Perianal actinomycetoma experience of 20 cases

© 2002

The International Society of Dermatology International Journal of Dermatology

2002,

41

, 491–493

491

Abstract

Background

Actinomycetoma is a chronic infection resulting from aerobic

Actinomycetes

.

The major agents are

Nocardia brasiliensis, Actinomadura madurae

, and

Streptomyces

somaliensis

. The most frequent topographies are the lower and upper limbs. The prognosis of

this disease is determined by several factors, such as etiologic agent, clinical topography, and

depth of disease (degree of involvement, visceral, and bone affection). The purpose of this

paper was to present our experience with actinomycetoma of the perianal region.

Methods

This study comprises 20 cases of perianal actinomycetoma, all of which were clinically

and microbiologically proven by direct examinations, cultures, and biopsies. Clinical responses

to the two principal treatment regimes used [combination of trimethoprim-sulfamethoxazole

(TMS/SMX) and diaminodiphenylsulfone (DDS) or amikacine plus TMS/SMX] are reported.

Results

Most of the cases were male (17/20, 85%), the mean age was 42.1 years, and the

farmers predominated (90%). The principal etiologic agent isolated was

N. brasiliensis

(85%).

Conclusions

Perianal actinomycetoma is a rare entity. Differential diagnosis with anal

sinuses, hydroadenitis, and cutaneous tuberculosis must be made in endemic areas by

performing mycologic tests and biopsies. Treatment depends on the etiologic agent involved

and the patient’s condition.

Blackwell Science, LtdOxford, UKIJDInternational Journal of Dermatology0011-9059Blackwell Science Ltd, 200241

Report

Perianal actinomycetomaChávez et al.

Perianal actinomycetoma experience of 20 cases

Guadalupe Chávez,

MD

, Roberto Estrada,

MD

, and Alexandro Bonifaz,

MB

From the Hospital General de Acapulco, Guerrero, and Hospital General de México, México City, México

Correspondence

Dr A. Bonifaz, Zempoala 60–101, Narvarte, CP 03020, México. E-mail: [email protected]

Introduction

Mycetoma is a chronic subcutaneous infection caused bytwo etiologies: true fungi (eumycetoma) and aerobic actino-mycetes (actinomycetoma), with the latter being the mostfrequent in Mexico. Its major causal agents are

Nocardiabrasiliensis

(85%) and

Actinomadura madurae

(10%). Itis considered to be an occupational disease, more frequentamong individuals involved in agricultural activities. Themost frequent clinical topography is the lower limbs, affectingmainly the feet and legs, and, to a lesser extent, the upperextremities (hands and arms); however, it may involve otherareas such as the back or head. The prognosis of mycetoma isdetermined by three factors: the etiologic agent, the anatomicarea involved, and the depth of involvement (bone andvisceral involvement). The purpose of this paper was to reportour experience with actinomycetomas in the perianal region,focusing on the clinical and microbiologic aspects.

1–5

Material and Methods

This paper included 12 cases of proven actinomycetoma from the

Dermatology Department of the General Hospital in Acapulco,

Guerrero, and eight cases from the Dermatology Department of

the General Hospital in Mexico City (part of the Ministry of Health).

The complete clinical history of each patient was obtained, and

all patients underwent a mycologic test consisting of direct

examinations with lugol solution in an attempt to observe and

classify the type of granule (grain). Cultures using the usual

Sabouraud dextrose agar, with isolated microorganisms, were

identified by their macroscopic and microscopic characteristics

and from biochemic tests. X-rays were taken to determine the

degree of bone involvement, and in some cases a biopsy was also

performed. A sample for bacteriologic culture was collected from

each patient to determine the associated bacterial flora.

A treatment regime was instituted for each patient. Eighteen

patients received 50–100 mg/day diaminodiphenylsulfone (DDS)

plus 80/400–160/800 mg/day trimetoprim-sulfamethoxazole

(TMS/SMX) at variable times. Two patients received two different

regimens: the first patient was given 15 mg/kg/day amikacin

(1 g/day) for three 21-day cycles with equal resting periods, plus

80/400 mg/day TMS/SMX. The second case (caused by

A. madura

)

was given 1 g streptomycin every other day to a total dose of 50 g,

plus 100–200 mg/day DDS.

Results

Of the 20 proven cases of perianal actinomycetic mycetomathe youngest patient was 21 years old and the oldest 68 (meanage = 42.1 years); 17 were male (85%) and only three were

IJD_1550.fm Page 491 Wednesday, August 14, 2002 8:40 AM

International Journal of Dermatology

2002,

41

, 491–493 © 2002

The International Society of Dermatology

492 Report

Perianal actinomycetoma

Chávez

et al.

female (15%). The majority were farmers devoted to agricul-tural activities such as the sowing and growing of variouscrops. Although the three females with the disease did housework, they were also involved in agricultural activities. There-fore, 18/20 (90%) cases were farmers. One patient had a jobin the city but came from a rural area, and another one was astudent in Mexico City (Table 1). It is important to state thatout of the 18 patients who came from rural areas, 14 reportedthat they defecated outdoors and sporadically wiped theirrectums with different objects, such as herbs, leaves, grainlesscorn cobs, branches, etc. Only two patients reported traumain the perianal area. The majority of the patients (16/20) camefrom the state of Guerrero (on the South Pacific coast ofMexico, 300–400 km from Mexico City) and the others camefrom nearby states (Morelos and Oaxaca).

In all cases the diagnosis of mycetoma was confirmedby mycologic and histopathologic examinations. The causalagent was isolated and identified in 18/20 cases. Of the isolatedcausal agents, 17/18 were

N. brasiliensis

and one was

A.madurae

; the remaining two cases were mycetomas with

Nocardia

-like granules. In the six cases in which histopathologywas performed the finding was nonspecific granuloma withabundant

Nocardia

-like, slightly basophilic granules. (Fig. 1)Associated bacterial flora was found in 8/20 cases;

Staphy-lococcus aureus

was identified in seven of these; and

Pseu-domonas aeruginosa

in one case.Regarding the treatment regimes, the 18 cases caused by

N. brasiliensis

(or with

Nocardia

-type granules) were givenDDS + TMS/SMX, with a variable length of treatment(usually until clinical cure was achieved). The earliest case torespond to treatment was after 6 months, while the latest caseresponded after 4.5 years; the mean treatment period was2.2 years. Only one extensive case caused by

N. brasiliensis

was given amikacin + TMS/SMX for three cycles, and oncetreatment with the former drug was complete, the patientreceived maintenance therapy with TMS/SMX for a further1.5 years. The case with mycetoma resulting from

A. madurae

received 50 g streptomycin (total dose), plus 100–200mg/day DDS; the latter drug was continued as maintenancetherapy at a tapered dose for 2.0 years. It is important tomention that five patients who were started on the first regimediscontinued therapy and were started on the first regimendiscontinued therapy and were lost to follow up, so theirresponses are unknown (Table 2).

Discussion

Actinomycetoma is a relatively common disease in Mexico,accounting for approximately 50% of subcutaneous mycoses;its major causal agent is

N. brasiliensis

and the main topo-graphy is the lower limbs.

3,4

This paper intends to highlightanother clinical topography which, based on our overall dataon mycetoma, accounts for 3.5% and is therefore importantto consider, especially as other morphologically similarconditions may occur at this location. We believe that the

Table 1 Demographic data

Age range: 21–68 years (mean = 42.1) Number of cases (%)

GenderMale 17 (85%)Female 3 (15%)

OccupationFarmer 15Housewife & farmer 3Worker 1Student 1

EtiologyNocardia brasiliensis 17Actinomadura madurae 1Not isolated* 2

Histopathology (biopsy)Unspecific granuloma plus Nocardia granules 6Not performed 12

*With Nocardia granules.

Figure 1 Direct examination of granule of Nocardia species (×40)

Table 2 Therapeutic results

Treatment regimen Result Cases (n)

TMS/SMX + DDS Cured 11Improved 2Unknown 5

Amikacin + TMS/SMX Cured 1Streptomycin + DDS Cured 1

TMS/SMX = Trimetoprim sulfamethoxazole; DDS = Diaminodiphenylsulfone; Unknown = patients who were lost to follow up.


© 2002

The International Society of Dermatology International Journal of Dermatology

2002,

41

, 491–493

493

Chávez

et al. Perianal actinomycetoma

Report

most important differential diagnoses should be hydrosoad-enitis (hydroadenitis), tuberculosis, and, mainly, anorectalsinuses. Thus, it is important to perform mycologic tests torule out mycetoma on every occasion in which any of thethree aforementioned conditions is suspected, particularly inpatients from endemic areas. (Fig. 2)

The occurrence of perianal mycetoma may be explained bydirect trauma, as reported by two of our patients. However,most important is the fact that upon wiping the rectum withvarious vegetables, many farmers may cause repeated smalltrauma in that area, leading to the inoculation of microorgan-isms. Most patients report this habit. The case of the studentwho had been born and had always lived in Mexico City,which is not an endemic area, is worth mentioning. This is acase we have published previously: the patient reported beinghomosexual and had the habit of introducing various objectsinto the anus, such as carrots, cucumbers, and radishes; wethink that this is a possible explanation for the disease, as allof these vegetables are tubercles that grow underground andtherefore must be rich in actinomycetes and fungi.

6

It should be emphasized that the perianal location not onlyrenders the treatment of mycetoma more difficult, as not onlymay the causal agents easily spread to the whole pelvic cavityand affect several organs such as the lower gastrointestinaltract and genitals, etc., but it also facilitates bacterial super-infection because of its proximity to the anus. This type ofbacteria usually decreases considerably once treatment hasbeen started, and only rarely does it become necessary toadminister an additional antibacterial agent. (Fig. 3)

As this paper is a collection of our experiences within acertain period of time, it does not intend to prove the efficacyof any treatment regime. The traditional DDS + TMS regimegenerally provides a good outcome.

7–9

However, we want toemphasize that, subjectively, treatment of perianal mycetomarequires more time, especially when comparing it to mycetomas

in other locations (i.e. feet, hands). It should be noted thatmost of the patients were cured, and that the few who showedclinical and mycologic improvement could be treated withother regimens such as amikacin + TMS/SMX or amoxicillin-clavulanic acid + DDS.

9–11

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Figure 2 Perianal actinomycetoma as a result of Nocardia brasiliensis

Figure 3 Perineal and perianal actinomycetoma as a result of Nocardia brasiliensis


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Perianal actinomycetoma experience of 20 cases