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UNIVERSITY” Sv.Kiril I Metodij” DENTISTRY FAKULTY SKOPJE , 2012 REVIEW PAPER: Rapid Maxillary Expansion Effects in Children with Nocturnal Enuresis KANDITATE: MENTOR: MIMOZA SELMANI Prof.dr.JULIANA GJORGOVA 1 Rapid Maxillary Expansion Effects in Children with Nocturnal Enuresis Introduction Primary nocturnal enuresis (NE) refers to involuntary loss of urine during sleep in patients who have never achieved a sustained period of dryness. Secondary NE is enuresis that develops after a patient has achieved a sustained period of bladder control.5 Children over the age of 4–6 years who wet their beds more than two nights per month are diagnosed as having NE.Although less than 5% of children with nocturnal enuresis have an organic basis for their wetting, the physical examination and history should carefully rule out any physical causes.15.Factors that have been observed to be associated with the occurrence of nocturnal enuresis include developmental delay, introduction of negative factors, heredity, small bladder capacity, and reduced nocturnal secretion of antidiuretic hormone.10 Treatment options include pharmacotherapy, the enuretic alarm, and complex regimens such as dry-bed training. Recently, desmopressin acetate (DDAVP) nasal spray has been found to be useful when temporary cessation of symptoms is necessary, such as for enuretic children attending school camps or sleeping over at friends' homes. Treatment of NE with imipramine has fallen into disfavor because of high relapse rate and risk of fatal over-dosing.8There were also recorded tonsylectomia and adenoidectomia for improvement or treatment of nocturnal enuresis,as paginal effects.1 2 3 25 16 17Nevertheles, no ash accurate mechanism. A previous retrospective study of 10 children with varying degrees of nocturnal enuresis has shown that one side effect of rapid maxillary expansion (RME) is spontaneous reduction in bedwetting at night. The aim of this prospective study was to analyze the effect of RME treatment (mean 2 weeks) in cases of chronic, long-standing nocturnal enuresis. Ten children, 8 to 13 years old, who had not responded to conventional medical treatment for bed-wetting, were referred from the pediatric department.9According to another study which were examined 10 cases,nocturnal enuresis ceased within a few months of maxillary expansion.There is growing consensus that upper airway obstruction is a causative factor in nocturnal enuresis.22 2 Nevertheles,similar studies on the 8 children who are liable to psychosocial stress condition and who had not responded to different conventional medical treatments.At the end of eight months observation, the mean rate of improvement in bed-wetting in the seven successful subjects was 74.2% after RME treatment.However, none of the subjects became completely dry.However, the long-term success rate is still questionable.23These three pilot studies shows that RME may have a positive effect on NE.The aim of this study was to determine the effect of orthodontic RME treatment in the short term on the NM in children with long-term resistance to medical therapy.The long-term success rate 10 years after RME, attitudes to treatment, and factors that influenced the success or failure were also important to analyze. MATERIALS AND METHODS Twenty-three subjects, 18 boys and 5 girls aged 6.7–15.0 years (mean = 10.1 years), took part in the study. The children and adolescents had been referred from pediatric specialists at Ryhov County Hospital in Jönköping, Sweden, or from other pediatric specialist clinics and had undergone a full pediatric and urologic examination. All were judged to be otherwise medically healthy. Assessment of ENT disorders was made by an ENT specialist at the hospital, and no respiratory problems were found. All children had primary enuresis, that is, enuresis had been ongoing without interruption since early childhood. Of the 23 children, 19 wet the bed every night, sometimes several times a night, and 4 children wet the bed several nights a week (Table1). Two children were daywetting. 3 Previous enuresis treatment included wetness alarm; behavioral therapy; Minirin (Ferring AB, Sweden), an antidiuretic hormone substitute; and acupuncture or paramedical methods. All children had earlier treatment with a wetness alarm and all except one had been treated with Minirin. All treatments had failed. Medical records were consulted, and anamneses were taken before treatment (Table1). Pre- and postexpansion casts were taken for occlusal classification and transverse arch measurements. Eleven children had a Class I occlusion, 11 had a Class II division 1, and 1 had a Class III occlusion. Seven children had crossbites—six were unilateral and one was bilateral. Lateral head films were also taken before treatment for cephalometric diagnosis (Table2.) and measurements of posterior airway related to age.11 Thirteen children were judged to be mouth breathers at the examination. Before treatment at the ENT department, all subjects underwent anterior rhinomanometry with and without a decongestant spray to determine nasal airway resistance (Rhinocomp, Cintec Invest AB, Sweden). Posttreatment (maximum of 1 month after completion of maxillary expansion) rhinomanometric examinations at the ENT department were difficult to schedule, and some patients had little interest in repeating the exam. Therefore, only 13 of the 23 patients underwent a follow-up rhinomanometric examination. Treatment consisted of orthodontic maxillary expansion using a Hyrax screw soldered to Ormco bands on the permanent first molars and first premolars of patients with these teeth (Figure 1), and on the first or second primary molars of patients who had unerupted first premolars. A parent activated the Hyrax screw each morning and evening, which gave an expansion of 0.45 mm a day. Expansion was made for a period of 2–2.5 weeks. The appliance was then replaced with a transpalatal arch in combination with a lingual arch made in contact with all teeth. This appliance was used as the retainer for 6 months. After that, the retainer was removed, the children were clinically monitored for 1 year, and the number of wet nights was recorded. A 10-year follow-up was done by administering a telephone questionnaire . Some of the questions were suggested by pediatric specialists. The interviews were conducted by one of the authors (USF). 4 RESULTS Of the 23 therapy-resistant children with primary NE, 11 improved and 12 reported no change after 10–14 days of orthodontic maxillary expansion (Figure2). Six became completely dry within 1 month after expansion and another five reported notable improvement.Improvement occurred more often in subjects younger than 10 years. Of the 14 subjects younger than age 10, 5 became completely dry, 3 improved, and 6 were unaffected. Of the nine subjects aged 10 years and older, only one became completely dry (Figure2.). The children with NE who showed improvement were investigated further to determine whether improvement was correlated with age, improved nasal resistance, sagittal and transversal occlusion, or anamnestic factors, such as family history or school performance (Table1.). No associations were found.Before the RME, one child in the dry group, one in the improved group, and two in the no change group were wetting the bed several nights a week. After the RME, NE decreased in the improved group so that three of the five children wet their bed only 1 or 2 nights a week. Expansion A mean expansion of 6.5 mm (range = 5–8 mm) was achieved. Seven children had a lateral crossbite at the start—six unilateral and one bilateral. The same amount of expansion was made for all children, irrespective of the presence of a crossbite or not. When expansion was complete, most children tended to have a scissors-bite in lateral segments. One year after the retainer was removed, transverse molar relations were normal in all children. Of the seven children with crossbites at the start, three became dry, one improved and three showed no change. 5 Rhinomanometry. Pharyngeal airways were assessed by the ENT department at the hospital, and no major deviations were found. Nasal resistance was reduced after expansion in 8 of the 13 measured subjects, 6 of whom were younger than 9 years. The mean improvement in nasal breathing was 28%. Of the 13 clinically diagnosed mouthbreathers, 2 became dry, 5 improved, and 6 showed no change. 10-year posttreatment interview. The dropout rate for the interviews 10 years after treatment was low (9%), as all but two subjects could be reached. The results of the 21 interviews were analyzed by group, that is, dry (n = 6), improved (n = 5), and no change (n = 10) during the 1-year observation period. Four members of the dry group believed RME had helped cure the enuresis, one subject said RME did not help, and one could not remember. Concerning their memories of the orthodontic appliance—whether RME was uncomfortable—five of the six subjects reported no problems and only one mentioned pain in the mouth and headache during expansion. No one was kept awake at night because of the appliance, and no one had to wake up at night to go to the bathroom. Questions on their present symptoms revealed that just one subject had to get up to urinate at night. No one had daytime symptoms, experienced obstipation, or was under medication for enuresis. Two of the six subjects in the dry group snored at night. Of the subjects that showed improvement after RME, two believed RME had helped, one thought RME had not helped, and two could not remember. One of the five subjects in this group thought the appliance was uncomfortable and made eating difficult. Four had no problems with the appliance. The appliance kept one of the subjects awake at night and one subject woke up at night to go to the bathroom. Concerning present symptoms, two had to urinate at night; no one had daytime symptoms or obstipation or was on medication. Two members of this group snored at night. 6 In the group that experienced no change in enuresis after RME, two thought RME improved the enuresis, six thought RME had not helped, and two could not remember. In this group, four had bad memories of the orthodontic appliance. They thought the appliance was large and clumsy, and they felt pain when the screws were tightened. The appliance kept one subject awake at night; two did not wake up, and seven could not remember. One had to wake up at night to go to the bathroom, three did not, and six could not remember. When this group was asked about current symptoms, four reported that they had to get up to urinate at night, nobody had any daytime symptoms or obstipation, and no one was on medication for enuresis. Two subjects reported that they snored.Ten years after the end of orthodontic treatment, 4 of the 21 interviewed subjects remembered that they were completely dry immediately after expansion. Fourteen became dry 1–3 years after RME. Three could not remember when they stopped wetting the bed. Seven still had to get up to urinate at night, no one had daytime problems or obstipation problems, and no one was on medication for enuresis. Six subjects reported that they snored. The enuresis stopped 1–3 years after RME in five subjects under the age of 10 years and nine subjects over the age of 10 years. DISCUSSION The results of RME treatment of 23 children in this study were similar to results of RME treatment of 10 children with NE in a previous pilot study.9 The results for these 33 children can be pooled for many reasons. The referrals came from the same pediatric clinics, and they were all treated at the same orthodontic clinic by the same team with identical appliances and routines for medical and dental registrations and follow-ups. Following are the pooled results for the 33 children with NE: 10 became completely dry within 1 month of RME, 8 showed notable improvement, and 15 experienced no change. More than 50% of the pooled group experienced immediate improvement (Figure 3.) compared with a spontaneous cure rate of 15% per year.15 7 In one study, transverse changes in the dentofacial structures and nasopharyngeal airway dimensions were analyzed statistically. The improvement of NE is hypothetically linked to improved breathing and thus to better saturation of blood. It is a well-known anatomical fact that there is an increase in the width of the nasal cavity immediately after expansion, particularly at the floor of the nose adjacent to the midpalatal suture,6 7 21 and RME positively affects nasal breathing by expanding the maxilla and the nasal cavity. Rhinomanometry and acoustic rhinometry provide the best methods for objective assessment of nasal airflow. However, care should be taken to calibrate the device properly, and the user should be completely familiar with rhinomanometers regarding the mathematical algorithm for resistance used by the accompanying software.18 More over, these two systems do not always reveal correlating results,24 and other factors, such as esophageal pressure, irrelevant to the nasal area may affect the measurements.19 Still, despite a notable increase in airflow, as measured by rhinomanometry before and after maxillary expansion, 45% of the subjects in our study were unaffected by RME. Thus, no absolute relationship seems to exist between airflow and enuresis. Breathing Although it has recently been argued that standard orthodontic radiographic methods suffice to evaluate breathing disorders,23 12 rhinomanometry with and without nasal decongestant spray both before and after maxillary expansion must be considered more accurate, especially for comparing measurements.9 We noted that the children with NE in our study had no ENT disorders and that respiratory functions were normal from the start. It could be speculated that RME relapses in the dental arch when the teeth tip due to masticatory function and cheek pressure. Osteoid formation and deposition in the median palatal suture might be more stable. It has been suggested that 9–12 months after RME, nasal airflow (nasal resistance) is still 80%– 90% of the maximal expansion effect.12 8 Crossbites In this study, 7 of 23 subjects had crossbites—six unilateral and one bilateral. Mean maxillary expansion was 6.5 mm (range = 5–8 mm). This exceeded the mean of 4.1 mm (range = 3–5) in the previous study.9 The subjects with no crossbite were thus dentally overexpanded in the maxilla, but they relapsed to normal after the expansion appliance was removed. Thus, expansion treatment was performed to improve or cure enuresis, even though transverse occlusions were often normal. Two RME studies have reported clinically stable, lasting effects in the arch.13 20 It may be assumed that these studies were made in cases with indications for maxillary expansion, that is, constricted or narrow arches or transverse crossbite occlusions. It is important to note that most subjects (16 of 23) in this study had normal arch perimeters and normal transverse occlusions. Thus, a full relapse to normal transverse occlusion was not unexpected afterward. Normal transverse occlusion does not seem to be a contraindication for 5–8-mm transverse maxillary expansion in an attempt to cure NE in children. Genetics Genetic factors are the most important in the etiology of nocturnal enuresis but somatic and psychosocial environmental factors have a major modulatory effect. Most commonly, nocturnal enuresis is inherited via an autosomal dominant mode of transmission with high penetrance (90%). All subtypes of nocturnal enuresis (primary, secondary, combined day/night wetting) are susceptible to comparable genetic influences. Certain syndromes of day wetting follow their own genetic mechanisms but this association with the genetics of nocturnal enuresis is not known.4 In fact, 60% of the patients in our sample (16 of 23) had a family history of enuresis (Table1)—12 of 23 were first-degree relatives—but no correlation with successful treatment was found. Success rate The success rate in this study of 23 children, where success was defined as dry or improved NE, was 48%, that is, somewhat less than in the pilot study by the same group (7 of 10). If the two samples are compiled, the success rate is 55% for the group of 33 children with NE after RME. 9 Noteworthy is the fact that the 10-year follow-up found the effects to be stable. This can be compared with an annual improvement of about 15%.2 In our opinion, RME treatment can be recommended, and the success rate is encouraging, especially considering that the children in our sample were long-standing chronic cases, were therapy resistant, and had no urogenital defects or respiratory disorders.Orthodontic expansion treatment is noninvasive, routine and rather quick and it has no negative side effects or complications. A strong relationship between NE and OSA, where 35% of a group of children with OSA also had NE, has been reported.1 When surgery was indicated in children with OSA, the NE resolved 60%–75% of the time.1Another hypothesis for the improvement of this specific sample can be the placebo effect. It is clear that coming to the dental school several times for the treatment and being taken care of might have positively affected the psychology of the children. However, the orthodontist is the final referral, and such a possible placebo effect should have expressed itself earlier, when previous treatment alternatives were introduced. The introduction of an orthodontic appliance in the mouth may also have a placebo effect. However, how long and to what extent the presence of an orthodontic appliance could be successful in the treatment of NE is an open question.23 CONCLUSIONS RME seems to positively affect NE. Results of orthodontic RME treatment in 23 children (aged 6–15 years old) with long-standing therapy-resistant primary enuresis were fairly immediate improvement in almost 50%.Six of the 21 subjects believed the orthodontic appliance had been uncomfortable and painful. This study demonstrated that RME treatment might cause relief for the enuretic children, although the long-term success rate is questionable and the underlying responsible mechanisms are still unclear. Therefore, further long-term follow-up research studies taking psychosocial factors into consideration are desirable in larger samples to build up a more reliable chain between responsible etiological factors and NE treatment modalities like RME. 10 REFERENCES 1. Basha S,Bialowas C,Ende K,Szeremeta W.Effectivenes of adenotonsillectomi in nocturnal enuresis secondary to obstructive sleep apnea.Laryngoskope.2005,115:1101-1103. 2. Guilleminault C,Eldridge FL,Simmons FB,Dement WC.Sleep apnea in eight children.J Pediatr.1976,58:23-30. 3. Guilleminault C.Stoohs R.Obstructive sleep apnea syndrome in children.J Pediatr.1990,17:46-51. 4. Gontard A,Schaumburg H,Hollman E,Eiberg H,Rittig S.The genetics of enuresis J.Urol 2001 Dec;166(6):2438-43 5. Harari M. Nocturnal enuresis. Aust Fam Physician. 1999;28(2):113-116. 6. Haas AJ. Rapid expansion of the maxillary dental arch and nasal cavity by opening the mid-palatal suture. Angle Orthod. 1961; 31:73–90. 7. Iseri H, Tekkaya E, Öztan Ö, Bilgiç S. Biomechanical effects of rapid maxillary expansion on the craniofacial skeleton, studied by the finite element method. Eur J Orthod. 1998; 20:347–356. 8. Jensen IN, Kristensen G. Alarm treatment: Analyses of response and relapse. Scand J Urol Nephrol Suppl. 1999;202:73-75. 9. Kurol J, Modin H, Bjerkhoel A.Orthodontic maxillary expansion and its effect on nocturnal enuresis.Angle Orthod.1998,68:225-232. 10. Lippincots Prim Care Pract.1997 Sep-Oct;1(4):399-407.Nocturnal enuresis in children.Sellinger VJ.Family Physician Associates,PA,of Tallahassee Pediatrics,Florida,USA. 11. Linder-Aronson S,Henrikson CO.Radiocephalometric analysis of anteroposterior nasopharyngeal dimensions in 6 to 12 year old mouth breathers compared with nose breathers.Practica Otorhinolaryngol.1973,212:19-29. 12. Lima, A. L. , R. M. Lima Filho , and A. M. Bolognese . Long-term clinical outcome of rapid maxillary expansion as the only treatment performed in Class I malocclusion. Angle Orthod 2005. 75:416–420. 11 13. Lagravere, M. O. , P. W. Major , and C. Flores-Mir . Long-term dental arch changes after rapid maxillary expansion treatment: a systematic review. Angle Orthod 2005. 75:151–157. 14. Memikoglu UT, Iseri H. Effects of a bonded rapid maxillary expansion appliance during orthodontic treatment. Angle Orthod. 1999; 69:251–256. 15. Neve us T,La”ckgren G,Tuvemo T,Hetta J,Hja”Ima”s K,Stenberg A.Enuresisbackground and treatment.SCAND j Urol Nephrol.2000,206:1-44. 16. Nowak KC,Weidder DJ.Pediatric nocturnal enuresis secondary to airway obstruction from cleft palate repair.ClinPediatr.1998,37:653-657. 17. Ng DK,Chau KW,Kwok KL.Nocturnal enuresis and obstructive sleep apnea in two children.Singapore Med J.2001,42:590-591. 18. Naito K, Miyata S, Saito S, Sakurai K, Takeuchi K. Comparison of perceptional nasal obstruction with rhinomanometric and acoustic rhinometric assessment. Eur Arch Otorhinolaryngol. 2001; 258:505–508. 19. Oliveira, N. L. Assessment of Rapid Maxillary Expansion and its Effects on the Nasal Cavity´s Size and Airway Resistance [master's thesis]. Chicago: University of Illinois; 2005. 20. Rushton, G. H. Nocturnal enuresis: Epidemiology, evaluation and currently available treatment options. J Pediatr 1989. 114:691. 21. Schumacher MJ. Nasal congestion and airway obstruction: the validity of available objective and subjective measures. Curr Allergy Asthma Rep. 2002; 2:245–251. 22. Timms, Donald J., Rapid maxillary expansion in the treatment of nocturnal enuresis. The Angle Orthodontist 1990:60(3):229-33. 23. Usumez S, Iseri h , Orhan M , Basciftci FA. Effect of rapid maxillary expansion on nocturnal enuresis.Angle Orthod.2003,73:532-538. 24. Virkkula P, Silvola J, Lehtonen H, Salmi T, Malmberg H. The effect of esophageal pressure monitoring on nasal airway resistance. Otolaryngol Head Neck Surg. 2001; 125:261–264. 25. Weider DJ,Hauri PJ.Nocturnal enuresis in children with upper airway obstruction.Int. J Pediatr. Otolaryngol.1985,9:173-182. 12 Table 1. Results from pretreatment interviews and medical records for the 23 children with enuresis 13 Table 2. Results of the pretreatment clinical and lateral head film examination of the 23 children with enuresis Figure 1.Maxillary expansion appliance using a Hyrax screw soldered to bands on the permanent first molars and first premolars 14 Figure 2.Changes in nocturnal enuresis in 23 children. Figure 3.Changes in nocturnal enuresis in 33 children. 15