Jpn. J. Med. Mycol. Vol. 43, 95 - 98, 2002 ISSN 0916-4804

Original Article

A Case of Confluent and Reticulated Papillomatosis

Responsive to Ketoconazole Cream

Taizo Hamaguchi 1, Machiko Nagase 2, Ryota Higuchi 2,

Iwao Takiuchi 2

1 Department of Dermatology , Showa University, Northern Yokohama Hospital 2 Department of Dermatology, Showa University, Fujigaoka Hospital

(Received: 31, July 2001. Accepted: 23, January 2002)

Abstract

We described here a case of a 43-year-old male who developed confluent and reticulated

papillomatosis (CRP) . The patient was found to be slightly obese and had no family history of such eruption. Numerous small red-brown erythemas were scattered over a wide area of the back and, in

many areas, the erythemas coalesced and formed a reticular pattern. The eruptions appeared 10 days

prior to the initial visit to our outpatient clinic. The Parker-KOH preparation of scraped scales revealed numerous round and budding non-clustering cells and no mycelial elements. Histological

examination showed subtle papillomatosis and sparse perivascular lymphohistiocytic infiltrations. Periodic acid schiff stain showed a few spores in the stratum corneum. Topical application of 2 %

ketoconazole cream produced complete resolution of the eruption in 7 days. The course and histological findings of our patient suggest the eruptions were developing CRP

lesions. Application of topical antifungal agents appears to be a beneficial initial treatment for early

CRP lesions. Key words: confluent and reticulated papillomatosis, Malassezia furfur, topical antifungal therapy

INTRODUCTION

Confluent and reticulated papillomatosis (CRP) is a distinct condition first described by Gougerot and Carteaud in 19271. Several causal factors have been postulated in the literature including endocrine disturbance 2~ and an abnormal response to the yeast Malassezia 3) . As the etiology of CRP remains unknown, various therapeutic modalities have been reported.

CASE REPORT

A 43-year-old Japanese male presented with eruptions on the back. The eruptions had developed 3-4 years earlier. The condition was exacerbated in summer, but cleared completely and spontaneously in winter. The patient had not previously sought medical treatment. The eruptions had reappeared 10 days before visiting

our hospital. Upon initial examination, he was found to be slightly obese (height, 168 cm; weight, 75 kg) and he had no family history of such eruption.

Numerous small red-brown erythemas were scattered over a wide area of the back. In many areas, erythemas coalesced and formed a reticular

pattern (Fig. 1) . The Parker-KOH-mounted examination revealed that the scales scraped from the erythemas had numerous round and budding cells without hyphae (Fig. 2). Histological examination of biopsy specimens taken from the lesions showed subtle papil-lomatous epidermis and compact hyperkeratosis. Only sparse lymphohistiocytic perivascular infiltrations were observed (Fig. 3) . Periodic acid schiff (PAS) stain showed a few spores in the stratum corneum.

Treatment with 2 % ketoconazole cream was

initiated after initial examination. The eruptions

began to clear up within a few days, with com-

plete clearance in 7 days (Fig. 4).

Address correspondence to: Iwao Takiuchi

Department of Dermatology, Showa University Fujigaoka

Hospital, 1-30 Fujigaoka, Aoba-ku, Yokohama, 227-8501 Japan.

96 真菌誌 第43巻 第2号 平成14年

DISCUSSION

Despite its clinically distinct appearance, CRP reveals only nonspecific papillomatosis and hyperkeratosis. As for the demographics of this disorder, a mean age at onset of 21 years') and 19 years'), and a female : male ratio of 2.5: 1 have been reported2). In addition, a considerable pro-

portion of CRP patients are obese, with or with-out endocrine disturbance. The eruptions of CRP are typically small hyperkeratotic erythematous

papules that coalesce to form reticular patterns that are predominantly localized at the mid-trunk. Diagnosis can be made from the distribu-tion and pattern of the eruption. Lee et al. 4l defined CRP according to the following features:

(1) hyperpigmented papules and plaques at least the central portion of the chest or back with confluence centrally and reticulation at the

periphery, (2) no demonstrable hyphae on either KOH preparation or histological testing, and (3)

papillomatosis, hyperkeratosis and acanthosis often with hypogranulosis and a sparse superficial

perivascular lymphocytic infiltrate histopatho-logically. Differential diagnosis of CRP must account for

prurigo pigmentosa, benign acanthosis nigricans, pseudoacanthosis nigricans, and tinea versicolor (TV). Prurigo pigmentosa is morphologically

similar to CRP, having reticulated hyperpig-

mented papules and plaques involving the central

portion of the chest and/or back. Prurigo

pigmentosa can be differentially diagnosed by

histologic findings and severe pruritus. The clini-

cal manifestation of velvety hyperpigmented

papules and plaques in a confluent pattern may

also mimic benign acanthosis nigricans and

pseudoacanthosis nigricans. The lack of involve-

ment of typically localized areas, such as the pos-

terior neck, axillae, and inguinal space,

distinguish CRP from benign acanthosis nigricans

and pseudoacanthosis nigricans.

Aggravation of the present patient's lesions in

the summer and his quick response to the topical

application of antifungal cream are disease char-

acteristics shared by TV and CRP. We were able

to make a final diagnosis of CRP because the re-

sponse to antifungal cream was somewhat quicker

Fig. 1. Clinical features prior to treatment

The patient presented with red-brown erythema scattered

across the upper back.

Fig. 2. Direct examination using the Parker KOH preparation

Large numbers of round and budding cells without hyphae

were apparent.

Fig. 3. Biopsy specimen of the lesion

Subtle papillomatous epidermis, hyperkeratosis and slight

lymphohistiocytic infiltrations of the dermis were observed.

Fig. 4. Clinical features posttreatment

Jpn. J. Med. Mycol. Vol. 43 (No. 2 ), 2002 97

than that expected in cases of TV, and no mycelial elements or numerous round and bud-

ding non-clustering cells were found on Parker-KOH mounting in our patient. Clusters of round

and budding cells and angular fragments of mycelial elements in the scales are consistent

findings of TV. Typically both spores and mycelial elements are found in PAS stain of the

biopsy specimen taken from TV lesions. On the other hand, PAS stain showed only a small num-

ber of spores in the case described. Roberts and Lachapelle 3) and Yesudian et al. 5~ reported CRP

cases in which PAS stains showed a small number of spores. The results of PAS stains are

not always correlated with the findings of KOH direct examination in CRP. The number of spores

may be decreased during the tissue processing. In the present case, the distribution and pattern

of eruption was consistent with CRP. However,

the majority of the eruptions were light brown erythema, and papillomatosis was only subtle on microscopic examination. Gougerot and

Carteaud 1) documented the initial eruption of CRP to comprise flat, slightly upheaving, rosy

papules, 2 to 4 mm in diameter, that progressed to brownish verrucous papules with a rough

surface that resembled verruca plana juvenilis. We speculated that the papules with a rough

surface seen in the present patient may corre-spond to the histological features of papil-

lomatosis. Because the individual eruption had

developed only 10 days earlier in this case, the eruption might have not been fully developed. The precise etiology of CRP is still unknown.

Endocrine disturbance, hereditary disorder, abnormal host response to Malassezia species 3, a

variant of amyloidosis 6), and keratinization defect') have all been proposed as possible etiological

factors. Accordingly, various therapeutic modali-ties have been applied to CRP depending on the

etiological hypothesis favored. Isotretinoin 4, 8) minocycline ~), calcipotriol 10), and weight reduc-

tion 11) have recently been reported to effectively treat CRP. Among these, minocycline has

become the most widely applied, attributable mainly to its effectiveness and safety. However,

none of the existing treatments have been

uniformly effective. The clinical resemblance and even coincidence between CRP and TV has led several authors 3, 5)

to propose that the Malassezia species may play a role in the development of CRP lesions.

Topical and systemic antifungal therapies including selenium sulfide 12), miconazole 13,14],

tolnaftate 15), and itraconazole 16) have produced

positive outcomes, but even in those cases in

which treatment was effective, the response was

limited and there was a high recurrence rate. Moreover, in the majority of the cases,

antifungals did not offer any benefits. Nordby 12} reviewed the treatment of reported CRP cases

with antifungals and concluded there was in fact no correlation between the KOH findings and

therapeutic response. Although recent findings do not support the Malassezia theory, in a small

number of the cases 3' 5,14) antifungal application completely eliminates CRP lesions as it did in

the present case. The course and histological findings of our patient suggest the eruptions were

developing CRP lesions, and we felt it prudent to start topical antifungal therapy, because not only it is safe but it also has only limited effects.

We conclude that application of topical

antifungal agents is appropriate as an initial treatment of early lesions of CRP and that it is,

at the present time, the most effective treatment for this condition. Physicians should consider use

of these agents in planning treatment planning for CRP.

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98 真菌誌 第43巻 第2号 平成14年

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