Jacobs D H, Macher A M, Handler R, Bennett J E, Collen M J, Gallin J I
Gastroenterology. 1984 Jul;87(1):201-3.
Patients with the hyperimmunoglobulin E-recurrent infection (Job's) syndrome, which is characterized by an elevated immunoglobulin E level, recurrent staphylococcal infections, and an abnormality of neutrophil chemotaxis, have been reported to have visceral Candida infections in addition to their more frequent pyogenic infections. We report a patient with Job's syndrome who presented with massive hematemesis secondary to esophageal cryptococcosis. A thorough evaluation for an occult neoplasm or extraesophageal cryptococcosis was negative. The patient received a 6-wk course of amphotericin B (970 mg) and 5-fluorocytosine with complete radiographic and endoscopic resolution of the lesion. He is doing well 18 mo after therapy. The patient was not anergic, and his response to T-cell mitogens, helper-to-suppressor T-cell ratio, total number of T cells, and immunoglobulin-producing capability were all normal. This case is unusual in that it is the first documentation of a cryptococcoma of the esophagus and underscores the importance of culturing abnormal specimens for unsuspected pathogens in unusual clinical circumstances.
高免疫球蛋白E复发性感染(乔布氏)综合征患者的特征为免疫球蛋白E水平升高、复发性葡萄球菌感染以及中性粒细胞趋化性异常,据报道,除了更常见的化脓性感染外,他们还会发生内脏念珠菌感染。我们报告了一名乔布氏综合征患者,该患者因食管隐球菌病出现大量呕血。对隐匿性肿瘤或食管外隐球菌病的全面评估结果为阴性。该患者接受了为期6周的两性霉素B(970毫克)和5-氟胞嘧啶治疗,病变在影像学和内镜检查中完全消退。治疗18个月后他情况良好。该患者并非无反应性,其对T细胞有丝分裂原的反应、辅助性T细胞与抑制性T细胞的比例、T细胞总数以及产生免疫球蛋白的能力均正常。该病例不同寻常之处在于它是食管隐球菌瘤的首例记录,并强调了在不寻常的临床情况下对异常标本进行培养以检测未被怀疑的病原体的重要性。