Rhinocerebral zygomycosis with pansinusitis in a 14-year-old girl with type 1 diabetes: a case report and review of the literature

Abstract

BackgroundZygomycosis is a rare life-threatening fungal infection affecting mostly patients with predisposing conditions such as diabetes mellitus, immunodeficiency, haemochromatosis or major trauma.MethodsWe describe a case of rhinocerebral zygomycosis in a girl with type 1 diabetes and review previous published cases and treatment options.ResultsA 14-year-old girl with type 1 diabetes mellitus occurred with dental pain, facial swelling, ecchymosis and left eye decreased visual acuity, unresponsive to antibiotic therapy. The coltures of the sinusal mucosa were positive for fungal species belonging to the Zygomycetes. She performed antifungal therapy with posaconazole (POS) with a very slow improvement and a poor glycemic control, leading to blindness of the left eye.ConclusionOur report adds further awareness on rhinocerebral zygomycosis and emphasizes on urgent diagnosis and timely management of this potentially fatal fungal infection through an adequate treatment.

Successful treatment of invasive mucormycosis with isavuconazole in pediatric patients

“…One clinical survey of twelve pediatric cases of invasive mucormycosis reported an overall mortality of 67%; for nonsoft tissue infections, the outcome was particularly dismal, with 88% mortality . A literature review specific to pediatric rhinocerebral zygomycosis, which included 28 cases, reported an overall mortality of 20% in patients with type 1 diabetes and 50% in those with other predisposing factors—mainly malignancy …”

Infectious diseases associated with pediatric type 1 diabetes mellitus: A narrative review

“…Zygomycosis is a rare life-threatening opportunistic fungal infection in humans, that can complicate both diabetes mellitus and immunodeficiency syndromes characterized by defects of the cell-mediated immunity ( 116 ). In particular, RhinOCerebral Mucormycosis (ROCM) is the most frequent form of mucormycosis in patients with diabetes mellitus ( 117 ).…”

“…Among the mechanisms involved in the higher incidence of mucormycosis in diabetic patients, there are the greater availability of glucose, a lower response of T-cells, and the increased expression of host receptors mediating the invasion of epithelial cells ( 119 ). Indeed, in the case of ketoacidosis, the fungal growth is favored by the low pH, due to hyperglycemia as well as higher levels of free iron ions ( 116 ). Furthermore, hyperglycemia and the associated metabolic acidosis, are responsible for impaired neutrophil function, neuropathies, and vascular insufficiency, which lead to a diminished resistance to infections and altered tissue response ( 116 ).…”

“…Indeed, in the case of ketoacidosis, the fungal growth is favored by the low pH, due to hyperglycemia as well as higher levels of free iron ions ( 116 ). Furthermore, hyperglycemia and the associated metabolic acidosis, are responsible for impaired neutrophil function, neuropathies, and vascular insufficiency, which lead to a diminished resistance to infections and altered tissue response ( 116 ).…”

“…Intravenous liposomal amphotericin B successfully penetrates the central nervous system, thus being used for invasive mucormycosis ( 122 ). Posaconazole is a new triazole with a broad antifungal spectrum and its administration seems to be associated with a higher survival rate ( 116 ). Di Coste et al.…”