Decayed, missing, and restored teeth in patients with Neurofibromatosis Type 1

Friedrich, Reinhard E; Reul, Anika

doi:10.4317/jced.54561

Cited by 8 publications

(12 citation statements)

References 31 publications

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“…The results suggest that NF1 as such does not cause susceptibility to caries (Table 1 ). A strict professional caries prevention is advised because gingival enlargements and motoric clumsiness may hamper the maintaining patient’s oral health and our results in agreement with recently published study of Friedrich et al [ 41 ].…”

Section: Resultssupporting

confidence: 92%

Craniofacial and oral alterations in patients with Neurofibromatosis 1

Visnapuu

Peltonen

Alivuotila

et al. 2018

Orphanet J Rare Dis

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AbtsractNeurofibromatosis type 1 (NF1) is one of the most common inherited syndromes. The literature on craniofacial alterations associated with NF1 has been limited and partially contradictory. This review is based on literature search and the results of the clinical study “Craniofacial and Oral Alterations and Speech in patients with Neurofibromatosis 1”, carried out at the University of Turku and Turku University Hospital, Finland in 2006-2012. By the end of 2012, a total of 110 NF1 patients, 54 female and 56 male patients, were examined.A part of our results confirms pre-existing understanding, a part is contradictory to previous considerations based mainly on case reports, and some are entirely novel. Specifically, our results confirmed that enlargement the mandibular canal is the most common abnormality of the mandible in patients with NF1. It should be noted, however, that this finding does not require treatment. Caries was not a major problem. In fact, it was less frequent in NF1 patients compared to reference population. These findings abrogate some previous perceptions. Novel findings of our project include periapical cemental dysplasia in females; short jaws, a finding which usually does not affect bite; and immunohistological analysis of oral mucosal abnormalities. Pioneering study on speech showed that various deviations were very common: As many as 94% of the participants showed some alterations.To conclude, the awareness of craniofacial alterations common in NF1would help avoiding unnecessary and even harmful involvement, e.g. of periapical cemental dysplasia or enlarged mandibular canal which do not require treatment.

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Section: Resultssupporting

confidence: 92%

Craniofacial and oral alterations in patients with Neurofibromatosis 1

Visnapuu

Peltonen

Alivuotila

et al. 2018

Orphanet J Rare Dis

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“…Freidrich and Reul showed lowest DMFT index in NF1 group when compared with reference group solely in mandibular nerve region. They reported missing teeth were more frequently registered with NF1 group 14 . In present case localized severe attachment loss and bone loss was seen with mandibular region when compared with maxillary region thus showing a predilection towards mandibular tooth loss.…”

Section: Discussionmentioning

confidence: 96%

Oral Rehabilitation in Neurofibromatosis Type 1- A Six Year Follow-Up Case Report

Pachrupe

Phadnaik

Gupta

2021

International Journal of Medical and Dental Sciences

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Neurofibromatosis is an autosomal dominant disorder that affects the bone, the nervous system, soft-tissue, and the skin. The expressivity of the disease is extremely variable, with manifestations ranging from mild lesions to several complications and functional impairment. The penetrance, otherwise, is 100%. Approximately 72% of Neurofibromatosis Type1 patients present with oral manifestations. These patients have an estimated 3-15% additional risk of malignant disease in their life-time. The following report presents a 30 year old lady diagnosed with Neurofibromatosis Type 1 in childhood who presented to department with complain of bleeding gums. Radiograph showed shallow sigmoid notch on left side, obtuse and thinned out left angle of mandible which are some additional features. A multidisciplinary approach of dental and medical specialties was taken here for oral rehabilitation and to manage and treat periodontal manifestations and other oral complaints. Case was followed for more than 6 years. Neurofibromatosis has no cure hence when such patients report to dental professionals it is up to them to act as primary care giver and instill positive frame of mind to help reduce stressful life events.

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“…Although a case of gingival neurofibroma in the attached gingiva has been reported [ 30 , 31 ], so far there is a lack of evidence regarding whether NF1 patients may present periodontal disease without tumors directly involving the oral cavity. In our study, as all NF1 patients did not have neurofibromas in the maxilla or mandible, our findings for the first time demonstrated that NF1 patients were at a high risk of periodontitis.…”

Section: Discussionmentioning

confidence: 99%

Age-dependent oral manifestations of neurofibromatosis type 1: a case–control study

Thota

Veeravalli

Manchala

et al. 2022

Orphanet J Rare Dis

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Introduction Most craniofacial manifestations of neurofibromatosis type 1 (NF1) are considered as a result of tumor compression. We sought to determine salivary changes, caries, and periodontal complications in NF1 patients without tumors in the oral cavity. Objective and methods Eleven NF1 patients without tumors in the oral cavity and 29 matched controls without NF1 were enrolled in this case–control study. Demographic information, medical history, and data of intraoral examinations, including the Decayed, Missing, and Filled Teeth (DMFT) scores and Russel’s periodontal index (PI), were recorded. The functional salivary analysis was performed for sialometry, salivary pH values, and amylase activity. Ingenuity Systems Pathway Analysis (IPA) was conducted to identify mutually activated pathways for NF1-associated oral complications. Results NF1 patients were associated with periodontitis (OR = 1.40, 95% CI = 1.06–1.73, P = 0.04), gingivitis (OR = 1.55, 95% CI = 1.09–2.01, P = 0.0002), and decreased salivary flow rates (OR = 1.40, 95% CI = 1.05–1.76, P = 0.005). Periodontal destruction, salivary changes, and dental caries in NF1 patients were age-dependent. Subgroup analyses based on age stratification suggested that salivary flow rates and salivary amylase activities were significantly low in NF1 patients aged over 20 years and that salivary pH values, PI and DMFT scores were significantly high among NF1- controls aged over 20. All oral complications were not significantly presented in NF1 patients aged below 20 years. IPA analyses suggested that cellular mechanisms underlying NF1-associated oral complications involved chronic inflammatory pathways and fibrosis signaling pathway. Conclusion NF1 patients without tumors in the oral cavity presented a comparatively high prevalence of age-dependent oral complications, including periodontal destruction and salivary gland dysfunction, which were associated with chronic inflammatory pathogenesis.

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Decayed, missing, and restored teeth in patients with Neurofibromatosis Type 1

Cited by 8 publications

References 31 publications

Craniofacial and oral alterations in patients with Neurofibromatosis 1

Craniofacial and oral alterations in patients with Neurofibromatosis 1

Oral Rehabilitation in Neurofibromatosis Type 1- A Six Year Follow-Up Case Report

Age-dependent oral manifestations of neurofibromatosis type 1: a case–control study

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