Keywords
spondyloarthritis
demyelinating lesions
anti-TNF-α therapy
Categories
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Abstract
Background: Hidradenitis suppurativa (HS) is an inflammatory skin disease primarily affecting large folds, which can be associated with spondyloarthritis (SpA). However, the rare coexistence of HS, SpA, and demyelinating lesions presents significant challenges in therapeutic decision-making, especially when resistant to first-line therapies. Case Presentation: We report the case of a 17‐year‐old boy with a three-year history of stage III hidradenitis suppurativa (HS) who presented with chronic inflammatory back pain for three months and a two-month history of a waddling gait. Initial treatments, including topical and oral antibiotics, failed to control his skin lesions. Physical examination revealed lumbar stiffness, positive sacroiliac tests, and brisk lower limb reflexes. Pelvic X-ray revealed bilateral stage III sacroiliitis, and brain MRI showed T2 and FLAIR hyperintensities suggestive of demyelinating lesions. Extensive workup, including cerebrospinal fluid analysis and normal levels of ANA, anti‐DNA, anti‐NMO, anti‐MOG antibodies, syphilitic and HIV serologies, vitamin B12, and folate, concluded to non-specific demyelinating lesions. A diagnosis of radiographic axial SpA and peripheral enthesitis associated with HS was established. Worsening skin condition prompted initiation of anti-TNF-α therapy (adalimumab) after multidisciplinary consultation. His gait disturbances improved with physiotherapy, with neurological evaluation favoring a functional origin. Conclusion: Managing concurrent HS and SpA with demyelinating lesions is challenging. Although anti-TNF-α therapy is effective for HS and SpA, its link to demyelinating diseases demands caution. Further research is needed to refine treatment strategies and improve patient outcomes.
References
1. Goldburg SR, Strober BE, Payette MJ. Hidradenitis suppurativa: epidemiology, clinical presentation, and pathogenesis. J Am Acad Dermatol. 2020;82:1045-58. doi: 10.1016/j.jaad.2019.08.090
2. Jemec GB, Heidenheim M, Nielsen NH. The prevalence of hidradenitis suppurativa and its potential precursor lesions. J Am Acad Dermatol. 1996;35:191-4. doi: 10.1016/s0190-9622(96)90321-7
3. Revuz JE, Canoui-Poitrine F, Wolkenstein P, Viallette C, Gabison G, Pouget F, et al. Prevalence and factors associated with hidradenitis suppurativa: results from two case-control studies. J Am Acad Dermatol. 2008;59:596-601. doi: 10.1016/j.jaad.2008.06.020
4. Jemec GBE. Clinical practice. Hidradenitis suppurativa. N Engl J Med. 2012;366:158-64. doi: 10.1056/NEJMcp1014163
5. Gau SY, Preclaro IAC, Wei JCC, Lee CY, Kuan YH, Hsiao YP, et al. Risk of psoriasis in people with hidradenitis suppurativa: a systematic review and meta-analysis. Front Immunol. 2022;13:1033844. doi: 10.3389/fimmu.2022.1033844
6. Lapins J, Ye W, Nyrén O, Emtestam L. Incidence of cancer among patients with hidradenitis suppurativa. Arch Dermatol. 2001;137:730-4.
7. Menter A. Recognizing and managing comorbidities and complications in hidradenitis suppurativa. Semin Cutan Med Surg. 2014;33(3 Suppl):S54-6. doi: 10.12788/j.sder.0093
8. Richette P, Molto A, Viguier M, Dawidowicz K, Hayem G, Nassif A, et al. Hidradenitis suppurativa associated with spondyloarthritis — results from a multicenter national prospective study. J Rheumatol. 2014;41:490-4. doi: 10.3899/jrheum.130977
9. Fauconier M, Reguiai Z, Barbe C, Colosio A, Eschard JP, Salmon JH, et al. Association between hidradenitis suppurativa and spondyloarthritis. Joint Bone Spine. 2018;85:593-7. doi: 10.1016/j.jbspin.2017.09.005
10. Rondags A, Arends S, Wink FR, Horváth B, Spoorenberg A. High prevalence of hidradenitis suppurativa symptoms in axial spondyloarthritis patients: a possible new extra-articular manifestation. Semin Arthritis Rheum. 2019;48:611-7. doi: 10.1016/j.semarthrit.2018.03.010
11. Scheinfeld N. Diseases associated with hidranitis suppurativa: part 2 of a series on hidradenitis. Dermatol Online J. 2013;19:18558.
12. Saunte DML, Jemec GBE. Hidradenitis suppurativa: advances in diagnosis and treatment. JAMA. 2017;318:2019-32. doi: 10.1001/jama.2017.16691
13. Zouboulis CC, Desai N, Emtestam L, Hunger RE, Ioannides D, Juhász I, et al. European S1 guideline for the treatment of hidradenitis suppurativa/acne inversa. J Eur Acad Dermatol Venereol. 2015;29:619-44. doi: 10.1111/jdv.12966
14. Hendricks AJ, Hsiao JL, Lowes MA, Shi VY. A comparison of international management guidelines for hidradenitis suppurativa. Dermatology. 2021;237:81-96. doi: 10.1159/000503605
15. Schneider-Burrus S, Witte-Haendel E, Christou D, Rigoni B, Sabat R, Diederichs G. High prevalence of back pain and axial spondyloarthropathy in patients with hidradenitis suppurativa. Dermatology. 2016;232:606-12. doi: 10.1159/000448838
16. Lowe MM, Naik HB, Clancy S, Pauli M, Smith KM, Bi Y, et al. Immunopathogenesis of hidradenitis suppurativa and response to anti-TNF-α therapy. JCI Insight. 2020;5:e139932. doi: 10.1172/jci.insight.139932
17. Sieper J, Poddubnyy D. Axial spondyloarthritis. Lancet. 2017;390:73-84. doi: 10.1016/S0140-6736(16)31591-4
18. Kimball AB, Kerdel F, Adams D, Mrowietz U, Gelfand JM, Gniadecki R, et al. Adalimumab for the treatment of moderate to severe hidradenitis suppurativa: a parallel randomized trial. Ann Intern Med. 2012;157:846-55. doi: 10.7326/0003-4819-157-12-201212180-00004
19. Kimball AB, Okun MM, Williams DA, Gottlieb AB, Papp KA, Zouboulis CC, et al. Two phase 3 trials of adalimumab for hidradenitis suppurativa. N Engl J Med. 2016;375:422-34. doi: 10.1056/NEJMoa1504370
20. Miller I, Lynggaard CD, Lophaven S, Zachariae C, Dufour DN, Jemec GBE. A double-blind placebo-controlled randomized trial of adalimumab in the treatment of hidradenitis suppurativa. Br J Dermatol. 2011;165:391-8. doi: 10.1111/j.1365-2133.2011.10339.x
21. Humira (adalimumab) injection, for subcutaneous use [prescribing information]. North Chicago (IL): AbbVie Inc.; 2025.
22. Ramiro S, Nikiphorou E, Sepriano A, Ortolan A, Webers C, Baraliakos X, et al. ASAS-EULAR recommendations for the management of axial spondyloarthritis: 2022 update. Ann Rheum Dis. 2023;82:19-34. doi: 10.1136/ard-2022-223296
23. Launer LJ. Epidemiology of white matter lesions. Top Magn Reson Imaging. 2004;15:365-7. doi: 10.1097/01.rmr.0000168216.98338.8d
24. Haller S, Kövari E, Herrmann FR, Cuvinciuc V, Tomm AM, Zulian GB, et al. Do brain T2/FLAIR white matter hyperintensities correspond to myelin loss in normal aging? A radiologic-neuropathologic correlation study. Acta Neuropathol Commun. 2013;1:14. doi: 10.1186/2051-5960-1-14
25. Wang ML, Zhang XX, Yu MM, Li WB, Li YH. Prevalence of white matter hyperintensity in young clinical patients. AJR Am J Roentgenol. 2019;213:667-71. doi: 10.2214/AJR.18.20888
26. Zou Q, Wang M, Zhang D, Wei X, Li W. White matter hyperintensities in young patients from a neurological outpatient clinic: prevalence, risk factors, and correlation with enlarged perivascular spaces. J Pers Med. 2023;13:525. doi: 10.3390/jpm13030525
27. Eikermann-Haerter K, Huang SY. White matter lesions in migraine. Am J Pathol. 2021;191:1955-62. doi: 10.1016/j.ajpath.2021.02.007
28. Wadhwa R, Wen W, Frankland A, Leung V, Sinbandhit C, Stuart A, et al. White matter hyperintensities in young individuals with bipolar disorder or at high genetic risk. J Affect Disord. 2019;245:228-36. doi: 10.1016/j.jad.2018.10.368
29. Garnier-Crussard A, Bougacha S, Wirth M, André C, Delarue M, Landeau B, et al. White matter hyperintensities across the adult lifespan: relation to age, Aβ load, and cognition. Alzheimers Res Ther. 2020;12:127. doi: 10.1186/s13195-020-00669-4
30. Remalante-Rayco P, Espiritu AI, Daghistani Y, Chim T, Atenafu E, Keshavarzi S, et al. Incidence and predictors of demyelinating disease in spondyloarthritis: data from a longitudinal cohort study. Rheumatology (Oxford). 2024;63:1980-6. doi: 10.1093/rheumatology/kead527
31. Taylor TRP, Galloway J, Davies R, Hyrich K, Dobson R. Demyelinating events following initiation of anti-TNFα therapy in the British Society for Rheumatology Biologics Registry in Rheumatoid Arthritis. Neurol Neuroimmunol Neuroinflamm. 2021;8:e992. doi: 10.1212/NXI.0000000000000992
32. Mercieca C, Vella N, Borg AA. Demyelination during anti-TNFα therapy for ankylosing spondylitis. Mod Rheumatol. 2012;22:303-7. doi: 10.1007/s10165-011-0498-4
33. Mohan N, Edwards ET, Cupps TR, Oliverio PJ, Sandberg G, Crayton H, et al. Demyelination occurring during anti-tumor necrosis factor alpha therapy for inflammatory arthritides. Arthritis Rheum. 2001;44:2862-9. doi: 10.1002/1529-0131(200112)44:12<2862::AID-ART474>3.0.CO;2-W
34. Seror R, Richez C, Sordet C, Rist S, Gossec L, Direz G, et al. Pattern of demyelination occurring during anti-TNF-α therapy: a French national survey. Rheumatology (Oxford). 2013;52:868-74. doi: 10.1093/rheumatology/kes375
35. Kemanetzoglou E, Andreadou E. CNS demyelination with TNF-α blockers. Curr Neurol Neurosci Rep. 2017;17:36. doi: 10.1007/s11910-017-0742-1
36. Kunchok A, Aksamit AJ, Davis JM, Kantarci OH, Keegan BM, Pittock SJ, et al. Association between tumor necrosis factor inhibitor exposure and inflammatory central nervous system events. JAMA Neurol. 2020;77:937-46. doi: 10.1001/jamaneurol.2020.1162
37. The Lenercept Multiple Sclerosis Study Group and The University of British Columbia MS/MRI Analysis Group. TNF neutralization in MS: results of a randomized, placebo-controlled multicenter study. Neurology. 1999;53:457-65.
38. van Oosten BW, Barkhof F, Truyen L, Boringa JB, Bertelsmann FW, von Blomberg BME, et al. Increased MRI activity and immune activation in two multiple sclerosis patients treated with the monoclonal anti-tumor necrosis factor antibody cA2. Neurology. 1996;47:1531-4. doi: 10.1212/wnl.47.6.1531
39. Lin S, Green HD, Hendy P, Heerasing NM, Chanchlani N, Hamilton B, et al. Clinical features and genetic risk of demyelination following anti-TNF treatment. J Crohns Colitis. 2020;14:1653-61. doi: 10.1093/ecco-jcc/jjaa104
40. Fernández-Espartero MC, Pérez-Zafrilla B, Naranjo A, Esteban C, Ortiz AM, Gómez-Reino JJ, et al. Demyelinating disease in patients treated with TNF antagonists in rheumatology: data from BIOBADASER, a pharmacovigilance database, and a systematic review. Semin Arthritis Rheum. 2011;40:330-7. doi: 10.1016/j.semarthrit.2010.06.004
41. Moyano B, Clemente A, Marín-Jiménez I, Martorell A. Clinical experience of the use of adalimumab in the management of hidradenitis suppurativa. Comparison of response rates with Crohn disease. Actas Dermosifiliogr. 2016;107 Suppl 2:43-50. doi: 10.1016/S0001-7310(17)30008-X
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Copyright (c) 2026 Makhlouf Yasmine, Wassim Tekaya, Boussaa Hiba, Miladi Saoussen, Souebni Leila, Ben Abdelghani Kaouther, Fazaa Alia, Laatar Ahmed (Author)