Literature DB >> 33498938

Prepubertal Periodontitis in a Patient with Combined Classical and Periodontal Ehlers-Danlos Syndrome.

Friedrich Stock1, Marcel Hanisch2, Sarah Lechner3, Saskia Biskup3, Axel Bohring1, Johannes Zschocke4, Ines Kapferer-Seebacher5.   

Abstract

We report an extremely rare case of combined classical and periodontal Ehlers-Danlos syndrome (EDS) with early severe periodontitis and a generalized lack of attached gingiva. A German family with classical EDS was investigated by physical and dental evaluation and exome and Sanger sequencing. Due to the specific periodontal phenotype in the affected child, an additional diagnosis of periodontal EDS was suspected. Physical and genetic examination of two affected and three unaffected family members revealed a family diagnosis of classical EDS with a heterozygous mutation in COL5A1 (c.1502del; p.Pro501Leufs*57). Additional to the major clinical criteria for classical EDS-generalized joint hypermobility, hyperelastic skin, and atrophic scarring -the child aged four years presented with generalized alveolar bone loss up to 80%, early loss of two lower incisors, severe gingival recession, and generalized lack of attached gingiva. Due to these clinical findings, an additional diagnosis of periodontal EDS was suspected. Further genetic analysis revealed the novel missense mutation c.658T>G (p.Cys220Gly) in C1R in a heterozygous state. Early severe periodontitis in association with generalized lack of attached gingiva is pathognomonic for periodontal EDS and led to the right clinical and genetic diagnosis in the present case.

Entities:  

Keywords:  C1R; Ehlers−Danlos syndrome; complement; periodontitis; type V collagen

Year:  2021        PMID: 33498938      PMCID: PMC7912441          DOI: 10.3390/biom11020149

Source DB:  PubMed          Journal:  Biomolecules        ISSN: 2218-273X


  21 in total

1.  Ehlers-Danlos syndromes: revised nosology, Villefranche, 1997. Ehlers-Danlos National Foundation (USA) and Ehlers-Danlos Support Group (UK).

Authors:  P Beighton; A De Paepe; B Steinmann; P Tsipouras; R J Wenstrup
Journal:  Am J Med Genet       Date:  1998-04-28

2.  The key role of the dental practitioner in early diagnosis of periodontal Ehlers-Danlos syndromes: a rare case report of siblings.

Authors:  Jorge Cortés-Bretón Brinkmann; Ignacio García-Gil; Diana Marina Lobato-Peña; Constanza Martínez-Mera; Maria Jesús Suárez-García; Jose María Martínez-González; Maria Rioboo
Journal:  Quintessence Int       Date:  2021       Impact factor: 1.677

3.  A Chinese family with periodontal Ehlers-Danlos syndrome associated with missense mutation in the C1R gene.

Authors:  Juan Wu; Jie Yang; Jie Zhao; Jingrong Wu; Xuan Zhang; Wai Keung Leung; Weibin Sun
Journal:  J Clin Periodontol       Date:  2018-11       Impact factor: 8.728

4.  Identification of the different salivary Interleukin-6 profiles in patients with periodontitis: A cross-sectional study.

Authors:  Gaetano Isola; Antonino Lo Giudice; Alessandro Polizzi; Angela Alibrandi; Paolo Murabito; Francesco Indelicato
Journal:  Arch Oral Biol       Date:  2020-11-30       Impact factor: 2.633

Review 5.  Role of complement in host-microbe homeostasis of the periodontium.

Authors:  George Hajishengallis; Toshiharu Abe; Tomoki Maekawa; Evlambia Hajishengallis; John D Lambris
Journal:  Semin Immunol       Date:  2013-05-16       Impact factor: 11.130

6.  Bi-allelic Alterations in AEBP1 Lead to Defective Collagen Assembly and Connective Tissue Structure Resulting in a Variant of Ehlers-Danlos Syndrome.

Authors:  Patrick R Blackburn; Zhi Xu; Kathleen E Tumelty; Rose W Zhao; William J Monis; Kimberly G Harris; Jennifer M Gass; Margot A Cousin; Nicole J Boczek; Mario V Mitkov; Mark A Cappel; Clair A Francomano; Joseph E Parisi; Eric W Klee; Eissa Faqeih; Fowzan S Alkuraya; Matthew D Layne; Nazli B McDonnell; Paldeep S Atwal
Journal:  Am J Hum Genet       Date:  2018-03-29       Impact factor: 11.025

7.  Prospective clinical investigations of children with periodontal Ehlers-Danlos syndrome identify generalized lack of attached gingiva as a pathognomonic feature.

Authors:  Ines Kapferer-Seebacher; Elizabeth Oakley-Hannibal; Ulrike Lepperdinger; Diana Johnson; Neeti Ghali; Angela F Brady; Glenda Sobey; Johannes Zschocke; Fleur S van Dijk
Journal:  Genet Med       Date:  2020-10-02       Impact factor: 8.822

8.  Ehlers-Danlos syndrome hypermobility type is associated with rheumatic diseases.

Authors:  Kyla R Rodgers; Jiang Gui; Mary Beth P Dinulos; Richard C Chou
Journal:  Sci Rep       Date:  2017-01-04       Impact factor: 4.379

9.  Periodontal Ehlers-Danlos syndrome is associated with leukoencephalopathy.

Authors:  Ines Kapferer-Seebacher; Quinten Waisfisz; Sylvia Boesch; Marieke Bronk; Peter van Tintelen; Elke R Gizewski; Rebekka Groebner; Johannes Zschocke; Marjo S van der Knaap
Journal:  Neurogenetics       Date:  2018-12-08       Impact factor: 2.660

10.  C1R Mutations Trigger Constitutive Complement 1 Activation in Periodontal Ehlers-Danlos Syndrome.

Authors:  Rebekka Gröbner; Ines Kapferer-Seebacher; Albert Amberger; Rita Redolfi; Fabien Dalonneau; Erik Björck; Di Milnes; Isabelle Bally; Veronique Rossi; Nicole Thielens; Heribert Stoiber; Christine Gaboriaud; Johannes Zschocke
Journal:  Front Immunol       Date:  2019-11-05       Impact factor: 7.561

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  2 in total

1.  Tissue Regeneration and Physiological Functional Recovery in Dental and Craniofacial Fields.

Authors:  Takehito Ouchi; Taneaki Nakagawa
Journal:  Biomolecules       Date:  2021-11-06

Review 2.  Oral manifestations of Ehlers-Danlos syndromes.

Authors:  Ulrike Lepperdinger; Johannes Zschocke; Ines Kapferer-Seebacher
Journal:  Am J Med Genet C Semin Med Genet       Date:  2021-11-06       Impact factor: 3.359

  2 in total

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