Segmental Stiff Skin Syndrome: A Rare Case Report from Indonesia

Background: This paper reports a segmental Stiff Skin Syndrome (SSS) case in a four-year-old girl.  SSS is a rare disease characterized by skin hardening and joint stiffness due to a mutation of the fibrillin-1 (FBN-1) gene encoding the fibrillin protein. The effective therapies for this disease are...

Full description

Saved in:
Bibliographic Details
Main Authors: Hafidzah Nurmastuti, Retno Danarti, Radijanti Anggraheni, Intan Arvianty, Devi Artami Susetiati, Niken Trisnowati
Format: Article
Language:Indonesian
Published: Department of Dermatology and Venereology, Faculty of Medicine, Universitas Airlangga 2025-03-01
Series:Berkala Ilmu Kesehatan Kulit dan Kelamin (Periodical of Dermatology and Venerology)
Subjects:
Online Access:https://e-journal.unair.ac.id/BIKK/article/view/64495
Tags: Add Tag
No Tags, Be the first to tag this record!
Description
Summary:Background: This paper reports a segmental Stiff Skin Syndrome (SSS) case in a four-year-old girl.  SSS is a rare disease characterized by skin hardening and joint stiffness due to a mutation of the fibrillin-1 (FBN-1) gene encoding the fibrillin protein. The effective therapies for this disease are limited. Case Report: The patient presented with hardened skin and a limping gait.  Clinically, there were hard, hyperpigmented patches with hypertrichosis on the skin of the left femur and left gluteal. The left coxae and left genu joint were in fixed flexion, causing a limping gait. The SSS histology revealed thickened collagen fibers, trapping adipocytes between them, but no inflammatory cells.  The UV-A phototherapy and physiotherapy session resulted in a modest improvement. Discussion: This case's segmental SSS diagnosis was based on clinical and histological findings. Establishing a diagnosis of SSS is a challenge for clinicians because it can resemble other skin disorders, particularly sclerosing diseases. The recommended management for SSS remains limited. In this case, after the UV-A phototherapy and physiotherapy, complaints of hardened skin were said to soften.
ISSN:1978-4279
2549-4082