Viewpoints in dermatology • Correspondence
Correspondence
Spreading of pre-existing segmental vitiligo after immunotherapy with house dust mite in a patient with atopic dermatitis doi: 10.1111/ced.12443 A 10-year-old Korean girl presented with a 3-year history of linearly arranged, hypopigmented patches on her left abdomen, and she was diagnosed with segmental vitiligo (SV) based on Wood light examination and the segmental distribution of the lesion. She also had a 7-year history of atopic dermatitis (AD). She underwent a 2-year course of excimer laser treatment for the SV, but the lesions did not respond well. The AD lesions were stable during the excimer laser treatment, but flared suddenly during treatment without progression of SV. The AD lesions were aggravated
(a)
even by conventional treatments for AD such as topical steroid and calcineurin inhibitors. Therefore, immunotherapy using house dust mite allergens was tried, as the patient was highly allergic to house dust mite by Multiple Allergen Simultaneous Test (MAST; Hitachi Chemical Diagnostics, Mountain view, CA, USA) allergy test. The patient received weekly injections of house dust mite allergen (Tyrosine-s; Allergy Therapeutics Ltd, Worthing, West Sussex, UK). Three months after immunotherapy, the AD lesions had improved, but the hypopigmented patches on the patient’s left abdomen started to increase in size along the ipsilateral dermatome (Fig. 1a,b), and new hypopigmented patches developed on the dorsum of her left foot (Fig. 1c,d). There have been a few previous reports of coexisting vitiligo and AD. A recent study showed a strong association
(b)
(d)
2005. Atopic dermatitis diagnosed
(c)
2012.7 flare-up of AD
2010.3 NBUVB 1 time/week
2012.12 vitiligo spreading
AD & SV Stable status
2010.10 Excimer laser 1 time/week
2009.12 Segmental vitiligo diagnosed Topical tacrolimus oint
2013.1 AD improvement
2012.9 Immunotherapy using house dust mite 1 time/wk total 13 times
Figure 1 (a) Linearly arranged, hypopigmented patches on the left abdomen before immunotherapy. (b) Hypopigmented patches
expanding from pre-existing segmental vitiligo (SV) along the ipsilateral segment after immunotherapy. (c) Coexistence of atopic dermatits (AD) and newly developed hypopigmented patches on the dorsum of the foot in the same direction as the pre-existing abdominal vilitigo lesions. (d) Clinical course of AD and SV during excimer laser therapy and immunotherapy.
920
Clinical and Experimental Dermatology (2015) 40, pp920–934
ª 2014 British Association of Dermatologists
Correspondence
between prepubertal nonsegmenal vitiligo (NSV) and AD, and a possible shared mechanism for both.1 However, the association of AD and SV has not been clearly established. To our knowledge, this is the first case report of spreading of SV after immunotherapy in a patient with AD. Such spreading is not thought to be associated with an autoimmune process, although SV has recently been reported to share an autoimmune mechanism with NSV.2,3 Although SV is usually explained by the neural hypothesis, this case supports a recent hypothesis that SV has a similar immunological mechanism to NSV in its development.3 A CD8+ melanocyte-specific T cellmediated immune response is known to play a role in SV with associated halo naevi, similar to NSV.4 It has been reported that dysfunction of humoral immunity may play a role in the progression of SV, whereas cellular immunity seems to be involved in NSV.5 T helper (Th)1/Th17 cellular immunity is thought to be involved in the pathogenesis of vitiligo, whereas AD is regarded as a Th2-skewed allergic skin disease. The mechanism of immunotherapy in AD is to downregulate Th2 response and to upregulate the Th1 pathway through allergen sensitization. Thus, our case shows that immunotherapy in AD could aggravate vitiligo via an immunotherapy-induced Th1 immune response. The presence of AD lesions over the dorsum of the patient’s left foot might indicate development of vitiligo lesions through koebnerization. However, this does not explain the spreading of vitiligo lesions on the left abdomen, which was not accompanied by AD. Furthermore, the AD lesions were bilateral and the vitiligo lesions were unilateral. In conclusion, our case suggests that immunotherapy for AD treatment could initiate spreading of SV, and that SV might have an overlapping immunological mechanism with NSV. S. Shin, J. Yo Shin, He Lee and S. H. Oh Department of Dermatology, Cutaneous Biology Research Institute, Yonsei University College of Medicine, 50 Yonsei-ro, Seodaemun-gu, Seoul 120-752, Korea E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 15 April 2014
References 1 Ezzedine K, Diallo A, Leaute-Labreze C et al. Pre- vs. postpubertal onset of vitiligo: multivariate analysis indicates atopic diathesis association in pre-pubertal onset vitiligo. Br J Dermatol 2012; 167: 490–5. 2 Attili VR, Attili SK. Segmental and generalized vitiligo: both forms demonstrate inflammatory histopathological features and clinical mosaicism. Indian J Dermatol 2013; 58: 433–8.
ª 2014 British Association of Dermatologists
3 van Geel N, Mollet I, Brochez L et al. New insights in segmental vitiligo: case report and review of theories. Br J Dermatol 2012; 166: 240–6. 4 van Geel NA, Mollet IG, De Schepper S et al. First histopathological and immunophenotypic analysis of early dynamic events in a patient with segmental vitiligo associated with halo nevi. Pigment Cell Melanoma Res 2010; 23: 375–84. 5 Lin X, Tang LY, Fu WW, Kang KF. Childhood vitiligo in China: clinical profiles and immunological findings in 620 cases. Am J Clin Dermatol 2011; 12: 277–81.
Identification and clinical consequences of a novel mutation in the gene for transglutaminase 1 in a patient with lamellar ichthyosis doi: 10.1111/ced.12456 We report a patient with lamellar ichthyosis, in whom a novel mutation was found in the gene for transglutaminase 1 (TGM1). The patient was a white girl, who was born prematurely by caesarean section at 34 weeks of gestation with birth weight of 2300 g and Apgar score of 9. The newborn was covered by a collodion membrane (Fig. 1a,b). She had ectropion and fixation of the lips in an O-shaped configuration, thickened fingernails and deformed ears. The collodion membrane was cracked and gradually desquamating. At 3 weeks of age, there was clinical improvement in the child’s condition. The collodion membrane was present only on the fingers and toes, causing constriction and ischaemia of the digits. The remaining skin was erythrodermic and covered with scales. The scales were large and multiangled, with a tendency to marginal desquamation (Fig. 1c). Eclabion persisted for 4 weeks and ectropion for 6 weeks. Later, the scales on the skin surface became finer and lighter, similar to bran flakes (Fig. 1d). When the child was 2 months old, treatment with acitretinum was introduced at an initial dosage of 1 mg/kg body weight per day in the first month of treatment, reducing to 0.65 mg/kg in the second month and 0.5 mg/kg in the third month. The therapy was stopped after 3 months of treatment because of raised levels of serum transaminases, calcium and alkaline phosphatase. The clinical symptoms observed in our patient during hospitalization did not allow us to precisely define the disorder. The bright scales and the erythrodermic underlying skin indicated a diagnosis of nonbullous congenital ichtyosiform erythroderma. However, the fact that the entire body, including the face, was affected, along with the size of the scales, their method of shedding (marginal shedding with central adherence), and the presence of ectropion and eclabion, indicated lamellar ichthyosis.1
Clinical and Experimental Dermatology (2015) 40, pp920–934
921
Correspondence
Spreading of pre-existing segmental vitiligo after immunotherapy with house dust mite in a patient with atopic dermatitis doi: 10.1111/ced.12443 A 10-year-old Korean girl presented with a 3-year history of linearly arranged, hypopigmented patches on her left abdomen, and she was diagnosed with segmental vitiligo (SV) based on Wood light examination and the segmental distribution of the lesion. She also had a 7-year history of atopic dermatitis (AD). She underwent a 2-year course of excimer laser treatment for the SV, but the lesions did not respond well. The AD lesions were stable during the excimer laser treatment, but flared suddenly during treatment without progression of SV. The AD lesions were aggravated
(a)
even by conventional treatments for AD such as topical steroid and calcineurin inhibitors. Therefore, immunotherapy using house dust mite allergens was tried, as the patient was highly allergic to house dust mite by Multiple Allergen Simultaneous Test (MAST; Hitachi Chemical Diagnostics, Mountain view, CA, USA) allergy test. The patient received weekly injections of house dust mite allergen (Tyrosine-s; Allergy Therapeutics Ltd, Worthing, West Sussex, UK). Three months after immunotherapy, the AD lesions had improved, but the hypopigmented patches on the patient’s left abdomen started to increase in size along the ipsilateral dermatome (Fig. 1a,b), and new hypopigmented patches developed on the dorsum of her left foot (Fig. 1c,d). There have been a few previous reports of coexisting vitiligo and AD. A recent study showed a strong association
(b)
(d)
2005. Atopic dermatitis diagnosed
(c)
2012.7 flare-up of AD
2010.3 NBUVB 1 time/week
2012.12 vitiligo spreading
AD & SV Stable status
2010.10 Excimer laser 1 time/week
2009.12 Segmental vitiligo diagnosed Topical tacrolimus oint
2013.1 AD improvement
2012.9 Immunotherapy using house dust mite 1 time/wk total 13 times
Figure 1 (a) Linearly arranged, hypopigmented patches on the left abdomen before immunotherapy. (b) Hypopigmented patches
expanding from pre-existing segmental vitiligo (SV) along the ipsilateral segment after immunotherapy. (c) Coexistence of atopic dermatits (AD) and newly developed hypopigmented patches on the dorsum of the foot in the same direction as the pre-existing abdominal vilitigo lesions. (d) Clinical course of AD and SV during excimer laser therapy and immunotherapy.
920
Clinical and Experimental Dermatology (2015) 40, pp920–934
ª 2014 British Association of Dermatologists
Correspondence
between prepubertal nonsegmenal vitiligo (NSV) and AD, and a possible shared mechanism for both.1 However, the association of AD and SV has not been clearly established. To our knowledge, this is the first case report of spreading of SV after immunotherapy in a patient with AD. Such spreading is not thought to be associated with an autoimmune process, although SV has recently been reported to share an autoimmune mechanism with NSV.2,3 Although SV is usually explained by the neural hypothesis, this case supports a recent hypothesis that SV has a similar immunological mechanism to NSV in its development.3 A CD8+ melanocyte-specific T cellmediated immune response is known to play a role in SV with associated halo naevi, similar to NSV.4 It has been reported that dysfunction of humoral immunity may play a role in the progression of SV, whereas cellular immunity seems to be involved in NSV.5 T helper (Th)1/Th17 cellular immunity is thought to be involved in the pathogenesis of vitiligo, whereas AD is regarded as a Th2-skewed allergic skin disease. The mechanism of immunotherapy in AD is to downregulate Th2 response and to upregulate the Th1 pathway through allergen sensitization. Thus, our case shows that immunotherapy in AD could aggravate vitiligo via an immunotherapy-induced Th1 immune response. The presence of AD lesions over the dorsum of the patient’s left foot might indicate development of vitiligo lesions through koebnerization. However, this does not explain the spreading of vitiligo lesions on the left abdomen, which was not accompanied by AD. Furthermore, the AD lesions were bilateral and the vitiligo lesions were unilateral. In conclusion, our case suggests that immunotherapy for AD treatment could initiate spreading of SV, and that SV might have an overlapping immunological mechanism with NSV. S. Shin, J. Yo Shin, He Lee and S. H. Oh Department of Dermatology, Cutaneous Biology Research Institute, Yonsei University College of Medicine, 50 Yonsei-ro, Seodaemun-gu, Seoul 120-752, Korea E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 15 April 2014
References 1 Ezzedine K, Diallo A, Leaute-Labreze C et al. Pre- vs. postpubertal onset of vitiligo: multivariate analysis indicates atopic diathesis association in pre-pubertal onset vitiligo. Br J Dermatol 2012; 167: 490–5. 2 Attili VR, Attili SK. Segmental and generalized vitiligo: both forms demonstrate inflammatory histopathological features and clinical mosaicism. Indian J Dermatol 2013; 58: 433–8.
ª 2014 British Association of Dermatologists
3 van Geel N, Mollet I, Brochez L et al. New insights in segmental vitiligo: case report and review of theories. Br J Dermatol 2012; 166: 240–6. 4 van Geel NA, Mollet IG, De Schepper S et al. First histopathological and immunophenotypic analysis of early dynamic events in a patient with segmental vitiligo associated with halo nevi. Pigment Cell Melanoma Res 2010; 23: 375–84. 5 Lin X, Tang LY, Fu WW, Kang KF. Childhood vitiligo in China: clinical profiles and immunological findings in 620 cases. Am J Clin Dermatol 2011; 12: 277–81.
Identification and clinical consequences of a novel mutation in the gene for transglutaminase 1 in a patient with lamellar ichthyosis doi: 10.1111/ced.12456 We report a patient with lamellar ichthyosis, in whom a novel mutation was found in the gene for transglutaminase 1 (TGM1). The patient was a white girl, who was born prematurely by caesarean section at 34 weeks of gestation with birth weight of 2300 g and Apgar score of 9. The newborn was covered by a collodion membrane (Fig. 1a,b). She had ectropion and fixation of the lips in an O-shaped configuration, thickened fingernails and deformed ears. The collodion membrane was cracked and gradually desquamating. At 3 weeks of age, there was clinical improvement in the child’s condition. The collodion membrane was present only on the fingers and toes, causing constriction and ischaemia of the digits. The remaining skin was erythrodermic and covered with scales. The scales were large and multiangled, with a tendency to marginal desquamation (Fig. 1c). Eclabion persisted for 4 weeks and ectropion for 6 weeks. Later, the scales on the skin surface became finer and lighter, similar to bran flakes (Fig. 1d). When the child was 2 months old, treatment with acitretinum was introduced at an initial dosage of 1 mg/kg body weight per day in the first month of treatment, reducing to 0.65 mg/kg in the second month and 0.5 mg/kg in the third month. The therapy was stopped after 3 months of treatment because of raised levels of serum transaminases, calcium and alkaline phosphatase. The clinical symptoms observed in our patient during hospitalization did not allow us to precisely define the disorder. The bright scales and the erythrodermic underlying skin indicated a diagnosis of nonbullous congenital ichtyosiform erythroderma. However, the fact that the entire body, including the face, was affected, along with the size of the scales, their method of shedding (marginal shedding with central adherence), and the presence of ectropion and eclabion, indicated lamellar ichthyosis.1
Clinical and Experimental Dermatology (2015) 40, pp920–934
921
