Acta Dermato-Venereologica 99-7CompleteContent | Page 18

685 SHORT COMMUNICATION Localized Pegfilgrastim-induced Neutrophilic Dermatosis with Tissue G-CSF Expression: A Mimicker of Sweet’s Syndrome Toshio ICHIKI 1 , Kazunari SUGITA 1 , Hiroyuki GOTO 1 , Masahisa SHINDO 2 and Osamu YAMAMOTO 1 1 Department of Medicine of Sensory and Motor Organs, Division of Dermatology, Faculty of Medicine, Tottori University, Tottori, and 2 Department of Dermatology, National Hospital Organization Hamada Medical Center, Hamada, Japan. E-mail: [email protected] Accepted Mar 8, 2019; E-published Mar 8, 2019 Pegfilgrastim is a granulocyte-colony stimulating factor (G-CSF) that has recently been developed by using the pegylation technology, whereby a polyethylene glycol moiety is attached to filgrastim. Although filgrastim- induced Sweet’s syndrome has been often reported, pegfilgrastim-induced Sweet’s syndrome is rare. Here we report an additional case of neutrophilic dermatosis induced by pegfilgrastim. Moreover, we have confirmed, for the first time, the local expression of G-CSF and we discuss the pathophysiology of the lesion. CASE REPORT A 59-year-old woman visited us for an evaluation of a red plaque on her right ankle (Fig. 1). She had been diagnosed with breast cancer (cT4bN3aM1, Stag4) at the age of 56 and had been re- peatedly treated with adjuvant chemotherapy and lenograstim (a conventional G-CSF) after a right mastectomy with a right axillary lymphadenectomy. To decrease the frequency of injections, leno- grastime was replaced by pegfilgrastim. Ten days after the initial pegfilgrastim administration, she observed an eruption on her left ankle. Physical examination revealed a red plaque with a strong induration of the outer side of the right ankle, partly covered with crusts and scales (Fig. 1). Her body temperature was 37.1°C, and she had no palpable lymph nodes. Pertinent laboratory findings included a white blood cell (WBC) count of 18.0 × 10 3 /µl with 91% neutrophils and 6% lymphocytes. C-reactive protein level was 2.77 mg/dl. Histopathologically, the biopsy specimen showed lobular neutrophilic panniculitis, without fat necrosis and a lack of nodular granulomatous phlebitis, and a dense mixed cellular infiltration, mainly composed of polymorphonulear leukocytes in the deep dermis and subcutis (Fig. 2a, b). There was also an aggregation of histiocytes (Fig. 2c). Immunohistochemically, G-CSF was detected in the cytoplasm of the infiltrating histiocytes (Fig. 2d). Recently, diagnostic criteria for the drug-induced Sweet’s syn- drome have been established, separately from those for the classic Sweet’s syndrome (1). According to these criteria, we could not make a diagnosis of the drug-induced Sweet’s syndrome because she was afebrile. Other conditions, including clinical findings, underlying cause and clinical course, fulfilled the criteria. The differential diagnosis should include erythema nodosum and ery­thema induratum. However, the present case shows a pre- dominantly lobular panniculitis and a lack of a granulomatous vasculitis of arterioles, lack of fat necrosis, and a lack of nodular granulomatous phlebitis exclude these diagnoses (2–4). Finally, we made a diagnosis of subcutaneous neutrophilic dermatosis comparable to Sweet’s syndrome. The lesion sponta- neously and rapidly cleared within 2 weeks without any special treatment except for the cessation of pegfilgrastim. Instead, treat- ment with lenograstim was restarted and similar symptoms have not been observed for over 1 year. DISCUSSION We have presented a case of subcutaneous neutrophilic dermatosis comparable to Sweet’s syndrome, induced by pegfilgrastim and the existence of G-CSF in the tis- sue. G-CSF-induced neutrophilic dermatoses, including Sweet’s syndrome, have been often reported (5). How­ ever, to the best of our knowledge, only 3 cases of Sweet’s syndrome (6–8) and one case of pyoderma gangrenosum (9) induced by pegfilgrastim have been reported so Fig. 1. A red plaque with strong induration of the outer side of the right ankle partly covered with crusts and scales. This is an open access article under the CC BY-NC license. www.medicaljournals.se/acta Journal Compilation © 2019 Acta Dermato-Venereologica. doi: 10.2340/00015555-3170 Acta Derm Venereol 2019; 99: 685–686