Oral Mucosal Lesions In Children With And Without Cleft Lip: A Case Control Study

Keywords

Cleft Lip; Oral Mucosa and Ulceration.

Introduction

Burning mouth syndrome can be defined as a complex disorder that is characterised by persistent burning sensation in the oral mucosa in the absence of any objective signs [1]. It can be characterised as chronic orofacial pain without any visible changes in the mucosa or presence of any lesions such as stomatodynia, glossodynia, neuropathic pain [2-4]. Burning mouth syndrome is found to be more prevalent in elderly women who are more prone to have hormonal imbalance. The condition is probably of multifactorial origin, [5-7] often idiopathic and a clear understanding about the exact etio-pathogenesis remains unclear [8, 9]. The most common sites which have been reported to be affected by burning mouth syndrome are the tongue, lips, hard palate and soft palate. The most common reported symptoms in addition to burning sensation are altered taste sensation and xerostomia or dry mouth [10].

As the etiopathogenesis of the disease remains unclear, there is no definitive cure for the disease, all the medication and treatment options are only palliative and for symptomatic relief. Classification of burning mouth syndrome:

Different classification types of burning mouth syndrome have been proposed by numerous people based on different diagnostic criteria. Lamey and Lewis have suggested classifying burning mouth syndrome into 3 subtypes according to pain intensity [11].

• Type I: Pain free waking - burning sensation developing in late morning with severity gradually increasing during the day. This affects 35% of the patients.

• Type II: Type II consists of continuous symptoms throughout the day.55 % of the patients are affected by this type.

• Type III: This type is characterised by intermittent symptoms with pain free periods during the day. This type affects the least number, only 10 % of the population [12-14]. Scala et al classified burning mouth syndrome into two categories.

• Primary : Idiopathic - local or systemic causes cannot be identified.

• Secondary : This type results from local and systemic factors.

Clinical features

It is extremely difficult to establish the true prevalence of Burning mouth syndrome as there are no definitive diagnostic criteria and poor awareness about the disease among oral health care professionals and dentists. The prevalence reported from various international studies ranges from 0.6- 15% [15]. Burning mouth syndrome most commonly affects middle aged and older women and the prevalence in such women increases upto 12-18% [12]. There have been no reported cases of burning mouth syndrome in children and adolescents [14]. Other epidemiological studies have reported a global prevalence of 0.5% [16].

The clinical features of burning mouth syndrome is highly variable and depends on each and every person thus making it extremely difficult to formulate a definite diagnostic criteria. The symptoms can mainly be burning or stinging sensation as well as tingling, numb feeling, altered sensation and metallic taste in the tongue. As mentioned earlier, the most common sites of the oral cavity affected would be the tongue, followed by anterior portion of hard palate and labial mucosa [1].

Etiology

The complex clinical behaviour of burning mouth syndrome has made it difficult to trace the etiology and pathogenesis of the disease. Salivary gland dysfunction also plays an important role in burning mouth syndrome cases. Some of the possible theories would be a) Abnormal interaction between sensory functions of facial and trigeminal nerve(17) b) Disturbances in the autonomic innervation and oral blood flow(18) c) Chronic anxiety or stress results in hormonal imbalance [19].

Diagnosis and treatment planning

Burning mouth syndrome is a challenging condition in terms of both diagnosis and management [20]. In general, in order to provide the best treatment possible [21, 22], these 3 approaches can be followed namely behavioural therapy, systemic medication and topical medication. Proper reassurance [23] and counselling regarding diet [24, 25] is of prime importance in treatment of burning mouth syndrome.

Burning mouth syndrome is a complex disorder making it difficult to diagnose as well as treat. A thorough understanding of its etiology and clinical features of the syndrome combined with better advancements in pharmacological interventions would help in better management of the syndrome [26, 27] This study throws on some light that focuses on determining the prevalence of burning mouth syndrome among the south Indian population - a first step in understanding the disorder. Thus, the aim of the study is to analyse the prevalence of burning mouth syndrome in patients visiting saveetha dental college.Previously our team has a rich experience in working on various research projects across multiple disciplines [28-42]. Now the growing trend in this area motivated us to pursue this project.

(17.8%), and least common were erythema multiforme (0.9%). [10].

Treating the child’s cleft conditions would be the most important both for the parent and the clinician as it forms the major part of food consumption. There are higher tendencies to miss out oral mucosal lesions like ulcers, coated tongue which could be due to a variety of factors. Assessing them would be of clinical significance as to whether these lesions need to be keenly diagnosed for children with cleft defects. However studies conducted in the south Indian population are limited. So the aim of the present study was to assess the presence of oral mucosal lesion in children with cleft lip and children without cleft lip. Previously our team has a rich experience in working on various research projects across multiple disciplines [11-25]. Now the growing trend in this area motivated us to pursue this project.

Materials and Methods

This is a retrospective study. This study was carried out in a hospital based university setting. This study was evaluated and ethically approved by an institutional ethical review committee. Retrospective data collected from 89,000 case records from June 2019 to March 2020. Informed consent was obtained from the parents or guardian before starting the treatment. Inclusion criteria were children with cleft lip, children aged from 6 months to 18 years, children with at least one or two erupted teeth, complete photographic and written records regarding the complete intra-oral examination of the patient. Age and gender matched controls i.e. children without cleft lip, were taken according to the relevant cases obtained from the inclusion criteria. The exclusion criteria were incomplete and censored dental records, children below the age of 6 months and improper photographs.

Total cases acquired for this study were patients 20 which includes 10 children with cleft lip and 10 children without cleft lip (age, gender matched controls). Selected case and control group were examined by three people; one reviewer, one guide and one researcher. Patient's case sheets were reviewed thoroughly. Cross checking of data including digital entry and intraoral photographs was done by an additional reviewer, and as a measure to minimise sampling bias, samples for the group were picked by the simple random sampling method. Digital entry of clinical examination and intraoral photographs were assessed. For both groups, presence of oral mucosal lesions were noted by a researcher, entered into Microsoft excel (MS Excel) and then transferred into Statistical Package for the Social Sciences (SPSS) Software for statistical analysis. A correlation test (Mann-Whitney test) was done between the children with cleft lip and children without cleft lip. The difference was statistically significant when the p-value was less than 0.05.

Results & Discussion

The final study sample size included a total of 20 children with 10 children with cleft lip (case group) and 10 children without cleft lip (control group). In this study, the control group was matched based on age and gender as similar to the case group. [Graph-1,2].

Absence of oral mucosal lesion was noticed in all children in both the groups i.e. children with and without cleft lip. On comparison of the results using Mann- Whitney test, the results were not statistically significant (p-value = 1) [Graph-3].

Oral mucosal lesions are commonly missed in the diagnostic period as the concerns over the actual cleft defect tend to be higher. The results of the current study shows that children in the case group i.e children with cleft lip did not have any oral mucosal lesions in their oral cavity. This result was contradictory to few studies which reported occurrence of mucosal lesions among children with cleft lip[26-27]. Amandeep Chopra et al [26] in 2014 reported that 19.5% of cleft lip patients had oral mucosal lesions. Previous study conducted by Ajith Krishnan et al [27] in 2010 reported that 5.33% of cases and 4.66% of controls had aphthous ulceration in the buccal mucosa , 1.33% of cleft lip cases and 2.66% of the controls had abscesses in the gingiva. The other result to be discussed is the absence of oral mucosal lesion in all the children of the control group i.e. children without cleft lip. Bezerra et al [28] found that childhood oral mucosal lesions among 0 to 5 five year old children to be 2.3% by observing their dental records. According to his study, the most common oral mucosal lesions were Born Nodules (37%) followed by candidiasis (25%) and least common was benign migratory glossitis (21%). Bessa et al found that incidence of childhood oral mucosal lesions among 0 to 4 year old children to be 24.9% and the most common lesions were geographic tongue (9.8%) followed by bite injuries (6.11%). Accidental biting during mastication, or consumption of hot food may cause traumatic ulceration. Iatrogenic damage caused by dental treatment also causes traumatic ulceration [29]. Prevalence of frictional keratosis ranges from 0.26 to 1.89% in children [29-32]. Prevalence of geographic tongue ranges between 0.37% and 14.3% in pediatric patients depending on the geographic area and may be up to 40.6% in children with systemic disease [30-38].

Preservation of primary teeth in the dental arch is important to guide the eruption of the permanent teeth in the optimal position. Grossly decayed primary teeth which are extracted before exfoliation causes space in the dental arch which causes malocclusion if space maintainer was not given [39, 40]. Bacteria play a vital role in the initiation and progression of dental caries which eventually causes pulpal and periapical disease [41]. Oral mucosal lesions interfere with oral hygiene measures as they cause mild discomfort while brushing the teeth subjecting to dental caries and periodontal disease in the children. Early childhood caries is the presence of one or more decayed which is cavitated or non cavitated, missing which is caused due to caries, or filled teeth in any primary tooth in a child younger than 71 months of age. Fluoride use has been recommended to prevent the dental caries [42]. Decreased concentration of fluoride also results in increased incidence of dental caries [43]. Saliva plays an important role in maintaining the oral health of an individual [44]. Untreated dental caries leads to pulpitis which is treated by means of root canal procedure - pulpectomy [45-49]. Chewable toothbrushes can be used to remove dental plaque and are more effective than manual brushing in children thereby decreasing the incidence of oral diseases [50-52].

Advantages of this study were that this was a case control study with age and gender matched controls to provide best results with high internal validity, reasonable data, Disadvantage of the study was that this was a unicentric study with geographic limitations, limited sample size and has lower external validity. Future scope for this study includes larger sample size which is not confined to a particular geographic area and to assess the oral mucosa and changes by clinically examining the cleft lip patients. Our institution is passionate about high quality evidence based research and has excelled in various fields [53-63]. We hope this study adds to this rich legacy.

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Figure 1:

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Graph 1: Bar graph represents the distribution of cases in case (children with cleft lip) and control group (children without cleft lip). (Y-axis represents number of patients; X-axis represents presence or absence of cleft lip) Note the equal distribution of cases in both the groups.

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Figure 2:

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Graph 2: Bar graph represents the distribution of cases in case (children with cleft lip) and control group (children without cleft lip). (Y-axis represents the number of patients; X-axis represents presence or absence of cleft lip; grey represents female; white represents male) Note the equal distribution of cases in both the groups.

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Figure 3:

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Graph 3: Bar graph represents the absence of oral mucosal lesions in children with cleft lip and children without cleft lip. (X-axis represents presence or absence of cleft lip; Y-axis represents the number of cases; green represents absence of oral mucosal lesions; blue represents presence of oral mucosal lesions). None of the cases in both the groups had oral mucosal lesions. (Mann-Whitney U test; p-value = 1 - not significant).

Conclusion

Within the limitations of the present study, there was no evidence of oral mucosal lesion in children with and without cleft lip.