Periodontitis associated with plasminogen deficiency: a case report

Abstract

BackgroundPlasminogen deficiency is a rare autosomal recessive disease, which is associated with aggressive periodontitis and gingival enlargement. Previously described treatments of plasminogen deficiency associated periodontitis have shown limited success. This is the first case report indicating a successful therapy approach consisting of a non-surgical supra- and subgingival debridement in combination with an adjunctive systemic antibiotic therapy and a strict supportive periodontal regimen over an observation period of 4 years.Case presentationThe intraoral examination of a 17-year-old Turkish female with severe plasminogen deficiency revealed generalized increased pocket probing depths ranging from 6 to 9 mm, bleeding on probing over 30%, generalized tooth mobility, and gingival hyperplasia. Alveolar bone loss ranged from 30% to 50%. Clinical attachment loss corresponded to pocket probing depths. Aggregatibacter actinomycetemcomitans, Porphyromonas gingivalis, Treponema denticola, Prevotella intermedia, Prevotella nigrescens and Eikenella corrodens have been detected by realtime polymerase chain reaction. Periodontal treatment consisted of full mouth disinfection and adjunctive systemic administration of amoxicillin (500 mg tid) and metronidazole (400 mg tid). A strict supportive periodontal therapy regimen every three month in terms of supra- and subgingival debridement was rendered. The reported therapy has significantly improved periodontal health and arrested disease progression. Intraoral examination at the end of the observation period 3.5 years after non-surgical periodontal therapy showed generalized decreased pocket probing depths ranging from 1 to 6 mm, bleeding on probing lower 30%, and tooth mobility class I and II. Furthermore, microbiological analysis shows the absence of Porphyromonas gingivalis, Prevotella intermedia and Treponema denticola after therapy.ConclusionAdjunctive antibiotic treatment may alter the oral microbiome and thus, the inflammatory response of periodontal disease associated to plasminogen deficiency and diminishes the risk of pseudomembrane formation and progressive attachment loss.This case report indicates that patients with plasminogen deficiency may benefit from non-surgical periodontal treatment in combination with an adjunctive antibiotic therapy and a strict supportive periodontal therapy regimen.

Ligneous Periodontitis in a Patient with Type 1 Plasminogen Deficiency: A Case Report and Review of the Literature

“…The fact that only 32% of patients who suffer from PLG Type 1 deficiency develop ligneous periodontitis strongly supports the notion that external triggers, i.e., trauma or infection, may play an Case Reports in Dentistry additional significant role in the pathogenesis of this disease [5,10,31]. The average age of patients reported in the literature with ligneous periodontitis is 12-18 years, while isolated cases have been reported in older patients; these differences may reflect variability in PLG activity due to different plasminogen gene mutations [32]. Histopathological examination was done to confirm the diagnosis.…”

“…Several therapeutic approaches to reduce the bacterial load to decrease the inflammation and thus the progression of the disease have been attempted; these procedures include scaling and root planing, chlorhexidine rinsing, administration of antibiotics, and periodontal surgery [9,25,30]. Neering et al have reported that patients with PLG-deficiency Type I may benefit from nonsurgical periodontal therapy including full mouth disinfection in combination with adjunctive antibiotic therapy and a strict supportive periodontal therapy regime every three months [32]. Most of the reports have been described as failures due to rapid gingival regrowth and progressive bone loss [5].…”

Fibrin is a critical regulator of neutrophil effector function at mucosal barrier sites

“…Indeed, such patients present with deposition of fibrin at various mucosal sites leading to ocular disease (conjunctivitis), oral mucosal disease (ligneous periodontitis), lung, vaginal and gastrointestinal tract involvement [3][4][5]. In the oral mucosa, local deposition of fibrin is hypothesized to lead to severe soft tissue and bone destruction around teeth and often loss of the entire dentition in adolescence [6][7][8][9]. Mucosal inflammation in areas surrounding the dentition and destruction of underlying bone are also the hallmarks of the common human oral mucosal disease, periodontitis [10,11].…”

Genetic Susceptibility Contributing to Periodontal and Cardiovascular Disease

“…Although the PD risk allele of rs4252120 was found to be strongly associated with lipoprotein(a) level and CAD risk (Qi et al 2012), LPA does not seem to represent a so-far-undetected genetic risk factor shared between PD and CAD/MI. The fact that PLG deficiency-a rare autosomal recessive disease (1.6 in 1 million individuals) caused by mutations of PLG-is known to be associated with severe early-onset PD (Neering et al 2015) suggests that PLG may be directly and causally related to AgP rather than due to linkage disequilibrium with the CAD-associated LPA SNPs.…”