work experience. A disadvantage experienced by the patient who was self-employed at the time of diagnosis was that he felt forced to start working again shortly after surgery, mainly for nancial reasons. Patients who became self-employed just after their diagnosis (one because of a company reorganization, the other because of health complaints) experienced bene ts of being self-employed: they could choose their own work rate and did not have to explain their health complaints to an employer.
Patients reported varying degrees of perceived job security. Some patients knew they would be able to maintain their job after treatment of their disease, while others were insecure about maintaining or obtaining a job if they told (future) employers about their disease. Patients also had concerns about nancial security. Particularly freelancers felt nancial pressure to work despite their disease and were worried about what would happen if they had to stop working. Some patients experienced di culties regarding their insurance policy, fearing that they would not to be able to get a new insurance due to their disease or being unable to pay the insurance premium.

Social relationships at work
Several patients reported issues in the relationship with colleagues due to their disease. Some perceived a lack of understanding from their colleagues, resulting in con icts and mockery, as well as limited support. Others did experience collegial support after having discussed their situation, which was considered di cult, but helpful by some patients.
Some patients also experienced issues in the relationship with their manager or supervisor, such as a lack of understanding. Discussing their situation with their manager led to a feeling of support at work and resulted in solutions for work-related problems for those who did discuss it.
Lastly, several self-employed patients considered collaborating with others or hiring employees to be able to keep doing their job.
In order to improve general understanding and support at work, patients recommended making a yer about pituitary disease that can be taken to work, so that their managers and colleagues can read reliable information about their disease and its consequences.

Task contents
Various task contents in uenced patients' ability to function at work, such as the work being mainly physically or mentally demanding. The included patients had predominantly mentally demanding jobs and reported that their return to work would have been more troublesome if they had had more physically demanding tasks. Moreover, patients in a managing position experienced more exibility regarding working hours and did not have to justify their absence compared to lower-ranked employees.
In general, exibility at work was considered important. Patients employed by a company that allowed exible working hours experienced that this was bene cial for their performance at work and for maintaining their job (participation). Likewise, self-employed patients considered the exibility concerning working pace and working hours as an advantage. Patients also mentioned that the ability to work from home was bene cial.

Working conditions
Issues in the corporate culture of a company affected patients' work experience. Several patients perceived that there was no room for employees to discuss their health problems, as they believed this would lead to di culties at work and they perceived a lack of understanding for any personal issues in their company. Moreover, due to a combination of concentration problems (cognitive complaints) and an overload of stimuli, some patients were bothered by a busy physical work environment, such as an openplan o ce.

Other environmental factors
Home environment Some patients experienced traveling from home to work as burdensome, as this in uenced their energy level. Financial savings also affected patients' work experience. Some patients could afford working less (participation), because they had a nancial buffer, or a partner with su cient income (non-professional support).

Social support
Patients perceived different degrees of social support by partners and relatives varied (non-professional support). Patients with little social support felt misunderstood by their surroundings and missed talking about their condition.
Professional support was provided by occupational health professionals, endocrinologists, and psychologists. Patients had diverse experiences with occupational health professionals and the Employee Insurance Agency (Dutch: Uitvoeringsinstituut Werknemersverzekeringen [UWV]). Some were satis ed with the care they had received. In their experience, their occupational physician was supportive, acknowledged their complaints, did not exert pressure, and offered su cient support and practical measures to adapt their work to their health problems. Others perceived little or no support from their occupational physician and reported a lack of actual interventions. In the patients' view, the lack of support originated from a lack of acknowledgement of their complaints, which was caused by a lack of understanding for and knowledge of pituitary conditions.
With regards to support by endocrinologists, some patients perceived a lack of information about their disease and expressed that early expectation management concerning their work situation would have been bene cial. The lack of information had resulted in frustration about their decreased work ability after surgery (emotional complaints).
Most patients had had some form of psychological support during the course of their disease. They found this helpful to cope with work-related problems and frustrations and to create a healthier work-life balance (participation).

Personal factors
Work-related personal factors Several personal factors reported by patients were strongly work-related. Especially patients with a high professional ambition found it di cult to accept that they were not able to do everything they used to do anymore, which resulted in frustration (emotional complaint). Some patients reported that having a great sense of duty or responsibility was the reason they continued going to work despite their health complaints.
The patients reported that work was a source of joy or satisfaction, which was a motivation to continue despite their health complaints. For some patients, however, the disease and its consequences made them doubt whether they still liked their job. Patients reported that knowing that you are good at your job, making people happy, and getting appreciation for your work resulted in higher job satisfaction, a feeling of ful lment, and more motivation. In contrast, physical and mental complaints (e.g. fatigue) contributed to lower job satisfaction and work motivation.

General personal factors
Personality Patients reported several personality traits that affected their work experience and work ability. Some patients reported having a high stress tolerance that enabled them to keep functioning at work despite their disease and having a busy job. For several patients, a desire to be active was a motivation to keep working. Moreover, perseverance was a character trait that helped patients to keep going to work. Some patients reported that a goal-oriented mindset helped them to deal with problems at work.

Acceptance of the consequences of their disease
Several patients experienced di culty accepting their current level of functioning and their changed work situation.
Coping styles Since their disease, some patients experienced a decreased ability to cope with pressure at work. Patients adopted various strategies to cope with their new situation. Several patients reported that dosing their activities and energy carefully and indicating and respecting their limits was important to cope with their fatigue. One patient carefully planned his medication intake on Friday, so that he could recover during the weekend from the drowsy feeling caused by the medication. In order to deal with memory and concentration problems (cognitive complaints), patients reported writing things down more often.
Moreover, putting things in perspective helped patients in coping with their health-related problems at work. They also perceived that consciously setting priorities helped to create structure in their work tasks and helped them improve their work-life balance (participation).

Lifestyle
Several patients modi ed their lifestyle in order to cope with their symptoms, such as a healthier diet, more physical exercise, yoga or meditation, and less alcohol consumption, the latter because some believed alcohol aggravated their attention problems (cognitive complaints).

Discussion
This qualitative study showed that patients suffering from pituitary disease experience limitations in performing work-related activities and restrictions in participation at work (at least partly) as a result of the physical and mental impairments related to their disease. In addition, we identi ed several workrelated and non-work-related contextual factors that in uence functioning at work, which can be classi ed according to the expanded ICF [25].
In this study, several work-related problems were identi ed that pertain to the part 'Functioning' of the ICF.
Due to fatigue and cognitive complaints, patients experienced limitations in activities such as reading and writing and reported making more mistakes at work and needing to take more time for work tasks. These problems were related to changes in work participation, varying from modi cations of their work tasks to working fewer hours or even losing their job. Moreover, for many patients, trying to meet the demands at work was at the expense of their functioning at home.
Besides issues in 'Functioning', various 'Contextual Factors' were identi ed, which could be categorized into environmental and personal factors according to the expanded ICF. Awareness of and attention for these factors by professionals could have a bene cial effect on patients' work experience, absenteeism, and presenteeism. In previous studies, age and comorbidity were reported to affect pituitary patients' functioning at work [15]. Although these factors were not reported by patients in the present study, patients did report several other important contextual factors. For instance, patients perceived a lack of understanding regarding personal or health issues, which not only occurred among colleagues and supervisors, but also among occupational health professionals. This emphasizes the need for awareness of the largely invisible long-term consequences of a pituitary disease. As some patients pointed out, yers or electronical resources providing information on pituitary disease and its consequences could be helpful to inform employers and colleagues about the disease. Other environmental factors that could be addressed to improve patients' functioning at work are the task contents, working conditions, and factors of the home environment (e.g., travel distance). No heavy physical tasks, su cient exibility, a quiet physical work environment, and a job close to home are important to function optimally at work. However, some environmental factors are less in uenceable, such as a patient's nancial situation. In the Netherlands, self-employed people will experience negative nancial consequences if they do not have a disability insurance and work less due to their disease, whereas employed people may be eligible for a disability pension if the Employee Insurance Agency judges they have a reduced work ability [30]. Besides these environmental factors, targeting personal factors (e.g. motivation, coping styles, and lifestyle) with psychological support, psychoeducation, and self-management interventions [31] could prove bene cial for patients' work experience and productivity. The focus of psychological interventions in this patient group is on recognition and acceptance of physical and mental changes after diagnosis, and on nding an optimal balance between work demands and available mental and physical resources.
Several aspects of health-related problems at work identi ed in this study, such as fatigue, a lack of understanding by employers, and concentration and memory problems, have also been reported for other chronic conditions [32][33][34][35], including traumatic brain injury [36][37][38], Q-fever [39], and sarcoidosis [40]. Since pituitary patients report a substantial burden of cognitive complaints on their functioning at work, these complaints, which are often not easily recognized by the patients' environment, require special attention in this population. Targeted interventions speci cally aimed at work rehabilitation, such as coaching, cognitive behavioral therapy, and multidisciplinary interventions, have been used previously in other chronic disease populations and could also prove bene cial in patients with pituitary disease [41]. In terms of the ICF, these interventions will mainly address the mental functions and work-related contextual factors.
The qualitative nature of the semi-structured interviews is a major strength of the present study. It allowed for a rich exploration of the patients' perspective taking into account the context and meaning of workrelated problems. Semi-structured interview studies, however, also have their limitations, including the risk that answers of participants are in uenced by the interaction with the interviewer. However, as the researchers conducting the interviews were experienced with interview studies, the risk of this bias was considered to be low. Furthermore, almost all participating patients were highly educated and had mentally rather than physically demanding jobs, which may limit the generalizability of the results.
Despite our intention to include a diverse group of patients, patients with a lower education level were less keen on participating. Moreover, following the guideline recommending to include six to ten participants [28], eight patients were included. This sample size might have been rather small considering the number of different pituitary diseases. However, it should be noted that different pituitary diseases have clinically and symptomatically quite a lot of similarities: many patients are treated surgically, and every tumor type can cause hypopituitarism, requiring hormone replacement therapy. Therefore, it can be assumed that the sample size was su cient to provide a valuable exploration of pituitary patients' work experiences. Finally, it should be noted that considerable overlap may exist between issues reported under impairments in mental functions, and general personal factors, as described in the expanded ICF by Heerkens et al. (2017) [26].
For future research it would be interesting to develop an ICF Core Set [42], describing the expanded ICF for pituitary disease, based on the results of the present study and taking into account the healthcare professionals' perspective. This would be helpful in clinical research evaluating work-related interventions, but also in clinical care for individual patients by improving communication and collaboration between healthcare professionals (e.g., endocrinologists, endocrine nurses) and occupational health professionals. Furthermore, future research should focus on the implementation of targeted interventions in pituitary patients, tailored to individual needs, in order to improve well-being and functioning at work.
In conclusion, the expanded ICF model for pituitary diseases described in the present study can bridge the gap between the approach of occupational health professionals and healthcare professionals involved in pituitary disease. Patients with pituitary disease report limitations in activities and restrictions in participation at work, which are in uenced by several environmental and personal factors. Healthcare professionals and occupational health professionals should be aware of these in uencing factors and should address those that are modi able in order to provide holistic, multidisciplinary patient-centered care. This can improve patients' well-being and functioning at work with the ultimate goal to improve HRQoL.

Declarations
Funding: This study was partially performed with nancial support from an ASPIRE young investigator research grant (grant number WI219567, P zer, New York, USA). P zer, however, had no involvement in the project; the views expressed in this paper are those of the authors only and do not re ect the views of P zer.

Con icts of interests:
The authors declare that they have no con ict of interest.
Ethics approval: This study was approved by the Medical Ethical Committee of the Leiden University Medical Center (P17.142) and was performed in accordance with the ethical standards as laid down in the 1964 Declaration of Helsinki and its later amendments or comparable ethical standards.
Informed consent: Informed consent was obtained from all individual participants included in the study.
Author's contributions: CDA, DJL, and NRB contributed to the study conception and design. Data collection was performed by CDA and DJL. The theoretical framework was discussed and agreed upon by MM, CDA, DJL, TPMVV and YFH. Data analysis was performed by MM. The rst draft of the manuscript was written by MM and CDA and all authors commented on previous versions of the manuscript. All authors read and approved the nal manuscript.