Hemophagocytic lymphohistiocytosis-a rare life threatening association with scrub typhus: case report

Arashdeep Virk, Mohit Singla, Pranay Trivedi, Prasun Bhattacharjee, Abhinav Tiwari
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Abstract

Scrub typhus is an important cause of acute febrile illness and one of the re-emerging infectious diseases in India particularly in southern Rajasthan. Hemophagocytic lympho-histiocytosis (HLH) results from an uncontrolled and ineffective hyperinflammatory response to a variety of triggers. HLH is further subdivided into primary and secondary type. We present a case of Scrub typhus which presented with multiple organ dysfunction syndrome (MODS) and secondary HLH which is a rare entity. A 9-month-old male child presented with high grade fever with encephalopathy. Examination showed hepatosplenomegaly, cervical Lymph adenopathy with eschar mark visible on abdomen. Scrub typhus was suspected on clinical grounds and further investigations were done. Serological diagnosis was strongly positive for scrub typhus. Initially, child did not respond to doxycycline, so further investigations were done to rule out HLH which fits into criteria of secondary HLH as per the revised HLH 2004 protocol. In view of secondary HLH and MODS methylprednisolone was added to treatment. Child responded to steroids and there was complete recovery. Scrub typhus patient with progressive MODS, in spite of appropriate antimicrobial therapy should raise the suspicion of secondary HLH which is rare but life-threatening condition and steroids plays an important role in the management of this condition.
嗜血细胞淋巴组织细胞增多症--一种罕见的与恙虫病相关的危及生命的疾病:病例报告
恙虫病是导致急性发热性疾病的重要原因,也是印度(尤其是拉贾斯坦邦南部)重新流行的传染病之一。嗜血细胞性淋巴组织细胞增生症(HLH)是由于对各种诱因产生失控和无效的高炎症反应所致。HLH 又分为原发性和继发性两种类型。我们介绍了一例恙虫病病例,该病例伴有多器官功能障碍综合征(MODS)和继发性HLH,这是一种罕见病。一名9个月大的男婴因高烧伴脑病就诊。检查结果显示肝脾肿大、颈淋巴腺病变,腹部可见焦痂。根据临床表现怀疑为恙虫病,并做了进一步检查。血清学诊断为恙虫病强阳性。最初,患儿对强力霉素没有反应,因此又做了进一步检查,以排除HLH,因为根据2004年修订的HLH方案,HLH符合继发性HLH的标准。鉴于继发性 HLH 和 MODS,治疗中增加了甲基强的松龙。患儿对类固醇治疗反应良好,已完全康复。尽管采用了适当的抗菌治疗,但恙虫病患者仍出现了进行性 MODS,这应引起对继发性 HLH 的怀疑。
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