Scheib J S, Quinet R J
Department of Internal Medicine, Ochsner Clinic, New Orleans, La.
South Med J. 1990 Jun;83(6):684-7. doi: 10.1097/00007611-199006000-00024.
A seropositive white man had follow-up for 16 years with a diagnosis of palindromic rheumatism. Treatment had included parenteral gold, methotrexate, prednisone, hydroxychloroquine sulfate, and penicillamine before diarrhea led to a biopsy-proven diagnosis of Whipple's disease. Clinical and radiographic criteria for ankylosing spondylitis were met. In addition to classic Whipple's arthropathy, he had the combined but singular findings of pancarpal destruction and cervical apophyseal fusion. HLA typing revealed the B7 antigen. This case illustrates the pitfalls in diagnosis of a chronic polyarthritis that has, as a typical feature, a long latency before manifesting its more specific signs and symptoms (ie, diarrhea, malabsorption, and hyperpigmentation). Care should be taken during evaluation of any disease with atypical and nonspecific features (eg, positive rheumatoid factor in a patient with polyarthritis) and one should continue to reevaluate the original impression while confirmatory evidence is lacking. Moreover, the roentgenographic findings of pancarpal narrowing, apophyseal fusion, and advanced iliofemoral joint disease, in addition to sacroiliitis and syndesmophyte formation, challenge the generally held notion that Whipple's arthropathy is a nondestructive joint disease.
一名血清学阳性的白人男性因复发性风湿症接受了16年的随访。治疗方法包括胃肠外注射金制剂、甲氨蝶呤、泼尼松、硫酸羟氯喹和青霉胺,后来腹泻导致经活检证实为惠普尔病。该患者符合强直性脊柱炎的临床和影像学标准。除了典型的惠普尔关节病外,他还出现了腕骨全部破坏和颈椎骨突融合这两种合并但独特的表现。人类白细胞抗原(HLA)分型显示其携带B7抗原。该病例说明了慢性多关节炎诊断中的陷阱,这种疾病的典型特征是在出现更具特异性的体征和症状(如腹泻、吸收不良和色素沉着)之前有很长的潜伏期。在评估任何具有非典型和非特异性特征的疾病(如多关节炎患者类风湿因子呈阳性)时应谨慎,在缺乏确诊证据的情况下,应持续重新评估最初的印象。此外,腕骨全部狭窄、骨突融合以及严重的髂股关节疾病的影像学表现,连同骶髂关节炎和韧带骨赘形成,对惠普尔关节病是一种非破坏性关节疾病这一普遍观点提出了挑战。