Zigomicosis y síndrome hemofagocítico en un paciente inmunocompetente: informe de caso / Zygomycosis and hemophagocytic lymphohistiocytosis in an immunocompetent patient: a case report

Introducción: la zigomicosis es una infección fúngica poco frecuente, con alta tasa de mortalidad y de mal pronóstico. Afecta principalmente a pacientes inmunocomprometidos. La asociación con el síndrome hemofagocítico es extremadamente inusual, más aún en pacientes inmunocompetentes, con pocos ejemplos registrados en la literatura. Caso clínico: se presenta el caso de un paciente masculino inmunocompetente de 40 años con diagnóstico de mucormicosis y síndrome hemofagocítico que evoluciona desfavorablemente, con fallo multiorgánico, a pesar de los esfuerzos médicos. Conclusión: la asociación de mucormicosis con síndrome hemofagocítico en un paciente inmunocompetente es extremadamente rara; existen pocos casos informados en Latinoamérica. Debemos tener presente esta asociación, ya que requiere un tratamiento agresivo y soporte vital avanzado. (AU)

Introduction: zygomycosis is a rare fungal infection that carries with high mortality rates. This poor prognosis, rapidly progressive infection mainly affects immunocompromised patients. The association with hemophagocytic lymphohistiocytosis is extremely unusual, even more in immunocompetent patients, with few cases reported. Case: we present the case of an immunocompetent male patient who was diagnosed with zygomycosis and hemophagocytic lymphohistiocytosis. Despite medical efforts he developed multiorganic failure. Conclusion: the association of mucormycosis with hemophagocytic lymphohistiocytosis in an immunocompetent patient is exceptional with few cases reported in Latin America. We must always suspect this association considering they require aggressive treatment and advanced life support. (AU)

Meningoencefalitis zigomicótica piogranulomatosa en un puercoespín de cola pren sil (Coendou prehensilis) / Piogranulomatous zygomycotic meningoencephalitisb in a prehensile tailed porcupine (Coendou prehensilis)

Con el término de zigomicosis (ZM) se designa un grupo de enfermedades causadas por hongos de la clase Zygomicetes, de los órdenes Mucorales y Entomophthorales, de varios géneros y especies. La presentación de ZM en animales ocurre esporádicamente en numerosas especies tanto en animales domésticos como silvestres (incluso en mamíferos marinos) y son pocos los casos comprobados de aislamiento e identificación del agente causal. Pocos hongos son considerados como neurotrópicos ya que tienden a localizarse en el SNC después de invadir el hospedador; sin embargo, la mayoría de los casos de meningitis o meningoencefalomielitis son parte de micosis sistémicas como la Candidiasis, la Criptococosis, la Aspergilosis o la ZM. El presente trabajo documenta el hallazgo de una meningoencefalitis zigomicotica secundaria a un trauma craneoencefálico en un puercoespín de cola prensil (Coendou prehensilis), siendo una enfermedad rara infrecuente en el sistema nervioso central tanto en humanos como en animales y adicionalmente sin reportes previos en esta especie.

With the word Zygomycosis (ZM) are designating a group of diseases caused by fungi of the class Zygomicetes, the orders Mucorales and Entomophthorales, of several genera and species. The presentation of ZM in animals occurs sporadically in many species both in domestic and wild animals (including marine mammals), and are checked few cases of isolation and identification of the causative agent. Few fungi are considered to be neurotropic because they tend to be located in the CNS after invading the host; however, most cases of meningitis or meningoencephalomyelitis are part of systemic fungal infection such as Candidiasis, Cryptococcosis, Aspergillosis or ZM. This paper documents the discovery of a zigomicotic meningoencephalomyelitis secondary to a head trauma in a prehensile-tailed porcupine (Coendou prehensilis), being a rare disease infrequent in the central nervous system both in human and animals and additionally without previous reports in this specie.

Rare mycoses of the oral cavity: a literature epidemiologic review.

Stomatologic fungal infections display different etiologies, pathogenesis, and clinical presentations. The incidence of rare mycoses of oral cavity is very low. These infections can involve both immunocompromised and immmunocompetent patients with common predisposing factors, such as diabetes or suffering from diseases causing immune system impairment. Oral mycoses can cause acute, chronic, and mucocutaneous lesions. Candidiasis is the most common mouth mycosis. Although occasionally primary mouth pathogens, Cryptococcus spp. or filamentous fungi (Aspergillus spp. and zygomycetes) can cause oral mycoses, with the oral localization more commonly secondary to a more serious systemic infection. The diagnosis of oral mycoses is based on clinical examination; for yeasts, culture is necessary to identify the etiologic agents; for filamentous fungi, in particular for zygomycetes and dimorphic, a definitive diagnosis can be made by histologic examination and pertinent stains with or without isolation of the fungus from the same site.

Zigomicosis cutánea primaria por Rhyzopus oryzae en una niña con leucemia linfoblástica aguda tipo B / Primary cutaneous zygomycosis by Rhyzopus oryzae in a girl diagnosed with an acute lymphoblastic leukemia B type

La Zigomicosis es una infección infrecuente causada por hongos oportunistas integrantes del orden Mucorales, que se presenta en pacientes de alto riego como en: leucemia, linfomas con neutropenia prolongada, cetoacidosis diabética, malnutrición severa, ruptura de la integridad de la barrera cutánea y terapia inmunosupresora. Se presenta un caso de Zigomicosis cutánea en una paciente pediátrica con leucemia linfoblástica aguda de tipo B, con severa neutropenia y tratamiento con corticoides. A los cinco días de su hospitalización desarrolló en el antebrazo (zona de punción venosa), una lesión indurada, eritematosa, que progresó y ulceró. A partir de exudados y biopsias del tejido subcutáneo se realizaron exámenes microscópicos directos con KOH, cultivos en agar Sabouraud y estudio histológico a través de técnicas convencionales de hematoxilinaeosina y PAS. Los análisis de los materiales clínicos revelaron la presencia de hifas hialinas, no tabicadas, gruesas, compatibles con un Zygomycete. En todos los tubos se obtuvo abundante desarrollo de un hongo filamentoso, identificado como Rhizopus oryzae. Posteriormente a la escisión quirúrgica y tratamiento con anfotericina B se obtuvo una evolución favorable del paciente hasta el presente.

Zygomycosis is an infrequent infection caused by opportunistic fungi which belong to the order Mucorales and which is present in high risk patients diagnosed with : leukemia, lymphomas with prolonged neutropenia, diabetic cetoacidosis, severe malnutrition, rupture of the entire cutaneous barrier and immunesuppressing therapy. This paper deals with a case of cutaneous Zygomycosis in a pediatric patient diagnosed with acute lymphoblastic leukemia B type, suffering a severe neutropenia and corticosteroid treatment. On the fifth day of hospitalization, her forearm (venous puncture zone) showed an indured, erimatose lesion which progressed and ulcerated. Collection of exudates and biopsies of subcutaneous tissue served to carry out direct microsco-pic examinations with KOH, cultures in Sabouraud Agar and a histologic study through conventional hematoxilin-eosin and PAS techniques. Analyses of the clinical materials revealed the presence of hyaline, not septated and broad hyphae suitable to a Zygomycete. In all the tubes there was an abundant development of filamentous fungus identified as Rhizopus oryzae. After the surgical scission and treatment with anfotericine B, the patient showed a favorable evolution up to now.

Emerging moulds: Fusarium, Scedosporium and Zygomycetes in transplant recipients.

PURPOSE OF REVIEW: The incidence of non-Aspergillus mould infections in transplant recipients has increased over the past decade. This paper will review recent data regarding the epidemiology, clinical manifestations, treatment and the outcome of infections caused by Fusarium spp., Scedosporium spp. and the Zygomycetes in transplant recipients. RECENT FINDINGS: Although the reasons for the increased incidence of non-Aspergillus mould infections in transplant recipients are not very clear, changes in transplant procedures, resulting in more severe immunosuppression, seem to play a major role, as suggested by recent epidemiologic data. Infections caused by these fungi have a tendency to be disseminated, and the prognosis is usually poor because the fungi are resistant to most of the available antifungal agents. New drugs, particularly the new azoles, may have a role in the treatment and prophylaxis of these infections, but the available data are still scant. SUMMARY: The understanding of the changing epidemiology of fungal infections and their implications for the prognosis of transplant recipients are important to better evaluate the impact of new transplant procedures.

[Zygomycosis following liver transplantation in adults: report of three cases]. / Zigomicose após transplante hepático em adultos: relato de três casos.

We report three cases of zygomycosis following liver transplant in a series of 300 patients. Diagnosis was determined via anatomicopathological examination (on necropsy in two cases and during surgery in one case). The disease had different manifestations: rhinomaxillary, gastrointestinal and, in one case, it compromised the liver artery anastomosis. In this case, surgical removal of the affected region and use of amphotericin B achieved resolution.

Cavernous sinus thrombosis caused by zygomycosis after unrelated bone marrow transplantation.

Invasive zygomycosis is a devastating fungal infection occurring as an opportunistic infection after bone marrow transplantation (BMT). Sinusitis can lead to fungal infection in immunosuppressed patients, and cavernous sinus thrombosis, an uncommon condition in immunocompetent patients, typically follows an infection involving the medial third of the face, nose, or paranasal sinuses. Patients undergoing unrelated-donor BMT (UD-BMT) are prone to develop life-threatening infections because of poor recovery of cellular immunity. Despite adequate clinical evaluation and treatment, the prognosis of patients with invasive fungal infections is dismal, especially when intracerebral structures are affected. We describe a case of a patient who underwent an UD-BMT and developed cavernous sinus thrombosis after sinusitis due to zygomycosis. Moreover, he also had disseminated fungal (Zygomycetes and Aspergillus) and viral (cytomegalovirus and adenovirus) infections.

Zigomicose: apresentações cutâneas e bucais / Zygomycosis: buccal and cutaneous presentations

A zigomicose é uma antes considerada rara, com tendência a aumentar, devido às condições de imunocomprometimento, em especial com o advento da Aids. Os autores apresentam revisão da literatura sobre as apresentações cutâneas e mucosas da doença. Tanto as manifestações tegumentares como bucais apresentam grande variabilidade nas lesões fundamentais. Para o diagnóstico são considerados o quadro clínico, os fatores predisponentes, a pesquisa fúngica e o exame histopatológico.