Meningeal sporotrichosis in an immunocompetent host: case report

IF 1.3 4区 医学 Q2 MEDICINE, GENERAL & INTERNAL Sao Paulo Medical Journal Pub Date : 2023-01-01 DOI:10.5327/1516-3180.141s1.682
Lara Teixeira Paiva, V. Ribeiro, Kaisy Nagella Alves, P. P. Christo, A. P. Gomes
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Abstract

Endemic in Brazil, sporotrichosis usually presents in its cutaneous form. Meningeal involvement is rare and generally affects immunocompromised hosts. Here we describe a case of meningeal sporotrichosis in an immunocompetent patient without concurrent skin lesions. A 78-year-old woman, previously healthy, developed cognitive deficits with fluctuating mental confusion over the course of three months, until she went to the emergency department with fatigue and stupor. Laboratory workup showed severe hyponatremia due to inappropriate ADH secretion. She was treated with fluid restriction and discharged. Two months later, she was referred to a neurologist because of persistent mental confusion in spite of controlled sodium. She had no headache, fever, seizures, or focal deficits. Cerebrospinal fluid (CSF) analysis revealed chronic meningitis: 232 cells (95% lymphocytes), elevated protein (3.021 mg/dl) and low glucose; cultures and polymerase chain reaction (PCR) for Mycobacterium were negative. Brain magnetic resonance imaging (MRI) revealed basal meningeal enhancement. She was hospitalized and empirically treated for meningeal tuberculosis, with no clinical nor CSF improvement after one month. In a careful history review, we found out that one year earlier she had been exposed to cats who died from sporotrichosis. One month after that, she had had a single skin lesion which spontaneously disappeared, followed by polyarthritis, which also resolved. At the time of our evaluation, she had no cutaneous lesion nor arthritis; CTs of the chest and abdomen were normal. PCR for Sporothrix in the CSF came out positive. She received liposomal amphotericin for 1 month, followed by a 6-months use of itraconazole. She fully recovered her cognitive capacities; follow-up MRI showed no alterations; her late CSF was nearly normal (5 cells, protein 55 mg/dl, no glucose consumption). Our report draws the attention to atypical neurological presentations of sporotrichosis, a treatable condition that may unrecognized.
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免疫正常宿主的脑膜孢子虫病:1例报告
孢子虫病在巴西流行,通常以皮肤形式出现。脑膜受累是罕见的,通常影响免疫功能低下的宿主。在这里我们描述一个病例脑膜炎孢子虫病在免疫能力的病人没有并发皮肤病变。一名78岁的女性,之前健康,在三个月的时间里出现了认知缺陷和波动的精神混乱,直到她因疲劳和昏迷而去了急诊室。实验室检查显示由于ADH分泌不当导致严重的低钠血症。患者经限制液体治疗后出院。两个月后,她被转介到神经科医生那里,尽管服用了控制钠的药物,但她仍然精神错乱。患者无头痛、发热、癫痫或局灶性缺陷。脑脊液(CSF)分析显示慢性脑膜炎:232个细胞(95%淋巴细胞),蛋白升高(3.021 mg/dl)和低血糖;分枝杆菌培养和聚合酶链反应(PCR)阴性。脑磁共振成像显示基底脑膜增强。她因脑膜结核住院并经验性治疗,一个月后临床和脑脊液均无改善。在仔细的历史回顾中,我们发现一年前她接触过死于孢子虫病的猫。一个月后,她出现了一处皮肤损伤,然后自然消失,接着出现了多发性关节炎,也消失了。在我们评估时,她没有皮肤病变,也没有关节炎;胸部和腹部ct检查正常。脑脊液孢子丝菌PCR阳性。她接受两性霉素脂质体治疗1个月,随后使用伊曲康唑6个月。她完全恢复了认知能力;随访MRI未见改变;晚期脑脊液基本正常(5个细胞,蛋白55 mg/dl,无葡萄糖消耗)。我们的报告引起了对孢子虫病的非典型神经学表现的关注,孢子虫病是一种可治疗但可能未被识别的疾病。
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来源期刊
Sao Paulo Medical Journal
Sao Paulo Medical Journal 医学-医学:内科
CiteScore
2.20
自引率
7.10%
发文量
210
审稿时长
6-12 weeks
期刊介绍: Published bimonthly by the Associação Paulista de Medicina, the journal accepts articles in the fields of clinical health science (internal medicine, gynecology and obstetrics, mental health, surgery, pediatrics and public health). Articles will be accepted in the form of original articles (clinical trials, cohort, case-control, prevalence, incidence, accuracy and cost-effectiveness studies and systematic reviews with or without meta-analysis), narrative reviews of the literature, case reports, short communications and letters to the editor. Papers with a commercial objective will not be accepted.
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