Primary cutaneous mucormycosis. Report of a case in a HIV patient

Primary cutaneous mucormycosis is an unusual mycotic infection associated to immunosupression. We present a 34 year-old woman with HIV infection with a necrotic primary mucormycosis of the skin associated to a venous catheter. She was treated with amphotericin B and surgical debridement.     

Pulmonary mucormycosis diagnosed by fine needle aspiration cytology. A case report

BACKGROUND: The usefulness of fine needle aspiration cytology (FNAC) in the diagnosis of lung lesions is well documented. Fungal lesions are among nonneoplastic lesions of the lung in which FNAC has proven a useful technique in both immunocompromised and immunocompetent patients. These include cryptococcosis, aspergillosis, histoplasmosis and coccidiodomycosis. Pulmonary mucormycosis, an aggressive fungal infection, is rarely diagnosed on FNAC. We report a case of isolated pulmonary mucormycosis diagnosed on FNAC. CASE: A 62-year-old renal transplant recipient with diabetes mellitus and hypertension, asymptomatic for four months, presented with tachypnea, generalized malaise and weakness. Radiologic studies showed an enlarging, cavitating lesion in the right lung. Computed tomography-guided fine needle aspiration performed on the lung lesion showed fungal profiles with broad, ribbonlike, aseptate hyphae with right-angled branching consistent with the Zygomycetes class of fungi, which includes Rhizopus and Mucor species. Fungal cultures confirmed the presence of Rhizopus. The patient underwent right pneumonectomy, was placed on liposomal amphotericin B therapy and discharged with good pulmonary status and stable kidney function. CONCLUSION: FNAC is a useful technique in the diagnosis of pulmonary mucormycosis.     

Primary Cutaneous Mucormycosis Caused by <Emphasis Type="Italic">Rhizopus oryzae</Emphasis>: A Case Report and Review of Literature

Mucormycosis is an invasive infection caused by opportunistic fungi. Rhizopus, Lichtheimia, Mucor and Rhizomucor are the most common isolated genera. Primary cutaneous mucormycosis is usually related to traumatic injuries, but immunocompromised cases are associated with underlying conditions such as diabetes mellitus and malignancies. The treatment of choice is surgical debridement and liposomal amphotericin B. We present a 40-year-old male with fever and a painful necrotic lesion on the middle back and history of poorly controlled diabetes mellitus. Rhizopus oryzae was isolated and identified using an internal transcribed spacer regions ITS1 and ITS2. An initial good response to treatment was observed; however, 7 days later a diabetic ketoacidosis due to poor adherence to treatment caused a lethal outcome.     

A Mucormycosis Case in a Cirrhotic Patient Successfully Treated with Posaconazole and Review of Published Literature

Mucormycosis is an invasive fungal infection associated with a high mortality rate, especially in immunocompromised hosts. Mucormycosis rarely occurs in cirrhotic patients. Here, we report a case of mucormycosis with underlying liver cirrhosis and diabetes mellitus. The patient suffered from maxillary sinusitis and osteomyelitis, and the infection was successfully treated with antifungal agents, surgical debridement, and hyperbaric oxygen therapy. The antifungal treatments used were liposomal amphotericin B, itraconazole, and posaconazole. Although our patient had liver cirrhosis (Child-Pugh classification B), no hepatic decompensation was developed during the treatment course of posaconazole. This is the first report of the safe and effective use of posaconazole for the treatment of mucormycosis in a cirrhotic patient.     

Gangrenous Cutaneous Mucormycosis Caused by Rhizopus oryzae: A Case Report and Review of Primary Cutaneous Mucormycosis in China Over Past 20 Years

Cutaneous mucormycosis is a rare opportunistic infection caused by zygomycetes that can be rapidly fatal if unrecognized. We describe the clinical, histopathological, fungal and molecular features of a case of gangrenous cutaneous mucormycosis. The patient presented with great necrosis on his right forearm at the site of detained intravenous cannula needle. He had type II diabetes and chronic renal insufficiency. KOH mount of black eschar showed many broad, aseptate fungal hyphae with right-angle branching. PAS staining of the tissue sample revealed similar broad hyphae in the dermis and cutis. Fungal culture and ITS sequence analysis identified this fungus as Rhizopus oryzae. As no organ involvement was detected, the patient was diagnosed with primary cutaneous mucormycosis. Considering the poor state of the patient, complete excision of the infectious tissue was performed without skin graft instead of amputation. At the same time, intravenous liposomal amphotericin B was given, starting from a small dosage and increased to a total dosage amount of 5.45 g. The wound recovered well with granulation. We emphasize that early recognition and prompt therapy including the control of the primary diseases were important. In this article, we also reviewed the features of primary cutaneous mucormycosis reported in China over the last 20 years.     

Mucormycosis in Mato Grosso, Brazil: A Case Reports, Caused by Rhizopus microsporus var. oligosporus and Rhizopus microsporus var. rhizopodiformis

We identified the etiological agents responsible for two fatal cases of rhinocerebral mucormycosis with the classical risk factor for uncontrolled type II diabetes mellitus. Their initial symptoms did not point immediately to the suspicion of mucormycosis. Case 1, caused by Rhizopus microsporus var. oligosporus, was a 52-year-old man who presented with a painful pimple on his nose, which evolved with swelling, erythema, and a central pustule on his right hemiface suspected to be cellulitis. After 7 days of antibiotic treatment, the patient worsened with signs of sepsis and the lesion evolved to necrosis involving all his right face. Case 2, caused by Rhizopus microsporus var. rhizopodiformis, was a 57-year-old woman placed on continuous therapy with azathioprine and corticoids after a renal transplant due to chronic arterial hypertension and uncontrolled type II diabetes mellitus. Because she was suspected to have sepsis, the patient was treated with broad-spectrum antibiotics and mechanical ventilation, yet she deteriorated. Because Candida spp. were isolated from urine and a BAL, she was treated with fluconazole for 10 days, then substituted by caspofungin. Two weeks later, she presented with exophthalmus of the left eye that was surrounded by a large inflammatory and necrotic area. Both patients were the diagnosed with mucormycosis via direct microscopy of necrotic material prior to their death.     

Primary Cutaneous Mucormycosis Presenting as a Giant Plaque: Uncommon Presentation of a Rare Mycosis

Mucormycosis is an uncommon opportunistic fungal infection caused by Zygomycetes. It usually affects immunocompromised, diabetic and trauma patients with infected wounds. We report a case of disseminated infection secondary to facial cutaneous mucormycosis caused by Saksenaea vasiformis in a diabetic patient who had a farming accident causing him severe head injury. The patient was treated with a combination of surgical debridement and antifungal therapy with liposomal amphotericin B, but he had a slow and fatal outcome. In cases of tissue necrosis following trauma involving wound contact with soil (i.e., potential fungal contamination), testing for the presence of Zygomycetes fungi such as S. vasiformis in both immunocompetent and immunocompromised patients is crucial. The reason is that this infection usually has a rapid progression and may be fatal if appropriate treatment is not administered.     

Successful Treatment of Pulmonary Mucormycosis Caused by <Emphasis Type="Italic">Cunninghamella bertholletiae</Emphasis> with High-Dose Liposomal Amphotericin B (10 mg/kg/day) Followed by a Lobectomy in Cord Blood Transplant Recipients

Infection caused by Cunninghamella bertholletiae carries one of the highest mortality rates among mucormycosis, and there are no reported cases that survived from the infection in allogeneic hematopoietic stem cell transplantation recipients occurring before neutrophil engraftment. Here, we present two cases of pulmonary mucormycosis caused by C. bertholletiae occurring before neutrophil engraftment after cord blood transplantation. Both were successfully treated with high-dose liposomal amphotericin B (10 mg/kg/day) combined with micafungin, which was then followed by neutrophil recovery, reduction in immunosuppressive agents, and a subsequent lobectomy. The intensive antifungal therapy immediately administered upon suspicion of mucormycosis greatly suppressed the infection in its early stage and was well tolerated despite its prolonged administration and simultaneous use of nephrotoxic agents after transplantation. Although the synergic effect of micafungin remains unclear, these cases highlight the importance of prompt administration of high-dose lipid polyene when suspecting mucormycosis in highly immunocompromised patients, which enables subsequent diagnostic and therapeutic interventions, resulting in a favorable outcome.     

Endogenous Fusarium Endophthalmitis During Treatment for Acute Myeloid Leukemia,Successfully Treated with 25-Gauge Vitrectomy and Antifungal Medications

Endogenous fungal endophthalmitis (EFE) caused by disseminated fusariosis is a rare condition that generally has a poor outcome, even with intensive therapy. Here, we describe a case in which this type of EFE was diagnosed with vitreous sampling and was successfully treated with 25-gauge vitrectomy and antifungals, including liposomal amphotericin B and voriconazole. A 16-year-old male patient undergoing treatment for acute myeloid leukemia complained of eye pain and blurred vision in his right eye. Treatment was initiated for a vitreous opacity, possibly associated with herpetic retinitis, but the patient worsened and he was referred to us. Right-eye visual acuity was limited to light perception. We suspected endogenous endophthalmitis and performed 25-gauge vitrectomy with antibiotic perfusion of ceftazidime, vancomycin, and voriconazole. Vitreous culturing revealed the presence of Fusarium solani species complex, and enhanced computed tomography revealed disseminated fusariosis lesions in the lung, spleen, and the soft tissue of the left upper arm. The patient received antifungal treatment with liposomal amphotericin B and voriconazole, and these conditions were eliminated. Visual acuity recovered to 20/400 after additional vitrectomy for tractional retinal detachment and was maintained at this level during the 6-month follow-up period. The success of our treatment allowed the capture of optical coherence tomography images of the retina during fusarium-associated endogenous endophthalmitis and the follow-up period. Furthermore, this case showed that immediate vitrectomy for suspected EFE and intensive treatment can lead to a good clinical outcome.     

Therapeutic Activity of Liposomal Amphotericin B in Systemic Mycoses

Abstract

Several clinical trials are presently underway to study various liposomal formulations of antineoplastics and antimicrobial agents (1–3). Both liposomal amphotericin B (L-AmpB) and various liposome formulations have been used in the treatment of experimental systemic fungal infections (4–6) and leishmaniasis (7,8) in animals. In most reports, an enhanced therapeutic index was observed. A uniform reduction in the toxic effects usually attributed to AmpB was reported and, in some cases, an enhanced therapeutic efficacy was also attained. We recently reported on the use of L-AmpB in 46 patients with systemic fungal infections (9). of that number, 31 had leukemia, 2 had lymphoma, 11 had other diagnoses (5 had received intensive immunosuppressive therapy following a heart transplantation, 2 had multiple myeloma, and the rest had other malignancies); 2 patients had no underlying disease. Forty-one of these patients had been previously treated with conventional AmpB. the fungal diagnosis was candidiasis in 21 patients, aspergillosis in 19, agents of mucormycosis in 3, and other diagnoses in the rest.     

El tratamiento de la mucormicosis (cigomicosis) en el siglo xxi

Infections due to zygomycetes, caused by mucorales and entomophthorales, are characterized by angioinvasion and invasion of neighboring organs or structures. Mucorales most commonly cause rhinocerebral, pulmonary, cutaneous or disseminated infection and its spread is favored by several diseases (such as diabetes or chronic kidney disease) and risk factors (neutropenia, immunosuppression, iron overload). They have a high mortality rate, and the key to success in their treatment are early diagnosis, prompt administration of antifungal treatment, and extensive surgical debridement. Currently, isavuconazole constitutes an option for the treatment of those mucormycosis refractory to liposomal amphotericin B. Due to its pharmacokinetic and pharmacodynamic characteristics and its low toxicity, it is also the best choice for maintenance therapy.     

急性淋巴细胞白血病患者侵袭性头状地霉感染1例及文献回顾

目的:通过报道1例急性淋巴细胞白血病患者侵袭性头状地霉感染的临床资料,并结合文献探讨头状地霉感染的临床特点、有效的诊断及治疗方法。方法:报道国内首例急性淋巴细胞白血病患者化疗后骨髓抑制期感染头状地霉病例,并对该病的诊断及治疗等进行系统文献回顾。结果:该白血病患者经血培养证实为头状地霉感染,并累及肺脏、肝脏和皮肤,治疗过程中先后采用卡泊芬净、脂质体两性霉素B和脂质体两性霉素B联合伏立康唑等治疗,虽然脂质体两性霉素B联合伏立康唑治疗患者体温正常,临床症状稍有改善,但是患者在化疗后40天放弃治疗并死于心肺功能衰竭。结论:头状地霉感染的发病率低,临床症状不够典型,诊断困难,预后差。根据患者的临床表现,结合血培养、GM实验、G实验和CT扫描等检查,可有助于诊断。头状地霉感染尚无非常有效的治疗方式,采用脂质体两性霉素B或两性霉素B联合伏立康唑或其他新的抗真菌药物可能获得一定的疗效,早期诊断、早期联合治疗和患者早期脱离粒缺状态是治疗成功的关键。     

Cutaneous Protothecosis in Patient with Diabetes Mellitus and Review of Published Case Reports

Protothecosis is an opportunistic infection caused by Prototheca, usually called as saprophytes, and is frequently found in natural and living surroundings with low virulence, but may cause chronic infection in immunocompromised individuals. We report a case of cutaneous protothecosis with zopfii var. portoricensis infection in a 66-year-old diabetic woman following hand surgery on middle right finger. Mycology study showed that smooth, creamy white, yeast-like colonies grown after necrotic tissue was cultured on Sabouraud dextrose agar at both 37 and 25°C. The organism was then identified as Prototheca zopfii var. portoricensis by molecular identification and also found from histopathology of the lesion. The lesion got improved with intravenous amphotericin B and itraconazole.     

Fluconazole Non-susceptible <Emphasis Type="Italic">Cryptococcus neoformans</Emphasis>, Relapsing/Refractory Cryptococcosis and Long-term Use of Liposomal Amphotericin B in an AIDS Patient

The treatment of cryptococcosis is hampered by inefficacy or intolerance to the recommended antifungal agents. A patient diagnosed with AIDS had multiple relapses of cryptococcal infection, which became refractory to antifungal agents during the course of therapy. During the follow-up, the patient developed renal toxicity due to amphotericin B use and non-susceptibility of isolated Cryptococcus neoformans to fluconazole was detected. Thereafter, antifungal treatment was performed exclusively with liposomal amphotericin B, reaching a cumulative dose of 19,180 mg over 46 months. The final relapse of cryptococcosis occurred during the maintenance phase with liposomal formulation in a once-weekly dose. Measurement of the minimum serum concentrations of amphotericin B, determined sequentially before and after this relapse, suggested the importance of monitoring drug levels when the liposomal formulation is used for a long period.     

Mucormycosis in a Patient with Acute Myeloid Leukemia Successfully Treated with Liposomal Amphotericin B Associated with Deferasirox and Hyperbaric Oxygen

We report a 38-year-old woman presenting with febrile neutropenia, acute myeloid leukemia (AML) and invasive mucormycosis. Bone marrow aspirate was characteristic of AML minimally differentiated (WHO classification 2008). Flow cytometric immunophenotyping analysis showed blasts positive for CD7, CD33, CD34, CD71, CD117, HLA-DR, MPO, and TdT, with normal karyotype (46, XX), and the absence of the FLT3-ITD and NPM1 mutations. The patient’s management included chemotherapy with cytarabine and idarubicin, and treatment with liposomal amphotericin B, deferasirox, hyperbaric oxygen therapy, and antibiotics. Nowadays, she is in complete hematological remission, and CT images of control are normal. Invasive mucormycosis is an uncommon and severe condition, which involves diagnosis and treatment challenges. Clinical features and predisposing factors should be highlighted in order to enhance the suspicion index, contributing to early diagnosis and disease control. Our aim is to report classical features of this uncommon condition and to emphasize usual management challenges.     

Isavuconazole in a Successful Combination Treatment of Disseminated Mucormycosis in a Child with Acute Lymphoblastic Leukaemia and Generalized Haemochromatosis: A Case Report and Review of the Literature

Invasive mucormycosis in immunocompromised children is a life-threatening fungal infection. We report a case of a 7-year-old girl treated for acute lymphoblastic leukaemia complicated by disseminated mucormycosis during induction therapy. Microscopic examination of surgically removed lung tissue revealed wide, pauci-septate hyphae suggesting a Mucorales infection. This diagnosis was confirmed immunohistochemically and by PCR analysis followed by a final identification of Cunninghamella sp. The patient was treated successfully with surgical debridement and antifungal combination therapy with amphotericin B, caspofungin and isavuconazole. The use of isavuconazole in a child was not previously reported. Additionally, case reports concerning pulmonary mucormycoses in paediatric population published after 2010 were reviewed. Nineteen out of 26 identified patients suffered from haematological diseases. Reported mortality reached 38.5%. By the fact of rising morbidity, unsatisfactory results of treatment and remaining high mortality of mucormycoses in immunocompromised patients, new therapeutic options are warrant. Isavuconazole, with its broad-spectrum activity, good safety profile and favourable pharmacokinetics, is a promising drug. However, further studies are necessary to confirm positive impact of isavuconazole on mucormycosis treatment in children.

     

多变根毛霉致面部皮肤毛霉病1例

报道1例由多变根毛霉引起的面部皮肤毛霉病.患者男,65岁,面部结节斑块伴痒半年余.皮损组织病理检查示真皮中下层有炎细胞及多核巨细胞浸润,并见粗大较短的无隔菌丝.经真菌培养和分子生物学鉴定,菌种鉴定为多变根毛霉.皮损经短时两性霉素B治疗后好转.     

Problemas clínicos en micología médica: problema número 45

A 43 year-old diabetic woman, who suffered chronic cough and brown expectoration, is presented in this clinical problem. X-ray exam and CT thorax scan showed a cavitary lung lesion, located at the upper field of the left lung. This lesion had 5 cm in diameter, with a thick wall and a spherical shadow inside. The diagnosis of chronic cavitary pulmonary coccidioidomycosis was based on the isolation of Coccidioides sp. from cultures of expectoration and bronchoalveolar lavage, and the detection of specific antibodies in immunodiffusion test and counterimmunoelectrophoresis with coccidiodin. Her diabetes was not well controlled. She was treated with intravenous amphotericin B and oral itraconazole, with good clinical response; after four months of treatment the patient abandoned clinical controls.We suppose that the patient presented a coccidioidal fungus ball, inside a chronic cavitary lesion due to pulmonary coccidiodomycosis. She came from an endemic zone of coccidioidomycosis in the Northwest of the Argentine Republic (Catamarca Province).     

Coronavirus Disease (Covid-19) Associated Mucormycosis (CAM): Case Report and Systematic Review of Literature

Severe coronavirus disease (COVID-19) is currently managed with systemic glucocorticoids. Opportunistic fungal infections are of concern in such patients. While COVID-19 associated pulmonary aspergillosis is increasingly recognized, mucormycosis is rare. We describe a case of probable pulmonary mucormycosis in a 55-year-old man with diabetes, end-stage kidney disease, and COVID-19. The index case was diagnosed with pulmonary mucormycosis 21 days following admission for severe COVID-19. He received 5 g of liposomal amphotericin B and was discharged after 54 days from the hospital. We also performed a systematic review of the literature and identified seven additional cases of COVID-19 associated mucormycosis (CAM). Of the eight cases included in our review, diabetes mellitus was the most common risk factor. Three subjects had no risk factor other than glucocorticoids for COVID-19. Mucormycosis usually developed 10–14 days after hospitalization. All except the index case died. In two subjects, CAM was diagnosed postmortem. Mucormycosis is an uncommon but serious infection that complicates the course of severe COVID-19. Subjects with diabetes mellitus and multiple risk factors may be at a higher risk for developing mucormycosis. Concurrent glucocorticoid therapy probably heightens the risk of mucormycosis. A high index of suspicion and aggressive management is required to improve outcomes.

     

Ocular Histoplasmosis

Case reports of three residents of Ontario with clinical histoplasmic chorioretinitis are presented. The diagnosis was made on the basis of the clinical appearance, the presence of calcified lesions in the chest, a negative skin test to tuberculin, and a positive skin test to toxoplasmin. All patients were treated with intravenous amphotericin B. Except for transitory elevation of blood urea nitrogen, there were no serious complications from the drug and in all cases the lesions in the eyes were improved. Histologic or cultural proof of the presence of fungus in the eye is not available, but clinical and laboratory findings can combine to point to the diagnosis of histoplasmosis. In such cases, since vision is at stake, treatment with amphotericin B should be considered.