Copyright © 2017 John Libbey Eurotext. Téléchargé par NYU LANGONE MED CTR SCH OF MED HEALTH SCIENCES LIBRARY le 19/01/2017.
HPV molecular biology typing showed HPV 5-DNA (Pr. G. Orth, Institut Pasteur, Paris), confirming the diagnosis of EV-like eruption. Search for EVER1 and EVER2 mutations in our patient (Dr M. Favre, Institut Pasteur, Paris) were negative. However, 2 messenger RNAs (mRNAs) from the EVER2 gene were studied; one of them carried a deletion, which could cause a truncated, ineffective protein. Several topical treatments were tried between 2000 and 2003, including retinoids and imiquimod (5% cream, 3 times/week) leading to partial regression. The patient stopped these treatments in 2003, because of poor efficiency and tolerance. Since 2008, the skin eruption has spontaneously and gradually disappeared totally. Regarding the immunovirological status, the patient was severely immunocompromised at the beginning of the eruption (CD4-T cell count: 79/L, viral load 2200 copies/ml); in 2000, when the EV-like eruption was diagnosed and as it was spreading, the CD4-T cell count was 172/L and the viral load was undetectable. Since 2001, the viral load has been undetectable, except for short periods. The CD4T cell count has gradually increased to reach 350/L in 2006. In HIV patients, about 40 cases of EV-like eruptions have been well described and published. The effect of AT on EV-like eruptions is still controversial. A summary of 20 patients for whom specific treatment of EV and the effect of AT are documented is shown in table 1. For 4 of these 20 patients, the eruption worsened with AT, which suggests the presence of Immune Reconstitution Inflammatory Syndrome (IRIS) [6, 10]. 14 patients had no improvement with AT [3-5, 9]. Two of them [7, 8] experienced a partial clearance, but these patients were also receiving antibiotics [7] or imiquimod [8]. We noticed a complete regression of the eruption in our patient, but this occurred several years after a virological and immune response to HAART. The longer follow-up of our patient may have contributed to this surprising result. Long-term effects of immune reconstitution may have been underestimated up to now. Most systemic or topical treatments usually used in EV were ineffective for these 20 patients. A few patients noticed a reduction (often with relapse) of the lesions [3, 8-10], as already reported.
4. Kroft EB, Melchers WJ, Blokx WA, de Hoop D, Warris A. A generalized skin eruption in a human immunodeficiency virus-infected boy. J Eur Acad Dermatol Venereol 2008; 2: 896-7. 5. Carré D, Dompmartin A, Verdon R, et al. Epidermodysplasia verruciformis in a patient with HIV infection: no response to highly active antiretroviral therapy. Int J Dermatol 2003; 42: 296-300. 6. Mermet I, Guerrini JS, Cairey-Remonnay S, et al. Cervical intraepithelial neoplasia associated with epidermodysplasia verruciformis HPV in an HIV-infected patient: a manifestation of immune restoration syndrome. Eur J Dermatol 2007; 17: 149-52. 7. Haas N, Fuchs PG, Hermes B, Henz BM. Remission of epidermodysplasia verruciformis-like skin eruption after highly active antiretroviral therapy in a human immunodeficiency virus-positive patient. Br J Dermatol 2001; 145: 669-70. 8. Lee KC, Risser J, Bercovitch L. What is the evidence for effective treatments of acquired epidermodysplasia verruciformis in HIV-infected patients? Arch Dermatol 2010; 146: 903-5. 9. Jacobelli S, Laude H, Carlotti A, et al. Epidermodysplasia verruciformis in human immunodeficiency virus-infected patients: a marker of human papillomavirus-related disorders not affected by antiretroviral therapy. Arch Dermatol 2011; 147: 590-6. 10. Darwich E, Darwich L, Ca˜ nadas MP, et al. New human papillomavirus (HPV) types involved in epidermodysplasia verruciformis (EV) in 3 HIV-infected patients: response to topical cidofovir. J Am Acad Dermatol 2011; 65: e43-5. doi:10.1684/ejd.2015.2601
Successful treatment of lichen spinulosus with topical adapalene A 69-year-old Japanese man presented with a 2-month history of multiple brown lesions on his trunk and limbs. Physical examination showed brownish, 2-4 mm follicular hyperkeratotic papules on the trunk and limbs (figure 1A). Some lesions were surrounded by a reddish halo. Routine
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B
Disclosure. Financial support: none. Conflict of interest: none. Department of Internal medicine and Infectious Diseases, Franc¸ois Quesnay General Hospital, 2 boulevard Sully, 78200 Mantes-la-Jolie, France
Anne Lise LECAPITAINE Véronique PERRONNE Franc¸oise GRANIER
1. Lutzner MA, Blanchet-Bardon C, Orth G. Clinical observations, virologic studies, treatment trials in patients with epidermodysplasia verruciformis, a disease induced by specific HPV. J Invest Dermatol 1984; 83: 18s-25s. 2. Ramoz N, Rueda LA, Bouadjar B, Montoya LS, Orth G, Favre M. Mutations in two adjacent novel genes are associated with epidermodysplasia verruciformis. Nat Genet 2002; 32: 579-81. 3. Rallis E, Paparizos V, Kyriakis K, Katsambas A. Treatment of epidermodysplasia verruciformis in human immunodeficiency virus-positive patients. J Eur Acad Dermatol Venereol 2009; 23: 195-6.
490
C
Figure 1. A) Multiple brownish keratotic papules on the trunk. B) Histopathological features of a papule: an enlarged follicle is seen, containing a orthokeratotic infundibular plug (hematoxylin-eosin, ×100). C) Substantial improvement of skin lesions after a 6-week treatment with topical 0.1% adapalene gel. EJD, vol. 25, n◦ 5, September-October 2015
Copyright © 2017 John Libbey Eurotext. Téléchargé par NYU LANGONE MED CTR SCH OF MED HEALTH SCIENCES LIBRARY le 19/01/2017.
laboratory tests (including HIV serology) were normal, except for a decreased red-blood cell count and increased serum glucose. None of his family members had similar lesions. Histological examination of lesional skin revealed an enlarged hair follicle containing an orthokeratotic keratin plug that protruded above the skin surface (figure 1B), and perifollicular mononuclear-cell infiltrate in the dermis. These clinico-pathological findings were consistent with lichen spinulosus (LS). Topical 0.1% adapalene gel was applied after diagnosis. Six weeks later, the skin lesions had improved with no significant local or systemic adverse effects, leaving residual follicular pigmentation (figure 1C). The lesions remained well controlled during the 6-month follow-up period. LS is characterized by asymptomatic follicular keratotic spiny papules symmetrically distributed on the neck, buttocks, abdomen and extensor aspects of the arms. The differential diagnosis of LS includes keratosis pilaris, follicular mucinosis, lichen nitidus and multiple minute digitate hyperkeratosis (MMDH). Keratosis pilaris affects the lateral aspects of the arms and thighs; histopathologically it is similar to LS, except that the perifollicular mononuclearcell infiltrate is sparse in most cases and that the keratin follicular plug does not protrude substantially above the skin surface. Follicular mucinosis is characterized clinically by well-demarcated erythematous papules or plaques with follicular prominence [1]. Since our patient had only a few papules over the lateral aspects of the arm and no mucin deposits in the hair follicles, we excluded the diagnoses of keratosis pilaris and follicular mucinosis. Lichen nitidus does not show hyperkeratosis or follicular plugging, histologically. MMDH has non-follicular hyperkeratosis; this diagnosis was excluded following the diagnostic algorithm by Caccetta et al. [2]. The cause and pathogenesis of LS remain unknown. It has been suggested that several factors, such as atopic diathesis, infections, id reaction to fungal infection, genetic factors, trauma and drug-induced reactions (including reactions to thallium, gold, diphtheria toxin, and arsphenamine) might be involved in the pathogenesis of LS [3, 4]. Until now, no standardized treatment has been established for LS. The common treatments include emollients and keratolytics [3]. Application of topical tacalcitol cream, a vitamin D analog, reportedly improved LS in 2 patients [4]. In one report, there was no improvement of LS with urea ointment but complete remission was subsequently obtained with topical tretinoin gel, which is a first-generation retinoid [4]. The author chose tretinoin gel, expecting a keratolytic effect on LS lesions [4]. Based on this successful result of topical tretinoin, we used adapalene in our patient with LS. Adapalene is the third generation of synthetic retinoids, containing naphthoic acid. Adapalene selectively binds to the retinoic acid receptors  and ␥, modulates abnormal desquamation of the skin, inhibits the differentiation of keratinocytes and has anti-inflammatory properties [5]. In addition, compared with other retinoids, adapalene causes less skin irritation due to its receptor selectivity. Compared with tretinoin, adapalene is better tolerated and shows better therapeutic effects on acne vulgaris [6], suggesting that it could also have better clinical efficacy on LS than tretinoin. Disclosure. Financial support: none. Conflict of interest: none. EJD, vol. 25, n◦ 5, September-October 2015
1 Department of Dermatology, Gunma University Graduate School of Medicine, 3-39-22 Showa, Maebashi, Gunma 371-8511, Japan 2 Sapporo Skin Clinic, Sapporo, Hokkaido 060-0063, Japan
Akihito UEHARA1 Masatoshi ABE1,2 Akira SHIMIZU1 Sei-ichiro MOTEGI1 Hiroo AMANO1 Osamu ISHIKAWA1
1. Cömert A, Akin O, Demirkesen C. Follicular mucinosis mimicking lichen spinulosus in an 11-year-old boy. Eur J Dermatol 2007; 17: 544-5. 2. Caccetta TP, Dessauvagie B, McCallum D, Kumarasinghe SP. Multiple minute digitate hyperkeratosis: a proposed algorithm for the digitate keratoses. J Am Acad Dermatol 2012; 67: e49-55. 3. Alan SB. Lichen spinulosus: Case report and overview. Cutis 1989; 43: 557-60. 4. Kim SH, Kang JH, Seo JK, Hwang SW, Sung HS, Lee D. Successful treatment of lichen spinulosus with topical tacalcitol cream. Pediatr Dermatol 2010; 27: 546-7. 5. Forman SB, Hudgins EM, Blaylock WK. Lichen spinulosus: excellent response to tretinoin gel and hydroactive adhesive applications. Arch Dermatol 2007; 143: 122-3. 6. Michel S, Jomard A, Demarchez M. Pharmacology of adapalene. Br J Dermatol 1998; 139: S3-7. doi:10.1684/ejd.2015.2597
Anaphylactic shock after the ingestion of jellyfish without a history of jellyfish contact or sting We read with interest the article entitled “Anaphylaxis caused by ingestion of jellyfish” by Imamura et al. in this journal [1]. They stated that their case subject was sensitized through the skin by jellyfish stings and that subsequent jellyfish intake induced anaphylaxis. However, we experienced a case of anaphylaxis after jellyfish ingestion without any history of jellyfish contact or sting. A 14-year-old boy developed a cough, urticaria and dyspnea 30 minutes after he ate a breakfast that included dried and salted jellyfish, which he rarely consumed. He had eaten other foods, such as rice, cucumber, and crab sticks, which he consumed daily without any incident. There was no past medical history other than a house-dust mite allergy. His mother had a history of anaphylaxis from pyrazolone derivatives. He had never been to the sea and had no previous episode of jellyfish contact or sting. Vital signs revealed tachycardia and hypotension. On physical examination, periorbital edema, wheezing and diffuse urticarial lesions were noted. Laboratory examinations - a complete blood count, serum electrolytes, liver function tests and serum creatinine level - were all normal. His symptoms suggested anaphylactic shock and he was treated with intramuscular epinephrine injection, volume resuscitation and infusions of corticosteroids and antihistamines. Following this, his symptoms dramatically abated without biphasic reaction. The patient’s total serum immunoglobulin E (IgE) concentration was 578 IU/mL. No specific IgE antibody against crab, cuttlefish, shrimp or tropomyosin was detected.
491
HPV molecular biology typing showed HPV 5-DNA (Pr. G. Orth, Institut Pasteur, Paris), confirming the diagnosis of EV-like eruption. Search for EVER1 and EVER2 mutations in our patient (Dr M. Favre, Institut Pasteur, Paris) were negative. However, 2 messenger RNAs (mRNAs) from the EVER2 gene were studied; one of them carried a deletion, which could cause a truncated, ineffective protein. Several topical treatments were tried between 2000 and 2003, including retinoids and imiquimod (5% cream, 3 times/week) leading to partial regression. The patient stopped these treatments in 2003, because of poor efficiency and tolerance. Since 2008, the skin eruption has spontaneously and gradually disappeared totally. Regarding the immunovirological status, the patient was severely immunocompromised at the beginning of the eruption (CD4-T cell count: 79/L, viral load 2200 copies/ml); in 2000, when the EV-like eruption was diagnosed and as it was spreading, the CD4-T cell count was 172/L and the viral load was undetectable. Since 2001, the viral load has been undetectable, except for short periods. The CD4T cell count has gradually increased to reach 350/L in 2006. In HIV patients, about 40 cases of EV-like eruptions have been well described and published. The effect of AT on EV-like eruptions is still controversial. A summary of 20 patients for whom specific treatment of EV and the effect of AT are documented is shown in table 1. For 4 of these 20 patients, the eruption worsened with AT, which suggests the presence of Immune Reconstitution Inflammatory Syndrome (IRIS) [6, 10]. 14 patients had no improvement with AT [3-5, 9]. Two of them [7, 8] experienced a partial clearance, but these patients were also receiving antibiotics [7] or imiquimod [8]. We noticed a complete regression of the eruption in our patient, but this occurred several years after a virological and immune response to HAART. The longer follow-up of our patient may have contributed to this surprising result. Long-term effects of immune reconstitution may have been underestimated up to now. Most systemic or topical treatments usually used in EV were ineffective for these 20 patients. A few patients noticed a reduction (often with relapse) of the lesions [3, 8-10], as already reported.
4. Kroft EB, Melchers WJ, Blokx WA, de Hoop D, Warris A. A generalized skin eruption in a human immunodeficiency virus-infected boy. J Eur Acad Dermatol Venereol 2008; 2: 896-7. 5. Carré D, Dompmartin A, Verdon R, et al. Epidermodysplasia verruciformis in a patient with HIV infection: no response to highly active antiretroviral therapy. Int J Dermatol 2003; 42: 296-300. 6. Mermet I, Guerrini JS, Cairey-Remonnay S, et al. Cervical intraepithelial neoplasia associated with epidermodysplasia verruciformis HPV in an HIV-infected patient: a manifestation of immune restoration syndrome. Eur J Dermatol 2007; 17: 149-52. 7. Haas N, Fuchs PG, Hermes B, Henz BM. Remission of epidermodysplasia verruciformis-like skin eruption after highly active antiretroviral therapy in a human immunodeficiency virus-positive patient. Br J Dermatol 2001; 145: 669-70. 8. Lee KC, Risser J, Bercovitch L. What is the evidence for effective treatments of acquired epidermodysplasia verruciformis in HIV-infected patients? Arch Dermatol 2010; 146: 903-5. 9. Jacobelli S, Laude H, Carlotti A, et al. Epidermodysplasia verruciformis in human immunodeficiency virus-infected patients: a marker of human papillomavirus-related disorders not affected by antiretroviral therapy. Arch Dermatol 2011; 147: 590-6. 10. Darwich E, Darwich L, Ca˜ nadas MP, et al. New human papillomavirus (HPV) types involved in epidermodysplasia verruciformis (EV) in 3 HIV-infected patients: response to topical cidofovir. J Am Acad Dermatol 2011; 65: e43-5. doi:10.1684/ejd.2015.2601
Successful treatment of lichen spinulosus with topical adapalene A 69-year-old Japanese man presented with a 2-month history of multiple brown lesions on his trunk and limbs. Physical examination showed brownish, 2-4 mm follicular hyperkeratotic papules on the trunk and limbs (figure 1A). Some lesions were surrounded by a reddish halo. Routine
A
B
Disclosure. Financial support: none. Conflict of interest: none. Department of Internal medicine and Infectious Diseases, Franc¸ois Quesnay General Hospital, 2 boulevard Sully, 78200 Mantes-la-Jolie, France
Anne Lise LECAPITAINE Véronique PERRONNE Franc¸oise GRANIER
1. Lutzner MA, Blanchet-Bardon C, Orth G. Clinical observations, virologic studies, treatment trials in patients with epidermodysplasia verruciformis, a disease induced by specific HPV. J Invest Dermatol 1984; 83: 18s-25s. 2. Ramoz N, Rueda LA, Bouadjar B, Montoya LS, Orth G, Favre M. Mutations in two adjacent novel genes are associated with epidermodysplasia verruciformis. Nat Genet 2002; 32: 579-81. 3. Rallis E, Paparizos V, Kyriakis K, Katsambas A. Treatment of epidermodysplasia verruciformis in human immunodeficiency virus-positive patients. J Eur Acad Dermatol Venereol 2009; 23: 195-6.
490
C
Figure 1. A) Multiple brownish keratotic papules on the trunk. B) Histopathological features of a papule: an enlarged follicle is seen, containing a orthokeratotic infundibular plug (hematoxylin-eosin, ×100). C) Substantial improvement of skin lesions after a 6-week treatment with topical 0.1% adapalene gel. EJD, vol. 25, n◦ 5, September-October 2015
Copyright © 2017 John Libbey Eurotext. Téléchargé par NYU LANGONE MED CTR SCH OF MED HEALTH SCIENCES LIBRARY le 19/01/2017.
laboratory tests (including HIV serology) were normal, except for a decreased red-blood cell count and increased serum glucose. None of his family members had similar lesions. Histological examination of lesional skin revealed an enlarged hair follicle containing an orthokeratotic keratin plug that protruded above the skin surface (figure 1B), and perifollicular mononuclear-cell infiltrate in the dermis. These clinico-pathological findings were consistent with lichen spinulosus (LS). Topical 0.1% adapalene gel was applied after diagnosis. Six weeks later, the skin lesions had improved with no significant local or systemic adverse effects, leaving residual follicular pigmentation (figure 1C). The lesions remained well controlled during the 6-month follow-up period. LS is characterized by asymptomatic follicular keratotic spiny papules symmetrically distributed on the neck, buttocks, abdomen and extensor aspects of the arms. The differential diagnosis of LS includes keratosis pilaris, follicular mucinosis, lichen nitidus and multiple minute digitate hyperkeratosis (MMDH). Keratosis pilaris affects the lateral aspects of the arms and thighs; histopathologically it is similar to LS, except that the perifollicular mononuclearcell infiltrate is sparse in most cases and that the keratin follicular plug does not protrude substantially above the skin surface. Follicular mucinosis is characterized clinically by well-demarcated erythematous papules or plaques with follicular prominence [1]. Since our patient had only a few papules over the lateral aspects of the arm and no mucin deposits in the hair follicles, we excluded the diagnoses of keratosis pilaris and follicular mucinosis. Lichen nitidus does not show hyperkeratosis or follicular plugging, histologically. MMDH has non-follicular hyperkeratosis; this diagnosis was excluded following the diagnostic algorithm by Caccetta et al. [2]. The cause and pathogenesis of LS remain unknown. It has been suggested that several factors, such as atopic diathesis, infections, id reaction to fungal infection, genetic factors, trauma and drug-induced reactions (including reactions to thallium, gold, diphtheria toxin, and arsphenamine) might be involved in the pathogenesis of LS [3, 4]. Until now, no standardized treatment has been established for LS. The common treatments include emollients and keratolytics [3]. Application of topical tacalcitol cream, a vitamin D analog, reportedly improved LS in 2 patients [4]. In one report, there was no improvement of LS with urea ointment but complete remission was subsequently obtained with topical tretinoin gel, which is a first-generation retinoid [4]. The author chose tretinoin gel, expecting a keratolytic effect on LS lesions [4]. Based on this successful result of topical tretinoin, we used adapalene in our patient with LS. Adapalene is the third generation of synthetic retinoids, containing naphthoic acid. Adapalene selectively binds to the retinoic acid receptors  and ␥, modulates abnormal desquamation of the skin, inhibits the differentiation of keratinocytes and has anti-inflammatory properties [5]. In addition, compared with other retinoids, adapalene causes less skin irritation due to its receptor selectivity. Compared with tretinoin, adapalene is better tolerated and shows better therapeutic effects on acne vulgaris [6], suggesting that it could also have better clinical efficacy on LS than tretinoin. Disclosure. Financial support: none. Conflict of interest: none. EJD, vol. 25, n◦ 5, September-October 2015
1 Department of Dermatology, Gunma University Graduate School of Medicine, 3-39-22 Showa, Maebashi, Gunma 371-8511, Japan 2 Sapporo Skin Clinic, Sapporo, Hokkaido 060-0063, Japan
Akihito UEHARA1 Masatoshi ABE1,2 Akira SHIMIZU1 Sei-ichiro MOTEGI1 Hiroo AMANO1 Osamu ISHIKAWA1
1. Cömert A, Akin O, Demirkesen C. Follicular mucinosis mimicking lichen spinulosus in an 11-year-old boy. Eur J Dermatol 2007; 17: 544-5. 2. Caccetta TP, Dessauvagie B, McCallum D, Kumarasinghe SP. Multiple minute digitate hyperkeratosis: a proposed algorithm for the digitate keratoses. J Am Acad Dermatol 2012; 67: e49-55. 3. Alan SB. Lichen spinulosus: Case report and overview. Cutis 1989; 43: 557-60. 4. Kim SH, Kang JH, Seo JK, Hwang SW, Sung HS, Lee D. Successful treatment of lichen spinulosus with topical tacalcitol cream. Pediatr Dermatol 2010; 27: 546-7. 5. Forman SB, Hudgins EM, Blaylock WK. Lichen spinulosus: excellent response to tretinoin gel and hydroactive adhesive applications. Arch Dermatol 2007; 143: 122-3. 6. Michel S, Jomard A, Demarchez M. Pharmacology of adapalene. Br J Dermatol 1998; 139: S3-7. doi:10.1684/ejd.2015.2597
Anaphylactic shock after the ingestion of jellyfish without a history of jellyfish contact or sting We read with interest the article entitled “Anaphylaxis caused by ingestion of jellyfish” by Imamura et al. in this journal [1]. They stated that their case subject was sensitized through the skin by jellyfish stings and that subsequent jellyfish intake induced anaphylaxis. However, we experienced a case of anaphylaxis after jellyfish ingestion without any history of jellyfish contact or sting. A 14-year-old boy developed a cough, urticaria and dyspnea 30 minutes after he ate a breakfast that included dried and salted jellyfish, which he rarely consumed. He had eaten other foods, such as rice, cucumber, and crab sticks, which he consumed daily without any incident. There was no past medical history other than a house-dust mite allergy. His mother had a history of anaphylaxis from pyrazolone derivatives. He had never been to the sea and had no previous episode of jellyfish contact or sting. Vital signs revealed tachycardia and hypotension. On physical examination, periorbital edema, wheezing and diffuse urticarial lesions were noted. Laboratory examinations - a complete blood count, serum electrolytes, liver function tests and serum creatinine level - were all normal. His symptoms suggested anaphylactic shock and he was treated with intramuscular epinephrine injection, volume resuscitation and infusions of corticosteroids and antihistamines. Following this, his symptoms dramatically abated without biphasic reaction. The patient’s total serum immunoglobulin E (IgE) concentration was 578 IU/mL. No specific IgE antibody against crab, cuttlefish, shrimp or tropomyosin was detected.
491
