Title: Health-Related Quality of Life in Pediatric Patients with Intestinal Failure without
Neurodevelopmental Delay: A Systematic Review and Meta-Analysis
Version: 2 Date: 18 Nov 2024
Reviewer's report:
Authors
Milo et al present a systematic review with meta-analysis that aimed to evaluate the available
data on health-related quality of life (HRQoL) in pediatric patients with intestinal failure,
excluding those with neurodevelopmental delay, compared with healthy controls.
With a growing population of children with intestinal failure and long-term parenteral
nutrition needs, understanding the impact of this disease on the HRQoL of patients is
imperative. The authors attempted to summarize the available evidence, excluding patients with
neurodevelopmental delay, which could independently affect their HRQOL.
The manuscript is clear and easy to follow, with adequate methodological design.
I have the following comments and suggestions for the authors:
Comments:
Introduction: The authors claim that “improved neonatal and surgical care has led to increased
survival rates for children with chronic IF on home parenteral nutrition.” However, the reference
used to make this statement comes from a study that assessed the longitudinal development of
HRQoL in 35 children with intestinal failure. The following study could further support this
statement and I suggest adding it as part of the references:
“Gattini D, Roberts AJ, Wales PW, Beath SV, Evans HM, Hind J, Mercer D, Wong T, Yap
J, Belza C, Huysentruyt K, Avitzur Y. Trends in Pediatric Intestinal Failure: A Multicenter,
Multinational Study. J Pediatr. 2021 Oct;237:16-23.e4. doi: 10.1016/j.jpeds.2021.06.025. Epub
2021 Jun 18. PMID: 34153281.”
Introduction/Aims: As stated in the results and discussion, this systematic review and meta-
analysis aimed to investigate the health-related quality of life (HRQOL) of pediatric patients
with intestinal failure (IF) without neurodevelopmental delay, in comparison to healthy controls.
I suggest clarifying that the review intended to compare the HRQoL of children with IF with
healthy controls (and not with other chronic GI conditions for example).
Methods: This systematic review excluded studies involving patients with neurodevelopmental
delay. Although the authors state that this includes genetic, metabolic, or neurological disorders,
neurodevelopmental delay should be explicitly defined. What about studies that included
patients with mild neurodevelopmental delay? Were these patients/studies also excluded?
Methods: One of the inclusion criteria of the systematic review is studies that assess HRQoL
using validated instruments. It is important to describe what the authors consider a validated
instrument. Did they intend to include measurement tools that were previously reported in
pediatrics? Or instruments validated in children with intestinal failure? Or instruments with
evidence of psychometric properties such as reliability, validity, and responsiveness in any
pediatric disease?
Methods/Results: In the results, the authors describe that some studies were excluded because
they involved other chronic conditions. It is not clear if they are referring to studies that are
not conducted on children with intestinal failure, or if they excluded studies of children with
intestinal failure that had other chronic conditions (i.e kidney disease, heart disease, etc). This
could be further clarified on the methods/result section.
Methods: PRISMA recommends presenting the full search strategies for all databases, registers,
and websites, including any filters and limits used. This can be added as a supplementary file.
Results: In the Flow diagram (Figure 1), there is no mention of studies excluded because they
included children with neurodevelopmental delay. Please clarify if this is because they were
excluded in the initial title/abstract screening or if no studies were finally excluded because of
this reason. Is it possible that some studies included patients with neurodevelopmental delay,
but this was not explicitly stated on their methods/results? If so, this should be included in the
discussion.
Discussion: Most of the included studies use the PedQL as a tool to measure HRQOL. However,
this tool was developed for the general pediatric population and does not include some relevant
characteristics of children with IF, such as the presence of a feeding tube, a stoma, long-term use
of a central line and HPN, etc. This is a limitation of the studies included that could be included
in the discussion.
Conclusions: The following phrase in the conclusion is not supported by the results of the
systematic review and should be moved to the discussion: “Inpatient education programs that
incorporate recreational and social activities could provide academic instruction and alleviate
the stress associated with prolonged medical care and hospitalization. Additionally, initiatives
aimed at improving awareness and social competences, such as self-efficacy and social skills
training, could be highly beneficial for this population.”
Title: Health-Related Quality of Life in Pediatric Patients with Intestinal Failure without
Neurodevelopmental Delay: A Systematic Review and Meta-Analysis
Version: 2 Date: 18 Nov 2024
Reviewer's report:
Authors
Milo et al present a systematic review with meta-analysis that aimed to evaluate the available
data on health-related quality of life (HRQoL) in pediatric patients with intestinal failure,
excluding those with neurodevelopmental delay, compared with healthy controls.
With a growing population of children with intestinal failure and long-term parenteral
nutrition needs, understanding the impact of this disease on the HRQoL of patients is
imperative. The authors attempted to summarize the available evidence, excluding patients with
neurodevelopmental delay, which could independently affect their HRQOL.
The manuscript is clear and easy to follow, with adequate methodological design.
I have the following comments and suggestions for the authors:
Comments:
Introduction: The authors claim that “improved neonatal and surgical care has led to increased
survival rates for children with chronic IF on home parenteral nutrition.” However, the reference
used to make this statement comes from a study that assessed the longitudinal development of
HRQoL in 35 children with intestinal failure. The following study could further support this
statement and I suggest adding it as part of the references:
“Gattini D, Roberts AJ, Wales PW, Beath SV, Evans HM, Hind J, Mercer D, Wong T, Yap
J, Belza C, Huysentruyt K, Avitzur Y. Trends in Pediatric Intestinal Failure: A Multicenter,
Multinational Study. J Pediatr. 2021 Oct;237:16-23.e4. doi: 10.1016/j.jpeds.2021.06.025. Epub
2021 Jun 18. PMID: 34153281.”
Introduction/Aims: As stated in the results and discussion, this systematic review and meta-
analysis aimed to investigate the health-related quality of life (HRQOL) of pediatric patients
with intestinal failure (IF) without neurodevelopmental delay, in comparison to healthy controls.
I suggest clarifying that the review intended to compare the HRQoL of children with IF with
healthy controls (and not with other chronic GI conditions for example).
Methods: This systematic review excluded studies involving patients with neurodevelopmental
delay. Although the authors state that this includes genetic, metabolic, or neurological disorders,
neurodevelopmental delay should be explicitly defined. What about studies that included
patients with mild neurodevelopmental delay? Were these patients/studies also excluded?
Methods: One of the inclusion criteria of the systematic review is studies that assess HRQoL
using validated instruments. It is important to describe what the authors consider a validated
instrument. Did they intend to include measurement tools that were previously reported in
pediatrics? Or instruments validated in children with intestinal failure? Or instruments with
evidence of psychometric properties such as reliability, validity, and responsiveness in any
pediatric disease?
Methods/Results: In the results, the authors describe that some studies were excluded because
they involved other chronic conditions. It is not clear if they are referring to studies that are
not conducted on children with intestinal failure, or if they excluded studies of children with
intestinal failure that had other chronic conditions (i.e kidney disease, heart disease, etc). This
could be further clarified on the methods/result section.
Methods: PRISMA recommends presenting the full search strategies for all databases, registers,
and websites, including any filters and limits used. This can be added as a supplementary file.
Results: In the Flow diagram (Figure 1), there is no mention of studies excluded because they
included children with neurodevelopmental delay. Please clarify if this is because they were
excluded in the initial title/abstract screening or if no studies were finally excluded because of
this reason. Is it possible that some studies included patients with neurodevelopmental delay,
but this was not explicitly stated on their methods/results? If so, this should be included in the
discussion.
Discussion: Most of the included studies use the PedQL as a tool to measure HRQOL. However,
this tool was developed for the general pediatric population and does not include some relevant
characteristics of children with IF, such as the presence of a feeding tube, a stoma, long-term use
of a central line and HPN, etc. This is a limitation of the studies included that could be included
in the discussion.
Conclusions: The following phrase in the conclusion is not supported by the results of the
systematic review and should be moved to the discussion: “Inpatient education programs that
incorporate recreational and social activities could provide academic instruction and alleviate
the stress associated with prolonged medical care and hospitalization. Additionally, initiatives
aimed at improving awareness and social competences, such as self-efficacy and social skills
training, could be highly beneficial for this population.”
Please review this paper.
user
Title: Health-Related Quality of Life in Pediatric Patients with Intestinal Failure without
Neurodevelopmental Delay: A Systematic Review and Meta-Analysis
Version: 2 Date: 18 Nov 2024
Reviewer's report:
Authors
Milo et al present a systematic review with meta-analysis that aimed to evaluate the available
data on health-related quality of life (HRQoL) in pediatric patients with intestinal failure,
excluding those with neurodevelopmental delay, compared with healthy controls.
With a growing population of children with intestinal failure and long-term parenteral
nutrition needs, understanding the impact of this disease on the HRQoL of patients is
imperative. The authors attempted to summarize the available evidence, excluding patients with
neurodevelopmental delay, which could independently affect their HRQOL.
The manuscript is clear and easy to follow, with adequate methodological design.
I have the following comments and suggestions for the authors:
Comments:
Introduction: The authors claim that “improved neonatal and surgical care has led to increased
survival rates for children with chronic IF on home parenteral nutrition.” However, the reference
used to make this statement comes from a study that assessed the longitudinal development of
HRQoL in 35 children with intestinal failure. The following study could further support this
statement and I suggest adding it as part of the references:
“Gattini D, Roberts AJ, Wales PW, Beath SV, Evans HM, Hind J, Mercer D, Wong T, Yap
J, Belza C, Huysentruyt K, Avitzur Y. Trends in Pediatric Intestinal Failure: A Multicenter,
Multinational Study. J Pediatr. 2021 Oct;237:16-23.e4. doi: 10.1016/j.jpeds.2021.06.025. Epub
2021 Jun 18. PMID: 34153281.”
Introduction/Aims: As stated in the results and discussion, this systematic review and meta-
analysis aimed to investigate the health-related quality of life (HRQOL) of pediatric patients
with intestinal failure (IF) without neurodevelopmental delay, in comparison to healthy controls.
I suggest clarifying that the review intended to compare the HRQoL of children with IF with
healthy controls (and not with other chronic GI conditions for example).
Methods: This systematic review excluded studies involving patients with neurodevelopmental
delay. Although the authors state that this includes genetic, metabolic, or neurological disorders,
neurodevelopmental delay should be explicitly defined. What about studies that included
patients with mild neurodevelopmental delay? Were these patients/studies also excluded?
Methods: One of the inclusion criteria of the systematic review is studies that assess HRQoL
using validated instruments. It is important to describe what the authors consider a validated
instrument. Did they intend to include measurement tools that were previously reported in
pediatrics? Or instruments validated in children with intestinal failure? Or instruments with
evidence of psychometric properties such as reliability, validity, and responsiveness in any
pediatric disease?
Methods/Results: In the results, the authors describe that some studies were excluded because
they involved other chronic conditions. It is not clear if they are referring to studies that are
not conducted on children with intestinal failure, or if they excluded studies of children with
intestinal failure that had other chronic conditions (i.e kidney disease, heart disease, etc). This
could be further clarified on the methods/result section.
Methods: PRISMA recommends presenting the full search strategies for all databases, registers,
and websites, including any filters and limits used. This can be added as a supplementary file.
Results: In the Flow diagram (Figure 1), there is no mention of studies excluded because they
included children with neurodevelopmental delay. Please clarify if this is because they were
excluded in the initial title/abstract screening or if no studies were finally excluded because of
this reason. Is it possible that some studies included patients with neurodevelopmental delay,
but this was not explicitly stated on their methods/results? If so, this should be included in the
discussion.
Discussion: Most of the included studies use the PedQL as a tool to measure HRQOL. However,
this tool was developed for the general pediatric population and does not include some relevant
characteristics of children with IF, such as the presence of a feeding tube, a stoma, long-term use
of a central line and HPN, etc. This is a limitation of the studies included that could be included
in the discussion.
Conclusions: The following phrase in the conclusion is not supported by the results of the
systematic review and should be moved to the discussion: “Inpatient education programs that
incorporate recreational and social activities could provide academic instruction and alleviate
the stress associated with prolonged medical care and hospitalization. Additionally, initiatives
aimed at improving awareness and social competences, such as self-efficacy and social skills
training, could be highly beneficial for this population.”
assistant