A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: a case report

A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: a case report

A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: A case report Erika Miti Yasui, BDS,a Richard...

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A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: A case report Erika Miti Yasui, BDS,a Richard Keiji Kimura, DDS,b Akemi Kawamura, DDS,c Shigehisa Akiyama, DDS, PhD,d and Ichijiro Morisaki, DDS, PhD,e Sa˜o Paulo, Brazil, and Osaka, Japan BANDEIRANTE UNIVERSITY AND OSAKA UNIVERSITY

Self-inflicted oral trauma occurs in a number of conditions with different etiologic and clinical characteristics. The management of such trauma also varies depending on the medical history of the patient; the etiology of the behavior; and the severity, frequency, and method of inflicting injury. This case report describes a modified oral screen placed in a 10-month-old female infant with cerebral palsy who had been having feeding problems caused by self-inflicted oral trauma. The modified oral screen effectively protected the wounds against further oral trauma to the lower lip and tongue without being fixed to the dentition. (Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2004;97:471-5)

Several strategies and appliances have been described to prevent and treat self-inflicted oral trauma, such as in comatose patients,1-3 Riga-Fede disease,4,5 Gaucher’s disease,6 and Lesch-Nyhan syndrome.7,8 Case reports involving infants and patients with cerebral palsy, however, make up a relatively small percentage of these studies. Self-inflicted oral trauma in infants is a very serious problem because it can create painful lesions that interfere with proper suckling and feeding and, thus, put the infant at risk of nutritional deficiency.9 In this report, we describe the management of selfinflicted oral trauma in a 10-month-old female infant with cerebral palsy through the use of a specially designed oral appliance.

erupted 2 months earlier. These biting episodes subsequently increased both in severity and frequency, resulting in a more serious wound. Because of the pain, she was unable to tolerate feeding with a nursing bottle, so nutrition was mainly supplied through a nasal feeding tube. An oral examination revealed partial eruption of the upper and lower incisors. The traumatic lesions were on the ventral and dorsal sides of the tip of the tongue and on the lower lip mucosa and were painful to the touch (Fig 1, A and B). Her breathing was exclusively nasal, but upper respiratory tract impairment was observed in the glossopharyngeal region. The patient was the second born from a premature delivery of opposite-sex twins, with delayed physical and mental development, a weight of 2584 kg, a stature of 40 cm, and very little trunk and head control. Her co-twin had been delivered without medical problems, with a weight of 2644 kg, a stature of 41 cm, and growth within the normal range.10

CASE REPORT A 10-month-old female infant clinically diagnosed with cerebral palsy was referred by her pediatrician and attended our clinic with oral trauma and bleeding. The medical history disclosed that the infant started having tonic biting motions when the mandibular and maxillary primary central incisors

a

Visiting Clinical Research Fellow, Bandeirante University, Sa˜o Paulo, Brazil. b Instructor, Division of Special Care Dentistry, Osaka University, Osaka, Japan. c Senior Resident, Division of Special Care Dentistry, Osaka University, Osaka, Japan. d Associate Professor, Division of Special Care Dentistry, Osaka University, Osaka, Japan. e Professor and Chief, Division of Special Care Dentistry, Osaka University, Osaka, Japan. Received for publication Apr 21, 2003; returned for revision Jun 25, 2003; accepted for publication Oct 1, 2003. 1079-2104/$ - see front matter Ó 2004 Elsevier Inc. All rights reserved. doi:10.1016/j.tripleo.2003.10.010

TREATMENT OF THE ORAL LESIONS The initial treatment entailed topical medication on the wounds with triamcinolone acetonide (Kenalog; BristolMyers Squibb, Tokyo, Japan) and by smoothing the incisal edges of the teeth. However, these procedures proved ineffective because of the tonic oral muscle movements, which continued to inflict trauma. Therefore, we hypothesized that the use of an oral appliance would prevent further trauma. Mandibular and maxillary arch impressions were made by using modeling compound in prefabricated trays made of resin because of its viscosity and rapid setting, and a working model was obtained. Then a pressure-laminated mouth guard, 0.8 mm in thickness, made of cellulose acetate butyrate (ERKO-DENT, Pfalzgrafenweiler, Germany) was fabricated (Fig 2, A). However, this appliance was also ineffective in that it was easily dislodged and the infant’s mother had difficulty in inserting and removing it. In addition, an increase in tonic muscular reflexes was 471

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Fig 2. Three kinds of oral appliances used in our patient. A, The Erkodur mouth guard fabricated for our patient. 1, Maxillary arch; 2, mandibular arch. B, The oral screen made of autopolymerizing acrylic resin. 1, Frontal view; 2, upper view. C, The modified oral screen made of Erkoflex. 1, Frontal view showing the exhaust holes; 2, upper view.

Fig 1. A, The patient at the first visit, with the nasal feeding tube in place. B, The initial intraoral examination disclosed severe ulceration on the tip of the tongue and the lower lip.

induced when the mouth guard was inserted in the patient’s mouth. We then decided to use an oral screen made of autopolymerizing acrylic resin (tray resin II; Shofu Inc, Kyoto, Japan). The impression of the oral vestibule in the occlusal position was taken by using a special tray (Fig 3, A and B), and an appliance was designed to fit the entire oral vestibule, in the manner described by Cheney11 (Fig 2, B). The oral screen was placed to verify proper fitting, and dental floss was attached to it for security. At the next examination, 4 days after insertion, the mother reported that the infant had adapted to the appliance well, but periodic episodes of displacement had occurred. Because we sought to improve the stability in the infant’s mouth, a modified design of a new oral screen was considered. An impression of the mucobuccal fold was taken by using tissue conditioner (Shofu Inc) in the custom tray; this helped to ensure that the appliance adapted appropriately to the oral structures. For the modified oral screen, we used a flexible material, ethylene vinyl acetate (4-mm Erkoflex mouth guard;

Erkodent). We extended the lateral aspect of the appliance and placed 2 exhaust holes on the body of the appliance to prevent it from being blown out of the mouth (Fig 2, C). Relief for the muscle and frenal attachments was carefully ensured at the initial placement of this appliance (Fig 4, A and B). Four days after the insertion of the new appliance, a significant reduction in injury to the oral tissues was observed. The mother reported her satisfaction with respect to the ease in removing and inserting the appliance, and the infant rapidly acclimated to the appliance with no further episodes of displacement. Periodic examinations were performed to monitor the tongue, lower lip, and the development of the dentition. Three weeks after the placement, the modified oral screen had to be remade because the elasticity of the material had degraded. Nonetheless, the intraoral examination showed that the wounds had healed completely, with no signs of further trauma (Fig 4, C).

DISCUSSION The patient in this report had severe self-inflicted oral trauma and additional concerns that required special attention. Rendering a clinical diagnosis and determin-

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Fig 3. The special tray for vestibular impression A, Labial view. B, Vestibular view.

ing the attendant treatment was especially difficult because the infant had a severe delay in neuromotor and intellectual development and was uncooperative. Four treatment strategies such as topical medication, incisal smoothing, mouth guard, and oral screen made of hard or flexible resin had been adapted to this patient; however, none of these resulted in a complete cure. In the infant’s medical history, the mother reported that the onset of the lesions coincided with the eruption of the primary incisors. On the basis of the results of the intraoral examination, we first sought to alleviate the patient’s problem through the concurrent smoothing of the edges of the incisors and the prescription of a topical medication. These treatments are effective for most of the patients with Riga-Fede disease. The condition known as Riga-Fede disease was first described by Riga in 1881, followed by Fede in 1890, and is characterized by ulceration on the ventral surface of the tongue caused by erupting incisions during suckling.4 In our patient, however, the lesions persisted after this treatment because she continued to inflict trauma to the oral

Fig 4. The modified oral screen. A, The clinical appearance of the patient with the modified oral screen in place. B, The oral screen in position in the oral vestibular space. C, Intraoral aspect of the patient after 3 weeks of therapy with the oral screen.

tissues, similar to the situation with the patient reported by Kozai et al.6 A proper understanding of the mechanism of selfinflicted oral trauma is essential to the treatment. Thus,

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with close observation of the patient, we noticed that she was experiencing strong tonic biting movements that had trapped the lower lip and tongue between the maxillary and mandibular incisors. The patient’s facial expression signified that she felt pain from this biting of the oral tissues. However, because of the severe neuromuscular dysfunction, she could not avoid biting the lower lip and tongue. From these observations, we made diagnosis of the lesions as self-inflicted oral trauma caused by uncontrolled and uncoordinated oral muscle movement. On the basis of this diagnosis, we decided to use a device that could hold her lip and tongue to prevent them from being bitten by the maxillary and mandibular incisors and allow the traumatized sites to heal. In our patient, the principal problem that we found in designing an effective appliance was related to its stability in the oral cavity. Most appliances used in the prevention and treatment of self-inflicted oral trauma must be seated on or wired to the dental surfaces—for example, resin splints, mouth guards,2 acrylic trays, and lip bumpers.14,15 It was not possible to use these appliances in our patient because of the lack of dental surfaces. Self-inflicted oral trauma can sometimes be prevented with the placement of a mouth guard, which is also indicated for use in sports and as an oral habit breaker.9 According to Hanson et al,16 the objectives of fabricating an appliance to prevent oral trauma are as follow:  To deflect traumatized tissues away from the occlusal table  To permit mandibular movement  To enable daily oral care  To heal the injured tissues  To be easily fabricated  To be comfortable  To not pose a risk to the patient. On the basis of these factors, we attempted to solve the problem by creating an oral screen made of self-cured acrylic resin by following the model described by Cheney.11 The oral screen is a myofunctional orthodontic appliance that is indicated to restore nasal breathing, to intercept premaxillary protrusion, and to prevent habits such as finger-sucking and tongue-thrusting. We decided to use the oral screen because it lay comfortably in the vestibular space between the lip and teeth and did not impinge on the alveolar mucosa. The patient tolerated the appliance well, but it was not resistant to dislodgment. Modifications of the design were performed on the basis of stability and retention to maintain the patient’s compliance; these included the extension of the posterior area, the placement of 2 exhaust holes on the appliance

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body, and the use of a flexible material. The primary retention of this appliance was accomplished by means of a labial bow and the strategically placed holes. The use of a flexible material permitted the appliance to function in concert with the oral muscle movements, preventing dislodgment and facilitating its manipulation by the child’s mother. The material deteriorates with time, but it can be remade simply and quickly and does not require a new impression of the arches, as do other appliances.2,12-15 In infants, taking impressions is always a problem because of limited access to the oral cavity and their uncooperativeness. Furthermore, in patients with cerebral palsy, this procedure provokes the patient’s spasticity, making it even more difficult to take an impression. Nevertheless, our patient adjusted to the appliance quickly and without incident. Healing of the soft tissue wounds progressed satisfactorily, and periodic follow-up examinations were scheduled to enable close observation of the patient. We are not sure how long the patient will have to use the oral screen because a longer follow-up period is necessary to confirm whether it was just a transient or an acute episode of self-inflicted oral trauma. The results nonetheless indicate that the new design of the oral screen was effective in preventing selfinflicted oral trauma in an infant with cerebral palsy. We thank Dr Clive Schneider Friedman for his kind help in revising the manuscript.

REFERENCES 1. Turley PK, Henson JL. Self-injurious lip-biting: etiology and management. J Pedod 1983;7:209-20. 2. Hayward JR, Trefz BR, Robert RC, Yellich GM. Soft plastic mouth guards for use in prevention of self-inflicted oral trauma. J Hosp Dent Pract 1979;13:36-7. 3. Sheller B. Self-inflicted oral trauma: report of case. Spec Care Dentist 1992;12:28-9. 4. Zaenglein AL, Chang MW, Meehan SA, Axelrod FB, Orlow SJ. Extensive Riga-Fede disease of the lip and tongue. J Am Acad Dermatol 2002;47:445-7. 5. Baghdadi ZD. Riga-Fede disease: report of a case and review. J Clin Pediatr Dent 2001;25:209-13. 6. Kozai K, Okamoto M, Nagasaka N. New tongue protector to prevent decubital lingual ulcers caused by tongue thrust with myoclonus. ASDC J Dent Child 1998;65:474-7. 7. Lee JH, Berkowitz RJ, Choi BJ. Oral self-mutilation in the LeschNyhan syndrome. ASDC J Dent Child 2002;69:66-9. 8. Rashid N, Yusuf H. Oral self-mutilation by a 17-month-old child with Lesch-Nyhan syndrome. Int J Paediatr Dent 1997;7:115-7. 9. Stokes AN, Croft GC, Gee D. Comparison of laboratory and intraorally formed mouth protectors. Endod Dent Traumatol 1987;3:255-8. 10. Scher AI, Petterson B, Blair E, Ellenberg JH, Grether JK, Haan E, et al. The risk of mortality or cerebral palsy in twins: a collaborative population-based study. Pediatr Res 2002;52:671-81. 11. Cheney EA. Treatment planning and therapy in the mixed dentition. Am J Orthod 1963;44:568-80.

ORAL SURGERY ORAL MEDICINE ORAL PATHOLOGY Volume 97, Number 4 12. Cehreli ZC, Olmez S. The use of a special mouthguard in the management of oral injury self-inflicted by a 4-year-old child. Int J Paediatr Dent 1996;6:277-81. 13. Fabiano JA, Thines TJ, Margarone JE. Management of selfinflicted oral trauma: report of case. Spec Care Dentist 1984;4: 214-5. 14. Saemundsson SR, Roberts MW. Oral self-injurious behavior in the developmentally disabled: review and a case. ASDC J Dent Child 1997;64:205-9, 228. 15. Sonnenberg EM. Treatment of self-induced trauma in a patient with cerebral palsy. Spec Care Dentist 1990;10:89-90.

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16. Hanson GE, Ogle RG, Giron L. A tongue stent for prevention of oral trauma in the comatose patient. Crit Care Med 1975;3:200-3. Reprint requests: Ichijiro Morisaki, DDS, PhD Division of Special Care Dentistry Osaka University Faculty of Dentistry 1-8 Yamadaoka, Suita-Osaka 565-0871 Japan [email protected]