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      Dental Findings in Patients With Non-surgical Hypoparathyroidism and Pseudohypoparathyroidism: A Systematic Review


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          Background: Dental aberrations have been mentioned in relation to non-surgical hypoparathyroidism (Ns-HypoPT) and pseudohypoparathyroidism (PHP). However, a systematic review of dental characteristics have not been performed. The present systematic review describes the dental findings in patients with Ns-HypoPT and PHP.

          Methods: Studies on Ns-HypoPT and PHP reporting dental features were eligible. A systematic literature search was conducted using four bibliographic databases (Web of Science, Scopus, Pubmed, and Embase) and was limited to studies written in English. Reviews, meta-analyses and letters were excluded. Both the research and reporting of results were based on PRISMA (preferred Reporting Items for Systematic Reviews and Meta-Analysis) guidelines.

          Results: Of 88 studies included, nine were cross-sectional, one was a prospective cohort study, 26 were case series, and 52 were case reports. The most frequently reported findings in patients with Ns-HypoPT were enamel opacities, enamel hypoplasia, hypodontia, and eruption disturbances. In patients with PHP, enamel hypoplasia, eruption disturbance, and deviation of the root morphology were the most frequently reported findings.

          Conclusion: An association between enamel hypoplasia and Ns-HypoPT and PHP is likely. The results should, however, be interpreted cautiously due to the limited number of high-quality studies. The present review confirms the need of further well-designed studies, such as large-scale studies, e.g., multicenter studies, to conclude on the reported associations between Ns-HypoPT/PHP and enamel hypoplasia.

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          Clinical variation of autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED) in a series of 68 patients.

          To define the clinical picture and course of the autosomal recessive disease called autoimmune polyendocrinopathy-candidiasis-ectodermal dystrophy (APECED), we report data from our 10-month to 31-year follow-up of 68 patients from 54 families, now 10 months to 53 years of age. The clinical manifestations varied greatly and included from one to eight disease components, 63 percent of the patients having three to five of them. The initial manifestation was oral candidiasis in 41 patients (60 percent), intestinal malabsorption in 6 (9 percent), and keratopathy in 2 (3 percent). All the patients had candidiasis at some time. The earliest endocrine component appeared at 19 months to 35 years of age. Hypoparathyroidism was present in 54 patients (79 percent), adrenocortical failure in 49 (72 percent), and gonadal failure in 15 (60 percent) of the female patients greater than or equal to 13 years of age and 4 (14 percent) of the male patients greater than or equal to 16 years of age. There were multiple endocrine deficiencies in half the patients. From 4 to 29 percent of the patients had periodic malabsorption, gastric parietal-cell atrophy, hepatitis, alopecia, vitiligo, or a combination of these conditions. Dental-enamel hypoplasia and keratopathy were also frequent but were not attributable to hypoparathyroidism. In the patients whose initial manifestation (other than candidiasis) was adrenal failure, the other components developed less often than in the remaining patients. We conclude that the clinical spectrum in patients with APECED is broad. The majority of patients have three to five manifestations, some of which may not appear until the fifth decade. Therefore, all patients need lifelong follow-up for the detection of new components of the disease.
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            Hypoparathyroidism in the adult: epidemiology, diagnosis, pathophysiology, target-organ involvement, treatment, and challenges for future research.

            Recent advances in understanding the epidemiology, genetics, diagnosis, clinical presentations, skeletal involvement, and therapeutic approaches to hypoparathyroidism led to the First International Workshop on Hypoparathyroidism that was held in 2009. At this conference, a group of experts convened to discuss these issues with a view towards a future research agenda for this disease. This review, which focuses primarily on hypoparathyroidism in the adult, provides a comprehensive summary of the latest information on this disease. Copyright © 2011 American Society for Bone and Mineral Research.
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              Strength of recommendation taxonomy (SORT): a patient-centered approach to grading evidence in the medical literature.

              A large number of taxonomies are used to rate the quality of an individual study and the strength of a recommendation based on a body of evidence. We have developed a new grading scale that will be used by several family medicine and primary care journals (required or optional), with the goal of allowing readers to learn one taxonomy that will apply to many sources of evidence. Our scale is called the Strength of Recommendation Taxonomy. It addresses the quality, quantity, and consistency of evidence and allows authors to rate individual studies or bodies of evidence. The taxonomy is built around the information mastery framework, which emphasizes the use of patient-oriented outcomes that measure changes in morbidity or mortality. An A-level recommendation is based on consistent and good-quality patient-oriented evidence; a B-level recommendation is based on inconsistent or limited-quality patient-oriented evidence; and a C-level recommendation is based on consensus, usual practice, opinion, disease-oriented evidence, or case series for studies of diagnosis, treatment, prevention, or screening. Levels of evidence from 1 to 3 for individual studies also are defined. We hope that consistent use of this taxonomy will improve the ability of authors and readers to communicate about the translation of research into practice.

                Author and article information

                Front Physiol
                Front Physiol
                Front. Physiol.
                Frontiers in Physiology
                Frontiers Media S.A.
                19 June 2018
                : 9
                [1] 1Section for Pediatric Dentistry, Department of Dentistry and Oral Health, Health, Aarhus University , Aarhus, Denmark
                [2] 2Department of Endocrinology and Internal Medicine, Aarhus University Hospital , Aarhus, Denmark
                [3] 3Center for Oral Health in Rare Diseases, Department of Maxillofacial Surgery, Aarhus University Hospital , Aarhus, Denmark
                [4] 4Faculté de Chirurgie Dentaire, Institut d'Etudes Avancées, USIAS, FMTS, RARENET Interreg V, Université de Strasbourg , Strasbourg, France
                [5] 5Pôle de Médecine et Chirurgie Bucco-Dentaires, Centre de Référence des Maladies Rares Orales et Dentaires, O-Rares, Hôpitaux Universitaires de Strasbourg , Strasbourg, France
                [6] 6Institut de Génétique et de Biologie Moléculaire and Cellulaire, Centre Européen de Recherche en Biologie et en Médecine, Université de Strasbourg, Centre National de la Recherche Scientifique UMR7104, Institut National de la Santé et de la Recherche Médicale U964 , Illkirch, France
                Author notes

                Edited by: Catherine Chaussain, Université Paris Descartes, France

                Reviewed by: Ana Carolina Acevedo, University of Brasília, Brazil; Elvire Le Norcy, Université Paris Descartes, France

                *Correspondence: Dorte Haubek dorte.haubek@ 123456dent.au.dk

                This article was submitted to Craniofacial Biology and Dental Research, a section of the journal Frontiers in Physiology

                Copyright © 2018 Hejlesen, Underbjerg, Gjørup, Bloch-Zupan, Sikjaer, Rejnmark and Haubek.

                This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

                Page count
                Figures: 2, Tables: 6, Equations: 0, References: 105, Pages: 15, Words: 11086
                Funded by: Tandlægeforeningen 10.13039/100008364
                Systematic Review

                Anatomy & Physiology
                hypoparathyroidism,pseudohypoparathyroidism,22q11 deletion syndrome,dental anomalies,enamel hypoplasia


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