Vol. 29 No. 04 Oct-Dec 2020

Ahsan Inayat                                  BDS

Muneeb Ahmed Lone                  BDS, FCPS

Inflammation that is associated with periodontium and its related structures can be managed in most of the patients but unfortunately, not all forms of periodontal disease respond to treatment. Periodontitis that is resistant to conventional periodontal therapy are recognized in certain medical conditions.1
Certainly one of the most challenging condition is Papillon-Lefèvre Syndrome. Papillon-Lefèvre Syndrome is a rare inherited autosomal recessive disorder which was first described by two french physicians, Papillon & Lefèvre in 1924.5
It usually causes severe destructive periodontal damage affecting both deciduous and the secondary dentition which can result in early loss of teeth. It affects the skin which is characterized by redness, hyperkeratosis of palm and sole and is caused by mutation in cathepsin-C gene.1 Hence it is a genetic defect that is located on chromosome 11q14.1-q14.3, which involves mutation of gene CTSC.3
The syndrome has an estimated prevalence of 1 to 4 cases per million. Incidence is seen in patient’s with consanguineous marriages.4 Such patients suffer from severe periodontal destruction as the primary teeth are lost as early as 4 years and most permanent teeth by age of 14 years.3-4 The etio-pathogenesis of PLS is
not completely understood however anatomic, microbial and viral agents as well as host response are suspected causative factors. Male and female are equally affected with no racial predominance.5 Here we report a case with clinical findings of such complex syndrome.
A 19-year-old male patient presented to the Prosthodontics department with the chief complaint of difficulty in chewing and bad breath. Past dental history revealed that the patient had early loss of deciduous teeth by the age of 5-6 years. He got his mandibular central incisors extracted a couple of years ago because he had difficulty in chewing with these teeth. Clinical examination revealed marked hyperkeratotic patches on dorsum of aspect hands (Fig 1). The nails of the hands were keratotic but were not malformed. His feet were not examined as the patient did not give consent to removal of his shoes; but informed that they were similar in appearance to his hands.
On intra-oral examination, there was generalized inflammation of the mucosa and grade III mobility in almost all the teeth (Fig 2). The Orthopantomogram (OPG) revealed advanced bone loss and a typical Papillon-Lefèvre Syndrome finding of floating teeth (Fig 3). No ultimate treatment exists for the prevention or management of periodontitis associated

Fig. 1: Hyperkeratotic patches on hands

Fig. 2: Intraoral view

with PLS.3 After discussing the various management options, patient opted for provision of conventional complete dentures after extraction of all the teeth. Owing to young age and better motor skills, such patients usually adapt easily and quickly to the prosthesis.4 More recently, implant-supported removable or fixed prosthesis,with and without bone augmentation,have been used as definitive treatment modality to rehabilitate such patients. However, only a few cases have been reported in literature and long-term clinical outcomes need to be established.5,6

Fig. 3: Panoramic radiograph showing advanced bone loss (teeth Floating in air)

Periodontitis is the more serious component of this disease. The deciduous teeth erupt at the usual time but then the onset of disease is heralded by gingivitis and a
florid Periodontitis. 2,4-8 There is no definitive treatment available for preventing Periodontitis although oral-hygiene instructions, root planning and scaling along with antibiotics can improve condition. In later stages, Patients with this disease may loose all of their teeth. Hence managing such a patient should involve a multidisciplinary team approach that involves a Periodontist, Prosthodontist, Dermatologist and Psychologist for treatment. 6,9

CONFLICT OF INTEREST

None declared

REFERENCES

1. Hart TC, Shapira L. Papillon-Lefèvre syndrome. Periodontol 2000;6:88-100 https://doi.org/10.1111/j.1600-0757.1994.tb00029.x
2. Sreeramulu B, Haragopal S, Shalini K, Sudha MD, Kiran G. The prosthodontic management of a young edentulous patient with the papillon lefevre syndrome-a rare case report. J Clin Diagn Res. 2012;6:1808-811 https://doi.org/10.7860/JCDR/2012/4884.2607
3. Ahmed B. Prosthodontic Rehabilitation of PapillonLefevre Syndrome. J Coll Phys Surg Pak J Coll Phy Sug Pak. 2014;24(Suppl 2):S132-34
4. Khan FY, Jan SM, Mushtaq M. Papillon-Lefèvre syndrome: Case report and review of the literature. J Indian Soci Periodontol. 2012;16:261. https://doi.org/10.4103/0972-124X.99273
5. Drucker DB, Marshall R, Bird PS. Aetiology of Papillon LeFèvre syndrome. Anaerobe. 2001;7:151-58 https://doi.org/10.1006/anae.2001.0369
6. Sreeramulu B, Shyam ND, Ajay P, Suman P. Papillon-Lefèvre syndrome: clinical presentation and management options. Clin Cosmet Investig Dent. 2015;7:75-81 https://doi.org/10.2147/CCIDE.S76080
7. Alam A, Ovais N ,Ahmed B, Prosthodontics Rehabilitation of Oral Function, J Coll Phy Surg Pak. 2016,26:872
8.  Zaman H. Papillon Lefevre Syndrome. J Pak Dent Assoc.1989; 5:25-33
9. Ahmad M, Hassan I, Masood Q. Papillon-lefevre syndrome. J Dermatolo case reports. 2009;3:53. https://doi.org/10.3315/jdcr.2009.1039

1. MDS Resident, Division of Prosthodontics, Dow University Health Sciences.
2. Assistant Professor, Department of Prosthodontics, Dr Irshad-ul-Ebad Khan Institute of Oral Health Sciences.
   Corresponding author: “Dr. Ahsan Inayat ” < ahsan_inayat@hotmail.com >

Ahsan Inayat                                  BDS

Muneeb Ahmed Lone                  BDS, FCPS

HOW TO CITE: Inayat A, Lone MA. Papillon-Lefevre syndrome. J Pak Dent Assoc 2020;29(4):269-270.
DOI: https://doi.org/10.25301/JPDA.294.269
Received: 12 May 2020, Accepted: 07 July 2020
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Abul Khair Zalan                                   BDS

Khadeejah Khalil Zubairy                    BDS

Anser Maxood                                       BDS, FRACDS

Manahil Niazi                                          BDS

Hira Zaman                                             BDS, MDS

Anika Gul                                                BDS

Miraat Anser                                           BDS

INTRODUCTION

Papillon-Lefèvre syndrome (PLS), was first described by two physicians, Papillon and Lefèvre, in France, 1924.1 It is an autosomal recessive disorder characterized by diffuse transgradient palmar-plantar hyperkeratosis, with premature loss of deciduous and permanent teeth, along with the calcification of the dura mater.2 Genetic studies of patients with PLS have mapped the major gene locus to chromosome 11q24-q21 and revealed mutation and loss of function of the cathepsin gene.3
It affects1- 4 people per 1 million population with no gender or racial predilection. With the eruption of deciduous teeth, diffuse hemorrhagic and hyperplastic gingivitis along with the loss of periodontal attachment develops. This results in teeth radiographically appearing as “floating” in the soft tissue. By 4 to 5 years of age, all primary teeth are lost or extracted with the gingiva returning to normal state of health until the eruption of permanent teeth. The permanent teeth also start
exfoliation just after eruption and are lost by the age of 14-15. There is dramatic alveolar bone destruction, often leaving atrophied jaws. Dermatological manifestations, usually occurs before four years of age, include hyperkeratosis of palms and soles, nail dystrophy, hyperhidrosis and keratinization on elbows and knees with the lesions appearing as white, yellow-like or red plaques or patches that then develop cracks, crusts, or deep fissures. The exact etiopathogenesis of this syndrome is not clearly understood and genetic, bacterial, or possible immunologic etiologies have been proposed. Many microbiologic studies and clinical
examinations after different treatment protocols have established a close association between the occurrence of Actinobacillus actinomycetemcomitans and destruction of periodontium in patients with PLS.4

CASE REPORT

A 14 years old male and his brother, a 13 years old male, otherwise healthy individuals, were seen at the outpatient department of Pediatric dentistry, children hospital, Pakistan Institute of Medical Sciences (PIMS), Islamabad. The presenting complaint of both the patients was falling of teeth immediately after their eruption. Further history revealed that the parents of the patients had a primary consanguineous marriage and a positive history of early shedding of teeth in
the family. For elaboration, the cases are numbered as 1 and 2.

CASE 1

On extraoral examination, the 14 years old male had normal facial features and decreased facial height with hyperkeratosis on palms and feet. Intraoral examination
revealed compromised oral hygiene with accumulation of plaque and food debris around the tooth surfaces in the mandibular arch while the maxillary arch was edentulous with markedly resorbed alveolar bone. A total number of

Fig.1: Orthopantamogram showing generalized alveolar bone loss to apical third of roots, Mandibular canines and first premolars in both quadrants are almost out of the socket (floating in air appearance) with little or no bony support. Mandibular third molars found erupting in both quadrants. Resorbed and completely edentulous maxillary ridge can also be seen.

four teeth were present in the mandible which were canines and first premolars on both sides of the arch. AggressivePeriodontitis was present and all of the teeth  had Grade III mobility (according to miller tooth mobility index 1985) which seemed to be “floating in the soft tissue” because of severe bone loss and exposed cementum. OPG radiograph was advised , which showed that the four teeth present in the mandibular arch had only 1-2 mm of bone support while third molars had not erupted yet. A complete blood picture was advised which was normal. On the basis of detailed

family history, clinical and radiographic evaluation, the diagnosis of Papillon lefevere syndrome (PLS) was confirmed. Consanguineous marriages is considered as a high risk factor for this disease.11 Treatment was started with instructions for the improvement of oral hygiene and 0.12% chlorhexidine was prescribed for two weeks to decrease the bacterial load.
Because of relatively late presentation to the dental department as compared to his younger brother, all of the teeth present in the mandibular arch were already loosened due to aggressive periodontitis which was accompanied with poor bone support and grade III mobility, hence considered nonsalvageable and were extracted. Patient was recalled after six weeks to re-evaluate the oral health and healing gums and soft tissues. The oral health and healing of gums was considered satisfactory. Provision of complete denture was planned. Primary impression was taken in alginate, a cast was formed. Special tray was made from the cast with acrylic to take secondary impression with impression compound.
Base plate was formed from the cast made up of secondary impression. Patient was subsequently recalled to take occlusal vertical dimension, trial and final insertion. Patient was then referred to dermatologist for the treatment of associated skin involvement. Follow up of one year showed good compliance and improved general health. Third molars were still not erupted. Implant retained prosthesis will be given after growth completion along with synthetic bone graft placement
because of severely resorbed maxillary and mandibular alveolar bone with deficient alveolar bone heights.

CASE 2

Extra-oral examination of 13 years old male patient showed normal facial features with hyperkeratosis of palms and soles. Intraoral examination revealed compromised oral hygiene with accumulation of plaque and food debris around the teeth present with marginal gingivitis. Loss of multiple teeth were seen in maxillary and mandibular arch. Exfoliated maxillary teeth were left central incisor, right second premolar

Fig.8: Radiograph showing generalized destruction of alveolar bone and multiple missing teeth in both upper and lower jaw.

and second and third molars on both sides, while exfoliated mandibular teeth were central incisors, lateral incisors, left second premolar and bilateral first and third molars. Multiple tilted/ drifted teeth were also present, although with no pathological mobility. OPG (orthopantomogram) radiograph was advised, which showed impacted mandibular right second premolar. Complete blood picture was advised which showed no findings. On the basis of detailed family history

extra-oral and intra-oral clinical and radiographic evaluation, a diagnosis of PLS was confirmed. Treatment was started with the instructions for the improvement of oral hygiene and 0.12% chlorhexidine gluconate mouth rinse prescription for two weeks.
Patient was then recalled for scaling and root planning. A provision of removable partial dentures was planned for the exfoliated maxillary and mandibular teeth. Acrylic dentures were made for both arches. After insertion of dentures, maintenance of meticulous oral hygiene instructions were was not given to prevent the
accumulation of pathological microbes which aggravate periodontal breakdown and teeth loss. Patient was referred to the dermatology department for the treatment of cutaneous lesions. More frequent recall schedule of 3-6 months was planned for scaling/root planning along with the evaluation of oral hygiene and periodontal health and repeatedly patient and parental counselling for maintenance of vigorous oral hygiene care to delay exfoliation of primary and permanent teeth till growth completion . Delaying shedding of teeth until growth completion helps in the attainment of optimal

alveolar bone height, which negates bone graft surgeries during placement of implant supported prosthesis. A good compliance with improved general health was noticed after one year of follow-up. Implant retained prosthesis will be given after growth completion.

DISCUSSION

The exact etiology and subsequent development of the Papillon lefevere syndrome is still not clear. Multiple factors including immunologic, microbiologic, and genetic appearance to be involved in the causation of PLS. Dysfunction of neutrophils (immunological) appearing as decreased phagocytic and chemotactic activities leading to deficient function of the monocytes, depression of helper/suppressor T cells ratio and decreased lymphocyte response to pathogens are considered to attribute in the development of this syndrome.5
Thus, increased susceptibility to infections is a common finding in this syndrome. The contributing organisms (microbiological) that have been established in PLS are Actinobacillus actinomycetemcomitans, Fusobacterium nucleatum, Porphyromonas gingivalis and Treponema denticola. Various studies have proposed that the mutation of the cathepsin-C gene (genetic) is also associated with the dental and dermatological abnormalities observed in this syndrome.
This genetic mutation is inherited as an autosomal recessive pattern. With both of the parents as carriers of the defective gene, the risk increases up to 25%.8 Consanguineous marriage is also contributory to PLS and considered as the high risk factor, which was observed in our case.11 Hence, familial counselling regarding genetics is very important in our country because of 20-50% of consanguineous marriages of total population.12,13
Dermatological and Periodontal changes basically characterizes Papillon Lefevre Syndrome. Cutaneous lesions include sharply demarcated palmoplantar hyperkeratosis while the periodontal changes includes severe aggressive periodontitis resulting in early loss of primary and permanent dentition, painful gums and multiple periodontal abscesses. Ullbro et al. did not observe any significant correlation between the severity of skin involvement and the severity
of the periodontal infection.9
In the current case all these dental findings and skin lesions were observed and attention has been given to the timely treatment and management of the syndrome for the better outcome in the later life. Other clinical features in PLS includes calcification of the dura mater, tentorium cerebelli, choroids plexus and falx cerebri. Intra-abdominal abscesses (pyogenic liver abscess) have also been reported.7 We could not observe any of these in the current case. Greither’s syndrome and Howel-Evans syndrome, acrodynia, cyclic neutropenia, hypophosphatasia, are the conditions which share similar signs and symptoms with PLS except periodontitis, so they were ruled out.1 Haim-Munk syndrome is an allelic variation of PLS but it shows additional clinical features including arachnodactyly and acro-osteolysis which are not present in PLS.6
The treatment plan necessitates multidisciplinary approach involving dermatologists and dental surgeon team (Pediatric dentists , Periodontists, Prosthodontists). Pediatric dentists should first recognize and timely manage patients with this syndrome for better outcomes in the later life after growth completion. Early diagnosis and prompt dental treatment guarantee better prognosis. For the treatment of dental manifestations, No specific treatment has been proposed but the main aim is to vigorously improve oral hygiene, eliminate the reservoir of micro-organisms and limit the periodontal destruction and allow teeth to be present and delay permanent teeth exfoliation until growth completion and attainment of optimal alveolar bone height. For this purpose, several modalities including conventional periodontal treatment (scaling and root planning); along with extraction of teeth with severe periodontitis and Grade-III mobility, followed by prosthodontic replacement is considered one of the mainstays of treatment. This was the treatment option adopted in our case. Oral hygiene instructions, a prescription of 0.12%
chlorhexidine gluconate mouth rinses and more frequent recalls for professional teeth cleansing and root planning along with one-week systemic antibiotics to control aggressive periodontitis and to prevent bacteremia and pyogenic liver abscesses should also be considered. Early extraction of non-salvageable teeth has also been recommended to prevent further periodontal destruction and bone loss thus allowing protection of a solid base for implantation of artificial dentures.10
Oral retinoids like isotretinoin, acitretin, and etretinate have also been suggested for the treatment of both the dental and the cutaneous defects observed in the PLS7 , but they show many adverse effects and hence not used in our study.
Treatment with retinoids before the eruption of permanent teeth results in a normal dentition, and is more efficacious if continued during the development of permanent teeth. PLS may also adversely affect the self-esteem and general health of children. So, a multidisciplinary approach addressing the social and psychological needs of these children is also necessary. Dental and dermatological evaluation and its early diagnosis and treatment by pediatric dentist along with
parental counseling regarding the genetics are an important part of the management strategy. Maintenance of good periodontal health is imperative in the management of PLS patients. So, a regular follow up of 3-6 months for ultrasonic scaling and evaluation and maintenance of vigorous oral hygiene is necessary to prevent microbial accumulation, periodontal breakdown and early teeth loss. Teeth loss before growth completion results in immature alveolar bone with marked alveolar ridge resorption, needs temporary restorations till adulthood and requires surgical bone grafting for final prosthesis.

CONCLUSION

Pediatric dentists should timely diagnose and manage patients with papillon lefevre syndrome.

CONFLICT OF INTEREST

None declared

REFERENCES

1. Sreeramulu B, Shyam ND, Ajay P, Suman P. Papillon-Lefèvre syndrome: clinical presentation and management options. Clin Cosmet Investig Dent. 2015;7:75-81. https://doi.org/10.2147/CCIDE.S76080
2. Gorlin RJ, Sedano H, Anderson VE. The syndrome of palmarplantar hyperkeratosis and premature periodontal destruction of the teeth. A clinical and genetic analysis of the Papillon-Lefèvre syndrome. J Pediatr. 1964;65:895-08 https://doi.org/10.1016/S0022-3476(64)80014-7
3. Hart TC, Hart PS, Bowden DW, et al, Mutations of the cathepsin C gene are responsible for Papillon-Lefèvre syndrome, J Medi Geneti 1999;36:881-87
4. Alsarheed M, Al-Sehaibany F. Combined orthodontic and periodontic treatment in a child with PapillonLefèvre syndrome. Saudi Med J. 2015;36:987-92 https://doi.org/10.15537/smj.2015.8.11437
5. Singla A, Sheikh S, Jindal SK, Brar R. Papillon Lefevre syndrome: Bridge between dermatologist and dentist. J Clin Exp Dent 2010;2:e43-6 https://doi.org/10.4317/jced.2.e43
6. Tasli L, Kacar N, Erdogan BS, Ergin S. Successful treatment of Papillon Lefevre syndrome with a combination of acitretin and topicalPUVA; a four year follow up. J Turk Acad Dermatol. 2009;3:1-4.
7. Pavankumar K. Papillon-Lefèvre syndrome: A case report. Saudi Dent J 2010;22:95-98 https://doi.org/10.1016/j.sdentj.2010.02.009
8. Hart TC, Shapira L. Papillon-Lefèvre syndrome. Periodontol 2000. 1994;6:88-100. https://doi.org/10.1111/j.1600-0757.1994.tb00029.x
9. Ullbro C, Crossner CG, Nederfors T, Alfadley A, Th estrup-Pedersen K Dermatologic and oral findings in a cohort of 47 patients with Papillon-Lefèvre syndrome. J Am Acad Dermatol 2003;48:345-51. https://doi.org/10.1067/mjd.2003.197
10. Janjua S.A., Khachemoune A. Papillon-Lefevre syndrome: case report and review of the literature. Dermatol. Online J. 2004;10:13
11. Shah AF, Tangade P, Agarwal S. Papillon-Lefevre syndrome: Reporting consanguinity as a risk factor. The Saudi Dent J. 2014;26:126-31.
    https://doi.org/10.1016/j.sdentj.2014.02.004
12. Modell B, Darr A. Science and society: genetic counselling and customary consanguineous marriage. Nat Rev Genet. 2002;3:225-9. https://doi.org/10.1038/nrg754
13. Vardi-Saliternik R, Friedlander Y, Cohen T.Consanguinity in a population sample of Israeli Muslim Arabs, Christian Arabs and Druze. Ann Hum Biol. 2002;29:422-31. https://doi.org/10.1080/03014460110100928

1. MDS (Resident), Department of Pediatric Dentistry, Children Hospital, Pakistan Institute Medical Sciences, Islamabad.
2. House Surgeon, Department of Pediatric Dentistry, Children Hospital, Pakistan Institute Medical Sciences, Islamabad.
3.  Dean of Dentistry, Head of Department, Department of Operative and Pediatric Dentistry, Pakistan Institute Medical Sciences, Islamabad.
4. House Surgeon, Department of Prosthodonti, Armed Institute of Dentistry, Rawalpindi.
5. Registrar, Department of Operative Dentistry, University College of Dentistry, University of Lahore.
6. FCPS (Resident) Department of Orthodontics, Sardar Begum Dental College, Peshawar.
7. MDS (Resident), Department of Pediatric Dentistry, Children Hospital, Pakistan Institute Medical Sciences, Islamabad.
   Corresponding author: “Dr. Abul Khair Zalan” < zalanjan@yahoo.com >

Abul Khair Zalan                                   BDS

Khadeejah Khalil Zubairy                    BDS

Anser Maxood                                       BDS, FRACDS

Manahil Niazi                                          BDS

Hira Zaman                                             BDS, MDS

Anika Gul                                                BDS

Miraat Anser                                           BDS

Papillon-Lefèvre syndrome (PLS) an autosomal recessive disorder characterized by diffuse transgradient palmar-plantar hyperkeratosis, with premature loss of deciduous and permanent teeth, along with the calcification of the dura mater. This results in teeth radiographically appearing as “floating” in the soft tissue. Genetic studies of patients with PLS have mapped the major gene locus to chromosome 11q24-q21 and revealed mutation and loss of function of the cathepsin gene. It affects 1- 4 people per 1 million population with no gender or racial predilection. Dermatological manifestations, usually occurs before four years of age, include hyperkeratosis of palms and soles, nail dystrophy, hyperhidrosis and keratinization on elbows and knees with the lesions appearing as white, yellow-like or red plaques or patches that then develop cracks, crusts, or deep fissures.
KEYWORD: Papillon-lefevre syndrome, hyperkeratosis
HOW TO CITE: Zalan AK, Zubairy KK, Maxood A, Niazi M, Zaman H, Gul A, Anser M. Papillon lefevre syndrome: Diagnosis and management in two affected siblings – A case report. J Pak Dent Assoc 2020;29(4):264-268.
DOI: https://doi.org/10.25301/JPDA.294.264
Received: 23 May 2020, Accepted: 24 August 2020
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Iftekhar Ahmed                       MBBS, MCPS, FCPS

Anwar Ali                                  BDS, FDSRCS

Marium Zaheer                       BDS, MDS

Ibraj Fatima

Nazeer Khan                      PhD

INTRODUCTION

Thar is the largest desert of Pakistan and seventh largest in the world.1 This desert has the blessing of being the only fertile desert in the world where the rainfall brings the dead land to life. That is the reason why people live not only in the outskirts of desert but also in the desert area.2 This semi-arid area seems not completely xeric because of the average rainfall of 200-300 mm which is usually erratic and non-consistent.3 The natural disaster is not always the drought but the poluuted water when available contains lots of impurities and sediments.4 This leads the human growth and development towards imbalanced proportions in majority of aspects of health. Thar desert is 120 meter elevated from sea level. The geographical proximity of the Great Rann of Kutch (the largest salt marsh) and
Arabian Sea in the South of Thar Desert render the underground water of Thar a marine environment.5 Furthermore, the alluvial plains of Indus River and the coast
make the underground water more salty.6 The volcano in the region of Rajisthan may be the source. Thar is a tropical area with maximum temperature as high as 48°C1 thus giving the clue of presence of coal beneath its burning lands.
Thar is the largest district of Sind.4 It has a total population of 1.65 million scattered over an area of 19,638 km2. Water, the most treasurable natural resource that is gifted to human beings, is under constantly increasing intimidation in this area. Therefore, inadequate access to safe drinking water has become the most critical and the most challenging environmental problem. Throughout history, people of this region have used groundwater as a source of drinking water and not only them, more than half of the world’s population depends on it for existence. Groundwater constitutes 97% of global freshwater and in many regions, groundwater springs are the single principal source for serving drinking water.7 Thus the accessibility of uncontaminated groundwater is most vital, as it serves as the critical component in every sphere of human life. Scarcity of drinking water and moreover the increased mineral and ion concentration in water are crucial factors in keeping the health of Thar people blow the level of satisfaction. For example the higher concentration of nitrate in groundwater is hazardous, predominantly for newborn babies affected by ‘blue baby syndrome.8 The local population pulls the underground water from wells for their drinking, cooking and everyday utilization1. The wells can be as deep as 3-90 m and pulling of water is aided by help of camels.
One of the most crippling biochemical diseases that emerge as a result of imbalanced ion concentration in water is fluorosis.9 That is caused by excess intake of fluoride ion. Along with many minerals present in water, one of the most important ions is fluoride ion.9
Fluoride is the ion of fluorine, the 17th most plentiful and most electronegative element on ecosphere. It is highly reactive that’s why it is found in combination of other elements such as calcium fluoride or fluorspar. Rocks and soil are the main reservoir of fluoride that leaches out in the underground water. Drinking water,
food, tea, air, medicaments and cosmetics are the sources of fluoride exposure in human beings. The drinking water or fresh water fluoride level (0.01-0.3 parts per million (ppm) is different from sea water fluoride level (1.2-1.5 ppm). Concentration of fluoride in water depends upon the temperature that is very high in Thar Desert and sedimentary rocks, lime stone, sand stones and volcano rocks present in this area.10
Fluorosis affect many systems in body primarily teeth and bones.11 The other systems include nervous system, reproductive system, liver, kidneys and cardiovascular system. Long term intake of fluoride during tooth development leads to enamel fluorosis. There is a linear relationship in dose and response. The recommended fluoride content in drinking water should not be more than 1 parts per million (ppm).12 It is of interest that the fluorosis occurs due to local effects
of fluoride on the mineralizing tooth germ, predominantly the maturation stage of enamel formation that is up to the age of 8 years.12 Fluorosis clinical spectrum ranges from mild discoloration of tooth surface to severe staining and pitting.12 There have been various studies mentioning the excessive level of fluoride preset in the drinking water of Thar district.1,13-16 The native individuals are at high risk of developing dental fluorosis. However, the literature search reveals scarcity of data on dental fluorosis.17,18 prevalence and its characteristics in Thar district. Therefore we design a study to evaluate the fluoride estimation in drinking water
and geometrical samples and to investigate the dental changes associated with increased fluoride intake that is the dental fluorosis in Thar.

METHODOLOGY

This was a descriptive comparative cross-sectional study. Ethical approval was obtained by the Institutional Review Board of Dow University of Health Sciences (Ph-D-06/ERB50/DUHS-08. The study was approved by Institutional Review Board, Dow University of Health Sciences. The study was conducted for dental florosis and sample size was calculated from WHO sample size calculator accordingly.
A total of 242 participants were included 121 in each group. List of residents was made with the help of map of houses. The study site for exposed was the village Sammo Rind that is situated 40 km in the South East of Chachro Town in Thar District. The unexposed group comprised of age and sex matched individuals from three villages (Pathan Goth, Screane Goth and Bi Khori Goth) of Gadap Town. Gadap Town is situated in North West Karachi along the Hub River at the provincial border between Sindh and Baluchistan. Using systematic random sampling every fifteenth resident was recruited and consent was taken. If consent was refused then next fifteenth resident was called upon. Likewise for the control gunexposed group, one out of thirty residents (age and sex matched) of three villages of Gadap Town was included. The study subjects’ of age of 12 years and above were brought to Karachi from the case and control sites. Both male and female subjects, living in the area since birth were included. The only exclusion criterion was the immigrants of other areas. Complete history with thorough clinical examination in proper clinical settings were conducted in a tertiary care hospital in Karachi. Oral cavity was examined by a senior dentist in dental unit Enamel abnormalities were recorded according to Dean HT classification (Table 2) of mottled enamel diagnosis and Thylstrup & Fejerskov Index (TF index) (Table 3). Moreover photographs were also taken. Findings were recorded in printed Performa. Samples of underground water were taken from study and unexposed group sites. For fluoride estimation in water and geometrical (rock, soil, clay etc) samples, Fluoride Ion Selective Electrode (FISE) method was applied. The total Ionic Strength Adjustment Buffer (TISAB) was used for pH and fragmentation of aluminum fluoride complexes which might otherwise lessen the fluoride activity.

RESULT

Data Analysis
A data base was developed on SPSS for windows version 22.0. Mean and standard deviation were calculated for quantitative variables like age, fluoride level etc.. A frequency with percentages was presented for qualitative variables like gender, fluorosis etc. Chi square test was applied and P-values <0.05 was taken as statistically significant.

Demographic Features
Of total 242 subjects, there were 121 exposed subjects and 121 unexposed subject. The mean age in exposed was 33.82± 14.10 years while the mean age in unexposed was found to be 33.33±11.67 years (Table-4). The mean difference

Table 1: Changes according to Deans index of the Cases and Controls

Table 2: Dean’s Index Classification

was insignificant (p=0.7686), Moreover, the total number of males in exposed subject was 90 while the total number of males in unexposed subjects was found to be 84, which constitute about 74.4% and 69.4% respectively. Majority of the exposed subjects (n=88; 72.7%) and unexposed (n=96; 79.3%) were married (Table-5).While the occupation of most of the exposed (n=49; 40.5%) and unexposed subject (n=43; 35.5%) were farming. However, responsibility of majority females were taking care of household constituting 25.6% (n=31) and 26.2% (n=32) of cases and controls respectively (Table-6).

Table 2a: Changes according to T.F Smooth Surfaces index

Table 2b: Changes according to T.F Occlusal Surfaces index

Table 3: Clinical Criteria and Scoring for the TFI (Thylstrup-Fejerskov Index)

Dental Changes
Out of 121 study subjects, all 121 subjects were found to have dental fluorosis that is 100%. In contrast, 82.6% in control area had normal teeth. According to Dean Index;

Dental fluorosis in a 14 years old boy

mild, moderate and severe changes were seen in 18 (14.9%), 69(57%) and 23(19%) exposed subject respectively (Table 1). Ch isqaure test was applied and P value found to be < 0.05.Given the p value <0.05 there is significant association between exposure to water having high flouride content and characterstics of enamel according to Dean index of classification. Moreover, evaluation according to T.F index grade for smooth surface index in 2, 3, 4, 5, 6, 7 and 8 changes were seen in 5(4.1%), 10(8.3%), 25(20.7%), 41(33.90%), 23(19%), 12(9.9%) and 4(3.3%), respectively with p value < 0.05 (Table 7a). TF scores for occlusal surface index for the same level were shown in table 2 b.Both T.F smooth surface index and occlusal surface index have statistically significant association with exposure group as
per given p value of <0.05.

Water fluoride estimation
The well water consumed by the population of Sammo Rind has been analyzed for its fluoride content and was found to be 6-8 mg/dl, as compared to control area where fluoride levels are found to be 0.30 mg/L. World Health Organization (WHO) recommends fluoride concentration level in drinking water at 1.5mg/L or it can be documented as 0.8-0.9 ppm. The remaining trace elements in this area were normal.

DISCUSSION

Pakistan is one among the countries where fluorosis is endemic. Thar Desert areas of Sindh province are among the affected areas. The groundwater, especially the water in unconfined aquifers, is the main source of drinking water for people living in the rural desert areas of Sindh. Groundwater is mostly available in dug-wells, generally at the depth of 30 to 75 meters, which is brackish and highly contaminated by the presence of fluoride. A high prevalence of fluorosis is present in such areas including dental, skeletal, musculoskeletal and other forms of fluorosis. There are numerous studies on the increased levels of fluoride in the ground water of Thar Desert. Many studies were conducted to find the prevalence of dental fluorosis in different parts of the country but there is a dearth of data on dental fluorosis in Thar Desert. Studies from Lahore, MianWali and Murdan and Quetta reported the prevalence of dental fluorosis.19-22 Study by Rizwan et al in 2012 in Lahore dental Hospital, patients coming for dental problems were examined and the frequency of dental fluorosis was found to be 12 %.20 This result is much lower than the result of our study, may be due to level of fluoride ion in drinking water is not as high as in Thar area. Moreover, in a tertiary care hospital patients come from near and far, not confining to a particular locality. Another reason of this vast difference may the fact that rural population usually live their whole lives in same place for decades while urban life is not so static, people move from one area to another area earlier as compared to rural life, hence changing the water for drinking.
Moreover, the source of water is not always ground water in urban life style. However, some portion of population get affected by ground water but other sources of fluoride for example tea, food, supplements, fluoride containing tooth pastes, mouth washes and beverages may contribute to develop fluorosis in urban population. Another study from Quetta by Sami et al reported the prevalence of dental fluorosis as 63.3% in school going children of Quetta, most of it was of moderate type.21 The prevalence rate is much higher in this study; the higher fluoride level in the region may be the cause. The prevalence found in Quetta school children was found to be close to our study result. A study conducted in MianWali and Murdan stated the prevalence of dental fluorosis as 98%, this result is in approximation with our result.19 On more study with this outcome in children of 12 year of age.23 As we examined the individuals not only from the Sammo Rind village but also the unexposed group of Gadap town of Karachi, the result showed that in spite of having similarity in eating style and life style of both groups of same ethnicity, the difference in the prevalence of dental fluorosis is due to the level of ground water fluoride ion concentration. Dental fluorosis was seen in 100% of subjects
and only in 17.4 % in unexposed group. Mild, moderate and severe changes were seen in 18 (14.9%), 69(57%) and 23(19%) exposed subjects according to Dean Index of grading system of dental fluorosis respectively wheareas the severity of flourosis in unexposed group was 0.8% only the majority of the positive patient in unexposed group fall into the category of either questionable (6.6%) or very mild (5.8%)The well water of Sammo Rind fluoride content was found to be 6-8 mg/dL, as compared to control area where fluoride levels are found to be 0.30 mg/L.

CONCLUSION

Fluorosis forays the Thar Desert so hard that no person had been found to have normal healthy teeth. The frequency of dental flourosis is significantly high in the area where flouride level in drinking water is higher than the recommended value when compared with the population drinking water with normal flouride.

REFERENCE

1. Rafique T, Naseem S, Bhanger MI, Usmani TH. Fluoride ion contamination in the groundwater of Mithi sub-district, the Thar Desert, Pakistan. Environmental Geology. 2008;56:317-26. https://doi.org/10.1007/s00254-007-1167-y
2. Rahmani AR, Soni R. Avifaunal changes in the Indian Thar desert. Journal of arid environments. 1997;36:687-703. https://doi.org/10.1006/jare.1996.0242
3. Lau WK, Kim KM. Fingerprinting the impacts of aerosols on longterm trends of the Indian summer monsoon regional rainfall. Geophysical Research Letters. 2010;37(16). https://doi.org/10.1029/2010GL043255
4. Bilal M, Liaqat MU, Cheema MJM, Mahmood T, Khan Q, editors. Spatial drought monitoring in Thar desert using satellite-based drought indices and geo informatics techniques. Multidisciplinary Digital Publishing Institute Proceedings; 2017. https://doi.org/10.3390/ecws-2-04948
5. Bhatt HB, Gohel SD, Singh SP. Phylogeny, novel bacterial lineage and enzymatic potential of haloalkaliphilic bacteria from the saline coastal desert of Little Rann of Kutch, Gujarat, India. 3 Biotech. 2018;8(1):53.https://doi.org/10.1007/s13205-017-1075-0
6. Edmunds WM, Smedley PL. Fluoride in natural waters. Essentials of medical geology: Springer; 2013. p. 311-36. https://doi.org/10.1007/978-94-007-4375-5_13
7. Gleick PH, Cain NL. The world’s water 2004-2005: the biennial report on freshwater resources: Island Press; 2004.
8. Soomro F, Rafique T, Michalski G, Ali SA, Naseem S, Khan MU. Occurrence and delineation of high nitrate contamination in the groundwater of Mithi sub-district, Thar Desert, Pakistan. Environmental earth sciences. 2017;76(10):355. https://doi.org/10.1007/s12665-017-6663-0
9. Sergio R-VJ, Luilli LC, Carlos R-LJ. Fluoride: its implications for public health. 2018.
10. Khatri G, Bahura C. Flouride in ground water sources of sardarshahar city of churu District in the thar desert of rajasthan. 2017. https://doi.org/10.20959/wjpr201710-9422
11. Simon MJ, Beil FT, Riedel C, Lau G, Tomsia A, Zimmermann EA, et al. Deterioration of teeth and alveolar bone loss due to chronic environmental high-level fluoride and low calcium exposure. Clinical oral investigations. 2016;20:2361-70. https://doi.org/10.1007/s00784-016-1727-1
12. Goodarzi F, Mahvi AH, Hosseini M, Nodehi RN, Kharazifard MJ, Parvizishad M. Fluoride concentration of drinking water and dental fluorosis: A systematic review and meta-analysis in Iran. Dental Hypotheses. 2016;7(3):81. https://doi.org/10.4103/2155-8213.190482
13. Husain V, Nizam H, Arain GM. Arsenic and fluoride mobilization mechanism in groundwater of Indus Delta and Thar Desert, Sindh, Pakistan. Int J Econ Env Geol. 2012;3:15-23.
14. Choubisa SL. Fluoride distribution in drinking groundwater in Rajasthan, India. Curr Sci. 2018;114:1851-57. https://doi.org/10.18520/cs/v114/i09/1851-1857
15. Rafique T, Naseem S, Ozsvath D, Hussain R, Bhanger MI, Usmani TH. Geochemical controls of high fluoride groundwater in Umarkot sub-district, Thar Desert, Pakistan. Science of the Total Environment. 2015;530:271-78. https://doi.org/10.1016/j.scitotenv.2015.05.038
16. Brahman KD, Kazi TG, Afridi HI, Naseem S, Arain SS, Ullah N. Evaluation of high levels of fluoride, arsenic species and other physicochemical parameters in underground water of two sub districts of Tharparkar, Pakistan: a multivariate study. Water research. 2013;47:1005-20. https://doi.org/10.1016/j.watres.2012.10.042
17. Rafique T, Ahmed I, Soomro F, Khan MH, Shirin K. Fluoride Levels in Urine, Blood Plasma and Serum of People Living in an Endemic Fluorosis Area in the Thar Desert, Pakistan. Journal of the Chemical Society of Pakistan. 2015;37(6).
18. Ahmed I, Sohail S, Hussain M, Khan N, Khan MH. MRI features of spinal Fluorosis: Results of an endemic community screening. Pakistan J Med Sci. 2013;29(1):177. https://doi.org/10.12669/pjms.291.3200
19. Siddiq K, Dost S, Naseem A, Arshad A, Ullah A. Prevalence of Dental Fluorosis in Mianwali and Mardan Districts. Journal of Cosmetics, Dermatological Sciences and Applications. 2011;1(03):106. https://doi.org/10.4236/jcdsa.2011.13016
20. Rizwan S, Rizwan M, Naveed A, Ahsan W. Incidence of dental fluorosis among the Patients visiting the university of Lahore Dental College/Hospital-A study. Pakistan Oral & Dental Journal. 2010;30(1).
21. Sami E, Vichayanrat T, Satitvipawee P. Caries with Dental Fluorosis and Oral Health Behaviour Among 12-Year School Children in Moderate-Fluoride Drinking Water Community in Quetta, Pakistan. Pakistan Journal of the College of Physicians and Surgeons Pakistan. 2016;26:744-47.
22. Tahir M, Rasheed H. Fluoride in the drinking water of Pakistan and the possible risk of crippling fluorosis. Drinking Water Engineering and Science. 2012;5:495-514. https://doi.org/10.5194/dwesd-5-495-2012
23. Khan AA, Whelton H, O’Mullane D. Is the fluoride level in drinking water a gold standard for the control of dental caries? Int Dent J. 2004;54:256-60. https://doi.org/10.1111/j.1875-595X.2004.tb00290.x

1. Professor, Department of General Medicine, D.I.M.C, Dow University of Health Sciences, Karachi.
2. Professor, Department of Ora Sugery, Dr Ishrat ul Ebad Khan Institute of Oral Health Sciences Dow University of Health Sciences, Karachi.
3. Assistant Professor, Department of Oral Pathology, Dr Ishrat ul Ebad Khan Institute of Oral Health Sciences Dow University of Health Sciences, Karachi.
4. MBBS (student) Undergraduate Student, Department of General Medicine, D.I.M.C, Dow University of Health Sciences, Karachi.
5. Professor, Department of Biostatistics, Jinnah Sindh Medical University.
   Corresponding author: “Dr. Iftekhar Ahmed” < dr_iftekharahmed@hotmail.com >

Iftekhar Ahmed                       MBBS, MCPS, FCPS

Anwar Ali                                   BDS, FDSRCS

Marium Zaheer                       BDS, MDS

Ibraj Fatima

Nazeer Khan                       PhD

OBJECTIVE: The objective of this study is to see the frequency of dental fluorosis in exposed area (Sammo Rind village of Thar District) and in unexposed area of Gadap town of Karachi and to find the association of fluoride level with dental fluorosis in both groups.
METHODOLOGY: The water samples were taken from both the sites and fluoride estimation in water and geometrical, was determined using Fluoride Ion Selective Electrode (FISE) method. By random selection 121 subjects from Sammo Rind village and 121 controls from Gadap Town Karachi were included in study after informed consent. The participants were clinically examined by an expert university teacher dentist in proper dental setups in examination lights.
RESULTS: A frequency of dental fluorosis of 100% was found among the study subjects with fluoride content as high as 6- 8 mg/dl as compared to prevalence of dental fluorosis 17.4% in unexposed group with water fluoride content as low as 0.30mg/dl.
CONCLUSIONS: The high level of underground water fluoride level and cent per cent dental fluorosis in Thar area is an alarming situation that should be addressed immediately at national level.
KEYWORDS: Exposed area, Dental Flurosis, Fluoride Level, Water Sample
HOW TO CITE: Ahmed I, Ali A, Zaheer M, Fatima I, Khan N. Frequency of dental fluorosis in population drinking water with high fluoride level in Thar. J Pak Dent Assoc 2020;29(4):259-263.
DOI: https://doi.org/10.25301/JPDA.294.259
Received: 19 June 2020, Accepted: 13 August 2020
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Saad Abdul Rehman                        BDS

Sadia Rizwan                                      BDS, FCPS

Syed Shah Faisal                               BDS, FCPS

Syed Sheeraz Hussain                     BDS, DCPS, MCPS, FCPS

INTRODUCTION

Curvature of occlusion from lateral view is a natural phenomenon in the dentition. In 1890, a researcher named Spee found it in human’s dentition, identified and drawn a line of occlusion which was termed as curve of spee. He utilized skulls with rubbed teeth to characterize this line.1 It is an assumed or visionary line, which is drawn and passes through the posterior teeth’s buccal cusp tips, follows the plane of occlusion. In the maxillary arch, usually this curve is convex and concave in the mandibular dentition.2 It is impossible to treat orthodontic cases without proper diagnosis and it requires multiple records in the form of extra oral pictures, intraoral pictures, radiographs which include Lateral cephalogram and panoramic radiograph and dental casts. Usually curve of spee is reduced and insignificant in the deciduous dentition, it increments to most extreme depth when second molar erupts in the oral cavity and come into the occlusion. Afterward it remained moderately stable till late youth and early adulthood.4The arrangement of the teeth along this line, connected to a few anatomical and useful components: joint tendency, overjet, molar cusp, and the quality and quantity of contacts.5
Andrew suggested that this curve should be flat to slightly bended. According to him it is one of the principle characteristic of normal occlusion. He also said that, there is always an expected propensity for this curve to exaggerate and deepen as time passes, and the reason behind it is that the mandible grows more and faster than the maxilla which causes the mandibular anterior dentition to be bound by the upper lip and anterior dentition.3
It leads to increase upward and backward forces, resulting in increased curve of spee and deepen the bite. It has been observed that those patients who has short face, will have exaggerated curve of spee depth. Flattening or over correction of this curve is one of the key objective in the treatment. Now a days, it is a very common practice to level and flatten this curve at the end of treatment. Few researches have been published regarding this curve, which evaluated the factors and conditions associated with increased curve of spee.6
There are multiple factors which effect this curve including dental and skeletal factors, especially vertical axis of the skeletal factors effect this curve.7 The cephalometric analysis helps to evaluate the face, cranium, cranial base, maxilla, mandible, dentition and alveolar process of maxilla and mandible in horizontal as well as vertical axis. The vertical relationship of these structures is as important as the horizontal and transverse relationships because it effects the treatment plan as well as the stability of the treatment.8
Vertical facial pattern of a face can be determined by number of linear and angular measurements. One of the important variable which is used to measure the pattern of facial divergence and vertical skeletal pattern is SNMP angle. It is usually formed by taking two horizontal lines which intersect each other to make an angle, those are SN line (Sella-Nasion) and Mandibular plane.9 On the basis of this angle, vertical skeletal pattern divided in to Hyperdivergent, Normodivergent and Hypodivergent cases. High angle case presents with hyperdivergent facial type, low angle case present with hypodivergent case and normal angle case present with normodivergent facial type.10 The key objectives of the study were to determine the curve spee depth in three different skeletal vertical patterns (Hypodivergent, Normodivergent and Hyperdivergent) and find out their association. As curve of spee correction is one of the goal of orthodontic treatment, this study will provide information regarding variation in curve of spee depth in different facial divergences which will definitely help orthodontists in treatment planning.

METHODOLOGY

Non Probability Consecutive method was used for sampling. By using WHO sample size calculator, taking statistics of curve of spee as 2.4052±0.679mm in margin of error 0.2, the calculated sample size should be atleast 45.20 Lateral cephalogram and Orthodontic models of mandibular arch of 110 patients with the mean age of 17.16±4 years, 26.4% (N=29) males with the mean age of 17.94±4.19 years and 73.6% (N=81) females with the mean age of 16.88±3.94 years, were obtained from the patient which had been visited to the Orthodontic’s Department. The ethical approval was obtained from the the institute (065/19).

The participants met the following criteria:

Inclusion criteria includes male and female patients patients with second molars erupted. Exclusion criteria include individuals having missing or impacted permanent canines/any teeth other than 3rd molars , any tooth anomaly of number, size, form and position as confirmed on clinical examination, having history of previous orthodontic treatment, any known systemic disease, history of facial trauma, cleft lip and Palate and history of surgical procedure of jaws. All the patients were examined by the researchers. Skeletal divergence defined by the mandibular plane (Lower border of mandible) to sella-nasion line angle (SN-MP). It was measured on lateral cephalogram (Fig I) and subjects were categorized.

Fig I: SN-MP Angle measurement on lateral cephalogram

Fig II: Measurement of depth of curve on mandibular cast

1. Hyperdivergent: If candidate having SNMP angle above 36 degree
2. Hypodivergent: If candidate having SNMP angle below 28 degree
3. Normodivergent: If candidate having SNMP angle between 28 to 36 degree

The depth of curve of Spee has been measured in such a way that a 6 inches stainless steel scale was placed on the occlusal surface of the dental cast of lower jaw which was making a tangent to the disto-buccal cusp tip of second molar to the incisal edges of the central incisors (Fig.II). It has been measured with the help of digital Vernier caliper. The measurements of right and left sides were noted and their mean was calculated.

STATISTICAL ANALYSIS

IBM SPSS Statistics 20th version used to analyze the data. Shapiro-Wilk’s test (p-value >0.05) showed that the curve of spee values were normally distributed for both males and females. The statistical analyses involved calculation of means and standard deviations of quantitative variables like age, curve of spee and frequencies & percentages of qualitative variables like sex and groups of vertical skeletal patterns / facial divergence. Intra-Class Correlation Coefficient has been used to check level of agreement between the measured and re-measured values by a researcher. One Way ANOVA test has been used to fulfill the objective.

RESULTS

The mean value of average depth curve of spee among all the patients was 2.48±0.65mm, the mean value of average depth of curve of spee in male patients was 2.48±0.55mm and in female it was nearly same as in male, 2.48±0.68mm. Out of 110 patients 20.9% (N= 23) were hypodivergent, 29.1% (N=32) were normodivergent and 50% (N=55) were hyperdivergent. The mean values of average depth of curve of spee among these three groups were 3.39±0.30mm, 2.62±0.23mm and 2.02±0.45mm (Table I). One-way ANOVA has been applied and it showed highly significant differences

Table I: Mean values of average depth of curve of spee among three different groups

in the values of curve of spee depth in different vertical skeletal patterns with the p-value of 0.000. Moving from

Table II: Difference between the groups

*The mean difference is significant at the 0.05 level.

Hypodivergent to Hyperdivergent cases, the curve of Spee depth reduces.

DISCUSSION

The assessment of the association of curve of Spee depth with the vertical skeletal variables was needed to study which leads to the alteration and deviation in the depth of the curve. It has been proven in the literature that the anterio-posterior and vertical position of the mandible are co-related to the curve of Spee depth in lower jaw. All the above mentioned variables are directly related to the treatment planning in orthodontics as well as treatment stability. Position and inclination of lower incisors are related to the curve of spee and vertical skeletal patterns of the subjects.11 Cheon et al conducted a study and found that there curve of Spee depth in lower jaw failed to show sexual dimorphism. He also found the significant correlation coefficients between curve of Spee depth in lower jaw and facial angle that is an indicator of mandible’s position in sagittal dimension which also effects the vertical facial patterns.12
Farella et al13 found that there is a significant influence of anterior and posterior facial height ratio on the depth of Spee’s curve in lower jaw and this ratio plays a vital role in the vertical skeletal facial patterns. In this study, it has been established that the curve is relatively exageratted in low angles (Hypodivergent) subjects as compare to the high angle (Hyperdivergent). The finding of lack of sexual dimorphism in curve of spee depth is in agreement with our findings. A possible reason and justification for the change in that curve among different facial divergent cases can be attributed to the occlusal forces exerted by the subjects, with the hypodivergent subjects exhibit comparatively higher occlusal forces.14,15
Patient with the increased SNMP values tend to have less over bite as compare to those with the reduced SNMP value. Batham et al16 studied that the subjects with deep bite had exaggerated curve of spee depth in the lower arch and reason behind it can be that when anterior teeth does not have vertical stop which leads to the continued eruption. The continued eruption causes the anterior aspect of the curve to deepen. Open bite cases usually associated with hyperdivergent skeletal pattern and deepbite cases usually come up with hypodivergent skeletal pattern. More or less same findings were observed by Trauten et al17 which showed that this curve is minimal in cases with open bite or open bite tendency and exaggerated in subjects with increased over bite. Trauten concluded that a reduced curve of Spee
seen in the hyper divergent subjects and exagerrated depth was observed in hypodivergent subjects. The results of Trauten and Batham were also supported by multiple studies done by Nanda, Shannon and Orthlieb that worked on the same variables as we did in this study.7,18 Furthermore Kumari et al19 found a negative correlation between the SNMP value and curve of Spee in lower arch, supporting our study in other way that cases with increased SNMP angle will have reduced curve of spee.
One more study has been done by Halimi et al5 and its results were not in favor with our study. They conducted study on 90 patients which were divided into three groups as Hypodivergent, Normodivergent, and Hyperdivergent that there is no significant difference in the curve of Spee among these groups. The target population of the present study was Orthodontic Patients, therefore, it is not easy to extrapolate the results on the general population. It is highly recommended
that further studies should be done with broad and large number of sample size with non-orthodontic cases so it becomes easy to apply the results on general population and other variables must be used for the diagnosis of skeletal pattern like FHMP (Frankfort Horizontal Mandibular Plane Angle) and y-axis angle.

CONCLUSION

There was a significant difference in the depth of curve of spee among hypodivergent, normodivergent and hyperdivergent patients.

CONFLICT OF INTEREST

None declared

REFERENCES

1. Sayar G, Oktay H. Assessment of curve of Spee in different malocclusions. Eur Oral Res. 2018;52:127. https://doi.org/10.26650/eor.2018.475
2. Marshall SD, Kruger K, Franciscus RG, Southard TE. Development of the mandibular curve of Spee and maxillary compensating curve: A finite element model. PloS one. 2019;14(5). https://doi.org/10.1371/journal.pone.0221137
3. Andrews LF. The six keys to normal occlusion. Am J Orthod. 1972;62:296-309. https://doi.org/10.1016/S0002-9416(72)90268-0
4. Al-Amiri HJ, Al-Dabagh DJ. Evaluation of the relationship between curve of Spee and dentofacial morphology in different skeletal patterns. J Baghdad Coll Dentis. 2015;325(2218):1-1. https://doi.org/10.12816/0015282
5. Halimi A, Benyahia H, Azeroual MF, Bahije L, Zaoui F. Relationships between facial divergence and DMD parameters. International orthodontics. 2017;15:698-707. https://doi.org/10.1016/j.ortho.2017.09.017
6. Nazruddin N, Tan YY. Evaluation of the Depth of the Curve of Spee, Overjet, and Overbite in Class I, Class II, and Class III Malocclusion Among Patients at University of North Sumatera Dental Hospital. In11th International Dentis Sci Meet (IDSM 2017) 2018 May. Atlantis Press. https://doi.org/10.2991/idsm-17.2018.27
7. Orthlieb JD. The curve of Spee: understanding the sagittal organization of mandibular teeth. Cranio 1997; 15: 333-340. https://doi.org/10.1080/08869634.1997.11746028
8. Livas C, Delli K, Spijkervet FKL, Vissink A, Dijkstra PU. Concurrent validity and reliability of cephalometric analysis using smartphone apps and computer software. Angle Orthod. 2019;89:6:889-96 https://doi.org/10.2319/021919-124.1
9. Khan WA, Faisal SS, Hussain SS. Correlation of Craniofacial Measurements between Cephalometric Radiographs and Facial Photographs. AnnalS Abbasi Shaheed Hosp Kar Medi Dent Coll. 2018;23:37-45.
10. Kakadiya JK, Kambalyal P, Singla M, Jingar J, Vishnoi P. Comparison of Incisor, Molar & Lower Anterior Facial Divergence in Hypodivergent, Hyperdivergent And Normodivergent Patient: A Study Modeland Cephalometric Study. Dent J Advan Stud. 2016;4:104-12. https://doi.org/10.1055/s-0038-1672054
11. Laird MF, Holton NE, Scott JE, Franciscus RG, Marshall SD, Southard TE. Spatial determinants of the mandibular curve of Spee in modern and archaic Homo. Am J Phys Anthropol. 2016;161: 226-36. https://doi.org/10.1002/ajpa.23020
12. Cheon SH, Park YH, Paik KS, Ahn SJ, Hayashi K, Yi WJ, Lee SP. Relationship between the curve of Spee and dentofacial morphology evaluated with a 3-dimensional reconstruction method in Korean adults. Am J Orthodont Dentofac Orthopedics. 2008;133:640-e7. https://doi.org/10.1016/j.ajodo.2007.11.020
13. Rozzi M, Mucedero M, Pezzuto C, Cozza P. Leveling the curve of Spee with continuous archwire appliances in different vertical skeletal patterns: A retrospective study. Am J Orthodont Dentofac Orthopedics. 2017;151:758-66. https://doi.org/10.1016/j.ajodo.2016.09.023
14. Proffit WR,Fields HW,Nixon WL. Occlusal forces in nor-maland long-face adults.J Dent Res 1981; 62: 566-571. https://doi.org/10.1177/00220345830620051201
15. Amjad N, Mahmood A, Masood RT, Nazir R. COMPARISON OF INCISORS INCLINATION IN HYPERDIVERGENT AND HYPODIVERGENT PATIENTS. Pak Oral Dent J. 2019;39:125-8.
16. Batham PR, Tandon P, Sharma VP, Singh A. Curve of Spee and its relationship with dentoskeletal morphology. J Indian Orthodo Soci. 2013;47:128-34. https://doi.org/10.1177/0974909820130303
17. Trouten JC, Enlow DH, Rabine M, Phelps AE, Swedlon D. Morphologic factors in openbite and deepbite. Angle Orthod 1983;53:192-211.
18. Shannon KR, Nanda R. Changes in the curve of Spee with treatment and at years post-treatment. Am J Orthod Dentofacial Orthop 2004;125:589-96. https://doi.org/10.1016/j.ajodo.2003.09.027
19. Kumari N, Fida M, Shaikh A. Exploration of variations in positions of upper and lower incisors, over jet, overbite, and irregularity index in orthodontic patients with dissimilar depths of curve of Spee. J Ay ub Med Coll. 2016;28:7 66-72.
20. Imai H, Yakushiji M, Shintani S. Longitudinal observation of the changes of the consecutive curve of the incisal edge, tip and cusp tip from deciduous and permanent dentition-Comparison between normal and crowded dental arch. Pediatric Pediatr Dent 2010;20:130-51. https://doi.org/10.1016/S0917-2394(10)70205-6

1.  FCPS II Resident, Department of Orthodontics, Karachi Medical and Dental College.
2. Assistant Professor, Department of Orthodontics, Dow University of Health Sciences.
3. Professor, Department of Orthodontics, Karachi Medical and Dental College.
4. Principal College of Dentistry, Professor & HOD Deparment of Orthodontics, Karachi Medical & Dental College.
   Corresponding author: “Dr. Saad Abdul Rehman” <saadmemon_786@hotmail.com>

Saad Abdul Rehman                        BDS

Sadia Rizwan                                      BDS, FCPS

Syed Shah Faisal                               BDS, FCPS

Syed Sheeraz Hussain                     BDS, DCPS, MCPS, FCPS

OBJECTIVE: To find the mean curve spee depth and association in three different skeletal vertical patterns (Hypodivergent, Normodivergent and Hyperdivergent).
METHODOLOGY: Orthodontic models of mandibular arch and Lateral cephalogram of 110 patients with the mean age of 17.16±4 years, 26.4% (N=29) males and 73.6% (N=81) females were taken from the patients who visited for the orthodontic treatment, to the Department of Orthodontics of Karachi Medical and Dental College. Skeletal divergence defined by the mandibular plane (Lower border of mandible) to sella-nasion line angle (SN-MP). It was measured on lateral cephalogram and Curve of spee depth measured on mandibular cast.
RESULTS:Out of 110 patients 20.9% (N= 23) were hypodivergent, 29.1% (N=32) were normodivergent and 50% (N=55) were hyperdivergent. The curve of spee among these three groups were 3.39±0.30mm, 2.62±0.23mm and 2.02±0.45mm. One-way ANOVA has been applied and it showed highly significant differences in the value of curve of spee depth among three vertical skeletal patterns with the p-value of 0.000. Moving from Hypodivergent to Hyperdivergent cases, the curve of Spee depth reduces.
CONCLUSION: It has been found that there is a significant difference in curve of spee among hypodivergent, normodivergent and hyperdivergent patients.
KEYWORDS: Dentistry, Curve of Spee, Vertical Skeletal Patterns, Orthodontic Diagnosis.
HOW TO CITE: Rehman SA, Rizwan S, Faisal SS, Hussain SS. Association of curve of spee with vertical skeletal patterns. J Pak Dent Assoc 2020;29(4):254-258.
DOI: https://doi.org/10.25301/JPDA.294.254
Received: 16 August 2020, Accepted: 04 September 2020
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Madiha Anwar                                     BDS

Beenish Fatima Alam                       BDS, MFDS, MSc

Syed Jaffar Abbas Zaidi                 MD, MFDS, MFD, MSc, FAIMER

INTRODUCTION

The aesthetic sense of a dentist needs to be developed from the point where the undergraduate student starts learning dental anatomy and tooth morphology.1 A dentist should have comprehensive knowledge regarding the morphology and function of teeth, as it provides the basis for a dentist to be able to restore the missing tooth structure with the help of restorative material.2 Along with the theoretical knowledge of tooth structure and its function, for proper restoration, it is crucial to have technical skills, manual dexterity and above all an artistic sense.3 Only then a clinician will be able to reproduce anatomical details to make the dental restoration appear as natural as possible.4 Psychomotor skills of dental students related to dental carving need to be developed early on so that manual dexterity in recreating adequate tooth form may be achieved for restorative clinical procedures and functional purposes.5,6
In pre-clinical years, dental anatomy and tooth morphology is the first subject that introduces dental students to human teeth’s structure and function.7 The knowledge of tooth morphology, alignment and occlusion is critical for identification of teeth as well as to relate the morphology of teeth with function in health and disease. At this initial stage, it is important to develop the psychomotor skills of the students which will, later on, help them in identifying the details of the tooth surface as well as the changes in the structure in disease or pathology.8 Oral anatomy and tooth morphology have been traditionally taught by didactic lectures, books, manuals, artificial tooth models and extracted teeth.9
Wheeler’s10 was one of the first who introduced tooth carving in wax blocks. In many dental schools, all over the world, tooth carving is an integral part of the curriculum where the students are required to carve a tooth from an oversized wax block or soap bar.4,9,11-14 The carved tooth approximates the dimensions of the natural teeth and it is
intended to re-create the different positive and negative anatomical features such as cusps, cingulum, ridges, fossae and grooves.15 The main purpose of tooth carving is to recreate accurate anatomical and morphological landmarks of the permanent and deciduous dentition for physiological function and harmony of teeth. The main ethos of dental wax carving is to nurture the fine art of waxing teeth by restoring them to their correct morphology and anatomy.
Every tooth in the dental arch has six surfaces and every surface of the tooth has unique distinguishing and identifying features. None of these surfaces is flat. Dental wax carving
is an exercise in learning to duplicate the contours and convexities and concavities unique to the tooth type and location. The skills involved in dental wax carving familiarizes dental students with hand instrumentation techniques and allows them to develop skills such as finger dexterity, grip, hand steadiness, aiming and reaction; all of which are essential for the high precision needed to practice dentistry.
Previously, at Bahria University Medical and Dental College, foundational knowledge of tooth morphology was traditionally presented in didactic lectures, textbooks and study guides while psychomotor skills of students were developed through two-dimensional line drawings of teeth and sketching projects of different views of the teeth in graph books. This traditional model of teaching was instilling only superficial understanding without any clinical significance or integration with clinical sciences. Feedback of students indicated that they were facing difficulty in interacting with patients as the first two years are predominantly technical introductory pre-clinical phase of dental education with no emphasis on developing psychomotor skills. Furthermore, they stated that the oral biology course was inadequate to prepare students from pre-clinical care to direct patient care. clinically relevant psychomotor skills were insufficiently being taught, thereby hindering student’s ability to apply learning in direct patient care.
Carving of the tooth was introduced in the current academic year to promote self-directed active learning and to bring clinical relevance to the subject of oral biology. The objectives of this study was to examine the efficacy of a newly introduced systematic dental carving technique for teaching tooth morphology that relies on psychomotor and kinaesthetic learning to facilitate student transition from pre-clinical care to the clinical dental practice.Moreover, the effectiveness of self-directed learning and accuracy of
self-assessment by the students was evaluated. The study also involved a brief survey about student’s perception of dental carving.

METHODOLOGY

The study was conducted on forty-two students studying in the first undergraduate year of Bahria University Medical and Dental College. The intended research population was
first year dental students and the entire population was included in the study by total population sampling so no formal size calculation was needed.16 All the participants signed consent forms and ethical approval was obtained from Institutional Review Board Reference No. (ERC 02/2019). Maxillary central incisor was chosen for this study. The students were randomly divided into two study groups: The 1st group comprised of 21 students who were taught tooth morphology with the help of lectures, plaster models, sketching and drawing of various views of teeth. This group was provided with a handout regarding the steps involved in carving and they were asked to carve the tooth without any demonstration or help. The handout was adapted from Wheeler’s tooth carving manual.10
The second group comprised of 21 students who were taught tooth morphology with the help of lectures, plaster models, sketching and drawing. Besides, they were given a step-by-step demonstration of tooth carving and handouts in their lab sessions.
The students were provided with a wax block that was 50 mm in length, 25 mm in width and 25 mm in depth. Both the groups had to carve a maxillary central incisor; Group I; had no previous demonstration about the steps, while Group II; carved the tooth after step by step practical demonstration given by the facilitator. The teeth were evaluated by both students and examiners. The teeth were coded, and examiners were randomly allocated carved teeth to ensure the integrity and validity of the results.

GRADING CHART

To standardize the grading of the teeth a grading chart was formulated based on the study conducted by Kilistoff et al17 (Table 1). This standardized chart had scored for different morphological features of the maxillary central incisor. The tooth was evaluated; 2 marks for each completed feature, 1 mark for complete feature and 0 for an absent feature.
The carved teeth of both groups were collected and coded. The carved teeth of 1st group were randomly divided into students of the 2nd group and vice versa. The students were asked to self-evaluate the teeth provided to them according to the grading chart. The teeth were then evaluated by two examiners who were randomly assigned carved teeth from either group. The examiners evaluated the teeth on the same grading chart

Table 1: Marking chart for tooth carving

*0= Feature is absent, 1= feature is present but it is incomplete or poorly defined, 2= Feature is clear and properly formed.

STUDENT’S SATISFACTION SURVEY

A questionnaire was modified by the authors from a study conducted by Abu Eid et al18 to gather student’s feedback regarding tooth carving. The survey consisted of two parts; the first part had the questions related to demographics of the students and the second part had twelve dichotomous questions related to tooth carving.

STATISTICAL ANALYSIS

Descriptive tests were applied to the demographics of the students, such as age and gender. Independent sample t-test was applied to compare the mean results of the scores given by the students and the examiners. A P-value of less than 0.005 was kept as significant. The frequency of the responses to the survey was calculated by applying descriptive tests.

RESULTS

The demographics of the participants are shown in Table 02. The marks that are given by the students and the faculty are shown in Table 03. The mean scores given by

Table 2: Demographics of the participants

Table 3: Marks given by the faculty and students

*P-value less than 0.05 is considered significant

Table 4: Student’s feedback form

morphological landmarks are exaggerated and easy to locate. These models can also help in reproducing the same features on the surface of the carved tooth. The plaster models were provided to the students and in response to the question regarding its helpfulness 61.9 % of students of the first group and 71.4% of students of the second group gave a positive response. This shows that enlarged tooth plaster models provide a three-dimensional teaching modality for tooth morphology that surpasses videos and other resources and can be used as a reference for tooth carving practice.
When the students were enquired whether they found the crown or root part difficult in carving, they had a mixed response. In general, students found carving the root part difficult as compared to a crown. This could be attributed to the fact that root is thinner than the crown part and hence more difficult to carve and manage as it can easily be chipped off during carving.13
The majority of the students in this study agreed that tooth carving should be taught in the first undergraduate year (71.4% and 61.9% respectively). Regarding the usefulness of the teaching methodology used, 71.4% of the students of the group I agreed that it helped improve their skills and knowledge, as it encouraged self-directed learning and experimenting. E-learning modalities like blended learning through learning management systems can provide the much-needed multi-modal approach of teaching dental anatomy(25). While 61.9% of the students of group II agreed that they preferred live demonstration of dental wax carving as it demonstrated step by step instructions. Self-assessment of students by evaluating each other’s carved teeth helped to familiarize to the marking criteria. Seventy-two per cent students of both the groups agreed that the marking criteria for dental wax carving was realistic, robust and comprehensive. Detailed analysis of all the anatomical features of a maxillary central incisor was included in the marking criteria.
A similar survey conducted by Eid et al18 on dental students reported similar results. Another survey conducted by Megahanand et al26 on dental graduates and practitioners
had a similar perception of tooth carving. They also agreed that tooth carving is important for clinical skills and should be an integral component of the undergraduate dental curriculum. A strong body of evidence including a systematic review conducted on the PRISMA guidelines suggested that practical demonstration of dental wax carving along with
the instructor was one of the most effective pedagogies in learning dental anatomy.13
Limitations of this study is that this study is based on a single institution. Large scale multi-centre studies are needed to generalize and validate the findings of this study. Based on these evidence-based recommendations, dental wax carving should be an integral component of the dental curriculum in Pakistan as it promotes early pre-clinical skills development commensurate with longitudinal integration and is an effective pedagogy for learning practical dental anatomy.

CONCLUSION

Based on results of this study, dental wax carving was found to be an effective pedagogical strategy to develop preclinical psychomotor skills of dental students by recreating tooth morphological characteristics in wax. The progress of students will be followed through their clinical skills and subsequently into their clinical work. It is therefore imperative
that tooth carving be included in the national dental curriculum so that longitudinal or spiral integration can take place.

CONFLICT OF INTEREST

None declared

REFERENCES

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1. Senior Lecturer, Department of Oral Biology, Bahria University Medical & Dental College, Karachi.
2. Assistant Professor, Department of Oral Biology, Bahria University Medical & Dental College, Karachi.
3. Assistant Professor, Department of Oral Biology, Dow Dental College, Dow University of Health Sciences,Karachi.
   Corresponding author: “Dr. Syed Jaffar Abbas Zaidi” < jaffar.zaidi@duhs.edu.pk >

Madiha Anwar                                     BDS

Beenish Fatima Alam                        BDS, MFDS, MSc

Syed Jaffar Abbas Zaidi                   MD, MFDS, MFD, MSc, FAIMER

OBJECTIVE: Dental wax carving exercise is a practical exercise to teach tooth morphology that develops psychomotor skills needed to practice clinical dentistry. This study aimed to determine the efficacy of dental wax carving as a teaching modality for tooth morphology and its assessment.
METHODOLOGY: Forty-two first-year dental students were randomly divided into two groups participated in this study from February 2019 till September 2019 at Bahria University Medical & Dental College. Learning resources related to dental wax carving were provided to the first group of 21 students while the second group of 21 students received practical demonstration. The students self-evaluated their dental carving based on a standardized checklist and two examiners then evaluated the dental wax carvings randomly. A student satisfaction survey was performed at the end of this study to gain feedback regarding the dental wax carving as a teaching methodology.
RESULTS: The mean scores given by self-assessment of students were significantly higher than those given by the examiners to both the groups. The group that was given a live demonstration of dental carving scored higher than the self-directed learners. Overall student feedback regarding dental carving was uniformly positive.
CONCLUSION: Dental carving skills develop manual dexterity and psychomotor skills for practicing clinical dentistry and should be an integral component of preclinical dental curriculum so that clinically relevant cognitive & psychomotor skills are incorporated. Based on this study, a live demonstration of dental wax carving skills should be the preferred teaching modality.
KEYWORDS: Dental carving, tooth carving, wax carving, psychomotor skills, dental anatomy, restorative dentistry
HOW TO CITE: Anwar M, Alam BF, Zaidi SJA. Tooth carving as a teaching modality in the study of tooth morphology: Students’ perception and performance. J Pak Dent Assoc 2020;29(4):249-253.
DOI: https://doi.org/10.25301/JPDA.294.249
Received: 11 April 2020, Accepted: 04 July 2020
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