Endophthalmitis of probable endogenous origin caused by Scedosporium boydii: A case report

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Informe 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" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1001 "Ancho" => 1300 "Tamanyo" => 163114 ] ] "descripcion" => array:1 [ "en" => "Fungal agents. (A) India ink preparation of cerebrospinal fluid showing an encapsulated yeast-like Cryptococcus neoformans (1000x). (B) Direct immunofluorescence in induced sputum showing the honeycombed-structures consistent with Pneumocystis jirovecii (400x). (C) Gomori-Grocott silver stain in induced sputum showing small fungal cells consistent with Histoplasma capsulatum (400x). (D) Bone marrow histopathology showing yeasts consistent with Histoplasma capsulatum (arrows) and Cryptococcus neoformans (Gomori-Grocott silver stain, 400x)." ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "Fernando Almeida-Silva, Lisandra Serra Damasceno, Maria Jose Buitrago Serna, Clara Valero, Leonardo Pereira Quintella, Rodrigo Almeida-Paes, Mauro de Medeiros Muniz, Rosely Maria Zancope-Oliveira" "autores" => array:8 [ 0 => array:2 [ "nombre" => "Fernando" "apellidos" => "Almeida-Silva" ] 1 => array:2 [ "nombre" => "Lisandra Serra" "apellidos" => "Damasceno" ] 2 => array:2 [ "nombre" => "Maria Jose Buitrago" "apellidos" => "Serna" ] 3 => array:2 [ "nombre" => "Clara" "apellidos" => "Valero" ] 4 => array:2 [ "nombre" => "Leonardo Pereira" "apellidos" => "Quintella" ] 5 => array:2 [ "nombre" => "Rodrigo" "apellidos" => "Almeida-Paes" ] 6 => array:2 [ "nombre" => "Mauro de Medeiros" "apellidos" => "Muniz" ] 7 => array:2 [ "nombre" => "Rosely Maria" "apellidos" => "Zancope-Oliveira" ] ] ] ] ] "idiomaDefecto" => "en" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1130140615000790?idApp=UINPBA00004N" "url" => "/11301406/0000003300000002/v2_201607020120/S1130140615000790/v2_201607020120/en/main.assets" ] "en" => array:20 [ "idiomaDefecto" => true "cabecera" => "Note" "titulo" => "Endophthalmitis of probable endogenous origin caused by Scedosporium boydii: A case report" "tieneTextoCompleto" => true "paginas" => array:1 [ 0 => array:2 [ "paginaInicial" => "122" "paginaFinal" => "125" ] ] "autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Virginia Vanzzini-Zago, Sonia Corredor-Casas, Abelardo Rodríguez-Reyes, Francisca Hernández-Hernández, Patricia Manzano-Gayosso, Rubén López Martínez, Alan Ricardo Alba Orozco" "autores" => array:7 [ 0 => array:4 [ "nombre" => "Virginia" "apellidos" => "Vanzzini-Zago" "email" => array:1 [ 0 => "vivanzzini@yahoo.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "Sonia" "apellidos" => "Corredor-Casas" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "d" "identificador" => "aff0020" ] ] ] 2 => array:3 [ "nombre" => "Abelardo" "apellidos" => "Rodríguez-Reyes" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "b" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "Francisca" "apellidos" => "Hernández-Hernández" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] 4 => array:3 [ "nombre" => "Patricia" "apellidos" => "Manzano-Gayosso" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] 5 => array:3 [ "nombre" => "Rubén López" "apellidos" => "Martínez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "c" "identificador" => "aff0015" ] ] ] 6 => array:3 [ "nombre" => "Alan Ricardo Alba" "apellidos" => "Orozco" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "e" "identificador" => "aff0025" ] ] ] ] "afiliaciones" => array:5 [ 0 => array:3 [ "entidad" => "Laboratorio de Microbiología del Hospital Asociación para evitar la Ceguera en México, Mexico" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Laboratorio de Patología del Hospital Asociación para Evitar la Ceguera en México, Mexico" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Departamento de Microbiología y Parasitología, Facultad de Medicina, Universidad Nacional Autónoma de México, México, D.F., Mexico" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Ophthalmic Pathology Chief, Instituto Mexicano de Oftalmología IAP Querétaro - Querétaro, Mexico" "etiqueta" => "d" "identificador" => "aff0020" ] 4 => array:3 [ "entidad" => "Becario del Servicio de Retina del Hospital Asociación para evitar la Ceguera en México, Mexico" "etiqueta" => "e" "identificador" => "aff0025" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Endoftalmitis de probable origen endógeno causada por Scedosporium boydii. Informe de un caso" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0025" "etiqueta" => "Fig. 5" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr5.jpeg" "Alto" => 679 "Ancho" => 1600 "Tamanyo" => 281136 ] ] "descripcion" => array:1 [ "en" => "(A) Cleistothecium observed after three weeks of incubation on Sabouraud–Emmons agar at room temperature (40×). (B) Oblong brown ascospores released after rupture of the cleistothecial wall (100×)." ] ] ] "textoCompleto" => "The few ocular infection cases shown to be caused by the Scedosporium apiospermum species complex have been a challenge to treat because of the delayed diagnoses, and poor response to antifungal drugs and surgical treatment.<a class="elsevierStyleCrossRef" href="#bib0100">1</a>Patients diagnosed with fungal endogenous endophthalmitis are associated with transient fungaemia and/or underlying systemic disorders. Patients with uncontrolled diabetes mellitus, long periods of antibiotic treatment, or immunosuppression therapy are at risk for endogenous endophthalmitis.<a class="elsevierStyleCrossRef" href="#bib0105">2</a> Conversely, there are some reports stating that corneal infection with the Scedosporium species complex is associated with traumatic injuries and concomitant exogenous endophthalmitis.<a class="elsevierStyleCrossRef" href="#bib0170">15</a>Some upper respiratory tract fungal infections have been associated with exogenous fungal endophthalmitis due to the loss of the natural mechanical barriers by destruction of the periorbital paranasal sinus bone walls. Candida species have been reported as a frequent cause of endogenous endophthalmitis via the haematogenous route in drug abusers, in patients under prolonged broad spectrum antibiotic therapy, or in patients after intestinal septic embolism.<a class="elsevierStyleCrossRefs" href="#bib0175">16,18</a>Aspergillus spp., Fusarium spp. and some pigmented fungi have also been associated with this pathology, and these fungi commonly induce a poor visual outcome.<a class="elsevierStyleCrossRefs" href="#bib0140">9,12</a> Endophthalmitis cases with both traumatic and non-traumatic origin have been described, indicating a severe septicaemia condition.<a class="elsevierStyleCrossRef" href="#bib0130">7</a> Here we report an endogenous fungal endophthalmitis case in a diabetic patient with a previous femur surgery, delayed diagnosis and a poor therapy outcome.Case reportA 69-year-old man diagnosed 6 years before with diabetes mellitus (not well controlled with glibenclamide due to the irregular intake of the medication), and with chronic arterial hypertension (treated irregularly with captopril) underwent surgery of the right femur 9 months before his first ophthalmic visit. He sought ocular medical assistance due to several symptoms: right ocular pain for the last 10 days, progressive loss of vision over the past 8 months and the presence of a central white spot on his right eye cornea for the last 4 days.The ocular examination showed that the right eye pupil was unresponsive to direct or consensual papillary reflex. Corneal edema, 100% hypopyon, hyperemic conjunctiva, and ciliar reaction of the limbus were present. The intraocular pressure (IOP) was 2mmHg, and the patient indicated no light perception. No other disorders were visible.Since our first ocular evaluation, we found that the vision and direct papillary reflex of the left eye were normal, and the anterior segment had no signs of inflammation. The IOP was 10mmHg, cup 0.65, and there were no signs of diabetic retinopathy. The patient was receiving treatment with timolol, brimonidine, dorzolamide, and latanoprost for open angle glaucoma in the left eye.The patient was hospitalized with a clinical diagnosis of endophthalmitis in the blind and painful right eye. An ultrasound showed total funnel retinal detachment in contact with the posterior lens capsule (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>); there were no mobile condensations in the vitreous cavity or diffuse inflammatory thickening of the choroid. The haematogenous abnormalities were leukocyte cell count 9000mm–3; 48.9mg/dl glucose; 53.6mg/dl blood urea; 1.47mg/dl blood creatinin.<elsevierMultimedia ident="fig0005"></elsevierMultimedia>All anti-glaucoma drugs for the left eye were discontinued as the patient did not have an optic disk glaucoma lesion. For the right eye, enucleation was successfully performed, and the post-operative course was satisfactory.The extracted tissue was sent for microbiological and histopathology studies. A Gram stain showed that no bacteria were present. The periodic acid Schiff (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>) and Grocott stains revealed thin septate hyphae, which were surrounded by histiocytes, neutrophils, and necrotic debris. The hyphae were located throughout the retina and choroid. Fibrosis and retinal atrophic changes (gliosis and loss of ganglion cells) were observed around the main lesion. The cornea presented with ulceration and polymorphonuclear leukocyte infiltration, but fungal cells were not observed in the corneal structures.<elsevierMultimedia ident="fig0010"></elsevierMultimedia>The blood agar and chocolate agar cultures were incubated in a 5% CO2 anaerobic atmosphere at 37°C. The culture on Sabouraud–Emmons agar was aerobically incubated at 27°C. Fungal growth was observed in all cultures after 5–8 days. On blood agar, several tan, cottony/powdered fungal colonies developed (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). On chocolate agar, gray, white cottony colonies were observed. On Sabouraud–Emmons agar, white cottony colonies, which turned to dark brown after 30 days, developed. No aerobic or anaerobic bacteria grew after 15 days of incubation at 37°C.<elsevierMultimedia ident="fig0015"></elsevierMultimedia>In order to study the macroscopic and microscopic morphology, a new culture and microculture from a monosporic subculture were performed on potato dextrose agar. For the PCR study, DNA from a monosporic culture in Sabouraud dextrose broth was extracted using a commercial kit (Exgene Plant SV, GeneAll®, Korea) following the manufacturer's instructions. For the PCR, the ITS1-ITS4 oligonucleotides were used as recommended by Luo and Mitchell.<a class="elsevierStyleCrossRef" href="#bib0160">13</a> The PCR components for each reaction were: 100ng of DNA, 2mM MgCl, 0.25mM dNTPs, 0.2μM oligonucleotides, and 3.5U Taq (Thermo Scientific, USA). An approximately 600bp fragment was obtained and then sequenced using the Big Dye Terminator version 3.1 Sequencing kit<a class="elsevierStyleCrossRef" href="#bib0165">14</a> on an ABI 3130xl Genetic Analyzer (Applied Biosystem HITACHI).On potato dextrose agar, the rapidly growing colony consisted of a fluffy aerial mycelium, which was at first white but then became cinnamon-brown (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>A). Microscopic examination showed hyaline hyphae (1–2μm in diameter), pyriform annelloconidia (4μm×6μm) borned singly on branched or simple elongate conidiophores (<a class="elsevierStyleCrossRef" href="#fig0020">Fig. 4</a>B and C). These characteristics were not significantly different among the three culture media used. After three weeks, brown to black round shaped-cleistothecia (140–200μm) with ellipsoidal, smooth-walled, pale yellow to golden brown ascospores (7μm×4.5μm), were observed (<a class="elsevierStyleCrossRef" href="#fig0025">Fig. 5</a>A and B). Tufts of conidiophores forming elongated synnemata of the Graphium type conidiation were also present. Considering these morphological characteristics, the isolate was suggested to be a member of the S. apiospermum species complex.<elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia>The identity of Scedosporium boydii was finally achieved based on the sequencing of the PCR amplicon of the internal transcribed spacer 1 and internal transcribed spacer 2. The sequenced fragments matched 100% to S. boydii (formerly named Pseudallescheria boydii<a class="elsevierStyleCrossRef" href="#bib0150">11</a>). The accession numbers are <a id="intr0005" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=JQ690940">JQ690940</a>, <a id="intr0010" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888398">AJ888398</a>, <a id="intr0015" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888383">AJ888383</a>, <a id="intr0020" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888394">AJ888394</a>, and <a id="intr0025" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888398">AJ888398</a>.DiscussionEndogenous endophthalmitis caused by the S. apiospermum species complex is not common and patients have typically a poor visual prognosis with a low likelihood of organ preservation. This disease has been diagnosed in patients with underlying systemic neutropenia, acute lymphocytic leukemia, Wegener's granulomatosis,<a class="elsevierStyleCrossRef" href="#bib0135">8</a> organ transplant patients, and immunocompetent patients with pulmonary fibrosis.<a class="elsevierStyleCrossRef" href="#bib0180">17</a>In the case described here, we think that uncontrolled diabetes mellitus was the main risk factor for the development of endophthalmitis, probably of endogenous origin. After a right femur surgery nine months prior to the ocular complaint, an unidentified infectious microorganism caused a transient blood infection with a septic embolus to the retina, which was the possible cause of the endogenous fungal endophthalmitis. The patient sought late for ophthalmic attention, which resulted in a delayed diagnosis. The fungus was present in the choroid and retina tissue smears, as well as in the cultures. Due to the retinal detachment, decreased ocular pressure, loss of light perception, the pain in the eye, and the risk of dissemination, enucleation was the better option.In other cases, when an early diagnosis of this disease has been established, a therapeutic regimen consisting of vitrectomy with intravitreal 0.1% voriconazole and oral voriconazole at regular doses has been indicated with successful outcomes.<a class="elsevierStyleCrossRef" href="#bib0190">19</a>In the present case, a very late diagnosis was established in a blind and painful eye; therefore, the patient was not treated with voriconazole. Enucleation was necessary for a complete removal of the infected tissues, preventing in this way a potential dissemination. The mycological and molecular studies confirmed S. boydii as the cause of the ocular mycosis.S. apiospermum is a complex of species that reproduce asexually, although sexual reproduction, as in this case, is also possible.<a class="elsevierStyleCrossRef" href="#bib0110">3</a> This holomorphic complex is genotypically diverse, and the techniques for identification and epidemiological studies are routinely used on pulmonary secretions obtained from cystic fibrosis patients.<a class="elsevierStyleCrossRef" href="#bib0145">10</a>S. boydii is one of the most clinically relevant species in the S. apiospermum complex and is also the most virulent.<a class="elsevierStyleCrossRef" href="#bib0125">6</a> Conventional phenotypic methods are of limited use for species differentiation within this complex, and precise identification requires molecular approaches.<a class="elsevierStyleCrossRefs" href="#bib0115">4,5</a> In the present case, the initial morphological identification was highly suggestive of a species of the S. apiospermum complex, but the molecular approaches were necessary to confirm the identity of the species. Endophthalmitis caused by species of the S. apiospermum complex is a devastating disease with a poor response to antifungal therapy and a poor visual outcome. To our knowledge, this is the first report of a S. boydii endophthalmitis in Mexico, which was confirmed by mycological molecular analyses.Conflicts of interestAuthors declare that there are no conflicts of interest." "textoCompletoSecciones" => array:1 [ "secciones" => array:9 [ 0 => array:3 [ "identificador" => "xres679576" "titulo" => "Abstract" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Background" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Case report" ] 2 => array:2 [ "identificador" => "abst0015" "titulo" => "Conclusions" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec685497" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres679577" "titulo" => "Resumen" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "abst0020" "titulo" => "Antecedentes" ] 1 => array:2 [ "identificador" => "abst0025" "titulo" => "Caso clínico" ] 2 => array:2 [ "identificador" => "abst0030" "titulo" => "Conclusiones" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec685498" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Case report" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Discussion" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 7 => array:2 [ "identificador" => "xack229159" "titulo" => "Acknowledgements" ] 8 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2014-10-05" "fechaAceptado" => "2015-07-24" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec685497" "palabras" => array:4 [ 0 => "Scedosporium apiospermum species complex" 1 => "Fungal endophthalmitis" 2 => "Mycosis" 3 => "Enucleation" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec685498" "palabras" => array:4 [ 0 => "Complejo de especies de Scedosporium apiospermum" 1 => "Endoftalmitis fúngica" 2 => "Micosis" 3 => "Enucleación" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:3 [ "titulo" => "Abstract" "resumen" => "BackgroundMycotic ocular infections caused by the Scedosporium apiospermum species complex are challenging to treat because of the delayed diagnoses and poor responses to antifungal drugs and surgical treatment. Case reportA case of a 69-year-old male patient with a history of diabetes mellitus type 2 and prior surgery on the right femur is described. In the 10 days prior to the ophthalmic consultation he started with ocular pain, adding to a previous and progressive loss of visual acuity in his right eye. The diagnosis of endophthalmitis of probable endogenous origin was established. Despite medical treatment, the patient's condition worsened and, due to the imminent risks, an enucleation was performed. Smears of the enucleation tissue revealed fungal cells, and the cultures yielded a fungus belonging to the S. apiospermum species complex, which was identified as Scedosporium boydii by morphological characteristics and sequencing of a PCR amplicon. ConclusionsA diagnosis of endophthalmitis of probable endogenous origin in the right eye was based on a previous right femur surgery. Potential risk to the patient led to enucleation." "secciones" => array:3 [ 0 => array:2 [ "identificador" => "abst0005" "titulo" => "Background" ] 1 => array:2 [ "identificador" => "abst0010" "titulo" => "Case report" ] 2 => array:2 [ "identificador" => "abst0015" "titulo" => "Conclusions" ] ] ] "es" => array:3 [ "titulo" => "Resumen" "resumen" => "AntecedentesLas infecciones micóticas oculares causadas por el complejo de especies de Scedosporium apiospermum son un reto en el tratamiento por su diagnóstico tardío y la pobre respuesta a los antimicóticos y al tratamiento quirúrgico. Caso clínicoSe describe el caso de un paciente de sexo masculino de 69 años con antecedentes de diabetes mellitus de tipo 2 y cirugía previa del fémur derecho. Diez días antes de la consulta oftalmológica comenzó con dolor ocular que se sumaba a una pérdida previa y progresiva de la capacidad visual en el ojo derecho. Se estableció el diagnóstico de endoftalmitis de posible origen endógeno. A pesar del tratamiento, el paciente no presentó mejoría y por los inminentes riesgos se decidió llevar a cabo una enucleación. El frotis a partir del tejido enucleado mostró células fúngicas y los cultivos revelaron un hongo perteneciente al complejo S. apiospermum, identificado como Scedosporium boydii por procedimientos morfológicos y por secuenciación de un amplicón de PCR. ConclusionesEl diagnóstico de endoftalmitis de probable origen endógeno en el ojo derecho se basó en el antecedente de una cirugía previa de fémur, cuyos riesgos para el paciente condujeron a una enucleación." "secciones" => array:3 [ 0 => array:2 [ "identificador" => "abst0020" "titulo" => "Antecedentes" ] 1 => array:2 [ "identificador" => "abst0025" "titulo" => "Caso clínico" ] 2 => array:2 [ "identificador" => "abst0030" "titulo" => "Conclusiones" ] ] ] ] "multimedia" => array:5 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 718 "Ancho" => 990 "Tamanyo" => 81730 ] ] "descripcion" => array:1 [ "en" => "A right eye ultrasound image showing a total retinal detachment contacting the posterior lens capsule." ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 709 "Ancho" => 951 "Tamanyo" => 206392 ] ] "descripcion" => array:1 [ "en" => "PAS stained section of the enucleated ocular tissue. Irregular, thin, septate hyphae and round fungal structures (arrows) with a background of necrotic detritus and neutrophils were observed (40×)." ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 876 "Ancho" => 900 "Tamanyo" => 105733 ] ] "descripcion" => array:1 [ "en" => "Primary isolation showing several white to tan, cottony fungal colonies on blood agar after 8 days of incubation." ] ] 3 => array:7 [ "identificador" => "fig0020" "etiqueta" => "Fig. 4" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr4.jpeg" "Alto" => 748 "Ancho" => 1600 "Tamanyo" => 157536 ] ] "descripcion" => array:1 [ "en" => "Macroscopic and microscopic morphology of the causative Scedosporium boydii. (A) A colony grown on potato dextrose agar is shown. 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Guest" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1007/s10792-007-9095-0" "Revista" => array:6 [ "tituloSerie" => "Int Ophthalmol" "fecha" => "2007" "volumen" => "27" "paginaInicial" => "391" "paginaFinal" => "394" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/17901927" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] "agradecimientos" => array:1 [ 0 => array:4 [ "identificador" => "xack229159" "titulo" => "Acknowledgements" "texto" => "We thank QFB Erika Córdova-Martínez (Departamento de Microbiología y Parasitología, Facultad de Medicina, UNAM) for her technical support with the molecular procedures and Oscar Albis Donado, MD (ophthalmologist) for his assistance revising the manuscript." "vista" => "all" ] ] ] "idiomaDefecto" => "en" "url" => "/11301406/0000003300000002/v2_201607020120/S1130140616000036/v2_201607020120/en/main.assets" "Apartado" => array:4 [ "identificador" => "7995" "tipo" => "SECCION" "es" => array:2 [ "titulo" => "Notas" "idiomaDefecto" => true ] "idiomaDefecto" => "es" ] "PDF" => "https://static.elsevier.es/multimedia/11301406/0000003300000002/v2_201607020120/S1130140616000036/v2_201607020120/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S1130140616000036?idApp=UINPBA00004N"]

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Endophthalmitis of probable endogenous origin caused by Scedosporium boydii: A case report

Endoftalmitis de probable origen endógeno causada por Scedosporium boydii. Informe de un caso

Virginia Vanzzini-Zagoa,

Corresponding author

vivanzzini@yahoo.com

Corresponding author.

, Sonia Corredor-Casasd, Abelardo Rodríguez-Reyesb, Francisca Hernández-Hernándezc, Patricia Manzano-Gayossoc, Rubén López Martínezc, Alan Ricardo Alba Orozcoe

a Laboratorio de Microbiología del Hospital Asociación para evitar la Ceguera en México, Mexico

b Laboratorio de Patología del Hospital Asociación para Evitar la Ceguera en México, Mexico

c Departamento de Microbiología y Parasitología, Facultad de Medicina, Universidad Nacional Autónoma de México, México, D.F., Mexico

d Ophthalmic Pathology Chief, Instituto Mexicano de Oftalmología IAP Querétaro - Querétaro, Mexico

e Becario del Servicio de Retina del Hospital Asociación para evitar la Ceguera en México, Mexico

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<span class="elsevierStyleItalic">Candida</span> species have been reported as a frequent cause of endogenous endophthalmitis via the haematogenous route in drug abusers&#44; in patients under prolonged broad spectrum antibiotic therapy&#44; or in patients after intestinal septic embolism&#46;<a class="elsevierStyleCrossRefs" href="#bib0175"><span class="elsevierStyleSup">16&#44;18</span></a><span class="elsevierStyleItalic">Aspergillus</span> spp&#46;&#44; <span class="elsevierStyleItalic">Fusarium</span> spp&#46; and some pigmented fungi have also been associated with this pathology&#44; and these fungi commonly induce a poor visual outcome&#46;<a class="elsevierStyleCrossRefs" href="#bib0140"><span class="elsevierStyleSup">9&#44;12</span></a> Endophthalmitis cases with both traumatic and non-traumatic origin have been described&#44; indicating a severe septicaemia condition&#46;<a class="elsevierStyleCrossRef" href="#bib0130"><span class="elsevierStyleSup">7</span></a> Here we report an endogenous fungal endophthalmitis case in a diabetic patient with a previous femur surgery&#44; delayed diagnosis and a poor therapy outcome&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0055">Case report</span><p id="par0020" class="elsevierStylePara elsevierViewall">A 69-year-old man diagnosed 6 years before with diabetes mellitus &#40;not well controlled with glibenclamide due to the irregular intake of the medication&#41;&#44; and with chronic arterial hypertension &#40;treated irregularly with captopril&#41; underwent surgery of the right femur 9 months before his first ophthalmic visit&#46; He sought ocular medical assistance due to several symptoms&#58; right ocular pain for the last 10 days&#44; progressive loss of vision over the past 8 months and the presence of a central white spot on his right eye cornea for the last 4 days&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">The ocular examination showed that the right eye pupil was unresponsive to direct or consensual papillary reflex&#46; Corneal edema&#44; 100&#37; hypopyon&#44; hyperemic conjunctiva&#44; and ciliar reaction of the limbus were present&#46; The intraocular pressure &#40;IOP&#41; was 2<span class="elsevierStyleHsp" style=""></span>mmHg&#44; and the patient indicated no light perception&#46; No other disorders were visible&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">Since our first ocular evaluation&#44; we found that the vision and direct papillary reflex of the left eye were normal&#44; and the anterior segment had no signs of inflammation&#46; The IOP was 10<span class="elsevierStyleHsp" style=""></span>mmHg&#44; cup 0&#46;65&#44; and there were no signs of diabetic retinopathy&#46; The patient was receiving treatment with timolol&#44; brimonidine&#44; dorzolamide&#44; and latanoprost for open angle glaucoma in the left eye&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">The patient was hospitalized with a clinical diagnosis of endophthalmitis in the blind and painful right eye&#46; An ultrasound showed total funnel retinal detachment in contact with the posterior lens capsule &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#59; there were no mobile condensations in the vitreous cavity or diffuse inflammatory thickening of the choroid&#46; The haematogenous abnormalities were leukocyte cell count 9000<span class="elsevierStyleHsp" style=""></span>mm<span class="elsevierStyleSup">&#8211;3</span>&#59; 48&#46;9<span class="elsevierStyleHsp" style=""></span>mg&#47;dl glucose&#59; 53&#46;6<span class="elsevierStyleHsp" style=""></span>mg&#47;dl blood urea&#59; 1&#46;47<span class="elsevierStyleHsp" style=""></span>mg&#47;dl blood creatinin&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">All anti-glaucoma drugs for the left eye were discontinued as the patient did not have an optic disk glaucoma lesion&#46; For the right eye&#44; enucleation was successfully performed&#44; and the post-operative course was satisfactory&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The extracted tissue was sent for microbiological and histopathology studies&#46; A Gram stain showed that no bacteria were present&#46; The periodic acid Schiff &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41; and Grocott stains revealed thin septate hyphae&#44; which were surrounded by histiocytes&#44; neutrophils&#44; and necrotic debris&#46; The hyphae were located throughout the retina and choroid&#46; Fibrosis and retinal atrophic changes &#40;gliosis and loss of ganglion cells&#41; were observed around the main lesion&#46; The cornea presented with ulceration and polymorphonuclear leukocyte infiltration&#44; but fungal cells were not observed in the corneal structures&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">The blood agar and chocolate agar cultures were incubated in a 5&#37; 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the rapidly growing colony consisted of a fluffy aerial mycelium&#44; which was at first white but then became cinnamon-brown &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>A&#41;&#46; Microscopic examination showed hyaline hyphae &#40;1&#8211;2<span class="elsevierStyleHsp" style=""></span>&#956;m in diameter&#41;&#44; pyriform annelloconidia &#40;4<span class="elsevierStyleHsp" style=""></span>&#956;m<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>6<span class="elsevierStyleHsp" style=""></span>&#956;m&#41; borned singly on branched or simple elongate conidiophores &#40;<a class="elsevierStyleCrossRef" href="#fig0020">Fig&#46; 4</a>B and C&#41;&#46; These characteristics were not significantly different among the three culture media used&#46; After three weeks&#44; brown to black round shaped-cleistothecia &#40;140&#8211;200<span class="elsevierStyleHsp" style=""></span>&#956;m&#41; with ellipsoidal&#44; smooth-walled&#44; pale yellow to golden brown ascospores &#40;7<span class="elsevierStyleHsp" style=""></span>&#956;m<span class="elsevierStyleHsp" style=""></span>&#215;<span class="elsevierStyleHsp" style=""></span>4&#46;5<span class="elsevierStyleHsp" style=""></span>&#956;m&#41;&#44; were observed &#40;<a class="elsevierStyleCrossRef" href="#fig0025">Fig&#46; 5</a>A and B&#41;&#46; Tufts of conidiophores forming elongated synnemata of the <span class="elsevierStyleItalic">Graphium</span> type conidiation were also present&#46; Considering these morphological characteristics&#44; the isolate was suggested to be a member of the <span class="elsevierStyleItalic">S&#46; apiospermum</span> species complex&#46;</p><elsevierMultimedia ident="fig0020"></elsevierMultimedia><elsevierMultimedia ident="fig0025"></elsevierMultimedia><p id="par0065" class="elsevierStylePara elsevierViewall">The identity of <span class="elsevierStyleItalic">Scedosporium boydii</span> was finally achieved based on the sequencing of the PCR amplicon of the internal transcribed spacer 1 and internal transcribed spacer 2&#46; The sequenced fragments matched 100&#37; to <span class="elsevierStyleItalic">S&#46; boydii</span> &#40;formerly named <span class="elsevierStyleItalic">Pseudallescheria boydii</span><a class="elsevierStyleCrossRef" href="#bib0150"><span class="elsevierStyleSup">11</span></a>&#41;&#46; The accession numbers are <a id="intr0005" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=JQ690940">JQ690940</a>&#44; <a id="intr0010" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888398">AJ888398</a>&#44; <a id="intr0015" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888383">AJ888383</a>&#44; <a id="intr0020" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888394">AJ888394</a>&#44; and <a id="intr0025" class="elsevierStyleInterRef" href="http://www.ncbi.nlm.nih.gov/entrez/query.fcgi?cmd=search&db=nucleotide&doptcmdl=genbank&term=AJ888398">AJ888398</a>&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0060">Discussion</span><p id="par0070" class="elsevierStylePara elsevierViewall">Endogenous endophthalmitis caused by the <span class="elsevierStyleItalic">S&#46; apiospermum</span> species complex is not common and patients have typically a poor visual prognosis with a low likelihood of organ preservation&#46; This disease has been diagnosed in patients with underlying systemic neutropenia&#44; acute lymphocytic leukemia&#44; Wegener&#39;s granulomatosis&#44;<a class="elsevierStyleCrossRef" href="#bib0135"><span class="elsevierStyleSup">8</span></a> organ transplant patients&#44; and immunocompetent patients with pulmonary fibrosis&#46;<a class="elsevierStyleCrossRef" href="#bib0180"><span class="elsevierStyleSup">17</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">In the case described here&#44; we think that uncontrolled diabetes mellitus was the main risk factor for the development of endophthalmitis&#44; probably of endogenous origin&#46; After a right femur surgery nine months prior to the ocular complaint&#44; an unidentified infectious microorganism caused a transient blood infection with a septic embolus to the retina&#44; which was the possible cause of the endogenous fungal endophthalmitis&#46; The patient sought late for ophthalmic attention&#44; which resulted in a delayed diagnosis&#46; The fungus was present in the choroid and retina tissue smears&#44; as well as in the cultures&#46; Due to the retinal detachment&#44; decreased ocular pressure&#44; loss of light perception&#44; the pain in the eye&#44; and the risk of dissemination&#44; enucleation was the better option&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">In other cases&#44; when an early diagnosis of this disease has been established&#44; a therapeutic regimen consisting of vitrectomy with intravitreal 0&#46;1&#37; voriconazole and oral voriconazole at regular doses has been indicated with successful outcomes&#46;<a class="elsevierStyleCrossRef" href="#bib0190"><span class="elsevierStyleSup">19</span></a></p><p id="par0085" class="elsevierStylePara elsevierViewall">In the present case&#44; a very late diagnosis was established in a blind and painful eye&#59; therefore&#44; the patient was not treated with voriconazole&#46; Enucleation was necessary for a complete removal of the infected tissues&#44; preventing in this way a potential dissemination&#46; The mycological and molecular studies confirmed <span class="elsevierStyleItalic">S&#46; boydii</span> as the cause of the ocular mycosis&#46;</p><p id="par0090" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">S&#46; apiospermum</span> is a complex of species that reproduce asexually&#44; although sexual reproduction&#44; as in this case&#44; is also possible&#46;<a class="elsevierStyleCrossRef" href="#bib0110"><span class="elsevierStyleSup">3</span></a> This holomorphic complex is genotypically diverse&#44; and the techniques for identification and epidemiological studies are routinely used on pulmonary secretions obtained from cystic fibrosis patients&#46;<a class="elsevierStyleCrossRef" href="#bib0145"><span class="elsevierStyleSup">10</span></a></p><p id="par0095" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">S&#46; boydii</span> is one of the most clinically relevant species in the <span class="elsevierStyleItalic">S&#46; apiospermum</span> complex and is also the most virulent&#46;<a class="elsevierStyleCrossRef" href="#bib0125"><span class="elsevierStyleSup">6</span></a> Conventional phenotypic methods are of limited use for species differentiation within this complex&#44; and precise identification requires molecular approaches&#46;<a class="elsevierStyleCrossRefs" href="#bib0115"><span class="elsevierStyleSup">4&#44;5</span></a> In the present case&#44; the initial morphological identification was highly suggestive of a species of the <span class="elsevierStyleItalic">S&#46; apiospermum</span> complex&#44; but the molecular approaches were necessary to confirm the identity of the species&#46; Endophthalmitis caused by species of the <span class="elsevierStyleItalic">S&#46; apiospermum</span> complex is a devastating disease with a poor response to antifungal therapy and a poor visual outcome&#46; To our knowledge&#44; this is the first report of a <span class="elsevierStyleItalic">S&#46; boydii</span> endophthalmitis in Mexico&#44; which was confirmed by mycological molecular analyses&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0065">Conflicts of interest</span><p id="par0100" class="elsevierStylePara elsevierViewall">Authors declare that there are no conflicts of interest&#46;</p></span></span>"
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          "titulo" => "Conflicts of interest"
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        7 => array:2 [
          "identificador" => "xack229159"
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          "titulo" => "References"
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    "pdfFichero" => "main.pdf"
    "tienePdf" => true
    "fechaRecibido" => "2014-10-05"
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          "clase" => "keyword"
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            0 => "<span class="elsevierStyleItalic">Scedosporium apiospermum</span> species complex"
            1 => "Fungal endophthalmitis"
            2 => "Mycosis"
            3 => "Enucleation"
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            0 => "Complejo de especies de <span class="elsevierStyleItalic">Scedosporium apiospermum</span>"
            1 => "Endoftalmitis f&#250;ngica"
            2 => "Micosis"
            3 => "Enucleaci&#243;n"
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    "resumen" => array:2 [
      "en" => array:3 [
        "titulo" => "Abstract"
        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Background</span><p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Mycotic ocular infections caused by the <span class="elsevierStyleItalic">Scedosporium apiospermum</span> species complex are challenging to treat because of the delayed diagnoses and poor responses to antifungal drugs and surgical treatment&#46;</p></span> <span id="abst0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Case report</span><p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">A case of a 69-year-old male patient with a history of diabetes mellitus type 2 and prior surgery on the right femur is described&#46; In the 10 days prior to the ophthalmic consultation he started with ocular pain&#44; adding to a previous and progressive loss of visual acuity in his right eye&#46; The diagnosis of endophthalmitis of probable endogenous origin was established&#46; Despite medical treatment&#44; the patient&#39;s condition worsened and&#44; due to the imminent risks&#44; an enucleation was performed&#46; Smears of the enucleation tissue revealed fungal cells&#44; and the cultures yielded a fungus belonging to the <span class="elsevierStyleItalic">S&#46; apiospermum</span> species complex&#44; which was identified as <span class="elsevierStyleItalic">Scedosporium boydii</span> by morphological characteristics and sequencing of a PCR amplicon&#46;</p></span> <span id="abst0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Conclusions</span><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A diagnosis of endophthalmitis of probable endogenous origin in the right eye was based on a previous right femur surgery&#46; Potential risk to the patient led to enucleation&#46;</p></span>"
        "secciones" => array:3 [
          0 => array:2 [
            "identificador" => "abst0005"
            "titulo" => "Background"
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          1 => array:2 [
            "identificador" => "abst0010"
            "titulo" => "Case report"
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          2 => array:2 [
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            "titulo" => "Conclusions"
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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">Las infecciones mic&#243;ticas oculares causadas por el complejo de especies de <span class="elsevierStyleItalic">Scedosporium apiospermum</span> son un reto en el tratamiento por su diagn&#243;stico tard&#237;o y la pobre respuesta a los antimic&#243;ticos y al tratamiento quir&#250;rgico&#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 describe el caso de un paciente de sexo masculino de 69 a&#241;os con antecedentes de diabetes mellitus de tipo 2 y cirug&#237;a previa del f&#233;mur derecho&#46; Diez d&#237;as antes de la consulta oftalmol&#243;gica comenz&#243; con dolor ocular que se sumaba a una p&#233;rdida previa y progresiva de la capacidad visual en el ojo derecho&#46; Se estableci&#243; el diagn&#243;stico de endoftalmitis de posible origen end&#243;geno&#46; A pesar del tratamiento&#44; el paciente no present&#243; mejor&#237;a y por los inminentes riesgos se decidi&#243; llevar a cabo una enucleaci&#243;n&#46; El frotis a partir del tejido enucleado mostr&#243; c&#233;lulas f&#250;ngicas y los cultivos revelaron un hongo perteneciente al complejo <span class="elsevierStyleItalic">S&#46; apiospermum</span>&#44; identificado como <span class="elsevierStyleItalic">Scedosporium boydii</span> por procedimientos morfol&#243;gicos y por secuenciaci&#243;n de un amplic&#243;n de PCR&#46;</p></span> <span id="abst0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conclusiones</span><p id="spar0030" class="elsevierStyleSimplePara elsevierViewall">El diagn&#243;stico de endoftalmitis de probable origen end&#243;geno en el ojo derecho se bas&#243; en el antecedente de una cirug&#237;a previa de f&#233;mur&#44; cuyos riesgos para el paciente condujeron a una enucleaci&#243;n&#46;</p></span>"
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          "en" => "<p id="spar0035" class="elsevierStyleSimplePara elsevierViewall">A right eye ultrasound image showing a total retinal detachment contacting the posterior lens capsule&#46;</p>"
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          "en" => "<p id="spar0040" class="elsevierStyleSimplePara elsevierViewall">PAS stained section of the enucleated ocular tissue&#46; Irregular&#44; thin&#44; septate hyphae and round fungal structures &#40;arrows&#41; with a background of necrotic detritus and neutrophils were observed &#40;40&#215;&#41;&#46;</p>"
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          "en" => "<p id="spar0055" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41; Cleistothecium observed after three weeks of incubation on Sabouraud&#8211;Emmons agar at room temperature &#40;40&#215;&#41;&#46; &#40;B&#41; Oblong brown ascospores released after rupture of the cleistothecial wall &#40;100&#215;&#41;&#46;</p>"
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      "titulo" => "References"
      "seccion" => array:1 [
        0 => array:2 [
          "identificador" => "bibs0005"
          "bibliografiaReferencia" => array:19 [
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                          "etal" => true
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Article information

ISSN: 11301406
Original language: English

DOI:10.1016/j.riam.2015.07.006

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2017 March	40	29	69
2017 February	125	1	126
2017 January	36	7	43
2016 December	47	6	53
2016 November	49	7	56
2016 October	36	16	52
2016 September	49	5	54
2016 August	35	5	40
2016 July	43	7	50
2016 June	1	0	1

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