Cherubism: Early Surgical Approach and Its Effects on the Quality of Life in a Pediatric Patient

Autores/as

  • Laryssa Thainá Mello Queiroz Cunha Department of oral diagnosis, Semiology and oral Pathology areas, Piracicaba Dental School, State University of Campinas (UNICAMP) https://orcid.org/0000-0003-1357-4129
  • Almira Oliveira Pereira Department of oral diagnosis, Semiology and oral Pathology areas, Piracicaba Dental School, State University of Campinas (UNICAMP)
  • Roniery Correia Santos Department of Psychology, IPOG - Instituto de Pós-Graduação e Graduação, Goiânia, Goiás, Brazil https://orcid.org/0000-0002-8481-1147
  • Eduardo Zancopé Department of Occlusion, Fixed Prosthesis and Dental Materials, School of Dentistry, Federal University of Uberlândia, Uberlândia, Minas Gerais, Brazil https://orcid.org/0000-0002-6567-4810
  • Sebastião Alves Pinto 5Pathologist, Goiano Institute of Oncology and Hematology, Goiânia, Goiás, Brazil
  • Valtuir Barbosa Félix Department of Oral Pathology, University Hospital of the Federal University of Alagoas, Maceió, Alagoas, Brasil https://orcid.org/0000-0002-2961-2487
  • Rubens Jorge Silveira Department of Oral and Maxillofacial Surgery and Traumatology, Pontifical Catholic University of Goiás (PUC - GOIÁS), https://orcid.org/0000-0002-3234-2158

DOI:

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

Palabras clave:

Cherubism, Facial deformities, Quality of life

Resumen

El querubismo es una osteodisplasia fibroósea poco frecuente que se caracteriza por la expansión simétrica de los maxilares, que suele producir deformidades faciales y deterioro funcional. Este estudio presenta el caso de un paciente pediátrico diagnosticado con querubismo que se sometió a una intervención quirúrgica temprana para mejorar la calidad de vida y reducir los impactos psicosociales asociados a la enfermedad. Se aplicó el cuestionario PedsQL™ 4.0 para evaluar la calidad de vida antes y después del procedimiento quirúrgico. Los resultados demostraron una mejora significativa en los dominios social y escolar, lo que refleja una reducción de la carga psicosocial del querubismo. Seis meses después del procedimiento, no se observaron complicaciones significativas ni signos de recurrencia, lo que destaca la eficacia de la intervención temprana. Estos hallazgos sugieren que un abordaje quirúrgico temprano es una estrategia valiosa para optimizar la calidad de vida y minimizar los efectos negativos del querubismo, en particular en niños en desarrollo social y académico.

Palabras clave: Querubismo; Deformidades faciales; Calidad de vida.

Citas

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.

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.

Liles SI, C. Hoppe I, Arnold L. Denosumab Therapy in Cherubism. Cleft Palate-Craniofacial Journal. 2022;1–9.

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.

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.

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(8):29.

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.

Jung KW, Yun JM, Lee JM, Choi IS. Sinonasal Manifestations of Severe Cherubism: A Case with 11-year Follow-up. Ear Nose Throat J. 2022;0(0):014556132211275.

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.

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. Quality of Life Research. 2019;28(10):2641–50.

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. Quality of Life Research. 2015;24(4):927–35.

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. Plast Reconstr Surg. 2016 Jul;138(1):175–87.

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.

Riatto BM, Riatto SG, Andrade LP. Querubismo E Qualidade De Vida: Impactos Físicos E Psicológicos. 2021;4:13–28.

Costabile C, Brunello MIB. Repercussões da inclusão escolar sobre o cotidiano de crianças com deficiência: um estudo a partir do relato das famílias. Revista de Terapia Ocupacional da Universidade de São Paulo. 2005 Sep 1;16(3).

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.

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.

Eiden S, Lausch E, Meckel S. Involution von Cherubismus im MRT unter Therapie mit Imatinib. RöFo - Fortschritte auf dem Gebiet der Röntgenstrahlen und der bildgebenden Verfahren. 2017 Jul 26;189(07):675–7.

Ricalde P, Ahson I, Schaefer ST. A Paradigm Shift in the Management of Cherubism? A Preliminary Report Using Imatinib. Journal of Oral and Maxillofacial Surgery. 2019 Jun;77(6):1278.e1-1278.e7.

Mukai T, Akagi T, Hiramatsu Asano S, 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 Apr 23;29(3):1089–101.

Son JH, Marshall DC, Valiathan M, Otteson T, Ferretti G, Grigorian PA, et al. Innovative Surgical Treatment of Severe Cherubism. Cleft Palate-Craniofacial Journal. 2019;56(1):90–3.

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.

Descargas

Publicado

2025-05-27

Cómo citar

1.
Thainá Mello Queiroz Cunha L, Oliveira Pereira A, Correia Santos R, Zancopé E, Alves Pinto S, Barbosa Félix V, et al. Cherubism: Early Surgical Approach and Its Effects on the Quality of Life in a Pediatric Patient. J Oral Diagn [Internet]. 27 de mayo de 2025 [citado 19 de junio de 2025];. Disponible en: https://www.joraldiagnosis.com/revista/article/view/326

Número

Sección

Case Report