Oral‐dental findings in dyskeratosis congenita

Yavuzyilmaz, Ezel; Yamalik, Nermin; Yetgin, Sevgi; Kansu, Özden

doi:10.1111/j.1600-0714.1992.tb01011.x

Cited by 32 publications

(29 citation statements)

References 19 publications

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“…Dental radiographs were not obtainable on the 3 year old with decreased ANC. Other previously reported findings of increased caries, hypodontia and thin enamel (Wald and Diner, 1974; Yavuzyilmaz et al , 1992; Brown, 2000) were not confirmed in this study. Given the heterogeneous nature of this patient group, these findings might be found in a larger study of patients and appropriate controls.…”

Section: Discussioncontrasting

confidence: 99%

“…Several case reports have described specific oral changes in association with DC. These include oral leukoplakia, increased dental caries, hypodontia, thin enamel structure, aggressive periodontitis, intraoral brown pigmentation, tooth loss, taurodontism and blunted roots (Wald and Diner, 1974; Loh et al , 1987; Yavuzyilmaz et al , 1992; Handley and Ogden, 2006). To establish the phenotypes of various IBMFS, including DC, the Clinical Genetics Branch of the National Cancer Institute (NCI) is extensively evaluating patients and family members with these disorders.…”

Section: Introductionmentioning

confidence: 99%

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Oral and dental phenotype of dyskeratosis congenita

et al. 2008

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Dyskeratosis congenita (DC) is an inherited bone marrow failure syndrome that is characterized by lacey reticular hyperpigmentation of the skin, dystrophic nails, mucous membrane leukoplakia and pancytopenia. Diagnosis may be delayed until clinical signs are apparent. Severe pancytopenia frequently causes early mortality of DC patients, who have an increased risk of developing oropharyngeal squamous cell carcinoma. Several case reports have described oral changes in DC, which include oral leukoplakia, increased dental caries, hypodontia, thin enamel structure, aggressive periodontitis, intraoral brown pigmentation, tooth loss, taurodontism and blunted roots. We determined the prevalence of these previously reported findings in a cohort of 17 patients with DC and 23 family members. The most common oral changes in DC patients were oral leukoplakia (65% of the entire DC population), decreased root/crown ratio (75% with sufficient tooth development) and mild taurodontism (57% with sufficient tooth development). From the clinical perspective, a diagnosis of DC or other inherited bone marrow failure syndrome should be considered in young persons with oral leukoplakia, particularly those with no history of smoking. Multiple permanent teeth with decreased root/crown ratios further suggest DC.

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Section: Discussioncontrasting

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Oral and dental phenotype of dyskeratosis congenita

et al. 2008

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“…Oral manifestations-Oral, dental, and periodontal manifestations reported are leukoplakia, decreased dental crown-to-root ratio, tooth taurodontism, and early-onset aggressive periodontal disease (Wald and Diner, 1974;Yavuzyilmaz et al, 1992;Atkinson et al, 2008). Atkinson et al detected oral leukoplakia in 65% of their study population, predisposing these individuals to oropharyngeal carcinoma (Atkinson et al, 2008).…”

Section: Treatment-optimal Treatment For Patients With Dkc Is Hct Fromentioning

confidence: 99%

Periodontal and other oral manifestations of immunodeficiency diseases

2016

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The list of immunodeficiency diseases grows each year as novel disorders are discovered, classified, and sometimes reclassified due to our ever-increasing knowledge of immune system function. Although the number of patients with secondary immunodeficiencies (SIDs) greatly exceeds those with primary immunodeficiencies (PIDs), the prevalence of both appears to be on the rise probably because of scientific breakthroughs that facilitate earlier and more accurate diagnosis. Primary immunodeficiencies in adults are not as rare as once thought. Globally, the main causes of secondary immunodeficiency are HIV infection and nutritional insufficiencies. Persons with acquired immune disorders such as AIDS caused by the Human Immunodeficiency Virus (HIV) are now living long and fulfilling lives as a result of highly active antiretroviral therapy (HAART). Irrespective of whether the patient’s immune deficient state is a consequence of a genetic defect or is secondary in nature, dental and medical practitioners must be aware of the constant potential for infections and/or expressions of autoimmunity in these individuals. The purpose of this paper is to review the most common conditions resulting from primary and secondary immunodeficiency states, how they are classified, and the detrimental manifestations of these disorders on the periodontal and oral tissues.

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“…However, intracellular edema is not pathognomonic for WSN; in fact, the same aspect is present in other lesions, such as the leukoedema which is quite similar to WSN, even if it shows no parakeratosis 6. When we manage a patient affected by WSN no treatment is required onto these lesions, except for a clinical condition of a plaque which extends onto the lip vermilion: in this kind of cases, we perform a surgical exeresis for aesthetic reasons 7.…”

Section: Introductionmentioning

confidence: 99%

Oral Infection by Staphylococcus Aureus in Patients Affected by White Sponge Nevus: A Description of Two Cases Occurred in the Same Family

Marrelli

Tatullo²,

Dipalma³

et al. 2012

Int. J. Med. Sci.

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Introduction. White Sponge Nevus (WSN) is a rare pathology with a pathogenesis on genetic basis, a benign course and a localization affecting the mucosal keratin.WSN is usually a symptomless pathology: when pain is present, some authors reported reduction of symptoms by taking penicillin or oral tetracycline rinses, suggesting that a bacterial overinfection could be at the base of possible painful symptoms.Case Report. We describe 2 patients affected by WSN, father and son: they presented two different oral diseases associated with an infection by Staphylococcus aureus. So, we have performed a careful oral hygiene to reduce infection in the oral cavity. In the following days we prescribed 2 rinses a day with a mouthwash containing chlorhexidine digluconate at two different percentages.Discussion. Early diagnosis of this lesion is important, because it allows us to exclude other more serious diseases. In the most part of cases, WSN requires no treatment because of its benign and asymptomatic behaviour: up to now, no protocol of treatment for this condition was standardized. Even if WSN is a painless condition, sometime a correlated painful symptomatology was reported.Conclusions. In our experience, we have achieved excellent results even with chlorhexidine digluconate rinses, considering that our treated cases were both infected by Staphylococcus aureus.We hypothesize that the corrugated plaques and the altered texture of the mucosa create the right conditions for the colonization and the development of microbial species such as saprophytic bacteria or fungal species.

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Oral‐dental findings in dyskeratosis congenita

Cited by 32 publications

References 19 publications

Oral and dental phenotype of dyskeratosis congenita

Oral and dental phenotype of dyskeratosis congenita

Periodontal and other oral manifestations of immunodeficiency diseases

Oral Infection by Staphylococcus Aureus in Patients Affected by White Sponge Nevus: A Description of Two Cases Occurred in the Same Family

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