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Abstract

Efficacy of bosentan in treatment of unresponsive cutaneous ulceration in disabling pansclerotic morphea in children.

Rosa Roldan, Guadalupe Morote, Maria del Carmen Castro, Maria Dolores Miranda, Jose Carlos Moreno and Eduardo Collantes
The Journal of Rheumatology December 2006, 33 (12) 2538-2540;
Rosa Roldan
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Guadalupe Morote
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Maria del Carmen Castro
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Maria Dolores Miranda
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Jose Carlos Moreno
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Eduardo Collantes
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Abstract

Disabling pansclerotic morphea (PM) of childhood is a rare and debilitating variant of localized scleroderma. We describe a 4-year-old girl with rapid progression of deep cutaneous fibrosis extending into the muscle fascia with disabling joint contractures of the hips, knees, ankles, and fingers and recalcitrant ischemic ulcerations. Within the first months of therapy with dual oral endothelin receptor antagonist bosentan (31.25 mg qd [corrected] for 4 weeks, then 31.25 mg bid) limb ulcers improved, with resolution of the widespread sclerotic skin lesions. Joint mobility improved, and a substantial decrease of skin thickness was noted. No side effects were noted. In the context of other data in scleroderma, bosentan may be a promising option in the treatment of PM.

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The Journal of Rheumatology
Vol. 33, Issue 12
1 Dec 2006
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Efficacy of bosentan in treatment of unresponsive cutaneous ulceration in disabling pansclerotic morphea in children.
Rosa Roldan, Guadalupe Morote, Maria del Carmen Castro, Maria Dolores Miranda, Jose Carlos Moreno, Eduardo Collantes
The Journal of Rheumatology Dec 2006, 33 (12) 2538-2540;

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Efficacy of bosentan in treatment of unresponsive cutaneous ulceration in disabling pansclerotic morphea in children.
Rosa Roldan, Guadalupe Morote, Maria del Carmen Castro, Maria Dolores Miranda, Jose Carlos Moreno, Eduardo Collantes
The Journal of Rheumatology Dec 2006, 33 (12) 2538-2540;
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