Treatment of Labial Ulcer Induced by Twisted and Delayed Eruption of Upper Right First Incisor in Patient with 9p Trisomy Syndrome：A Case Report

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  • TAKAHASHI Atsushi
    Clinics of Dentistry for the Disabled, Tohoku University Hospital
  • NAGANUMA Yukihiro
    Clinics of Dentistry for the Disabled, Tohoku University Hospital
  • MATSUZAKA Kumi
    Clinics of Dentistry for the Disabled, Tohoku University Hospital
  • KITAOKA Aki
    Clinics of Dentistry for the Disabled, Tohoku University Hospital
  • SASAKI Keiichi
    Clinics of Dentistry for the Disabled, Tohoku University Hospital
  • IGARI Kazuko
    Clinics of Dentistry for the Disabled, Tohoku University Hospital

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Other Title
  • 上顎中切歯萌出異常による慢性的な口唇粘膜潰瘍への対応に苦慮した9pトリソミー症候群の1例

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<p>The 9p trisomy syndrome is a chromosomal abnormality that is clinically characterized by short stature, microcephaly, and delayed bone development, including craniofacial and digital dysmorphic features, as well as varying degrees of intellectual disability. We describe here a 22-year-old woman with 9p trisomy syndrome who exhibited an upper labial ulcer induced by twisted and delayed eruption of the upper right first incisor. She demonstrated a high-arched palate, shortened superior labial frenum, malocclusion, three supernumerary teeth, and three impacted teeth, as well as moderate periodontitis. She had a clinical history of hypocalcemia induced by pseudohypoparathyroidism at the age of 6, resulting in an enamel hypoplasia line on both first upper premolars.</p><p>To treat the ulcer, it seemed effective for this patient with disability to modify the shape of the upper incisor, in order to relive pressure between the upper lip and upper incisor. When the ulcer had recovered, improvement of the twisted upper incisor via orthodontic therapy could follow. To prevent aggravation, maintenance of oral hygiene by her guardians was also needed.</p><p>After several attempts at training her to adapt to dental care, we determined that her cooperation might be inadequate for dental treatment. We provided a glass ionomer cement covering to widen the surface of the upper first incisor and asked her guardian to keep her mouth clean by daily brushing. At first, the ulcer showed repeated episodes of improvement and aggravation. Thus, to further reduce the pressure between the lip and teeth we again covered both the upper first and second incisors with glass ionomer cement. As a result, the ulcer recovered and did not recur. Although orthodontic therapy could not be applied because of the patient’s inadequate cooperation, she accepted professional mechanical cleaning of the teeth. Her oral hygiene gradually improved. Regular dental check-ups are necessary for maintenance of oral hygiene and for early detection of oral disease.</p>

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