Letter to the Editor
Tolosa-Hunt syndrome with spontaneous remission and recurrence
Síndrome de Tolosa-Hunt con remisión espontánea y recurrencia
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Se observa alteración de la señal en el seno cavernoso derecho<span class="elsevierStyleBold">.</span> B) RM cerebral y de órbitas. Secuencia T1 axial con gadolinio. Se observa realce asimétrico del ápex orbitario y pared lateral del seno cavernoso derecho.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "E. Carreón, S. Muñiz, D. Di Capua, J. Porta-Etessam" "autores" => array:4 [ 0 => array:2 [ "nombre" => "E." "apellidos" => "Carreón" ] 1 => array:2 [ "nombre" => "S." "apellidos" => "Muñiz" ] 2 => array:2 [ "nombre" => "D." "apellidos" => "Di Capua" ] 3 => array:2 [ "nombre" => "J." 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"apellidos" => "Marta Moreno" ] ] ] ] ] "idiomaDefecto" => "en" "Traduccion" => array:1 [ "es" => array:9 [ "pii" => "S0213485316000220" "doi" => "10.1016/j.nrl.2015.12.010" "estado" => "S300" "subdocumento" => "" "abierto" => array:3 [ "ES" => true "ES2" => true "LATM" => true ] "gratuito" => true "lecturas" => array:1 [ "total" => 0 ] "idiomaDefecto" => "es" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S0213485316000220?idApp=UINPBA00004N" ] ] "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2173580817301529?idApp=UINPBA00004N" "url" => "/21735808/0000003300000001/v1_201803061529/S2173580817301529/v1_201803061529/en/main.assets" ] "en" => array:15 [ "idiomaDefecto" => true "cabecera" => "<span class="elsevierStyleTextfn">Letter to the Editor</span>" "titulo" => "Tolosa-Hunt syndrome with spontaneous remission and recurrence" "tieneTextoCompleto" => true "saludo" => "Dear Editor," "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "68" "paginaFinal" => "70" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "E. Carreón, S. Muñiz, D. Di Capua, J. Porta-Etessam" "autores" => array:4 [ 0 => array:3 [ "nombre" => "E." "apellidos" => "Carreón" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 1 => array:3 [ "nombre" => "S." "apellidos" => "Muñiz" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "D." "apellidos" => "Di Capua" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:4 [ "nombre" => "J." "apellidos" => "Porta-Etessam" "email" => array:2 [ 0 => "jporta@yahoo.com" 1 => "jesusport@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Neurología, Hospital Universitario Virgen de la Arrixaca, Murcia, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Neurología, Unidad de Cefaleas, Hospital Clínico Universitario San Carlos, Madrid, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Síndrome de Tolosa-Hunt con remisión espontánea y recurrencia" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 948 "Ancho" => 2025 "Tamanyo" => 184518 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Brain MRI scan: gadolinium-enhanced T1-weighted coronal sequence showing signal alterations in the right cavernous sinus. (B) Brain and orbital MRI scan: gadolinium-enhanced T1-weighted axial sequence revealing asymmetrical contrast uptake in the orbital apex and the lateral wall of the right cavernous sinus.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Tolosa-Hunt syndrome (THS) is an idiopathic condition included in the differential diagnosis of painful ophthalmoplegia. The condition is caused by granulomatous infiltration of the lateral wall of the cavernous sinus and characterised by one or more episodes of unilateral orbital pain associated with paralysis of one or more oculomotor nerves. Magnetic resonance imaging (MRI) reveals granulomatous infiltration of the cavernous sinus. The condition is treated with corticosteroids, leading to the complete resolution of symptoms within 72<span class="elsevierStyleHsp" style=""></span>hours. Up to half of patients experience recurrences even several months or years after the first episode; recurrences are usually ipsilateral. However, spontaneous remission may also occur. We present a case of spontaneous remission and subsequent recurrence of THS.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Our patient was a 31-year-old Peruvian woman who had been living in Spain for the past 5 years. She had a history of idiopathic right-sided peripheral facial paralysis in 2009, which resolved completely, and a 2-year history of episodic migraine without aura. The patient was receiving oral contraceptives. She visited the emergency department in October 2011 due to a one-week history of binocular diplopia and headache. Diplopia became worse with right horizontal gaze and did not change with distance. Headache was right hemicranial and pulsatile; it was associated with photophobia and had similar features to those of her usual episodes of migraine, increasing progressively until reaching a score of 8/10 on the visual analogue scale. The patient also reported paraesthesia in the area around the mouth and the right side of the jaw. No proptosis or any other eye abnormalities were reported.</p><p id="par0015" class="elsevierStylePara elsevierViewall">In August of the same year, the patient had experienced a similar episode, which lasted a week and resolved spontaneously, leaving no sequelae. The results from the physical examination and the patient's vital signs were normal. A neuro-ophthalmological examination revealed anisocoria (right pupil: 4<span class="elsevierStyleHsp" style=""></span>mm; left pupil: 3<span class="elsevierStyleHsp" style=""></span>mm); changes in illumination did not affect pupil size. Both direct and consensual light reflexes were normal. We also observed ptosis in the right eye and dysconjugate gaze in the primary gaze position; esotropia in the right eye and hypertropia in the left (positive cover-uncover test, negative alternate cover test). Examination of ocular movements revealed limited abduction and supradextroversion of the right eye; diplopia was most marked in these gaze positions. In the following days, the right eye also displayed limited adduction, infradextroversion, and infralaevoversion. The rest of the neurological examination (including examination of the eye fundus, visual acuity, and the remaining cranial nerves) found no abnormalities. In summary, the results of the neuro-ophthalmological evaluation were compatible with paralysis of the right third and sixth cranial nerves.</p><p id="par0020" class="elsevierStylePara elsevierViewall">In view of her symptoms, the patient was diagnosed with recurrent painful ophthalmoplegia. A complete blood count and biochemical study (including determination of the erythrocyte sedimentation rate [ESR] and an angiotensin-converting enzyme [ACE] test) only revealed hypertriglyceridaemia. The results from serology tests for Borrelia and syphilis and an immunological study (antinuclear, anti-neutrophil cytoplasmic, and anti-thyroid autoantibodies) were negative. A CSF analysis (biochemical analysis, culture, and Venereal Disease Research Laboratory test) disclosed no abnormalities. Post-contrast MRI sequences displayed asymmetrical uptake in the orbital apex and the lateral wall of the right cavernous sinus (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). No asymmetries were found in the calibre or signal intensity of the optic nerve.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">The patient was diagnosed with THS based on her symptoms and neuroimaging results, and after other aetiologies were ruled out. We started treatment with intravenous methylprednisolone dosed at 1<span class="elsevierStyleHsp" style=""></span>g every 24<span class="elsevierStyleHsp" style=""></span>hours for 3 days, followed by decreasing doses of oral prednisone. Within 24<span class="elsevierStyleHsp" style=""></span>hours from treatment onset, headache and diplopia improved progressively, eventually resolving completely.</p><p id="par0030" class="elsevierStylePara elsevierViewall">THS is a rare, idiopathic, inflammatory process of the cavernous sinus. The syndrome affects both sexes equally and is more frequent in middle-aged individuals. Some authors, however, have reported cases of THS in children.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">1</span></a> THS is characterised by unilateral orbital pain that may radiate to the retro-orbital, frontal, temporal, and even occipital regions. Pain is associated with diplopia due to involvement of one or more oculomotor nerves, and may also affect the ipsilateral first or second branches of the trigeminal nerve.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">2,3</span></a> Cranial nerve involvement may occur simultaneously or up to 2 weeks after headache. Involvement of other cranial nerves, such as cranial nerve II, the third branch of cranial nerve V, cranial nerve VII, or the ipsilateral sympathetic pupillary pathway, has also been described.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">4</span></a> Bilateral cranial nerve involvement is very infrequent. In 2004, the International Headache Society reviewed the 1998 diagnostic criteria for headache disorders.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Brain and orbital MRI is the imaging technique of choice in patients with painful ophthalmoplegia. Patients with THS display an enlarged cavernous sinus which appears as isointense signal on T1/T2-weighted sequences and gadolinium uptake in that area. A lack of MRI findings, however, does not rule out a diagnosis of THS: the literature reports some cases of THS in patients with typical symptoms and normal neuroimaging results.<a class="elsevierStyleCrossRef" href="#bib0105"><span class="elsevierStyleSup">6</span></a> An MRI study of the cavernous sinus and orbital apex is highly sensitive in detecting the characteristic inflammatory lesions observed in THS and is very useful for following up these patients.<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Corticosteroids are the treatment of choice according to the diagnostic criteria for THS.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">5</span></a> There are no conclusive data on the most appropriate dose, frequency of administration, treatment duration, administration route, or type of corticosteroid. In clinical practice, these patients are usually treated either with an intravenous bolus of methylprednisolone dosed at 500-1000<span class="elsevierStyleHsp" style=""></span>mg/day for 3-5 days or with oral prednisone dosed at 1<span class="elsevierStyleHsp" style=""></span>mg/kg per day.<a class="elsevierStyleCrossRef" href="#bib0115"><span class="elsevierStyleSup">8</span></a> Treatment duration ranges from a few weeks to several months and should be adapted to each patient's clinical response. Recurrence is less likely in patients receiving high corticosteroid doses (500-1000<span class="elsevierStyleHsp" style=""></span>mg/day for several days, followed by daily doses of 1<span class="elsevierStyleHsp" style=""></span>mg/kg for several weeks).<a class="elsevierStyleCrossRef" href="#bib0120"><span class="elsevierStyleSup">9</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">THS must be diagnosed by exclusion, which requires adequate differential diagnosis.<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">10</span></a> Patients should undergo thorough testing, including a complete blood count (ESR and ACE), chest radiography, serology and immunology tests, a lumbar puncture, and a neuroimaging study. Differential diagnosis should include other entities associated with painful ophthalmoplegia and/or affecting the cavernous sinus, superior orbital fissure, or orbit,<a class="elsevierStyleCrossRefs" href="#bib0125"><span class="elsevierStyleSup">10,11</span></a> as well as those conditions manifesting with headache and ophthalmoplegia or fluctuating/episodic diplopia (<a class="elsevierStyleCrossRef" href="#tbl0005">Table 1</a>).<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">12</span></a></p><elsevierMultimedia ident="tbl0005"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">In the present case, a history of episodic migraine combined with a previous self-limiting episode of similar characteristics initially pointed to ophthalmoplegic migraine. However, MRI findings and the resolution of pain and ophthalmoplegia within 12<span class="elsevierStyleHsp" style=""></span>hours from treatment onset led us to conclude that the patient had recurrent THS.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Although spontaneous remission does occur at times, recurrence is more frequent (40%-50% of patients). Recurrences are usually ipsilateral, and to a lesser extent contralateral; bilateral recurrence is infrequent. Follow-up of these patients is recommended due to the frequency of THS recurrence. Recurrences normally occur within the first months following the initial episode, although the literature reports cases of recurrences over a decade after the first episode.<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">14</span></a> Clinical and radiological follow-up of these patients is essential. Such conditions as sarcoidosis, vasculitis, and lymphoma may initially respond to treatment with corticosteroids; in these cases, subsequent progression of the disease will lead us to the correct diagnosis.<a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">15</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Spontaneous resolution of THS, as in our case, is infrequent and has rarely been reported in the recent literature. The cases of spontaneous THS resolution published in the literature were reported before corticosteroids were established as the treatment of choice for THS. New diagnostic criteria, combined with recent advances in neuroimaging, will very likely enable earlier diagnosis of THS and administration of corticosteroid treatment, resulting in fewer cases of spontaneous resolution. However, spontaneous resolution of THS is not unlikely. The condition should therefore be suspected in patients experiencing an episode of self-limiting painful ophthalmoplegia and subsequent recurrence of symptoms.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Carreón E, Muñiz S, Di Capua D, Porta-Etessam J. Síndrome de Tolosa-Hunt con remisión espontánea y recurrencia. Neurología. 2018;33:68–70.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 948 "Ancho" => 2025 "Tamanyo" => 184518 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A) Brain MRI scan: gadolinium-enhanced T1-weighted coronal sequence showing signal alterations in the right cavernous sinus. (B) Brain and orbital MRI scan: gadolinium-enhanced T1-weighted axial sequence revealing asymmetrical contrast uptake in the orbital apex and the lateral wall of the right cavernous sinus.</p>" ] ] 1 => array:9 [ "identificador" => "tbl0005" "etiqueta" => "Table 1" "tipo" => "MULTIMEDIATABLA" "mostrarFloat" => true "mostrarDisplay" => false "fuente" => "<span class="elsevierStyleItalic">Source</span>: Modified from Porta-Etessam et al.<a class="elsevierStyleCrossRef" href="#bib0140"><span class="elsevierStyleSup">13</span></a>" "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at1" "detalle" => "Table " "rol" => "short" ] ] "tabla" => array:1 [ "tablatextoimagen" => array:1 [ 0 => array:2 [ "tabla" => array:1 [ 0 => """ <table border="0" frame="\n \t\t\t\t\tvoid\n \t\t\t\t" class=""><tbody title="tbody"><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Myasthenia gravis \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Neuromyotonia \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Superior oblique myokymia \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Thyroid-associated orbitopathy \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Decompensated heterophoria \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Convergence spasm \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Cyclic spasms \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Intermittent ophthalmoplegia \t\t\t\t\t\t\n \t\t\t\t</td></tr><tr title="table-row"><td class="td-with-role" title="table-entry ; entry_with_role_rowhead " align="left" valign="top">Tolosa-Hunt syndrome \t\t\t\t\t\t\n \t\t\t\t</td></tr></tbody></table> """ ] "imagenFichero" => array:1 [ 0 => "xTab1688282.png" ] ] ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Disorders manifesting with episodic or fluctuating diplopia</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib0080" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The Tolosa-Hunt syndrome in children: a case report" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "C. 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| Year/Month | Html | Total | |
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| 2024 November | 14 | 3 | 17 |
| 2024 October | 61 | 2 | 63 |
| 2024 September | 58 | 6 | 64 |
| 2024 August | 54 | 7 | 61 |
| 2024 July | 63 | 9 | 72 |
| 2024 June | 80 | 5 | 85 |
| 2024 May | 71 | 7 | 78 |
| 2024 April | 57 | 8 | 65 |
| 2024 March | 72 | 4 | 76 |
| 2024 February | 80 | 4 | 84 |
| 2024 January | 96 | 8 | 104 |
| 2023 December | 80 | 5 | 85 |
| 2023 November | 186 | 11 | 197 |
| 2023 October | 126 | 13 | 139 |
| 2023 September | 81 | 7 | 88 |
| 2023 August | 142 | 13 | 155 |
| 2023 July | 129 | 8 | 137 |
| 2023 June | 147 | 16 | 163 |
| 2023 May | 168 | 9 | 177 |
| 2023 April | 165 | 7 | 172 |
| 2023 March | 145 | 11 | 156 |
| 2023 February | 105 | 8 | 113 |
| 2023 January | 114 | 20 | 134 |
| 2022 December | 117 | 16 | 133 |
| 2022 November | 143 | 27 | 170 |
| 2022 October | 147 | 17 | 164 |
| 2022 September | 136 | 33 | 169 |
| 2022 August | 137 | 16 | 153 |
| 2022 July | 109 | 15 | 124 |
| 2022 June | 103 | 10 | 113 |
| 2022 May | 123 | 26 | 149 |
| 2022 April | 122 | 11 | 133 |
| 2022 March | 137 | 10 | 147 |
| 2022 February | 182 | 12 | 194 |
| 2022 January | 185 | 11 | 196 |
| 2021 December | 136 | 17 | 153 |
| 2021 November | 168 | 19 | 187 |
| 2021 October | 110 | 20 | 130 |
| 2021 September | 85 | 17 | 102 |
| 2021 August | 143 | 13 | 156 |
| 2021 July | 144 | 15 | 159 |
| 2021 June | 139 | 15 | 154 |
| 2021 May | 172 | 15 | 187 |
| 2021 April | 383 | 28 | 411 |
| 2021 March | 225 | 13 | 238 |
| 2021 February | 189 | 20 | 209 |
| 2021 January | 191 | 29 | 220 |
| 2020 December | 173 | 22 | 195 |
| 2020 November | 171 | 12 | 183 |
| 2020 October | 112 | 12 | 124 |
| 2020 September | 171 | 23 | 194 |
| 2020 August | 199 | 17 | 216 |
| 2020 July | 143 | 16 | 159 |
| 2020 June | 177 | 24 | 201 |
| 2020 May | 154 | 23 | 177 |
| 2020 April | 137 | 16 | 153 |
| 2020 March | 148 | 16 | 164 |
| 2020 February | 147 | 18 | 165 |
| 2020 January | 125 | 8 | 133 |
| 2019 December | 125 | 26 | 151 |
| 2019 November | 117 | 15 | 132 |
| 2019 October | 102 | 8 | 110 |
| 2019 September | 131 | 4 | 135 |
| 2019 August | 60 | 2 | 62 |
| 2019 July | 48 | 11 | 59 |
| 2019 June | 90 | 5 | 95 |
| 2019 May | 218 | 31 | 249 |
| 2019 April | 131 | 8 | 139 |
| 2019 March | 48 | 12 | 60 |
| 2019 February | 38 | 4 | 42 |
| 2019 January | 35 | 3 | 38 |
| 2018 December | 28 | 6 | 34 |
| 2018 November | 31 | 6 | 37 |
| 2018 October | 67 | 16 | 83 |
| 2018 September | 56 | 4 | 60 |
| 2018 August | 13 | 1 | 14 |
| 2018 July | 17 | 2 | 19 |
| 2018 June | 18 | 0 | 18 |
| 2018 May | 13 | 3 | 16 |
| 2018 April | 15 | 3 | 18 |
| 2018 March | 17 | 5 | 22 |
| 2018 February | 5 | 6 | 11 |
| 2018 January | 1 | 4 | 5 |
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