先天性無痛無汗症児に発症した下顎骨骨髄炎 : 歯髄診断に透過型光電脈波法(TLP)を応用した1例  [in Japanese] Mandibular Osteomyelitis in the Presence of Congenital Insensitivity to Pain with Anhidrosis : A case in which transmitted-light plethysmography (TLP) was used for pulp diagnosis  [in Japanese]

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Author(s)

    • 三輪 全三 MIWA Zenzo
    • 東京医科歯科大学大学院,医歯学総合研究科,口腔機能再構築学系,口腔機能発育学講座, 小児歯科学分野 Section of Developmental Oral Health Science, Department of Orofacial Development and Functions, Division of Oral Health Sciences, Graduate School of Medical and Dental Science, Tokyo Medical and Dental University
    • 柿野 聡子 KAKINO Satoko
    • 東京医科歯科大学大学院,医歯学総合研究科,口腔機能再構築学系,口腔機能発育学講座, 小児歯科学分野 Section of Developmental Oral Health Science, Department of Orofacial Development and Functions, Division of Oral Health Sciences, Graduate School of Medical and Dental Science, Tokyo Medical and Dental University
    • 上原 奈緒子 UEHARA Naoko
    • 東京医科歯科大学大学院,医歯学総合研究科,口腔機能再構築学系,口腔機能発育学講座, 小児歯科学分野 Section of Developmental Oral Health Science, Department of Orofacial Development and Functions, Division of Oral Health Sciences, Graduate School of Medical and Dental Science, Tokyo Medical and Dental University
    • 土橋 なつみ TSUCHIHASHI Natsumi
    • 東京医科歯科大学大学院,医歯学総合研究科,口腔機能再構築学系,口腔機能発育学講座, 小児歯科学分野 Section of Developmental Oral Health Science, Department of Orofacial Development and Functions, Division of Oral Health Sciences, Graduate School of Medical and Dental Science, Tokyo Medical and Dental University
    • 今村 由紀 IMAMURA Yuki
    • 東京医科歯科大学大学院,医歯学総合研究科,口腔機能再構築学系,口腔機能発育学講座, 小児歯科学分野 Section of Developmental Oral Health Science, Department of Orofacial Development and Functions, Division of Oral Health Sciences, Graduate School of Medical and Dental Science, Tokyo Medical and Dental University
    • 黒原 一人 KUROHARA Kazuto
    • 東京医科歯科大学大学院,医歯学総合研究科,顎顔面頸部機能再建学系,顎顔面機能修復学講座,顎顔面外科学分野 Section of Maxillofacial Surgery, Department of Maxillofacial Reconstructionand Function, Division of Maxillofacial/Neck Reconstruction Graduate School of Medical and Dental Science, Tokyo Medical and Dental University
    • 高木 裕三 TAKAGI Yuzo
    • 東京医科歯科大学大学院,医歯学総合研究科,口腔機能再構築学系,口腔機能発育学講座, 小児歯科学分野 Section of Developmental Oral Health Science, Department of Orofacial Development and Functions, Division of Oral Health Sciences, Graduate School of Medical and Dental Science, Tokyo Medical and Dental University

Abstract

先天性無痛無汗症(CIPA)は遺伝性感覚・自律神経ニューロパチーのⅣ型であり,精神発達遅滞を伴う常染色体劣性遺伝性疾患である。無随のC 線維や細い有随のA<i>δ </i>線維が欠如するために,痛覚と発汗機能が欠如している。しかし,太い有髄のA<i>β </i>線維は存在しており触覚などはあるとされている。自傷行為による舌,口唇や手指の咬傷,歯の自己抜去まれに下顎骨骨髄炎などが多くみられ,歯科的な治療,ケアが重要な疾患である。今回,無痛無汗症と診断されている11 歳5 か月の女児で歯の自己抜去が原因で下顎骨骨髄炎を発症し,2 か月間に6 歯を喪失し同部位の腐骨除去手術を行った症例を経験した。術前に当科で腐骨の除去範囲および抜去する歯の決定のために歯髄の生死診断を行った。EPT(歯髄電気診)では,測定したすべての歯が無反応であり,歯髄診断が無効であったが,これは本疾患における歯髄感覚の伝達機構(C 線維およびA<i>δ </i>線維)が欠如するためと思われた。これに代わって,歯髄血流の有無が測定可能なTLP(透過型光電脈波)法を用いて歯髄血流の有無を測定した。その結果,腐骨と境界領域にある下顎右側第2 小臼歯は血流が確認でき生活歯と診断され,抜去せずに保存したところ,手術より10 か月後における当該歯と下顎骨のエックス線検査およびTLP 法による検査では予後の経過は良好であった。

Congenital insensitivity to pain with anhidrosis (CIPA) is a type IV hereditary sensory and autonomic neuropathy associated with an autosomal recessive genetic disease with mental retardation.CIPA individuals lack pain sensation and perspiration function due to the absence of unmyelinated Cand thin myelinated A<i>δ </i>fibers. However, since thick myelinated A<i>β </i>fibers are present, tactile sensation is considered to be functioning. Patients with this disease show bite wounds to the tongue, lips,and fingers due to self-mutilation, and conduct self tooth extraction that occasionally causes mandibular osteomyelitis ; therefore, it is a disease for which dental treatment and care are important.In this study, an 11-year and 5-month-old girl, diagnosed with CIPA, exhibited mandibular osteomyelitis due to self tooth extraction. Six teeth were lost in 2 months, and sequestrectomy was performed in the area.Before surgery, dental pulp vitality was assessed in our department to determine the removal range of mandibular sequesters and indicate which teeth required extraction. Since all of the teeth measured were non-responsive to a conventional electric pulp test (EPT) this dental pulp vitality diagnostic test was considered useless, and the presence of pulp pulse waves was examined using transmitted-light plethysmography (TLP), in which the presence of pulpal blood flow could be objectively measured.As a result, the presence of pulp pulse waves in the mandibular right second premolar, located in the border region of sequesters, was confirmed. The tooth was diagnosed as vital, and was preserved without performing tooth extraction. TLP appears to be very useful for the diagnosis of teeth in patients with congenital insensitivity to pain with anhidrosis. At 10 months after surgery, a favorable clinical course has been shown based on TLP and X-ray examinations of the preserved tooth and mandibular bone.

Journal

  • The Japanese Journal of Pediatric Dentistry

    The Japanese Journal of Pediatric Dentistry 49(1), 41-46, 2011-03-25

    Japanese Society of Pediatric Dentistry

References:  14

Codes

  • NII Article ID (NAID)
    10028185996
  • NII NACSIS-CAT ID (NCID)
    AN00116228
  • Text Lang
    JPN
  • Article Type
    NOT
  • ISSN
    05831199
  • Data Source
    CJP  J-STAGE 
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