Oral self-inflicted accidental trauma in patients with neurological disorders: a case report of dental management in infants with cerebellar hypoplasia

Self-inflicted oral injuries, accidental or otherwise, can cause major consequences. Measures need to be taken to protect individuals from chronic self-injurious behaviour; however, there are no official guidelines on the subject. The purpose of this article is to show the case of a 1-year-old patient with neurological disorders who, following the eruption of deciduous teeth, had self-inflicted a traumatic ulcer on his tongue and lower lip. Following a multidisciplinary approach involving several operating units of our hospital to make a diagnosis, an oral device was designed to completely cover the dental elements to prevent recurrence of the trauma and to prevent further worsening of the injuries already caused. The purpose of this work is to demonstrate that although the surgical approach, such as extraction of the dental elements, may be the quickest solution in situations similar to the one presented, the high biological cost and irreversibility of the result lead to seeking alternatives and more conservative solutions such as the one described.

Self-inflicted trauma; Oral cavity; Neurological

[1] Pigno MA, Funk JJ. Prevention of tongue biting with a removable oral device: a clinical report. The Journal of Prosthetic Dentistry. 2000; 83: 508–510.

[2] Kocsard E, Schwarz L, Stephen BS, D’abrera VSE. Morsicatio buccarum. The British Journal of Dermatology. 1962; 74: 454–457.

[3] Frikha F, Bahloul E, Mesrati H, Sellami K, Amouri M, Turki H. Morsicatio buccarum et labiorum. Annals of Dermatology and Venereology. 2019; 146: 594–595. (In French)

[4] Fardi K, Topouzelia N, Kotsanos N. Lesch-Nyham syndrome: a preventive approach to self-mutilation. International Journal of Paediatric Dentistry. 2003; 13: 51–56.

[5] Cusumano FJ, Penna KJ, Panossian G. Prevention of self-mutilation in patients with Lesch-Nyhan syndrome: review of literature. ASDC Journal of Dentistry for Children. 2001; 68: 175–178.

[6] Cehreli ZC, Olmez S. The use of a special mouthguard in the management of oral injury self-inflicted by a 4-year-old child. International Journal of Paediatric Dentistry. 1996; 6: 277–281.

[7] Limeres J, Feijoo JF, Baluja F, Seoane JM, Diniz M, Diz P. Oral self-injury: an update. Dental Traumatology. 2013; 29: 8–14.

[8] Kwon IJ, Kim SM, Park HK, Myoung H, Lee JH, Lee SK. Successful treatment of self-inflicted tongue trauma patient using a special oral appliance. International Journal of Pediatric Otorhinolaryngology. 2015; 79: 1938–1941.

[9] Siragusa M, Ferri R, Russo R, Lentini M, Schepis C. Self-inflicted lesions of the mouth and lips in mentally retarded young subjects. European Journal of Dermatology. 2013; 23: 843–848.

[10] Sorghabi W. Oral self-mutilation: a repetitive tongue trauma. Dutch Journal of Dentistry (NTVT). 2020; 127: 231–235. (In Dutch)

[11] Zaenglein AL, Chang MW, Meehan SA, Axelrod FB, Orlow SJ. Extensive Riga-Fede disease of the lip and tongue. Journal of the American Academy of Dermatology. 2002; 47: 445–447.

[12] Wiechers C, Buchenau W, Arand J, Oertel A, Peters K, Müller-Hagedorn S, et al. Mandibular growth in infants with Robin sequence treated with the Tübingen palatal plate. Head & Face Medicine. 2019; 15: 17.

[13] Yasui EM, Kimura RK, Kawamura A, Akiyama S, Morisaki I. A modified oral screen appliance to prevent self-inflicted oral trauma in an infant with cerebral palsy: a case report. Oral Surgery, Oral Medicine, Oral Pathology, Oral Radiology, and Endodontology. 2004; 97: 471–475.

[14] Pitak-Arnnop P, Auychai P, Subbalekha K, Malakul A, Meningaud J, Neff A. An approach to a repeated self-biting tongue injury in a toddler. Paediatric Archives. 2021; 28: 576–579. (In French)

[15] Shapira J, Birenboim R, Shoshani M, Abdel‐Kader A, Behar O, Moskovitz M, et al. Overcoming the oral aspects of ­self-mutilation: description of a method. Special Care in Dentistry. 2016; 36: 282–287.

[16] Mark DG, Granquist EJ. Are prophylactic oral antibiotics indicated for the treatment of intraoral wounds? Annals of Emergency Medicine. 2008; 52: 368–372.

[17] Ragazzini G, Delucchi A, Calcagno E, Servetto R, Denotti G. A modified intraoral resin mouthguard to prevent self-mutilations in Lesch-Nyhan patients. International Journal of Dentistry. 2014; 2014: 396830.

[18] Poets CF, Maas C, Buchenau W, Arand J, Vierzig A, Braumann B, et al. Multicenter study on the effectiveness of the pre-epiglottic baton plate for airway obstruction and feeding problems in Robin sequence. Orphanet Journal of Rare Diseases. 2017; 12: 46.

[19] Tinastepe N, Küçük BB, Oral K. Botulinum toxin for the treatment of bruxism. CRANIO®. 2015; 33: 292–299.

[20] Lee SJ, McCall WD, Kim YK, Chung SC, Chung JW. Effect of botulinum toxin injection on nocturnal bruxism: a randomized controlled trial. American Journal of Physical Medicine & Rehabilitation. 2010; 89: 16–23.

[21] Long H, Liao Z, Wang Y, Liao L, Lai W. Efficacy of botulinum toxins on bruxism: an evidence-based review. International Dental Journal. 2012; 62: 1–5.

[22] Nadelman P, Bedran N, Magno MB, Masterson D, de Castro ACR, Maia LC. Premature loss of primary anterior teeth and its consequences to primary dental arch and speech pattern: a systematic review and meta-analysis. International Journal of Paediatric Dentistry. 2020; 30: 687–712.

[23] Yamanaka R, Soga Y, Moriya Y, Okui A, Takeuchi T, Sato K, et al. Management of lacerated and swollen tongue after convulsive seizure with a mouth protector: interprofessional collaboration including dentists in intensive care. Acta Medica Okayama. 2014; 68: 375–378.

[24] Kiat-Amnuay S, Koh SH, Powner DJ. An occlusal guard for preventing and treating self-inflicted tongue trauma in a comatose patient: a clinical report. The Journal of Prosthetic Dentistry. 2008; 99: 421–424.

[25] Bartkowska P, Komisarek O. Scar management in patients after cleft lip repair—systematic review cleft lip scar management. Journal of Cosmetic Dermatology. 2020; 19: 1866–1876.

[26] Sitzia E, Santarsiero S, Tucci FM, De Vincentiis G, Galeotti A, Festa P. Balloon dilation and rapid maxillary expansion: a novel combination treatment for congenital nasal pyriform aperture stenosis in an infant. Italian Journal of Pediatrics. 2021; 47: 189.

[27] Letieri ADS, Martins ML, Ferreira Filho JCC, Agostini M, de Araújo Castro GFB. Gradual deprogramming of self-inflicted oral trauma habit in a child with cerebral palsy. Journal of Dentistry for Children. 2021; 88: 58–61.