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Purtscher-like retinopathy in a paediatric patient with haemolytic uraemic syndrome: A case report and literature review
Retinopatía Purtscher-like en paciente pediátrico con síndrome urémico hemolítico: reporte de caso y revisión de la literatura
F. Benvenuto
, S. Guillen, L. Marchiscio, J. Falbo, A. Fandiño
Corresponding author
Ophthalmology Service - SAMIC Pediatric, Hospital «Prof. Dr. Juan P. Garrahan», Buenos Aires, Argentina
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Benvenuto, S. Guillen, L. Marchiscio, J. Falbo, A. Fandiño" "autores" => array:5 [ 0 => array:4 [ "nombre" => "F." "apellidos" => "Benvenuto" "email" => array:1 [ 0 => "dr.benvenutofranco@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "S." "apellidos" => "Guillen" ] 2 => array:2 [ "nombre" => "L." "apellidos" => "Marchiscio" ] 3 => array:2 [ "nombre" => "J." "apellidos" => "Falbo" ] 4 => array:2 [ "nombre" => "A." "apellidos" => "Fandiño" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Ophthalmology Service - SAMIC Pediatric, Hospital «Prof. Dr. Juan P. Garrahan», Buenos Aires, Argentina" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "<span class="elsevierStyleItalic">Corresponding author</span>." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Retinopatía <span class="elsevierStyleItalic">Purtscher-like</span> en paciente pediátrico con síndrome urémico hemolítico: reporte de caso y revisión de la literatura" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 702 "Ancho" => 905 "Tamanyo" => 60982 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Presence of pericardial fluid (yellow arrow).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall">Haemolytic uraemic syndrome (HUS) is one of the leading causes of acute renal failure in children. It occurs most frequently between the first and tenth year of life, with a frequency of 1–3 cases per 100,000 children.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> There is a classic triad defined by the presence of haemolytic anaemia, thrombocytopenia and acute renal failure. It can be grouped into 2 forms, typical or atypical, depending on whether or not it is associated with diarrhoea due to infection with verotoxin-producing <span class="elsevierStyleItalic">Escherichia coli</span> (0157:H7). The pathophysiology consists of pro-inflammatory and pro-thrombotic changes with altered coagulation associated with endothelial damage.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Purtscher retinopathy is characterised by visual loss and retinal lesions such as cotton-wool exudates, intraretinal haemorrhages, retinal pallor that occur after chest or head trauma. When the aetiology is not trauma, it is called <span class="elsevierStyleItalic">Purtscher-like</span> and can be seen in pancreatitis, thrombotic microangiopathy such as HUS and autoimmune diseases.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,5</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Ocular involvement in pediatric patients with a diagnosis of HUS has been reported in few cases, and is even rarer in a condition that did not require dialysis treatment.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical case</span><p id="par0020" class="elsevierStylePara elsevierViewall">Patient aged 8 years, previously healthy with no relevant pathological history and complete vaccination, who presented with vomiting and diarrhoea of 72 h of evolution, with bloody stools and fever of 38  °C in the last 48 h.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The patient had pale skin and mucous membranes, unsteady gait and tremor in the limbs, and was hypotensive with a blood pressure of 64/47 mmHg. Admission was decided, initiating treatment with physiological saline solution 40 ml/kg and ceftriaxone 1500 mg. Blood cultures, complete blood count and urine were taken. The presence of blurred vision led to interconsultation with the Ophthalmology Department.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Initial ophthalmological examination produced visual acuity (VA) of 4/10 in both eyes (BE), anterior segment without abnormalities while fundus examination showed vascular tortuosity, superficial intraretinal haemorrhages in flame, peripapillary retinal whitening and cotton-wool exudates with peripapillary predominance (Purtscher "flecken") in BE (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0035" class="elsevierStylePara elsevierViewall">After the first 24 h of hospitalisation the following parameters were recorded: temperature 37.2 °C, blood pressure 110/72 mmHg and diuresis for 24 h was 100 ml. In the control laboratory the values were: haematocrit 31%, haemoglobin 11.6 g/dl, leucocytes 28,730 ml, platelets 21,000 ml, lactate dehydrogenase 3784 U/l, blood creatine 2.4 mg/dl, uric acid 11 mg/dl and urea 238 mg/dl. Proteinuria, hematuria and peripheral blood schistocytes were also observed.</p><p id="par0040" class="elsevierStylePara elsevierViewall">Verotoxin analysis of fecal material was positive. Together with hemolysis and acute renal failure, a diagnosis of HUS was made.</p><p id="par0045" class="elsevierStylePara elsevierViewall">The patient remained admitted for water and electrolyte supplementation and treatment with amlodipine 2.5 mg/every 24 h, erythropoietin 2000 UI/24 h, bisacodyl 5 mg/24 h, omeprazole 20 mg/24 h and folic acid 1 mg/24 h. During hospitalisation, control ultrasound scans were performed, showing kidneys of normal size although the presence of pericardial effusion was found, which evolved favourably with treatment (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0050" class="elsevierStylePara elsevierViewall">The patient was discharged after 2 weeks of hospitalisation, without requiring dialysis treatment. Due to the diagnosis of HUS and the ocular fundus findings, with no history of trauma, the picture was interpreted as <span class="elsevierStyleItalic">Purtscher-like</span> retinopathy. One month after the first check-up, a new check-up was performed, showing a VA of 10/10 in BE, absence of peripapillary whitening and an almost complete decrease in retinal haemorrhages, vascular tortuosity and soft Purtscher "flecken" exudates (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>).</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Discussion</span><p id="par0055" class="elsevierStylePara elsevierViewall">Purtscher first published haemorrhages and retinal whitening in 2 patients with a history of severe head trauma.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The same findings were later described in patients with severe chest trauma, long bone fractures or no history of trauma but with a diagnosis of different entities such as pancreatitis and collagen diseases. It has been postulated that these characteristic findings may be secondary to thrombotic microangiopathy with endothelial damage and increased platelet consumption.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Purtscher-like</span> retinopathy is a rare entity, occurring in 0.24 patients per 1,000,000 per year.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> The pathophysiology of this retinopathy is associated with endothelial damage with occlusion in the pre-capillary arteries by leukocytes, platelets, fibrin and complement aggregates. This would be the cause of the findings usually observed as Purtscher "flecken" cotton-wool exudates, retinal haemorrhages, macular edema and cherry red pseudo-stain.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">The present patient was diagnosed at the clinical and laboratory level with HUS, and reported visual disturbances from the first hospital visit. Due to the rapid diagnosis and prompt treatment of HUS, the patient did not require dialysis. Fundus findings were consistent with <span class="elsevierStyleItalic">Purtscher-like</span> retinopathy. The pathogenesis of HUS that explains this is the result of thrombotic microangiopathy with endothelial damage probably induced by the exotoxin.<a class="elsevierStyleCrossRefs" href="#bib0040"><span class="elsevierStyleSup">8,9</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Purtscher-like</span> retinopathy is a rare ophthalmological condition associated with severe multi-systemic diseases. We have found no reports in the literature of this retinopathy due to HUS in patients who have not required dialysis treatment. Fundus examination in patients with this disease is of great help in the diagnosis of this retinopathy.</p></span><span id="sec1015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect1041">Conflict of interests</span><p id="par1061" class="elsevierStylePara elsevierViewall">The authors declare no conflict of interest.</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Funding</span><p id="par0075" class="elsevierStylePara elsevierViewall">The authors declare that they have not received any funding for this work.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:10 [ 0 => array:3 [ "identificador" => "xres1600808" "titulo" => "Abstract" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0005" ] ] ] 1 => array:2 [ "identificador" => "xpalclavsec1434530" "titulo" => "Keywords" ] 2 => array:3 [ "identificador" => "xres1600807" "titulo" => "Resumen" "secciones" => array:1 [ 0 => array:1 [ "identificador" => "abst0010" ] ] ] 3 => array:2 [ "identificador" => "xpalclavsec1434529" "titulo" => "Palabras clave" ] 4 => array:2 [ "identificador" => "sec0005" "titulo" => "Introduction" ] 5 => array:2 [ "identificador" => "sec0010" "titulo" => "Clinical case" ] 6 => array:2 [ "identificador" => "sec0015" "titulo" => "Discussion" ] 7 => array:2 [ "identificador" => "sec1015" "titulo" => "Conflict of interests" ] 8 => array:2 [ "identificador" => "sec0020" "titulo" => "Funding" ] 9 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-08-25" "fechaAceptado" => "2020-10-14" "PalabrasClave" => array:2 [ "en" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Keywords" "identificador" => "xpalclavsec1434530" "palabras" => array:4 [ 0 => "Haemolytic uraemic syndrome" 1 => "Paediatric retina" 2 => "Purtscher retinopathy" 3 => "Purtscher-like retinopathy" ] ] ] "es" => array:1 [ 0 => array:4 [ "clase" => "keyword" "titulo" => "Palabras clave" "identificador" => "xpalclavsec1434529" "palabras" => array:4 [ 0 => "Síndrome urémico hemolítico" 1 => "Retina pediátrica" 2 => "Retinopatía de Purtscher" 3 => "Retinopatía <span class="elsevierStyleItalic">Purtscher-like</span>" ] ] ] ] "tieneResumen" => true "resumen" => array:2 [ "en" => array:2 [ "titulo" => "Abstract" "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0020" class="elsevierStyleSimplePara elsevierViewall">An 8-year-old boy presented with fever, vomits, bloody diarrhoea, and blurred vision. The patient was diagnosed with Haemolytic Uraemic Syndrome (HUS) due to the symptoms and a positive Verotoxin stool test. Funduscopic examination showed retinal involvement in both eyes, peri-papillary paleness, retinal haemorrhages, and soft "Purtscher Fleckens” exudates. A favourable outcome was achieved after hospital admission and systemic treatment. Dialysis treatment was not needed due the preserved diuresis. Although Purtscher-like retinopathy is very uncommon, ocular examination is mandatory in patients with pancreatitis, autoimmune diseases, and thrombotic microangiopathies, such as HUS.</p></span>" ] "es" => array:2 [ "titulo" => "Resumen" "resumen" => "<span id="abst0010" class="elsevierStyleSection elsevierViewall"><p id="spar0025" class="elsevierStyleSimplePara elsevierViewall">Se presenta el caso clínico de un paciente de 8 años de edad con un cuadro febril, vómitos, diarrea con sangre y visión borrosa. El paciente fue diagnosticado de Síndrome Urémico Hemolítico (SHU) por clínica y detección de Verotoxina en materia fecal. La exploración fundoscópica mostró compromiso retinal en ambos ojos, palidez peripapilar, hemorragias retinianas y exudados blandos en forma de Purtscher Flecken. La evolución fue favorable tras la internación y tratamiento sistémico, no requiriendo diálisis por diuresis conservada. La exploración oftalmológica de estos pacientes es fundamental para el estudio del paciente, ya que, a pesar de ser infrecuente, la retinopatía <span class="elsevierStyleItalic">Purtscher-like</span> puede verse en cuadros de pancreatitis, enfermedades autoinmunes y microangiopatías trombóticas como el SHU.</p></span>" ] ] "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Benvenuto F, Guillen S, Marchiscio L, Falbo J, Fandiño A. Retinopatía <span class="elsevierStyleItalic">Purtscher-like</span> en paciente pediátrico con síndrome urémico hemolítico: reporte de caso y revisión de la literatura. Arch Soc Esp Oftalmol. 2021;96:607–610.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1436 "Ancho" => 905 "Tamanyo" => 171106 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Fundus of right (A) and left (B) eyes showing intraretinal haemorrhages, vascular tortuosity, retinal pallor and mainly soft exudates with Purtscher <span class="elsevierStyleItalic">flecken</span> appearance.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Fig. 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 702 "Ancho" => 905 "Tamanyo" => 60982 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Presence of pericardial fluid (yellow arrow).</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1457 "Ancho" => 905 "Tamanyo" => 224196 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Fundus right (A) and left (B) showing the almost complete absence of intraretinal haemorrhages and soft exudates.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:9 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Acute renal failure" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "S.P. 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