Ecthyma gangrenosum in an infant with neutropenia
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IMAGES IN HEMATOLOGY
Ecthyma gangrenosum in an infant with neutropenia Nanae Kikuchi1 · Hiro Nakao2 · Yukio Matsumoto3 · Kazue Yoshida3 · Masashi Okai4 · Toshinao Kawai4 · Mitsuru Kubota2 · Akira Ishiguro1,5 Received: 7 August 2020 / Revised: 9 November 2020 / Accepted: 10 November 2020 © Japanese Society of Hematology 2020
Keywords Ecthyma gangrenosum · Neutropenia · Pseudomonas aeruginosa · Immunocompromised patient Abbreviations ANC Absolute neutrophil count EG Ecthyma gangrenosum A previously healthy 1-year-old girl presented with fever and two skin lesions on her right arm. Five days prior to admission, she had fever and small papules that developed into a necrotic ulcer on her right shoulder the next day. Two days prior to admission, a similar ulcer developed on the right upper arm. There was no family history of immunodeficiency or hematological disease. Physical examination revealed two necrotic ulcers on the right arm that were 2 cm and 1 cm in size, with a black eschar and erythematous rim (Fig. 1a). There were no skin lesions in the anogenital or axillary areas. Laboratory tests showed a leukocyte count of 6500/μL and absolute neutrophil count (ANC) of 65/μL. The results of immunological screening, including serum levels of immunoglobulins and complement (C3 and C4), and lymphocyte subsets were unremarkable. Wound culture yielded Pseudomonas aeruginosa, and the condition was diagnosed as ecthyma gangrenosum (EG). Blood cultures were negative. Pathology of the ulcer showed a broadly ulcerated zone of necrotic dermis * Hiro Nakao nakao‑[email protected] 1
Center for Postgraduate Education and Training, National Center for Child Health and Development (NCCHD), Tokyo, Japan
2
Department of General Pediatrics and Interdisciplinary Medicine, National Center for Child Health and Development, 2‑10‑1 Okura, Setagaya‑ku, Tokyo 157‑8535, Japan
3
Division of Dermatology, NCCHD, Tokyo, Japan
4
Division of Immunology, NCCHD, Tokyo, Japan
5
Division of Hematology, NCCHD, Tokyo, Japan
and subcutis with neutrophilic infiltration. ANC values were 65–1688/μL during hospitalization. Bone marrow aspirate exhibited hypercellularity with myeloid hyperplasia without excess of blasts; however, there was a decrease in segmented neutrophils. Neutrophil-specific antibody was not detected. There were no mutations in ELANE, HAX-1, WAS, CSF3R, or VPS45.L4. Intravenous infusion of cefepime was initiated on the first day of hospitalization, and debridement was performed on the first and third days of hospitalization. After these treatments, the patient’s condition became stable and the necrotic area decreased (Fig. 1b, c). Intravenous antibiotic therapy was continued for 3 weeks. She still demonstrated chronic neutropenia (ANC 173–640/μL) 1 year later, but without serious infections. These observations indicated that she had chronic benign neutropenia. EG is characterized by often fatal cutaneous infections caused by Gram-negative organisms, usually P. aeruginosa. EG commonly occurs in immunocompromised patients, for
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