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A case of primary disseminated rhinosporidiosis and dapsone-induced autoimmune hemolytic anemia: A therapeutic misadventure

机译:主要筛选的鼻孢子虫病和双酮诱导的自身免疫溶血性贫血案例:治疗性误解

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摘要

Rhinosporidiosis, a chronic inflammatory disease, which is caused by the aquatic microorganism Rhinosporidium seeberi, is endemic in India and in many other regions of the tropics. It primarily infects mucocutaneous surfaces of nose, nasopharynx, and conjunctiva through transepithelial invasion. However, over the centuries, atypical involvement of other body parts, especially viscera, bone, subcutaneous layers, genitals, the tracheobronchial tree, and even the skull has been, though rarely, reported. This chronic granulomatous infection is notorious for its propensity for recurrence following autoinoculation and poor response to most of the anti-microbials except dapsone. Surgical excision followed by cauterization remains the treatment of choice when an operation is feasible. We herein report a case of an immunocompetent person with primary disseminated dermato-pulmonary rhinosporidiosis, which created significant diagnostic dilemma at the beginning, got complicated due to dapsone-induced direct anti-globulin test-positive autoimmune hemolytic anemia, and finally responded to prolonged multidrug therapy with liposomal amphotericin B, ketoconazole and cycloserine. This report establishes the importance of tissue diagnosis in rhinosporidiosis and even, in resource-poor set-ups, a simple histopathological diagnosis can promote an early and affordable accurate diagnosis, and subsequently, a proper therapeutic intervention.
机译:鼻孢子虫病,慢性炎症疾病,由水生微生物鼻孢菌虫虫,在印度流行,在热带地区的许多其他地区。它主要通过Transepearlial侵袭感染鼻咽,鼻咽和结膜的粘膜皮肤表面。然而,几个世纪以来,其他身体部位的非典型涉及,尤其是内脏,骨,皮下层,生殖器,气管弯曲树,甚至是头骨,虽然很少报道。这种慢性肉芽肿性感染对于自身入侵后复发性和对大多数抗微生物的反应差的倾向是臭名昭着的。手术切除后跟烧灼仍然是当操作是可行的时选择的选择。我们在此情况下,我们报告了一种具有初级散发的皮肤肺鼻窦病虫病的免疫活性剂的案例,它在开始时产生了显着的诊断困境,由于双酮诱导的直接抗球蛋白试验阳性免疫溶血性贫血具有复杂,并且最终应对延长的多药用脂质体两性霉素B,酮康唑和环丝氨酸治疗。本报告规定了组织诊断在鼻孢子虫病中的重要性,甚至在资源差的设置中,简单的组织病理学诊断可以促进早期和实惠的准确诊断,随后进行适当的治疗干预。

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