The preservation of human ova for future fertilization has been made available to healthy women in 2011–2012. This treatment, dubbed elective egg freezing (EEF), is undertaken primarily by highly educated unpartnered women without children, concerned of age-related fertility decline. In Israel, treatment is available to women aged 30–41. However, unlike many other fertility treatments, EEF is not state subsidized. The public discourse of EEF funding in Israel is the focus of the present study.The article analyzes three sources of data: press presentations of EEF; a Parliamentary Committee discussion dedicated to EEF funding; interviews with 36 Israeli women who have undertaken EEF.Numerous speakers raised the issue of equity, claiming that reproduction was a state interest and therefore, a state responsibility, including securing equitable treatment to Israeli women of all economic strata. Highlighting the generous funding of other fertility treatments, they claimed that EEF was inequitable, discriminating against poorer single women, who could not afford it. Few actors, however, rejected state funding as intervention in women’s reproductive lives and called for reconsideration of the local reproductive imperative.The invocation of equity by Israeli users of EEF, clinicians and some policy makers as grounds for a call to fund a treatment that serves a well-established subpopulation seeking to relieve a social rather than a medical problem, illustrates the profound context-embeddedness of notions of health equity. More generally, it may suggest that using an inclusive language in a discourse of equity may potentially be invoked so as to promote the interests of a particular subpopulation. Equity in health commonly looks into access and treatment of major, potentially life-threatening medical conditions, most often focusing on underprivileged populations. The present article investigates a somewhat different landscape. First, the scrutinized service is that of elective egg freezing (EEF), a fertility preservation treatment aiming to enable healthy women conceive at a later age, when motherhood is more compatible with their lives. While addressing a life-changing social and existential concern – involuntary childless – the problem at hand is social rather than medical. Second, the relevant clientele that normally undertakes the treatment is by and large pretty well-off: healthy, well-educated women in their thirties, mostly covered by comprehensive health insurance in wealthy countries. Third, ample studies show that the majority of women who undertake EEF do not return to the clinic in order to use the preserved ova. From an equity perspective, in the field of the present research, the state of Israel, public health insurance is universal and includes, among other services, the world’s most generous coverage for fertility treatments. It is against this backdrop that the lack of funding for EEF is of distinct local significance and a cause of frustration for numerous consumers, activists and some policy-makers. This article probes the public discourse surrounding EEF funding in Israel, focusing on the issue of equity, as a somewhat exceptional use of the notion, exploring its local significance alongside a potentially more general lesson to be learned on equity. The case at hand illustrates how the notion of health equity and the use of inclusive language might be invoked so as to eventually promote the interests of a particular subpopulation, which generally belongs in a fairly resource-rich segment of society.Health policies are context-dependent, varying greatly across countries, social classes and insurers, with various bodies granting wider services in some areas than in others e.g., –5). Loca. Loca5])The technology of egg vitrification enables flash freezing of a woman’s ova, so as to be thawed and fertilized later on in the woman’s life. The treatment was originally offered in the early 2000s to young cancer patients, aiming to preserve their fertility beyond chemotherapy’s damages. Since 2011–2012, treatment is also offered for non-medical reasons, primarily, on grounds of anticipated age-related fertility decline ;), thereEEF is part of the expanding range of assisted reproductive technologies (ART) that are being increasingly used in the shaping of people’s lives and family ties. With the proliferation of these technologies, state regulation and issues of equity in this private domain have expanded. The impact of related policies goes beyond individuals, enhancing and potentially modifying social perceptions of key social notions such as parenthood and normativity alongside fairness and equity.Feminists are divided about EEF. Whereas some argue that the new technology expands gender and class equity, others contend it widens social gaps e.g., , 23–25).–25.23–25The surge in uptake may overshadow equity challenges posed by local policies. Treatment cost is a major impediment for many women. Unlike egg freezing due to medical reasons, which is often subsidized , EThough scholars debate the clarity of the division between medical and non-medical reasons for egg freezing and the related funding gaps –41, lay Marrying and having children are key to normative adulthood in Israel , 47. In In this local context, EEF flourishes. Israel’s Ministry of Health (MoH) ratified the application of egg freezing for non-medical reasons already in 2011, when EEF was still labelled experimental internationally . The staBeyond the financial burden, the lack of subsidy is symbolically significant. Given the similarity between EEF and IVF, the policy actually allocates funds by the immediate purpose of the treatment rather than the actual procedure that is being performed. Notably, when asked, Israeli students supported private funding of EEF, wishing to keep reproductive choice private, beyond state or employers’ reach .Despite the private funding, EEF flourishes in Israel as well. Already in 2015, most local IVF units were performing EEF  and demaPress materials were selected as following:The paper presents a discourse analysis by synthesizing three types of materials: 1. Press articles 2. A Parliamentary committee discussion and 3. Ethnographic interviews with Israeli women who have undertaken EEF. The specific sources are:i.Dviri, M. You turned 35 and have not yet had egg freezing? You should read this, July 1, 2021.ii.Passo, D. Why I decided to freeze eggs: 35 years Danielle Passo shares her fertility preservation, January 21, 2021.iii.Kahane, A. Egg freezing: the methods that will help you stop the biological clock, September 15, 2020.The first three articles that topped google search of the terms: “Ynet egg freezing” in Hebrew, in December 15, 2021. Ynet is one of Israel’s most popular news and general content portal in the Hebrew language , 65. Theiv.Gal, S. Should you put all the eggs in one basket? January 31, 2021.v.Mashiach, S. Anat Kamm, egg freezing is liberation from the clock tyranny, December 1, 2021.vi.Efrati, I. The number of women undertaking egg freezing has grown tenfold in 6 years, February 6, 2018.2.Knesset (Israeli Parliament) Committee for the Advancement of Women’s Status on EEF funding, held in November 30, 2021, lasting over 2 h. The meeting was fully videotaped and posted on the internet, with participants’ names and titles fully listed.A discussion in the 3.Interview material from a bi-national U.S.-Israeli qualitative study of women who have undertaken EEF or MEF, conducted between 2014 and 2016 . In IsraThe second set of articles topped google search for “Haaretz, egg freezing.” Haaretz is Israel’s oldest daily newspaper, generally identified as highbrow and liberal, with 4–6% of local readership . The thrAll three types of materials – articles, Parliamentary discussion and interviews – were analyzed with special focus on funding and related issues. Press articles were read and re-read and funding related sections were outlined. These sections were probed for emerging themes and a code was devised to capture these themes. The articles were then coded accordingly. Though various themes appeared in numerous items, each article’s framework was maintained and findings were presented chronologically per articles. The Parliamentary discussion, which was wholly devoted to the subject of EEF funding, was watched several times. The various arguments were identified and grouped according to their underlying rationale and the speaker’s role and position vis-à-vis EEF. The interviews were transcribed and a coding scheme was co-developed on the basis of emerging themes and patterns that were identified through careful review and comparison of all interview transcripts. Descriptive statistical analysis of the participants’ socio-demographic data was also compiled.These materials neither exhaust nor represent Israel’s discourse on EEF and equity. In fact, they rather have several limitations: the press articles are few; the Parliamentary discussion is but a single event; the interviewed women are also few and have undertaken EEF several years ago, when the technology was newer and dearer. Moreover, all the interviewed women were Jewish. Though this religiously monolithic profile is typical of Israel’s EEF clientele, it is nonetheless incomplete. Still, taken together, the various sources reveal several themes of interest, underlying notions of equity, healthcare and reproduction in contemporary Israel.In case the woman meets a partner later in life, preserving unfertilized ova enables fertilization by the partner’s sperm, so the embryo carries the genetics of both parents... If the eggs were frozen after fertilization, no further procedure is needed before transfer to the womb. The disadvantage is that the embryo’s genetic makeup has been determined, even if the woman has found a new partner .Cost and equity were not mentioned.The more EEF proliferated in Israel, the greater the media attention it received. In Ynet, Israel’s popular news and general content website, the top-ranking article in a Google search conducted in 2022, was published in September 15, 2020, 6 months into the Covid pandemic and 3 days before the Jewish New Year, a highly sensitive moment for many single women in Israel . ThYou’ll surely meet someone eventually and you’ll fall in love and get married, but meanwhile, you should do it, just to be on the safe side ... After all, you’ll soon turn 35.The author decribed how she kept hearing about EEFin the morning TV shows, in articles reviewing the EEF trend, in posts of celebrities who decided to freeze. Sometimes I feel that everyone is out there, queuing in front of the giant, scary freezer.After many conversations, including with her mother, the author defied:I am afraid, that’s the truth. I am afraid of being pregnant, let alone of having a baby come out of me, and I’m afraid that … I’ll be an anxious mother … Anyway, I don’t recall anyone pressuring Sarah, our Matriarch, to have egg freezing, and there she was, conceiving at 100. So for now, I can be left alone. Until then, I’ll go on being a terrific aunt, and rest assured that my nephews greatly enjoy my gifts, just saying.Thus, despite the amplifying effect of the pandemic and the imminent High Holiday, the author publicized her decision to forego EEF. Still, she softened her rejection by attributing it to personal fears rather than a critical ideology. Moreover, she positioned herself within the Jewish collectivity by invoking “Sarah, our Matriarch” and eventually presented her motherly side by “being a terrific aunt.” The rejection of EEF was thus construed as a personal particularity rather than a critique of the technology or the policy. The article revolved around maternity. Cost and equity were not mentioned.The second article was published 2 days later, a day before the Jewish New Year , depictiA decade after the ratification of EEF, not all women of reproductive age are aware that the woman’s fertility potential is time restricted.Taking for granted that motherhood is genetic and universally desired, the expert emphasized that women’s fertility was fast decaying. EEF cost or equity were not mentioned here, too.The third article, authored by a fertility clinician, overviewed the reasons to undertake EEF. It opened with a praising: “The State of Israel is an international fertility empire and a reproductive superpower” . Yet, thHaaretz’s egg freezing articles were topped by a 2018 (Efrati) front page article, published under the title: “The number of women who have their eggs preserved has risen tenfold within six years”. Alongside the growing demand for EEF, the journalist cited women’s gratitude for the opportunity to “gain control over my life” and become mothers later on, when they will have met a partner. Experts were cited praising EEF as a solution for childless unpartnered women, who previously “had to choose between becoming a mother now or give up the motherhood dream.” A clinician decsribed births following EEF by women who had met a partner after performing the procedure, “showing that women who undertake EEF have not necessarily given up on couplehood.” A member of Knesseth who has undertaken EEF herself, enthusiastically called for funding, as did a practitioner, who described a “sweeping trend” towards EEF among religious women. The calls for funding did not mention equity.turns us into walking wombs and prompts questions. Does EEF eternalize gender inequality, as men have no such ‘expiry date’? will EEF become a prerequisite for dating? Or make women wait longer, for they have ‘an insurance’?The issue of cost was mentioned twice. Two media celebrities, who had shared their EEF experiences online, highlighted EEF’s financial burden. Additionally, a professor of economics criticized “the state’s obsession with fertility, which allocates resources irrationally … We are one of the world’s densest countries and prosperity and welfare are injured by pronatalism. We should definitely not encourage natality!” At the same time, he accepted single women’s claim: “if the state is pronatalist, then it should go all the way. They are right, from their own perspective, but you don’t correct one distortion with another.” The journalist summarized by juxtaposing EEF’s benefits “for women in their early thirties who want to start a family somewhat later” alongside its clinical and financial challenges and invited debate and subtler policy making.The second article, published in January 31, 2021 , overvieThe Israeli woman’s womb does not belong to her; it belongs to the state, as a greenhouse for future soldiers or an organ that needs subsidy to be set aside for several years, so its container, the woman, can serve the army for longer years … Why should we let, not to say invite the state into our womb?... the state can justly claim that if it funds the pregnancy, it controls it … left wing activists should cry out: ‘Get out of our womb, the state of Israel!’”In a matter of hours, Haaretz published a response article, written by Israel’s IVF pioneer gynecologist . Today’s discussion is convened in order to clarify the importance of subsidizing [EEF].The participants raised various arguments in favor of subsidy: it would cohere with the state’s prioritization of natality, it would save the state “at least 4.8 million shekels ($1.5 million) a year” in unsuccessful IVF cycles to reproductively older women; EEF for women in their early 20s would reduce costs of genetic testing, as well. One clinician construed EEF as “preventive medicine, which is always better than curative treatment;” another one, as “a feminist issue today, like contraception in previous generations.” Maayan Adam, who had shared her EEF experience online, tied EEF to women’s confidence “to take a job, even in the army, to sign for longer years,” thereby reducing domestic economic violence, for women will no longer depend on their partners’ wealth.Maayan Adam, a media celebrity who had shared her EEF experience online, approached a member of Knesset (MK) and proposed to discuss EEF funding. In November 30, 2021, the Parliament Committee for the Advancement of Women’s Status dedicated a meeting to the subject. The discussion chair was a female MK from the liberal Labor party. The participants included fertility experts, two women who had shared their EEF experiences online, a former MK who had undertaken EEF and had previously called for funding, representatives of Israel’s HMOs, a representative of an NGO for fertility treatments in Israel, the fertility preservation director of Puah , was topped by a Ynet article entitled: “A [women’s magazine’s] journalist in a Parliament discussion: ‘Only a woman who has money can freeze eggs. This is a privileged procedure’” . The subAs elsewhere, nearly all the Israeli women who had undertaken EEF were well-educated, unpartnered and childless when they embarked on EEF in their later 30s . Still, all its citizens, so yes, the state should fund egg freezing!A country like ours, that believes in democracy and equality, should support women … if not full funding, at least some help. As my mother said: to have all the options open to The view of fertility as a state interest was repeatedly illustrated by its exceptional IVF funding, against which, the lack of subsidy for EEF became unacceptable. Gali’s approach was typical:What annoyed me the most is that I had to pay, because I’m having precisely the same treatment like someone who’s undergoing insemination [=embryo transfer]! … If I’d fertilized my body, I wouldn’t have to pay! And the state encourages birth! I’m doing, like, egg freezing for my fertility, why should I pay the full cost?Gilly, a school teacher, attributed the unfairness to the users not being “a couple, or a family by the book,” while Hagit, a system analyst, followed the same line and attributed EEF inequity as part of a wider discrimination:single women are always discriminated against, in tax, in the way society looks at them. It’s hard enough as it is, and such funding could contribute to the equality.Rose, a senior manager, politicized the inequity by contrasting it with the state’s generosity towards women in other life circumstances:so frustrating.There are many women, well-educated women, who say: “I won’t have children until I can support them” … That’s why I’ve waited, built a career … and am now making a lot of money … But other women … cannot pay astronomical sums … And here stands an orthodox woman, who keeps bearing children … and she gets this whole treatment for fifty dollars, because for her it’s not EEF but IVF, and this does get funded! And this demoralizes the woman who works full time and encourages the one who doesn’t!!... It is Debbie summed up: “I feel like a second-class citizen.”Nearly all the Israeli participants who had undertaken EEF agreed that the procedure should be funded, at least in part. Sigal, 40, an educational counselor, found private funding unfair:It all depends on education. The attitude towards fertility should change, there should be more choice, free choice. There’s too much intervention.Yael, a project manager, saw EEF as a personal endeavor of mature women and as such, rightly self-funded:See, at this age, you are already sort of ‘spoiled.’ So if you want to have egg freezing, you should pay for it.Sharon, a high-tech lawyer, was reluctant as she did not think that EEF addressed a medical problem. Two other women, both newcomers to Israel, rooted their ambivalence in Israel’s wider reproductive scene. Emily, 35, from the UK, claimed that “the pressure here is very strong, so it’s very difficult to say ‘I don’t want children’, extremely difficult.” Jennifer, a doctor from the U.S., was the only one who questioned Israel’s IVF policy up front:I’m not sure if the state should share [EEF] cost. There’s also a question about IVF and how much the state should fund... And there’s a broader question if one can be a parent at any age, things like that. Of course, when it comes to one’s private case, no one cares and just wants everything for oneself.On the whole then, EEF users, who called for state subsidy, hailed reproduction as a state interest, as manifested in the exceptional IVF policy and therefore claimed that the state must ensure that every woman could conceive, irrespective of her economic and family circumstances. From this perspective, lack of funding was perceived as inequitable. In contradistinction, the few women who voiced reluctance about EEF subsidy, questioned existing policies and ideologies, including the maternal imperative, but have not invoked the issue of equity.Other women were, however, more ambivalent regarding state subsidy. Interestingly, these more critical women have not mentioned fairness or equity. Moriah, a graduate student, hesitated, out of concern that EEF funding might burden the state budget and compromise women’s reproductive autonomy. However, eventually, she thought that HMOs were making money out of childbearing and therefore should fund EEF. Adi, 36, an actress, called for a broader change. She, too, did not mention equity:Starting with the more reluctant views, we note that speakers who disproved of EEF subsidy raised a range of arguments to ground their opinions, e.g., condemnation of the interference in women’s reproductive lives, critique of Israel’s ‘distorted’ IVF policy and “obsession with fertility,” enhancing the local bio-maternal imperative. At the center of the discourse, the speakers have placed broader issues related to the local reproductive policy and ideology. None of these speakers has referred to inequity as grounds for rejecting subsidy.In contrast, advocates of state subsidy repeatedly brought up the issue of equity. Portraying Israel as a singular microcosm where childbearing is of exceptional import to individuals as well as the state, these actors endorsed inclusive language when claiming that the state was responsible of ensuring that local women of all material and family circumstances could access the treatment they needed in order to conceive. Subsidy supporters construed EEF funding as a civil issue and as such, deserving public funding in order to be equitable. The exceptional funding of IVF served as proof of the unfairness of denying state support from women who required EEF. Any link between a woman’s financial standing and maternal plans was repudiated as illegitimate. The fact that the majority of the potential users were singles – a life situation widely viewed in child-centered Israel , as elseA historical perspective might help elucidate the seeming contradiction vs. IVF policy. Israel’s IVF policy was formed in the 1980s, when, the country’s population was less than half its current size . TodayIf this is indeed the case, then refraining from EEF subsidy, as decided by the MoH Basket Committee, may be highly adaptive. Economically, it saves state resources, at least in the short term; politically, EEF is not as entrenched in local expectations and its funding has never stirred a major public protest; symbolically, the denial of funding may gently signal a shift in the state’s demographic interest.How should we read the fact that equity was mentioned exclusively by subsidy advocates? On its face, this is obvious: people who seek greater equity demand state support in order to secure equitable access to service. The case at hand is, however, atypical. EEF is a treatment serving young healthy women seeking to relieve a problem that is social rather than medical. The nature of the clientele was indeed visible on the public scene: the women who have undertaken EEF were journalists, celebrities and a former MK, alongside clinicians in lucrative practices. One might argue that this social profile reflects the private funding, which bars women of lesser means from the costly treatment.Relevant statistics might shed some light. First, Israel’s poorest women concentrate in Arab and Jewish ultra-orthodox communities. Though more religious and even Ultra-orthodox Jewish women undertake EEF , women iThe economic aspect of this generalization might be challenged by recalling that in the aforementioned bi-national U.S.-Israel study, over half the Israeli participants had their EEF funded by their parents. This finding might be reconciled with the current argument in several ways. First, cost of living in Israel is extremely high and even well-educated single women who work full time may find it hard to save for exceptional expenses like EEF. Second, judging by the interviews, parental funding did not necessarily represent the woman’s financial limitations. In some cases, it was rather a symbolic statement of parents’ support of their daughter’s decision. Third, several women were actually unsure whether they wanted to have children and pursued EEF largely because of their parents’ requests. Fourth, some women maintained very close ties with their parents, rendering economic boundaries somewhat blurred. Either way, the parents’ funding also attests to the relative strength of the women’s families of origin. Indeed, some of the women in the study were working in less remunerative jobs as they somewhat relied on their parents’ wealth. Beyond these explanations lies the decrease in EEF cost, which has roughly halved since the time of the bi-national study, rendering the procedure affordable to a wider clientele. Within this perspective, a state subsidy might be seen as directed to serve a relatively well-educated well-earning segment of the population.To further contextualize the subsidy demand, one should consider the wider context of Israel’s healthcare. Though offering full national coverage, the service is far from equitable. Some sectors have been experiencing unequal access and care for years . Infant Against this backdrop, calling for EEF subsidy in the name of equity raises several questions: How pressing is EEF vis-à-vis other local health needs and resources? How difficult is it for potential local users to fund EEF on their own, now that prices have dropped? What message would EEF subsidy convey to Israeli citizens regarding the state’s view of the acuteness of childbearing? These questions would stand out especially sharply given the lack of subsidy for EEF in public healthcare systems elsewhere, including in countries much wealthier than Israel (with the noted recent exception of France). Indeed, IVF is funded in Israel more generously than elsewhere. Yet, IVF funding dates back to the early 1980s, it aims to relieve a medical problem and is more efficient in terms of actual natality. All these aspects will gain further significance with the current challenges brought about by the formidable demographic growth.And yet, one can well understand the perspective of Israeli women who call for EEF subsidy. Studies have shown that women who undertake EEF in settings where IVF is reimbursed, feel discriminated against by what they view as policy inconsistency , 40. ThiIs there a more general lesson to be learned about equity from the highly particular case of EEF in Israel? The bi-national egg freezing study may be instructive. When invited to recommend improvements to treatment, U.S. study participants suggested a whole range of complementary services, like stand-alone EEF clinics, after working-hours information sessions and injection classes, diverse staff specializing in EEF for single, lesbian, ethnic, and religious minority women, transportation and home care on day of egg retrieval, in-house psychologists specializing in EEF needs of single women, especially ones left by their partners. Alongside, insurance coverage and fee reduction were also mentioned. Israeli participants, in contrast, have concentrated almost exclusively on the need for public subsidy .The sharp difference outlines each group’s distinct perception of ideal healthcare. In the U.S., where healthcare is primarily private and commercial, the participants have listed a range of supportive services, aimed at improving women’s treatment experiences. The Israeli women, in contrast, have all been used to public healthcare, which, despite considerable erosion, forms the backbone of local health services. U.S. participants have thus basically accepted EEF as a privately funded service but called for an improved treatment envelope. Israeli women have not even envisioned such services, but were appalled that they had to pay. The women’s expectations thus reflected the systems they have been using, with American participants expecting more comprehensive and supportive service than Israelis, who had lower expectations but protested against inequitable access.Equity thus emerges in its context embeddedness. People’s views of what is fair and equitable are cast by their experience-based perceptions. What is considered inequitable and raises public resentment in one context might not be considered an option at all in another. The very use of the concept of ‘equity’ as a strategic campaigning tool may therefore teach us about local systems’ scope and priorities. “Health equity – underpinned by social justice – needs to be embedded at the structural level to support effective prioritization,” contended van Roode et al. . Yet, ‘sFuture studies may assess the impact of existing EEF funding policy on local women and on local notions of health and equity. The answers may inspire a wider investigation of the health policy-equity nexus and its sensitivity as reflecting and influencing local notions underlying healthcare funding.The present study reveals the crucial weight of local contexts in the shaping of perceptions of desirable healthcare and subsequently, of notions of health equity. Thus, in one context, private funding of a specific service may be taken for granted as ‘natural’ while in another context, paying for the same service out of one’s own pocket – even a much lower sum – may create a sense of discrimination among users and invoke calls for equity in the name of fairness and social justice. As such, the circumstances in which calls for equity are invoked may serve as a sensitive tool for learning about local ideals of healthcare and about public expectations from local services, as well as the diversity of notions of state responsibility, fairness and equity. At times, state actual responses to such calls for equity and the rift between these responses and public declarations may also point at emerging, possibly not-yet-declared changes in state policies and interests.


The editorial shares a collective vision for Kyiv becoming a regional resource for teaching, practicing, and advancing neurophotonics - with a recipe for developing a summer school program as a forward-looking way to start. IBRO summer school we had in Valdivia, Chile. It was called “Dynamic imaging in neuroscience.” The Valdivia location was chosen by the IBRO president at that time, Pierre Magistretti, because it offered affordable full boarding and travel for Latin American students. The other and equally important reason was a strong local base thanks to Felipe Barros and his laboratory at the Centro de Estudios Científicos.I love summer schools. As a student, I never got a chance to attend one, but as a teacher I have contributed to many related to neurophotonics. Summer schools combine all of my favorite things: summer, the excitement of doing and teaching immersive science, the joy of seeing friends and colleagues, and interesting destinations. My favorite so far was an So, here is a recipe for an awesome summer school:1.Choose a location with (a) a strong and committed local base but (b) away from the main roads to drive down the registration fees.2.Find sponsors among nonprofit organizations and industry.3.Attract world experts to serve as faculty.4.Select applicants following the principle of inclusivity while giving priority to regional students for whom this school would provide a unique, affordable opportunity.5.Get together and enjoy science! Well, I skipped over a few details, but this is the essence.Pavel Belan, Nana Voitenko, and Olga Garaschuk who have a collective vision of Kyiv becoming a regional resource for teaching, practicing, and advancing neurophotonics.The secret ingredient for 1a  is local afficionados and program builders. On that note, I’d like to introduce to you my new Ukrainian friends Kyiv Academic University (KAU). Pavel is a physicist by training who specializes in biophysics applied to synaptic transmission and neuronal excitability. His research relies heavily on optical tools such as confocal microscopy and FRET. His vision is to build a thriving, multidisciplinary program in Kyiv that would bring together technology and biology experts under a single umbrella sharing the same neuroscience mission. Such program would attract and retain local talent. After all, there’s no place like home. A strong program combining neuroscience and photonics would be a place to come back to after spending some time abroad.Pavel Belan chairs the Department of Biomedicine and Neurosciences of Ukrainian Global University, a new global network that aims to connect Ukrainian students and scholars with educational institutions around the world. We want a strong connection to international community, but we also have to avoid a brain drain. A local Neurophotonics Center in Kyiv serving as a base for an international summer school would achieve multiple goals: push the development of the local base, serve the region, strengthen ties with world experts, raise the standards of local high education. Our ultimate ambition is to lead. Ukraine is a big country. We must leverage the depth of talent available to us at home.”“At KAU, we believe in transdisciplinary approach,” says Pavel. “We apply physical and chemical approaches to develop new methods and then apply these methods for solving biological problems. Recently, KAU became a partner of the Nana Voitenko is on a mission to establish the Dobrobut Academy Medical School that will be a new kind of medical school in Ukraine. In current practice, Ukrainian medical universities often are disconnected from research centers and hospitals, so students have no exposure to medical research and can receive a diploma without ever seeing a patient. Doesn’t sound like a good idea? Well, because it isn’t. In the West, medical school is followed by years of residency in a real-world clinical setting. Ukraine, however, inherited a Soviet-era system, where researchers and clinicians hardly participate in medical education. Nana is determined to bring a change.Nana’s own research revolves around calcium imaging in sensory and spinal cord neurons for understanding the mechanisms of chronic pain. She imagines a future where a local neurophotonics resource will serve her medical students. “I see Dobrobut Academy Medical School adopting the Western model of medical education to generate a cadre of world-class medical experts in Ukraine,” says Nana. “Like in the US, some of them will be MD/PhD students who eventually will lead their own laboratories combining medical practice with research. With growing importance of optical tools in biomedical research, related to brain and beyond, building a body of local neurophotonics expertise will play an important role in building and sustaining medical excellence.”Olga Garaschuk is a Ukrainian-born neuroscientist living and working in Germany. A professor at University of Tübingen, Olga is widely recognized for her work on two photon calcium imaging in living, behaving brains. In 2003, she published a seminal paper in PNAS that opened the door for delivery of fluorescent calcium probes to many neurons in deep brain tissue at once; today we call it “bulk loading” of synthetic probes. This technique has enabled, for the first time, to image activity of neuronal networks not in a dish but in the brain where they belong! Remember, this was before the advent of genetically encoded fluorescent probes.EMBO training courses for live two-photon imaging. As a co-initiator of the Ukrainian Academic International Network (UKRAINET) and the President of German–Ukrainian Academic Society, she organized and participated in several summer schools in Ukraine, the last taking place in Lviv in August 2019. Olga and Nana were busy planning the next one when the COVID pandemic arrived, putting their plans on hold.Olga is a sought-after teacher and frequent participant of summer schools and short courses in neuroimaging. She has organized and taught several “But wait,”—you may say—“forget about COVID, there is a war in Ukraine. Why are we talking about neurophotonics programs and summer schools in the war zone?” Yes, there is a war. Something that was unthinkable just a few months ago has become a brutal reality. But Ukrainian people are resilient. They look to the future, and so should we. Besides, building programs takes time, so we’d better get started!Here are a few additional useful links:ScienceForUkraine is an academic community group of volunteers who collect and disseminate information about support for Ukrainian students and scholars.UAScience.reload is an initiative of the Ukrainian diaspora who advocate for “preserving, updating and reloading” the Ukrainian scientific community.


Surf therapy is increasingly used as a health intervention, but evidence of its mental health benefits remains unclear. This longitudinal mixed-method study assessed the usability and acceptability of a novel online data collection tool and process to measure the impact of a surf programme on acute and chronic mental wellbeing. Fifteen women attending a 6-week surf programme in the UK were asked to complete a tool consisting of video recordings, word association and the Short Warwick-Edinburgh Mental Well-Being scale (SWEMWBS). Usability and acceptability were assessed through focus groups and quantitative data. The data generated in the focus groups, video recordings and word association were analysed via reflexive thematic analysis, and SWEMWBS presented descriptively. Participants perceived the tool as easy to use due to the completion time and its functionalities, and useful for self-reflection. Facilitating conditions such as timing and location, areas for improvement such as increased privacy, accessibility, incentivisation, and factors impacting data generated were further identified. Data collected covered both acute and chronic mental wellbeing and showed a positive relationship between surf and mental wellbeing. Further research is needed to confirm these findings in diverse populations, identify potential moderators, and confirm the validity of this tool and process. Being or practising a physical activity in blue space is increasingly shown as associated with an improvement in wellbeing ,2,3. SurThe mechanistic pathway through which surf therapy or any other physical activity could lead to an improvement in mental health remains unclear. A systematic review investigating the link between physical activity and cognitive and mental health in youth  did not There is no consensus yet about what constitutes mental wellbeing, how to best measure it and what theory should be the basis for policy making ,15. AlthHowever, measurement of mental wellbeing can be challenging. Linton and colleagues  found 99Thus, the primary objective of this study was to evaluate the usability and acceptability of an online data collection tool and process to evaluate mental wellbeing. A secondary objective was to assess the impact of the 6-week surf programme on participant acute and chronic mental wellbeing.The study participants are women, from different ethnic backgrounds, some living in deprived areas. Gender, ethnicity, age, economic status, and cultural backgrounds have been identified as moderators in the use of and access to technology and perceived skills ,29. TherThis study, conducted at The Wave, used an exploratory longitudinal cohort mixed-method design, whereby both qualitative and quantitative measurements were taken with the same study participants at defined timepoints to evaluate the impact of a surf programme . The expThis study used a purposive convenience sampling through a planned surf programme cohort at The Wave . The proThe programme consisted of weekly surf sessions for 6 weeks from May to July 2022. The first and second sessions were held two weeks apart due to public holidays. The participants were provided with all the equipment needed  upon their arrival.As privacy was requested by the participants, only female surf instructors were involved with this cohort, and the sessions were held in a section of the lake where the participants mixed as little as possible with the public. The sessions were run by two surf instructors and lasted approximately 1.5 h, including 30 min of coaching out of the water for training and safety purposes. In the water, the participants were within their depth (able to stand up in the water) and could decide to take the waves as they come, helped by the instructors as needed. In line with the activities of the community groups involved in this cohort, the goal of the surf programme was to improve wellbeing through outdoor physical activity. There were no set individual or group goals, so the participants could set their own pace and rest on the shore whenever needed. The research team were present during each session.p = 0.000)  here and everybody feeling as though they can contribute [was] really helpful”.(P11)Although most of the participants preferred to complete the survey on site straight after the session as shown by the quantitative data, the choice to complete it at home is still warranted to facilitate different types of participants.“when I did it at home, I really, I felt my answers […] more comfortable for me”.(P13)The level of perceived privacy was also evoked as a factor that would impact their answers.“you get no distraction or anything so you could be more open”, “If I had been in a booth, I would probably have taken more time thinking about what to say”.(P7)The idea of having a dedicated space on-site to complete the survey was welcomed. This space would need to provide privacy within a low-noise and low-traffic environment, especially to reduce the possibility of being overheard and made feel uncomfortable when recording videos.“Big thing for me would be to have a space with no one behind you. Not necessarily that people can hear you, but so there is no one around you”.(P7)Although the tool was perceived as easy to use, there were several suggestions to make it more accessible. To increase PEOU, the participants should be given the choice of media to use . Participants uncomfortable with expressing themselves, typing or spelling could be provided with word suggestions.“the word map, I wonder if you could turn that into like a live thing, so people could click on them? Like you say, if it’s prohibitive finding or spelling or whatever, you could choose”.(P9)Social influence was also identified as a determinant in the intent to use the tool, with a higher study participation acceptance during the presentation to the initial group, than when individuals joined the programme after the first week. Furthermore, observations throughout data collection identified that seeing participants completing the survey on-site seemed to remind and encourage others to do so as well. Providing privacy was seen as a facilitating condition that would also decrease the impact of social influence on the participants’ answers.Beyond this, the social norms were not perceived as having an impact on the data generated. The participants, when comfortable with the video media, were not self-conscious with their physical appearance on the video recordings: “there is no expectation you know, not just for Friday night…” (P9), “I actually quite like looking at it, cause I look like I am quite really overexcited, […] like I had the nicest time” (P9).Low perceived self-efficacy with digital tools was also identified as a factor in the PEOU:“for those who are not computer savvy” (P14), “Most of us are not [very technical]”.Implementing facilitating conditions would compensate for lower self-efficacy with digital tools, and therefore increase PEOU.Several additional factors influenced the data collected through the online tool. Although participants were given the option to complete the survey after leaving the site, this was not always possible due to their other commitments. One participant mentioned that “[they] were a bit like hurried up, because [they] had to all go” (P7) and that she “definitely had to give short answers as [she] wanted to quickly get through it” (P7), suggesting that the lack of time had an impact on the depth and breadth of answers provided. Although self-reflection was perceived as an opportunity, the nature of the survey and the questions could also trigger difficult feelings.“Yes, so it’s just, actually, sometimes those questions, although you could think, ‘Oh, they might trigger some kinds of people’, really, actually, they might help also, that people to understand, actually, this is a positive for me”.(P11)Events during the sessions, even minor, could also impact the answers to the survey. Two participants having experienced minor injuries skipped the video recording on this occasion, although they completed the rest of the survey without a noticeable impact on their answers.Lastly, participants were made aware of the study purpose, and expressed that they were already convinced of the positive impact of this surf programme on mental wellbeing. One participant recognized the existence of a research-effect bias and tried to mitigate this at her level.“I think sometimes I was a bit… not biased, but I was like I really want this research to work”.(P6)For these reasons, the answers provided to this survey may not be a full and accurate reflection of the participants’ mental wellbeing state.As expected, the videos captured the participants’ acute or ‘in the moment’ feelings. The participants mentioned changes that they perceived in their mental wellbeing, the physical impact of the surf session, or the events causing the feelings expressed.The feelings expressed are characteristic of positive affect in the hedonic wellbeing dimension, with quotes such as “I feel really happy”, “exhilarated”, “positive”, “energized”, “proud of myself”, “my mood has lifted”, “having that amazing buzz when you catch a wave”, “being in the water, it’s just sort of emptied the mind of anxiety”, “walking up the walk from the car park to the wave I started to get sort a lot of butterflies”, “[…] that buzz about doing something completely different out of the routine”, “watching all the women smile and have a laugh” mentioned. Where negative affect was expressed, it linked to physical sensations , the end of the programme , or events during the session .Participants also expressed feelings characteristic of eudaimonic wellbeing and linked to the experience. Feelings of individual or collective growth and sense of achievement, effort and social connectedness were identified, with participants “learn[ing] something new each time”, sharing “the unified goal of standing up on these boards”, experiencing an “amazing feeling to finally being able to feel confident to stand up”, and “seeing everyone’s faces just with the improvement and the confidence”.Participants expressed that they noticed a change in their mental wellbeing over time, either in the hedonic or the eudaimonic wellbeing domains. Participants expressed that they felt “a bit more mindful, more so than last week”, “much calmer”, “much better this week”, and reflected on “how heavy [their] thoughts have been in the last few weeks, and now [being] just absolutely buzzing”.Another emergent theme in these video recordings was the physical impact of the surf session. The participants described a positive impact, with participants feeling “a bit achy at the moment but really good”, “happy tired”, “exhausted in like a brilliant way that only this sort of exercise can give you”. Participants also felt like “[they]’ve worked out”, “[had] a lot of energy” and were “managing to do 23,000 steps that day”.The weather was mostly favourable during the sessions. The sunny weather was evoked as a reason for positive affect. The less favourable weather was evoked in the participants’ in a positive way: “even though it was raining, and the weather wasn’t that great it did not put anyone off”. The weather during the session did not seem to impact the participant’s acute feelings to a significant extent. Weather conditions are reported in the Out of the 100 words collected, 81% are evocative of hedonic wellbeing feelings such as happiness, excitement, satisfaction, relaxation, etc. 13% of the words are related to eudaimonic wellbeing, covering the constructs of growth, mindfulness, effort, and self-realization. The last 6% evoke the physical impact of the experience. The results are presented in The descriptive statistics are shown in The SWEMWBS mean score was 24.3 (SD 7.0) at the first measurement, and 28.5 (SD 4.2) at the last measurement.The purpose of this study was to assess the usability and acceptability of a novel online tool and data collection process to evaluate the impact of a 6-week surf programme on acute and chronic mental wellbeing. The tool was used on average 2.3 times out of the average of 4.1 sessions attended by the study participants and of the 4 measurement timepoints planned, a completion rate of 57.5%. This is lower than observed in ESM using mobile devices  . This isThe themes emerging from the focus groups show that the tool was perceived as quick and easy to use despite a relatively low perceived self-efficacy with digital tools, and useful as a means for self-reflection. The use of this tool could be further increased by enhancing its PEOU and PU and implementing further facilitating conditions, especially increasing privacy when completing the video recordings on site. It should be highlighted that providing word suggestions to increase accessibility, as suggested during the focus groups, would come with potential bias from a research point of view, as the participants may choose words that look more socially desirable but do not truly represent their views. Several factors impacting the data generated by the participants, such as lack of time, events during the sessions, potential research bias, or mental wellbeing state were further identified.The data generated through the video recordings, word association and SWEMWBS show that these are potential complementary measurement tools to assess the different dimensions and constructs of mental wellbeing. There was moderate redundancy between the themes emerging from the video recordings (mainly positive affect related to the experience related to hedonic wellbeing), and constructs measured via the SWEMWBS  . There wFuture research should seek to investigate the usability and acceptability in varied study populations and explore further the reasons of non-use to ensure that this tool and data collection process are adapted to measure mental wellbeing outcomes in the context of this surf programme. Additionally, there is a need to assess the validity of the measurement tools to evaluate the impact of surf on mental wellbeing in a population more representative of the targeted population, before assessing their experimental validity, as suggested in the proposed draft Edinburgh Framework for quantitative measurement research .p < 0.000) and emotional wellbeing (t(81) = −3.87, p < 0.000) measured by the validated Stirling Children’s Wellbeing Scale [Regarding the impact of the surf programme, the data generated through the video recordings support the causal relationship between the surf session and the acute feelings of mental wellbeing. This included feelings of positive affect, social connectedness, growth, and sense of achievement. The quantitative results also show an improvement in mental wellbeing with an increase of the SWEMWBS score from a mean of 24.3 (SD 7.0) to 28.5 (SD 4.2). These results are similar to those found in the existing surf therapy literature. Marshall and colleagues  found inng Scale ,66. Thesng Scale . The stuHowever, a positive relationship and causal inference between the surf programme and the improvement in mental wellbeing cannot be ascertained from the quantitative results in the present study due to non-random sampling, no pre-intervention measurement, no measurement of confounders such as physical activity and socio-economic levels affecting the internal and external validity . FurtherThe constructs of the TAM and the UTAUT emerged from the qualitative data. Further research is needed to establish the predictive values of these variables in this context. There is a lack of literature about intramethod mixing used in health intervention evaluations which prevents an in-depth discussion in relation to the existing knowledge base. However, for all the moderators studied in the TAM and in the context of ESM using mobile devices , PU was A strength of the current study resides in the fact that it was conducted in free-living conditions, which may provide better information and informed decisions on whether the tool has good reliability . The conIt is unavoidable that the interpretation of the data is influenced by the researcher’s background and beliefs as for any qualitative analysis . To aid However, several limitations affect the credibility and transferability of the results.The first focus group may have been too large and diverse to allow all participants to express their views, thereby limiting the breadth and depth of information collected . In addiAlthough the research team implemented different measures to increase credibility , one main limitation is the lack of investigator triangulation and member checking due to time constraints, whereby the coding was performed by one researcher and the transcripts and coding were not checked by the participants ,72.Furthermore, the credibility of the results related to the primary and secondary objectives may have been affected by the participants’ existing involvement in physical activity programs to improve mental wellbeing, and by the interviewer effect, where the participants relate to the interviewer’s background ,73. BothThere may have been an element of social desirability bias in the answers to the survey, especially in the video recordings where the participants were aware that the research team will watch them. Lastly, due to a purposive convenience sample, there may be little transferability of the findings outside of this study population. The PEOU, PU, BI, actual use and data generated are likely dependent on age and gender .Another limitation of this study is that the participants’ level of self-esteem was not measured. As some participants found their self-reflection in the video recordings uncomfortable, it is possible that self-esteem could have an impact on the breadth and depth of data generated via the video media.Furthermore, although the participants freely agreed to participate in the study, it is possible that not all participants have the willingness (be prepared to) or eagerness  to share their feelings .This study provides evidence that this intramethod tool and data collection process were perceived as easy to use and useful by a group of women and could be further improved for future use. This tool and data collection process allowed the acute and chronic mental wellbeing to be evaluated closer to the time of the experience, and captured data related to both the hedonic and eudaimonic dimensions of mental wellbeing. The data indicates that mental wellbeing may be supported by participating in the surf programme. However, further research is needed to confirm these findings in diverse populations, identify potential moderators impacting the use of the tool and the data generated, and confirm the validity of this tool and process for experimental use.


Two popular models of trustworthiness have garnered support over the years. One has postulated three aspects of trustworthiness as state-based antecedents to trust. Another has been interpreted to comprise two aspects of trustworthiness. Empirical data shows support for both models, and debate remains as to the theoretical and practical reasons researchers may adopt one model over the other. The present research aimed to consider this debate by investigating the factor structure of trustworthiness. Taking items from two scales commonly employed to assess trustworthiness, we leveraged structural equation modeling to explore which theoretical model is supported by the data in an organizational trust context. We considered an array of first-order, second-order, and bifactor models. The best-fitting model was a bifactor model comprising one general trustworthiness factor and ability, benevolence, and integrity grouping factors. This model was determined to be essentially unidimensional, though this is qualified by the finding that the grouping variables accounted for significant variance with for several organizational outcome criteria. These results suggest that respondents typically employ a general factor when responding to items assessing trustworthiness, and researchers may be better served treating the construct as unidimensional or engaging in scale parceling of their models to reflect this response tendency more accurately. However, the substantial variance accounted by the grouping variables in hierarchical regression suggest there may be contexts in which it would be acceptable to consider the theoretical factors of ability, benevolence, and integrity independent of general trustworthiness. Interpersonal trust is a fundamental component of relationships established both within the workplace  and in dDespite the prevalence of ability was defined as the perception that the trustee can perform one or more specific tasks. Perceived benevolence was described as the extent to which the trustee is perceived to have the trustor’s best interest in mind. Perceived integrity was defined as the extent to which the trustee values are perceived to be in alignment with the trustor’s values. Trust was defined as the extent to which the trustor is willing to be vulnerable to the trustee without being able to directly monitor their actions. Finally, actuating this willingness was defined as risk-taking in the relationship . Although research and theory indicate general tendencies, trust, and risk-taking in the relationship are important constructs, they are beyond the scope of the current study, which focuses on the theoretical composition and assessment of trustworthiness. Later empirical research demonstrated perceived ability, benevolence, and integrity have strong, positive intercorrelations with each other . In his structural model, McAllister demonstrated that both forms of trust were related, and that affect-based trust was positively associated with actor interaction frequency.Alternatively, the other popular theoretical conceptualization of trust is a two-factor model. worthiness as antecedents to one’s willingness to be vulnerable to another . A total of 66.32% participants identified as male, 33.05% identified as female, and 0.06% identified as a gender identity not listed on the survey.For determining sample size for our analyses, we relied on the rules of thumb described by worthiness if one leverages the nomenclature strongly disagree) to seven (strongly agree). The Cronbach’s alpha estimates from the current study are presented in Perceived trustworthiness in the participants’ supervisor was measured using 17 items from strongly disagree) to five (strongly agree). An example is “I would be willing to let my boss have complete control over my future in the company.”We used Mayer and Davis’ scale  to measunot at all) to seven (entirely). A sample item is “To what extent are you satisfied with the total contribution made by your supervisor?”We assessed participants’ ratings of their supervisor’s performance using McAllister’s four-item scale . Items wnot at all characteristic) to five (very characteristic). An example is “Volunteering to do things not formally required by the job.”We used Kelloway et al.’s scale  to assesnever) to five (very often). A sample item is “Covered up your mistakes.”We assessed participants’ tendency to engage in intentional behavior that was harmful to their organization’s interests using Kelloway et al.’s scale . This waYes/No criterion. The second item, “How strongly do you feel about leaving the organization within the next 12 months?” was assessed using a five-point scale ranging from one (very strongly) to five . The final item, “How likely is it that you will leave the organization in the next 12 months?” was assessed using a five-point scale ranging from one (very likely) to five .We used three items previously employed by post-hoc metrics employed for data cleaning included checking for missing data and correct responses to attention check items, as well as completion time for survey pages .We then created a three-factor model representing 2χ) fit index was utilized to reveal the degree of fit between nested models, as it has a testable significance assessment  were used in assessment of the relative fit of the models. Lastly, we extracted the estimated factor scores from the best fitting bifactor model and used the variables to predict the criterion variables of perceived supervisor performance, organizational citizenship behaviors, counterproductive work behaviors, and turnover intentions.We chose three changes in fit statistics to determine the relative fit of each model. First, the chi-square . First, we conducted CFAs on the trustworthiness scales separately, to ensure the proposed structures fit the data well before combining the scales. The 2χ(116) = 358.80, p < 0.001, CFI = 0.93, TLI = 0.91, RMSEA = 0.07, SRMR = 0.05. The 2χ(43) = 310.74, p < 0.001, CFI = 0.89, TLI = 0.85, RMSEA = 0.11, SRMR = 0.07. Modification indices indicated most of the affect-based items cross loaded onto the cognition-based trustworthiness factor. The last cognition-based item cross-loaded onto the affect-based trustworthiness factor, but it was deemed close enough to the established cutoffs. As such, all items from both scales were incorporated into subsequent analyses.The reliability estimates and intercorrelations for all variables are illustrated in Next, we fit our models to the data. Fit results of all models are illustrated in post-hoc because the measurement model was the best-fitting model with no higher order latent traits. This model fit the data poorly and significantly worse than Model 7; thus, it was rejected. Based on these analyses, a bifactor model that employs grouping factors based on Mayer et al.’s model of trustworthiness is most representative of the factor structure for these items. Results for this bifactor model with factor loadings are illustrated in Models 6 and 7, the bifactor models, both showed good fit and had improved fit over Model 0. Due to having the same degrees of freedom, Models 6 and 7 could not be compared directly, but Model 7 demonstrated a better chi-squared value and improved CFI. Therefore, Model 7 was retained as the final model. Model 8, an alternative bifactor model with all five factors from both scales , was considered r = 0.99, p < 0.01). Taken together, these results suggest that modeling the factor structure of the data is similar enough to a unidimensional model and that treating it as such is reasonable in practice.We conducted further analysis to ensure Model 7 was not overfitting the data, as this is a notable risk with bifactor models . To thisRegardless of the results for the bifactor indices, we examined the predictive validity of the general factor as well as the grouping factors of Model 7 in relation to established criteria related to trustworthiness. We performed a hierarchical regression with the first step including the general factor and the second step adding the grouping factors to determine if the grouping factors predicted any additional variance after controlling for the general factor. As illustrated in 2χ = 36.57, p < 0.001), but this may have been influenced by the sample size, as fitting larger models to even moderate sample sizes can positively bias chi-square results  and cognition- and affect-based trust. As such, using On this note, we have followed the speculation of many scientists before us in assuming that An additional limitation is that we did not include Tomlinson and colleagues’ model , 2020 inWe found the bifactor model, with a general trustworthiness factor and three orthogonal grouping factors, showed the best model fit for the ability, benevolence, and integrity factors described by The original contributions presented in the study are included in the article/This study was reviewed and approved by Air Force Research Laboratory Institutional Review Board for involving human participants. Written informed consent for participation was not required for this study in accordance with the national legislation and the institutional requirements.This project was initially conceived through a collaboration between ML, GA, and AC, with the initial idea proposed by GA. ML created the online study, conducted data collection, and completed data analysis. GA and AC drafted the Introduction and Discussion sections of the manuscript, while ML wrote the Methods and Results. ML led further drafting and editing of the entire manuscript with contributions from both GA and AC. All authors contributed to the article and approved the submitted version.This research was supported, in part, by the Air Force Research Laboratory (contract # FA-8650-16-D-6616) facilitating study design, implementation, data collection, analysis, and write up.The views expressed are those of the authors and do not necessarily reflect the official policy or position of the Department of the Air Force, the Department of Defense, or the U.S. government.ML is employed at General Dynamics Information Technology, Inc.The remaining authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Phelan-McDermid syndrome (PHMDS)/22q13.3 deletion syndrome is a rare genetic disorder associated with autism spectrum disorder (ASD), intellectual disability (ID), and bipolar disorder. While numerous cases have been reported describing successful pharmacological treatment of bipolar disorder in PHMDS, there is currently little guidance available on how to organize and execute such treatment. The aim of the current case study was to explore how pharmacological treatment of bipolar disorder in PHMDS may be organized and evaluated in an outpatient setting. Through a complex process of try and fail, including systematic evaluation of any change to the intervention and never implementing more than one change at the time, the patient gradually improved, regaining his communicative and adaptive skills. Four years passed from referral to this result was achieved. Organizing assessment and treatment as a collaborative effort involving specialized mental health professionals, professional caregivers and the patient’s family proved feasible. Many of the challenges present in assessment of psychiatric disorder in individuals with ASD and ID are likely to be present also in evaluation of treatment effects, particularly in disorders where symptoms occur in phases. The approach described in the current paper may contribute to reducing the impact of these challenges. While individuals with autism spectrum disorder (ASD) and intellectual disability (ID) are susceptible to the same set of psychiatric disorders as the population in general (Rosen 606232) is a rare, genetic disorder caused by a haploinsufficiency of the SHANK3 gene resulting from either a point mutation or an heterozygous deletion on the distal long arm of chromosome 22  or 22q13.3 deletion syndrome . The initial assessment included use of several conventional assessment tools: the Mini Neuropsychiatric Interview (MINI)  . MLPA  confirmed the deletion, with a proximal breakpoint between intron 3 and 9 in SHANK3, extending to and including ACR and RABL2B.At the time of the referral “Jonathan” was a young man in his late twenties. He had previous diagnoses of ASD and moderate ID. PHMDS was diagnosed during the present assessment using genome-wide array comparative genomic hybridization (aCGH) , showing an approximately 50et al. Jonathan showed developmental delay from an early age, walking and speaking late. He received a diagnosis of ID in preschool age, which was specified at moderate ID in his early teens. ASD was also diagnosed in his early teens. Jonathan developed complex verbal utterances around the age of six, but his verbal communication was characterised by a lack of perspective taking and a tendency to talk only about the things that interested him. His verbal language included frequent use of idiosyncrasies and professional care staff would often struggle to understand him until they had worked with him for some time. Jonathan had some adaptive skills, but needed help with most things in daily life, including personal hygiene and preparing meals. Jonathan had repetitive motor behaviours typically seen in ASD in childhood, including hand flapping. However, possibly atypical for PHMDS , which had to be reduced to a lower dosage (1 mg) due to Jonathan developing bradykinesia as a side effect. Jonathan also had digestive problems, including frequent obstipation, as well as pollen allergies.A few years prior to the current referral, Jonathan had undergone a mental health assessment concluding that he had a major depressive episode. During this episode, he displayed more negative mood and greater difficulties with emotion regulation, becoming more easily frustrated and irritable. He also had sleep difficulties, spoke less than usual and showed less initiative. During this episode, Jonathan was prescribed risperidone , they were organized twice yearly. Meetings included discussion of the status quo, evaluation of any change implemented since the last meeting, and agreeing on changes in the time leading up to the next meeting. The intellectual disability nurse from the outpatient clinic visited Jonathan several times between meetings, advising his care staff. There was also frequent contact between Jonathan’s family and his caregivers.The treatment plan was made in collaboration between Jonathan’s family, the professional caregivers in his home, and the specialized outpatient clinic. Assessment and treatment was organized as a series of meetings between Jonathan’s mother and sister, the two professional caregivers who had the main responsibility for his care, and a psychiatrist and an intellectual disability nurse from the specialized outpatient clinic. Initially, these meetings took place every three months. Later, when Jonathan’s condition had become more stable , Jonathan’s professional caregivers focused on adapting demands, tasks and activities to Jonathan’s current status and symptom load. This included Jonathan participating in his regular daily activities; going to a day centre, going for walks, car and bike rides, cleaning his yard, watching TV, getting visits from family, and participating in planning meals. Because Jonathan showed a reduced number of initiatives, his caregivers strove to facilitate Jonathan making choices and validating  was initiated in an attempt to alleviate psychotic symptoms shortly after referral.Jonathan was referred to the outpatient clinic in September. Due to his previous reaction to risperidone, aripripazole . It was introduced in a dosage of 50 mg and then gradually increased during the following autumn until a dosage of 150 mg x2 was reached.Following a review of the literature on treatment of bipolar disorder in PHMDS (e.g. Egger  mg was given as optional medication in instances of severe restlessness. The effect of this, however, was unclear. This use of risperidone was therefore discontinued in May. The dosage of Lamotrigine was increased in April, reaching 200mg given twice daily.Jonathan’s sleeping patterns seemed to be unaffected by the introduction of melatonin, and it was therefore discontinued in March. However, shortly after this, Jonathan developed more symptoms of restlessness and difficulties sleeping and apparent symptoms of hypomania – i.e. not as severe as previous manic episodes. Thus, in April Melatonin was re-instated and risperidone 1et al.  mg. While Jonathan’s symptoms were unaffected by this, he did not show any initial side-effects and the dosage was increased twice. At a total dosage of 15 mg olanzapine for two consecutive days, with the addition of 5 mg nitrazepam for sleep, Jonathan slept for more than 12 h and mania symptoms gradually subsided over a few days. Nitrazepam was discontinued after three full nights’ sleep.In June, Jonathan again developed symptoms of mania. He had several days with no sleep, continually wandering around his apartment, communicating little and seeming driven and intense. He showed severely disorganized behaviour, and had difficulties cooperating in basic tasks such as getting dressed, eating, going to the toilet and having a bath. Symptoms equalled an YMRS score of 44, which in typically developing individuals would indicate severe mania (Young  mg + 5 mg. Due to the severity of the manic episodes and the risk of new episodes, it was agreed to keep Jonathan’s pharmacological treatment, sleeping pattern and general surroundings as stable as possible during the following autumn and winter. Zopiklone was introduced in an attempt to further stabilise Jonathan’s sleeping patterns, but seemed to have little effect and was changed to alimemazine 10 mg, then 20 mg. Melatonin was discontinued. In a further attempt to ensure stable sleeping patterns, the entire olanzapine dosage was moved to the evening. At this point, Jonathan thus received 10 mg. olanzapine in the evenings, lamotrigine 200 mg x2, alimemazine 20 mg, and aripripazole 5 mg, as well as pantoprazole sodium for gastrointestinal reflux, and cetirizine for his allergies.In August Jonathan ate well, slept more or less as usual and again communicated more verbally. Unusually for him, however, he still seemed anxious when left alone. Olanzapine dosage was reduced to 5 mg to 2.5 mg in September and discontinued in December. It was also suspected that the use of alimemazine gave Jonathan slight hang-overs and might negatively affect his cognitive capacity. Alimemazine was therefore gradually discontinued without any apparent negative changes to Jonathan’s sleeping patterns. At the end of the year, Jonathan’s medication comprised olanzapine 10 mg, lamotrigine 200 mg x2, pantoprazole sodium and cetirizine.It was unclear whether the remaining dosage of aripripazole was helpful to Jonathan, but due to the apparently accentuated risk of developing mania during summer it was agreed not to make any changes to his medication during the spring. The crisis management plan was updated, but Jonathan did not develop mania symptoms in the summer; he slept well and did not show increased restlessness. Aripripazole dosage was reduced from 5 mg, resulting in Jonathan seeming slightly more socially present and aware of his surroundings, more frequently commenting on things he finds interesting, and making more conversational initiatives. However, a full evaluation of this change will not be possible until the summer of the next year.In case aripripazole had been working as a protective factor for mania, it was agreed not to make any further changes to Jonathan’s medication during spring. The crisis management plan was updated, but did not come into effect, as Jonathan did not develop symptoms of mania. In the autumn, olanzapine was further reduced to 7.5et al. et al. Four years after referral, Jonathan has regained most of his previous functioning and he has had two consecutive summers without symptoms of severe psychopathology. The return of adaptive and communicative functioning has been gradual, with no leaps associated with changes to his medication. In general, these changes seem to have followed a pattern where change of medication first seemed to affect symptom load, and improvement of adaptive and communicative skills followed gradually. The current psychopharmacological treatment, with concurrent use of one mood stabilizer and an atypical antipsychotic, is in line with previous descriptions of pharmacological treatment of bipolar disorder in PHMDS (Verhoeven Identification and treatment of bipolar disorder in PHMDS required a systematic, collaborative effort involving the patient’s family and his professional caregivers, as well as specialized mental health professionals. The organization of treatment as a series of meetings where pharmacological and other interventions were planned and evaluated worked well, easing communication between the involved parties. Noticeably, four years passed from referral until the involved parties considered treatment to have been successful.While the pharmacological treatment administered is typical for bipolar disorder, the manner in which it was organized and evaluated was not. Drugs were initiated at a lower dosage than usual, and dosages were increased more slowly. Even if individuals with PHMDS and others with ASD and ID are often unable to express how they subjectively experience changes to pharmacological treatment, they are likely to be affected by side effects or other experiences of discomfort similarly to others. Furthermore, even for positive effects, individuals with co-occurring ASD and ID may not be able to understand what is happening to them and positive treatment effects may thus be experienced as strange or frightening for these individuals. This suggests that it may be important to allow for enough time to pass before evaluating the effect of medication change, to avoid actual effects and side effects being confused with behavioural change associated with the subjective experience of the change itself.two years following his last manic episode. This further underlines the importance of making enough time before evaluating pharmacological intervention in mental health problems in PHMDS. The exception to this pattern was during episodes of mania, where effects of increased antipsychotics and sleep medication were more or less instant when reaching a certain dosage. Furthermore, the use of an atypical antipsychotic as prophylaxis to prevent development of new episodes of mania could not be adequately evaluated until the following summer had passed, as it was during summers the patient seemed to be at the highest risk. In the evaluation of changes to pharmacological treatment, it was also necessary to take account of contextual factors that might have influenced the patient’s behaviour. Together, this underlines that treatment of bipolar disorder in PHMDS is a time-consuming process, as well as the importance of using a systematic approach to symptom monitoring and evaluation of treatment effects.Effects of treatment in the current case were rarely immediately observable. Rather, these effects seemed to follow a pattern where there was an initial, gradual effect on symptom load, which was followed by gradual improvements to adaptive and communicative functioning. Improvements in communicative and adaptive functioning in the current patient have been gradual for et al. et al. et al. SHANK3 gene. Consistent with previous findings, the patient in the current study had a smaller deletion. While the pharmacological aspects of treatment of bipolar disorder has been described in previous studies (Rowland et al. et al. et al. et al. et al. et al. Recent findings indicate that bipolar disorder may be one of the most commonly co-occurring conditions in individuals with PHMDS (Kolevzon et al. et al. et al. et al. Some studies have described the bipolar disorder occurring in individuals with PHMDS as ‘atypical’ (Verhoeven Treatment was organized as a collaborative effort, with family, professional caregivers, and specialized mental health professionals participating on an equal footing. Regular meetings provided opportunities to evaluate prior interventions and discuss future ones, as well as the prioritizing the sequence of these. These discussions also ensured that all involved parties contributed with their specific knowledge of the patient, observations and experience of the status quo. The regularity of these meetings was seen both as aiding in communication and reducing the risk of misunderstandings and conflict, by everyone involved meeting face to face and neither family nor professional caregivers having to rely on second-hand information from the specialized mental health professionals. This collaboration is likely to have been particularly important in attempts to make sense of the changes to the patient’s stereotypic behaviour and his insistence on sameness.et al. et al. ongoing risk of misinterpretation involving either diagnostic overshadowing, or wrongly attributing features of ASD or ID to the co-occurring psychiatric disorder. This suggests that measures undertaken to overcome these risks during assessment need to be applied throughout the entire course of treatment in bipolar disorder in ASD and ID. Continually striving to unite the views of family members, professional caregivers, and mental health professionals, may be one approach to reduce these risks.In our view, the current organization served as an important means to overcome some of the inherent challenges to evaluating the effects of psychopharmacological treatment in individuals with ASD and ID, and may therefore serve as a template for other mental health professionals facing these challenges. This may be particularly relevant in cases involving psychiatric disorders where symptoms occur in phases, because challenges inherent to both diagnostic assessment and evaluation of treatment in individuals with ASD and ID (Rosen