|Year : 2014 | Volume
| Issue : 4 | Page : 486-488
Rhino-oculo-cerebral aspergillus and mucor co-infections in an immunocompromised patient with type 2 diabetes mellitus
Kalidas Rit1, Rajdeep Saha2, Rupali Dey1, Gautam Barik3
1 Department of Microbiology, Institute of Post-Graduate Medical Education and Research, 244 AJC Bose Road, Kamarhati, Kolkata, India
2 Department of Microbiology, National Medical College, Kamarhati, Kolkata, India
3 Department of Microbiology, College of Medicine and Sagar Dutta Hospital, Kamarhati, Kolkata, India
|Date of Web Publication||25-Jun-2014|
70B T.C. Mukherjee Street, PO: Rishra, Dist: Hooghly - 712 248, West Bengal
Source of Support: None, Conflict of Interest: None
Mucormycosis are pathogenic moulds of the mucorales species usually occurring in immunocompromised patients or in patients with uncontrolled diabetes mellitus. Aspergillosis is the clinical condition caused by Aspergillus species and may cause an invasive disease with high case fatality rate, especially in immunosuppressed patients. A 46-year-old male patient with Type 2 diabetes mellitus with underlying malignancy presented with proptosis of left eye. Combined infections of Mucor and Aspergillus were diagnosed by means of computed tomography (CT) scan and biopsy. Treatment with Amphotericin B and Voriconazole was started, the patient died within 3 months, from multi-organ failure.
Keywords: Aspergillosis, diabetes mellitus, immunocompromised, mucormycosis
|How to cite this article:|
Rit K, Saha R, Dey R, Barik G. Rhino-oculo-cerebral aspergillus and mucor co-infections in an immunocompromised patient with type 2 diabetes mellitus. Med J DY Patil Univ 2014;7:486-8
|How to cite this URL:|
Rit K, Saha R, Dey R, Barik G. Rhino-oculo-cerebral aspergillus and mucor co-infections in an immunocompromised patient with type 2 diabetes mellitus. Med J DY Patil Univ [serial online] 2014 [cited 2020 Jan 29];7:486-8. Available from: http://www.mjdrdypu.org/text.asp?2014/7/4/486/135278
| Introduction|| |
Aspergillosis is the clinical condition caused by the Aspergillus species, most often A. fumigatus. Mucormycosis is an opportunistic, aggressive infection caused by organisms belonging to the class of Phycomycetes.  They occur almost exclusively in immunocompromised patients like uncontrolled diabetics, those on chemotherapy and steroids.  Despite advances in diagnosis and management, mortality rates are still high. Reports of combined Mucor and Aspergilus infections limited to rhino-oculo-cerebral (ROC) regions are very rare. , Here we report a case of rhino-oculo-cerebral aspergillosis and mucormycosis in an uncontrolled Type 2 diabetic patient with underlying malignancy.
| Case Report|| |
A 46-year-old man presented in the ophthalmology outpatients' department (OPD) with proptosis of the left eye for the last three months along with ptosis and diminution of vision for last one month. The patient was poorly built and debilitated, confused, and disoriented. There was presence of a firm swelling over the left maxillary region, and bloody nasal discharge [Figure 1]. The patient was a known diabetic, hypertensive and there was a history of epistaxis six months back which persisted for one month. There was a history of nasal blockage and polypectomy operation done at a sub-divisional hospital but nasal blockage persisted. Radiological examination revealed opacification of the left maxillary sinus and destruction of bony walls. At the time of presentation random blood sugar level was 290 mg/dl and BP was 160/96 mm Hg. After proper control of blood sugar and hypertension, exploration of maxillary sinus was done by modified Caldwell-Luc operation and the tissue was collected. Histopathological examination revealed presence of squamous cell carcinoma. Patient was subsequently treated by combination of radiotherapy and surgery. After a one-month symptom-free period the patient presented with throbbing left-sided headache, pain and tenderness over the left maxillary sinus area and blocked left nostril. CT scan showed a large heterogeneous diffuse soft-tissue mass involving both the maxillary antrums and part of the nasopharynx with left orbital proptosis [Figure 2]. Sellar and parasellar regions were normal. Examination revealed complete opthalmoplegia and inflamed edematous anterior chamber of right eye. X-ray of the adjoining area showed soft opacity with erosion of superior, middle and inferior margin of the left maxillary antrum. Blood test results revealed leucocytic count 16,400/cmm with C-reactive protein 85 mg/l and post-prandial blood sugar level of 463 mg/dl. Blood urea and creatinine levels were 57 mg/dl and 3.6 mg/dl respectively. Urine dipstick analysis identified the presence of ketones and blood gas analysis revealed metabolic acidosis. Sinoscopy confirmed severely inflamed maxillary sinus and material collected by sinoscopy procedure revealed septate hyphae with dichotomous branching suggestive of Aspergillus species. Further culture of collected material identified the species as Aspergillus flavus. Material from the left wall of the middle turbinate of the left nasal cavity also revealed growth of Aspergillus flavus [Figure 3]. Intravenous infusion of Voriconazole was started and continued for seven days without much improvement of clinical condition. After 15 days there was occurrence of bloody nasal discharge. Histopathological staining showed broad nonseptate hyphae of Mucor [Figure 4]. Intravenous infusion of Amphotericin B was started. He tolerated Amphotericin B very poorly and ultimately after 17 days of therapy the patient expired.
|Figure 1: Ptosis of left eye with bloody nasal discharge from left nostril|
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|Figure 2: CT scan showing a heterogeneous diffuse soft-tissue mass involving both maxillary antrums|
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| Discussion|| |
Here we have presented a rare combined infection of mucormycosis and aspergillosis in an immunocompromised patient with uncontrolled diabetes mellitus. The diagnosis was confirmed when causative agents were disclosed by both histopathology and culture. Mucormycosis is an opportunistic suppurative infection which spreads by direct as well as hematogenous dissemination and is associated with vascular invasion resulting in thrombosis, embolism, and infarction.  The predisposing factors for mucormycosis are uncontrolled diabetes mellitus (particularly in patients having ketoacidosis), underlying malignancies, renal failure, organ transplant, long-term immunosuppressive therapy, and cirrhosis.  Our patient had uncontrolled diabetes with ketoacidosis which is a well-known predisposing factor for mucormycosis. Although several genera are associated with this disease the most common forms are Rhizopus, Rhizomucor and Absidia. Rhizopus is the predominant pathogen accounting for 90% cases of rhinocerebral mucormycosis.  Uncontrolled diabetes mellitus can alter the normal immunological response of patients to infections. Such patients have decreased granulocytic phagocytic activity with altered polymorphonuclear leucocyte response. Reports have suggested that the ability of the serum of immunocompromised patients to inhibit Rhizopus in vitro is reduced, which makes them suitable hosts for opportunistic fungal infections. Furthermore, the acidosis and hyperglycemia provide an excellent environment for the fungus to grow. 
Aspergillosis is a well-known fungal infection most commonly caused by A. fumigatus. Invasive aspergillosis of the ROC region may spread to the adjoining regions appearing as yellow or black necrotic ulcers.  If diagnosis and therapeutic interventions are delayed it may result in massive tissue destruction and, eventually death. Aspergillosis most commonly affects patients with poorly controlled diabetes with immunodeficiency, like that of our case.
This case report of combined aspergillosis and mucormycosis of the ROC region is of importance because of its rarity. Mairano et al., presented a case of combined infection in a patient affected by Castleman disease. 
The principles of management are complete treatment of underlying medical disease, correction of hypoxia, acidosis, hyperglycemia, and electrolyte abnormalities.  The mainstay of treatment is systemic Amphotericin B, preferably liposomal preparation.  In this case, the patient was treated with IV Amphotericin B and Voriconazole, but because of poor general health and compromised renal function, patient tolerance to both the drugs was poor and ultimately he succumbed to his disease.
| References|| |
|1.||Tryfon S, Stanopoulos I, Kakavelas E, Nikolaidou A, Kioumis I. Rhino cerebral mucormycosis in a patient with latent diabetes mellitus: A case report. J Oral Maxillofac Surg 2002;60:328-30. |
|2.||Yohai RA, Bullock JD, Aziz AA, Markert RJ. Survival factors in rhino-orbital-cerebral mucormycosis. Surv Ophthalmol 1994;39:3-22. |
|3.||Maiorano E, Favia G, Capodiferro S, Montagna MT, Lo Muzio L. Combined mucormycosis and aspergillosis of the oro-sinonasal region in a patient affected by Castleman disease. Virchows Arch 2005;446:28-33. |
|4.||Alfano C, Chiummariello S, Dessy LA, Bistoni G, Scuderi N. Combined Mucormycosis and Aspergillosis of the Rhinocerebral Region. In Vivo 2006;20:311-15. |
|5.||Khan AR, Khan MU, Ullah S, Ullah H, Shah J. Rhinocerebral mucormycosis. J Coll Physicians Surg Pak 2002;12:639-41. |
|6.||Romett JL, Newman RK. Aspergillosis of the nose and paranasal sinuses. Laryngoscope 1982;92:764-6. |
|7.||Spellberg B, Edwards J Jr., Ibrahim A. Novel perspectives on mucormycosis: Pathophysiology. presentation, and management. Clin Microbiol Rev 2005;18:556-69. |
|8.||Armstrong D. Treatment of opportunistic fungal infections. Clin Inf Dis 1993;16:1-9. |
[Figure 1], [Figure 2], [Figure 3], [Figure 4]