J.ophthalmol.(Ukraine).2022;4:62-67.
http://doi.org/10.31288/oftalmolzh202246267
Received: 28.04.2022; Accepted: 09.06.2022; Published on-line: 24.08.2022
Diagnostic assessment and treatment of cherubism as prevention of ocular complications
R. S. Nazaryan 1, N. N. Mikhailenko 1, Yu. Yu. Yaroslavska 1, A. V. Bondarenko 1, V. V. Gargin 1, M. V. Lytvynenko 2, O. V. Artiomov 3, D. A. Oliynyk 2, T. V. Tregub 2
1 Kharkiv National Medical University; Kharkiv (Ukraine)
2 Odesa National Medical University; Odesa (Ukraine)
3 SI "The Filatov Institute of Eye Diseases and Tissue Therapy of the NAMS of Ukraine"; Odesa (Ukraine)
TO CITE THIS ARTICLE: Nazaryan RS, Mikhailenko NN, Yaroslavska YuYu, Bondarenko AV, Gargin VV, Lytvynenko MV, Artiomov OV, Oliynyk DA, Tregub TV. Diagnostic assessment and treatment of cherubism as prevention of ocular complications. J.ophthalmol.(Ukraine).2022;4:62-67. http://doi.org/10.31288/oftalmolzh202246267
This is an initiative study. Cherubism as a form of fibrous dysplasia of jaw bones is a congenital hereditary abnormality of osseous tissue maturation. Although the disease requires mostly maxillofacial surgery and orthodontic care, patients also have to be consulted by an ophthalmologist. The purpose of this study was to share our experience in the diagnostic assessment and treatment of cherubism, a rare form of fibrous dysplasia that is commonly misdiagnosed, which results in inadequate treatment and may lead to serious ocular complications. We present our experience of diagnostic assessment and treatment of four patients with cherubism for one to twenty years. The diagnosis of cherubism became evident after histological study of the material excised during surgery, because postoperative histology showed that changes were not cancerous (adamantinoma), but dysplastic. In addition, new foci developed or old foci recurred some time after surgery. Consequently, true jaw tumors must be correctly differentiated from dental cysts and from dysplastic bone lesions. Patients were followed up once every 6 months. Over the period of follow-up, ocular involvement was observed in the woman who had been diagnosed in childhood with congenital mixed astigmatism. Given our observations, one may make a conclusion that cherubism, a rare form of fibrous dysplasia, is a hereditary or familial disorder, and regresses after completion of puberty and puberty-related changes in hormonal milieu. Timely orthodontic care (and, if required, maxillofacial surgery care) will facilitate preventing the progression of the disease and its ophthalmological consequences. Special attention of ophthalmologists should be paid to cherubism patients with maxillary lesions.
Keywords: fibrous dysplasia, cherubism, cyst-like jaw lesions, orbit
References
1.Morice A, Joly A, Ricquebourg M, et al. Cherubism as a systemic skeletal disease: evidence from an aggressive case. BMC Musculoskelet Disord. 2020;21(1):564. Published 2020 Aug 21.
2.Chrcanovic BR, Guimarães LM, Gomes CC, Gomez RS. Cherubism: a systematic literature review of clinical and molecular aspects. Int J Oral Maxillofac Surg. 2021;50(1):43-53.
3.Machado RA, Pontes H, Pires FR, et al. Clinical and genetic analysis of patients with cherubism. Oral Dis. 2017;23(8):1109-1115.
4.Argyris PP, Gopalakrishnan R, Hu Y, Reichenberger EJ, Koutlas IG. Clinicopathologic and Molecular Characteristics of Familial Cherubism with Associated Odontogenic Tumorous Proliferations. Head Neck Pathol. 2018;12(1):136-144.
5.Chen Wongworawat Y, Jack D, Inman JC, et al. Regional Lymph Node Enlargement in Clinically Severe Cherubism. ClinPathol. 2019;12:2632010X19861107. Published 2019 Jul 4.
6.Popova TM, Kryvenko LS, Tishchenko OV, Nakonechna OA, Podrigalo LV, Nessonova TD, et al. Effect of Electronic Cigarettes on Oral Microbial Flora. J Pharm Nutr Sci. 2021;11(1):54-64.Yoo SH, Pineles SL, Jarrahy R, Velez FG. Ophthalmic manifestations of cherubism. J AAPOS. 2015;19(1):70-72.
7.Carroll AL, Sullivan TJ. Orbital involvement in cherubism. Clin Exp Ophthalmol. 2001;29(1):38-40.
8.Lacorzana J, Prieto-Moreno CG. Cherubism with orbital involvement. Indian J Ophthalmol. 2020;68(11):2534.
9.Nechyporenko AS, Radutny R, Alekseeva VV, Titova G, Gargin V. Complex Automatic Determination of Morphological Parameters for Bone Tissue in Human Paranasal Sinuses. Open Bioinformatics J. 2021;14(1):130-137.
10.Radutniy R, Nechyporenko A, Alekseeva V, Titova G, Bibik D, Gargin VV. Automated measurement of bone thickness on SCT sections and other images. In: Proceedings of the 2020 IEEE 3rd International Conference on Data Stream Mining and Processing, DSMP 2020; 2020. p. 222-6.
11.Ahmadi AJ, Pirinjian GE, Sires BS. Optic neuropathy and macular chorioretinal folds caused by orbital cherubism. Arch Ophthalmol. 2003;121(4):570-573.
12.Mello LGM, Saraiva FP, Monteiro MLR. Angioid streaks and optic disc drusen in cherubism: a case report. Arq Bras Oftalmol. 2020;83(6):535-537.
13.Colombo F, Cursiefen C, Neukam FW, Holbach LM. Orbital involvement in cherubism. Ophthalmology. 2001;108(10):1884-1888.
14.Gargin V, Radutny R, Titova G, Bibik D, Kirichenko A, Bazhenov O. Application of the computer vision system for evaluation of pathomorphological images. 2020 IEEE 40th International Conference on Electronics and Nanotechnology, ELNANO 2020 - Proceedings; 2020. 469-473,
15.Nechyporenko AS, Reshetnik VM, Alekseeva VV, Yurevych NO, Nazaryan RS, Gargin VV. Implementation and analysis of uncertainty of measurement results for lower walls of maxillary and frontal sinuses. In: 2020 IEEE 40th International Conference on Electronics and Nanotechnology, ELNANO 2020 - Proceedings; 20202020. p. 460-3.
16.Nechyporenko A, Reshetnik V, Shyian D, Alekseeva V, Radutny R, Gargin V. Solutions to the 3d model problem of pressure measurement in the area of maxillary sinus anastomosis. In: CEUR Workshop Proceedings [Internet]; 20202020. p. 275-84.
17.Palamar OI, Lukach EV, Maletskyi AP, Poliakova SI, Kravets OV, Serezhko YO, et al. Clinical, morphological, CT and MRI characteristics of anterior skull base and orbital tumors. Oftalmol Zh. 2020(5):62-74.
18.Son JH, Marshall DC, Valiathan M, et al. Innovative Surgical Treatment of Severe Cherubism. Cleft Palate Craniofac J. 2019;56(1):90-93.
19.Bobrova NF, Umanets MM, Dembovetska GM, Bratishko AI. Reconstructive duet in a pole-to-pole surgery for ocular trauma: A pediatric case study. Oftalmol Zh. 2021;90(2):55-60.
20.Mirmohammadsadeghi A, Eshraghi B, Shahsanaei A, Assari R. Cherubism: report of three cases and literature review. Orbit. 2015;34(1):33-37.
21.Lyndin M, Gluschenko N, Sikora V, et al. Morphofunctional features of articular cartilage structure. Folia Med Cracov. 2019;59(3):81-93.
22.Kuzenko Y, Mykhno O, Sikora V, Bida V, Bida O. Dental terminology "discoloration" or "pigment dystrophy" - a review and practical recommendations. Pol Merkur Lekarski. 2022;50(295):65-67.
23.Font RL, Blanco G, Soparkar CN, Patrinely JR, Ostrowski ML. Giant cell reparative granuloma of the orbit associated with cherubism. Ophthalmology. 2003;110(9):1846-1849.
24.Polyvianna Y, Chumachenko D, Chumachenko T. Computer aided system of time series analysis methods for forecasting the epidemics outbreaks. 2019 15th International Conference on the Experience of Designing and Application of CAD Systems, CADSM 2019:1-4.
Disclosures
Conflict of Interest: The authors declare that there is no conflict of interest that could influence their opinion regarding the subject or materials of this manuscript.
Sources of Support: none.