Disc electrophorethic pattern of gingival collagen isolated from a patient with palmoplantar hyperkeratosis

Shoshan, S.; Finkelstein, Shifra; Rosenzweig, Kurt A.

doi:10.1111/j.1600-0765.1970.tb00725.x

Cited by 17 publications

(10 citation statements)

References 11 publications

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“…This damage may have been secondary to the inflammatory lesion, or due to a primary defect in its formation. According to Shoshan et al (1970), periodontal involvement in PLS may result from functional imbalance of collagenolytic activity in this region.…”

Section: Discussionmentioning

confidence: 99%

“…Hyperhydrosis of palms and/or soles, and non-typical nail changes also occur with varying frequency (Haneke 1979), PLS is an inherited disease with an autosomal recessive basis, and its fre-quency in the general population has been estimated at about one to four per million (Gorlin et al 1964), The cause of the di.sease is still unknown, but a number of local factors have been implicated. The syndrome was first thought to be a combination of ecto-and mesodermal malformations (Wannenmacher 1938), Possible defects of cementum (Smith & Rosenzweig 1967) and gingival epithelium (Gorlin et al 1964, Lyberg 1982, Schroeder et al, 1983, as well as a functional imbalance of coUagenolytic activity in the periodontal ligament (Shoshan et al, 1970) have also been suggested as aetiological factors. Recent studies of the subgingival plaque from PLS patients revealed a predominance of gram-anaerobic rods, including B. gingivalis.…”

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confidence: 99%

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Ultrastructure of the periodontal lesion in a case of Papillon‐Lefèvre syndronne (PLS)

Vrahopoulos

Barber

Liakoni

et al. 1988

J Clinic Periodontology

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In this study, 11 permanent teeth and their associated soft tissues from an 11-year-old boy with PLS were examined. Plaque, cementum and periodontal tissues were examined by scanning (SEM) and transmission electron microscopy (TEM). Except for depressed lymphocyte transformation, there were no abnormal haematological data. Local findings included abnormally thin cementum, extensive destruction of the periodontal ligament were still attached to the root, and severe inflammation of the soft tissues. Few bacteria were found in any of the soft tissue layers. The apical border plaque was restricted to gram- cocci and rods. The features observed in this case of PLS may indicate primary defects of cementum or ligament attachment, or disruption of fibroblast and cementoblast function due to the rapid advance of the disease process. Lack of bacterial invasion in the pocket soft tissue casts doubt on its involvement in the present case of severe periodontitis. The restricted range of morphotypes observed suggests a limited range of associated organisms. Further research is required to clarify the rôle of the host response and to identify the organisms involved.

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Section: Discussionmentioning

confidence: 99%

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confidence: 99%

Ultrastructure of the periodontal lesion in a case of Papillon‐Lefèvre syndronne (PLS)

Vrahopoulos

Barber

Liakoni

et al. 1988

J Clinic Periodontology

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“…Case no. 1 A large family of Jews from Cochin, India with 15 members affected by Papillon-Lefevre syndrome has been extensively studied over the years (Shoshan et al 1970, Hacham-Zadeh et al 1979, Shapira et ai. 1985.…”

Section: Case Reportsmentioning

confidence: 99%

Partial expression of the Papillon‐Lefèvre syndrome in 2 unrelated families

Soskolne

Stabholz

Dyke

et al. 1996

J Clinic Periodontology

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The Papillon-Lefèvre syndrome (PLS) is a rare, autosomal recessive trait that is characterized by palmar plantar keratosis (PPK) and severe, early onset periodontitis, affecting both deciduous and permanent dentitions. The clinical presentation of PLS is variable; the disease occurs so infrequently as to limit clinical cases for study. The exception is a few families with extensive consanguinity in which numerous cases occur. Of particular interest to mapping the genetic origin of the syndrome is the co-expression of the major traits of hyperkeratosis and periodontitis, and their severity. In this paper, we report 2 families with multiple affected individuals from geographically remote areas. A large extended family, from the Cochin region of India, currently residing in Israel, in which there is documented consanguinity and a family from the southwest region of Germany. In each family, 1 individual presents with hyperkeratotic lesions with the complete absence of periodontal lesions. Further, the difference in severity of the hyperkeratotic lesions between families is marked, and one sibling in the German family expressed rapid, early onset periodontitis in the absence of PPK. The genetic nature and penetrance of the genetic defect are discussed.

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“…They can cleave collagens, gelatins, elastins, fibronectins and other substrates (Woessner 1991) and also play an important role in pathological events such as periodontitis and alveolar bone destruction (Ryan et al 1996). Interestingly, the electrophoretic pattern of gingival collagen from a PLS patient showed similarity to the pattern of gingival collagen after digestion by animal collagenases (Shoshan et al 1970). Cheung et al (1982) found an elevated level of collagen synthesis by gingival fibroblasts derived from a patient with PLS, which may be interpreted as an attempt to substitute for digested components involved in the disease process.…”

Section: Discussionmentioning

confidence: 90%

Localisation of a gene for Papillon-Lefèvre syndrome to chromosome 11q14-q21 by homozygosity mapping

et al. 1997

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Papillon-Lefèvre syndrome is an autosomal recessively inherited palmoplantar keratoderma of unknown aetiology associated with severe periodontitis leading to premature loss of dentition. Three consanguineous families, two of Turkish and one of German origin, and three multiplex families, one of Ethiopian and two of German origin, with 11 affected and 6 unaffected siblings in all were studied. A targeted genome search was initially attempted to several candidate gene regions but failed to demonstrate linkage. Therefore a genome-wide linkage scan using a combination of homozygosity mapping and traditional linkage analysis was undertaken. Linkage was obtained with marker D11S937 with a maximum two-point lod score of Zmax = 6.1 at recombination fraction theta = 0.00 on chromosome 11q14-q21 near the metalloproteinase gene cluster. Multipoint likelihood calculations gave a maximum lod score of 7.35 between D11S901 and D11S1358. A 9.2-cM region homozygous by descent in the affected members of the three consanguineous families lies between markers D11S1989 and D11S4176 harbouring the as yet unknown Papillon-Lefèvre syndrome gene. Haplotype analyses in all the families studied support this localisation. This study has identified a further locus harbouring a gene for palmoplantar keratoderma and one possibly involved in periodontitis.

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Disc electrophorethic pattern of gingival collagen isolated from a patient with palmoplantar hyperkeratosis

Cited by 17 publications

References 11 publications

Ultrastructure of the periodontal lesion in a case of Papillon‐Lefèvre syndronne (PLS)

Ultrastructure of the periodontal lesion in a case of Papillon‐Lefèvre syndronne (PLS)

Partial expression of the Papillon‐Lefèvre syndrome in 2 unrelated families

Localisation of a gene for Papillon-Lefèvre syndrome to chromosome 11q14-q21 by homozygosity mapping

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