阴茎硬化性淋巴管炎4例分析

目的:提高对阴茎硬化性淋巴管炎的认识和诊断水平。方法:通过总结临床阴茎硬化性淋巴管炎的临床资料,分析阴茎硬化性淋巴管炎的误诊原因。结果:阴茎硬化性淋巴管炎比较少见,临床上多易误诊。结论:阴茎硬化性淋巴管炎是良性的自限性疾病,诊断明确后治疗相对简单,临床医生要加强对其的认识,减少误诊误治。     

Rickettsia sibirica isolation from a patient and detection in ticks, Portugal

We report the first isolation of Rickettsia sibirica (strain mongolotimonae) from the blood of a patient and detection by polymerase chain reaction (PCR) of the rickettsia in a Rhipicephalus pusillus tick collected from a dead mongoose (Herpestes ichneumon) in the Alentejo region, Portugal. We describe also the first PCR detection of a new Rickettsia strain that is related to R. sibirica.     

从丝虫病淋巴管(结)炎的特殊临床表现分析其发病原因

在马来丝虫病流行区调查时，曾收集到有特殊临床表现的淋巴管（结）炎病例，其中有离开流行区即停止发作的，有服海群生后导致发作的，有患腿继发感染并严重化脓后获痊愈的，也有继发细菌感染而诱发的。作者举出７个典型病例，试图对丝虫病流行区常见的急性淋巴管（结）炎的病因进行分析，作为一个方面的论证资料。     

Granulomas obstruct lymphatics in all layers of the intestine in Crohn's disease

After an earlier study defining immunolabeled lymphoid follicles, obstructed lymphatics, and granulomas of the diseased ilea of 24 Crohn's disease patients, we chose to trace the lymphatics of these cases and 10 additional by serial sectioning. Particular attention was given to establishing physical continuity between granuloma‐obstructed lymphatics and lymphatics with ‘lymphocytic thrombi’. Formalin‐fixed paraffin‐embedded tissue blocks from resected diseased ilea and proximal colons from 34 patients were reviewed. Patients were 13 men and 21 women, aged 14–60 years. Duration of disease ranged from 1 month to 10 years. Immunohistochemistry employed D2‐40 antibody to label lymphatics and anti‐CD68 to label granulomas. Twenty‐nine of the 34 (85%) resection tissues had lymphangectasia, in mucosa, submucosa, and subserosa. In 53% of the specimens, lymphatics of the various layers were obstructed by granulomas that filled the lumina. In 44%, 15/34, there were also distended lymphatics that were totally plugged with lymphocytes. In 10 of the 15, serial sections revealed continuity between the lymphocyte‐plugged lymphatics and the endolymphatic granulomatous obstruction downstream. In 5 of the cases, D2‐40 immunostaining revealed redundant lymphatic endothelium interwoven with the granuloma cells. Granulomas totally obstruct lymphatics in all layers of the intestine in Crohn's disease. Upstream of these obstructions, lymphatics are distended with lymphocytes. The degree and extent of this potentially irreversible ‘lymphangitis nodosa’ have undoubtedly confounded treatment regimens and clinical trials. There currently are no imaging methods to demonstrate the lymphangitis, nor treatments to resolve it.     

Metastatic Crohn’s disease accompanying granulomatous vasculitis and lymphangitis in the vulva

Metastatic Crohn’s disease (CD) is an extremely rare extragastrointestinal manifestation of CD, and is characterized histopathologically by the presence of non-caseating granulomatous inflammation. Granulomatous vasculitis and lymphangitis have rarely been documented in metastatic CD. Herein, we report the first documented case of metastatic CD accompanied by both granulomatous vasculitis and lymphangitis in the vulva. A 35-year-old Japanese female with CD presented with multiple small nodules in her vulva. Biopsy was performed under a clinical diagnosis of genital warts. A histopathological study revealed marked lymphangiectasia in the papillary dermis. Within the dilated lymphatics, lymphocytes and aggregates of macrophages were present, which are typical features of granulomatous lymphangitis. Tiny non-caseating granulomas and granulomatous vasculitis were also observed. Accordingly, a diagnosis of metastatic CD accompanied by both granulomatous vasculitis and lymphangitis was made. The occurrence of cutaneous lesions in patients with CD is well known. Albeit extremely rare, lymphangiectasia has been reported in the vulva of CD patients that clinically mimicked viral warts, as in the present case. The diagnosis of metastatic CD in the present case was not difficult because characteristic histopathological features were present, and a clinical history of CD was available. However, a few cases of genital swelling associated with granulomatous inflammation prior to a diagnosis of gastrointestinal CD have been documented. Therefore, granulomatous vasculitis and lymphangitis in the external genitals should be considered as potential indication of metastatic CD even in cases without a history of gastrointestinal CD.