Predisposing

Journal of Pediatric Psychology, 2015, 1–23
doi: 10.1093/jpepsy/jsv076
Systematic Review
Systematic Review: Predisposing, Precipitating,

Perpetuating, and Present Factors Predicting
Anticipatory Distress to Painful Medical
5 Procedures in Children
Nicole M. Racine,1 MA, Rebecca R. Pillai Riddell,1,2 PHD, Maria Khan,1
BA, Masa Calic,1 BA, Anna Taddio,2,3 PHD, and Paula Tablon,1 BA
1
Department of Psychology, York University, 2Hospital for Sick Children, and 3University of Toronto
10 All correspondence concerning this article should be addressed to Rebecca R. Pillai Riddell, PHD, Department of
Psychology, York University, 4700 Keele Street, OUCH Laboratory, 2004/6 Sherman Health Sciences Building,
Toronto, ON, M3J 1P3, Canada. E-mail: [email protected]
Received January 12, 2015; revisions received July 15, 2015; accepted July 17, 2015
Abstract
15 Objective To conduct a systematic review of the factors predicting anticipatory distress to painful
medical procedures in children. Methods A systematic search was conducted to identify studies
with factors related to anticipatory distress to painful medical procedures in children aged 0–18
years. The search retrieved 7,088 articles to review against inclusion criteria. A total of 77 studies
were included in the review. Results Thirty-one factors were found to predict anticipatory dis-
20 tress to painful medical procedures in children. A narrative synthesis of the evidence was con-
ducted, and a summary figure is presented. Conclusions Many factors were elucidated that con-
tribute to the occurrence of anticipatory distress to painful medical procedures. The factors that
appear to increase anticipatory distress are child psychopathology, difficult child temperament,
parent distress promoting behaviors, parent situational distress, previous pain events, parent antic-
25 ipation of distress, and parent anxious predisposition. Longitudinal and experimental research is
needed to further elucidate these factors.
Key words: anxiety; children; infancy; pain; parents; systematic review.
Introduction and exhibit more intense pain responses (Taddio,

30 Healthy children experience frequent medical proce- Shah, Gilbert-MacLeod, & Katz, 2002). Anticipatory
dures such as immunization and blood draws (Public distress and fear of medical procedures have also been 45
Health Agency of Canada, 2006). Many young chil- identified as concerns in preschool and school-aged
dren experience high levels of pain and distress during children. One study found that 22% of 4–6-year-old
these procedures, and adequate pain management children experience serious distress during the prepa-
35 strategies are seldom used (Lisi, Campbell, Pillai ratory phase of an immunization (Jacobson et al.,
Riddell, Garfield, & Greenberg, 2013). Many children 2001). Another recent study found that more than 50
also experience distress and anxiety before the proce- half of children under the age of 8 years have needle
dure even begins (Blount, Sturges, & Powers, 1990). fear (Taddio et al., 2012). This finding is particularly
This is called anticipatory distress. Anticipatory dis- concerning as anticipatory distress has been associated
40 tress has been identified as occurring as early as in- with several negative sequelae (Bijttebier &
fancy. Newborn infants who have been exposed to Vertommen, 1998; Palermo & Drotar, 1996; Tsao 55
several painful procedures can learn to anticipate pain et al., 2004; Wright, Yelland, Heathcote, Ng, &
C The Author 2015. Published by Oxford University Press on behalf of the Society of Pediatric Psychology.
V
All rights reserved. For permissions, please e-mail: [email protected] 1
2 Racine et al.
Wright, 2009). These negative outcomes could lead to child behavior, and child cognitions), and present fac-
avoidance of painful medical procedures and reduced tors (e.g., health care professional behavior). This re-
compliance with preventative medical care (Taddio view also evaluated the included studies for risk of 60
et al., 2012). Despite the important implications of an- bias and identified methodological limitations of cur-
5 ticipatory distress to painful medical procedures for rent studies. Promising directions for future research
children, little empirical work has investigated the fac- in this area are outlined.
tors that lead to its development.
Several models in the developmental literature have
outlined the pathways that lead to the development of Method
10 maladaptive anxiety and anxiety-related problems Criteria for Considering Studies for This Review 65
(Cicchetti & Cohen, 1995; Rachman, 1977; Vasey & Types of Studies
Dadds, 2001). Within the pediatric pain literature, Studies examining factors that are related to or predict
some work has examined the preprocedural child fac- anticipatory distress (anxiety, fear, distress) to painful
tors that impact a child’s pain response (Kleiber & medical procedures that were published in peer-
15 McCarthy, 2006; Young, 2005); however, these mod- reviewed journals were considered for inclusion. 70
els focus on pain responses rather than anxiety and an- Although the goal of the study was to examine antici-
ticipatory distress. Previous models share a common patory distress, not pain, pain studies that measured
emphasis on the transactional and developmental na- anticipatory distress, anxiety or fear were included in
ture of anxiety or fear over time and highlight the dy- the review. Given the study of fear and anxiety is a
20 namic interaction between the individual child and bourgeoning area in the field of pediatric pain and the 75
his/her environment. The four “Ps” of case formula- goal was not to summarize treatment efficacy, non-
tion (predisposing, precipitating, perpetuating, and randomized studies were included in this review and
protective factors) also provide a useful framework formed the preponderance of the literature base.
for organizing the factors that may contribute to the Nonrandomized studies were included following
25 development of anticipatory distress (Barker, 1988; guidelines of the Cochrane Collaboration that a sys- 80
Carr, 1999; Winters, Hanson, & Stoyanova, 2007). tematic review should include the best available study
Predisposing factors are those that put a child at risk designs with the least risk of bias (Reeves, Deeks,
of developing a problem (in this case, high anticipa- Higgins, & Wells, 2011; Higgins & Green, 2011).
tory distress). These may include genetics, life events, Randomized controlled trials were included when ap-
30 or temperament. Precipitating factors refer to a spe- propriate; however, the variables predicting anticipa- 85
cific event or trigger to the onset of the current prob- tory distress were the focus, not the treatment effect.
lem. Perpetuating factors are those that maintain the Pharmacological (e.g., sedatives) and physical (e.g.,
problem once it has become established. Finally, pro- needle type) predictors of anticipatory distress were
tective factors are strengths of the child or reduce the not examined in this review. All studies were exam-
35 severity of problems and promote healthy and adap- ined for potential sources of bias. 90
tive functioning. Another “P” that can be relevant in
case formulation are “present” factors, that is, those
that are operating during the time of the event-eliciting Types of Participants
distress. Present factors are relevant due to the empha- To be considered in the review, the study had to exam-
40 sis on “procedure” or context in the literature. ine a painful procedure in children from birth through
Additionally, factors that are considered protective 18 years of age. The study also had to measure antici-
can be collapsed within predisposing, perpetuating, patory distress (including anxiety/fear rated before or 95
and present factors. after the procedure or in some cases pain scores prior
The objective of this review is to summarize the to the application of pain) to a painful medical proce-
45 findings of studies that examine factors that predict dure or operation (laboratory pain tasks were ex-
anticipatory distress to painful medical procedures in cluded from the review). Exclusion criteria for studies
children. This systematic review is a qualitative syn- were: no painful medical procedure, incorrect age 100
thesis and summarizes the findings from the search in (i.e., not children 0 to 18 years), and studies where no
a summary figure. The goal of the summary figure factor was analyzed for its relationship to anticipatory
50 (Figure 2) is to provide an overview for researchers distress.
and clinicians of the current literature as well as high-
light gaps in the literature. Based on the developmen- Types of Measures of Anticipatory Distress
tal psychopathology perspective, factors in this review Studies that used an objective behavioral measure, ob- 105
were hypothesized to fall under the four Ps of case for- server reported (e.g., parent, nurse, physician, and re-
55 mulation: predisposing (e.g., genetics and tempera- search assistant), or self-report measure of distress
ment), precipitating (e.g., negative pain experiences), prior to a painful medical procedure or operation
perpetuating (e.g., parent behavior, parent anxiety, were included in this review. In addition to distress
Predisposing, Precipitating, Perpetuating 3
prior to painful medical procedures, for the purpose of November 20, 2013. Separate search strategies and
this review, the term anticipatory distress was opera- terms were developed for each of the databases. 30
tionalized to be an umbrella term that also included Search results were limited to publication years
ratings of fear or anxiety about a procedure provided (1946þ) and age group (children 0–18 years). Search
5 after the procedure or operation as well as a retrospec- terms related to anticipatory distress, medical proce-
tive report of anxiety/fear about a procedure. In cir- dures, pain procedures, and children were systemati-
cumstances where more than one measure of cally paired (see Supplementary Appendix 1). A 35
anticipatory distress was provided, self-report mea- manual database search was also conducted for new
sures of anxiety, fear, and distress were prioritized. articles published after 2013 to update the search in
10 Behavioral measures and observer report measures November 2014. Prior meta-analyses and reference
were used when self-report was not available or was lists from identified studies were also reviewed.
not developmentally appropriate. Additionally, mea- Authors of studies that could not be found were 40
sures that were most specific to anxiety and fear were contacted.
used. For example, using the State Trait Anxiety
15 Inventory over a general distress measure. Measures Data Collection and Analysis
of anticipatory distress most proximal to the painful Selection of Studies
medical procedure were used. For preoperative stud- Three authors (N.R., R.P.R., A.T.) and the librarian
ies, ratings in holding areas or during induction were from a tertiary hospital identified studies through 45
used rather than during separation from parents as not database searching as described above, and duplicates
20 to confound fear and distress of the medical procedure were removed using reference management software
with fear and distress from separation. (Endnote X7). Two review authors (N.M.R. and
R.R.P.R.) initially screened 1,000 abstracts to pilot
Search Methods for Identification of Studies the initial search strategy. Five review authors 50
A review protocol was not registered for this review. (N.M.R., R.R.P.R., P.T., M.C., and M.K.) screened ti-
A librarian from a tertiary hospital with specialized tles and abstracts of studies from the final database
25 training in conducting systematic reviews conducted a searches for inclusion in the review based on predeter-
systematic search in MEDLINE, EMBASE, and EBM mined inclusion and exclusion criteria listed above.
Reviews—Cochrane Central Register of Controlled Figure 1 provides the Preferred Reporting Items for 55
Trials and PsycINFO to include articles indexed as of Systematic Reviews and Meta-Analyses (PRISMA)
Figure 1. Included study flow chart following PRISMA guidelines.

4 Racine et al.
(Moher, Liberati, Tetzlaff, & Altman, 2009) chart bias” was indicated when one of the four criteria was 55
outlining the flow of study selection. missing, not mentioned, or did not meet the criteria
for low or high risk of bias. To be evaluated as low
Data Extraction and Management risk of bias, all the criteria had to be rated as low.
Four authors conducted data extraction independently
5 for all included studies using a data extraction form Data Synthesis
created by the lead author designed for this review, Because of the diversity of medical procedures, out- 60
which was approved by the senior author (R.R.P.R.). come measures used, and participant ages included in
The lead author conducted training sessions with the the studies, a meta-analysis was not deemed appropri-
review authors to explicitly outline the exclusion crite- ate for this review and, rather, a narrative synthesis
10 ria and how to use the data extraction form. Decision- framework (Popay et al., 2005) was applied.
making reliability for study inclusion was evaluated Influenced by developmental psychopathology theory 65
for 20% of all studies screened. Percent agreement, and by the four Ps for case conceptualization (Vasey
calculated as the percentage of studies that were & Dadds, 2001; Winters, Hanson, & Stoyanova,
agreed upon between two authors, ranged from 0.83 2007), this review categorized factors related to antici-
15 to 0.95 indicating strong inter-rater agreement. patory distress to painful medical procedures as pre-
disposing, precipitating, perpetuating, or present 70
Assessment of Risk of Bias factors. Present factors were chosen (instead of protec-
A nuanced approach was necessary as the purpose of tive factors) due to the emphasis on “procedure” or
this review was not to evaluate treatment outcomes or contextual factors in the literature. Additionally, fac-
to make recommendations about practice. The state of tors that could be considered protective factors were
20 the literature in the area of anticipatory distress is collapsed within predisposing, perpetuating, and pre- 75
such that the preponderance of research is observa- sent factors as it made more conceptual sense based
tional, not experimental, in nature. However, assess- on how these factors were operationalized in the medi-
ment of risk of bias within observational studies was cal literature. For the purposes of this review, predis-
deemed necessary despite the lack of randomization. posing factors were operationalized as inherent
25 Risk of bias was assessed for the 77 included studies variables that increase the child’s risk for anticipatory 80
using the Cochrane Collaboration methodology for distress, for example, preexisting aspects of the child
systematic reviews (Higgins & Green, 2011). The ma- such as age, gender, or temperament as well as socio-
jority of the studies included in the review (70 studies) demographic variables of the parent or environment.
were not randomized controlled trials. In the Risk of Precipitating factors were conceptualized as factors
30 Bias tool created by the Cochrane Collaboration, the that lead to the onset of anticipatory distress to painful 85
first three criteria (random sequence generation, allo- medical procedures such as a negative pain event or
cation concealment, and blinding of participants) are previous experience with pain. Perpetuating or main-
only relevant for randomized controlled trials. As taining factors (Carr, 1999) were factors that likely
such, for observational and retrospective studies, only extend or preserve the problem such as parent behav-
35 the last four criteria were used to make judgments: ior that maintains the child’s distress both inside and 90
blinding of outcome assessment, incomplete outcome outside the medical procedure. Finally, present factors
data, selective reporting, and other sources of bias. were variables that occurred at the time of the proce-
This adaptation was based on the decision not to pe- dure and could positively or negatively influence the
nalize nonrandomized observational studies for being child’s anticipatory distress. It should be noted that
40 evaluated against criteria for randomization. The predisposing, precipitating, perpetuating, and present 95
Cochrane collaboration recently launched a risk of factors are not mutually exclusive categories as some
bias tool for nonrandomized studies of interventions factors may apply to multiple categories. These factors
(Sterne, Higgins, & Reeves, 2014); however, at the may also interact to compound anticipatory distress.
time of this manuscript, trainings were only beginning For the purposes of the review, the lead author and se-
45 to be offered and the tool was not yet widely used. nior author categorized each factor for parsimony and 100
Established tools to evaluate risk of bias in nonran- ease of interpretation.

domized studies were also considered (e.g., Down &
Black, 1998). However, given the number of studies in
the review, an abbreviated tool was selected. Two au- Results
50 thors evaluated risk of bias and consensus decisions Results of the Search
were made where authors disagreed. All studies were The search strategy retrieved 7,088 abstracts to review
classified as high, unclear, or low risk of bias. If one of against the inclusion criteria. Four individuals 105
the criteria was rated as “high,” the overall study rat- screened the initial 7,088 abstracts against inclusion
ing was considered to be high risk. “Unclear risk of criteria. Based on these criteria, the full article was
retrieved for 159 studies. Eighty-two articles for which overall findings synthesis have been presented in
the full text was retrieved were excluded from the re- Tables I–IV as well as in Figure 2.
view. A total of 77 full-text studies were included in
the review. The review process followed the Preferred Predisposing Factors 55
5 Reporting Items for Systematic Reviews and Meta- Child
Analyses (PRISMA) guidelines (Moher et al., 2009; As seen in Table I, 10 variables were identified for
Figure 1). Tables I–IV provide detailed overviews of child predisposing factors (Table I). Results suggest
the included studies including age range, sample size, that the data regarding age were inconclusive with al-
country of origin, procedure, design, and risk of bias most half the studies showing no age effect, and the 60
10 rating. In summary, most included studies were majority of the other studies suggesting younger chil-
observational, from North America, encompassed a dren have higher anticipatory distress. The overall risk
broad age range, and were based on surgical or nee- of bias for age was unclear (Table I). For gender, al-
dle-related procedures. The most common procedures though results were varied, most studies (20/26) re-
included surgery or operative procedures (29), immu- ported there was no effect of gender on anticipatory 65
15 nizations/injections (13), dental procedures (11), and distress in children, while six studies found that girls
venipuncture procedures (8). A total of 15,106 partici- experienced more anticipatory distress than boys. The
pants were included in the review. overall risk of bias for gender was unclear (Table I).
In terms of the types of outcome measures, 43 mea- Four studies examined the effect of race on anticipa-
sured anxiety, 15 measured fear, 13 measured distress, tory distress, with the majority suggesting no effect. 70
20 and 6 measured baseline pain. The majority of out- The overall risk of bias was unclear (Table I). Birth or-
come measures were self-report (35), while the re- der was not found to have an effect on child anticipa-
maining outcome measures were behavioral (11) or tory distress as indicated by three studies. The effect of
observer reported (31). number of siblings and sibling order was investigated
by three studies and was found to have no effect. 75
Risk of Bias Child illness and child intelligence were both investi-
25 Risk of bias was assessed for the 77 studies included in gated by one study and were both found to positively
the review using the Cochrane Collaboration method- predict child anticipatory distress.
ology for systematic reviews. As all but 7 studies were For child psychopathology, the overall findings (6/8)
nonrandomized controlled trials, 15 studies were eval- supported the positive relationship between preexist- 80
uated to have high risk of bias, 16 studies had unclear ing child psychopathology and increased child antici-
30 risk of bias, and 46 studies had low risk of bias patory distress to painful medical procedures. The
(Tables I–IV). overall risk of bias rating was unclear (Table I). For
child temperament, the overall findings (8/11 studies)
support a positive relationship between difficult child 85
Factors Included in the Review temperament and increased child anticipatory distress.
The list of factors that predict anticipatory distress to The overall risk of bias score for temperament was
painful medical procedures can be found in the left- unclear (Table I). Finally, there were inconclusive re-
35 most column of Tables I–IV. Overall, there were 31 sults for child attachment from two low risk of bias
factors that were examined for their relationship to studies. One study (Horton et al., 2015) indicated that 90
predict anticipatory distress. infants with avoidant infant attachment had lower an-
ticipatory distress where as another study (Lumley,
Melamed, & Abeles, 1993) found no effect.
Overall Findings
The overall goal of the review was to synthesize the lit-
40 erature on factors that predict anticipatory distress to Parent
painful medical procedures. Following data extrac- A total of 12 studies provided evidence for parent pre- 95
tion, the lead and senior authors synthesized the re- disposing factors that are associated with child antici-
sults into the summary figure. The summary figure patory distress to painful medical procedures. Results
(Figure 2) includes most of the information from are found in Table I. Overall findings suggest that par-
45 Tables I to IV and highlights the contribution of pre- ent anxious predisposition, and pain experience or
disposing, precipitating, perpetuating, and present fac- fear of pain were all associated with increased antici- 100
tors influencing the child’s anticipatory distress. Only patory distress. The results for parent education and
factors with two studies or more that were similar in coping style were found to be inconclusive.
nature were included in Figure 2. Moreover, in the
50 summary figure, factors were subcategorized accord- Health Professional
ing to child, parent, health care professional, and/or No health professional factors were found under the
contextual domains. Finally, the risk of bias and predisposing domain. 105
6
Table I. Predisposing Factors of Anticipatory Anxiety
Study Age (years) N Country Procedure Design Risk of Result Summary of Results summary
bias bias
Child predisposing factors

1. Age (43 studies; N ¼ 9,890)
Bevan et al. (1990) 2–10 134 Canada Preop E Unclear Unclear (25 Inconclusive
Broome and Hellier (1987) 5–11 84 USA Medical R Low low, 8 high,
Caldwell-Andrews, Kain, Mayes, Kerns, and Ng 2–12 289 USA Preop O Unclear 10 unclear)
(2005)
Carpenter (1992) 4–18 73 USA Venipuncture O Low
Chen, Craske, Katz, Schwartz, and Zeltzer 3–18 55 USA LP RCT High
(2000)
Chorney & Kain (2009) 2–10 293 USA Preop O Low
Chorney, Torry, McLaren, Chen, and Kain 2–10 293 USA Preop O Unclear
(2009)
Dahlquist, Power, Cox, and Fernbach (1994) 2–7 63 USA BMA O Low
8–17
Dahlquist et al. (2001) 5–15 45 USA Intramuscular in- O Low
jection and LP
Dahlquist and Pendley (2005) 2.4–5.1 29 USA Immunization RCT High
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low
Fukuchi et al. (2005) 2–12 78 Brazil Preop O Unclear
Hatava, Olsson, and Lagerkranser (2000) 2–10 160 Sweden Preop E Unclear
Holm-Knudsen, Carlin, and McKenzie 0–14 2,122 Australia Preop O Unclear
(1998)
Hosey et al. (2006) 2–14 407 UK Dental O Low
Howe et al. (2011) 4.9–16.2 23 USA Injection, Finger O Unclear
sticks
Kain, Mayes, Weisman, and Hofstadter (2000) 3–10 60 USA Preop O High
Lilley, Craig, and Grunau (1997) 0.17–1.5 75 Canada Immunization O Low
Mahoney, Ayers, and Seddon (2010) 7–16 50 UK Venipuncture O Low
Melamed, Meyer, Gee, and Soule (1993) 4–12 46 USA Preop O Low
Olak et al. (2013) 8–10 344 Estonia Dental R Low
Taddio et al. (2012) 6–17 1,024 Canada Immunization R High
Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low
Kain, Mayes, O’Connor, and Cicchetti (1996) 2–10 163 USA Preop O Low þ
Tickle et al. (2009) 5–9 799 UK Dental O High þ
Al-Jundi and Mahmood (2010) 2–12 118 Jordan Dental O High 1
Bijttebier and Vertommen (1998) 2.75–12.75 47 Belgium Venipuncture O High 1
Carr, Lemanek, and Armstrong (1998) 3–12 62 USA Allergy skin testing O Low 1
Claar, Walker, and Smith (2002) 8–18 100 USA EGD O Low 1
deVos et al. (2012) M ¼ 3.1 18 USA Immunotherapy O High 1
Injections
Field, Alpert, Vega-Lahr, Goldstein, and Perry 4–10 56 USA Preop O Low 1
(1988)
(continued)
Racine et al.
Table I. Continued
bias bias
Fox and Newton (2006) 5–17 38 UK Dental RCT Low 1

Goodenough, Champion, Laubreaux, Tabah, 3–17 117 Australia Venipuncture O Unclear 1
and Kampel (1998)
Jacobson et al. (2001) 1–6 150 USA Immunization O Low 1
Lumley, Melamed, and Abeles (1993) 4–10 50 USA Preop O Low 1
Ortiz et al. (2014) 8–16 437 Mexico Dental O Unclear 1
McMurtry, Noel, Chambers, and McGrath 5–10 100 Canada Venipuncture O Low 1
(2011)
Mekarski and Richardson (1997) 2.5–13 324 Canada Dental O Low 1
Messeri, Caprilli, and Busoni (2004) 2–14 39 Italy Preop O Low 1
Siaw, Stephens, and Holmes (1986) 3.5–12.8 30 USA Preop O Unclear 1
Predisposing, Precipitating, Perpetuating
Thompson (1994) 8–12 43 USA Preop O Low 1

Wright, Stewart, and Finley (2010) 3–6 61 Canada Preop E Low 1
Wright, Stewart, and Finley (2013) 3–6 61 Canada Preop O Low 1
2. Gender (26 studies; N ¼ 6,483)
Al-Jundi and Mahmood (2010) 2–12 118 Jordan Dental O High 1 Unclear (18 No effect
Bearden, Feinstein, and Cohen (2012) 3–5 90 USA Immunization O Low 1 low, 6 high, 2
Bijttebier and Vertommen (1998) 2.75–12.75 47 Belgium Venipuncture O High 1 unclear)
Carr, Lemanek, and Armstrong (1998) 3–12 62 USA Allergy test O Low 1
Colares, Franca, Ferreira, Amorim Filho, and 5–12 970 Brazil Dental R Low 1
Oliverira (2013)
Dahlquist et al. (2001) 5–15 45 USA Intramuscular in- O Low 1
jection and LP
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low 1
Fortier, Martin, MacLaren Chorney, Mayes, and 11–18 59 USA Preop O Low 1
Kain (2011)
Fox and Newton (2006) 5–17 38 UK Dental RCT Low 1
Gazal and Mackie (2007) 2–12 201 UK Dental O Low 1
Goodenough et al. (1998) 3–17 117 Australia Venipuncture O Unclear 1
Hanas et al. (2002) 1–15 41 Sweden Insulin injection RCT High 1
Horton et al. (2015) 1–1.5 130 Canada Immunization O Low 1
Kain et al. (2000) 3–10 60 USA Preop O High 1
Ortiz et al. (2014) 8–16 437 Mexico Dental O Unclear 1

Broome and Hellier (1987) 5–11 84 USA Medical O Low þGirls
Logan and Rose (2004) 12–18 102 USA Preop O Low þGirls
McMurtry et al. (2011) 5–10 100 Canada Venipuncture O Low þGirls
Olak et al. (2013) 8–10 344 Estonia Dental R Low þGirls
Taddio et al. (2012) 6–17 1,024 Canada Immunization R High þGirls
7
Tickle et al. (2009) 5–9 799 UK Dental O High þGirls

(continued)
8
Table I. Continued
bias bias
3. Child psychopathology (8 studies; N ¼ 2,053)

Davidson et al. (2006) 3–12 1,250 Australia Preop O Low þ Unclear (6 low, Positively predicts an-
Ericsson, Wadsby, and Hultcrantz (2006) 5–15 92 Sweden Preop RCT High þ 2 high) ticipatory distress.
Fortier et al. (2011) 11–18 59 USA Preop O Low þ
Hosey et al. (2006) 2–14 407 UK Dental O Low þ
Kain et al. (2000) 3–10 60 USA Preop O High þ
Wright, Stewart, and Finley (2013) 3–6 61 Canada Preop O Low þ
Kiley and Polillio (1997) School age 74 USA Immunization E Low 1
4. Temperament (11 studies; N ¼ 2,235)
Arnrup, Broberg, Berggren, and Bodin (2003) 4–12 86 Sweden Dental E High þ Unclear (7 low, Positively predicts an-
Chen et al. (2000) 3–18 55 USA LP RCT High þ 3 high, 1 ticipatory distress.
Cropper et al. (2011) 4–7 84 UK GA (Cochlear O Low þ unclear)
Implant)
Fortier et al. (2011) 11–18 59 USA Preop O Low þ
Jacobson et al. (2001) 1–6 150 USA Immunization O Low þ
Kain et al. (1996) 2–10 163 USA Preop O Low þ
Lee and White-Traut (1996) 3–7 137 USA Venipuncture O Unclear þ
Horton et al. (2015) 1–1.5 130 Canada Immunization O Low 1
5. Race (4 studies; N ¼ 296)
Broome and Hellier (1987) 3–15 140 USA Medical R Low 1 Unclear (3 low, No effect
Kain et al. (2000) 3–10 60 USA Preop O High 1 1 high)
Melamed et al. (1993) 4–12 46 USA Preop O Low þ
6. Birth order (3 studies; N ¼ 1,352)
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low 1 Low (3 low) No effect
Fortier et al. (2011) 11–18 59 USA Preop O Low 1
7. Number of siblings/sibling order (3 studies; N ¼ 1,369)
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low 1 Unclear (2 low, No effect
Fortier et al. (2011) 11–18 59 USA Preop O Low 1 1 high)
Kain et al. (2000) 3–10 60 USA Preop O High 1
8. Child illness (1 study; N ¼ 80)
Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low þ Low CNS disease positively
predicts.
9. Attachment (2 studies; N ¼ 180)

Horton et al. (2015) 1–1.5 130 Canada Immunization O Low Low Inconclusive
(continued)
Racine et al.
Table I. Continued
bias bias
10. Intelligence (1 study; N ¼ 60)

Kain et al. (2000) 3–10 60 USA Preop O High þ High Positively predicts an-
ticipatory distress.
Parent predisposing factors
1. Anxious predisposition (4 studies; N ¼ 1,532)
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low þ Low Positively predicts an-
Kain et al. (1996) 2–10 163 USA Preop O Low þ ticipatory distress.
Messeri, Caprilli, and Busoni (2004) 2–14 39 Italy Preop O Low þ
Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low 1

2. Beliefs about coping and coping style (2 studies; N ¼ 349)
Caldwell-Andrews et al. (2005) 2–12 289 USA Preop O Unclear Unclear Inconclusive.
3. Pain experience and fear of pain (3 studies; N ¼ 1,185)
Ellerton and Merriam (1994) 3–15 75 Canada Preop R Unclear þ High (1 unclear, Positively predicts an-
Arnup (2003) 4–12 86 Sweden Dental E High 1 2 high) ticipatory distress.
Taddio et al. (2012) 6–17 1,024 Canada Immunization R High þ
4. Parental education (2 studies; N ¼ 1,029)
Colares et al. (2013) 5–12 970 Brazil Dental R Low þ Low (2 low) Inconclusive
5. Parent gender (1 study; N ¼ 437)

Ortiz et al. (2014) 8–16 437 Mexico Dental O Unclear þ Unclear Mothers associated
with more anticipa-
tory distress.
Contextual predisposing factors
1. Previous hospitalization of child or sibling (5 studies; N ¼ 1,451)
Broome and Hellier (1987) 5–11 84 USA Medical R Low 1 Unclear (4 low, No effect of
Field et al. (1988) 4–10 56 USA Preop O Low 1 1 high) previous
deVos et al. (2012) M ¼ 3.1 18 USA Immunotherapy O High 1 hospitalization.
Injections
Broome and Hellier (1987) 5–11 84 USA Medical R Low þ
(sib
ling)
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low þ
2. Other contextual factors (4 studies; N ¼ 3,079)
Colares et al. (2013) 5–12 970 Brazil Dental R Low þ (no visits)
9
10 Racine et al.
Contextual
study; E ¼ experimental study; R ¼ retrospective study; RCT ¼ randomized controlled trial; Preop ¼ preoperative; LP ¼ lumbar puncture; BMA ¼ bone marrow aspiration; CNS ¼ central nervous
Note. þ Factor has a positive relationship with anticipatory distress; Factor has a negative relationship with anticipatory distress; 1 ¼ no effect or significant relationship; O ¼ observational
Two factors were identified as contextual predisposing
dental visits posi-

Results summary
factors: previous hospitalization of the child or sibling
tively predict.
(five studies) and other contextual factors (four stud-
No/irregular
ies). For previous hospitalization, four of the five stud- 110
ies found no effect of previous hospitalization on child
anticipatory distress; however, one study (Broome &
Unclear (2 low, Hellier, 1987) found that hospitalization of a sibling
(but not of self) was associated with higher anticipa-
Summary of
tory distress. The overall risk of bias for this factor 115
2 high)
was unclear. In terms of other contextual factors, the

overall findings showed that having never visited a
bias
dentist and having irregular visits to the dentist are

positively associated with child anticipatory distress.
Other demographic variables had mixed results. The 120
evidence for this factor was unclear.
þ (visits)
1(demo
1 (SES)
hics)
grap
Result
Precipitating Factors
Child
Two broad factors were identified as precipitating fac-
Risk of
High
High
tors that contributed to the onset of anticipatory dis-

Low
125
bias
tress to painful medical procedures (Table II). The first

factor was general and specific negative pain events
Design
(33 studies). The results showed that 17 studies found

O
O
that previous negative experiences positively predict

child anticipatory distress, whereas 12 studies found 130
no effect of previous painful events. Four studies indi-
cated that history of painful procedures was in fact as-
sociated with decreased child anticipatory distress.
Procedure
Overall, the evidence points toward a positive rela-

Dental
1,250 Australia Preop
Preop
tionship between previous pain events and child antic- 135

ipatory distress based on unclear risk of bias.
Country
The second precipitating child factor that was iden-

tified was previous child/adolescent behavior (five
USA
UK
studies). Overall, results indicate that previous pain

behavior positively predicts child anticipatory distress. 140
799
60
The risk of bias was unclear.

Age (years) N
Parent
No parent factors were found under the precipitating
3–12
3–10
5–9
domain.
Health Professional 145

No health professional factors were found under the
precipitating domain.
Contextual
No contextual factors were found under the precipi-
tating domain. 150
Davidson et al. (2006)
Table I. Continued
Tickle et al. (2009)
Perpetuating Factors
Kain et al. (2000)
Child
As listed in Table III, four factors were identified as
child perpetuating factors: child knowledge (seven
system.
Study
studies), child coping style (four studies), child cogni- 155

tions (three studies), and other child behaviors (two
Table II. Precipitating Factors of Anticipatory Anxiety
Study Age (years) N Country Procedure Design Risk of Result Summary of Results
bias bias summary
Child precipitating factors

1. General and specific negative pain events (33 studies; N ¼ 5,186)
Al-Jundi and Mahmood (2010) 2–12 118 Jordan Dental O High þ Unclear (20 Positively pre-
Bijttebier and Vertommen (1998) 2.75–12.75 47 Belgium Venipuncture O High þ low, 7 high, 6 dicts anticipa-
Caes et al. (2014) 0.6–15 28 Canada BMA or LP O Unclear þ unclear) tory distress.
Carillo-Diaz, Crego, Armfield, and Romero (2013) 8–18 179 Spain Dental R Unclear þ
Colares et al. (2013) 5–12 970 Brazil Dental R Low þ

Cropper et al. (2011) 4–7 84 UK Preop O Low þ
Ellerton and Merriam (1994) 3–15 75 Canada Preop R Unclear þ

Hatava, Olsson, and Lagerkranser (2000) 2–10 160 Sweden Preop E Unclear þ
Jacobson et al. (2001) 1–6 150 USA Immunization O Low þ
Kain et al. (1996) 2–10 163 USA Preop O Low þ
Lee and White-Traut (1996) 3–7 137 USA Venipuncture O Unclear þ
Lumley, Melamed, and Abeles (1993) 4–10 50 USA Preop O Low þ (quality)
Noel, McMurtry, Chambers, and McGrath (2010) 5–10 48 Canada Venipuncture O Low þ
Olak et al. (2013) 8–10 344 Estonia Dental R Low þ
Pillai Riddell et al. (2011) 0–1 731 Canada Immunization O Low þ
Taddio et al. (2002) Newborns 66 Canada Venipuncture, O Low þ
(>1 Vitamin K
month) injections

Arnrup et al. (2003) 4–12 86 Sweden Dental E High 1
Broome, Lillis, McGahee, and Bates (1994) 3–15 14 USA LP O Low 1
Carr, Lemanek, and Armstrong (1998) 3–12 62 USA Allergy testing O Low 1
Chorney & Kain (2009) 2–10 293 USA Preop O Low 1
Ericcson, Wadsby, and Hultcrantz (2006) 5–15 92 Sweden Preop RCT High 1
Dahlquist et al. (2001) 5–15 45 USA Intramuscular O Low 1
injection and
LP
Goubet, Clifton, and Shah (2001) 0–0.04 12 USA Heel-lance O Low 1
Owens and Todt (1984) Newborns 20 USA Heel-lance O Low 1
(>1
month)
McMurtry et al. (2011) 5–10 100 Canada Venipuncture O Low 1
Mahoney, Ayers, and Seddon (2010) 7–16 50 UK Venipuncture O Low
deVos et al. (2012) M ¼ 3.1 18 USA Immunotherapy O High
Injections
11
(continued)
12 Racine et al.
studies). For child knowledge, the results were incon- 50

45
40
35
Note. þ Factor has a positive relationship with anticipatory distress; Factor has a negative relationship with anticipatory distress; 1, no effect or significant relationship; O ¼ observational study;
clusive. This was based on unclear evidence. The evi-
dence for child coping style was inconclusive based on
dicts anticipa-
tory distress.
Positively pre-
unclear risk of bias. For child cognitions (three stud-
ies), overall results suggest that child cognitions in-
summary
Results
cluding high threat appraisal, lower perceived control, 55
and high aversion to the procedure were all associated
with higher child anticipatory distress, based on stud-
ies with unclear risk of bias. Finally, the evidence for
Unclear (4 low,
other child behaviors (two studies) was inconclusive
1 unclear)
Summary of
as studies highlighted different child behaviors associ-
E ¼ experimental study; R ¼ retrospective study; RCT ¼ randomized controlled trial; Preop ¼ preoperative; LP ¼ lumbar puncture; BMA ¼ bone marrow aspiration.
60
ated with increased or decreased anticipatory distress.
bias
Parent
Four factors were identified as parent perpetuating
Result
factors: parent behavior (7 studies), parent situational

1

þ
þ
þ
þ
distress (19 studies), parent anticipation of child dis- 65

tress (5 studies), and parent self-efficacy/attitudes (2
Unclear
Unclear
Risk of
High
studies) (Table III). Overall findings suggest that most

Low
Low
Low
Low
bias
parent behavior, parent situational distress, and par-

ent anticipation of child distress were associated with
Design
RCT
increased anticipatory distress (Table III). 70

O
O
O
O
O
O
Insulin injection
Insulin injection
Health professional
Immunization
Venipuncture
and Finger
No health professional factors were found under the

Procedure
perpetuating domain.
sticks
Preop
Preop
Preop
Contextual
Australia
Australia
Country
No contextual factors were found under the perpetu-

Canada
Sweden
75
ating domain.
USA
USA
USA
2,122
1,250
Present Factors
150
100
23
41
59
N
Child
As listed in Table IV, one factor was identified for
Age (years)
child present factors: idiosyncratic needs. One study 80

4.9–16.2
(Ameringer, Elswick Jr, Shockey, & Dillon, 2013)

11–18
1–15
0–14
3–12
5–10
showed that fatigue and nausea were positively associ-

1–6
ated with child anticipatory distress prior to chemo-

therapy with a low risk of bias.
2. Previous pain behavior (5 studies; N ¼ 3,681)
Holm-Knudsen, Carlin, and McKenzie (1998)
Parent 85
One parent present factor was identified: parental
presence during a painful medical procedure. The
overall results for this factor are inconclusive (Table
IV). The risk of bias for this factor was unclear.
McMurtry et al. (2011)
Davidson et al. (2006)
Jacobson et al. (2001)
Health Professional 90
Table II. Continued
Fortier et al. (2011)

Hanas et al. (2002)
Howe et al. (2011)
One factor was identified as a health professional pre-

sent factor: health professional behavior (three stud-
ies). Overall, evidence suggests that distress promoting
behavior by health care professionals is associated
Study
with higher child anticipatory distress. The overall 95

risk of bias was unclear.
Table III. Perpetuating Factors of Anticipatory Anxiety
Study Age (years) N Country Procedure Design Risk of bias Result Summary of bias Results summary
Child perpetuating factors

1. Child knowledge (7 studies; N ¼ 1,850)
Claar, Walker, and Barnard (2002) 8–17 100 USA EGD O Low 1 Unclear (5 low, 2 Inconclusive
Crandall, Lammers, Senders, Braun, and Savedra 7–13 60 USA Preop E Low 1 unclear)
(2008)
Claar, Walker, and Smith (2002) 8–18 100 USA EGD O Low 1
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low
Hatava, Olsson, and Lagerkranser (2000) 2–10 160 Sweden Preop E Unclear
Siaw, Stephens, and Holmes (1986) 3.5–12.8 30 USA Preop O Unclear
2. Child coping style (4 studies; N ¼ 174)

Bijttebier and Vertommen (1998) 2.75–12.75 47 Belgium Venipuncture O High þ Unclear (1 high, 3 Inconclusive
Field et al. (1988) 4–10 56 USA Preop O Low 1 low)
Smith, Ackerson, and Blotcky (1989) 6–18 28 USA BMA and LP O Low 1
Thompson (1994) 8–12 43 USA Preop O Low
3. Other child behaviors (2 studies; N ¼ 368)
Chorney & Kain (2009) 2–10 293 USA Preop O Low þ Unclear (1 low, 1 More research
Kain et al. (1998) 2–12 75 USA Preop RCT Unclear unclear) needed.
4. Child cognitions (3 studies; N ¼ 352)
Carillo-Diaz et al. (2013) 8–18 179 Spain Dental R Unclear þ (expectancy) Unclear (2 low, 1 Negative child cog-
Claar, Walker, and Smith (2002) 8–18 100 USA EGD O Low þ unclear) nitions positively
Carpenter (1992) 4–18 73 USA Venipuncture O Low _ predict child an-
Carillo-Diaz et al. (2013) 8–18 179 Spain Dental R Unclear þ (appraisal) ticipatory
distress.
Parent perpetuating factors
1. Parent behavior (7 studies; N ¼ 1,962)
Blount, Sturges, and Powers (1990) 5–13 22 USA BMA or LP O Unclear þ Unclear (5 low, 2 Parent behaviour is
Chorney et al. (2009) 2–10 293 USA Preop O Unclear þ, (distraction) unclear) associated with
Dahlquist Power, Cox, and Fernbach (1994) 2–7, 8–17 63 USA BMA O Low þ anticipatory dis-
Dahlquist et al. (2001) 5–15 45 USA Intramuscular in- O Low þ tress. Direction
jection and LP dependent on
Lisi et al. (2013) 0–1 760 Canada Immunization O Low þ type of behavior.
Noel et al. (2010) 5–10 48 Canada Venipuncture O Low þ
Pillai Riddell et al. (2011) 0–1 731 Canada Immunization O Low 1
2. Parent situational distress (19 studies; N ¼ 4,998)
Arnrup et al. (2003) 4–12 86 Sweden Dental E High þ Unclear (7 high, 9 Positively predicts
Bearden et al. (2012) 3–5 90 USA Immunization O Low þ low, 3 unclear) anticipatory
Bevan et al. (1990) 2–10 134 Canada Preop E Unclear þ distress.
Caes et al. (2014) 0.6–15 28 Canada BMA or LP O Unclear þ
Colares et al. (2013) 5–12 970 Brazil Dental R Low þ
Dahlquist Power, Cox, and Fernbach (1994) 2–7, 8–17 63 USA BMA O Low þ
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low þ
Gazal and Mackie (2007) 2–12 201 UK Dental O Low þ
13
(continued)
14
Table III. Continued

Study Age (years) N Country Procedure Design Risk of bias Result Summary of bias Results summary
Hatava, Olsson, and Lagerkranser (2000) 2–10 160 Sweden Preop E Unclear þ
LaMontagne, Hepworth, Johnson, and Cohen 8–17 90 USA Preop O High þ
(1996)
Messeri, Caprilli, and Busoni (2004) 2–14 39 Italy Preop O Low þ
Olak et al. (2013) 8–10 344 Estonia Dental R Low þ
Tourigny (1992) 2–10 50 Canada Preop O High þ
Al-Jundi and Mahmood (2010) 2–12 118 Jordan Dental O High 1
Dahlquist and Pendley (2005) 2.4–5.1 29 USA Immunization RCT High 1
Hosey et al. (2006) 2–14 407 UK Dental O Low 1
Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low 1
3. Parent anticipation of child distress (5 studies; N ¼ 742)
Jacobson et al. (2001) 1–6 150 USA Immunization O Low þ Unclear (1 unclear, Positively predicts
Ortiz et al. (2014) 8–16 437 Mexico Dental O Unclear þ 4 low) anticipatory
Lumley, Melamed, and Abeles (1993) 4–10 50 USA Preop O Low þ distress.
Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low þ
Srivastava, Betts, Rosenberg, and Kainer (2001) 0–6.5 25 Australia Micturating O Low 1
cystoure
throgram
4. Parent self-efficacy/attitude toward procedure (2 studies; N ¼ 236)
Arnrup et al. (2003) 4–12 86 Sweden Dental E High 1 Unclear More research is
Jacobson et al. (2001) 1–6 150 USA Immunization O Low 1 needed.
Note. þ Factor has a positive relationship with anticipatory distress; Factor has a negative relationship with anticipatory distress; 1, no effect or significant relationship; O ¼ observational study;
E ¼ experimental study; R ¼ retrospective study; RCT ¼ randomized controlled trial; Preop ¼ preoperative; LP ¼ lumbar puncture; BMA ¼ bone marrow aspiration;
EGD ¼ esophagogastroduodenoscopy.
Racine et al.
Table IV. Present Factors of Anticipatory Anxiety
Study Age N Country Procedure Design Risk Result Summary of bias Results summary
(years) of bias
Child present factors

1. Idiosyncratic needs (1 study; N ¼ 9)
Ameringer et al. (2013) 13–18 9 USA Chemotherapy O Low þ Low More research is
needed.
Parent present factors
1. Parent presence (6 studies; N ¼ 2,159)
Al-Jundi and Mahmood (2010) 2–12 118 Jordan Dental O High 1 Unclear (3 low, 2 Inconclusive
Bevan et al. (1990) 2–10 134 Canada Preop E Unclear 1 high, 1 unclear)
Tourigny (1992) 2–10 50 Canada Preop O High þ
Messeri, Caprilli, and Busoni (2004) 2–14 39 Italy Preop O Low

Kain et al. (2006) 2–12 568 USA Preop O Low ,þ
Health care professional factors
1. Health professional behavior (3 studies; N ¼ 386)
Noel et al. (2010) 5–10 48 Canada Venipuncture O Low þ Unclear (2 low, 1 Distress promoting
Chorney et al. (2009) 2–10 293 USA Preop O Unclear þ unclear) behaviour posi-
Dahlquist et al. (2001) 5–15 45 USA Intramuscular in- O Low þ (nurse) tively predicts
jection and LP 1 (MD) child anticipa-
tory distress.
Contextual present factors
1. Environmental factors (15 different studies; N ¼ 4,926)
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low admission type Unclear (9 low, 4 More research
Holm-Knudsen, Carlin, and McKenzie (1998) 0–14 2,122 Australia Preop O Unclear induction location high, 2 unclear) needed.
Kain, Wang, Mayes, Krivutza, and Teague 2–7 70 USA Preop RCT High reduced sensory
(2001) stimulation
Mekarski and Richardson (1997) 2.5–13 324 Canada Dental O Low þ dental work severity
deVos et al. (2012) M ¼ 3.1 18 USA Immunotherapy O High 1 injection personnel
Injections
Horton et al. (2015) 1–1.5 130 Canada Immunization O Low 1 number of needles
Davidson et al. (2006) 3–12 1,250 Australia Preop O Low þ longer procedure
deVos et al. (2012) M ¼ 3.1 18 USA Immunotherapy O High þ intervals between
Injections needles
Holm-Knudsen, Carlin, and McKenzie (1998) 0–14 2,122 Australia Preop O Unclear 1 fasting time
Al-Jundi and Mahmood (2010) 2–12 118 Jordan Dental O High þ referral reason
Dahlquist Power, Cox, and Fernbach (1994) 2–7, 8–17 63 USA BMA O Low 1 time since diagnosis
Dahlquist et al. (2001) 5–15 45 USA Intramuscular in- O Low 1 time since diagnosis
jection and LP
Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low 1 time since diagnosis
Dahlquist and Pendley (2005) 2.4–5.1 29 USA Immunization RCT High time since diagnosis
Holm-Knudsen, Carlin, and McKenzie (1998) 0–14 2,122 Australia Preop O Unclear 1 type of case
Ortiz et al. (2014) 8–16 437 Mexico Dental O Unclear 1 procedure
Wright, Stewart, and Finley (2010) 3–6 61 Canada Preop E Low 1 surgery type
Wollin et al. (2004) 5–12 120 Australia Preop O Low þ various factors
15
(continued)
16 Racine et al.
Note. þ, factor has a positive relationship with anticipatory distress; , factor has a negative relationship with anticipatory distress; 1, no effect or significant relationship; O ¼ observational study;
Contextual
Results summary
One broad factor, environmental factors, was identi-
fied for contextual present factors. Fifteen studies in-
vestigated the effects of various contextual factors on 100
child anticipatory distress during the painful medical
procedure (e.g., type of admission and severity of pro-
Summary of bias cedure). Results vary based on the study.
Discussion
E ¼ experimental study; R ¼ retrospective study; RCT ¼ randomized controlled trial; Preop ¼ preoperative; LP ¼ lumbar puncture; BMA ¼ bone marrow aspiration.
The purpose of this review was to summarize the find- 105
ings of studies that examine the factors that predict
anticipatory distress to painful medical procedures in
children. The overarching goal of this review was to
1 number of needles
1 preadmission visit
qualitatively synthesize the literature on the factors

that predict anticipatory distress to painful medical 110
procedures into a summary figure using predisposing,
precipitating, perpetuating, and present factors as a
Result
framework. The following paragraphs will discuss key

findings and patterns from the summary figure
(Figure 2) of the review in the context of methodologi- 115
of bias
High
cal differences and risk of bias within studies. Only

Low
Design Risk
factors with two or more included studies of a similar

nature that can be found in the summary figure will be
discussed. Finally, clinical implications, areas for fu-
O
ture research based on the summary figure and limita- 120

tions of the review will be highlighted.
Immunotherapy
Injections
Procedure
Predisposing Factors
Child
Preop
There were some interesting patterns among the child

predisposing factors. First, there is clear evidence that 125
Country
child psychopathology and difficult, fearful, or shy

USA
USA
child temperament are individual child factors that in-

crease the risk of child anticipatory distress. This find-
ing is in line with developmental literature suggesting
18
59
that children who have internalizing or externalizing

N
130
problems have more difficulty regulating their affect
M ¼ 3.1
(Bradley, 2003). Preexisting psychopathology or diffi-

(years)
11–18
Age
cult temperament may be important factors to screen

for prior to a medical procedure or surgery to have an
understanding of how a child might respond or cope 135
with the procedure. The risk of bias subsuming this
factor was generally unclear because there were 6 low
and 2 high-rated studies included.
Second, gender does not appear to play an impor-
tant role in predicting anticipatory distress. Although 140
some studies did find that girls experience higher an-
ticipatory distress to medical procedures, the majority
of studies did not find an effect. The studies that found
Table IV. Continued
Fortier et al. (2011)

deVos et al. (2012)
an effect for girls had participants closer to pubertal

age, which may have played a role. 145
Some predisposing child factors yielded inconclu-
sive results. Despite the large body of research (43
Study
studies) that examined the effect of age on child antici-

patory distress, the research on this factor does not
Figure 2. Summary figure of results. FS ¼ findings synthesis; ROB ¼ risk of bias; þ ¼ factor has a positive relationship with
anticipatory distress; ¼ factor has a negative relationship with anticipatory distress; 1 ¼ no effect or significant relation-
ship; ? ¼ inconclusive results; U ¼ unclear risk of bias; L ¼ low risk of bias; H ¼ high risk of bias; # ¼ number of studies.
18 Racine et al.
seem to converge. Almost half the studies showed no not found to have an effect on child anticipatory dis- 55
effect of age, while the other half suggests younger chil- tress (only previous hospitalization of siblings did).
dren experience higher anticipatory distress. The type It may be that hospitalization itself is not suffi-
of medical procedure did not seem to systematically cient to lead to the development of fear but
5 differ between the two groups. Although the studies rather that negative experiences or vicarious fear are
that did not find an effect of age were more likely to much more salient. The risk of bias for this factor 60
have a low risk of bias, it is difficult to make conclu- was unclear as there was a mix of high and low
sions based on this. Methodological factors may also rated studies. Few predisposing contextual
have contributed to differences in results as the major- factors have been examined and more research is
10 ity of studies did not examine a discrete age range but needed.
rather averaged over large age ranges of up to 15 years.
Examining a restricted age range may also have con-
tributed to the lack of an effect. At this point, the re- Precipitating Factors 65
sults on age remain largely inconclusive, although the Child

15 results from this review point toward younger children Two broad factors were identified as factors that con-
experiencing more anticipatory distress than older chil- tributed to the onset of anticipatory distress to painful
dren. This is in line with the literature that indicates medical procedures. The first factor is general and spe-
that younger children are more likely to be fearful and cific negative pain events. Although the overall result 70
distressed and that this fear may increase and decrease is that previous negative pain events predict anticipa-
20 over the course of childhood (American Psychiatric tory distress, this was not uniformly the case across
Association, 2013). It may also be the case that the re- studies. Some reasons for this include risk of bias and
lationship between age and anticipatory distress is sample size. The studies that found an effect of previ-
nonlinear or co-varies with other factors. Future longi- ous negative procedures largely had low risk of bias 75
tudinal or cross-sectional studies could provide some and large sample sizes. The studies that found a nega-
25 insight into whether age is an important factor in pre- tive relationship between previous pain events and an-
dicting child anticipatory distress. ticipatory distress were methodologically different in
that they all involved short routine medical procedures
such as insulin injections (Hanas et al., 2002; Howe, 80
Parent Ratcliffe, Tuttle, Dougherty, & Lipman, 2011) and
Two parent predisposing factors that emerged as pre- immunotherapy injection (deVos et al., 2012). These
dicting increased child anticipatory distress are parent types of procedures provide repeated exposure to the
30 anxious predisposition and previous parent pain experi- stimuli whereby eventually extinction of the fear oc-
ence. The fact that a parent’s own anxiety and fear/ curs. Typically, developing children do not usually 85
experiences with pain are related to the child’s anticipa- have daily exposure to needles or surgery to facilitate
tory distress directly supports the transmission of anxi- extinction, which may explain the difference in finding
ety from parent to child. Previous work has for the studies. Furthermore, previous work in child
35 hypothesized the mechanisms by which this occurs, anxiety has demonstrated that direct conditioning is
such as through modeling and information transmission only one pathway to the development of anxiety prob- 90
(Rachman, 1977; Vasey & Ollendick, 2000). It may be lems in children (Vasey & Dadds, 2001). According to
that parents are discussing or demonstrating their fear retrospective reports of adults with phobia, model-
of pain as it relates to painful medical procedures, im- ing and information transmission were the most com-
40 pacting the anticipatory distress of their children. Future mon modes of fear acquisition with a minority
experimental research could examine how transmission reporting direct conditioning experiences (Vasey & 95
of fear of painful medical procedures occurs to develop Ollendick, 2000). This highlights that, although direct
targets for intervention. Risk of bias was variable across conditioning of a general or specific negative
factors ranging the full gamut from low to unclear to pain event may precipitate anticipatory distress for
45 high. Of note, the anxious predisposition has low risk of some children, multiple factors are at play, including
bias; thus, there is increased confidence in this finding. the frequency and severity of the painful medical 100
Inconclusive results were found for the impact of procedure.
parent education level on child anticipatory distress. The second factor that was identified as a child
The difference in finding may be due to the differences precipitating factor is previous child/adolescent
50 in education levels included in the studies. More re- behavior (five studies). There is evidence that a
search is needed in this area. child or adolescent’s previous behavior during a pain- 105
ful medical procedure will predict anticipatory distress
Context at a future medical procedure. The risk of bias
Two contextual factors emerged under the predispos- was rated as unclear due to one study with an unclear
ing domain. First, previous hospitalization was overall rating.
Perpetuating Factors (Vasey & Dadds, 2001). These results highlight the
Child importance of engaging parents in interventions to
Four child factors were identified as maintaining child help reduce child anticipatory distress. The risk of bias
anticipatory distress. First, child maladaptive cogni- for the parent perpetuating factors was unclear, indi-
5 tions were found to positively predict distress prior to cating reduced confidence in these findings. 60
a procedure with overall low risk of bias studies.
Overall, children who perceived less control expected
an aversive experience and appraised procedures as Present Factors
more threatening were more inclined to be distressed Parent
10 prior to those procedures. Given that child threat ap- A trend toward family-centered care has led to the in-
praisal and perceived control predict child anticipa- crease of parental presence within pediatric healthcare
tory distress, this highlights the importance of settings. Although parent presence during child hospi- 65
teaching children cognitive and behavioral coping tal stays have been associated with positive outcomes
strategies to manage their anticipatory distress. The (Wright, Stewart, & Finley, 2010), parent presence
15 overall risk of bias is unclear, reducing our confidence during a painful medical procedure in this review had
in these findings. inconclusive results. However, two studies pointed to
In terms of having more knowledge about the pro- mechanisms that may underlie the effect of parental 70
cedure, the overall results were inconclusive; however, presence. Kain, Caldwell-Andrews, Maranets, Nelson,
three studies did show a decrease in anticipatory dis- and Mayes (2006) found that the presence of a calm
20 tress. The method, type of information, and develop- parent reduces preoperative anxiety, while the pres-
mental level of the knowledge provided may be ence of an overly anxious parent does not. Given the
important variables in whether the knowledge pre- transactional and individual factors that predict antici- 75
sented works. The overall risk of bias was unclear for patory distress, clinical recommendations for parental
this factor, reducing our confidence in the findings. presence during a procedure should be based on char-
25 There was inconclusive evidence for child coping style acteristics of the parent and their ability to provide
with unclear risk of bias, reducing confidence in these calm support rather than the blanket assumption that
findings. There are specific child behaviors such as us- all parents should consistently be present or not pre- 80
ing nonprocedural talk, humor, and talking to a par- sent. More research should investigate the conditions
ent that were related to child coping before a under which parental presence is beneficial in reducing
30 procedure, while verbal resistance was found to be child anticipatory distress.
positively associated to child preoperative anxiety.
The child behavior factor had an overall unclear risk
of bias, reducing our confidence in these findings.
More research on the child behaviors that are associ- Health Professional
Our synthesis demonstrates that health care profes- 85
35 ated with coping before a painful medical procedure
sional behavior does play an important role in predict-
will help inform targets for intervention.
ing child anticipatory distress. Given the crucial role
that healthcare professionals can play in the experi-
Parent ence of children and families during painful medical
When examining the parent factors that perpetuate a procedures (Mahoney, Ayers, & Seddon, 2010), re- 90
child’s anticipatory distress to painful medical proce- searchers should continue to examine distress reducing
40 dures, an important pattern emerges. Across three fac- behaviors such as distraction that could be taught to
tors examined, there was evidence that parent factors health care professionals, as well as parents, as an in-
play a key role in maintaining the distress of children tervention to reduce procedural distress prior to a
during painful medical procedures. Parent behavior painful medical procedure. 95
during the procedure, parent situational distress/state

45 anxiety, and parent anticipation of child distress had
overall results predictive of child anticipatory distress. Contextual
Although the findings of the studies were not com- Fifteen studies investigated the effects of various con-
pletely uniform, the majority of studies highlighted the textual factors on child anticipatory distress during
role that parents play in continuing child anticipatory the painful medical procedure. It is difficult to synthe-
50 distress. It has been argued that, particularly for in- size this research due to the varied contextual factors; 100
fants and young children, the caregiver is the most im- however, some environmental factors (e.g., induction
portant context in the pediatric pain setting (Pillai location and sensory stimulation) do seem to impact
Riddell & Racine, 2009). Parental responding (model- anticipatory distress. More research is needed to deter-
ing, overprotection, reinforcement, and encourage- mine which contextual factors should be addressed in
55 ment) plays a key role in the development of anxiety clinical practice. 105
20 Racine et al.
Author’s Conclusions and Clinical Implications knowledge of these proposed factors should be incor-
As outlined in developmental psychopathology theory porated in randomized trials that test the efficacy of
(Cicchetti & Cohen, 1995), the development of antici- treatments of anticipatory distress. Having large age
patory distress occurs through a dynamic interplay of ranges in studies and not controlling for factors such
5 factors, including individual child factors, parent fac- as psychopathology (parent/child), previous pain ex- 60
tors, health professional factors, and their environ- periences (parent/child) and parent soothing behav-
ment. There is no unique pathway that leads to the iors/coping strategies will continue to limit the value
development of anticipatory distress but rather the in- of randomized controlled trials because they do not at-
teraction of predisposing, precipitating, perpetuating, tempt to accommodate the inherent variability of pain
10 and present factors over time leads to the onset and responses and the causes for the variability (Pillai 65
maintenance of distress. Riddell et al., 2013).
In this review, we examined 31 factors that predict
anticipatory distress. Children with preexisting anxi- Supplementary Data
ety and a difficult temperament were more likely to Supplementary data can be found at: http://www.jpepsy.oxfordjournals.org/.
15 have anticipatory distress. Parents and children should
examine patterns of past child behavior during painful Acknowledgments
procedures to better support and prepare children
The authors thank Noam Bin-Noon for her contribution to 70
with these risk factors. Parent anxiety and parent’s
this review in conducting some of the initial article screen-
previous experiences with pain are also important pre- ing, Elizabeth Uleryk for performing the data searches and
20 dictors of anticipatory distress. This suggests that par- Zhaodi Culbreath for assistance with figures.
ents need to be aware of their own subjective
experience of medical procedures and how to manage
Funding
their own anxiety in medical contexts. Past pain events
and previous child behavior are indicators of future This research was supported by awards to Dr. Pillai Riddell 75
25 anticipatory distress. Using adequate pain manage- from the Canadian Institutes of Health Research (MOP
ment is of utmost importance in reducing the likeli- 84511), the Ontario Ministry of Research and Innovation
(ER05-08-219), the Canadian Foundation for Innovation,
hood of conditioning fear and anxiety. Child and
and the York Research Chairs Program. Ms. Racine received
parent emotional and cognitive factors serve to main- awards from the Canadian Institutes of Health Research, the 80
tain or fuel anticipatory distress. These areas will be Government of Ontario, the Lillian and Don Wright
30 important targets for interventions. Finally, health Foundation, and the Canadian Pain Society. Ms. Racine is
professionals should be wary of engaging in distress also a trainee member of Pain in Child Health (PICH), a
promoting behavior such as verbal reassurance and strategic research training initiative of the Canadian
criticism and are encouraged to use coping promoting Institutes of Health Research. 85
behavior such as talking about things other than the Conflicts of interest: None declared.
35 procedure and engaging in distraction.
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Journal of Pediatric Psychology, 2015, 1–23

Systematic Review: Predisposing, Precipitating,

Key words: anxiety; children; infancy; pain; parents; systematic review.

Introduction and exhibit more intense pain responses (Taddio,

Figure 1. Included study flow chart following PRISMA guidelines.

Established tools to evaluate risk of bias in nonran- ease of interpretation.

Table I. Predisposing Factors of Anticipatory Anxiety

Child predisposing factors

Fox and Newton (2006) 5–17 38 UK Dental RCT Low 1

Thompson (1994) 8–12 43 USA Preop O Low 1

Ortiz et al. (2014) 8–16 437 Mexico Dental O Unclear 1

Tickle et al. (2009) 5–9 799 UK Dental O High þGirls

3. Child psychopathology (8 studies; N ¼ 2,053)

9. Attachment (2 studies; N ¼ 180)

10. Intelligence (1 study; N ¼ 60)

Tyc et al. (2002) 2–7 80 USA Radiation Therapy O Low 1

5. Parent gender (1 study; N ¼ 437)

dental visits posi-

was unclear. In terms of other contextual factors, the

dentist and having irregular visits to the dentist are

tors that contributed to the onset of anticipatory dis-

tress to painful medical procedures (Table II). The first

(33 studies). The results showed that 17 studies found

that previous negative experiences positively predict

Overall, the evidence points toward a positive rela-

tionship between previous pain events and child antic- 135

The second precipitating child factor that was iden-

studies). Overall, results indicate that previous pain

The risk of bias was unclear.

Health Professional 145

Tickle et al. (2009)

studies), child coping style (four studies), child cogni- 155

Child precipitating factors

Colares et al. (2013) 5–12 970 Brazil Dental R Low þ

Ellerton and Merriam (1994) 3–15 75 Canada Preop R Unclear þ

Tickle et al. (2009) 5–9 799 UK Dental O High þ

studies). For child knowledge, the results were incon- 50

as studies highlighted different child behaviors associ-

factors: parent behavior (7 studies), parent situational

distress (19 studies), parent anticipation of child dis- 65

studies) (Table III). Overall findings suggest that most

parent behavior, parent situational distress, and par-

increased anticipatory distress (Table III). 70

No health professional factors were found under the

No contextual factors were found under the perpetu-

child present factors: idiosyncratic needs. One study 80

(Ameringer, Elswick Jr, Shockey, & Dillon, 2013)

showed that fatigue and nausea were positively associ-

ated with child anticipatory distress prior to chemo-

Fortier et al. (2011)

One factor was identified as a health professional pre-

with higher child anticipatory distress. The overall 95

Child perpetuating factors

2. Child coping style (4 studies; N ¼ 174)

Table III. Continued

Child present factors

qualitatively synthesize the literature on the factors

framework. The following paragraphs will discuss key

cal differences and risk of bias within studies. Only