Neuro-leishmaniasis with cauda equina syndrome and cranial nerve palsy: a rare manifestation of recurrent atypical visceral leishmaniasis.

IF 3.4 3区 医学 Q2 INFECTIOUS DISEASES BMC Infectious Diseases Pub Date : 2024-11-06 DOI:10.1186/s12879-024-10082-z
Karthick Kumar Vaitheeswaran, Baidhnath Kumar Gupta, Rahul Krishnan G, Manish Soneja, Naval K Vikram, Upendra Baitha, Amandeep Singh, Naveet Wig, Mudsser Azam, Ruchi Singh, Ajay Garg, Nishikant Damle, Yamini Dharmashaktu
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Abstract

Background: Visceral leishmaniasis (VL) is a neglected tropical disease primarily affecting Brazil, East Africa, and India, with India accounting for 18% of the global burden. While VL typically presents with systemic symptoms like fever, weight loss, and splenomegaly, it can occasionally manifest atypically, posing significant diagnostic challenges. Neurological presentations of VL are extremely rare, making them difficult to suspect and diagnose. Cases where VL predominantly presents with neurological symptoms are particularly novel, underscoring the need for heightened awareness of such atypical manifestations in endemic regions.

Clinical case: A 38-year-old man with history of recurrent atypical VL presented with diffuse lower back pain, progressive tingling, numbness, weakness in the lower extremities, and double vision for one month. Clinical and radiological evaluations suggested cauda equina syndrome and cranial nerve palsy, accompanied by generalized lymphadenopathy, subcutaneous nodules, and skin papules. The differential diagnosis initially included disseminated tuberculosis, histoplasmosis, and lymphoma. Cerebrospinal fluid (CSF) analysis revealed an inflammatory syndrome. Histopathology of lymph node and bone marrow revealed Leishmania amastigotes and subcutaneous nodule and skin biopsy revealed inflammatory cells with granulomas. Furthermore, the qPCR test on DNA from a subcutaneous nodule, lymph node, and CSF was positive for Leishmania kinetoplast DNA. The species was further confirmed as Leishmania donovani through ITS-based PCR amplification and sequencing. Finally, a diagnosis of relapse of VL with lymph node, cutaneous, and neurological involvement, including abducens nerve palsy and cauda equina syndrome, was established. He was treated with combination of liposomal amphotericin B and miltefosine, along with intrathecal hyaluronidase, resulting in significant improvement.

Conclusion: Unlike previously reported cases with both systemic and neurological symptoms, our patient predominantly presented with neurological manifestations, making this a unique and novel presentation of VL. This case highlights diagnostic challenges and management of atypical VL, emphasizing neurological involvement and successful therapeutic strategies.

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神经利什曼病伴有马尾综合征和颅神经麻痹:复发性非典型内脏利什曼病的罕见表现。
背景:内脏利什曼病(VL)是一种被忽视的热带疾病,主要影响巴西、东非和印度,其中印度的发病率占全球发病率的 18%。虽然内脏利什曼病通常表现为发热、体重减轻和脾肿大等全身症状,但偶尔也会有不典型的表现,这给诊断带来了巨大挑战。VL 的神经系统表现极为罕见,因此难以怀疑和诊断。VL主要表现为神经系统症状的病例尤为新颖,突出表明在流行地区需要提高对此类不典型表现的认识:临床病例:一名 38 岁的男子曾反复出现非典型 VL,并伴有弥漫性下背痛、进行性刺痛、麻木、下肢无力和复视一个月。临床和放射学评估提示马尾综合征和颅神经麻痹,伴有全身淋巴结病、皮下结节和皮肤丘疹。最初的鉴别诊断包括播散性结核、组织胞浆菌病和淋巴瘤。脑脊液(CSF)分析表明患者患有炎症综合征。淋巴结和骨髓的组织病理学检查发现了利什曼原虫,皮下结节和皮肤活检发现了带有肉芽肿的炎性细胞。此外,对皮下结节、淋巴结和 CSF 的 DNA 进行的 qPCR 检测显示,利什曼原核 DNA 呈阳性。通过基于 ITS 的 PCR 扩增和测序,进一步确认了该病原体为多诺万利什曼病。最后,他被确诊为 VL 复发,并伴有淋巴结、皮肤和神经系统受累,包括外展神经麻痹和马尾综合征。他接受了两性霉素 B 脂质体和米替福新的联合治疗,并使用了鞘内透明质酸酶,结果病情明显好转:结论:与之前报道的同时伴有全身症状和神经系统症状的病例不同,我们的患者主要表现为神经系统症状,因此是一种独特而新颖的 VL 表现。本病例突出了非典型 VL 的诊断挑战和管理,强调了神经系统受累和成功的治疗策略。
本文章由计算机程序翻译,如有差异,请以英文原文为准。
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来源期刊
BMC Infectious Diseases
BMC Infectious Diseases 医学-传染病学
CiteScore
6.50
自引率
0.00%
发文量
860
审稿时长
3.3 months
期刊介绍: BMC Infectious Diseases is an open access, peer-reviewed journal that considers articles on all aspects of the prevention, diagnosis and management of infectious and sexually transmitted diseases in humans, as well as related molecular genetics, pathophysiology, and epidemiology.
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