Chen Helen, Elhawi Mary, Tarbox Michelle
Department of Dermatology, Texas Tech University Health Sciences Center, Lubbock, USA.
Cureus. 2025 Jul 25;17(7):e88725. doi: 10.7759/cureus.88725. eCollection 2025 Jul.
Congenital linear scleroderma (CLS) is a rare connective tissue disorder characterized by unilateral linear induration that can lead to restricted mobility and limb deformities. Various treatments have been proposed, including D-penicillamine, phototherapy, oral vitamin D, and immunosuppressants such as methotrexate (MTX), cyclosporine, and interleukin-6 (IL-6) inhibitors. We present the case of a 14-year-old female patient with refractory CLS, whose condition continued to worsen despite treatment with tocilizumab, mycophenolate mofetil (MMF), and MTX. She was subsequently started on a glucagon-like peptide-1 (GLP-1) receptor agonist and began to experience improved mobility in her left arm and decreased skin hardening. To our knowledge, this is the first reported case suggesting the potential role of GLP-1 receptor agonists in slowing the progression of localized scleroderma, possibly due to their anti-fibrotic and anti-inflammatory effects.
先天性线状硬皮病（CLS）是一种罕见的结缔组织疾病，其特征为单侧线状硬结，可导致活动受限和肢体畸形。已提出多种治疗方法，包括D-青霉胺、光疗、口服维生素D以及免疫抑制剂，如甲氨蝶呤（MTX）、环孢素和白细胞介素-6（IL-6）抑制剂。我们报告了一例14岁难治性CLS女性患者的病例，尽管使用托珠单抗、霉酚酸酯（MMF）和MTX进行治疗，其病情仍持续恶化。随后她开始使用胰高血糖素样肽-1（GLP-1）受体激动剂，左臂活动能力开始改善，皮肤硬化减轻。据我们所知，这是首例提示GLP-1受体激动剂可能在减缓局限性硬皮病进展中发挥作用的报道病例，这可能归因于其抗纤维化和抗炎作用。
