Published online Sep 26, 2022. doi: 10.12998/wjcc.v10.i27.9768
Peer-review started: January 11, 2022
First decision: March 15, 2022
Revised: March 26, 2022
Accepted: August 14, 2022
Article in press: August 14, 2022
Published online: September 26, 2022
Giant cellulitis-like Sweet syndrome (SS) is a rare subtype of SS, and reports of the combined histiocytoid type of pathology are scarce. Here, we report a case of SS with distinctive clinical presentations and which was difficult to distinguish from cellulitis. By sharing this case and a discussion of the related literature in detail, we aim to provide clinicians with new insights into the characteristics of histiocytoid giant cellulitis-like (HGC)-SS and the pathogenesis of SS.
A 52-year-old male was admitted after experiencing progressive fatigue for 1 mo and tongue swelling with pain for 1 d. He was diagnosed with myelodysplastic syndrome (MDS) and angioneurotic edema of the tongue and floor of the mouth. However, 7 d after examination by sternal aspiration, a violaceous, tender, and swollen nodule developed at the site, with poorly demarcated erythema of the surrounding skin. Considering his profile of risk factors, the diagnosis of cellulitis was made and he was administered broad-spectrum antibiotics. When the lesion continued to worsen and he developed chills and fever, pathogenic and dermatopathological examination led to the diagnosis of HGC-SS. Treatment with prednisone led to the fever being relieved within 24 h and the skin lesion being resolved within 1 wk. The patient refused intensive treatment and was instead given thalidomide, erythropoietin, stanozolol, and supportive care. The prednisone was gradually tapered, with no signs of recurrence, but he died 2 mo later of severe pneumonia.
HGC-SS demonstrates unique manifestation. SS and leukemia cutis share cytological origin. Myelofibrosis and SS are adverse prognostic factors for MDS.
Core Tip: We describe the case of a 52-year-old male with myelodysplastic syndrome who developed histiocytoid giant cellulitis-like Sweet syndrome (SS) at a sternal puncture site. The patient had unique clinical presentations that have never been reported before, and the disease profile was difficult to distinguish from cellulitis. Cellulitis that has failed to respond to broad-spectrum anti-microbial therapy requires a skin biopsy. For patients with concurrent myeloid neoplasm and SS, intensive treatment is necessary since SS and myeloid leukemia cutis may be different stages of the same disease and both indicate a poor prognosis.