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.

Effect of Wenyang Huashi Prescription on Inflammatory Factors and Immune Function of Patients After Allergic Fungal Rhinosinusitis / 中国实验方剂学杂志

Objective::To assess the efficacy of Wenyang Huashi prescription on rehabilitation of patients after allergic fungal rhinosinusitis (AFRS) operation, and its effect on inflammatory factors and immune function. Method::One hundred and forty-four patients were randomly divided into control group and observation group by random number table, with 72 cases in each group. Patients in control group was treated with Messerklinger to remove lesions completely, and got anti-infective therapy for three days, prednisone acetate tablets, 10 mg/time, 2 times/days, fluticasone propionate inhaled aerosol for a continued 4 weeks, 2 sprays/time, 1 time/day, itraconazole capsules at the first, fifth and ninth week after treatment (one course of treatment was 7 days), 0.2 g/time, 1 time/day. In addition to the therapy of meloxicam tablets, patients in observation group were also given local rinsing and oral administration of Wenyang Huashi prescription. The courses of treatment of both groups were 12 weeks. Before the operation and at the fourth and 12th week after the operation, subjective conditions were assessed. And before the operation and at the 12th, 24th and 48th week after the operation, Lund-Kennedy detected by endoscopic sinus surgery and nasal and paranasal sinus outcomes test-20 (SNOT-20) were scored. And the relapse was recorded during 48 weeks after the operation. And levels of tumor necrosis factor-alpha (TNF-alpha), interleukin-1bet (IL-1bet), nuclear factor kappa B (NF-kappa B), intercellular adhesion molecule in peripheral blood-1 (ICAM-1), T lymphocyte subsets (CD3+, CD4+, CD8+ and CD4+ /CD8+) were detected. Result::By rank sum test, the clinical efficacy in observation group was better than that in control group (Z=2.016, P<0.05). At the fourth and 12th week after the operation, scores of nasal obstruction, dizziness, runny nose and dysosmia in observation group were lower than those in control group (P<0.01). And scores of lund-kennedy and SNOT-20 were lower than those in control group (P<0.01). And levels of TNF-α, IL-1β, NF-κB and ICAM-1 were lower than those in control group (P<0.01). After treatment, levels of CD3+, CD4+ /CD8+ and CD4+ were higher than those in control group, and level of CD8+ was lower than that in control group (P<0.05). During the one-year follow-up, the relapse rate in observation group was 7.81%(5/64), which was lower than 22.58%(14/62) in control group (χ2=5.363, P<0.05). Conclusion::Wenyang Huashi prescription can improve short-term and long-term symptoms, improve the comprehensive curative effect, the quality of life and the immune function of the body, alleviate inflammatory injury, reduce the recurrence rate and improve the prognosis, and so is worth further study and use.

Rinosinusitis fúngica invasiva aguda: actualización en pacientes adultos y pediátricos / Acute invasive fungal rhinosinusitis: update in adults and pediatric patients

RESUMEN La rinosinusitis fúngica invasiva aguda (RSFIA) es una enfermedad poco frecuente caracterizada por una infiltración fúngica de la submucosa y vasos sanguíneos de las cavidades nasal y paranasal. Afecta a pacientes con grados variables de inmunosupresión, destacando entre estas patologías subyacentes la diabetes mellitus y las neoplasias malignas hematológicas. Presenta una alta tasa de mortalidad, pudiendo reducirse significativamente si el diagnóstico y el tratamiento se realizan precozmente. Este artículo tiene por objetivo presentar una revisión actualizada de la literatura respecto a la presentación clínica, microbiología, factores de riesgos, métodos diagnósticos, tratamiento y pronóstico de la RSFIA, tanto en adultos como en niños.

ABSTRACT Acute invasive fungal rhinosinusitis (AIFS) is a rare disease characterized by fungal infiltration of the submucosa and blood vessels of the nasal y paranasal cavities. It affects almost exclusively patients with different degrees of immunosuppression, with underlying pathologies such as diabetes mellitus and hematological malignancies. AIFS has a high mortality rate, but it can be significantly reduced if the diagnosis and treatment are carried out early in the course of disease. This article aims to present an updated literature review regarding clinical presentation, microbiology, risk factors, diagnostic methods, treatment and prognosis of AIFS, both in adults and children.

A retrospective review of 46 cases of chronic invasive fungal rhinosinusitis / 中华传染病杂志

Objective To investigate the clinical characteristics of chronic invasive fungal rhinosinusitis.Methods Clinical features and outcomes of 46 proven cases of chronic invasive fungal rhinosinusitis admitted in Huashan Hospital,Fudan University from January 2009 to December 2016 were retrospectively reviewed.Results Of the 46 patients enrolled,left sphenoid sinus,ethmoid sinus and maxillary sinus were affected in 24,23 and 20 cases,respectively,while right maxillary sinus,ethmoid sinus and sphenoid sinus were affected in 18,16 and 15 eases,respectively.Left and right frontal sinus were affected in 9 and 6 cases,respectively.The central nervous system and orbit were the most commonly affected sites in external nasal involvements,noted in 22 cases respectively.Left sphenoid (17 cases) and ethmoid sinus (15 cases) involvements were most common in central nervous system affected patients.Left sphenoid (14 cases) and ethmoid sinus (13 cases) involvements were most common in orbit affected patients.Aspergillus species were the primary pathogens observed in 42 eases.Zygomycete,candida and dark filamentous fungus were observed in two,one and one case,respectively.Pathologically,37 of the cases were chronic nongranulomatous type and the left 9 were chronic granulomatous type who were all immunocompetent hosts.The initial symptoms usually included headache,dizziness and nasal discomforts including nasal obstruction and purulent secretion.The chief complaints usually included headache,dizziness,and visual disturbances including blurred vision,vision loss or even blindness.Antifungal treatment combined with surgical interventions for removal or drainage focus lesions achieved significant effect,and 42 patients were cured.Conclusions Chronic invasive fungal rhinosinusitis should be taken into consideration in the presence of nasal discomforts or nonspecific symptoms such as headache and dizziness.The possibility of chronic invasive fungal rhinosinusitis should be cautious after the emergence of vision abnormalities.

Expression and significance of Maspin and IKKα in sinusoidal mucosa of rats with fungal rhinosinusitis / 中国比较医学杂志

Objective To investigate the expression and significance of Maspin and IKKα in nasosinusoidal mucosa of rats with fungal rhinosinusitis (FRS).Methods A total of 40 SD rats were used to establish the FRS model, and randomly divided into nasal obstruction group, FRS group, immunosuppressive group and invasive FRS group, 10 rats in each group.Another 10 normal rats were used as control group.Mice in the control group were fed with normal diet.In the nasal obstruction group, the mice had only hemostatic cotton stuffed in the nasal cavity and injection of 0.9% NaCl in the abdominal and nasal cavities.In the FRS group, the mice were injected Aspergillus fumigatus spore suspension into the nasal cavity and 0.9% NaCl i.p.The mice of the immunosuppressive group were given cyclophosphamide i.p.and 0.9% NaCl injection into the nasal cavity.The invasive FRS group was injected with cyclophosphamide i.p.and Aspergillus fumigatus spore suspension into the nasal cavity.The serum levels of interleukin-6 (IL-6) and tumor necrosis factor-α (TNF-α) were measured by enzyme-linked immunosorbent assay (ELISA).The expression of Maspin and IKKα in nasosinusoidal mucosa was detected by immunohistochemical staining.The expression of Maspin mRNA and IKKα mRNA in the nasosinusoidal mucosa was detected by fluorescence quantitative PCR.Results The serum levels of IL-6 and TNF-α in different groups were significantly different (P 0.05).Theresult of immunohistochemical staining showed that the protein expression of Maspin in the FRS group and invasive FRS group was significantly lower than that in the control group, nasal obstruction group and immunosuppressive group, while the expression of IKKα protein was significantly higher than that of control group, nasal obstruction group and immunosuppressive group (P< 0.05).The protein expression of Maspin in the invasive FRS group was significantly lower than that in the FRS group, by contrast, the expression of IKKα protein was significantly higher (P< 0.05).The PCRresult revealed that the expression levels of Maspin and IKKα mRNA were (0.217 ± 0.013) and (0.193 ± 0.012), significantly lower than that in the control, obstruction and immunosuppressive groups [(0.309 ± 0.021), (0.302 ± 0.017), and (0.293 ± 0.02)] (P< 0.05), while the expressions level of IKKα mRNA were significantly higher [(0.319 ± 0.043), (0.384 ± 0.048) vs (0.169 ± 0.015), (0.171 ± 0.018), and (0.175 ± 0.019)] (P< 0.05).Conclusions Down-regulation of Maspin expression after IKKα activation is the main cause of the onset of FRS, which may also be one of the mechanisms of invasive FRS.

Multiple primary pathologies in a patient with primary hyperparathyroidism

@#<p style="text-align: justify;">Co-existence of multiple pathologies affecting a patient poses a challenge in the diagnosis and management. A rare combination of early tongue cancer, maxillary fungal rhinosinusitis, calcified thyroid nodule and primary hyperparathyroidism in a 70-year-old female is described. The evidences used for disease probabilities based on diagnostic results and effectivess of treatment modalities are presented. The decision-making process to come up with an individualized management is discussed.</p>

Fungal rhinosinusitis: A clinicomycological perspective.

Purpose: Chronic rhinosinusitis (CRS) is a widely prevalent condition globally as well as in India. The spectrum of fungal involvement in CRS runs from benign colonisation to potentially life-threatening invasive disease. Successful treatment of such mycotic infections largely depends on the accurate identification of the pathogen, early and appropriate intervention by surgical clearance, supported with antifungal medication as per standard regimen. Thus, this study was undertaken to determine the prevalence of fungal rhinosinusitis (FRS), and to analyse its clinicomycological profile. Materials and Methods: Fifty-two patients with clinical suspicion of CRS attending a tertiary care hospital during a one-year period were included in this retrospective analysis. The sinonasal specimens were subjected to microscopy by KOH mount and fungal culture as per standard mycological technique. Tissue specimens were also subjected to histopathological examination. Results: Male to female ratio was 1.25:1; age varied from 14 years to 62 years with majority of patients (37%) belonging to age group 21-40 years. The prevalence of FRS was 44%, and 74% of it was caused by Aspergillus sp. Aspergillus flavus (A. flavus) (52%) was the most prevalent fungus isolated. Allergic fungal rhinosinusitis (AFRS) was the most common presentation (79%). Conclusion: FRS is a continuous spectrum of disease varying in presentation, treatment and long-term sequelae. Correct identification of the fungus remains essential for appropriate treatment.

Unusual causes of fungal rhinosinusitis: A study from a tertiary care centre in South India.

Purpose: The frequency of mycotic infections of the nose and paranasal sinuses has been increasing over the past three decades. Apart from the common causes of fungal rhinosinusitis such as Aspergillus species and Penicillium species, there have been reports of rare and unusual fungi isolated from India and other countries. Objective: The objective of this study is to fi nd out the prevalence of fungal infections of the nose and paranasal sinuses caused by unusual fungal isolates at a tertiary care teaching hospital in South India. Materials and Methods: Duration of the study period was from April 2009 to March 2010. Specimens were collected from the nose and paranasal sinuses of all clinically and radiologically diagnosed cases of rhinosinusitis. All the clinical specimens were processed by standard methods for fungal culture. This included initial screening by 10% potassium hydroxide, inoculation of the specimen onto Sabouraud dextrose agar and incubation at 25°C and 37°C, followed by slide culture and other special techniques wherever necessary. Histopathological examination was also performed for the specimens. Results: A total of 60 specimens were received for fungal culture from cases of rhinosinusitis during the period, out of which 45 showed no growth. There were nine cases of Aspergillus fl avus, 1 each of Aspergillus fumigatus and Penicillium species. The rest four specimens grew rare fungal isolates, i.e. Acremonium sp., Scedosporium apiospermun, Cladosporium cladosporioides and Lasiodiplodia theobromae. Histopathological fi ndings were also positive for these four cases. Conclusion: Apart from the common causes, unusual fungal pathogens were isolated from cases of rhinosinusitis during the study period, which is in accordance with similar reports from other parts of India and some other countries.

Fungal rhinosinusitis: A prospective study in a University hospital of Uttar Pradesh.

Background: To assess the purpose of fungal rhinosinusitis in a University hospital and to correlate histopathological findings with culture results for accurate clinical classification of the disease. Materials and Methods: One-hundred suspected patients were included in the study. Data was collected in a brief predetermined format. Samples like nasal lavages, sinus secretions, and tissue specimens were processed and examined by microbiology culture using recommended techniques. Slide culture was done to observe the microscopic morphology. Histopathological examination was done by H and E stain and PAS stain for classification. Results: Out of 100 cases of rhinosinusitis, 21 cases were culture-positive for fungal rhinosinusitis. On the basis of histopathological findings, 14 cases (66.67%) were found to be of non-invasive fungal rhinosinusitis. Aspergillus flavus was the most common fungal isolate. Conclusion: Mycological profile of rhinosinusitis in Lucknow was thus evaluated. Histopathological and microbiological findings reported 21 cases of fungal rhinosinusitis among 100 suspected cases of rhinosinusitis.

Rinosinusitis alérgica por Curvularia inaequalis (Shear) Boedijn / Allergic rhinosinusitis by Curvularia inaequalis (Shear) Boedijn

Curvularia inaequalis (Shear) Boedijn is a fungus dematiaceo, saprophyte and plant pathogen found mainly in tropical and subtropical areas, associated with various organic substrates. Rarely been identified in systemic infections, skin and there is only one report of allergic rhinosinusitis described above. A case of allergic fungal rhinosinusitis by Curvularia inaequalis (Shear) Boedijn in which diagnosis was considered the signs and symptoms, sinus CT and cultivation of mucin.The patient was treated with endoscopic surgical toilet, plus use of inhaled steroids and itraconazole systemic. With good clinical response, is asymptomatic at one year.

Curvularia inaequalis (Shear) Boedijn es un hongo dematiáceo, saprófito y fitopatógeno, presente principalmente en áreas tropicales y subtropicales, asociado a distintos sustratos orgánicos. Se ha identificado escasamente en infecciones sistémicas, cutáneas y sólo existe una comunicación de un caso de rinosinusitis alérgica descrito anteriormente. Presentamos el caso clínico de un paciente con una rinosinusitis alérgica fúngica por Curvularia inaequalis (Shear) Boedijn en cuyo diagnóstico se consideró los síntomas y signos clínicos, la TAC de senos paranasales y el cultivo de la mucina. El paciente fue tratado con un aseo quirúrgico por vía endoscópica, además del uso de corticoesteroides inhalatorios e itra-conazol sistémico. Presentó una buena respuesta clínica, encontrándose asintomático a un año del tratamiento.

Clinical Characteristics and Treatment of Fungal Rhinosinusitis / 대한이비인후과학회지

Fungal rhinosinusitis was once considered a rare disorder but is now reported with increasing frequency throughout the world. This entity is now thought to comprise five subtypes. Acute invasive fungal rhinosinusitis, chronic invasive fungal rhinosinusitis, and chronic granulomatous invasive fungal rhinosinusitis make up the invasive group, whereas noninvasive fungal rhinosinusitis is composed of fungal ball and fungus related eosinophilic rhinosinusitis including allergic fungal rhinosinusitis (AFRS). These five subtypes are distinct entities with different clinical, histological, and radiologic features. The diagnosis of each category is important for optimum therapy and predicting the course. However, consensus on terminology, pathogenesis, and optimal management is lacking. The distinction of granulomatous from chronic invasive type is not beyond controversy as both types have a chronic course and predominant orbital involvement. AFRS, eosinophilic fungal rhinosinusitis, and esinophilic mucin rhinosinusitis are imprecise and require better definition. The clear differentiation and definition of categories of fungal rhinosinusitis is related to the development of a management protocol of each category. Prompt diagnosis and initiation of appropriate therapy are essential to avoid a protracted or fatal outcome.

Non-surgical Management of Fungal Sinusitis

This article attempts to review the current understanding of non-surgical management of fungal rhinosinusitis. Fungal rhinosinusitis can be divided into an invasive or non-invasive form based on the clinical, radiologic, and histologic manifestations. For the proper management of acute fulminant invasive fungal sinusitis, antifungal medications, complete surgical debridement, and reversal of the underlying state of immune dysfunction are necessary. Antifungal medication and complete extirpation of the disease are required in the treatment of chronic invasive fungal sinusitis or granulomatous fungal sinusitis. Sinus fungal ball can be adequately treated through a complete surgical removal of the fungal ball and irrigation of the involved sinus. Post-operative antifungal therapy is rarely necessary unless the patient suffers from a compromised immune function. Treatment of an allergic fungal rhinosinusitis is marked by a high rate of recidivism. Earlier 'radical' surgery has currently been replaced with a combination of conservative surgery and medical therapy, including immunomodulation with corticosteroids or immunotherapy.