Cases reported "Papillon-Lefevre Disease"

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1/7. Microbiological features of Papillon-Lefevre syndrome periodontitis.

    Papillon-Lefevre syndrome patients exhibit hyperkeratosis palmo-plantaris and severe periodontitis. The syndrome is an autosomal recessive trait, but the mechanism of periodontal destruction is not known. This report presents the clinical and microbiological features of an 11-year old girl with Papillon-Lefevre syndrome. Clinical examination included conventional periodontal measurements and radiographic analysis. In samples from 3 deep periodontal lesions, the occurrence of major suspected periodontopathic bacteria was determined by selective and non-selective culture and polymerase chain reaction (PCR) identification, and the presence of cytomegalovirus and Epstein-Barr type 1 virus by a nested-PCR detection method. 10 of 22 available teeth demonstrated severe periodontal breakdown. Major cultivable bacteria included actinobacillus actinomycetemcomitans (3.4% of total isolates), prevotella nigrescens (16.4%), fusobacterium nucleatum (14.3%) and peptostreptococcus micros (10.6%). A. actinomycetemcomitans, P. nigrescens, porphyromonas gingivalis and eikenella corrodens were identified by PCR analysis. The patient's non-affected parents and older brother revealed several periodontal pathogens but not A. actinomycetemcomitans. The viral examination demonstrated cytomegalovirus and Epstein-Barr type 1 virus in the subgingival sample of the Papillon-Lefevre syndrome patient. The father and brother yielded subgingival cytomegalovirus but not Epstein-Barr type 1 virus. We hypothesize that human herpesviruses in concert with A. actinomycetemcomitans play important roles in the development of Papillon-Lefevre syndrome periodontitis.
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2/7. Combined mechanical and antibiotic periodontal therapy in a case of Papillon-Lefevre syndrome.

    BACKGROUND: Papillon Lefevre syndrome (PLS) is a rare entity and, as such, it is almost impossible to evaluate an effective therapy in a randomized controlled study. The amount of success reported after therapy for prepubertal periodontitis (PP) in PLS is highly variable from case to case. The goal of this case report is to evaluate the effects of a combined mechanical and antibiotic periodontal therapy regimen in the management of PLS. methods: A male patient was diagnosed as suffering from PP associated with PLS at the age of 7 years. He showed hyperkeratosis of the palms and soles, as well as advanced periodontal disease already affecting permanent teeth with maximal probing depth and vertical attachment loss of 12 mm and 11 mm, respectively. Subgingival debridement was performed with simultaneous administration of oral 250 mg amoxicillin 3 times daily and 250 mg metronidazole twice daily for one week. Clinical parameters were assessed and subgingival plaque was collected from all teeth prior to therapy and 7 and 26 months after treatment. Selective cultures for A. actinomycetemcomitans were incubated for each individual tooth and DNA probe analysis was performed for various periodontal pathogens. RESULTS: Prior to combined mechanical and antibiotic treatment, all teeth but one harbored actinobacillus actinomycetemcomitans subgingivally. However, at 7 and 26 months after therapy A. actinomycetemcomitans could be detected neither by culture nor by dna probes. Clinical parameters improved markedly and teeth erupting after therapy did not exhibit attachment loss of more than 1.5 mm during the observation period. CONCLUSIONS: Eradication (suppression beneath detection levels) of A. actinomycetemcomitans seems to play a significant role in the successful treatment of localized prepubertal periodontitis in PLS.
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3/7. Papillon-Lefevre syndrome: serum immunoglobulin g (IgG) subclass antibody response to periodontopathic bacteria. A case report.

    BACKGROUND: Papillon-Lefevre syndrome (PLS) is a rare autosomal recessive disorder which is characterized by palmar-plantar hyperkeratosis and rapid periodontal destruction of both primary and permanent dentitions. In this case report, we present clinical features, and microbiological and immunological findings of 40 month-old Thai male PLS patient. methods: Microbiological examinations consisted of bacterial culture methods utilizing selective media, morphological identification, and biochemical tests. In addition, the specific serum IgG subclass antibody titers reactive with etiologic periodontal bacteria were determined by the dot-blot immunological analysis and enzyme linked immunosorbent assay (ELISA). RESULTS: The examinations revealed that the patient harbored 3 major suspected periodontopathic microorganisms, A. actinomycetemcomitans, P. gingivalis, and P. intermedia. The patient's serum IgG1, IgG2, and IgG3, but not IgG4, titers against A. actinomycetemcomitans were dramatically increased. The predominant IgG subclass was IgG1. In contrast, the IgG titers against other tested bacteria, P. gingivalis, P. intermedia, and F. nucleatum, appeared to be similar to those of a healthy control. CONCLUSIONS: A. actinomycetemcomitans seems to play a pivotal role in the bacteria-host interaction in PLS periodontal pathogenesis. Response of the specific serum IgG subclass antibody titers against the A. actinomycetemcomitans antigen has been demonstrated. This association warrants further investigation.
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4/7. Treatment of Papillon-Lefevre syndrome periodontitis.

    BACKGROUND, AIMS: Conventional mechanical treatment of Papillon-Lefevre syndrome periodontitis has a poor prognosis. This report describes an effective antimicrobial treatment of rapidly progressing periodontitis in an 11-year old girl having Papillon-Lefevre syndrome. METHOD: Clinical examination included conventional periodontal measurements and radiographic analysis. Occurrence of major suspected periodontopathic bacteria was determined by selective and non-selective culture and by polymerase chain reaction (PCR) identification. Presence of cytomegalovirus and Epstein-Barr type 1 virus was determined by a nested-PCR detection method. Therapy included scaling and root planing, oral hygiene instruction, and systemic amoxicillin-metronidazole therapy (250 mg of each/3 times daily/10 days) which, based on follow-up microbiological testing, was repeated after 4 months. Supportive periodontal therapy took place at 2 visits during a 16-month period. RESULTS: At baseline, 10 of 22 available teeth demonstrated severe periodontal breakdown. At 16 months, probing and radiographic measurements revealed no teeth with additional attachment loss, and several teeth exhibited significant reduction in gingivitis and pocket depth, increase in radiographic alveolar bone height and clinical attachment level, and radiographic evidence of crestal lamina dura. Baseline subgingival microbiota included actinobacillus actinomycetemcomitans (3.4% of total isolates), prevotella nigrescens (16.4%), Fusobacteriumnucleatum (14.3%) and peptostreptococcus micros (10.6%), as well as cytomegalovirus and Epstein-Barr type 1 virus. At termination of the study, culture and PCR examinations showed absence of A. actinomycetemcomitans, P. micros and herpesviruses, and P. nigrescens and F.nucleatum each comprised less than 0.1 % of subgingival isolates. CONCLUSION: This study suggests that controlling the periodontopathic microbiota by appropriate antibiotic and conventional periodontal therapy can arrest Papillon-Lefevre syndrome periodontitis.
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5/7. Orthodontic treatment in a patient with Papillon-Lefevre syndrome.

    BACKGROUND: Report of a combined periodontal and orthodontic treatment in a patient with Papillon-Lefevre syndrome (PLS). methods: A patient with PLS was treated orthodontically 26 months after the start of a combined mechanical and antibiotic therapy. Clinical periodontal parameters were obtained 26 (t1), 60 (t2), and 79 (t3) months after anti-infective therapy. The deepest site of each tooth was sampled for microbiological analysis at 26 and 60 months. Periodontal maintenance therapy was provided every 6 weeks. After a stable periodontal situation was achieved, orthodontic treatment, consisting of space opening for the upper canines with a multibracket appliance and coil springs, was carried out. In the lower jaw, crowding was resolved by an orthodontic mesialization of the canines. RESULTS: Twenty-six months (t1) after the beginning of the combined mechanical and antibiotic therapy, 6% of the sites exhibited 4 mm probing depth (PD) with bleeding on probing (BOP) or PD > or =5 mm. Sixty months (t2) after therapy the number of sites with 4 mm PD with BOP or PD > or =5 mm had increased to 17%, and 79 months after therapy (t3) 13% of all sites were similarly affected. From 26 to 60 months, a slight mean clinical attachment level (CAL) gain was observed, whereas the mean PD increased. From 60 to 79 months, there was a mean PD reduction. However, a significant mean attachment loss was also noted. After 26 months (t1), rna probes failed to detect A. actinomycetemcomitans, P. gingivalis, or T. forsythensis from any site. Thirty-four months later (t2), subgingival recolonization was observed. A. actinomycetemcomitans was detected by rna probes at three sites. At 26 and 60 months (t1, t2), trypticase-soy with serum, bacitracin, and vancomycin (TSBV) culture failed to detect A. actinomycetemcomitans at any of the sampled sites. Eighty-two months after the beginning of therapy (t4), none of the applied methods could detect A. actinomycetemcomitans from the pooled samples from the deepest pockets of each quadrant or the oral mucosa. In the present case, concomitant orthodontic treatment with a fixed appliance could be performed without further pronounced periodontal deterioration. Space for eruption of the canines and premolars was created, in addition to an alignment of the teeth. CONCLUSION: After a successful combined mechanical and antibiotic periodontal therapy of the PLS periodontitis, moderate orthodontic tooth movements may be possible within a complex interdisciplinary treatment regimen.
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6/7. in vitro studies of monocyte function in two siblings with Papillon-Lefevre syndrome.

    blood monocytes were isolated from two siblings with Papillon-Lefevre syndrome (PLs) and compared to corresponding cells from their healthy cousin. The number of monocytes isolated were within normal limits in all three test participants. Aggregating tendency was increased when PLs monocytes were cultured in the presence of autologous sera. The monocyte ability of specific immune phagocytosis was decreased in PLs patients. The monocyte morphology, non-specific phagocytosis, lysosomal enzyme activities, and response to E. coli endotoxin were similar in patients and control.
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7/7. Clinical, bacteriological, and immunological examinations and the treatment process of two Papillon-Lefevre syndrome patients.

    Papillon-Lefevre syndrome (PLS) is a rare disease associated with the early onset of periodontal breakdown in deciduous and permanent dentition. The etiology of the destruction has not been completely clarified. Two female patients (ages 4 and 7 years) with severe destruction of the periodontal structures were examined. Except for palmar and plantar hyperkeratosis, dermatologic examination revealed no other medical disorders. On immunological analysis, measurement of serum antibody titers to 7 periodontopathic bacteria including porphyromonas gingivalis and actinobacillus actinomycetemcomitans was performed by enzyme-linked immunosorbent assay (ELISA). Further immunoblot analysis of A. actinomycetemcomitans and microbial culture of samples collected from deep periodontal pockets and mouthrinse solution were performed. The serum of the two patients showed high IgG titer against A. actinomycetemcomitans. Immunoblot results of the two patients against sonicated extract of A. actinomycetemcomitans Y4 strain exhibited a similar pattern. The band pattern differed from that observed in other forms of early onset periodontitis patients or periodontally healthy subjects. Moreover, A. actinomycetemcomitans colonies were cultured in high percentages from the pocket samples. Antibiotic therapy was instituted in addition to conventional periodontal therapy. In the younger patient, all deciduous teeth were extracted as part of the treatment and A. actinomycetemcomitans was no longer detected. All four permanent first molars and 8 permanent incisors subsequently erupted with healthy periodontium. However, the older patient did not improve after periodontal and antibiotic (minocycline and erythromycin) treatments and A. actinomycetemcomitans was consistently detected. ofloxacin medication finally eliminated A. actinomycetemcomitans from the periodontal pockets. This antibiotic was also associated with reduced gingival inflammation and probing depth.(ABSTRACT TRUNCATED AT 250 WORDS)
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