Aoun Jessica, Habib Robert, Charaffeddine Khalil, Taraif Suad, Loya Asif, Khalifeh Ibrahim
Department of Pathology and Laboratory Medicine, American University of Beirut Medical Center, Beirut, Lebanon.
Department of Internal Medicine, Outcomes Research Unit, American University of Beirut Medical Center, Beirut, Lebanon.
PLoS Negl Trop Dis. 2014 Oct 23;8(10):e3255. doi: 10.1371/journal.pntd.0003255. eCollection 2014 Oct.
Caseating granulomas are often associated with a mycobacterial infection (TB) and are thought to be exceedingly rare in cutaneous leishmaniasis (CL). However, no large series has accurately documented the incidence of caseating granulomas in CL.
A multiregional cohort consisting of 317 patients with CL [Syria (157), Pakistan (66), Lebanon (47), Saudi Arabia (43), Ethiopia (2) and Iran (2)] was reviewed. Clinical [age, sex, disease duration, lesion type and geographic and anatomic location] and microscopic data [presence of and type of granuloma, Ridley's parasitic index (PI) and pattern (RP)] were documented. Presence of microorganisms was evaluated using special stains (GMS, PAS, AFB and Gram) and polymerase chain reaction (PCR) for TB and CL. All cases included in this study were confirmed as CL by PCR followed by restriction fragment length polymorphism analysis for molecular speciation and were negative for other organisms by all other studies performed. Categorical and continuous factors were compared for granuloma types using Chi-square, t-test or Mann-Whitney test as appropriate.
Granulomas were identified in 195 (61.5%) cases of CL and these were divided to 49 caseating (25.2%), 9 suppurative (4.6%) and 137 tuberculoid without necrosis (70.2%). Caseating and tuberculoid granuloma groups were significantly different in terms of the geographical source, with more cases harboring caseating granulomas in Saudi Arabia (p<0.0001). Histologically, both groups were also different in the distribution of their RP (p<0.0001) with a doubling RP3 in caseating granulomas (31% vs. 15%) as opposed to doubling of RP5 in tuberculoid granuloma group (38% vs. 19%). Time needed to achieve healing (RP5) was notably shorter in tuberculoid vs. caseating group (4.0 vs. 6.2 months). Parasitic Index, CL species and other considered variables did not differ for the granuloma type groups.
In our multiregional large cohort, a notable 18.2% of all CL cases harbored caseating granulomas therefore; CL should be considered part of the differential diagnosis for cases with caseating granulomas in endemic regions, especially considering that the regions included in our cohort are also endemic for TB. Of note, cases of CL with caseating granulomas also showed a slower healing process, with no association with specific species, which may be due to worse host immune response in such cases or to a more aggressive leishmania strains.
干酪样肉芽肿常与分枝杆菌感染(结核病)相关,被认为在皮肤利什曼病(CL)中极为罕见。然而,尚无大型系列研究准确记录CL中干酪样肉芽肿的发病率。
对一个多地区队列进行了回顾,该队列由317例CL患者组成[叙利亚(157例)、巴基斯坦(66例)、黎巴嫩(47例)、沙特阿拉伯(43例)、埃塞俄比亚(2例)和伊朗(2例)]。记录了临床[年龄、性别、病程、病变类型以及地理和解剖位置]和显微镜检查数据[肉芽肿的存在及类型、里德利寄生虫指数(PI)和模式(RP)]。使用特殊染色(GMS、PAS、抗酸杆菌和革兰氏染色)以及针对结核病和CL的聚合酶链反应(PCR)评估微生物的存在情况。本研究纳入的所有病例均通过PCR确诊为CL,随后进行限制性片段长度多态性分析以进行分子分类,并且通过所有其他检测均未检测到其他生物体。根据情况,使用卡方检验、t检验或曼 - 惠特尼检验对肉芽肿类型的分类和连续因素进行比较。
在195例(61.5%)CL病例中发现了肉芽肿,这些肉芽肿分为49例干酪样(25.2%)、9例化脓性(4.6%)和137例无坏死的结核样(70.2%)。干酪样肉芽肿组和结核样肉芽肿组在地理来源方面存在显著差异,沙特阿拉伯有更多病例存在干酪样肉芽肿(p<0.0001)。在组织学上,两组在RP分布方面也存在差异(p<0.0001),干酪样肉芽肿中RP3翻倍(31%对15%),而结核样肉芽肿组中RP5翻倍(38%对19%)。结核样肉芽肿组实现愈合(RP5)所需时间明显短于干酪样肉芽肿组(4.0个月对6.2个月)。肉芽肿类型组的寄生虫指数、CL种类及其他考虑变量无差异。
在我们的多地区大型队列中,所有CL病例中有18.2%存在干酪样肉芽肿;因此,在流行地区,对于有干酪样肉芽肿的病例,应将CL纳入鉴别诊断,特别是考虑到我们队列中的地区也是结核病的流行区。值得注意的是,伴有干酪样肉芽肿的CL病例愈合过程也较慢,与特定种类无关,这可能是由于此类病例中宿主免疫反应较差或利什曼原虫菌株更具侵袭性。