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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 3  |  Issue : 3  |  Page : 94-96

Combined welsh and modified ramam regimen in the management of an actinomycetoma patient with sulfonamide drug sensitivity


Department of Dermatology, AIIMS, Raebareli, Uttar Pradesh, India

Date of Submission29-Dec-2021
Date of Decision05-Feb-2022
Date of Acceptance05-Feb-2022
Date of Web Publication03-Nov-2022

Correspondence Address:
K Geetha
Department of Dermatology, AIIMS, Raebareli, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jascp.jascp_33_21

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  Abstract 


Mycetoma is a chronic, suppurative granulomatous infection which has been declared as a neglected tropical disease by the World Health Organization. The clinical manifestations include the classical triad of swelling, sinus tracts, and discharge. It is of two types, eumycetoma (true fungi) or actinomycetoma (filamentous aerobic bacteria), and is more prevalent in tropical and subtropical areas. Here is a case report of mycetoma patient who presented to the outpatient department with the classical triad of symptoms. Histopathological confirmation of actinomycetoma was made and started with Welsh regimen. As he developed drug sensitivity to cotrimoxazole, it was stopped. He was continued with amikacin as per Welsh regimen along with doxycycline 100 mg twice daily instead of cotrimoxazole for 3 cycles with complete cure. Regular monitoring with hemogram, renal function tests, and audiogram were done before and after each cycle. There are multiple reports of treatment of actinomycetoma by various antibiotics such as cotrimoxazole, dapsone, streptomycin, trimethoprim, rifampicin, and amoxicillin–clavulanic acid combination. This case report highlights the importance of individualization of treatment regimen based on the clinical scenario.

Keywords: Actinomycetoma, doxycycline, Welsh regimen


How to cite this article:
Geetha K. Combined welsh and modified ramam regimen in the management of an actinomycetoma patient with sulfonamide drug sensitivity. J Appl Sci Clin Pract 2022;3:94-6

How to cite this URL:
Geetha K. Combined welsh and modified ramam regimen in the management of an actinomycetoma patient with sulfonamide drug sensitivity. J Appl Sci Clin Pract [serial online] 2022 [cited 2022 Dec 6];3:94-6. Available from: http://www.jascp.org/text.asp?2022/3/3/94/360447




  Introduction Top


Mycetoma is an endemic disease in many developing countries and is caused by actinomycetes or filamentous fungi with the involvement of underlying fascia and bones. It has been commonly reported among farmers and field workers, who walk barefoot as the causative organisms are mainly present in the soil and thorny vegetation. The most common causative organisms of actinomycotic mycetoma are aerobic bacteria like Nocardia brasiliensis, Actinomadura madurae, Actinomadura pelletieri, and Streptomyces somaliensis, whereas eumycotic mycetoma is mainly caused by true fungi Madurella Mycetomatis.[1]

The disease course starts with subcutaneous swelling, which gradually enlarges with involvement of the deeper tissues, which connects to the surface through multiple sinuses that may discharge fluid containing grains. These granules or grains are microcolonies of causative agents mainly used in differentiating the two types.[2] Grains may vary in size, color, and microscopic features based on the causative organism. Grains in actinomycetoma are white, yellow, and red colored, but in eumycetoma, mostly black and rarely white or yellow in color. Mycetoma mostly occurs on the feet and hands in almost nearly 84% of cases followed by involvement of trunk, legs, perineum, head, neck, and other sites. It usually presents with secondary bacterial infection by Staphylococcus aureus or Streptococcus pyogenes. The lesions can be graded as small when the involvement is <5 cm without bone involvement, moderate when the size is 5 cm to 10 cm with bone involvement, and massive if the size is >10 cm with bone involvement and secondary bacterial infection. This grading is usually taken into consideration for deciding the therapeutic part.[3]


  Case Report Top


A 48-year-old male, farmer by occupation, came to our skin outpatient department with complaints of swelling with multiple discharging sinuses over the dorsal, medial, and lateral aspect of the right foot of 2 years duration. It started as a single asymptomatic nodule over the dorsal aspect of the right foot that gradually enlarged in size with the development of multiple painful discharging sinuses after few months. He had taken multiple courses of antibiotics and analgesics for temporary relief on and off. On examination of the right foot, there was hardened swelling occupying the dorsal, medial, and lateral sides with multiple discharging nodules. The regional lymph nodes were not enlarged. Gross examination of the discharge showed the presence of yellow granules. X-ray of the right foot showed no bony abnormality. The patient history and physical findings were suggestive of mycetoma. Histological examination revealed suppurative granulomas in the subcutaneous tissue. The diagnosis of actinomycotic mycetoma was made based on the appearance of Gram-positive fine thread-like branching bacteria in the Gram-stained smear and a negative finding in the modified Kinyoun's stain with 1% sulfuric acid and Ziehl–Neelsen stain.

He was started treatment with Welsh regimen comprising injection amikacin 15 mg/kg/iv divided into 2 doses along with oral trimethoprim–sulfamethoxazole (TS) (7 and 35 mg/kg/day, respectively) for 21 days. As he developed drug sensitivity to cotrimoxazole after few days, it was stopped. Later, amoxiclav 635 mg two times daily was added. After 10 days of starting amoxiclav, he developed gastric intolerance, following which it was stopped. Then, taking into of modified Ramam regimen, doxycycline 100 mg was added. Then, he was continued with amikacin as per Welsh regimen along with doxycycline 100 mg twice daily instead of cotrimoxazole for 3 cycles with a significant improvement [Figure 1]. Regular monitoring with hemogram, renal function tests, and audiogram were done before and after each cycle.
Figure 1: Actinomycetoma before and after treatment

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  Discussion Top


The various antibiotics that have been used in the treatment of actinomycotic mycetoma includes Sulfonamides DDS (4,4-diaminodiphenyl-sulfone), TS, amikacin sulfate-TS, rifampicin, netilmicin-TS, minocycline, amoxicillin-clavulanate, linezolid, fosfomycin, imipenem, meropenem, and moxifloxacin. Among these, sulfamethoxazole with trimethoprim (co-trimoxazole) is always the gold standard and is being used as first-line therapy either singly or in combination with other drugs such as penicillin, dapsone, or aminoglycoside.[4] Apart from this, there are multiple regimens such as Ramam regimen, modified Ramam, Welsh regimen, and modified Welsh regimen that are found beneficial in the treatment of actinomycetoma [Table 1].[5] Earlier in 1987, Welsh introduced a regimen with good therapeutic response comprising amikacin alone and in combination with TMP-SMX in those patients with resistant actinomycotic mycetoma and with systemic involvement. Damle et al. in 2008 introduced the modified Welsh regimen with added rifampicin.[6],[7] Ramam et al. described a two-step regimen which constituted of an intensive phase with penicillin, gentamicin, and cotrimoxazole for 5–7 weeks, followed by maintenance therapy with amoxicillin and cotrimoxazole continued 5–6 months after clinical remission. It was later modified to gentamicin (1.5 mg/kg IV) plus TMP-SMX (two DS tablets) given twice daily for 4 weeks followed by continuation of TMP-SMX plus doxycycline (100 mg twice daily). This modification has reduced the duration of intensive phase and also made treatment more affordable while maintaining the efficacy. Achieving clinical cure of mycetoma is a major therapeutic challenge and the drug combinations and the number of cycles of Welsh regimen can be modified based on the disease involvement and patient response.[8]
Table 1: Regimens used in actinomycetoma

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Clinical and therapeutic information on this neglected tropical disease is needed to elaborate public health strategies to reduce disease burden. Collecting regional data helps refine diagnostic and treatment protocols for case management. More case series with larger sample sizes and prospective follow-up are necessary to determine the best therapeutic protocol. Environmental studies to determine primary reservoirs of the causative bacteria and case mapping may additionally help in the development of effective preventive strategies and focus efforts on high-burden areas.[9]

Involvement of bones and other viscera can occur if actinomycetoma is left untreated. Patients devoid of osseous involvement generally responds well to medical therapy. In general, the cure rate for actinomycetoma treated with medication varies from 60% to 90%. Treatment must be adequately monitored with laboratory investigations to rule out drug toxicity.[10] The treatment regime can be modified and individualized as per the clinical presentation of the patient for providing a better safety and efficacy.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Agarwal P, Jagati A, Rathod SP, Kalra K, Patel S, Chaudhari M. Clinical features of mycetoma and the appropriate treatment options. Res Rep Trop Med 2021;12:173-9.  Back to cited text no. 1
    
2.
Tilak R, Singh S, Garg A, Bassi J, Tilak V, Gulati AK. A case of Actinomycotic mycetoma involving the right foot. J Infect Dev Ctries 2009;3:71-3.  Back to cited text no. 2
    
3.
Scolding P, Fahal A, Yotsu RR. Drug therapy for Mycetoma. Cochrane Database Syst Rev 2018;2018:CD013082.  Back to cited text no. 3
    
4.
Welsh O, Al-Abdely HM, Salinas-Carmona MC, Fahal AH. Mycetoma medical therapy. PLoS Negl Trop Dis 2014;8:e3218.  Back to cited text no. 4
    
5.
Relhan V, Mahajan K, Agarwal P, Garg VK. Mycetoma: An update. Indian J Dermatol 2017;62:332-40.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
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. 6
    
7.
Mathews S, Jadhav R, Reza A, Karim T. Actinomycetoma-the welsh regimen in a rural Indian scenario. Indian J Surg 2012;74:480-2.  Back to cited text no. 7
    
8.
Ramam M, Bhat R, Garg T, Sharma VK, Ray R, Singh MK, et al. A modified two-step treatment for actinomycetoma. Indian J Dermatol Venereol Leprol 2007;73:235-9.  Back to cited text no. 8
[PUBMED]  [Full text]  
9.
Cárdenas-de la Garza JA, Welsh O, Cuéllar-Barboza A, Suarez-Sánchez KP, De la Cruz-Valadez E, Cruz-Gómez LG, et al. Clinical characteristics and treatment of actinomycetoma in northeast Mexico: A case series. PLoS Negl Trop Dis 2020;14:e0008123.  Back to cited text no. 9
    
10.
Arenas R, Fernandez Martinez RF, Torres-Guerrero E, Garcia C. Actinomycetoma: An update on diagnosis and treatment. Cutis 2017;99:E11-5.  Back to cited text no. 10
    


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