# Intraoral Involvement in Linear Scleroderma En Coup De Sabre: A Case Report

**Authors:** Nuha A Alkanhal, Haifa AlKhodier

PMC · DOI: 10.7759/cureus.55886 · Cureus · 2024-03-10

## TL;DR

This case report describes a rare instance of linear scleroderma in a child that affected the mouth and led to dental and muscular complications.

## Contribution

The paper presents a rare case of oral involvement in linear scleroderma en coup de sabre in a pediatric patient.

## Key findings

- The patient exhibited hypotrophied mylohyoid muscle and reduced alveolar bone height.
- The patient had a deviated midline and was classified as high risk for dental caries.
- Comprehensive dental treatment was successfully completed after multidisciplinary consultation.

## Abstract

Localized scleroderma is a connective tissue disorder that causes excessive collagen deposition and skin fibrosis. It can be subdivided into morphea and linear scleroderma. En coup de sabre (ECDS) is a rare variant of linear scleroderma typically found among children. It is usually treated with methotrexate and corticotherapy in addition to folic acid supplements. To date, few cases of ECDS have been reported with oral involvement. This case report discusses a seven-year-old girl with linear scleroderma ECDS who was referred to the dental clinic to evaluate muscular hypotrophy on the floor of the mouth. Upon clinical and radiographic examination, the patient had hypotrophied mylohyoid muscle, reduced alveolar bone height on the affected side, and a deviated midline to the affected side as well. Furthermore, the patient was classified as having a high caries risk. After consultation with the primary physician regarding treatment modalities and options, the patient completed her comprehensive dental treatment at the Dental Department at King Faisal Specialist Hospital and Research Centre in Riyadh, Saudi Arabia.

## Linked entities

- **Chemicals:** methotrexate (PubChem CID 4112), folic acid (PubChem CID 135398658)
- **Diseases:** linear scleroderma (MONDO:0043294), morphea (MONDO:0019562), dental caries (MONDO:0005276)

## Full-text entities

- **Diseases:** hypotrophied mylohyoid muscle (MESH:D019042), skin fibrosis (MESH:D005355), caries (MESH:D003731), muscular hypotrophy (MESH:D009135), collagen (MESH:D003095), ECDS (MESH:D012594)
- **Species:** Homo sapiens (human, species) [taxon 9606]

## Full text

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## Figures

6 figures with captions in the complete paper: https://tomesphere.com/paper/PMC11003326/full.md

## References

20 references — full list in the complete paper: https://tomesphere.com/paper/PMC11003326/full.md

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Source: https://tomesphere.com/paper/PMC11003326