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CASE REPORT
Year : 2022  |  Volume : 23  |  Issue : 1  |  Page : 83-86

Actinomycetoma in a child: A rare occurrence


Department of Dermatology and STD, University College of Medical Sciences and GTB Hospital (University of Delhi), Delhi, India

Date of Submission08-Jul-2021
Date of Decision12-Nov-2021
Date of Acceptance19-Nov-2021
Date of Web Publication31-Dec-2021

Correspondence Address:
Archana Singal
Department of Dermatology and STD, University College of Medical Sciences and GTB Hospital (University of Delhi), Delhi - 110 095
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/ijpd.ijpd_106_21

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  Abstract 


Mycetoma is a chronic, suppurative, localized, and destructive infection involving skin, subcutaneous tissue, fascia, muscle and sometimes invade the underlying bone. The infection is prevalent in tropical countries and involves chiefly adults. Etiologically, it is classified into eumycetoma and actinomycetoma caused by fungus and bacteria respectively. Clinically, mycetoma presents as a triad of firm to soft swelling with multiple sinuses and discharging grains of different colors. Mycetoma is reported rarely in children. We report an 11-year-old boy diagnosed with actinomycetoma of the left foot with classical clinical, histological, and radiological features.

Keywords: Actinomycetoma, child, discharging sinuses, eumycetoma, grains


How to cite this article:
Yadav S, Singal A. Actinomycetoma in a child: A rare occurrence. Indian J Paediatr Dermatol 2022;23:83-6

How to cite this URL:
Yadav S, Singal A. Actinomycetoma in a child: A rare occurrence. Indian J Paediatr Dermatol [serial online] 2022 [cited 2022 Jan 16];23:83-6. Available from: https://www.ijpd.in/text.asp?2022/23/1/83/334660




  Introduction Top


Mycetoma is a chronic suppurative granulomatous infection affecting skin, subcutaneous tissue, fascia, muscles and sometimes involves the underlying bone. The infection generally follows traumatic inoculation of the causative micro-organism over the exposed and trauma-prone sites. Although mycetoma has been reported from all over the world, the disease is more prevalent in mycetoma belt (tropical and subtropical regions especially Sudan, Mexico, India, and Venezuela).[1] It is endemic in India affecting mostly people of low socioeconomic strata and manual workers such as farmers and laborers. Mycetoma may be caused by the filamentous bacteria (actinomycetoma) or fungi (eumycetoma). Clinically, it presents as firm to soft swelling with multiple sinuses discharging grains of different colors which is characteristic of mycetoma. Worldwide, Madurella mycetomatis is the most common cause of this affliction while In India, Nocardia species are the commonest causes of actinomycetoma with the exception of northern regions (particularly Rajasthan) where eumycetoma occurs more frequently.  Actinomadura pelletieri Scientific Name Search more common in regions with heavy rainfall and Nocardia brasilliensis and Streptomyces somaliensis occur predominantly in dry deserted areas.


  Case Report Top


An 11-year-old girl, resident of Delhi, presented with diffuse, ill-defined, circumferential swelling overlying left ankle joint extending up to forefoot with multiple discharging sinuses of 3 months duration. The child had a history of preceding trauma at this site for which surgical intervention was done about 8 weeks back by a general practitioner. Examination revealed ill-defined, firm, nontender, indurated diffuse swelling with intervening scarring and sinuses discharging pus on manipulation but no grains were extracted. Contracture was present over the dorsum of foot along the ankle joint (intervention site) leading to restriction of movement of affected foot mainly dorsiflexion and plantar flexion and difficulty in walking [Figure 1] and [Figure 2]. Rest of the physical and systemic examination revealed no abnormality. Hematological and biochemical parameters of the child were within the normal range. X-ray of the joint showed focal non expansible lytic lesion with no sclerotic margin at metaphysis of tibia and no cortical defect. Computerized tomography scan revealed diffuse osteopenia with lytic lesions in tibia, multiple soft tissue swelling and abscesses [Figure 3]. A wedge biopsy was performed from the lesion and was sent for culture and histological examination. The culture grew no organism after 6-weeks of inoculation. The hematoxylin and eosin-stained sections demonstrated suppurative mixed cell granuloma composed of neutrophils, lymphocytes and plasma cells surrounding an intensely eosinophilic collection of micro-organisms arranged in a radiate, star-like, club shaped configurations known as Splendore–Hoeppli Phenomenon [Figure 4].
Figure 1: Ill-defined circumferential swelling over left ankle with multiple discharging sinus

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Figure 2: Contracture over the dorsal surface of the left foot involving the ankle joint and difficulty in dorsiflexion

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Figure 3: Computerized tomography scan of the left foot showing ddiffuse osteopenia with lytic lesion in tibia, soft tissue swelling and abscesses, characteristic of mycetoma

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Figure 4: Splendore-Hoeppli phenomenon demonstrating suppurative mixed cell granuloma composed of neutrophils, lymphocytes and plasma cells surrounding an intensely eosinophilic collection of micro-organisms (H and E, ×400)

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Based on the classical clinical features supplemented by radiological and histopathological findings, a final diagnosis of actinomycetoma was made. The patient was started on injectable amoxycillin-clavulanic acid (375 mg thrice daily) and amikacin (250 mg once daily) along with oral trimethoprim and sulfamethoxazole (400 mg) plus trimethoprim (80 mg) twice daily in an intensive phase of 4 weeks followed by oral amoxiclav and cotrimoxazole in the maintenance phase for 6 months. Subsequent monthly follow-up showed no active discharge along with appreciable decrease in the circumference of the lesion [Figure 5]. After 6 months of treatment, the patient's mobility at ankle joint is significantly restored with a considerable decrease in the size of swelling.
Figure 5: Reduction in the swelling over the foot as well as healing sinuses after 6 weeks of treatment

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  Discussion Top


Mycetoma is a chronic suppurative infection affecting skin, subcutaneous tissue, and bone prevalent in tropical and subtropical regions between latitudes 15°S and 30°N known as the mycetoma belt[1] due to high humidity and short rainy season. The disease is endemic in the Indian subcontinent and was added to the list of neglected tropical disease by WHO in 2013.[2] It is an infrequent disease in children with reported incidence ranging between 3% and 4.5% in various studies. A large multicenter Mexican series of 2105 patients with mycetoma seen over a period of 30 years, only 78 children (3.7%) <15 years of age, were reported and Bonifaz and his associates in 2007, reported on 15 mycetoma patients with age <15 years seen over a period of 25 years.[3],[4] Mycetoma are caused by various species of fungi and bacteria. Etiologically, it is classified into actinomycetoma and eumycetoma with former accounting for nearly 75% cases in India in adults as well as children. Actinomycetoma is caused by the aerobic species of actinomycetes belonging to the genera Nocardia, Streptomyces, and Actinomadura with Nocardia brasiliensis, Actinomadura madurae, Actinomadura pelletieri, and Streptomyces somaliensis being the most common. Eumycotic mycetoma is caused by variety of fungi, the most common being Madurella mycetomatis.[5] Clinically, mycetoma is characterised by triad of subcutaneous swelling, multiple sinuses and discharge containing grains mainly over the trauma prone exposed sites predominantly the lower limbs due to traumatic subcutaneous inoculation but rarely can involve shoulder, face, neck, thorax and abdomen as well.[6] The clinical presentation and further extent of the disease involvement is considerably same in children as in adults.[3],[4] Disease evolves over period of months to years after inoculation via minor trauma. If left undiagnosed and untreated, the disease has the potential of causing deformity and disabilities and accelerates the rate of drop outs in children from school. However, amputation rates are lower, probably because of the shorter duration of disease and earlier reporting to hospital.[4] Therefore, prompt diagnosis and treatment are of utmost importance. Various diagnostic modalities are directed at identification of species and bony involvement which are similar in children as well as adults and are implicated to decides the drugs, mode and duration of therapy.

Examination of the grain macro and microscopically obtained spontaneously or on manually expression and their color helps in the identification of the causative organism [Table 1]. Direct microscopy using gram-stained preparation of discharge or fine needle aspirate helps in identifying fine filaments for actinomycotic etiology and short and broad hyphae for eumycetoma.[7] The culture is done on blood agar/egg-containing media for actinomycetes and sabouraud dextrose agar for fungal culture. Histopathology shows suppurative granuloma surrounding the broad radially arranged hyphae which is further surrounded by mixed inflammatory infiltrate comprising of lymphocytes, neutrophils, plasma cells, and eosinophils.[8],[9] Polymerase chain reaction permits the identification of specific species and phylogenetic relationship and can be performed directly on the biopsy specimen. Radioimaging is an important diagnostic tool to differentiate between actinomycetoma and eumycetoma as illustrated in the [Table 2].[10]
Table 1: Causative organism with corresponding colour of grains in mycetoma

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Table 2: Radiological features in mycetoma

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Our patient had discharging sinuses without grains and histology was consistent with actinomycetoma.

Actinomycetoma is amendable to treatment provided the therapy is instituted early. Usually, combination therapy is provided to avoid drug resistance. Welsh regimen comprises of cyclic doses of amikacin 15 mg/kg/day in two divided doses in cycles of 21 days (1–3 cycles given) with cotrimoxazole DS BD continuously for 35–105 days. In modified welsh regimen, rifampicin is added as a third drug.[10] Other drugs that can be used with variable results are dapsone, streptomycin, ciprofloxacin, and nelitmicin. Cases unresponsive to medical therapy can be taken for surgery.[8] In our experience, as in the present case, combination treatment with amoxycillin clavulanic acid, amikacin, and cotrimoxazole works is a safe and effective treatment modality.


  Conclusion Top


We would like to conclude that actinomycetoma though rare can present in child with significant morbidity requiring early intervention to prevent further deeper extension and subsequent complications.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Relhan V, Mahajan K, Agarwal P, Garg VK. Mycetoma: An update. Indian J Dermatol 2017;62:332-40.  Back to cited text no. 1
[PUBMED]  [Full text]  
2.
van de Sande W, Maghoub ES, Fahal AH, Goodfellow M, Welsh O, Zijlstra E. The mycetoma knowledge gap: Identification of research priorities. PloS Negl Trop Dis 2014;8:e2667.  Back to cited text no. 2
    
3.
Bonifaz A, Ibarra G, Saúl A, Paredes-Solis V, Carrasco-Gerard E, Fierro-Arias L. Mycetoma in children: Experience with 15 cases. Pediatr Infect Dis J 2007;26:50-2.  Back to cited text no. 3
    
4.
Fahal AH, Sabaa AH. Mycetoma in children in Sudan. Trans R Soc Trop Med Hyg 2010;104:117-21.  Back to cited text no. 4
    
5.
Reis CM, Reis-Filho EG. Mycetomas: An epidemiological, etiological, clinical, laboratory and therapeutic review. An Bras Dermatol 2018;93:8-18.  Back to cited text no. 5
    
6.
Welsh O, Vera-Cabrera L, Salinas-Carmona MC. Mycetoma. Clin Dermatol 2007;25:195-202.  Back to cited text no. 6
    
7.
Afroz N, Khan N, Siddiqui FA, Rizvi M. Eumycetoma versus actinomycetoma: Diagnosis on cytology. J Cytol 2010;27:133-5.  Back to cited text no. 7
[PUBMED]  [Full text]  
8.
Ahmed AA, van de Sande W, Fahal AH. Mycetoma laboratory diagnosis: Review article. PLoS Negl Trop Dis 2017;11:e0005638.  Back to cited text no. 8
    
9.
Nenoff P, van de Sande WW, Fahal AH, Reinel D, Schöfer H. Eumycetoma and actinomycetoma – An update on causative agents, epidemiology, pathogenesis, diagnostics and therapy. J Eur Acad Dermatol Venereol 2015;29:1873-83.  Back to cited text no. 9
    
10.
Damle DK, Mahajan PM, Pradhan SN, Belgaumkar VA, Gosavi AP, Tolat SN, et al. Modified Welsh regimen: A promising therapy for actinomycetoma. J Drugs Dermatol 2008;7:853-6.  Back to cited text no. 10
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]
 
 
    Tables

  [Table 1], [Table 2]



 

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