A
Disclosure. Financial support: none. Conflict of interest: none.
B
1
Department of Dermatology, Department of Dermatopathology, University Hospital of Geneva, 4 Rue Gabrielle-Perret-Gentil, 1211 Geneva, Switzerland 2
C D
E
F
Figure 1. Psoriasiform hyperplasia of the epidermis with a mild superficial perivascular lymphocytic infiltrate (A) and overlying “geometric” parakeratosis (B). C) follicular lesions coalescing into erythematosquamous plaques on the trunk. D) yellowish-orange color of the palms; E, F) clinical remission after surgery.
tar keratoderma. In 1980, Griffiths proposed a classification of PRP into five types [1]. An HIV-associated type has recently been described [2]. The origin of PRP is unknown, although infections, genetic and metabolic factors have been suggested. It is usually a chronic, long-lasting disease and the drugs classically prescribed for therapy (acitretin, methotrexate, TNF blockers or other biologics) are needed for several months. Clinical remission was achieved in eight adults with methotrexate (oral or injected) at the dose of 10-30 mg/week [3]; by contrast, Allison et al. reported a complete failure in five patients [4]. Associations of PRP with malignancies have been reported, either hematologic (leukemia) or solid (renal-cell carcinoma, hepatocellular carcinoma, laryngeal carcinoma, adenocarcinoma of the lung, bronchogenic carcinoma, colon carcinoma, metastatic adenocarcinoma, etc.) [5-10]. In most of these cases, an improvement and clearance of the dermatosis was obtained after tumor removal [5-8, 10]. In our case, the patient had PRP leading to the discovery of a benign cerebral meningioma; noticeably, a dramatic regression of the eruption was observed after surgical removal of the tumor. Methotrexate had been used for only three weeks, therefore a beneficial effect of this drug on the skin lesions seems unlikely. We thus speculate a direct association between the PRP and the meningioma. To our knowledge, this is the first case of PRP associated with a benign tumor.
274
Sandrine QUENAN1 Gurkan KAYA2 Emmanuel LAFFITTE1
1. Griffiths WA. Pityriasis rubra pilaris. Clin Exp Dermatol 1980; 5: 105-12. 2. Miralles ES, Nunez M, De Las Heras ME, et al. Pityriasis rubra pilaris and human immunodeficiency virus infection. Br J Dermatol 1995; 133: 990-3. 3. Dicken CH. Treatment of classic pityriasis rubra pilaris. J Am Acad Dermatol 1994; 31: 997-9. 4. Allison DS, El-Azhary RA, Calobrisi SDDicken CH. Pityriasis rubra pilaris in children. J Am Acad Dermatol 2002; 47: 386-9. 5. Batchelor RJ, Yung A, Merchant W, Goodfield MJ. Pityriasis rubra pilaris as the initial presentation of renal cell carcinoma? Clin Exp Dermatol 2005; 30: 442-3. 6. Garretson CB, Machan ML, Krejci-Manwaring J, Aires D, TonkovicCapin V. Letter: Adenocarcinoma of the lung associated with pityriasis rubra pilaris. Dermatol Online J 2011; 17: 14. 7. Kloos C, Muller UA, Hoffken K, et al. [Paraneoplastic pityriasis rubra pilaris in metastatic adenocarcinoma without diagnosable primary]. Dtsch Med Wochenschr 2002; 127: 437-40. 8. Kurzydlo AM, Gillespie R. Paraneoplastic pityriasis rubra pilaris in association with bronchogenic carcinoma. Australas J Dermatol 2004; 45: 130-2. 9. Sharma S, Weiss GR, Paulger B. Pityriasis rubra pilaris as an initial presentation of hepatocellular carcinoma. Dermatology 1997; 194: 166-7. 10. Vitiello M, Miteva M, Romanelli P, et al. Pityriasis rubra pilaris: was it the first manifestation of colon cancer in a patient with preexisting psoriasis? J Am Acad Dermatol 2013; 68: e43-4. doi:10.1684/ejd.2015.2532
Chromoblastomycosis caused by Phialophora verrucosa on the hand The fungi that form black colonies on culture medium due to melanin pigment in the cell wall are collectively referred to as dematiaceous fungi. Dematiaceous fungal infections with a verrucous or plaque appearance and histopathologically sclerotic or muriform cells are classified as chromoblastomycosis. Phialophora verrucosa (P. verrucosa) is one of the chromoblastomycosis-causing fungi [1]. It is common in the environment but infrequently causes an infection due to its low pathogenicity [2]. Here, we report a case of chromoblastomycosis caused by P. verrucosa. A 77-year-old male patient had presented to his former hospital with a small, growing, hyperkeratotic nodule on the back of his right hand, one year previously (figure 1A). As a farmer, he often handled soil with his bare hands. He had no immunocompromising condition, had not taken any immunosuppressive drugs and had no HIV infection. The nodule on the back of his right hand had grown to a EJD, vol. 25, n◦ 3, May-June 2015
A
D
B
C
E
F
10.0 µ m
Figure 1. Crusted erythematous plaque 5 cm in size at the initial visit to his former hospital (A). At the initial visit to our hospital, the 5 cm erythematous plaque had a horseshoe periphery that rose around its center (B). Three months after itraconazole treatment (C). A skin biopsy showed pseudoepithelial hyperplasia (D). Sclerotic cells within a multinucleated giant cell (E). They were stained with HE. Original magnification × 12.5 (D) and × 400 (E). Slide culture showed flask-shaped phialide, cup-shaped collarette (Length/Width = L/W = 0.8), and hypha that produced ecru and elliptic (2.54.5 × 1.5-3.0m) microconidia (F).
crusted erythematous plaque, 5 cm in size. Differential diagnoses were deep cutaneous mycosis, atypical mycobacterial disease and squamous cell carcinoma. His former doctor examined him but did not reach a diagnosis. The patient came to Kanazawa University Hospital two months after his first visit to his former hospital. The 5cm erythematous plaque had a horseshoe periphery that rose around its center (figure 1B). We performed a skin biopsy of the erythematous plaque. Histopathological examination of the biopsied skin showed pseudoepitheliomatous hyperplasia and infiltration of various subsets of leukocytes, including lymphocytes, neutrophils, plasma cells, eosinophils and multinucleated giant cells in the dermis (figure 1D). Additionally, we observed chain fungal spores and sclerotic cells (figure 1E). The isolated fungus KMU8930 produced black colonies, which were ash gray in the middle of the surface and greenish black at the back, in potato dextrose agar at 27 ◦ C and 37 ◦ C. However, the colonies did not grow at 40 ◦ C. Slide culture showed flask-shaped phialide, cup-shaped collarette (Length/Width = L/W = 0.8), and hypha that produced ecru and elliptic (2.5-4.5 × 1.5-3.0 m) microconidia (figure 1F). We made a diagnosis of chromobastomycosis caused by P. verrucosa based on the clinical presentation, histopathology and fungal culture results. The strain KMU8930 matched the D1/D2 domain sequence 565bp of large subunit ribosomal DNA with known data of P. verrucosa. The patient was started on oral itraconazole, 100mg/day and he then recovered in about five months (figure 1C). Chromoblastomycosis is caused by dematiaceous fungi, including Fonsecaea pedrosoi (F. pedrosoi), P. verrucosa, F. compacta, Cladophialophora carrionii and Rhinocladialla aquaspersa. Chromoblastomycosis has the distinctive clinical presentation of a warty papule that slowly enlarges from a hypertrophic plaque. In some cases, the plaque is flat in the initial phase and expands slowly, with central scarring. The histological hallmark of chromoblastomycoEJD, vol. 25, n◦ 3, May-June 2015
sis is the formation of sclerotic cells with small, round, thick-walled and brownish fungus elements in the affected tissue [1]. The primary causative fungus in chromoblastomycosis is F. pedrosoi, with P. verrucosa less frequent. In fact, Minotto et al. reported that, in 100 cases diagnosed with chromoblastmycosis in Brazil, 96% were caused by F. pedrosoi and 4% by P. verrucosa [3]. P. verrucosa is a wood-rotting fungus that occurs in nature. It has many opportunities to be transmitted to humans but infrequently causes an infection because of low pathogenicity [2]. However, Hoffmann reported a case of superficial infection with P. verrucosa on the face, which spread to the paranasal sinuses and palate [4]. The main treatment of chromoblastomycosis is antifungal chemotherapy. Itraconazole or terbinafine is often successful. Additionally, topical heat therapy may also be effective cases of P. verrucosa. Ishibashi et al. reported that a disposable pocket warmer was effective, since it rises up to 63 ◦ C and lasts 12 h and P. verrucosa cannot grow over 40 ◦ C [5]. The temperature should be kept approximately between 40 ◦ C and 43 ◦ C to avoid low-temperature burn. Chromoblastomycosis commonly recurs after treatment. Therefore, patients should be carefully monitored at least twice after clinical healing and two consecutive negative microscopic and cultural examinations should be confirmed [6]. Disclosure. Financial support: none. Conflict of interest: none. 1 Department of Dermatology, Faculty of Medicine, Kanazawa University, 13-1 Takaramachi, Kanazawa, Ishikawa 920-8641, Japan 2 Department of Dermatology, Nanto Municipal Hospital, Toyama, Japan 3 Department of Dermatology, 4 Division of Dermatomycology (Novartis Pharma), Research Institute of Medical Science, Kanazawa Medical University, Ishikawa, Japan
Akiko TAKEUCHI1,2 Kazufumi ANZAWA3,4 Takashi MOCHIZUKI3,4 Kazuhiko TAKEHARA1 Yasuhito HAMAGUCHI1
1. Lopez Martinez R, Mendez Tovar LJ. Chromoblastmycosis. Clin Dermatol 2007; 25: 188-94. 2. Felger CE, Friedman L. Experimental cerebral chromoblastomycosis. J Infect Dis 1962; 111: 1-7. 3. Minotto R, Verajao Bernardi CD, Mallmann LF, et al. Chromoblastomycosis: a review of 100 cases in the state of Rio Grande do Sui, Brazil. J Am Acad Dermato 2001; 44: 585-92. 4. Hofmann H, Choi SM, Wilsmann-Theis D, et al. Invasive chromoblastmycosis and sinusitis due to Phialophora verrucosa in a child from northern Africa. Mycoses 2005; 48: 456-61. 5. Ishibashi M, Numata Y, Tagami H, et al. Successful treatment of cutaneous botryomycosis with a combination of minocycline and topical heat therapy. J Am Acad Dermatol 1984; 10: 615-9. 6. Tintelnot K. Therapy of infections caused by dematiaceous fungi. Mycoses 1997; 40: 91-6. doi:10.1684/ejd.2015.2581
275
Disclosure. Financial support: none. Conflict of interest: none.
B
1
Department of Dermatology, Department of Dermatopathology, University Hospital of Geneva, 4 Rue Gabrielle-Perret-Gentil, 1211 Geneva, Switzerland 2
C D
E
F
Figure 1. Psoriasiform hyperplasia of the epidermis with a mild superficial perivascular lymphocytic infiltrate (A) and overlying “geometric” parakeratosis (B). C) follicular lesions coalescing into erythematosquamous plaques on the trunk. D) yellowish-orange color of the palms; E, F) clinical remission after surgery.
tar keratoderma. In 1980, Griffiths proposed a classification of PRP into five types [1]. An HIV-associated type has recently been described [2]. The origin of PRP is unknown, although infections, genetic and metabolic factors have been suggested. It is usually a chronic, long-lasting disease and the drugs classically prescribed for therapy (acitretin, methotrexate, TNF blockers or other biologics) are needed for several months. Clinical remission was achieved in eight adults with methotrexate (oral or injected) at the dose of 10-30 mg/week [3]; by contrast, Allison et al. reported a complete failure in five patients [4]. Associations of PRP with malignancies have been reported, either hematologic (leukemia) or solid (renal-cell carcinoma, hepatocellular carcinoma, laryngeal carcinoma, adenocarcinoma of the lung, bronchogenic carcinoma, colon carcinoma, metastatic adenocarcinoma, etc.) [5-10]. In most of these cases, an improvement and clearance of the dermatosis was obtained after tumor removal [5-8, 10]. In our case, the patient had PRP leading to the discovery of a benign cerebral meningioma; noticeably, a dramatic regression of the eruption was observed after surgical removal of the tumor. Methotrexate had been used for only three weeks, therefore a beneficial effect of this drug on the skin lesions seems unlikely. We thus speculate a direct association between the PRP and the meningioma. To our knowledge, this is the first case of PRP associated with a benign tumor.
274
Sandrine QUENAN1 Gurkan KAYA2 Emmanuel LAFFITTE1
1. Griffiths WA. Pityriasis rubra pilaris. Clin Exp Dermatol 1980; 5: 105-12. 2. Miralles ES, Nunez M, De Las Heras ME, et al. Pityriasis rubra pilaris and human immunodeficiency virus infection. Br J Dermatol 1995; 133: 990-3. 3. Dicken CH. Treatment of classic pityriasis rubra pilaris. J Am Acad Dermatol 1994; 31: 997-9. 4. Allison DS, El-Azhary RA, Calobrisi SDDicken CH. Pityriasis rubra pilaris in children. J Am Acad Dermatol 2002; 47: 386-9. 5. Batchelor RJ, Yung A, Merchant W, Goodfield MJ. Pityriasis rubra pilaris as the initial presentation of renal cell carcinoma? Clin Exp Dermatol 2005; 30: 442-3. 6. Garretson CB, Machan ML, Krejci-Manwaring J, Aires D, TonkovicCapin V. Letter: Adenocarcinoma of the lung associated with pityriasis rubra pilaris. Dermatol Online J 2011; 17: 14. 7. Kloos C, Muller UA, Hoffken K, et al. [Paraneoplastic pityriasis rubra pilaris in metastatic adenocarcinoma without diagnosable primary]. Dtsch Med Wochenschr 2002; 127: 437-40. 8. Kurzydlo AM, Gillespie R. Paraneoplastic pityriasis rubra pilaris in association with bronchogenic carcinoma. Australas J Dermatol 2004; 45: 130-2. 9. Sharma S, Weiss GR, Paulger B. Pityriasis rubra pilaris as an initial presentation of hepatocellular carcinoma. Dermatology 1997; 194: 166-7. 10. Vitiello M, Miteva M, Romanelli P, et al. Pityriasis rubra pilaris: was it the first manifestation of colon cancer in a patient with preexisting psoriasis? J Am Acad Dermatol 2013; 68: e43-4. doi:10.1684/ejd.2015.2532
Chromoblastomycosis caused by Phialophora verrucosa on the hand The fungi that form black colonies on culture medium due to melanin pigment in the cell wall are collectively referred to as dematiaceous fungi. Dematiaceous fungal infections with a verrucous or plaque appearance and histopathologically sclerotic or muriform cells are classified as chromoblastomycosis. Phialophora verrucosa (P. verrucosa) is one of the chromoblastomycosis-causing fungi [1]. It is common in the environment but infrequently causes an infection due to its low pathogenicity [2]. Here, we report a case of chromoblastomycosis caused by P. verrucosa. A 77-year-old male patient had presented to his former hospital with a small, growing, hyperkeratotic nodule on the back of his right hand, one year previously (figure 1A). As a farmer, he often handled soil with his bare hands. He had no immunocompromising condition, had not taken any immunosuppressive drugs and had no HIV infection. The nodule on the back of his right hand had grown to a EJD, vol. 25, n◦ 3, May-June 2015
A
D
B
C
E
F
10.0 µ m
Figure 1. Crusted erythematous plaque 5 cm in size at the initial visit to his former hospital (A). At the initial visit to our hospital, the 5 cm erythematous plaque had a horseshoe periphery that rose around its center (B). Three months after itraconazole treatment (C). A skin biopsy showed pseudoepithelial hyperplasia (D). Sclerotic cells within a multinucleated giant cell (E). They were stained with HE. Original magnification × 12.5 (D) and × 400 (E). Slide culture showed flask-shaped phialide, cup-shaped collarette (Length/Width = L/W = 0.8), and hypha that produced ecru and elliptic (2.54.5 × 1.5-3.0m) microconidia (F).
crusted erythematous plaque, 5 cm in size. Differential diagnoses were deep cutaneous mycosis, atypical mycobacterial disease and squamous cell carcinoma. His former doctor examined him but did not reach a diagnosis. The patient came to Kanazawa University Hospital two months after his first visit to his former hospital. The 5cm erythematous plaque had a horseshoe periphery that rose around its center (figure 1B). We performed a skin biopsy of the erythematous plaque. Histopathological examination of the biopsied skin showed pseudoepitheliomatous hyperplasia and infiltration of various subsets of leukocytes, including lymphocytes, neutrophils, plasma cells, eosinophils and multinucleated giant cells in the dermis (figure 1D). Additionally, we observed chain fungal spores and sclerotic cells (figure 1E). The isolated fungus KMU8930 produced black colonies, which were ash gray in the middle of the surface and greenish black at the back, in potato dextrose agar at 27 ◦ C and 37 ◦ C. However, the colonies did not grow at 40 ◦ C. Slide culture showed flask-shaped phialide, cup-shaped collarette (Length/Width = L/W = 0.8), and hypha that produced ecru and elliptic (2.5-4.5 × 1.5-3.0 m) microconidia (figure 1F). We made a diagnosis of chromobastomycosis caused by P. verrucosa based on the clinical presentation, histopathology and fungal culture results. The strain KMU8930 matched the D1/D2 domain sequence 565bp of large subunit ribosomal DNA with known data of P. verrucosa. The patient was started on oral itraconazole, 100mg/day and he then recovered in about five months (figure 1C). Chromoblastomycosis is caused by dematiaceous fungi, including Fonsecaea pedrosoi (F. pedrosoi), P. verrucosa, F. compacta, Cladophialophora carrionii and Rhinocladialla aquaspersa. Chromoblastomycosis has the distinctive clinical presentation of a warty papule that slowly enlarges from a hypertrophic plaque. In some cases, the plaque is flat in the initial phase and expands slowly, with central scarring. The histological hallmark of chromoblastomycoEJD, vol. 25, n◦ 3, May-June 2015
sis is the formation of sclerotic cells with small, round, thick-walled and brownish fungus elements in the affected tissue [1]. The primary causative fungus in chromoblastomycosis is F. pedrosoi, with P. verrucosa less frequent. In fact, Minotto et al. reported that, in 100 cases diagnosed with chromoblastmycosis in Brazil, 96% were caused by F. pedrosoi and 4% by P. verrucosa [3]. P. verrucosa is a wood-rotting fungus that occurs in nature. It has many opportunities to be transmitted to humans but infrequently causes an infection because of low pathogenicity [2]. However, Hoffmann reported a case of superficial infection with P. verrucosa on the face, which spread to the paranasal sinuses and palate [4]. The main treatment of chromoblastomycosis is antifungal chemotherapy. Itraconazole or terbinafine is often successful. Additionally, topical heat therapy may also be effective cases of P. verrucosa. Ishibashi et al. reported that a disposable pocket warmer was effective, since it rises up to 63 ◦ C and lasts 12 h and P. verrucosa cannot grow over 40 ◦ C [5]. The temperature should be kept approximately between 40 ◦ C and 43 ◦ C to avoid low-temperature burn. Chromoblastomycosis commonly recurs after treatment. Therefore, patients should be carefully monitored at least twice after clinical healing and two consecutive negative microscopic and cultural examinations should be confirmed [6]. Disclosure. Financial support: none. Conflict of interest: none. 1 Department of Dermatology, Faculty of Medicine, Kanazawa University, 13-1 Takaramachi, Kanazawa, Ishikawa 920-8641, Japan 2 Department of Dermatology, Nanto Municipal Hospital, Toyama, Japan 3 Department of Dermatology, 4 Division of Dermatomycology (Novartis Pharma), Research Institute of Medical Science, Kanazawa Medical University, Ishikawa, Japan
Akiko TAKEUCHI1,2 Kazufumi ANZAWA3,4 Takashi MOCHIZUKI3,4 Kazuhiko TAKEHARA1 Yasuhito HAMAGUCHI1
1. Lopez Martinez R, Mendez Tovar LJ. Chromoblastmycosis. Clin Dermatol 2007; 25: 188-94. 2. Felger CE, Friedman L. Experimental cerebral chromoblastomycosis. J Infect Dis 1962; 111: 1-7. 3. Minotto R, Verajao Bernardi CD, Mallmann LF, et al. Chromoblastomycosis: a review of 100 cases in the state of Rio Grande do Sui, Brazil. J Am Acad Dermato 2001; 44: 585-92. 4. Hofmann H, Choi SM, Wilsmann-Theis D, et al. Invasive chromoblastmycosis and sinusitis due to Phialophora verrucosa in a child from northern Africa. Mycoses 2005; 48: 456-61. 5. Ishibashi M, Numata Y, Tagami H, et al. Successful treatment of cutaneous botryomycosis with a combination of minocycline and topical heat therapy. J Am Acad Dermatol 1984; 10: 615-9. 6. Tintelnot K. Therapy of infections caused by dematiaceous fungi. Mycoses 1997; 40: 91-6. doi:10.1684/ejd.2015.2581
275
