Main Article Content
small fiber neuropathy, sarcoidosis, infliximab, treatment, SFNSL
Background and aim: Small fiber neuropathy (SFN) may present as complication in sarcoidosis.(1) SFN can potentially result into a large range of symptoms with a high impact on quality of life.(2) Although treatment of the underlying disease of SFN is paramount, little research has been performed to investigate SFN improvement as consequence of sarcoidosis treatment. This retrospective study investigates whether there is an association between the anti-inflammatory effects of infliximab and SFN-symptoms
Methods: The Small Fiber Neuropathy Screening List (SFNSL) was used to measure changes in SFN symptoms during infliximab treatment. Maximal standardized uptake value (SUVmax) from Fluordeoxyglucose Positron Emission Tomography (FDG-PET) was used as a measure for inflammatory activity.
Results: 36 sarcoidosis patients were eligible for analysis. SFNSL-score showed a mean decrease of -1,9 points (p = 0.446). SUVmax did improve with a mean of -3.7 (p<0.001). No correlation between a decrease of SUVmax and SFNSL screening list could be found (p=0.610).
Conclusions: Our data reveal no association between anti-inflammatory effect of infliximab and SFN-related symptoms in patients with sarcoidosis, which contradicts previous case-reports and case-series.(3–6) Given the major negative impact of SFN-related symptoms on the quality of life in patients with sarcoidosis, it is necessary that the possible beneficial effect of anti-inflammatory therapy will be further addressed in future prospective studies.1
2. Bakkers M, Faber CG, Hoeijmakers JGJ, Lauria G, Merkies ISJ. Small Fibers, Large Impact: Quality of Life in Small-Fiber Neuropathy. Vol 49., 2014.
3. Grunewald J, Grutters JC, Arkema E V., Saketkoo LA, Moller DR, Müller-Quernheim J. Sarcoidosis. Nature Reviews Disease Primers 2019; 5.
4. Valeyre D, Prasse A, Nunes H, Uzunhan Y, Brillet P-Y, Müller-Quernheim J. Sarcoidosis. Vol 103., 2014.
5. Birnie DH, Sauer WH, Bogun F, Cooper JM, Culver DA, Duvernoy CS, et al. HRS expert consensus statement on the diagnosis and management of arrhythmias associated with cardiac sarcoidosis. Heart Rhythm 2014; 11:1304–1323. P
6. Baughman RP, Drent M, Kavuru M, Judson MA, Costabel U, Du Bois R, et al. Infliximab therapy in patients with chronic sarcoidosis and pulmonary involvement. American Journal of Respiratory and Critical Care Medicine 2006; 174:795–802.
7. Voortman M, Hendriks CMR, Elfferich MDP, Bonella F, Møller J, De Vries J, et al. The Burden of Sarcoidosis Symptoms from a Patient Perspective. Lung 2019; 197:155–161.
8. Voortman M, Fritz D, Vogels OJM, Eftimov F, Van De Beek D, Brouwer MC, et al. Small fiber neuropathy: A disabling and underrecognized syndrome. Current Opinion in Pulmonary Medicine 2017; 23:447–457.
9. Cox CE, Donohue JF, Brown CD, Kataria YP, Judson MA. Health-related quality of life of persons with sarcoidosis. Chest 2004; 125:997–1004. Published online: 2004.
10. De Vries J, Wirnsberger RM. Fatigue, quality of life and health status in sarcoidosis. Sarcoidosis 2010:92–104.
11. Parambil JG, Tavee JO, Zhou L, Pearson KS, Culver DA. Efficacy of intravenous immunoglobulin for small fiber neuropathy associated with sarcoidosis. Respiratory Medicine 2011; 105:101–105.
12. Hoitsma E, Faber C, Santen-Hoeufft M van, Vries J de, Reulen J, Drenth M. Improvement of small fiber neuropathy in a sarcoidosis patient after treatment with infliximab. Sarcoidosis Vasculitis and Diffuse Lung Diseases 2006; 23:73–77.
13. Van Velzen M, Heij L, Niesters M, Cerami A, Dunne A, Dahan A, et al. Ara 290 for treatment of small fiber neuropathy in sarcoidosis. Expert Opinion on Investigational Drugs 2014; 23:541–550.
14. Costabel U, Hunninghake GW, Statement S. EDITORIAL ATS / ERS / WASOG statement on sarcoidosis. Eur Respir J 1999; 14:735–737.
15. Hoitsma E, De Vries J, Drent M. The small fiber neuropathy screening list: Construction and cross-validation in sarcoidosis. Respiratory Medicine 2011; 105:95–100.
16. Voortman M, Beekman E, Drent M, Hoitsma E, De Vries J. Determination of the smallest detectable change (SDC) and the minimal important difference (MID) for the small fiber neuropathy screening list (SFNSL) in sarcoidosis. Sarcoidosis Vasculitis and Diffuse Lung Diseases 2018; 35:333–341.
17. Pinker K, Riedl C, Weber WA. Evaluating tumor response with FDG PET: updates on PERCIST, comparison with EORTC criteria and clues to future developments. European Journal of Nuclear Medicine and Molecular Imaging 2017; 44:55–66. Published online: 2017.
18. Vorselaars ADM, Crommelin HA, Deneer VHM, Meek B, Claessen AME, Keijsers RGM, et al. Effectiveness of infliximab in refractory FDG PET-positive sarcoidosis. European Respiratory Journal 2015; 46:175–185.
19. Keijsers RG, Verzijlbergen FJ, Oyen WJ, Van Den Bosch JM, Ruven HJ, Van Velzen-Blad H, et al. 18F-FDG PET, genotype-corrected ACE and sIL-2R in newly diagnosed sarcoidosis. European Journal of Nuclear Medicine and Molecular Imaging 2009; 36:1131–1137.
20. Wijnen PA, Cremers JP, Nelemans PJ, Erckens RJ, Hoitsma E, Jansen TL, et al. Association of the TNF-α G-308A polymorphism with TNF-inhibitor response in sarcoidosis. European Respiratory Journal 2014; 43:1730–1739.
21. Tavee JO, Karwa K, Ahmed Z, Thompson N, Parambil J, Culver DA. Sarcoidosis-Associated Small Fiber Neuropathy in a Large Cohort: Clinical Aspects and Response to IVIG and Anti-TNF Alpha Treatment. Vol 126. Elsevier Ltd, 2017.
22. Vis R, van de Garde EMW, Meek B, Korenromp IHE, Grutters JC. Randomised, placebo-controlled trial of dexamethasone for quality of life in pulmonary sarcoidosis. Respiratory Medicine 2020; 165.