A Case of Coffin-Lowry Syndrome

DOI
  • Kawakami Maiko
    Department of Pediatric Dentistry, Hiroshima University Graduate School of Biomedical Sciences
  • Suzuki Junji
    Department of Pediatric Dentistry, Hiroshima University Graduate School of Biomedical Sciences
  • Okada Mitsugi
    Department of Pediatric Dentistry, Hiroshima University Graduate School of Biomedical Sciences
  • Soda Yoshiko
    Department of Pediatric Dentistry, Hiroshima University Graduate School of Biomedical Sciences
  • Hayashi Fumiko
    Department of Pediatric Dentistry, Hiroshima University Graduate School of Biomedical Sciences
  • Miura Kazuo
    Department of Pediatric Dentistry, Hiroshima University Graduate School of Biomedical Sciences
  • Kozai Katsuyuki
    Clinic of Pediatric Dentistry, Hiroshima University Dental Hospital

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Other Title
  • 歯槽骨吸収を伴うCoffin-Lowry症候群の1症例

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Abstract

We encountered a boy with Coffin-Lowry syndrome which is an X-linked inherited disease. In the present study, we examined the situation by studying a cast model, using X-ray photographs including cephalometric analysis and the polymerase chain reaction (PCR) method for the detection of periodontal pathogens<BR>1. The patient was considered as being atypical case of the Coffin-Lowry syndrome with mental retardation, asmall stature, low weight, pectus carinatum, thoracolumber kyphosis and hyperplastic terminals of each finger.<BR>2. Lateral cephalometric analysis revealed that the patient showed less growth of the oral and maxillofacial region, and in particular, N-Ans was over 3 SD smaller than that of the control.<BR>3. Dental X-ray examinations showed noticeable root resorption of the upper central incisors and horizontal alveolar bone resorption around the lower central incisors. Furthermore, PCR analysis revealed that several putativeperiodontal pathogens, such as P. gingivalis and A. actinomycetemcomitans harbored in the oral cavity of the patient.

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