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Isolated trigeminal sensory neuropathy during etanercept therapy: A case report
G Model BONSOI-4606; No. of Pages 2
ARTICLE IN PRESS Joint Bone Spine xxx (2017) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the Editor Isolated trigeminal sensory neuropathy during etanercept therapy: A case report
a r t i c l e
i n f o
Keywords: Neuropathy Trigeminal nerve Etanercept Spondyloarthritis TNF␣ Inhibitor
Several neurologic side effects were reported while using TNF␣ blockers. Their clinical features are various and the pathological mechanisms are not well explained. Herein, we describe, the first case to the best of our knowledge of trigeminal neuropathy induced by etanercept. A 33-year-old-man had an 8 years history of refractory axial spondyloarthritis to previous therapy with NSAID. Treatment with etanercept, 50 mg per week, was started then he developed coxitis. He was clinically improved within 15 weeks of treatment. Five months later, the patient developed a progressive bilateral jaw and upper lip numbness. On neurological examination, he had light touch and pinprick hypoesthesia in both maxillary nerve distributions, with decreased thermoalgic sensation in the same area. There had no sensory or motor loss in the other areas of trigeminal nerve. Corneal reflex was conserved with entirely normal function of the other cranial nerves. There was no herpes or zoster eruption. The oral soft tissues were clinically healthy and decreased dental sensitivity was noted. Laboratory tests did not show high C-reactive protein level. There was no monoclonal band on protein electrophoresis. The level of sugar, vitamin B 12 and thyroid hormones were within normal limits. HIV, hepatitis C, syphilis and Lyme disease were excluded by appropriate tests. Immunologic screening showed no evidence of disregulation, no antibodies to DNA nor ANCA or SSA or SSB or cryoglobulinemia. Labial salivary gland biopsy was also normal. A panoramic radiography, a computed tomography and a cerebral MRI were performed and showed no abnormality. Thus, the recent introduction of etanercept, the bilateral character of the neuropathy and the absence of evidence against alternative diagnosis, were considered sufficient to suggest that etanercept treatment resulted in the development of trigeminal neuropathy and so that the treatment was discontinued. This was followed by quick recovery in only 10 days. Considering the major disability of the disease while interrupting the biological
therapy, TNF␣ antagonist was reinitiated at half dose resulting in the same symptoms, as in previous episode. It is often difficult to assess the likelihood of a causal connection between a drug exposure and side effects but according to Miller et al. [1], it seems to be legible that etanercept is the cause for our patient’s symptoms. The most common clinical patterns of neurologic adverse events due to TNF␣ antagonists are demyelinating peripheral or central neuropathies [2–5]. The involvement of cranial nerves is rare and only optic neuritis was reported [6,7], trigeminal neuropathy had not been described. The group of drugs involved in trigeminal sensory neuropathy as a side effect includes isoniazid, phynetoin, mefloquin [8] and interferon [9]; it also can be caused by a tumor, infections or autoimmune diseases [10]. This case highlights the importance of analyzing the causality of TNF␣ antagonists in side effects before a definitive assertion with a necessary exhaustive investigation. Disclosure of interest The authors declare that they have no competing interest. References [1] Miller FW, Hess EV, Clauw DJ, et al. Approaches for identifying and defining environmentally associated rheumatic disorders. Arthritis Rheum 2000;43:243. [2] Stübgen J-P. Tumor necrosis factor-alpha antagonists and neuropathy. Muscle Nerve 2008;37:281–92. [3] Birnbaum J, Bingham CO. Non-length-dependent and length-dependent smallfiber neuropathies associated with tumor necrosis factor (TNF)-inhibitor therapy in patients with rheumatoid arthritis: expanding the spectrum of neurological disease associated with TNF-inhibitors. Semin Arthritis Rheum 2014;43:638–47. [4] Shin I-SJ, Baer AN, Kwon HJ, et al. Guillain-Barré and Miller Fisher syndromes occurring with tumor necrosis factor ␣ antagonist therapy. Arthritis Rheum 2006;54:1429–34. [5] Deepak P, Stobaugh DJ, Sherid M, et al. Neurological events with tumour necrosis factor alpha inhibitors reported to the Food and Drug Administration Adverse Event Reporting System. Aliment Pharmacol Ther 2013;38:388–96. [6] Winthrop KL, Chen L, Fraunfelder FW, et al. Initiation of anti-TNF therapy and the risk of optic neuritis: from the safety assessment of biologic Therapy (SABER) Study. Am J Ophthalmol 2013;155:183–9. [7] Yokoyama W, Takada K, Miyasaka N, et al. Myelitis and optic neuritis induced by a long course of etanercept in a patient with rheumatoid arthritis. BMJ Case Rep 2014 [bcr-2014-205779]. [8] Watt-Smith S, Mehta K, Scully C. Mefloquine-induced trigeminal sensory neuropathy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:163–5. [9] Marey-López J, Sousa CP. Trigeminal sensory neuropathy related to interferonalpha treatment. Muscle Nerve 2006;33:581–2. [10] Szczudlik P, Kierdaszuk B, Bakon´ L, et al. Idiopathic trigeminal sensory neuropathy. A case report. Neurol Neurochir Pol 2013;47:499–501.
http://dx.doi.org/10.1016/j.jbspin.2017.05.023 ´ e´ franc¸aise de rhumatologie. 1297-319X/© 2017 Published by Elsevier Masson SAS on behalf of Societ
Please cite this article in press as: Rekik S, et al. Isolated trigeminal sensory neuropathy during etanercept therapy: A case report. Joint Bone Spine (2017), http://dx.doi.org/10.1016/j.jbspin.2017.05.023
G Model BONSOI-4606; No. of Pages 2 2
ARTICLE IN PRESS Letter to the Editor / Joint Bone Spine xxx (2017) xxx–xxx
Sonia Rekik a,∗ Sirine Daldoul a Soumaya Boussaid a Zakaria Saiid b Mohamed Ben Amor c Mohamed Elleuch a a Service de rhumatologie, hôpital La Rabta, La Rabta Jebbari, 1007 Tunis, Tunisia b Service de neurologie, institut de neurologie La Rabta, 1007 Tunis, Tunisia
c
Service d’oto-rhino-laryngologie, hôpital La Rabta, 1007 Tunis, Tunisia ∗ Corresponding
author. E-mail address: [email protected] (S. Rekik) Accepted 31 March 2017 Available online xxx
Please cite this article in press as: Rekik S, et al. Isolated trigeminal sensory neuropathy during etanercept therapy: A case report. Joint Bone Spine (2017), http://dx.doi.org/10.1016/j.jbspin.2017.05.023
ARTICLE IN PRESS Joint Bone Spine xxx (2017) xxx–xxx
Available online at
ScienceDirect www.sciencedirect.com
Letter to the Editor Isolated trigeminal sensory neuropathy during etanercept therapy: A case report
a r t i c l e
i n f o
Keywords: Neuropathy Trigeminal nerve Etanercept Spondyloarthritis TNF␣ Inhibitor
Several neurologic side effects were reported while using TNF␣ blockers. Their clinical features are various and the pathological mechanisms are not well explained. Herein, we describe, the first case to the best of our knowledge of trigeminal neuropathy induced by etanercept. A 33-year-old-man had an 8 years history of refractory axial spondyloarthritis to previous therapy with NSAID. Treatment with etanercept, 50 mg per week, was started then he developed coxitis. He was clinically improved within 15 weeks of treatment. Five months later, the patient developed a progressive bilateral jaw and upper lip numbness. On neurological examination, he had light touch and pinprick hypoesthesia in both maxillary nerve distributions, with decreased thermoalgic sensation in the same area. There had no sensory or motor loss in the other areas of trigeminal nerve. Corneal reflex was conserved with entirely normal function of the other cranial nerves. There was no herpes or zoster eruption. The oral soft tissues were clinically healthy and decreased dental sensitivity was noted. Laboratory tests did not show high C-reactive protein level. There was no monoclonal band on protein electrophoresis. The level of sugar, vitamin B 12 and thyroid hormones were within normal limits. HIV, hepatitis C, syphilis and Lyme disease were excluded by appropriate tests. Immunologic screening showed no evidence of disregulation, no antibodies to DNA nor ANCA or SSA or SSB or cryoglobulinemia. Labial salivary gland biopsy was also normal. A panoramic radiography, a computed tomography and a cerebral MRI were performed and showed no abnormality. Thus, the recent introduction of etanercept, the bilateral character of the neuropathy and the absence of evidence against alternative diagnosis, were considered sufficient to suggest that etanercept treatment resulted in the development of trigeminal neuropathy and so that the treatment was discontinued. This was followed by quick recovery in only 10 days. Considering the major disability of the disease while interrupting the biological
therapy, TNF␣ antagonist was reinitiated at half dose resulting in the same symptoms, as in previous episode. It is often difficult to assess the likelihood of a causal connection between a drug exposure and side effects but according to Miller et al. [1], it seems to be legible that etanercept is the cause for our patient’s symptoms. The most common clinical patterns of neurologic adverse events due to TNF␣ antagonists are demyelinating peripheral or central neuropathies [2–5]. The involvement of cranial nerves is rare and only optic neuritis was reported [6,7], trigeminal neuropathy had not been described. The group of drugs involved in trigeminal sensory neuropathy as a side effect includes isoniazid, phynetoin, mefloquin [8] and interferon [9]; it also can be caused by a tumor, infections or autoimmune diseases [10]. This case highlights the importance of analyzing the causality of TNF␣ antagonists in side effects before a definitive assertion with a necessary exhaustive investigation. Disclosure of interest The authors declare that they have no competing interest. References [1] Miller FW, Hess EV, Clauw DJ, et al. Approaches for identifying and defining environmentally associated rheumatic disorders. Arthritis Rheum 2000;43:243. [2] Stübgen J-P. Tumor necrosis factor-alpha antagonists and neuropathy. Muscle Nerve 2008;37:281–92. [3] Birnbaum J, Bingham CO. Non-length-dependent and length-dependent smallfiber neuropathies associated with tumor necrosis factor (TNF)-inhibitor therapy in patients with rheumatoid arthritis: expanding the spectrum of neurological disease associated with TNF-inhibitors. Semin Arthritis Rheum 2014;43:638–47. [4] Shin I-SJ, Baer AN, Kwon HJ, et al. Guillain-Barré and Miller Fisher syndromes occurring with tumor necrosis factor ␣ antagonist therapy. Arthritis Rheum 2006;54:1429–34. [5] Deepak P, Stobaugh DJ, Sherid M, et al. Neurological events with tumour necrosis factor alpha inhibitors reported to the Food and Drug Administration Adverse Event Reporting System. Aliment Pharmacol Ther 2013;38:388–96. [6] Winthrop KL, Chen L, Fraunfelder FW, et al. Initiation of anti-TNF therapy and the risk of optic neuritis: from the safety assessment of biologic Therapy (SABER) Study. Am J Ophthalmol 2013;155:183–9. [7] Yokoyama W, Takada K, Miyasaka N, et al. Myelitis and optic neuritis induced by a long course of etanercept in a patient with rheumatoid arthritis. BMJ Case Rep 2014 [bcr-2014-205779]. [8] Watt-Smith S, Mehta K, Scully C. Mefloquine-induced trigeminal sensory neuropathy. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2001;92:163–5. [9] Marey-López J, Sousa CP. Trigeminal sensory neuropathy related to interferonalpha treatment. Muscle Nerve 2006;33:581–2. [10] Szczudlik P, Kierdaszuk B, Bakon´ L, et al. Idiopathic trigeminal sensory neuropathy. A case report. Neurol Neurochir Pol 2013;47:499–501.
http://dx.doi.org/10.1016/j.jbspin.2017.05.023 ´ e´ franc¸aise de rhumatologie. 1297-319X/© 2017 Published by Elsevier Masson SAS on behalf of Societ
Please cite this article in press as: Rekik S, et al. Isolated trigeminal sensory neuropathy during etanercept therapy: A case report. Joint Bone Spine (2017), http://dx.doi.org/10.1016/j.jbspin.2017.05.023
G Model BONSOI-4606; No. of Pages 2 2
ARTICLE IN PRESS Letter to the Editor / Joint Bone Spine xxx (2017) xxx–xxx
Sonia Rekik a,∗ Sirine Daldoul a Soumaya Boussaid a Zakaria Saiid b Mohamed Ben Amor c Mohamed Elleuch a a Service de rhumatologie, hôpital La Rabta, La Rabta Jebbari, 1007 Tunis, Tunisia b Service de neurologie, institut de neurologie La Rabta, 1007 Tunis, Tunisia
c
Service d’oto-rhino-laryngologie, hôpital La Rabta, 1007 Tunis, Tunisia ∗ Corresponding
author. E-mail address: [email protected] (S. Rekik) Accepted 31 March 2017 Available online xxx
Please cite this article in press as: Rekik S, et al. Isolated trigeminal sensory neuropathy during etanercept therapy: A case report. Joint Bone Spine (2017), http://dx.doi.org/10.1016/j.jbspin.2017.05.023