Literature DB >> 8910840

Early-onset periodontitis associated with Weary-Kindler syndrome: a case report.

C B Wiebe1, J G Silver, H S Larjava.   

Abstract

Weary-Kindler syndrome is a rare and poorly understood genetic disorder that has manifestations of both epidermolysis bullosa and poikiloderma congenitale. There are approximately 70 cases documented in the past 40 years but no cases appear in the dental literature, although dental findings have been discussed superficially in dermatological and pediatric publications. This case reports on the periodontal findings and treatment for a 16-year-old female diagnosed with the syndrome. Early exfoliation of deciduous teeth, severe periodontal bone loss around many permanent teeth, and fragile bleeding gingiva were key features. Microbiological testing revealed an absence of Actinobacillus actinomycetemcomitans and low levels of other commonly accepted periodontal pathogens. Tests for inflammation, including AST and elastase, were positive prior to therapy and greatly decreased after mechanical root instrumentation. A beneficial effect of non-surgical periodontal therapy was observed in the short-term follow-up.

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Year:  1996        PMID: 8910840     DOI: 10.1902/jop.1996.67.10.1004

Source DB:  PubMed          Journal:  J Periodontol        ISSN: 0022-3492            Impact factor:   6.993


  8 in total

1.  Loss of kindlin-1, a human homolog of the Caenorhabditis elegans actin-extracellular-matrix linker protein UNC-112, causes Kindler syndrome.

Authors:  Dawn H Siegel; Gabrielle H S Ashton; Homero G Penagos; James V Lee; Heidi S Feiler; Kirk C Wilhelmsen; Andrew P South; Frances J D Smith; Alan R Prescott; Vesarat Wessagowit; Noritaka Oyama; Masashi Akiyama; Daifullah Al Aboud; Khalid Al Aboud; Ahmad Al Githami; Khalid Al Hawsawi; Abla Al Ismaily; Raouf Al-Suwaid; David J Atherton; Ruggero Caputo; Jo-David Fine; Ilona J Frieden; Elaine Fuchs; Richard M Haber; Takashi Harada; Yasuo Kitajima; Susan B Mallory; Hideoki Ogawa; Sedef Sahin; Hiroshi Shimizu; Yasushi Suga; Gianluca Tadini; Kikuo Tsuchiya; Colin B Wiebe; Fenella Wojnarowska; Adel B Zaghloul; Takahiro Hamada; Rajeev Mallipeddi; Robin A J Eady; W H Irwin McLean; John A McGrath; Ervin H Epstein
Journal:  Am J Hum Genet       Date:  2003-06-03       Impact factor: 11.025

2.  Odontogenic ameloblast-associated and amelotin are novel basal lamina components.

Authors:  Juliana Dos Santos Neves; Rima M Wazen; Shingo Kuroda; Sylvia Francis Zalzal; Pierre Moffatt; Antonio Nanci
Journal:  Histochem Cell Biol       Date:  2012-01-10       Impact factor: 4.304

3.  Localization and potential function of kindlin-1 in periodontal tissues.

Authors:  Giorgio Petricca; Mari Leppilampi; Guoqiao Jiang; Gethin R Owen; Colin Wiebe; Yizeng Tu; Leeni Koivisto; Lari Häkkinen; Chuanyue Wu; Hannu Larjava
Journal:  Eur J Oral Sci       Date:  2009-10       Impact factor: 2.612

Review 4.  Oral Alterations in Heritable Epidermolysis Bullosa: A Clinical Study and Literature Review.

Authors:  Alessandro Polizzi; Simona Santonocito; Romeo Patini; Vincenzo Quinzi; Stefano Mummolo; Rosalia Leonardi; Alberto Bianchi; Gaetano Isola
Journal:  Biomed Res Int       Date:  2022-05-31       Impact factor: 3.246

Review 5.  Kindler syndrome and periodontal disease: review of the literature and a 12-year follow-up case.

Authors:  Colin B Wiebe; Giorgio Petricca; Lari Häkkinen; Guoqiao Jiang; Chuanyue Wu; Hannu S Larjava
Journal:  J Periodontol       Date:  2008-05       Impact factor: 6.993

6.  Management of a granulomatous lesion in a patient with Kindler's Syndrome.

Authors:  Anuradha Bhatsange; Yugandhara Khadse; Sabina Deshmukh; Swapnil Karwa
Journal:  J Indian Soc Periodontol       Date:  2018 Jan-Feb

7.  Clinical practice guidelines: Oral health care for children and adults living with epidermolysis bullosa.

Authors:  Susanne Krämer; James Lucas; Francisca Gamboa; Miguel Peñarrocha Diago; David Peñarrocha Oltra; Marcelo Guzmán-Letelier; Sanchit Paul; Gustavo Molina; Lorena Sepúlveda; Ignacio Araya; Rubén Soto; Carolina Arriagada; Anne W Lucky; Jemima E Mellerio; Roger Cornwall; Fatimah Alsayer; Reinhard Schilke; Mark Adam Antal; Fernanda Castrillón; Camila Paredes; Maria Concepción Serrano; Victoria Clark
Journal:  Spec Care Dentist       Date:  2020-11

8.  Kindler's syndrome: A rare case report.

Authors:  Neelam Suman; Simrat Kaur; Supreet Kaur; Vandana Sarangal
Journal:  Contemp Clin Dent       Date:  2014-04
  8 in total

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