Correspondence
The histological findings of both patients on skin biopsy were similar, namely, subepidermal blistering with abundant neutrophilic infiltrate. Using direct immunofluorescence, linear deposition of IgA antibodies was seen at the dermoepidermal junction in both cases (Fig. 1d, Fig. 2d). For patient 2, immunoblotting detected antibodies against the 97-KDa antigen. In both cases, bacterial infection was excluded by culture of swabs taken from the blisters. Patient 1 was started on cloxacillin 25 mg/kg/day for a year, with good results. Subsequently, the treatment was changed to salazopyrine until the age of 7 years, with progressive reduction in the number of attacks until their complete disappearance at 12 years of age. Patient 2 began treatment with erythromycin 50 mg/ kg/day for 3 weeks. It was necessary to repeat the course of antibiotics 8 weeks later owing to reactivation of the disease. The condition then cleared, and to date, the patient has been asymptomatic. The pathogenesis of LABD is still unknown. IgA autoantibodies produce chemotaxis of eosinophils and basophils, which are responsible for the formation of the subepidermal blisters as a result of degranulation and release of lysozyme and beta-glucuronidase.3 Several authors have reported using oral antibiotics for LABD, which produced complete response in most of the cases (Table 1). There are few published reports providing scientific evidence on the use of erythromycin.4,5 The beneficial effects of antibiotics on LABD may be due to the inhibition of neutrophil chemotaxis and release of lysozyme and b-glucuronidase, which result in the antiinflammatory effects of these drugs, as has also been seen in in vitro assays. A similar phenomenon occurs with salazopyrine. In our first case, a long-lasting clinical response was obtained with the combined use of both treatments. In our second case, the new attack rapidly responded to a new course of oral antibiotic, and subsequently, the patient has been free of the disease for several months. In conclusion, the existence of a growing number of cases reporting response to antibiotics in LABD lays the foundations for a treatment that is as effective as traditional therapies. We consider that therapy with antibiotics, particularly with erythromycin because of its low index of side-effects in paediatric patients, should be the first step in the treatment of LABD, as the clinical response can be rapid and long-lasting. M. Navedo de las Heras University Clinic of Navarra, Pamplona, Navarra, Spain E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 10 May 2013
References 1 Lara I, Pope E. Autoinmune blistering diseases in children. Semin Cutan Med Surg 2010; 29: 85–91.
2014 British Association of Dermatologists
2 Olivera JE, S anchez–Valverde F, Espa~ na A et al. Deficit de lipasa pancre atica y enfermedad IgA lineal en la infancia. An Esp Pediatr 1996; 44: 73–5. 3 Yeh SW, Ahmed B, Sami N et al. Blistering disorders: diagnosis and treatment. Dermatol Ther 2003; 16: 214– 23. 4 Farrant P, Darley C, Carmichael A. Is erythromycin an effective treatment for chronic bullous disease of childhood? A national survey of members of the British society for paediatric dermatology. Pediatric Dermatol 2008; 4: 479–82. 5 Cooper SM, Powell J, Wojnarowska F. Linear IgA disease: successful treatment with erythromycin. Clin Exp Dermatol 2002; 27: 677–9. 6 Siegfried EC, Sirawan S. Chronic bullous disease of childhood: successful treatment with dicloxacillin. J Am Acad Dermatol 1998; 39: 797–800. 7 Skinner RB Jr, Rotondo CK, Schneider MA et al. Treatment of chronic bullous dermatosis of childhood with oral dicloxacillin. Pediatr Dermatol 1995; 12: 65–6. 8 Denguezli M, Ben Nejma B, Nouira R et al. IgA linear bullous dermatosis in children: a series of 12 Tunisian patients. Ann Dermatol Venereol 1994; 12: 888–92. 9 Alajlan A, Al-Khawajah M, Al-Sheikh O et al. Treatment of linear IgA bullous dermatosis of childhood with flucloxacillin. J Am Acad Dermatol 2006; 54: 652–6.
Sarcoidosis associated with cosmetic fillers doi: 10.1111/ced.12262
Approximately 25% of cutaneous sarcoidosis biopsies may show refractile material, suggesting that in some patients, foreign material may be the nidus for granuloma formation.1 We report a patient with systemic sarcoidosis who developed specific lesions in facial areas injected with cosmetic fillers 16 years previously. A 70-year-old woman presented with a 10-day history of facial subcutaneous nodular lesions. Sixteen years previously, an unknown cosmetic filler had been injected into her face. On physical examination, 5 – 6 nodular, non-erythematous, subcutaneous indurated lesions were seen in both the malar and submandibular areas (Fig. 1a). The lesions were non-tender, 10 – 20 mm in diameter, and covered with normal-appearing skin. A few days after the onset of the facial lesions, the patient developed several painful subcutaneous erythematous nodules in both forearms and both legs (Fig. 1b). Laboratory investigations showed normal levels for peptidyl-dipeptidase and calcium (0.42 lkat/L and 2.42 mmol/L respectively). Ocular evaluation was also normal. Chest radiography showed bilateral hilar lymphadenopathy. Positron emission tomography–computed
Clinical and Experimental Dermatology (2014) 39, pp395–397
397
Correspondence
tomography showed enlarged hilar bilateral and mediastinal lymph nodes and bilateral pulmonary opacities. Pulmonary function tests were normal. On histological examination of a biopsy taken from a nodular lesion on the forearm, a predominantly septal panniculitis was seen, consistent with erythema nodosum (EN). A biopsy taken from a facial nodular lesion howed noncaseating epithelioid granulomas with epithelioid
(a)
cells, and multinucleated giant cells containing empty roundish vacuoles of variable size (Fig. 2). The vacuoles were also found in the extracellular space, and were nonbirefringent under polarized light. The microscopic appearance of the foreign material was highly suggestive of silicone. Dermal filler injections are becoming increasingly popular for cosmetic purposes. However, all injectable products used as dermal filler substances can cause foreign-body granulomas, with an incidence ranging from 0.01 to 1.0%. Granulomas can occur at any time from 6 months to many years after the injection.2 The incidence of true sarcoidosis within this granulomatous reactions is not known. Only six patients (including ours) with systemic sarcoidosis and granulomatous lesions in areas injected with cosmetic fillers have been reported to date.2-5 Three of these six cases were induced by a-interferon (IFN), suggesting that the development of granulomatous
(a)
(b)
(b)
Figure 2 Biopsy taken from a facial nodular lesion, showing sar-
Figure 1 (a) Non-tender nodular lesions on the face, in areas
where cosmetic fillers had been injected 16 years previously; (b) erythema nodosum lesions located on the leg.
398
Clinical and Experimental Dermatology (2014) 39, pp395–397
coid granulomas with multinucleated giant cells containing intracytoplasmic roundish vacuoles of variable size. The vacuoles were non-birefringent under polarized light, and were also found in the extracellular space. Haematoxylin and eosin, original magnification (a) 9 10; (b) 9 20.
2014 British Association of Dermatologists
Correspondence
cutaneous lesions of sarcoidosis in areas injected with cosmetic fillers could be more common in IFN-induced sarcoidosis than in sarcoidosis not triggered by IFN. Although foreign bodies or filler substances are not the cause of systemic sarcoidosis, the filler particles did influence the distribution of the lesions when our patient developed sarcoidosis. In patients with granulomatous reactions to cosmetic fillers, sarcoidosis must be excluded, particularly in IFN-treated patients. We treated our patient with potassium iodide, and the EN lesions healed in a few weeks. The facial nodular lesions later slowly disappeared without any treatment. To date, the patient remains asymptomatic. ,2 R. M. Penın,3 I. Figueras,1 J Marcoval,1 J. Ma~ na M. Labori2 and R. Llatjos3 Departments of 1Dermatology, 2Internal Medicine and 3Pathology, Bellvitge Hospital, C/Feixa Llarga s/n, L’Hospitalet de Llobregat, 08907 Barcelona, Spain E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 21 October 2013
AA is an autoimmune-mediated disease, making it one of the most common autoimmune diseases.1 Infection with human immunodeficiency virus (HIV) has been associated with autoimmune diseases, and several reports describe induction of AA by HIV infection.2 We report a case of AU that resolved spontaneously after HIV infection. This is, to our knowledge, the first report of HIV-induced remission of AA. A 46-year-old patient had sustained recurrent attacks of patchy AA on his scalp since the age of 6 years, progressing to AT at the age of 20 years. When he was 26 years old, he developed AU, which was recalcitrant to treatment. He had tattoos applied to compensate for eyebrow loss.
(a)
References J, Moreno A et al. Foreign bodies in 1 Marcoval J, Ma~ na granulomatous cutaneous lesions of patients with systemic sarcoidosis. Arch. Dermatol 2001; 137: 427–30. 2 Descamps V, Landry J, Frances C et al. Facial cosmetic filler injections as possible target for systemic sarcoidosis in patients treated with interferon for chronic hepatitis C: two cases. Dermatology 2008; 217: 81–4. 3 Sidwell RU, Johnson McL, Francis N et al. Cutaneous sarcoidal granulomas developing after Artecoll facial cosmetic filler in a patient with newly diagnosed systemic sarcoidosis. Clin Exp Dermatol 2006; 31: 208–11. 4 Fischer J, Metzler G, Schaller M. Cosmetic permanent fillers for soft tissue augmentation: a new contraindication for interferon therapies. Arch Dermatol 2007; 143: 507–10. 5 Dal Sacco D, Cozzani E, Parodi A, Rebora A. Scar sarcoidosis after hyaluronic acid injection. Int J Dermatol 2005; 44: 411–12. (b)
Remission of long-standing alopecia universalis after human immunodeficiency virus infection doi: 10.1111/ced.12260
Alopecia areata (AA) is a common disorder, which usually presents as patches of hair loss on the scalp or beard. However, in 5% of the patients,1 the disease may progress to alopecia totalis (AT), affecting all scalp hair, or even alopecia universalis (AU), affecting all scalp and body hair. AU is highly resistant to treatment, and spontaneous remission occurs only rarely.1
2014 British Association of Dermatologists
Figure 1 (a) Almost complete regrowth of hair on the scalp after
human immunodeficiency virus infection with decreased CD4 counts in a patient with alopecia universalis. (b) Regrowth of eyebrow hair, covering the black tattoo used to compensate for the long-standing eyebrow loss caused by the patient’s disease. Picture was taken when the patient was 46 years old, 40 years after alopecia areata had started.
Clinical and Experimental Dermatology (2014) 39, pp395–399
399
The histological findings of both patients on skin biopsy were similar, namely, subepidermal blistering with abundant neutrophilic infiltrate. Using direct immunofluorescence, linear deposition of IgA antibodies was seen at the dermoepidermal junction in both cases (Fig. 1d, Fig. 2d). For patient 2, immunoblotting detected antibodies against the 97-KDa antigen. In both cases, bacterial infection was excluded by culture of swabs taken from the blisters. Patient 1 was started on cloxacillin 25 mg/kg/day for a year, with good results. Subsequently, the treatment was changed to salazopyrine until the age of 7 years, with progressive reduction in the number of attacks until their complete disappearance at 12 years of age. Patient 2 began treatment with erythromycin 50 mg/ kg/day for 3 weeks. It was necessary to repeat the course of antibiotics 8 weeks later owing to reactivation of the disease. The condition then cleared, and to date, the patient has been asymptomatic. The pathogenesis of LABD is still unknown. IgA autoantibodies produce chemotaxis of eosinophils and basophils, which are responsible for the formation of the subepidermal blisters as a result of degranulation and release of lysozyme and beta-glucuronidase.3 Several authors have reported using oral antibiotics for LABD, which produced complete response in most of the cases (Table 1). There are few published reports providing scientific evidence on the use of erythromycin.4,5 The beneficial effects of antibiotics on LABD may be due to the inhibition of neutrophil chemotaxis and release of lysozyme and b-glucuronidase, which result in the antiinflammatory effects of these drugs, as has also been seen in in vitro assays. A similar phenomenon occurs with salazopyrine. In our first case, a long-lasting clinical response was obtained with the combined use of both treatments. In our second case, the new attack rapidly responded to a new course of oral antibiotic, and subsequently, the patient has been free of the disease for several months. In conclusion, the existence of a growing number of cases reporting response to antibiotics in LABD lays the foundations for a treatment that is as effective as traditional therapies. We consider that therapy with antibiotics, particularly with erythromycin because of its low index of side-effects in paediatric patients, should be the first step in the treatment of LABD, as the clinical response can be rapid and long-lasting. M. Navedo de las Heras University Clinic of Navarra, Pamplona, Navarra, Spain E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 10 May 2013
References 1 Lara I, Pope E. Autoinmune blistering diseases in children. Semin Cutan Med Surg 2010; 29: 85–91.
2014 British Association of Dermatologists
2 Olivera JE, S anchez–Valverde F, Espa~ na A et al. Deficit de lipasa pancre atica y enfermedad IgA lineal en la infancia. An Esp Pediatr 1996; 44: 73–5. 3 Yeh SW, Ahmed B, Sami N et al. Blistering disorders: diagnosis and treatment. Dermatol Ther 2003; 16: 214– 23. 4 Farrant P, Darley C, Carmichael A. Is erythromycin an effective treatment for chronic bullous disease of childhood? A national survey of members of the British society for paediatric dermatology. Pediatric Dermatol 2008; 4: 479–82. 5 Cooper SM, Powell J, Wojnarowska F. Linear IgA disease: successful treatment with erythromycin. Clin Exp Dermatol 2002; 27: 677–9. 6 Siegfried EC, Sirawan S. Chronic bullous disease of childhood: successful treatment with dicloxacillin. J Am Acad Dermatol 1998; 39: 797–800. 7 Skinner RB Jr, Rotondo CK, Schneider MA et al. Treatment of chronic bullous dermatosis of childhood with oral dicloxacillin. Pediatr Dermatol 1995; 12: 65–6. 8 Denguezli M, Ben Nejma B, Nouira R et al. IgA linear bullous dermatosis in children: a series of 12 Tunisian patients. Ann Dermatol Venereol 1994; 12: 888–92. 9 Alajlan A, Al-Khawajah M, Al-Sheikh O et al. Treatment of linear IgA bullous dermatosis of childhood with flucloxacillin. J Am Acad Dermatol 2006; 54: 652–6.
Sarcoidosis associated with cosmetic fillers doi: 10.1111/ced.12262
Approximately 25% of cutaneous sarcoidosis biopsies may show refractile material, suggesting that in some patients, foreign material may be the nidus for granuloma formation.1 We report a patient with systemic sarcoidosis who developed specific lesions in facial areas injected with cosmetic fillers 16 years previously. A 70-year-old woman presented with a 10-day history of facial subcutaneous nodular lesions. Sixteen years previously, an unknown cosmetic filler had been injected into her face. On physical examination, 5 – 6 nodular, non-erythematous, subcutaneous indurated lesions were seen in both the malar and submandibular areas (Fig. 1a). The lesions were non-tender, 10 – 20 mm in diameter, and covered with normal-appearing skin. A few days after the onset of the facial lesions, the patient developed several painful subcutaneous erythematous nodules in both forearms and both legs (Fig. 1b). Laboratory investigations showed normal levels for peptidyl-dipeptidase and calcium (0.42 lkat/L and 2.42 mmol/L respectively). Ocular evaluation was also normal. Chest radiography showed bilateral hilar lymphadenopathy. Positron emission tomography–computed
Clinical and Experimental Dermatology (2014) 39, pp395–397
397
Correspondence
tomography showed enlarged hilar bilateral and mediastinal lymph nodes and bilateral pulmonary opacities. Pulmonary function tests were normal. On histological examination of a biopsy taken from a nodular lesion on the forearm, a predominantly septal panniculitis was seen, consistent with erythema nodosum (EN). A biopsy taken from a facial nodular lesion howed noncaseating epithelioid granulomas with epithelioid
(a)
cells, and multinucleated giant cells containing empty roundish vacuoles of variable size (Fig. 2). The vacuoles were also found in the extracellular space, and were nonbirefringent under polarized light. The microscopic appearance of the foreign material was highly suggestive of silicone. Dermal filler injections are becoming increasingly popular for cosmetic purposes. However, all injectable products used as dermal filler substances can cause foreign-body granulomas, with an incidence ranging from 0.01 to 1.0%. Granulomas can occur at any time from 6 months to many years after the injection.2 The incidence of true sarcoidosis within this granulomatous reactions is not known. Only six patients (including ours) with systemic sarcoidosis and granulomatous lesions in areas injected with cosmetic fillers have been reported to date.2-5 Three of these six cases were induced by a-interferon (IFN), suggesting that the development of granulomatous
(a)
(b)
(b)
Figure 2 Biopsy taken from a facial nodular lesion, showing sar-
Figure 1 (a) Non-tender nodular lesions on the face, in areas
where cosmetic fillers had been injected 16 years previously; (b) erythema nodosum lesions located on the leg.
398
Clinical and Experimental Dermatology (2014) 39, pp395–397
coid granulomas with multinucleated giant cells containing intracytoplasmic roundish vacuoles of variable size. The vacuoles were non-birefringent under polarized light, and were also found in the extracellular space. Haematoxylin and eosin, original magnification (a) 9 10; (b) 9 20.
2014 British Association of Dermatologists
Correspondence
cutaneous lesions of sarcoidosis in areas injected with cosmetic fillers could be more common in IFN-induced sarcoidosis than in sarcoidosis not triggered by IFN. Although foreign bodies or filler substances are not the cause of systemic sarcoidosis, the filler particles did influence the distribution of the lesions when our patient developed sarcoidosis. In patients with granulomatous reactions to cosmetic fillers, sarcoidosis must be excluded, particularly in IFN-treated patients. We treated our patient with potassium iodide, and the EN lesions healed in a few weeks. The facial nodular lesions later slowly disappeared without any treatment. To date, the patient remains asymptomatic. ,2 R. M. Penın,3 I. Figueras,1 J Marcoval,1 J. Ma~ na M. Labori2 and R. Llatjos3 Departments of 1Dermatology, 2Internal Medicine and 3Pathology, Bellvitge Hospital, C/Feixa Llarga s/n, L’Hospitalet de Llobregat, 08907 Barcelona, Spain E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 21 October 2013
AA is an autoimmune-mediated disease, making it one of the most common autoimmune diseases.1 Infection with human immunodeficiency virus (HIV) has been associated with autoimmune diseases, and several reports describe induction of AA by HIV infection.2 We report a case of AU that resolved spontaneously after HIV infection. This is, to our knowledge, the first report of HIV-induced remission of AA. A 46-year-old patient had sustained recurrent attacks of patchy AA on his scalp since the age of 6 years, progressing to AT at the age of 20 years. When he was 26 years old, he developed AU, which was recalcitrant to treatment. He had tattoos applied to compensate for eyebrow loss.
(a)
References J, Moreno A et al. Foreign bodies in 1 Marcoval J, Ma~ na granulomatous cutaneous lesions of patients with systemic sarcoidosis. Arch. Dermatol 2001; 137: 427–30. 2 Descamps V, Landry J, Frances C et al. Facial cosmetic filler injections as possible target for systemic sarcoidosis in patients treated with interferon for chronic hepatitis C: two cases. Dermatology 2008; 217: 81–4. 3 Sidwell RU, Johnson McL, Francis N et al. Cutaneous sarcoidal granulomas developing after Artecoll facial cosmetic filler in a patient with newly diagnosed systemic sarcoidosis. Clin Exp Dermatol 2006; 31: 208–11. 4 Fischer J, Metzler G, Schaller M. Cosmetic permanent fillers for soft tissue augmentation: a new contraindication for interferon therapies. Arch Dermatol 2007; 143: 507–10. 5 Dal Sacco D, Cozzani E, Parodi A, Rebora A. Scar sarcoidosis after hyaluronic acid injection. Int J Dermatol 2005; 44: 411–12. (b)
Remission of long-standing alopecia universalis after human immunodeficiency virus infection doi: 10.1111/ced.12260
Alopecia areata (AA) is a common disorder, which usually presents as patches of hair loss on the scalp or beard. However, in 5% of the patients,1 the disease may progress to alopecia totalis (AT), affecting all scalp hair, or even alopecia universalis (AU), affecting all scalp and body hair. AU is highly resistant to treatment, and spontaneous remission occurs only rarely.1
2014 British Association of Dermatologists
Figure 1 (a) Almost complete regrowth of hair on the scalp after
human immunodeficiency virus infection with decreased CD4 counts in a patient with alopecia universalis. (b) Regrowth of eyebrow hair, covering the black tattoo used to compensate for the long-standing eyebrow loss caused by the patient’s disease. Picture was taken when the patient was 46 years old, 40 years after alopecia areata had started.
Clinical and Experimental Dermatology (2014) 39, pp395–399
399
