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Patient-Reported Burden of a Neuroendocrine Tumor (NET) Diagnosis: Results From the First Global Survey of Patients With NETs

Patient-Reported Burden of a Neuroendocrine Tumor (NET) Diagnosis: Results From the First Global... abstract original report Patient-Reported Burden of a Neuroendocrine Tumor (NET) Diagnosis: Results From the First Global Survey of Patients With NETs Purpose Despite the considerable impact of neuroendocrine tumors (NETs) on patients’ daily lives, the journey of the patient with a NET has rarely been documented, with published data to date being limited to small qualitative studies. NETs are heterogeneous malignancies with nonspecific symptomology, leading to extensive health care use and diagnostic delays that affect survival. A large, international patient survey was conducted to increase understanding of the experience of the patient with a NET and identify unmet needs, with the aim of improving disease awareness and care worldwide. Methods An anonymous, self-reported survey was conducted (online or on paper) from February to May Simron Singh 2014, recruiting patients with NETs from > 12 countries as a collaboration between the International Dan Granberg Neuroendocrine Cancer Alliance and Novartis Pharmaceuticals. Survey questions captured information on sociodemographics, clinical characteristics, NET diagnostic experience, disease impact/management, Edward Wolin interaction with medical teams, NET knowledge/awareness, and sources of information. This article Richard Warner reports the most relevant findings on patient experience with NETs and the impact of NETs on health care Maia Sissons system resources. Teodora Kolarova Results A total of 1,928 patients with NETs participated. A NET diagnosis had a substantially negative Grace Goldstein impact on patients’ personal and work lives. Patients reported delayed diagnosis and extensive NET-related Marianne Pavel health care resource use. Patients desired improvement in many aspects of NET care, including availability of a wider range of NET-specific treatment options, better access to NET experts or specialist centers, and a Kjell Oberg more knowledgeable, better-coordinated/-aligned NET medical team. John Leyden Conclusion This global patient-reported survey demonstrates the considerable burden of NETs with regard to symptoms, work and daily life, and health care resource use, and highlights considerable unmet needs. Author affiliations appear at the end of this article. Further intervention is required to improve the patient experience among those with NETs. Supported by Novartis J Glob Oncol 3. © 2016 by American Society of Clinical Oncology Licensed under the Creative Commons Attribution 4.0 License Pharmaceuticals and conducted as an equal collaboration between the 4,5,7 International Neuroendocrine INTRODUCTION toward increasing incidence is evident. This Cancer Alliance and Novartis. increasing global incidence, together with the Neuroendocrine tumors (NETs) are a diverse Medical editorial writing considerable number of people living with NETs assistance was provided by group of malignancies that arise from neuroendo- (estimated prevalence: 35 per 100,000), high- 1-3 ApotheCom Associates crine cells throughout the body. Although NETs (Yardley, PA) and was funded lights the importance of improving awareness of are uncommon, incidence rates continue to in- by Novartis. this disease, increasing accurate diagnosis, and crease, partly because of greater awareness of the Authors’ disclosures of 3,4 optimizing disease management. disease and increased accuracy of diagnosis. potential conflicts of interest and contributions are found at The incidence of NETs in the United States has NETs are generally graded on the basis of mitotic the end of this article. increased five-fold over the past 30 years, with five count and proliferative index. NETs can be di- Corresponding author: Simron out of every 100,000 people being diagnosed with vided into functional (clinically symptomatic, typ- Singh, MD, MPH, FRCP(C), NETs annually. Although NET incidence varies ically characterized by the hormones they secrete) Odette Cancer Center—Sunnybrook across geographic regions (higher in the Nether- and nonfunctional (silent/nonsecretory, free of Hospital, 2075 Bayview Ave, lands [4.9 per 100,000] and Canada [5.86 per hormone-related symptoms, and generally iden- Room T2-047, Toronto, ON 5 6 1,10 100,000] ; lower in Japan [2.10 per 100,000] tified incidentally) tumors, although current M4N 3M5, Canada; e-mail: simron.singh@sunnybrook.ca. and Taiwan [1.51 per 100,000] ), an overall trend evidence indicates that diagnosis and treatment 43 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology © 2016 by American Society of Clinical Oncology Licensed under the Creative Commons Attribution 4.0 License strategies should follow the same principles re- in support of people living with NETs, collaborated gardless of functional status. The symptoms with Novartis Pharmaceuticals to conduct the first associated with NETs may be nonspecific or ab- global survey to gather data about the experience 1,2,12 sent until more advanced stages, often lead- of patients with NETs from multiple countries. To 2,12 ing to delays in diagnosis. Indeed, NET-related our knowledge, this has been the first endeavor to symptoms may persist for long periods (median, collect information on the patient experience with NETs. The aim of this patient-reported survey was 9.2 years) before an accurate diagnosis is made, to increase understanding of the views and expe- thus potentially placing a substantial burden on riences of patients with NETs, including their both the patient and the health care system. needs and challenges with regard to diagnosis, Because of the nonspecific nature of symptoms, management of NETs, interactions with medical there seems to be no clear pathway of care for teams, knowledge and awareness levels of the patients with NETs; patients may be seen by disease, and information sources. multiple specialists and undergo extensive and repetitive testing, leading to varying and potentially The findings of this survey expand the knowledge conflicting treatment recommendations and con- base regarding NETs from a patient perspective, tributing to delays in an accurate diagnosis. The with the goal of improving disease awareness resulting psychological and emotional burden and patient care. Collecting and reporting such may at least partially contribute to the reported patient-centered data, in addition to directing im- worse health-related quality of life (HRQoL) out- provements in patient experience and care prac- comes among patients with NETs compared with tice, can have overarching beneficial effects on the 14 18 the general population. health care system as a whole. Despite the considerable impact of this disease on In this publication, we present findings most rel- patients’ daily lives, the journey of the patient with evant to the impact of NETs on patients’ daily lives a NET has rarely been documented, with pub- and on the health care system, and highlight un- lished data being limited to small qualitative met needs from the patient perspective. 15,16 studies. These studies highlight the lack METHODS of a standard care pathway for patients with NETs Study Design and Participants and identify a number of potential challenges, including difficulty in achieving a NET diagnosis, Patients were primarily recruited online via the use limited access to information about NETs and to of flyers, Web site postings, e-mails, and social media NET-specific treatment centers, and inadequate channels of the INCA member organizations/patient ongoing support. advocacy groups. For transparency, the Novartis logo was clearly displayed on all survey materials to Patient experience and patient-reported out- indicate that Novartis was involved in the survey, comes are becoming increasingly important end and all materials stated that the sponsoring com- points in oncology. The Institute for Healthcare pany or the INCA patient group partners may use Improvement has introduced an initiative, called the data for disease awareness purposes. the Triple Aim, to help address insufficiencies in health care and thus optimize health care system Survey Details performance. These linked goals are to improve Survey domains and key questions were initially the patient experience of care, improve the health generated at a roundtable meeting of the INCA of populations, and reduce the per capita cost of leadership held during the 10th annual Euro- health care. It is believed that these goals must pean Neuroendocrine Tumor Society confer- be addressed simultaneously to achieve health ence in Barcelona, Spain, in March 2013. Hall & care reform, and one facet of this effort is to em- Partners, a research organization, was used to power individuals and their families in the context construct a detailed survey to assess patients’ ex- of their health care. As oncology care moves periences with NETs on the basis of the meeting toward a more patient-centered care focus, it is discussions and a review of the NET literature. important that clinicians understand the experi- Fourteen NET patient health consumer groups ence of NETs from the perspective of the patient. within INCA had direct input into question devel- The International Neuroendocrine Cancer Alliance opment, and the final questionnaire was reviewed (INCA) consists of a network of 18 independent and edited by all members of INCA and by Novartis charitable organizations and patient groups for between May and October 2013. This anonymous individuals with NETs from 15 countries around survey was developed to be primarily conducted the world. INCA, which aims to be the global voice online, with an approximate time for completion of 44 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Table 1 – Summary of Patient-Reported Sociodemographics and Clinical Characteristics 25 minutes on the basis of length. Paper surveys were also generated and distributed via patient Patient Sociodemographics and Clinical Patients advocacy groups and health care professionals Characteristics (N = 1,928) (HCPs) to reach patients without Internet access. Patient-reported sociodemographics Both the online and paper surveys were available Region, % in eight languages: Bulgarian, Dutch, English, Americas 48 French, German, Japanese, Norwegian, and sim- Europe 40 plified Chinese. Hall & Partners fielded the survey from February to May 2014 and analyzed the Asia 5 results. Oceania 7 Age (mean), years 56.8 Patient-reported data on sociodemographic infor- mation (eg, age, sex, educational background), Age distribution (years), % clinical characteristics (eg, NET type, years since ,40 8 diagnosis, functional status, tumor grade), burden 40-49 17 of NETs on daily life and work, desired improve- 50-59 32 ments, and information sources were collected. In this survey, data were purposefully self-reported to 60-69 31 maximally reflect the patient’s voice. The ques- 70+ 12 tionnaire covered patients’ overall awareness of Female, % 64 NETs, the diagnostic experience throughout their Educational level, % disease course, burden of disease, ongoing man- Not university educated 55 agement, interactions with health care providers, Bachelor degree or higher 45 experience with and access to NET treatments, and resources used for education about NETs. Ques- Caregiver, % tions were categorized as follows: initial screening, Yes 65 patient’s current status, diagnosis, quality of life, No 35 NET management, NET treatment, NET education, Work status, % and demographics (the full survey appears in the Employed Data Supplement). With the exception of certain questions designed to gather numerical informa- Full-time 25 tion, survey questions were closed ended; partic- Part-time 8 ipants were provided options from which to choose. Self-employed 6 When patients were asked to rate a particular Retired 31 parameter,responsesincludedthegradeddescrip- Medical disability 18 tors not at all, somewhat, very, and extremely, and Not employed/homemaker/student 11 when asked the degree to which they (dis)agreed with a particular statement, the descriptors strongly Patient-reported clinical characteristics and somewhat were added to response choices. NET type, % Gastrointestinal 54 Data Analyses Pancreas 22 Global data were analyzed using the MERLIN Lung 12 (Merlinco, London, UK) survey software package. Thymus 1 Survey responses were summarized using de- scriptive statistics, including means, medians, Other 8 and percentages. Statistical significance is shown Unknown 5 at a 95% CI level (P , .05). In some instances, Tumor grade, %* responses are presented as the top two responses (eg, somewhat agree/strongly agree; a moderate amount/a lot). RESULTS Unknown/do not remember 35 Patient Characteristics (Continued on following page) A total of 1,928 patients with NETs participated in the survey, with the majority being recruited through patient advocacy groups (37%) and 45 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Table 1 – Summary of Patient-Reported Sociodemographics and Clinical Characteristics NETs (22%) and lung NETs (12%). Of the patients (Continued) who knew their functional status (n = 1,551), 55% reported having functional disease. Of those who Patient Sociodemographics and Clinical Patients Characteristics (N = 1,928) knew their tumor grade (n = 1,242), 91% reported having grade 1 or 2 tumors; 35% of all patients either Functional status, %† did not know or had not been told their tumor grade. Functional 44 Nonfunctional 15 Diagnostic Delays and Use of Health Care Asymptomatic 21 Resources Unknown/do not remember 19 Mean patient-reported time from first symptom Time since diagnosis (mean), years 5.2 onset to diagnosis was 52 months; 29% of patients required > 5 years for a NET diagnosis, and 58% Time since diagnosis (years), % of patients had metastases at the time of diagnosis. ,559 Patients saw a mean of 6.2 HCPs across a mean of 5-9 27 11.8 doctor visits in the period of first onset of 10-14 8 symptoms before receiving their NET diagnosis 15+ 6 (Figs 1A and 1B). Metastatic disease at the time of 58 On average, approximately three HCPs were in- diagnosis, % volved in the ongoing management of patients with Surgical removal of primary tumor since 70 NETs, with oncologists/hematologists (70%) and diagnosis, % general practitioners (58%) seen most often after diagnosis. Annually, patients reported having a Abbreviations: Ki-67, protein encoded by the MKI67 gene; NET, neuroendocrine tumor. *Grade 1: NETs are relatively slow growing, Ki-67 index < 2%; grade 2: NETs have a less predictable, mean of 5.5 NET-related tests, with 28% receiving moderately aggressive course, Ki-67 index 3%–20%; grade 3: NETs can be highly aggressive, Ki-67 tests six or more times per year after diagnosis. index . 20%. †Functional NETs: produce symptoms caused by the secretions of hormones (eg, flushing, diarrhea, Impact of NETs on Patients’ Personal and Work wheezing, cramping); nonfunctional NETs: do not secrete hormones, but they may cause symptoms Lives caused by the tumors’ growth (eg, pain, intestinal blockage, bleeding). Living with NETs seemed to have a considerable online sources (51%). These patients were re- impact on patients’ personal and work lives. Al- cruited from . 12 countries in the Americas though on a scale of 1 to 6 (1 = very poor health, (United States [n = 758], Canada [n = 164], and 6 = excellent health), patients reported a mean other countries in North, Central, and South Amer- health score of 4, a relatively large proportion ica [n = 6]), Asia (Japan [n = 81], Singapore, and (37%) reported very poor to fair health. Patients other countries in Asia [n = 18]), Europe (Germany reported experiencing numerous ongoing NET- [n = 311], United Kingdom [n = 156], France [n = related symptoms, such as general fatigue/muscle 117], Norway [n = 54], Belgium [n = 29], Bulgaria fatigue/weakness (56%), diarrhea (48%), and [n = 18], and other countries in Europe [n = 78]), abdominal pain or cramping (41%; Fig 2). Many and Oceania (n = 138). The exact number of of these symptoms occurred on a daily basis. countries could not be determined because some Most survey participants (71%) reported a mod- respondents identified the general region in which erate to substantial negative impact of NETs on they lived rather than a specific country. Overall, their daily life, including overall energy levels the majority of respondents were from North Amer- (70%), finances (50%), and the ability to perform ica (n = 922; 48%) and Europe (n = 763; 40%). everyday household chores (45%) or care for family (39%; Fig 3A). A large proportion of pa- Patient-reported sociodemographic parameters tients believed that living with NETs substantially and clinical characteristics are summarized in affected (a moderate amount/a lot) their emo- Table 1. The mean age was 56.8 years, and 64% tional health (60%), the emotional health of were women. Close to half of the patients (45%) family/friends (48%), and their relationships with had a bachelor’s degree or higher, and two-thirds family/friends (34%/34%). had a caregiver (ie, a close family member or friend to help them manage day-to-day NET-associated Most patients (92%) reported making one or more activities). Only 39% of patients were currently work- lifestyle changes as a result of NETs, including ing (full-/part-time employment or self-employed) dietary changes (58%), increased time/money and 18% had a self-identified medical disability spent on traveling to or from their medical appoint- (not further specified). GI NETs were the most ments (52%/51%), and seeing a therapist for common NET type (54%), followed by pancreatic emotional aspects of the disease (20%; Fig 3B). 46 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Fig 1 – (A)Thenumberofhealthcare Mean number of HCPs seen before a NET diagnosis: 6.2 professionals (HCPs) seen before receiving a neuroendocrine tumor (NET) diagnosis. Base 12 12 population: all respondents (N = 1,928). Question: Approximately how many HCPs (including all doctors, 1 2 345 6–9 10–19 20+ Don't specialists, and nurses) were know/can't involved in your diagnosis of remember a NET? Please consider all those you saw from the time Number of HCPs you first experienced symptoms to the time you Mean number of health care visits made before a NET diagnosis: 11.8 received the diagnosis of a NET. (B) The number of healthcare visitsmade before receiving a neuroendocrine tumor (NET) diagnosis. Base 12 population: all respondents (N = 1,928). Question: Approximately how many different visits to HCPs 1–2 3–4 5–6 7–8 9–10 11–15 16+ Don't (including all doctors, know/can't specialists, and nurses) did remember you have to make? Please Number of Health Care Visits consider all those you saw from thetimeyou first experienced symptoms to the time you received the NETs had a significant impact on work life. Almost 439.35] or 182 km [median, 40; range, 1 to diagnosis of a NET. half of patients (49%) reported taking days off from 9,656; standard deviation, 503.38]). work, 27% asked their employer to make accom- modations, and 24% reduced their work hours DISCUSSION because of NETs (Fig 3C). Of the 440 patients not To our knowledge, this is the first large, global currently employed or unable to work because of study that attempts to characterize the impact medical disability, 82% reported having to stop of NETs from the patient perspective. To date, working as a direct result of their NETs. there is a scarcity of published data on the experiences and perspectives of patients with Access Issues NETs, which may be in part due to the rarity of this cancer as well as difficulties in collecting Despite the high frequency of physician visits patient-reported data from an international per- and medical tests, survey findings revealed nu- spective. For this survey, the extensive use of merous unmet needs with regard to NET man- online social media sources through local/ agement. The majority of patients desired better regional advocacy groups enabled recruitment availability of a wider range of NET-specific of a large number of patients with NETs in a treatment options (60%) and access to NET relatively short period of time. experts or a NET specialist center (56%). Close to half believed that more knowledgeable NET Findings from this survey brought to light the medical providers (47%) and a better coordinated/ substantial impact of NETs on patients’ daily aligned NET medical team (45%) would improve lives, including their physical and emotional NET care (Fig 4). In addition, many patients well-being, financial stressors, and impaired lacked access to treatments they know exist interactions with family and friends. The NET- (Table 2). In all cases, the percentage of patients related burden resulted in lifestyle changes with knowledge of particular treatment options and substantial financial strain and had a neg- was greater than the percentage of patients with ative impact on patients’ ability to work. Although access to those options. Patients also reported validated HRQoL assessment tools were not having to travel an extended distance to see their used in this survey, the findings are consistent NET medical providers (mean distance, 126 miles with previously conducted surveys in Norway and [median, 25; range, 1 to 9,942; standard deviation, the United States, which reported worse HRQoL 47 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Patients (%) Patients (%) Fig 2 – Neuroendocrine tumor General fatigue, muscle fatigue, weakness (n = 1,088) 56 (NET) symptoms Diarrhea (n = 924) 41 48 experienced, often on Abdominal pain or cramping (n = 783) 41 41 a daily basis. Base population: all respondents Skin reactions: flushing, rash, redness, thinning (n = 706) 46 37 (n values as indicated in Sweating, headaches/dizziness, nausea (n = 589) 41 31 figure). Questions: Which of Anxiety, palpitations (n = 492) 35 26 the following symptoms, if Breathlessness/wheezing (n = 471) 52 24 any, do you suffer from as a result of your NET? Select Changes in blood pressure (n = 428) 45 22 all that apply. How Heartburn/reflux (n = 401) 21 frequently do you suffer Weight loss (n = 399) 38 21 from each of the following symptomsas a result of your Memory loss and/or confusion (n = 373) 53 19 NET? Steatorrhea (increase in fat content in stools) (n = 368) 46 19 Weight gain; large, round face; excessive fat torso (n = 289) 75 15 Percentage of the total population Vision problems (n = 226) 67 12 of patients experiencing each symptom. Osteoporosis (n = 136) 82 7 Within each subgroup of patients Rectal bleeding (n = 96) who experienced a particular symptom, the percentage who Jaundice (n = 27) experienced that symptom on a constant or daily basis. Other (n = 125) 0 10203040506070 Patients (%) scores in patients with NETs versus the general outcome if they are well informed and assertive 19,20 18,22 population, as expected. A previous US sur- regarding their treatment. Since 2001, vey of cancer survivors also reported poorer when a report by the Institute of Medicine pro- posed that patient-centeredness should be one HRQoL in this group compared with adults with- of the aims of the US health care system, out cancer. increasing efforts have been made to provide This survey also highlighted numerous unmet more patient-centered care, in part by custom- needs for patients with NETs, including the desire izing that care according to the individual pa- for better access to NET-specific medical teams tient’s needs and values and placing patients in (ie, NET experts and specialist centers), as well as control of their own care. The findings from NET-specific treatments and information. A need this survey emphasize the need for more stan- for increased awareness of NETs among the med- dardized pathways of NET patient care that ical community was also demonstrated, reflected address ongoing issues and challenges identi- by a desire for patients to have better access to fied by patients. Patients often reported a sub- more knowledgeable NET medical providers as stantial delay in diagnosis and extensive use of well as a better coordinated NET medical team. health care resources (involving large numbers Indeed, the finding that 35% of patients did not of HCP visits and numerous tests) and also know or had not been told their tumor grade may voiced the desire for better coordinated care reflect a lack of communication between patients from medical providers. Lack of these elements and physicians regarding NETs. These findings potentially contributes to excessive use of health are aligned with results of previous survey-based care resources, affecting both patients and the oncology studies, particularly with regard to ac- health care system. This scenario is the inverse of cess to information about NETs from physicians, the triple aim promoted by the Institute for Health- NET-specific treatment centers, and disease- care Improvement report on patient-centered 15,16 17,23 specific support. Clearly, there is a need for care. Success with patient-centered ap- better coordination and accessibility of care for proaches to NET care (improved outcomes patients with NETs. and quality of care) are increasingly being demonstrated in multidisciplinary specialist It has been established that not only are pa- centers. tients more likely to be compliant with treatment if they are actively involved in their own health Our survey has several potential limitations that care, but also they are more likely to have a better should be taken into consideration, including 48 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Fig 3 – (A) Negative impact of A My overall energy levels 70 neuroendocrine tumors My emotional health 60 (NETs) on daily life. Base: My ability to participate in leisure activities all respondents (N = 1,928). Question: How My attitude towards daily life much has each of the My social life following areas of your life been negatively affected, if 51 My ability to travel at all, by your NET? Top two My finances 50 box scores are shown (a The emotional health of those close to me moderate amount/a lot). (B) (family, friends, etc.) Lifestyle changes caused My ability to perform everyday household chores by having a NET. Base: all My ability to care for my family 39 respondents (N = 1,928). Question: Since you were My relationship with my spouse/partner 35 diagnosed with your NET, My relationships with my friends 34 have you had to make any of My relationships with my family the following changes? (other than a spouse/partner) Please select all that apply. My ability to perform my job 16 (C) Impact of NETs on work 0 10203040506070 life. *For example, flexible work schedule, work from Patients (%) home, adaptive devices, opportunities for rest. Base Make dietary changes 58 population: respondents Spend a lot of time on travel to/from who are working full time/ medical appointments part time or are self- Increase spending on travel for employed (n = 741). medical appointments Question: Has your NET Stop or cut back on physical activities impacted you at work in any Stop or cut back on social life of the following ways? Make exercise changes 43 Please select all that apply. Cut back on leisure purchases HCP, health care (vacations, etc.) professional. Increase spending on nutritional products See a therapist for emotional aspects of disease Stop or cut back on caregiving for family/friend Stop or cut back on childcare 9 Other 5 0 10203040506070 Patients (%) I have had to take days off from work I have had to ask my employer to make accommodations* I have had to work reduced hours I have had to stop working altogether for a period of time Colleagues have looked at me differently I have had to take a less demanding job within the company I have had to switch to a new job 6 Seek disability 6 The HCPs who treat my cancer have advised me to stop working Other None of the above 23 0 10203040506070 Patients (%) 49 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Fig 4 – Patients’ beliefs regarding A wider range of NET treatment options 60 improvements that would Better access to NET experts/medical help with ongoing centers that specialize in NET neuroendocrine tumor More information about/more opportunity to participate in NET clinical trials (NET) management. Base population: all respondents More knowledgable NET medical providers 47 (N = 1,928). Question: More treatments available in my country Which of the following that I see in other countries would help with the ongoing A better coordinated/aligned team of NET medical providers management of your NET? More information brochures from my NET medical providers Other/none of the above 11 0 10203040506070 Patients (%) recall bias, given the fact that findings are solely These limitations notwithstanding, a key strength based on patient responses to closed-ended of this global patient-reported survey is that find- questions and not confirmed by medical re- ings directly reflect patients’ perspectives on cords. In addition, the survey was qualitative NETs. Having an understanding of how patients in nature; standardized, validated HRQoL as- experience their disease is unique and of signif- sessments were not used. Finally, most patients icant value, because it directly aligns with the new were recruited online (Web sites, 31%; social movement in health care toward patient-centered 17,23 media, 20%) and through patient advocacy care. A concerted effort from clinicians and groups (37%). Respondents may have been HCPs at various levels is needed to effectively more likely to be highly engaged, motivated interpret the data and successfully implement care seekers. There was also a preponderance appropriate care processes to bring these im- of female respondents, and nearly half of the provements in the quality of patient-centered care participants (45%; Table 1) had higher educa- to fruition. Regional and country-specific ana- tion levels. As such, the survey population may lyses of these survey data have also been con- not be fully representative of a heterogeneous ducted; INCA is currently working with their local NET population. However, comparison of the organizations to use this information to help better disease characteristics (site of origin, func- inform NET patient care according to geographic tional status, and grade/differentiation) of the region. patient population that participated in this sur- vey to those included in pivotal phase 3 clini- In conclusion, this large, global patient survey demonstrates the considerable burden of NETs cal trials on NETs revealed similarities (Data 26-32 Supplement). with respect to symptoms, impact on work and Table 2 – Availability of Neuroendocrine Tumor Treatments Treatments I Have Received/Am Currently Treatments I Have Heard Of* Treatments I Have Access To† Receiving‡ Surgery 90†‡ 76‡ 67 Chemotherapy 75†‡ 38‡ 22 Drug therapy other than chemotherapy 79†‡ 56‡ 47 PRRT 74†‡ 28‡ 17 Interventional radiology 65†‡ 30‡ 16 Observation 74†‡ 53‡ 44 Other/none of the above 13†‡ 77 NOTE. Data presented as %. Base: all respondents (N = 1,928). Question: This is a list of available NET treatments. Please select those you have heard of, those you have access to (meaning they are available to you), and those you have received/are currently receiving for the treatment of your NET. Abbreviation: NET, neuroendocrine tumor; PRRT, peptide receptor radionuclide therapy. *†‡Symbols appearing next to a given value indicate significant differences, P, .05, between the patient group specified in that column compared with those specified in the other columns. 50 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology daily life, and health care resource use and and insights regarding best practices for disease highlights unmet needs and ongoing challenges. management. Taken together, this survey provides valuable findings that maybeusedinfutureresearchto DOI: 10.1200/JGO.2015.002980 gain a better understanding of this rare disease Published online on jgo.org on June 8, 2016. AUTHOR CONTRIBUTIONS Grace Goldstein COO of The Carcinoid Cancer Foundation, Inc.; reports that Conception and design: Maia Sissons, Teodora Kolarova, the Foundation has received grants from Novartis, Ipsen, Grace Goldstein, John Leyden Advanced Accelerator Applications, and Lexicon Collection and assembly of data: Maia Sissons, Pharmaceuticals Teodora Kolarova, Grace Goldstein, John Leyden Travel, Accommodations, Expenses: Novartis Data analysis and interpretation: All authors Manuscript writing: All authors Marianne Pavel Final approval of manuscript: All authors Honoraria: Ipsen, Lexicon Pharmaceuticals, Novartis, Pfizer AUTHORS’ DISCLOSURES OF Consulting or Advisory Role: Ipsen, Lexicon Pharmaceuticals, POTENTIAL CONFLICTS OF INTEREST Novartis, Pfizer The following represents disclosure information provided by Research Funding: Novartis authors of this manuscript. All relationships are considered Travel, Accommodations, Expenses: Ipsen, Novartis compensated. Relationships are self-held unless noted. I = Kjell Oberg Immediate Family Member, Inst = My Institution. Relation- No relationship to disclose ships may not relate to the subject matter of this manuscript. For more information about ASCO’s conflict of interest policy, John Leyden please refer to www.asco.org/rwc or ascopubs.org/jco/site/ifc. President of the Unicorn Foundation; reports that the Simron Singh Foundation has received support from Novartis Australia, Honoraria: Novartis, Pfizer, Ipsen Pfizer Australia, and Ipsen Travel, Accommodations, Expenses: Novartis, Pfizer, Ipsen Dan Granberg ACKNOWLEDGMENT We thank the patients who participated in this survey No relationship to disclose and our patient advocacy partners throughout the world Edward Wolin who contributed to this project, including The Unicorn Honoraria: Novartis, Ipsen, Celgene, Advanced Accelerator Foundation (Australia), vzw NET & MEN Kanker Applications (Belgium), The Association of Cancer Patients and Consulting or Advisory Role: Novartis, Ipsen, Celgene, Friends (APOZ, Bulgaria), Carcinoid-Neuroendocrine Advanced Accelerator Applications Tumour Society (CNETS, Canada), Association des Patients Porteurs de Tumeurs Endocrines Diverses (APTED, Richard Warner France), Netzwerk Neuroendokrine Tumoren (Germany), Honoraria: Novartis, Pfizer, Lexicon Pharmaceuticals PanCAN (Japan), Unicorn Foundation NZ (New Zealand), Consulting or Advisory Role: Novartis, Pfizer, Lexicon CarciNor (Norway), Carcinoid & Neuroendocrine Tumor Pharmaceuticals Society (CNETS, Singapore), NET Patient Foundation Research Funding: Lexicon Pharmaceuticals and The Association for Multiple Endocrine Neoplasia Disorders (AMEND, United Kingdom), and The Maia Sissons Carcinoid Cancer Foundation and Caring for Carcinoid Research Funding: Novartis, Ipsen, Pfizer Foundation (United States). Hall & Partners, a Teodora Kolarova research organization, fielded the survey and analyzed the No relationship to disclose results. Affiliations Simron Singh, University of Toronto, Toronto, Ontario, Canada; Dan Granberg and Kjell Oberg, Uppsala University Hospital, Uppsala, Sweden; Edward Wolin, Montefiore Einstein Center for Cancer Care, Bronx, NY; Richard Warner, Mount Sinai School of Medicine,NewYork,NY;MaiaSissons,NETPatientFoundation,HockleyHeath,UnitedKingdom;TeodoraKolarova,APOZ&Friends, Sofia, Bulgaria; Grace Goldstein, The Carcinoid Cancer Foundation, Inc., White Plains, NY; Marianne Pavel,ChariteU ´ ni- versitats ¨ medizin Berlin, Berlin, Germany; and John Leyden, The Unicorn Foundation, Mosman, New South Wales, Australia. REFERENCES 1. Oberg KE: Gastrointestinal neuroendocrine tumors. Ann Oncol 21:vii72-vii80, 2010 (suppl 7) 2. Vinik AI, Woltering EA, Warner RR, et al: NANETS consensus guidelines for the diagnosis of neuroendocrine tumor. Pancreas 39:713-734, 2010 51 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology 3. Yao JC, Hassan M, Phan A, et al: One hundred years after “carcinoid”: Epidemiology of and prognostic factors for neuroendocrine tumors in 35,825 cases in the United States. J Clin Oncol 26:3063-3072, 2008 4. Korse CM, Taal BG, van Velthuysen MLF, et al: Incidence and survival of neuroendocrine tumours in the Netherlands according to histological grade: Experience of two decades of cancer registry. Eur J Cancer 49:1975-1983, 2013 5. Hallet J, Law CH, Cukier M, et al: Exploring the rising incidence of neuroendocrine tumors: A population-based analysis of epidemiology, metastatic presentation, and outcomes. Cancer 121:589-597, 2015 6. Ito T, Sasano H, Tanaka M, et al: Epidemiological study of gastroenteropancreatic neuroendocrine tumors in Japan. J Gastroenterol 45:234-243, 2010 7. Tsai HJ, Wu CC, Tsai CR, et al: The epidemiology of neuroendocrine tumors in Taiwan: A nation-wide cancer registry- based study. PLoS One 8:e62487, 2013 8. Oberg K, Castellano D: Current knowledge on diagnosis and staging of neuroendocrine tumors. Cancer Metastasis Rev 30:3-7, 2011 (suppl 1) 9. Rindi G, Kloppel G, Couvelard A, et al: TNM staging of midgut and hindgut (neuro) endocrine tumors: A consensus proposal including a grading system. Virchows Arch 451:757-762, 2007 10. Gastrointestinal Carcinoid Tumours Treatment (PDQ ). http://www.cancer.gov/types/gi-carcinoid-tumors/patient/ gi-carcinoid-treatment-pdq 11. Modlin IM, Moss SF, Gustafsson BI, et al: The archaic distinction between functioning and nonfunctioning neu- roendocrine neoplasms is no longer clinically relevant. Langenbecks Arch Surg 396:1145-1156, 2011 12. Boudreaux JP, Klimstra DS, Hassan MM, et al: The NANETS consensus guideline for the diagnosis and management of neuroendocrine tumors: Well-differentiated neuroendocrine tumors of the jejunum, ileum, appendix, and cecum. Pancreas 39:753-766, 2010 13. Metz DC, Choi J, Strosberg J, et al: A rationale for multidisciplinary care in treating neuroendocrine tumours. Curr Opin Endocrinol Diabetes Obes 19:306-313, 2012 14. Beaumont JL, Zhimei L, Choi S, et al: Health-related quality of life of patients with neuroendocrine tumor compared to the United States general population. Pancreas 39:271, 2010 (abstr) 15. Feinberg Y, Law C, Singh S, et al: Patient experiences of having a neuroendocrine tumour: A qualitative study. Eur J Oncol Nurs 17:541-545, 2013 16. Griffiths J, Willard C, Burgess A, et al: Meeting the ongoing needs of survivors of rarer cancer. Eur J Oncol Nurs 11:434- 441, 2007 17. Institute for Healthcare Improvement: IHI Triple Aim Initiative. http://www.ihi.org/Engage/Initiatives/TripleAim/Pages/ default.aspx 18. Browne K, Roseman D, Shaller D, et al: Analysis & commentary. Measuring patient experience as a strategy for improving primary care. Health Aff (Millwood) 29:921-925, 2010 19. Haugland T, Vatn MH, Veenstra M, et al: Health related quality of life in patients with neuroendocrine tumors compared with the general Norwegian population. Qual Life Res 18:719-726, 2009 20. Beaumont JL, Cella D, Phan AT, et al: Comparison of health-related quality of life in patients with neuroendocrine tumors with quality of life in the general US population. Pancreas 41:461-466, 2012 21. Weaver KE, Forsythe LP, Reeve BB, et al: Mental and physical health-related quality of life among U.S. cancer survivors: Population estimates from the 2010 National Health Interview Survey. Cancer Epidemiol Biomarkers Prev 21:2108-2117, 2012 22. Clancy CM: How patient-centered healthcare can improve quality. http://www.psqh.com/marapr08/ahrq.html 23. Committee on Quality Healthcare in America, Institute of America: Crossing the Quality Chasm: A New Health System for the 21st Century. 1-360, 2015. https://www.nationalacademies.org/hmd/~/media/Files/Report%20Files/2001/ Crossing-the-Quality-Chasm/Quality%20Chasm%202001%20%20report%20brief.pdf 24. Berwick DM: A user’s manual for the IOM’s ‘Quality Chasm’ report. Health Aff (Millwood) 21:80-90, 2002 25. Singh S, Law C: Multidisciplinary reference centers: The care of neuroendocrine tumors. J Oncol Pract 6:e11-e16, 26. Rinke A, Muller HH, Schade-Brittinger C, et al: Placebo-controlled, double-blind, prospective, randomized study on the effect of octreotide LAR in the control of tumor growth in patients with metastatic neuroendocrine midgut tumors: A report from the PROMID Study Group. J Clin Oncol 27:4656-4663, 2009 27. Caplin ME, Pavel M, Cwikła JB, et al: Lanreotide in metastatic enteropancreatic neuroendocrine tumors. N Engl J Med 371:224-233, 2014 28. Yao JC, Fazio N, Singh S, et al: Everolimus for the treatment of advanced, non-functional neuroendocrine tumours of the lung or gastrointestinal tract (RADIANT-4): A randomised, placebo-controlled, phase 3 study. Lancet 387:968- 977, 2016 52 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology 29. Yao JC, Shah MH, Ito T, et al: Everolimus for advanced pancreatic neuroendocrine tumors. N Engl J Med 364:514- 523, 2011 30. Pavel ME, Hainsworth JD, Baudin E, et al: Everolimus plus octreotide long-acting repeatable for the treatment of advanced neuroendocrine tumours associated with carcinoid syndrome (RADIANT-2): A randomised, placebo- controlled, phase 3 study. Lancet 378:2005-2012, 2011 31. Raymond E, Dahan L, Raoul JL, et al: Sunitinib malate for the treatment of pancreatic neuroendocrine tumors. N Engl J Med 364:501-513, 2011 32. Strosberg J, Wolin E, Chasen B, et al: 177-Lu-Dotatate significantly improves progression-free survival in patients with midgut neuroendocrine tumours: Results of the phase III NETTER-1 trial. Presented at: European Cancer Congress 2015, Vienna, Austria, September 25-29, 2015 (abstr 6LBA) 33. Davies E, Cleary PD: Hearing the patient’s voice? Factors affecting the use of patient survey data in quality im- provement. Qual Saf Health Care 14:428-432, 2005 APPENDIX Presented at the 7th Annual Symposium of the North American Neuroendocrine Tumor Society, Nashville, TN, October 10-11, 2014; the European Neuroendocrine Tumor Society (ENETS) 12th Annual Conference, Barcelona, Spain, March 11-13, 2015; and the Oncology Nursing Society 40th Annual Congress, Orlando, FL, April 23-26, 2015. 53 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology http://www.deepdyve.com/assets/images/DeepDyve-Logo-lg.png Journal of Global Oncology Wolters Kluwer Health

Patient-Reported Burden of a Neuroendocrine Tumor (NET) Diagnosis: Results From the First Global Survey of Patients With NETs

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Wolters Kluwer Health
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Copyright (C) 2017 The Authors. Published by Wolters Kluwer Health, Inc. All rights reserved.
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2378-9506
DOI
10.1200/JGO.2015.002980
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Abstract

abstract original report Patient-Reported Burden of a Neuroendocrine Tumor (NET) Diagnosis: Results From the First Global Survey of Patients With NETs Purpose Despite the considerable impact of neuroendocrine tumors (NETs) on patients’ daily lives, the journey of the patient with a NET has rarely been documented, with published data to date being limited to small qualitative studies. NETs are heterogeneous malignancies with nonspecific symptomology, leading to extensive health care use and diagnostic delays that affect survival. A large, international patient survey was conducted to increase understanding of the experience of the patient with a NET and identify unmet needs, with the aim of improving disease awareness and care worldwide. Methods An anonymous, self-reported survey was conducted (online or on paper) from February to May Simron Singh 2014, recruiting patients with NETs from > 12 countries as a collaboration between the International Dan Granberg Neuroendocrine Cancer Alliance and Novartis Pharmaceuticals. Survey questions captured information on sociodemographics, clinical characteristics, NET diagnostic experience, disease impact/management, Edward Wolin interaction with medical teams, NET knowledge/awareness, and sources of information. This article Richard Warner reports the most relevant findings on patient experience with NETs and the impact of NETs on health care Maia Sissons system resources. Teodora Kolarova Results A total of 1,928 patients with NETs participated. A NET diagnosis had a substantially negative Grace Goldstein impact on patients’ personal and work lives. Patients reported delayed diagnosis and extensive NET-related Marianne Pavel health care resource use. Patients desired improvement in many aspects of NET care, including availability of a wider range of NET-specific treatment options, better access to NET experts or specialist centers, and a Kjell Oberg more knowledgeable, better-coordinated/-aligned NET medical team. John Leyden Conclusion This global patient-reported survey demonstrates the considerable burden of NETs with regard to symptoms, work and daily life, and health care resource use, and highlights considerable unmet needs. Author affiliations appear at the end of this article. Further intervention is required to improve the patient experience among those with NETs. Supported by Novartis J Glob Oncol 3. © 2016 by American Society of Clinical Oncology Licensed under the Creative Commons Attribution 4.0 License Pharmaceuticals and conducted as an equal collaboration between the 4,5,7 International Neuroendocrine INTRODUCTION toward increasing incidence is evident. This Cancer Alliance and Novartis. increasing global incidence, together with the Neuroendocrine tumors (NETs) are a diverse Medical editorial writing considerable number of people living with NETs assistance was provided by group of malignancies that arise from neuroendo- (estimated prevalence: 35 per 100,000), high- 1-3 ApotheCom Associates crine cells throughout the body. Although NETs (Yardley, PA) and was funded lights the importance of improving awareness of are uncommon, incidence rates continue to in- by Novartis. this disease, increasing accurate diagnosis, and crease, partly because of greater awareness of the Authors’ disclosures of 3,4 optimizing disease management. disease and increased accuracy of diagnosis. potential conflicts of interest and contributions are found at The incidence of NETs in the United States has NETs are generally graded on the basis of mitotic the end of this article. increased five-fold over the past 30 years, with five count and proliferative index. NETs can be di- Corresponding author: Simron out of every 100,000 people being diagnosed with vided into functional (clinically symptomatic, typ- Singh, MD, MPH, FRCP(C), NETs annually. Although NET incidence varies ically characterized by the hormones they secrete) Odette Cancer Center—Sunnybrook across geographic regions (higher in the Nether- and nonfunctional (silent/nonsecretory, free of Hospital, 2075 Bayview Ave, lands [4.9 per 100,000] and Canada [5.86 per hormone-related symptoms, and generally iden- Room T2-047, Toronto, ON 5 6 1,10 100,000] ; lower in Japan [2.10 per 100,000] tified incidentally) tumors, although current M4N 3M5, Canada; e-mail: simron.singh@sunnybrook.ca. and Taiwan [1.51 per 100,000] ), an overall trend evidence indicates that diagnosis and treatment 43 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology © 2016 by American Society of Clinical Oncology Licensed under the Creative Commons Attribution 4.0 License strategies should follow the same principles re- in support of people living with NETs, collaborated gardless of functional status. The symptoms with Novartis Pharmaceuticals to conduct the first associated with NETs may be nonspecific or ab- global survey to gather data about the experience 1,2,12 sent until more advanced stages, often lead- of patients with NETs from multiple countries. To 2,12 ing to delays in diagnosis. Indeed, NET-related our knowledge, this has been the first endeavor to symptoms may persist for long periods (median, collect information on the patient experience with NETs. The aim of this patient-reported survey was 9.2 years) before an accurate diagnosis is made, to increase understanding of the views and expe- thus potentially placing a substantial burden on riences of patients with NETs, including their both the patient and the health care system. needs and challenges with regard to diagnosis, Because of the nonspecific nature of symptoms, management of NETs, interactions with medical there seems to be no clear pathway of care for teams, knowledge and awareness levels of the patients with NETs; patients may be seen by disease, and information sources. multiple specialists and undergo extensive and repetitive testing, leading to varying and potentially The findings of this survey expand the knowledge conflicting treatment recommendations and con- base regarding NETs from a patient perspective, tributing to delays in an accurate diagnosis. The with the goal of improving disease awareness resulting psychological and emotional burden and patient care. Collecting and reporting such may at least partially contribute to the reported patient-centered data, in addition to directing im- worse health-related quality of life (HRQoL) out- provements in patient experience and care prac- comes among patients with NETs compared with tice, can have overarching beneficial effects on the 14 18 the general population. health care system as a whole. Despite the considerable impact of this disease on In this publication, we present findings most rel- patients’ daily lives, the journey of the patient with evant to the impact of NETs on patients’ daily lives a NET has rarely been documented, with pub- and on the health care system, and highlight un- lished data being limited to small qualitative met needs from the patient perspective. 15,16 studies. These studies highlight the lack METHODS of a standard care pathway for patients with NETs Study Design and Participants and identify a number of potential challenges, including difficulty in achieving a NET diagnosis, Patients were primarily recruited online via the use limited access to information about NETs and to of flyers, Web site postings, e-mails, and social media NET-specific treatment centers, and inadequate channels of the INCA member organizations/patient ongoing support. advocacy groups. For transparency, the Novartis logo was clearly displayed on all survey materials to Patient experience and patient-reported out- indicate that Novartis was involved in the survey, comes are becoming increasingly important end and all materials stated that the sponsoring com- points in oncology. The Institute for Healthcare pany or the INCA patient group partners may use Improvement has introduced an initiative, called the data for disease awareness purposes. the Triple Aim, to help address insufficiencies in health care and thus optimize health care system Survey Details performance. These linked goals are to improve Survey domains and key questions were initially the patient experience of care, improve the health generated at a roundtable meeting of the INCA of populations, and reduce the per capita cost of leadership held during the 10th annual Euro- health care. It is believed that these goals must pean Neuroendocrine Tumor Society confer- be addressed simultaneously to achieve health ence in Barcelona, Spain, in March 2013. Hall & care reform, and one facet of this effort is to em- Partners, a research organization, was used to power individuals and their families in the context construct a detailed survey to assess patients’ ex- of their health care. As oncology care moves periences with NETs on the basis of the meeting toward a more patient-centered care focus, it is discussions and a review of the NET literature. important that clinicians understand the experi- Fourteen NET patient health consumer groups ence of NETs from the perspective of the patient. within INCA had direct input into question devel- The International Neuroendocrine Cancer Alliance opment, and the final questionnaire was reviewed (INCA) consists of a network of 18 independent and edited by all members of INCA and by Novartis charitable organizations and patient groups for between May and October 2013. This anonymous individuals with NETs from 15 countries around survey was developed to be primarily conducted the world. INCA, which aims to be the global voice online, with an approximate time for completion of 44 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Table 1 – Summary of Patient-Reported Sociodemographics and Clinical Characteristics 25 minutes on the basis of length. Paper surveys were also generated and distributed via patient Patient Sociodemographics and Clinical Patients advocacy groups and health care professionals Characteristics (N = 1,928) (HCPs) to reach patients without Internet access. Patient-reported sociodemographics Both the online and paper surveys were available Region, % in eight languages: Bulgarian, Dutch, English, Americas 48 French, German, Japanese, Norwegian, and sim- Europe 40 plified Chinese. Hall & Partners fielded the survey from February to May 2014 and analyzed the Asia 5 results. Oceania 7 Age (mean), years 56.8 Patient-reported data on sociodemographic infor- mation (eg, age, sex, educational background), Age distribution (years), % clinical characteristics (eg, NET type, years since ,40 8 diagnosis, functional status, tumor grade), burden 40-49 17 of NETs on daily life and work, desired improve- 50-59 32 ments, and information sources were collected. In this survey, data were purposefully self-reported to 60-69 31 maximally reflect the patient’s voice. The ques- 70+ 12 tionnaire covered patients’ overall awareness of Female, % 64 NETs, the diagnostic experience throughout their Educational level, % disease course, burden of disease, ongoing man- Not university educated 55 agement, interactions with health care providers, Bachelor degree or higher 45 experience with and access to NET treatments, and resources used for education about NETs. Ques- Caregiver, % tions were categorized as follows: initial screening, Yes 65 patient’s current status, diagnosis, quality of life, No 35 NET management, NET treatment, NET education, Work status, % and demographics (the full survey appears in the Employed Data Supplement). With the exception of certain questions designed to gather numerical informa- Full-time 25 tion, survey questions were closed ended; partic- Part-time 8 ipants were provided options from which to choose. Self-employed 6 When patients were asked to rate a particular Retired 31 parameter,responsesincludedthegradeddescrip- Medical disability 18 tors not at all, somewhat, very, and extremely, and Not employed/homemaker/student 11 when asked the degree to which they (dis)agreed with a particular statement, the descriptors strongly Patient-reported clinical characteristics and somewhat were added to response choices. NET type, % Gastrointestinal 54 Data Analyses Pancreas 22 Global data were analyzed using the MERLIN Lung 12 (Merlinco, London, UK) survey software package. Thymus 1 Survey responses were summarized using de- scriptive statistics, including means, medians, Other 8 and percentages. Statistical significance is shown Unknown 5 at a 95% CI level (P , .05). In some instances, Tumor grade, %* responses are presented as the top two responses (eg, somewhat agree/strongly agree; a moderate amount/a lot). RESULTS Unknown/do not remember 35 Patient Characteristics (Continued on following page) A total of 1,928 patients with NETs participated in the survey, with the majority being recruited through patient advocacy groups (37%) and 45 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Table 1 – Summary of Patient-Reported Sociodemographics and Clinical Characteristics NETs (22%) and lung NETs (12%). Of the patients (Continued) who knew their functional status (n = 1,551), 55% reported having functional disease. Of those who Patient Sociodemographics and Clinical Patients Characteristics (N = 1,928) knew their tumor grade (n = 1,242), 91% reported having grade 1 or 2 tumors; 35% of all patients either Functional status, %† did not know or had not been told their tumor grade. Functional 44 Nonfunctional 15 Diagnostic Delays and Use of Health Care Asymptomatic 21 Resources Unknown/do not remember 19 Mean patient-reported time from first symptom Time since diagnosis (mean), years 5.2 onset to diagnosis was 52 months; 29% of patients required > 5 years for a NET diagnosis, and 58% Time since diagnosis (years), % of patients had metastases at the time of diagnosis. ,559 Patients saw a mean of 6.2 HCPs across a mean of 5-9 27 11.8 doctor visits in the period of first onset of 10-14 8 symptoms before receiving their NET diagnosis 15+ 6 (Figs 1A and 1B). Metastatic disease at the time of 58 On average, approximately three HCPs were in- diagnosis, % volved in the ongoing management of patients with Surgical removal of primary tumor since 70 NETs, with oncologists/hematologists (70%) and diagnosis, % general practitioners (58%) seen most often after diagnosis. Annually, patients reported having a Abbreviations: Ki-67, protein encoded by the MKI67 gene; NET, neuroendocrine tumor. *Grade 1: NETs are relatively slow growing, Ki-67 index < 2%; grade 2: NETs have a less predictable, mean of 5.5 NET-related tests, with 28% receiving moderately aggressive course, Ki-67 index 3%–20%; grade 3: NETs can be highly aggressive, Ki-67 tests six or more times per year after diagnosis. index . 20%. †Functional NETs: produce symptoms caused by the secretions of hormones (eg, flushing, diarrhea, Impact of NETs on Patients’ Personal and Work wheezing, cramping); nonfunctional NETs: do not secrete hormones, but they may cause symptoms Lives caused by the tumors’ growth (eg, pain, intestinal blockage, bleeding). Living with NETs seemed to have a considerable online sources (51%). These patients were re- impact on patients’ personal and work lives. Al- cruited from . 12 countries in the Americas though on a scale of 1 to 6 (1 = very poor health, (United States [n = 758], Canada [n = 164], and 6 = excellent health), patients reported a mean other countries in North, Central, and South Amer- health score of 4, a relatively large proportion ica [n = 6]), Asia (Japan [n = 81], Singapore, and (37%) reported very poor to fair health. Patients other countries in Asia [n = 18]), Europe (Germany reported experiencing numerous ongoing NET- [n = 311], United Kingdom [n = 156], France [n = related symptoms, such as general fatigue/muscle 117], Norway [n = 54], Belgium [n = 29], Bulgaria fatigue/weakness (56%), diarrhea (48%), and [n = 18], and other countries in Europe [n = 78]), abdominal pain or cramping (41%; Fig 2). Many and Oceania (n = 138). The exact number of of these symptoms occurred on a daily basis. countries could not be determined because some Most survey participants (71%) reported a mod- respondents identified the general region in which erate to substantial negative impact of NETs on they lived rather than a specific country. Overall, their daily life, including overall energy levels the majority of respondents were from North Amer- (70%), finances (50%), and the ability to perform ica (n = 922; 48%) and Europe (n = 763; 40%). everyday household chores (45%) or care for family (39%; Fig 3A). A large proportion of pa- Patient-reported sociodemographic parameters tients believed that living with NETs substantially and clinical characteristics are summarized in affected (a moderate amount/a lot) their emo- Table 1. The mean age was 56.8 years, and 64% tional health (60%), the emotional health of were women. Close to half of the patients (45%) family/friends (48%), and their relationships with had a bachelor’s degree or higher, and two-thirds family/friends (34%/34%). had a caregiver (ie, a close family member or friend to help them manage day-to-day NET-associated Most patients (92%) reported making one or more activities). Only 39% of patients were currently work- lifestyle changes as a result of NETs, including ing (full-/part-time employment or self-employed) dietary changes (58%), increased time/money and 18% had a self-identified medical disability spent on traveling to or from their medical appoint- (not further specified). GI NETs were the most ments (52%/51%), and seeing a therapist for common NET type (54%), followed by pancreatic emotional aspects of the disease (20%; Fig 3B). 46 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Fig 1 – (A)Thenumberofhealthcare Mean number of HCPs seen before a NET diagnosis: 6.2 professionals (HCPs) seen before receiving a neuroendocrine tumor (NET) diagnosis. Base 12 12 population: all respondents (N = 1,928). Question: Approximately how many HCPs (including all doctors, 1 2 345 6–9 10–19 20+ Don't specialists, and nurses) were know/can't involved in your diagnosis of remember a NET? Please consider all those you saw from the time Number of HCPs you first experienced symptoms to the time you Mean number of health care visits made before a NET diagnosis: 11.8 received the diagnosis of a NET. (B) The number of healthcare visitsmade before receiving a neuroendocrine tumor (NET) diagnosis. Base 12 population: all respondents (N = 1,928). Question: Approximately how many different visits to HCPs 1–2 3–4 5–6 7–8 9–10 11–15 16+ Don't (including all doctors, know/can't specialists, and nurses) did remember you have to make? Please Number of Health Care Visits consider all those you saw from thetimeyou first experienced symptoms to the time you received the NETs had a significant impact on work life. Almost 439.35] or 182 km [median, 40; range, 1 to diagnosis of a NET. half of patients (49%) reported taking days off from 9,656; standard deviation, 503.38]). work, 27% asked their employer to make accom- modations, and 24% reduced their work hours DISCUSSION because of NETs (Fig 3C). Of the 440 patients not To our knowledge, this is the first large, global currently employed or unable to work because of study that attempts to characterize the impact medical disability, 82% reported having to stop of NETs from the patient perspective. To date, working as a direct result of their NETs. there is a scarcity of published data on the experiences and perspectives of patients with Access Issues NETs, which may be in part due to the rarity of this cancer as well as difficulties in collecting Despite the high frequency of physician visits patient-reported data from an international per- and medical tests, survey findings revealed nu- spective. For this survey, the extensive use of merous unmet needs with regard to NET man- online social media sources through local/ agement. The majority of patients desired better regional advocacy groups enabled recruitment availability of a wider range of NET-specific of a large number of patients with NETs in a treatment options (60%) and access to NET relatively short period of time. experts or a NET specialist center (56%). Close to half believed that more knowledgeable NET Findings from this survey brought to light the medical providers (47%) and a better coordinated/ substantial impact of NETs on patients’ daily aligned NET medical team (45%) would improve lives, including their physical and emotional NET care (Fig 4). In addition, many patients well-being, financial stressors, and impaired lacked access to treatments they know exist interactions with family and friends. The NET- (Table 2). In all cases, the percentage of patients related burden resulted in lifestyle changes with knowledge of particular treatment options and substantial financial strain and had a neg- was greater than the percentage of patients with ative impact on patients’ ability to work. Although access to those options. Patients also reported validated HRQoL assessment tools were not having to travel an extended distance to see their used in this survey, the findings are consistent NET medical providers (mean distance, 126 miles with previously conducted surveys in Norway and [median, 25; range, 1 to 9,942; standard deviation, the United States, which reported worse HRQoL 47 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Patients (%) Patients (%) Fig 2 – Neuroendocrine tumor General fatigue, muscle fatigue, weakness (n = 1,088) 56 (NET) symptoms Diarrhea (n = 924) 41 48 experienced, often on Abdominal pain or cramping (n = 783) 41 41 a daily basis. Base population: all respondents Skin reactions: flushing, rash, redness, thinning (n = 706) 46 37 (n values as indicated in Sweating, headaches/dizziness, nausea (n = 589) 41 31 figure). Questions: Which of Anxiety, palpitations (n = 492) 35 26 the following symptoms, if Breathlessness/wheezing (n = 471) 52 24 any, do you suffer from as a result of your NET? Select Changes in blood pressure (n = 428) 45 22 all that apply. How Heartburn/reflux (n = 401) 21 frequently do you suffer Weight loss (n = 399) 38 21 from each of the following symptomsas a result of your Memory loss and/or confusion (n = 373) 53 19 NET? Steatorrhea (increase in fat content in stools) (n = 368) 46 19 Weight gain; large, round face; excessive fat torso (n = 289) 75 15 Percentage of the total population Vision problems (n = 226) 67 12 of patients experiencing each symptom. Osteoporosis (n = 136) 82 7 Within each subgroup of patients Rectal bleeding (n = 96) who experienced a particular symptom, the percentage who Jaundice (n = 27) experienced that symptom on a constant or daily basis. Other (n = 125) 0 10203040506070 Patients (%) scores in patients with NETs versus the general outcome if they are well informed and assertive 19,20 18,22 population, as expected. A previous US sur- regarding their treatment. Since 2001, vey of cancer survivors also reported poorer when a report by the Institute of Medicine pro- posed that patient-centeredness should be one HRQoL in this group compared with adults with- of the aims of the US health care system, out cancer. increasing efforts have been made to provide This survey also highlighted numerous unmet more patient-centered care, in part by custom- needs for patients with NETs, including the desire izing that care according to the individual pa- for better access to NET-specific medical teams tient’s needs and values and placing patients in (ie, NET experts and specialist centers), as well as control of their own care. The findings from NET-specific treatments and information. A need this survey emphasize the need for more stan- for increased awareness of NETs among the med- dardized pathways of NET patient care that ical community was also demonstrated, reflected address ongoing issues and challenges identi- by a desire for patients to have better access to fied by patients. Patients often reported a sub- more knowledgeable NET medical providers as stantial delay in diagnosis and extensive use of well as a better coordinated NET medical team. health care resources (involving large numbers Indeed, the finding that 35% of patients did not of HCP visits and numerous tests) and also know or had not been told their tumor grade may voiced the desire for better coordinated care reflect a lack of communication between patients from medical providers. Lack of these elements and physicians regarding NETs. These findings potentially contributes to excessive use of health are aligned with results of previous survey-based care resources, affecting both patients and the oncology studies, particularly with regard to ac- health care system. This scenario is the inverse of cess to information about NETs from physicians, the triple aim promoted by the Institute for Health- NET-specific treatment centers, and disease- care Improvement report on patient-centered 15,16 17,23 specific support. Clearly, there is a need for care. Success with patient-centered ap- better coordination and accessibility of care for proaches to NET care (improved outcomes patients with NETs. and quality of care) are increasingly being demonstrated in multidisciplinary specialist It has been established that not only are pa- centers. tients more likely to be compliant with treatment if they are actively involved in their own health Our survey has several potential limitations that care, but also they are more likely to have a better should be taken into consideration, including 48 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Fig 3 – (A) Negative impact of A My overall energy levels 70 neuroendocrine tumors My emotional health 60 (NETs) on daily life. Base: My ability to participate in leisure activities all respondents (N = 1,928). Question: How My attitude towards daily life much has each of the My social life following areas of your life been negatively affected, if 51 My ability to travel at all, by your NET? Top two My finances 50 box scores are shown (a The emotional health of those close to me moderate amount/a lot). (B) (family, friends, etc.) Lifestyle changes caused My ability to perform everyday household chores by having a NET. Base: all My ability to care for my family 39 respondents (N = 1,928). Question: Since you were My relationship with my spouse/partner 35 diagnosed with your NET, My relationships with my friends 34 have you had to make any of My relationships with my family the following changes? (other than a spouse/partner) Please select all that apply. My ability to perform my job 16 (C) Impact of NETs on work 0 10203040506070 life. *For example, flexible work schedule, work from Patients (%) home, adaptive devices, opportunities for rest. Base Make dietary changes 58 population: respondents Spend a lot of time on travel to/from who are working full time/ medical appointments part time or are self- Increase spending on travel for employed (n = 741). medical appointments Question: Has your NET Stop or cut back on physical activities impacted you at work in any Stop or cut back on social life of the following ways? Make exercise changes 43 Please select all that apply. Cut back on leisure purchases HCP, health care (vacations, etc.) professional. Increase spending on nutritional products See a therapist for emotional aspects of disease Stop or cut back on caregiving for family/friend Stop or cut back on childcare 9 Other 5 0 10203040506070 Patients (%) I have had to take days off from work I have had to ask my employer to make accommodations* I have had to work reduced hours I have had to stop working altogether for a period of time Colleagues have looked at me differently I have had to take a less demanding job within the company I have had to switch to a new job 6 Seek disability 6 The HCPs who treat my cancer have advised me to stop working Other None of the above 23 0 10203040506070 Patients (%) 49 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology Fig 4 – Patients’ beliefs regarding A wider range of NET treatment options 60 improvements that would Better access to NET experts/medical help with ongoing centers that specialize in NET neuroendocrine tumor More information about/more opportunity to participate in NET clinical trials (NET) management. Base population: all respondents More knowledgable NET medical providers 47 (N = 1,928). Question: More treatments available in my country Which of the following that I see in other countries would help with the ongoing A better coordinated/aligned team of NET medical providers management of your NET? More information brochures from my NET medical providers Other/none of the above 11 0 10203040506070 Patients (%) recall bias, given the fact that findings are solely These limitations notwithstanding, a key strength based on patient responses to closed-ended of this global patient-reported survey is that find- questions and not confirmed by medical re- ings directly reflect patients’ perspectives on cords. In addition, the survey was qualitative NETs. Having an understanding of how patients in nature; standardized, validated HRQoL as- experience their disease is unique and of signif- sessments were not used. Finally, most patients icant value, because it directly aligns with the new were recruited online (Web sites, 31%; social movement in health care toward patient-centered 17,23 media, 20%) and through patient advocacy care. A concerted effort from clinicians and groups (37%). Respondents may have been HCPs at various levels is needed to effectively more likely to be highly engaged, motivated interpret the data and successfully implement care seekers. There was also a preponderance appropriate care processes to bring these im- of female respondents, and nearly half of the provements in the quality of patient-centered care participants (45%; Table 1) had higher educa- to fruition. Regional and country-specific ana- tion levels. As such, the survey population may lyses of these survey data have also been con- not be fully representative of a heterogeneous ducted; INCA is currently working with their local NET population. However, comparison of the organizations to use this information to help better disease characteristics (site of origin, func- inform NET patient care according to geographic tional status, and grade/differentiation) of the region. patient population that participated in this sur- vey to those included in pivotal phase 3 clini- In conclusion, this large, global patient survey demonstrates the considerable burden of NETs cal trials on NETs revealed similarities (Data 26-32 Supplement). with respect to symptoms, impact on work and Table 2 – Availability of Neuroendocrine Tumor Treatments Treatments I Have Received/Am Currently Treatments I Have Heard Of* Treatments I Have Access To† Receiving‡ Surgery 90†‡ 76‡ 67 Chemotherapy 75†‡ 38‡ 22 Drug therapy other than chemotherapy 79†‡ 56‡ 47 PRRT 74†‡ 28‡ 17 Interventional radiology 65†‡ 30‡ 16 Observation 74†‡ 53‡ 44 Other/none of the above 13†‡ 77 NOTE. Data presented as %. Base: all respondents (N = 1,928). Question: This is a list of available NET treatments. Please select those you have heard of, those you have access to (meaning they are available to you), and those you have received/are currently receiving for the treatment of your NET. Abbreviation: NET, neuroendocrine tumor; PRRT, peptide receptor radionuclide therapy. *†‡Symbols appearing next to a given value indicate significant differences, P, .05, between the patient group specified in that column compared with those specified in the other columns. 50 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology daily life, and health care resource use and and insights regarding best practices for disease highlights unmet needs and ongoing challenges. management. Taken together, this survey provides valuable findings that maybeusedinfutureresearchto DOI: 10.1200/JGO.2015.002980 gain a better understanding of this rare disease Published online on jgo.org on June 8, 2016. AUTHOR CONTRIBUTIONS Grace Goldstein COO of The Carcinoid Cancer Foundation, Inc.; reports that Conception and design: Maia Sissons, Teodora Kolarova, the Foundation has received grants from Novartis, Ipsen, Grace Goldstein, John Leyden Advanced Accelerator Applications, and Lexicon Collection and assembly of data: Maia Sissons, Pharmaceuticals Teodora Kolarova, Grace Goldstein, John Leyden Travel, Accommodations, Expenses: Novartis Data analysis and interpretation: All authors Manuscript writing: All authors Marianne Pavel Final approval of manuscript: All authors Honoraria: Ipsen, Lexicon Pharmaceuticals, Novartis, Pfizer AUTHORS’ DISCLOSURES OF Consulting or Advisory Role: Ipsen, Lexicon Pharmaceuticals, POTENTIAL CONFLICTS OF INTEREST Novartis, Pfizer The following represents disclosure information provided by Research Funding: Novartis authors of this manuscript. All relationships are considered Travel, Accommodations, Expenses: Ipsen, Novartis compensated. Relationships are self-held unless noted. I = Kjell Oberg Immediate Family Member, Inst = My Institution. Relation- No relationship to disclose ships may not relate to the subject matter of this manuscript. For more information about ASCO’s conflict of interest policy, John Leyden please refer to www.asco.org/rwc or ascopubs.org/jco/site/ifc. President of the Unicorn Foundation; reports that the Simron Singh Foundation has received support from Novartis Australia, Honoraria: Novartis, Pfizer, Ipsen Pfizer Australia, and Ipsen Travel, Accommodations, Expenses: Novartis, Pfizer, Ipsen Dan Granberg ACKNOWLEDGMENT We thank the patients who participated in this survey No relationship to disclose and our patient advocacy partners throughout the world Edward Wolin who contributed to this project, including The Unicorn Honoraria: Novartis, Ipsen, Celgene, Advanced Accelerator Foundation (Australia), vzw NET & MEN Kanker Applications (Belgium), The Association of Cancer Patients and Consulting or Advisory Role: Novartis, Ipsen, Celgene, Friends (APOZ, Bulgaria), Carcinoid-Neuroendocrine Advanced Accelerator Applications Tumour Society (CNETS, Canada), Association des Patients Porteurs de Tumeurs Endocrines Diverses (APTED, Richard Warner France), Netzwerk Neuroendokrine Tumoren (Germany), Honoraria: Novartis, Pfizer, Lexicon Pharmaceuticals PanCAN (Japan), Unicorn Foundation NZ (New Zealand), Consulting or Advisory Role: Novartis, Pfizer, Lexicon CarciNor (Norway), Carcinoid & Neuroendocrine Tumor Pharmaceuticals Society (CNETS, Singapore), NET Patient Foundation Research Funding: Lexicon Pharmaceuticals and The Association for Multiple Endocrine Neoplasia Disorders (AMEND, United Kingdom), and The Maia Sissons Carcinoid Cancer Foundation and Caring for Carcinoid Research Funding: Novartis, Ipsen, Pfizer Foundation (United States). Hall & Partners, a Teodora Kolarova research organization, fielded the survey and analyzed the No relationship to disclose results. Affiliations Simron Singh, University of Toronto, Toronto, Ontario, Canada; Dan Granberg and Kjell Oberg, Uppsala University Hospital, Uppsala, Sweden; Edward Wolin, Montefiore Einstein Center for Cancer Care, Bronx, NY; Richard Warner, Mount Sinai School of Medicine,NewYork,NY;MaiaSissons,NETPatientFoundation,HockleyHeath,UnitedKingdom;TeodoraKolarova,APOZ&Friends, Sofia, Bulgaria; Grace Goldstein, The Carcinoid Cancer Foundation, Inc., White Plains, NY; Marianne Pavel,ChariteU ´ ni- versitats ¨ medizin Berlin, Berlin, Germany; and John Leyden, The Unicorn Foundation, Mosman, New South Wales, Australia. REFERENCES 1. Oberg KE: Gastrointestinal neuroendocrine tumors. Ann Oncol 21:vii72-vii80, 2010 (suppl 7) 2. Vinik AI, Woltering EA, Warner RR, et al: NANETS consensus guidelines for the diagnosis of neuroendocrine tumor. 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Strosberg J, Wolin E, Chasen B, et al: 177-Lu-Dotatate significantly improves progression-free survival in patients with midgut neuroendocrine tumours: Results of the phase III NETTER-1 trial. Presented at: European Cancer Congress 2015, Vienna, Austria, September 25-29, 2015 (abstr 6LBA) 33. Davies E, Cleary PD: Hearing the patient’s voice? Factors affecting the use of patient survey data in quality im- provement. Qual Saf Health Care 14:428-432, 2005 APPENDIX Presented at the 7th Annual Symposium of the North American Neuroendocrine Tumor Society, Nashville, TN, October 10-11, 2014; the European Neuroendocrine Tumor Society (ENETS) 12th Annual Conference, Barcelona, Spain, March 11-13, 2015; and the Oncology Nursing Society 40th Annual Congress, Orlando, FL, April 23-26, 2015. 53 Volume 3, Issue 1, February 2017 jgo.org JGO – JournalofGlobal Oncology

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Journal of Global OncologyWolters Kluwer Health

Published: Jun 8, 2016

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