A rare case of Sweet syndrome secondary to melioidosis

Abstract

BackgroundMelioidosis is an emerging infection in South Asia caused by Burkholderia pseudomallei with various clinical presentations that include pneumonia, bacteraemia, arthritis, and deep-seated abscesses. Various cutaneous manifestations have been described in association with melioidosis. However Sweet Syndrome secondary to melioidosis has not been reported in the literature. Herein we describe the first case of Sweet syndrome secondary to melioidosis.Case presentationA 53-year-old previously healthy Sri Lankan female presented with high-grade fever, painful oral ulcers, odynophagia and multiple bilateral cervical lymphadenopathies for 1 month. She also had a loss of appetite and weight. She had oral ulcers and bilateral blepharitis. Dermatological examination revealed multiple tender papules with a mamillated appearance and targetoid lesions with a yellowish centre over the face, upper trunk and upper limbs. She also had multiple tender subcutaneous nodules over the extensor aspect of upper limbs. Her inflammatory markers were significantly elevated. Aspirate from a submental lymph node abscess revealed the growth of Burkholderia pseudomallei. Melioidosis antibody titer was > 10,240. The histology of the skin lesions of the face and left forearm showed a prominent neutrophilic infiltrate in the dermis and the morphological features were in favour of Sweet syndrome with panniculitis. She was started on intravenous meropenem 2 g daily and showed rapid clinical improvement with the disappearance of skin lesions as well as a reduction in inflammatory markers.ConclusionSweet syndrome is an uncommon inflammatory disorder known to be associated with upper respiratory tract and gastrointestinal infections, malignancies and the use of certain drugs. Melioidosis is an emerging infection with various cutaneous manifestations. This is the first case of melioidosis causing the secondary sweet syndrome. It emphasizes the importance of considering melioidosis as a potential aetiology in patients with Sweet syndrome.

New Practical Aspects of Sweet Syndrome

“…A 2020 Thai study of 88 patients with nontuberculous mycobacterial (NTM) infections reported the prevalence of SS to be 3.4%, and concluded SS to be the most common reactive dermatosis in NTM infections [120]. The incidence of SS in other bacterial infections is less well characterized, although SS has been reported in the setting of infection with Burkholderia pseudomallei [121], Erysipelothrix rhusiopathiae [122], Mycobacterium tuberculosis [123], and Treponema pallidum [124,125]. Viruses have also been reported in association with SS, with Hepatitis C [126], human immunodeficiency virus [127,128], and severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) [129][130][131] being recently reported as culprits.…”

A rare case of meliodosis presenting as lupus panniculitis with intermittent fever

“…Patients with melioidosis can get various types of skin lesions as well as skin abscesses [9]. There are cases reported with Sweet syndrome and erythema nodosum associated with melioidosis [10,11]. However, cases of melioidisis presenting as panniculitis, especially as lupus or lobular panniculitis, are very rare.…”

Neutrophilic Dermatoses: a Clinical Update

“…Classic SS encompasses idiopathic SS and SS associated with infections, vaccinations, inflammatory disorders, and pregnancy. Upper respiratory infections are most commonly cited as an infectious trigger; however, a range of bacterial, viral, and fungal infections have been implicated [24][25][26][27][28][29][30][31]. Since 2019, SS has been reported following coronavirus disease 2019 (COVID-19) and rarely following vaccination with the Oxford-AstraZeneca, Moderna, and Pfizer-BioNTech vaccines [32 •, 33-36].…”