Successful Treatment of Balamuthia mandrillaris Amoebic Infection with Extensive Neurological and Cutaneous Involvement

Instituto de Medicina Tropical Alexander von Humboldt, Universidad Peruana Cayetano Heredia, Lima, Peru.
Clinical Infectious Diseases (Impact Factor: 8.89). 07/2010; 51(2):e7-11. DOI: 10.1086/653609
Source: PubMed


Granulomatous amoebic encephalitis caused by Balamuthia mandrillaris is an uncommon infection for which there is no optimal therapy. We describe a young, female patient who presented with extensive
cutaneous and neurological involvement and who recovered after receiving prolonged treatment with miltefosine, fluconazole,
and albendazole.

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Available from: Alfonso Martín Cabello-Vílchez, Jul 04, 2015
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    • "There is precedence for other agents that pass poorly into CSF, such as macrolides, penetrating well into brain parenchyma (Nau et al. 2010; Jaruratanasirikul et al. 1996). Despite our patient's measured drug levels, miltefosine has shown promise in treating other patients with Balamuthia GAE, in combination with other drugs (Bravo et al. 2011; Martínez et al. 2010; Centers for Disease Control and Prevention 2010). It is possible that the observed success of miltefosine in reported GAE cases may be due to superior penetration into brain parenchyma when compared to CSF. "
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    ABSTRACT: Balamuthia mandrillaris, a free-living ameba, causes rare but frequently fatal granulomatous amebic encephalitis (GAE). Few patients have survived after receiving experimental drug combinations, with or without brain lesion excisions. Some GAE survivors have been treated with a multi-drug regimen including miltefosine, an investigational anti-leishmanial agent with in vitro amebacidal activity. Miltefosine dosing for GAE has been based on leishmaniasis dosing because no data exist in humans concerning its pharmacologic distribution in the central nervous system. We describe results of limited cerebrospinal fluid (CSF) and serum drug level testing performed during clinical management of a child with fatal GAE who was treated with a multiple drug regimen including miltefosine. Brain biopsy specimens, CSF, and sera were tested for B. mandrillaris using multiple techniques, including culture, real-time polymerase chain reaction, immunohistochemical techniques, and serology. CSF and serum miltefosine levels were determined using a liquid chromatography method coupled to tandem mass spectrometry. The CSF miltefosine concentration on hospital admission day 12 was 0.4 μg/mL. The serum miltefosine concentration on day 37, about 80 h post-miltefosine treatment, was 15.3 μg/mL. These are the first results confirming some blood-brain barrier penetration by miltefosine in a human, although with low-level CSF accumulation. Further evaluation of brain parenchyma penetration is required to determine optimal miltefosine dosing for Balamuthia GAE, balanced with the drug's toxicity profile. Additionally, the Balamuthia isolate was evaluated by real-time polymerase chain reaction (PCR), demonstrating genetic variability in 18S ribosomal RNA (18S rRNA) sequences and possibly signaling the first identification of multiple Balamuthia strains with varying pathogenicities.
    Full-text · Article · Sep 2015 · Parasitology Research
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    • "CT scan and MRI typically show multifocally enhanced mass-like lesions with ring enhancement, edema, and hydrocephalus mimicking brain abscess, brain metastasis, or intracerebral hematoma [3]. A small number of solitary mass lesion was also reported [8-10,25,26] which mimicked a brain tumor with no obvious sign of encephalitis, similar to our case. Interestingly, a single mass in most cases situated in the anterior cranial fossa. "
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    ABSTRACT: Balamuthia mandrillaris is one of the 4 amebas in fresh water and soil that cause diseases in humans. Granulomatous amebic encephalitis (GAE), caused by B. mandrillaris, is a rare but life-threatening condition. A 4-year-old, previously healthy, Thai girl presented with progressive headache and ataxia for over a month. Neuroimaging studies showed an infiltrative mass at the right cerebellar hemisphere mimicking a malignant cerebellar tumor. The pathological finding after total mass removal revealed severe necrotizing inflammation, with presence of scattered amebic trophozoites. Cerebrospinal fluid (CSF) obtained from lumbar puncture showed evidence of non-specific inflammation without identifiable organisms. A combination of pentamidine, sulfasalazine, fluconazole, and clarithromycin had been initiated promptly before PCR confirmed the diagnosis of Balamuthia amebic encephalitis (BAE). The patient showed initial improvement after the surgery and combined medical treatment, but gradually deteriorated and died of multiple organ failure within 46 days upon admission despite early diagnosis and treatment. In addition to the case, 10 survivors of BAE reported in the PubMed database were briefly reviewed in an attempt to identify the possible factors leading to survival of the patients diagnosed with this rare disease.
    Full-text · Article · Jun 2013 · The Korean Journal of Parasitology
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    • "Recently, however, three patients, an ∼60-year-old patient from California, a 6-year-old girl, also from California, and a 70- year-old female from New York, survived the infection after treatment with a combination of pentamidine isethionate, sulfadiazine, azithromycin/clarithromycin, fluconazole, and 5-fluorocytosine [12] [21]. Currently, several patients with balamuthiasis are being treated with the above regimen as well as miltefosine [7] [8], which has been found to have strong inhibitory effects in vitro on Balamuthia [42] and has been used successfully to cure this infection [30]. Other survivors include two Peruvian patients with cutaneous lesions who became well after prolonged therapy with albendazole and itraconazole [6]. "
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    ABSTRACT: Members of the free-living amebic genera Acanthamoeba, Balamuthia, and Naegleria are known to cause infections of the central nervous system (CNS) of humans and other animals. Several species of Acanthamoeba cause an insidious and chronic disease, granulomatous amebic encephalitis (GAE), principally in immunocompromised hosts including persons infected with HIV/AIDS. Additionally, Acanthamoeba spp. also causes infection of the human cornea, Acanthamoeba keratitis. B. mandrillaris, the only known species of Balamuthia, causes GAE in both immunocompromised and immunocompetent hosts. Both Acanthamoeba and B. mandrillaris also cause a disseminated disease including the lungs, skin, kidneys, and uterus. N. fowleri, on the other hand, infects immunocompetent children and young adults leading to an acute and fulminating, necrotizing primary amebic meningoencephalitis. This review describes the biology of the amebae, clinical manifestations, diagnosis including molecular identification, immunological, and epidemiological features associated with the infections caused by these amebae.
    Full-text · Article · Jan 2010 · Journal of neuroparasitology
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