Congenital cranio-facial abnormalities in paediatric population: a systematic review on temporomandibular disorders

The aim of this systematic review was to explore the prevalence of temporomandibular disorders (TMD) in patients affected by congenital craniofacial disorders (CCD). Out of 292 papers, 9 studies were included with 561 participants. The included studies analyzed at least 15 patients treated for temporomandibular disorders, evaluating at least one clinical outcomes such as orofacial pain and/or muscular diseases, malocclusion, skeletal abnormality and/or deformities concerning temporomandibular joint, upper/lower jaw bone malformations. These findings provide an evidence of an association between temporomandibular disorders and congenital craniofacial disorders. The evidence is supported by the findings of a small number of papers in the literature, some of which were of just fair quality. To be definitive in this regard, additional research with a sizable sample size and control group are required.

Cranio-facial abnormalities; Congenital; Orofacial pain; Pediatric; Temporomandibular disorders

[1] Sicard L, O’Hana D, Khonsari RH, Kaddour Brahim A. Bilateral dislocation of the temporomandibular joint in children. Journal of Oral and Maxillofacial Surgery. 2018; 76: 2307–2315.

[2] Di Francesco F, Lanza A, di Blasio M, Vaienti B, Cafferata EA, Cervino G, et al. Application of botulinum toxin in temporomandibular disorders: a systematic review of randomized controlled trials (RCTs). Applied Sciences. 2022; 12: 12409.

[3] Young A. Idiopathic condylar resorption: the current understanding in diagnosis and treatment. The Journal of Indian Prosthodontic Society. 2017; 17: 128–135.

[4] Hodzic Z, Törnwall J, Leikola J, Heliövaara A, Suojanen J. Alloplastic temporomandibular joint reconstruction in congenital craniofacial deformities. Journal of Craniofacial Surgery. 2021; 32: e548–e551.

[5] Manocha S, Farokhnia N, Khosropanah S, Bertol JW, Santiago J Junior, Fakhouri WD. Systematic review of hormonal and genetic factors involved in the nonsyndromic disorders of the lower jaw. Developmental Dynamics. 2019; 248: 162–172.

[6] Minervini G, Del Mondo D, Russo D, Cervino G, D’Amico C, Fiorillo L. Stem cells in temporomandibular joint engineering: state of art and future persectives. The Journal of Craniofacial Surgery. 2022; 33: 2181.

[7] Wright JW Jr, Wright JW 3rd, Hicks G. Polytomography and congenital anomalies of the ear. Annals of Otology, Rhinology & Laryngology. 1982; 91: 480–484.

[8] Pitirri MK, Durham EL, Romano NA, Santos JI, Coupe AP, Zheng H, et al. Meckel’s cartilage in mandibular development and dysmorphogenesis. Frontiers in Genetics. 2022; 13: 871927.

[9] Wolford LM, Perez DE. Surgical management of congenital deformities with temporomandibular joint malformation. Oral and Maxillofacial Surgery Clinics of North America. 2015; 27: 137–154.

[10] Kaneyama K, Segami N, Hatta T. Congenital deformities and developmental abnormalities of the mandibular condyle in the temporomandibu-lar joint. Congenital Anomalies. 2008; 48: 118–125.

[11] Minervini G, Franco R, Marrapodi MM, Fiorillo L, Cervino G, Cicciù M. Prevalence of temporomandibular disorders (TMD) in pregnancy: a systematic review with meta-analysis. Journal of Oral Rehabilitation. 2023. [Preprint].

[12] Fiorillo L, De Stefano R, Cervino G, Crimi S, Bianchi A, Campagna P, et al. Oral and psychological alterations in haemophiliac patients. Biomedicines. 2019; 7: 33.

[13] Minervini G, Franco R, Marrapodi MM, Mehta V, Fiorillo L, Badnjević A, et al. The association between COVID-19 related anxiety, stress, depression, temporomandibular disorders, and headaches from childhood to adulthood: a systematic review. Brain Sciences. 2023; 13: 481.

[14] Quinzi V, Paskay LC, Manenti RJ, Giancaspro S, Marzo G, Saccomanno S. Telemedicine for a multidisciplinary assessment of orofacial pain in a patient affected by eagle’s syndrome: a clinical case report. The Open Dentistry Journal. 2021; 15: 102–110.

[15] Minervini G, Mariani P, Fiorillo L, Cervino G, Cicciù M, Laino L. Prevalence of temporomandibular disorders in people with multiple sclerosis: a systematic review and meta-analysis. CRANIO®. 2022; 1–9.

[16] Ramly EP, Yu JW, Eisemann BS, Yue O, Alfonso AR, Kantar RS, et al. Temporomandibular joint ankylosis in pediatric patients with craniofacial differences: causes, recurrence and clinical outcomes. Journal of Craniofacial Surgery. 2020; 31: 1343–1347.

[17] Sadrabad MJ, Ameli N, Kianpour M, Ghorbani R, Sohanian S. The relationship of temporomandibular disorders with class II malocclusion as a risk factor. APOS Trends in Orthodontics. 2021; 11: 41–47.

[18] Minervini G, Fiorillo L, Russo D, Lanza A, D’Amico C, Cervino G, et al. Prosthodontic treatment in patients with temporomandibular disorders and orofacial pain and/or bruxism: a review of the literature. Prosthesis. 2022; 4: 253–262.

[19] Saccomanno S, Quinzi V, D’Andrea N, Albani A, Coceani Paskay L, Marzo G. Traumatic events and eagle syndrome: is there any correlation?A systematic review. Healthcare. 2021; 9: 825.

[20] Pirttiniemi P, Peltomäki T, Müller L, Luder HU. Abnormal mandibular growth and the condylar cartilage. European Journal of Orthodontics. 2009; 31: 1–11.

[21] Allori AC, Chang CC, Fariña R, Grayson BH, Warren SM, McCarthy JG. Current concepts in pediatric temporomandibular joint disorders: part 1. etiology, epidemiology, and classification. Plastic and Reconstructive Surgery. 2010; 126: 1263–1275.

[22] Resnick CM. Temporomandibular joint reconstruction in the growing child. Oral and Maxillofacial Surgery Clinics of North America. 2018; 30: 109–121.

[23] Schiffman E, Ohrbach R, Truelove E, Look J, Anderson G, Goulet J, et al. Diagnostic criteria for temporomandibular disorders (DC/TMD) for clinical and research applications: recommendations of the international RDC/TMD consortium network* and orofacial pain special interest group†. Journal of Oral & Facial Pain and Headache. 2014; 28: 6–27.

[24] Özkan NC, Ozkan F. The relationship of temporomandibular disorders with headaches: a retrospective analysis. Agri. 2011; 23: 13–17.

[25] Deregibus A, Ferrillo M, Piancin MG, Domini MC, De Sire A, Castroflorio T. Are occlusal splints effective in reducing myofascial pain in patients with muscle-related temporomandibular disorders? A randomized-controlled trial. Turkish Journal of Physical Medicine and Rehabilitation. 2021; 67: 32–40.

[26] Di Francesco F, De Marco G, Capcha EB, Lanza A, Cristache CM, Vernal R, et al. Patient satisfaction and survival of maxillary overdentures supported by four or six splinted implants: a systematic review with meta-analysis. BMC Oral Health. 2021; 21: 247.

[27] Duman S, Vural H. Evaluation of the relationship between malocclusions and sleep-disordered breathing in children. CRANIO®. 2022; 40: 295–302.

[28] Page MJ, McKenzie JE, Bossuyt PM, Boutron I, Hoffmann TC, Mulrow CD, et al. The PRISMA 2020 statement: an updated guideline for reporting systematic reviews. BMJ. 2021; 372: n71.

[29] Wells A, Shea B, O’Connell D, Peterson J, Welch V, Losos M, et al. The newcastle-ottawa scale (NOS) for assessing the quality of nonrandomised studies in meta-analyses. Available at: https://www.ohri.ca/programs/clinical_epidemiology/oxford.asp (Accessed: 17 February 2014).

[30] Poon CC, Meara JG, Heggie AA. Hemifacial microsomia: use of the OMENS-plus classification at the royal children’s hospital of melbourne. Plastic and Reconstructive Surgery. 2003; 111: 1011–1018.

[31] Church MW, Eldis F, Blakley BW, Bawle EV. Hearing, language, speech, vestibular, and dentofacial disorders in fetal alcohol syndrome. Alcoholism, Clinical and Experimental Research. 1997; 21: 227–237.

[32] Isberg A, Isacsson G, Nah K. Mandibular coronoid process locking: a prospective study of frequency and association with internal derangement of the temporomandibular joint. Oral Surgery, Oral Medicine, Oral Pathology. 1987; 63: 275–279.

[33] Isberg A, Eliasson S. A cephalometric analysis of patients with coronoid process enlargement and locking. American Journal of Orthodontics and Dentofacial Orthopedics. 1990; 97: 35–40.

[34] Steinberg B, Nelson VS, Feinberg SE, Calhoun C. Incidence of maxillofacial involvement in arthrogryposis multiplex congenita. Journal of Oral and Maxillofacial Surgery. 1996; 54: 956–959.

[35] Ferri J, Schlund M, Touzet-Roumazeille S, Sciote JJ, Nicot R. Temporomandibular joint dysplasia in cranio-maxillofacial dysplasia: a retrospective study. Guideline treatment proposal. Journal of Craniofacial Surgery. 2021; 32: 1014–1021.

[36] Castro T, de Paula Martins Santos C, de Oliveira Lira Ortega A, Gallottini M. Oral characteristics and medical considerations in the dental treatment of individuals with Williams syndrome. Special Care in Dentistry. 2019; 39: 108–113.

[37] Bendixen KH, Gjørup H, Baad-Hansen L, Dahl Hald J, Harsløf T, Schmidt MH, et al. Temporomandibular disorders and psychosocial status in osteogenesis imperfecta—a cross-sectional study. BMC Oral Health. 2018; 18: 35.

[38] Małgorzata KM, Małgorzata P, Kinga S, Jerzy S. Temporomandibular joint and cervical spine mobility assessment in the prevention of temporomandibular disorders in children with osteogenesis imperfecta: a pilot study. International Journal of Environmental Research and Public Health. 2021; 18: 1076.

[39] De Rossi SS, Greenberg MS, Liu F, Steinkeler A. Temporomandibular disorders. Medical Clinics of North America. 2014; 98: 1353–1384.

[40] Gauer RL, Semidey MJ. Diagnosis and treatment of temporomandibular disorders. American Family Physician. 2015; 91: 378–386.

[41] Ingawalé S, Goswami T. Temporomandibular joint: disorders, treatments, and biomechanics. Annals of Biomedical Engineering. 2009; 37: 976–996.

[42] Türp JC, Strub JR. Prosthetic rehabilitation in patients with temporo-mandibular disorders. The Journal of Prosthetic Dentistry. 1996; 76: 418–423.

[43] Plesh O, Curtis D, Levine J, Mccall Jr WD. Amitriptyline treatment of chronic pain in patients with temporomandibular disorders. Journal of Oral Rehabilitation. 2000; 27: 834–841.

[44] Ribeiro JAM, de Resende CMBM, Lopes ALC, Farias-Neto A, Carreiro ADFP. Association between prosthetic factors and temporomandibular disorders in complete denture wearers. Gerodontology. 2014; 31: 308–313.

[45] Masotti C, Oliveira KG, Poerner F, Splendore A, Souza J, Freitas RDS, et al. Auriculo-condylar syndrome: mapping of a first locus and evidence for genetic heterogeneity. European Journal of Human Genetics. 2008; 16: 145–152.

[46] Ozcelik TB, Pektas ZO. Management of chronic unilateral temporomandibular joint dislocation with a mandibular guidance prosthesis: a clinical report. The Journal of Prosthetic Dentistry. 2008; 99: 95–100.

[47] De Boever JA, Carlsson GE, Klineberg IJ. Need for occlusal therapy and prosthodontic treatment in the management of temporomandibular disorders. Part II. Tooth loss and prosthodontic treatment. Journal of Oral Rehabilitation. 2000; 27: 647–659.

[48] Mor N, Tang C, Blitzer A. Temporomandibular myofacial pain treated with botulinum toxin injection. Toxins. 2015; 7: 2791–2800.

[49] Carlsson GE. Some dogmas related to prosthodontics, temporomandibular disorders and occlusion. Acta Odontologica Scandinavica. 2010; 68: 313–322.