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. 2024 Aug;41(8):3278-3298.
doi: 10.1007/s12325-024-02915-9. Epub 2024 Jul 3.

Eteplirsen Treatment for Duchenne Muscular Dystrophy: A Qualitative Patient Experience Study

Affiliations

Eteplirsen Treatment for Duchenne Muscular Dystrophy: A Qualitative Patient Experience Study

Joel Iff et al. Adv Ther. 2024 Aug.

Abstract

Introduction: Duchenne muscular dystrophy (DMD) is characterized by rapid functional decline. Current available treatment options aim to delay disease progression or stabilize physical function. To aid in healthcare providers' understanding of the symptoms of disease that impact patients' experience, this study explored children's physical functioning, activities of daily living (ADLs), and health-related quality of life (HRQoL) after receiving eteplirsen, a weekly infusion indicated for individuals with DMD with exon 51 skip-amenable mutations.

Methods: Fifteen caregivers of male individuals with DMD participated in a 60-min, semi-structured interview. Open-ended questioning explored changes in the children's condition or maintenance in abilities since eteplirsen initiation.

Results: Children with DMD (age 7-15 years [mean 10.9]; steroid treatment at interview, n = 8; time since eteplirsen initiation 3-24 months [mean 14.9]) were described by caregivers as ambulatory (n = 9) and non-ambulatory (n = 6). Caregivers of ambulatory children reported improvements or maintenance of walking ability (n = 7/9), running (n = 6/9), and using stairs (n = 4/9). Continued decline in using stairs was reported by two caregivers. In upper-limb functioning, improvements or maintenances in fine-motor movements were reported by nearly half of all caregivers (n = 7/15), with one caregiver noting a continued decline. Subsequent improvements or maintenances in ADLs were described. Improvements or maintenances in fatigue (n = 9/15), muscle weakness (n = 7/15), and pain (n = 6/15) were reported, although some caregivers described a continued decline (n = 3/15 fatigue, n = 1/15 muscle weakness, n = 2/15 pain). Importantly, most caregivers who reported maintenances in ability perceived this as a positive outcome (n = 6/9).

Conclusion: This exploratory study indicated that most caregivers perceived improvements or maintenances in aspects of their child's physical functioning, ADLs, and HRQoL since eteplirsen initiation, which they perceived to be a positive outcome.

Keywords: Duchenne muscular dystrophy; Eteplirsen; Health-related quality of life; Qualitative.

Plain language summary

Duchenne muscular dystrophy (DMD) is a rare disease characterized by progressive muscle weakness. Early on, this weakness presents as difficulty walking, but eventually children lose the ability to walk, develop spinal curvature, and experience problems with the heart and lung muscles. People with DMD are missing a key protein in their bodies called dystrophin. Eteplirsen is a weekly, intravenous treatment approved to treat people with a specific DMD genetic misspelling. The goal of the treatment is to slow down the disease and delay the time to losing ability to walk or needing help breathing. Fifteen caregivers of children living with DMD participated in a 60-min telephone interview. Caregivers were asked questions about the child’s DMD symptoms and how those symptoms impact the child’s daily life. Caregivers discussed their child’s experience while receiving eteplirsen treatment and changes since the start of treatment. Caregivers described their child’s muscle weakness and how this has affected their movements (e.g., using stairs, running or walking). Since starting eteplirsen treatment, all caregivers reported some improvement or maintenance in parts of their child’s physical functioning, activities of daily living (e.g., sports/leisure, getting dressed and self-care), and symptoms (e.g., muscle weakness, pain and fatigue), even though some decline was also reported (e.g., physical functioning, getting dressed, self-care, muscle weakness, pain and fatigue). The results provide insights into physical functioning and quality of life of children with DMD who are receiving eteplirsen. However, more research is needed to fully understand the impact of eteplirsen on these experiences.

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Conflict of interest statement

Joel Iff, Ihor Sehinovych, and Carolyn McNeill are employees of Sarepta Therapeutics, Inc., and may hold stock/options. Chloe Carmichael, Stephanie McKee, and Helen Kitchen are employees and stockholders of Clarivate, a healthcare research company that provides consultancy to pharmaceutical companies, including Sarepta Therapeutics, Inc. Carolina Tesi-Rocha reports consulting fees (Avexis, Biogen, Sarepta Therapeutics, Inc., Novartis, NS Pharma) and is a site investigator for clinical trials: Avexis, Biogen, Cytokinetics, Genzyme, Pfizer, PTC Therapeutics, Roche, Sarepta Therapeutics, Inc., Scholar Rock, and Janssen. Erik Henricson has received consulting fees from Sarepta Therapeutics, Inc., Santhera Pharmaceuticals, Pfizer, Eprirum Bio, Capricor, Catabasis, Mallinkrodt, Bristol-Myers Squibb, PTC Therapeutics, PepGen, and GSK and has received speaker honoraria from Parent Project Muscular Dystrophy, Muscular Dystrophy Association, and ENMC. Francesco Muntoni has received consultant fees and speaker honoraria from Sarepta Therapeutics, Inc. He is a member of the Pfizer SAB and, relevant for DMD, has received consultancies from Dyne Therapeutics, Roche, and PTC Therapeutics.

Figures

Fig. 1
Fig. 1
Relationship between time on eteplirsen treatment (months) and patient age at treatment initiation (years). *The age at treatment initiation starts at 5 years old. Although children with DMD were only eligible for the interview study if aged ≥ 7 years, they may have been receiving eteplirsen for 3–24 months prior
Fig. 2
Fig. 2
Changes in ambulatory status from treatment initiation to interview screening. Data confirmed by SareptAssist clinical records; At interview screening, caregivers were provided with the following brief, plain-language descriptions of the ambulatory stages as part of a demographic screener: early ambulatory (can walk and rise independently); late ambulatory (can walk but has lost the ability to rise); early non-ambulatory (loss of ability to walk but is not on ventilation); late non-ambulatory (loss of ability to walk and is on ventilation)
Fig. 3
Fig. 3
Thematic map of the Duchenne muscular dystrophy (DMD) patient experience. Arrows represent hypothesized relationships between concepts. ADLs activities of daily living, DMD Duchenne muscular dystrophy, HRQoL health-related quality of life
Fig. 4
Fig. 4
Physical functioning since eteplirsen initiation. *Changes to lower limb movements were only assessed for caregivers of children who were ambulatory at the time of interview (n = 9/15)
Fig. 5
Fig. 5
Activities of daily living and signs/symptoms of DMD since eteplirsen initiation. ADL activities of daily living, DMD Duchenne muscular dystrophy
Fig. 6
Fig. 6
Cognitive-behavioral functioning and emotional well-being concepts since eteplirsen initiation

References

    1. Ryder S, Leadley R, Armstrong N, et al. The burden, epidemiology, costs and treatment for Duchenne muscular dystrophy: an evidence review. Orphanet J Rare Dis. 2017;12:1–21. 10.1186/s13023-017-0631-3 - DOI - PMC - PubMed
    1. Broomfield J, Hill M, Guglieri M, Crowther M, Abrams K. Life expectancy in Duchenne muscular dystrophy: reproduced individual patient data meta-analysis. Neurology. 2021;97:e2304–14. 10.1212/WNL.0000000000012910 - DOI - PMC - PubMed
    1. Bach JR, Martinez D, Saulat B. Duchenne muscular dystrophy: the effect of glucocorticoids on ventilator use and ambulation. Am J Phys Med. 2010;89:620–4.10.1097/PHM.0b013e3181e72207 - DOI - PubMed
    1. Birnkrant DJ, Bushby K, Bann CM, et al. Diagnosis and management of Duchenne muscular dystrophy, part 1: diagnosis, and neuromuscular, rehabilitation, endocrine, and gastrointestinal and nutritional management. Lancet Neurol. 2018;17:251–67. 10.1016/S1474-4422(18)30024-3 - DOI - PMC - PubMed
    1. Powell PA, Carlton J. A comprehensive qualitative framework for health-related quality of life in Duchenne muscular dystrophy. Qual Life Res. 2023;32:1–12.10.1007/s11136-022-03240-w - DOI - PMC - PubMed