Identifying the unmet health needs of patients with congenital hypogonadotropic hypogonadism using a web-based needs assessment: implications for online interventions and peer-to-peer support

Orphanet Journal of Rare Diseases

Nelly Pitteloud³  Diane Morin⁴  Richard Quinton¹  Andrew A Dwyer² 

[1] University of Newcastle-upon-Tyne, Institute of Genetic Medicine and the Royal Victoria Infirmary, Newcastle-upon-Tyne NE1 3BZ, UK;University of Lausanne, Institut universitaire de formation et de recherche en soins and the Endocrinology, Diabetes & Metabolism Service of the Centre Hospitalier Universitaire Vaudois, 46 Rue du Bugnon, BH19.317, Lausanne CH-1011, Switzerland;University of Lausanne and the Endocrinology, Diabetes & Metabolism Service of the Centre Hospitalier Universitaire Vaudois, 46 Rue du Bugnon, Lausanne 1011, Switzerland;University of Lausanne Institut universitaire de formation et de recherche en soins, Biopole 2 - Route de la Corniche 10, Lausanne 1010, Switzerland

关键词: Nursing;    Patient-centered care;    Health promotion;    Rare diseases;    E-health;    Internet;    Community based participatory research;    Kallmann syndrome;    Congenital hypogonadotropic hypogonadism;   

Others  :  861620
DOI  :  10.1186/1750-1172-9-83

 received in 2014-03-21, accepted in 2014-06-04,  发布年份 2014

【 摘 要 】

Background

Patients with rare diseases such as congenital hypogonadotropic hypogonadism (CHH) are dispersed, often challenged to find specialized care and face other health disparities. The internet has the potential to reach a wide audience of rare disease patients and can help connect patients and specialists. Therefore, this study aimed to: (i) determine if web-based platforms could be effectively used to conduct an online needs assessment of dispersed CHH patients; (ii) identify the unmet health and informational needs of CHH patients and (iii) assess patient acceptability regarding patient-centered, web-based interventions to bridge shortfalls in care.

Methods

A sequential mixed-methods design was used: first, an online survey was conducted to evaluate health promoting behavior and identify unmet health and informational needs of CHH men. Subsequently, patient focus groups were held to explore specific patient-identified targets for care and to examine the acceptability of possible online interventions. Descriptive statistics and thematic qualitative analyses were used.

Results

105 male participants completed the online survey (mean age 37 ± 11, range 19–66 years) representing a spectrum of patients across a broad socioeconomic range and all but one subject had adequate healthcare literacy. The survey revealed periods of non-adherence to treatment (34/93, 37%) and gaps in healthcare (36/87, 41%) exceeding one year. Patient focus groups identified lasting psychological effects related to feelings of isolation, shame and body-image concerns. Survey respondents were active internet users, nearly all had sought CHH information online (101/105, 96%), and they rated the internet, healthcare providers, and online community as equally important CHH information sources. Focus group participants were overwhelmingly positive regarding online interventions/support with links to reach expert healthcare providers and for peer-to-peer support.

Conclusion

The web-based needs assessment was an effective way to reach dispersed CHH patients. These individuals often have long gaps in care and struggle with the psychosocial sequelae of CHH. They are highly motivated internet users seeking information and tapping into online communities and are receptive to novel web-based interventions addressing their unmet needs.

【 授权许可】

   
2014 Dwyer et al.; licensee BioMed Central Ltd.

【 图 表 】

【 参考文献 】

• [1]EURORDIS: The voice of 12,000 patients: Experiences and expectations of rare disease patients on diagnosis and care in Europe. France: Boulogne-Billancourt; 2009.
• [2]Holtzclaw Williams P: Policy framework for rare disease health disparities. Policy Polit Nurs Pract 2011, 12:114-118.
• [3]Cohen JS, Biesecker BB: Quality of life in rare genetic conditions: a systematic review of the literature. Am J Med Genet A 2010, 152A:1136-1156.
• [4]Huyard C: What, if anything, is specific about having a rare disorder? Patients' judgements on being ill and being rare. Health Expect 2009, 12:361-370.
• [5]Nettleton S, Watt I, O'Malley L, Duffey P: Understanding the narratives of people who live with medically unexplained illness. Patient Educ Couns 2005, 56:205-210.
• [6]Fox S: Peer-to-peer healthcare: Many people - especially those living with chronic or rare diseases - use online connections to supplement professional medical advice. Washington, D.C: Pew Internet; 2011.
• [7]Tozzi AE, Mingarelli R, Agricola E, Gonfiantini M, Pandolfi E, Carloni E, Gesualdo F, Dallapiccola B: The internet user profile of Italian families of patients with rare diseases: a web survey. Orphanet J Rare Dis 2013, 8:76. BioMed Central Full Text
• [8]Ayme S, Kole A, Groft S: Empowerment of patients: lessons from the rare diseases community. Lancet 2008, 371:2048-2051.
• [9]Fromantin M, Gineste J, Didier A, Rouvier J: [Impuberism and hypogonadism at induction into military service. Statistical study]. Probl Actuels Endocrinol Nutr 1973, 16:179-199.
• [10]Brioude F, Bouligand J, Trabado S, Francou B, Salenave S, Kamenicky P, Brailly-Tabard S, Chanson P, Guiochon-Mantel A, Young J: Non-syndromic congenital hypogonadotropic hypogonadism: clinical presentation and genotype-phenotype relationships. Eur J Endocrinol 2010, 162:835-851.
• [11]Mitchell AL, Dwyer A, Pitteloud N, Quinton R: Genetic basis and variable phenotypic expression of Kallmann syndrome: towards a unifying theory. Trends Endocrinol Metab 2011, 22:249-258.
• [12]Huffer V, Scott WH, Connor TB, Lovice H: Psychological studies of adult male patients with sexual infantilism before and after androgen therapy. Ann Intern Med 1964, 61:255-268.
• [13]Smith N, Quinton R: Kallmann syndrome. BMJ 2012, 345:e6971.
• [14]Aydogan U, Aydogdu A, Akbulut H, Sonmez A, Yuksel S, Basaran Y, Uzun O, Bolu E, Saglam K: Increased frequency of anxiety, depression, quality of life and sexual life in young hypogonadotropic hypogonadal males and impacts of testosterone replacement therapy on these conditions. Endocr J 2012, 59:1099-1105.
• [15]Lasaite L, Ceponis J, Preiksa RT, Zilaitiene B: Impaired emotional state, quality of life and cognitive functions in young hypogonadal men. Andrologia 2013. Dec 8. doi:10.1111/and.12199. [Epub ahead of print]
• [16]Pitteloud N, Hayes FJ, Dwyer A, Boepple PA, Lee H, Crowley WF Jr: Predictors of outcome of long-term GnRH therapy in men with idiopathic hypogonadotropic hypogonadism. J Clin Endocrinol Metab 2002, 87:4128-4136.
• [17]Liu PY, Baker HW, Jayadev V, Zacharin M, Conway AJ, Handelsman DJ: Induction of spermatogenesis and fertility during gonadotropin treatment of gonadotropin-deficient infertile men: predictors of fertility outcome. J Clin Endocrinol Metab 2009, 94:801-808.
• [18]Warne DW, Decosterd G, Okada H, Yano Y, Koide N, Howles CM: A combined analysis of data to identify predictive factors for spermatogenesis in men with hypogonadotropic hypogonadism treated with recombinant human follicle-stimulating hormone and human chorionic gonadotropin. Fertil Steril 2009, 92:594-604.
• [19]Raivio T, Falardeau J, Dwyer A, Quinton R, Hayes FJ, Hughes VA, Cole LW, Pearce SH, Lee H, Boepple P, Crowley WF Jr, Pitteloud N: Reversal of idiopathic hypogonadotropic hypogonadism. N Engl J Med 2007, 357:863-873.
• [20]Dunbar-Jacob J, Erlen JA, Schlenk EA, Ryan CM, Sereika SM, Doswell WM: Adherence in chronic disease. Annu Rev Nurs Res 2000, 18:48-90.
• [21]Funnell MM, Anderson RM: MSJAMA: the problem with compliance in diabetes. JAMA 2000, 284:1709.
• [22]Finkelstein JS, Klibanski A, Neer RM, Greenspan SL, Rosenthal DI, Crowley WF Jr: Osteoporosis in men with idiopathic hypogonadotropic hypogonadism. Ann Intern Med 1987, 106:354-361.
• [23]Popat VB, Calis KA, Vanderhoof VH, Cizza G, Reynolds JC, Sebring N, Troendle JF, Nelson LM: Bone mineral density in estrogen-deficient young women. J Clin Endocrinol Metab 2009, 94:2277-2283.
• [24]Ding EL, Song Y, Malik VS, Liu S: Sex differences of endogenous sex hormones and risk of type 2 diabetes: a systematic review and meta-analysis. JAMA 2006, 295:1288-1299.
• [25]Corona G, Monami M, Rastrelli G, Aversa A, Sforza A, Lenzi A, Forti G, Mannucci E, Maggi M: Type 2 diabetes mellitus and testosterone: a meta-analysis study. Int J Androl 2011, 34:528-540.
• [26]Brand JS, van der Tweel I, Grobbee DE, Emmelot-Vonk MH, van der Schouw YT: Testosterone, sex hormone-binding globulin and the metabolic syndrome: a systematic review and meta-analysis of observational studies. Int J Epidemiol 2011, 40:189-207.
• [27]Yialamas MA, Dwyer AA, Hanley E, Lee H, Pitteloud N, Hayes FJ: Acute sex steroid withdrawal reduces insulin sensitivity in healthy men with idiopathic hypogonadotropic hypogonadism. J Clin Endocrinol Metab 2007, 92:4254-4259.
• [28]Laitinen EM, Hero M, Vaaralahti K, Tommiska J, Raivio T: Bone mineral density, body composition and bone turnover in patients with congenital hypogonadotropic hypogonadism. Int J Androl 2012, 35:534-540.
• [29]Wallerstein NB, Duran B: Using community-based participatory research to address health disparities. Health Promot Pract 2006, 7:312-323.
• [30]COST Action BM1105 [http://www.gnrhnetwork.eu/ webcite]
• [31]Young J: Approach to the male patient with congenital hypogonadotropic hypogonadism. J Clin Endocrinol Metab 2012, 97:707-718.
• [32]Clinical trials registry of the U.S. National Institutes of Health. [http://clinicaltrials.gov/ webcite]
• [33]Chew LD, Bradley KA, Boyko EJ: Brief questions to identify patients with inadequate health literacy. Fam Med 2004, 36:588-594.
• [34]Chew LD, Griffin JM, Partin MR, Noorbaloochi S, Grill JP, Snyder A, Bradley KA, Nugent SM, Baines AD, Vanryn M: Validation of screening questions for limited health literacy in a large VA outpatient population. J Gen Intern Med 2008, 23:561-566.
• [35]Pender NJ, Murdaugh C, Parsons MA: Health promotion in nursing practice. In Book Health promotion in nursing practice. 6th edition. Pearson: Upper Saddle River NJ; 2010.
• [36]Saldana J: Coding Manual for Qualitative Researchers. Thousand Oaks, CA: Sage; 2009.
• [37]Kruer MC, Steiner RD: The role of evidence-based medicine and clinical trials in rare genetic disorders. Clin Genet 2008, 74:197-207.
• [38]Watson MS, Epstein C, Howell RR, Jones MC, Korf BR, McCabe ER, Simpson JL: Developing a national collaborative study system for rare genetic diseases. Genet Med 2008, 10:325-329.
• [39]Eysenbach G, Wyatt J: Using the Internet for surveys and health research. J Med Internet Res 2002, 4:E13.
• [40]Close S, Smaldone A, Fennoy I, Reame N, Grey M: Using information technology and social networking for recruitment of research participants: experience from an exploratory study of pediatric Klinefelter syndrome. J Med Internet Res 2013, 15:e48.
• [41]Ramo DE, Prochaska JJ: Broad reach and targeted recruitment using Facebook for an online survey of young adult substance use. J Med Internet Res 2012, 14:e28.
• [42]Shahani S, Braga-Basaria M, Basaria S: Androgen deprivation therapy in prostate cancer and metabolic risk for atherosclerosis. J Clin Endocrinol Metab 2008, 93:2042-2049.
• [43]Hudelson P, Kolly V, Perneger T: Patients' perceptions of discrimination during hospitalization. Health Expect 2010, 13:24-32.
• [44]Kritz M, Gschwandtner M, Stefanov V, Hanbury A, Samwald M: Utilization and perceived problems of online medical resources and search tools among different groups of European physicians. J Med Internet Res 2013, 15:e122.
• [45]PubMed Central [http://www.ncbi.nlm.nih.gov/pmc/ webcite]
• [46]Hartzler A, Pratt W: Managing the personal side of health: how patient expertise differs from the expertise of clinicians. J Med Internet Res 2011, 13:e62.
• [47]Lorig K, Ritter PL, Plant K, Laurent DD, Kelly P, Rowe S: The South Australia health chronic disease self-management Internet trial. Health Educ Behav 2013, 40:67-77.
• [48]Archambault PM: WikiBuild: a new application to support patient and health care professional involvement in the development of patient support tools. J Med Internet Res 2011, 13:e114.
• [49]Gupta S, Wan FT, Newton D, Bhattacharyya OK, Chignell MH, Straus SE: WikiBuild: a new online collaboration process for multistakeholder tool development and consensus building. J Med Internet Res 2011, 13:e108.
• [50]World Health Organization (WHO): Ottawa Charter for Health Promotion. In First International Conference on Health Promotion. Ottawa, Canada: World Helath Organization; 1986.
• [51]Pender NJ, Murdaugh CL, Parsons MA: Health Promotion in Nursing Practice. 6th edition. Upper Saddle River, NJ: Prentice Hall; 2010.
• [52]Ho AY, Berggren I, Dahlborg-Lyckhage E: Diabetes empowerment related to pender's health promotion model: a meta-synthesis. Nurs Health Sci 2010, 12:259-267.
• [53]Greenhalgh T: Patient and public involvement in chronic illness: beyond the expert patient. BMJ 2009, 338:b49.
• [54]Howe J: The rise of crowdsourcing. Wired Magazine 2006, 14:1-4.
• [55]Swan M: Crowdsourced health research studies: an important emerging complement to clinical trials in the public health research ecosystem. J Med Internet Res 2012, 14:e46.
• [56]Proudfoot J, Parker G, Manicavasagar V, Hadzi-Pavlovic D, Whitton A, Nicholas J, Smith M, Burckhardt R: Effects of adjunctive peer support on perceptions of illness control and understanding in an online psychoeducation program for bipolar disorder: a randomised controlled trial. J Affect Disord 2012, 142:98-105.
• [57]van der Wulp I, De Leeuw JR, Gorter KJ, Rutten GE: Effectiveness of peer-led self-management coaching for patients recently diagnosed with Type 2 diabetes mellitus in primary care: a randomized controlled trial. Diabet Med 2012, 29:e390-e397.
• [58]Seybert H: Internet use in households and by individuals in 2011. In Book Internet use in households and by individuals in 2011 (Editor ed.^eds.). City: European Commission; 2011.
• [59]Napoli PM, Obar JA: Mobile Leapfrogging and Digital Divide Policy: Assessing the limitations of mobile Internet access. In Book Mobile Leapfrogging and Digital Divide Policy: Assessing the limitations of mobile Internet access. (Editor ed.^eds.). City: New America Foundation; 2013.
• [60]Bedgood R, Sadurski R, Schade RR: The use of the internet in data assimilation in rare diseases. Dig Dis Sci 2007, 52:307-312.