Introduction

Cerebral palsy (CP) is defined by Rosenbaum as "a group of permanent developmental disorders of movement and posture, causing functional limitation. These are attributed to non-progressive disturbances produced in the developing fetal or infant brain" [1 p564]. It is usually diagnosed after two years and is the most common cause of physical disability in children. It has an estimated prevalence of 2 per 1000 live births, with multiple risk factors, each contributing to a different degree of severity in various individuals [1]. CP can be classified according to its distribution in the body and postural and movement characteristics, which guides the diagnosis and treatment of the child and allows for differentiated interventions or approaches [1-3]. The diagnosis depends on several elements, including the identification of prenatal, perinatal, and postnatal risk factors, as well as different social and cultural agents that may influence the development of this health condition, and there is still a wide field of exploration of this topic [4-8].

For their part, risk factors associated with health conditions have been studied in different fields for years. The World Health Organization has defined them as "any trait, characteristic, or exposure of an individual that increases his or her likelihood of disease or injury" [9 p106]. This article follows the previous concept and the proposal of Rutter, who classified them into three categories: 1. Biological, which refers to the physical nature of the events; 2. Organic, which refers to medical diagnoses; and 3. Environmental refers to cultural, social, and economic resources. In a complementary manner, Rutter proposes the following classification, which corresponds to the level of influence each risk factor may have on the person: 1. Proximal risk factors; 2. Medial risk factors; and 3. Distal risk factors [10,11].

In particular, risk factors associated with CP have been considered globally from three categories: prenatal, perinatal, and postnatal. The literature review by Himmelmann et al. [12] found a significant association between the development of CP and organic factors such as chorioamnionitis, maternal urinary tract infections, cytomegalovirus and neurotropic virus infection, and hyperbilirubinemia. In addition, it found that intrauterine growth deviations and multiple pregnancies represent a risk factor for CP. It is worth highlighting that from the environmental factors, their study reports an association between social deprivation and the development of CP in children.

Freire et al. [5] found that gestational hypertension and preeclampsia are risk factors for developing CP in children. This factor generates a limitation in the availability of nutrients and oxygen to the brain and restriction intrauterine growth. The above is due to the placental dysfunction associated with these conditions.

Derived from literature reports, Włodarczyk et al. [13] refer to the following factors as those associated with the risk of developing CP: maternal viral infections (STORCH), threatened abortion, gestational hypertension, pelvic delivery, preterm birth, low birth weight, placental insufficiency, forceps use, neonatal jaundice, respiratory distress syndrome, and other neonatal complications such as seizures, intraventricular hemorrhage, and sepsis.

The study conducted in Moldova by Bufteac et al. [14] found a correlation between the development of CP and the following risk factors: alcohol consumption during pregnancy, maternal arterial hypertension, maternal age over 35 years, maternal epilepsy, multiple pregnancies, breech delivery, cord circulation, and hyperbilirubinemia. From the environmental factors, the study draws attention to the results related to the rural origin and home-assisted childbirth, given their significance.

In addition to some organic and biological factors, such as APGAR less than seven at minute five and short stature for gestational age, Jöud et al. [4] found an understudied risk factor to be considered: maternal obesity.

Stoknes et al. [6] found an increased risk for CP as the number of associated factors increases; they state that the four main combinations of risk factors associated with CP are maternal illness and preterm labor, preterm labor and induction, maternal illness and induction, and maternal illness and APGAR less than seven at five minutes. This last combination yielded the highest correlation in their study.

Although CP has been extensively studied worldwide and significant associations have been found with risk factors, mainly biological and organic, it is not easy to find literature that reports questionnaires designed to collect information on pre-, peri- and postnatal biological, organic, and environmental factors that may be related to the development of this condition in children. The latter becomes a knowledge gap that requires viable and well-founded solutions. This aspect is essential since prevention and intervention strategies can be appropriately oriented to the extent that the risk factors associated with lesions occurring in the immature brain are identified.

For this reason, there is a need to design an instrument to collect this information for approaching the child and family, including assessment, professional reasoning, diagnosis, and establishing and implementing intervention plans.

Considering the above, the objective of this research was to design and validate, in its appearance, a questionnaire to collect information on risk factors in children with cerebral palsy in order to have an instrument through which these factors can be taken into account, from the biological, organic and environmental fields, and provide the health team with a valuable tool to provide information and guide decision-making around the child with CP and his family (Annex: FR-PC Questionnaire).

Methodology

A quantitative study of diagnostic test evaluation was developed through a design phase and another phase of face validation of a questionnaire. The latter was carried out through the participation of 43 mothers and ten health professionals, who formed the sample of the present study, that is, a non-probabilistic sample by convenience.

In this regard, it is essential to note that there is a wide variety of criteria for determining the sample size of test validation studies and, even more so, for face or appearance validation processes. Some authors suggest ten participants per item, some suggest five, and others suggest between two and three participants for each item contained in a broad instrument [15].

The design phase took into account the recommendations issued by Corral [16], Alonso et al. [17], and by Muñiz et al. [18] in terms of four premises: every questionnaire must be a reasonable and comprehensible document, sensitive to change concerning the phenomenon it measures, with justifiable and reasonable basic assumptions and clearly defined components. Similarly, this phase included four moments: 1) Determining the purpose of the instrument; 2) Defining the type of instrument to be used; 3) Conceptualizing the construct; 4) Operationalizing the construct.

Concerning the purpose of the instrument, it is expected to have a primary input through which the health team will have information on biological, organic, and environmental factors that will guide decision-making for the child with CP and his or her family.

The instrument analyzed in this research work is a questionnaire that allows the application of a series of questions on possible factors related to the development of CP. The questionnaire includes a mixed response type since it contemplates several response options, with closed, semi-closed, and open alternatives [16,17,19].

The conceptualization phase of the construct was carried out through a literature search guided by the PIOM strategy [20,21], which included the following keywords in Spanish and their correspondence in English: "Cerebral Palsy," "Questionnaires," "Risk Factors," "Validation Study (DeCS)."

This research considered the "Risk Factors that may be associated with Cerebral Palsy" as all those events or conditions of a natural, organic, or environmental order that occur before, during, or after birth and predispose an individual to develop cerebral palsy. According to their influence level, these can be proximal, medial, or distal.

According to the above, and bearing in mind both the purpose of the instrument and Rutter's proposal, Table 1 presents the operationalization of the construct.

Table 1

Operationalization of the construct FR-PC questionnaire

After defining the construct and developing its operationalization, the formulation of each question (item) and response options continued, particularly for those with a closed or semi-closed structure. Thus, this led to the design of the questionnaire, oriented towards collecting useful information regarding factors that, proximally or distally, directly or indirectly, may represent a risk for the development of CP. The form contains 29 items or questions and is designed to be filled out by health professionals, following a structure that allows its alignment with the processes corresponding to the child's clinical history (Annex: FR-PC Questionnaire).

The face validation stage of the questionnaire involved the participation of 43 mothers and ten health professionals, selected by convenience, taking into account the following inclusion criteria:

Mothers: women who have been mothers residing in Colombia, with a basic primary school level that allows the completion of the face validation format of the questionnaire.

Health professionals: therapists, nurses, doctors, and psychologists, residents of Colombia, and with expertise in managing children's health conditions.

As part of the face validation process, two criteria were evaluated for each of the questions in the questionnaire: clarity and comprehension. About the above, the rating was established on an ordinal measurement scale, from 1 to 3, with its corresponding indicator (Table 2). It should be noted that the format for face validation recorded by professionals includes the 29 questions proposed by the FR-PC questionnaire. In contrast, the format applied to the mothers did not consider 3 of these, given their level of specificity (questions # 22, 23, and 24).

Table 2

Criteria for face validation of the FR-PC questionnaire

In order to allow the analysis of the information, the format for face validation of the FR-PC questionnaire was sent via e-mail to the professionals and was applied to the mothers. Following the procedure used in the study by Suárez-Acuña et al. [22], 80% acceptability was established as the minimum value to give rise to the acceptance of the question concerning each criterion evaluated; for questions with acceptance percentages below 80%, a new review process was projected, in order to obtain an adjusted version.

This research is part of the project "Cerebral Palsy, General Movements, Motor Development in Infants and Family Coping Strategies," endorsed by the Ethics and Research Committee of the Universidad de La Sabana, with code ENF-58-2020. The research took into account both the ethical and environmental guidelines established by the Universidad de La Sabana and the international guidelines related to the ethics of scientific writing and those to safeguard the integrity and confidentiality of the people who participated in the study, considering the principles of privacy and autonomy, established by the Declaration of Helsinki and the guidelines of Resolution 8430 of 1993 of the Colombian Ministry of Health.

The FR-PC questionnaire will be the instrument to be used to collect data related to biological, organic, and environmental risk factors of the children who are part of the study "Cerebral Palsy, General Movements, Motor Development in Infants and Family Coping Strategies."

Results

In general, the design of the FR-PC questionnaire was carried out, which response to the proposal on risk factors issued by Rutter, to the available evidence on the subject, and the premises and construction phases of Corral [16] and Alonso et al. [17].

The analysis of the face or perceived validity of the FR-PC questionnaire reported a total of 12 items with a lack of clarity and difficulty in understanding, identified as having obtained a percentage of acceptance lower than 80% (Tables 3 and 4).

15.4% of the questions in the questionnaire were unclear, and 19.2% of the questions in the instrument were difficult for the mothers to understand (Table 3).

Table 3

Analysis of the face validation of the FR-PC questionnaire - mothers

Note. Questions 22,23, and 24 were not included for mothers, given their level of specificity.

From the professionals' perspective, 34.5% of the questions in the questionnaire were reported as lacking clarity and being difficult to understand (Table 4).

Table 4

Analysis of the face validity of the FR-PC questionnaire - professionals

In general, mothers reported five questions without clarity and difficulties understanding (# 5 - 7 - 10 - 13 - 28). On the part of the professionals, 11 questions arose with a need for more clarity and difficulties in understanding (6 - 7 - 10 - 13 - 14 - 15 - 16 - 17 - 18 - 27 - 28). Therefore, considering that some questions were reported with difficulties by both mothers and professionals, 12 questions were revised and adjusted, equivalent to 41.4% of the total number of questions in the FR-PC questionnaire. This adjustment took into account the comments and suggestions recorded by the participants in the study (Table 5).

Table 5

Adjusting questions that are unclear and difficult to understand according to the face validation process with the participation of mothers and professionals

Discussion

The design and validation of instruments for identifying risk factors in the diagnosis of CP is an essential aspect of research, evidence-based practice, health promotion, and decision-making and, as a primary goal, for the development of the child and his or her family.

According to the literature review process conducted within this study, it is evident that there are prenatal, perinatal, and postnatal biological, organic, and environmental risk factors that have been studied for many decades about the influence they may exert on the development of CP [7]. However, there needs to be more knowledgeable about the experience of research processes that have focused on the design and validation of a questionnaire aimed at collecting information on risk factors that could be associated with the development of CP.

Regarding the risk mentioned above factors, Wlodarczyk et al. [13] describe the main ones the following: 1. Prenatal: multiple pregnancies, vaginal bleeding, and chronic maternal infections; 2. perinatal: hypoxia, cesarean sections, premature rupture of membranes, and premature contractions; and 3. postnatal: respiratory failure, pneumonia, ischemic events, and sepsis. In addition, Tharaldsen et al. [23] report that APGAR score, congenital malformations, and prematurity should also be taken into account among the risk factors since they have been shown to have a strong association with the development of cerebral palsy in Norwegian children.

The design of the FR-PC questionnaire took into account the variables mentioned above because they are proximal factors, some of them biological and others organic, for the development of CP (Table 1).

The study by Bufteac et al. [14] proposes considering risk factors related to the mother, labor, and delivery. In doing so, it draws attention to biological, organic, and environmental factors, which have been included in the FR-PC questionnaire from different levels of influence (Table 1). Among these, maternal age, the mother's educational level, exposure to alcohol, and socioeconomic level are considered necessary, as well as maternal diseases, type of delivery, premature rupture of membranes, out-of-hospital deliveries, assisted vaginal deliveries, gestational age, APGAR score, multiple gestations, and birth weight.

On the other hand, Graham et al. [3] report neonatal asphyxia and preterm birth as the main risk factors associated with the development of cerebral palsy. Mainly, gestational age at birth is a variable inversely related to the possibility of developing cerebral palsy. The lower the gestational age at birth, the greater the possibility of developing cerebral palsy. For example, the risk in infants born before 28 weeks gestation is approximately 50 times higher than that of full-term infants. Both asphyxia and gestational age are proximal order variables, which are identified through the application of the FR-PC questionnaire. According to their category, these have been incorporated into the organic and biological factors, respectively (Table 1).

For term infants, variables include placental abnormalities, fetal growth retardation, neonatal depression with a low APGAR score, neonatal hyperbilirubinemia and jaundice, infections, and perinatal asphyxia, among others, are factors that have been associated with the possibility of developing CP [3]. Therefore, the FR-PC questionnaire considers questions aimed at collecting information on proximal organic factors, represented in this case by pre-, peri- and postnatal complications that may affect the development of the immature brain (Table 1).

According to Schneider et al. [7], maternal age is a factor to be considered within the broad spectrum of possible CP-related variables. Both advanced maternal and young maternal age are conditions related to the development of CP. This element is also part of the structure of the FR-PC questionnaire.

In addition to the biological and organic factors mentioned above, Chen et al. [24] draw attention to the possible relationship between parental education and the development of CP. The FR-PC questionnaire has items that will allow the collection of information on environmental factors, including variables such as the mother's level of education, occupation, access to public services, and household income, among others.

Based on the above, the FR-PC questionnaire has gathered evidence-based information that aims to relate biological, organic, and environmental factors of proximal, medial, or distal order to the development of CP. According to the results of the appearance validation process of the FR-PC questionnaire, questions 5, 6, 7, 10, 13, 14, 15, 16, 17, 18, 27, and 28 were adjusted because mothers and professionals reported them as being unclear and/or difficult to understand.

Limitations

On the one hand, the sample size could have been larger; on the other hand, more literature was needed to contrast the study results with other similar studies.

Conclusions

Based on the present study, a questionnaire (FR-PC Questionnaire) was designed and validated to collect information on possible risk factors that may be related to the development of CP. After its adjustment, the FR-PC Questionnaire is a clear and understandable tool that gathers pre-, peri- and postnatal biological, organic, and environmental elements that facilitate the organization and integration of data from the child's clinical history with CP for decision-making. Likewise, the FR-PC Questionnaire may be used in training, academic or research processes.

It is important to note that identifying the risk factors that influence the diagnosis of PC allows relevant decisions to be made, such as those related to the promotion of the health of individuals and groups. It also helps to guide the intervention processes between the interdisciplinary health team and the children and their families.

The research team intends to continue the present study through the content validation of the FR-PC Questionnaire.

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Notes

Author notes

*Correspondence: María Eugenia Serrano-Gómez. Email: maria.serrano4@unisabana.edu.co