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Dlo130076.Pdf Letters Figure. Clinical Image of Pityriasis Rosea Box. Proposed Diagnostic Criteria for Pityriasis Roseaa A patient is diagnosed as having pityriasis rosea if: 1. On at least one occasion or clinical encounter, he/she has all the essential clinical features and at least one of the optional clinical features, and 2. On all occasions or clinical encounters related to the eruption, he/ she does not have any of the exclusional clinical features. The essential clinical features are: 1. Discrete circular or oval lesions, 2. Scaling on most lesions, and 3. Peripheral collarette scaling with central clearance on at least two lesions. The optional clinical features are: 1. Truncal and proximal limb distribution, with less than 10% of lesions distal to mid-upper-arm and mid-thigh, 2. Orientation of most lesions along skin cleavage lines, and 3. A herald patch (not necessarily the largest) appearing at least Skin lesions present on the groin, genitals, and pubic mound show multiple oval, two days before eruption of other lesions, noted from patient sharply defined, coalescent, scaly, annular plaques surrounded by erythema. history or from clinical observation. The exclusional clinical features are: Approximately 20% of patients present with atypical or vari- 1. Multiple small vesicles at the center of 2 or more lesions, ant forms of PR, which are less readily recognized than typical 2. Two or more lesions on palmar or plantar skin surfaces, and 3. Clinical or serologic evidence of secondary syphilis. eruptions and may pose a diagnostic challenge.2,3 The mor- phologic characteristics of the eruption may be papular, ve- a This outline was first published by Chuh and Zawar6 and is reproduced here sicular, purpuric or hemorrhagic, or urticarial. Very small le- with permission. sions will be observed in papular PR, and PR with enormous plaques is known as pityriasis rosea gigantea of Darier. A mor- phologic variant characterized by atypical large patches that tend to be few in number and coalescent has been described. 2. Chuh A, Zawar V, Lee A. Atypical presentations of pityriasis rosea: case In this variant, commonly referred to as pityriasis circinata et presentations. J Eur Acad Dermatol Venereol. 2005;19(1):120-126. marginata of Vidal or limb-girdle PR, the eruption generally ap- 3. Brzezinski P, Sinjab AT. Pityriasis rosea in a 12-month-old infant. Our Dermatol pears in the axillae, the groin, or both, with the trunk and ex- Online. 2012;3(2):119-122. 4,5 tremities usually spared. A simple classification for atypical 4. Chukwudi NL. Comment: does pityriasis rosea koebnerize? Our Dermatol pityriasis rosea has been proposed by Chuh and Zawar (Box).6 Online. 2013;4(2):191. In our patient, the eruption fulfills all 3 essential clinical 5. Zawar V, Jerajani H, Pol R. Current trends in pityriasis rosea. Expert Rev features (discrete annular lesions, scaling, and peripheral col- Dermatol. 2010;5(3):325-333. larette scaling with central clearance on at least 2 lesions), all 6. Chuh A, Zawar V. Case reports and studies on pityriasis rosea: from number of patients to meta-analyses and diagnostic criteria. Our Dermatol Online.2012; 3 optional clinical features (relative truncal distribution, ori- 3(2):141-142. entation along skin cleavage lines, and herald patch), and none of the exclusional clinical features. This case has clinical fea- Cutaneous Hemophagocytosis After Alemtuzumab tures of localized PR, papular PR, and pityriasis circinata et mar- Injection in a Patient With Sézary Syndrome ginata of Vidal. It should also be noted that the involvement Alemtuzumab, a CD52 monoclonal antibody, is increasingly of penile and scrotal skin is rarely reported in PR. Physicians used for treating advanced cutaneous T-cell lymphomas in- should be aware of the wide spectrum of PR variants so that cluding Sézary syndrome (SS). While injection site reactions appropriate management and reassurance can be offered. are common, the finding of localized cutaneous hemophago- cytosis at the injection site without systemic hemophagocy- Piotr Brzezinski, MD, PhD tosis is rare. Anca Chiriac, MD, PhD Report of a Case | A woman in her 60s presented with a 2-year his- Author Affiliations: Dermatological Clinic, Sixth Military Support Unit, Ustka, Poland (Brzezinski); Department of Dermatology, Nicolina Medical Center, Iasi, tory of SS (clinical stage IVA [T4NXM0B2]). After multiple regi- Romania (Chiriac). mens failed, she was treated with subcutaneous alemtuzumab Corresponding Author: Piotr Brzezinski, MD, PhD, Department of injections (10 mg each) thrice weekly for 10 weeks and experi- Dermatology, Sixth Military Support Unit, os Ledowo 1N, 76-270 Ustka, Poland enced complete remission. However, her disease recurred 7 ([email protected]). months after therapy was completed. She restarted treatment Published Online: July 30, 2014. doi:10.1001/jamadermatol.2013.10505. with alemtuzumab and 1 month later developed large, tender, Conflict of Interest Disclosures: None reported. indurated plaques on the left lower abdomen and right thigh at 1. Chuh AA, Lee A, Molinari N. Case clustering in pityriasis rosea: a multicenter epidemiologic study in primary care settings in Hong Kong. Arch Dermatol. her injection sites (Figure, A). Analysis of a right thigh biopsy 2003;139(4):489-493. specimen (Figure, B) showed a deep dermal and subcutaneous jamadermatology.com JAMA Dermatology September 2014 Volume 150, Number 9 1021 Copyright 2014 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/01/2021 Letters Figure. Clinical and Pathologic Images From a Case of Cutaneous Hemophagocytosis After Subcutaneous Alemtuzumab Injection A B A, Clinical image of erythematous to brown indurated plaque on right lateral thigh following subcutaneous injection with alemtuzumab. B, Pathologically, a deep dermal infiltrate consisting of histiocytes, neutrophils, extravasated erythrocytes, and scattered atypical lymphocytes was present; histiocytes with phagocytosed neutrophils, lymphocytes, and erythrocytes were seen (hemophagocytosis; arrowheads) (hematoxylin-eosin, original magnification ×400). infiltrate of lymphocytes, histiocytes, and neutrophils consis- papular eruption. In virus-induced cases, the cutaneous tent with injection site reaction. Numerous histiocytes contained appearance may reflect the underlying cause. Typical histo- phagocytosed lymphocytes, erythrocytes, and/or neutrophils pathologic characteristics include a dermal perivascular lym- without features of vasculitis or vasculopathy. Immunohisto- phohistiocytic infiltrate, nuclear debris, extravasated eryth- chemical analysis showed a normal CD4:CD8 ratio of 3:1. The re- rocytes, and, rarely, hemophagocytosis in skin lesions.1 sults of blood tests and in situ hybridization assays for Epstein- We describe herein a case of localized cutaneous hemo- Barr virus (EBV)–encoded RNA and EBV DNA were negative. phagocytosis in an injection site reaction without evident sys- The dose of alemtuzumab was decreased, and then the full temic involvement. To our knowledge, only 4 cases of cutane- dose was resumed with dexamethasone premedication, and ous hemophagocytosis have been previously reported, with an finally alemtuzumab therapy was discontinued altogether ow- underlying condition identified in 3 of these 4 cases (lymphoma, ing to persistent, painful injection site reaction. The indu- autoimmune disease).2,3 A viral trigger was suspected but not rated erythematous plaques slowly resolved under treat- confirmed in the fourth case. In all cases, there was no evidence ment with clobetasol, 0.05%, ointment. The patient of extracutaneous hemophagocytosis, with the isolated skin subsequently started a clinical trial of therapeutic PI-3 (phos- findings possibly representing a form of leukocytoclastic vas- phatidylinositide 3) kinase inhibitor. culitis. Features of vasculitis were not identified in our case. Peripheral T-cell lymphomas (PTCLs) have also been asso- Discussion | Hemophagocytosis (the engulfment of erythro- ciated with development of HPS. Reactivation of EBV is thought cytes, their precursors, and occasionally white blood cells by to play a role, although it is unclear whether EBV-infected ma- typically benign histiocytes) involves multiple sites (spleen, lignant T cells initiate HPS or if latent EBV infection predis- bone marrow, lymph nodes), usually in association with he- poses to HPS.4 The development of HPS was reported in 2 of mophagocytic syndrome (HPS). Characterized by high fever, 14 patients with relapsed or refractory PTCL treated with ale- cytopenias, liver dysfunction, and coagulopathy, HPS can oc- mtuzumab and was attributed to EBV reactivation in the set- cur primarily (eg, inherited defects of cellular toxic effects) or ting of PTCL.5 Alemtuzumab has never been linked to HPS in secondarily (infections, autoimmune/rheumatologic dis- mycosis fungoides or SS and chronic lymphocytic leukemia eases, and malignant conditions, particularly lymphomas).1 (CLL). Recent data have shown that soluble CD52 detected in Secondary HPS may result from dysfunctional cytotoxic T and plasma of patients with CLL can form immune complexes with natural killer cells, leading to excess cytokine production and alemtuzumab.6 However, an underlying immune complex– uncontrolled activation of lymphocytes and histiocytes.1 mediated mechanism seems unlikely in our case in which the Cutaneous findings are seen in 6% to 65% of HPS cases. reaction was localized to the injection site and there was no
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