Lehur A C, Zielinski M, Pluvy J, Grégoire V, Diamantis S, Bleibtreu A, Rioux C, Picard A, Vallois D
Infectious and Tropical Diseases Department, University Hospital Bichat-Claude Bernard, APHP, 46 rue Henri Huchard, 75018, Paris, France.
Otorhinolaryngology Department, University Hospital Bichat-Claude Bernard, APHP, Paris, France.
BMC Infect Dis. 2017 May 5;17(1):328. doi: 10.1186/s12879-017-2419-4.
Disseminated Histoplasmosis (DH) is a rare manifestation of Acquired Immune Deficiency Syndrome (AIDS) in European countries. Naso-maxillar osteolysis due to Histoplasma capsulatum var. capsulatum (Hcc) is unusual in endemic countries and has never been reported in European countries. Differential diagnoses such as malignant tumors, cocaine use, granulomatosis, vasculitis and infections are more frequently observed and could delay and/or bias the final diagnosis.
We report the case of an immunocompromised patient infected by Human Immunodeficiency Virus (HIV) with naso-maxillar histoplasmosis in a non-endemic country. Our aim is to describe the clinical presentation, the diagnostic and therapeutic issues. A 53-year-old woman, originated from Haiti, was admitted in 2016 for nasal deformation with alteration of general condition evolving for at least 6 months. HIV infection was diagnosed in 2006 and classified at AIDS stage in 2008 due to cytomegalovirus infection associated with pulmonary histoplasmosis. At admission, CD4 cell count was 9/mm. Surgical biopsies were performed and ruled out differential or associated diagnoses. Mycological cultures identified Hcc and Blood Polymerase Chain Reaction (PCR) for Hcc was positive. The patient was given daily Amphothericin B liposomal infusion during 1 month. Hcc PCR became negative in the blood under treatment, and then oral switch by itraconazole was introduced. Antiretroviral treatment was reintroduced after a 3-week histoplasmosis treatment. Normalization of naso-maxillar mucosa enabled a palatal prosthesis.
Naso-maxillar histoplasmosis is extremely rare; this is the first case ever reported in a non-endemic country. Differential diagnoses must be ruled out by conducting microbiologic tools and histological examinations on surgical biopsies. Early antifungal treatment should be initiated in order to prevent DH severe outcomes.
播散性组织胞浆菌病（DH）在欧洲国家是获得性免疫缺陷综合征（AIDS）的一种罕见表现。荚膜组织胞浆菌荚膜变种（Hcc）引起的鼻上颌骨骨质溶解在该病流行国家并不常见，在欧洲国家从未有过报道。诸如恶性肿瘤、可卡因使用、肉芽肿病、血管炎和感染等鉴别诊断更为常见，可能会延迟和/或影响最终诊断。
我们报告一例在非流行国家感染人类免疫缺陷病毒（HIV）并患有鼻上颌骨组织胞浆菌病的免疫功能低下患者。我们的目的是描述其临床表现、诊断和治疗问题。一名53岁来自海地的女性于2016年因鼻畸形及全身状况改变入院，这种情况已持续至少6个月。2006年诊断出HIV感染，2008年因与肺组织胞浆菌病相关的巨细胞病毒感染而被归类为AIDS期。入院时，CD4细胞计数为9/mm。进行了手术活检，排除了鉴别诊断或相关诊断。真菌培养鉴定出Hcc，血液聚合酶链反应（PCR）检测Hcc呈阳性。患者接受了为期1个月的每日脂质体两性霉素B输注。治疗期间血液中的Hcc PCR转为阴性，随后改用伊曲康唑口服。在组织胞浆菌病治疗3周后重新开始抗逆转录病毒治疗。鼻上颌骨黏膜恢复正常后安装了腭托。
鼻上颌骨组织胞浆菌病极为罕见；这是在非流行国家报道的首例病例。必须通过对手术活检进行微生物学检测和组织学检查来排除鉴别诊断。应尽早开始抗真菌治疗以预防DH的严重后果。
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