A Case of Chromomycosis Treated by Surgical Therapy Combined With Preceded Oral Administration of Terbinafine To Reduce The Size of The Lesion

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Tokai J Exp Clin Med., Vol. 37, No. 1, pp.

6-10, 2012

A Case of Chromomycosis Treated by Surgical Therapy Combined


with Preceded Oral Administration of Terbinafine to Reduce the
Size of the Lesion
Kana TAMURA*1, Takashi MATSUYAMA*1, Eiichiro YAHAGI*1,
Tomoko KOJIMA*1, Emiko AKASAKA*1, Akio KONDO*1, Norihiro IKOMA*1,
Tomotaka MABUCHI*1, Shiho TAMIYA*1, Akira OZAWA*1
and Takashi MOCHIZUKI*2
Department of Dermatology, Tokai University School of Medicine
*2
Department of Dermatology, Kanazawa Medical University

*1

(Received May 27, 2011; Accepted November 4, 2011)

Chromomycosis is a chronic fungal disease of the skin and subcutaneous tissues caused by a group of dematiaceous black fungi [1]. Small lesions can be removed with excision, but other cases are difficult to treat.
We report a case of chromomycosis caused by Fonsecaea pedrosoi (F. pedrosoi). The case involved a 74-yearold man, who had noted a lesion on the back of the right thigh, that was gradually enlarging and reaching up
to 30 cm in diameter, in 20-years. From microscopic examination, sclerotic cells were seen. We diagnosed
this case as chromomycosis caused by F. pedrosoi on mycological examination. The patient was initially
treated with oral terbinafine (250mg/day) as the lesion was very large. After the 18 months treatment, the
size of the lesion reduced to 1cm, then the remaining lesion was excised.
Key words: chromomycosis, Fonsecaea pedrosoi, terbinafine, surgery

INTRODUCTION
Chromomycosis is a term applied to infection with
some black dematiaceous fungi and is characterized
by sclerotic pigmented bodies intermediate between
a yeast and hyphal form in the tissues. Infections
have occurred throughout the world. The fungi are
thought to be present in soil, wood and vegetable
debris. Chromomycosis is particularly seen in farmers
and agricultural workers, because the infection occurs
primarily in skin following trauma [2]. Chromomycosis
is treated with surgery, antifungal agents or local
thermotherapy. The preferred treatment of small and
localized lesions is usually surgical excision with wide
surgical margins to prevent local recurrence. However,
surgical treatment is often not feasible for deep lesions
or for those with extensive cutaneous involvement. For
deep or extensive lesions, prolonged treatment with
systemic antifungal agents alone or in combination
provides the best chance of cure [1].
Here, we report a case of a chromomycosis with a
large lesion, which were treated by oral terbinafine to
reduce size and subsequently excised.
CASEREPORT
A 74-year-old fisherman presented with a 20-year
history of a slowly enlarging lesion on the back of his
right thigh. It had been diagnosed as tinea corporis
by local dermatologists and treated with topical ketoconazole and terbinafine. However, the eruption had

enlarged despite treatment, and he was referred to our


hospital.
On clinical finding, there was an annular erythema
approximately 30 30 cm in size. The border of the
ring was 2 cm in width, slightly rose, discontinuous
and accompanied with scales on the surface (Fig. 1a).
The scales showed sclerotic cells in direct mycological exams with 10% KOH (Fig. 2a). Laboratory data
were all within normal limits except for positive HCV
antibody. There were no significant findings on the abdominal ultrasonography, and computed tomography
showed an old cerebral infarction.
The skin biopsy was carried out on the edge of
the erythema. The histological finding was showed
acanthosis, epithelioid cell granuloma including multinucleated giant cells, and neutrophilic infiltration in
the dermis (Fig. 3a). Sclerotic cells were recognized on
PAS staining(Fig. 3b). Examination of the mycological
culture on Sabourauds dextrose agar at room temperature showed growths of heaped and fuzzy colonies
with black surface (Fig. 2b). Slide culture showed the
fungal morphology to be of the Rhinocladiella-type
(Fig. 2c). The ITS-RFLP pattern matched for Fonsecaea
pedrosoi (rDNA type 2) [3]. The diagnosis of chromomycosis was made at by mycological evaluation that
confirmed the presence of fungal cells, identified in
the following culture as Fonsecaea pedrosoi (F. pedrosoi).
Treatment with terbinafine of 250mg/day was started. In 10 weeks, the lesion began to disappear partially.
18 months after the initiation of terbinafine, there was

Kana TAMURA, Department of Dermatology, Tokai University School of Medicine, 143 Shimokasuya, Isehara, Kanagawa 259-1193, JapanTel: +81-463-931121 Fax: +81-463-91-9387Email: kana.m@is.icc.u-tokai.ac.jp

K. TAMURA et al. / A Case of Chromomycosis

c
Fig. 1 Clinical features (on the back
of patient's right thigh)

a First examination

An annular erythema approximately 30 30 cm
in size. The border of the
ring was 2 cm in width,
slightly rose, discontinuous
and accompanied with
scales on the surface.

b 18 months after treatment

Pigmentation and 3 erythematous lesions, each of
1 cm in size, with scaly
surface.

c 3 months after surgical
excision

Postoperative lesion showing no sign of recurrence.

a good clinical response, with mostly pigmentation left


and three erythematous lesions 1-cm in diameter on
the edge of the original lesion (Fig. 1b). At this point,
scales did not show sclerotic cells in direct mycological
examination with 10% KOH. Treatment with terbinafine was continued for a total of 32 months, but there
was little clinical improvement (the period of terbinafine treatment is not exactly accurate as the medication was interrupted during a hospital admission for a
bone fracture and he had been non-compliant at this
period). The remaining lesions we excised with a 5
mm margin. When the excised specimen was cultured,
F. pedrosoi was negative in the pigmented lesion and
positive in the erythematous lesion. There was no clinical recurrence three months after surgical excision (Fig.
1c).
DISCUSSION
Chromomycosis is a subcutaneous mycosis caused
by the dematiaceous fungi Fonsecaea, Phialophora and
Cladophialophora [4]. It is seen worldwide, but is most
commonly seen in tropical and subtropical regions [1].
In Japan, it has been reported that F. pedrosoi is the
most common causative fungus, accounting for 87%

of the disease in this country [5]. It starts as a scaly


papule, often following superficial trauma, which
slowly expands into a verrucous nodule or plaque [6].
The course of the infection is chronic, slowly progressive, and it is often asymptomatic [4].
An Ichushi-Web database (Japan Medical Abstracts
Society, JAMAS, http//www.jamas.or.jp/) search was
conducted using the key word chromomycosis for
papers published from 1997 to 2009 in Japan. As a
result, there have been 64 reports, including our case,
and a total of 35 cases (54.6%) were caused by F.
pedrosoi. From the 35 cases [5, 7-35, 37-39] that was
caused by F. pedrosoi, the male to female ratio of these
cases were 1 to 0.94. The age of patients ranged from
36 to 89 years of age with the greatest number of patients in the 60-69 years age group. Lesions were most
commonly found on the arms (40%).
Oral antifungal agents for chromomycosis include
fluconazole, flucytosine, ketoconazole, terbinafine
and itraconazole. In the 1990
s, terbinafine and itraconazole were the most popular agents. The reported
response rate of these agents in chromomycosis was
60% for itraconazole and 66.7% for terbinafine [36].
As patient in this case was on many other medications

K. TAMURA et al. / A Case of Chromomycosis

Fig. 2 Mycological examination



a First examination.

Microscopic examination (10% KOH, 200)
The scales showed sclerotic cells.

b Examination of the mycological culture on
Sabouraud's dextrose agar at room temperature
for 30 days

Heaped and fuzzy colonies with a black surface.
(8.5 cm dish)

c Slide culture (400)

Fungal morphology to be of the Rhinocladiellatype.

for hepatitis C, cerebral infarction, hypertension; ICTZ


was not selected to avoid potential interaction with
Ca-blocker for hypertension. 1997 was the year when
terbinafine became avilable in Japan. Since 1997, 17
reported cases of chromomycosis have been treated
with terbinafine, of which 9 cases were caused by F.
pedrosoi. Many reported cases did not respond to terbinafine 125mg/day, prompting either a dose increase to
250mg/day, a change in medication, surgical removal
or addition of local thermotherapy [5, 36].
Local thermotherapy has few side effects but
requires prolonged treatment and perseverance of
patients [37]. We did not use local thermotherapy because the patient refused this treatment.
Surgical removal of lesions is considered the most
preferred treatment [38]. It is difficult to surgically
remove large lesions due to the possibility of scarring.
If the lesion is small and localized, we can excise the
lesion with a 5 mm margin [36]. The maximum excised area in past reports was 3.7 3.5 cm in size [39].
At the initial presentation of our case, the lesion was
30 cm in size and thus, the operative risk was high.
Terbinafine at 250mg/day orally was considered to be
the treatment of choice compared with surgical resection, local thermotherapy and itraconazole. As there
was little clinical improvement after 32 months of
terbinafine administration, we excised the remaining
lesion and there has been no evidence of recurrence to
date.
There have been 35 cases of chromomycosis caused
by F. pedrosoi, reported in Japan from 1997 to 2009.

Among 35 reports, 15 cases has been managed surgically, 8 cases treated with only antifungal agent, 6
cases treated by combination of antifungal agent and
local thermotherapy, 2 cases is other treatments and
4 cases are unknown. 11 cases among the surgically
removed 15 cases have showed no recurrence (Table 1).
However, only one case out of the orally treated 8 cases
has been treated successfully.
Chromomycosis recur easily, and is often intractable.
We stopped oral terbinafine 3 weeks after the surgery
because culture of the excised pigmented lesion was
negative. There was no recurrence three months after
surgical excision. Oral administration of terbinafin
250mg/day could reduce in size, however, it could not
completely cure [5, 36, 38]. Monotherapy with orally
administered antifungal agent may not lead to a complete cure. Combination therapy should be considered
taking into account the state of the lesion. Our case
report demonstrated that in the case of large lesions,
oral terbinafine may be administered first to reduce
the size of the lesions prior to surgical removal.
(This report was presented at the 25 th Annual
M e e t i ng of Jap a n O rg a n i z a t i on of C l i n ic a l
Dermatologists)

K. TAMURA et al. / A Case of Chromomycosis

Fig. 3 Histological features



a Acanthosis, epithelioid cell granuloma including multinucleated giant cells and neutrophilic infiltration in the
dermis. (H-E staining, 40)

b Sclerotic cells are found within giant cells. (PAS staining, 400)

Table 1 Summary of Japanese cases of chromomycosis caused by F. pedrosoi which were surgically removed.
Patient
No.

Age

Sex

Site of
Lesion

71

knee

2.1 2.5 cm

69

buttock

13 cm

62

foot

71

cheek

68

waist

6 cm

62

hand

2.5 1.7 cm

59

back

3.0 3.5 cm

87

cheek

1.1 0.8 cm

60

arm

1 cm

10

62

foot

3.6 2.5 cm

11

36

arm

2.0 1.8 cm

12

67

arm

1.5 cm

13

84

buttock

2.0 cm

Size

14

78

arm

3.7 3.5 cm

15

74

thigh

30 30 cm

Preoperation
ND
antifungal
agents
local
thermotherapy
antifungal
agents
local
thermotherapy
ND
antifungal
agents
local
thermotherapy
antifungal
agents
local
thermotherapy

antifungal
agents
local
thermotherapy
antifungal
agents

Not done: ND
Not mentioned:

Postoperation

Efffect

Reference
No.

antifungal
agents
antifungal
agents
local
thermotherapy

no
recurrence

11

12

ND

no
recurrence

13

antifungal
agents

recurrence

14

ND

15

ND

no
recurrence

16

17

no
recurrence
no
recurrence
no
recurrence
no
recurrence
no
recurrence

18

20

antifungal
agents
local
thermotherapy

no
recurrence

22

antifungal
agents

no
recurrence

39

antifungal
agents

no
recurrence

this case

antifungal
agents

antifungal
agents

19

37
21

K. TAMURA et al. / A Case of Chromomycosis


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