Hypereosinophilia with systemic thrombophlebitis Hiroyuki Kanno MD a, * , Naohisa Ouchi MD a , Masatoshi Sato MD c , Tsukasa Wada MD b , Takashi Sawai MD a a Department of Pathology, Iwate Medical University School of Medicine, Morioka 020-8505, Japan b Department of Neurosurgery, Iwate Medical University School of Medicine, Morioka 020-8505, Japan c Department of Orthopedic Surgery, Hanamaki General Hospital, Hanamaki 025-0075, Japan Received 22 September 2004; accepted 18 March 2005 Summary A 34-year-old Japanese woman developed subcutaneous induration in the left thigh, then showed extreme eosinophilia, and died of hemorrhagic infarction of the brain. Autopsy revealed endocarditis with eosinophil infiltration and systemic thrombophlebitis, including pulmonary veins and intrahepatic branches of the portal vein. Arterial structure was relatively preserved. She had no clinical history of asthma and had anti–ascarid IgE antibody at postmortem serological examination; thus, her disease does not fulfill the diagnostic criteria of Churg-Strauss syndrome and idiopathic hyper- eosinophilic syndrome (HES). Her organ involvement is, however, consistent with that of HES; thus, her pathophysiological conditions would resemble those of HES. Systemic thrombophlebitis without arterial lesion in patients with hypereosinophilia has never been reported, and this case would broaden the spectrum of vascular lesions in these patients. D 2005 Elsevier Inc. All rights reserved. 1. Introduction Peripheral blood eosinophilia can be associated with a variety of diseases, ranging from parasitic infection to bronchial asthma, or etiology may be unknown. Eosino- philic leukocytes promote the allergic inflammation through the release of various mediators, and the heart and blood vessels are the major target of tissue injury by eosinophils [1]. In the disease-developing process, eosinophil cationic granule proteins such as major basic protein (MBP) are potent stimuli of platelet activation and aggregation [2] and are able to alter the clotting process by interfering with endothelial cell surface thrombomodulin [3]. Furthermore, eosinophils release reactive oxygen species and directly injure the endothelial cells [4]. Two disease entities with eosinophilia causing the injury of circulatory system, Churg-Strauss syndrome (CSS) [5] and idiopathic hypereosinophilic syndrome (HES) [1,6], have been established. In CSS, granulomatous angitis with eosinophilia, the broad range of systemic vessels, from small arteries to veins, is affected. The HES presents a heterogeneous group of disorders with organ system dysfunction. The most serious lesion in HES is the heart injury such as endocarditis and myocarditis, and subsequent thromboembolism leads to multiple organ dysfunctions [1]. Here, we report a case of systemic thrombophlebitis including pulmonary and liver vessels with peripheral blood eosinophilia, which fulfill the diagnostic criteria of neither CSS nor HES. 0046-8177/$ – see front matter D 2005 Elsevier Inc. All rights reserved. doi:10.1016/j.humpath.2005.03.017 T Corresponding author. E-mail address: hirokan@iwate-med.ac.jp (H. Kanno). Keywords: Hypereosinophilia; Vasculitis; Phlebitis; Endocarditis Human Pathology (2005) 36, 585 – 589 www.elsevier.com/locate/humpath