Mycetoma in Somalia: an updated literature review

Abstract

Introduction:

Mycetoma is a chronic disabling inflammatory disease characterized by the formation of granulomatous masses in subcutaneous tissue extending to the skin, deep tissues, and bones.

Findings:

An exhaustive and detailed exploration of medical literature unveiled only 11 published articles addressing mycetoma in Somalia since 1927. Among these, six were case reports involving 12 patients, and three were case series encompassing 163 patients, resulting in a total of 175 reported cases. Notably, two articles in French lacking abstracts were excluded from the review. Considering Somalia shares geographical and environmental traits with other mycetoma-endemic regions, a heightened prevalence of the disease is anticipated in the country. It is essential to recognize, however, that mycetoma is not a notifiable disease, and the accuracy of disease reporting in Somalia is compromised due to various factors and consequently, the actual number of mycetoma cases in Somalia is likely to surpass the figures presented in this report.

Conclusion:

Ongoing research, international collaboration, and efforts to improve healthcare accessibility and awareness are essential. Encourage country to make mycetoma a notifiable disease with the integration of data collection in the national health information system. Addressing the environmental determinants, describe true burden, disability, impact of quality life, and implementing preventive measures can contribute to mitigating the impact of mycetoma in Somalia.

Introduction

Mycetoma is a chronic disabling inflammatory disease characterized by the formation of granulomatous masses in subcutaneous tissue extending to the skin, deep tissues, and bones^[1-3]. This disease can be caused by specific fungi (eumycetoma) or bacteria (actinomycetoma)^[4,5]. Globally, mycetoma exhibits a distinct geographical distribution, primarily endemic in tropical and subtropical regions^[6-8]. In Africa, it thrives in the “mycetoma belt,” encompassing the Sahel region, East Africa, and parts of West Africa, with heightened incidences reported in Sudan, Senegal, and Mauritania^[9-12]. The Middle East, particularly Yemen and Saudi Arabia, also document endemic cases^[13,14]. India and Pakistan report mycetoma endemicity in specific regions, while Southeast Asia, including Thailand and Indonesia, acknowledges sporadic cases^[15-17]. In Latin and South America, Mexico highlights significant mycetoma cases, particularly of the actinomycetoma type^[18-21]. Although the United States and Europe are not endemic, isolated cases are reported, often linked to a history of travel or residence in endemic regions^[22,23].

Mycetoma distribution aligns with environmental conditions, such as soil types and climate^[24,25]. Warm, arid to, semi-arid climates with alkaline soil provide ideal settings for the proliferation of mycetoma-causing agents ^[24,25]. Individuals with frequent exposure to soil and thorns, notably agricultural workers, have a higher risk of mycetoma development^[26,27]. Limited access to healthcare and diagnostic facilities poses challenges for accurate diagnosis and reporting of mycetoma cases among affected individuals^[28,29].

Recognized as a neglected tropical disease (NTD) by the World Health Organization (WHO), mycetoma impact extends beyond direct health consequences, affecting the quality of life and economic productivity of those in endemic regions^[30,31].

It is crucial to acknowledge the dynamic nature of mycetoma distribution and prevalence, which is subject to changes influenced by environmental changes, population movements, and advancements in diagnostic capabilities^[32]. Ongoing research and international collaboration play a crucial role in enhancing our understanding of mycetoma and its evolving global distribution.

Mycetoma risk factors in Somalia

Somalia’s geographical location, topography, and climate make it part of the mycetoma belt along with other endemic countries. Many specific factors in Somalia contribute to the risk of mycetoma, including its geographical location. Being located in the Horn of Africa, it is within the mycetoma belt, a region known for the high prevalence of mycetoma cases. The disease is particularly endemic in certain geographical zones, and Somalia’s position in this belt increases the risk of mycetoma transmission.

The diverse topography of Somalia, including plains, plateaus, and highlands, provides various ecological niches where the causative agents of mycetoma can thrive. The presence of specific fungi and bacteria in the environment, coupled with favorable conditions, contributes to the persistence of mycetoma in different regions of the country.

The challenging arid to semi-arid climate in Somalia also plays a role in the prevalence of mycetoma. The dry conditions during the extended dry season create environments where soil-borne pathogens responsible for mycetoma can survive and persist. Additionally, the wet season, especially in the southern regions, can create conditions conducive to the growth and transmission of mycetoma-causing agents.

Somalia’s reliance on agriculture, which is significantly impacted by the country’s climate and periodic droughts, may contribute to mycetoma risk. Agricultural workers, who often come into contact with contaminated soil and water sources, may be at higher risk of contracting mycetoma. The periodic droughts and environmental challenges in Somalia can strain the healthcare infrastructure, limiting access to proper medical care for mycetoma patients. Limited healthcare resources may result in delayed diagnoses and inadequate treatment, contributing to the persistence of the disease^[28,33-35].

Understanding these factors is crucial for public health initiatives aimed at preventing and managing mycetoma in Somalia. Efforts to improve healthcare accessibility, promote awareness, and address the environmental determinants of mycetoma can contribute to reducing the impact of this neglected tropical disease in the region.

The literature review

To identify relevant studies, a systematic search was conducted across multiple databases. Boolean operators (AND, OR) were used to refine the search, ensuring comprehensive coverage of studies.The following databases were searched: PubMed/Medline, Cochrane CENTRAL and Cochrane CDSR, Embase, Web of Science Core Collection, BIOSIS, Scopus, regional database, African Journals Online, IndMED and selected databases of the Global Index Medicus [AIM (AFRO), IMEMR (EMRO), IMSEAR (SEARO), LILACS (AMRO/OPAS)], Ovid Global Health, clinical trial registries (clinicaltrials.gov and WHO ICTRP). No language restrictions were applied (Table 1).

Table 1.

The published articles on mycetoma in Somalia in the medical literature search

Authors	Published date	Patients no.	Study date	Reference
Doğan et al	2024	10; 8 males, 2 females	Not reported	[36]
Zimmer et al	2023	1 female	2023	[37]
Cazzato et al	2021	1 male	2021	[38]
El Muttardi et al	2010	1 female	2010	[39]
Fasciana	2018	1 male	2018	[40]
Destombes et al	1971	103	1959-1964	[41]
Orio et al	1963	50	1954-1963	[42]
Rosati	1961	2	1961	[43]
Polidori	1936	5	1936	[44]
Gelonesi	1927	2	1927	[45]

The search revealed several mycetoma cases. Ten mycetoma cases were reported from Somalia in 2024. In this series, there were eight male patients and two females. Their ages ranged between 20 and 81 years. The disease duration ranged between three months and 31 years. All of them lived in rural areas and engaged in livestock care or farming. Mycetoma was reported in the lower limb in seven patients, two patients had thoracolumbar region, and one patient had hand mycetoma. The diagnosis was clinically in four patients, and in six patients, the diagnosis was confirmed by biopsy. The treatment was planned empirically or according to the biopsy results. Several medications were given that included Itraconazole 100 mg with amoxicillin-clavulanic acid 1 G bd or Monocycline 100 mg bd or trimethoprim-sulfamethoxazole bd combinations for at least 1 year. One patient with knee mycetoma underwent surgical excision for suspicion of malignancy. Recurrence was reported in two patients after one year. The types of mycetoma and treatment outcomes were not reported in this series^[36]. While recently in certain specialized centers use PCR for molecular identification. However, all these techniques are invasive, time-consuming, and risk complications like bleeding, sepsis, and disease spread. Additionally, these procedures are not field-friendly or suitable for epidemiological surveys, requiring patients to travel to specialized centers for disease confirmation and treatment. In remote rural areas, fine-needle aspiration cytology (FNAC) can diagnose mycetoma without the invasiveness of surgical biopsies. However, operator-dependent sensitivity and specificity are significant.^[46] While most acceptable current treatment options for actinomycetoma are cycles of the combination of amikacin sulfate 15 mg/kg twice daily for 3 weeks and cotrimoxazole 1.5 mg/kg twice daily for 5 weeks until cure. Ketoconazole 800 mg/day is the recommended drug, for eumycetoma, itraconazole 400 mg/day can be used for patients show poor response and/or developed severe side effects. The mean duration of treatment is 9 months.^[47]

In 2023, a report from Germany on a 25-year-old pregnant female immigrant from Somalia was published. She presented with nodular, indolent progressive swelling of the right distal-medial lower leg and multiple nodular changes on the fingers of both hands. A surgical biopsy showed no evidence of mycobacteria by conventional microbiology or specific polymerase chain reactions. A postpartum magnetic resonance imaging of the right lower leg showed the radiographic “dot in circle sign.” Further, histopathological examination revealed multiple branched fungal hyphae forming typical grains. Specific mycological polymerase chain reaction confirmed the diagnosis of Biatriospora mackinnonii (formerly classified as Nigrograna mackinnonii), a causative agent of Eumycetoma. This infiltrative mycosis is endemic in sub-Saharan Africa. Itraconazole (200 mg twice a day) was initiated. A follow-up magnetic resonance imaging after 6 months confirmed a clear regression of the findings^[37].

An 11-year-old boy, primarily from Djibouti, Somalia, with unspecified lesions affecting the malleolar and peri-malleolar region of the right foot, was reported in 2021. He had symptomless, painless diffuse nodular lesions that affected the skin and subcutaneous tissue, their color varied from yellow to red, hard in consistency, with multiple subcutaneous abscesses, which, after rupture, resulted in skin sinuses^[38]. The biopsy histopathological examination showed multiple grains of Streptomyces somaliensis surrounded by intense inflammatory granulomas and fibrosis, and that was confirmed by grains culture. He was started on Amikacin sulphate in combination with Trimethoprim-sulfamethoxazole, and after three months of therapy, the lesions began to respond and shrink in diameter^[38].

A 24-year-old immunocompetent Somali man who moved to different African countries and settled in Sicily, Italy, presented with a painless, slowly progressive massive foot mycetoma. His condition started when he was 13 years old. At presentation, the foot was badly affected and warm, with multiple draining sinuses and limited toes movements. His laboratory tests were within normal limits. Imaging examinations revealed marked soft tissue swelling, bone sclerosis and cavities of the five metatarsal and toe. A sole surgical biopsy was done and showed chronic granulomatous inflammation with PAS-positive grains. Grains culture revealed evidence of Actinomadura madurae confirmed by PCR and sequencing of 16S rRNA and hsp65 genes. He was started on a combination of antibiotics, and the lesion began to regress in size in 4 months^[40].

From the United Kingdom, a 4-year eumycetoma of the left foot in a 16-year-old Somalian girl was reported. She has been a resident of the UK for over a year. The patient underwent aggressive surgical debridement with a 6-month course of anti-fungal medication^[39].

A large case series of 103 mycetoma patients from Somalia in the period 1959 to 1964 was reported by the Pasteur Institute. Grains were seen in 94 of them and this, added to cultural features, allowed the diagnosis of 60 pathogens as follows: 44 Madurella mycetomi, 1 Leptosphaeria senegalensis, 1 Pyrenochaeta romeroi (Madurella griseea), 3 Allescheria boydii, 1 Fusarium sp., 3 Neotestudina (Zopfia) rosatii, and 1 unidentified; 34 were actinomycetes: 24 Streptomyces somaliensis, 4 Actinomadura madurae, 3 A. pellefieri and 3 Nocardia spp. Most patients presented late with advanced disease.

There was an association between the causative microorganism’s geographical distribution and the climate. In central Somalia, M. mycetomi was prevented, Streptomyces somaliensis, organisms reported from more desert areas, and white grain mycetomata and those caused by Nocardia spp. occurred in more humid areas^[41]. The study reported on two new fungi. One, obtained in culture, was called Neotestudina (Zopfia) rosatii. The three patients affected lived in Mudugh (two in El Bur). The other fungus was not identified. It was also recovered from El Bur, and one with similar microscopic characters has been seen in Chad and “territoire français des Afars et des Issas.” Both fungi are desert species. The authors recommended health education to raise awareness and early treatment^[41].

Orio and his associates reported on 50 mycetoma patients from Somalia seen in the period 1954 and 1962. The agents isolated were Streptomyces somaliensis (n = 23), Madurella mycetomi (n = 20) and Streptomyces madurae (n = 4). A small grains mycetoma (Nocardid) could not be identified specifically. A case of mycetoma due to Leptosphaeria senegalensis and a case due to Pyrenochaeta romeroi were histopathologically identified^[42].

Two Somali patients with mycetoma of the foot yielded soft whitish brown grains at operation were reported in 1961. The culture of these grains gave rise to a hitherto undescribed Ascomycete with many of the characteristics of the genus Testudina; the authors described the fungus and proposed the binomial Neotestudina rosatii nov. gen., nov. sp., for it^[43].

Polidori was reported in 1936 on mycetoma patients seen at Mogadishu Civil Hospital, South Somalia. He collected and reported on several cases of madura foot and other mycotic affections that included five mycetoma patients. Most patients had massive disease at presentation, and the lesions were ulcerated offensively with massive secretion. The patients were unable to walk, ill, and anemic. Large doses of potassium iodide were the treatment of choice but rarely effective. In many cases, amputation was necessary. For patients with early small lesions, wide local excisions were performed. The mycetoma type, diagnosis and treatment outcomes were not reported^[44].

In 1927, Gelonesi reported on two patients with mycetoma in Southern Somalia. He considered the causative microorganisms as parasites of Madura Foot. Both patients had black grains. He described the causative microscopic appearances and named them Mucor mycetoma and Aspergillus mycetomi villabruzzii^[45].

In the literature search, two articles in French without abstracts were found, and they were excluded.^[48,49].

Conclusion

In conclusion, this updated literature review on mycetoma in Somalia reveals a limited but valuable pool of information comprising 11 published articles. The identified cases encompassed a range of presentations, from common occurrences in rural areas to unique instances in immigrants, reflecting the diverse nature of mycetoma cases within the Somali population. The historical context, dating back to studies in the 1920s, emphasizes the persistence of mycetoma in Somalia. Despite advancements in healthcare, mycetoma remains a challenge, especially in remote areas where access to medical care is limited.

The geographical and environmental context of Somalia, positioned within the mycetoma belt, underscores the heightened risk of mycetoma occurrence in the country. The diverse topography, challenging arid climate, reliance on agriculture, and periodic droughts contribute to the conducive conditions for mycetoma-causing agents to thrive. These factors, combined with limited healthcare resources, further complicate the accurate diagnosis and reporting of mycetoma cases.

However, it is crucial to acknowledge the limitations of the existing literature, such as the small number of published articles and the challenges posed by two excluded French articles without abstracts. The lack of a standardized reporting system for mycetoma in Somalia further complicates efforts to estimate the actual prevalence accurately.

In light of these findings, ongoing research, international collaboration, and efforts to improve healthcare accessibility and awareness are essential. Develop and implement awareness campaigns aimed at educating communities about mycetoma’s symptoms, transmission, and preventive measures. Use accessible platforms like community centers, local radio, social media, and schools to reach wider audiences, emphasizing early detection and healthcare-seeking behavior.

Encourage country to make mycetoma a notifiable disease with the integration of data collection in the national health information system. Addressing the environmental determinants, describe true burden, disability, impact of quality life, and implementing preventive measures can contribute to mitigating the impact of mycetoma in Somalia. As the understanding of mycetoma evolves globally, continuous vigilance and research are imperative to adapt strategies for the dynamic nature of this neglected tropical disease.

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Authors’ contributions

All authors listed on the manuscript contributed significantly in the manuscript. All authors involved in the writing of the manuscript at draft and any revision stages, and have read and approved the final version.

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Data availability statement

All data were available within manuscript.