Letter to the Editor
Tonic pupil as an initial manifestation of Sjögren's syndrome
Pupila tónica como manifestación inicial de un síndrome de Sjögren
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Tandón Cárdenes, Luisina Curutchet Mesner" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Yesica" "apellidos" => "Miranda Bacallado" "email" => array:2 [ 0 => "yesi_mirand@hotmail.com" 1 => "yesimb86@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" => "Ayoze" "apellidos" => "González Hernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Luis A." "apellidos" => "Tandón Cárdenes" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Luisina" "apellidos" => "Curutchet Mesner" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Sección de Neurología, Hospital Universitario de Gran Canaria Dr. Negrín, Las Palmas de Gran Canaria, Las Palmas, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Universitario de Gran Canaria Dr. Negrín, Las Palmas de Gran Canaria, Las Palmas, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Pupila tónica como manifestación inicial de un síndrome de Sjögren" ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Tonic pupil is a common cause of anisocoria, characterized by greater mydriasis in brightness, with a poor response to light and a better response to convergence. Its origin is usually idiopathic, as part of Adie syndrome. However, it may be related to other diseases, being part of the onset of these in rare occasions. Because of it, it is necessary to pay special attention to the existence of outliers, as that could result the diagnose of certain entities that could benefit from a specific treatment.</p><p id="par0010" class="elsevierStylePara elsevierViewall">We report the case of a 51-year-old woman with a history of rheumatoid arthritis and autoimmune hepatitis, who presented at the Neuro-ophthalmology unit with a 2-month history pupillary anisocoria. In the targeted anamnesis, the patient did not report any visual or neurological symptoms and did not complain about subjective oral or ocular dryness. On physical examination, it was observed that, in light conditions, the right pupil measured 7<span class="elsevierStyleHsp" style=""></span>mm in diameter and the left 3<span class="elsevierStyleHsp" style=""></span>mm, while in dark conditions, the right measured 7<span class="elsevierStyleHsp" style=""></span>mm and the left 6<span class="elsevierStyleHsp" style=""></span>mm. The right pupil was unresponsive to light reflex and slightly responsive to convergence. 30<span class="elsevierStyleHsp" style=""></span>min after pilocarpine 0.125% had been instilled in both eyes, the right pupil diameter became 2<span class="elsevierStyleHsp" style=""></span>mm, while the left remained at 3<span class="elsevierStyleHsp" style=""></span>mm. The rest of the neurological examination, including muscle stretch reflexes, was normal. A complete blood test was requested, showing slightly increased liver enzymes, with no other relevant data. An autoimmunity study was carried out, showing positivity for rheumatoid factor, <span class="elsevierStyleItalic">antinuclear antibodies</span>(ANA) (with 1/640 titres) and anti-Ro/anti-La. The value of anti-CCP was within normal limits. Syphilis serology was negative. A brain MRI showed no significant findings. Given the autoimmune study outcome, a Schirmer test was conducted, which showed a bilateral dry eye and a salivary gland scintigraphy with no tracer uptake at submaxillary level, confirming the diagnosis of Sjögren's syndrome.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Tonic pupil occurs in the context of parasympathetic denervation of the ciliary ganglion emerging fibres, followed by an aberrant reinnervation. A prevalence of 2 cases per 1000 inhabitants has been estimated.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> In the context of Adie syndrome, the idiopathic origin is the most common. This is characterized by a tonic pupil associated with involvement of the peripheral nervous system (lower limbs hyperreflexia). It occurs most frequently in females, between 20 and 40 years of age (ratio 2–3:1).<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Its onset is related to viral infections that affect the parasympathetic system.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2–6</span></a> Sometimes hemifacial anhidrosis (Ross-Adie syndrome) is also added.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7,8</span></a> However, it is also described in various conditions, such as neurosyphilis, paraneoplastic syndromes, Guillain–Barre syndrome and systemic inflammatory diseases.<a class="elsevierStyleCrossRefs" href="#bib0095"><span class="elsevierStyleSup">9,10</span></a> Its occurrence as the initial manifestation of these is very rare. The diagnosis is made when a pupil anisocoria is evident, greater in brightness, with a mydriatic pupil responding poorly to light reflex and convergence. It can be confirmed with pilocarpine instillation (1/8%), observing an increased cholinergic sensitivity in the tonic pupil. The use of the solution is recommended at 0.0625% in some recent publications. In cases where it occurs in young women unilaterally associated with hyperreflexia, without other manifestations, it is not necessary to perform a larger study, since the most likely aetiology is Adie syndrome. However, when there are outliers, as occurring in men, in an unusual presentation age, absence of hyperreflexia, bilateral involvement, or other neurological symptoms, it is necessary to look for secondary causes broadening the aetiology, including vascular, inflammatory, infectious, neo and paraneoplastic causes. Therefore, according to the anamnesis, a brain MRI, lumbar puncture, chest radiography, autoimmunity study and serological study should be assessed. In our case, the age of onset was higher than expected in Adie syndrome, there was involvement of the peripheral system, and there was also a history of rheumatoid arthritis. For this reason, it was decided to request an autoimmune study, which gave a positive ANA result at high titres, as well as some positive anti-Ro and anti-La antibodies. This combination evidenced the possibility of Sjogren's syndrome, so the Schirmer test was performed, which confirmed the presence of bilateral ocular dryness and a salivary gland scintigraphy, which confirmed the involvement of these. Thus, the patient fulfilled 3 criteria for the possible diagnosing of Sjögren's syndrome: keratoconjunctivitis sicca, xerostomia and autoimmune disease laboratory data (ANA, anti-Ro, and anti-La). In addition, a salivary gland dysfunction became evident through the scintigraphy, which would favour the diagnosis. There have been reports of tonic pupil cases in Sjögren's syndrome, although not usually as first manifestation. In those cases, a high level of suspicion would be necessary.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> There is no effective treatment for tonic pupil. The use of bifocals or topical pilocarpine or eserine, may be recommended.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> In our case, it was associated with rheumatoid arthritis (between 4 and 31% is associated with secondary Sjögren's syndrome); however, in these cases there is often anti-CCP positivity, which was not the case in our patient, suspecting a primary Sjögren's syndrome with hepatic and articular involvement. In conclusion, the most common cause of tonic pupil is Adie Syndrome, but whenever outliers appear, consideration should be given to a secondary source. A high level of suspicion would still be necessary to advance the diagnosis of diseases that could benefit from a specific treatment. In the case of Sjögren's syndrome, the tonic pupil may be an initial manifestation of the disease, so it should be included as part of the differential diagnosis in the presence of outliers.</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: Miranda Bacallado Y, González Hernández A, Tandón Cárdenes LA, Curutchet Mesner L. Pupila tónica como manifestación inicial de un síndrome de Sjögren. Med Clin (Barc). 2016;146:515–516.</p>" ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Adie's syndrome: some new observations" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "H.S. Thompson" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:5 [ "tituloSerie" => "Trans Am Opthalmol Soc" "fecha" => "1977" "volumen" => "75" "paginaInicial" => "587" "paginaFinal" => "626" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0060" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "A varicella-induced pupil abnormality" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "A.R. 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