Date Published: March 13, 2015
Publisher: Public Library of Science
Author(s): Ahmed Fahal, EL Sheikh Mahgoub, Ahmed Mohamed EL Hassan, Angom Osman Jacoub, Doaa Hassan, Joseph M. Vinetz. http://doi.org/10.1371/journal.pntd.0003587
Abstract: Mycetoma is a unique neglected tropical disease which is endemic in what is known as the “mycetoma belt”. The disease has many devastating impacts on patients and communities in endemic area and is characterised by massive deformity, destruction and disability. Mycetoma is commonly seen in the foot and hand and less frequent in other parts of the body. Mycetoma of the head and neck is a rarity and is associated with high morbidity and even mortality if not treated early. In this communication we report on 49 patients with head and neck mycetoma followed up at the Mycetoma Research Centre in Khartoum. Most of the reported patients had actinomycetoma and the majority were young adult males from mycetoma endemic areas in the Sudan. Most of them were students, farmers and workers. Prior to presentation the majority had long disease duration and the cause was multifactorial. Advanced disease with massive lesion, deformity and disability was the common presentation. There was no obvious history of local trauma, familial tendency or other predisposing factor identified in this group of patients. MRI and CT scan were the most accurate diagnostic tools to determine the disease extent. The treatment outcome was rather poor and characterised by low cure rate, poor outcome and high follows-up dropout. Such a gloomy outcome calls for structured and objective health education programs.
Partial Text: Mycetoma is one of the neglected tropical diseases, characterised by massive deformity, disability and can be fatal if not managed properly and timely [1–3]. It is a chronic, specific, granulomatous, progressive subcutaneous inflammatory disease that spreads to involve the skin, deep structures and bones [4,5]. The disease is caused by true fungi or by certain bacteria and hence it is usually classified into eumycetoma and actinomycetoma, respectively [6,7]. Madurella mycetomatis is the commonest eumycetoma causative agent while Streptomyces somaliensis and Nocardiae are the common causative organisms for actinomycetoma [8–10]. Mycetoma has a definite geographic distribution and it is endemic in what is known as the “Mycetoma Belt” that includes Sudan, Senegal, Somalia, South India, South America and Mexico; however, it is reported in many other countries [11–17]. The infection usually progresses slowly over many years and it is commonly painless and that may contribute to the late presentation of many patients [2,18]. The painless subcutaneous mass, multiple sinuses and discharge with grains is distinctive of this infection  Young adult males in the age range 20–40 years are more frequently affected [2,4]. Farmers, workers and students are affected most but no occupation is exempted [2,5].
This descriptive, cross-sectional hospital based study was conducted at the Mycetoma Research Centre (MRC), University of Khartoum, Khartoum, Sudan. The study included 49 patients with confirmed head and neck mycetoma seen in the period January 1991 and October 2014. The diagnosis of mycetoma was confirmed by careful interview, meticulous clinical examinations and standard investigations.
The 49 studied patients with confirmed head & neck mycetoma constituted 0.76% of the total MRC patient population seen during the study duration. In the present study, 33 patients (67.3%) had actinomycetoma and 16 (32.7%) had eumycetoma. There were 39 males (79.6%) and 10 females (20.4%). Their ages ranged between 9 and 67 years with a mean age of 27.9 ± 14.7 years. 31(63.3%) of the patients were under 40 years-old at presentation and 23(46.9%) were in the age group 1–20 years. Only five patients (10.2%) were more than 50 years of age at presentation.
The incidence of mycetoma of the head and neck region is infrequent. Review of the medical literature revealed only few reports on mycetoma in this site [10,29–31], and although Sudan is considered the mycetoma homeland, only few reports on head and neck mycetoma were reported. Lynch in 1964, reported on 1860 mycetoma patients and of these only 18 patients (0.96%) had head and neck mycetoma . Mahgoub in 1977 reported an incidence of 3% of head and neck mycetoma . In 1986, Gumaa and her associates reported on 15 out 400 patients with mycetoma (3.75%) involving the head and neck region. This communication is in line with the fact that, mycetoma at this region is a rarity.