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Effects of Enzyme Replacement Therapy for Primary Teeth in a Patient with Infantile Hypophosphatasia

  • Rena Okawa1,*,
  • Saaya Matayoshi1
  • Rina Kariya1
  • Yuko Ogaya1
  • Ryota Nomura1
  • Kazuhiko Nakano1

1Department of Pediatric Dentistry, Osaka University Graduate School of Dentistry, Osaka, Japan

DOI: 10.17796/1053-4625-44.5.9 Vol.44,Issue 5,September 2020 pp.348-351

Published: 01 September 2020

*Corresponding Author(s): Rena Okawa E-mail: rokawa@dent.osaka-u.ac.jp

Abstract

Hypophosphatasia (HPP) is a skeletal disorder characterized by hypomineralization of bone, with early exfoliation of primary teeth. Alkaline phosphatase enzyme replacement therapy (ERT) has been shown to improve bone hypomineralization for patients with HPP, although its dental effects are unknown. A 20-monthold Japanese boy diagnosed with infantile HPP was referred to our clinic because of early exfoliation of primary teeth. The patient had been followed by a pediatrician since the age of 3 months, due to slow weight gain. At the age of 12 months, primary incisors showed sudden exfoliation; at the age of 19 months, a diagnosis of HPP was made based on bone and dental manifestations. ERT was initiated at the age of 21 months. The patient demonstrated stable periodontal conditions of primary molars that erupted after initiation of ERT, due to improved alveolar bone and tooth mineralization. Thus, ERT may improve both dental and systemic conditions.

Keywords

Hypophosphatasia; Enzyme replacement therapy; Primary teeth; Early exfoliation; Poor weight gain

Cite and Share

Rena Okawa,Saaya Matayoshi,Rina Kariya,Yuko Ogaya,Ryota Nomura,Kazuhiko Nakano. Effects of Enzyme Replacement Therapy for Primary Teeth in a Patient with Infantile Hypophosphatasia. Journal of Clinical Pediatric Dentistry. 2020. 44(5);348-351.

References

1. Whyte MP, Zhang F, Wenkert D, et al. Hypophosphatasia: validation and expansion of the clinical nosology for children from 25 year experience with 173 pediatric patients. Bone 75: 229-239, 2015

2. Whyte MP. Hypophosphatasia–aetiology, nosology, pathogenesis, diagnosis and treatment. Nat Rev Endocrinol 12: 233-246, 2016.

3. Whyte MP, Wenkert D, Zhang F. Hypophosphatasia: Natural history study of 101 affected children investigated at one research center. Bone 93: 125–138, 2016.

4. Whyte MP. Hypophosphatasia: An overview for 2017. Bone 102: 15-25, 2017.

5. Mornet E. Hypophosphatasia. Metabolism 82: 142-155, 2018.

6. Michigami T, Ohata Y, Fujiwara M, et al. Clinical practice guidelines for hypophosphatasia. Clin Pediatr Endocrinol 29: 9-24, 2020.

7. Okawa R, Nakano K, Matsumoto M, et al. Oral manifestations of patients with hypophosphatasia. Ped Dent J 22:155-162, 2012.

8. Bloch-Zupan A. Hypophosphatasia: diagnosis and clinical signs–a dental surgeon perspective. Int J Paediatr Dent 26: 426-438, 2016.

9. Okawa R, Kitaoka T, Saga K, et al. Report of two dental patients diagnosed with hypophosphatasia. J Clin Case Rep 6; 2, 2016.

10. Whyte MP, Greenberg CR, Salman NJ, et al. Enzyme-replacement therapy in life-threatening hypophosphatasia. N Engl J Med 366(10); 904–913, 2012.

11. Whyte MP, Madson KL, Phillips D, et al. Asfotase alfa therapy for children with hypophosphatasia. JCI Insight 1(9), e85971, 2016.

12. Whyte MP, Rockman-Greenberg C, Ozono K, et al. Asfotase alfa treatment improves survival for perinatal and infantile hypophosphatasia. J Clin Endocrinol Metab 101: 334-342, 2016.

13. Okazaki Y, Kitajima H, Mochizuki N, et al. Lethal hypophosphatasia successfully treated with enzyme replacement from day 1 after birth. Eur J Pediatr 175(3), 433–437, 2016.

14. Kitaoka T, Tajima T, Nagasaki K, et al. Safety and efficacy with asfotase alfa in patients with hypophosphatasia: Results from a Japanese clinical trial. Clin Endocrinol 87: 10-19, 2017.

15. McDonald RE, Avery DR, Dean JA: Eruption of the teeth: Local, systemic, and congenital factors that influence the process, Edited by McDonald R, Avery DR, Dean JA, Dentistry for the child and adolescent 8th, Mosby, Missouri, pp.176-179, 2004.

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