Tonic pupil as an initial manifestation of Sjögren's syndrome

Miranda Bacallado, Yesica; González Hernández, Ayoze; Tandón Cárdenes, Luis A.; Curutchet Mesner, Luisina

doi:10.1016/j.medcle.2016.07.009

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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" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Ayoze" "apellidos" => "González Hernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "Luis A." "apellidos" => "Tandón Cárdenes" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Luisina" "apellidos" => "Curutchet Mesner" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "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" => "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.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 7mm in diameter and the left 3mm, while in dark conditions, the right measured 7mm and the left 6mm. The right pupil was unresponsive to light reflex and slightly responsive to convergence. 30min after pilocarpine 0.125% had been instilled in both eyes, the right pupil diameter became 2mm, while the left remained at 3mm. 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, antinuclear antibodies(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.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">1</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">1</a> Its onset is related to viral infections that affect the parasympathetic system.<a class="elsevierStyleCrossRefs" href="#bib0060">2–6</a> Sometimes hemifacial anhidrosis (Ross-Adie syndrome) is also added.<a class="elsevierStyleCrossRefs" href="#bib0085">7,8</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">9,10</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">10</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">1</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." 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Montagna" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Ital J Neurol Sci" "fecha" => "1997" "volumen" => "18" "paginaInicial" => "293" "paginaFinal" => "295" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/9412854" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000014600000011/v1_201609250103/S2387020616303813/v1_201609250103/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000014600000011/v1_201609250103/S2387020616303813/v1_201609250103/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020616303813?idApp=UINPBA00004N"]

ISSN: 2387-0206

Launched in 1943, Medicina Clínica is a fortnightly journal aimed at the promotion of clinical research and practice among internal medicine and other specialists. The key characteristics of Medicina Clínica are the scientific and methodological rigor of its manuscripts, the topicality of its contents, and, especially, its practical focus with highly useful information for clinical practice. Medicina Clínica is predominantly interested in publishing original research manuscripts, which are rigorously selected according to their quality, originality, and interest, and also in continued medical education-oriented manuscripts, which are commissioned by the journal to relevant authors (Editorials, Reviews, and Diagnosis and Treatment). These manuscripts contain updated topics with a major clinical or conceptual relevance in modern medicine. The journal adheres to the standards of academic research publications in all aspects including peer-review and ethical principles. Medicina Clínica is included in the General and Internal Medicine category of Thomson Reuters.

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Current Contents/Clinical Medicine, Journal Citation Reports, SCI-Expanded, Index Medicus/Medline, Excerpta Medica/EMBASE, IBECS, IME, MEDES, PASCAL, SCOPUS, SciVerse ScienceDirect

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Impact factor

The Impact Factor measures the average number of citations received in a particular year by papers published in the journal during the two preceding years.

© Clarivate Analytics, Journal Citation Reports 2022

Impact factor 2023

2.6

Citescore

CiteScore measures average citations received per document published.

Citescore 2023

3.1

SJR

SRJ is a prestige metric based on the idea that not all citations are the same. SJR uses a similar algorithm as the Google page rank; it provides a quantitative and qualitative measure of the journal's impact.

SJR 2023

0.371

SNIP

SNIP measures contextual citation impact by wighting citations based on the total number of citations in a subject field.

SNIP 2023

0.511

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Journal Information

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Vol. 146. Issue 11.

Pages 515-516 (June 2016)

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