Pulmonary thrombotic microangiopathy caused by neoplastic urothelial carcinoma cells

Matute Guerrero, Almudena; Abanses Moreno, Paula; Villasmil Chaparro, Isabel

doi:10.1016/j.medcle.2022.10.013

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B) CKAE1-AE3 immunohistochemical technique: microscopic image of the lung parenchyma, cross section of an artery, reveals a group of positive tumour cells in its lumen (red arrow). The colour of the image is only visible in the electronic version." ] ] ] "textoCompleto" => "Tumour thrombotic microangiopathy is a rare clinicopathological entity related to malignant neoplasms, difficult to diagnose ante mortem. It is characterised by pulmonary complications with rapid progression of dyspnoea and pulmonary hypertension leading to a poor prognosis.<a class="elsevierStyleCrossRef" href="#bib0005">1</a> We report the case of a pulmonary tumour thrombotic microangiopathy (PTTM) due to urothelial cancer.This is a 57-year-old man with no medical history who came to the emergency department for chest pain and dyspnoea. The electrocardiogram showed an S1Q3T3 pattern and the blood test showed elevated d-dimer levels (14,000ng feu/mL). Computed tomography was negative for pulmonary embolism (PE), and echocardiography showed signs of right ventricular overload. Given a high suspicion of PE, anticoagulation treatment was started. Doppler imaging of the lower limbs ruled out thrombosis in the venous territory and the study was completed with a ventilation-perfusion scintigraphy that showed repletion defects in subsegmental vessels, suggestive but not conclusive of PE. After 72h, thrombocytopenia (13,000mm3), anaemia (Hb 10.9g/dl), elevated total bilirubin (3.34mg/dl) and LDH (719U/l) were evident, and low haptoglobin (<2.56mg/dl, reference values 32−197mg/dl), so anticoagulation was discontinued due to risk of haemorrhage. Given the suspicion of thrombotic microangiopathy, a peripheral blood smear was performed which showed schistocytes, concordant with the clinical suspicion of thrombotic thrombocytopenic purpura, ADAMST13 was requested and the patient was admitted to the intensive care unit to start plasmapheresis and respiratory support. Eight hours after admission to the intensive care unit, emergency intubation was performed due to increased work of breathing, with subsequent cardiorespiratory arrest and unsuccessful cardiopulmonary resuscitation manoeuvres, resulting in death.An autopsy was requested which revealed microvascular pulmonary tumour embolism with images of tumour thrombi in medium calibre arteries of both lungs, metastasis of micropapillary carcinoma with immunophenotype consistent with urothelial origin (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>) and dilatation of right cavities secondary to underlying disease (metastatic tumour PE).<elsevierMultimedia ident="fig0005"></elsevierMultimedia>PTTM is a rare clinicopathological entity characterised by microscopic tumour embolization and fibrocellular proliferation of the intima of small arterioles, first described in 1990 by von Hervay et al. The most common origin is stomach cancer, followed by lung, breast, pancreatic and oesophageal cancer.<a class="elsevierStyleCrossRef" href="#bib0010">2</a>The most common symptoms are progressive dyspnoea, cough, and haemoptysis, attributed to pulmonary arterial hypertension and right heart failure, leading to a misdiagnosis of idiopathic pulmonary arterial hypertension.Pathological findings in the biopsy are essential for diagnostic confirmation. The radiological diagnosis has not been established because the findings are usually minimal or non-specific.<a class="elsevierStyleCrossRef" href="#bib0015">3</a>This case shows the complexity of PTTM diagnosis and its aggressive course once symptoms appear. Ante mortem diagnosis and treatment is essential before the patient develops pulmonary hypertension, so it is important to suspect microangiopathy, pulmonary hypertension, absence of macroscopic pulmonary emboli and sub-segmental defects on perfusion scintigraphy.<a class="elsevierStyleCrossRef" href="#bib0020">4</a>" "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:1 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Fig. 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1063 "Ancho" => 2007 "Tamanyo" => 456398 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "(A) H&E technique: microscopic image of lung parenchyma showing the alveoli and, on the right, a longitudinal section of an artery with the presence of a cluster of tumour cells in its lumen (red arrow). B) CKAE1-AE3 immunohistochemical technique: microscopic image of the lung parenchyma, cross section of an artery, reveals a group of positive tumour cells in its lumen (red arrow). The colour of the image is only visible in the electronic version." ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary tumor thrombotic microangiopathy: a systematic review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "R.H. Godbole" 1 => "R. Saggar" 2 => "N. Kamangar" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1177/2045894019851000" "Revista" => array:5 [ "tituloSerie" => "Pulm Circ." "fecha" => "2019" "volumen" => "9" "paginaInicial" => "1" "paginaFinal" => "13" ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0010" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary tumor thrombotic microangiopathy with pulmonary hypertension" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "A. Von Herbay" 1 => "A. Illes" 2 => "R. Waldherr" 3 => "H.F. Otto" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1002/1097-0142(19900801)66:3<587::aid-cncr2820660330>3.0.co;2-j" "Revista" => array:6 [ "tituloSerie" => "Cancer." "fecha" => "1990" "volumen" => "66" "paginaInicial" => "587" "paginaFinal" => "592" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/2163747" "web" => "Medline" ] ] ] ] ] ] ] ] 2 => array:3 [ "identificador" => "bib0015" "etiqueta" => "3" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary tumor thrombotic microangiopathy caused by prostate carcinoma" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "D. Katayama" 1 => "K. Kuriyama" 2 => "T. Kinoshita" 3 => "K. Nagai" 4 => "H. Hongyo" 5 => "K. Kishimoto" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.1177/2058460116662300" "Revista" => array:3 [ "tituloSerie" => "Acta Radiol Open." "fecha" => "2016" "volumen" => "5" ] ] ] ] ] ] 3 => array:3 [ "identificador" => "bib0020" "etiqueta" => "4" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Pulmonary tumor thrombotic microangiopathy: case report and literature review" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:1 [ 0 => "C.K. Chen" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.6705/j.jacme.201809_8(3).0006" "Revista" => array:6 [ "tituloSerie" => "J Acute Med." "fecha" => "2018" "volumen" => "8" "paginaInicial" => "127" "paginaFinal" => "131" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/32995214" "web" => "Medline" ] ] ] ] ] ] ] ] ] ] ] ] ] "idiomaDefecto" => "en" "url" => "/23870206/0000016000000005/v1_202303052139/S2387020623000554/v1_202303052139/en/main.assets" "Apartado" => array:4 [ "identificador" => "43309" "tipo" => "SECCION" "en" => array:2 [ "titulo" => "Letters to the Editor" "idiomaDefecto" => true ] "idiomaDefecto" => "en" ] "PDF" => "https://static.elsevier.es/multimedia/23870206/0000016000000005/v1_202303052139/S2387020623000554/v1_202303052139/en/main.pdf?idApp=UINPBA00004N&text.app=https://www.elsevier.es/" "EPUB" => "https://multimedia.elsevier.es/PublicationsMultimediaV1/item/epub/S2387020623000554?idApp=UINPBA00004N"]

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

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2.6

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3.1

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0.371

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0.511

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Vol. 160. Issue 5.