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884

Our case is particularly interesting because the patient showed a rapid multiorgan involvement. Peculiar characteristics of IVLBCL result in tumour cells involving all types of organs, mainly bone marrow, the central nervous system and skin, with insult to organs resulting from transient or permanent vascular occlusion.4 Even if the lymphoma is usually disseminated extensively at presentation and the overall mortality rate is thought to be > 80%,5 in case of cutaneous involvement, such as our own, skin biopsy is a quick, minimally invasive and highly informative procedure. However, it is not possible to treat all cases ante-mortem. Studies suggest that IVLBCL responds remarkably well to rituximab-containing chemotherapy (R-CHOP).4,6 If possible, also high-dose chemotherapy with autologous hematopoietic stem cell rescue or aggressive combined therapy with high doses of methotrexate should be considered.6 However, in our case, the rapid course and aggressiveness did not allow for any treatments.

Funding source The authors declare that there are no financial or personal relationships that could inappropriately influence (or bias) the author’s decisions, work, or manuscript.

Acknowledgements None. L. Feci,1* M. Pellegrino,1 V. Mancini,2 P. Taddeucci,1 E. Trovato,1 C. Miracco,2 M. Fimiani1 1

Dermatology Section, Department of Clinical Medicine and Immunology, Siena University, 2Pathological Anatomy Section, Department of Human Pathology and Oncology, University of Siena, Siena, Italy *Correspondence: L. Feci. E-mail: [email protected]

References 1 Zuckerman D, Seliem R, Hochberg E. Intravascular lymphoma: the oncologist’s “great imitator”. Oncologist 2006; 11: 496–502. 2 Nakamura S, Ponzoni M, Campo E. Intravascular large B-cell lymphoma. In: Swerdlow SH, Campo E, Harris NL et al., eds. WHO classification of tumours of haematopoietic and lymphoid tissues. IARC Press, Lyon, France, 2008: 252–253. 3 Kong YY, Dai B, Sheng WQ et al. Intravascular large B-cell lymphoma with cutaneous manifestations: a clinicopathologic, immunophenotypic and molecular study of three cases. J Cutan Pathol 2009; 36: 865–870. 4 Ferreri AJ, Campo E, Seymour JF et al. Intravascular lymphoma: clinical presentation, natural history, management and prognostic factors in a series of 38 cases, with special emphasis on the ‘cutaneous variant’. Br J Haematol 2004; 127: 173–183. 5 Murase T, Yamaguchi M, Suzuki R et al. Intravascular large B-cell lymphoma (IVLBCL): a clinicopathologic study of 96 cases with special reference to the immuno phenotypic heterogeneity of CD5. Blood 2007; 109: 478–485. 6 Shimada K, Kinoshita T, Naoe T et al. Presentation and management of intravascular large B-cell lymphoma. Lancet Oncol 2009; 10: 895–902. DOI: 10.1111/jdv.13047

JEADV 2016, 30, 852–909

Successful treatment of lichen amyloidosus with oral alitretinoin Editor Lichen amyloidosus and macular amyloidosis are distinct manifestations of primary cutaneous amyloidosis. Skin lesions are typically accompanied by severe itching. Amyloid deposits in the papillary dermis are the typical histologic feature and diagnostic of the disease. No evidence-based standard treatment is available up to now. A variety of different treatment options such as topical corticosteroids, dimethyl sulfoxide (DMSO), acitretin, dermabrasion or ablative CO2 laser have been reported.1 Here, we describe a marked and durable improvement of clinical symptoms in a patient with long-lasting and recalcitrant lichen amyloidosus upon treatment with oral alitretinoin. A 66-year-old patient presented with pruritic papules on his arms, back and legs. His skin was notably dry. Skin biopsies taken during the course of the disease recurrently showed acanthosis with focal hypergranulosis and mild papillomatosis. Eosinophilic material was found in the upper dermis, which showed affinity to the pan-cytokeratin marker MNF-116. The histologic analysis of active skin lesions confirmed the clinical diagnosis of lichen amyloidosis (Fig. 1). A large spectrum of treatments such as topical steroids, 0.1% tacrolimus ointment, emollients, PUVA therapy and DMSO showed limited success. We finally decided to try oral retinoids. Since we were concerned that acitretin or etretinate would exacerbate the patient’s sebostasis and xerosis, we decided to start oral alitretinoin at 30 mg per day. The pruritus was effectively improved and the hyperkeratotic papules flattened within one month on therapy (Fig. 2a and b). Oral alitretinoin was initially given for six subsequent months before the patient stopped the treatment self-dependently. The disease remained stable over the next 4 months, but deteriorated in the follow-up. Re-implementation of alitretinoin 30 mg per day proved to be effective in both improving the pruritus and the skin lesions. Cutaneous amyloidosis has been divided into two forms: macular amyloidosis and lichen amyloidosus, but also indeterminate variants have been described. Macular amyloidosis occurs as hyperpigmented patches or plaques mainly on the back, whereas lichen amyloidosis typically appears as firm or hyperkeratotic papules on the extremities, specifically the legs. In both types, lesions are extremely pruritic. The amyloid deposits consist mainly of cytokeratin 5 originating from adjacent basal keratinocytes and are located predominantly in the papillary dermis.2 Several reports describe good responses to treatment with retinoids such as acitretin and etretinate.3–5 Most retinoids, however, lead to dryness of the skin, which may eventually deteriorate the itch. Therefore, we decided to use alitretinoin (9-cis-retinoid acid). It is a retinoid receptor pan-agonist with

© 2015 European Academy of Dermatology and Venereology

Letters to the Editor

885

(a)

(b)

Figure 1 Amyloid deposits in the papillary dermis. (a) Haematoxylin and eosin stain. (b) Pan-cytokeratin staining with MNF-116, which targets a variety of cytokeratins.

(a)

(b)

Figure 2 The status of the skin of the patient before and after treatment with alitretinoin is shown.

the capacity to bind all six known retinoid receptors. In contrast to isotretinoin or acitretin, it exerts strong anti-inflammatory and immune-modulatory effects without suppressing the activity of the sebaceous glands. It is used mainly for the treatment of chronic hand eczema, leading to durable remissions. Additional clinical efficacy has been reported in a variety of other cutaneous diseases such as ichthyosis or Darier’s disease.6,7 In the case presented here, alitretinoin showed rapid and robust activity in treating the papules without aggravating the xerosis. Taken together, we identify alitretinoin as a novel effective treatment option for cutaneous amyloidosis, specifically in patients with strong pruritus and sebostatic skin conditions.

4 Choi JY, Sippe J, Lee S. Acitretin for lichen amyloidosus. Australas J Dermatol 2008; 49: 109–113. 5 Ozcan A, Senol M, Aydin NE, Karaca S. Amyloidosis cutis dyschromica: a case treated with acitretin. J Dermatol 2005; 32: 474–477. 6 Lynde C, Cambazard F, Ruzicka T et al. Extended treatment with oral alitretinoin for patients with chronic hand eczema not fully responding to initial treatment. Clin Exp Dermatol 2012; 37: 712–717. 7 Ruzicka T, Lynde CW, Jemec GB et al. Efficacy and safety of oral alitretinoin (9-cis retinoic acid) in patients with severe chronic hand eczema refractory to topical corticosteroids: results of a randomized, doubleblind, placebo-controlled, multicentre trial. Br J Dermatol 2008; 158: 808–817. DOI: 10.1111/jdv.13048

J. K. Tietze,* M. V. Heppt, M. J. Flaig, P. Thomas Department of Dermatology and Allergy, Ludwig-Maximilian University, Munich, Germany *Correspondence: J. K. Tietze. E-mail: [email protected]. de

References 1 Schreml S, Szeimies RM, Landthaler M, Babilas P. Cutaneous amyloidosis. Hautarzt 2011; 62: 56–61. 2 Apaydin R, Gurbuz Y, Bayramgurler D, Muezzinoglu B, Bilen N. Cytokeratin expression in lichen amyloidosus and macular amyloidosis. J Eur Acad Dermatol Venereol 2004; 18: 305–309. 3 Carlesimo M, Narcisi A, Orsini D et al. A case of lichen amyloidosus treated with acitretin. Clin Ter 2011; 162: e59–e61.

JEADV 2016, 30, 852–909

An old lady with Pediculosis pubis on the head hair Editor Three species of lice are specific ectoparasites for human, Pediculus humanus capitis (head lice), P. humanus corporis (body lice) and Phtirus pubis (pubic lice). The head louse lives mainly on the human scalp. It has never demonstrated a real vectorial role.1,2 The body louse is the primary vector of the bacterial

© 2015 European Academy of Dermatology and Venereology

Successful treatment of lichen amyloidosus with oral alitretinoin.

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