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Primary trochlear headache. A periorbital pain with a specific diagnosis and treatment
Cefalea troclear primaria. Un dolor periorbitario con diagnóstico y tratamiento específicos
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Un dolor periorbitario con diagnóstico y tratamiento específicos" "tienePdf" => "es" "tieneTextoCompleto" => "es" "tieneResumen" => array:2 [ 0 => "es" 1 => "en" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "150" "paginaFinal" => "152" ] ] "titulosAlternativos" => array:1 [ "en" => array:1 [ "titulo" => "Primary trochlear headache. A periorbital pain with a specific diagnosis and treatment" ] ] "contieneResumen" => array:2 [ "es" => true "en" => true ] "contieneTextoCompleto" => array:1 [ "es" => true ] "contienePdf" => array:1 [ "es" => true ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figura 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1012 "Ancho" => 1000 "Tamanyo" => 81829 ] ] "descripcion" => array:1 [ "es" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Maniobra de exploración de la tróclea. A) Al palpar el ángulo superointerno de la órbita se desencadena el dolor. B) Este dolor se intensifica con la supraducción de la mirada.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "P. Sánchez Ruiz, C. Martín Villaescusa, A. Duat Rodríguez, V. Cantarín Extremera, M.L. 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A periorbital pain with a specific diagnosis and treatment" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "150" "paginaFinal" => "152" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "P. Sánchez Ruiz, C. Martín Villaescusa, A. Duat Rodríguez, V. Cantarín Extremera, M.L. Ruiz-Falcó Rojas" "autores" => array:5 [ 0 => array:4 [ "nombre" => "P." "apellidos" => "Sánchez Ruiz" "email" => array:1 [ 0 => "prado_sanchez_ruiz@hotmail.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" => "C." "apellidos" => "Martín Villaescusa" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 2 => array:3 [ "nombre" => "A." 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Hospital Infantil Niño Jesús, 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" => "Cefalea troclear primaria. Un dolor periorbitario con diagnóstico y tratamiento específicos" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1012 "Ancho" => 1000 "Tamanyo" => 89678 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">trochlea examination maneuver. A) palpation of upper inner angle of the orbit triggers pain. B) said pain intensifies in supraduction gaze position.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Pain associated to headaches occurs mainly due to the activation of peripheral nociceptive receptors. It has recently been described that receptors in the periocular structures could perform as originating source and modulators of new headaches.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The case of a teenage patient diagnosed with migraine is described. Said patient consulted due to headache associating periorbital pain.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinic case</span><p id="par0010" class="elsevierStylePara elsevierViewall">Female, 13, with previous diagnostic of migraine, referred by the Emergency Dept. due to disabling headache refractory to painkillers with 10 days evolution. The patient referred atypical pain located in the left supraorbital inner angle with hemicranial and ipsolateral irradiation, insidious onset and progressively increasing intensity. Said headache coursed with episodes alternating high intensity pain (about 5<span class="elsevierStyleHsp" style=""></span>min duration) with moderate residual pain. Since onset the patient referred moderate photophobia as well as abdominalgia with incoercible vomiting. The patient did not exhibit trigeminal-autonomous symptomatology or visual alterations. Arterial pressure and cardiac frequency as well as neurological examination and study with devices were normal. Ophthalmological examination discarded visual acuity deficit and normal ocular fundus. Reactivity and pupil reflex as well as ocular motility were also normal. However, palpation of the trochlea in the upper inner angle of the left eye produced increased pain intensity giving rise to paleness, sweating and vomiting. Due to the refractory nature of the pain, a study with basic analytics and thyroid function was requested with normal results. Cerebral resonance was carried out on the first day of admittance to discard secondary origin of the headache. The study was expanded with angioresonance sequences in order to discard venous sinus thrombosis.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Due to the unilateral characteristic of the pain and suspecting trigeminal-autonomous headache, carbamazepine and subsequently indometacin were prescribed without improvement. Before being admitted, the patient had received topiramate treatment as prophylaxis for migraine. Finally, after ocular examination findings, topical injection of anesthetic with lidocaine and corticoids was indicated (5<span class="elsevierStyleHsp" style=""></span>mg triamcinolone) in the left trochlear region, giving rise to remarkable improvement. Forty-eight hours later, the patient was discharged without digestive symptoms and hardly any orbital or hemicranial pain. In a follow-up visit 3 months later the patient remained asymptomatic without requiring additional infiltrations.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">The trochlea is a cartilage structure located in the superomedial extreme of the orbit, containing the tendon of the upper oblique muscles and innervated by sensitive ophthalmic nerve ramifications.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Its location is very close to the supra-and infra-trochlear nerves. The trochlea is important because its sensitive innervation generates nociceptive afferents on the trigeminal nerve,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> thus becoming an activation point for various types of headaches.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2,4</span></a> The pain originated by the trochlea is periorbital with frontal and ipsolateral irradiation in up to two thirds of cases.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The latest international classification of headaches<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> includes headaches associated to trochleitis described by the group of Yangüela et al.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In this type of headache, the friction produced by the tendon of the upper oblique muscle when moving under the trochlea produces trochlear inflammation or trochleitis.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Even though the etiology thereof is generally idiopathic,<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> the literature describes cases of selfimmune, rheumatological, traumatic or tumoral origin.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> In fact, a recent publication reported the case of trochleitis associated to sinusitis in a pediatric patient.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> In some cases trochleitis could be bilateral<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,7,8</span></a> and could associate diplopia in vertical gaze or supraduction limitations.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Yangüela et al. also described other patients with trochlea-originated headaches without associated inflammation.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> This clinic condition is known as primary trochlear headache and, even though it has been observed that it is more frequent than trochleitis<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> it is not yet included in the latest headache classification.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The clinic of these patients is similar to that of trochleitis patients although pain is more intense and disabling.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Referred pain could have abrupt or insidious onset (25% or 75%, respectively), with chronic (90%) or intermittent pattern (10%), in which the majority of patients describe continuous baseline pain with highly significant pain outbreaks.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> During exacerbation, pain could be daily in up to 85% of cases and could become more acute in sleep in up to 50% of cases (due to the eye movements of the REM sleep phase), which makes it necessary to visit the Emergency Department in up to 70% of cases.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In contrast with trochleitis, there is no limitation of ocular motility or associated diplopia. Diagnostic is by exclusion. In this entity it is believed that the physiopathological origin of the pain derives from the mechanical irritation occurring in the supra-and infra-trochlear nerves due to close anatomical contact with the trochlea.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Accordingly, the characteristics of the headache described in the present clinic case appeared to be compatible with said condition. Activities such as reading that induces greater use of the upper oblique muscle could produce an increase of pain, constituting a trigger factor although a clear trigger is not always apparent.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In this case, the patient had no visual deficit and denied any recent activity that could have triggered the clinic.</p><p id="par0035" class="elsevierStylePara elsevierViewall">It is important to take into account that nociceptive stimuli of the trochlea could trigger and perpetuate other types of headaches, making diagnostic more difficult. In primary trochlear headache it has been observed that up to 60% of patients could associate other types of headaches.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> The literature has described cases of paroxysmal hemicranial with trochlear disease.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> The importance of all this lies in that headaches associated to trochleodynia will not improve if the trochlea is not treated.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3,9</span></a> This explains that in the present case the patient associated digestive symptomatology and photophobia to periorbital pain, making it more typical of migraine. Accordingly, it is important to explore the trochlea in all pediatric patients with periorbital pain because some maneuvers could act as pain triggers (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). It must be taken into account that said maneuvers can also be positive in patients with other orbital diseases such as tumors, myositis or thyroid disease.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">It is recommendable to screen for a secondary diseases in patients with trochlear pain, including rheumatologic profile, thyroid autoimmunity<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> well as|as, orbital imaging test to evidence trochlear inflammation or tumor infiltration.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> Some authors have considered that if anamnesis, physical examination do, not indicate any other systemic process, it might not be, required to carry out supplementary studies10because etiology is idiopathic in the majority of cases.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,3</span></a> In the present case, image test was taken to discard intracranial disease, trochlear inflammation. Analytical studies were requested after the diagnostic had a clear direction.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Treatment for patients with primary trochlear pain with or without trochlear inflammation consists in the injection of nonsteroid anti-inflammatories, with the option of applying local corticoids in refractory cases.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> The addition of a local anesthetic enables a quick confirmation test. Said treatment usually produces a remarkable improvement of symptoms within 48<span class="elsevierStyleHsp" style=""></span>h and in up to 90% of patients<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> due to the fact that it diminishes nociceptive afferents on the trigeminal nerve. If relapses occur, new injections can be carried out.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,8</span></a> Descriptions of side effects include minor local hematomas.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2,8</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">In view of the above and even though literature is scarce and headaches with trochlear pain are not very frequent,<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> this entity must be taken into account in all periorbital headaches, including pediatric patients. Trochlear examination is increasingly important in children who may not be able to describe the characteristics of their pain in full detail. To date, not many pediatric cases have been described in the literature, probably because said entity is infradiagnosed. It is yet to be determined whether pediatric patients have different characteristics in what concerns origin, treatment and prognosis of this condition when larger series are published.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflict of interests</span><p id="par0055" class="elsevierStylePara elsevierViewall">No conflict of interests was described by the authors.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1312675" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1211554" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1312676" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1211555" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinic case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0020" "titulo" => "Conflict of interests" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2019-04-05" "fechaAceptado" => "2019-08-06" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1211554" "palabras" => array:4 [ 0 => "Migraine" 1 => "Orbital pain" 2 => "Steroid injection" 3 => "Trochlear headache." ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1211555" "palabras" => array:4 [ 0 => "Migraña" 1 => "Dolor orbitario" 2 => "Inyección de esteroides" 3 => "Cefalea troclear." ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Primary trochlear headache is a little-known cause of periorbital headache described in adults. It can involve very disabling pain. In addition, it can be associated with other types of headaches, making them even more difficult to identify. To diagnose this pathology, it is necessary that the examination of the trochlea be incorporated into the usual clinical practice of the patient with headache, which will allow the establishment of an adequate treatment.</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">The case is presented of an adolescent patient with a diagnosis of migraine, who was admitted with a disabling headache secondary to a primary trochlear headache.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">La cefalea troclear primaria es una causa de cefalea periorbitaria descrita en adultos, poco conocida. Esta entidad puede dar lugar a un dolor muy invalidante. Además, puede asociarse a otro tipo de cefaleas, dificultando aún más su identificación. Para diagnosticar esta patología es necesario que la exploración de la tróclea sea incorporada en la práctica clínica habitual del paciente con cefalea, lo cual permitirá poder establecer un tratamiento adecuado.</p><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Se presenta el caso de una paciente adolescente con diagnóstico de migraña que ingresó por un cuadro de cefalea invalidante secundaria a una cefalea troclear primaria.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Sánchez Ruiz P, Martín Villaescusa C, Duat Rodríguez A, Cantarín Extremera V, Ruiz-Falcó Rojas ML. Cefalea troclear primaria. Un dolor periorbitario con diagnóstico y tratamiento específicos. Arch Soc Esp Oftalmol. 2020;95:150–152.</p>" ] ] "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1012 "Ancho" => 1000 "Tamanyo" => 89678 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">trochlea examination maneuver. A) palpation of upper inner angle of the orbit triggers pain. B) said pain intensifies in supraduction gaze position.</p>" ] ] ] "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" => "Trochleitis and migraine headache" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "J. Yangüela" 1 => "J.A. Pareja" 2 => "N. López" 3 => "M.S. 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