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Corticodependent bilateral trochleitis
Trocleítis bilateral corticodependiente
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Fernández Jiménez-Ortiz, S.M. Escribá de la Fuente, R. Sampedro Yáñez, B. Maroto Rodríguez, N. Toledano Fernández, B. Villarrubia Torcal" "autores" => array:6 [ 0 => array:4 [ "nombre" => "H." "apellidos" => "Fernández Jiménez-Ortiz" "email" => array:1 [ 0 => "hectorfjo@gmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "S.M." "apellidos" => "Escribá de la Fuente" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "R." "apellidos" => "Sampedro Yáñez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 3 => array:3 [ "nombre" => "B." "apellidos" => "Maroto Rodríguez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 4 => array:3 [ "nombre" => "N." "apellidos" => "Toledano Fernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 5 => array:3 [ "nombre" => "B." "apellidos" => "Villarrubia Torcal" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Medicina Física y Rehabilitación, Hospital de Nuestra Señora del Prado, Hospital General Talavera de la Reina, Toledo, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Trocleítis bilateral corticodependiente" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 790 "Ancho" => 2500 "Tamanyo" => 157475 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Fig. " "rol" => "short" ] ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Trochleitis is inflammation of the trochlea and peritrochlear region, specifically the superior oblique (SO) muscle sheath.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It was described in 1984 by Tyschen.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Trochleodynia is considered to be a group of disorders characterized by pain in the trochlear region. One or more of the following structures are affected: cartilaginous trochlea, SO muscle, tendon and fibrovascular sheath of the SO, and the surrounding nerves that provide nociceptive information, mainly the supraorbital and supratrochlear nerves.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Three etiopathogenic theories have been proposed:<ul class="elsevierStyleList" id="lis0005"><li class="elsevierStyleListItem" id="lsti0005"><span class="elsevierStyleLabel">1</span><p id="par0010" class="elsevierStylePara elsevierViewall">Neuropathic pain due to repetitive mechanical stimulation.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a></p></li><li class="elsevierStyleListItem" id="lsti0010"><span class="elsevierStyleLabel">2</span><p id="par0015" class="elsevierStylePara elsevierViewall">Neuromuscular pain that postulates the existence of myofascial trigger points.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p></li><li class="elsevierStyleListItem" id="lsti0015"><span class="elsevierStyleLabel">3</span><p id="par0020" class="elsevierStylePara elsevierViewall">Inflammatory pain supported by anatomopathological findings of perivascular lymphocytic infiltration in surgical biopsies.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p></li></ul></p><p id="par0025" class="elsevierStylePara elsevierViewall">The association of trochleitis and migraine is widely documented.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a> Trochleodynia may exacerbate migraine or impair its treatment, or it may occur in isolation. Treatment of trochlear pain is sometimes overlooked, as most patients often have coexisting headache disorders.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical case</span><p id="par0030" class="elsevierStylePara elsevierViewall">A 29-year-old woman visited the emergency department after two months of peritrochlear pain in both eyes. The pain was intense, oppressive, non-radiating, non-paroxysmal and with poor response to naproxen and paracetamol. She did not report a decrease in visual acuity, but there was a worsening in frequency and intensity of her migraine.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Personal history: grade I obesity, migraines without aura without preventive treatment, extrinsic asthma treated with occasional salbutamol and hemolytic uraemic syndrome without current treatment (she had received eculizumab two years earlier). The pain during her visit to the emergency department was reported as 9 out of 10 on the visual scale, without relieving after administration of intravenous analgesia (intravenous diclofenac). It was decided to admit her for study and she was initially treated as migraine status.</p><p id="par0040" class="elsevierStylePara elsevierViewall">The neurological examination did not reveal any focality or suspicion of other associated symptoms. During admission she was treated with diclofenac and paracetamol orally and intravenously, barely relieving the pain to 6/10 on the visual scale. After 24 hours, treatment was started with systemic corticosteroids 1 mg/kg body weight, which was also ineffective and did not reduce the pain to any extent.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The study of the patient with nuclear magnetic resonance of the skull, spine and orbits showed a slight right trochlear inflammation (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A lumbar puncture was performed (without pathological findings) and an autoimmune and infectious study was negative.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">During admission, the first periocular infiltration of triamcinolone (dose 20 mg) was performed in the upper inner canthus of both eyes (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). The pain subsided after periocular infiltrations of triamcinolone for 3 weeks. Due to the worsening in supraversion, prisms of 3 diopters prismatic lower base were prescribed in both eyes, which relieved the pain slightly (2 points on the visual scale) and allowed half an hour of daily reading. In this patient there was an association already described with occasional migraines, so amitriptyline was prescribed at a dose of 50 mg daily, which achieved mild relief of the trochleodynia.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0055" class="elsevierStylePara elsevierViewall">The differential diagnosis so far was idiopathic orbital inflammation, sarcoidosis, localized myositis and ophthalmic migraine. The study for these pathologies was negative; normal angiotension converting enzyme, negative IgG4, absence of inflammation in orbital muscle bellies and absence of response to migraine treatment.</p><p id="par0060" class="elsevierStylePara elsevierViewall">The infiltrations continued without incident until the last bilateral one in which she presented severe vasovagal symptoms. Given the persistence of disabling pain in the right eye, it was decided, together with the patient, to perform a surgical exploration, take biopsies and inject right peritrochlear dexamethasone.</p><p id="par0065" class="elsevierStylePara elsevierViewall">The intervention consisted of dissection of the SO tendon in the nasal region of the right eyeball (only symptomatic at that time), biopsy of the tendon and perilesional tissue and infiltration of methylprednisolone (40 mg) (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The anti-pathological study was reported as "fibroadipose tissue. No specificity or malignancy".</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0070" class="elsevierStylePara elsevierViewall">The patient received a total of 5 infiltrations in the left eye and 6 in the right eye, the last one performed in the operating room. Currently she does not exhibit habitual trochleodynia, she is wearing prisms of 3 prismatic dioptres prismatic lower base of both eyes, although she does report slight limitation for near visual efforts due to trochleodynia.</p><p id="par0075" class="elsevierStylePara elsevierViewall">In the case of new decisive ones, it was proposed to continue the infiltrations until intolerable adverse effects appear. Peritrochlear injection of platelet-rich plasma, whose usefulness in tendinitis in other anatomical regions has been documented, has also been proposed as a treatment for compassionate use.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0080" class="elsevierStylePara elsevierViewall">The anatomical specificity of pain in trochleitis suggests SO tendinitis as an initial possibility. The vast majority of trochleitis are idiopathic, however we must not forget that it can be a manifestation of underlying diseases.</p><p id="par0085" class="elsevierStylePara elsevierViewall">It has been suggested that the study of the anatomical pattern of involvement suggests SO tendinitis as an initial possibility, which could correlate with regional variability in protein expression.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> Consistent with this hypothesis is the finding that only 2% of idiopathic inflammatory diseases affect the SO,<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> suggesting a different antigenic target for these diseases.</p><p id="par0090" class="elsevierStylePara elsevierViewall">Isolated SO myositis is a very rare entity. Tychsen reported 13 cases of trochleitis with SO myositis without motility restriction and successfully treated with oral steroids.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> SO myositis has been described in Wegener's granulomatosis, sarcoidosis, idiopathic inflammatory disease. Symptomatic muscle involvement in sarcoidosis has been reported in only 1.4-2.3% of cases,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> and has been divided into three entities: palpable nodules, acute and chronic myositis.</p><p id="par0095" class="elsevierStylePara elsevierViewall">The authors consider that the improvement in the condition after surgery was due to better access to the trochlea and that sedation of the patient in the operating room allowed higher doses of corticosteroid and anesthetic to be instilled, although this point is not supported by the literature as this is the first published case of which we are aware.</p><p id="par0100" class="elsevierStylePara elsevierViewall">There are documented series in the literature of up to 86 patients followed for 70 months. Five percent of cases presented relapses up to 70 months after the onset of the picture and one of the patients required 17 infiltrations.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> However, the present case is exceptional as we have hardly any other case of chronic bilateral idiopathic trochleitis.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The possible association of hemolytic uraemic syndrome with trochleitis remains to be determined, as well as the role of the indurated tissue found intraoperatively.</p><p id="par0105" class="elsevierStylePara elsevierViewall">The following protocol is recommended for routine management of trochleitis:<ul class="elsevierStyleList" id="lis0010"><li class="elsevierStyleListItem" id="lsti0020"><span class="elsevierStyleLabel">-</span><p id="par0110" class="elsevierStylePara elsevierViewall">Study with neuroimaging test, ideally nuclear magnetic resonance but assessing computed axial tomography according to availability in each center to rule out masses, vascular malformations or myositis.</p></li><li class="elsevierStyleListItem" id="lsti0025"><span class="elsevierStyleLabel">-</span><p id="par0115" class="elsevierStylePara elsevierViewall">Initial treatment with non-steroidal anti-inflammatory drugs at usual doses for 14 days (naproxen 550 mg every 12 hours).</p></li><li class="elsevierStyleListItem" id="lsti0030"><span class="elsevierStyleLabel">-</span><p id="par0120" class="elsevierStylePara elsevierViewall">If pain persists: infiltrations with local corticosteroids which can be repeated only if partially effective. Do not repeat in the absence of response.</p></li><li class="elsevierStyleListItem" id="lsti0035"><span class="elsevierStyleLabel">-</span><p id="par0125" class="elsevierStylePara elsevierViewall">In case of persistent pain or atypical picture an etiological study is recommended in accordance with the rest of the examination to rule out systemic diseases: C-reactive protein and glomerular sedimentation rate, angiotensin converting enzyme and chest X-ray (sarcoidosis), antinuclear antibodies (connective tissue diseases), anti-DNA (lupus), thyroid profile and thyroid-stimulating immunoglobulin (dysthyroid orbitopathy), rheumatoid factor (rheumatoid arthritis, lupus), anti-SSA (Ro) and anti-SSB (La) (Sjögren’s), C-ANCA antibodies (Wegener’s granulomatosis), lupus serology, acquired immunodeficiency virus, Lyme disease and Mantoux.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p></li></ul></p><p id="par0130" class="elsevierStylePara elsevierViewall">Trochleitis in pediatric age is rare, however its association with sinusitis and trauma (even banal) has been described, which can lead to inflammatory acquired Brown’s syndrome.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Depending on the nature of the trauma, neuroimaging tests should be evaluated to rule out more serious lesions.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> Its initial treatment is identical to that of adults with ibuprofen in a weight-adjusted dose for 15 days, with excellent prognosis in most cases.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusions</span><p id="par0135" class="elsevierStylePara elsevierViewall">Trochleitis or trochleodynia is a characteristic clinical picture with which any ophthalmologist should be familiar. Atypical presentations require an exhaustive etiological study as well as the application of more drastic therapeutic measures, which may require, as in the present case, a greater number of infiltrations than expected and even planning a surgical intervention for muscle biopsy.</p></span><span id="sec1050" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect1110">Conflict of interests</span><p id="par1165" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1726723" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1524942" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1726724" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1524943" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinical case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conclusions" ] 8 => array:2 [ "identificador" => "sec1050" "titulo" => "Conflict of interests" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-09-21" "fechaAceptado" => "2020-12-26" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1524942" "palabras" => array:4 [ 0 => "Trochleitis" 1 => "Trochleodynia" 2 => "Corticosteroids" 3 => "Strabismus surgery" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1524943" "palabras" => array:4 [ 0 => "Trocleítis" 1 => "Trocleodinia" 2 => "Corticosteroides" 3 => "Cirugía de estrabismo" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Trochleitis is usually a transient and non-disabling inflammation of the trochlea of superior oblique. The case is presented of a difficult to manage bilateral trochleitis in a 29-year-old woman. After an exhaustive aetiological study with neuro-imaging tests, as well as an analysis of autoimmunity and infection, no underlying cause was found. Multiple injections of corticosteroids were required in both eyes, with a partial effect. Surgical intervention was finally decided in order to visually examine the trochlea, take biopsies, and inject methylprednisolone. These were effective in relieving the symptoms. This case is exceptional due to it involving both eyes and its severity, and represented a therapeutic challenge for the clinical team.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">La trocleítis es habitualmente una inflamación transitoria, unilateral y no incapacitante de la tróclea del oblicuo superior. Presentamos el caso de una trocleítis bilateral en mujer de 29 años de evolución tórpida. Tras estudio etiológico exhaustivo con pruebas de neuroimagen, análisis de autoinmunidad e infeccioso no se encontró ninguna causa subyacente. Precisó múltiples infiltraciones en ambos ojos con efecto parcial. Finalmente se decidió intervención quirúrgica para exploración visual de la tróclea, toma de biopsias e infiltración de metilprednisolona que resultaron eficaces en el alivio sintomático. Este caso es excepcional por su bilateralidad y severidad, suponiendo un desafío terapéutico para el equipo clínico.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Fernández Jiménez-Ortiz H, Escribá de la Fuente SM, Sampedro Yáñez R, Baroto Rodríguez B, Toledo Fernández N, Villarrubia Torcal B. Trocleítis bilateral corticodependiente. Arch Soc Esp Oftalmol. 2022;97:340–343.</p>" ] ] "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1117 "Ancho" => 2167 "Tamanyo" => 110796 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 1246 "Ancho" => 1583 "Tamanyo" => 244390 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 790 "Ancho" => 2500 "Tamanyo" => 157475 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Fig. " "rol" => "short" ] ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Bilateral idiopathic trochleitis as a cause of frontal cephalgia" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "P. 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