Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report
Introduction: Tolosa-Hunt syndrome (THS) is an inflammatory disease with painful ophthalmoplegia and unilateral periorbital headache as detailed by the International Classification of Headache Disorders, 3rd edition (ICHD-3). Azathioprine has been suggested as a second-line treatment in refractory T...
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| Format: | Article |
| Language: | English |
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Universitas Airlangga
2022-01-01
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| Series: | Aksona |
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| Online Access: | https://e-journal.unair.ac.id/aksona/article/view/35336 |
| _version_ | 1797448677044781056 |
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| author | Devi Ariani Sudibyo Mohammad Saiful Ardhi |
| author_facet | Devi Ariani Sudibyo Mohammad Saiful Ardhi |
| author_sort | Devi Ariani Sudibyo |
| collection | DOAJ |
| description | Introduction: Tolosa-Hunt syndrome (THS) is an inflammatory disease with painful ophthalmoplegia and unilateral periorbital headache as detailed by the International Classification of Headache Disorders, 3rd edition (ICHD-3). Azathioprine has been suggested as a second-line treatment in refractory THS when oral corticosteroid only gives a partial response. Case: Two cases of THS, 45-year-old and 41-year-old women with unilateral headache, drooping of the left upper eyelid, and diplopia. They presented with complete ophthalmoplegia and ophthalmic division of trigeminal nerve disturbance. Magnetic resonance imaging (MRI) showed thickening of the left cavernous sinus, suggesting THS, while the other was normal. Corticosteroid (prednisone 1-1,5 mg/day) was given orally for the first two weeks, and according to the Numeric Pain Rating Scale (NPRS), pain intensity was reduced from severe to moderate. As a second-line treatment, azathioprine (2 mg/kg/day) was given afterward, with a significant reduction in pain intensity and remission of ophthalmoplegia within seven days. Azathioprine was used as an immunosuppressive agent and was continued for another three months without any deterioration in neurological deficits. The levels of complement 3, 4 (C3, C4), and C-reactive protein (CRP) were normal in both patients, with a slight increase in erythrocyte sedimentation rate (ESR) and equivocal values on antinuclear antibody (ANA) results. Other differentials of THS were eliminated from history-taking, physical examination, and proper investigations. Conclusion: Azathioprine as a second-line treatment can be used instead of an oral corticosteroid for refractory cases of THS with fewer side effects. Complete remission of ophthalmoplegia and a significant reduction in pain intensity was obtained. |
| first_indexed | 2024-03-09T14:13:47Z |
| format | Article |
| id | doaj.art-78575ecbe8214e828943f56794cb574e |
| institution | Directory Open Access Journal |
| issn | 2807-7970 |
| language | English |
| last_indexed | 2024-03-09T14:13:47Z |
| publishDate | 2022-01-01 |
| publisher | Universitas Airlangga |
| record_format | Article |
| series | Aksona |
| spelling | doaj.art-78575ecbe8214e828943f56794cb574e2023-11-29T05:12:29ZengUniversitas AirlanggaAksona2807-79702022-01-0121303310.20473/aksona.v2i1.18633353Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case ReportDevi Ariani Sudibyo0https://orcid.org/0000-0003-3411-9256Mohammad Saiful Ardhi1https://orcid.org/0000-0002-8699-2631Department of Neurology, Faculty of Medicine, Universitas Airlangga; Dr. Soetomo General Hospital, Surabaya, IndonesiaDepartment of Neurology, Faculty of Medicine, Universitas Airlangga; Dr. Soetomo General Hospital, Surabaya, IndonesiaIntroduction: Tolosa-Hunt syndrome (THS) is an inflammatory disease with painful ophthalmoplegia and unilateral periorbital headache as detailed by the International Classification of Headache Disorders, 3rd edition (ICHD-3). Azathioprine has been suggested as a second-line treatment in refractory THS when oral corticosteroid only gives a partial response. Case: Two cases of THS, 45-year-old and 41-year-old women with unilateral headache, drooping of the left upper eyelid, and diplopia. They presented with complete ophthalmoplegia and ophthalmic division of trigeminal nerve disturbance. Magnetic resonance imaging (MRI) showed thickening of the left cavernous sinus, suggesting THS, while the other was normal. Corticosteroid (prednisone 1-1,5 mg/day) was given orally for the first two weeks, and according to the Numeric Pain Rating Scale (NPRS), pain intensity was reduced from severe to moderate. As a second-line treatment, azathioprine (2 mg/kg/day) was given afterward, with a significant reduction in pain intensity and remission of ophthalmoplegia within seven days. Azathioprine was used as an immunosuppressive agent and was continued for another three months without any deterioration in neurological deficits. The levels of complement 3, 4 (C3, C4), and C-reactive protein (CRP) were normal in both patients, with a slight increase in erythrocyte sedimentation rate (ESR) and equivocal values on antinuclear antibody (ANA) results. Other differentials of THS were eliminated from history-taking, physical examination, and proper investigations. Conclusion: Azathioprine as a second-line treatment can be used instead of an oral corticosteroid for refractory cases of THS with fewer side effects. Complete remission of ophthalmoplegia and a significant reduction in pain intensity was obtained.https://e-journal.unair.ac.id/aksona/article/view/35336azathioprinediseasepain intensityrefractorytolosa-hunt syndrome |
| spellingShingle | Devi Ariani Sudibyo Mohammad Saiful Ardhi Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report Aksona azathioprine disease pain intensity refractory tolosa-hunt syndrome |
| title | Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report |
| title_full | Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report |
| title_fullStr | Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report |
| title_full_unstemmed | Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report |
| title_short | Azathioptrine in Refractory Tolosa-Hunt Syndrome: Two Case Report |
| title_sort | azathioptrine in refractory tolosa hunt syndrome two case report |
| topic | azathioprine disease pain intensity refractory tolosa-hunt syndrome |
| url | https://e-journal.unair.ac.id/aksona/article/view/35336 |
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