A beautiful struggle: Parent-perceived impact of short bowel syndrome on child and family wellbeing

Neumann, Marie L.; Allen, Jessica Y.; Kakani, Swapna; Ladner, Amy; Rauen, Meghan Hall; Weaver, Meaghann S.; Mercer, David F.

doi:10.1016/j.jpedsurg.2021.09.039

Cited by 9 publications

(16 citation statements)

References 46 publications

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“…Per la costruzione dei questionari si è partiti da un'analisi della letteratura che ha consentito di individuare le aree di QoL maggiormente indagate nelle rilevazioni effettuate con il setting di pazienti oggetto della ricerca. Tenuto conto del numero limitato di contributi sul tema e della variabilità di strumenti utilizzati nelle rilevazioni [12,18,19,[35][36][37], si è deciso di sviluppare un questionario ad hoc, limitato nel numero degli item e rivolto al paziente, cui è stato affiancato un secondo questionario rivolto al caregiver. Per la parte relativa alla perdita di produttività si è fatto invece riferimento ad altri strumenti, già validati, adattati per la patologia.…”

Section: Struttura Dell'indagine E Sviluppo Dei Questionariunclassified

[Burden of Short Bowel Syndrome in Italy: Direct and Indirect Costs and Quality of Life]

Mennini

Bini

Paoletti

et al. 2023

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OBJECTIVE: The Short Bowel Syndrome (SBS) is the most common cause of chronic intestinal failure (CIF) due to benign disease. The prevalence in Europe was estimated about 1.4 cases per million and few information are available for Italy. Home parenteral nutrition (HPN) is the primary and lifesaving treatment for patients with CIF. The parenteral nutrition (PN) has a great impact on the quality of life (QoL) of patients and the role of the caregiver is crucial for the disease management. The aim of this study was to evaluate the economic burden of SBS in Italy and to assess the impact of the disease and the parenteral nutrition on the quality of life of patients.METHODS: The total burden of SBS in Italy was assessed both in terms of costs and QoL using different tools. A prevalence-based cost of illness (COI) model was developed to estimate direct costs (PN cost, central venous catheter insertion cost, monitoring cost, hospitalization cost) and indirect costs (absenteeism, presenteeism, unemployment, abandonment of work due to the disease and economic benefits granted by the National Social Security Institute) associated with patient with SBS in Italy. The total annual costs were calculated considering a micro-costing approach, thus associating the average costs per patient with the prevalence of the disease. A systematic literature review (SLR) was conducted to collect epidemiological and direct cost data related to the patients with SBS. Hospital costs were estimated using the national Hospital Information System. Indirect costs were estimated using a human capital approach; therefore, the productivity loss was estimated both for patients and caregivers. A survey was conducted to obtain data about productivity loss and quality of life of patients and caregivers. The questionnaires were completed by clinicians, who were asked to indirectly report the experience of patients with SBS in parenteral nutrition currently being treated at their referral center. In a subsequent phase, a focus group was conducted to collect further information on QoL for patients and caregivers based on the experience of the KOLs involved. The QoL was evaluated considering a Likert scale.RESULTS: The prevalence of patients with gastrointestinal disease in HPN was estimated equal to 9.4 and 2.3 patients per million inhabitants for adults (age >18) and pediatric (age 0-18 years) patients, respectively. Knowing that SBS is the main cause of CIF due to benign disease, constituting 75% among adults and 56% among children, the number of adults with SBS in HPN present in Italy were 420, while the number of children with SBS in HPN were 77. Regarding direct costs, the mean total annual cost associated with adult and pediatric patient with SBS in Italy was estimated equal to € 36,434 and € 46,682, respectively. Parenteral nutrition accounted for 91% of the mean total cost estimated for the adult and for 87% of the mean total cost estimated for pediatric patient. Concerning indirect costs, the mean total annual cost per adult patient was estimated equal to € 51,093 (81% related to the productivity loss because of the abandonment of work due to the disease), while the mean total cost per pediatric patient was estimated equal to € 3,201 (60% related to caregiver’s presenteeism and 40% attributable to caregiver’s absenteeism). Finally, the average total annual cost per adult SBS patient in Italy was estimated at € 87,527 (42% of direct costs and 58% of indirect costs), whereas for pediatric patients it was estimated at € 49,882 (94% of direct costs and 6% of indirect costs). Overall, the weighted average cost for an SBS patient in Italy was estimated at € 81,712 (47% of direct costs and 53% of indirect costs). The analysis conducted on the QoL of patients with SBS in PN has shown that QoL was perceived as low (mean value equal to 5). The greatest impact on QoL was due to the disease (mean value equal to 9), while PN appeared to have less impact (mean value equal to 6).CONCLUSIONS: The analysis provides an estimate of the total burden associated with patients with SBS in Italy both in terms of cost and QoL. The cost associated with parenteral nutrition and indirect costs represent the main drivers of the total cost estimated for a patient with SBS in Italy. Based on the experience of the KOL involved in this study it was also found that the disease has a great impact on the QoL of these patients.

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Section: Struttura Dell'indagine E Sviluppo Dei Questionariunclassified

[Burden of Short Bowel Syndrome in Italy: Direct and Indirect Costs and Quality of Life]

Mennini

Bini

Paoletti

et al. 2023

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“…46 In the setting of SBS, a patient/family centered approach to research was performed to investigate quality of life and family management for children with SBS and their families. 50 The authors argue that their communitydriven research on quality of life contributes nuance to the narrative of quality of life for children with SBS and their families. Future research related to SBS should explicitly engage patients and caregivers as partners in knowledge production at every stage of the research process.…”

Section: Pfcc Outcome and Researchmentioning

confidence: 99%

“…The mandate for PCOR has gained momentum in recent years, particularly with the establishment of the Patient‐Centered Outcomes Research Institute in 2010 46 . In the setting of SBS, a patient/family centered approach to research was performed to investigate quality of life and family management for children with SBS and their families 50 . The authors argue that their community‐driven research on quality of life contributes nuance to the narrative of quality of life for children with SBS and their families.…”

Section: Pfcc Outcome and Researchmentioning

confidence: 99%

Advocating for a patient‐ and family centered care approach to management of short bowel syndrome

Kumpf

Neumann

Kakani³

2023

Nut in Clin Prac

Self Cite

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Patient-and family centered care (PFCC) is a model of providing healthcare that incorporates the preferences, needs, and values of the patient and their family and is built on a solid partnership between the healthcare team and patient/family. This partnership is critical in short bowel syndrome (SBS) management since the condition is rare, chronic, involves a heterogenous population, and calls for a personalized approach to care. Institutions can facilitate the practice of PFCC by supporting a teamwork approach to care, which, in the case of SBS, ideally involves a comprehensive intestinal rehabilitation program consisting of qualified healthcare practitioners who are supported with the necessary resources and budget. Clinicians can engage in a range of processes to center patients and families in the management of SBS, including fostering whole-person care, building partnerships with patients and families, cultivating communication, and providing information effectively. Empowering patients to self-manage important aspects of their condition is an important component of PFCC and can enhance coping to chronic disease.Therapy nonadherence represents a breakdown in the PFCC approach to care, especially when nonadherence is sustained, and the healthcare provider is intentionally misled. An individualized approach to care that incorporates patient/family priorities should ultimately enhance therapy adherence. Lastly, patients/families should play a central role in determining meaningful outcomes as it relates to PFCC and shaping the research that affects them. This review highlights needs and priorities of patients with SBS and their families and suggests ways to address gaps in existing care to improve outcomes.

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“…The beauty is seeing the amazing strength and tenacity of a tiny, fragile, human being and the compassion of a praying nurse, or a doctor that says they believe in and have seen miracles. The beauty is finding hope when all hope seems lost 47 …”

Section: Figurementioning

confidence: 99%

“…The beauty is finding hope when all hope seems lost. 47 TPN basically means when you are totally down and out, and probably facing death, it means a new beginning, a chance for a new beginning, and it gives you hope. It means life, yet it is not life as you knew it knew it before, and it's a whole different world.…”

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confidence: 99%

Rhoads Research Lecture—Reflections from a clinician scientist: The power of patient voice

Winkler

2022

J Parenter Enteral Nutr

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Qualitative research is a scientific method that systematically examines a phenomenon with the purpose of understanding and describing human experiences, exploring meanings and patterns, and illuminating the patient's lived experience. The Rhoads Research Lecture will highlight the power of patient voice and its importance to clinicians and researchers in addressing key clinical needs that are most relevant to patients receiving nutrition support. The subjective experience of patients who are dependent on home parenteral nutrition (HPN) will be shared, including how patients view HPN, define their quality of life (QOL), and describe the meaning of food in the context of being intravenously fed. As a result of these exploratory studies, the HPN patient‐reported outcome questionnaire (HPN‐PROQ) was developed and validated. Incorporating the HPN‐PROQ in practice empowers patients to identify and communicate QOL and HPN therapy goals and clinicians to delve deeper in the provision of holistic and empathetic care.

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A beautiful struggle: Parent-perceived impact of short bowel syndrome on child and family wellbeing

Cited by 9 publications

References 46 publications

[Burden of Short Bowel Syndrome in Italy: Direct and Indirect Costs and Quality of Life]

[Burden of Short Bowel Syndrome in Italy: Direct and Indirect Costs and Quality of Life]

Advocating for a patient‐ and family centered care approach to management of short bowel syndrome

Rhoads Research Lecture—Reflections from a clinician scientist: The power of patient voice

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