I
MYCOSES
35, 197-199 (1992)
ACCEPTED: JULY 30, 1991
CASEREPORT
Dermatophyte infection in ichthyosis vulgaris *
Dermatophytose bei Ichthyosis vulgaris” G. Agostini, V. Geti, Elisa M. Difonzo and B. Giannotti Key words. Tinea pedis, tinea manuum, dermatophytosis, ichthyosis vulgaris. Schlusselworter. Tinea pedis, Tinea manuum, Dermatophytose, Ichthyosis vulgaris.
Summary. A 31-year-old male who had been suffering from ichthyosis vulgaris developed hyperkeratosic lesions on the palms and soles and ungual alterations of both hands and feet. These lesions were resistant to topical corticosteroid and emollient cream treatments and were found to be caused by Trichophyton rubrum. Association of ichthyosis vulgaris and dermatophytosis- both very common disorders-is to our knowledge a very rare event.
garis since the age of one. He reported some improvement in his condition after puberty: only lesions on the trunk and lower limbs (Fig. 1) remained, along with widespread “dryness” of the skin. This was more severe during the winter, and more evident on the palms and soles where
Zusammenfassung. Bei einem 3 1-jahrigen Patienten mit Ichthyosis vulgaris waren seit einigen Jahren hyperkeratotische Veranderungen der Palmae und Plantae sowie Nagelanomalien aufgetreten. Die Behandlung mit topischen Kortikosteroiden und Fettsalben lienen die Lasionen unverandert. Die mykologischen Untersuchungen fuhrten zur Isolierung von Trichophyton rubrum. Das gleichzeitige Auftreten von Ichthyosis vulgaris und Dermatophytosen (beide haufige Krankheiten) ist nach unserem Wissen sehr selten. Case report Antonio, a 3 I-year-old office worker whose hobby is tennis, had been suffering from ichthyosis vulDepartment of Dermatology, University of Florence, Florence, Italy. Correspondence: Prof. Dr Elisa Margherita Difonzo, Istituto di Clinica Dermosifilopatica, Via degli Alfani 37, 1-50121 Firenze, Italy. *Paper presented at the XIth Congress of ISHAM, Montreal, Canada, 1991.
Figure 1. Ichthyosis vulgaris lesions on the lower limbs: brownish scales, adherent at the centre.
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G. ACOSTINI ET AL.
the grooves were deeper (“ichthyotic hand and foot”). Greyish hyperkeratotic areas had developed 5 years earlier on his palms and soles (Figs 2 and 3). The family physician diagnosed the lesions as palmo-plantar ichthyosis and treated them, unsuccessfully, with topical corticosteroids and emollient creams. For 3 years the patient had also noticed changes in the nails of the 1st and 3rd finger of the left hand, and 1st and 2nd toe of the left foot (Fig. 4);the free edges of the nails appeared destroyed and there was an accumulation of friable keratinic matter. When the patient came under our observation, the clinical picture of the lesions, and especially the nails, was highly suggestive of a dermatophytic infection and, therefore, mycological tests were performed. Laboratory results
Direct microscope examination, after maceration in 20% KOH, revealed numerous septated and
Figure 4. Nail involvement with distal onychomycosis.
branched filaments in matter taken from the palms, soles and the infected nails. The culture test on Sabouraud glucose agar with penicillin G, streptomycin and actidione resulted in the isolation of Trichophyton rubrum whereas the mycological tests on scales taken from the trunk and lower limbs were negative. Routine blood chemistry was normal. Treatment and clinical course
Treatment was initiated with itraconazole (100 mg per day). Within 40 days the lesions on the palms and soles were cured and significant improvement of the nails was achieved. The treatment was suspended because the patient complained of gastro-intestinal intolerance (nausea). He continued topical (bifonazole) treatment for the nail infection for 2 months and this proved successful. Figure 2. Hyperkeratosis of the palms, with cracks and fissures more evident on the left hand; note the marked grooves.
Discussion
Figure 3. Bilateral hyperkeratosis of the soles.
The modified epidermal turnover with thickening of the horny layer, qualitative and quantitative changes in surface lipids, functional changes in the sebaceous and sudoriferous glands and other major local defence factors that we can observe in patients with ichthyosis vulgaris and other congenital forms of ichthyosis seem to favour the onset of superficial fungus, and especially dermatophytic infections. To our knowledge and experience [ 1-31, findings of dermatophytosis during the course of ichthyosis are relatively rare, in spite of the fact that ichthyosis vulgaris is a very common disease [4]. I n the past 6 years we have examined 2085 patients with dermatophytoses: only 3 of these mycoses 35, 197- 199 ( 1992)
DERMATOPHYTOSIS IN ICHTHYOSIS
had ichthyosis vulgaris and presented hyperkeratotic tinea pedis. On the other hand, there are many reports of associations between tinea pedisltinea manuum and other keratinization disorders, especially hereditary palmo-plantar keratoderma [5-81. In these cases, the most frequently isolated aetiological agent is Trichophyton mentagrophytes which seems to have a high keratinase activity [9]. Epidermal changes of ichthyosis vulgaris do not seem to predispose for fungal infections. In the case of infection, however, the marked hyperkeratosis of ichthyotic origin seems to prevent intensive inflammation. Therefore, dermatophyte infection of ichthyotic palms and soles is difficult to treat and diagnose.
2 3
4
5 6
7
References
8 9
1 Koblenzer, P. J. & Miller, M. E. (1972) Lamellar ichthyosis, recurrent Trichophyton rubrum infections; familial
199
abnormality of chemotaxis of polymorphonuclear leukocytes. Arch. Dennatol. 106, 755-756. Kamalam, A. & Thambiah, A. S. (1982) Genetic ichthyosis and Trichophyton rubrum infection in infants. Mykosen 25, 281-283. Shelley, E. D., Shelley, W. B. & Schafer, R. L. (1989) Generalized Trichophyton rubrum infection in congenital ichthyosiform erythroderma. 3. Am. Acad. Dennatol. 20, 1133-1 134. Ebling, F. J. G., Marks, R. & Rook, A. (1986) Ichthyosis vulgaris. In: Rook, A., Wilkinson, D. S., Ebling, F. J. G., Champion, R. H. & Burton, J. L. (eds) Textbook of Dermatology. 4th edn. Oxford: Blackwell Scientific Publications, pp. 1416-1418. Nielsen, P. G. (1984) Dermatophyte infections in hereditary palmo-plantar keratoderma. Dennatologica 168, 238-241. Nielsen, P. G. (1984) The importance of the vehicle in the treatment of dermatophytosis in hereditary palrnoplantar keratoderma. Mykosen 27, 227-230. Nielsen, P. G. (1985) Skin pH on soles in patients with hereditary palmoplantar keratoderma and pathogenicity of dermatophytes. Mykosen 28, 3 10-312. Nielsen, P. G. (1988) Hereditary palmoplantar keratoderma and dermatophytosis. Int. 3. Dermatol. 27,223-229. Yu, R. J., Harmon, S. R. & Blank, F. (1969) Isolation and purification of an extracellular keratinase of Trichophyton mentagrophytes. 3. Bactm'ol. 96, 1435- 1436.
MYCOSES
35, 197-199 (1992)
ACCEPTED: JULY 30, 1991
CASEREPORT
Dermatophyte infection in ichthyosis vulgaris *
Dermatophytose bei Ichthyosis vulgaris” G. Agostini, V. Geti, Elisa M. Difonzo and B. Giannotti Key words. Tinea pedis, tinea manuum, dermatophytosis, ichthyosis vulgaris. Schlusselworter. Tinea pedis, Tinea manuum, Dermatophytose, Ichthyosis vulgaris.
Summary. A 31-year-old male who had been suffering from ichthyosis vulgaris developed hyperkeratosic lesions on the palms and soles and ungual alterations of both hands and feet. These lesions were resistant to topical corticosteroid and emollient cream treatments and were found to be caused by Trichophyton rubrum. Association of ichthyosis vulgaris and dermatophytosis- both very common disorders-is to our knowledge a very rare event.
garis since the age of one. He reported some improvement in his condition after puberty: only lesions on the trunk and lower limbs (Fig. 1) remained, along with widespread “dryness” of the skin. This was more severe during the winter, and more evident on the palms and soles where
Zusammenfassung. Bei einem 3 1-jahrigen Patienten mit Ichthyosis vulgaris waren seit einigen Jahren hyperkeratotische Veranderungen der Palmae und Plantae sowie Nagelanomalien aufgetreten. Die Behandlung mit topischen Kortikosteroiden und Fettsalben lienen die Lasionen unverandert. Die mykologischen Untersuchungen fuhrten zur Isolierung von Trichophyton rubrum. Das gleichzeitige Auftreten von Ichthyosis vulgaris und Dermatophytosen (beide haufige Krankheiten) ist nach unserem Wissen sehr selten. Case report Antonio, a 3 I-year-old office worker whose hobby is tennis, had been suffering from ichthyosis vulDepartment of Dermatology, University of Florence, Florence, Italy. Correspondence: Prof. Dr Elisa Margherita Difonzo, Istituto di Clinica Dermosifilopatica, Via degli Alfani 37, 1-50121 Firenze, Italy. *Paper presented at the XIth Congress of ISHAM, Montreal, Canada, 1991.
Figure 1. Ichthyosis vulgaris lesions on the lower limbs: brownish scales, adherent at the centre.
198
G. ACOSTINI ET AL.
the grooves were deeper (“ichthyotic hand and foot”). Greyish hyperkeratotic areas had developed 5 years earlier on his palms and soles (Figs 2 and 3). The family physician diagnosed the lesions as palmo-plantar ichthyosis and treated them, unsuccessfully, with topical corticosteroids and emollient creams. For 3 years the patient had also noticed changes in the nails of the 1st and 3rd finger of the left hand, and 1st and 2nd toe of the left foot (Fig. 4);the free edges of the nails appeared destroyed and there was an accumulation of friable keratinic matter. When the patient came under our observation, the clinical picture of the lesions, and especially the nails, was highly suggestive of a dermatophytic infection and, therefore, mycological tests were performed. Laboratory results
Direct microscope examination, after maceration in 20% KOH, revealed numerous septated and
Figure 4. Nail involvement with distal onychomycosis.
branched filaments in matter taken from the palms, soles and the infected nails. The culture test on Sabouraud glucose agar with penicillin G, streptomycin and actidione resulted in the isolation of Trichophyton rubrum whereas the mycological tests on scales taken from the trunk and lower limbs were negative. Routine blood chemistry was normal. Treatment and clinical course
Treatment was initiated with itraconazole (100 mg per day). Within 40 days the lesions on the palms and soles were cured and significant improvement of the nails was achieved. The treatment was suspended because the patient complained of gastro-intestinal intolerance (nausea). He continued topical (bifonazole) treatment for the nail infection for 2 months and this proved successful. Figure 2. Hyperkeratosis of the palms, with cracks and fissures more evident on the left hand; note the marked grooves.
Discussion
Figure 3. Bilateral hyperkeratosis of the soles.
The modified epidermal turnover with thickening of the horny layer, qualitative and quantitative changes in surface lipids, functional changes in the sebaceous and sudoriferous glands and other major local defence factors that we can observe in patients with ichthyosis vulgaris and other congenital forms of ichthyosis seem to favour the onset of superficial fungus, and especially dermatophytic infections. To our knowledge and experience [ 1-31, findings of dermatophytosis during the course of ichthyosis are relatively rare, in spite of the fact that ichthyosis vulgaris is a very common disease [4]. I n the past 6 years we have examined 2085 patients with dermatophytoses: only 3 of these mycoses 35, 197- 199 ( 1992)
DERMATOPHYTOSIS IN ICHTHYOSIS
had ichthyosis vulgaris and presented hyperkeratotic tinea pedis. On the other hand, there are many reports of associations between tinea pedisltinea manuum and other keratinization disorders, especially hereditary palmo-plantar keratoderma [5-81. In these cases, the most frequently isolated aetiological agent is Trichophyton mentagrophytes which seems to have a high keratinase activity [9]. Epidermal changes of ichthyosis vulgaris do not seem to predispose for fungal infections. In the case of infection, however, the marked hyperkeratosis of ichthyotic origin seems to prevent intensive inflammation. Therefore, dermatophyte infection of ichthyotic palms and soles is difficult to treat and diagnose.
2 3
4
5 6
7
References
8 9
1 Koblenzer, P. J. & Miller, M. E. (1972) Lamellar ichthyosis, recurrent Trichophyton rubrum infections; familial
199
abnormality of chemotaxis of polymorphonuclear leukocytes. Arch. Dennatol. 106, 755-756. Kamalam, A. & Thambiah, A. S. (1982) Genetic ichthyosis and Trichophyton rubrum infection in infants. Mykosen 25, 281-283. Shelley, E. D., Shelley, W. B. & Schafer, R. L. (1989) Generalized Trichophyton rubrum infection in congenital ichthyosiform erythroderma. 3. Am. Acad. Dennatol. 20, 1133-1 134. Ebling, F. J. G., Marks, R. & Rook, A. (1986) Ichthyosis vulgaris. In: Rook, A., Wilkinson, D. S., Ebling, F. J. G., Champion, R. H. & Burton, J. L. (eds) Textbook of Dermatology. 4th edn. Oxford: Blackwell Scientific Publications, pp. 1416-1418. Nielsen, P. G. (1984) Dermatophyte infections in hereditary palmo-plantar keratoderma. Dennatologica 168, 238-241. Nielsen, P. G. (1984) The importance of the vehicle in the treatment of dermatophytosis in hereditary palrnoplantar keratoderma. Mykosen 27, 227-230. Nielsen, P. G. (1985) Skin pH on soles in patients with hereditary palmoplantar keratoderma and pathogenicity of dermatophytes. Mykosen 28, 3 10-312. Nielsen, P. G. (1988) Hereditary palmoplantar keratoderma and dermatophytosis. Int. 3. Dermatol. 27,223-229. Yu, R. J., Harmon, S. R. & Blank, F. (1969) Isolation and purification of an extracellular keratinase of Trichophyton mentagrophytes. 3. Bactm'ol. 96, 1435- 1436.
