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 Table of Contents  
ORIGINAL ARTICLE
Year : 2019  |  Volume : 6  |  Issue : 2  |  Page : 114-119

Maternal factor effect on the infants with cleft lip and cleft palate: A case–control study


1 Masters of Nursing, Khoy University of Medical Sciences, Khoy, Iran
2 Masters of Nursing, School of Nursing and Midwifery, Tabriz University of Medical Sciences, Tabriz, Iran
3 Department of Epidemiology and Biostatistics, School of Public Health, Iran University of Medical Sciences, Tehran, Iran

Date of Web Publication7-Aug-2019

Correspondence Address:
Dr. Mehdi Mokhtari
Department of Epidemiology and Biostatistics, School of Public Health, Iran University of Medical Sciences, Tehran
Iran
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jclpca.jclpca_21_18

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  Abstract 

Objectives: Oral clefts are the most common congenital abnormalities that have various causes. The aim of this study was to investigate maternal factors influencing the event of this disease among newborns. Methods: This study was a case–control research. To investigate the effects of maternal factors on cleft lip and palate, medical records of 26 infants with cleft lip and palate as well as those of 52 healthy infants without this abnormality born in Qamar Bani Hashem Hospital in Khoy during 2015–2017 were studied. The required data were collected through interviews and reviewing their medical records. Results: The odds ratio of having a baby with cleft lip and cleft palate among mothers with a history of using cosmetics, being at a high age, use of psychiatric drugs, passive smoking, family history of the disease, and use of analgesics were, respectively, 1/9 (confidence interval [CI] =1.03–4.2), 2/2 (CI = 1.01–4.1), 1/7 (CI = 1.1–4.8), 2/08 (CI = 1.2–5.1), 2/8 (CI = 1.3–5.2), and 1/5 (CI = 1.01–4.9) times as much as the mothers of healthy infants. Conclusion: Lifestyle modification is the important factor in the prevention of cleft lip and cleft palate. Inclusion of preventive programs in prenatal maternal care programs may prevent or reduce the incidence of this disease.

Keywords: Case–control study, cleft lip, cleft palate, infants, maternal factors


How to cite this article:
Purabdollah M, Sanaeeifar M, Alipoor S, Bahrami A, Saadati H, Mokhtari M. Maternal factor effect on the infants with cleft lip and cleft palate: A case–control study. J Cleft Lip Palate Craniofac Anomal 2019;6:114-9

How to cite this URL:
Purabdollah M, Sanaeeifar M, Alipoor S, Bahrami A, Saadati H, Mokhtari M. Maternal factor effect on the infants with cleft lip and cleft palate: A case–control study. J Cleft Lip Palate Craniofac Anomal [serial online] 2019 [cited 2019 Aug 21];6:114-9. Available from: http://www.jclpca.org/text.asp?2019/6/2/114/264094


  Introduction Top


Cleft lip and cleft palate are among the most common congenital abnormalities that usually appear as isolated. These clefts are divided into two separate categories according to embryonic origin and genetic studies: in the first category, the primary palate is involved in utero and it causes cleft lip or cleft palate, and in the second category, the secondary palate is involved in utero causing cleft palate alone.[1],[2]

The prevalence of cleft palate and cleft lip depends on several factors including race,[3],[4] geographical region,[3] and economic level.[5] The highest incidence of the disease is among Native Americans (3.7 per 1,000 live births) and the lowest is among blacks (0.3 per 1000 live births).[3],[6] Several studies conducted in Iran have estimated a prevalence of 0.97 abnormalities per 1000 live births in Gorgan over a 6-year period and 2.14 per 1000 live births in Tehran during 5 years.[7]

Regarding different symptoms of the disease, the etiology of this disease seems to be a combination of genetic (30%) and environmental (70%) factors.[8] The major environmental factors affecting on cleft palate and cleft lip are smoking,[9] alcohol consumption, use of corticosteroids, antiepileptic drugs, folic acid deficiency, and diabetes during pregnancy.[10] Syndromic abnormalities are mainly observed in the form of cleft palate or at least cleft lip alone.[11]

Cleft lip and cleft palate are among common syndromes and congenital abnormalities that bring about much psychological and economic harm to the families and cause speech and hearing disorders to the newborns.[12]

Having a baby with an abnormal face may cause emotional stress for the family. Few studies have been done on identifying environmental factors affecting the families, especially behaviors of mothers in the birth of babies with cleft lip and cleft palate. What was proposed here as the aim of this study was to identify maternal factors affecting the incidence of cleft lip and cleft palate and the likelihood of preventing them by genetic consultations and amendment of family lifestyle as well as calculation of risk probability and informing the health-care providers of the short time to delivery so as to effectively prevent the occurrence of this disorder.


  Methods Top


This case–control study was done in the Department of Pediatrics in Qamar Bani Hashem Hospital in the city of Khoy in 2018. The study population included the newborns with congenital cleft lip and cleft palate. Having this disease was the only inclusion criterion for the study group, and the disease had been confirmed by pediatricians. The case group included 26 newborns with cleft lip and cleft palate that had been selected during a 3-year period from 2015 to 2017among the patients registered at the hospital.

The control group included the newborns without cleft lip and cleft palate who had no known cause of the disease. The selection criteria for the control group included the matching of date of birth, gender, and lack of cleft lip and cleft palate, and once they were selected, their medical records were examined.

Khoy had only one hospital to provide obstetric services; therefore, all the cases and controls were selected from that hospital.

To collect the data, a survey form (checklist) as well as telephone and in-person interviews was used. Its validity and reliability had been confirmed by pediatricians and conducting a pilot study. With regard to the aim of the study to determine the maternal factors influencing the incidence of cleft lip and cleft palate and due to the low prevalence of the disease, the sample size was obtained from all available cases in the hospital that had been born during 2015–2017.

After identifying the case and control groups and getting the consent of the hospital authorities to make phone calls to the parents of the infants in both the groups, they were called and their informed consent was obtained. The time of meetings was then determined, and the in-person interviews were conducted by two trained nurses. Three mothers in the case group and five mothers in the control group were not willing to be interviewed face to face. Hence, telephone interviews were done with them, and the required data were collected.

The questions in the survey form to be asked each group included demographic information, gestational age, consanguineous marriage, family, economic situation, economic status and diet, smoking and drug abuse by the mother and other family members who were living together, history of psychiatric drug consumption, the extent of using cosmetics, number of children, the sick infant's place of birth in the family, and the family history of cleft lip and cleft palate.

The parents were given moral obligation for confidentiality of the children's and their parents' information. The collected data were statistically analyzed by IBM SPSS statistic version-16 (SPSS Inc, Chicago) software. The statistical indices used in this study included descriptive and analytical (odds ratio, McNemar's test, and conditional logistic regression) ones.


  Results Top


Of the 26 infants with cleft lip and cleft palate, 19 were boys (73.1%) and 7 were girls (26.9%), and the same ratio was selected for the control group. The mean age of mothers was 26 ± 3 in the case group and 23 ± 2 in the control group, and the mean ages of fathers in the case and control groups were 31 ± 3 and 27 ± 3, respectively.

14.8% of the patients' mothers were in the age group 35 and older, while 11.1% of the control group mothers were older than 35 years of age. The results showed a significant relationship between gestational age and the incidence of congenital abnormalities.

The average weight of the infants with cleft lip and cleft palate in the case group was 3250 g, and 21.8% (7 infants) of them had been born weighing <2500 g. However, in the control group only, 12.5% of the infants (8 infants) had been born weighing <2500 g and this showed a significant difference.

Four patients (16%) with cleft lip and cleft palate were living in the city, while 22 of them (84%) were living in rural areas. This indicated a significant relationship between the location and the incidence of the disease.

In terms of education level, none of the mothers in the case group had a university degree, and most of them (61.6%) had primary and secondary education levels. In contrast, 13.5% of the mothers in the control group had university degrees, and comparing these two groups did not show a statistically significant difference [Table 1].
Table 1: Distribution education, employment, and residence between two groups

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The number of consanguineous marriages in the case group was 14 (53.8%), but it was 12 in the control group (23%). This showed no statistically significant relationship between family history of the disease and the incidence of cleft lip and cleft palate. In terms of the impact of education and employment status of the mothers in both the groups, no significant difference was observed [Table 1].

In the separate logistic regression model, the following results were obtained after examining the relationship between the independent variables and cleft lip and cleft palate:

The odds ratio of having a baby with cleft lip and cleft palate among mothers with a history of using cosmetics, being at a high age, use of psychiatric drugs, smoke, family history of the disease, and use of analgesics were, respectively, 1/9 (CI = 1.03–4.2), 2/2 (CI = 1.01–4.1), 1/7 (CI = 1.1–4.8), 2/8 (CI = 1.2–5.1), 2/8 (CI = 1.3–5.2), and 1/5 (CI = 1.01–4.9) times as much as the mothers of healthy infants. Comparing the frequency distribution of the variables studied in both the groups and the odds ratios can be observed in [Table 2].
Table 2: Distribution of variables and logistic regression to estimate the odds ratio based on the model

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To examine the impact of using cosmetics on the incidence of congenital cleft lip and cleft palate disorders, all mothers were asked about the amount, type, and time of using cosmetics. Eighteen mothers with sick babies (69.2%) and 25 mothers in the control group (48.1%) stated that they had had a daily use of cosmetics during pregnancy, and the chance of having a sick baby among the mothers who used cosmetics was more than those who were not exposed to these substances.

None of the mothers surveyed reported smoking, and to examine the impact of smoke, passive smoking was evaluated. The results showed that 16 mothers in the case group (61.5%) had a history of smoking by their family members. Thus, they had experienced smoking into passive smoking. Hence, a significant relationship was observed for maternal exposure to smoking.

Most mothers in the case group had a history of taking psychiatric and hypnotic (benzodiazepines) drugs (65.3%) during pregnancy because they were already suffering from some kind of psychiatric disorder that had a significant relationship with the incidence of the disease compared to the control group.

To investigate the effects of radiation on this congenital abnormality, due to the unavailability of sufficient evidence on the extent and duration of radiation exposure, the results were obtained based on medical prescriptions given by the mothers' doctors during their pregnancy. Hence, the results showed that regardless of the effects of radiation caused by the ultrasound that all mothers stated that they had experienced at least once during their pregnancy and also the available evidence confirmed it, 42.3% of the mothers in the case group (11 mothers) and 32.6% in the control group (17 mothers) had a history of exposure to radiation for various medical reasons. The odds ratio of the disease among the newborns in the case group was 1.5 times as much as the infants in the control group, but no statistically significant relationship was found between them.


  Discussion Top


These results of this study have shown several significant differences in general terms between the groups, but since the subgroups were so small, it was not possible to identify which particular factors were responsible.

One of the most important jaw and facial abnormalities is cleft lip and palate from which ≥10 million people worldwide are suffering.[13] These abnormalities can affect lips alone, lips and palate, or palate alone and usually follow a multifactorial inheritance pattern.[8]

As the results of our study show, consanguineous marriage and positive family history of the disease existed in 53.8% of the cases. Accordingly, it can be concluded that genetic factors play an important role in the development of this disease. Kohli et al showed that the family were living in the rural, due to the specific cultures and different life styles might be at-risk families and give them necessary education to reduce the spread of the disease.[14]

53.8% of all newborns in the case group had a positive family history of the disease, and this showed the impact of positive genetic factors in the incidence of the disease that was similar to the results of various studies.[15],[16] In a study conducted in Estonia, the rate had been reported to be about 19%.[15] The studies done on twins and the families with a family history of the disease indicated the positive impact of genetic factors on the incidence of cleft palate and cleft lip.[16],[17]

The risk of the cleft lip and cleft palate in newborns whose mothers had a history of passive smoking during pregnancy was 2.8 times higher than those in the control group. The comparison of these two groups showed a statistically significant relationship between the groups and the results were consistent with the statistics provided in most studies. Lammer et al. showed that combined absence of Glutathion S-transferases (GSTM1 and GSTT1) enzymes among the offspring of smoking mothers was associated with a nearly 6 times increased risk for cleft lip (6.3; 1.3–42). A similar increased risk for cleft palate was associated with the absence of GSTM1 but not for the absence of GSTT1.[18],[19]

Results of studying this variable showed that mothers who had higher exposure to smoking in the home experienced higher psychological pressures, and this had ultimately a direct relationship with increased risk of abnormalities among newborns. In their study, Sousa et al. pointed out this issue as well as the role of the mothers' mental instability and their desire to use drugs and smoking in the increased risk of abnormalities among newborns.[20]

The mean age of the individuals in this study was 27.2 for the case group and 23.1 for the control group. This shows a significant relationship between increased maternal age and having babies with the disease. These values showed consistency with the results of various studies conducted in this area.[17],[21],[22]

In addition to the relationship between increased maternal age and the chance of infants with the cleft lip and palate, paternal age was also a risk factor. Results of several studies suggested an increased risk of this adverse event due to the increased paternal age.[17],[22] In a study conducted in Estonia, paternal age over 30 years had been reported as a factor to increase infant abnormalities.[15]

65.6% of the mothers in the case group had a history of taking psychiatric drugs during pregnancy and previous years. This had a significant relationship with the occurrence of this abnormality as a result of taking benzodiazepines. This was consistent and compliant with the results of a majority of studies that confirmed the relationship between taking psychiatric drugs and an increased risk of giving birth to children with cleft lip and cleft palate.[23],[24],[25] It was due to the impact of stress on families, and our study showed that the risk of the disease among these individuals was 1.7 times as much as the control group.

The average birth weights of the infants in the case and control groups were 3250 g and 3440 g, respectively, and 28.1% of the infants in the case group were weighing <2500 g, while in the control group, 14.6% of the infants were weighing <2500 g. These findings were similar to the results of a study by Sadri and inconsistent with the results of Jamilian's study in which the infants weighing <2500 g in the patient group consisted 50% of the cases.[23]

This might be justified according to people's different cultural and economic levels on which basis people under different conditions influenced by their community and resources change their education and may act based on the culture of their society. The results of this study showed that people might work or continue their education according to their family's desire or the conditions of their society, and this could be involved in their knowledge and awareness about the promotion of their own or community's health.

42.3% (n = 11) of the women in the case group and 32.6% (n = 17) in the control group had a history of radiation exposure, and the chance of the disease occurrence among the newborns in the case group was 1.5 times more than in the control group, but the relationship was not statistically significant. This was inconsistent with the results of the study by Sadler,[2] and the reason might be the low sample size of our study. Therefore, studying the risk factors for the disease in densely populated areas with high sample volumes is recommended.

In a study conducted on the integrated effects of genetic and environmental factors in the incidence of cleft lip and cleft palate, Dixon et al. indicated that in women who had a positive genetic history of developing cleft lip and cleft palate, exposure to environmental risk factors would increase the chance of having an affected child by 2–4 times. In their study, one of the risk factors for the disease was the use of cosmetics by the mothers during pregnancy and that was consistent with the results of our study.[26]

The present study showed that in women who used cosmetics in various forms and for a long time during pregnancy, the chance of having an affected child was 1.9 times more than in other women in their group. This could be attributed to ethnic traditions because most mothers with sick children were among those who tended to use cosmetics very much, especially cosmetics of traditional types.

Among the patients' mothers, 17 reported irrational use of antibiotics during the last trimester of pregnancy and 11 said that they had been taking pain relievers. The results of this study showed that the chance of the disease in this group was greater than in the other group and these were consistent with the results of several other studies.[27],[28]


  Conclusion Top


According to the findings of this study, it can be concluded that most environmental factors affecting the risk of sick babies were rooted in maternal risk factors, and with regard to maternal factors affecting the incidence of cleft lip and cleft palate and through the inclusion of relevant preventive programs in maternal care during pregnancy, the disease can be significantly prevented.

Limitations

One limitation of this study was the possibility of recall bias and direct impact of economic situation on the incidence of mental stress and lifestyle. In addition, given the impact of ethnic and genetic factors on the pathogenesis of this disease, doing other studies in different parts of the country is recommended. Another limitation of the present study might be the types of parental involvement based on their different levels of education and culture when responding to the questions.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
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Acuña-González G, Medina-Solís CE, Maupomé G, Escoffie-Ramírez M, Hernández-Romano J, Márquez-Corona Mde L, et al. Family history and socioeconomic risk factors for non-syndromic cleft lip and palate: A matched case-control study in a less developed country. Biomedica 2011;31:381-91.  Back to cited text no. 17
    
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