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A Case Report of Successful Treatment of Recalcitrant Childhood Localized Scleroderma with Infliximab and Leflunomide.

Ferguson ID, Weiser P, Torok KS - Open Rheumatol J (2015)

Bottom Line: Herein we report successful treatment of an adolescent Caucasian female with severe progressive localized scleroderma (mixed subtype, including generalized morphea and linear scleroderma of the trunk/limb) using infliximab and leflunomide.The patient demonstrated improvement after the first 9 months of therapy based on her clinical examination, objective measures, and patient and parent global assessments.Larger longitudinal studies or case series are needed to confirm and further investigate infliximab's role in localized scleroderma.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, Division of Rheumatology, Children's Hospital of Pittsburgh of the University of Pittsburgh Medical Center, 4401 Penn Avenue, Pittsburgh, PA 15224, USA.

ABSTRACT
Herein we report successful treatment of an adolescent Caucasian female with severe progressive localized scleroderma (mixed subtype, including generalized morphea and linear scleroderma of the trunk/limb) using infliximab and leflunomide. The patient demonstrated improvement after the first 9 months of therapy based on her clinical examination, objective measures, and patient and parent global assessments. Infliximab is a potential treatment option for pediatric localized scleroderma patients who have progression of disease or who are unable to tolerate the side effect profile of more standard systemic therapy. Larger longitudinal studies or case series are needed to confirm and further investigate infliximab's role in localized scleroderma.

No MeSH data available.


Related in: MedlinePlus

Pre- and Post-treatment photographs of the hands. (a) Pre-treatment photographs (left panel) demonstrate diffuse swelling offingers and limited range of motion. The patient was unable to make a fist. (b) Post-treatment photographs (right panel) demonstratedmarked decrease in swelling and the ability to close her fist.
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Figure 3: Pre- and Post-treatment photographs of the hands. (a) Pre-treatment photographs (left panel) demonstrate diffuse swelling offingers and limited range of motion. The patient was unable to make a fist. (b) Post-treatment photographs (right panel) demonstratedmarked decrease in swelling and the ability to close her fist.

Mentions: On our initial examination, she demonstrated a variety of findings, including both active disease and damage features. Most notably were two overall cutaneous and deep tissue patterns. One consisted of diffuse large coalescing plaque morphea lesions covering her trunk, with mild erythematous borders, yellow/white waxy indurated centers and moderate to severe skin thickness throughout. The other consisted of deep linear bands of thick skin traveling down the majority of her upper and lower extremities, concentrated on the extensor surfaces, spreading out and affecting the digits with diffuse limitation range of motion of both large and small joints. An associated general edema was also demonstrated of her peripheral extremities. Please see pre-treatment photographs in Figs. (2, 3) and pre- infliximab treatment durometer readings (skin hardness) and joint function in Table 1.


A Case Report of Successful Treatment of Recalcitrant Childhood Localized Scleroderma with Infliximab and Leflunomide.

Ferguson ID, Weiser P, Torok KS - Open Rheumatol J (2015)

Pre- and Post-treatment photographs of the hands. (a) Pre-treatment photographs (left panel) demonstrate diffuse swelling offingers and limited range of motion. The patient was unable to make a fist. (b) Post-treatment photographs (right panel) demonstratedmarked decrease in swelling and the ability to close her fist.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4493649&req=5

Figure 3: Pre- and Post-treatment photographs of the hands. (a) Pre-treatment photographs (left panel) demonstrate diffuse swelling offingers and limited range of motion. The patient was unable to make a fist. (b) Post-treatment photographs (right panel) demonstratedmarked decrease in swelling and the ability to close her fist.
Mentions: On our initial examination, she demonstrated a variety of findings, including both active disease and damage features. Most notably were two overall cutaneous and deep tissue patterns. One consisted of diffuse large coalescing plaque morphea lesions covering her trunk, with mild erythematous borders, yellow/white waxy indurated centers and moderate to severe skin thickness throughout. The other consisted of deep linear bands of thick skin traveling down the majority of her upper and lower extremities, concentrated on the extensor surfaces, spreading out and affecting the digits with diffuse limitation range of motion of both large and small joints. An associated general edema was also demonstrated of her peripheral extremities. Please see pre-treatment photographs in Figs. (2, 3) and pre- infliximab treatment durometer readings (skin hardness) and joint function in Table 1.

Bottom Line: Herein we report successful treatment of an adolescent Caucasian female with severe progressive localized scleroderma (mixed subtype, including generalized morphea and linear scleroderma of the trunk/limb) using infliximab and leflunomide.The patient demonstrated improvement after the first 9 months of therapy based on her clinical examination, objective measures, and patient and parent global assessments.Larger longitudinal studies or case series are needed to confirm and further investigate infliximab's role in localized scleroderma.

View Article: PubMed Central - PubMed

Affiliation: Department of Pediatrics, Division of Rheumatology, Children's Hospital of Pittsburgh of the University of Pittsburgh Medical Center, 4401 Penn Avenue, Pittsburgh, PA 15224, USA.

ABSTRACT
Herein we report successful treatment of an adolescent Caucasian female with severe progressive localized scleroderma (mixed subtype, including generalized morphea and linear scleroderma of the trunk/limb) using infliximab and leflunomide. The patient demonstrated improvement after the first 9 months of therapy based on her clinical examination, objective measures, and patient and parent global assessments. Infliximab is a potential treatment option for pediatric localized scleroderma patients who have progression of disease or who are unable to tolerate the side effect profile of more standard systemic therapy. Larger longitudinal studies or case series are needed to confirm and further investigate infliximab's role in localized scleroderma.

No MeSH data available.


Related in: MedlinePlus