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Clinical and Laboratory Studies of Severe Periodontal Disease in an Adolescent Associated With Hypophosphatasia. A Case Report
Author(s) -
Watanabe Hisashi,
Umeda Makoto,
Seki Tomoko,
Ishikawa Isao
Publication year - 1993
Publication title -
journal of periodontology
Language(s) - English
Resource type - Journals
SCImago Journal Rank - 2.036
H-Index - 156
eISSN - 1943-3670
pISSN - 0022-3492
DOI - 10.1902/jop.1993.64.3.174
Subject(s) - hypophosphatasia , medicine , dental alveolus , deciduous teeth , aggressive periodontitis , porphyromonas gingivalis , cementum , urine , molar , periodontitis , alkaline phosphatase , dentistry , chemistry , dentin , biochemistry , enzyme
H ypophosphatasia is a congenital disease characterized by defective bone mineralization, deficiency of alkaline phosphatase (ALP) activity, increased excretion of the phosphoethanolamine (PEA) in the urine, and premature loss of the deciduous teeth. A male hypophosphatasia patient (aged 15 years 6 months) with premature exfoliation of the deciduous teeth was referred to our hospital because of severe periodontal destruction in the permanent dentition. Blood and urine tests as well as oral and periodontal examinations were performed. Serum antibody titers against 7 periodontopathic bacteria by the enzyme‐linked immunosorbent assay (ELISA), monocyte and neutrophil chemotaxis measurements, and cellular immunity tests were also performed. Low levels of ALP in serum and PEA in the urine were found. Monocyte and neutrophil chemotaxis exhibited normal values. Slightly depressed CD2+, CD3+, and CD4+ and slightly elevated activity of NK cells were found. An elevated level of serum antibody to Porphyromonas gingivalis was observed. Oral radiographic examination showed a mirror pattern of alveolar bone loss which is similar to that seen in localized juvenile periodontitis. Periodontal treatment of this patient was carried out for 4 years. The severely affected sites, the lower right and left first molars and the upper right first molar, had to be extracted. However, the other sites were well maintained. The serum IgG level against P. gingivalis was decreased after 4 years of periodontal treatment. Infection with P. gingivalis was suspected to be associated with the destruction of this patient's hypophosphatasia, but other dental abnormalities such as abnormal enamel, dentin, and cementum formation may also have contributed to the periodontal pathology. J Periodontol 1993;64:174–180 .