Isolated palate ulcer due to mucormycosis

Mucormycosis is a rare but serious fungal infection that rapidly attacks and kills its untreated victims, who are often immunocompromised. It is one of the most fulminant and often fatal mycotic infections known to human beings. Rhinocerebral mucormycosis is the commonest presentation and its extension to the orbit and brain is quite usual but the palatal involvement is a rare and late occurrence. Isolated location of mucormycosis on the palate in an immunocompetent host is an unusual clinical entity. Here we report a case of deep hard palate ulcer due to mucormycosis in a 56-year-old man without any predisposing factor. He was successfully treated with a combination of surgical debridement and systemic liposomal amphotericin B administration for six weeks. By presenting this case report we would like to emphasis that mucormycosis should be included in the differential diagnosis of the hard palate ulcers even in immunocompetent patient.     

Rhinocerebral mucormycosis: A deadly disease on the rise

According to WHO, there will be epidemic of diabetes world over and India is going to be ‘A diabetes capital of the world’ by 2025. With the increasing incidence of diabetes, the associated complications are also bound to increase. Rhinocerebral mucormycosis is one of them. Rhinocerebral Mucormycosis is an opportunistic, fulminating fungal infection, caused by Rhizopus species of order of mucorales, frequently seen in diabetic and immunocompromised patients. Mucormycosis has a very high mortality rate. Early diagnosis and treatment with Amphotericin-B is the key to combat this disease successfully. We have seen 13 cases in last 3 years (2002–2005) in our area. This incidence is significant, as this type of cases were rarely seen before 2002, in this geographical area. We present an account of these cases; treatment strategies adopted, review of literature, and highlight ‘the role of ENT surgeon in diagnosis and management of this dreadful disease’.     

Mucormycosis in Iran: a systematic review

Fungi in the order Mucorales cause acute, invasive and frequently fatal infections in susceptible patients. This study aimed to perform a systematic review of all reported mucormycosis cases during the last 25 years in Iran. After a comprehensive literature search, we identified 98 cases in Iran from 1990–2015. The mean patient age was 39.8 ± 19.2 years. Diabetes was the most common underlying condition (47.9%), and 22.4% of the patients underwent solid organ or bone marrow transplantation. The most common clinical forms of mucormycosis were rhinocerebral (48.9%), pulmonary (9.2%) and cutaneous (9.2%). Eight cases of disseminated disease were identified. Overall mortality in the identified cases was 40.8%, with the highest mortality rate in patients diagnosed with disseminated infection (75%). The mortality rate in rhinocerebral infection patients was significantly lower (45.8%). Rhinocerebral infection was the most common clinical manifestation in diabetes patients (72.9%). Patients were diagnosed using various methods including histopathology (85.7%), microscopy (12.3%) and culture (2.0%). Rhizopus species were the most prevalent (51.7%), followed by Mucor species (17.2%). Sixty‐nine patients were treated with a combination of surgery and antifungal therapy (resulting survival rate, 66.7%). Owing to the high mortality rate of advanced mucormycosis, early diagnosis and treatment may significantly improve survival rates. Therefore, increased monitoring and awareness of this life‐threatening disease is critical.     

Successful Treatment of Rhinocerebral Mucormycosis with Liposomal Amphotericin B and Surgery in Two Diabetic Patients with Renal Dysfunction

Abstract

The zygomycetes are a class of fungi that can cause a variety of infections in humans. Rhinocerebral mucormycosis is a rare disease and usually affects diabetic or immunosuppressed patients. The disease progresses rapidly and is usually fatal despite aggressive surgical and medical therapy. We report the management of two cases of rhino-sinusal and orbital mucormycosis in diabetic patients on treatment with corticosteroids, and mild renal impairment, successfully treated with a combination of aggressive surgical debridement and liposomal amphotericin B.     

Rhinocerebral phycomycosis

Rhinocerebral mucormycosis is a rare opportunistic fungal infection which may prove fatal. It involves the nose, paranasal sinuses, orbit and may spread intracranially. It is usually associated with metabolic disorders, especially ketosis. Present paper describes our experience of managing five such cases, two of which died. An early diagnosis and prompt agressive therapy in the form of local surgical correction of underlying disorder plus administration of amphotericin B are essential.     

Successful treatment of rhinocerebral mucormycosis with liposomal amphotericin B and surgery in two diabetic patients with renal dysfunction

The zygomycetes are a class of fungi that can cause a variety of infections in humans. Rhinocerebral mucormycosis is a rare disease and usually affects diabetic or immunosuppressed patients. The disease progresses rapidly and is usually fatal despite aggressive surgical and medical therapy. We report the management of two cases of rhino-sinusal and orbital mucormycosis in diabetic patients on treatment with corticosteroids, and mild renal impairment, successfully treated with a combination of aggressive surgical debridement and liposomal amphotericin B.     

Delaying diagnostic procedure significantly increases mortality in patients with invasive mucormycosis

Invasive mucormycosis is an uncommon but increasing life‐threatening fungal infection. The present study investigated clinical characteristics and mortality among patients diagnosed as invasive mucormycosis infection. We retrospectively reviewed a total of 24 histologically proven cases of invasive mucormycosis at two tertiary care referral hospitals between November 2005 and February 2014. Overall survival was 50% (n = 12). The time between onset of symptom and diagnostic procedure proved to be associated with mortality (P = 0.009). In addition, preexisting renal failure and thrombocytopenia demonstrated trends toward a poor outcome in our study (P = 0.089 and 0.065, respectively). On multivariate regression analysis, delayed diagnostic procedure (more than 16 days after the onset of symptoms) was an independent predictor of mortality (OR= 12.34, 95% CI, 1.43–10.64; P = 0.022). Mucormycosis is a destructive fungal infection that is associated with high mortality rates, ranging from 40% to 100% depending on the form of disease. When a clinician suspects invasive mucormycosis infection, an early diagnostic procedure performed within 16 days from the onset of symptom and early initiation of antifungal therapy will lead to successful management of this highly fatal disease.     

Localized muscular mucormycosis in a child with acute leukemia

Mucormycosis is a rare fungal infection of childhood, occurring mainly in patients with chronic illnesses such as diabetes and malignancies. The fungus seldom grows in culture and confirmation of the diagnosis depends on histologic examination of infected tissues. To date, the reported natural history of the disease has been rapid progression and a fatal outcome. Therefore, the importance of early diagnosis by tissue biopsy and early treatment with surgical debridement and systemic antifungal therapy cannot be overemphasized. The pulmonary system is the most common site for mucormycosis in patients with leukemia. We report what we believe to be the first successfully treated case of isolated muscular mucormycosis occurring in a child with biphenotypic acute leukemia. The diagnosis was made promptly by tissue examination at the time of surgical debridement. The patient was also given systemic amphotericin-B therapy.      

Invasive rhino-orbital fungal sinusitis following dental manipulation

Rhinocerebral mucormycosis is a fulminant fungal infection of the nose and paranasal sinuses in immunocompromised patients. But mucormycosis following dental manipulation in immunocompetent patients leading to orbital involvement is rare. The objective of this study was to highlight the variable presentations of mucormycosis. We had two immunocompetent patients, who had undergone some dental treatment by quacks, later developed fulminant mucormycosis of the paranasal sinuses and blindness. The endoscopic sinus surgery and Amphotericin B chemotherapy resulted in a good outcome. This disease requires an aggressive approach of combined endoscopic sinus surgery and Amphotericin B to increase the chances of survival in these patients.     

Chronic rhinocerebral mucormycosis: a rare case report and review of the literature

Rhinocerebral mucormycosis is an invasive infection caused by filamentous fungi of the Mucoraceae family. The rhinocerebral form of the disease represents the most common form and has two distinct clinical entities. The common presentation consists of a rapidly progressive infection with high mortality rate, while the other presentation is that of a chronic infection with lower mortality. In the present paper we report a rare case of chronic rhinocerebral mucormycosis. An 85‐year‐old male with a 6‐month history of purulent and odorous nasal discharge, and sporadic episodes of epistaxis and anosmia, presented to the outpatient department of our clinic. Initial cultures were positive only for Pseudomonas aeruginosa. The patient was unresponsive to ciprofloxacin treatment, developing necrotic areas of the nasal septum suspicious for rhinocerebral mucormycosis. Admission to the ENT clinic followed, with histopathologic evaluation of the vomer bone confirming the diagnosis. The patient was treated with amphotericin B and was discharged 3 weeks later on oral posaconazole therapy. Chronic rhinocerebral mucormycosis may present with atypical symptoms or coinfection with another agent. A high degree of clinical suspicion is required for correct diagnosis and prompt initiation of appropriate treatment.     

Primary chronic mucormycosis

Mucormycosis generally presents as an acute fulminating infection which is seen in diabetics or in patients with underlying debilitating disease. Here we report a rare case of chronic primary mucormycosis. This case merits consideration as the presentation was chronic and indolent and also there was no underlying primary cause.     

Successful management of hepatic mucormycosis in an acute lymphoblastic leukaemia patient: a case report and review of the literature

We present a case of hepatic mucormycosis in a 9‐year‐old boy with acute lymphoblastic leukaemia. Despite long‐term use of combined liposomal amphotericin B and posaconazole therapy, the lesion persisted and could only be treated by surgical excision. After surgery, antifungal treatment was continued with posaconazole. On follow‐up, the patient had two episodes of ascending cholangitis which were responsive to intravenous antibiotics. He is doing well at the moment in remission for 2.5 years. Mucormycosis was long regarded as a fatal infection with poor prognosis. With early medical and surgical management, survival rates increase. Isolated hepatic mucormycosis is rare and only seven cases were reported in the literature up to now. We wanted to emphasise the role of early surgery in patients with hepatic mucormycosis in view of the literature.     

Overview and outcome of mucormycosis among children with cancer: Report from the Children's Cancer Hospital Egypt

Mucormycosis represents a real challenge in immunocompromised patients. This study aimed to describe the clinical characteristics, treatment outcome and infection‐related mortality in our patients at the Children's Cancer Hospital 57357, Cairo, Egypt. This is a retrospective study during the period 2007‐2017. Data analysis included demographic data, risk factors, diagnostic workup, treatment and outcome. During the study period, 45 patients developed proven mucormycosis according to EORTC/MSG criteria (2008). Ninety percentof cases were of haematological malignancies. Liposomal amphotericin B was the mainstay of treatment. Posaconazole was used as secondary prophylaxis in 35% of cases. Combination antifungal was used in three cases with progressive mucormycosis. Surgical intervention was achievable in 50% of cases. Therapy was successful in 35 patients (66%). Complications related to mucormycosis were seen in five cases with disfigurement and perforated hard palate. Chemotherapy delay with subsequent relapse of primary malignancy was reported in one case. Mucormycosis‐related mortality was 33% (15 cases). Mucormycosis is a major cause of mortality among patients with haematological malignancies. Early diagnosis of Mucormycosis infection, with rapid initiation of appropriate antifungal therapy and surgical intervention, whenever feasible, is the backbone of mucormycosis treatment.     

Prior invasive pulmonary and cerebellar mucormycosis is not a primary contraindication to perform an autologous stem cell transplatation in leukemia

Mucormycosis infections, caused by fungi of the families Rhizopus, Mucor or Absidia, are typically rapidly progressive and often fatal. We report a 27-year-old male with acute myeloid leukemia (AML) developing an invasive pulmonary-CNS mucormycosis during the neutropenic period after salvage induction chemotherapy; the infection was successfully controlled with surgery and antifungal therapy. The patient received two courses of consolidation chemotherapy and underwent autologous stem cells transplantation (ASCT) while receiving secondary antifungal systemic prophylaxis with liposomal Amphotericin B (L-AMB, Ambisome). There was no clinical, radiological or microbiological evidence of mycotic reactivation during the bone marrow transplantation (BMT) procedure.     

Rhinocerebral mucormycosis,risk factors and the type of oral manifestations in patients referred to a University Hospital in Tabriz,Iran 2007‐2017

Inadequate data are available on the global epidemiology of mucormycosis, mainly derived from the evaluation of specific population groups. Rhinocerebral mucormycosis is an invading and fatal mycosis, particularly among diabetic patients. In the present study, patients hospitalised in Imam Reza Hospital in Tabriz, from 2007 to 2017, were evaluated. The hospital information system (HIS) was used to collect the records of the patients. A total of 42 patients with a diagnosis of mucormycosis were included in the study, 40 cases (95%) of which had a diagnosis of the rhinocerebral form. Of these 40 patients, 21 (52.5%) and 19 (47.5%) were male and female, respectively. Seven cases (17.5%) of rhinocerebral mucormycosis were due to dental procedures. The most predisposing factor in the patients was diabetes with 36 (90%) cases. In our study, the role of tooth extraction in patients with uncontrolled diabetes was identified as an important factor. It may show the important role of dentists in preventing of the disease in diabetic patients.     

Mucormycotic pseudoaneurysm of the common carotid artery with tracheal involvement

Mucormycosis is an emerging and fatal fungal infection. A high index of suspicion and the knowledge of its potential manifestations are essential for early diagnosis. We describe a patient with acute lymphoblastic leukaemia (L2 subtype) who developed a neck mass following a course of induction chemotherapy. Doppler ultrasonography and angiography of the neck revealed a pseudoaneurysm of the right common carotid artery. The patient then developed haemoptysis. Surgical exploration revealed a necrotic right common carotid artery with anteromedial pseudoaneurysm and adjacent tracheal wall perforation. Local debridement and tracheal repair were performed. Nonseptate hypheal invasion (mucormycosis) was found on the microscopic examination of the excised arterial wall. A subsequent recurrence of pseudoaneurysm was treated with local surgical debridement and intravenous amphotericin B (Fungizone) administration. Although rare, clinicians should be aware of these possible presenting features of mucormycosis as early diagnosis and treatment may potentially improve the survival.     

Mucormycosis in paediatric patients: demographics, risk factors and outcome of 12 contemporary cases

Mucormycosis is associated with high morbidity and mortality and is perceived as an emerging fungal infection. However, contemporary paediatric data are limited. We present a series of paediatric cases of mucormycosis reported from Germany and Austria collected within a voluntary epidemiological survey through standardised, anonymized case report forms. Twelve cases were reported between January 2004 and December 2008 (six men; mean age: 12.6 years, range: 0.1-17 years). Mucormycosis was proven in nine, and probable in three cases. Isolates included Lichtheimia (syn. Absidia pro parte, Mycocladus) (five), Rhizopus (three) and Mucor (one) species. Infection was limited to soft tissue in three cases, the lung in two cases, and an infected thrombus in one case; rhinocerebral disease was found in three cases, and pulmonary-mediastinal, pulmonary-cerebral and soft tissue-cerebral involvement in one case each. All three patients with isolated soft tissue infection were cured, whereas seven of the remaining patients died (one patient without follow-up). The overall mortality rate was 67%. While these data cannot provide conclusive data on incidence and disease burden of mucormycosis in paediatric patients, they reflect the continuing threat of these infections to immunocompromised patients and the need for improved diagnosis and management.     

Orbital and paranasal sinusal involvements by Rhinocerebral Mucormycosis

Rhinocerebral Mucormycosis is known for its sudden affliction of the nose, paranasal sinuses and the brain. Though the sinuses are commonly involved, the clinical manifestations are often overlooked because of their vagueness. The mycotic infection affects all the paranasal sinuses with minimal involvement of the frontal sinuses. Orbital extension is very common from the ethmoidal sinuses.     

Oral mucormycosis in patients with haematologic malignancies in a bone marrow transplant unit

Mucormycosis is an aggressive and life‐threatening opportunistic fungal infection, which predominantly affects immunocompromised patients. It typically manifests in rhinocerebral, pulmonary or disseminated forms in patients with immunosuppressive conditions. Mucormycosis limited to the oral cavity is rare, and to the best of our knowledge only seven cases have previously been reported in English literature. We present five consecutive cases of oral mucormycosis in patients with leukaemia, and provide a literature review.     

Focus on invasive mucormycosis in paediatric haematology oncology patients: a series of 11 cases

Mucormycosis has emerged as an increasingly important infection in oncology centres with high mortality, especially in severely immunocompromised patients. We carried out a retrospective study of 11 children with mucormycosis treated in seven French oncology‐haematology paediatric wards during the period from 1991 to 2011. Lichtheimia corymbifera and Mucor spp. were the predominant pathogens. Treatment regimens included antifungal therapy, reversal of underlying predisposing risk factors and surgical debridement. Although mucormycosis is associated with high mortality, this infection could be cured in eight of our cases of severely immunocompromised paediatric cancer patients.