Developmental Defects of Enamel and Dental Anomalies in Children with Skin Diseases

doi:10.21203/rs.3.rs-4712611/v1

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Developmental Defects of Enamel and Dental Anomalies in Children with Skin Diseases

https://doi.org/10.21203/rs.3.rs-4712611/v1

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Objectives

The common embryologic neural origin of the ectoderm includes the epidermal layer of the skin and the amelodentinal (the enamel and dentine) components of the teeth, which result in a variety of conditions affecting both skin and dentition to different degrees. The aim of the study was to evaluate the occurrence of dental anomalies among pediatric patients with skin diseases.

Materials and Methods

Patients with skin disease (4–16 ages, n = 71) were included in study group and 41 age-gender-matched children were designated as control group. Enamel defects were evaluated with the MIH Index. Panoramic radiographs were taken, and dental anomalies seen in the radiographs (hypodontia, hyperdontia, taurodontism, short root anomaly) were recorded.

Results

MIH was found to be significantly higher in chronic skin diseases group. DDE-not-related MIH, and other dental anomalies were found to be statistically significantly higher in the genodermatoses group.

Conclusion

Current study confirms a connection between MIH and chronic skin diseases, as well as a relationship between genodermatoses, DDD-not-related MIH, and other anomalies.

Clinical Relevance:

Dental screening and management should be taken into account as part of the patient's overall health care plan, and clinicians should be aware of the potential oral health issues associated with skin diseases.

MIH

Dental anomalies

Skin disease

Atopic dermatitis

Genodermatosis

Amelogenesis is a complex process that begins with the secretion of enamel matrix proteins, followed by mineralization, and finally, the maturation of the entire structure [1]. Factors affecting amelogenesis and causing developmental defects of enamel (DDE) are traditionally classified as either hereditary or acquired [2]. Causes of DDE include environmental intoxications such as fluoride and dioxins, trauma, localized infections, irradiation, chemotherapy, infectious diseases, perinatal and postnatal problems, and malnutrition [3]. Demarcated opacities in the first permanent molars, a type of DDE, have been well-known and regarded as a separate entity by pediatric dentists since they were first defined by Weerheijm in 2001 as molar incisor hypomineralization (MIH) [4]. In recent years, epidemiological studies on enamel defects have focused on MIH. Despite numerous investigations into potential factors, the etiology of MIH remains unclear. It is likely that MIH is caused not by a single factor but by a combination of multiple factors [2].

Developmental alterations of teeth affect not only the enamel but also the structure, number, size, and shape of teeth [5]. From initiation to the final basic features such as shape, size, and structure, teeth are typical examples of organs in which genes predominantly determine the progress of development, while environmental factors play a minor role [6]. Every disease reflects the interactions between genes and the environment [7]. In monogenic disorders, a single mutation can lead to a specific phenotype [7]. However, the spectrum and degree of manifestations depend on numerous environmental factors [7]. The common embryologic neural origin of the ectoderm includes the epidermal layer of the skin and the amelodentinal (the enamel and dentine) components of the teeth, resulting in various conditions affecting both the skin and dentition to different degrees [8].

Dermatitis, also known as eczema, is a chronic relapsing inflammatory skin condition that causes dry skin, redness, and itchiness. Its etiology is not clear, but recent research indicates that its pathogenesis is complex, resulting from a combination of genetic and environmental factors that induce skin barrier dysfunction, cutaneous and systemic immune dysregulation, skin microbiota dysbiosis, and a strong genetic influence. Specific types of dermatitis are more common in certain age groups. For example, atopic dermatitis (AD) is much more common in children than in adults [9].

Psoriasis is one of the most frequent chronic and inflammatory skin diseases in childhood. Polygenic susceptibility and environmental triggers are believed to be involved in the pathogenesis of psoriasis. Like AD, psoriasis presents with scaly and dry skin [9].

Nevus is a nonspecific medical term for a visible, circumscribed, chronic lesion of the skin or mucosa. The term nevus is applied to a number of conditions caused by neoplasias and hyperplasias of melanocytes, as well as a number of pigmentation disorders, both hypermelanotic (containing increased melanin, the pigment responsible for skin color) and hypomelanotic (containing decreased melanin) [9].

Mastocytosis encompasses a group of rare clinical entities that are characterized by abnormal growth and, usually, a low accumulation of clonal and morphologically abnormal mast cells within one or more organs. Childhood-onset mastocytosis is generally detectable during the first year of life. This type of mastocytosis is most often restricted to the skin [9].

Morphea, or localized scleroderma, is an inflammatory disease that primarily affects the skin and underlying tissues, leading to sclerosis of the affected tissues. Besides genetic predisposition, there are environmental triggers such as traumatic factors, radiation, medications, and infections [9].

The skin is the organ of the human body that is most commonly involved in monogenic diseases. More than one-third of all genetic diseases affect the integument. Currently, more than 350 genodermatoses have been identified with functional insights [9].

Genetically inherited skin diseases affect not only the skin but also the oral tissues. For example, in ectodermal dysplasia (ED), dental anomalies like hypodontia are a core clinical feature [9].

The ichthyoses are a large, heterogeneous group of skin cornification disorders. They can be inherited or acquired and result in defective keratinocyte differentiation and abnormal epidermal barrier formation. The resultant skin barrier dysfunction leads to increased transepidermal water loss and inflammation. Disordered cornification is clinically characterized by skin scaling with various degrees of thickening, desquamation (peeling), and erythema (redness) [9].

Xeroderma pigmentosum is one of the rare genetically inherited skin diseases that causes sensitivity to ultraviolet radiation from sunlight, resulting in pigmentation changes in the skin, and can lead to skin cancers [9]. Additionally, epidermolysis bullosa (EB) is a very rare disease caused by mutations of proteins in the dermo-epidermal junction. The disease has four phenotypic subgroups: Epidermolysis Bullosa Simplex (EBS), Junctional Epidermolysis Bullosa (JEB), Dystrophic Epidermolysis Bullosa (DEB), and Kindler Syndrome (KEB). Junctional EB is notable for its effects on teeth, known as amelogenesis imperfecta (AI). AI may be a phenotypic enamel manifestation in syndromes like JEB. All the genes related to JEB are involved in ameloblast cell adhesion, which is critical for normal enamel mineralization [9].

Albinism includes a group of genetically inherited autosomal recessive conditions typically characterized by a congenital reduction or absence of melanin pigment biosynthesis, which gives the natural color to the skin, iris, and hair. Traditionally, albinism has been classified according to clinical phenotype, with the two main categories being oculocutaneous albinism (OCA) types 1, 2, and 3, and ocular albinism [9].

Our literature search showed that the overall relationship between dermatological conditions and the developmental anomalies of teeth is not fully explored. Considering that the skin shares a common embryological background with enamel, the factors that cause any skin disorder may also cause developmental anomalies in the teeth [10]. Hence, the aim of the study was to evaluate the occurrence of dental anomalies (DDE, MIH, hypodontia, hyperdontia, taurodontism, root malformations, short root anomaly) among pediatric patients with skin diseases.

Ethical Approval

and Study Population: Ethical approval was obtained prior to the study from the Marmara University Faculty of Dentistry Clinical Research Ethics Committee on June 6, 2017, with approval number #2017 − 117. This study was performed in accordance with the Helsinki Declaration. Informed consent was obtained from the parents of all the children involved in the study.

Sample Size Calculation: GPower statistical software (v.3.1.9.6., Universitat Kiel, Kiesl, Germany) was used to calculate the sample size. The calculation was based on the findings of the study by Hernandez et al., which showed a statistically significant relationship between AD and MIH [22]. The researchers investigated the relationship between different diseases and MIH and reported that MIH was observed significantly more in the population with AD (OR: 90.9; CI: 33.4–247.1; p < 0.001). Accordingly, for the two-tailed sample size calculation of the present study, the significance level alpha was set at 0.05, and the critical z level was determined to be 1.95 at a power level of 98%. In line with these parameters, it was concluded that the sample size should be at least 97, with at least 45% in the study group. Considering a 10% dropout risk, the minimum sample size was determined to be 107.

Study Design

We conducted the present comparative observational study during September 2017–May 2018. Seventy-one patients with skin disease (between 4 and 14 years old) who applied to Marmara University Department of Dermatology fulfilled the inclusion criteria described below.

Inclusion Criteria:

Pediatric patients aged 4–14 who applied to the dermatology clinic where chronic and genetic skin diseases are followed up.
Children who complied with the implementation of the procedures required for the investigation.

Exclusion Criteria:

Patients with acute dermatological conditions.
Patients not between the ages of 4 and 14.
Children who did not comply with the implementation of the procedures required for the investigation.

Forty-one healthy children who applied to Marmara University Department of Pediatric Dentistry for the first time, matched by age and gender with the study group, were designated as the control group. The health status of the patients, indicating the absence of skin diseases, was confirmed based on their medical records and parents’ self-proclamation.

Clinical Examination

Patients eligible for inclusion in this study were identified during the clinical examinations at the Marmara University Department of Dermatology Pediatric Follow-up Clinic while they were being examined for their dermatological condition. The patients' diagnoses and medications were verified and confirmed by a dermatology specialist (ADY), and the archive records of Marmara University Dermatology Department were scanned for the patients' past history.

Questionnaire

A questionnaire form was filled out by parents to evaluate the family’s demographics, oral hygiene habits, and the prenatal, natal, and postnatal histories of the patient.

Although the first intraoral examinations were performed at the dermatological outpatient clinic, the second intraoral examinations were conducted in the dental settings of the pediatric dentistry clinic. These examinations were performed by a single trained examiner (EO) under the same dental unit and reflector, with a smooth-surfaced standard mouth mirror (#5) and tactile inspection with a standard dental probe (PCP11). We evaluated enamel defects with the Modified Developmental Defects of Enamel Index (mDDE Index), which is the preferred index in many epidemiological studies [11]. This index includes normal, demarcated opacities (white/cream or yellow/brown), diffuse opacities (diffuse lines, diffuse patchy, diffuse confluent, confluent/patchy, + and loss of enamel), and hypoplasia (pits, missing enamel, or any other defects). Elfrink et al. stated that intraoral dental photographs can be used to evaluate DDE [12]. Thus, to facilitate repeated evaluations, dental photographs were taken. An intraoral digital camera (Canon EOS 750D) and macro lens (Canon EF 100mm f/2.8L IS USM, Tokyo, Japan) fixed on the camera with an owl bracket were used to take intraoral photographs. The camera was used in “Auto” mode for each shot.

Because of the deficiencies of the mDDE Index in recording atypical restorations or caries, post-eruptive enamel breakdown, MIH-related tooth loss, and amelogenesis imperfecta, we used the MIH Index for the repeated examinations [13]. Teeth affected by MIH and DDE-related MIH were evaluated using the criteria approved by the European Academy of Paediatric Dentistry (EAPD) in 2015 [14]. These criteria include no visible enamel defects, enamel defects that are not related to MIH (diffuse opacities, hypoplasia, amelogenesis imperfecta, hypomineralization defect), white/creamy and yellow/brown demarcated opacities, post-eruptive enamel breakdown, atypical MIH-specific restorations and caries, tooth loss due to MIH, and unscored conditions [14]. The intra-examiner Kappa values of the examiner who diagnosed MIH and DDE-not-related MIH ranged from 0.83 to 0.89. The high degree of agreement was retained during re-examination periods.

We had some limitations for taking photos of all patients. For ichthyosis patients, we couldn’t use a dental retractor because of their angular cheilitis. Additionally, we couldn’t take photos of a four-year-old child because of her dental anxiety. Due to these limitations, we used the initial evaluation’s mDDE Index scores and renamed them to the MIH Index.

Panoramic radiographs were taken, and dental anomalies observed in the radiographs (hypodontia, hyperdontia, taurodontism, short root anomaly) were recorded.

The diseases of the children in the study group were grouped as genodermatoses and chronic skin diseases under the guidance of the dermatology specialist (ADY). Then, statistical analyses were performed to compare the obtained data between the control group and the study group and its subgroups.

Statistical Analyses:

SPSS version 21.0 software (IBM, Chicago, Illinois, USA) was used for statistical analysis. Descriptive analyses of numerical variables were calculated as the mean and standard deviation. The chi-square and Fisher's exact tests were used for the analysis of categorical variables. The Shapiro-Wilk test was used for normality analysis. We used the Student's t-test or Mann-Whitney U test to compare the numerical data of two groups based on the normality analysis. Values equal to or less than 0.05 (p ≤ 0.05) were interpreted as statistically significant.

A total of 71 pediatric patients with dermatological diseases were included in the study. The diagnoses were as follows: dermatitis in 34, nevus in 9, psoriasis in 8, epidermolysis bullosa in 7, ichthyosis group of disorders in 5, ectodermal dysplasia in 2, xeroderma pigmentosum in 2, morphea in 2, mastocytosis in 1, and albinism in 1 patient.

The mean age of the study group and the control group was 9.05 ± 2.47 and 8.51 ± 2.66, respectively (p = 0.19). There were 40 (56%) girls and 31 (44%) boys in the study group, and 21 (51%) girls and 20 (49%) boys in the control group (p = 0.6). The study group had a statistically significantly shorter breastfeeding duration than the control group (p = 0.009); systemic disease and allergy in the first three years of life were found to be statistically significantly higher in the study group than in the control group (p = 0.06 and p = 0.024, respectively) [Supplementary Table].

The children in the study group had a statistically significantly higher incidence of diseases in the first 3 years of life compared to the control group (p = 0.006). There were 29 patients in the study group who had a disease in the first 3 years of life. Of these patients, 15 had skin disease, 4 had allergic asthma, 8 had allergies, 3 had an infectious disease, and 2 had been operated on for various reasons. In the control group, there were 7 patients who had a disease in the first 3 years of life. While three of these patients were operated on for cryptorchidism, inguinal hernia, and eye canal occlusion, others had an infectious disease. It was observed that 40.5% of the diseases in the first 3 years of life among the children in the study group were existing skin diseases.

MIH was detected in 23% (n = 16) of the study group. Although this rate was approximately twice the 12% MIH rate in the control group (n = 5), there was no statistically significant difference between the groups (p = 0.17) [Table I]. DDE-not-related MIH was found to be statistically significantly higher in the study group than in the control group (p = 0.01). Additionally, other dental anomalies (hypodontia, hyperdontia, taurodontism, and root malformations) were found to be statistically significantly higher in the study group than in the control group (p = 0.03).

We evaluated the study group in two subgroups: chronic skin diseases (n = 54) and genodermatoses (n = 17) [Table II]. Although there was no statistically significant difference between subgroups in terms of MIH, there was a statistically significant difference in favor of the genodermatoses group in terms of DDE-not-related MIH (p = 0.002) and other dental anomalies (p < 0.001). Table III shows the comparison of the genodermatoses group with the control group. In this table, DDE-not-related MIH and other dental anomalies show high statistical significance in favor of the genodermatoses group.

Table IV shows the evaluation of dental anomalies in the chronic skin disease group and the control group. In this table, MIH and DDE-not-related MIH percentages are statistically significantly higher in the chronic skin disease group than in the control group (p = 0.004, p = 0.2, respectively) [Table IV].

Table V shows the different features that were diagnosed in the patients with skin diseases. Dermatitis was found to be the most frequent skin disease (47.9%), and almost one third of these patients (29.5%) had MIH and 11.8% had DDE-not-related MIH [Table V]. Other dental anomalies (17.5%) in dermatitis patients included three patients with hypodontia and one with primary failure of eruption in the lower molars.

This study encompasses the dental findings of pediatric patients visiting dermatology clinics. We believe it is clinically significant as it collectively evaluates the oral and dental health of children with dermatological problems. Interactions of multiple genes, epigenetic factors, and environmental influences not only affect the development of the skin but also the shape of individual teeth, groups of teeth, and the dentition as a whole [15]. To date, more than 200 genes involved in embryonic development, morphogenesis, and differentiation of tooth processes have been identified [16].

There are several dermatologic disorders that have a genetic etiology or a genetic predisposition, called “genodermatoses” [17]. Most of the genodermatoses cause anomalies that affect the size, number, shape, or structure of the tooth [8].

One of the genodermatoses is ectodermal dysplasia (ED), and in many EDs, dental defects such as hypodontia and oligodontia of the primary and permanent teeth represent a core clinical feature [18]. The oral findings of the ED patients in the current study are consistent with the literature.

Current information regarding dental anomalies in epidermolysis bullosa (EB) is based on case reports, most of which describe patients with generalized enamel hypoplasia [19–21], one with failure of tooth eruption [19], one with a localized enamel defect [22], and one with amelogenesis imperfecta [23]. In the current study, in addition to generalized DDE, amelogenesis imperfecta, and failure of tooth eruption, hypodontia and taurodontism were also detected (Figure I). Fig I Panoramic radiograph of a patient with epidermolysis bullosa. Hypodontia and taurodontism in molar teeth were detected in a patient diagnosed with amelogenesis imperfecta

The oral manifestations of xeroderma pigmentosum (XP) are mainly related to the occurrence of malignant tumors in the lips, tongue, and buccal mucosa [9]. In the current study, DDE in XP was revealed for the first time, but there was no finding of oral malignancy in our patients as in the literature.

Dental anomalies in ichthyosis patients are based on case reports. There is limited knowledge about the oral manifestations of ichthyosis. In some studies, the presence of supernumerary teeth [24], hypodontia [25], and DDE [26] were reported. Basel-Vanagaite et al. described conical (deciduous) teeth or notched and pitted permanent teeth in three individuals with ichthyosis and hypotrichosis [27]. In the current study, in addition to hypodontia and DDE, short root anomaly was revealed for the first time (Figure II). Fig II Short root anomaly was detected in the panoramic radiograph of a patient with ichthyosis

Varied dental features have been reported in albinism patients. In a case of oculocutaneous albinism (OCA), the patient had an upper maxillary lateral incisor showing features of both dens invaginatus (dens in dente) and dens evaginatus [28]. In another case, enamel hypoplasia has been reported in brothers with OCA in both primary and permanent dentition [29]. In the current study, MIH was found in an albinism patient; the oral findings of the albinism patient did not match the literature.

In the current study, dental anomalies in the genodermatoses group were compared with both the chronic skin disease group and the healthy control group. DDE-not-related MIH, such as amelogenesis imperfecta, and other dental anomalies, such as hypodontia, were found to be significantly higher statistically in the genodermatoses group, in line with the literature. This confirms that such anomalies are frequently seen in genetically-based diseases.

The concept of multifactorial etiology is more prominent in chronic skin diseases. In the current study, dermatitis constituted 63% of the chronic skin disease group. There are studies in the literature investigating the relationship between atopic dermatitis and dental defects. In the study about the effect of AD on dentition published by Tan et al. in 2022, they found that enamel hypoplasia (33%) and hypodontia (13%) were reported [30]. Sodré in 2020 found 17.4% of the demarcated opacities in AD patients [31]. However, both of them were cross-sectional studies in which they did not have a control group to compare their results with a healthy population. In the study by Periqua about dental manifestations of AD in 2017, it was shown that anatomical dental abnormalities, including agenesia, hypoplasia, black-stain, tooth fusion, dental ankylosis, and odontogenic tumors, were seen in 13/90 (14.4%) of AD patients [32]. Hernandez revealed, for the first time, a statistically significant relationship between AD and the presence of MIH. In Hernandez’s study, it was found that 45% of children with MIH had AD [12]. The results of the dermatitis patients in the current study also support the literature (Figure III). Fig III MIH was detected of a patient with atopic dermatitis

Although there are well-documented reports of psoriatic lesions appearing on oral soft tissues (lips, tongue, palate, buccal mucosa, and gingiva), there is only one case report focused on oral hard tissues, describing a patient with psoriasis who has oligodontia and DDE [33]. Similar to the results of this case report, half of the psoriasis patients in the current study had dental defects (25% MIH, 12.5% DDE-not-related MIH, and 12.5% hypodontia) (Figure IV). Fig IV hypodontia was detected in the panoramic radiograph of a patient with psoriasis

Oral mucosa and dental involvement have only been rarely described in morphea [34]. In a survey of the reported 25 cases of morphea associated with oral and dental manifestations, besides the typical cutaneous features, the patients presented unerupted teeth, root development defects, root atrophy, and short roots [34]. In the current study, there was no involvement of morphea lesions in the mouth in either of our morphea patients, and, contrary to the literature, there was no root problem. However, the current study revealed MIH in a morphea patient for the first time (Figure V). Fig V MIH was detected of a patient with morphea

It has been reported that more than 50% of patients with mastocytosis have bone pathology. Apart from a case report documenting mast cell infiltration in the jawbone [35], no other reports regarding the oral findings of this disease have been found in the literature. Due to the lack of sufficient data in the literature and the small number of mastocytosis patients in the current study, it is not possible to discuss the results.

Reports describing intraoral anomalies associated with nevi are few. In a review, dental anomalies observed in 13 patients with oral linear epidermal nevus were reported as odontoma in one, and hypoplasia and hypodontia in the other [36]. In both cases, linear lesions were intraorally placed. Many inherited and acquired diseases of the skin or mucosa manifest themselves in a linear configuration. The embryological basis of the distribution pattern of these lines remains, so far, an enigma, although it has been reported that they may represent visible embryonic migration paths of clones of ectodermal cells that carry genetic disorders [37]. In some of our patients, the lesions were in a linear configuration, but in none of our patients were the lesions adjacent to oral tissues. In the current study, 55% of patients with nevi had different types of enamel defects, but none of them had any dental anomalies like hypodontia, as reported in the literature.

According to the current study, while DDE-not-related MIH and dental anomalies predominated in the genodermatoses group, the opposite was the case in chronic skin diseases, where MIH was found to be significantly higher.

The progression of dermatitis group diseases in the world and the perspective of researchers on this disease are not much different from the perspective of pedodontists on MIH. In prevalence studies, results differ for both diseases in different parts of the world [38, 39]. Interestingly, both diseases are not easily diagnosed [40, 41]. It is thought that environmental and genetic effects play a role in the etiology of both diseases [2, 9]. In recent years, these diseases have attracted more attention from researchers, and null mutations in the filaggrin (FLG) gene have been established as a strong genetic risk factor for AD [42]. Therefore, future studies with larger sample sizes of patients with dermatologic diseases are necessary to confirm and expand upon our findings, providing a better understanding of the dental characteristics associated with these disorders and to understand what affects our teeth and skin separately or together.

Strength and Limitations of this Study:

Oral findings of skin diseases are generally included as case reports in the literature. However, this study is the first to reveal the relationship between skin diseases and dental anomalies by comparing them with a healthy control group. On the other hand, this study has some limitations. The current study was conducted in Istanbul, a metropolis with a population of approximately 20 million. It was challenging to encourage families who regularly visit dermatology clinics to come to another hospital and participate in our study. Despite our efforts, many families with children suffering from skin diseases chose not to participate. Children with skin diseases, which cover the largest area of the body and are the most visible organ, were often shy and had a lower quality of life. Although cooperation was generally good, the children were hesitant about the possibility of dental treatment.

The current study confirmed a connection between MIH and chronic skin diseases, as well as a relationship between genodermatoses, DDE-not-related MIH, and other anomalies. Children with skin diseases may have poor oral health and esthetics. To comprehend the underlying mechanisms and create management plans for dental anomalies in patients with skin diseases, more research is required. Dental screening and management should be taken into account as part of the patient's overall health care plan, and clinicians should be aware of the potential oral health issues associated with skin diseases.

Author contribution: Conceptualization: [AEO], [ADY], [AM]; Methadology: [AEO], [ADY], [AM]; Formal analysis and investigation: [AEO], [AM]; Writing--- original draft preparation [AEO]; Writing--- review and editing [AEO], [ADY], [AM]; Supervision: [AM].

Funding: This research was supported by Marmara University Scientific Research Projects Commission Presidency, SAG-C-DRP-090518-0218.

Data availability: The datasets generated during and/or analysed during the current study are available from corresponding author on reasonable request.

Ethics approval: The ethics approval was achieved prior to study from Marmara University Faculty of Dentistry Clinical Research Ethics Committee on 06.2017 with # 2017-117. This study was performed under the Helsinki Declaration.

Consent of participate: Informed consent was obtained from the parents of all the children for the procedure.

Competing interests: The authors declare no competing interests.

Anthonappa RP, King NM (2015) Enamel Defects in the Permanent Dentition: Prevalence and Etiology. In: Drummond B, Kilpatrick N (eds) Planning and Care for Children and Adolescents with Dental Enamel Defects. Springer, Berlin
Alaluusua S (2010) Aetiology of Molar-Incisor Hypomineralisation: A systematic review. Eur Arch Paediatr Dent 11(2):53-58. https://doi.org/10.1007/BF03262713
Torlińska-Walkowiak N, Majewska KA, Sowińska A, Kędzia A, Opydo-Szymaczek J (2023) Developmental enamel defects and dental anomalies of number and size in children with growth hormone deficiency. Sci Rep 13(1):14707. https://doi.org/10.1038/s41598-023-41892-x
Weerheijm KL, Jälevik B, Alaluusua S (2001) Molar-incisor hypomineralisation. Caries Res 35(5):390-1. https://doi.org/10.1159/000047479
Neville BW, Damm DD, Allen CM, et al (2016) Oral and Maxillofacial Pathology. 16th ed. Elsevier, St. Louis, MO
Thesleff I (2014) Current understanding of the process of tooth formation: transfer from the laboratory to the clinic. Aust Dent J 59 Suppl 1:48-54. https://doi.org/10.1111/adj.12102
Itin PH, Burger B (2010) Genodermatosen in der täglichen Praxis - die Rolle des Dermatologen [Genodermatoses for practitioners--principles and concepts]. Ther Umsch 67(9):483-485. https://doi.org/10.1024/0040-5930/a000081
Freiman A, Borsuk D, Barankin B, Sperber GH, Krafchik B (2009) Dental manifestations of dermatologic conditions. J Am Acad Dermatol 60(2):289-298. https://doi.org/10.1016/j.jaad.2008.09.056
Odom RB, James WD, Berger TG (2000) Andrews's Diseases of the Skin, Clinical Dermatology. 9th ed. WB Saunders, Philadelphia, pp 1069-1071
Hernandez M, Boj J, Espasa E, Planells P, Peretz B (2018) Molar-Incisor Hypomineralization: Positive Correlation with AD and Food Allergies. J Clin Pediatr Dent 42(5):344-348. https://doi.org/10.17796/1053-4625-42.5.4
Clarkson J, O'Mullane D (1989) A modified DDE Index for use in epidemiological studies of enamel defects. J Dent Res 68(3):445-450. https://doi.org/10.1177/00220345890680030201
Elfrink ME, Veerkamp JS, Aartman IH, Moll HA, Ten Cate JM (2009) Validity of scoring caries and primary molar hypomineralization (DMH) on intraoral photographs. Eur Arch Paediatr Dent 10 Suppl 1:5-10. https://doi.org/10.1007/BF03262693
Weerheijm KL (2003) Molar incisor hypomineralisation (MIH). Eur J Paediatr Dent 4(3):114-120
Ghanim A, Elfrink M, Weerheijm K, Mariño R, Manton D (2015) A practical method for use in epidemiological studies on enamel hypomineralisation. Eur Arch Paediatr Dent 16(3):235-246. https://doi.org/10.1007/s40368-015-0178-8
Brook AH (2009) Multilevel complex interactions between genetic, epigenetic and environmental factors in the aetiology of anomalies of dental development. Arch Oral Biol 54 Suppl 1:S3-S17. https://doi.org/10.1016/j.archoralbio.2009.09.005
Abu-Hussein M, Watted N, Yehia M, Prof P, Iraqi F (2015) Clinical genetic basis of tooth agenesis. J Dent Med Sci 14:68-77
Rimoin DL, Connor JM, Pyeritz RE (1996) Emery and Rimoin's Principles and Practices of Medical Genetics. 3rd ed. Churchill Livingstone, New York, NY, pp 1383-1385
Deshmukh S, Prashanth S (2012) Ectodermal Dysplasia: A Genetic Review. Int J Clin Pediatr Dent 5:197-202. https://doi.org/10.5005/jp-journals-10005-1165
Brooks JK, Bare LC, Davidson J, Taylor LS, Wright JT (2008) Junctional epidermolysis bullosa associated with hypoplastic enamel and pervasive failure of tooth eruption: Oral rehabilitation with use of an overdenture. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 105:e24–e28
Sadler E, et al (2005) Dental alterations in junctional epidermolysis bullosa – report of a patient with a mutation in the LAMB3-gene. J Dtsch Dermatol Ges 3:359-363
Wright JT, Fine JD, Johnson L (1994) Dental caries risk in hereditary epidermolysis bullosa. Pediatr Dent 16:427-432
Wright JT, Fine JD, Johnson LB, Steinmetz TT (1993) Oral involvement of recessive dystrophic epidermolysis bullosa inversa. Am J Med Genet 47:1184-1188
Javed AP, Shenai P, Chatra L, Veena KM, Rao PK, Prabhu R (2013) Occurrence of epidermolysis bullosa along with Amelogenesis imperfecta in a female patient of India. Dent Res J (Isfahan) 10(6):813-816
Rodrigues GF, Losso AR, dos Reis Moraes R, Duarte ML, Macedo RG, de Araújo Castro GF (2023) Complexity of dental management of a pediatric patient with lamellar ichthyosis: case report. Research, Society and Development 12(4):e23912441211
Pranitha V, Thimma Reddy BV, Daneswari V, Sudhanwan ND (2014) Lamellar Ichthyosis - A case report. J Clin Diagn Res 8(11):ZD01-2
Nair K, Kodhandram GS (2016) Oral manifestations of lamellar ichthyosis: A rare case report. Indian J Paediatr Dermatol 17:283. https://doi.org/10.4103/2319-7250.184428
Basel-Vanagaite L, Attia R, Ishida-Yamamoto A, et al (2007) Autosomal recessive ichthyosis with hypotrichosis caused by a mutation in ST14, encoding type II transmembrane serine protease matriptase. Am J Hum Genet 80(3):467-477
Suprabha BS (2005) Premolarized double dens in dente in albinism—a case report. J Indian Soc Pedod Prev Dent 23:156-158
Odom RB, James WD, Berger TG (2000) Andrews's Diseases of the Skin Clinical Dermatology. 9th ed. WB Saunders, Philadelphia, pp 1069-1071
Tan SY, Leong SW, Hsu CS, Chandran NS (2022) Association of Moderate-Severe AD with Dental Anomalies. Indian J Dermatol 67(5):539-542. https://doi.org/10.4103/ijd.ijd_375_22
Stofella Sodré C, Ferreira DC, Vieira MS, et al (2021) Clinical oral profile of pediatric patients with AD: A cross-sectional study. Oral Dis 27(7):1834-1846. https://doi.org/10.1111/odi.13721
Perugia C, Saraceno R, Ventura A, et al (2017) AD and dental manifestations. G Ital Dermatol Venereol 152(2):122-125. https://doi.org/10.23736/S0392-0488.16.05224-X
Rai P, Kumar G, Chaudhary M (2010) Psoriatic triad in a patient presenting with oligodontia. J Indian Soc Pedod Prev Dent 28(4):297-301. https://doi.org/10.4103/0970-438
Tang MM, Bornstein MM, Irla N, Beltraminelli H, Lombardi T, Borradori L (2012) Oral mucosal morphea: a new variant. Dermatology 224(3):215-220. https://doi.org/10.1159/000337554
Castling B, Smith AT, Myers B (2006) Involvement of the jaw bones in systemic mastocytosis. Br J Oral Maxillofac Surg 44(2):87-88. https://doi.org/10.1016/j.bjoms.2005.03.021
Tesi D, Ficarra G (2010) Oral linear epidermal nevus: a review of the literature and report of two new cases. Head Neck Pathol 4(2):139-143. https://doi.org/10.1007/s12105-010-0165-7
Bolognia JL, Orlow JJ, Glick SA, et al (1994) Lines of Blaschko. J Am Acad Dermat 31:157–190
Hadi HA, Tarmizi AI, Khalid KA, Gajdács M, Aslam A, Jamshed S (2021) The Epidemiology and Global Burden of Atopic Dermatitis: A Narrative Review. Life (Basel) 11(9):936. https://doi.org/10.3390/life11090936
Lygidakis NA, Garot E, Somani C, Taylor GD, Rouas P, Wong FSL (2022) Best clinical practice guidance for clinicians dealing with children presenting with molar-incisor-hypomineralisation (MIH): an updated European Academy of Paediatric Dentistry policy document. Eur Arch Paediatr Dent 23(1):3-21. https://doi.org/10.1007/s40368-021-00668-5
Jälevik B, Szigyarto-Matei A, Robertson A (2019) Difficulties in identifying developmental defects of the enamel: a BITA study. Eur Arch Paediatr Dent 20(5):481-488. https://doi.org/10.1007/s40368-019-00431-x
Weidinger S, Nosbaum A, Simpson E, Guttman E (2022) Good practice intervention for clinical assessment and diagnosis of atopic dermatitis: Findings from the atopic dermatitis quality of care initiative. Dermatol Ther 35(3):e15259. https://doi.org/10.1111/dth.15259
Novak N, Baurecht H, Schäfer T, Rodriguez E, Wagenpfeil S, Klopp N, Heinrich J, Behrendt H, Ring J, Wichmann E, Illig T, Weidinger S (2008) Loss-of-function mutations in the filaggrin gene and allergic contact sensitization to nickel. J Invest Dermatol 128(6):1430-1435. https://doi.org/10.1038/sj.jid.5701190

Table I: Evaluation of Anomaly Types Percentages in Study and Control Groups
Defect Types	Study group (n=71)		Control group (n=41)		p⁺
Defect Types	+	-	+	-	p⁺
MIH	16 (23%)	55 (77%)	5 (12%)	36 (88%)	0.17
DDE not related MIH	18 (25%)	53 (75%)	3 (7%)	38 (93%)	0.01
Other Anomalies	11 (15%)	60 (85%)	1 (2%)	40 (98%)	0.03
⁺: Ki Square Test

Table II: Evaluation of Anomaly Types Percentages in Study Groups Subgroups
Defect types	Genodermatosis (n=17)		Chronic Skin diseases (n=54)		p⁺
Defect types	+	-	+	-	p⁺
MIH	1 (6%)	16 (94%)	15 (28%)	39 (72%)	0.059
DDE not related MIH	9 (53%)	8 (47%)	9 (17%)	45 (83%)	0.002
Other Anomalies	7 (41%)	10 (59%)	4 (7%)	50 (93%)	<0.001
⁺: Ki Square Test

Table III: Evaluation of Anomaly Types in Genodermotosis Group and Control group
Defect types	Genodermatosis (n=17)		Control group (n=41)		p⁺
Defect types	+	-	+	-	p⁺
MIH	1 (6%)	16 (94%)	5 (12%)	36 (88%)	0.47
DDE not related MIH	9 (53%)	8 (47%)	3 (7%)	38 (93%)	<0.001
Other Anomalies	7 (41%)	10 (59%)	1 (2%)	40 (98%)	<0.001
⁺: Ki Square Test

Table IV: Evaluation of Anomaly Types in Chronic Skin Diseases Group and Control group
Defect types	Chronic Skin diseases (n=54)		Control group (n=41)		p⁺
Defect types	+	-	+	-	p⁺
MIH	15 (28%)	39 (72%)	5 (12%)	36 (88%)	0.004
DDE not related MIH	9 (17%)	45 (83%)	3 (7%)	38 (93%)	0.02
Other Anomalies	4 (7%)	50 (93%)	1 (2%)	40 (98%)	0.08
⁺: Ki Square Test

Table V: Skin Diseases in the Study Group and the Types and Percentages of Anomalies Seen in These Diseases
Diseases	MIH	DDE not related MIH	Other anomalies
Dermatitis (n=34)	10 (29.5%)	4 (11.8%)	3 (17.5%)
Psoriasis (n=8)	2 (25%)	1 (12.5%)	1 (12.5%)
Morphea (n=2)	1 (50%)
Mastocytosis (n=1)		1 (100%)
Nevus (n=9)	2 (22.2%)	3 (33%)
Ectodermal Dysplasia (n=2)			2 (100%)
Epidermolysis Bullosa (n=7)		7 (100%)	3 (43%)
Xeroderma Pigmentosum (n=2)		1 (50%)
Ichthyosis (n=5)		1 (20%)	2 (40%)
Albinism (n=1)	1 (100%)

No competing interests reported.

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Developmental Defects of Enamel and Dental Anomalies in Children with Skin Diseases

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