CHEK2 (n = 8), ATM (n = 1), or PALB2 (n = 3). Women reported a wide range of feelings, both positive  and negative . All women applied strategies of emotion-focused coping to deal with this lifelong situation. Appraisal and evaluation of the affected mother’s coping might influence the patient’s own behavior and coping style. These results could be used during and after post-test genetic counseling to provide more needs-oriented counseling, and to help women in adjusting to and coping with being a pathogenic variant carrier.Hereditary breast cancer accounts for approximately 30% of newly diagnosed breast cancer (BC) cases. Pathogenic variants in moderate-risk BC genes (MBCG) differ from those in high-risk genes in terms of associated cancer risks, affected organs, and available preventive options. Little is known about how MBCG pathogenic variant carriers who have attended post-test genetic counseling perceive their situation, how they cope with their situation, and which support needs they might have. Problem-centered, guided, individual interviews were conducted with twelve women carrying pathogenic variants in MBCG. The interview analysis was based on Mayring’s qualitative content analysis. The women were between 29 and 59 years old and carried pathogenic variants in the risk genes  BRCA1 or BRCA2 in approximately 24% of cases .Another woman stated: “Yes, although I had expected the genetic test result to be positive, I have to say I was a bit shocked when I received this information. And then you are, I guess, not that receptive” .At the same time, six out of the twelve participants (50%), both survivors and previvors, also experienced feelings of relief at the time of the disclosure of the genetic test result. Four of them (33%) experienced both relief and overwhelm. One of them describes this confluence: “On the one hand, the relief. Yes, it is something. You can explain now why it was like that with my mother. But of course, there is also this shock. Oh my God, now you have it too. What will happen, if?” .The different reasons for the participants feeling relief are described in BRCA1/2 pathogenic variants. One participant felt relief because she finally had an explanation and confirmation of her increased risk. One previvor was very relieved not to have BRCA1/2 and because she finally had something concrete to hold onto. One woman was relieved because, with her having a pathogenic variant, her daughter and sister were included into the multimodal intensified breast surveillance program of the university hospital.Three women felt relief because—against their expectation—their test result was negative for The three participants (25%) who did not feel overwhelmed reported feelings of certainty and indifference regarding the moment of the genetic test disclosure. They all had experienced women suffering from BC in their nuclear family. One participant said: “But you think to yourself, ‘Yes…well…okay…you were expecting it. Just go home. Have a cup of coffee (laughing)’” .Further feelings arose during the PTGC. Three previvors experienced grief through the memory of deceased loved ones at the moment of genetic test disclosure: “So, at the first moment, I immediately had to think of my sister because she had that, too” .Two women, both survivors, explained that the genetic test disclosure was not as stressful and upsetting as the previous BC diagnosis had been: “That means this whole thing, the breast cancer diagnosis the year before, that was a lot more upsetting and awful” .One participant was not accompanied by friends or family and felt lonely during disclosure.Concerning the overall situation, seven women (58%) experienced feelings of fear or concern . These fOn the other hand, five participants (42%), all survivors, expressed rather feelings of calmness and acceptance towards the overall situation. One participant stated: “I’ll now tell you quite honestly. In my case, there is nothing I can do about it now” .Another woman shared this view: “I do everything that can be done. I regularly go for checkups” .“They [the children] have blocked this” , one participant said. Another woman stated: “They [the relatives] could not understand this” . Four women (33%) expressed a high need for safety, which was expressed in the fact that they would be willing to do anything (including risk-reducing surgery) to minimize their cancer risk in the future. One woman explained: Furthermore, four women (33%), two survivors and two previvors, experienced a feeling of a lack of understanding by their relatives regarding their way of coping with the pathogenic variant. “And I say very clearly, if there is something, operate straight away, full stop” .For three women (25%), all survivors with a family history of BC, it was important to distance themselves from the feelings and needs of other women who were in similar situations. One explained right at the beginning of the interview that she might not be the right person to talk to because she was different from others due to her individual medical family history. Another woman shared this view: “Then I’ll just say what I consider important, so that maybe afterwards you can understand why I might have very different needs than any of the other women you’re interviewing” .“the burden of passing the gene on to the children”  and the fear associated with it. She also pointed out: Concerning the feelings associated with being a carrier of a pathogenic variant, one participant spoke about “It’s also about not having any resentment towards your parents, and I think that’s very, very important, especially with such a disease that is inherited” .Another participant talked about her father’s feelings about being a carrier of a pathogenic variant: “Well, he [the participant’s father] doesn’t say it like that, he always says he didn’t have that much time. But I do think so. Yes, I think he also has a bit of a guilty conscience” .Furthermore, the women’s feelings might also be influenced by the behavior or statements of  relatives. One woman describes the repelling behavior of her niece after the genetic test disclosure: “Well, for example, my niece/I have a problem there now. We got along great with each other (...). Therefore, I was so disappointed and hurt. That was really hard” .Women with pathogenic variants for hereditary BC must cope with this situation their whole life. One aim of this study was to find out how these women manage to cope with their pathogenic variant and the lifelong uncertainty and to identify resources that they draw on. Coping was defined and classified according to the Transactional Model of Stress and Coping by Lazarus and Folkmann  into emoAll twelve women mentioned resources or coping strategies assigned to emotion-focused coping. A very important aspect was the support from their partner, family, and friends, which was mentioned by eight participants (67%). Another very important aspect mentioned by six women (50%) was strategies that can be summarized as self-care strategies. This includes meditation, mindfulness, and consideration of one’s own needs and desires and not only those of others. Further emotion-focused coping strategies were psychotherapy and self-help groups, sports and nature-related activities, religion, and acceptance and repression.Ten women (83%) described strategies classified as problem-focused coping. Taking part in the multimodal intensified breast surveillance program of the university hospital was mentioned by nine out of twelve women (75%). Another important aspect was health-promoting behavior such as healthy eating or exercise. Health-promoting strategies were mentioned by four (33%) participants. In addition, for two women (17%), the direct confrontation with the pathogenic variant and the disease was important to cope with their situation. Further problem-focused coping strategies included willingness to undergo (risk-reducing) surgery and becoming more aware of their own health and more sensitive towards body changes.Five women (42%), all survivors, mentioned aspects that reframe or reappraise the situation in positive ways and that can be classified as meaning-based coping. Three (25%) were happy to be able to share the knowledge and experience that they gained due to their BC disease with their children and other women; one of them even considered writing a book about her experiences. Two women stated that, because of their own BC disease, they became aware of what is good for themselves, and that one should enjoy life to the fullest and be grateful. Two became aware of their own personal strengths in the way they handled the overall situation. Another woman explained that it was only through psycho-oncology counseling during her BC disease that she was able to address pre-existing underlying psychological conditions that she did not deal with before.Except for one woman, all participants had a family history of BC, either in their nuclear (67%) or extended (25%) family. This experience might have influenced the women’s feeling, views, and coping style. Three women (25%) whose mothers died of cancer mentioned that they had already dealt with the topics of disease and death very intensively: “For me, it [death] is just part of it because my parents passed away very early. For me, it’s just an ordinary topic” .Another aspect of the family medical history was the evaluation and appraisal of the patient’s mother’s way of dealing with her own diagnosis of BC. Three women expressed a negative opinion about their mothers’ way of dealing with their cancer disease. One woman explained: “This generation doesn’t go to see a doctor. This is a generation that closes its eyes and sticks its head in the sand. (…) That’s why I deal with those things completely differently than my mother. And I am simply a completely different person in such matters” .On the other hand, one woman expressed a positive opinion about how her mother dealt with the BC disease: “Well, I am not afraid at all of actually getting sick. Not at all. My mom did a good job” .Five women (42%) indicated that they did not need either personal psycho-oncological counseling or support through self-help groups. For two of them, one survivor and one previvor, and both with a medical family history of BC, talking to family members was more important than talking to counselors or therapists. One of them said: “It was offered to me, to seek psychological help. But I did not want that for myself. Well, I am someone, I say of myself, I can handle that quite well because I have seen it until the end with my mother. (…) Family is more important to me, before I talk to someone else about it” .The other one had a very close relationship with her sister, who also carried a MBCG pathogenic variant and had been affected by BC, and with whom she talked a lot about the pathogenic variant and how to deal with the BC disease. The other three women did not mention an intensive exchange with their family but had no need for psycho-oncological counseling or support that was offered through a self-help group, either.Five out of twelve women (42%) mentioned that they had already seen a therapist or counselor. Two of them, both survivors, shared very positive experiences about it: “Also with the cancer, we also talked about that. So, there were many things, that had to be sorted out. And that helped me quite well” .This participant even stated that she would still be interested in further specialized counseling as offered by the university hospital, but that it was too far away from her hometown to make use of this offer. The other woman revealed: “And I perceived that [the personal psycho-oncological counseling] really helpful during that time, again and again and again” .Both were not interested in self-help groups. The other three women attended counseling once in the past but reported that they had no need for personal psycho-oncological support regarding the pathogenic variant. Two of them, one survivor and one previvor, were interested in self-help groups instead. One of them was a previvor without any contact with other pathogenic variant carriers in the family and the other woman was a survivor, who described very positive experiences regarding self-help groups during her BC disease: “Well, after my experience with psychologists, that’s quite nice, but I believe that if you talk to other affected people, you can, yes, much more relate to that. You have much more empathy and you get told much more what you can do, right?” .Two interviewees indicated that they would be interested in both psycho-oncological support and self-help groups. One was a survivor and the other one was a previvor, and both experienced the disease through their mothers and had close relationships with their families.Eventually, some interviewees added general comments regarding the topic of psycho-oncological counseling. Three of them considered it important to have local services and that there was no need to travel to the university hospital. One woman emphasized that it was important for the psycho-oncology counselor to have medical professional knowledge.The aim of this study was to explore the feelings, coping styles, and support needs of women confronted with a pathogenic variant in a MBCG. After conducting an initial systematic literature search in five databases , a qualitative approach was chosen to gain first insights into the feelings and coping situations of these women.BRCA1/2 pathogenic variant carriers can be perceived to be as threatening as being diagnosed with BC. Augestadt et al. [BRCA1 and BRCA2 pathogenic variants at the time of cancer diagnosis without receiving genetic counselling prior to genetic testing. Many of these women already felt overwhelmed and mentally overloaded when being offered genetic testing because of the amount of information that they received. The results of our study indicate that not only the diagnosis of a severe disease and the offer of genetic testing can be overwhelming and stressful, but also the disclosure of the genetic test result. This is in line with Leonarczyk and Mawn [BRCA1/2 previvors. The participants also experienced the diagnosis of the BRCA1/2 pathogenic variant as profoundly traumatic and overwhelming. The participants of our study also described that—due to this overwhelm—they were not able to fully assimilate the genetic and medical information provided orally during the PTGC. According to Kessels [The results of the study revealed a wide range of feelings, both positive and negative. Overwhelm and being emotionally and/or mentally overloaded at the moment of the disclosure of the genetic test result were mentioned by almost all participants, regardless of whether they were survivors or previvors. Studies have shown that feelings of overwhelm and emotional overload may arise when severe diseases are diagnosed. Samson et al.  indicatet et al.  conducteand Mawn , who con Kessels  and Brow Kessels , recall  Kessels ,40 and c Kessels . There iBRCA1/2 pathogenic variants. Since BRCA1/2 pathogenic variants are associated with a higher cancer risk, it may comfort some women that the test result is negative. This finding is in line with Reyes et al. [ATM and/or CHEK2 pathogenic variants. Participants of this study also reported relief due to the relatively lower risk of developing BC compared to individuals with pathogenic variants in high-risk genes, such as BRCA1/2. This information might be comforting and helpful for MBCG pathogenic variant carriers and may be communicated during the PTGC. However, Waltz et al. [BRCA1/2 as a reference or gold standard in communicating other BC-related genes and its possible implications for patients. They found out that the use of BRCA1/2 as “anchors” may confuse patients and impact their understanding of the uncertainty. Hence, this should be paid attention to when using BRCA1/2 as a reference at the PTGC. Both feelings, overwhelm and relief, can occur at the same time and demonstrate the complexity of feelings that can occur and with which the genetic counselor may be confronted during the PTGC.A further feeling associated with the disclosure of the genetic test result that was identified was relief that—other than expected—the genetic test result was negative for s et al. , who recz et al.  examinedSome feelings arose only or mainly in different subgroups. Some survivors expressed feelings of calmness or acceptance while previvors expressed fear and concern, especially when they had already experienced the BC disease through their mother. Grief and sadness were experienced during disclosure by those who had already lost close family members because of BC. This variety of feelings indicates that the feelings of affected women may differ from those of unaffected women and that the family medical history might have an influence on their needs and feelings as well. As a consequence, information and counseling might need to be adapted to these specific situations to make sure that all feelings and needs are appropriately addressed.BRCA1/2 pathogenic variant carriers. In a study by Lynch et al. [Concerning the feelings associated with being a carrier of a pathogenic variant, several feelings came up during the interviews: feelings of guilt, the burden of inheriting a pathogenic variant, the parents’ bad conscience, and the blaming of the female relatives were described by the participants. This is in line with other studies focusing on h et al. , particih et al.  and Granh et al.  use the h et al.  focused BRCA1/2 [All participants of this study used coping strategies that can be classified as emotion-focused coping according to the definition of Lazarus and Folkman , e.g., sBRCA1/2 ,22,49. TBRCA1/2 , uncertaBRCA1/2  in a quaBRCA1/2 . They foBRCA1/2  determinBRCA1/2 , social BRCA1/2 pathogenic variant carriers indicating that participation in a multimodal intensified breast surveillance program might lead to fear and worry caused by  abnormal findings, follow-up tests, and biopsies. Werner-Lin [BRCA1/2 pathogenic variant, cancer-related distress decreased significantly after having both RRM and risk-reducing salpingo-oophorectomy. The participants of our study mostly experienced the multimodal intensified breast surveillance program as helpful and positive. One reason for this difference might be the age of our participants, with only one woman under the age of 30. This young previvor’s concerns were further elaborated in Stracke et al. [In addition to emotion-focused coping, Lazarus and Folkman define problem-focused coping as the strategies that people use to manage or solve the problem that is causing the distress . The mosrner-Lin  and Hoskrner-Lin  use the rner-Lin , in unafe et al.  and weree et al. ,56. Anote et al. ,58.A further coping style that was mentioned by 42% of the participants—all survivors—was meaning-focused coping. As with emotion-focused coping, meaning-focused coping appears to be used more in situations that are chronic and cannot be changed, such as chronic illness, and it might be used when initial coping efforts fail . StudiesThe results of our study indicate that a family history of BC—especially in the nuclear family—might also have an impact on the coping style and the attitudes and views of the participants. Some women expressed a positive or negative opinion about how their mother dealt with the BC disease. This appraisal and evaluation of the mother’s coping might influence the patient’s own behavior and coping style. Studies have found that a family history of BC influences BC prevention decisions among healthy women at elevated risk of BC ,63. FurtBRCA1/2 pathogenic variant carriers by Hughes and Phelps [BRCA1/2 pathogenic variant. In addition to the calls, they encouraged the use of text messaging and/or email and concluded that a mixed-medium intervention might be preferable and the most effective. Segal et al. [BRCA1/2 pathogenic variant carriers to their offspring and also suggested that one type of support may not be suitable for all women. Eventually, a quantitative study on the support needs of unaffected women with a family history of BC found that demographic variables did not predict the interest in attending a support group [The participants had very diverse and heterogeneous views on the topics of psycho-oncological support and self-help. While some were interested in both and expressed a relevant need, others were rather reserved, preferring their family as a primary contact. This diversity was also found in a qualitative study on the support needs of affected and unaffected d Phelps , indicatd Phelps . Farrelyd Phelps  suggestel et al.  investigrt group .There are a few limitations of our study. We interviewed only one woman without children, and only one woman without a family history of BC. We recommend further studies to gain deeper insights into the feelings, coping styles, and support needs of these women. Furthermore, quantitative studies are needed to explore potential differences between the different subgroups and between the different MBCG pathogenic variants. A further limitation is the selection bias. As purposive sampling was employed, the participants might have already been interested in sharing their personal experiences. Another potential limitation is the sample size, which included just twelve participants. However, a theoretical saturation was postulated since it was possible to assign more than 80% of the codings from the 12th interview to pre-existing codes. Furthermore, the results of this study may not be transferable across different settings or to institutions with other counseling standards and processes.One of the strengths of our study is the qualitative approach, which enabled us to explore a wide range of feelings, living contexts, and circumstances on a one-to-one basis. Thanks to the protected settings of the individual interviews, the participants were able to open up and share very private thoughts and experiences and the interviewer was able to react spontaneously to statements and emotions. Another strength is the heterogeneity of the study sample: the women interviewed had pathogenic variants in different MBCG, represented both survivors and previvors, and some had completed their family planning while others had not. This provided a first in-depth insight into a broad spectrum of different perspectives, life situations, feelings, and opinions from this specific target group.The aim of this study was to obtain a closer insight into the situation of women carrying pathogenic variants in MBCG. We were able to explore feelings during and following the disclosure of the genetic test result, women’s personal coping strategies, possible influences of familial medical history, and the women’s attitudes towards psycho-oncological support and self-help. This study demonstrates that women with pathogenic variants in MBCG experience a wide range of feelings at the time of the disclosure of the genetic test result during the PTGC and continue to have a wide range of feelings several months or years after the disclosure. Negative feelings such as overwhelm, fear and concern, grief, and guilt, as well as positive feelings such as relief, calmness, and acceptance, might occur, even at the same time. This should be taken into account and might be addressed by counselors and physicians at the PTGC or at the annual multimodal intensified breast surveillance appointments, if necessary. To deal with this lifelong situation and the lifelong uncertainty posed by the pathogenic variant, all participants mentioned resources or coping strategies that can be assigned to emotion-focused coping, and most participants also described strategies that are classified as problem-focused coping. Some affected women mentioned aspects that can be classified as meaning-based coping. Women with a family history of BC—especially in the nuclear family—might express a positive or negative opinion about how their mothers dealt with the BC disease. This appraisal and evaluation of their mother’s coping might influence their own behavior and coping style. These valuable insights into the women’s feelings and coping mechanisms might help to improve the PTGC sessions, to make counselling more patient-centered, and to provide more needs-oriented information for carriers of pathogenic variants in MBCG. Concerning the topics of psycho-oncological support and self-help, the participants had a very diverse and heterogeneous view. Hence, psychological support and the possibility to join self-help groups should be offered to women carrying pathogenic variants in MBCG at the PTGC as well as at follow-up appointments.


Steve J Seligman (2/4/1931–11/11/2021) has passed away at the age of 90 years. In 2011, he joined our laboratory at the Rockefeller University, New York, as an Adjunct Professor, his primary affiliation remaining New York Medical College, Valhalla, where he had been a Research Professor since 2001. Most of his career was spent at SUNY, Downstate Medical Center, Brooklyn, where he had been an Associate (1968–1981), Full Professor of Medicine (1981–2000), and then finally Professor Emeritus (2000–2021). Before that, he was a Fellow (1961–1963) and Assistant Professor in Medicine (1963–1968) at UCLA (1961–1963). He trained as a Resident at Cornell Bellevue Hospital (1956–1957), Tufts Boston City Hospital (1959–1960), and Boston Veterans Administration (1960–1961), after attending medical school at NYU (1952–1956) and college at Harvard University (1948–1952). Importantly, he also served with the Public Health Service as a USPHS Epidemic Intelligence Officer at UCLA and USC from 1957 to 1959. These two years triggered his life-long interest in the global epidemiology of infectious diseases.During a conversation about life-threatening adverse reactions to the yellow fever (YF) vaccine, Adolfo Garcia-Sastre  recommended that he contact us. In 2011, Steve wrote his first letter to us: “Until 2001, yellow fever vaccine was considered the safest of the live virus vaccines. However, cases began to be reported of reactions to the vaccine termed yellow fever vaccine-associated viscerotropic disease (YEL-AVD). Estimates of the incidence of YEL-AVD are about 0.3–0.4 per 100,000. The disease is multisystemic and resembles yellow fever caused by a wild-type virus. Viral sequences isolated from the patients have not shown any significant differences from the vaccine. Two risk groups had been identified: vaccine recipients ≥ 60 and those who had been thymectomized because of thymomas. I have recently described a third group, women of child-bearing age in whom so far the disease has invariably been fatal in contrast to the 21% case fatality rate in men ≥ 60”.Lancet of the first papers describing severe diseases caused by the live attenuated vaccine [What Steve did not know is that we had already collected samples from some of these rare and tragic cases following the publication in the  vaccine , 2. Our  vaccine –6. We im vaccine , before Lancet based on the lessons learnt from the use of live attenuated YF vaccine [Lancet, but one issue later, Rachel Barwick Eidex summarized the total of 23 cases of YF vaccine-associated viscerotropic disease (YEL-AVD) reported since the first known case in 1996 [In 2004, Steve first expressed his concerns regarding the safety of live flavivirus vaccines in a brief review in the  vaccine , 9, whic vaccine . Also in in 1996 . Four of in 1996 . Over th in 1996 –15. In p in 1996 ?Unsurprisingly, given our “monogenic” bias , we starThe answer for these three risk groups emerged one year later, in 2020, when autoantibodies against type I IFNs were discovered to cause life-threatening COVID-19 pneumonia . Women wVaccine, it is clear that it can serve as a road map for the human genetic and immunological discoveries we made with him years later [Looking again at Fig. rs later . It now rs later .Fig. 1SHow could a small epidemiological study of the clinical features of only 64 patients predict so accurately the genetic and immunological determinants of adverse reactions to the YFV 17D virus? Perhaps the answer lies in Steve’s own words in 1996: “Persons who are effective in research have skills that differ from those of effective teachers and clinicians. These skills include the ability to generate research ideas, to access sufficient research resources, to obtain satisfaction from long-term results that is sufficient to continue frustrating tasks, and to persist in publishing and disseminating their results” . Steve’sVirology in 1959 aged 28 years [In his last e-mail to us a few months ago, entitled “What’s happening with me,” Steve wrote that “Unfortunately, I am not in good health and am living in an independent living facility. I don’t feel up to participating in conference calls. However, I am on the lookout for reports in which there is likely to have been an underlying genetic defect and will try to prepare something for us to submit. Thanks for giving me the opportunity to participate in work with your team.” At the age of 90, right up to the end, he continued working to help us understand the root cause of infectious diseases, including adverse reactions to the YF vaccine, in particular, a field to which he had made key contributions. He also hoped to write a review summarizing his 20-year-long successful quest. This obituary is not as accurate and detailed a story of this journey as he would have written. It is of particular note that, having published his first paper in 28 years , it was Steve was also a great father, grandfather, and husband. The kindest of men. A scientist, physician, and teacher who gave his all to trying to help sick people get better. He also had three core values at heart. The first was respect for science. He was dismayed at the dismissiveness of so many regarding the incredible scientific research performed of late, which has saved countless lives. The second was respect for civil rights. He was always concerned that everyone should be treated equally in every respect and fought for this in every way possible. And, the third was respect for the planet. There is so much that we can accomplish through our own actions as people to ensure that future generations have the opportunity to enjoy Earth.Steve will be sorely missed in our laboratory and by the biomedical community at large. He is survived by his sons, William and Marc Seligman, grandchildren Marisa and Evan Seligman, and his wife Hannelore Seligman, together with her children and their families: Andrew Mackles and his spouse Kristen Petterson-Mackles, their children Jeremy and Benjiman Mackles, and Paul and Ronnie Mackles, and their son Trevor Mackles.


Unaddressed, this can result in poor long-term bereavement outcomes. This paper describes the development and evaluation of Good Grief M = 16.63) participated in the evaluation. Grief intensity (Texas Revised Inventory of Grief), meaning-making (Grief and Meaning Reconstruction Inventory), trauma coping  and unmet needs (Bereaved Cancer Needs Instrument) measures were administered pre-program and 3-months post-program. Acceptability was measured after each session and at the program’s conclusion. Appropriateness was measured at 3-month follow-up. Thirteen participants were interviewed three months post-program on their perceptions of the program.One hundred and nine Australian AYAs (68% female; age: 12–25 years, Participants reported high program satisfaction, engagement with psychosocial sessions, and enjoyment of recreational activities. Significant improvements were observed in trauma coping abilities and reductions in unmet needs for managing emotions, social support, respite, future planning, and accessing information and support domains. No change was evident in grief intensity or meaning-making as measured quantitatively. Interviews supported these quantitative findings but also identified evidence of personal growth, a component of meaning-making.Good Grief is a highly acceptable and beneficial intervention that addresses the unique needs of AYAs bereaved by familial cancer. The death of a parent or sibling can be a distressing and traumatic event at any life stage. However, during adolescence and young adulthood  – a time characterised by emotional, psychological and physical development and identity formation – such a loss can be particularly stressful and impactful, having short- and long-term effects on wellbeing –7. BereaRegarding cancer bereavement specifically, a systematic review of the child and AYA sibling and offspring literature in this area was recently conducted by Hoffmann, Kaiser, and Kersting . They foPatterson and Rangganadhan  identifiAlthough there is a clear need for psychosocial support for bereaved AYAs (both those bereaved by cancer or due to other causes) to assist them effectively cope with and adjust to loss, there is a dearth of bereavement interventions for AYAs. Those that do exist are mostly targeted at children and younger AYAs  –21 ratheDesign and measurement issues also exist in the literature. As highlighted in several systematic reviews of the current literature –24, many. The GG program was designed to provide a safe and supportive environment for AYAs to achieve the following outcomes: increased coping abilities to better manage grief; reduced unmet information, respite, social support, psychological support, and practical support needs; and increased post-loss meaning-making . The name ‘Good Grief’ was chosen for the program in consultation with some of Canteen’s clients, encapsulating the idea that the grief process is a natural, normal and healthy  outcome following the loss of a loved one.To minimise the likelihood of negative short- and long-term consequences, it is essential that evidence-based and age-appropriate support is available to AYAs who have experienced the death of a parent or sibling due to cancer. To address this, we developed Good Grief (GG) – a 3-day, 2-night psychosocial programThe following theoretical and therapeutic frameworks were used in developing the GG program and informing its outcomes.The DPM posits that within the context of daily life, bereaved individuals engage in coping processes in relation to two kinds of stressor – loss and restoration , 29. LosStroebe and Schut  postulatThe constructivist approach theorises that an individual’s understanding of their identity is formed through “the stories that we construct about ourselves and share with others” . In the Research pioneered by Neff  defines In the bereavement literature, a continuing bond is defined as “the presence of an ongoing inner relationship with the deceased person by the bereaved individual” , and is This paper describes the development and evaluation of the Good Grief (GG) program – a manualised camp-based program based on the DPM and aimed at meeting the social support, coping and respite needs of AYAs who have had a parent or sibling die from cancer.An advisory group comprising of clinicians with expertise in working with cancer bereaved AYAs, AYA psycho-oncology researchers, and bereaved young people, guided the program design and development. The dual process model of coping with bereavement (DPM) was selected as the primary guide for the development of the program. With this model in mind, GG was designed to encourage active oscillation between the loss and restoration orientations during the camp; moving between therapeutic grief work and recreation/respite activities throughout the camp. This model was also explicitly discussed over the course of the program to assist participants to understand the grief process in greater depth and normalise their experience. In the development of this program we were particularly considerate of the developmental stage of those attending the camp. Therapeutic activities adopted for this program were based on approaches that attend to the formation of identity and values, and normalise rather than pathologise grief responses. Such approaches include acceptance and commitment therapy (ACT), constructivist therapies such as narrative therapy and Kristen Neff’s concept of self-compassion . We alsoDuring the program, participants were given the opportunity to share stories about their deceased loved one. A modified version of the Life Imprint  .GG is a two-night three-day camp-based program to support AYAs following the death of a parent or sibling from cancer. GG aims to provide a space for young people to: increase their coping abilities in order to better manage their grief, address unmet needs  that often arise following the death of a loved one due to cancer, and make meaning from their loss. Moreover, GG provides a place for young people to share about the life and legacy of their loved one with their peers – to acknowledge the impact their deceased family member has had on their lives, and to honour, grieve and remember them with peers who are going through a similar experience. GG has six psychosocial sessions  impacted by cancer (themselves or in their family), were recruited to the program via multiple methods, including flyers, emails, phone calls and face-to-face conversations. The program was provided at no cost to attendees. Information sheets were sent to participants, informing them of the purpose of the program’s evaluation, and that participation was voluntary and would not affect their ability to attend the program or their relationship with Canteen. Consent/assent was received from all participants and a parent/guardian if the participant was under age 18. Data were collected via paper surveys and interviews. AYAs completed demographics questions and baseline outcome measures at the beginning of the program (T0). Missing demographic data was obtained from Canteen’s client management system. They completed a program satisfaction survey at the conclusion of the program (T1). In the T2 survey, completed three months post-program, participants responded to a measure assessing usefulness, relevance and practicability and outcome measures. AYAs also completed session engagement surveys at the immediate conclusion of each program session. Participants were assigned an ID number which was used to match their surveys at each timepoint . AYAs who consented to be contacted for an interview were contacted after T2, at which point an interview was scheduled if they agreed to take part.To understand the impact of this program we used aspects of a framework for implementation outcomes  to assesSee Table AYAs who consented to be interviewed participated in a 20-min semi-structured phone interview with a researcher which was recorded and transcribed for accuracy. Interview questions explored AYAs’ personal experience of the program in greater depth than surveys, with a focus on expectations of the program, skills and knowledge acquired, and areas for program improvement.Where more than 20% of a participant’s data was missing for a scale or score calculation, their data was excluded from that analysis. Where up to 20% of a participant’s data was missing for a scale or score, a prorated scale or score was calculated using the average of valid items.AYAs’ session engagement scores represent the average of their helpfulness, meaningfulness and interestingness scores for that session. It should be noted that attendees at some programs did not complete ratings for the Honouring Ceremony due to those sessions finishing late, or facilitators not wanting to disrupt the emotional flow of the session.Helpful and Very helpful responses, divided by the total number of responses. The same was done for overall program satisfaction .For AYAs’ ratings of overall program helpfulness and topic usefulness/relevance/practicability, the percentage endorsing the program/topic as helpful represents the combined number of AYAs’ responses to the open-ended satisfaction questions  were categorised into key topics.Measure totals were calculated. For the TRIG, responses are summed to produce an overall score , with higher scores representing greater grief intensity . For theTo determine the effect of time on participants’ outcome scores, a one-way repeated measures multivariate analysis of variance (MANOVA) was conducted with time  as the independent variable and grief intensity , meaning-making , trauma coping abilities , and unmet needs  as dependent variables. For measures that had subscales and were found to be significant, further exploratory analysis was completed.A summary of participant interview responses was prepared under the key interview topics of program acceptability, meeting expectations, program benefits, personal growth, and improved coping strategies. Semantic coding of each response was completed and then codes were organised into categories . The most common categories are presented under the key topic headings, with selected quotes as illustrative examples. One person conducted detailed coding of interviews, which was examined by another reviewer for accuracy.Mage = 16.63, range 12 to 25 years; out of 151 total attendees (72%) who attended a GG program participated in the program evaluation. The majority of AYA participants were female (68%), born in Australia (97%), and currently studying (82%). Seven percent of AYA participants identify as Aboriginal or Torres Strait Islander, and 6% reported they spoke a language other than English at home. Most AYA participants had experienced the death of a parent . See Table One hundred and nine bereaved AYAs , followed by Honouring Ceremony , Coping and Resources , Sharing Memories , and Introduction of Loved One . Understanding Grief was the lowest scoring session .AYAs reported high levels of session engagement. Life Imprint was the highest scoring session . All but one AYA (99%) stated they would recommend the program to another young person whose parent or sibling has died from cancer. AYA ratings of satisfaction with facilitators ranged from 86 to 98% for all items, with high internal consistency (α > 0.8).Reflecting on the overall program at its completion, 99% of participants agreed that GG was helpful or very helpful, and 99% reported they were mostly or very satisfied with the program. High enjoyment of recreational activities was also reported  mentioned the psychosocial sessions, with 14 specifically discussing the Honouring Ceremony session. This was followed by making new friends and meeting people (n = 29), recreation activities (n = 21), and the supportive and social environment of the program (n = 10).Of the 108 AYAs who completed the measures immediately post-program (T1), in the open-ended questions section when asked what they most useful aspects of the program, the psychosocial sessions were mentioned most (n = 80). Of these, one-third discussed the Honouring Ceremony session, 12 specifically discussed the Coping and Resources session, and 11 mentioned the Life Imprint session. Following this, 11 AYAs mentioned being able to talk to and share their experiences with others.When AYAs were asked about the why they would recommend the program, the most common responses were because it was helpful (n = 33), it provided an opportunity to meet other young people going through a similar experience (n = 26), they gained useful knowledge/skills (n = 22), and it was an enjoyable/positive experience (n = 13).When detailing improving the program, the greatest number of respondents (n = 50) mentioned making changes to the program structure. Of these AYAs, 20 reported wanting the program to be longer in duration and 19 reported wanting changes to be made to sessions . This was followed by young people suggesting having more or different recreational activities (n = 16). Eighteen AYAs stated that they did not believe any changes should be made to the program.When asked about suggestions they have for Reflecting on the usefulness, relevance and practicability of the program topics for their day-to-day lives when asked as part of the three-month post-program survey, all program topics were perceived to be helpful by most young people  = 6.73, p < .001, partial η2 = .31. Analysis of the individual dependent variables was conducted. To account for multiple comparisons, a Bonferroni correction was applied. Analysis revealed no difference in total TRIG from T0 to T2  = 3.14, p = .08, partial η2 = .05). However, the GMRI Personal Growth subscale  = 6.73, p = .01, partial η2 = .10), PACT Coping Flexibility Scale  = 10.26, p = .002, partial η2 = .14) and total BCNI  = 16.64, p < .001, partial η2 = .21) were all significant at an adjusted alpha of 0.0125. Compared with T0, participants at T2 reported significantly greater personal growth-related meaning-making , trauma coping abilities , and lower unmet needs . These analyses were repeated with outliers removed. All significant results were retained with the exception of the GMRI Personal Growth subscale, which became non-significant (p = .04) at the Bonferroni adjusted alpha. To see whether our non-significant results could have occurred due to a lack of statistical power, we conducted power analyses using G*Power .”“It’s an experience that will change your life, and will go through the sections of acknowledging, of reflecting, and honouring in a really thoughtful way. And you'll meet wonderful people who will be some of your greatest friends, and that you’ll have with you for, hopefully, the rest of your life.”The high satisfaction reported in surveys was also reflected in interviews, with the majority  reported that GG had met their expectations. Common expectations discussed were learning skills to express grief, meeting others with similar experiences, and fun and respite. The remaining two interviewees reported their expectations were only partially met, with reasons including being unable to make friends, feeling that there was not equal opportunity given for group members to share in the group space, or learning fewer coping skills than expected.Most interviewees  identified making friends as a key benefit lasting well beyond the program, and nearly two-thirds reported having met others with similar experiences to their own who could understand what they were going through, with one AYA stating “that’s something really huge that I gained from that camp, was the friends I made” and “everyone in there could relate to what I was going through” and another explaining:“if you're trying to talk to your friends or someone [else] about it they don't understand how to explain and talk to you about things. They'll find it awkward and avoid it, but all these people [at the GG program] know where you're coming from”.Developing skills in expressing grief openly to others was also identified as a key benefit by just over half of the young people: “if I’m struggling with something, I will tell the friends that I met [on GG] or I’ll let my dad know… I’m more accepting of the fact that I need to ask for help”. Finally, two of thirteen interviewees mentioned feeling closer to their loved one as a result of the program, with one young person describing “I wanted to feel closer to Mum and also, sort of understand how I was feeling…I think that’s the biggest thing that I took from the camp, is that I can still have a connection with Mum”.Interviews also reflected several program benefits that young people experienced during the program and in the months afterwards. Ten out of the thirteen AYAs interviewed (77%) talked about feeling less alone as a result of the program and identifying with other participants, with one AYA stating:I think it's definitely made me a lot more stronger and a lot more aware of other people around me… it's just changed a lot of aspects of my life, really. How I treat other people. And how I expect to be treated by other people”.“Another young person said:I think there's a process of maturation that occurs, and I think that it makes you a more…- it makes you a wiser person than you necessarily would be for your age, and I think it makes you a more compassionate person. I think that it's… fast tracked the whole maturation process.”“Interviews additionally reflected AYA participants’ awareness of personal growth following their loss, with eleven of thirteen interviewees (85%) citing that they felt different since their loved one’s death. AYAs reported this personal growth occurring in a number of areas, including maturation, gaining of new perspective and assuming more responsibility, with one AYA stating:I’ve learnt to be able to cry and it’s okay to show your emotions”, and others said:“I kind of used things that I have done with dad previously [to cope]. Whenever I contribute to an activity I kind of remember, it helps me get over it a little bit. Like whenever I go fishing or camping, like even the scent of the tents kind of remind me of him a little bit”.“I think sometimes we can get a little bit lost through life and through just day to day things, but the camp really gave us a chance to sort of sit down with our feelings, and sort of go, ‘This is what's going on. This is how I feel. It's okay to feel this way’”.Improvement of coping skills and abilities was also supported by interview data, with all interviewees mentioning various coping strategies they had learned from the program. These included talking about their emotions with others, accepting their grief and acknowledging the uniqueness of the individual grief experience, openness to facing grief rather than avoiding it, writing letters to their loved one and keeping their loved one’s memory alive. Illustrating this, one AYA stated that “This paper provides evidence that GG was well received by young people bereaved due to cancer and that it significantly improved their psychosocial wellbeing. In particular, significant improvements were found in the core program outcomes, which had been identified as critical for preventing poor bereavement outcomes in this cohort. Our findings show that young people who attend the program experience the benefits that it was designed to deliver. It is expected that these benefits will provide AYAs with greater resilience and internal and external resources to walk through their grief experience and adjust to life following the death of their loved one.Young people were highly satisfied with the program, with strong convergence of survey and interview results. Almost all young people found the program helpful, satisfying and would recommend it to others. They also reported high enjoyment of recreational activities and satisfaction with facilitators. AYAs reported high engagement with all psychosocial sessions and for seven of the nine program topics at least 80% of young people reported them as helpful for their day-to-day lives. Regarding the helpfulness of the GG program’s topics, the examination of strengths and values shared with the deceased loved one, and the exploration of the shared grief experience between participants was the most highly endorsed topic. Supporting this finding, McDonald et al.  reporteda very good opportunity to learn coping skills. And to learn more about why you feel the way you feel and grief as a whole”. The PACT’s conceptualisation of coping as the flexible use of strategies relating to both facing the trauma experienced  and moving forward with one’s life  aligns with the DPM’s hypothesis of optimal adjustment to bereavement  back and I needed a set time or a set thing telling me what to do… I think, for me, it was just that I actually did get to spend that time reflecting… Which I, in all honesty, I hadn’t really done”.“Another possibility is that perhaps no change in these symptoms should be expected. On reflection, as this is a measure of grief symptoms that for the most part constitute a normal and even healthy response to loss [No change was observed in grief intensity, with post-hoc power analysis suggesting that this finding is unlikely to be due to insufficient power. Whilst we do not know with certainty why there was no change, it may have been that some young people had avoided engaging with grief prior to the program, and so the program may have provided a space for them to engage with their grief, potentially increasing or sustaining their grief intensity scores in the months following GG. Illustrating this possibility, in an interview, one AYA reported, to loss , then peWithout a control group, we were unable to account for potential changes in outcomes over time due to natural growth and recovery processes unrelated to the intervention, or due to participants potentially receiving other forms of support from Canteen. Additionally, whilst McDonald et al.  found thGG is a sustainable community-based program to which healthcare providers can refer young people who have experienced the death of a family member to cancer. This is especially important given these young people’s unmet needs and that the provision of such services is generally out of scope for primary, secondary and tertiary healthcare. Reflecting GG’s long-term sustainability, since its initial piloting, GG has been run 10 times, and has informed the development of an online grief program alternative for AYAs who cannot attend GG in person. This has been especially important during travel and social distancing restrictions associated with COVID-19. Furthermore, GG offers AYAs therapeutic opportunities beyond those that can be provided through typical one-to-one counselling services – namely peer support and the lived experience of the DPM.Healthcare services and hospitals may be unaware that their patients have adolescent or young adult children, so when providing end-of-life care to cancer patients, they should implement systems to assist with the identification of AYA family members who may benefit from referral to community-based support programs like GG  either Given the indelible impact that a parent or sibling’s death to cancer can have on young people, it is critical that theoretically driven and evidence-based programs addressing this impact are available and accessible to bereaved young people. The current study provides strong evidence for the acceptability, appropriateness and effectiveness of the GG program in meeting this need and demonstrates the vital role community organizations have in the provision of such support.


Open access funding enabled and organized by Projekt DEAL.The article “Orthorexic eating behavior and body dissatisfaction in a sample of young females”, written by Friederike Barthels, Julia Kisser and Reinhard Pietrowsky, was originally published electronically on the publisher’s internet portal on 15 August 2020 without open access. With the author(s)’ decision to opt for Open Choice, the copyright of the article changed on 18 May 2021 to © The Author(s) 2021 and the article is forthwith distributed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article is included in the article’s Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit The original article has been corrected.


According to an influential concept, humans acquire spatial knowledge about their environment in three distinct stages: landmark knowledge is acquired first, then route knowledge, and finally survey knowledge. The stage concept has been challenged by studies which observed that in a wayfinding paradigm, route, and survey knowledge emerge at the same time and; therefore, were seemingly acquired in parallel. However, this experimental evidence is not conclusive because the above studies suffered from a ceiling effect. The present study was designed to overcome the ceiling effect by increasing the complexity of the wayfinding task. We asked 60 young participants to find their way through an urban environment rendered in virtual reality, and assessed their landmark, route, and survey knowledge after each of ten trials. We found that all three types of knowledge gradually increased from the first to the last trial. We further found that correlations between the three types of knowledge increased from trial to trial. This outcome disagrees profoundly with the stage concept, but is compatible with the parallel concept. Specifically, it is in accordance with the view that landmark, route, and survey knowledge are acquired by multiple overlapping and interacting processes: those processes may start out more or less independently in the first trial but, due to common constraints or synergies, may gradually increase their cooperation during subsequent trials. Once this is accomplished, we acquire route knowledge, i.e., we learn the sequence of direction choices at intersections. This sequence can be coded directly, e.g., as “left, then straight, then right”  which, in our experience, is easy to master.We are aware of only one study that provides credible experimental evidence against the stage concept and for the parallel concept. It documented that landmarks are easier to remember when metric information about the connecting hallways is provided, which suggests that at least landmark and route knowledge are acquired in parallel  participated in this study. They were recruited by written and by online postings, as well as by personal contacts on the campus of the German Sport University Cologne. All participants were without physical or cognitive deficits by self-report. Persons who wore eyeglasses or contact lenses in their daily life continued to wear them in the experiment. This study was approved by the Ethics Committee of the German Sport University Cologne. All participants signed an informed consent statement before testing began, and were not paid for their contribution to our study.Participants learned to find their way through a virtual reality city (VR City), developed by a commercial provider with the Unity™ game engine. A large-scale downtown area with numerous streets, buildings, props, and a community park were displayed on three integrated 46-in. TV screens, located at eye level see Fig. . The midParticipants progressed forward through this environment by walking at their self-determined speed on a non-motorized, gait-powered treadmill ; to turn left or right, they pressed a switch attached to the left or right handrail, respectively, which led to a left- or rightward rotation of the virtual environment at a constant angular velocity. As a precaution against falls, participants wore a harness that was attached to the ceiling. Participants wore their daily clothes as they were in daily life. The middle monitor was in line with the participants’ medio-sagittal plane, and the other two monitors were positioned to the left and right at an angle of 106°, as if participants were actually moving in the VR City. Participants were then told that they will go along three different routes according to the experimenter’s instruction. Every route involved three landmarks . Route A involved 8 decision points, route B 10 and route C 12 decision points, for a total of 30 decision points  × 10 = 130 min.Each learning phase began at a common starting point in the public park of VR City. Route A led participants from that point towards a bakery store. After reaching that destination, participants were teleported back to the starting point and subsequently walked along route B to a book shop. After reaching that destination, they were again teleported back to the starting point and walked along route C to a red house. The same three routes were used in the learning phase of all trials, i.e., participants experienced ten times the sequence route A—route B—route C.The learning phase of the first two trials was experimenter-guided: as participants approached an intersection, they were told exactly which direction they should take. The learning phase of the remaining eight trials was self-guided: as participants approached an intersection, they called out which direction they planned to take, and the experimenter only corrected them if necessary. Thus, participants received instant error feedback and never took a wrong direction.Each testing phase consisted of a recognition and a sequence test, both administered on a laptop PC, followed by a map and a direction test, both administered on a touchscreen tablet PC. The same four tests were administered in the testing phase of all trials.First-person views of eighteen intersections were displayed sequentially on a computer screen Fig. a. Nine oThe same eighteen intersections were presented simultaneously on the monitor for 5 min Fig. b. ParticThe public park of VR City was presented in top view on the touch screen, along with the common starting point of all three routes. Participants were asked to use a touch pen and draw on that touchable screen the three routes which they had just walked Fig. c. They w.The destination of the first route was displayed on the touch screen in top view. Participants were told to use a touch pen again and draw a line from the street median towards the destination of the second route. They were then asked to draw a line towards the destination of the third route, and finally to draw a line towards the starting point Fig. d. The deWe considered the recognition test to be a selective indicator of landmark knowledge. We; therefore, calculated landmark knowledge in percent asSimilarly, we considered the direction test to be a selective indicator of survey knowledge. Therefore, we calculated survey knowledge in percent as:The remaining two tests are not as selective, however. The sequence test is often regarded as a measure of route knowledge  between predicted and actual scores.The onset of knowledge acquisition was quantified, separately for each type of knowledge, by fitting the knowledge scores with a set of models. Model 1 presumed that knowledge was acquired from the first trial on; to satisfy this model, scores were fitted by a single regression line. Model 2 presumed that knowledge was acquired from the second trial on; to satisfy this model, scores from trials 1 and 2 were fitted by a horizontal line The association between different types of knowledge was determined by calculating the bivariate correlations between landmark and route knowledge, landmark and survey knowledge, as well as route and survey knowledge. This was done separately for each trial.Figure Landmark knowledge scores were fitted by model 1 with RMSE = 22.924, which was lower than RMSE of all other models. Route knowledge scores were fitted by model 1 with RMSE = 0.6563 which was again lower than RMSE of all other models. Likewise, survey knowledge scores were fitted by model 1 with RMSE = 29.4707, which was once more lower than RMSE for all other models. We; therefore, found no evidence for the view that acquisition of any type of knowledge began later than on the very first trial.d < 0.167, all three p > 0.05; homoscedasticity: all three per inspection), we calculated a linear regression for each knowledge pair. The outcome without bootstrapping and with bootstrapping (B = 1000) was very similar; here we report the data with bootstrapping. p values are Bonferroni–Holm corrected for multiple testing. We thus yielded for the landmark—route pair a regression slope of 0.083 , which was significantly different from zero . For the route—survey pair, we yielded a slope of 0.042 , which again was significantly different from zero for . For the survey—landmark pair, we yielded a slope of 0.027 , which also was significantly different from zero . Thus, correlations increased significantly from trial to trial for all three knowledge pairs.Figure The present study deals with the acquisition of spatial knowledge in a wayfinding task that was complex enough to avoid ceiling effects. Tests of landmark, route, and survey knowledge were administered at the end of every trial, and revealed that landmark, route, and survey knowledge developed gradually and in parallel across all ten trials. Our findings should be interpreted with caution, however: they refer to the acquisition of spatial knowledge specifically in a wayfinding task, and may not fully extend to the acquisition of spatial knowledge in a map-studying task, or in other tasks see “”.By fitting different acquisition-onset models to our data, we documented that our findings are best compatible with the view that acquisition of spatial knowledge started from the first trial on; this was the case for all three types of knowledge. This outcome disagrees with the stage concept, which stipulates that only landmark knowledge is acquired during the initial trials, only route knowledge during subsequent trials, and only survey knowledge during yet later trials see “”. InsteaParallel acquisition of landmark, route, and survey knowledge could be interpreted as evidence that all three types of knowledge are different manifestations of the same memory system, i.e., of generic spatial memory. Alternatively, parallel acquisition could be interpreted as evidence that the three types of knowledge rely on distinct memory systems, and that knowledge acquisition in all three systems was triggered by the same event, namely, by exposure to the first trial. However, both views are difficult to reconcile with the data in Fig. It is interesting to note that correlations between landmark and route knowledge increased from trial to trial at a higher rate than those between landmark and survey knowledge, and those between route and survey knowledge. In terms of the cooperative systems hypothesis, this finding suggests that the mutual cooperation between the memory systems for landmark and route knowledge is closer than their cooperation with the memory system for survey knowledge. It has indeed been proposed that the processing requirements for landmark and route knowledge are particularly similar (Chrastil, It might be considered as a limitation of our study that the three routes were walked in a fixed order by all participants, rather than in a balanced order to avoid serial order effects. Note, however, that three routes that start at a common location are equivalent to a single route which repeatedly passes through a common location. Such looping routes are not unusual in real life, e.g., on an extensive, but not meticulously planned shopping trip. Our route design therefore reflects serial contingencies also found in natural wayfinding.Another limitation of our study is that route knowledge was quantified as the common factor of two tests, rather than as the score on a single test. This was necessary because available wayfinding literature offers no agreed-upon test that is sensitive to route knowledge only.In conclusion, our data provide evidence for the parallel concept of spatial knowledge acquisition, using a wayfinding task that is unhampered by ceiling effects. Furthermore, our data suggest that landmark, route, and survey knowledge may be acquired by multiple overlapping processes, rather than by three independent processes or by a single process.


Introduction: Recovery has become a catalyst for much organisational and cultural change within mental health services. Recovery involves the service user living the best life of their choice despite the presence of mental health challenges. In contrast, recovery of families remains under-developed with minimal attention given to the unique support families may require in their own recovery journeys. This paper aims to place focus on the topic through a systematic review of the literature into family recovery interventions in mental health; Method and Analysis: A PRISMA compliant systematic review was initiated. It included how the reviewers retrieved and selected studies for the systematic review. It outlined the inclusion/exclusion criteria and how these were further developed through the PICO framework. It also outlined how the reviewers assessed issues of bias and quality, as well as the process of data synthesis; Results: Three studies were included in this review. Each focusing on family recovery interventions across the lifespan: Kidstime to family toolkits to family psychoeducation. The benefits and challenges of each intervention to the family were synthesised along with a list of four family recovery enablers that are vital for the implementation of such family recovery interventions; Discussion/Implications for Practice: The results highlight the paucity of quality literature available for family recovery interventions. All three studies scored poorly in terms of quality, with one particular study (Nagi and Davies 2015) lacking quotations from participants to back up their claims. From this study, a number of actions need to be implemented, specifically around the enablers needed to allow for family recovery interventions to be fully implemented. In recent years, mental health services in Ireland have adopted a recovery-orientated paradigm/model . The con“…a deeply personal, unique process of changing one’s attitudes, values, feelings, goals, skills, and/or roles. It is a way of living a satisfying, hopeful, and contributing life even within the limitations caused by illness. Recovery involves the development of new meaning and purpose in one’s life as one grows beyond the catastrophic effects of mental illness”  (p. 21).Eolas [The notion of personal recovery as a journey, that focuses on quality of life and meaning beyond a broader set of psychosocial parameters is distinct to the idea of clinical recovery which focuses more on examining psychopathology, symptom alleviation and functionality among others . These tEolas , developSharing the Vision” [Sharing the Vision” advises that peers, i.e., service users and their families, should be core to the development and running of mental health services [A Vision for Change” [Sharing the Vision”, many documents have been cited and built on this recommendation [“ Vision”  is the cservices . Since t Change” , the preendation ,13,14.The Care Act 2014” (England) [The Carer Recognition Act” [The Social Welfare (Carer’s Allowance) Regulations” [The Carers and Direct Payments Act” (Northern Ireland) [The Carers Leave Act” [At least 182,884 people identify as family carers in Ireland . While fEngland) , offerinion Act” , states lations”  and “TheIreland) , respectave Act” .Sharing the Vision, should offer a range of support options to families as well as service users from psychoeducation to group-based aides and peer support. Amongst these services fits’ recovery interventions for families. It is with this in mind that this review was carried out.A modern and progressive mental health service, as outlined in A National Framework for Recovery in Mental Health” [Family Recovery Guidance Document 2018–2020” [In families where a member has a mental health issue, the family also require support for their own recovery”, [This review was required for a number of reasons. Firstly, while it was acknowledged in both national ,17,34,35 Health” , the “FaAs such, a systematic review of the available literature was conducted to address the following objectives:To identify what recovery interventions exist for families of loved ones attending mental health services that focus on the family member in their own right.To identify key enablers of family recovery interventions.The review was compliant with the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) standardised reporting guidelines  PRISMA iIt is now recognised as best practice when conducting a systematic review, to also create a protocol. This is useful for several reasons including (1) avoiding duplication of work, (2) maintaining accountability and (3) maintaining the rigor associated with systematic reviews. All protocols for systematic reviews are registered with Cochrane once published and a serial number assigned. The reviewers created a protocol for this systematic review. However, we were unsuccessful in registering it. As such, the protocol for this systematic review was not published or registered with Cochrane.Initially, a generic inclusion/exclusion criterion  was deveA primary search was undertaken using ten databases: CINAHL, JSTOR, OVID SP, PsycARTICLES, PsycINFO, PubMed, Science Direct, Web of Science, Wiley Online Library and EBSCHO host. Following this, secondary searches were undertaken using repositories and sources such as ResearchGate. Finally, to strengthen the review process further, a third round of searching was employed using the references from already included papers .A search time limit of 10 years (2010–2020) was adhered to by both reviewers to ensure that only the most up-to-date, best-available evidence was collected. The search terms used included: “family”, “supporter”, “spouse”, “partner”, “significant other”, “caregiver”, “relative”, “sibling”, “parent”, “adult”, “mental”, “psychiatric”, “mental illness”, “psychiatric disorder”, “mental ill health”, “intervention”, “group work”, “therapy”, “education”, “support”, “recovery”, “wellbeing”, “self-care”, “burden” and “quality of life”.There were three rounds of screening employed for this systematic review. Round one encompassed a review of titles from the search results. When reviewing titles, the reviewers saved any documents relating to family recovery into a round one folder. This folder was further divided to show documents that were found through databases and those found within repositories and other sources. Round two involved reading abstracts/executive summaries of all articles saved in the round one folder. During this time, duplicates were removed and saved in the duplicate folder within the round two folder. The initial inclusion/exclusion criteria  were appA data extraction form was developed. Study data were extracted and placed in a Microsoft Word document for purpose of data collation. This was completed by both reviewers to ensure accuracy. Disagreements between authors were resolved through the re-analysis of the particular paper in question, along with a discussion on the next steps. A trial of the data extraction form was conducted on the first two studies that met the inclusion/exclusion criteria to ensure that it captured all aspects and intricacies of the findings. Data extracted from studies included: authors, year, geographical location, study aim, sample, sample size, age range, setting, methodological approach and theoretical orientation of the given study.selection bias, performance bias and attrition bias. Disagreements between reviewers were resolved by consensus.As part of this review, bias was assessed by both reviewers  as part of the quality appraisal process. During the assessment, the reviewers assessed bias under the headings: An adaptation of  criticalThe reviewers adopted a process of thematic analyses  to analyInitially, there were 94 hits that specifically related to family recovery interventions within mental health services. This was narrowed down to 41 hits based on the research question. Further restrictions were imposed by the inclusion/exclusion criteria. After implementing the more detailed inclusion/exclusion criteria , Three sThe studies consisted of three research papers, all of which employed a variety of qualitative methodologies, service settings and category of family members. Two studies included parents within their sample with one paper respectively including partners  and siblFrom the included studies, three family recovery interventions were identified. These included a family recovery toolkit , family Included studies mentioned several benefits to the aforementioned family recovery interventions. These included: education, social inclusion and facilitation of discussions on difficult topics. Such benefits are now discussed.trusted” information provided in it by service providers [Two out of the three interventions discussed within this review have educational components to their respective intervention ,50. Ref.roviders . Such inLike , ref. 5 reportedDespite the lack of educational interventions associated with the  study. E“I’d say, this is a bit silly but I like, I quite like the games and like the easier way of understanding it.” This increased knowledge allowed the children involved to speak more freely with their understanding peers regarding their familial situation resulting from parental mental ill-health and their fears of becoming mentally unwell themselves .“When I started the Kidstime project, I felt like I couldn’t really express myself, because I know that people often thought that because my mum had mental illness I may have mental illness, so I didn’t want to say anything, because I didn’t want to seem odd or say anything inappropriate, so I kept to myself. So when I started coming to this project, you realize that, not necessarily, because when you know that other people have the same problems as you, and they look normal, they seem normal, that’s its okay to come out and just, you know, express yourself a bit more. So I just felt like I wouldn’t necessarily, after learning about the illness I felt like I wouldn’t necessarily become mentally ill, so it’s okay for me to express myself.” Only one study described a recovery intervention that surrounded a social activity with others within the mental health community: Kidstime . This so“We play games and we just talk about life and then if someone has an idea and then we talk about that.” According to , having Two studies in this review discussed how their representative initiative (recovery toolkit and Kidstime) acted as catalysts for these family members to discuss difficult topics ,51. Acco“Home was sad, Kidstime was fun. That’s what I looked forward to. I looked forward to having fun, you know being a child. But at home you have to be an adult, look after yourself, look after mum, look after the house, give her medication; at Kidstime you’re having fun. You’re being looked after and you’re not looking after others…. there are people there who are paying attention to you and you can go and speak to because you probably can’t speak to your mum because you know she’s not well she probably won’t understand. But Kidstime was time for the kids; I think that’s why it’s called Kidstime.” The above family recovery interventions are also affiliated with several challenges that impacts on not just the service but those participating in such interventions Such challenges were themed and include: hidden interventions, practicalities and age-appropriate interventions. These are now presented.Two out of the three included studies cited hidden interventions as a challenge to such family recovery interventions in mental health ,50. AccoTwo of the included studies highlighted practicalities as a potential challenge to implementing such family recovery interventions ,51. AccoRef.  was the “I would like more people my age around.” “The only downside, what I was going to say before but I didn’t want to say anything, was that, I felt more attention was paid to the younger kids than to the 15, 16 year olds.” The three studies included in the review also identified several enablers of family recovery interventions based on their research into the three family recovery interventions. Such enablers surround the inclusion of written information, access, supports and finally how decisions are made to attend/take part in interventions discussed above. These are now discussed.trusted” source [One of the included papers cited the use of written information as a way to support family recovery moving forward . Within ” source . The imp“……even just pointers to that, where you can go on and ask questions because a lot of people have, like I say great knowledge of how to deal with things and it may not be the right ones for you but it it’s ideas isn’t it, to trigger you.” Participants from all three studies reviewed all mentioned, to some degree, the issue of access either to information ,50 or to“I think maybe, that if there had have been a pack when I was going through it…., maybe the outcome for my son would have been different than it is now.” However, unlike the previous study, ref.  participOnly one study had recommendations regarding support . AlthougOnly one of the studies reviewed  discusse“I felt shy, I wondered what it was going to do, what it was about. Now I know. It is about having fun and mental illness so when my mum or dad get ill, you can help them.” There was evidence found in the review to suggest that family recovery interventions have multiple positive effects for families however, these are off-set by perceived barriers to being able to access such supports in terms of gate-keeping of information by service providers, the necessity of the service provider’s input in terms of supporting people through the recovery intervention and age-appropriateness of these interventions. Within the review findings, key enablers were identified that address such barriers and more. These enablers, if implemented correctly will allow all categories of family members to feel more involved in the entire recovery process. Findings from the review suggest that recovery interventions can take many forms but at a basic level should include offering consistent, reliable written information to families, ensuring information provided is accessible at all stages of the recovery journey particularly at the beginning and that recovery interventions are not conditional, i.e., that provision of recovery interventions should not take away from face-to-face interactions with service providers.To the author’s knowledge, no previous reviews have been carried out focusing on family recovery interventions . The finFamilies consistently articulate a need for written information ,13 and fLess clear are articulations of reasons why family members do or do not engage in interventions. While this was found to be one of the potential enablers of recovery from this review, it is not possible to extrapolate this as a definite enabler of family recovery.This review is possibly the first to examine the topic of family recovery interventions under the specific parameters set out at the beginning of the review. We used robust methods to identify, select, appraise and synthesise the articles included in the review. We followed accepted standardised reporting guidelines to ensure rigour in the reporting of the research. The review included articles on people identifying as families across the lifespan, of adults with mental health difficulties.The findings of the review are limited in the generalisable conclusions that can be drawn due to the poor quality of the included appraised articles across all domains in our chosen critical appraisal tool. The articles included in the review are all drawn from the UK which limits generalisability across different mental health settings globally. The  article The risk of bias was accessed using an adapted version of a protocol developed by  and is pThe findings of the review assist to bridge a gap in the literature and provide evidence on the types of interventions available to families in mental health services. It also highlights gaps that may exist in the provision of services to families in these settings. This information is important not only for future research on understanding family’s needs in the recovery process but also for identifying the types of recovery interventions mental health practitioners should be developing and facilitating in order to ensure the provision of high-quality, effective services to families in mental health settings.Understanding what families need to support their own recovery journey as distinct from the recovery journey of their loved one in mental health settings and knowing how to put this into practice, is a necessary aim for mental health practitioners. The findings from this review could assist mental health practitioners to address practical gaps in service provision for families within their services using an evidence-informed approach.Practical applications of this review, such as ensuring families are given appropriate information at an early stage of their recovery journey, should be easily addressed and acted upon. Added to this, the fact that national policy in Ireland supports the views put forward by families in this systematic review should ensure that mental health practitioners feel empowered to activate these changes within their workplace settings.As articulated by families themselves, there are a number of areas that could benefit from more in-depth research to assist in strengthening the evidence-base for family interventions and their constructs. This includes looking at why information may be gate-kept from families which could otherwise greatly assist them in navigating their recovery journey and that of their loved ones, what investment is needed from mental health services to adequately support families in their recovery journeys and what the unique aspect of recovery interventions for distinct family groups are, e.g., young family members, adult family members, LQBTQI+ family members and so on.With the journey towards recovery-focused mental health services for service users fully underway and becoming embedded in organisational and cultural ways across services, it is now incumbent on services to invest similarly in supporting families in their recovery journeys.The results of this review mirror much of the cultural and organisational attitudes towards families of those with mental health challenges. The results are created from few and poorly reported studies which imply a paucity of attention given to family recovery. As such, as service moves forward and develops, one must be cognisant that the family has their own recovery journey to follow too. Added to this, the process of recovery for families is somewhat similar to that of the service user, albeit with some key distinctions. This is most clearly evident through the enablers of family recovery listed above. Finally, this review also highlights that more attention is needed on a practical and research level into the process of family recovery within a mental health context.