Palatal mucormycosis in an immune-competent infant following dengue haemorrhagic fever. A rare disease entity, treated with a challenging course of IV liposomal amphotericin B for 270+ days; longest duration reported from Sri Lanka

Author:

Pathiraja Hashan1,Munasighe Rakitha2,Gunapala Rasika2,Fernando Jerrad2,Gunaratne Sandini2,Pemasiri Chethana3,Jayasekera Primali3

Affiliation:

1. University of Kelaniya

2. Lady Ridgeway Hospital for Children

3. Department of Mycology, Medical Research Institute

Abstract

Abstract

Background Mucormycosis is an emerging global illness with significant morbidity and mortality. Causative fungi can spread through inhalation of sporangiospores or direct inoculation through damaged skin or mucosa in susceptible patients, especially those with impaired immune systems. Here we report a rare occurrence of the disease in an immunocompetent infant following dengue haemorrhagic fever, highlighting the treatment course which is the longest duration reported from Sri Lanka so far. Case presentation A 4-month-old infant was admitted to the medical intensive care unit (ICU) after experiencing dengue haemorrhagic fever, which was complicated by multi-organ dysfunction and required intubation. After recovery, he was found to have a necrotic lesion in the palate, which was confirmed to be mucormycosis following biopsy. Computed tomography (CT) and Magnetic resonance imaging (MRI) scans showed erosions of the hard palate and involvement of the paranasal sinuses, orbital floor and soft palate without brain or eye lesions. The child was started on intravenous (IV) liposomal amphotericin B and required several debridement surgeries. Radiological studies showed persistent active bone lesions and antifungals were continued until radiological, mycological and clinical clearance was achieved. Intravenous amphotericin B was given for 270+ days required central lines and was complicated by venous thrombosis. A palatal prosthesis was inserted until a definite palatal repair is done. The child is clinically well and thriving. Laboratory evaluations showed normal IgG and subclasses, IgA, IgM, and IgE levels, HIV testing was negative, nitroblue tetrazolium test(NBT) was normal and metabolic screening was negative. Conclusion Though mucormycosis usually occur in children with immunosuppression or metabolic syndromes, it can occur in immunocompetent children, especially following a critical illness with high lactic acid levels, as in our case. It’s important to manage these children under multi-disciplinary care and complete treatment until there is evidence of radiological clearance to achieve better outcome.

Publisher

Springer Science and Business Media LLC

Reference18 articles.

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3. Disease entities in mucormycosis;Serris A;J Fungi (Basel),2019

4. Rhino-orbito-cerebral mucormycosis infection in a 4-year-old Egyptian girl;Sorour A;Med Mycol Case Rep,2022

5. Australia and New Zealand mycoses interest group of the australasian society for infectious diseases. Mucormycosis in Australia: contemporary epidemiology and outcomes;Kennedy KJ;Clin Microbiol Infect,2016

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