Article Data

  • Views 458
  • Dowloads 193

Systematic reviews

Open Access

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

  • Giuseppe Minervini1
  • Maria Maddalena Marrapodi2
  • Gabriele Cervino3
  • Rocco Franco4
  • Alessandro Lanza1
  • Marco Cicciù5
  • Fabrizio Di Francesco1,*,

1Multidisciplinary Department of Medical-Surgical and Odontostomatological Specialties, University of Campania “Luigi Vanvitelli”, 80121 Naples, Italy

2Department of Woman, Child and General and Specialist Surgery, University of Campania “Luigi Vanvitelli”, 80121 Naples, Italy

3Department of Biomedical and Dental Sciences and Morphofunctional Imaging, School of Dentistry, University of Messina, 98125 Messina, Italy

4Department of Biomedicine and Prevention, University of Rome “Tor Vergata”, 00100 Rome, Italy

5Department of Biomedical and Surgical and Biomedical Sciences, Catania University, 95123 Catania, Italy

DOI: 10.22514/jocpd.2023.049 Vol.47,Issue 5,September 2023 pp.12-18

Submitted: 20 February 2023 Accepted: 11 April 2023

Published: 03 September 2023

*Corresponding Author(s): Fabrizio Di Francesco E-mail:


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

Cite and Share

Giuseppe Minervini,Maria Maddalena Marrapodi,Gabriele Cervino,Rocco Franco,Alessandro Lanza,Marco Cicciù,Fabrizio Di Francesco. Congenital cranio-facial abnormalities in paediatric population: a systematic review on temporomandibular disorders. Journal of Clinical Pediatric Dentistry. 2023. 47(5);12-18.


[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: (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.

Abstracted / indexed in

Science Citation Index Expanded (SciSearch) Created as SCI in 1964, Science Citation Index Expanded now indexes over 9,500 of the world’s most impactful journals across 178 scientific disciplines. More than 53 million records and 1.18 billion cited references date back from 1900 to present.

PubMed (MEDLINE) PubMed comprises more than 35 million citations for biomedical literature from MEDLINE, life science journals, and online books. Citations may include links to full text content from PubMed Central and publisher web sites.

Biological Abstracts Easily discover critical journal coverage of the life sciences with Biological Abstracts, produced by the Web of Science Group, with topics ranging from botany to microbiology to pharmacology. Including BIOSIS indexing and MeSH terms, specialized indexing in Biological Abstracts helps you to discover more accurate, context-sensitive results.

Google Scholar Google Scholar is a freely accessible web search engine that indexes the full text or metadata of scholarly literature across an array of publishing formats and disciplines.

JournalSeek Genamics JournalSeek is the largest completely categorized database of freely available journal information available on the internet. The database presently contains 39226 titles. Journal information includes the description (aims and scope), journal abbreviation, journal homepage link, subject category and ISSN.

Current Contents - Clinical Medicine Current Contents - Clinical Medicine provides easy access to complete tables of contents, abstracts, bibliographic information and all other significant items in recently published issues from over 1,000 leading journals in clinical medicine.

BIOSIS Previews BIOSIS Previews is an English-language, bibliographic database service, with abstracts and citation indexing. It is part of Clarivate Analytics Web of Science suite. BIOSIS Previews indexes data from 1926 to the present.

Journal Citation Reports/Science Edition Journal Citation Reports/Science Edition aims to evaluate a journal’s value from multiple perspectives including the journal impact factor, descriptive data about a journal’s open access content as well as contributing authors, and provide readers a transparent and publisher-neutral data & statistics information about the journal.

Scopus: CiteScore 2.0 (2022) Scopus is Elsevier's abstract and citation database launched in 2004. Scopus covers nearly 36,377 titles (22,794 active titles and 13,583 Inactive titles) from approximately 11,678 publishers, of which 34,346 are peer-reviewed journals in top-level subject fields: life sciences, social sciences, physical sciences and health sciences.

Submission Turnaround Time