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mandrillaris</span> has been little known&#44; and it can only be concluded that soil and polluted water exposures were mentioned frequently in the cases&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">Here&#44; the authors reported a 15-year-old boy presented with a red plaque on the nasal dorsum over one month&#44; with slight itching&#46; He denied fever or taking any medicine lately&#46; His family and travel history were normal&#44; but he used to swim in the wild pond with ambiguous trauma on the nose&#46; Dermatologic examination revealed a well-circumscribed red plaque with a slightly raised border and mild infiltration with a few scales on the surface &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#44; involving about two-thirds of his nasal dorsum&#46; No enlarged cervical lymph node was detected&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The biopsy was taken from the red plaque&#44; and the histopathology showed mixed inflammatory cells infiltration with the predominance of lymphocytes&#44; histocytes&#44; eosinophils&#44; and multinuclear giant cells&#44; with numerous granulomas observed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; However&#44; both Periodic Acid-Schiff &#40;PAS&#41; and acid-fast staining were negative&#44; as well as fungal culture and atypical mycobacteriosis detection&#46; Even so&#44; oral Itraconazole was prescribed&#44; 200&#8239;mg&#47;day&#46; A month later&#44; he had a &#8220;cold&#8221; with a slight cough and an enlarged lesion involving almost the whole nasal dorsum &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#46; Computed Tomography &#40;CT&#41; scan of the lungs was negative&#44; and a second biopsy showed a similar histological profile &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46; As the investigation of <span class="elsevierStyleItalic">Mycobacterium tuberculosis</span> on lesion sample by PCR-reversed dot hybridization had been negative&#44; the treatment was switched to oral clarithromycin 500&#8239;mg&#47;day and doxycycline 200&#8239;mg&#47;day&#44; combined with Itraconazole 400&#8239;mg&#47;day&#46; Another month later&#44; high-grade fever and repeated nosebleeds occurred&#46; He got into a coma and was hospitalized in another hospital five days later&#46; A cranial CT scan showed multiple abnormal low-density foci in bilateral frontal&#44; parietal lobes&#44; and right occipital lobes&#46; His cerebrospinal fluid sample was sent to Guangzhou Vision medicals Inc&#46; for pathogen detection via Next-Generation Sequence &#40;NGS&#41;&#44; which confirmed the presence of <span class="elsevierStyleItalic">B&#46; mandrillaris</span> genomic sequences expressed as 88&#46;17&#37; of the whole parasites&#44; which suggests that <span class="elsevierStyleItalic">B&#46; mandrillaris</span> was the main pathogen&#46; Though Itraconazole and Linezolid intravenously were given&#44; he died about 2-weeks later&#44; unfortunately&#46; The authors confirmed that <span class="elsevierStyleItalic">B&#46; mandrillaris</span> infection already existed in the embedding skin biopsy tissue from initial red plaques by NGS&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">Only about 7 cases of <span class="elsevierStyleItalic">B&#46; mandrillaris</span> infection were reported per year worldwide&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The patients can be divided into two types by different clinical manifestations&#46; One type is mainly from the United States&#44; which presents with encephalitis without skin lesions&#44; and usually dies within months&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4</span></a> The other one is observed mainly in China<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and Peru&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> where the skin is usually involved first and then gradually develops into encephalitis&#46; Up to 50&#37; of cases were children&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Wang et al&#46; reported 28 Chinese cases&#44; most of them seemed to be less aggressive with higher survival rate &#40;44&#37;&#41;&#44; which may be related to the timely treatment before infection developed into GAE&#46; Similarly&#44; the present case also presented with skin lesions&#44; and it took about half a year before encephalitis&#46; Unfortunately&#44; the authors didn&#39;t diagnose in time and prevent the infection from rapidly worsening&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">It is difficult to diagnose <span class="elsevierStyleItalic">B&#46; mandrillaris</span> infection early&#46; Traumatic history and contact history of dirty water or soil should be of concern&#46; Most cases showed a common indurate plaque without ulceration or pustule secretion&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Microscopically&#44; the amoeba looks like histiocytes in morphology and is difficult to be identified&#46; Conventional PAS and acid-fast staining are unhelpful&#44; and the infection is easily misdiagnosed as lethal midline granuloma&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Immunohistochemical staining with anti<span class="elsevierStyleItalic">-Balamuthia</span> antibody is useful&#44; while the antibody is not commercially available yet&#46; The next-generation sequence technique is recommended&#44; which can get a higher positive rate for more kinds of microbes&#44; especially some rare pathogen&#44; from different samples&#44; such as tissues&#44; blood&#44; body fluids&#44; and secretions&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The treatment is troublesome for physicians&#44; and there is no standard strategy till now&#46; The miltefosine may benefit patients and should be considered in any treatment plan&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Besides&#44; lincomycin&#44; azithromycin&#44; and interferon-&#947; may be useful&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Except for drug treatments&#44; Doyle et al&#46; reported a case successfully treated with excision of the infected brain tissue&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> but it is not the first choice for most encephalitis&#46; Clinically it is imperative for us to make early diagnoses and discover effective treatments to save more lives&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">Our project was supported by the National Natural Science Foundation of China &#40;Grant No&#46;81773316&#41;&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors&#39; contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Kang Tao&#58; Conception and planning of the study&#59; critical review of the literature&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; elaboration and writing of the manuscript&#59; critical review of the manuscript&#59; approval of the final version of the manuscript&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ting Wang&#58; Approval of the final version of the manuscript&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; critical review of the manuscript&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Lian Zhang&#58; Approval of the final version of the manuscript&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; critical review of the manuscript&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Xi-Chuan Yang&#58; Approval of the final version of the manuscript&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; critical review of the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Zhi-Fang Zhai&#58; Conception and planning of the study&#59; critical review of the literature&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; elaboration and writing of the manuscript&#59; critical review of the manuscript&#59; approval of the final version of the manuscript&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41;&#44; Clinical image showing a red well-circumscribed plaque on his nasal dorsum with slightly raised border and a few scales on the surface&#46; &#40;B&#41;&#44; The histopathology examination of the first biopsy shows cutaneous mixed inflammatory cells infiltration&#44; including lymphocytes&#44; histocytes&#44; eosinophils and multinuclear giant cells&#44; with numerous granulomas observed&#46; &#40;Hematoxylin &#38; eosin&#44; &#215;100&#41;&#46;</p>"
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Tropical/Infectoparasitary Dermatology
Fatal Balamuthia mandrillaris infection with red plaques on the nasal dorsum as the first presentation
Kang Tao, Ting Wang, Lian Zhang, Xi-Chuan Yang, Zhi-Fang Zhai
Corresponding author
zhaizf1004@163.com

Corresponding author.
Department of Dermatology and Venereology, Southwest Hospital, Army Medical University, Chongqing, China
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41;&#44; Clinical image showing a red well-circumscribed plaque on his nasal dorsum with slightly raised border and a few scales on the surface&#46; &#40;B&#41;&#44; The histopathology examination of the first biopsy shows cutaneous mixed inflammatory cells infiltration&#44; including lymphocytes&#44; histocytes&#44; eosinophils and multinuclear giant cells&#44; with numerous granulomas observed&#46; &#40;Hematoxylin &#38; eosin&#44; &#215;100&#41;&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Introduction</span><p id="par0005" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleItalic">Balamuthia mandrillaris</span> infection is a rare and fatal disease caused by <span class="elsevierStyleItalic">B&#46; mandrillaris</span> amoeba&#44; which was first isolated from a pregnant mandrill monkey died from encephalitis in 1986&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> It causes Central Nervous System &#40;CNS&#41; disease called Granulomatous Amebic Encephalitis &#40;GAE&#41;&#46; Most patients rapidly developed fatal encephalitis&#44; and the lethality rate was up to 90&#37; in the United States from 1974 to 2016&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The epidemiology of <span class="elsevierStyleItalic">B&#46; mandrillaris</span> has been little known&#44; and it can only be concluded that soil and polluted water exposures were mentioned frequently in the cases&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Case report</span><p id="par0010" class="elsevierStylePara elsevierViewall">Here&#44; the authors reported a 15-year-old boy presented with a red plaque on the nasal dorsum over one month&#44; with slight itching&#46; He denied fever or taking any medicine lately&#46; His family and travel history were normal&#44; but he used to swim in the wild pond with ambiguous trauma on the nose&#46; Dermatologic examination revealed a well-circumscribed red plaque with a slightly raised border and mild infiltration with a few scales on the surface &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>A&#41;&#44; involving about two-thirds of his nasal dorsum&#46; No enlarged cervical lymph node was detected&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">The biopsy was taken from the red plaque&#44; and the histopathology showed mixed inflammatory cells infiltration with the predominance of lymphocytes&#44; histocytes&#44; eosinophils&#44; and multinuclear giant cells&#44; with numerous granulomas observed &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>B&#41;&#46; However&#44; both Periodic Acid-Schiff &#40;PAS&#41; and acid-fast staining were negative&#44; as well as fungal culture and atypical mycobacteriosis detection&#46; Even so&#44; oral Itraconazole was prescribed&#44; 200&#8239;mg&#47;day&#46; A month later&#44; he had a &#8220;cold&#8221; with a slight cough and an enlarged lesion involving almost the whole nasal dorsum &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>A&#41;&#46; Computed Tomography &#40;CT&#41; scan of the lungs was negative&#44; and a second biopsy showed a similar histological profile &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>B&#41;&#46; As the investigation of <span class="elsevierStyleItalic">Mycobacterium tuberculosis</span> on lesion sample by PCR-reversed dot hybridization had been negative&#44; the treatment was switched to oral clarithromycin 500&#8239;mg&#47;day and doxycycline 200&#8239;mg&#47;day&#44; combined with Itraconazole 400&#8239;mg&#47;day&#46; Another month later&#44; high-grade fever and repeated nosebleeds occurred&#46; He got into a coma and was hospitalized in another hospital five days later&#46; A cranial CT scan showed multiple abnormal low-density foci in bilateral frontal&#44; parietal lobes&#44; and right occipital lobes&#46; His cerebrospinal fluid sample was sent to Guangzhou Vision medicals Inc&#46; for pathogen detection via Next-Generation Sequence &#40;NGS&#41;&#44; which confirmed the presence of <span class="elsevierStyleItalic">B&#46; mandrillaris</span> genomic sequences expressed as 88&#46;17&#37; of the whole parasites&#44; which suggests that <span class="elsevierStyleItalic">B&#46; mandrillaris</span> was the main pathogen&#46; Though Itraconazole and Linezolid intravenously were given&#44; he died about 2-weeks later&#44; unfortunately&#46; The authors confirmed that <span class="elsevierStyleItalic">B&#46; mandrillaris</span> infection already existed in the embedding skin biopsy tissue from initial red plaques by NGS&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Discussion</span><p id="par0020" class="elsevierStylePara elsevierViewall">Only about 7 cases of <span class="elsevierStyleItalic">B&#46; mandrillaris</span> infection were reported per year worldwide&#46;<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> The patients can be divided into two types by different clinical manifestations&#46; One type is mainly from the United States&#44; which presents with encephalitis without skin lesions&#44; and usually dies within months&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4</span></a> The other one is observed mainly in China<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> and Peru&#44;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> where the skin is usually involved first and then gradually develops into encephalitis&#46; Up to 50&#37; of cases were children&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Wang et al&#46; reported 28 Chinese cases&#44; most of them seemed to be less aggressive with higher survival rate &#40;44&#37;&#41;&#44; which may be related to the timely treatment before infection developed into GAE&#46; Similarly&#44; the present case also presented with skin lesions&#44; and it took about half a year before encephalitis&#46; Unfortunately&#44; the authors didn&#39;t diagnose in time and prevent the infection from rapidly worsening&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">It is difficult to diagnose <span class="elsevierStyleItalic">B&#46; mandrillaris</span> infection early&#46; Traumatic history and contact history of dirty water or soil should be of concern&#46; Most cases showed a common indurate plaque without ulceration or pustule secretion&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Microscopically&#44; the amoeba looks like histiocytes in morphology and is difficult to be identified&#46; Conventional PAS and acid-fast staining are unhelpful&#44; and the infection is easily misdiagnosed as lethal midline granuloma&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Immunohistochemical staining with anti<span class="elsevierStyleItalic">-Balamuthia</span> antibody is useful&#44; while the antibody is not commercially available yet&#46; The next-generation sequence technique is recommended&#44; which can get a higher positive rate for more kinds of microbes&#44; especially some rare pathogen&#44; from different samples&#44; such as tissues&#44; blood&#44; body fluids&#44; and secretions&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">The treatment is troublesome for physicians&#44; and there is no standard strategy till now&#46; The miltefosine may benefit patients and should be considered in any treatment plan&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Besides&#44; lincomycin&#44; azithromycin&#44; and interferon-&#947; may be useful&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Except for drug treatments&#44; Doyle et al&#46; reported a case successfully treated with excision of the infected brain tissue&#44;<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">10</span></a> but it is not the first choice for most encephalitis&#46; Clinically it is imperative for us to make early diagnoses and discover effective treatments to save more lives&#46;</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">Our project was supported by the National Natural Science Foundation of China &#40;Grant No&#46;81773316&#41;&#46;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Authors&#39; contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Kang Tao&#58; Conception and planning of the study&#59; critical review of the literature&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; elaboration and writing of the manuscript&#59; critical review of the manuscript&#59; approval of the final version of the manuscript&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Ting Wang&#58; Approval of the final version of the manuscript&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; critical review of the manuscript&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Lian Zhang&#58; Approval of the final version of the manuscript&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; critical review of the manuscript&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Xi-Chuan Yang&#58; Approval of the final version of the manuscript&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; critical review of the manuscript&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">Zhi-Fang Zhai&#58; Conception and planning of the study&#59; critical review of the literature&#59; obtaining&#44; analysis&#44; and interpretation of the data&#59; elaboration and writing of the manuscript&#59; critical review of the manuscript&#59; approval of the final version of the manuscript&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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        "resumen" => "<span id="abst0005" class="elsevierStyleSection elsevierViewall"><p id="spar0015" class="elsevierStyleSimplePara elsevierViewall"><span class="elsevierStyleItalic">Balamuthia mandrillaris</span> infection is a rare infectious disease around the world&#44; with high rates of morbidity and mortality&#46; Its early and correct diagnosis is a big challenge for us&#44; and without it the delay in starting effective treatment can lead to the development of encephalitis&#46; This is a report of a case of <span class="elsevierStyleItalic">Balamuthia mandrillaris</span> infection in a Chinese boy&#44; with red plaques on the nasal dorsum as the first presentation&#44; who finally developed into fatal encephalitis&#46; The authors have reviewed the related literature and share the special skin features in order to favor the early diagnosis of the disease and increase the chances of survival&#46;</p></span>"
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          "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">&#40;A&#41;&#44; Clinical image showing a red well-circumscribed plaque on his nasal dorsum with slightly raised border and a few scales on the surface&#46; &#40;B&#41;&#44; The histopathology examination of the first biopsy shows cutaneous mixed inflammatory cells infiltration&#44; including lymphocytes&#44; histocytes&#44; eosinophils and multinuclear giant cells&#44; with numerous granulomas observed&#46; &#40;Hematoxylin &#38; eosin&#44; &#215;100&#41;&#46;</p>"
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Article information
ISSN: 03650596
Original language: English
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