Sublingual Abnormalities in Systemic Sclerosis

Tracy M Frech, John D Pauling, Maureen A Murtaugh, Katherine Kendall, Robyn T Domsic

Research output: Contribution to journalArticle

7 Citations (Scopus)

Abstract

BACKGROUND: Sublingual frenulum abnormalities have been observed in systemic sclerosis (SSc), but the clinical significance of such features is not known. The goal of this project was to devise a reliable bedside tool to confirm the presence of sublingual frenulum abnormalities in SSc and explore potential associations with patient demographics and gastrointestinal clinical phenotype.

METHODS: A working group was created to develop a semiquantitative assessment tool for assessing sublingual abnormalities, the Sublingual Abnormalities Index (SAI). Sublingual frenulum thickness, frenulum length, sublingual buccal mucosa pallor, and the presence of oral telangiectasia were each individually scored using 0- to 2-point Likert scales and a composite score of the 4 domains created by summation of the individual scores. Assessment of the sublingual region of 21 patients with SSc and 8 control subjects was undertaken. An image of the sublingual frenulum was obtained using prespecified camera settings to allow assessment of interrater reliability with 2 independent blinded assessors.

RESULTS: Scores for each of the SAI domains differed between control subject and SSc population (P = 0.0003). Patients with SSc had a composite SAI score of 4.3 (SD, 0.37). None of the control subjects had a composite SAI score of more than 2. There was excellent interrater reliability between clinician assessment and each blinded assessor (Cohen κ's of 0.72 and 0.82, respectively).

CONCLUSIONS: This feasibility study confirms the presence of clinical sublingual abnormalities in SSc, which can be categorized using a simple scoring chart with moderate to near-perfect interrater agreement. The functional significance and pathogenesis of this abnormality warrant further study.

Original languageEnglish
Pages (from-to)19-21
Number of pages3
JournalJCR : Journal of Clinical Rheumatology
Volume22
Issue number1
DOIs
Publication statusPublished - Jan 2016

Fingerprint

Systemic Scleroderma
Pallor
Mouth Floor
Telangiectasis
Mouth Mucosa
Feasibility Studies
Demography
Phenotype
Population

Keywords

  • Feasibility Studies
  • Female
  • Humans
  • Lingual Frenum
  • Male
  • Middle Aged
  • Mouth Mucosa
  • Reproducibility of Results
  • Scleroderma, Systemic
  • Severity of Illness Index
  • Tongue Diseases
  • Journal Article
  • Research Support, N.I.H., Extramural

Cite this

Sublingual Abnormalities in Systemic Sclerosis. / Frech, Tracy M; Pauling, John D; Murtaugh, Maureen A; Kendall, Katherine; Domsic, Robyn T.

In: JCR : Journal of Clinical Rheumatology , Vol. 22, No. 1, 01.2016, p. 19-21.

Research output: Contribution to journalArticle

Frech, Tracy M ; Pauling, John D ; Murtaugh, Maureen A ; Kendall, Katherine ; Domsic, Robyn T. / Sublingual Abnormalities in Systemic Sclerosis. In: JCR : Journal of Clinical Rheumatology . 2016 ; Vol. 22, No. 1. pp. 19-21.
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T1 - Sublingual Abnormalities in Systemic Sclerosis

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AU - Pauling, John D

AU - Murtaugh, Maureen A

AU - Kendall, Katherine

AU - Domsic, Robyn T

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N2 - BACKGROUND: Sublingual frenulum abnormalities have been observed in systemic sclerosis (SSc), but the clinical significance of such features is not known. The goal of this project was to devise a reliable bedside tool to confirm the presence of sublingual frenulum abnormalities in SSc and explore potential associations with patient demographics and gastrointestinal clinical phenotype.METHODS: A working group was created to develop a semiquantitative assessment tool for assessing sublingual abnormalities, the Sublingual Abnormalities Index (SAI). Sublingual frenulum thickness, frenulum length, sublingual buccal mucosa pallor, and the presence of oral telangiectasia were each individually scored using 0- to 2-point Likert scales and a composite score of the 4 domains created by summation of the individual scores. Assessment of the sublingual region of 21 patients with SSc and 8 control subjects was undertaken. An image of the sublingual frenulum was obtained using prespecified camera settings to allow assessment of interrater reliability with 2 independent blinded assessors.RESULTS: Scores for each of the SAI domains differed between control subject and SSc population (P = 0.0003). Patients with SSc had a composite SAI score of 4.3 (SD, 0.37). None of the control subjects had a composite SAI score of more than 2. There was excellent interrater reliability between clinician assessment and each blinded assessor (Cohen κ's of 0.72 and 0.82, respectively).CONCLUSIONS: This feasibility study confirms the presence of clinical sublingual abnormalities in SSc, which can be categorized using a simple scoring chart with moderate to near-perfect interrater agreement. The functional significance and pathogenesis of this abnormality warrant further study.

AB - BACKGROUND: Sublingual frenulum abnormalities have been observed in systemic sclerosis (SSc), but the clinical significance of such features is not known. The goal of this project was to devise a reliable bedside tool to confirm the presence of sublingual frenulum abnormalities in SSc and explore potential associations with patient demographics and gastrointestinal clinical phenotype.METHODS: A working group was created to develop a semiquantitative assessment tool for assessing sublingual abnormalities, the Sublingual Abnormalities Index (SAI). Sublingual frenulum thickness, frenulum length, sublingual buccal mucosa pallor, and the presence of oral telangiectasia were each individually scored using 0- to 2-point Likert scales and a composite score of the 4 domains created by summation of the individual scores. Assessment of the sublingual region of 21 patients with SSc and 8 control subjects was undertaken. An image of the sublingual frenulum was obtained using prespecified camera settings to allow assessment of interrater reliability with 2 independent blinded assessors.RESULTS: Scores for each of the SAI domains differed between control subject and SSc population (P = 0.0003). Patients with SSc had a composite SAI score of 4.3 (SD, 0.37). None of the control subjects had a composite SAI score of more than 2. There was excellent interrater reliability between clinician assessment and each blinded assessor (Cohen κ's of 0.72 and 0.82, respectively).CONCLUSIONS: This feasibility study confirms the presence of clinical sublingual abnormalities in SSc, which can be categorized using a simple scoring chart with moderate to near-perfect interrater agreement. The functional significance and pathogenesis of this abnormality warrant further study.

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KW - Female

KW - Humans

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KW - Male

KW - Middle Aged

KW - Mouth Mucosa

KW - Reproducibility of Results

KW - Scleroderma, Systemic

KW - Severity of Illness Index

KW - Tongue Diseases

KW - Journal Article

KW - Research Support, N.I.H., Extramural

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DO - 10.1097/RHU.0000000000000339

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VL - 22

SP - 19

EP - 21

JO - JCR : Journal of Clinical Rheumatology

JF - JCR : Journal of Clinical Rheumatology

SN - 1076-1608

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ER -