Increased risk of pyogenic liver abscess in children with Papillon-Lefevre syndrome

Oğuzkurt, Pelin; Tanyel, Feridun Cahit; Büyükpamukçu, Nebìl; Hiçsönmez, Akgün

doi:10.1016/s0022-3468(96)90420-0

Cited by 30 publications

(36 citation statements)

References 4 publications

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“…22 These 2 factors likely contribute to the development of the liver abscess. 8 In our patient, the inflamed gingiva was the likely point of entry of S aureus that led to bacteremia and subsequently the liver abscess. Different immunologic defects have been described in patients with PLS.…”

Section: Discussionmentioning

confidence: 62%

“…The disease is characterized by diffuse PPK and juvenile periodontitis. 5,8,9 PPK usually arises during the first 4 years of life with sharply demarcated erythematous hyperkeratosis more pronounced on the soles and possibly extending to the dorsa of the hands and feet. 10 In winter, PPK may worsen, causing painful fissures that limit ambulation.…”

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

“…5,8 In 1 study of 16 patients with pyogenic liver abscess, 2 patients were found retrospectively to have PLS. 8 To date, 4 reports of 5 patients with PLS who developed pyogenic liver abscess have appeared in the English literature. 5,6,8,20 Pyogenic liver abscess usually results from the seeding of the liver by pathogenic bacteria through a hematogenous route.…”

Section: Discussionmentioning

confidence: 99%

“…8 To date, 4 reports of 5 patients with PLS who developed pyogenic liver abscess have appeared in the English literature. 5,6,8,20 Pyogenic liver abscess usually results from the seeding of the liver by pathogenic bacteria through a hematogenous route. 21 The most common etiologic agent is S aureus, and most often a solitary abscess is found.…”

Section: Discussionmentioning

confidence: 99%

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Pyogenic Liver Abscess and Papillon-Lefèvre Syndrome: Not a Rare Association

Almuneef¹,

Khenaizan

Ajaji³

et al. 2003

Pediatrics

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ABSTRACT. Papillon-Lefèvre syndrome is a rare, autosomal recessive disease comprising palmoplantar keratoderma and periodontitis. Pyogenic liver abscess is an increasingly recognized complication. We report a new case of this association and review the current literature. Pediatrics 2003;111:e85-e88. URL: http://www.pediatrics. org/cgi/content/full/111/1/e85; Papillon-Lefèvre syndrome, liver abscess.

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Section: Discussionmentioning

confidence: 62%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

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Pyogenic Liver Abscess and Papillon-Lefèvre Syndrome: Not a Rare Association

Almuneef¹,

Khenaizan

Ajaji³

et al. 2003

Pediatrics

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A novel mutation in the cathepsin C (CTSC)gene in Iranian family withPapillon‐Lefevresyndrome

Ghanei

Abbaszadegan

Forghanifard

et al. 2021

Clinical & Exp Dental Res

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Objectives: In this study, we analyzed the whole exomes of CTSC gene in a family with history of PLS.Materials and methods: Genomic DNA was extracted from peripheral blood and genotype analysis was performed. The mutated protein sequence was used to find the best possible tertiary structure for homology modeling. The homology modeling of the novel mutation was then performed using the online Swiss-Prot server. The results were also analyzed for to verify its validity.Results: The analysis of CTSC gene elucidated a novel insertion GAC. The novel mutation was proved by analyzing 50 healthy control volunteers. Modeling of the novel found mutation in CTSC gene revealed structural defects that may have caused the functional abnormalities. Conclusions:The structural analysis of the mutated protein model identifies changes in the stereo-chemical and the energy level of the mutated protein. Since this protein play a role in the activation of granule serine proteases from cytotoxic T lymphocytes, natural killer cells, mast cells, such structural defects may lead to its malfunction causing dysfunctioning of immune defense mechanisms.

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Review Paper: Dental Treatment for Patients with Papillon-Lefèvre Syndrome (PLS)

Ullbro

Twetman

2007

Eur Arch Paediatr Dent

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Increased risk of pyogenic liver abscess in children with Papillon-Lefevre syndrome

Cited by 30 publications

References 4 publications

Pyogenic Liver Abscess and Papillon-Lefèvre Syndrome: Not a Rare Association

Pyogenic Liver Abscess and Papillon-Lefèvre Syndrome: Not a Rare Association

A novel mutation in the cathepsin C (CTSC)gene in Iranian family withPapillon‐Lefevresyndrome

Review Paper: Dental Treatment for Patients with Papillon-Lefèvre Syndrome (PLS)

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