Painful trigeminal neuropathy caused by severe mandibular resorption and nerve compression in a patient with systemic sclerosis: Case report and literature review

Author(s):  
Debra Krimsky Fischoff ◽  
David Sirois
2019 ◽  
Vol 5 (2) ◽  
pp. 159-164 ◽  
Author(s):  
Jan-Gerd Rademacher ◽  
Chris Wincup ◽  
Björn Tampe ◽  
Peter Korsten

Background: Systemic sclerosis is a rare autoimmune disease characterized by skin and organ fibrosis, and vasculopathy. Raynaud’s phenomenon is almost universally present in systemic sclerosis and can be the most debilitating symptom. Raynaud’s phenomenon may lead to the development of digital ulcers, potentially complicated by infection, tissue necrosis, and auto-amputation. Recommended treatments have variable efficacy. Methods: We report the case of a 30-year-old woman with diffuse systemic sclerosis suffering from severe Raynaud’s phenomenon and digital ulcers with digital tissue necrosis who was treated with combination therapy of an endothelin receptor antagonist and phosphodiesterase 5 inhibitor. In addition, we reviewed the literature on the topic. Results: Previous therapy with calcium-channel blockers, intravenous iloprost, and bosentan had all failed to control symptoms. We added sildenafil in combination with bosentan and observed a rapid and sustained treatment effect. Raynaud’s phenomenon severity, number of attacks, and attack duration decreased within 2 weeks of initiating treatment. Furthermore, this resulted in the healing of established digital ulcers. Conclusion: Our case report suggests that combination therapy may be a feasible treatment for the most severely affected and refractory patients. In our literature review, we found one retrospective study and three additional cases with similarly encouraging results.


2006 ◽  
Vol 16 (3) ◽  
pp. 183-187 ◽  
Author(s):  
Kenji Takagi ◽  
Shinichiro Nishio ◽  
Kimiko Akimoto ◽  
Takumi Yoshino ◽  
Shinichi Kawai

2019 ◽  
Vol In Press (In Press) ◽  
Author(s):  
Serkan Erkus ◽  
Ozkan Kose ◽  
Adil Turan ◽  
Onder Kalenderer

Author(s):  
Emmanuel Murwanashyaka ◽  
Alex Buteera ◽  
Emmanuel Bukara ◽  
Lynnette Kyokunda-Tumwine ◽  
Emmanuel Rudakemwa

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