증 례 자폐환자의자해로인한구강내손상 : 증례보고 지은혜 이효설 최형준 김성오 최병재 손흥규 이제호 연세대학교치과대학소아치과학교실 Abstract SELF-INJURIOUS BEHAVIOR IN A PATIENT WITH AUTISM : A CASE REPORT Eunhye Ji, Hyo-seol Lee, Hyung-Jun Choi, Seong-Oh Kim, Byung-Jai Choi, Heung-Kyu Son, Jae-Ho Lee Department of Pediatric Dentistry, College of Dentistry, Yonsei University Self-injurious behavior (SIB) has been defined as the deliberate destruction or alteration of body tissue without conscious suicidal intent. It occurs in conjunction with a variety of psychiatric disorders as well as various developmental disabilities and some syndromes. The behavior is destructive and causes concern and distress to all involved in the care and treatment of the affected individual. A 13-year-old girl with autism, mental retardation and delayed development was reffered from her pediatrician because of severe and painful lower lip biting. An intraoral examination revealed a diffuse swelling of lower lip. It was covered with necrotic slough and the ulcer and scarring of the lower lip was observed. We chose to use an oral removable prosthesis for Conservative treatment. It was decided to use a soft silicone mouthguard in the maxillary arch. Initially, she could not tolerate the appliance inside her mouth but soon adapted with the appliance. After one month, she lost the mouth guard and started lip biting. So we made mouth guard again. There are no standard methods for preventing self-injurious behavior in a patient who is developmentally disabled. Appropriate preventive methods must be developed for each individual patient based on close observation and clinical findings. Behavior modification techniques, pharmacological treatment, extraction of teeth, orthognathic surgery and intra/extra oral appliances can be performed for adjust self-injurious behavior. A suitable oral guard could be tried initially before employing more invasive approaches. Key words : Autism, self-injurious behavior(sib), mouthguard Ⅰ. 서론 교신저자 : 이제호 120-752 서울특별시서대문구신촌동 134 연세대학교치과대학소아치과학교실 Tel: 02-2228-3173 Fax: 02-392-7420 E-mail: leejh@yuhs.ac 자해행위는자살의의도는없이자신의신체일부를무의식적이거나고의로손상시키는것 1) 으로미세한손상에서부터, 불구를유발할수있는손상까지다양하게나타난다. 흔히자해행위는머리및몸때리기, 피부절단혹은손가락깨물기등의형태로나타나며눈, 성기및구강내자해행 원고접수일 : 2012.05.01 / 원고최종수정일 : 2012.05.22 / 원고채택일 : 2012.06.05-10 -
대한장애인치과학회지 8(1) 2012 위가포함된다. 자해행위의유병율에관해정확히알려진바는없으나여성에서좀더호발하며일반인의경우 100,000명중 750 명정도의비율로추정되고정신지체가있는환자의경우 7.7% 에서 22.8% 의비율로나타나며심한정신지체가있는환자의경우 40% 까지그빈도가증가하는것으로알려져있다 2,3). 자해의원인은명확히밝혀져있지않고매우복잡한것으로생각되고있는데추정되는원인으로는 Cornelia de Lange 증후군 4,5), Gilles de La Tourettes 증후군 6), Lesch-Nyhan 증후군 4) 같은유전질환이있는경우, 선천적으로통증을느끼지못하는감각신경병 4) 을앓는경우, 뇌염같은감염성질환 4,7) 에걸린경우, 자폐 2,8,9) 나정신지체 5) 가있는경우에발생하는것으로알려져있다. 이와같이자해증상은발육장애나여러증후군과관련하여다양한정신질환관연관되어나타난다. 이러한자해행위는대부분통증을느끼면서도지속적으로스스로의신체에손상을가하기때문에이를막기위한적극적인개입이필요하고다양한분야에서의접근이필요하다. 본증례에서는정신지체를동반한자폐증환아로서자해로인한하순의심각한손상을입었으나 soft mouthguard 를이용하여증상의악화를막을수있었고양호한하순의치유과정을보였다. Ⅱ. 증례 13세 9개월된여아로입술을계속깨무는증상이나아지지않는다는주소로내원하였으며전신병력상자폐, 정신지체, 발육지연등을보이고있었고이로인해소아정신과에서계속치료를받고있는상태였다. 본환아는이전에도 가족등주변사람에게폭력을행사하거나자해행위로인하여수차례입원병력이있었고본원소아치과에내원전에도입술을깨무는증상으로인해소아청소년과에서약처방을받고 2주간소독처치를받았으나증상이나아지지않고더심해지는양상을보였다고하였다. 구강내검사시하순의전반적인부종이있었으며괴사성조직으로덮혀있고궤양이형성되어있는것을볼수있었다 (Fig. 1). 자해로인한하순손상의방지를위한보존적처치로상악에 soft silicone mouthguard를계획하고인상채득을시행하였다. 인상채득시환아의협조를구할수없어환아를 unit chair에누운상태로강제속박을하고개구기를이용하여강제로입을벌린후인상채득을하였다. 다음날 mouth guard를제작하여장착하였고 mouthguard의삼킴을방지하기위해치실을묶어주었다. Mouthguard 장착시처음에는환아가거부하였으나주변으로관심을돌리자곧적응하였고보호자에따르면장치를잘사용하였다고하였다. 1개월검진시장치를분실하고다시입술을깨무는증상이나타나장치를재제작하였고현재까지잘사용하고있다. Ⅲ. 총괄및고찰자폐성장애는 1934년 Kanner에의해최초로소개된질환으로자폐증상의발현시기는 36개월이전이며사회적대인관계영역의문제에서감정표현의장애, 대인관계에서의무관심, 부적절한고착등을나타내며기이한이상행동영역에서과도한불안, 자기파괴적행동, 이상언어행동등을보인다. 또한주변환경에과도하게저항하면서공격적인행동이나다른부적응행동을보이기도하는것으로알려져있다. 이러한자폐환자는 10,000명중 1.3~16명의비율로발생하고자폐환자의 70% 가자해를하는것으로보고 Fig. 1. Ulcer, necrotic tissue from the lower lip at the initial examination. - 11 -
Fig. 2. Soft silicone mouthguard with dental floss. Fig. 3. Follow-up after 1 month. Almost wound healing following appliance therapy but starting biting lower lip after lost the appliance. 되고있다 10,11). 자폐환자의경우의사소통의어려움과통증인지의변화로자해행위를차단하기가어렵다. 본환자의경우정신지체를동반한자폐로진단을받았고약물요법을위해소아정신과에서약처방을계속받고있는상태였으나자해증상이개선되지않아추가적인다른처치가필요할것으로생각되었다. 자해행위의치료방법으로는약물을이용한치료방법, 행동조절을통한조절방법, 물리적억제방법등이사용될수있고구강내손상을예방하기위해구강내또는구강외장치를사용하거나악교정수술, 관련치아의발거등이제안되고있다. 약물을이용한치료의경우주로 dopaminergic, opiate, serotonin system에대한연구가많으나도파민수용체나세로토닌수용체에만관여하는약제의경우한계가존재한다. 세로토닌-도파민길항제들은다른항정신병약물에비해비교적일관된효과들이보고되고있다 12). 자해행위를대체할만한적절한행동을유도하거나각개인에맞는강화인자를부여하는등의긍정적강화와같은행동조절을이용한방법이이용될수있으나이는증상이경미한자해증상을보이는경우에는가능하다. 그러나자폐환자의경우의사소통의어려움이있고개인에맞는적절한강화인자를찾는것이어렵다 2). 긍정적강화를통한행동개선을시도할경우적절한행동을했을때 30분간격으로칭찬을하고보상인자를주는것이필요하고보호자의시간투자와인내심이필요하다. 물리적속박이필요할경우최소한의억제를하는동시에효율적인보호를하는것이중요한데억제정도는자해의심각성정도에따라결정되어야한다 2). 팔이나머리에자해를가하는경우헬멧이나 arm-board, 속박용의복등을이 용한신체적속박을사용할수있으나이역시자해행위가심한경우에는큰효과가없는것으로보고되고있다 13). 구강내장치나구강외장치의경우 bite block을이용하여전치부개방교합을유도하거나 facemask, chin-cup 같은장치와함께구내, 외탄성고무를이용한장치, 교정용 band 나기성금속관에 soldering한 lip bumper, 혀와입술을보호할수있는다양한형태의 shield 등을사용할수있다 14). 또한가장흔하게사용할수있는구강내장치로 soft mouthguard를사용할수있는데 mouthguard의경우제작이간편하고환자혹은보호자가장치를제거하는것이용이하다. 또한장치의청소와구강위생에도유리하며장치의파손으로인한 2차적인구강내손상의염려가없어안전하다는장점이있다 15). 그러나심한치주질환이있는경우사용이어렵고장치의표면이쉽게마모되어재제작이필요할수있으며치열이계속변화하는어린이의경우재제작이계속필요하다는단점이있다. 또한제작과정중환자의큰교합력으로인해인상채득이어려울수있고유지력과견고성이떨어져효과적인자해예방에한계가있을수있다 16). 다른자해예방방법으로보존적인술식들이실패하는경우손상을야기하는해당치아를모두발거하는방법 4) 이사용될수있는데 Rashid 와 Yusuf 17) 에따르면전치아의발거가극단적이기는하나완전하게자해행위를차단할수있는방법이라고하였고보존적인방법들은연령증가와함께증상이심해질수록성공률이낮아져해당치아의발거가필요하다는주장 12) 도있다. 또한상, 하악골의악교정수술을이용하여인위적으로개방교합을형성시켜주는방법을사용할수있는데 Macpherson 18) 등은악교정수술을이용하여만족스러운 - 12 -
대한장애인치과학회지 8(1) 2012 결과를얻었다고보고하기도하였다. 본증례의환자는우선가장보존적인방법이라고생각되는 mouthguard의제작에대해보호자에게설명하였고이장치가실패할경우다른종류의가철성장치제작을고려해보기로하였다. 보호자에게 mouthguard의탈착법및관리에대해설명을하고환아의공격적인행동으로인해 mouthguard의유지가어렵거나환아스스로장치를제거할경우다시내원할것을설명드렸으나검진날짜에는내원을하지않았고 1개월뒤장치분실로인해재내원시하순의상처는치유가된상태였고환아의거부반응도미약하여장치를잘사용했음을확인할수있었다. 환자의병력상자해나폭력으로인한수차례의입원경험이있고증상의완화와악화가반복될수있어장기적인관찰이필요하며장치뿐아니라약물치료가계속병행되어야할것으로생각된다. 자해행위를방지하기위한표준화된방법은아직정해져있지않으나각환자의자해양상및정도에따라적절한접근이필요하다. 가능한약물치료와행동조절, 교상방지장치등을이용한보존적인방법이시행된후효과가없을경우치아발거나악교정수술등의방법이고려될수있을것이다. 유전질환이나정신지체, 자폐등으로인한자해환자들에대한관심이높아지면서이러한환자들에게치과의사로서의역할또한증가하고있다. 따라서치과의사로서구강영역의자해행위에대한원인및손상의원인을정확히파악하고적절한치료법을찾으려는노력이필요할것으로생각된다. Ⅳ. 요약자폐증및정신지체가있는환자의입술깨물기증상을 mouthguard를사용하여성공적으로조절하였다. 치과적으로접근가능한구강내자해증상의치료는가철성혹은고정성장치를이용하거나관련치아의발치, 악교정수술등을시도할수있으며각환자에맞는적절한치료법을선택하는것이중요하다. 자해행동이일시적으로개선되었다해도재발의가능성이높으므로장기적인관리와소아정신과와의협진하에정신심리학적환경개선및약물치료가병행되어야할것이다. 참고문헌 1. Lucavechi T, Barberia E, Maroto M, et al.: Selfinjurious behavior in a patient with mental retardation: review of the literature and a case report. Quintessence Int, 38:e393-398, 2007. 2. Medina AC, Sogbe R, Gomez-Rey AM, et al.: Factitial oral lesions in an autistic paediatric patient. Int J Paediatr Dent, 13:130-137, 2003. 3. Saemundsson SR, Roberts MW: Oral self-injurious behavior in the developmentally disabled: review and a case. ASDC J Dent Child, 64:205-209, 228, 1997. 4. Fardi K, Topouzelis N, Kotsanos N: Lesch- Nyhan syndrome: a preventive approach to selfmutilation. Int J Paediatr Dent, 13:51-56, 2003. 5. Cauwels RG, Martens LC: Self-mutilation behaviour in Lesch-Nyhan syndrome. J Oral Pathol Med, 34:573-575, 2005. 6. Shimoyama T, Horie N, Kato T, et al.: Tourette s syndrome with rapid deterioration by self-mutilation of the upper lip. J Clin Pediatr Dent, 27:177-180, 2003. 7. Chen LR, Liu JF: Successful treatment of selfinflicted oral mutilation using an acrylic splint retained by a head gear. Pediatr Dent, 18:408-410, 1996. 8. Johnson CD, Matt MK, Dennison D, et al.: Preventing factitious gingival injury in an autistic patient. J Am Dent Assoc, 127:244-247, 1996. 9. Mobily PR, Herr KA, Kelley LS: Cognitive-behavioral techniques to reduce pain: a validation study. Int J Nurs Stud, 30:537-548, 1993. 10. Wing L: The definition and prevalence of autism: A review. Eur Child Adolesc Psychiatry, 2:61-74, 1993. 11. Howlin P: Behavioural techniques to reduce selfinjurious behaviour in children with autism. Acta Paedopsychiatr, 56:75-84, 1993. 12. 김지희, 최병재, 김성오, 최형준, 손흥규, 이제호 : Lesch-Nyhan 증후군환아의자해에의한혀의외상. 대한소아치과학회지, 35:532-537, 2008. 13. Eguchi S, Tokioka T, Motoyoshi A, et al.: A selfcontrollable mask with helmet to prevent self finger-mutilation in the Lesch-Nyhan syndrome. Arch Phys Med Rehabil, 75:709-710, 1994. 14. Jeong TS, Lee JH, Kim S, et al.: A preventive approach to oral self-mutilation in Lesch-Nyhan syndrome: a case report. Pediatr Dent, 28:341-344, 2006. 15. Finger ST, Duperon DF: The management of self-inflicted oral trauma secondary to encephalitis: a clinical report. ASDC J Dent Child, 58:60- - 13 -
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