Pictorial Essay 35

Oligodontia in a Teenager with Down Syndrome – A Case Study Zahnmedizinische Aspekte der Behandlung von Anodontie bei einem Teenager mit Down Syndrom

Authors

W. Reiche1, L. Parkanyi2, G. Braunitzer2, K. Nagy2, K. Karpati2

Affiliations

1



2

Key words ▶ Down syndrome ● ▶ dental implants ● ▶ oligodontia ● ▶ orthodontic treatment ● ▶ minimally invasive treatment ●

Introduction

Bibliography DOI  http://dx.doi.org/ 10.1055/s-0034-1384525 Published online: October 22, 2014 Klin Padiatr 2015; 227: 35–37 © Georg Thieme Verlag KG Stuttgart · New York ISSN 0300-8630 Correspondence Dr. Wilfried Reiche Reiche & Partner Zahnmedizinisches ­Kompetenzzentrum Porschestraße 74 38440 Wolfsburg Germany Tel.:  + 49/536/118 810 Fax:  + 49/536/1188 117 [email protected]



For a long time, Down syndrome patients with oligodontia have not been rehabilitated at all or just insufficiently, by the application of bridges and/or removable prostheses. It is very difficult to find Down-specific correction guidelines or protocols in the literature, while such protocols developed specifically for children and teenagers living with this disease do not exist at all. This is obviously a hiatus that might easily keep dentists from treating these patients beyond basic care. Orthodontic correction with subsequent implant insertion is an obvious solution, however, the circumstances often limit the range of applicable methods. The authors present the case of a teenage patient whose condition ruled out invasive orthodontic treatment (even though it was indicated), and implant insertion had to be carried out in a minimally invasive manner too. Trisomy 21 is the most common of the chromosomal disorders. It is a chromosomal numerical aberration caused in 95 % of the cases by meiotic nondisjunction resulting in a chromosome count of 47. In 4 % of the cases the extra chromosomal material is present not as an extra chromosome but as a translocation of the long arm of chromosome 21 to chromosome 22 or 14. 1 % of the patients are mosaics, having a mixture of 46- and 47-chromosome cells. Maternal age is a significant factor of incidence: the disease occurs in 1 in 1 550 live births in women under 20 years of age, in contrast to 1 in 25 live births in mothers over 45 [3]. Clinically the disease is characterised by a very typical phenotype: the patients are oligophrenic to varying degrees, they have a flat face profile and epicanthal folds, and in about 40 % of the cases cardiac malformations are also present. Due primarily to cardiac malformations and susceptibility to various infections the life expectancy of these patients used to be short. Despite that fact that the causes for the immune defi-

ciency Trisomy 21 have not yet been clearly identified, it is true that life expectancy of this group of patients is increasing, even in severe cases such as myeloid leukemia (ML-DS) [6]. Today, however, about 80 % of them reach their fiftieth birthday [10], which makes their quality of life an issue to be seriously considered, and the management of their often disabling dental problems, is one way to address that issue. Factors that are relevant for dental treatment planning include brachycephaly, skeletal and/or dental dysgnathia, hypodontia, oligodontia, macroglossia, muscle hypotonia, hypermobility, joint luxations and immune deficiency [5]. Decreased compliance and poor ability to maintain oral hygiene also complicate the situation [1]. Oligodontia is among the most prevalent dental problems in this group of patients [8]. In spite of that, only a few studies discuss the treatment of this problem [7, 9], and we know about only one study that deals with the implant treatment of a teenage Down syndrome patient. The aim of this Pictorial Essay is to show that oral rehabilitation with dental implants in such cases is indeed possible, even in unfavourable circumstances.

Case report



The treatment of an 18-year-old patient with Down syndrome is presented. Dental status assessment revealed multiple agenesis of the teeth 12, 17, 18, 22, 27, 28, 31, 38, 41 and 48, dysgnathia (ANGLE class III as recrudescence alio loco after multiband treatment), macroglossia with habitual tongue position, lateral and frontal open bite with occlusion on the first molars ▶  Fig. 1), and persisting deciduous teeth (53, 63, ( ● 65, 71 and 81). The compliance of the patient in this case could be categorised as average, and parental support/compliance as excellent. In view of the parental compliance it was assumed

Reiche W et al. Oligodontia in a Teenager …  Klin Padiatr 2015; 227: 35–37

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Schlüsselwörter ▶ Down Syndrom ● ▶ Zahnimplantate ● ▶ Oligodontie ● ▶ kieferorthopädische ● ­Behandlung ▶ minimal invasive Behandlung ●

 Reiche & Partner, Zahnmedizinisches Kompetenzzentrum, Wolfsburg, Germany  Faculty of Dentistry, University of Szeged, Szeged, Hungary.

36 Pictorial Essay

that the guidelines for oral hygiene and domestic measures would be observed. The necessity of consequent cooperation was pointed out to the parents on several occasions. An invasive surgical correction of the dysgnathia and subsequent orthodontic multiband treatment to correct the non-occlusion and the open bite were planned, as well as the retrusion of the canine teeth in the upper jaw and holding the space in the frontal position of the lower jaw. After successful orthodontic treatment implants in the positions 12, 22, 31 and 41 were to be inserted. However, this (basically invasive) treatment plan was ruled out once we learned that the patient had already undergone multiple heart surgeries under general anaesthesia. His chronic pneumonia and recurring infections, which were accompanied by an elevated risk of endocarditis, confirmed our decision. The treatment plan was adjusted so that the dysgnathia would be treated exclusively with non-invasive methods, thus accepting a possibly suboptimal outcome. The orthodontic treatment was initiated by extracting the teeth 53, 63 and 65 to open the gaps of 12 and 22 through retorsion from 13 and 23. Teeth 71 and 81 were left in situ to serve as ‘space holders’. The ultimate goal of the orthodontic treatment was the correction of the occlusal open bite. This was carried out ▶  Fig. 2). Since the patient by the application of class III elastics ( ● was unable to adjust the bands several times a day by himself, this task was accomplished mainly by his father and the nursing staff of the workshop for disabled people. The results after one and a half year of non-invasive treatment exceeded our expectations. The bite was almost perfectly closed and the planned implants could now be inserted under analgosedation and antibiotic prophylaxis. Lateral bone augmentation was indicated, but minimal invasiveness was also observed, therefore techniques like lateral bone augmentation with iliac crest transplants and/or guided bone regeneration were not considered. Rather, specially designed thin implants by the Spanish firm BTI (Tiny® Implants) were applied. Through the use of the expander set that comes as an element of the implant system it became possible to prepare the implant alveolus properly and with only slightly traumatic bone splitting. To avoid recrudescence, the multiband appliance remained in its position and was only removed shortly before the insertion of the superstructures. The healing of the implants as well as the prosthetic phase of the treatment went without ▶  Fig. 3). During the entire span of the treatment complications ( ● the patient attended check-ups and dental ­cleanings every 4

Reiche W et al. Oligodontia in a Teenager …  Klin Padiatr 2015; 227: 35–37

Fig. 2  Example for class III Elastics (another patient-case).

Fig. 3  X-Ray after insertion of the implants.

week. As a result, oral hygiene could be kept at a satisfactory level throughout. Gingival lesions were not observed during the treatment. The therapy of the patient was accomplished about 2 years ago ▶  Fig. 4 and ●  ▶  Fig. 5) and he still follows a strict 3-month recall ( ● protocol. Both the prosthetic reconstruction and the natural teeth are in an excellent condition, in which the parents’ dedication to keeping up a high level of oral hygiene possibly plays an important role.

Discussion



Given the often low level of cooperation and difficulties that arise from the systemic factors of the disease, the dental rehabilitation of patients with Down syndrome often poses a challenge. At the same time it is known that oligodontia is the most frequent dental problem in Down syndrome, that the life expectancy of these patients is relatively long today, and that dental rehabilitation by implant treatment significantly enhances patient-perceived quality of life also in otherwise healthy patients [2]. These factors point to the importance of dental rehabilitation in this patient group, even in the face of difficulties. Sometimes a regular implant therapy is counterindicated, like in the presented case. Because of the medical history of the patient we had to adjust our original invasive approach, and we had to find a minimally invasive way, which meant that the surgical correction of dysgnathia had to be given up completely, and we had to find an implant system that enabled us to work with

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Fig. 1  Dental status before treatment.

Pictorial Essay 37 Conclusion

Fig. 4  Situation after the treatment.

In summary, although oligodontia in Down syndrome may be a challenge for the dentist, it is far from unmanageable. Even in a case where only a limited set of methods was at our disposal as a standard implantation approach could not be taken, an optimal outcome could be reached. The most important technical lesson we learned from this case is that a noninvasive orthodontic method can yield just as good results as an invasive one in a case when an invasive approach seems to be indicated. Of course, this is not generalisable, but we suggest that noninvasive alternatives should always be considered, especially in this population of patients, who suffer from all the different aspects of their condition anyway. The choice of methods, however, is merely a technical issue, and even the best choice can result in poor outcome if patient compliance is not good. The compliance of Down syndrome patients is rarely promising in such terms. Therefore it is essential to establish a very good rapport with caretakers, as their understanding and adherence may be the factor the outcome hinges on.

Conflict of interest: The authors have no conflict of interest to disclose. Literature

Fig. 5  Situation after the treatment.

­ arrow bone without bone augmentation in a minimally invan sive way. We thought that the noninvasive correction of dysgnathia would lead to a suboptimal result, but to our surprise class III elastics turned out to be an alternative to surgery. We do not believe that without the excellent compliance of the parents and nursing staff this would have been possible. The application of the specially designed implants yielded the expected results. The implantation and subsequent healing went without complications. The implants have now survived more than 2 years and no signs of deterioration are seen, in which, again, excellent oral hygiene and the outstanding compliance of all concerned play a major role.

1 Al Habashneh R, Al-Jundi S, Khader Y et al. Oral health status and reasons for not attending dental care among 12- to 16-year-old children with Down syndrome in special needs centres in Jordan. Int J Dent Hyg 2012; 10: 259–264 2 Curtis DA, Sharma AB, Finzen FC. The use of dental implants to improve quality of life for edentulous patients. J Calif Dent Assoc 2008; 36: 275–280 3 Kumar V, Abbas AK, Fausto N et al. Robbins Basic Pathology. 9th ed. Philadelphia: Elsevier Saunders, 2012 4 Lustig JP, Yanko R, Zilberman U. Use of dental implants in patients with Down syndrome: a case report. Spec Care Dentist 2002; 22: 201–204 5 de Moraes ME, de Moraes LC, Dotto GN et al. Dental anomalies in patients with Down syndrome. Braz Dent J 2007; 18: 346–350 6 Reinhardt D, Reinhardt K, Neuhoff C et al. GATA1-Mutations-assoziierte Leukämien bei Kindern mit Trisomie 21-Mosaik. Klin Padiatr 2012; 224: 153–155 7 Ribeiro CG, Siqueira AF, Bez L et al. Dental implant rehabilitation of a patient with down syndrome: a case report. J Oral Implantol 2011; 37: 481–487 8 Russell BG, Kjaer I. Tooth agenesis in Down syndrome. Am J Med Genet 1995; 55: 466–471 9 Soares MR, de Paula FO, Chaves M et al. Patient with Down syndrome and implant therapy: a case report. Braz Dent J 2010; 21: 550–554 10 World Health Organisation. Genes and Chromosomal Diseases. 2013; Available: http://www.who.int/genomics/public/geneticdiseases/en/ index1.html

Reiche W et al. Oligodontia in a Teenager …  Klin Padiatr 2015; 227: 35–37

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