Impact of oral health related quality of life of children and adolescents with sickle cell anemia: a cross-sectional study

doi:10.21203/rs.3.rs-3240252/v1

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Impact of oral health related quality of life of children and adolescents with sickle cell anemia: a cross-sectional study

https://doi.org/10.21203/rs.3.rs-3240252/v1

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Backgroud The purpose of this study was to evaluate the impact of oral health conditions on the quality of life (QoL) of children and adolescents with sickle cell anemia (SCA).

Methods: The sample consisted of 76 children and adolescents aged 8–14 years. For inclussion in the study they were required to have a diagnosis of HbSS SCA in their medical records, without a pain crisis or any dental emergency in the last three months. The children and adolescents with SCA were from the Hematology and Hemotherapy Center of Maranhão (HEMOMAR), a state health institution responsible for governmental policies related to hematology and transfusion medicine in the State of Maranhão, Brazil. Demographic characteristics, socioeconomic status, oral hygiene, caries, malocclusion, and oral health-related quality of life (OHRQoL) were assessed. OHRQoL was assessed using the Child Perceptions Questionnaire (CPQ8-10 and CPQ11-14). Descriptive statistics, Student's t and Mann−Whitney tests were performed (α = 5%).

Results: The brown race was the most prevalent for both age groups (8–10 years − 63.2% and 11–14 years – 57.9%). The predominant monthly family income for both age groups was below $106. Visible plaque and gingival bleeding were higher in children with aged 8–10 years. Dental caries significantly impacted the quality of life of adolescents through the domain “oral symptom” (p = 0.031). The malocclusion significantly impacted the quality of life of adolescents (“total score”, p = 0.026; “social well-being”, p = 0.045).

Conclusion: Oral health impairment negatively affected the QoL of adolescents with SCA.

sickle cell anemia

oral health

quality of life

children

adolescents

Multiple organ morbidity and a high risk of early mortality [1]. It is a chronic genetic disease that is the result of abnormalities in the structure of hemoglobin. SCA occurs through a genetic mutation in which glutamic acid is replaced by valine in the sixth position of the beta chain of hemoglobin, creating hemoglobin S [2,3]. SCA primarily affects African descendants, and as a result, migration has spread worldwide [4].

As a result of repeated vaso-occlusion events throughout their lives, people with SCA can present with systemic manifestations, such as osteoarticular pain, cerebrovascular accident, acute chest syndrome, priapism, avascular necrosis of the bone, osteopenia, osteoporosis, high susceptibility to infections, sleep disorders, and fatigue. In addition, other manifestations can occur during childhood and adolescence and can be expressed in the maxillofacial complex. These include maxillary prognathism, malocclusion, hypercementosis, delayed dental eruption, oral mucosal pallor, orofacial pain, asymptomatic pulp necrosis, enamel and dentin hypoplasia, and depapillated tongue [5–8]. These changes are not unique to SCA. However, they can characterize oral impairments in individuals with SCA and aid in diagnosis [9].

Oral health-related quality of life (OHRQoL) is defined as “the impact of oral disorders on aspects of everyday life that are important to patients and persons, with those impacts being of sufficient magnitude, whether in terms of severity, frequency, or duration, to affect an individual’s perception of their life overall” [10]. Studies have shown that oral impairments affect the quality of life (QoL) of children. For example, a population-based study showed that untreated dental caries significantly affected the QoL of the public [11]. Furthermore, a systematic review [12] assessed whether oral conditions affect the QoL of adolescents. Malocclusion, dental trauma, and dental caries are the primary factors responsible for the negative impact on daily life.

The pathology of SCA is not responsible for the poor oral condition that affects individuals but for the negligence of caregivers in the oral health care of their children. Similarly, SCA can compromise children's QoL [13], and the deterioration of the oral condition of individuals with SCA can explain the negative impacts on QoL and involve symptoms, functional, emotional, and social impairments [14–16]. QoL focuses on individuals and encourages comprehensive, observant, and qualified healthcare.

This study aimed to evaluate the impact of oral health conditions on the QoL of children and adolescents with SCA. This study hypothesizes that oral health impairments in children and adolescents with SCA have a greater negative impact on QoL.

Study design

This study followed the guidelines of the Reporting of Observational Studies in Epidemiology [17] guidelines. This cross-sectional study was conducted to identify differences in OHRQoL between SCA children and adolescents with or without oral health impairments. The study considered oral health conditions such as visible plaques, gingival bleeding, caries, and malocclusion.

This study was carried out in 76 children and adolescents between 8 and 14 years of age. The children and adolescents with SCA were from the Hematology and Hemotherapy Center of Maranhão (HEMOMAR), a state health institution responsible for governmental policies related to hematology and transfusion medicine in the State of Maranhão, Brazil. This study was conducted from September to November 2020.

Sample size calculation

A sample size calculation was performed to compare the mean impact between the two groups. For children, a standard deviation (sd) of 6.2 [14], alpha of 5%, beta of 10%, and a minimum difference of 5 points to be detected between groups were considered. A sample “n” of 34 children was obtained. For adolescents, sd of 3.95 [14], alpha of 5%, beta of 10%, and a minimum difference of 5 points to be detected between groups were considered. A sample of n of 15 adolescents was obtained. The sample calculation was performed on the website: estatistica.bauru.usp

Inclusion and exclusion criteria

Children and adolescents aged 8−14 years were included in the study. They were required to have a diagnosis of HbSS SCA in their medical records, without a pain crisis or any dental emergency in the last three months.

Oral health status

To evaluate oral health, the decayed, missing, and filled permanent teeth index and decayed, indicated extraction, and filled deciduous teeth index [18] were applied. We also applied visible plaque and gingival bleeding index [19]. The latter was evaluated by passing a periodontal probe through the gingival margin.

The Dental Aesthetic Index (DAI) [20] was used to detect dentofacial abnormalities by evaluating dentition, space, and occlusion. The results of the DAI with a value ≤ 25 indicated that there was no abnormality or mild malocclusion with no or mild need for treatment. Values between 26 and 30 represent definite malocclusion requiring elective treatment. Values between 31 and 35 represent severe malocclusion that requires orthodontic treatment. For values ≥ 36, malocclusion is considered severe or debilitating, which makes orthodontic treatment essential. In this study, the DAI was divided into non-malocclusion (DAI ≤ 25) and malocclusion (DAI ≥26).

Child perceptions questionnaire

The long-form Child Perceptions Questionnaire (CPQ) was used in interviews with children aged 8−10 years (CPQ8-10) and adolescents aged 11−14 years (CPQ11-14) [21,22]. The CPQ was used to assess the influence of oral health status on the QoL of the participants. CPQ8-10 and CPQ11-14 each contained four domains: oral symptoms, functional limitations, emotional well-being, and social well-being, with 25 and 37 questions, respectively. The response categories were 0 = never, 1 = once or twice, 2 = sometimes, 3 = frequently, and 4 = almost every day or every day. The total scores for the respective questionnaires ranged from 0 to 100 and 0 to 148, with higher scores denoting a greater negative impact on QoL. CPQ8-10 records the frequency of events in the last month and CPQ11-14 in the last 3 months. Before the questionnaire was completed, the respondents were informed that there were no right or wrong answers and that they should be sincere when answering. Furthermore, respondents responded without the assistance of a caregiver.

Examiner calibration

A single rater (GMLM) performed data collection. The evaluator was a dental surgeon with 6 years of experience in the patient care domain at HEMOMAR. They had experience performing dental treatment on individuals of all age groups with hematologic diseases and evaluated dental caries, visible plaque, and gingival bleeding. The evaluator was calibrated only for DAI.

The calibration process was divided into three stages and was conducted by a gold standard examiner to apply the DAI (MCF). The first stage consisted of a theoretical phase (exposure of photographs representing the diagnostic criteria of different malocclusions) and a diagnostic exercise phase using oral photographic projections of different malocclusions. In the second stage (15 days later), the oral photographs were shown to the evaluator (GMLM), who performed the diagnosis once again. Kappa statistics were used to analyze the intra-examiner agreement (K = 0.875). The third stage included a clinical evaluation of the two children to diagnose the different malocclusions that comprise the DAI. The children were reexamined after 15 days. Kappa statistics were used to analyze the intra-examiner agreement (K = 0.855).

Evaluations were performed in the dental office using a clinical mirror and millimeter periodontal probe (WHO).

Statistical analysis

Descriptive statistics, Student's t and Mann-Whitney tests were performed (α = 5%). This study compared the impact on OHRQoL between SCA children and adolescents with or without oral health impairments. The Statistical Package for the Social Sciences (SPSS, version 21.0 SPSS Inc., Chicago, IL, USA) was used for all analyses.

The mean age of the children and adolescents was 9.0 and 12.7 years, respectively. The brown race was the most prevalent for both age groups. Most of the children and adolescents with SCA lived in rural areas (76.3% of the children and 63.2% of the adolescents). Families of children (71.1%) and adolescents (50%) with SCA had a monthly family income below of 106 dollars (Table 1).

Table 1

Demographic, periodontal and dental health-related characteristics (visible plaque, gingival bleeding, caries, and malocclusion) in children (8−10 years) (n = 38) and adolescents (11–14 years) (n = 38) with SCA.
	SCA 8–10 years	SCA 11–14 years
	n (%)	n (%)
Demographic characteristics
Age (years)	8 years	11 years
	13 (34.2)	8 (21.1)
	9 years	12 years
	12 (31.6)	8 (21.1)
	10 years	13 years
	13 (34.2)	8 (21.1)
	---	14 years
	---	14 (36.8)
Sex
Male	15 (39.5)	20 (52.6)
Female	23 (60.5)	18 (47.4)
Ethnicity
Black	9 (23.7)	3 (15.8)
Brown	24 (63.2)	22 (57.9)
White	4 (10.5)	8 (21.1)
Other^d	1 (2.6)	1 (2.6)
Area
Urban	9 (23.7)	14 (36.8)
Rural	29 (76.3)	24 (63.2)
Monthly family income*
< $106	27 (71.1)	19 (50.0)
From $106 to $317	9 (23.7)	16 (42.1)
> $317	2 (5.2)	1 (2.6)
Periodontal health	Mean (sd)	Mean (sd)
Visible plaque	56.61 (31.64)	15.21 (18.29)
Gingival bleeding	30.89 (27.94)	7.61 (10.87)
Dental health	n (%)	n (%)
Caries^e
No	5 (13.2)	13 (34.2)
Yes	33 (86.8)	25 (65.8)
Malocclusion^f
No	29 (76.3)	29 (76.3)
Yes	9 (23.7)	9 (23.7)
^dYellow+ indigenous; ^eCaries and restored but decayed teeth.
^fMeasured using the Dental Aesthetic Index. sd: standard deviation.
* Missing data (for 2 adolescents).

Table 2 shows the effects of dental caries and malocclusion on the QoL of children and adolescents with SCA. Dental caries significantly impacted the quality of life of adolescents through the domain “oral symptoms” (p = 0.031). The malocclusion significantly impacted the quality of life of the adolescents by means of “total score” (p = 0.026), and domain “social well-being” (p = 0.045).

Table 2

Total and domain scores of CPQ8-10 and CPQ11-14 in children (8−10 years) (n = 38) and adolescents (11−14 years) (n = 38) with SCA.
	CPQ8-10
	CARIES			MALOCCLUSION
	Yes (n = 33)	No (n = 5)		Yes (n = 9)	No (n = 29)
	Mean (sd) Median; 25%-75%	Mean (sd) Median; 25%-75%	p	Mean (sd) Median; 25%-75%	Mean (sd) Median; 25%-75%	p
Total score	27.09 (15.36) 27.00; 15.50–40.50	22.60 (13.11) 26.00; 10.00-33.50	^∝0.540	28.00 (15.95) 26.00; 17.00–38.00	26.03 (14.95) 26.00; 12.00–40.00	^∝0.736
Domains
Oral symptoms	6.97 (3.85) 7.00; 4.50–9.50	7.40 (4.22) 6.00; 4.00-11.50	^∝0.819	8.33 (4.72) 9.00; 5.00-11.50	6.62 (3.52) 6.00; 4.50-9.00	^∝0.247
Functional limitations	6.33 (4.25) 8.00; 3.00-9.50	7.80 (6.34) 10.0; 1.50–13.00	^∝0.504	6.78 (3.60) 8.00; 4.50-9.00	6.45 (4.79) 8.00; 2.50–10.00	^*0.959
Emotional well-being	6.06 (4.98) 6.00; 1.00-9.50	4.20 (4.92) 4.00; 0.00-8.50	^*0.399	5.67 (6.67) 5.00; 0.00–9.00	5.86 (4.43) 6.00; 1.00-9.50	^*0.602
Social well-being	7.73 (6.12) 8.00; 1.50–11.50	3.20 (2.68) 2.00; 1.00–6.00	^*0.134	7.22 (4.12) 8.00; 4.00-10.50	7.10 (6.49) 7.00; 0.50–12.50	^*0.742
	CPQ11-14
	CARIES			MALOCCLUSION
	Yes (n = 25)	No (n = 13)		Yes (n = 9)	No (n = 29)
	Mean (sd) Median; 25%-75%	Mean (sd) Median; 25%-75%	p	Mean (sd) Median; 25%-75%	Mean (sd) Median; 25%-75%	p
Total score	25.60 (17.11) 16.00; 13.00-39.50	18.62 (17.86) 15.00; 5.00–25.00	0.230	33.11 (14.27) 30.00; 23.00-45.50	20.14 (17.41) 14.00; 8.00–34.00	0.026
Domains
Oral symptoms	8.04 (4.61) 8.00; 5.00-10.75	4.58 (3.90) 3.50; 2.00–9.00	^∝0.031	9.33 (4.87) 10.00; 6.00-12.50	6.00 (4.31) 6.00; 2.00–8.00	^∝0.088
Functional limitations	6.48 (5.36) 7.00; 1.00–10.00	4.92 (5.30) 3.50; 0.50-7.00	^*0.411	7.67 (5.03) 9.00; 2.50–11.00	5.35 (5.37) 3.50; 0.50-8.00	^*0.156
Emotional well-being	3.74 (3.39) 4.00; 1.50-6.00	5.17 (4.55) 4.00; 0.50–10.50	^*0.716	5.56 (4.07) 4.00; 3.00-10.50	3.77 (3.70) 2.50; 0.50-6.00	^*0.170
Social well-being	8.61 (7.71) 7.00; 0.50–15.50	5.50 (7.03) 4.00; 0.00-7.50	^*0.159	10.56 (6.29) 8.00; 6.00–15.00	6.50 (7.75) 3.50; 0.00-12.50	^*0.045
^∝ Teste t de Student; * Mann−Whitney test; Interquartile range: 25%-75%.

This study confirmed the hypothesis that the oral health impairments of adolescents with SCA have a significant impact on their QoL. Dental caries significantly impacted the lives of adolescents with SCA, through “oral symptoms”. Malocclusion, in turn, affected the lives of adolescents with SCD, primarily through “social well-being”. Without considering the type of oral impairment presented by the groups, Fernandes et al. [14] found that the most significantly compromised domains were “oral symptoms” and “functional limitations.”

The worst impacts experienced by adolescents with SCA on QoL may be due to neglect of oral care or the impossibility of regular dental care. The healthcare facility where the patients were treated had a dental department dedicated to sickle cell patients. However, most children and adolescents with SCA live in rural areas with limited access to technical resources and SCA professionals. A considerable proportion of this population still lives in extreme poverty [23], a common factor in the health of the general population [24].

A significant contingent of participants with SCA presented with caries, corroborating the findings of Singh et al. [25]. In the present study, the higher prevalence of caries in children could also be explained by lower socioeconomic conditions [26–28]. Of the families of the 38 sickle children, 27 had a monthly family income below 106 dollars; of the 38 sickle adolescents, 19 had a monthly family income below 106 dollars.

Regarding other clinical factors indicating oral health impairment, visible plaques and gingival bleeding had higher mean numbers in children with SCA aged 8−10 years than in adolescents (11–14 years) with SCA. Oral hygiene directly affects visible plaque formation and gingival bleeding. Therefore, it is likely that oral hygiene is more inadequate in children than in adolescents with SCA.

Regarding gingival bleeding, different studies agree that it is unknown whether SCA affects periodontal tissues [29] by exacerbating the inflammatory process arising from the presence of bacterial biofilms. However, the increased gingival bleeding observed in children with SCA may be explained by inflammation and oxidative stress factors related to SCA and periodontal disease. An imbalance between the production of oxidants and antioxidant capacity is a critical factor in endothelial cell dysfunction and inflammation [30]. Therefore, the inflammatory response may be persistent in participants with SCA. This leads to prolonged inflammation that can affect periodontal tissues, such as the gingiva, and exacerbates their response to the minimal accumulation of dental bacterial biofilms [31].

In the present study, malocclusion was less prevalent in children and adolescents with SCA, disagreeing with the findings of Costa et al. [5], Basyouni et al. [8], and Pashine et al. [32]. Similar to the present study, these studies also evaluated malocclusion using DAI. Basyouni et al. [8] reported that severe malocclusion is more prevalent in patients with SCA. In a case-control study [38], mild and moderate malocclusions were more frequent in the SCA group. Based on these findings, malocclusion may be related to craniofacial abnormalities, muscle imbalance, lack of lip seal, and changes in bone base [5,8]. Changes in the jawbone can be related to erythroblastic hyperplasia and compensatory medullary expansion due to vaso-occlusion and are a source of chronic progressive disability, such as avascular necrosis [33].

In this study, malocclusion explained difficulties in socialization of adolescents. This shows the need for oral health programs beyond prevention and treatment to restore the shape of the tooth destroyed by carious lesions, but of orthodontic therapies that resolve malocclusion. However, professionals must consider vaso-occlusive events in these individuals, as increased hematopoiesis in the medullary spaces leads to increased trabecular space in the bone. An increase in the trabecular space can reduce bone density, making it more brittle and possibly more susceptible to bone defects [34].

In addition to the impacts of systemic impairments, particularly vaso-occlusive crises [3,35], children and adolescents with SCA experience physical and social impacts due to oral impairments. This finding reinforces the need for more mindful and qualified comprehensive healthcare. Establishing these measures will reduce complications, which can further weaken the general condition of these individuals.

Considering that there are still very few studies evaluating OHRQoL in individuals with SCA [14–16], this study reinforces the existing literature and strengthens the evidence for the impairments experienced by individuals. In this study, the oral symptoms could be confounded with the intensity of SCA symptoms. Therefore, individuals who had a pain crisis on the day of data collection were excluded from the sample.

This study had some limitations, such as the convenience sampling process, which may have resulted in selection bias. Information bias may be present in studies with subjective evaluation, as research subjects may not precisely remember past events, such as the severity and frequency of the impacts experienced daily. Therefore, longitudinal studies are needed to confirm and strengthen the evidence on OHRQoL in individuals with SCA. Evaluating the influence of oral conditions on the impact on the lives of these children will allow the establishment of more comprehensive health policies and provide better QoL for this population.

Based on the findings, it is concluded that oral health impairment in adolescents with SCA negatively interferes with their QoL.

Acknowledgements

None.

Author contributions

“Conceptualization, G.M. and R.T.; methodology, M.F.; validation, M.F and G,M.; investigation, C.C.; A.M and M.M.; data curation, D.B..; writing—original draft preparation, M.F. and G.M..; visualization, R.T. and G.M.; writing—review and editing, R.T. and M.F. All authors have read and agreed to the published version of the manuscript.”

Funding

To Foundation for Support to Research and Scientific and Technological Development of Maranhão—FAPEMA (POS-GRAD-02355/21).

Data Availability

The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.

Ethics approval and consent to participate

This study was approved by the Ethics Committee of The UNICEUMA (no. 4.236.886/2020). The authors declare that the investigations were carried out following the rules of the Declaration of Helsinki of 1975 (https://www.wma.net/what-we-do/medical-ethics/declaration-of-helsinki/), revised in 2013. Informed consent was obtained from all patients or those responsible for the patients in writing.

Consent for publication

Not applicable.

Competing Interest

The authors have no conflicts of interest to declare.

Author details

¹ Post graduated Program in Dentistry Programa of Ceuma University, São Luís, Maranhão, Brasil

² Post graduated Program in Dentistry Programa of Maranhao Federal University , São Luís, Maranhão, Brasil

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No competing interests reported.

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Impact of oral health related quality of life of children and adolescents with sickle cell anemia: a cross-sectional study

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Abstract

Introduction

Methods

Study design

Sample size calculation

Inclusion and exclusion criteria

Oral health status

Child perceptions questionnaire

Examiner calibration

Statistical analysis

Results

Discussion

Conclusions

Declarations

References

Additional Declarations

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