Correspondence
to a more favourable outcome, and may enable the avoidance of potentially toxic treatments. S. L. Bell,1 A. N. Patel,1 I. H. Leach,2 and S. N. Cohen1 Department of 1Dermatology and 2Histopathology, Nottingham University Hospitals NHS Trust, Queen’s Medical Centre, Nottingham, UK E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 4 November 2013
References 1 Judge MR, McLean WHI, Munro CS. Pityriasis rubra pilaris. Disorders of keratinization. In Rook’s Textbook of Dermatology, 8th edn (Burns T, Breathnach S, Cox N, Griffiths C, eds). Oxford: Wiley-Blackwell, 2010: 19.76–19.81. 2 Plana A, Carrascosa JM, Vilavella M et al. Pityriasis rubra pilaris-like reaction induced by imatinib. Clin Exp Dermatol 2013; 38: 520–2. 3 Joint Formulary Committee. British National Formulary. 64th edn. London: BMJ Group and Pharmaceutical Press, British National Formulary group and RPS Publishing, 2012; 121. 4 McNamara D. Diagnosis: malignant melanoma presenting as paraneoplastic pityriasis rubra pilaris (Case of the month). Skin Allergy News, 2010. Available at: www.nxtbook.com/nxtbooks/elsevier/san_201003/index. php?startid=35 (accessed 2 November 2013). 5 Tannenbaum CB, Billick RC, Srolovitz H. Multiple cutaneous malignancies in a patient with pityriasis rubra pilaris and focal acantholytic dyskeratosis. J Am Acad Dermatol 1996; 35: 781–2.
A linear lichenoid eruption following isotretinoin therapy
ating the eccrine glands. Interestingly, several of the inflammatory foci were reminiscent of lichen nitidus, with focal collections of inflammatory cells within adjacent claw-like rete ridges (Fig. 1b). The oral isotretinoin was discontinued, and the patent was prescribed a potent topical steroid. The eruption improved 1 month later, and a diagnosis of a linear lichenoid eruption (lichen planus; LP) secondary to isotretinoin was made. Linear LP was first described by Marie-Guillame Alphonse Devergie in 1854 (see review by Auner1). Since then, there have been 25 case reports of linear LP in the literature; the average age of onset of linear LP was 39 years, and 63% of patients were male. Most cases resolved after 7 months, and mucosal involvement was seen in 30%. There are three case reports in the literature of linear lichenoid drug eruptions. Two of these cases were due to valsartan, and the third to nicergoline.2,3
(a)
(b)
doi: 10.1111/ced.12281 A 20-year-old man presented with a unilateral, hyperpigmented, papular, scaly eruption on his trunk (Fig. 1a), following the lines of Blaschko. The eruption was relatively asymptomatic, and there was no nail or mucosal involvement. The eruption had occurred 1 month into treatment with oral isotretinoin for acne. A punch biopsy was taken from the eruption on the patient’s abdomen. On histological examination, the features seen consisted largely of a ‘burnt-out’ lichenoid dermatosis, with subsequent pigment incontinence. At low power, melanin pigment incontinence and relatively sparse inflammation was visible. At higher power, several foci of lichenoid inflammation were apparent, and a single dense collection of lymphocytes was seen to be perme-
2014 British Association of Dermatologists
Figure 1 (a) A lichenoid eruption following the lines of Blaschko;
(b) lichenoid inflammatory infiltrate (haematoxylin and eosin, original magnification 910).
Clinical and Experimental Dermatology (2014) 39, pp395–405
405
Correspondence
Linear LP is thought to be due to mosaicism, whereby a postzygotic somatic mutation leads to a clone of keratinocytes, which are more at risk of being affected by a dermatosis such as LP. An external antigen, such as a drug or a virus, then leads to the development of a dermatosis within the lines of Blaschko.4 There are no specific features to differentiate LP from a lichenoid drug eruption, although parakeratosis, eosinophils and plasma cells are more frequently seen in a lichenoid drug eruption.5 Certain medications are more typically associated with lichenoid drug eruptions, including penicillamine, gold, angiotensin-converting enzyme inhibitors, anti-malarials, thiazide diuretics, beta-blockers and nonsteroidal antiinflammatory agents.3 The pathogenesis of such eruptions is thought to be due to an alteration of the cell surface molecules by the drug, making them more antigenic and eliciting a lichenoid drug reaction.3 Isotretinoin has been reported to cause many cutaneous eruptions, aside from its association with hypervitaminosis A.5 There is one case report of the vulvovaginal–gingival variant of LP associated with isotretinoin,5 but there have been no reports of a linear lichenoid drug eruption following isotretinoin, and in fact, isotretinoin has been used successfully to treat LP.3 Our patient developed this eruption 1 month after starting treatment with isotretinoin, and it subsequently improved on discontinuation of the drug. We diagnosed this as a linear lichenoid drug eruption secondary to isotretinoin. We believe that this is the first report of a linear lichenoid eruption following treatment with isotretinoin. A. D. Scott,1 A. Robinson,2 and L. C. Fuller1 1 Department of Dermatology, Chelsea and Westminster NHS Foundation Trust, London, UK; and 2St John’s Institute, St Thomas’ Hospital, London, UK E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 13 October 2013
(a)
(b)
References 1 Auner JF. Lichen planus unilateralis. J Cutan Dis 1917; 25: 166–70. 2 Gencoglan G, Ceylan C, Kazandi AC. Linear lichenoid drug eruption induced by valsartan. Clin Exp Dermatol 2009; 34: 334–5. 3 Munoz MA, Perez-Bernal AM, Camacho FM. Lichenoid drug eruption following the Blaschko lines. Dermatology 1996; 193: 66–7. 4 Dominguez MVG, Mateu AV, Viera R, Solano JL, Sintes RN, Salmeron MTG. Linear lichen planus and hepatitis C. Dermatol Online J 2006; 1: 17. 5 Boyd AS, King LE. Lichenoid drug reaction from isotretinoin therapy. Cutis 2001; 68: 301–3.
Novel mutation in the PTCH1 gene in a patient with Gorlin syndrome with prominent clinical features doi: 10.1111/ced.12291 We report a novel PTCH1 gene mutation in a girl with multiple basal cell carcinomas (BCCs) that had been present since birth. A 2-year-old girl was referred to a paediatric dermatology department with a history of congenital hydrocephalus, macrocephaly and ulnar polydactyly on her left hand and both feet. She had frontal bossing, ocular hypertelorism and multiple papules 2–4 mm in diameter, located mainly on the hands and feet (Fig. 1a,b). No palmar– plantar pits were evident. Psychomotor development was appropriate for her age, and results of neurological, ophthalmological and intraoral examinations were normal. Imaging tests (magnetic resonance imaging, orthopantomography and serial bone scans) excluded the presence of associated brain abnormalities, odontogenic cysts or skeletal abnormalities. Histopathological examination of various papules confirmed the diagnosis of BCC. During
(c)
Figure 1 (a) Multiple basal cell carcinomas on the back of the patient’s left hand; (b) dermoscopy of a lesion on the hand, showing a
well-demarcated lesion, with multiple blue-grey globules; (c) multiple acrochordon-like basal cell carcinomas on the left-side of the neck.
406
Clinical and Experimental Dermatology (2014) 39, pp395–406
2014 British Association of Dermatologists
to a more favourable outcome, and may enable the avoidance of potentially toxic treatments. S. L. Bell,1 A. N. Patel,1 I. H. Leach,2 and S. N. Cohen1 Department of 1Dermatology and 2Histopathology, Nottingham University Hospitals NHS Trust, Queen’s Medical Centre, Nottingham, UK E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 4 November 2013
References 1 Judge MR, McLean WHI, Munro CS. Pityriasis rubra pilaris. Disorders of keratinization. In Rook’s Textbook of Dermatology, 8th edn (Burns T, Breathnach S, Cox N, Griffiths C, eds). Oxford: Wiley-Blackwell, 2010: 19.76–19.81. 2 Plana A, Carrascosa JM, Vilavella M et al. Pityriasis rubra pilaris-like reaction induced by imatinib. Clin Exp Dermatol 2013; 38: 520–2. 3 Joint Formulary Committee. British National Formulary. 64th edn. London: BMJ Group and Pharmaceutical Press, British National Formulary group and RPS Publishing, 2012; 121. 4 McNamara D. Diagnosis: malignant melanoma presenting as paraneoplastic pityriasis rubra pilaris (Case of the month). Skin Allergy News, 2010. Available at: www.nxtbook.com/nxtbooks/elsevier/san_201003/index. php?startid=35 (accessed 2 November 2013). 5 Tannenbaum CB, Billick RC, Srolovitz H. Multiple cutaneous malignancies in a patient with pityriasis rubra pilaris and focal acantholytic dyskeratosis. J Am Acad Dermatol 1996; 35: 781–2.
A linear lichenoid eruption following isotretinoin therapy
ating the eccrine glands. Interestingly, several of the inflammatory foci were reminiscent of lichen nitidus, with focal collections of inflammatory cells within adjacent claw-like rete ridges (Fig. 1b). The oral isotretinoin was discontinued, and the patent was prescribed a potent topical steroid. The eruption improved 1 month later, and a diagnosis of a linear lichenoid eruption (lichen planus; LP) secondary to isotretinoin was made. Linear LP was first described by Marie-Guillame Alphonse Devergie in 1854 (see review by Auner1). Since then, there have been 25 case reports of linear LP in the literature; the average age of onset of linear LP was 39 years, and 63% of patients were male. Most cases resolved after 7 months, and mucosal involvement was seen in 30%. There are three case reports in the literature of linear lichenoid drug eruptions. Two of these cases were due to valsartan, and the third to nicergoline.2,3
(a)
(b)
doi: 10.1111/ced.12281 A 20-year-old man presented with a unilateral, hyperpigmented, papular, scaly eruption on his trunk (Fig. 1a), following the lines of Blaschko. The eruption was relatively asymptomatic, and there was no nail or mucosal involvement. The eruption had occurred 1 month into treatment with oral isotretinoin for acne. A punch biopsy was taken from the eruption on the patient’s abdomen. On histological examination, the features seen consisted largely of a ‘burnt-out’ lichenoid dermatosis, with subsequent pigment incontinence. At low power, melanin pigment incontinence and relatively sparse inflammation was visible. At higher power, several foci of lichenoid inflammation were apparent, and a single dense collection of lymphocytes was seen to be perme-
2014 British Association of Dermatologists
Figure 1 (a) A lichenoid eruption following the lines of Blaschko;
(b) lichenoid inflammatory infiltrate (haematoxylin and eosin, original magnification 910).
Clinical and Experimental Dermatology (2014) 39, pp395–405
405
Correspondence
Linear LP is thought to be due to mosaicism, whereby a postzygotic somatic mutation leads to a clone of keratinocytes, which are more at risk of being affected by a dermatosis such as LP. An external antigen, such as a drug or a virus, then leads to the development of a dermatosis within the lines of Blaschko.4 There are no specific features to differentiate LP from a lichenoid drug eruption, although parakeratosis, eosinophils and plasma cells are more frequently seen in a lichenoid drug eruption.5 Certain medications are more typically associated with lichenoid drug eruptions, including penicillamine, gold, angiotensin-converting enzyme inhibitors, anti-malarials, thiazide diuretics, beta-blockers and nonsteroidal antiinflammatory agents.3 The pathogenesis of such eruptions is thought to be due to an alteration of the cell surface molecules by the drug, making them more antigenic and eliciting a lichenoid drug reaction.3 Isotretinoin has been reported to cause many cutaneous eruptions, aside from its association with hypervitaminosis A.5 There is one case report of the vulvovaginal–gingival variant of LP associated with isotretinoin,5 but there have been no reports of a linear lichenoid drug eruption following isotretinoin, and in fact, isotretinoin has been used successfully to treat LP.3 Our patient developed this eruption 1 month after starting treatment with isotretinoin, and it subsequently improved on discontinuation of the drug. We diagnosed this as a linear lichenoid drug eruption secondary to isotretinoin. We believe that this is the first report of a linear lichenoid eruption following treatment with isotretinoin. A. D. Scott,1 A. Robinson,2 and L. C. Fuller1 1 Department of Dermatology, Chelsea and Westminster NHS Foundation Trust, London, UK; and 2St John’s Institute, St Thomas’ Hospital, London, UK E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 13 October 2013
(a)
(b)
References 1 Auner JF. Lichen planus unilateralis. J Cutan Dis 1917; 25: 166–70. 2 Gencoglan G, Ceylan C, Kazandi AC. Linear lichenoid drug eruption induced by valsartan. Clin Exp Dermatol 2009; 34: 334–5. 3 Munoz MA, Perez-Bernal AM, Camacho FM. Lichenoid drug eruption following the Blaschko lines. Dermatology 1996; 193: 66–7. 4 Dominguez MVG, Mateu AV, Viera R, Solano JL, Sintes RN, Salmeron MTG. Linear lichen planus and hepatitis C. Dermatol Online J 2006; 1: 17. 5 Boyd AS, King LE. Lichenoid drug reaction from isotretinoin therapy. Cutis 2001; 68: 301–3.
Novel mutation in the PTCH1 gene in a patient with Gorlin syndrome with prominent clinical features doi: 10.1111/ced.12291 We report a novel PTCH1 gene mutation in a girl with multiple basal cell carcinomas (BCCs) that had been present since birth. A 2-year-old girl was referred to a paediatric dermatology department with a history of congenital hydrocephalus, macrocephaly and ulnar polydactyly on her left hand and both feet. She had frontal bossing, ocular hypertelorism and multiple papules 2–4 mm in diameter, located mainly on the hands and feet (Fig. 1a,b). No palmar– plantar pits were evident. Psychomotor development was appropriate for her age, and results of neurological, ophthalmological and intraoral examinations were normal. Imaging tests (magnetic resonance imaging, orthopantomography and serial bone scans) excluded the presence of associated brain abnormalities, odontogenic cysts or skeletal abnormalities. Histopathological examination of various papules confirmed the diagnosis of BCC. During
(c)
Figure 1 (a) Multiple basal cell carcinomas on the back of the patient’s left hand; (b) dermoscopy of a lesion on the hand, showing a
well-demarcated lesion, with multiple blue-grey globules; (c) multiple acrochordon-like basal cell carcinomas on the left-side of the neck.
406
Clinical and Experimental Dermatology (2014) 39, pp395–406
2014 British Association of Dermatologists
