Running head: INTEGRATIVE LITERATURE REVIEW 1

Integrative Literature Review

Christine Randazzo
Bon Secours Memorial College of Nursing
Karen G. Mellott PhD, MS, RN
NUR 4122: Nursing Research
November 13, 2019
“I Pledge…”
INTEGRATIVE LITERATURE REVIEW 2

Abstract
Purpose: The purpose of this integrative review is to evaluate the psychosocial experiences of

family members of infants born with cleft lip and/or palate (CL/P). Research was gathered to

answer the PICOT question: “How Do Family Members of Infants Born with CL/P Perceive

Psychosocial Experiences of the Diagnosis Throughout their Lives?”

Introduction/Background: CL/P is a common craniofacial anomaly worldwide. The diagnosis of

CL/P can present many challenges. Having a child with a health condition is a burden to

families, thus, it is important to research how to support their needs.

Design and Search Methods: Research was collected using PubMed and Google Scholar online

databases. Articles were chosen based on specific criteria. Information was analyzed to support

perceptions of the psychosocial experiences of family members to children with CL/P.

Results and Findings: Findings show two common themes amongst the research articles: A lack

of support, and differences in needs between mothers and fathers. Mothers experience problems

related to low self-esteem, while fathers have more internalizing problems.

Limitations: This review is not exhaustive, as it analyzes five articles and the researcher has

minimal experience. Further limitations include potential bias and small sample sizes, making

generalizability difficult.

Implications for Practice: This review can be used in practice by providing the necessary support

to those involved with CL/P. Needs vary amongst family members, and it is important to

address them to provide the best outcomes.

Recommendations: Future research is needed in order to build literature on the topic of

psychosocial effects related to CL/P.

INTEGRATIVE LITERATURE REVIEW 3

Integrative Literature Review

The purpose of this integrative literature review is to evaluate scholarly literature to

determine the psychosocial impact that cleft lip and/or cleft palate (CL/P) has on family

members. Cleft lip and/or palate is one of the most common craniofacial anomalies worldwide

and is caused by abnormal facial development during gestation (Guest, Costa, McCarthy,

Cunniffe, & Stock 2019). The abnormality contributes to initial problems such as difficulties

with feeding and the need for surgery to repair the lip and palate within the first year to 18

months of life. (Nidey, Uribe, Marazita, & Wehby, 2015). Problems with speech, hearing,

dental, psychological, and learning issues may occur later in life, contributing to financial issues

for families (Nidey et al., 2015). Cleft lip and/or palate can have a significant psychosocial

impact and present many challenges for those born with it, along with challenges for the family

members who care for them (Guest et al., 2019; Nidey et al., 2015; Stock & Rumsey, 2015;

Stock, Stoneman, Cunniffe, & Rumsey 2016; Ueki et al., 2019).

Researchers have studied challenges faced by individuals with CL/P, but there is little

evidence that identifies the psychosocial challenges faced by those individuals’ extended family

members. The aim of this integrative review is to analyze the available literature to the

researcher’s PICOT question: “How Do Family Members of Infants Born with CL/P Perceive

Psychosocial Experiences of the Diagnosis Throughout their Lives?”

Design/Research Methods

The research design is an integrative review. The method utilized by the researcher

began with a preliminary search using PubMed and Google Scholar. The search words included

cleft lip and palate, family, parent health, psychosocial, and support. The number of articles
INTEGRATIVE LITERATURE REVIEW 4

generated from the initial search was approximately 2,090. Limits to the search process were set

to full text, peer reviewed quantitative and qualitative research studies, English language, and

publishing dates within the past five years (2015 and above). Meta-analysis and systematic

reviews were not considered for this integrative review. Any articles that did not meet the

requirements for the search process were excluded. The five articles that were selected have

different study designs as some are qualitative studies and some are quantitative studies. All

articles have similarities in content, and results and are relevant to the PICOT question.

Findings/Results

The findings and results of the five reviewed studies assess the psychosocial effects that

family members experience having a child born with CL/P. A detailed summary of the research

is noted in Appendix 1. Two common themes noted in the research were lack of psychosocial

support and differences in needs between mothers and fathers.

Lack of Support

Lack of support for parents and extended family members of children with CL/P is a

common theme in three of the five articles. When families find out they are going to have a

child born with CL/P, they face several psychosocial challenges. The theme, “Lack of Support”

is defined as a lack of support from healthcare providers as well as peers in social settings.

Many families felt there was insufficient support from others and that it would be beneficial to

improve support services.

Guest et al. (2019) evaluated the impact that grandparents have in the lives of their

grandchildren with CL/P. The purpose of this study was to explore the experiences and support

needs of grandparents of children born with CL/P. The study was a qualitative phenomenology
INTEGRATIVE LITERATURE REVIEW 5

design that included 12 grandparents, ages 53 to 79 years old, who had a grandchild born with

CL/P. Data were collected via individual telephone interviews that were audio-recorded,

transcribed verbatim, and analyzed using thematic analysis. Thematic analysis is a method used

to analyze qualitative data and involves identifying and organizing key themes within data sets

(Guest et al., 2019).

The themes identified were emotional impact of CL/P on grandparents, grandparents’

social experiences, their involvement, information needs, and their support needs. Participants

described experiencing negative emotions following the news of their grandchild’s diagnosis and

negative encounters in public (Guest et al., 2019). Participants also felt they did not receive

enough information about their grandchild’s condition, and that only hearing information from

the parents was not reliable. Grandparents’ expressed that it would have been nice to have

increased contact with healthcare professionals and to be informed on how to best support the

family, help with feeding, and have access to peer support (Guest et al., 2019).

Stock and Rumsey (2015) studied the support needs of fathers as previous studies have

focused mostly on the impact of CL/P on mothers. The purpose of this study was to explore the

impact of a child born with CL/P from the fathers’ perspective. The study was a qualitative

phenomenology design that included 15 fathers, ages 31 through 58 years old, of children born

with CL/P. Data were collected and recorded via individual free response telephone interviews.

Thematic analysis was used to analyze the interview data which generated four themes using

Braun and Clarke’s guidelines (Stock & Rumsey, 2015).

The four themes identified were variations in care and support, appraisals of the cleft,

perceptions of treatment, and looking back and moving forward. Most fathers received upsetting

reports of their child’s diagnosis and stated the information was delivered in a very insensitive
INTEGRATIVE LITERATURE REVIEW 6

way (Stock & Rumsey, 2015). Fathers also found that following birth, healthcare providers had

poor knowledge about CL/P and lacked confidence when caring for their newborn (Stock &

Rumsey, 2015). Fathers talked about how there is little support available for men in general, and

that having a baby born with a complication made this more apparent. Fathers also stated that

individual support would have been valuable as they felt they had to remain strong and positive

for their wife and child; this led to fathers having no outlet for their own emotions (Stock &

Rumsey, 2015).

Stock, Stoneman, Cunniffe, and Rumsey (2016) studied the support needs of siblings of

children born with CL/P. The purpose of this study was to gain a better understanding of the

challenges and benefits associated with being an unaffected sibling of a child born with CL/P,

according to the perspectives of parents and siblings (Stock et al., 2016). The study was a

qualitative phenomenology design that included 13 participants. Participants were either a

sibling of an individual born with CL/P, or a parent of at least one child born with CL/P and one

child born without CL/P. Data were collected via individual, free-response telephone interviews.

Each interview was recorded and transcribed verbatim, then analyzed using thematic analysis.

Three themes were identified following Braun and Clarke’s guidelines (Stock et al., 2016).

The three themes were perceptions of positive and negative impacts, factors affecting the

degree of impact, and support for families. Siblings reported having anxiety that stemmed from

not understanding the diagnosis and treatment process of CL/P (Stock et al., 2016). Parents

expressed it would have been beneficial for siblings to be more involved in the treatment process

of CL/P, and that there is a need for support from professionals to help explain CL/P to the

unaffected child. Parents also reported that the unaffected sibling was often left out of the

healthcare system, and including them would be valuable to the entire family (Stock et al., 2016).
INTEGRATIVE LITERATURE REVIEW 7

Differences in Needs Between Mothers and Fathers

Differences in needs between mothers and fathers were discussed in three of the five

articles. Previous studies have focused solely on the needs of the mother or the child born with

CL/P and have not included fathers (Stock & Rumsey, 2016). However, the evidence reviewed

relates to the needs of fathers, which is an important perspective to be considered. The definition

of this theme is that mothers and fathers have different takes regarding psychosocial needs when

caring for children born with CL/P.

Nidey et al. (2015) studied how parents are impacted by children with CL/P. The

purpose of this study was to identify predictors of psychosocial well-being of parents of affected

children with oral clefts. The study used a quantitative design: Descriptive Observational design

can be assumed but is not clearly stated in the article. The study used validated instruments to

measure social avoidance and distress, fear of negative evaluation scale, self-esteem, and

interpersonal support (Nidey et al., 2015). Participants included 287 parents of children with oral

clefts. Data were collected during interviews with parents using the same instruments across the

board. Parents were asked to complete four self-administered instruments for measuring

psychosocial status and social support (Nidey et al., 2015). Regression analysis was used to

evaluate psychosocial outcomes.

One of the measurement tools used was the Social Avoidance and Distress Scale,

developed to evaluate anxiety and distress of individuals in social situations. Another

measurement tool was the Fear of Negative Evaluation, developed to evaluate the respondent’s

apprehension of being judged negatively by others. A third measurement tool was the

Rosenberg Self-Esteem, which was used to evaluate the participants’ feelings of self-acceptance
INTEGRATIVE LITERATURE REVIEW 8

and self-worth. The final measurement tool was the Interpersonal Support Evaluation List, which

measured the participant’s perception of social support.

The psychosocial instruments used provided continuous measures and estimated

regressions of the instrument scores one at a time over the explanatory variables of interest using

ordinary least squares. Regressions were estimated for the total scores of each of the four

instruments as well as the scores of six subscales. Differences were found between fathers’ and

mothers’ psychosocial status, as fathers showed greater self-esteem and reduced concern of

negative judgement, while mothers reported having someone to talk to about their problems.

Nidley et al., (2015) generated statistically significant results which showed that fathers

had higher self-esteem than mothers (P = 0.01) and lower concern of being negatively judged by

others (P < 0.0001); however fathers had lower perception of having someone to talk to about

their problems (P = 0.01). High household income was associated with greater self-esteem and

perception of social support (<0.05). Parents of male affected children had greater perception of

social support than parents of female affected children (P = 0.04). No significant differences in

parental psychosocial status measures were found by cleft type. Similarly, there is little evidence

of changes with child age, except for an increase in parental distress and decline in self-esteem

during mid-adolescence (age 15-17 years) (Nidley et al., 2015).

Ueki et al. (2019) studied the experiences of parents with children born with CL/P. The

purpose of this study was to explore the psychosocial differences and resilience of the fathers

and mothers of children with CL/P. The design of this study was a quantitative design and it can

be assumed that Descriptive design was used, although was not clearly stated. Participants

included 64 couples of parents with children with CL/P. Participants came on site to the clinic
INTEGRATIVE LITERATURE REVIEW 9

where self-administered cross-sectional questionnaires were completed then submitted to a

lockbox or by mail within one month (Ueki et al., 2019).

The questionnaire included parent and child demographic characteristics, difficulties with

CL/P, and the Scale to Measure Resilience in Child Care (SMRCC). The SMRCC is a validated

self-report measure of the parent’s resilience in terms of successfully adapting in difficult

situations related to CL/P (Ueki et al., 2019). The difficulties with CL/P were assessed with 12

unique items that were based on results of the authors’ prior research that clarified difficulties

parents have with children with CL/P. Results showed the parents had worries, concerns, and

anxiety about parenting a child with CL/P. Each item was rated on a five-point Likert-style scale

that ranged from 1 (strongly disagree) to 5 (strongly agree) (Ueki et al., 2019).

Paired t-tests were used to clarify data from the SMRCC. A p-value of <0.05 indicated

statistical significance, and statistical analysis was performed. It was found that mothers of

children with CL/P feel more guilt: For example, “I think that the child’s disease is my fault”

showed statistical significance with a p-value of <0.001. On the other hand, fathers had greater

resilience with problem-solving skills with a p-value of 0.020 and acceptance of CL/P with a p-

value of 0.002 (Ueki et al., 2019).

Stock & Rumsey (2015) studied the psychosocial impact CL/P has on fathers as previous

studies have focused mostly on the impact of CL/P on mothers. It was found that there were

variations in care and support in fathers compared to mothers, and that further backing is needed

to support both. Stock & Rumsey (2015) mentioned that although experiences voiced by fathers

do have similarities with previous research on mothers, it is also important that the fathers’ views

and needs are considered.

INTEGRATIVE LITERATURE REVIEW 10

Discussion/Implications

The articles reviewed correlate well with the PICOT question in evaluating the

psychosocial experiences in relation to CL/P. Similarities and differences were noticed between

all five articles. Similarities between all articles included how family members perceived the

impact of CL/P as well as mentioning positive and negative aspects of their experiences.

Another major similarity amongst all articles was that they provided insight into experiences of

populations who are not normally studied and found that these populations need resources and

support. Some differences among the studies were that three were qualitative studies, and two

were quantitative studies. The qualitative studies were similar to each other as they all used

thematic analysis, but differed from the quantitative studies as they discussed statistically

significant data.

Implications for practice include having resources provided to all family members to help

them manage the psychosocial aspect of a CL/P diagnosis. Treating the family as a whole rather

than on an individual level would be beneficial to all that are involved with CL/P children and

lead to desirable outcomes for everyone. Encouraging fathers, siblings, and grandparents to be

involved in treatments and included in the learning for CL/P care would increase knowledge,

awareness, and self-esteem.

All five articles indicated that future research should be done in order to expand on what

is already known and to fill in the gaps of what is not known. Since there are not many studies

related to the extended family members of those with CL/P, conducting further research to

identify the needs of these populations would lead to better outcomes of psychosocial status.

Different family members may have different needs, and it is important to address them
INTEGRATIVE LITERATURE REVIEW 11

individually while including them in learning as a whole (Guest et al., 2019; Nidey et al., 2015;

Stock & Rumsey, 2015; Stock et al., 2016; Ueki et al., 2019).

Limitations

Limitations of this integrative review included minimal experience in research

knowledge, only using five articles less than five years old, and that this was not an exhaustive

review. Common limitations found in the research articles included the lack of background

knowledge and small population samples indicating an inability to generalize to further

populations. Further limitations included the potential for bias, as a few studies had self-

selecting participants for the sample size. Both qualitative and quantitative studies did not

indicate specific design, which had to be assumed based off of knowledge of this researcher.

Conclusion

Findings from the research of this integrative review conclude how family members

perceive psychosocial experiences in relation to a child born with CL/P. The results of these

studies are relatable to the PICOT question: “How Do Family Members of Infants Born with

CL/P Perceive Psychosocial Experiences of the Diagnosis Throughout their Lives?” Future

studies can use the current information to produce further research in order to provide families

with the necessary support and education. Providing accurate information to families will

promote the greatest outcomes for children with CL/P as well as those involved in their care.
INTEGRATIVE LITERATURE REVIEW 12

References
Guest, E., Custa, B., McCarthy, G., Cunniffe, C., & Stock, N.M. (2019). The experiences and

support needs of grandparents of children born with cleft lip and/or palate. The Cleft Palate--

Craniofacial Journal, 56 (9)1181-1186. doi:10.1177/1055665619850709

Nidey, M., Moreno Uribe, L.M., Marazita M.M., & Webby, G.L. (2015). Psychosocial well-

being of parents of children with oral clefts. Child: Care, Health and Development, 42(1)

42-50. doi: 10.1111/cch.12276

Stock, N.M., & Rumsey, N. (2015). Parenting a child with a cleft: the father’s perspective. The

Cleft Palate-Craniofacial Journal, 52 (1) 31-43. doi: 10.1597/13-035

Stock, N.M., Stoneman, K., Cunniffe, C., & Rumsey, N. (2016). The psychosocial impact of

cleft lip and/or palate on unaffected siblings. The Cleft Palate-Craniofacial Journal, 53 (6)

670-682. doi:10.1597/15-148

Ueki, S., Fujita, Y., Kitao, M., Kumagai, Y., Ike, M., Niinomi, K., … Fujiwara, C. (2019).

Resilience and difficulties of parents of children with a cleft lip and palate. Japan

Journal of Nursing Science, 16, 232-237. doi: 10.1111/jjns.12231

INTEGRATIVE LITERATURE REVIEW 13

Appendix 1: Summary of the Literature Tables

Table 1
Article Reference Guest, E., Custa, B., McCarthy, G., Cunniffe, C., & Stock, N.M. (2019).
(APA formatting) The experiences and support needs of grandparents of children born with
cleft lip and/or palate. The Cleft Palate-Craniofacial Journal, 56 (9)
1181-1186. doi:10.1177/1055665619850709
Background PS: Grandparents play an increasingly significant role of involvement in
their grandchildren’s lives and further research is needed to address
Purpose grandparents’ concerns and psychosocial impact.

P: The purpose of this study was to explore the experiences and support
Significance needs of grandparents of children born with cleft lip and/or palate
statement (CL/P).

S: Significance is building literature on the research question and to

detect the psychosocial impact of CL/P on extended family members in
order to meet their current and future needs.
Conceptual or Yes or No: No conceptual or theoretical framework was used.
Theoretical
Framework used Name of Framework: none

Method-- Design / Design: Qualitative (No specific type was identified, but can assume
phenomenology)

Philosophical Philosophical View: Inductive qualitative approach

Underpinnings
Sample / Sample: 12 (11 female) grandparents who had a grandchild born with
CL/P age ranged from 53 to 79 years old, White British or White
Scottish ethnicity.
Setting /
Setting: Scotland
Ethical
Considerations Ethical: No specific mention of ethical approval. Participants were sent
a participant information sheet and a copy of the consent form. It was
made clear that participants had the right to withdraw from the study
prior to data analysis. Participants provided verbal consent.
Major Variables 1. No variables as this was a qualitative study.
Studied and their
Definition
(Brief summary)
Measurement Measures/Tools:
Tool(s) (List each 1. Thematic analysis
one separately)
INTEGRATIVE LITERATURE REVIEW 14

Data Collection Data Collection: Individual telephone interviews were conducted over
Method (brief the phone to eliminate travel costs and geographical barriers.
summary)
Data Analysis Statistics:
(describe Interviews were audio-recorded, transcribed verbatim, and analyzed
techniques, such as using inductive thematic analysis. This involves identifying and
specific statistical organizing key themes within qualitative data sets.
tests for
quantitative
studies (i.e. t-test, Interpretation methods: Each transcript was analyzed independently by
pearson’s r) or the first and second authors. These analyses were then compared, and
interpretation overall findings were discussed with the third author until full agreement
methods for was reached.
qualitative studies
(i.e. content
analysis,
Colaizzi’s method)
Findings / • Five key themes were identified.
Discussion 1. Emotional impact of CL/P on grandparents
(summarize major 2. Grandparents’ social experiences
points with brief 3. Grandparents’ involvement
statements using 4. Grandparents’ information needs
bullet points) 5. Grandparents’ support needs
• The findings of this study provide novel insight into the experiences of
an underexplored population in the context of CL/P.

Appraisal/Worth • Grandparents who participated in this study reported similar

to practice experiences and support needs.
• This small-scale study provides important insight into the
experiences and support needs of grandparents of children born
with CL/P.
• Clinicians and charitable organizations could consider how
existing resources could be made more accessible for wider
family members.
This research provides insight to the PICOT question of how family
members of infants born with CL/P perceive psychosocial experiences of
the diagnosis up to the first year of life.
INTEGRATIVE LITERATURE REVIEW 15

Table 2
Article Reference Nidey, M., Moreno Uribe, L.M., Marazita M.M., Webby, G.L. (2015).
(APA formatting) Psychosocial well-being of parents of children with oral clefts. Child:
Care, Health and Development, 42(1) 42-50.doi: 10.1111/cch.12276
Background PS:
Parents of children with oral clefts can be impacted in several ways
Purpose psychosocially, but minimal evidence exists to support this.
P:
The purpose of this study was to identify predictors of psychosocial well-
Significance being of parents of affected children with oral clefts.
statement S: Significance is building literature on the research and to further
understand the psychosocial impact of CL/P on all members of families
Conceptual or Yes or No: No
Theoretical
Framework used Name of Framework: n/a
Method-- Design / Design: Quantitative study using validated psychosocial instruments to
measure social avoidance and distress, fear of negative evaluation scale,
self esteem and interpersonal support
Philosophical
Underpinnings Philosophical View: n/a
Sample / Sample: 287 parents (171 mothers and 116 fathers) of children with oral
clefts. Participants were identified and recruited at three craniofacial
clinics: University of Iowa Craniofacial Clinic, St Louis Children’s
Setting / Hospital, and Cleft Craniofacial Center at Children’s Hospital of
Pittsburg.
Ethical
Considerations Setting: studies were completed either at the study site where they were
recruited or at their home during visits by study staff.

Ethical: Participants gave consent in person following IRB approved

protocols.
Major Variables 1. Independent variable: parents psychosocial well being
Studied and their 2. Dependent variables:
Definition a. Distress and avoidance
(Brief summary) b. Belonging
c. Self-esteem
d. Tangible items
e. Appraisal
Measurement Measures/Tools:
Tool(s) (List each 1. Social Avoidance and Distress scale (SAD)
one separately) a. The SAD scale was developed to evaluate anxiety and
Data Collection distress of individuals in social situations
Method (brief 2. Fear of Negative Evaluation (FNE)
summary) a. The FNE scale was developed to evaluate respondent’s
apprehension of being judged negatively by others (Likert
scale)
INTEGRATIVE LITERATURE REVIEW 16

3. Rosenberg Self-Esteem (RSE)

a. The RSE was used to evaluate the participants’ feelings of
self-acceptance and self-worth (Likert scale)
4. Interpersonal Support Evaluation List (ISEL)
a. The ISEL was developed to measure the participant’s
perception of social support (Likert scale)

Data Collection: Data were collected by trained study staff during

interviews with parents using similar instruments across all sites. Parents
were asked to complete four self-administered instruments for measuring
psychosocial status and social support, along with giving information
about demographics and socioeconomic status.
Data Analysis Statistics: The psychosocial instruments used provided continuous
(describe measures, and estimated regressions of the instrument scores one at a
techniques, such as time over the explanatory variables of interest using ordinary least
specific statistical squares. Regressions were estimated for the total scores of each of the
tests for four instruments as well as the scores of six subscales.
quantitative
studies (i.e. t-test, Interpretation methods: n/a
pearson’s r) or
interpretation
methods for
qualitative studies
(i.e. content
analysis,
Colaizzi’s method)
Findings / • There were differences between fathers and mothers
Discussion psychosocial status. Fathers showed greater self-esteem and
(summarize major reduced concern about negative judgement of others, white
points with brief mothers reported more on having someone to talk to about
statements using their problems than fathers.
bullet points) • Parents with higher household income have greater self-esteem
and perception of social support than those in poor households.
Results showed that a family-based treatment approach as opposed to
only focusing on the affected child would be supportive and beneficial to
families.
Appraisal/Worth • It is important to understand the psychosocial outcomes of
to practice parents of children with oral clefts because we can better prepare
healthcare professionals to treat families.
• Future studies are needed to understand how families as a unit are
impacted.
• Engaging parents by looking at their psychosocial status in
holistic family-based treatment approaches may enhance family
well-being.
INTEGRATIVE LITERATURE REVIEW 17

This research provides insight to the PICOT question of how family

members of infants born with CL/P perceive psychosocial experiences of
the diagnosis up to the first year of life.
INTEGRATIVE LITERATURE REVIEW 18

Table 3
Article Reference Stock, N.M., Rumsey, N. (2015). Parenting a child with a cleft: the
(APA formatting) father’s perspective. The Cleft Palate-Craniofacial Journal, 52 (1). doi:
10.1597/13-035
Background PS: The birth of a child with a health condition puts a significant burden
on families as parents often feel shocked and overwhelmed with the
Purpose potential challenges their child may face.

P: The purpose is to explore the impact of a child born with cleft lip
Significance and/or palate from the perspective of the father.
statement
S: Significance is to build knowledge to understand the psychosocial
impact that CL/P has on extended family members (not just the
individual and/or the mother) so they can be provided support
appropriately.
Conceptual or Yes or No: Yes
Theoretical
Framework used Name of Framework: Pragmatic framework
Method-- Design / Design: Qualitative (No specific type was identified, but can assume
phenomenology)

Philosophical Philosophical View: inductive data-driven approach

Underpinnings
Sample / Sample:
15 fathers of children born with CL/P ages 31 through 58 years (39
average). All fathers identified as white British.
Setting /
Setting: U.K.
Ethical
Considerations Ethical: Ethical approval was obtained from the University of the West
of England Research Ethics Committee and also followed by the British
psychological Society’s Code of Ethics and Conduct throughout.
Participants were made aware of key components including
confidentiality and the right to withdraw. All participants were given
pseudonyms to ensure anonymity.
Major Variables No variables as this was a qualitative study.
Studied and their
Definition
(Brief summary)
Measurement Measures/Tools:
Tool(s) (List each 1. Thematic analysis
one separately) Data Collection:.
Data Collection Individual free response recorded telephone interviews were conducted
Method (brief until no new information was being obtained.
summary)
INTEGRATIVE LITERATURE REVIEW 19

Data Analysis Statistics: Interviews were conducted by the first author who has a
(describe background in psychology and is trained in interview techniques. The
techniques, such as interviewer prepared prompts about key topics to elicit further
specific statistical information, but other wise participants had freedom to relay their own
tests for stories. Interviews lasted between 40 minutes and one hour.
quantitative
studies (i.e. t-test, Interpretation methods: an inductive, data-driven approach was taken
pearson’s r) or using Braun and Clarke’s guidelines including becoming familiar with
interpretation the data, identifying interesting features of the data, searching for
methods for themes, reviewing themes, defining and naming themes, and producing
qualitative studies the report. Emerging themes were initially identified by the first author,
(i.e. content then checked and discussed until both authors came to an agreement.
analysis,
Colaizzi’s method)
Findings / • Four key themes were identified, each with multiple
Discussion subthemes.
(summarize major 1. Variations in care and support
points with brief a. Responses of non-specialist healthcare professionals
statements using b. Social experiences and acceptance
bullet points) c. Care provided by cleft specialists
d. A lack of support for fathers
2. Appraisals of the cleft
a. No limits
b. Aetiology
c. Making comparisons
3. Perceptions of treatment
a. Operations
b. Barriers and facilitators to treatment
c. A long journey
4. Looking back and moving forward
a. Personal growth
b. Giving something back
c. The future
These themes provide evidence for the need of addressing support needs
of fathers in both clinical practice and applied research.
Appraisal/Worth • Longitudinal research needs to be done in order to more
to practice accurately capture the journey families take when a child is
born with CL/P using this research as supportive information.
• This article supports the greater need for resources and
awareness of the psychosocial impact CL/P has on fathers and
extended family members.
• It is important to consider the fathers’ viewpoints and needs
when providing support to families of children with CL/P.
• This research provides insight to the PICOT question of how
family members of infants born with CL/P perceive
INTEGRATIVE LITERATURE REVIEW 20

psychosocial experiences of the diagnosis up to the first year of

life.
INTEGRATIVE LITERATURE REVIEW 21

Table 4
Article Reference Stock, N.M., Stoneman, K., Cunniffe, C., Rumsey, N. (2016). The
(APA formatting) psychosocial impact of cleft lip and/or palate on unaffected siblings. The
Cleft Palate-Craniofacial Journal, 53 (6). doi: 10.1597/15-148
Background PS: Sibling relationships are unique social connections that can impact
psychosocial adjustment. Unaffected siblings of children with long-term
Purpose chronic illnesses are at risk for poorer psychological functioning.
Research has been done to investigate the impact CL/P has on affected
individuals and their parents, but not much has been explored about close
Significance family members.
statement
P: The purpose of this study was to gain a better understanding of the
lived experiences of unaffected siblings of children born with CL/P to
learn how to best support them.

S: Significance is to understand how siblings are impacted

psychosocially and contribute to further research to meet the needs of
this population.
Conceptual or Yes or No: Yes
Theoretical
Framework used Name of Framework: pragmatic framework
Method-- Design / Design: Qualitative (No specific type was identified, but can assume
phenomenology)
Philosophical View: philosophical underpinnings were not identified in
Philosophical the article.
Underpinnings
Sample / Sample: A total of 13 participants who were either a sibling of an
individual born with CL/P or a parent of at least one child born with
CL/P and one without. Siblings age ranged from 11 and 31 years old
Setting / (average 17), and parents from ages 23 to 60 (average 48).

Ethical Setting: Participants were recruited from multiple regions across the
Considerations U.K.

Ethical: The study was reviewed and approved by the Ethics Committee
for the Department of Health and Social Sciences at the University of the
West of England in Bristol, U.K. This study was also reviewed by the
Advisory Panel for the CLAPA Regional Coordinators Project. It was
also mentioned that the British Psychological Society Code of Ethics and
Conduct was adhered to throughout the study. Participants were self-
selecting, were explained purpose of study and ability to withdraw, and
gave written consent. Pseudonyms were also given to maintain
confidentiality.
Major Variables No variables as this was a qualitative study.
Studied and their
Definition
INTEGRATIVE LITERATURE REVIEW 22

(Brief summary)
Measurement Measures/Tools:
Tool(s) (List each 1. Thematic analysis
one separately)
Data Collection
Method (brief Data Collection: Individual, free-response telephone interviews were
summary) conducted and guided by an open-ended, semi-structured interview
schedule compiled by the authors using existing literature and
experiences of families affected by CL/P.
Data Analysis Statistics:
(describe Each interview was audio recorded and transcribed verbatim; then
techniques, such as analyzed separately by the first and second authors using thematic
specific statistical analysis. This method is used to identify and organize patterns within a
tests for data set.
quantitative
studies (i.e. t-test,
pearson’s r) or Interpretation methods: Thematic analysis was used in accordance with
interpretation Braun and Clarke’s guidelines following the steps of becoming familiar
methods for with the data, identifying interesting features of the data, searching for
qualitative studies themes, reviewing themes, defining and naming themes, and producing
(i.e. content the report. Themes were checked between the two coders until they
analysis, reached an agreement on level of importance.
Colaizzi’s method)
Findings / • Three key themes were identified, and further categorized in
Discussion subthemes.
(summarize major 1. Perceptions of positive and negative impacts
points with brief a. Sibling rivalry
statements using b. The sibling bond
bullet points) c. Sibling anxiety
d. Positive impacts

2. Factors affecting the degree of impact

a. Birth order
b. Age gap
c. Gender
d. Treatment burden
e. Social support network
f. Socioeconomic status
g. Parental coping
h. Sibling involvement

3. Support for families

a. Support for parents
b. Support for siblings
c. Mode of support
INTEGRATIVE LITERATURE REVIEW 23

The findings of this study provide insight into a population that is

typically overlooked in both CL/P research and practice.
Appraisal/Worth • This study helps to better understand the experiences and support
to practice need of neglected patient groups while capturing multiple
perspectives.
• It is desirable for healthcare teams to include all members of the
family to reach desirable outcomes for everyone.
Normalizing siblings to reduce anxiety and allow them to be involved in
treatments encouraging them to learn and be included in caring for CL/P
INTEGRATIVE LITERATURE REVIEW 24

Table 5
Article Reference Ueki, S., Fujita, Y., Kitao, M., Kumagai, Y., Ike, M., Niinomi, K., …
(APA formatting) Fujiwara, C. (2019). Resilience and difficulties of parents of children
with a cleft lip and palate. Japan Journal of Nursing Science, 16, 232-
237.
Background PS: Parents experience a significant psychosocial impact having children
born with CL/P including worry, guilt, conflict, and anxiety.
Purpose
P: The purpose of this study was to explore the psychological differences
and resilience of the fathers and mothers of children with CL/P.
Significance
statement S: The significance is building literature on the research question to
determine psychosocial impact of CL/P on family members in order to
best support their needs.
Conceptual or Yes or No: No
Theoretical
Framework used Name of Framework: n/a
Method-- Design / Design: Quantitative, self-administered cross-sectional questionnaire

Philosophical View: n/a

Philosophical
Underpinnings
Sample / Sample: 64 couples of parents with children with CL/P

Setting: Outpatient clinic in Japan

Setting /
Ethical: Ethical approval was granted from the institutional review
Ethical board of the research hospital. Participants were given an anonymous
Considerations questionnaire along with an information sheet stating the purpose and
method of the study and privacy protection.

Major Variables 1. Independent variable: parents’ emotional difficulties and

Studied and their resilience
Definition 2. Dependent variables:
(Brief summary)
a. Support from surrounding persons – “I have a supporter whom
I can count on, no matter what”
b. Problem-solving skills – “I can come with anything by using
my experience”
c. Recognition and acceptance – “I think I can manage some
difficulty if I face it”

Measurement Measures/Tools:
Tool(s) (List each • Cross-sectional questionnaire
one separately) • Likert-style scale
INTEGRATIVE LITERATURE REVIEW 25

Data Collection • Scale to Measure Resilience in Child Care (SMRCC)

Method (brief
summary) Data Collection:.
Participants came to the clinic to complete questionnaires, which were
then submitted to a lockbox in the waiting area or sent by mail within
one month.
Data Analysis Statistics:
(describe Paired t-tests were used to clarify data from the SMRCC. A p-value of
techniques, such as <0.05 indicated statistical significance, and statistical analysis was done
specific statistical by using IBM SPSS Statistics for Windows v. 20.0
tests for
quantitative Interpretation methods: n/a
studies (i.e. t-test,
pearson’s r) or
interpretation
methods for
qualitative studies
(i.e. content
analysis,
Colaizzi’s method)
Findings / • Mothers of children with CL/P feel a certain degree of guilt,
Discussion which healthcare providers need to support via programs.
(summarize major • Fathers had greater resilience with problem solving skills and
points with brief recognition and acceptance of CL/P. They should be provided
statements using additional knowledge about the disease and be taught skills to
bullet points) increase parenting ability.
Appraisal/Worth • It is important to understand psychosocial differences between
to practice mothers’ and fathers’ challenges with their children with CL/P.
• Support and resources should be provided to families based on
their needs.
• This research provides insight to the PICOT question of how
family members of infants born with CL/P perceive psychosocial
experiences of the diagnosis up to the first year of life.