Epidemiology of pediatric schistosomiasis in hard-to-reach areas and populations: A scoping review protocol

Phyllis Munyiva Isaiah; Marta Sólveig Palmeirim; Peter Steinmann

doi:10.12688/f1000research.126884.1

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Epidemiology of pediatric schistosomiasis in hard-to-reach areas and populations: A scoping review protocol

[version 1; peer review: 2 approved with reservations]

Phyllis Munyiva Isaiah ^1,2, Marta Sólveig Palmeirim^1,2, Peter Steinmann^1,2

PUBLISHED 21 Oct 2022

Author details Author details

¹ Swiss Centre for International Health, Swiss Tropical and Public Health Institute, Allschwil, Switzerland
² University of Basel, Basel, Switzerland

Phyllis Munyiva Isaiah
Roles: Conceptualization, Methodology, Visualization, Writing – Original Draft Preparation, Writing – Review & Editing

Marta Sólveig Palmeirim
Roles: Conceptualization, Writing – Review & Editing

Peter Steinmann
Roles: Conceptualization, Supervision, Writing – Review & Editing

OPEN PEER REVIEW

REVIEWER STATUS

This article is included in the Sociology of Health gateway.

This article is included in the Neglected Tropical Diseases collection.

Abstract

Background: Schistosomiasis is a neglected tropical disease (NTD) that affects millions of people. Children are the most vulnerable group to developing overt disease. An estimated 779 million people are at risk of schistosomiasis and 50 million preschool-age children (PSAC) need treatment. PSAC are not currently targeted by national chemotherapy campaigns due to a lack of suitable pediatric formulations of praziquantel. The Pediatric Praziquantel Consortium has developed an orally dispersible praziquantel formulation (arpraziquantel) and is facilitating its adoption for schistosomiasis control by endemic countries through the ADOPT program – an implementation research program that paves the way for the large-scale delivery of the child-friendly formulation to treat schistosomiasis in preschool-aged children in endemic countries. A key challenge for comprehensive NTD control including schistosomiasis is reaching all at-risk populations, including those hard to reach. Main access barriers include geographic, social and economic conditions.
Objective: This scoping literature review aims to document the epidemiology of schistosomiasis in children under 6 years of age living in hard-to-reach areas and populations.
Methods: This review will adopt the five-stage scoping review process of identifying the research question, identifying relevant studies, study selection, charting data and collating, summarizing and reporting results. Electronic databases including Medline, Web of Science, Embase (Ovid), LILACS and African Journals OnLine (AJOL) will be searched for relevant articles. Two independent reviewers will screen identified articles using a two-stage approach of reviewing the title/abstract and then the full text of provisionally retained articles. Relevant literatures will be downloaded into EndNote X9 to maintain and manage citation and facilitate the overall review process. A meta-analysis will be conducted if indicated.
Relevance: The results will provide insights into the burden of schistosomiasis among marginalized PSAC, aiming to produce evidence on the need for inclusion of this population when designing the expansion of preventive chemotherapy programs.

Keywords

Schistosomiasis, Prevalence, Epidemiology, Pre-School Aged Children, Pediatric, Hard-to-reach, Praziquantel

Corresponding author: Phyllis Munyiva Isaiah

Competing interests: No competing interests were disclosed.

Grant information: The authors declare that no grants were involved in supporting this work. The first author is the recipient of a personal stipend by the Amt für Ausbildungsbeiträge (AfA) Basel-Stadt. The last author is a member of the Pediatric Praziquantel Consortium.
The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

Copyright: © 2022 Isaiah PM et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

How to cite: Isaiah PM, Palmeirim MS and Steinmann P. Epidemiology of pediatric schistosomiasis in hard-to-reach areas and populations: A scoping review protocol [version 1; peer review: 2 approved with reservations]. F1000Research 2022, 11:1203 (https://doi.org/10.12688/f1000research.126884.1) First published: 21 Oct 2022, 11:1203 (https://doi.org/10.12688/f1000research.126884.1) Latest published: 20 Jan 2023, 11:1203 (https://doi.org/10.12688/f1000research.126884.2)

Abbreviations and acronyms

DALYs: Disability-Adjusted Life Years

NTD: Neglected Tropical Disease

PSAC: Preschool-aged Children

S. haematobium: Schistosoma haematobium

S. mansoni: Schistosoma mansoni

SAC: School-aged children

SDG: Sustainable Development Goal

Introduction

Schistosomiasis is a neglected tropical disease (NTD) that affects millions of people worldwide. It is estimated that 779 million people are at risk of developing this disease.¹^,² Children are the most vulnerable group to developing overt disease with 50 million preschool-age (PSAC) children in need of treatment.³ Adults are most affected by the consequences of chronic infection.⁴ Schistosomiasis is cause by an infection with a Schistosoma species parasite.⁵ There are several species, the most common being S. mansoni and S. haematobium. In a human host, the adult parasites release eggs into the environment through faeces (S. mansoni) and urine (S. haematobium). An intermediate host snail living in stagnant water is involved in the transmission cycle. Human infections take place through skin penetration when in contact with stagnant water where Schistosoma spp. larvae are present. The disease mainly affects the poorest communities without access to safe water and improved sanitation, or exposed to water during occupational and domestic activities.⁶^,⁷ Symptoms of chronic schistosomiasis include anaemia, cognitive impairment, deficits in linear growth leading to chronic under-nutrition (stunting) as well as acute under-nutrition (wasting),⁵ genital lesions,⁸ and irreversible organ damage as a result of fibrosis.⁹ The morbidity caused by schistosomes is commonly associated with moderate-to-heavy intensities of infection as measured by the density of excreted eggs, and is progressive.¹⁰ In addition to these health consequences, schistosomiasis is associated with negative economic and social impacts.¹¹ Estimates indicate that schistosomiasis causes an annual loss of 4.5 million disability-adjusted life years (DALYs).¹²

Although NTD control programs have been established in many endemic countries, the rolling out of schistosomiasis control and elimination components is still limited.¹³ Periodic deworming with praziquantel, the strategy recommended by WHO for the control of schistosomiasis, is available primarily to school-aged children (SAC; 5-15 years old) who can be reached efficiently through school-based programs.¹⁰ In addition to previous studies,¹⁴ the 2022 WHO guideline on control and elimination of human schistosomiasis¹⁵ has identified important treatment gaps in this strategy, including that infected PSAC are largely left untreated.

This could be because for a long time the PSAC group has been categorized as a low risk group for schistosomiasis infection¹⁶ and its impact on the health of this age group, although unknown, is often considered negligible.¹⁷ In addition, logistical and operational difficulties in collecting samples from PSAC for diagnosis, especially in hard-to-reach areas, lack of sensitive diagnostics for light schistosomiasis infections and a paucity of data on risk factors in PSAC have further biased schistosomiasis research to focus on SAC and adults over the years.¹⁶ Lastly, current donations of the main drug to treat schistosomiasis – praziquantel – are restricted to SAC.

Treatment equity is not currently achieved for schistosomiasis control as hundreds of millions of the worlds’ most vulnerable, most disadvantaged people, including PSAC, are still left behind. This is true especially for people whose incomes are below the federal poverty threshold, who live in vulnerable social and economic situation such as undocumented persons, socially excluded groups due to language, religious and other societal barriers¹⁸ and in the remotest, hardest to reach parts of endemic countries. These hard-to-reach populations are often ethnic minorities, island and fishing communities and migrant populations and other minority or marginalized populations, hindering the attainment of the Sustainable Development Goal (SDG) 3 and the commitment of global leaders to ensure that “no one is left behind” from development progress.⁷^,¹⁹

Objective

This scoping literature review aims to document the epidemiology of pediatric schistosomiasis (children under 6 years of age) living in hard-to-reach areas and populations. In this review, hard-to-reach populations are defined following Shaghaghi et al., as: i) migrants/island and fishing communities/nomads, ii) those living in remote physical and geographical location, and iii) those living in vulnerable social and economic situation such as minority groups, undocumented persons, socially excluded groups due to language and religious barriers.¹⁸

The aim is to produce evidence on the need for inclusion of this population when designing the expansion of preventive chemotherapy to also cover those living in hard-to–reach areas. This is in accordance with the WHO guideline of 2022 on control and elimination of human schistosomiasis¹⁵ recommendation on expansion of preventive chemotherapy to cover all in need.

Review question

The condition, context, and population (CoCoPop) framework to inform the review objective is shown in Table 1.

Table 1. Condition, context, and population framework.

Condition	Context	Population
Schistosomiasis infection	Hard-to-reach populations	Children under 6 years

Methods

This review will adopt the five-stage scoping review process guideline recommended by Arksey et. al.,²⁰ taking into consideration the modifications recommended by Peters et al.²¹

Evidence searches

Electronic literature databases including Medline, Web of Science, Embase (Ovid), LILACS and African Journals OnLine (AJOL) will be searched for published scientific studies on pediatric schistosomiasis in hard-to reach areas, using a pre-determined search strategy (Table 2). With the guidance of a librarian (University Medical Library- University of Basel), we first developed and optimized a search strategy for PubMed. This search was then translated using the SR-accelerator tool²² developed by BOND university to generate the equivalent search terms for Embase (Ovid) and Web of Science.

Table 2. Searching databases and strategies.

a.) PubMed Search Query
(Schistosomiasis [Mesh] OR Schistosom*[tiab] OR Bilharzi*[tiab] OR “blood fluke*”[tiab] OR “snail fever*”[tiab] OR “Katayama fever*”[tiab]) AND (Child [Mesh] OR child [tw] OR children [tw] OR Infant [Mesh] OR infan*[tw] OR newborn*[tw] OR new-born*[tw] OR baby [tw] OR babies [tw] OR suckling*[tw] OR toddler*[tw] OR childhood [tw] OR schoolchild*[tw] OR childcare [tw] OR child-care [tw] OR young [ti] OR youngster*[tw] OR preschool [tw] OR pre-school [tw] OR kid [tw] OR kids [tw] OR boy [tw] OR boys [tw] OR girl*[tw] OR pre-adolescen*[tw] OR schoolage*[tw] OR school-age*[tw] OR schoolboy*[tw] OR schoolgirl*[tw] OR Pediatrics [Mesh] OR Pediatric*[tw] OR Paediatric*[tw] OR (child [all] NOT child [au]) OR children*[all] OR schoolchild*[all] OR “under 5”[tw] OR “<5 year*”[all] OR “<=5 year*”[all] OR infan*[all] OR pediat*[all] OR paediat*[all] OR neonat*[all] OR toddler*[all] OR preteen*[all] OR newborn*[all] OR postneonat*[all] OR postnatal*[all] OR puberty [all] OR preschool*[all] OR suckling*[all] OR juvenile [all] OR “new born*”[all] OR new-born*[all] OR neo-nat*[all] OR neonat*[all] OR perinat*[all] OR underag*[all] OR “under age”[all] OR “under aged”[all] OR youth*[all] OR kinder*[all] OR pubescen*[all] OR prepubescen*[all] OR prepuberty [all] OR “school age”[all] OR “stratified by age”[all] OR schoolage [all] OR “school ages”[all] OR schoolage*[all] OR “one year old”[ti] OR “two year old”[ti] OR “three year old”[ti] OR “four year old”[ti] OR “five year old”[ti] OR “six year old”[ti] OR “1 year old”[ti] OR “2 year old”[ti] OR “3 year old”[ti] OR “4 year old”[ti] OR “5 year old”[ti] OR “6 year old”[ti] OR “two years old”[ti] OR “three years old”[ti] OR “four years old”[ti] OR “five years old”[ti] OR “six years old”[ti] OR “2 years old”[ti] OR “3 years old”[ti] OR “4 years old”[ti] OR “5 years old”[ti] OR “6 years old”[ti]) NOT (animals [Mesh] NOT humans [Mesh])

b.) Web of Science Search Query
(ALL=Schistosomiasis OR (TI=Schistosom* OR AB=Schistosom*) OR (TI=Bilharzi* OR AB=Bilharzi*) OR (TI="blood fluke*" OR AB="blood fluke*") OR (TI="snail fever*" OR AB="snail fever*") OR (TI="Katayama fever*" OR AB="Katayama fever*")) AND (ALL=Child OR ALL=child OR ALL=children OR ALL=Infant OR ALL=infan* OR ALL=newborn* OR ALL=new-born* OR ALL=baby OR ALL=babies OR ALL=suckling* OR ALL=toddler* OR ALL=childhood OR ALL=schoolchild* OR ALL=childcare OR ALL=child-care OR TI=young OR ALL=youngster* OR ALL=preschool OR ALL=pre-school OR ALL=kid OR ALL=kids OR ALL=boy OR ALL=boys OR ALL=girl* OR ALL=pre-adolescen* OR ALL=schoolage* OR ALL=school-age* OR ALL=schoolboy* OR ALL=schoolgirl* OR ALL=Pediatrics OR ALL=Pediatric* OR ALL=Paediatric* OR (ALL=child NOT AU=child) OR ALL=children* OR ALL=schoolchild* OR ALL="under 5" OR ALL="<5 year*" OR ALL="<=5 year*" OR ALL=infan* OR ALL=pediat* OR ALL=paediat* OR ALL=neonat* OR ALL=toddler* OR ALL=preteen* OR ALL=newborn* OR ALL=postneonat* OR ALL=postnatal* OR ALL=puberty OR ALL=preschool* OR ALL=suckling* OR ALL=juvenile OR ALL="new born*" OR ALL=new-born* OR ALL=neo-nat* OR ALL=neonat* OR ALL=perinat* OR ALL=underag* OR ALL="under age" OR ALL="under aged" OR ALL=youth* OR ALL=kinder* OR ALL=pubescen* OR ALL=prepubescen* OR ALL=prepuberty OR ALL="school age" OR ALL="stratified by age" OR ALL=schoolage OR ALL="school ages" OR ALL=schoolage* OR TI="one year old" OR TI="two year old" OR TI="three year old" OR TI="four year old" OR TI="five year old" OR TI="six year old" OR TI="1 year old" OR TI="2 year old" OR TI="3 year old" OR TI="4 year old" OR TI="5 year old" OR TI="6 year old" OR TI="two years old" OR TI="three years old" OR TI="four years old" OR TI="five years old" OR TI="six years old" OR TI="2 years old" OR TI="3 years old" OR TI="4 years old" OR TI="5 years old" OR TI="6 years old") NOT (ALL=animals NOT ALL=humans)

C.) Embase (Ovid) Search Strategy
(exp Schistosomiasis/ OR Schistosom*.tw. OR Bilharzi*.tw. OR “blood fluke*”.tw. OR “snail fever*”.tw. OR “Katayama fever*”.tw.) AND (exp Child/ OR child.mp. OR children.mp. OR exp Infant/ OR infan*.mp. OR newborn*.mp. OR new-born*.mp. OR baby.mp. OR babies.mp. OR suckling*.mp. OR toddler*.mp. OR childhood.mp. OR schoolchild*.mp. OR childcare.mp. OR child-care.mp. OR young.ti. OR youngster*.mp. OR preschool.mp. OR pre-school.mp. OR kid.mp. OR kids.mp. OR boy.mp. OR boys.mp. OR girl*.mp. OR pre-adolescen*.mp. OR schoolage*.mp. OR school-age*.mp. OR schoolboy*.mp. OR schoolgirl*.mp. OR exp Pediatrics/OR Pediatric*.mp. OR Paediatric*.mp. OR (child.af. NOT child.au.) OR children*.af. OR schoolchild*.af. OR “under 5”.mp. OR “<5 year*”.af. OR “<=5 year*”.af. OR infan*.af. OR pediat*.af. OR paediat*.af. OR neonat*.af. OR toddler*.af. OR preteen*.af. OR newborn*.af. OR postneonat*.af. OR postnatal*.af. OR puberty.af. OR preschool*.af. OR suckling*.af. OR juvenile.af. OR “new born*”.af. OR new-born*.af. OR neo-nat*.af. OR neonat*.af. OR perinat*.af. OR underag*.af. OR “under age”.af. OR “under aged”.af. OR youth*.af. OR kinder*.af. OR pubescen*.af. OR prepubescen*.af. OR prepuberty.af. OR “school age”.af. OR “stratified by age”.af. OR schoolage.af. OR “school ages”.af. OR schoolage*.af. OR “one year old”.ti. OR “two year old”.ti. OR “three year old”.ti. OR “four year old”.ti. OR “five year old”.ti. OR “six year old”.ti. OR “1 year old”.ti. OR “2 year old”.ti. OR “3 year old”.ti. OR “4 year old”.ti. OR “5 year old”.ti. OR “6 year old”.ti. OR “two years old”.ti. OR “three years old”.ti. OR “four years old”.ti. OR “five years old”.ti. OR “six years old”.ti. OR “2 years old”.ti. OR “3 years old”.ti. OR “4 years old”.ti. OR “5 years old”.ti. OR “6 years old”.ti.) NOT (exp animals/NOT exp humans/)

d.) AJOL Search Strategy
‘schistosomiasis AND children’

e.) LILACS search Strategy
schistosomiasis AND (children OR Preschool child*)

Published grey literature, WHO literature databases and reports, and documentation obtained from schistosomiasis experts working in relevant organizations such as Kenya Medical Research Institute (KEMRI), Schistosomiasis Control Initiative Foundation (SCIF), Schistosomiasis Consortium for Operational Research and Evaluation (SCORE), the Foundation for Innovative New Diagnostics (FIND), Sight Savers, Hellen Keller and Merck KGaA will also be included in this review. Experts will be actively contacted with an invitation to share relevant documents and references.

All identified references will be screened independently by two reviewers (Phyllis Munyiva Isaiah and Marta Palmeirim) using a two-stage approach.

First, a manual search will be conducted on the initial hit list by reviewing the title and abstracts to identify schistosomiasis studies conducted among PSAC. Second, we will review the full texts of the shortlisted articles to identify studies conducted in hard-to-reach areas and populations.

Additional references will then be retrieved by manually searching the bibliographies of identified articles. All relevant literatures will be downloaded into EndNote X9 to maintain and manage citation and facilitate the overall review process.

Inclusion/exclusion criteria

We will include cross-sectional, cohort and case control studies on schistosomiasis in children under 6 years old and living in hard-to-reach areas or belonging to hard-to-reach populations. Similar studies done on women of reproductive age and adults; or studies that did not apply cross-sectional or cohort or case control design (e.g. case reports) will be excluded from the review.

Risk of bias and quality assessment

Reviewers will assess all included studies independently for possible bias by using the Joanna Briggs Institute (JBI) Prevalence Critical Appraisal Tool.²³ All selected studies will be assessed using the 10 quality control items suggested by the tool. A score of 1 will be awarded for each item fulfilled while a 0 score will be awarded for each unfilled control item. Score aggregates will then be generated and converted into a low, moderate and high quality classification.²⁴ Poor quality studies will be excluded, clearly documenting the reason for exclusion.

Data extraction and synthesis

Data will be extracted from included documents and exported to a predefined summary template in Microsoft excel 2016. Extracted data will include:

i. Bibliographic information (first author, journal/document, year of publication)
ii. Year and country of study
iii. Sample size
iv. Study population age and sex
v. Prevalence and/or incidence of schistosomiasis
vi. Mean eggs per gram of feces or infection intensity classification (if available)
vii. Specific schistosome species observed
viii. Diagnostic method
ix. Type of hard-to-reach population/area

Data analysis

Extracted data will be analyzed using IBM SPSS statistics V.24. Descriptive statistics will be performed to allow for narrative synthesis. Weighted population mean outcomes will then be calculated for prevalence among PSAC.²⁵ To calculate pooled prevalence estimates (PPE), the inverse variance heterogeneity (IVhet) model²⁶ in MetaXL will be used for the selected studies, to ensure that statistical error is not underestimated.²⁴ The level of heterogeneity on selected studies will be evaluated using Cochran’s Q and I² statistics. This will be done by stratifying our data according to schistosomiasis prevalence and the region where the studies were conducted to determine heterogeneity between subgroups and within-groups.²⁴ Forest plots will be used to show the estimated prevalence (95% confidence interval).

Dissemination of results

All findings will be published in a scientific article in a peer reviewed journal. The findings of this review will be reported in accordance with Preferred Reporting Items for Systematic Reviews and Meta-Analyses–Extension for Scoping Reviews (PRISMA –ScR) guidelines.

Study status

Electronic databases have been searched using the above-mentioned search strategy for articles. The reviewers are currently screening the articles for duplicates.

Data availability

No data are associated with this article.

Acknowledgments

We acknowledge Dr. Thomas Fürst (University Medical Library- University of Basel) for his input and guidance in developing and optimizing search strategies used in this review.

References

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Version 2

VERSION 2 PUBLISHED 21 Oct 2022

Author details Author details

¹ Swiss Centre for International Health, Swiss Tropical and Public Health Institute, Allschwil, Switzerland
² University of Basel, Basel, Switzerland

Phyllis Munyiva Isaiah
Roles: Conceptualization, Methodology, Visualization, Writing – Original Draft Preparation, Writing – Review & Editing

Marta Sólveig Palmeirim
Roles: Conceptualization, Writing – Review & Editing

Peter Steinmann
Roles: Conceptualization, Supervision, Writing – Review & Editing

Competing interests

No competing interests were disclosed.

Grant information

The authors declare that no grants were involved in supporting this work. The first author is the recipient of a personal stipend by the Amt für Ausbildungsbeiträge (AfA) Basel-Stadt. The last author is a member of the Pediatric Praziquantel Consortium.
The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript.

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https://doi.org/10.12688/f1000research.126884.2

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© 2022 Isaiah PM et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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Isaiah PM, Palmeirim MS and Steinmann P. Epidemiology of pediatric schistosomiasis in hard-to-reach areas and populations: A scoping review protocol [version 1; peer review: 2 approved with reservations] F1000Research 2022, 11:1203 (https://doi.org/10.12688/f1000research.126884.1)

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Reviewer Report 10 Jan 2023

Zhou Xiao-Nong, Chinese Center for Disease Control and Prevention, National Institute of Parasitic Diseases, Shanghai, China; School of Global Health, Chinese Center for Tropical Dsieases Research, Shanghai Jiao Tong University School of Medicine, Shanghai, China

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https://doi.org/10.5256/f1000research.139341.r156448

The authors are trying to review epidemiology of pediatric schistosomiasis in hard-to-reach areas and populations, the results will provide evidences to designing the extension of preventive chemotherapy (PC) programs in the global schistosomiasis control program, by using the five-stage scoping review process. It is worthwhile for publication of the protocol due to big gaps are existed in the previous global NTDs program that focus only on the population over 6 years old. But there are several limitations to design the protocol based on the objectives of the study, which can overcome by revision of the study protocol.

The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.
The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.
In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.
In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.
Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Is the rationale for, and objectives of, the study clearly described?

Yes
Is the study design appropriate for the research question?

Partly
Are sufficient details of the methods provided to allow replication by others?

Partly
Are the datasets clearly presented in a useable and accessible format?

Partly

Competing Interests: No competing interests were disclosed.

Reviewer Expertise: Epidemiolgoy of schistosomiasis

I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard, however I have significant reservations, as outlined above.

CITE

Report a concern

Author Response 20 Jan 2023

Phyllis Isaiah, University of Basel, Basel, Switzerland

20 Jan 2023

Author Response

Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the ... Continue reading Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.

Question 2: The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.

Response: Thank you for pointing this out. This has been amended in the protocol.

Question 3: In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.

Response: Thank you for pointing this out. This has been amended in the protocol. We had only one objective.

Question 4: In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.

Response: Thank you for pointing this out. This has been amended in the protocol. An annex (a data collection table) has been attached.

Question 5: Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Response: Thank you for pointing this out. This has been amended in the protocol.
Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.

Question 2: The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.

Response: Thank you for pointing this out. This has been amended in the protocol.

Question 3: In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.

Response: Thank you for pointing this out. This has been amended in the protocol. We had only one objective.

Question 4: In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.

Response: Thank you for pointing this out. This has been amended in the protocol. An annex (a data collection table) has been attached.

Question 5: Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Response: Thank you for pointing this out. This has been amended in the protocol.
Competing Interests: No competing interests disclosed Close
Report a concern
Respond or Comment

COMMENTS ON THIS REPORT

Author Response 20 Jan 2023

Phyllis Isaiah, University of Basel, Basel, Switzerland

20 Jan 2023

Author Response

Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the ... Continue reading Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.

Question 2: The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.

Response: Thank you for pointing this out. This has been amended in the protocol.

Question 3: In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.

Response: Thank you for pointing this out. This has been amended in the protocol. We had only one objective.

Question 4: In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.

Response: Thank you for pointing this out. This has been amended in the protocol. An annex (a data collection table) has been attached.

Question 5: Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Response: Thank you for pointing this out. This has been amended in the protocol.
Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.

Question 2: The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.

Response: Thank you for pointing this out. This has been amended in the protocol.

Question 3: In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.

Response: Thank you for pointing this out. This has been amended in the protocol. We had only one objective.

Question 4: In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.

Response: Thank you for pointing this out. This has been amended in the protocol. An annex (a data collection table) has been attached.

Question 5: Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Response: Thank you for pointing this out. This has been amended in the protocol.
Competing Interests: No competing interests disclosed Close
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Views

Reviewer Report 15 Dec 2022

Dirk Engels, Uniting to Combat NTDs Support Centre, Geneva, Switzerland

Approved with Reservations

https://doi.org/10.5256/f1000research.139341.r156449

The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of pre-school age children to be reached. My main remark would be: with such a substantial scoping review, why limiting the study to hard-to-reach populations only? I suggest to expand to all eligible studies reporting on schistosomiasis in PSAC and treat hard-to-reach populations as a sub-analysis. In this way as-broad-as-possible information is captured in one scoping review, leaving all options open for an access plan

Is the rationale for, and objectives of, the study clearly described?

Yes
Is the study design appropriate for the research question?

Yes
Are sufficient details of the methods provided to allow replication by others?

Yes
Are the datasets clearly presented in a useable and accessible format?

Yes

Competing Interests: No competing interests were disclosed.

Reviewer Expertise: Tropical Diseases

CITE

Report a concern

Author Response 20 Jan 2023

Phyllis Isaiah, University of Basel, Basel, Switzerland

20 Jan 2023

Author Response

Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of ... Continue reading Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of pre-school age children to be reached. My main remark would be: with such a substantial scoping review, why limiting the study to hard-to-reach populations only? I suggest to expand to all eligible studies reporting on schistosomiasis in PSAC and treat hard-to-reach populations as a sub-analysis. In this way as-broad-as-possible information is captured in one scoping review, leaving all options open for an access plan

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.
Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of pre-school age children to be reached. My main remark would be: with such a substantial scoping review, why limiting the study to hard-to-reach populations only? I suggest to expand to all eligible studies reporting on schistosomiasis in PSAC and treat hard-to-reach populations as a sub-analysis. In this way as-broad-as-possible information is captured in one scoping review, leaving all options open for an access plan

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.
Competing Interests: No competing interests. Close
Report a concern
Respond or Comment

COMMENTS ON THIS REPORT

Author Response 20 Jan 2023

Phyllis Isaiah, University of Basel, Basel, Switzerland

20 Jan 2023

Author Response

Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of ... Continue reading Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of pre-school age children to be reached. My main remark would be: with such a substantial scoping review, why limiting the study to hard-to-reach populations only? I suggest to expand to all eligible studies reporting on schistosomiasis in PSAC and treat hard-to-reach populations as a sub-analysis. In this way as-broad-as-possible information is captured in one scoping review, leaving all options open for an access plan

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.
Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of pre-school age children to be reached. My main remark would be: with such a substantial scoping review, why limiting the study to hard-to-reach populations only? I suggest to expand to all eligible studies reporting on schistosomiasis in PSAC and treat hard-to-reach populations as a sub-analysis. In this way as-broad-as-possible information is captured in one scoping review, leaving all options open for an access plan

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.
Competing Interests: No competing interests. Close
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Version 2

VERSION 2 PUBLISHED 21 Oct 2022

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Dirk Engels, Uniting to Combat NTDs Support Centre, Geneva, Switzerland
Zhou Xiao-Nong, National Institute of Parasitic Diseases, Shanghai, China; Shanghai Jiao Tong University School of Medicine, Shanghai, China

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6 Views

03 Feb 2023 | for Version 2

Dirk Engels, Uniting to Combat NTDs Support Centre, Geneva, Switzerland

6 Views Cite this report Responses(0)

Approved

I have seen the clarifications by the authors and they are sufficient for me to amend my previous status to “Approved”

Competing Interests

No competing interests were disclosed.

I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.

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6 Views

27 Jan 2023 | for Version 2

6 Views Cite this report Responses(0)

Approved

All my comments have been addressed well, but two minor issues need to be revised.

Under the Introduction, the sentence of "Estimates indicate that schistosomiasis causes an annual loss of 4.5 million disability-adjusted life years (DALYs)" need to check a proper reference on the number of the DALYs, so the number of DALYs may be changed?
In the Table 1, "Schistosomiasis infection" changes as "Schistosomes infection" or "Schistosoma spp. infection".

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Epidemiolgoy of schistosomiasis

I confirm that I have read this submission and believe that I have an appropriate level of expertise to confirm that it is of an acceptable scientific standard.

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10 Views

10 Jan 2023 | for Version 1

10 Views Cite this report Responses(1)

Approved With Reservations

The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.
The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.
In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.
In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.
Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Is the rationale for, and objectives of, the study clearly described?

Yes
Is the study design appropriate for the research question?

Partly
Are sufficient details of the methods provided to allow replication by others?

Partly
Are the datasets clearly presented in a useable and accessible format?

Partly

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Epidemiolgoy of schistosomiasis

Respond to this report

Responses (1)

Author Response

20 Jan 2023

Phyllis Isaiah, University of Basel, Basel, Switzerland

Question 1: The scope of the population to be studied: based on the objectives to provide evidence to designing the extension of PC program, it is essential to understand the current situation of all pre-school age children (PSAC) versus PSAC living in the hard-to-reach areas. Therefore, in the protocol, it is suggested to include the data from whole PSAC population, then to compare the epidemic features between PSAC living in the hard-to-reach areas and all of PSAC in endemic areas.

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.

Question 2: The definition of hard-to-reach areas and hard-to-reach population should be different, but the authors only provide the definition of hard-to-reach population, which covers three types of features. For my understanding, the PSAC population living in the hard-to reach areas is the part of hard-to-reach population. In addition, those living in rich areas but always in the migrate status, such as boat family, etc. So it is suggested to give definitions both of hard-to-reach areas as well as hard-to-reach populations.

Response: Thank you for pointing this out. This has been amended in the protocol.

Question 3: In the objective part, it is suggested to firstly provide a general objective with two to three specific aims, to indicate detail objectives of the study designing. Then followed by the definitions and scope of the review, in order to clearly describe the final outcomes of the study.

Response: Thank you for pointing this out. This has been amended in the protocol. We had only one objective.

Question 4: In the Methods part, it is suggested to provide the detail information of the data analysis, due to there is vague information of the statistical analysis approach provided in the current version. It is also suggested to provide the data collection tables as attached file, which is very important for researchers to collect data and form the datasets as well as to perform the data handling by using unified SOPs in different countries where studying.

Response: Thank you for pointing this out. This has been amended in the protocol. An annex (a data collection table) has been attached.

Question 5: Under the Methods part, data extraction and synthesis, point vi. “Mean eggs per gram f feces or infection intensity classification (if available) “ is only suitable for S. mansoni infection, but not suitable for S. haematobium. So please revise this sentence by consideration of both species of schistosomes.

Response: Thank you for pointing this out. This has been amended in the protocol.

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Competing Interests

No competing interests disclosed

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Reviewer Report

18 Views

15 Dec 2022 | for Version 1

Dirk Engels, Uniting to Combat NTDs Support Centre, Geneva, Switzerland

18 Views Cite this report Responses(1)

Approved With Reservations

Is the rationale for, and objectives of, the study clearly described?

Yes
Is the study design appropriate for the research question?

Yes
Are sufficient details of the methods provided to allow replication by others?

Yes
Are the datasets clearly presented in a useable and accessible format?

Yes

Competing Interests

No competing interests were disclosed.

Reviewer Expertise

Tropical Diseases

Respond to this report

Responses (1)

Author Response

20 Jan 2023

Phyllis Isaiah, University of Basel, Basel, Switzerland

Question 1: The study is relevant as it is clearly important, especially in view of the ADOPT programme for pediatric praziquantel, to have best estimates of the global number of pre-school age children to be reached. My main remark would be: with such a substantial scoping review, why limiting the study to hard-to-reach populations only? I suggest to expand to all eligible studies reporting on schistosomiasis in PSAC and treat hard-to-reach populations as a sub-analysis. In this way as-broad-as-possible information is captured in one scoping review, leaving all options open for an access plan

Response: Thank you for the feedback.

A recent study by Kalinda et al., 2020 conducted a systematic review of schistosomiasis infection burden among PSAC. We also anticipate that in view of the availability of pediatric praziquantel, intervention need will shift to ensure the very marginalized PSAC are covered, hence the focus on hard-to-reach areas and population. We have however compared the results of the systematic review by Kalinda and colleagues with our results in the actual scoping review manuscript.

View more View less

Competing Interests

No competing interests.

Alongside their report, reviewers assign a status to the article:

Approved - the paper is scientifically sound in its current form and only minor, if any, improvements are suggested

Approved with reservations - A number of small changes, sometimes more significant revisions are required to address specific details and improve the papers academic merit.

Not approved - fundamental flaws in the paper seriously undermine the findings and conclusions

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Epidemiology of pediatric schistosomiasis in hard-to-reach areas and populations: A scoping review protocol

Abstract

Keywords

Abbreviations and acronyms

Introduction

Objective

Review question

Table 1. Condition, context, and population framework.

Methods

Table 2. Searching databases and strategies.

Risk of bias and quality assessment

Data extraction and synthesis

Data analysis

Dissemination of results

Study status

Data availability

Acknowledgments

References

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