What is the Prevalence of Undiagnosed Nevoid Basal Cell Carcinoma Syndrome in Children With an Odontogenic Keratocyst?

Karhade, Deepti Shroff; Afshar, Salim; Padwa, Bonnie L.

doi:10.1016/j.joms.2019.01.045

Cited by 10 publications

(15 citation statements)

References 8 publications

Supporting

Mentioning

Contrasting

Order By: Relevance

“…Moreover, left undiagnosed or untreated, OKCs may result in substantial bone loss and subsequent pathologic fractures. 4 , 12 Notwithstanding its importance, dental screening for GGS has received scant attention in the analytic literature.…”

Section: Discussionmentioning

confidence: 99%

Dental Screening Including Panoramic Radiograph for Gorlin-Goltz Syndrome in Patients With Multiple Basal Cell Carcinomas

et al. 2022

View full text Add to dashboard Cite

Purpose To answer the following clinical research question: “Among patients with multiple basal cell carcinomas (mBCCs), can panoramic radiograph (PaR) facilitate the diagnosis of Gorlin-Goltz syndrome (GGS)?” Methods This retrospective study enrolled mBCCs subjects who presented to a German tertiary care center between 1 January 2015 and 31 December 2021. The primary predictor was presence of syndromic mBCCs, and the main outcomes were jaw cysts and odontogenic keratocysts (OKCs). Descriptive, bi- and multivariate statistics, diagnostic test evaluation, and number needed to screen (NNS) were computed at α = 95%. Results The sample comprised 527 mBCCs patients (36.1% females; 6.8% GGS; 5.5% OKCs; mean age, 74.5 ± 15.8 years [range, 15-102]). There was a significant association between syndromic mBCCs and jaw cysts ( P < .0001; NNS = 2 [95% CI, CI, 1.1 to 1.4]). In the adjusted logistic model, PaR identified GGS via radiographic diagnosis of jaw cysts in case of 1) age ≤ 35 years, 2) ≥ 5 BCCs, and 3) ≥ 1 high-risk BCCs. Nearly every jaw cyst identified by PaR was OKCs ( P = .01; 95% CI, 3.1 to 3,101.4; NNS = 1.3 [95% CI, .9 to 2]). The post hoc power was 100%. Conclusions Dental screening with the use of PaR for mBCCs patients, especially those aged ≤35 years, or with ≥5 BCCs, or ≥1 high-risk BCCs, may be helpful in detection and identification of GGS through recognition of OKCs.

show abstract

Section: Discussionmentioning

confidence: 99%

Dental Screening Including Panoramic Radiograph for Gorlin-Goltz Syndrome in Patients With Multiple Basal Cell Carcinomas

et al. 2022

View full text Add to dashboard Cite

show abstract

“…Syndromic OKC only about 5% of all OKC cases. 45 Multiple OKCs in children need to be considered for association with nevoid basal carcinoma syndrome (NBCCS). This syndrome is genetically inherited with various expressions, such as multiple nevoid basal cell carcinomas, odontogenic keratocysts, congenital skeletal defects, ectopic calcifications, plantar and palmar pits, central nervous system and ocular lesions, and hypertelorism.…”

Section: Review Article Discussionmentioning

confidence: 99%

“…5,46 Patients with NBCSS are usually younger, under 20 years with a median age of 15 years when diagnosed with OKC. 45,47 The most common location of OKC was in the mandibular posterior region as shown in Figure 3. This is in accordance with the findings by Bhat, A et al 48 The location of the lesion is an important consideration in the interpretation process because each lesion has a predilection for a particular area.…”

Section: Review Article Discussionmentioning

confidence: 99%

Imaging of odontogenic keratocyst of the jaw by panoramic radiography: a scoping review

Sulastri

Firman

Epsilawati

2021

JRDI

View full text Add to dashboard Cite

Objectives: This review article is aimed to determine the imaging of odontogenic keratocyst of the jaw by panoramic radiography. Review: This research is a descriptive research using the scoping review method based on the Preferred Reporting Items for Systematic Review and Meta-analysis Scoping Review (PRISMA-Scr) which was conducted from February 2021 to April 2021. The search for literature related to the research topic was carried out through database of scientific articles on PMC NCBI, Pubmed NCBI, and Garuda as well as hand searching. The identified articles were screened by checking for duplicates, reading the titles and abstracts, and reading the entire articles. A total of 16 articles were included out of 161 articles in total. Conclusion: Imaging of odontogenic keratocyst of the jaw by panoramic radiography is most commonly found in the 1st and 2nd decades of life and in males, the lesions are unilocular or multilocular radiolucent and have an envelopmental shape in which the outline of the cyst surrounds the entire unerupted tooth. OKCs have well-defined with sclerotic or scalloped margins, most often occur in the posterior mandible and often associated with impacted 3rd molars, root resorption was a rare occurrence.

show abstract

“…Odontogenic keratocysts are characterized by aggressive growth and high recurrence rates, which, depending on the surgical modality used in their treatment, could reach even 32.3% (Al‐Moraissi et al., 2017). Another striking feature of these lesions is the fact that they are among the most consistent manifestations of nevoid basal cell carcinoma (NBCC) syndrome (Karhade et al., 2019; Lo Muzio, 2008). This condition is directly related to mutations of the Patched (PTCH) gene, which is a part of the Sonic Hedgehog (Shh) signaling pathway.…”

Section: Introductionmentioning

confidence: 99%

Gene polymorphisms in odontogenic keratocysts and ameloblastomas: A systematic review

et al. 2021

View full text Add to dashboard Cite

Objectives:The aim of this systematic review was to critically analyze available data on gene polymorphisms in odontogenic keratocysts (OKC) and ameloblastomas, including their possible relationship with clinical and histological features of these lesions. Materials and Methods: A comprehensive search of Web of Science Scopus, PubMed, Cochrane Central Register of Controlled Trials and EMBASE was conducted using relevant key terms and supplemented by a gray literature search. Quality assessment of included studies was performed using criteria from the Strengthening the Reporting of Genetic Association (STREGA) statement. Results: Ten studies were included in the final review. Survivin -31G/C, interleukin IL-1α -889 C/T, p53 codon 72 G/C, tumor necrosis factor TNFα (−308G>A) and itsreceptor TNF-R1 (36A>G), glioma-associated oncogene homolog 1 rs2228224 and matrix metalloproteinase 2 rs243865 gene polymorphisms were reported to be associated with OKC. For ameloblastomas, p53 codon 72 G/C, X-ray repair crosscomplementing protein 1-codons 194 and 399 and matrix metalloproteinase 9 rs3918242 gene polymorphisms were identified as risk factors. It was not possible to establish a relationship between specific polymorphisms and clinical and histological features of investigated lesions. Conclusions:Several gene polymorphisms might be considered as a risk factor for the development of these lesions. Future studies should investigate whether these polymorphisms might be used to identify patients with increased risk of recurrence or aggressive disease.

show abstract

What is the Prevalence of Undiagnosed Nevoid Basal Cell Carcinoma Syndrome in Children With an Odontogenic Keratocyst?

Cited by 10 publications

References 8 publications

Dental Screening Including Panoramic Radiograph for Gorlin-Goltz Syndrome in Patients With Multiple Basal Cell Carcinomas

Dental Screening Including Panoramic Radiograph for Gorlin-Goltz Syndrome in Patients With Multiple Basal Cell Carcinomas

Imaging of odontogenic keratocyst of the jaw by panoramic radiography: a scoping review

Gene polymorphisms in odontogenic keratocysts and ameloblastomas: A systematic review

Contact Info

Product

Resources

About