Osteomielite maxilar associada ao COVID-19: mucormicose ou não? Uma série de casos / Maxillary osteomyelitis associated with COVID-19: mucormycosis or not? A series of cases

Aim: A series of cases have been presented involving the oral cavity focusing on the presentation, diagnosis and treatment of mucormycosis that can form a basis for successful therapy. Background: The management of severe coronavirus disease (COVID-19) in conjunction with comorbidities such as diabetes mellitus, hematological malignancies, organ transplants, and immunosuppression have led to a rise of mucormycosis which is an opportunistic infection. Cases Description: The various forms that have been enlisted till date are rhino-cerebral, rhino-orbital, gastrointestinal, cutaneous, and disseminated mucormycosis. From the dentistry and maxillofacial surgery perspective, the cases depicting extension of mucormycosis into the oral cavity have been less frequently recorded and thus, require a detailed study. The patients that reported to our private practice had non-tender swelling, draining sinuses and mobility of teeth. A similarity was observed in the clinical signs both in osteomyelitis and mucormycosis. Thus, a histopathological examination was used to establish the definitive diagnosis. Conclusion: Mucormycosis is a life threatening pathology that requires intervention by other branches to make an early diagnosis and commence the treatment. The characteristic ulceration or necrosis is often absent in the initial stage and thus, histopathological examination and radiographic assessment are required to formulate a definitive diagnosis. Early intervention is a necessity to avoid morbidity. The treatment involves surgical debridement of the necrotic infected tissue followed by systemic antifungal therapy. Mucormycosis has recently seen a spike in its prevalence, post the second-wave of coronavirus pandemic in India. It was seen commonly in patients with compromised immunity, diabetes mellitus, hematological malignancies, or on corticosteroid therapy. Mucormycosis invading the palate mostly via maxillary sinus has been less frequently described. In the post-COVID era the features associated with mucormycosis involving oral cavity, should warrant a possible differential diagnosis and managed appropriately. (AU)

Objetivo: Apresentar uma série de casos com enfâse na apresentação, diagnóstico e tratamento da mucormicose oral, assim como uma revisão sistemática que sirva como base para estabelecimento de terapias de sucesso. Introdução: A forma severa da infecção por coronavirus (COVID-19) associada a diabetes mellitus, doenças hematológicas malignas, transplante de órgãos e imunossupressão levaram a um aumento das infecções oportunistas de mucormicose. Descrição dos Casos: As diversas apresentações clínicas que foram descritas até o momento são a rinocerebral, rino-orbital, gastrointestinal, cutânea e mucormicose disseminada. No que concerne a odontologia e a cirurgia maxillofacial, os casos que apresentam extensão de mucormicose para cavidade oral tem sido menos reportados e assim requerem mais estudos. Os pacientes que compareceram a nossa clínica apresentavam aumento de volume endurecido, drenagem de fluidos dos seios maxilares e mobilidade dentária. Clinicamente tanto a osteomielite quanto a mucormicose apresentaram-se de forma semelhante. Assim, análise histopatológica foi utilizada para estabelecimento do diagnóstico definitivo. Conclusão: A mucormicose é uma patologia grave que requer intervenção precoce para estabelecimento do tratamento. A ulceração e necrose características usualmente estão ausentes nos estágios iniciais da lesão, assim análise histopatológica e radiográfica são necessárias para o diagnóstico final. Intervenção precoce é necessária para diminuir a morbidade. O tratamento envolve o debridamento cirúrgico da área necrosada seguida de terapia antifúngica sistêmica. Recentemente, houve um aumento nos casos de mucormicose, após a Segunda onda da pandemia de COVID-19 na índia. Os casos acometiam principalmente pacientes imunocomprometidos, com diabetes mellitus, doenças hematológicas malignas e em uso de corticosteróides. A mucormicose invadindo o palato pelos seios maxilares foi raramente descrita. Na era pós-COVID a mucormicose envolvendo a cavidade oral deve entrar no painel de diagnósticos diferenciais para que o tratamento adequado possa ser instituído precocemente.(AU)

Riesgo de mortalidad en infecciones invasivas por Mucor SPP. en niños con enfermedades hemato-oncológicas: revisión sistemática / Mortality risk of invasive Mucor spp. infections in children with cancer: A systematic review

Los estudios sobre la infección fúngica invasiva (IFI) por Mucor spp. en pacientes pediátricos con patología hematooncológica, son de baja solidez científica, lo que dificulta conocer en profundidad sus características y evolución. Con el objetivo de analizar la evolución fatal de esos pacientes, se llevó a cabo esta revisión sistemática (RS). Material y métodos: La búsqueda bibliográfica se realizó con fecha 23 de marzo de 2023, en las principales bases de datos (Medline (a través de Pubmed), Embase (a través de Embase-Elsevier), The Cochrane Library (a través de Wiley), Cinahl (a través de Ebsco HOST), SCI-EXPANDED, SciELO (a través de la WOS) y Scopus (a través de Scopus-Elsevier), libre (mediante el motor Google) y revisando las citas de los artículos incluidos. Resultados: Se rescataron 1393 artículos, de los cuales se descartaron 1386 por diversas razones. Mediante el análisis de los textos completos, finalmente se incluyeron 7 estudios. Todos los estudios eran series de casos (nivel 4). La mediana de la frecuencia de muerte observada fue de 36,6% (Q1 20% - Q347%). Conclusiones: Esta RS mostró en niños con patología hemato-oncológica, que la mortalidad por IFI por Mucor spp. alcanzó a casi un tercio de los pacientes (AU)

Studies on invasive fungal infection (IFI) by Mucor spp. in pediatric patients with cancer have a low level of evidence, which makes it difficult to elucidate its characteristics and progression. To analyze the fatal outcome of these patients, this systematic review (SR) was conducted. Material and methods: A literature search was carried out on March 23, 2023, in the following main databases (Medline (via Pubmed), Embase (via Embase-Elsevier), The Cochrane Library (via Wiley), Cinahl (via Ebsco HOST), SCI-EXPANDED, SciELO (via the WOS) and Scopus (via Scopus-Elsevier). Additionally, a complementary search was carried out using free search engines (such as Google) and by reviewing the references of the included articles. Results: A total of 1393 articles were retrieved, of which 1386 were excluded for various reasons. After a thorough analysis of the full-text articles, 7 studies were ultimately included in the review. All studies were case series (level 4). The median observed death rate was 36.6% (IQR, 20% - 47%). Conclusions: This SR showed that in children with hematological-oncological disease, mortality due to IFI by Mucor spp. affected almost one third of the patients (AU)

Mucormicosis bronquial, un hallazgo inusual en la broncoscopia / Bronchial mucormycosis, an unusual finding on bronchoscopy

La mucormicosis es una infección causada por hongos oportunistas pertenecientes al orden de los mucorales. Desde la aparición de la pandemia por SARS-CoV-2 se han emitido alertas sobre el incremento en la presentación de esta infección. La presentación de mucormicosis traqueo-bronquial representa el 37% de la afectación pulmonar. A continuación, se desarrolla el caso clínico de una pa-ciente diabética con neumonía severa por SARS-CoV-2, con evolución desfavorable, quien es llevada a broncoscopia, encontrando pseudomembranas blanquecinas que obs-truían la luz del bronquio fuente izquierdo, confirmándose histopatológicamente la presencia de mucorales. Se realiza reporte del presente caso para reconocer la apa-rición de infecciones fúngicas en pacientes que presen-tan diabetes mal controlada e infección por SARS-CoV-2 que tienen una evolución tórpida. La presencia de seudo membranas blancas puede ser un signo de alarma para tener este tipo de infecciones dentro de las posibilidades diagnósticas

Mucormycosis is an infection caused by opportunistic fungi belonging to the order Mucorales. Since the SARS CoV 2 pandemic, warnings have been issued about the increase in the presentation of this infection. The presentation of tracheobronchial mucormycosis represents 37% of pulmonary involvement.We present the clinical case of a diabetic patient with severe pneumonia due to SARS VOC 2, with unfavorable evolution, who was taken to bronchoscopy and found whitish pseudomembranes obstructing the lumen of the left main bronchus, confirming histopathologically the presence of mucormycosis.A report of the present case is made to recognize the appearance of fungal infections in patients with poorly controlled diabetes and SARSe COV 2 infection, who have a torpid evolution. The presence of white pseudomembranes should be an alarm sign to have this type of infections within the diagnostic possibilities

Infección por Aspergillus flavus y Rhizopus oryzae complex en paciente con diabetes mellitus / Infection due to Aspergillus flavus and Rhizopus oryzae complex in a patient with diabetes mellitus

La sinusitis micótica es una condición patológica que puede presentarse en pacientes con diabetes mellitus y estar asociada a una crisis hiperglucémica. Es una entidad agresiva con complicaciones locales que incluyen afectación de la órbita y el sistema nervioso central, y compromiso vascular. A pesar del tratamiento quirúrgico y antimicótico, la mortalidad es de hasta el 75 %. Se describe el caso de una paciente con diagnóstico de cetoacidosis diabética y signos de oftalmoplejía unilateral que llevaron al estudio con resonancia magnética del sistema nervioso central; se encontraron signos de sinusitis, meningitis y cerebritis. Los estudios microbiológicos iniciales fueron negativos, y los biomarcadores galactomanano sérico y el antígeno de Cryptococcus también fueron negativos. Tras el manejo quirúrgico, se llegó a la identificación de Aspergillus flavus y Rhizopus spp. en el tejido de los senos paranasales. La paciente recibió tratamiento con posaconazol y, tras dos meses de seguimiento, había presentado mejoría clínica. La infección fúngica dual y la infección por A. flavus son entidades poco frecuentes y de relevancia clínica, sin casos presentados previamente en nuestro país por lo que este corresponde a un caso de interés clínico.

Fungal sinusitis is a pathology that can occur in patients with diabetes mellitus and be associated with a hyperglycemic crisis. It is an aggressive entity with local complications that include involvement of the orbit or the central nervous system, and vascular involvement. Despite surgical and antifungal treatment, mortality raises up to 75%. We report the case of a female patient with a diagnosis of diabetic ketoacidosis and signs of unilateral ophthalmoplegia, which led to the study with magnetic resonance imaging of the central nervous system, finding signs of sinusitis, meningitis, and cerebritis. Initial microbiological studies were negative, and biomarkers such as serum galactomannan and Cryptococcus antigen were also negative. After surgical management and the identification of Aspergillus flavus and Rhizopus spp. in sinus tissue, the patient received treatment with posaconazole and after two months of follow-up she presented clinical improvement. Dual fungal infection and infection by A. flavus are uncommon and clinically relevant entities, with no cases previously reported in our country, therefore this corresponds to a case of clinical interest.

Mucormicosis invasora en pacientes con COVID-19: reporte de 2 casos / Invasive mucormycosis during COVID-19 infection. Report of two cases

COVID-19 disease is associated with a significant number of opportunistic infections, including invasive fungal infections such as mucormycosis. The prevalence of the latter is rare, estimated to be between 0.005 and 1.7 per million inhabitants. Risk factors include hematological diseases, Diabetes Mellitus with poor metabolic control, solid organ transplantation, neutropenia, and prolonged administration of systemic corticosteroids. We report two males aged 60 and 75 years with pulmonary and tracheobronchial invasive mucormycosis, respectively. Both patients had a deficient metabolic control of their diabetes as a predisposing risk factor added to severe COVID-19 infection. High suspicion and early diagnosis are essential for prompt treatment, especially considering the associated high morbidity and mortality of this fungal infection.

A hospital-based observational study on clinical and radiographic findings in COVID-19 associated rhinomaxillary mucormycosis: revealing the pandora box

Mucormycosis is a rare, rapidly spreading, fulminant, opportunistic infection that is caused by a group of filamentous molds. During the second wave of COVID-19 India reported most of the cases of mucormycosis which is termed as COVID-19-associated mucormycosis (CAM). Aim: The purpose of this study is to describe and understand the clinical and radiographic findings related to COVID-19 associated rhinomaxillary mucormycosis. Methods: In this observational study 76 individuals with proven rhinomaxillary mucormycosis were included. The demographic profile, predisposing factors, anatomic structures involved, oral manifestations, radiographic findings management, and 90-day mortality were recorded and analyzed. Results: Among 76 individuals with COVID-19-associated rhinomaxillary mucormycosis diabetes mellitus was present in 93.42% of cases. Almost all patients received corticosteroids during COVID-19 treatment. The maxilla was most commonly involved in around 98.6% of cases. Interestingly 1 case involving the mandible was noted and the maxillary sinus was the most commonly involved. Mortality occurred in 1.31% (n=1) of cases. Conclusion: Diabetes was the most common predisposing factor. Administration of corticosteroids was evident. A considerable number of patients developed diabetes during the treatment of COVID-19. Early signs and oral manifestations of rhinomaxillary mucormycosis play a pivotal role in the early diagnosis and prompt treatment to reduce mortality and morbidity in COVID-19 associatedrhinomaxillary mucormycosis patients

Expert consensus on diagnosis and management of mucormycosis in China / 中华内科杂志

Mucormycosis is an important invasive fungal disease that is difficult to diagnose and treat, and has a high mortality rate. To improve the diagnosis and treatment of mucormycosis by clinicians, the Medical Mycology Society of Chinese Medicine and Education Association engaged multidisciplinary experts to compile this expert consensus. This consensus refers to the latest international guidelines for diagnosis and treatment of mucormycosis, combined with the characteristics and treatment needs of mucormycosis in China and covers the following eight aspects to provide reference for Chinese clinicians: pathogenic agents, high-risk factors, clinical types, imaging manifestations, etiological diagnosis, clinical diagnosis, treatment, and prevention.

COVID-19 associated rhinomaxillary mucormycosis: a case series and review of literature

During COVID-19 pandemic, fulminant deep fungal infection started emerging in India, known as Mucormycosis. This type of mucormycosis was termed as COVID-19 associated mucormycosis (CAM). These patients had previous history of COVID-19 infection. Such cases were mainly reported in immunocompromised patients such as patients with poorly controlled diabetes and chronic renal diseases etc. Rhinomaxillary mucormycosis is an aggressive, fulminant, fatal deep fungal infection of head and neck region. Early diagnosis and prompt treatment can reduce the mortality and morbidity associated with the disease; hence we present case series of rhinomaxillary mucormycosis to create awareness amongst dental surgeons

Mucormicosis periostomal posoperatoria, a propósito de un caso / Mucormycosis periostomal: case report

Resumen Introducción: La mucormicosis en una enfermedad infrecuente y oportunista que afecta, principalmente, a pacientes inmunocomprometidos. Pocas veces se han reportado casos de afectación periostomal. Clínicamente puede ser confundida con otras patologías, pudiendo tener una evolución fulminante, por lo que un adecuado y pronto diagnóstico son necesarios para una instauración precoz del tratamiento. Caso Clínico: Se presenta el caso de una paciente de 62 años inmunocomprometida, que tras complicaciones quirúrgicas evoluciona con mucormicosis periostomal de la pared abdominal. A pesar de un tratamiento quirúrgico con múltiples resecciones de tejido asociado a antifúngico local y sistémico, la paciente fallece, concordante a la letalidad expresada en la literatura.

Introduction: Mucormycosis is a rare and opportunistic disease that mainly affects immunocompromised patients. Few cases of peristomal involvement have been reported. Clinically it can be confused with other pathologies and may have a fulminant evolution, so an adequate and prompt diagnosis is necessary for an early establishment of treatment. Clinical Case: We present the case of a 62-year-old immunocompromised patient who, after surgical complications, evolves with periostomal mucormycosis of the abdominal wall. Despite surgical treatment with multiple tissue resections, associated with local and systemic antifungal agents, the patient died, consistent with the lethality expressed in the literature.

Mucormicosis asociada a COVID-19: revisión de la literatura para odontólogos / COVID-19 associated mucormycosis: a literature review for dentists

La mucormicosis es una infección fúngica rara, con alta morbilidad y mortalidad. Se presenta principalmente en pa- cientes con diabetes mellitus no controlada, inmunocompro- metidos, con tratamiento crónicos con esteroides, entre otros. Actualmente, se cree que la pandemia de COVID-19 y los tratamientos con corticosteroides podrían estar implicados en el aumento de casos de esta micosis. Este hongo invade el sistema vascular, ocluyendo el flujo sanguíneo arterial y generando una rápida trombosis e isque- mia, lo que provoca la necrosis de los tejidos duros y blandos, con invasión rápida a los tejidos circundantes. Hay varias formas clínicas. En la cavidad bucal se presenta la variante rino-orbito-cerebral, que afecta el paladar en forma de lesión eritematosa o grisácea que puede progresar hacia la formación de una masa necrótica o ulceración con muy escaso sangrado de mucosa. Se manifiesta con síntomas típicos de una rinosinusitis con fiebre y dolor en las piezas dentarias superiores. El tratamiento consta de tres pilares fundamentales: el diagnóstico, un manejo adecuado de las comorbilidades y la combinación de las terapias antifúngica y quirúrgica. Desde el año 2020, la mucormicosis asociada a COVID-19 pasó a ser un evento de notificación obligatoria inmediata al Sistema Nacional de Vigilancia de la Salud (SNVS2.0) me- diante el Sistema Integrado de Información Sanitaria Argen- tina (SISA). Es importante destacar que se han reportado casos de mu- cormicosis luego de extracciones dentales; lo que impulsa a afianzar los conocimientos sobre esta enfermedad, extremar las medidas preventivas e incentivar el diagnóstico precoz en la atención odontológica, debido a la rapidez en la evolución de la patología (AU))

Mucormycosis is a rare fungal infection, with high mor- bidity and mortality. It occurs mainly in patients with uncon- trolled diabetes mellitus, immunocompromised, on chronic treatment with steroids, among others. Currently, it is believed that the COVID-19 pandemic and the corticosteroid treatments could be one of the causes of increased cases. This fungus invades the vascular system, occluding arteri- al blood flow and generating rapid thrombosis and ischemia, which causes necrosis of hard and soft tissues, with rapid in- vasion to the surrounding tissues. There are several clinical forms. In the oral cavity, the rhino-orbito-cerebral variant presents itself affecting the pal- ate in the form of an erythematous or grayish lesion that can progress towards the formation of a necrotic mass or ulcera- tion with very little mucosal bleeding. It manifests itself with typical symptoms of rhinosinusitis, with fever and pain in the upper teeth. The treatment consists of three fundamental pillars: diag- nosis, proper management of comorbidities and the combina- tion of antifungal and surgical therapies. Since 2020, COVID-19 associated mucormycosis became an event of mandatory immediate notification to the National Health Surveillance System (SNVS2.0,) through the Argentina Integrated Health Information System (SISA). It is important to emphasize that mucormycosis cases had been reported following tooth extractions, which drives to strengthen knowledge about this disease, extreme preventive measures and encourage early diagnosis in dental care, due to the speed of the evolution of the pathology (AU))

Maxilectomia em paciente diabética tipo II, diagnosticada com Mucormicose rinomaxilar, com posterior reabilitação protética: relato de caso / Maxillectomy in a type II diabetic patient, diagnosed with rhinomaxillary mucormycosis, with subsequent prosthetic rehabilitation: case report / Maxilectomía en paciente diabética tipo II diagnosticada de mucormicosis rinomaxilar, con posterior rehabilitación protésica: reporte de caso

A mucormicose é uma infecção fúngica angioinvasiva que afeta uma ampla faixa etária, geralmente imunodeprimidos, sem predileção por gênero ou raça e com alta taxa de mortalidade. Essa infecção inicia se no nariz, devido à inalação dos esporos, podendo-se espalhar pelos seios paranasais, órbita e estruturas intracranianas. As características clínicas incluem parestesia perinasal, celulite periorbitária, rinorréia, obstrução nasal, epistaxe e diminuição de peso. O tratamento efetivo dessa comorbidade compoe uma combinação de manejo clínico e medicamentoso, conjuntamente com desbridamento cirúrgico radical do tecido infectado e/ou necrótico. O caso clínico descrito nesse trabalho refere-se a uma paciente diagnosticada com Mucormicose rino maxilar e diabetes do tipo II, a qual foi submetida a procedimento cirúrgico de hemimaxilectomia do lado esquerdo com posterior reabilitação protética para selamento de comunicação buco-naso-sino-etmoidal... (AU)

Mucormycosis is an angioinvasive fungal infection that affects a wide age group, usually immunocompromised, with no gender or race predilection, and with a high mortality rate. This infection starts in the nose, due to the inhalation of spores, and can spread through the paranasal sinuses, orbit and intracranial structures. Clinical features include perinasal paresthesia, periorbital cellulitis, rhinorrhea, nasal obstruction, epistaxis and weight loss. Effective treatment of this comorbidity comprises a combination of clinical and drug management, together with radical surgical debridement of infected and/or necrotic tissue. The clinical case described in this work refers to a patient diagnosed with Mucormycosis Rhinomaxilla and Type II diabetes, who underwent a surgical procedure of left hemimaxillectomy with subsequent prosthetic rehabilitation for sealing of the bucco nasal-sino-ethmoidal communication... (AU)

La mucormicosis es una micosis angioinvasiva que afecta a un amplio grupo de edad, habitualmente inmunodeprimidos, sin predilección de género ni raza, y con una alta tasa de mortalidad. Esta infección comienza en la nariz, debido a la inhalación de esporas, y puede extenderse a través de los senos paranasales, la órbita y las estructuras intracraneales. Las características clínicas incluyen parestesia perinasal, celulitis periorbitaria, rinorrea, obstrucción nasal, epistaxis y pérdida de peso. El tratamiento eficaz de esta comorbilidad comprende una combinación de manejo clínico y farmacológico, junto con un desbridamiento quirúrgico radical del tejido infectado y / o necrótico. El caso clínico descrito en este trabajo se refiere a una paciente diagnosticada de Mucormicosis Rinomaxilar y diabetes Tipo II, que fue sometida a un procedimiento quirúrgico de hemimaxilectomía izquierda con posterior rehabilitación protésica para sellar la comunicación buco-nasal-sino-etmoidal... (AU)

COVID-19 y rinosinusitis invasiva aguda secundaria a mucormicosis: a propósito de 2 casos en Colombia / Covid-19 and acute invasive rhinosinusitis secondary to mucormycosis: report of two cases in Colombia

Introducción: la mucormicosis es una infección micótica poco frecuente, también conocida como zigomicosis o phycomycosis, de baja prevalencia, pero con alta mortalidad, por lo regular ocasionada por estados de inmunosupresión como los que ocurren después de infecciones por COVID-19, teniendo esta asociación patológica una alta tasa de mortalidad y secuelas en la salud de los que la padecen. Presentación de los casos: son dos casos de mucormicosis rinocerebral asociados con COVID-19 y se describen el manejo y los desenlaces.

Introduction:mucormycosis is a rare fungal infection, also known as zygomycosis or phycomycosis. It features low prevalence and high mortality rate. It usually affects patients with altered immunity precipitated by COVID-19 infection. This co-infection is associated with high mortality rate and sequelae in affected patients. Case report: we present two cases of rhino-cerebral mucormycosis co-infection in COVID-19 patients, the provided treatment, and outcomes.

Corticosteroid therapy associated with COVID-19: an ally or a risk factor for the establishment of secondary fungal co-infections?

Corticosteroid therapy to combat inflammation caused by SARS-CoV-2 seems to be a risk factor for developing secondary fungal co-infections. PubMed and ScienceDirect databases were searched, with the following word groups: [(aspergillosis OR mucormycosis OR candidiasis) AND (coronavirus disease) AND (corticoids). The selected articles present the main risk factors related to the establishment of secondary fungal co-infections in COVID-19 patients. Corticosteroid therapy used to combat inflammation caused by SARS-CoV-2 has been shown to be strongly associated with the establishment of mucormycosis and aspergillosis. Mucormycosis has been the main fungal co-infection related to corticosteroid therapy, causing a high number of deaths in COVID-19 patients. Diabetes mellitus was the most prevalent comorbidity, especially for the establishment of mucormycosis. Dexamethasone use seems to be associated with mucormycosis emergence and death. However, aspergillosis showed a greater relationship with patient recovery. Thus, risk factors such as diabetes mellitus, combined with corticosteroid use, have shown a relationship to the establishment of mucormycosis. The corticosteroids used in COVID-19 patients should be individually analyzed, considering the patient's medical history and the risk/benefit ratio of the use of these drugs.

Mucormicosis orbitorrinocerebral: el manejo más allá de la cirugía. Informe de caso clínico y revisión de la literatura / Rhino-orbital-cerebral mucormycosis: management beyond surgery. Clinical case report and literature review

Introducción: se presenta el caso clínico de un paciente con mucormicosis orbitorrinocerebral y revisión de la literatura. Caso clínico: se trata de hombre de 45 años que consultó por síntomas nasosinusales manejado ambulatoriamente con múltiples antibióticos sin mejoría, por lo cual se realizó manejo quirúrgico con toma de muestras que revelaron hifas compatibles con mucormicosis. Cuando acudió a la institución presentaba extensión de la infección a todas las cavidades nasales y parte de la base del cráneo. En la búsqueda de la inmunosupresión, se encontró una diabetes de novo de difícil control. Discusión: la mucormicosis orbitorrinocerebral es una enfermedad altamente invasiva que requiere un manejo multidisciplinario, cirugías seriadas y extendidas, antimicóticos tópicos y sistémicos. El pronóstico mejora y la evolución puede ser favorable cuando se logra controlar la causa de la inmunosupresión, en este caso la diabetes. Conclusiones: es requisito fundamental realizar un tratamiento multidisciplinario en el abordaje de estos pacientes, tanto en la especialidad clínica como en la quirúrgica, psicosocial, nutricional y de rehabilitación.

Introduction: It is the clinical case of a patient with rhino-orbital-cerebral mucormycosis and the review of the literature. Clinical case: We present the case of a previously healthy young man who started with sinonasal symptoms and hyphae compatible with mucormycosis were found in the initial samples of outpatient care. When he came to the institution, the infection had spread to all the nasal cavities and part of the skull base. Discussion: Searching for baseline immunosuppression, only difficult-to-control diabetes and de novo diagnosis were found. It required multidisciplinary management, serial and extended surgeries, topical, venous and oral antifungals. The prognosis improved and the evolution became favorable when glycemic control was achieved. Conclusion: It is a fundamental requirement to carry out a multidisciplinary work for the approach to these patients, both in the clinical, surgical, psychosocial, nutritional and rehabilitation specialties.

Loss of an eye to mucormycosis following corticosteroid therapy for COVID-19

Mucormycosis is a rare, sometimes severe fungal infection that has emerged as a possible complication of COVID-19. We report a case of a non-diabetic, apparently immunocompetent patient diagnosed with rhino-orbital-cerebral mucormycosis shortly after COVID-19 treatment with dexamethasone. The patient received optimized systemic antifungal therapy and extensive surgical treatment. So far, four months after the last hospital discharge, the patient has been in good general condition. This case is a dramatic reminder that beneficial corticosteroid therapy in general inevitably carries a risk of opportunistic infection, and corticosteroid therapy for COVID-19 risks orbital-rhinocerebral mucormycosis that clinicians should watch for with vigilance.

Clinical analysis of 3 cases of mucormycosis in children with acute lymphoblastic leukemia and literature review / 中华儿科杂志

Objective: To investigate the clinical features, diagnosis,treatment and prognosis of children with acute lymphoblastic leukemia complicated with mucormycosis, and to improve the understanding of the disease. Methods: The clinical data of 3 children with acute lymphoblastic leukemia (ALL) complicated with mucormycosis treated at the First Affiliated Hospital of Zhengzhou University between October 2020 and January 2021 were analyzed retrospectively. Literature search and review covered the China national knowledge infrastructure, Wanfang database and Pubmed using the keywords of "acute lymphoblastic leukemia" and "mucormycosis" up to June 2021. Results: Case 1, a 12-year-old boy, was diagnosed with ALL, developed fever and chest pain during induction therapy. The Metagenomic next-generation sequencing (mNGS) testing of alveolar perfusion fluid suggested infection with Rhizopus oryzae. Amphotericin B combined with posaconazole was applied and amphotericin B was removed after improvement. Bone destruction was indicated by CT. Amphotericin B was applied again. Case 2, a 4-year-old boy, with a history of pallor and tetter, was diagnosed with ALL. He developed cough and fever during induction therapy. mNGS of blood suggested infection with Rhizomucor pusillus. Amphotericin B combined with voriconazole was applied, but the situation was not significantly improved. The disseminated infection occurred. Amphotericin B combined with posaconazole was applied and vacuum sealing drainage was performed. Case 3, a 2-year-old girl, was diagnosed with ALL, developed fever and cough during induction therapy. Rhizomucor pusillus was indicated by mNGS. Amphotericin B combined with posaconazole was used, and posaconazole was stopped after improvement. Follow-up until June 2021, the condition of the 3 children improved. There was no recurrent Mucor infection, and the primary hematopathy was in complete remission. According to the literature, 7 reports were found in Chinese journals, while 17 reports were found in English literature, 25 cases have been reported. Among a total of 28 children, 11 cases rhino-orbito-cerebral mucormycosis, four pulmonary mucormycosis, 2 cutaneous mucormycosis, 2 gastrointestinal mucormycosis and 9 disseminated mucormycosis. There were 17 cases developed infection during induction chemotherapy, 8 cases during maintenance therapy, 3 cases after hematopoietic stem cell transplantation. Voriconazole was used in 15 cases; 19 cases were treated with combined surgery, 7 cases were treated with drugs only, 2 cases were untreated; 21 cases showed improvement after treatment. Death occurred in seven cases. Conclusions: ALL complicated with mucormycosis often occurs in the stage of induction therapy. The clinical features lacked specificity, mNGS can help find the pathogen and provide evidence for diagnosis. Surgical treatment also could be combined when necessary, which is helpful to improve the prognosis.

Clinical features of children with Cunninghamella spp. infection: a case report and literature review / 南方医科大学学报

We report a case of mucormycosis induced by Cunninghamella spp. infection in a ten-year-old girl with acute lymphoblastic leukemia, who developed fever and respiratory symptoms after chemotherapy and was diagnosed with invasive fungal disease. Peripheral blood DNA sequences were analyzed using metagenomic next-generation sequencing (mNGS), and by comparison with the Pathogens Metagenomics Database (PMDB), we identified Cunninghamella spp. with sequence number 514 as the pathogen. The patient was treated with amphotericin B combined with posaconazole and showed a favorable response. We searched Pubmed, Embase, CNKI, and Wanfang database for reports of cases of Cunninghamella spp. infection in children and retrieved 22 reported cases (including 12 males) with a median age of 13.5 (3-18) years. In these 22 cases, hematological malignancy was the most common underlying condition (19/22), and most of patients experienced an acute onset and rapid progression with respiratory symptoms (14/20) and fever (16/20) as the most common symptoms. CT imaging often showed unilateral lesions with varying imaging findings, including pulmonary nodules or masses, infiltrative changes, and pleural effusion. Definite diagnoses were established in 18 of the cases, and 4 had probable diagnoses; the lungs and skin were the most frequent organs compromised by the infection. A definite diagnosis of Cunninghamella spp. infection still relied on histopathological examination and fungal culture, but the molecular techniques including PCR and mNGS had shown potentials in the diagnosis. Almost all the cases received antifungal treatment after diagnosis (21/22), and 13 patients also underwent surgeries. Death occurred in 9 (42%) of the cases at a median of 19 (4-54) days after onset of the signs or symptoms. The patients receiving antifungal therapy combined with surgery had a high survival rate (9/13, 69%) than those with antifungal therapy alone (3/8, 37%). Invasive fungal disease is a common complication in immunoco-mpromised patients, but Cunninghamella spp. infection is rare and has a high mortality rate. In cases highly suspected of this disease, active diagnosis and early treatment are critical to improve the survival outcomes of the patients.