Cherubism: early surgical approach and its effects on the quality of life in a pediatric patient

Autores

DOI:

https://doi.org/10.5327/2525-5711.326

Palavras-chave:

Cherubism, Facial deformities, Quality of life

Resumo

Querubismo é uma osteodisplasia fibro-óssea rara caracterizada pela expansão simétrica dos maxilares, frequentemente levando a deformidades faciais e comprometimento funcional. Este estudo apresenta o caso de um paciente pediátrico diagnosticado com querubismo que foi submetido a intervenção cirúrgica precoce para melhorar a qualidade de vida e reduzir os impactos psicossociais associados à doença. O questionário PedsQL™ 4.0 foi aplicado para avaliar a qualidade de vida antes e depois do procedimento cirúrgico. Os resultados demonstraram uma melhora significativa nos domínios social e escolar, refletindo uma redução na carga psicossocial do querubismo. Seis meses após o procedimento, nenhuma complicação significativa ou sinais de recorrência foram observados, destacando a eficácia da intervenção precoce. Esses achados sugerem que uma abordagem cirúrgica precoce é uma estratégia valiosa para otimizar a qualidade de vida e minimizar os efeitos negativos do querubismo, particularmente em crianças em desenvolvimento social e acadêmico.

Palavras-chave: Querubismo; Deformidades faciais; Qualidade de vida.

Referências

Tekin AF, Ünal ÖF, Göksel S, Özcan İ. Clinical and radiological evaluation of cherubism: a rare case report. Radiol Case Rep. 2020;15(4):416-9. https://doi.org/10.1016/j.radcr.2020.01.003

Moss W, Pires G, Zahid R, Tyrell R, Rhemtulla I, Gociman B. Severe cherubism treated with curettage, osteotomy, and bony repositioning: a case series of three patients. Plast Reconstr Surg Glob Open. 2022;10(1):e4079. https://doi.org/10.1097/GOX.0000000000004079

Liles SI, Hoppe IC, Arnold L. Denosumab therapy in cherubism. Cleft Palate Craniofacial J. 2023;60(12):1665-73. https://doi.org/10.1177/10556656221113891

Lahfidi A, Traore WM, Diallo ID, Lrhorfi N, Elhaddad S, Allali N, et al. Cherubism: a rare case report with literature review. Radiol Case Rep. 2022;17(10):3971-3. https://doi.org/10.1016/j.radcr.2022.07.063

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. https://doi.org/10.1016/j.ijom.2020.05.021

Garlick JW, Willis RN, Donato DP, Gociman B. Cherubism in a 4-year-old boy managed with tumor curettage, mandibular osteotomies and repositioning. Plast Aesthet Res. 2018;5:29. https://doi.org/10.20517/2347-9264.2018.36

Holley TJ, Giannini PJ, Narayana N, Desa VP. Early detection of cherubism with eventual bilateral progression: a literature review and case report. Oral Surg Oral Med Oral Pathol Oral Radiol. 2019;127(3):e77-83. https://doi.org/10.1016/j.oooo.2018.08.011

Jung KW, Yun JM, Lee JM, Choi IS. Sinonasal manifestations of severe cherubism: a case with 11-year follow-up. Ear Nose Throat J. 2025;104(1_suppl):257S-62S. https://doi.org/10.1177/01455613221127588

Papadaki ME, Lietman SA, Levine MA, Olsen BR, Kaban LB, Reichenberger EJ. Cherubism: best clinical practice. Orphanet J Rare Dis. 2012;7(Suppl. 1):S6. https://doi.org/10.1186/1750-1172-7-S1-S6

Haraldstad K, Wahl A, Andenæs R, Andersen JR, Andersen MH, Beisland E, et al. A systematic review of quality of life research in medicine and health sciences. Qual Life Res. 2019;28(10):2641-50. https://doi.org/10.1007/s11136-019-02214-9

Geirdal AØ, Saltnes SS, Storhaug K, Åsten P, Nordgarden H, Jensen JL. Living with orofacial conditions: psychological distress and quality of life in adults affected with Treacher Collins syndrome, cherubism, or oligodontia/ectodermal dysplasia-a comparative study. Qual Life Res. 2015;24(4):927-35. https://doi.org/10.1007/s11136-014-0826-1

Tapia VJ, Epstein S, Tolmach OS, Hassan AS, Chung NN, Gosman AA. Health-related quality-of-life instruments for pediatric patients with diverse facial deformities: a systematic literature review. Plast Reconstr Surg. 2016;138(1):175-87. https://doi.org/10.1097/PRS.0000000000002285

Shapiro DN, Waljee J, Ranganathan K, Buchman S, Warschausky S. Using the patient reported outcomes measurement information system to evaluate psychosocial functioning among children with craniofacial anomalies. Plast Reconstr Surg. 2015;135(6):1673-9. https://doi.org/10.1097/PRS.0000000000001269

Riatto BM, Riatto SG, Andrade LP. Querubismo e qualidade de vida: impactos físicos e psicológicos. Revista Diálogos em Saúde. 2021;4(1):13-28.

Costabile C, Brunello MIB. Repercussões da inclusão escolar sobre o cotidiano de crianças com deficiência: um estudo a partir do relato das famílias. Rev Ter Ocup Univ São Paulo. 2005;16(3):124-30.

Kueper J, Tsimbal C, Olsen BR, Kaban L, Liao EC. SH3BP2-related fibro-osseous disorders of the maxilla and mandible: a systematic review. Int J Oral Maxillofac Surg. 2022;51(1):54-61. https://doi.org/10.1016/j.ijom.2021.04.001

Gupta S, Singh K, Garg A, Bhandari PS, Sah SK, Reichenberger E, et al. Clinicoradiologic follow up of cherubism with aggressive characteristics: a series of 3 cases. Oral Surg Oral Med Oral Pathol Oral Radiol. 2019;128(5):e191-201. https://doi.org/10.1016/j.oooo.2019.01.082

Eiden S, Lausch E, Meckel S. Involution von cherubismus im MRT unter therapie mit imatinib. RoFo. 2017;189(7):675-7. https://doi.org/10.1055/s-0043-105074

Ricalde P, Ahson I, Schaefer ST. A paradigm shift in the management of cherubism? A preliminary report using imatinib. J Oral Maxillofac Surg. 2019;77(6):1278.e1-1278.e7. https://doi.org/10.1016/j.joms.2019.02.021

Mukai T, Akagi T, Asano SH, Tosa I, Ono M, Kittaka M, et al. Imatinib has minimal effects on inflammatory and osteopenic phenotypes in a murine cherubism model. Oral Dis. 2023;29(3):1089-101. https://doi.org/10.1111/odi.14073

Son JH, Marshall DC, Valiathan M, Otteson T, Ferretti G, Grigorian PA, et al. Innovative surgical treatment of severe cherubism. Cleft Palate Craniofac J. 2019;56(1):90-3. https://doi.org/10.1177/1055665618774008

Holley TJ, Giannini PJ, Narayana N, Desa VP. Early detection of cherubism with eventual bilateral progression: a literature review and case report. Oral Surg Oral Med Oral Pathol Oral Radiol. 2019;127(3):e77-83. https://doi.org/10.1016/j.oooo.2018.08.011

Publicado

2025-05-27

Como Citar

1.
Cunha LTMQ, Pereira AO, Santos RC, Zancopé E, Pinto SA, Félix VB, et al. Cherubism: early surgical approach and its effects on the quality of life in a pediatric patient. J Oral Diagn [Internet]. 27º de maio de 2025 [citado 8º de outubro de 2025];10. Disponível em: https://www.jordi.com.br/revista/article/view/326

Edição

Seção

Relato de Caso