A Case Series of Adult and Pediatric Basidiobolomycosis in Saudi Arabia: An Emerging Rare Disease

IF 2.2 4区 医学 Q2 MEDICINE, GENERAL & INTERNAL
Fahdah Al-Mutairi, Adel Almaymuni, Bayan Hafiz, Alanoud Alghanem, Nada Rabie, Mohammed Samannodi, Nabeel Khojah, Ishaq Mudawi, Abdulkareem Alshamrani, Tahani Almeleebia, Wala Felemban, Daliah Abdulhafeez, Rehab Fadaq, Khalid Al Johani, Sara Tayeb, Heba Jalloun
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Abstract

Background: Basidiobolomycosis is a fungal infection caused by Basidiobolus ranarum. It is primarily located in the tropical areas. It is commonly affecting the gastrointestinal tract and rarely presents with systemic affection.

Patients and Methods: The records of fifteen patients, including pediatrics, from various geographic regions (western and southern regions of the Kingdom of Saudi Arabia) who were diagnosed with basidiobolomycosis in two tertiary hospitals over the last fourteen consecutive years from January 2010 to February 2023 were included. A detailed history and data for each patient were reviewed and evaluated.

Results: A total of fifteen patients were found to have basidiobolomycosis, of which fourteen were found to have gastrointestinal basidiobolomycosis (GIB). They presented with abdominal pain, fever, nausea, vomiting, diarrhea, and loss of weight. One of them with an orbital infection presented with periorbital swelling, hotness, and redness. The diagnosis was confirmed after histopathological examination with typical features of granulomatous reaction, dense infiltrate of eosinophils, and fungal structures. Seven patients were treated with antifungals alone, such as voriconazole or itraconazole. Eight patients were treated by surgical resection followed by antifungal medication. Ten patients demonstrated good clinical response and completed their treatment course, while one patient showed a slight improvement and is still receiving treatment and follow-up. There have been four reported cases of death. The length of the therapy was scheduled based on how the patients responded clinically to the intervention.

Conclusions: We present the series with the most patients collected to date, and it is shown that GIB is often misdiagnosed as cancer or inflammatory bowel disease. Histological analysis confirms the diagnosis of basidiobolomycosis. The choice of therapy is still up for debate: medical intervention alone or surgical resection followed by antifungal medication.

Abstract Image

沙特阿拉伯成人和儿童担子孢子菌病病例系列:一种新出现的罕见疾病
背景:担子孢子菌病是由担子孢子菌引起的真菌感染。它主要位于热带地区。它通常影响胃肠道,很少表现出全身的影响。患者和方法:纳入了2010年1月至2023年2月连续14年在两家三级医院诊断为担子孢子菌病的不同地理区域(沙特阿拉伯王国西部和南部地区)的15例包括儿科在内的患者的记录。对每位患者的详细病史和数据进行了回顾和评估。结果:15例患者发现担子黑菌病,其中14例发现胃肠道担子黑菌病(GIB)。患者表现为腹痛、发热、恶心、呕吐、腹泻和体重减轻。其中1例眼眶感染,表现为眼眶周围肿胀、发热、红肿。经组织病理学检查,诊断为肉芽肿反应,嗜酸性粒细胞密集浸润,真菌结构。7例患者单独使用伏立康唑或伊曲康唑等抗真菌药物治疗。8例患者行手术切除加抗真菌药物治疗。10例临床反应良好,完成疗程,1例稍有好转,仍在接受治疗和随访。已有4例死亡报告。治疗时间的长短是根据患者对干预的临床反应来确定的。结论:我们提供了迄今为止收集的大多数患者的系列,并表明GIB经常被误诊为癌症或炎症性肠病。组织学分析证实担子孢子菌病的诊断。治疗的选择仍在争论中:单独的医学干预还是手术切除后的抗真菌药物治疗。
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来源期刊
CiteScore
5.30
自引率
0.00%
发文量
274
审稿时长
3-8 weeks
期刊介绍: IJCP is a general medical journal. IJCP gives special priority to work that has international appeal. IJCP publishes: Editorials. IJCP Editorials are commissioned. [Peer reviewed at the editor''s discretion] Perspectives. Most IJCP Perspectives are commissioned. Example. [Peer reviewed at the editor''s discretion] Study design and interpretation. Example. [Always peer reviewed] Original data from clinical investigations. In particular: Primary research papers from RCTs, observational studies, epidemiological studies; pre-specified sub-analyses; pooled analyses. [Always peer reviewed] Meta-analyses. [Always peer reviewed] Systematic reviews. From October 2009, special priority will be given to systematic reviews. [Always peer reviewed] Non-systematic/narrative reviews. From October 2009, reviews that are not systematic will be considered only if they include a discrete Methods section that must explicitly describe the authors'' approach. Special priority will, however, be given to systematic reviews. [Always peer reviewed] ''How to…'' papers. Example. [Always peer reviewed] Consensus statements. [Always peer reviewed] Short reports. [Always peer reviewed] Letters. [Peer reviewed at the editor''s discretion] International scope IJCP publishes work from investigators globally. Around 30% of IJCP articles list an author from the UK. Around 30% of IJCP articles list an author from the USA or Canada. Around 45% of IJCP articles list an author from a European country that is not the UK. Around 15% of articles published in IJCP list an author from a country in the Asia-Pacific region.
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