Cutaneous mucormycosis: a case report

Cutaneous mucormycosis is a rare opportunistic fungal infection. It usually is benign in immunocompetent patients, but it can lead to devastating consequences in immunocompromised patients. Immediate diagnosis and treatment are crucial in preventing fatality. We describe a case of cutaneous mucormycosis in a man with a history of non-Hodgkin lymphoma in an effort to raise diagnostic suspicion of this life-threatening infection and prevent a fatal outcome.     

伊曲康唑治疗1例原发性皮肤毛霉病

应用伊曲康唑治疗１例罕见的由冻土毛霉黄色（或土黄）型所致的原发性皮肤毛霉病,并对其治疗前后的临床、真菌学及组织病理改变进行了观察。临床、真菌学痊愈。斯皮仁诺对毛霉目某些真菌是敏感的,对原发性皮肤毛霉病的治疗是安全有效的。     

不规则毛霉感染引起的皮肤毛霉病20例回顾性分析

目的通过对不规则毛霉感染病例的回顾性分析,探讨不规则毛霉感染引起皮肤毛霉病的流行病学、易感因素、临床表现、诊断方法及治疗策略,提高临床医生的警惕性及诊治经验。方法对检索到的20例不规则毛霉感染病例资料进行总结、分析。结果 20例不规则毛霉感染患者均表现为皮肤毛霉病,仅1例同时伴有肺部感染。15例来自中国,1例有糖尿病史,1例有白血病史,10例有外伤、手术或叮咬史。皮损几乎均出现在暴露部位,其中鼻面部13例,四肢7例,病程多呈慢性。早期表现为红斑、丘疹、结节,后期可发展成溃疡、坏死、黑痂及骨质破坏。经真菌镜检、培养、组织病理、ITS区测序等检查证实为不规则毛霉。两性霉素B及其脂质体单独或联合伊曲康唑治疗预后好。结论不规则毛霉感染多发生于免疫正常者,外伤、烧伤、手术及叮咬是最主要诱发因素。感染主要累及鼻面部及四肢皮肤暴露部位,以红斑、坏死为主要表现,极少累及内脏。真菌培养及组织病理检查可以诊断该病,ITS区测序可明确鉴定到种。及早诊断并给予清创及两性霉素B是治疗成功的关键。     

The emergence of mucormycosis as an important opportunistic fungal infection: five cases presenting to a tertiary referral center for mycology

BACKGROUND: Mucormycosis, a rare opportunistic fungal infection, is re-emerging in importance with the increase in prevalence of immunosuppressive states, both as a result of therapy and disease. METHODS: We report five cases of mucormycosis diagnosed by the Dermatology Department and managed jointly with the Medical and Surgical Services of "Dr Manuel Gea Gonzalez" General Hospital in Mexico City, a tertiary referral center for mycology. We also review the current literature including recent advances in medical therapy. RESULTS: Four of the five cases were of the rhino-orbital-cerebral variant, commonly associated with significant mortality, and one of these patients died despite early diagnosis and aggressive management. The fifth case was primary cutaneous mucormycosis and this patient survived infection without relapse. Diabetic ketoacidosis predisposed to infection in four cases and the other was associated with advanced human immunodeficiency virus infection. Radiologic imaging was important in cases of facial involvement in order to evaluate the extent of disease and possible intracranial involvement. All cases were managed with systemic antifungals and surgical debridement, together with the treatment of predisposing factors. CONCLUSIONS: These cases illustrate the need for early clinical recognition and prompt therapy, as well as the requirement for tissue biopsy in order to demonstrate the characteristic morphologic features of this fungal agent in the absence of positive mycology culture results. This report also highlights that, although rhino-orbital-cerebral mucormycosis requires effective multidisciplinary management, the disease not uncommonly presents to dermatologists for diagnosis.     

毛霉病七例临床与病理分析

目的 探讨毛霉病的临床表现、预后和病理表现之间可能存在的关系。方法 回顾性分析1989-2006年我院诊治的7例经真菌培养证实为毛霉病患者的临床资料和病理切片。结果 7例中鼻脑毛霉病1例、皮肤毛霉病6例，其中2例为毛霉性坏死性筋膜炎。鼻脑毛霉病和毛霉性坏死性筋膜炎患者病情进展快，均以死亡告终；其皮损组织病理表现呈混合性炎症细胞浸润，坏死明显，可见大量菌丝和侵血管现象。4例以浸润性红斑为主要表现的皮肤毛霉病患者病情进展慢，有2例已治愈；其皮损组织病理呈肉芽肿性炎症，菌丝较少，未见明显的侵血管现象。7例均未见明确的菌丝侵神经现象。结论 毛霉病皮损组织病理表现呈混合性炎症细胞浸润，菌丝多，出现明显侵血管现象者预后较差。     

Palatal necrosis in an AIDS patient: a case of mucormycosis

We report a case of rhinocerebral mucormycosis presenting in a patient with AIDS and review the literature on mucormycosis occurring in the setting of HIV disease. Mucormycosis in HIV is rare. However, it can be the presenting opportunistic infection in AIDS. Predisposing factors for Mucor infection in HIV disease include low CD4 count, neutropenia, and active intravenous drug use. Mucormycosis can present in the basal ganglia, the skin, the gastrointestinal tract, the respiratory tract, or may be disseminated. The disease may develop insidiously or may progress rapidly with a fulminant course. Therapy usually consists of surgical debridement/excision accompanied by intravenous amphotericin B.     

Cutaneous mucormycosis

We report two cases of primary cutaneous mucormycosis in immunocompromised patients. Although their clinical presentation showed nonspecific necrotic ulcers, the histopathologic features were diagnostic. In both cases, broad, branching, nonseptate hyphae were found in the dermis and subcutaneous tissue. In one case the hyphae could be observed in the epidermis. To our knowledge, this is the only reported case of primary cutaneous mucormycosis with hyphae detectable in the epidermis.     

Cutaneous mucormycosis in a heart transplant patient

We present the first case of cutaneous mucormycosis reported in a patient who had undergone a heart transplant operation. This appeared to be a localized infection without a predisposing local factor such as a surgical adhesive and without evidence of dissemination. Prompt treatment with intravenous amphotericin B resulted in an apparent cure.     

2型糖尿病合并鼻窦部毛霉菌感染1例

毛霉菌病是一种急性霉菌感染,常具有致死性。鼻脑型毛霉菌病多见于糖尿病患者,其他严重的免疫缺陷患者中也可见到。本文报告1例糖尿病合并鼻眶部毛霉菌患者,经早期诊断及手术治疗,预后良好。提示糖尿病患者出现坏死性鼻窦炎并伴随急性神经功能障碍时应考虑鼻脑型毛霉菌病的可能。早期诊断和治疗是预后的关键因素。     

Rhino-orbito-cerebral mucormycosis caused by Rhizopus microsporus var. microsporus in a diabetic patient with COVID-19

COVID-19 disease caused by the SARS-CoV-2 coronavirus causes a wide range of clinical manifestations, ranging from mild to severe, with the main ones affecting the respiratory tract, such as pneumonia. In patients with greater severity, the high frequency of bacterial and fungal coinfection stands out, a situation related both to the patient's pre-existing comorbidities and due to the hospitalization itself. Cases of mucormycosis associated with COVID-19 were highlighted in the lay and scientific media, with the increase in mycosis cases being directly and indirectly attributed to the viral infection. This report describes a case of rhino-orbito-cerebral mucormycosis in a diabetic patient hospitalized for COVID-19, whose diagnosis was confirmed by identifying the agent Rhizopus microsporus var. microsporus through culture for fungi and PCR examination.     

Primary Cutaneous Mucormycosis in an Extremely Preterm Infant Successfully Treated with Liposomal Amphotericin B

Cutaneous mucormycosis is a rare but often fatal invasive fungal infection that occurs most commonly in patients with diabetes, malignancy, and other immunocompromising conditions. We report an extremely preterm (<28 weeks) baby boy who developed polymicrobial sepsis and primary cutaneous mucormycosis within his first 10 days of life. He was successfully treated with medical management alone since he was not a candidate for surgery. Successful treatment of cutaneous mucormycosis without surgical debridement has been reported on only two other occasions. This case highlights the importance of rapid and thorough evaluation of skin lesions when evaluating preterm infants and other immunocompromised patients, even when other sources of infection have been identified.     

Primary cutaneous mucormycosis in a trauma patient

We report a rare case of primary cutaneous mucormycosis caused by Rhizopus oryzae that occurred in an immunocompetent trauma patient. The patient had encrusted erythematous plaques with pustules on the left shin, which had been abraded in a traffic accident. Histologic examination revealed widespread granulomatous inflammation and characteristic broad, non-septate hyphae with right-angle branching in the dermis. The infection was cured with intravenous amphotericin B therapy.     

原发性皮肤毛霉病

报告1例原发性皮肤毛霉病.患者男,57岁.右前臂埋置静脉套管针17 d后局部出现红肿,迅速坏死并扩大.患者患2型糖尿病及慢性肾功能不全.皮肤科检查示右前臂大片坏死区,上覆黑色焦痂及渗出.取黑痂及渗液行真菌镜检,镜下可见大量粗大、无分隔、垂直分枝的菌丝,培养第2天即有棉花糖样菌落生长,初步诊断为皮肤毛霉病.皮损行组织病理检查,PAS染色示真皮及皮下组织可见较多粗大、无分隔菌丝,确诊为皮肤毛霉病.给予两性霉素B脂质体泵入、坏死组织清创术、术后外敷两性霉素B脂质体溶液及外用人成纤维细胞生长因子.患者伤口愈合良好,随访4个月无复发.     

Successful treatment of a patient with cutaneous co-infection caused by Mucor irregularis and Klebsiella pneumoniae

The authors report a rare case of primary cutaneous mucormycosis caused by Mucor irregularis and cutaneous Klebsiella pneumoniae infections in a 67-year-old Chinese woman. After the administration of liposomal amphotericin B combined with cefoperazone/sulbactam sodium, the patient recovered. Invasive fungal infection combined with cutaneous bacterial infection should receive attention.     

Primary cutaneous mucormycosis caused by Mucor irregularis in an elderly person

Primary cutaneous mucormycosis is a rare but often lethal severe fungal infection, which usually occurs in immunocompromised patients. We report a case of primary cutaneous mucormycosis caused by Mucor irregularis in an elderly patient. Seven months after the surgical dissection of the involved skin, cutaneous mucormycosis recurred at the peripheral edge of the skin graft. Shortly subsequent to the administration of liposomal amphotericin B, the remaining skin lesion was excised again. M. irregularis is rarely but increasingly reported as a cause of mucormycosis in immunocompetent individuals, especially in Asian farmers. M. irregularis may be largely disseminated in the soils of Asia and thus the trivial trauma at the time of farm work may be a trigger for the onset. These cases tend to leave severe cosmetic damage even in healthy individuals, although the vital prognosis is not affected.     

Mucormicosis rinocerebral fulminante

—We present the case of a 53-year-old male who presented with a rapidly progressing rhinocerebral mucormycosis, causing a large centrofacial necrotic plaque and the death of the patient, despite the quick implementation of intravenous amphotericin B. The patient presented numerous factors which predisposed him to the disease, as he was a malnourished patient who had been treated with chemotherapy two months before for a cancer of the larynx; he also presented with pancytopenia and was being treated for pneumonia with broad-spectrum antibiotics. The multiple predisposing factors may have been the cause of the fulminant course of the mucormycosis.     

Cutaneous infarction. Manifestation of disseminated mucormycosis.

We describe the second reported case of cutaneous infarction as a manifestation of disseminated mucormycosis. The lesion, which closely resembled ecthyma gangrenosum, occurred in a leukemic patient who was on a regimen of broad-spectrum antibiotics. In this case as in the only other reported case, Mucor pusillus was the cause.     

Primary cutaneous mucormycosis: a diagnosis to consider

Primary cutaneous mucormycosis is a deep fungal infection, mainly seen in diabetics and immunocompromised subjects. Rapid diagnosis and therapy are necessary to avoid fatal outcome. We describe the complete histopathological and microbiological studies of primary cutaneous mucormycosis in a 74-year-old man with several risk factors, such as chronic obstructive pulmonary disease, respiratory acidosis, hemolytic anemia, myelodysplastic syndrome and iatrogenic diabetes, due to corticosteroid therapy. He developed two cutaneous necrotic scars on his left leg. Mucormycosis was suspected and specimens from surgical débridement were histopathologically and microbiologically studied confirming the clinical diagnosis. Amphotericin B was given topically and intravenously resulting in complete healing of the ulcer. Risk factors and microbiological studies are compared with those in the current literature. It is necessary in certain cases to suspect mucormycosis infections in diabetics, immunocompromised subjects and even in healthy individuals. Rapid diagnosis and treatment are important, but they should be based on complete histopathological and microbiological studies, to establish the genus of the causal agent.     

Genetic identification and detection of human pathogenic Rhizopus species, a major mucormycosis agent, by multiplex PCR based on internal transcribed spacer region of rRNA gene

BACKGROUND: Mucormycosis is an invasive opportunistic infection caused by fungi belonging to the order Mucorales. Due to the lack of laboratory tests, the diagnosis of mucormycosis is notoriously difficult. Added with its rapid progression as well as the debilitated state of the patients who contract the disease, mortality is extremely high. OBJECTIVE: The goal of this study was to genetically identify human pathogenic Rhizopus species, a major mucormycosis agent, by the internal transcribed spacer (ITS) region of rRNA gene. METHODS: Primers were designed to identify five Rhizopus species known to cause human disease by multiplex PCR. PCR was done not only with test strains and clinical isolates, but also with clinical samples from cutaneous mucormycosis patients. Sporangiospore morphology was observed by scanning electron microscopy to confirm the correlation of phenotypic and genotypic features. RESULTS: Multiplex PCR identified five Rhizopus species including Rhizopus oryzae, where R. azygosporus could only be distinguished from R. microsporus by certain polymorphisms that were present in its sequence. When this multiplex PCR was applied to clinical samples from three mucormycosis patients (paraffin sections from all and sera from one patient), Rhizopus DNA corresponding to the isolated pathogens were specifically detected. CONCLUSION: While fungal DNA detection from clinical samples is a rigorously studied area, this is the first report to genetically identify and detect Rhizopus species from human mucormycosis specimens. This may expand the possibility of this multiplex PCR system not only to identify isolated fungi, but also as a screening method for visceral mucormycosis.     

The first case of cutaneous mucormycosis caused by Rhizopus azygosporus

A rapidly enlarging leg ulcer appeared in a 54-year-old woman with systemic lupus erythematosus receiving aggressive immunosuppressive therapy. Skin biopsy revealed proliferation of hyphae in the midst of a neutrophilic abscess. Culture yielded Rhizopus azygosporus. As no organ involvement was detected by thorough examination, the patient was diagnosed as having primary cutaneous mucormycosis. Although intravenous amphotericin B therapy seemed to be very effective, it had to be discontinued due to nephrotoxicity. She unfortunately died of subsequent disseminated fungal infection and cerebral infarction in which the primary cause could not be determined. Minimum inhibitory concentrations of several antifungal drugs to the isolate were examined and amphotericin B proved to be the only agent that may potentially reach the effective plasma concentration. This is the first case report of cutaneous mucormycosis caused by R. azygosporus.