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Sclerodermatomyositis, ocular manifestations
Esclerodermatomiositis con manifestaciones oculares predominantes
M. Pedroza-Seresa,
Corresponding author
mpedrozaseres@gmail.com

Corresponding author.
, J.C. Serna-Ojedaa, L.F. Flores-Suárezb
a Instituto de Oftalmología «Conde de Valenciana», Mexico City, Mexico
b Clínica de Vasculitis Sistémicas Primarias, Instituto Nacional de Enfermedades Respiratorias, Mexico City, Mexico
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myositis&#44; Gottron sign &#40;erythematous or purple plates appearing in the dorsal area of the inter-phalanx&#44; metacarpian-phalanx&#44; elbows and knee joints&#41; and calcinosis&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Some ocular expressions have been described including heliotrope erythema &#40;hyperpigmentation&#44; scaling and palpebral edema&#41;&#44; avascular areas in the conjunctiva&#44; uveitis&#44; scleritis and glaucoma&#44; as well as retinopathy with cotton-like lesions&#44; retinal hemorrhages&#44; retinal edema and optic nerve paleness&#46;<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5&#8211;8</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Diagnostic is based on clinical findings&#44; data compatible with inflammatory myopathy and antibody pattern&#46; The auto-antibodies reported in this conditions include anti-ribonucleoprotein &#40;anti-RNP&#41; which are associated with this overlap in variable percentages &#40;generally under 30&#37;&#41; whereas nearly 90&#37; of patients with scleroderma are positive to antinuclear antibodies &#40;AAN&#41;&#44; and 35&#37; are positive for rheumatoid factor&#46;<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2&#44;9&#44;10</span></a> Treatment includes systemic glycocorticoids although generally an immunosuppressant agent is also required&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Clinic case report</span><p id="par0025" class="elsevierStylePara elsevierViewall">Female&#44; 57&#44; who consulted due to poor vision and intermittent episodes of throbbing pain in both eyes during 20 years&#46; She was diagnosed in another institution with idiopathic panuveitis and treated with topical steroids and midriatics with apparent clinical improvement&#46; Two years prior to consulting at the authors&#8217; practice&#44; the patient debuted with pain in proximal and distal inter-phalanx&#44; metacarpo-phalanx and shoulder joints without morning stiffness and with proximal myalgia in thoracic limbs&#46; In addition&#44; she exhibited dyspnea with median effort and dysphagia against solids as well as lesions in the forehead which increased with exposure to the sun&#46; Pathological personal history included hypertension with 7 years evolution&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Ophthalmological examination produced visual acuity in the right eye &#40;RE&#41; of 20&#47;200 that improved to 20&#47;80 and 20&#47;100 in the left eye &#40;LE&#41; that improved to 20&#47;80&#46; Biomicroscopy revealed 360&#176; scleral thinning&#44; cornea with discrete peripheral thinning&#44; anterior chamber with cellularity 2&#43; and clear lens in the RE &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The LE showed sclera without alterations&#44; anterior chamber with cellularity 2&#43;&#44; posterior synechiae and clear lens &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; RE ocular fundus did not show alterations whereas the LE ocular fundus identified 2&#43; vitreous cellularity &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">General physical examination found Gottron&#39;s papules&#44; heliotrope erythema&#44; changes suggesting Raynaud in the distal phalanx of hands&#44; increased skin consistency in those areas&#44; Heberden and Bouchard nodules and abnormal capillaroscopy &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>&#41;&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">The relevant lab results were&#58; anti-centromere antibodies 1&#58;1&#46;280 &#40;normal&#58; 1&#58;20&#41;&#44; AAN positive &#40;59&#46;7<span class="elsevierStyleHsp" style=""></span>U&#47;ml&#44; with normal &#60;10<span class="elsevierStyleHsp" style=""></span>U&#47;ml&#41;&#44; PCR 20&#46;7<span class="elsevierStyleHsp" style=""></span>mg&#47;l &#40;normal&#58; &#60;3&#46;8&#41;&#44; VSG 38<span class="elsevierStyleHsp" style=""></span>mm&#47;h&#44; CPK 310&#46;7<span class="elsevierStyleHsp" style=""></span>U&#47;l &#40;normal&#58; up to 140&#41;&#44; rheumatoid factor&#44; antibodies against Ro and La negative and normal blood biometry&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Considering the clinic and serological findings&#44; the patient was diagnosed with probable sclerodermatomyositis&#44; based on typical dermatological changes&#44; myalgia&#44; high CPK&#44; distal cutaneous induration&#44; abnormal capillaroscopy and esophagic symptoms&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">In the most recent follow-up&#44; the patient was in treatment with prednisone at a dose of 1<span class="elsevierStyleHsp" style=""></span>mg&#47;kg&#47;day&#44; that was gradually diminished every week&#44; azathioprine 100<span class="elsevierStyleHsp" style=""></span>mg daily&#44; 1&#37; prednisolone drops and reduction dose and midriatics every 12<span class="elsevierStyleHsp" style=""></span>h&#46; The patient remained clinically stable with cellularity diminished to 0&#43; in anterior chamber and vitreous of authorities&#44; and controls extraocular expressions in one year follow-up&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Discussion</span><p id="par0055" class="elsevierStylePara elsevierViewall">The importance of the present case is due both to the infrequent appearance of overlap syndromes and to the scarcity of literature about ocular expressions thereof&#44; in contrast with other self immune dermatological diseases&#46; A study by Akikusa et al&#46; reporting the ocular expressions of 108 patients with juvenile dermatomyositis found that 38&#37; had heliotrope erythema&#44; 18&#46;3&#37; had cataracts mainly attributed to the chronic use of glycocorticoids&#44; 7&#46;4&#37; had palpebral atrophic scars and 4&#46;9&#37; had blepharitis&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> However&#44; retinopathy associated to dermatomyositis is rare&#46; It was first reported in 1958 by Venkatesh et al&#46;&#44; with 2 sclerodermatomyositis cases with proliferative retinopathy&#46; However&#44; to the best of the authors&#8217; knowledge&#44; the case of the present patient is the first to be published with non-necrotizing anterior scleritis and non-granulomatous bilateral anterior uveitis with posterior expressions&#46;<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> The long history of ocular symptoms diagnosed 20 years before must be noted&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">This paper has described some inflammatory ocular expressions which are not typical in a sclerodermatomyositis case and that have contributed to the differential diagnostic&#46; As said disease is rare&#44; its identification requires significant diagnostic suspicion as well as an extensive clinic and laboratory approach that includes full ophthalmological examinations&#46; The prognostic role of ocular findings is yet to be defined&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0070">Conflict of interests</span><p id="par0065" class="elsevierStylePara elsevierViewall">No conflict of interests was declared by the authors&#46;</p></span></span>"
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          "titulo" => "Introduction"
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          "titulo" => "Clinic case report"
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        6 => array:2 [
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          "clase" => "keyword"
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            0 => "Sclerodermatomyositis"
            1 => "Overlap syndrome"
            2 => "Antinuclear antibodies"
            3 => "Sclera"
            4 => "Myositis"
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        ]
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          "clase" => "keyword"
          "titulo" => "Palabras clave"
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            0 => "Esclerodermatomiositis"
            1 => "S&#237;ndrome de superposici&#243;n"
            2 => "Anticuerpos antinucleares"
            3 => "Esclera"
            4 => "Miositis"
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        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Background</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Sclerodermatomyositis is an overlap syndrome of myositis and scleroderma&#44; with dermatological&#44; muscular and joint involvement&#44; but may also present with ocular manifestations&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Clinical case</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A 57 year-old woman presented with ophthalmological manifestations&#44; including scleral thinning 360&#176;&#44; and the presence of cells in the anterior and posterior chamber&#46; Oriented physical examination and laboratory studies led to the diagnosis&#44; with the need for systemic treatment&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conclusion</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Sclerodermatomyositis is a rare disease&#46; Its diagnosis needs thorough clinical and laboratory studies&#44; and its management should be multidisciplinary when inflammatory ocular manifestations may be present&#46;</p></span>"
        "secciones" => array:3 [
          0 => array:2 [
            "identificador" => "abst0005"
            "titulo" => "Background"
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          1 => array:2 [
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        "titulo" => "Resumen"
        "resumen" => "<span id="abst0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Antecedentes</span><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">La esclerodermatomiositis es un s&#237;ndrome de superposici&#243;n que tiene manifestaciones dermatol&#243;gicas&#44; musculares y articulares&#44; y que puede presentar afecci&#243;n ocular&#46;</p></span> <span id="abst0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Caso cl&#237;nico</span><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Se presenta el caso de una mujer de 57 a&#241;os en quien la exploraci&#243;n oftalmol&#243;gica hizo evidente adelgazamiento escleral 360 grados&#44; celularidad anterior y v&#237;trea&#46; La exploraci&#243;n f&#237;sica orientada y los estudios de laboratorio permitieron sustentar el diagn&#243;stico de esclerodermatomiositis&#44; por lo que se trat&#243; de forma sist&#233;mica&#46;</p></span> <span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusi&#243;n</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">La esclerodermatomiositis es una enfermedad rara&#44; cuyo diagn&#243;stico implica estudio cl&#237;nico y de laboratorio&#44; y su manejo debe ser multidisciplinario&#44; donde las manifestaciones oculares inflamatorias pueden estar presentes&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as&#58; Pedroza-Seres M&#44; Serna-Ojeda JC&#44; Flores-Su&#225;rez LF&#46; Esclerodermatomiositis con manifestaciones oculares predominantes&#46; Arch Soc Esp Oftalmol&#46; 2017&#59;92&#58;334&#8211;337&#46;</p>"
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Article information
ISSN: 21735794
Original language: English
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