The Burden of Spinal Muscular Atrophy on Informal Caregivers
Abstract
:1. Introduction
2. Materials and Methods
Statistical Analysis
3. Results
4. Discussion
5. Conclusions
Supplementary Materials
Author Contributions
Funding
Acknowledgments
Conflicts of Interest
References
- Alías, L.; Barceló, M.; Bernal, S.; Martínez-Hernández, R.; Also-Rallo, E.; Vázquez, C.; Santana, A.; Millán, J.; Baiget, M.; Tizzano, E. Improving detection and genetic counseling in carriers of spinal muscular atrophy with two copies of theSMN1gene. Clin. Genet. 2013, 85, 470–475. [Google Scholar] [CrossRef] [PubMed]
- Finkel, R.S.; McDermott, M.P.; Kaufmann, P.; Darras, B.T.; Chung, W.K.; Sproule, D.M.; Kang, P.B.; Foley, A.R.; Yang, M.L.; Martens, W.B.; et al. Observational study of spinal muscular atrophy type I and implications for clinical trials. Neurology 2014, 83, 810–817. [Google Scholar] [CrossRef] [PubMed]
- Kolb, S.J.; Coffey, C.S.; Yankey, J.W.; Pt, D.K.K.; Arnold, W.D.; Rutkove, S.B.; Swoboda, K.J.; Reyna, S.P.; Sakonju, A.; Darras, B.T.; et al. Natural history of infantile-onset spinal muscular atrophy. Ann. Neurol. 2017, 82, 883–891. [Google Scholar] [CrossRef] [PubMed]
- Lager, C.; Kroksmark, A.K. Pain in adolescents with spinal muscular atrophy and Duchenne and Becker muscular dystrophy. Eur. J. Paediatr. Neurol. 2015, 19, 537–546. [Google Scholar] [CrossRef]
- Bach, J.R. Noninvasive Respiratory Management of Patients with Neuromuscular Disease. Ann. Rehabil Med. 2017, 41, 519–538. [Google Scholar] [CrossRef] [Green Version]
- Rouault, F.; Christie-Brown, V.; Broekgaarden, R.; Gusset, N.; Henderson, D.; Marczuk, P.; Schwersenz, I.; Bellis, G.; Cottet, C. Disease impact on general well-being and therapeutic expectations of European Type II and Type III spinal muscular atrophy patients. Neuromuscul. Disord. 2017, 27, 428–438. [Google Scholar] [CrossRef]
- Van Exel, J.; Bobinac, A.; Koopmanschap, M.; Brouwer, W. The invisible hands made visible: Recognizing the value of informal care in healthcare decision-making. Expert Rev. Pharm. Outcomes Res. 2008, 8, 557–561. [Google Scholar] [CrossRef]
- Hoefman, R.J.; Van Exel, J.; Brouwer, W. How to Include Informal Care in Economic Evaluations. Pharm. Econ. 2013, 31, 1105–1119. [Google Scholar] [CrossRef]
- Oliva-Moreno, J.; Trapero-Bertran, M.; Peña-Longobardo, L.M.; Del Pozo-Rubio, R. The Valuation of Informal Care in Cost-of-Illness Studies: A Systematic Review. Pharm. Econ. 2017, 35, 331–345. [Google Scholar] [CrossRef]
- Krol, M.; Papenburg, J.; Van Exel, J. Does Including Informal Care in Economic Evaluations Matter? A Systematic Review of Inclusion and Impact of Informal Care in Cost-Effectiveness Studies. Pharm. Econ. 2015, 33, 123–135. [Google Scholar] [CrossRef]
- Peña-Longobardo, L.M.; Rodríguez-Sánchez, B.; Oliva-Moreno, J.; Aranda-Reneo, I.; López-Bastida, J. How relevant are social costs in economic evaluations? The case of Alzheimer’s disease. Eur. J. Health Econ. 2019, 20, 1207–1236. [Google Scholar] [CrossRef] [PubMed]
- Klug, C.; Schreiber-Katz, O.; Thiele, S.; Schorling, E.; Zowe, J.; Reilich, P.; Walter, M.C.; Nagels, K.H. Disease burden of spinal muscular atrophy in Germany. Orphanet J. Rare Dis. 2016, 11, 1–9. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- López-Bastida, J.; Peña-Longobardo, L.M.; Aranda-Reneo, I.; Tizzano, E.; Sefton, M.; Oliva-Moreno, J. Social/economic costs and health-related quality of life in patients with spinal muscular atrophy (SMA) in Spain. Orphanet J. Rare Dis. 2017, 12, 1–7. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Armstrong, E.P.; Malone, D.C.; Yeh, W.S.; Dahl, G.J.; Lee, R.L.; Sicignano, N. The economic burden of spinal muscular atrophy. J. Med. Econ. 2016, 19, 822–826. [Google Scholar] [CrossRef]
- Droege, M.; Sproule, D.; Arjunji, R.; Gauthier-Loiselle, M.; Cloutier, M.; Dabbous, O. Economic burden of spinal muscular atrophy in the United States: A contemporary assessment. J. Med. Econ. 2020, 23, 70–79. [Google Scholar] [CrossRef] [Green Version]
- Tetafort, A.; Couray-Targe, S.; Harmand, S.; Ansolabehere, X.; Maurel, F.; Hammes, F. Assessing the Real-World Hospital Economic Burden of Spinal Muscular Atrophy (SMA) In France. Value Health 2017, 20, A553. [Google Scholar] [CrossRef]
- Qian, Y.; McGraw, S.; Henne, J.; Jarecki, J.; Hobby, K.; Yeh, W.S. Understanding the experiences and needs of individuals with Spinal Muscular Atrophy and their parents: A qualitative study. BMC Neurol. 2015, 15, 1–12. [Google Scholar] [CrossRef] [Green Version]
- Bauer, J.; Sousa-Poza, A. Impact of informal care on caregivers employment, health and family. Popul. Ageing 2015, 8, 113–145. [Google Scholar] [CrossRef] [Green Version]
- Triantafillou, J.; Naiditch, M.; Repkova, K.; Stiehr, K.; Carretero, S.; Emilsson, T. Informal care in the long-term care system. Eur. Overv. Pap. 2011. Available online: https://www.euro.centre.org/downloads/detail/768 (accessed on 23 October 2020).
- Peña-Longobardo, L.M.; Aranda-Reneo, I.; Oliva-Moreno, J.; Litzkendorf, S.; Durand-Zaleski, I.; Tizzano, E.F.; Bastida, J.L. The Economic Impact and Health-Related Quality of Life of Spinal Muscular Atrophy. An Analysis across Europe. Int. J. Environ. Res. Public Health 2020, 17, 5640. [Google Scholar] [CrossRef]
- López-Bastida, J.; Oliva-Moreno, J.; Linertová, R.; Serrano-Aguilar, P. Social/economic costs and health-related quality of life in patients with rare diseases in Europe. Eur. J. Health Econ. 2016, 17, 1–5. [Google Scholar] [CrossRef] [Green Version]
- Munsat, T.L.; Davies, K.E. International SMA Consortium Meeting (26–28 June 1992, Bonn, Germany). Neuromuscul. Disord. 1992, 2, 423–428. [Google Scholar] [CrossRef]
- Zerres, K.; Rudnik-Schoneborn, S. Natural history in proximal spinal muscular atrophy. Clinical analysis of 445 patients and suggestions for a modification of existing classifications. Arch. Neurol. 1995, 52, 518–523. [Google Scholar] [CrossRef] [PubMed]
- Van den Berg, B.; Brouwer, W.B.; Koopmanschap, M.A. Economic valuation of informal care. An overview of methods and applications. Eur. J. Health Econ. 2004, 5, 36–45. [Google Scholar] [CrossRef] [PubMed]
- Arora, S.; Goodall, S.; Viney, R.; Einfeld, S. Using Discrete-Choice Experiment Methods to Estimate the Value of Informal Care: The Case of Children with Intellectual Disability. Pharm. Econ. 2018, 37, 501–511. [Google Scholar] [CrossRef]
- Eckert, L.; Gupta, S.; Amand, C.; Gadkari, A.; Mahajan, P.; Gelfand, J.M. The burden of atopic dermatitis in US adults: Health care resource utilization data from the 2013 National Health and Wellness Survey. J. Am. Acad. Dermatol. 2018, 78, 54–61.e1. [Google Scholar] [CrossRef]
- Johansson, G.; Mushnikov, V.; Bäckström, T.; Engström, A.; Khalid, J.M.; Wall, J.; Hoti, F. Exacerbations and healthcare resource utilization among COPD patients in a Swedish registry-based nation-wide study. BMC Pulm. Med. 2018, 18, 17. [Google Scholar] [CrossRef] [Green Version]
- Murage, M.J.; Anderson, A.; Oliveria, S.A.; Casso, D.; Ojeh, C.K.; Muram, T.M.; Merola, J.F.; Araujo, A.B. Healthcare resource utilization and costs among psoriasis patients treated with biologics, overall and by disease severity. J. Med. Econ. 2018, 21, 745–754. [Google Scholar] [CrossRef]
- Oldenkamp, M.; Bültmann, U.; Wittek, R.P.; Stolk, R.P.; Hagedoorn, M.; Smidt, N. Combining informal care and paid work: The use of work arrangements by working adult-child caregivers in the Netherlands. Heal. Soc. Care Community 2017, 26, e122–e131. [Google Scholar] [CrossRef] [Green Version]
- Hébert, R.; Bravo, G.; Préville, M. Reliability, Validity and Reference Values of the Zarit Burden Interview for Assessing Informal Caregivers of Community-Dwelling Older Persons with Dementia. Can. J. Aging/La Rev. Can. Vieil. 2000, 19, 494–507. [Google Scholar] [CrossRef]
- Van den Berg, B.; Spauwen, P. Measurement of informal care: An empirical study into the valid measurement of time spent on informal caregiving. Health Econ. 2006, 15, 447–460. [Google Scholar] [CrossRef] [Green Version]
- Cavazza, M.; BURQOL-RD Research Network; Kodra, Y.; Armeni, P.; De Santis, M.; López-Bastida, J.; Linertová, R.; Oliva-Moreno, J.; Serrano-Aguilar, P.; Posada, M.; et al. Social/economic costs and health-related quality of life in patients with Duchenne muscular dystrophy in Europe. Eur. J. Health Econ. 2016, 17, 19–29. [Google Scholar] [CrossRef]
- Péntek, M.; Gulácsi, L.; Brodszky, V.; Baji, P.; Boncz, I.; Pogány, G.; López-Bastida, J.; Linertová, R.; Oliva-Moreno, J.; Serrano-Aguilar, P.; et al. Social/economic costs and health-related quality of life of mucopolysaccharidosis patients and their caregivers in Europe. Eur. J. Health Econ. 2016, 17, 89–98. [Google Scholar] [CrossRef]
- Kuhlmann, A.; the BURQOL-RD Research Network; Schmidt, T.; Treskova, M.; López-Bastida, J.; Linertová, R.; Oliva-Moreno, J.; Serrano-Aguilar, P.; Posada, M.; Kanavos, P.; et al. Social/economic costs and health-related quality of life in patients with juvenile idiopathic arthritis in Europe. Eur. J. Health Econ. 2016, 17, 79–87. [Google Scholar] [CrossRef]
- Oliva-Moreno, J.; Pena-Longobardo, L.M.; Mar, J.; Masjuan, J.; Soulard, S.; Gonzalez-Rojas, N.; Becerra, V.; Casado, M.A.; Torres, C.; Yebenes, M.; et al. Determinants of Informal Care, Burden, and Risk of Burnout in Caregivers of Stroke Survivors: The CONOCES Study. Stroke 2018, 49, 140–146. [Google Scholar] [CrossRef]
- Olazarán, J.; Agüera-Ortiz, L.; Argimón, J.M.; Reed, C.; Ciudad, A.; Andrade, P.; Dilla, T. Costs and quality of life in community-dwelling patients with Alzheimer’s disease in Spain: Results from the GERAS II observational study. Int. Psychogeriatr. 2017, 29, 2081–2093. [Google Scholar] [CrossRef] [PubMed]
- Rapp, T.; Andrieu, S.; Chartier, F.; Deberdt, W.; Reed, C.; Belger, M.; Vellas, B. Resource Use and Cost of Alzheimer’s Disease in France: 18-Month Results from the GERAS Observational Study. Value Health 2018, 21, 295–303. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Farrar, M.A.; Carey, K.A.; Paguinto, S.G.; Chambers, G.; Kasparian, N.A. Financial, opportunity and psychosocial costs of spinal muscular atrophy: An exploratory qualitative analysis of Australian carer perspectives. BMJ Open 2018, 8, e020907. [Google Scholar] [CrossRef] [PubMed]
- Von Gontard, A.; Rudnik-Schoneborn, S.; Zerres, K. Stress and coping in parents of children and adolescents with spinal muscular atrophy. Klin. Padiatr. 2012, 224, 247–251. [Google Scholar] [CrossRef]
- Wan, H.W.Y.; Carey, K.A.; D’Silva, A.; Kasparian, N.A.; Farrar, M.A. “Getting ready for the adult”: How adults with spinal muscular atrophy perceive and experience healthcare, transition and well-being. Orphanet J. Rare Dis. 2019, 14, 1–13. [Google Scholar] [CrossRef]
- Brazil, K.; Kaasalainen, S.; Williams, A.; Dumont, S. A Comparison of Support Needs Between Rural and Urban Family Caregivers Providing Palliative Care. Am. J. Hosp. Palliat. Med. 2013, 31, 13–19. [Google Scholar] [CrossRef]
- Brazil, K.; Kaasalainen, S.; Williams, A.; Rodriguez, C. Comparing the experiences of rural and urban family caregivers of the terminally ill. Rural. Remote. Health 2013, 13, 2250. [Google Scholar] [PubMed]
- Rodwell, C.; Aymé, S. Rare disease policies to improve care for patients in Europe. Biochim. Biophys. Acta (BBA)-Mol. Basis Dis. 2015, 1852, 2329–2335. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Evangelista, T.; Hedley, V.; Atalaia, A.; Johnson, M.; Lynn, S.; Le Cam, Y.; Bushby, K. The context for the thematic grouping of rare diseases to facilitate the establishment of European Reference Networks. Orphanet J. Rare Dis. 2016, 11, 1–8. [Google Scholar] [CrossRef] [PubMed] [Green Version]
- Cannizzo, S.; Lorenzoni, V.; Palla, I.; Pirri, S.; Trieste, L.; Triulzi, I.; Turchetti, G. Rare diseases under different levels of economic analysis: Current activities, challenges and perspectives. RMD Open 2018, 4, e000794. [Google Scholar] [CrossRef]
- Goodrich, K.; Kaambwa, B.; Al-Janabi, H. The inclusion of informal care in applied economic evaluation: A review. Value Health 2012, 15, 975–981. [Google Scholar] [CrossRef] [Green Version]
- De Vries, L.M.; Van Baal, P.H.M.; Brouwer, W.B.F. Future Costs in Cost-Effectiveness Analyses: Past, Present, Future. Pharm. Econ. 2019, 37, 119–130. [Google Scholar] [CrossRef] [Green Version]
- Jiménez-Martín, S.; Prieto, C.V. The trade-off between formal and informal care in Spain. Eur. J. Health Econ. 2012, 13, 461–490. [Google Scholar] [CrossRef]
- Muramatsu, N.; Yin, H.; Campbell, R.T.; Hoyem, R.L.; Jacob, M.A.; Ross, C.O. Risk of nursing home admission among older americans: Does states’ spending on home- and community-based services matter? J. Gerontol. B. Psychol. Sci. Soc. Sci. 2007, 62, S169–S178. [Google Scholar] [CrossRef]
- Viitanen, T.K. Informal and Formal Care in Europe; Institute for the Study of Labor: Bonn, Germany, 2007; Available online: http://ftp.iza.org/dp2648.pdf (accessed on 23 October 2020).
- European Commission. The 2018 Ageing Report: Economic and Budgetary Projections for the EU Member States (2016–2070); Publications Office of the European Union: Luxembourg, 2018. [Google Scholar]
- Finkel, R.S.; Mercuri, E.; Darras, B.T.; Connolly, A.M.; Kuntz, N.L.; Kirschner, J.; Chiriboga, C.A.; Saito, K.; Servais, L.; Tizzano, E.; et al. Nusinersen versus Sham Control in Infantile-Onset Spinal Muscular Atrophy. New Engl. J. Med. 2017, 377, 1723–1732. [Google Scholar] [CrossRef] [Green Version]
- Mendell, J.R.; Al-Zaidy, S.; Shell, R.; Arnold, W.D.; Rodino-Klapac, L.R.; Prior, T.W.; Lowes, L.; Alfano, L.; Berry, K.; Church, K.; et al. Single-Dose Gene-Replacement Therapy for Spinal Muscular Atrophy. New Engl. J. Med. 2017, 377, 1713–1722. [Google Scholar] [CrossRef]
- Keam, S.J. Risdiplam: First Approval. Drugs 2020, 80, 1853–1858. [Google Scholar] [CrossRef]
- Tizzano, E.F.; Finkel, R. Spinal muscular atrophy: A changing phenotype beyond the clinical trials. Neuromuscul. Disord. 2017, 27, 883–889. [Google Scholar] [CrossRef] [PubMed]
- Mercuri, E.; Finkel, R.S.; Muntoni, F.; Wirth, B.; Montes, J.; Main, M.; Mazzone, E.S.; Vitale, M.; Snyder, B.; Quijano-Roy, S.; et al. Diagnosis and management of spinal muscular atrophy: Part 1: Recommendations for diagnosis, rehabilitation, orthopedic and nutritional care. Neuromuscul. Disord. NMD 2018, 28, 103–115. [Google Scholar] [CrossRef] [PubMed] [Green Version]
Characteristic/Outcome | Type I (n = 11) | Type II (n = 42) | Type III (n = 15) | Total (n = 68) |
---|---|---|---|---|
Caregivers | ||||
Sex female, n (%) | 8 (70) | 34 (81) | 13 (87) | 55 (81) |
Age a, mean (SD) | 42.1 (11.7) | 39.5 (9.0) | 39.3 (7.6) | 39.9 (9.1) |
Employed/self-employed, n (%) | 7 (64) | 24 (57) | 10 (66) | 41 (60) |
Marital status a, n (%) | ||||
Single | 7 (70) | 6 (14) | 1 (7) | 14 (21) |
Married or cohabiting | 3 (30) | 32 (76) | 13 (93) | 48 (73) |
Divorced/separated | 0 (0) | 4 (10) | 0 (0) | 4 (6) |
Zarit b, mean (SD) | 25.1 (23.2) | 34.8 (13.3) | 29.6 (17.1) | 31.9 (16.5) |
Patients | ||||
Sex female, n (%) | 9 (82) | 20 (48) | 7 (47) | 36 (53) |
Age c, mean (SD) | 9.8 (7.7) | 7.7 (4.6) | 6.6 (4.2) | 7.0 (5.2) |
Education d, n (%) | ||||
Educated at an ordinary school | 7 (64) | 3 (8) | 7 (46) | 17 (26) |
Educated at an ordinary centre with special support | 2 (18) | 22 (56) | 6 (40) | 30 (46) |
Educated at a special needs education centre | 0 (0) | 3 (8) | 0 (0) | 3 (5) |
Home schooled | 0 (0) | 1 (3) | 1 (7) | 2 (3) |
Nursery school | 0 (0) | 8 (21) | 1 (7) | 9 (14) |
Not receiving education | 2 (18) | 2 (5) | 0 (0) | 4 (6) |
Genetic test confirmation of SMA (yes), n % | 9 (82) | 35 (83) | 12 (80) | 56 (82) |
Activity | Type I | Type II | Type III | All | ||||
---|---|---|---|---|---|---|---|---|
Main Caregiver | All Caregivers | Main Caregiver | All Caregivers | Main Caregiver | All Caregivers | Main Caregiver | All Caregivers | |
Basic hygiene, dressing | 1.82 (1.01) | 3.23 (2.18) | 1.26 (1.11) | 1.62 (1.2) | 0.58 (0.36) | 0.91 (0.58) | 1.2 (1.04) | 1.73 (1.48) |
Feeding the patient | 1.61 (1.88) | 2.5 (2.67) | 1.33 (1.45) | 1.71 (1.92) | 0.45 (0.63) | 0.69 (1.09) | 1.18 (1.43) | 1.61 (1.97) |
Bathing or showering | 0.86 (0.53) | 1.61 (1.16) | 0.7 (0.6) | 1 (0.96) | 0.38 (0.36) | 0.71 (0.64) | 0.66 (0.56) | 1.03 (0.96) |
Helping the patient to move | 1.18 (0.9) | 2.05 (1.21) | 2.15 (2.38) | 2.75 (2.71) | 0.74 (1.32) | 1.39 (2.61) | 1.68 (2.08) | 2.34 (2.54) |
Total BADL | 5.47 (2.71) | 9.38 (4.77) | 5.44 (3.71) | 7.08 (4.42) | 2.15 (2.34) | 3.71 (4.52) | 4.72 (3.54) | 6.71 (4.78) |
Cooking and preparing special meals | 0.82 (0.72) | 1.55 (1.27) | 0.65 (0.82) | 0.91 (1.12) | 0.18 (0.36) | 0.25 (0.49) | 0.57 (0.75) | 0.87 (1.1) |
Administering drugs/minor cures | 1.18 (0.84) | 1.98 (1.58) | 1.07 (1.19) | 1.35 (1.47) | 0.64 (1.31) | 0.83 (1.53) | 0.99 (1.17) | 1.33 (1.52) |
Other IADL a | 0.03 (0.1) | 0.03 (0.1) | 0.02 (0.14) | 0.05 (0.3) | 0 (0) | 0 (0) | 0.02 (0.12) | 0.03 (0.24) |
Total IADL | 2.03 (1.12) | 3.55 (2.17) | 1.74 (1.59) | 2.31 (2.18) | 0.83 (1.3) | 1.08 (1.51) | 1.59 (1.5) | 2.24 (2.16) |
Other activities directly related to the disease | 0.8 (0.73) | 1.39 (1.25) | 0.68 (1.12) | 1.09 (1.72) | 0.17 (0.36) | 0.8 (1.49) | 0.59 (0.96) | 1.08 (1.59) |
All activities | 8.3 (3.6) | 14.32 (6.94) | 7.86 (4.8) | 10.48 (6.41) | 3.14 (2.42) | 5.59 (4.73) | 6.89 (4.62) | 10.02 (6.68) |
Informal Caregiving Time a (All Time of Care, Main Caregiver Plus Other Caregivers) | |||
High Burden of Care (more than 10 h per day) | Intermediate Burden of Care (between 5 and 10 h per day) | Low Burden of Care (less than 5 h per day) | |
dy/dx (Standard Error) | dy/dx (Standard Error) | dy/dx (Standard Error) | |
Type of SMA (reference category = type III) | |||
Type I | 0.36 * (0.17) | −0.05 (0.04) | −0.31 * (0.15) |
Type II | 0.30 ** (0.10) | −0.04 (0.04) | −0.26 ** (0.08) |
n | 64 | ||
LR chi2 | 35.9 | ||
Pseudo R2 | 0.26 | ||
Informal caregiving time a (main caregiver’s time) | |||
dy/dx (Standard Error) | dy/dx (Standard Error) | dy/dx (Standard Error) | |
Type of SMA (reference category = type III) | |||
Type I | 0.39 * (0.20) | −0.05 (0.05) | −0.34 * (0.17) |
Type II | 0.44 ** (0.12) | −0.06 (0.06) | −0.38 ** (0.09) |
n | 64 | ||
LR chi2 | 20.87 | ||
Pseudo R2 | 0.15 |
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Aranda-Reneo, I.; Peña-Longobardo, L.M.; Oliva-Moreno, J.; Litzkendorf, S.; Durand-Zaleski, I.; Tizzano, E.F.; López-Bastida, J. The Burden of Spinal Muscular Atrophy on Informal Caregivers. Int. J. Environ. Res. Public Health 2020, 17, 8989. https://doi.org/10.3390/ijerph17238989
Aranda-Reneo I, Peña-Longobardo LM, Oliva-Moreno J, Litzkendorf S, Durand-Zaleski I, Tizzano EF, López-Bastida J. The Burden of Spinal Muscular Atrophy on Informal Caregivers. International Journal of Environmental Research and Public Health. 2020; 17(23):8989. https://doi.org/10.3390/ijerph17238989
Chicago/Turabian StyleAranda-Reneo, Isaac, Luz María Peña-Longobardo, Juan Oliva-Moreno, Svenja Litzkendorf, Isabelle Durand-Zaleski, Eduardo F. Tizzano, and Julio López-Bastida. 2020. "The Burden of Spinal Muscular Atrophy on Informal Caregivers" International Journal of Environmental Research and Public Health 17, no. 23: 8989. https://doi.org/10.3390/ijerph17238989
APA StyleAranda-Reneo, I., Peña-Longobardo, L. M., Oliva-Moreno, J., Litzkendorf, S., Durand-Zaleski, I., Tizzano, E. F., & López-Bastida, J. (2020). The Burden of Spinal Muscular Atrophy on Informal Caregivers. International Journal of Environmental Research and Public Health, 17(23), 8989. https://doi.org/10.3390/ijerph17238989