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Study of aggressive carcinomas through orbital metastasis
Estudio de carcinomas agresivos a través de metástasis orbitarias
G. Espinosa-Barberia,b,
, C. Alba Lineroc, F.J. Galván Gonzáleza, E. Álvarez Gonzáleza, A. Rey Lópezd, F. Medina Riveroa
Corresponding author
a Servicio de Oftalmología, Hospital Universitario de Gran Canaria Doctor Negrín, Las Palmas de Gran Canaria, Spain
b Escuela de Doctorado y Posgrado, Universidad Las Palmas de Gran Canaria, Las Palmas de Gran Canaria, Spain
c Hospital Clínic de Barcelona, Instituto de Oftalmología, Barcelona, Spain
d Servicio de Anatomía Patológica, Hospital Universitario de Gran Canaria Doctor Negrín, Las Palmas de Gran Canaria, Spain
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(A y B) Exploración externa con pérdida de la mirada conjugada, ptosis y movimientos oculares no congruentes. TC de órbitas (C) en el que existe infiltración metastásica bilateral de ambos músculos rectos horizontales.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "G. Espinosa-Barberi, C. Alba Linero, F.J. Galván González, E. Álvarez González, A. Rey López, F. Medina Rivero" "autores" => array:6 [ 0 => array:2 [ "nombre" => "G." "apellidos" => "Espinosa-Barberi" ] 1 => array:2 [ "nombre" => "C." "apellidos" => "Alba Linero" ] 2 => array:2 [ "nombre" => "F.J." "apellidos" => "Galván González" ] 3 => array:2 [ "nombre" => "E." "apellidos" => "Álvarez González" ] 4 => array:2 [ "nombre" => "A." "apellidos" => "Rey López" ] 5 => array:2 [ "nombre" => "F." 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A case report" "tienePdf" => "en" "tieneTextoCompleto" => "en" "tieneResumen" => array:2 [ 0 => "en" 1 => "es" ] "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "257" "paginaFinal" => "259" ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Schwannoma de párpado, a propósito de un caso" ] ] "contieneResumen" => array:2 [ "en" => true "es" => true ] "contieneTextoCompleto" => array:1 [ "en" => true ] "contienePdf" => array:1 [ "en" => true ] "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" => 1389 "Ancho" => 1267 "Tamanyo" => 623826 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">(A) Medium magnification shows peripheral nerve remains in the external portion (arrow) (H&E, 100×). 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"apellidos" => "Medina Rivero" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Servicio de Oftalmología, Hospital Universitario de Gran Canaria Doctor Negrín, Las Palmas de Gran Canaria, Spain" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Escuela de Doctorado y Posgrado, Universidad Las Palmas de Gran Canaria, Las Palmas de Gran Canaria, Spain" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Hospital Clínic de Barcelona, Instituto de Oftalmología, Barcelona, Spain" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Servicio de Anatomía Patológica, Hospital Universitario de Gran Canaria Doctor Negrín, Las Palmas de Gran Canaria, Spain" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Estudio de carcinomas agresivos a través de metástasis orbitarias" ] ] "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" => 1653 "Ancho" => 2083 "Tamanyo" => 285700 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Case 2. (A and B) External examination with loss of conjugated gaze, ptosis and incongruent ocular movements. Orbit CT (C) showing bilateral metastatic infiltration of both horizontal rectus muscles.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Orbital tumoral disease comprises a large group of pathologies that could produce a broad range of symptoms and signs that must be taken into account when examining ophthalmological patients. The prevalence of lesions varies according to different published series, with the literature comprising reviews such as that by Bonavolontà et al.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> reporting predominance of benign pathologies, with 38% of patients exhibiting malign lesions, predominantly non-Hodgkin lymphoma (12%), basal cell carcinoma (3%) and orbital metastases (3%). The most frequent primary tumours are breast, lung, melanoma and prostate.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">In up to 15% of cases, ocular clinic is the first expression of the systemic disease, most common expressions being diplopia (49%), pain (42%), los of visual acuity (VA) (30%) and proptosis (29%).<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">The objective of treatment should be to preserve vision and diminish pain, above all considering that the majority of said tumours associate a mean survival of 9.3-18 months as from metastasis identification, regardless of histological type.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinic case reports</span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Case 1</span><p id="par0020" class="elsevierStylePara elsevierViewall">Male, 63, with history of arterial hypertension, dyslipidemia and ischaemic cardiopathy, with a tobacco index (IPA) of 50 packs/year, who consulted due to VA loss in the right eye (RE), vertical binocular diplopia and tumoration in the right orbital region with 15 days evolution. The patient referred hyporexia and loss of weight exceeding 10<span class="elsevierStyleHsp" style=""></span>kg in the past 6 months, as well as lower limbs weakness the past week.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Ophthalmological examination showed that VA was 20/32 in both eyes (BE). External examination showed a solid palpable consistent mass located superiorly and laterally to the right lateral orbit edge, superior palpebral ptosis and supraversion restriction in RE (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). The excursion of the upper eyelid from the extreme lower gaze up to the extreme upper gaze was 7<span class="elsevierStyleHsp" style=""></span>mm, and the margin-reflex distance 1 (MRD1) was 2<span class="elsevierStyleHsp" style=""></span>mm. Exophthalmometry with Hertel exophthalmometer was 25<span class="elsevierStyleHsp" style=""></span>mm in the RE and 18<span class="elsevierStyleHsp" style=""></span>mm in the left eye (LE). Anterior segment (BSA) featured the presence of corticonuclear cataract in BE. The rest of the ophthalmological examination and supplementary tests produced normal results.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">On the basis of said findings, orbital computerized tomography (CT) was taken, enabling the identification of a lesion measuring a maximum axial diameter of 4.9<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>3.4<span class="elsevierStyleHsp" style=""></span>cm, with intraconal extension from the upper external margin of the orbit due to destruction of the frontal bone, hindering the anterior displacement of the ocular globe (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>A). Thoracoabdominal CT confirmed the presence of disseminated pulmonary neoplasia compatible with stage IV (T3N3M1C), with multiple bone metastases in the spinal column invading the spinal canal and exerting macular compression at different levels (D3, D8-D9, L1) (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>B–D).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">Due to the extension of the mass towards soft tissue and easy accessibility through percutaneous approach, the patient was referred to Pathological Anatomy for cytology study with thin needle puncture-aspiration, reported as metastasis of microcytic bronchogenic carcinoma (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>E). The patient was admitted for administration of chemotherapy scheme (carboplatin and etoposide) and palliative radiotherapy.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Case 2</span><p id="par0040" class="elsevierStylePara elsevierViewall">Female, 69, diagnosed one year earlier with infiltrating ductal breast cancer in stage T2N0M0, with positive hormonal receptors and in current treatment with chemotherapy and hormonal therapy, who visited due to binocular diplopia in all gaze positions with 2 months evolution.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ophthalmological examination showed VA of 20/40 in RE and 20/32 in LE in addition to motility restriction in all gaze positions and a 22<span class="elsevierStyleHsp" style=""></span>mm non-pulsating proptosis under exophthalmometry (Hertel exophthalmometer) in the RE. LE exhibited diminished ocular movements and 18<span class="elsevierStyleHsp" style=""></span>mm proptosis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>A and B).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">BSA examination showed intense conjunctival chemosis in the RE. ocular pressure readings were within the normal ranges, with forced duction test for BE. Ocular fundus (OF) did not exhibit optic nerve compromise or visible chorioretinal alterations, while optical coherence tomography (OCT) produced normal results with preserved foveolar profiles.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Due to suspected vascular lesion of the carotid-cavernous fistula type with orbital compromise, neuroimaging tests were requested. CT showed infiltration of bilateral extraocular muscles by tumoral mass, larger at the right aside, without compromising both optic nerves in axial sections (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>C).</p><p id="par0060" class="elsevierStylePara elsevierViewall">PAAF biopsy of the intra-orbital mass was taken, reported as pleomorphic cells compatible with metastasis secondary to breast carcinoma. The extension study revealed metastatic compromise towards lungs and lumbar vertebrae. Due to the poor prognosis of the patient, palliative treatment with corticoids was prescribed in addition to continuing hormone therapy, although the patient died 2 months after diagnosis due to the orbital disease.</p></span></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0045">Discussion</span><p id="par0065" class="elsevierStylePara elsevierViewall">The orbital region can be affected by a range of pathologies, the most prevalent of which are inflammatory and, within these, Graves’ disease, followed by neoplastic diseases.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> Secondary tumours and/or metastatic disease express in only 2-3% of patients with cancer, although it is considered to be an infradiagnosed pathology due to nonspecific clinic, poor vital prognosis and difficult access. Accordingly, said lesions could be defined as relatively infrequent.<a class="elsevierStyleCrossRefs" href="#bib0055"><span class="elsevierStyleSup">1,4</span></a> Tumours with higher metastasis risk include breast cancer in females and prostate or lung cancer in males.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> These are rapidly growing and potentially destructive lesions, as can be appreciated in the imaging tests of the first case. The fact of presenting said lytic image indicates aggressiveness which increased suspicions on malignity.</p><p id="par0070" class="elsevierStylePara elsevierViewall">Patients could present nonspecific symptoms from early stages. The main signs include proptosis with double vision as a symptom,<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5,6</span></a> both of which were observed in both cases, with the most constant symptom being exophthalmos due to occupation of the intraorbital space. As a consequence of the broad range of symptoms, age of expression as well as higher incidence in females, differential diagnostic with other more frequent pathologies should be performed, such as dysthyroid orbitopathy and vascular malformations as initially suspected in the second case, where other causes could be excluded due to the neuroimaging tests.</p><p id="par0075" class="elsevierStylePara elsevierViewall">In the diagnostic of both patients, the history of the first (marked tobacco smoking habit) as well as the second patient (mammary carcinoma) pointed towards suspected tumour from the beginning. In these cases, the CT study was crucial because in several locations there is no visible compromise of the OF or in echography. Said neuroimaging test has demonstrated to be a useful tool not only for diagnosing said pathology but also for enabling the identification of bone changes and confirming follow-up.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a></p><p id="par0080" class="elsevierStylePara elsevierViewall">In both cases, atypical data could be observed. In the first patient, the orbital disease was the starting point of the systemic study and lung lesion diagnostic, as only in 10% of cases metastatic disease clinic constitutes the first expression of a primary tumour, and in 70% of cases it is a unique metastasis.<a class="elsevierStyleCrossRef" href="#bib0080"><span class="elsevierStyleSup">6</span></a> A peculiarity to take into account in the second patient is the presence of extraocular musculature infiltration in the context of breast carcinoma because it is a relatively infrequent condition in clinic practice.<a class="elsevierStyleCrossRefs" href="#bib0085"><span class="elsevierStyleSup">7–9</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">For confirmation diagnostic, PAAF constitutes an excellent and hardly invasive option that facilitates the identification of the type and possible location of the primary tumour, although it is important to have an accessible area in order to avoid false negatives, in which case an open incisional biopsy can be taken.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> In the present patients, said technique did not involve added complications and results were readily available.</p><p id="par0090" class="elsevierStylePara elsevierViewall">By way of conclusion, it can be stated that the cases presented herein are atypical described forms of metastatic orbital disease. In the first case, the orbital compromise was the first expression of the disease and greatly facilitated the primary tumour diagnostic. The second case presented rectus muscles infiltration, an infrequent finding. Due to the general deterioration of said patients and the advanced stage of the disease, as described above it was decided to establish palliative treatment in order to improve their quality of life during the survival period.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0050">Conflict of interests</span><p id="par0095" class="elsevierStylePara elsevierViewall">The authors state that they have no commercial interest and have not received financial support.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres1184814" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1104924" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1184815" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1104925" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:3 [ "identificador" => "sec0010" "titulo" => "Clinic case reports" "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0015" "titulo" => "Case 1" ] 1 => array:2 [ "identificador" => "sec0020" "titulo" => "Case 2" ] ] ] 6 => array:2 [ "identificador" => "sec0025" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflict of interests" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2018-07-23" "fechaAceptado" => "2018-10-29" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1104924" "palabras" => array:5 [ 0 => "Orbital metastases" 1 => "Lung cell carcinoma" 2 => "Breast cancer" 3 => "Diplopia" 4 => "Proptosis" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1104925" "palabras" => array:5 [ 0 => "Metástasis orbitaria" 1 => "Carcinoma de pulmón" 2 => "Cáncer de mama" 3 => "Diplopía" 4 => "Proptosis" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Two clinical cases of orbital metastasis are presented. The first is a 63 year-old male with a recent loss of visual acuity associated with binocular diplopia. Neuroimaging tests revealed a tumour of aggressive features with intraconal extension and bone invasion. The systemic study led to the diagnosis of stage IV small cell bronchogenic carcinoma. The second case is a 69 year-old woman with breast cancer in early stages that showed alterations in ocular motility and proptosis. Computed tomography showed tumour infiltration of extraocular musculature. The anatomo-pathological study confirmed the diagnosis of orbital metastasis. Orbital metastatic disease is a relatively infrequent entity with varied clinical presentation and an unfavourable prognosis. It constitutes a diagnostic challenge that should be suspected in patients with risk factors for tumour disease and imaging techniques used to define the extent and severity of the condition.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Se presentan dos casos clínicos de metástasis orbitaria. El primero es el de un varón de 63 años con pérdida de agudeza visual reciente asociada a diplopía binocular. Las pruebas de neuroimagen revelan una tumoración de características agresivas con extensión intraconal e invasión ósea. El estudio sistémico nos lleva al diagnóstico de carcinoma broncogénico microcítico en estadio IV. El segundo caso corresponde a una mujer de 69 años con cáncer de mama en estadios precoces que presenta alteraciones de motilidad ocular y proptosis. Mediante tomografía computarizada se evidencia infiltración tumoral de musculatura extraocular. El estudio anatomopatológico confirma el diagnóstico de metástasis orbitaria.</p><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">La enfermedad metastásica orbitaria es una entidad relativamente infrecuente, con presentación clínica variada y pronóstico desfavorable. Constituye un reto diagnóstico que se debe sospechar en pacientes con factores de riesgo para enfermedad tumoral y recurrir a las técnicas de imagen para definir la extensión y severidad del cuadro.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Espinosa-Barberi G, Alba Linero C, Galván González FJ, Álvarez González E, Rey López A, Medina Rivero F. Estudio de carcinomas agresivos a través de metástasis orbitarias. Arch Soc Esp Oftalmol. 2019;94:252–256.</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" => 2082 "Ancho" => 2083 "Tamanyo" => 628450 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">External examination, showing loss of conjugated gaze, supraversion limitation and adduction in the right eye.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 2086 "Ancho" => 2083 "Tamanyo" => 548775 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Case 1. Orbit CT c/c (A) with solid and lytic mass on the right orbit, compromising the lateral rectus. Thoracoabdominal CT showing 5<span class="elsevierStyleHsp" style=""></span>cm right side pulmonary mass (B) that extends to displace the trachea (C) and occupies the vertebral canal at D9 (D). Anatomopathological study with Papanicolaou staining cytology (E) showing geomorphic cells with very little cytoplasm, increased nucleus and necrotic areas.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1653 "Ancho" => 2083 "Tamanyo" => 285700 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">Case 2. (A and B) External examination with loss of conjugated gaze, ptosis and incongruent ocular movements. 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