Phoma and Acremonium invasive fungal rhinosinusitis in congenital acute lymphocytic leukemia and literature review
Invasive rhinocerebral fungal infections are a difficult and often fatal problem in children with hematologic malignancies, with increasing reports of rare pathogens. We describe a case of congenital acute lymphoblastic leukemia (ALL) and invasive fungal rhinosinusitis involving Acremonium and Phoma species, not previously reported in invasive pediatric fungal rhinosinusitis, and review all published cases of human Phoma infections. A literature and institutional review for Phoma spp. was completed including patient demographics, infection site, immune status, treatment and outcome. A female neonate with acute lymphoblastic leukemia presented with hyperleukocytosis and advanced invasive Phoma and Acremonium spp. rhinosinusitis. Despite aggressive medical and surgical therapy, the disease progressed to a rhinocerebral infection with a fatal outcome. Twenty cases of Phoma spp. were found in a complete literature search, including 6 females and 14 males from 18months to 77years old. Infections were superficial in fifteen patients and involved deeper tissue in five patients, with sites including cutaneous, subcutaneous and deep tissue sites (eye, lung, extremity deep tissue compartments). This case highlights the risks and grave prognosis of fulminant invasive fungal rhinosinusitis in the context of congenital leukemia, and the potential for rare fungal species in neonates with immunosuppression.
[Show abstract] [Hide abstract] ABSTRACT: The etiologic agents of many invasive fungal infections are saprobes and opportunistic pathogens. Some of these fungi are darkly pigmented due to melanin production and traditionally have been named 'dematiaceous'. The melanised fungi cause a wide array of clinical syndromes ranging from superficial to deep-seated infections. Diagnosis relies on histopathological examination of clinical specimens and of examination of cultures. Sequencing is recommended for accurate species identification, especially for unusual or newly described pathogens. In cases of mycetoma and chromoblastomycosis, pathognomonic histological findings are very useful and the Fontana-Masson stain, specific for melanin, usually confirms the diagnosis. There are no standardized therapies but voriconazole, posaconazole and itraconazole demonstrate the most consistent in vitro activity against this group of fungi. Oral itraconazole has been considered the drug of choice, given the extensive clinical experience with this drug. However, voriconazole may presumably be superior for central nervous system infections due to its ability to achieve good cerebrospinal fluid levels. Posaconazole is a well-tolerated alternative drug, backed by less clinical experience but with excellent salvage treatment results after failure of other antifungals. Amphotericin B has been useful as alternative therapy in some cases. Combination antifungal therapy is recommended for cerebral abscesses when surgery is not possible and for disseminated infections in immunocompromised patients. This article is protected by copyright. All rights reserved.0Comments 34Citations
- "Pycnidia are black, globose, subglobose, or pyriform and either submerged or on the surface of agar. Conidia (pycnidiospores) are produced from the phialides that line the inner wall of pycnidia and are hyaline, one-celled, elliptical, rod shaped, or curved [164,464]. A PCR assay for detecting P. exigua DNA in deparaffinized lung-biopsy material has been developed . "
- "Pycnidia are black, globose, subglobose, or pyriform and either submerged or on the surface of agar. Conidia (pycnidiospores) are produced from the phialides that line the inner wall of pycnidia and are hyaline, one-celled, elliptical, rod shaped or curved [164,460]. A PCR assay for detecting Phoma exigua DNA in deparaffinized lung biopsy material has been developed . "
- [Show abstract] [Hide abstract] ABSTRACT: Most human mycoses are incited by fungi that replicate by the production of asexual propagules (usually called conidia), which are readily dispersed throughout nature and also in vitro. Fungi that are acquired in this manner include a diversity of yeasts and dermatophytes, as well as those whose acquisition is primarily pulmonary, such as the thermally dimorphic primary pathogens, and several opportunistic genera such as Aspergillus, Fusarium and others. Recent reports, however, indicate an increasing number of infections, ranging from superficial to systemic, that are due to coelomycetous fungi. In these fungi, the asexual spores are produced within fruiting bodies referred to as a conidiomata. As these conidia are less frequently airborne, the method of acquisition is commonly by traumatic implantation of plant material or by a diversity of contaminated fomites, rather than by inhalation. These infections are also more commonly seen in immunocompromised individuals. In this work, we have attempted to provide a historial overview as well as the current taxonomy for this group of fungi, an up-to-date assessment of the scope of infections caused by various coelomycetous genera, their clinical entities, useful tools for isolation and identification, and general guidelines for the appropriate management and treatment of these mycoses.0Comments 2Citations