Ab-Rahman Hasliana Azrah, Wong Pooi-Fong, Rahim Hafiz, Abd-Jamil Juraina, Tan Kim-Kee, Sulaiman Syuhaida, Lum Chai-See, Syed-Omar Syarifah-Faridah, AbuBakar Sazaly
Tropical Infectious Diseases Research and Education Centre (TIDREC), Faculty of Medicine, University of Malaya, 50603 Kuala Lumpur, Malaysia ; Department of Medical Microbiology, Faculty of Medicine, University of Malaya, 50603 Kuala Lumpur, Malaysia ; Department of Pharmacology, Faculty of Medicine, University of Malaya, 50603 Kuala Lumpur, Malaysia.
Department of Pharmacology, Faculty of Medicine, University of Malaya, 50603 Kuala Lumpur, Malaysia.
Springerplus. 2015 Nov 2;4:665. doi: 10.1186/s40064-015-1463-z. eCollection 2015.
HPS is a potentially life-threatening histiocytic disorder that has been described in various viral infections including dengue. Its involvement in severe and fatal dengue is probably more common but is presently under recognized.
A 38-year-old female was admitted after 5 days of fever. She was deeply jaundiced, leukopenic and thrombocytopenic. Marked elevation of transaminases, hyperbilirubinemia and hypoalbuminemia were observed. She had deranged INR values and prolonged aPTT accompanied with hypofibrinogenemia. She also had splenomegaly. She was positive for dengue IgM. Five days later she became polyuric and CT brain image showed gross generalized cerebral edema. Her conditions deteriorated by day 9, became confused with GCS of 9/15. Her BMAT showed minimal histiocytes. Her serum ferritin level peaked at 13,670.00 µg/mL and her sCD163 and sCD25 values were markedly elevated at 4750.00 ng/mL and 4191.00 pg/mL, respectively. She succumbed to the disease on day 10 and examination of her tissues showed the presence of dengue virus genome in the bone marrow.
It is described here, a case of fatal dengue with clinical features of HPS. Though BMAT results did not show the presence of macrophage hemophagocytosis, other laboratory features were consistent with HPS especially marked elevation of ferritin, sCD163 and sCD25. Detection of dengue virus in the patient's bone marrow, fifteen days after the onset of fever was also consistent with the suggestion that the HPS is associated with dengue virus infection.
The findings highlight HPS as a possible complication leading to severe dengue and revealed persistent dengue virus infection of the bone marrow. Detection of HPS markers; ferritin, sCD163 and sCD25, therefore, should be considered for early recognition of HPS-associated dengue.
噬血细胞性淋巴组织细胞增生症(HPS)是一种潜在的危及生命的组织细胞疾病,在包括登革热在内的各种病毒感染中均有描述。它在严重和致命的登革热中的参与可能更为常见,但目前尚未得到充分认识。
一名38岁女性在发热5天后入院。她有深度黄疸、白细胞减少和血小板减少。观察到转氨酶显著升高、高胆红素血症和低白蛋白血症。她的国际标准化比值(INR)异常,活化部分凝血活酶时间(aPTT)延长,并伴有纤维蛋白原血症。她还出现脾肿大。她的登革热IgM呈阳性。五天后,她出现多尿,脑部CT图像显示广泛性脑水肿。到第9天,她的病情恶化,意识模糊,格拉斯哥昏迷量表(GCS)评分为9/15。她的骨髓活检组织形态学分析(BMAT)显示仅有少量组织细胞。她的血清铁蛋白水平峰值达到13,670.00µg/mL,可溶性CD163(sCD163)和可溶性CD25(sCD25)值分别显著升高至4750.00ng/mL和4191.00pg/mL。她在第10天死于该病,对其组织的检查显示骨髓中存在登革热病毒基因组。
本文描述了一例具有HPS临床特征的致命性登革热病例。虽然BMAT结果未显示巨噬细胞噬血现象,但其他实验室特征与HPS一致,尤其是铁蛋白、sCD163和sCD25的显著升高。在发热15天后,在患者骨髓中检测到登革热病毒也支持HPS与登革热病毒感染相关的观点。
这些发现突出了HPS作为严重登革热可能并发症的可能性,并揭示了骨髓中持续存在的登革热病毒感染。因此,应考虑检测HPS标志物铁蛋白、sCD163和sCD25,以便早期识别与HPS相关的登革热。