A case report of pulmonary tumor thrombotic microangiopathy (PTTM) caused by esophageal squamous cell carcinoma

Fujishiro, Takeshi; Shuto, Kiyohiko; Shiratori, Toru; Kono, Tuguaki; Akutsu, Yasunori; Uesato, Masaya; Hoshino, Isamu; Murakami, Kentaro; Imanishi, Shunsuke; Tochigi, Toru; Yonemori, Yoko; Matsubara, Hisahiro

doi:10.1007/s10388-013-0382-8

Cited by 25 publications

(29 citation statements)

References 22 publications

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“…Fujishiro et al reported 103 patients with PTTM, among whom the most frequent primary cancer was gastric (58 cases), followed by lung (10 cases), breast (7 cases), cancer of unknown primary (f5 cases), ovarian (5 cases) and bladder (4 cases). 2 As shown in table 1, imatinib was effective in three cases of signet-ring cell carcinoma, including our case, but was weaker in cases of colon (patient 2) and breast (patients 4 and 5) cancer. The mechanism is still unclear, but it has been suggested that production of growth factors, including PDGF, might be different depending on the tumour characteristics.…”

Section: Discussionsupporting

confidence: 54%

“…1 In fact, it has been reported that only 7 of 103 patients were pathologically diagnosed with PTTM while still alive. 2 Platelet-derived growth factor (PDGF) stimulation has been reported to be involved in the proliferation and anti-apoptosis of pulmonary artery endothelial cells and smooth muscle cells in patients with pulmonary hypertension (PH). [3][4][5] PDGF is also involved in the progression of PTTM.…”

Section: Introductionmentioning

confidence: 99%

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Imatinib dramatically alleviates pulmonary tumour thrombotic microangiopathy induced by gastric cancer

Kubota¹,

Shinozaki

Imai

et al. 2017

BMJ Case Reports

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Pulmonary tumour thrombotic microangiopathy (PTTM) is a rare complication of cancer, which can be lethal due to progressive pulmonary hypertension (PH). Several case reports have demonstrated that imatinib, a platelet-derived growth factor receptor–tyrosine kinase inhibitor, can improve severe PH in patients with PTTM.We describe the case of a 56-year-old woman. Her mean pulmonary arterial pressure (mPAP) was 47 mm Hg, and her dyspnoea worsened rapidly over several days. Although pulmonary embolism was not observed on CT, enlargement of the para-aortic lymph nodes was detected. Gastro-oesophageal endoscopy revealed signet-ring cell carcinoma. We diagnosed her as having PTTM based on her clinical course, and started treatment with imatinib. Five days after its administration, her mPAP decreased dramatically. She was discharged and lived without symptoms of PH until her death due to systemic metastasis of carcinoma. In some cases of PTTM, imatinib may be an effective therapeutic option for PH.

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Section: Discussionsupporting

confidence: 54%

Section: Introductionmentioning

confidence: 99%

Imatinib dramatically alleviates pulmonary tumour thrombotic microangiopathy induced by gastric cancer

Kubota¹,

Shinozaki

Imai

et al. 2017

BMJ Case Reports

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“…A study has estimated the median survival as 5 days from admission to hospital 6. PTTM may be identified using video‐assisted thoracoscopic surgery, trans‐bronchial biopsy, right heart catheterization, and CT‐guided biopsy 7…”

Section: Discussionmentioning

confidence: 99%

Subacute right heart failure revealing three simultaneous causes of post‐embolic pulmonary hypertension in metastatic dissemination of breast cancer

et al. 2016

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A 72‐year‐old woman with history of breast cancer only treated surgically was referred to our department for pulmonary hypertension (PH) suspicion. Echocardiogram revealed elevated right ventricular systolic pressure. Computed tomography (CT) angiogram showed no pulmonary embolism (PE), but lung scan revealed two ventilation‐perfusion mismatch areas. Right cardiac catheterization established precapillary PH. Despite treatment with PH specific therapy (sildenafil, ambrisentan, and epoprostenol), her condition worsened rapidly with acute right heart failure (RHF). She died 22 days after admission. Post‐mortem microscopic examination showed a rare combination of PH etiologies consistent with metastasis of breast cancer in pulmonary vasculature including the rare pulmonary tumour thrombotic microangiopathy (PTTM).

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“…Only a few cases exist in the literature of patients with PTTM diagnosed prior to death (8)(9)(10)(11)(12). Miyano et al reported a patient with previously resected gastric cancer in whom PTTM was suggested by elevated serum VEGF and D-dimer levels (8).…”

Section: Discussionmentioning

confidence: 99%

Fatal Pulmonary Tumor Thrombotic Microangiopathy Caused by Undiagnosed Metastatic Gastric Adenocarcinoma

Endicott-Yazdani

Ghazi

Armstrong

et al. 2015

Baylor University Medical Center Proceedings

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Pulmonary tumor thrombotic microangiopathy (PTTM) is a rare cause of rapidly progressive dyspnea in a background of metastatic cancer. Gastric adenocarcinoma is the neoplasm most frequently associated with PTTM. Unfortunately, PTTM is difficult to identify clinically and is most commonly a postmortem diagnosis. We present the case of a woman with no previous diagnosis of cancer who presented with rapidly progressive shortness of breath. She was diagnosed with severe pulmonary arterial hypertension and rapidly succumbed to her illness. A postmortem diagnosis of PTTM was established based on autopsy results. P ulmonary tumor thrombotic microangiopathy (PTTM) is a rare and usually fatal cause of rapidly progressive dyspnea, caused by malignancy (1, 2). It was fi rst described by von Herbay et al, who identifi ed fi brocellular proliferation of the intima of small pulmonary arterial vessels in patients with metastatic carcinoma (2). Th is report describes a patient who presented with rapidly progressive dyspnea and pulmonary hypertension and was subsequently found at autopsy to have PTTM from an undiagnosed metastatic gastric adenocarcinoma. CASE REPORTA 52-year-old woman was transferred from an outside hospital with a 1.5-month history of minimally productive cough and a 2-week history of progressive dyspnea. She complained of nausea, vomiting, and vague abdominal pain. Her medical history was signifi cant for untreated diabetes and hypertension. Her only medications were famotidine and esomeprazole as needed for gastroesophageal refl ux. Review of systems was positive for night sweats, fatigue, a 10-lb weight loss over 6 months, anorexia, and paroxysmal nocturnal dyspnea. Her temperature was 95.3°F; pulse, 80 beats per minute; blood pressure, 151/94 mm Hg; respiratory rate, 20 breaths per minute; and oxygen saturation, 99% on 15 L/minute with a nonrebreather mask. Physical exam revealed an obese woman in moderate respiratory distress, with jugular venous distention, a prominent P2 heart sound, few bilateral basilar crackles with deep inspiration, and mild epigastric tenderness but no palpable masses or ascites.Abnormal laboratory results included a hemoglobin level of 10.2 g/dL; platelets, 96 K/μL; albumin, 2.9 g/dL; and alkaline phosphatase, 189 U/L. Arterial blood gas analysis demonstrated a pH of 7.40; the patient's partial pressure of carbon dioxide was 41 mm Hg, partial pressure of oxygen in arterial blood, 141 mm Hg, and bicarbonate, 25.1 mmol/L. A transthoracic echocardiogram revealed a dilated right atrium and right ventricle and moderate to severe pulmonary hypertension with an estimated right ventricular systolic pressure of 66 mm Hg and a D-shaped septum. A ventilation-perfusion scan showed a very low probability of a pulmonary embolism, and lower extremity venous Doppler studies were negative. A right heart catheterization demonstrated moderate pulmonary hypertension nonresponsive to inhaled nitric oxide, with a pulmonary artery pressure of 70/30 mm Hg, mean 37 mm Hg, a pulmonary capillary wedge pressu...

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A case report of pulmonary tumor thrombotic microangiopathy (PTTM) caused by esophageal squamous cell carcinoma

Cited by 25 publications

References 22 publications

Imatinib dramatically alleviates pulmonary tumour thrombotic microangiopathy induced by gastric cancer

Imatinib dramatically alleviates pulmonary tumour thrombotic microangiopathy induced by gastric cancer

Subacute right heart failure revealing three simultaneous causes of post‐embolic pulmonary hypertension in metastatic dissemination of breast cancer

Fatal Pulmonary Tumor Thrombotic Microangiopathy Caused by Undiagnosed Metastatic Gastric Adenocarcinoma

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