- Email: [email protected]
Successful treatment of self-inflicted tongue trauma patient using a special oral appliance
G Model
PEDOT-7716; No. of Pages 4 International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case report
Successful treatment of self-inflicted tongue trauma patient using a special oral appliance§ Ik Jae Kwon a, Soung Min Kim a,*, Hee Kyung Park b, Hoon Myoung a, Jong Ho Lee a, Suk Keun Lee c a
Department of Oral and Maxillofacial Surgery, Dental Research Institute, School of Dentistry, Seoul National University, Seoul, South Korea Department of Oral Medicine and Oral Diagnosis, Dental Research Institute, School of Dentistry, Seoul National University, Seoul, South Korea c Department of Oral Pathology, College of Dentistry, Gangneung-Wonju National University, Gangneung, South Korea b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 18 May 2015 Received in revised form 3 August 2015 Accepted 5 August 2015 Available online xxx
A 7-year-old male presented with a painful ulcerative lesion on the right lateral tongue and left lower buccal mucosa due to self-inflicted trauma. Antibiotic medication and use of a mouthwash agent were not effective. We made a special oral appliance to cover the maxillary arch and teeth to protect the tongue. The patient showed immediate improvement and did not suffer from any complications. Invasive procedures such as biopsy were not needed. We believe that accurate clinical diagnosis is important and treatment with an oral appliance is effective in self-inflicted oral trauma in children. ß 2015 Elsevier Ireland Ltd. All rights reserved.
Keywords: Oral appliance Self-inflicted trauma Oral self-injury Tongue biting
1. Introduction Self-inflicted oral injury is a behavioral disturbance that consists of deliberate destruction of an oral lesion [1]. Self-injury is an important health care factor that can affect individuals of any age or gender and its incidence is increasing among pediatric patients [1]. This is a very serious problem for pediatric patients because it can create painful lesions that interfere with proper suckling and feeding and, thus, put the patient at risk of nutritional deficiency [2]. Tongue biting is the most common mode of self-inflicted injury, most frequently involving the oral and perioral regions [1]. Tongue biting is a distressing situation for the child and a challenging problem for the clinician. There are many etiological factors contributing to tongue biting including developmental disability, which may be associated with a syndrome such as Lesch–Nyhan [3] or Moebius [4], neuropathy causing insensitivity to pain [5] and cerebral palsy [2]. However, in this case, the patient had no relevant medical history.
§ This study was supported by Basic Science Research and International Research & Development Program through the National Research Foundation of Korea (NRF) funded by the Ministry of Education (NRF-2010-0012214) & the Ministry of Science, ICT & Future Planning (2014K1A3A9A01033785). * Corresponding author. Tel.: +82 2 2072 0213; fax: +82 2 766 4948. E-mail address: [email protected] (S.M. Kim).
Several strategies and appliances have been described for the prevention and treatment of self-inflicted oral trauma. Treatment may be classified into 4 groups: psychological treatment, pharmacological treatment, intraoral devices, and surgical procedures [1]. Establishing widely applicable clinical treatment protocols is difficult. The majority of English literature on oral self-mutilation is in the form of case reports, rendering diagnosis, elimination of other pathological diseases and selection of treatment options difficult. In this report, we present a case of oral self-inflicted tongue trauma in a 7-year-old patient and discuss the diagnosis and successful treatment of oral self-injury. 2. Case presentation A 7-year-old male was referred to the Department of Oral and Maxillofacial Surgery at Seoul National University Hospital, for the surgical biopsy under the tongue cancer suspicion. The patient suffered from a four-week history of ulcerative lesion on right lateral tongue and had no relevant medical history. Clinically, traumatic erythematous and ulcerative lesion on right lateral border of the tongue was observed with slight swelling and pain (Fig. 1A). The ulcerative lesion was positioned near the upper and lower first molar, which had been the main cause of the trauma (Fig. 1B). The patient had mixed dentition with permanent anterior
http://dx.doi.org/10.1016/j.ijporl.2015.08.013 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
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PEDOT-7716; No. of Pages 4 2
I.J. Kwon et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
Fig. 1. Initial intraoral examination of the right lateral tongue (A), dorsal part of tongue (B), and left lower anterior buccal mucosa (C).
Fig. 2. A dental model was fabricated and the construction bite was 5 mm in thickness (A), the modified oral appliance using acrylic resin with Adams clasps and ‘C’ clasps (B), application of the modified oral appliance (C).
Fig. 3. Clinical aspect of the gradually healing tongue after one week (A), continuous improvement with decreased symptom after two months (B), and the lesion was completely healed after six months (C).
teeth and a first molar. He also had a similar ulcerative lesion on the left lower anterior buccal mucosa (Fig. 1C). Based on both tongue and mucosal leisons, the diagnosis was traumatic ulcer. Before visiting our hospital, he was treated at another hospital with antibiotic medication and mouth-wash agents. Unfortunately, those treatments were not effective and the lesion appeared to be worsen. We decided that tongue biting caused the lesion after careful clinical examination. We did not conduct any invasive surgical procedures such as biopsy or excision, but instead constructed an oral appliance to cover the maxilla arches and teeth to protect the tongue against further tissue damage. Impressions with alginate materials and occlusal registrations with baseplate wax were made and models were poured in dental stone. Construction bite was 5 mm in thickness (Fig. 2A). A modified oral appliance was fabricated using acrylic resin and hard wires for retention containing Adams clasps and ‘C’ clasps on both sides (Fig. 2B). The appliance was designed to fully cover the maxilla teeth and adjusted to occlusion with 5-mm thickness (Fig. 2C). We periodically evaluated the stability of occlusion and changes in
dentition. The appliance was worn full-time except during mealtimes. During the initial treatment of approximately 2 weeks, NSAIDs were prescribed to reduce pain and inflammation. The patient and his parents were educated and very cooperative for our avoiding risks such as swallowing, heavy force or broken resin. This patient was evaluated regularly. After one week, the ulcerative lesion gradually improved (Fig. 3A). Specifically, the left lower buccal lesion healed rapidly, indicating it was a traumatic lesion. We encouraged the patient to wear the appliance as long as possible, preferably 12–14 h a day. During follow-up, the lesion continually improved and symptoms including pain and swelling decreased in two months (Fig. 3B). After six months, tongue biting had ceased and the ulcerative lesion was completely healed (Fig. 3C). The patient’s general health had improved because he could eat comfortably. Subsequently, invasive procedures were not performed and other pathological benign or malignant lesions were excluded. We advised the patient to wear the appliance for as long as possible and periodically adjusted it due to dentition changes.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
G Model
PEDOT-7716; No. of Pages 4 I.J. Kwon et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
3. Discussion Position of the ulcer and change of occlusions, which is cause of emotional change, are remarkable diagnostic points, although diagnosis of self-inflicted oral trauma is quite difficult and challenging issues for clinician. Moreover, if the patient is pediatric, differential diagnosis will be harder, because they usually don’t understand their condition exactly and hard to control and communicate. Also they sometimes have uncontrolled oral habits which are cause of self-trauma. Although there are few well-developed diagnostic tools and treatment protocols for oral injury, some researchers emphasize detecting risk factors, early diagnosis and oral habits control [1]. Between six and seven year old, permanent teeth are starting to erupt with permanent first molar and incisors, so the occlusions are changing [6]. At this mixed dentition, oral health and conditions, such as occlusions, are related to emotional status, resulted to psychotic problems [7]. Psychotic problems are one of the major causes of selfinjurious behavior [8]. In our 7-year-old case, he had two ulcerative lesions in right posterior tongue (Fig. 1B) and left lower anterior buccal mucosa (Fig. 1C). They were positioned to near permanent first molars and incisors each other. The position near the permanent teeth indicated that oral self-injury was cause of the ulcer and it might change the emotional status. Our oral appliance was customized for individuals, so it is necessary to take impressions with the reconstruction bite registrations. So uncooperative patients, such as Cerebral Palsy or infants cannot be indicated for this special appliance. And for a 2 year old child, there are no posterior teeth usually, so other preventive types of selfinflict for only anterior teeth, such as wet gauze biting, can be recommended. The patient in this report had severe self-inflicted oral trauma and additional concerns that required special attention. The lesion showed significant improvement after the acrylic appliance was placed on the teeth. Biopsy was not necessary, because the traumatic lesion improved as the etiologic factor was eliminated. The patient underwent periodic follow-up to evaluate the other teeth as they erupted. In the future, the appliance can either be extended or adjusted for erupted or extracted teeth. Rapid healing proved that physical restraint is an effective method to prevent self-injury directly in an individualized manner. Severe and repeated tongue biting injury can cause ulceration. Several authors reported that the potential for local infection is high and might result in a generalized systemic inflammatory response [9]. Therefore, to determine the treatment strategy, accurate diagnosis is important. The cause of ulcerative lesions and the risk factors of tongue injury should be evaluated before invasive procedures such as biopsy are performed. If the risk factors are evaluated correctly, the response of treatment can be rapid. If not, other potential causes such as malignancy can be further evaluated. In the present case, the patient was young and habitual tongue biting was observed on clinical examination. Therefore, non-invasive treatment using a restrictive oral device was selected. In our case, after one week, the lesion’s clinical aspect improved and six months later the site of injury was healed completely. In previously reported cases, if an oral appliance was working properly, the lesion was improved clinically approximately 2–3 weeks later and after 3 months the lesion was almost completely healed [4,10]. Conversely, if the lesion did not improve despite restricting oral habits after 2–3 weeks, additional diagnostic methods such as biopsy may be necessary. Several methods have been proposed for the treatment of selfinflicted oral injuries, including behavior modification, pharmacological agents, psychosocial therapy and oral appliances such as acrylic splints, tongue stents or mouth guards [9]. Among these potential treatments, many authors chose oral appliances as the
3
first remedy. Variable types of oral appliances have been introduced, such as hard acrylic resin splints [11,12], thermoplastic splints [13], or mouth guards [3]. Furthermore, some clinicians demonstrated that modified oral self-injury appliance provided better protection and behavior control. For example, to prevent traumatic macroglossia, tongue guards bended separately and embedded in the acrylic appliance are used [14]. Several clinicians used a combination of thermoplastic splints and a mouth guard for infants [2]. To choose the form of treatment, we considered the effectiveness, stability and retention to maintain patient compliance. Additionally an appliance should prevent tissue damage and protect the oral cavity including the tongue, buccal cheek, teeth and gingiva. A hard acrylic resin splint with Adams clasps and ‘C’ clasps had good stability, retention and maintenance. We used a construction bite 5 mm in thickness, which was expected to help occlusal stability and harmony of myofascial function. In addition, the design that covered the maxillary arch and teeth protected the teeth and surrounding tissue. We focused the thickness of appliance, and the 5.0-mm thickness can accommodate some space for tongue posture and teeth covering resin will prevent the direct contact between teeth and tongue surface. The appliance also could be modified and adjusted for patients changing occlusion during adolescence. After one year follow up, the appliance was still functioning properly for tissue protection and habit prevention. Our special oral appliance is suitable to pediatric patients who can be cooperative in noninvasive dental care such as teeth brushing and impression. Both primary and permanent dentitions, including mixed dentition, are suitable for this oral device. As hard acrylic resin and proper clasps give sufficient retention, patients can retain their oral device easily. Furthermore, the elastic properties of clasps allow removing the device easily. Our young patient could wear and take off our oral device easily by himself after education. Additionally, the patient experienced no complications or discomfort. Our oral appliance design was effective for treatment of oral self-injury. 4. Conclusion In the present case report we emphasized the importance of a behavioral approach for treating self-mutilation in pediatric patient. Accurate examination and evaluation of unconscious biting habits must be performed. A customized oral appliance to prevent tongue biting was our treatment of choice. This technique was a more conservative therapy, which in turn made more invasive treatments unnecessary. Conflict of interest statement There are no conflicts of interest related to this article to declare. References [1] J. Limeres, J.F. Feijoo, F. Baluja, J.M. Seoane, M. Diniz, P. Diz, Oral self-injury: an update, Dent. Traumatol. 29 (1) (2013) 8–14. [2] E.M. Yasui, R.K. Kimura, A. Kawamura, S. Akiyama, I. Morisaki, A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: a case report, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 97 (4) (2004) 71–75. [3] M. Romero Maroto, C. Ruiz Duque, G. Vincent, I. Garcia Recuero, A. Romance, Management of oral lesions in Lesch–Nyham syndrome, J. Clin. Pediatr. Dent. 38 (3) (2014) 247–249. [4] L.F. Guimara˜es, M.E. Janini, A´.S.B. Vieira, L.C. Maia, L.G. Primo, Self-inflicted oral trauma in a baby with Moebius syndrome, J. Dent. Child. 74 (3) (2007) 224–227. [5] M.M.S. Schalka, M.S.N.P. Correˆa, A.L. Ciamponi, Congenital insensitivity-to-pain with anhidrosis (CIPA): a case report with 4-year follow-up, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 101 (6) (2006) 769–773.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
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[6] S. Bayrak, E. Sen Tunc, N. Tuloglu, A. Acikgoz, Timing of permanent teeth eruption in Turkish children, J. Clin. Pediatr. Dent. 37 (2) (2012) 207–211 (2012). [7] T.S. Barbosa, P.M. Castelo, M.S. Leme, M.B.D. Gavia˜o, Associations between oral health-related quality of life and emotional statuses in children and preadolescents, Oral Dis. 18 (7) (2012) 639–647. [8] R.M. Winchel, M. Stanley, Self-injurious behavior: a review of the behavior and biology of self-mutilation, Am. J. Psychiatry 148 (3) (1991) 306–317. [9] S. Kiat-amnuay, S.H. Koh, D.J. Powner, An occlusal guard for preventing and treating self-inflicted tongue trauma in a comatose patient: a clinical report, J. Prosthet. Dent. 99 (6) (2008) 421–424. ¨ lmez, The use of a special mouthguard in the management of oral [10] Z.C. C¸ehreli, S. O injury self-inflicted by a 4-year-old child, Int. J. Paediatr. Dent. 6 (4) (1996) 277–281.
[11] M.C. Munerato, S.P. Moure, V. Machado, F.G. Gomes, Self-mutilation of tongue and lip in a patient with simple schizophrenia, Clin. Med. Res. 9 (1) (2011) 42–45. [12] A. Arhakis, N. Topouzelis, E. Kotsiomiti, N. Kotsanos, Effective treatment of selfinjurious oral trauma in Lesch–Nyhan syndrome: a case report, Dent. Traumatol. 26 (6) (2010) 496–500. [13] A. Suchak, M. Mars, A unique presentation of severe tongue biting in 10 monthold twins with a novel approach to management, Eur. Arch. Paediatr. Dent. 11 (3) (2010) 149–154. [14] B. Karayazgan-Saracoglu, D. Kecik, Utilization of a modified bite guard for preventing traumatic macroglossia, J. Craniofac. Surg. 22 (1) (2011) 238–240.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
PEDOT-7716; No. of Pages 4 International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
Contents lists available at ScienceDirect
International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Case report
Successful treatment of self-inflicted tongue trauma patient using a special oral appliance§ Ik Jae Kwon a, Soung Min Kim a,*, Hee Kyung Park b, Hoon Myoung a, Jong Ho Lee a, Suk Keun Lee c a
Department of Oral and Maxillofacial Surgery, Dental Research Institute, School of Dentistry, Seoul National University, Seoul, South Korea Department of Oral Medicine and Oral Diagnosis, Dental Research Institute, School of Dentistry, Seoul National University, Seoul, South Korea c Department of Oral Pathology, College of Dentistry, Gangneung-Wonju National University, Gangneung, South Korea b
A R T I C L E I N F O
A B S T R A C T
Article history: Received 18 May 2015 Received in revised form 3 August 2015 Accepted 5 August 2015 Available online xxx
A 7-year-old male presented with a painful ulcerative lesion on the right lateral tongue and left lower buccal mucosa due to self-inflicted trauma. Antibiotic medication and use of a mouthwash agent were not effective. We made a special oral appliance to cover the maxillary arch and teeth to protect the tongue. The patient showed immediate improvement and did not suffer from any complications. Invasive procedures such as biopsy were not needed. We believe that accurate clinical diagnosis is important and treatment with an oral appliance is effective in self-inflicted oral trauma in children. ß 2015 Elsevier Ireland Ltd. All rights reserved.
Keywords: Oral appliance Self-inflicted trauma Oral self-injury Tongue biting
1. Introduction Self-inflicted oral injury is a behavioral disturbance that consists of deliberate destruction of an oral lesion [1]. Self-injury is an important health care factor that can affect individuals of any age or gender and its incidence is increasing among pediatric patients [1]. This is a very serious problem for pediatric patients because it can create painful lesions that interfere with proper suckling and feeding and, thus, put the patient at risk of nutritional deficiency [2]. Tongue biting is the most common mode of self-inflicted injury, most frequently involving the oral and perioral regions [1]. Tongue biting is a distressing situation for the child and a challenging problem for the clinician. There are many etiological factors contributing to tongue biting including developmental disability, which may be associated with a syndrome such as Lesch–Nyhan [3] or Moebius [4], neuropathy causing insensitivity to pain [5] and cerebral palsy [2]. However, in this case, the patient had no relevant medical history.
§ This study was supported by Basic Science Research and International Research & Development Program through the National Research Foundation of Korea (NRF) funded by the Ministry of Education (NRF-2010-0012214) & the Ministry of Science, ICT & Future Planning (2014K1A3A9A01033785). * Corresponding author. Tel.: +82 2 2072 0213; fax: +82 2 766 4948. E-mail address: [email protected] (S.M. Kim).
Several strategies and appliances have been described for the prevention and treatment of self-inflicted oral trauma. Treatment may be classified into 4 groups: psychological treatment, pharmacological treatment, intraoral devices, and surgical procedures [1]. Establishing widely applicable clinical treatment protocols is difficult. The majority of English literature on oral self-mutilation is in the form of case reports, rendering diagnosis, elimination of other pathological diseases and selection of treatment options difficult. In this report, we present a case of oral self-inflicted tongue trauma in a 7-year-old patient and discuss the diagnosis and successful treatment of oral self-injury. 2. Case presentation A 7-year-old male was referred to the Department of Oral and Maxillofacial Surgery at Seoul National University Hospital, for the surgical biopsy under the tongue cancer suspicion. The patient suffered from a four-week history of ulcerative lesion on right lateral tongue and had no relevant medical history. Clinically, traumatic erythematous and ulcerative lesion on right lateral border of the tongue was observed with slight swelling and pain (Fig. 1A). The ulcerative lesion was positioned near the upper and lower first molar, which had been the main cause of the trauma (Fig. 1B). The patient had mixed dentition with permanent anterior
http://dx.doi.org/10.1016/j.ijporl.2015.08.013 0165-5876/ß 2015 Elsevier Ireland Ltd. All rights reserved.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
G Model
PEDOT-7716; No. of Pages 4 2
I.J. Kwon et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
Fig. 1. Initial intraoral examination of the right lateral tongue (A), dorsal part of tongue (B), and left lower anterior buccal mucosa (C).
Fig. 2. A dental model was fabricated and the construction bite was 5 mm in thickness (A), the modified oral appliance using acrylic resin with Adams clasps and ‘C’ clasps (B), application of the modified oral appliance (C).
Fig. 3. Clinical aspect of the gradually healing tongue after one week (A), continuous improvement with decreased symptom after two months (B), and the lesion was completely healed after six months (C).
teeth and a first molar. He also had a similar ulcerative lesion on the left lower anterior buccal mucosa (Fig. 1C). Based on both tongue and mucosal leisons, the diagnosis was traumatic ulcer. Before visiting our hospital, he was treated at another hospital with antibiotic medication and mouth-wash agents. Unfortunately, those treatments were not effective and the lesion appeared to be worsen. We decided that tongue biting caused the lesion after careful clinical examination. We did not conduct any invasive surgical procedures such as biopsy or excision, but instead constructed an oral appliance to cover the maxilla arches and teeth to protect the tongue against further tissue damage. Impressions with alginate materials and occlusal registrations with baseplate wax were made and models were poured in dental stone. Construction bite was 5 mm in thickness (Fig. 2A). A modified oral appliance was fabricated using acrylic resin and hard wires for retention containing Adams clasps and ‘C’ clasps on both sides (Fig. 2B). The appliance was designed to fully cover the maxilla teeth and adjusted to occlusion with 5-mm thickness (Fig. 2C). We periodically evaluated the stability of occlusion and changes in
dentition. The appliance was worn full-time except during mealtimes. During the initial treatment of approximately 2 weeks, NSAIDs were prescribed to reduce pain and inflammation. The patient and his parents were educated and very cooperative for our avoiding risks such as swallowing, heavy force or broken resin. This patient was evaluated regularly. After one week, the ulcerative lesion gradually improved (Fig. 3A). Specifically, the left lower buccal lesion healed rapidly, indicating it was a traumatic lesion. We encouraged the patient to wear the appliance as long as possible, preferably 12–14 h a day. During follow-up, the lesion continually improved and symptoms including pain and swelling decreased in two months (Fig. 3B). After six months, tongue biting had ceased and the ulcerative lesion was completely healed (Fig. 3C). The patient’s general health had improved because he could eat comfortably. Subsequently, invasive procedures were not performed and other pathological benign or malignant lesions were excluded. We advised the patient to wear the appliance for as long as possible and periodically adjusted it due to dentition changes.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
G Model
PEDOT-7716; No. of Pages 4 I.J. Kwon et al. / International Journal of Pediatric Otorhinolaryngology xxx (2015) xxx–xxx
3. Discussion Position of the ulcer and change of occlusions, which is cause of emotional change, are remarkable diagnostic points, although diagnosis of self-inflicted oral trauma is quite difficult and challenging issues for clinician. Moreover, if the patient is pediatric, differential diagnosis will be harder, because they usually don’t understand their condition exactly and hard to control and communicate. Also they sometimes have uncontrolled oral habits which are cause of self-trauma. Although there are few well-developed diagnostic tools and treatment protocols for oral injury, some researchers emphasize detecting risk factors, early diagnosis and oral habits control [1]. Between six and seven year old, permanent teeth are starting to erupt with permanent first molar and incisors, so the occlusions are changing [6]. At this mixed dentition, oral health and conditions, such as occlusions, are related to emotional status, resulted to psychotic problems [7]. Psychotic problems are one of the major causes of selfinjurious behavior [8]. In our 7-year-old case, he had two ulcerative lesions in right posterior tongue (Fig. 1B) and left lower anterior buccal mucosa (Fig. 1C). They were positioned to near permanent first molars and incisors each other. The position near the permanent teeth indicated that oral self-injury was cause of the ulcer and it might change the emotional status. Our oral appliance was customized for individuals, so it is necessary to take impressions with the reconstruction bite registrations. So uncooperative patients, such as Cerebral Palsy or infants cannot be indicated for this special appliance. And for a 2 year old child, there are no posterior teeth usually, so other preventive types of selfinflict for only anterior teeth, such as wet gauze biting, can be recommended. The patient in this report had severe self-inflicted oral trauma and additional concerns that required special attention. The lesion showed significant improvement after the acrylic appliance was placed on the teeth. Biopsy was not necessary, because the traumatic lesion improved as the etiologic factor was eliminated. The patient underwent periodic follow-up to evaluate the other teeth as they erupted. In the future, the appliance can either be extended or adjusted for erupted or extracted teeth. Rapid healing proved that physical restraint is an effective method to prevent self-injury directly in an individualized manner. Severe and repeated tongue biting injury can cause ulceration. Several authors reported that the potential for local infection is high and might result in a generalized systemic inflammatory response [9]. Therefore, to determine the treatment strategy, accurate diagnosis is important. The cause of ulcerative lesions and the risk factors of tongue injury should be evaluated before invasive procedures such as biopsy are performed. If the risk factors are evaluated correctly, the response of treatment can be rapid. If not, other potential causes such as malignancy can be further evaluated. In the present case, the patient was young and habitual tongue biting was observed on clinical examination. Therefore, non-invasive treatment using a restrictive oral device was selected. In our case, after one week, the lesion’s clinical aspect improved and six months later the site of injury was healed completely. In previously reported cases, if an oral appliance was working properly, the lesion was improved clinically approximately 2–3 weeks later and after 3 months the lesion was almost completely healed [4,10]. Conversely, if the lesion did not improve despite restricting oral habits after 2–3 weeks, additional diagnostic methods such as biopsy may be necessary. Several methods have been proposed for the treatment of selfinflicted oral injuries, including behavior modification, pharmacological agents, psychosocial therapy and oral appliances such as acrylic splints, tongue stents or mouth guards [9]. Among these potential treatments, many authors chose oral appliances as the
3
first remedy. Variable types of oral appliances have been introduced, such as hard acrylic resin splints [11,12], thermoplastic splints [13], or mouth guards [3]. Furthermore, some clinicians demonstrated that modified oral self-injury appliance provided better protection and behavior control. For example, to prevent traumatic macroglossia, tongue guards bended separately and embedded in the acrylic appliance are used [14]. Several clinicians used a combination of thermoplastic splints and a mouth guard for infants [2]. To choose the form of treatment, we considered the effectiveness, stability and retention to maintain patient compliance. Additionally an appliance should prevent tissue damage and protect the oral cavity including the tongue, buccal cheek, teeth and gingiva. A hard acrylic resin splint with Adams clasps and ‘C’ clasps had good stability, retention and maintenance. We used a construction bite 5 mm in thickness, which was expected to help occlusal stability and harmony of myofascial function. In addition, the design that covered the maxillary arch and teeth protected the teeth and surrounding tissue. We focused the thickness of appliance, and the 5.0-mm thickness can accommodate some space for tongue posture and teeth covering resin will prevent the direct contact between teeth and tongue surface. The appliance also could be modified and adjusted for patients changing occlusion during adolescence. After one year follow up, the appliance was still functioning properly for tissue protection and habit prevention. Our special oral appliance is suitable to pediatric patients who can be cooperative in noninvasive dental care such as teeth brushing and impression. Both primary and permanent dentitions, including mixed dentition, are suitable for this oral device. As hard acrylic resin and proper clasps give sufficient retention, patients can retain their oral device easily. Furthermore, the elastic properties of clasps allow removing the device easily. Our young patient could wear and take off our oral device easily by himself after education. Additionally, the patient experienced no complications or discomfort. Our oral appliance design was effective for treatment of oral self-injury. 4. Conclusion In the present case report we emphasized the importance of a behavioral approach for treating self-mutilation in pediatric patient. Accurate examination and evaluation of unconscious biting habits must be performed. A customized oral appliance to prevent tongue biting was our treatment of choice. This technique was a more conservative therapy, which in turn made more invasive treatments unnecessary. Conflict of interest statement There are no conflicts of interest related to this article to declare. References [1] J. Limeres, J.F. Feijoo, F. Baluja, J.M. Seoane, M. Diniz, P. Diz, Oral self-injury: an update, Dent. Traumatol. 29 (1) (2013) 8–14. [2] E.M. Yasui, R.K. Kimura, A. Kawamura, S. Akiyama, I. Morisaki, A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: a case report, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 97 (4) (2004) 71–75. [3] M. Romero Maroto, C. Ruiz Duque, G. Vincent, I. Garcia Recuero, A. Romance, Management of oral lesions in Lesch–Nyham syndrome, J. Clin. Pediatr. Dent. 38 (3) (2014) 247–249. [4] L.F. Guimara˜es, M.E. Janini, A´.S.B. Vieira, L.C. Maia, L.G. Primo, Self-inflicted oral trauma in a baby with Moebius syndrome, J. Dent. Child. 74 (3) (2007) 224–227. [5] M.M.S. Schalka, M.S.N.P. Correˆa, A.L. Ciamponi, Congenital insensitivity-to-pain with anhidrosis (CIPA): a case report with 4-year follow-up, Oral Surg. Oral Med. Oral Pathol. Oral Radiol. Endod. 101 (6) (2006) 769–773.
Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013
G Model
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Please cite this article in press as: I.J. Kwon, et al., Successful treatment of self-inflicted tongue trauma patient using a special oral appliance, Int. J. Pediatr. Otorhinolaryngol. (2015), http://dx.doi.org/10.1016/j.ijporl.2015.08.013