Analysis of Mineral Density of Calcified Tissues in Children with X-Linked Hypophosphatemic Rickets and Hypophosphatasia Using Cone Beam Computed Tomography Data

ORIGINAL ARTICLE
Dmitriy A. Lezhnev, Elena V. Vislobokova, Larisa P. Kiselnikova, Natalia A. Sholokhova, Margarita V. Smyslenova, Viktor P. Truten

 
International Journal of Biomedicine. 2021;11(1):53-57.
DOI: 10.21103/Article11(1)_OA11
Originally published March 5, 2021

Abstract: 

The purpose of the present cohort study was a quantitative assessment of the enamel, dentin, and alveolar bone mineral density (BMD) using Cone Beam Computed Tomography (CBCT) scans in patients with X-linked hypophosphatemic rickets (HLHR) and hypophosphatasia (HPP) and a comparison with the data obtained from the control group.
Methods and Results: The unrepresentative, non-random sample included 30 CBCT scans of children with genetically and biochemically confirmed XLHR (OMIM #307800) and HPP (OMIM: 146300, 241510, 241500, and 146300). X-ray examination and dental care were carried out in the Radiology Diagnostics Department and Pediatric Dentistry Department at Moscow State University of Medicine and Dentistry named after AI Evdokimov. The mineral density of calcified tissues (enamel, dentin, and alveolar bone) was evaluated using i-CAT Vision TM software options on reconstructed CBCT axial views.
The images of all XLHR and HPP patients visualized large pulp chambers with prominent pulp horns extending to the dentin-enamel junction. The present study revealed poor alveolar bone mineralization in patients with HPP and XLHR. Analysis of CBCT scans showed a significant dentine hypodensity in XLHR patients, which may contribute to the emergence of multiple, spontaneous, periapical abscesses spreading rapidly in the jawbone.
Conclusion: Data obtained could be used for planning dental treatment of patients with XLHR and HPP.

Keywords: 
X-linked hypophosphatemic rickets • hypophosphatasia • mineral density • cone beam computed tomography
References: 
  1. Opsahl Vital S, Gaucher C, Bardet C, Rowe PS, George A, Linglart A, Chaussain C. Tooth dentin defects reflect genetic disorders affecting bone mineralization. Bone. 2012 Apr;50(4):989-97. doi: 10.1016/j.bone.2012.01.010. 
  2. Lee BN, Jung HY, Chang HS, Hwang YC, Oh WM. Dental management of patients with X-linked hypophosphatemia. Restor Dent Endod. 2017 May;42(2):146-151. doi: 10.5395/rde.2017.42.2.146. 
  3. Bianchi ML. Hypophosphatasia: an overview of the disease and its treatment. Osteoporos Int. 2015 Dec;26(12):2743-57. doi: 10.1007/s00198-015-3272-1. 
  4. Whyte MP. Hypophosphatasia: An overview For 2017. Bone. 2017 Sep;102:15-25. doi: 10.1016/j.bone.2017.02.011. 
  5. Sabandal MM, Robotta P, Bürklein S, Schäfer E. Review of the dental implications of X-linked hypophosphataemic rickets (XLHR). Clin Oral Investig. 2015 May;19(4):759-68. doi: 10.1007/s00784-015-1425-
  6. Bloch-Zupan A. Hypophosphatasia: diagnosis and clinical signs - a dental surgeon perspective. Int J Paediatr Dent. 2016 Nov;26(6):426-438. doi: 10.1111/ipd.12232. 
  7. de Moura PM, Hallac RR, Seaward JR, Kane AA, Aguiar M, Raggio R, Gutfilen B. Objective and subjective image evaluation of maxillary alveolar bone based on cone beam computed tomography exposure parameters. Oral Surg Oral Med Oral Pathol Oral Radiol. 2016 May;121(5):557-65. doi: 10.1016/j.oooo.2016.01.019. 
  8. Yeung AWK, Jacobs R, Bornstein MM. Novel low-dose protocols using cone beam computed tomography in dental medicine: a review focusing on indications, limitations, and future possibilities. Clin Oral Investig. 2019 Jun;23(6):2573-2581. doi: 10.1007/s00784-019-02907-y.
  9. Zhang J, Tian Y. Liu Y, Liu Q. Alveolar mineral density measurement using CBCT images. Neuroscience and biomedical engineering. 2017;5(1): 44-49. doi : 10.2174/2213385205666170612114301
  10. Ribeiro TR, Costa FW, Soares EC, Williams JR Jr, Fonteles CS. Enamel and dentin mineralization in familial hypophosphatemic rickets: a micro-CT study. Dentomaxillofac Radiol. 2015;44(5):20140347. doi: 10.1259/dmfr.20140347.
  11. Hayashi-Sakai S, Sakamoto M, Hayashi T, Kondo T, Sugita K, Sakai J, Shimomura-Kuroki J, Ike M, Nikkuni Y, Nishiyama H. Evaluation of permanent and primary enamel and dentin mineral density using micro-computed tomography. Oral Radiol. 2019 Jan;35(1):29-34. doi: 10.1007/s11282-018-0315-2. 
  12. Pauwels R, Jacobs R, Singer SR, Mupparapu M. CBCT-based bone quality assessment: are Hounsfield units applicable? Dentomaxillofac Radiol. 2015;44(1):20140238. doi: 10.1259/dmfr.20140238. 
  13. Van Dessel J, Huang Y, Depypere M, Rubira-Bullen I, Maes F, Jacobs R. A comparative evaluation of cone beam CT and micro-CT on trabecular bone structures in the human mandible. Dentomaxillofac Radiol. 2013;42(8):20130145. doi: 10.1259/dmfr.20130145.
  14. Skrinar A, Dvorak-Ewell M, Evins A, Macica C, Linglart A, Imel EA, Theodore-Oklota C, San Martin J. The Lifelong Impact of X-Linked Hypophosphatemia: Results From a Burden of Disease Survey. J Endocr Soc. 2019 May 7;3(7):1321-1334. doi: 10.1210/js.2018-00365.
  15. Foster BL, Nociti FH Jr, Somerman MJ. The rachitic tooth. Endocr Rev. 2014 Feb;35(1):1-34. doi: 10.1210/er.2013-1009.

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Received December 30, 2020.
Accepted February 5, 2021.
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