A fatal case with pulmonary tumor thrombotic microangiopathy (PTTM) originating from adenoid cystic carcinoma in sublingual gland

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  • Hayashi Kazuki
    Department of Head and Neck Surgery, Kobe City Medical Center General Hospital
  • Shinohara Shogo
    Department of Head and Neck Surgery, Kobe City Medical Center General Hospital
  • Suehiro Atsushi
    Department of Head and Neck Surgery, Kobe City Medical Center General Hospital
  • Kishimoto Ippei
    Department of Head and Neck Surgery, Kobe City Medical Center General Hospital
  • Harada Hiroyuki
    Department of Head and Neck Surgery, Kobe City Medical Center General Hospital
  • Sato Yuki
    Department of Respiratory Medicine, Kobe City Medical Center General Hospital
  • Uehara Keiichiro
    Department of Clinical Pathology, Kobe City Medical Center General Hospital

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  • 肺腫瘍血栓性微小血管症（PTTM）による呼吸不全のため急激に死の転機をたどった舌下腺腺様囊胞癌症例

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Pulmonary tumor thrombotic microangiopathy (PTTM) is characterized by intimal proliferation of micro tumors in pulmonary small arteries. Most cases of this disease reported in the literature show fatal outcomes. It is difficult to diagnose PTTM before pathologic autopsy because a CT scan or chest X-ray cannot give a clear image of blood clots or thrombosis.<br>We report a case with PTTM originating from adenoid cystic carcinoma in the sublingual gland, who died soon after being admitted to hospital.<br>A 53-year-old man was referred to our hospital with a one-month history of submucous oral floor tumor. Salivary duct carcinoma was suspected by fine-needle aspiration cytology (FNAC), and an open biopsy was planned to confirm the pathology. During the operation, the pulse oximeter revealed desaturation at around 84% and he was admitted as an emergency.<br>Contrast CT did not show any airway stenosis, infiltrative shadows, pleural effusion or pulmonary artery thrombosis responsible for the desaturation even though there were several metastatic lesions in the bilateral lung fields. There were no signs of restraint disorder or obstructed disorder on the lung function tests.<br>Cardiac ultrasonography showed no symptoms of heart disease. Blood flow scintigraphy revealed peripheral low perfusion of blood in both his lungs even though ventilation scintigraphy showed enough diffusion of air. Finally, trans-bronchial lung biopsy (TBLB) was performed and a diagnosis of PTTM was established.<br>Anticoagulant therapy and chemotherapy were started on the 19th day after admission, but the patient died of acute respiratory failure on the 25th day.

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