著しい開口障害を呈したRett syndromeの1例
Rett syndrome is found exclusively in young females, and is a progressive neurologic disease characterized by mental retardation, repeated hand-rubbing behavior, and related symptoms. We report a case of trismus that was treated in a patient with Rett syndrome. A 14-year-old girl (height, 120 cm; we...
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| Published in | 日本口腔外科学会雑誌 Vol. 59; no. 4; pp. 250 - 253 |
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| Main Authors | , , , , , |
| Format | Journal Article |
| Language | Japanese |
| Published |
社団法人 日本口腔外科学会
20.04.2013
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| Subjects | |
| Online Access | Get full text |
| ISSN | 0021-5163 2186-1579 |
| DOI | 10.5794/jjoms.59.250 |
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| Abstract | Rett syndrome is found exclusively in young females, and is a progressive neurologic disease characterized by mental retardation, repeated hand-rubbing behavior, and related symptoms. We report a case of trismus that was treated in a patient with Rett syndrome. A 14-year-old girl (height, 120 cm; weight 20 kg) presented at Asahi University Hospital in December 2006 because of trismus. Her medical history included epilepsy, patent ductus arteriosus, microcephaly, hypertension, and suspected Rett syndrome since 6 years of age. Trismus had been present for several years. Examination revealed mental retardation, scoliosis, manual stereotyped behavior, developmental disease of the feet, and difficulty in independently walking. Her face was symmetric, with a developed mandible angle. Flaredout maxillary front teeth with attrition and jaw opening of 15 mm were confirmed. Radiologic examination showed hypertrophy of both coronoid processes and slight adhesion in the left temporomandibular joint. Hypertrophy of both coronoid processes was diagnosed clinically, and both were removed. The intraoperative jaw opening was 45 mm. The jaw opening has been maintained at 32 mm as of 5 years 9 months after surgery. The case was considered to involve trimus caused by coronoid process enlargement due to development of the masseter and temporal muscles secondary to bruxism. |
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| AbstractList | Rett syndrome is found exclusively in young females, and is a progressive neurologic disease characterized by mental retardation, repeated hand-rubbing behavior, and related symptoms. We report a case of trismus that was treated in a patient with Rett syndrome. A 14-year-old girl (height, 120 cm; weight 20 kg) presented at Asahi University Hospital in December 2006 because of trismus. Her medical history included epilepsy, patent ductus arteriosus, microcephaly, hypertension, and suspected Rett syndrome since 6 years of age. Trismus had been present for several years. Examination revealed mental retardation, scoliosis, manual stereotyped behavior, developmental disease of the feet, and difficulty in independently walking. Her face was symmetric, with a developed mandible angle. Flaredout maxillary front teeth with attrition and jaw opening of 15 mm were confirmed. Radiologic examination showed hypertrophy of both coronoid processes and slight adhesion in the left temporomandibular joint. Hypertrophy of both coronoid processes was diagnosed clinically, and both were removed. The intraoperative jaw opening was 45 mm. The jaw opening has been maintained at 32 mm as of 5 years 9 months after surgery. The case was considered to involve trimus caused by coronoid process enlargement due to development of the masseter and temporal muscles secondary to bruxism. |
| Author | 細原, 政俊 住友, 伸一郎 田中, 四郎 玄, 景華 式守, 道夫 江原, 雄一 |
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| References | 13) 山田一尋：咬合挙上に伴うラット下顎骨咬筋深層付着部の変化に関する組織学的研究．日矯歯誌 44: 611-626, 1985. 1) Rett A : On a unusual brain atrophy syndrome in hyperammonemia in childhood. Wien Med Wochenschr. 116: 723-726, 1966. 17) 久賀陽子，古賀千尋，他：両側性筋突起過形成症の1例．日口外誌 43: 567-569, 1997. 3) 独） 国立精神・神経医療研究センター http://www.ncnp.go.jp/. Accessed April 10, 2008. 4) 保母妃美子，田村文誉，他：Rett症候群の口腔機能に関する研究．障害者歯科 32: 197, 2011. 11) 佐藤修一，川村 仁，他：両側筋突起過形成により顎運動障害を呈した2症例．口科誌 38: 219-228, 1989. 8) 吉田直人，吉田治彦，他：レット症候群患者の1例口へのかかわりと流涎，手もみ行動の変化について．障害者歯科 24: 456, 2003. 20) Fitzpatrick BN : Bilateral hyperplasia of the mandibular coronoid process. Oral Surg Oral Med Oral Pathol 29: 184-90, 1970. 18) Van Hoof RF and Bestig WF : coronoid process enlargement. Brit J Oral Surg 10: 339, 1973. 14) Yamada K, Hanada K, et al : U1trastructural studies on the migration mechanism of muscle attachment. J Dent Res 65: 792, 1986. 22) 加納浩之，小林正治，他：再手術を要した両側性筋突起過形成の1例．日顎変形誌 18: 49-54, 2008. 9) 工藤逸郎：第2章口腔外科診断法 2現症．上野 正，伊藤秀夫編：最新口腔外科学．第3版，医歯薬出版，東京，1993，15-28頁 6) 稲田朱美，落合智子，他：知的障害を伴う混合歯列期小児に行った咬合誘導の2症例．障害者歯科 29: 63-70, 2008. 21) Gerbino G, Bianchi SD, et al : Hyperplasia of the mandibular coronoid process: long-term follow-up after coronoidotomy. J Craniomaxillofac Surg 25: 169-73, 1997. 5) 堤香奈子，村上旬平，他：Rett症候群児・者の口腔に関する家族への意識調査．障害者歯科 32: 261, 2011. 12) Ginestet G, Dupuis A, et al : Constriction des machoires dorigine coronoid-malaire. Rev Stomat 58: 233-237, 1957. 7) 黒下礼奈，中川貴美子，他：ブラキシズムを伴うRett症候群患児への対応と咬筋活動分析．障害者歯科 28: 500, 2007. 15) Yamada K, Hanada K, et al : A light microscope autoradiographic study on the migration mechanism of muscle attachment during growth. J Bone ＆ Mineral Metab 5: 44-50, 1987. 19) Rivas PH : Reduction of mouth opening due to mandibular coronoid progess enlargement. Report of a case. Oral Surg Oral Med Oral Pathol 47: 131-134, 1979. 2) Amir RE, Van den Veyver IB, et al : Rett syndorome is caused by mutations in X-lined MECP2, encoding methyl-CpG-binding protein 2. Nat Genet 23: 185-188, 1999. 10) Shachelford RT and Brown WH : Osteochondroma of the coroniod process of the mandible. Surg Gynecol Obsteric 77: 51-54, 1943. 16) Yamada K, Hanada K, et al : An ultrastructural study of changes in the tendinous attachment of the deep layer of masseter muscle to the rat mandible during bite raising. Anat Ant 15: 44-50, 1987. |
| References_xml | – reference: 21) Gerbino G, Bianchi SD, et al : Hyperplasia of the mandibular coronoid process: long-term follow-up after coronoidotomy. J Craniomaxillofac Surg 25: 169-73, 1997. – reference: 16) Yamada K, Hanada K, et al : An ultrastructural study of changes in the tendinous attachment of the deep layer of masseter muscle to the rat mandible during bite raising. Anat Ant 15: 44-50, 1987. – reference: 9) 工藤逸郎：第2章口腔外科診断法 2現症．上野 正，伊藤秀夫編：最新口腔外科学．第3版，医歯薬出版，東京，1993，15-28頁． – reference: 5) 堤香奈子，村上旬平，他：Rett症候群児・者の口腔に関する家族への意識調査．障害者歯科 32: 261, 2011. – reference: 11) 佐藤修一，川村 仁，他：両側筋突起過形成により顎運動障害を呈した2症例．口科誌 38: 219-228, 1989. – reference: 7) 黒下礼奈，中川貴美子，他：ブラキシズムを伴うRett症候群患児への対応と咬筋活動分析．障害者歯科 28: 500, 2007. – reference: 17) 久賀陽子，古賀千尋，他：両側性筋突起過形成症の1例．日口外誌 43: 567-569, 1997. – reference: 13) 山田一尋：咬合挙上に伴うラット下顎骨咬筋深層付着部の変化に関する組織学的研究．日矯歯誌 44: 611-626, 1985. – reference: 12) Ginestet G, Dupuis A, et al : Constriction des machoires dorigine coronoid-malaire. Rev Stomat 58: 233-237, 1957. – reference: 8) 吉田直人，吉田治彦，他：レット症候群患者の1例口へのかかわりと流涎，手もみ行動の変化について．障害者歯科 24: 456, 2003. – reference: 18) Van Hoof RF and Bestig WF : coronoid process enlargement. Brit J Oral Surg 10: 339, 1973. – reference: 2) Amir RE, Van den Veyver IB, et al : Rett syndorome is caused by mutations in X-lined MECP2, encoding methyl-CpG-binding protein 2. Nat Genet 23: 185-188, 1999. – reference: 4) 保母妃美子，田村文誉，他：Rett症候群の口腔機能に関する研究．障害者歯科 32: 197, 2011. – reference: 6) 稲田朱美，落合智子，他：知的障害を伴う混合歯列期小児に行った咬合誘導の2症例．障害者歯科 29: 63-70, 2008. – reference: 20) Fitzpatrick BN : Bilateral hyperplasia of the mandibular coronoid process. Oral Surg Oral Med Oral Pathol 29: 184-90, 1970. – reference: 10) Shachelford RT and Brown WH : Osteochondroma of the coroniod process of the mandible. Surg Gynecol Obsteric 77: 51-54, 1943. – reference: 19) Rivas PH : Reduction of mouth opening due to mandibular coronoid progess enlargement. Report of a case. Oral Surg Oral Med Oral Pathol 47: 131-134, 1979. – reference: 22) 加納浩之，小林正治，他：再手術を要した両側性筋突起過形成の1例．日顎変形誌 18: 49-54, 2008. – reference: 1) Rett A : On a unusual brain atrophy syndrome in hyperammonemia in childhood. Wien Med Wochenschr. 116: 723-726, 1966. – reference: 3) 独） 国立精神・神経医療研究センター http://www.ncnp.go.jp/. Accessed April 10, 2008. – reference: 14) Yamada K, Hanada K, et al : U1trastructural studies on the migration mechanism of muscle attachment. J Dent Res 65: 792, 1986. – reference: 15) Yamada K, Hanada K, et al : A light microscope autoradiographic study on the migration mechanism of muscle attachment during growth. J Bone ＆ Mineral Metab 5: 44-50, 1987. |
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| SubjectTerms | Rett 症候群 筋突起過形成症 開口障害 |
| Title | 著しい開口障害を呈したRett syndromeの1例 |
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