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Non-sinonasal isolated facio-orbital mucormycosis – A case report
Journal de Mycologie Me´dicale 28 (2018) 538–541
Available online at
ScienceDirect www.sciencedirect.com
Case report/Cas clinique
Non-sinonasal isolated facio-orbital mucormycosis – A case report S.K. Swain a,*, M.C. Sahu a, A. Banerjee b a b
Department of Otorhinolaryngology, IMS and SUM hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinganagar, 751003 Bhubaneswar, Odisha, India Department of Anesthesia, IMS and SUM hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinganagar, 751003 Bhubaneswar, Odisha, India
A R T I C L E I N F O
A B S T R A C T
Article history: Received 3 January 2018 Received in revised form 30 April 2018 Accepted 3 May 2018
Mucormycosis is a rare clinical entity, often affect immunocompromised patients. It is an emergency situation and has poor prognosis. Prompt diagnosis with tissue biopsy, local control of the disease by aggressive surgical debridement and appropriate systemic antifungal treatment improve the prognosis and survival of the patients. Treatment of mucormycosis needs antifungal agents such as Amphotericin B and wide surgical debridement. Early diagnosis and treatment is often needed for survival of the patients. We describe a rare case of mucormycosis affecting facio-orbital area without involving sinonnasal cavity.
C 2018 Elsevier Masson SAS. All rights reserved.
Keywords: Mucormycosis Facio-orbital Amphotericin B Surgical debridement
1. Introduction Mucormycosis is a rare and dreaded disease caused by a fungus of the order Mucorales. It has high morbidity and mortality [1]. Most commonly Rhizopus species are causative organisms for the mucormycosis. Diabetic ketoacidosis and neutropenia are common predisposing conditions. This is a life-threatening infection which has remarkable affinity for arteries [2]. This fungus often dissects internal elastic lamina from the media of the blood vessels, resulting extensive damage to the endothelium and lead to thrombosis. Mucormycosis is classified into different forms as per anatomic sites like rhino maxillary, central nervous system, cutaneous, pulmonary, disseminated and miscellaneous. The rhino-orbito-cerebral is the most common variety of mucormycosis [3]. The most common sites of mucormycosis infections are paranasal sinuses (39%), lungs (24%), skin (19%), brain (9%), gastrointestinal (7%) forms and other miscellaneous types are extremely rare [4]. The ideal treatment need correction of underlying risk factors, antifungal treatment with Amphotericin B and aggressive surgery. Here we are reporting a case nonsinonasal mucormycosis which only affecting the orbit and facial area. 2. Case report A 56-year-old lady attended the outpatient department of Otorhinolaryngology with a swelling (3 cm 3 cm) in the right * Corresponding author. Department of Otorhinolaryngology, IMS and SUM Hospital, Bhubaneswar, Odisha, India. E-mail address: [email protected] (S.K. Swain). https://doi.org/10.1016/j.mycmed.2018.05.003 C 2018 Elsevier Masson SAS. All rights reserved. 1156-5233/
facio-orbital area (Fig. 1) since 2 months. She was known case of diabetic mellitus and hypertensive patients. She was under treatment with oral hypoglycemic agents like metformin and glipizide but her blood sugar was poorly controlled (HbA1c 10.6%). Diagnostic nasal endoscopy showed normal nasal cavity and nasopharynx. Ophthalmological consultation revealed loss of the vision in the right eye. Computed tomography (CT) scan of the nose and paranasal sinuses revealed normal nasal cavity but a mass involving right orbit and adjacent facial area (Fig. 2). A small piece of biopsy sample was taken from the facio-orbital mass (Fig. 3) which showed the picture of mucormycosis with some foci of nonseptate fungal hyphae with right angled hyphal branches (Fig. 4). She had undergone radical excision of the mass along with orbital exenteration followed by parenteral liposomal Amphotericin B (5 mg/kg). Patient follow-up showed no evidence of recurrence after six months of surgery. 3. Discussion Mucormycosis is caused by saprophytic fungi of many genera related to phycomycetes (zygomycetes) and order Mucorales [5]. The fungus has great affinity towards to arteries and adheres to the arterial wall. It grows along the internal elastic lamina of blood vessels causing thrombosis, ischemia and necrosis of the surrounding tissues. Mucorales are abundantly seen in soil, decaying vegetables, animal excreta and foodstuffs. They grow rapidly in humid environment and the sporangiospores are released and spread as airborne propagules. In India, the air borne spores are more during transition from summer to rainy season as it may be ideal for fungal growth [6]. Mucormycosis is a rare clinical entity and often affect immunocompromised patients.
S.K. Swain et al. / Journal de Mycologie Me´dicale 28 (2018) 538–541
Fig. 1. A mass (3 cm 3 cm) present at the right side facio-orbit region.
The organisms causing Mucormycosis is a fungus, primarily of the genus Rhizopus or Mucor. Mucormycosis may be of rhino-orbitalcerebral and pulmonary infections and these types of the diseases may lead to emergency situation to the patients [7]. In our case, the disease is affecting only orbit and nearby facial area without involving nose and sinuses. These are rapidly growing organisms, characterized by ribbon like hyphae with no or only few septae. The genera affecting human are Cunninghamella, Absidia (Lichtheimia), Mucor, Rhizomucor, Rhizopus and one the basis of geographical distribution, Saksenaea and Apophysomyces [8]. This aggressive and highly destructive fungal infections more often seen in immunocompromised hosts like patients with
539
hematological malignancies or those taking hematopoietic stem cells transplantation. Diabetic mellitus patients with ketoacidosis and transfusion/dyserythropoetic iron overloaded patients are also important risk groups. The most common predisposing factors associated with mucormycosis are uncontrolled diabetes mellitus especially with history of ketoacidosis [9]. Neutropenia, immunosuppressive therapy, acquired immune deficiency syndrome, malnutrition, dialysis, hematological malignancy and organ transplantation are often predisposing factors. Mucormycosis universally affects immunocompromised patients. The potent T-cell depleting agents used for immunosuppression as in organ transplantation are often leads to high risk for causing mucormycosis. Although mucormycosis is ubiquitous and rapidly spread, it seldom affects an immunologically competent patient. Therefore mucormycosis occurs among patients with serious underlying conditions like diabetic mellitus, leukemia, organ transplantations, acquired immunodeficiency syndrome, severe burn and immunosuppressive medications. Around 70 to 80% of mucormycosis patients have diabetes mellitus [10]. Radiological imaging will not establish but suggest the diagnosis of mucormycosis. CT or MRI are helpful for Facial and cerebral involvement of mucormycosis and determine loco-regional extension towards the orbit and brain and also identify the cerebral thrombosis [11]. Cavernous sinus involvement is best assessed by MRI [12]. Difficulties in diagnosis and followed by antifungal treatment with resistance to many commonly prescribed antifungal agents leads to high mortality in certain group of patients [13]. There is no such definitive diagnosis for the mucormycosis except histopathological confirmation by seeing the non-septate hyphae of the affected tissue [3]. Necrosis of the surrounding tissue prevents antifungal drugs like Amphotericin B to enter the site of infection [14]. Early debridement of the affected tissue gives better prognosis for successful treatment in mucormycosis [15]. Liposomal Amphotericin B treatment has better tissue penetration and considered as first line treatment [4]. However, monotherapy like surgery alone or Amphotericin B alone has limited efficacy whereas the combination therapy with
Fig. 2. CT scan (Fig 2a coronal cut and Fig 2b axial cut) picture showing mass at right facio-orbit area without involving nose and paranasal sinuses. a: coronal cut; b: axial cut.
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S.K. Swain et al. / Journal de Mycologie Me´dicale 28 (2018) 538–541
period. Liposomal form better concentrates in macrophages including deep infected tissue with higher CNS penetration [19]. Posaconazole a new type of triazole is useful in failure cases with better efficacy in vitro and in vivo with good tolerance and very fewer side effects. Isavuconazole, a new type of extended spectrum of triazole which act against molds, yeast and dimorphic fungi and also approved for the treatment of invasive aspergillosis and Mucormycosis [20]. Total duration of antifungal drug treatment in mucormycosis varies with evolution, but at least advised for 12 weeks [9]. For prevention of the mucormycosis, the risk factors should be properly addressed. The mucormycosis is often seen in patients of uncontrolled diabetes mellitus which need a very good control for preventing this dreaded disease. 4. Conclusion
Fig. 3. Facio-orbital mass opened for taking biopsy.
liposomal Amphotericin B or Posaconazole and surgical debridement are recommended treatment [16]. Amphotericin B has potential toxicity on renal function and so its dose should be individually adjusted between 0.5 mg/kg/day to 1.0 mg/kg/day on the basis of body weight and renal functions of the patients. The total cumulative dosage of Amphotericin B is 2 to 4 g often advocated to the adult patient. Hyperbaric oxygen therapy and certain cytokines like GM-CSF and interferon gamma act as adjuvant treatment and these are under assessment. Hyperbaric oxygen therapy has fungi static effect and helps revascularization of the necrotic or ischemic tissue [17]. The survival rate of mucormycosis patients has been improved by new antifungal agents and combination therapy. Despite advancement in treatment modality and care, the overall mortality rate of mucormycosis patients remains high due to delay in diagnosis. A delayed diagnosis of mucormycosis of more than five days is associated with increased chance of death [18]. For successful outcome, correction of predisposing factors, timely diagnosis, surgical debridement and systemic anti fungal are needed in combination. The systemic Amphotericin B has its own side effects which need vigilant monitoring. Amphotericin B in classic desoxycholate form in the dose of 1–1.5 mg/kg/day or more preferably in liposomal form of Amphotericin B is highly useful for mucormycosis. The liposomal form has less nephrotoxicity and also allows higher doses in 5 to 15 mg/kg/day for prolonged
Fig. 4. Histopathology microphotograph showing broad non-septate hyphae (yellow arrow mark showed) with 908 branching (Eosin stain and 400 magnification).
Mucormycosis is a rare and dreaded fungal infection. It rapidly progressive disease with fatal outcome unless diagnosed early and treated promptly. Delayed diagnosis and treatment may lead to fatal complications and death. Treatment includes both medical and surgical. It needs an early and adequate treatment with liposomal Amphotericin B; surgical resection and control of risk factors can improve the prognosis. Prognosis depends on multiple factors and need early initiation of treatment. A multi-disciplinary approach often needed and consists of otorhinolaryngologists, ophthalmologists, neurologists and dentists for successful management of patients with mucormycosis. A rare location for mucormycosis like isolated facio-orbital area may be a site which should be kept in mind. Disclosure of interest The authors declare that they have no competing interest. References [1] Katragkou A, Walsh TJ, Roilides E. Why is mucormycosis more difficult to cure than more common mycoses? Clin Microbiol Infect 2014;20:74–81. [2] Bavikar P, Mehta V. Rhino-orbital-cerebral mucormycosis: a fatal complication of uncontrolled diabetes mellitus. Cureus 2017;9:e1841. [3] Viterbo S, Fasolis M, Garzino-Demo P, et al. Management and outcomes of three cases of rhino-cerebral mucormycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:69–74. [4] Chamilos G, Lewis RE, Kontoyiannis DP. Delaying amphotericin B-based frontline therapy significantly increases mortality among patients with hematologic malignancy who have zygomycosis. Clin Infect Dis 2008;47:503–9. [5] Pinto ME, Manrique HA, Guevara X, et al. Hyperglycemic hyperosmolar state and rhino-orbital mucormycosis. Diabetes Res Clin Pract 2011;91:37–9. [6] Babu SV, Venkatesh U, Prasannaraj T, Shivaprakash KV, Prathima S. Sinonasal mucormucosis: a series of seven cases. Clin Rhinol 2012;5:25–7. [7] Kauffman CA, Malani AN. Zygomycosis: an emerging fungal infection with new options for management. Curr Infect Dis Rep 2007;9:435. [8] De Hoog GS, Guarro J, Gene J, Figueras MJ. Atlas of clinical fungi. Utrecht, The Netherlands: Centraalbureau voor Schimmelcultures; 2011. [9] Wali U, Balkhair A, Al-Mujaini A. Cerebro-rhino orbital mucormycosis: an update. J Infect Public Health 2012;5:116–26. [10] Hsiao YC, Tsan KW, Wang TY. Rhinocerebral mucormycosis in diabetes: a case report. J Intern Med Taiwan 2002;13:160–4. [11] Lmekki S, Zaki Z, El Alami MN. Mucormycose rhinoce´re´brale. Med Mal Infect 2012;42:171–3. [12] Mohammadi R, Nazeri M, Sayedayn SM, Ehteram H. A successful treatment of rhinocerebral mucormycosis due to Rhizopus oryzae. J Res Med Sci 2014;19:72–4. [13] Skiada A, Pagano L, Groll A, et al. Zygomycosis in Europe: analysis of 230 cases accrued by the registry of the European Confederation of Medical Mycology (ECMM) Working Group on Zygomycosis between 2005 and 2007. Clin Microbiol Infect 2011;17:1859–67. [14] Lanternier F, Sun HY, Ribaud P, et al. Mucormycosis in organ and stem cell transplant recipients. Clin Infect Dis 2012;54:1629–36. [15] Lai MC, Zhang W, Yang Z, et al. First case report of isolated penile mucormycosis in a liver transplantation recipient. Int J Infect Dis 2014;29:208–10. [16] Chakrabarti A, Das A, Sharma A, et al. Ten years’ experience in zygomycosis at a tertiary care centre in India. J Infect 2001;42:261–6.
S.K. Swain et al. / Journal de Mycologie Me´dicale 28 (2018) 538–541 [17] Almannai M, Imran H, Estrada B, Siddiqui AH. Successful treatment of rhinoorbital mucormycosis with posaconazole and hyperbaric oxygen therapy. Pediatr Hemato Oncol 2013;30:184–6. [18] Farojov R, Aydın O, Yılmaz C, Iakobadze Z, Do ganay L, Camlı D, et al. Rhinoorbita-maxillary mucormycosis after liver transplantation: a case report. Transplant Proc 2016;48:3210–3.
541
[19] Kim JG, Park HJ, Park JH, et al. Importance of immediate surgical intervention and antifungal treatment for rhinocerebral mucormycosis: a case report. J Korean Assoc Oral Maxillofac Surg 2013;39:246–50. [20] Graves B, Orla Morrissey C, Wei A, Coutsouvelis J, Ellis S, Pham A, et al. Isavuconazole as salvage therapy for mucormycosis. Med Mycol Case Rep 2016;11:36–9.
Available online at
ScienceDirect www.sciencedirect.com
Case report/Cas clinique
Non-sinonasal isolated facio-orbital mucormycosis – A case report S.K. Swain a,*, M.C. Sahu a, A. Banerjee b a b
Department of Otorhinolaryngology, IMS and SUM hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinganagar, 751003 Bhubaneswar, Odisha, India Department of Anesthesia, IMS and SUM hospital, Siksha ‘‘O’’ Anusandhan University, K8, Kalinganagar, 751003 Bhubaneswar, Odisha, India
A R T I C L E I N F O
A B S T R A C T
Article history: Received 3 January 2018 Received in revised form 30 April 2018 Accepted 3 May 2018
Mucormycosis is a rare clinical entity, often affect immunocompromised patients. It is an emergency situation and has poor prognosis. Prompt diagnosis with tissue biopsy, local control of the disease by aggressive surgical debridement and appropriate systemic antifungal treatment improve the prognosis and survival of the patients. Treatment of mucormycosis needs antifungal agents such as Amphotericin B and wide surgical debridement. Early diagnosis and treatment is often needed for survival of the patients. We describe a rare case of mucormycosis affecting facio-orbital area without involving sinonnasal cavity.
C 2018 Elsevier Masson SAS. All rights reserved.
Keywords: Mucormycosis Facio-orbital Amphotericin B Surgical debridement
1. Introduction Mucormycosis is a rare and dreaded disease caused by a fungus of the order Mucorales. It has high morbidity and mortality [1]. Most commonly Rhizopus species are causative organisms for the mucormycosis. Diabetic ketoacidosis and neutropenia are common predisposing conditions. This is a life-threatening infection which has remarkable affinity for arteries [2]. This fungus often dissects internal elastic lamina from the media of the blood vessels, resulting extensive damage to the endothelium and lead to thrombosis. Mucormycosis is classified into different forms as per anatomic sites like rhino maxillary, central nervous system, cutaneous, pulmonary, disseminated and miscellaneous. The rhino-orbito-cerebral is the most common variety of mucormycosis [3]. The most common sites of mucormycosis infections are paranasal sinuses (39%), lungs (24%), skin (19%), brain (9%), gastrointestinal (7%) forms and other miscellaneous types are extremely rare [4]. The ideal treatment need correction of underlying risk factors, antifungal treatment with Amphotericin B and aggressive surgery. Here we are reporting a case nonsinonasal mucormycosis which only affecting the orbit and facial area. 2. Case report A 56-year-old lady attended the outpatient department of Otorhinolaryngology with a swelling (3 cm 3 cm) in the right * Corresponding author. Department of Otorhinolaryngology, IMS and SUM Hospital, Bhubaneswar, Odisha, India. E-mail address: [email protected] (S.K. Swain). https://doi.org/10.1016/j.mycmed.2018.05.003 C 2018 Elsevier Masson SAS. All rights reserved. 1156-5233/
facio-orbital area (Fig. 1) since 2 months. She was known case of diabetic mellitus and hypertensive patients. She was under treatment with oral hypoglycemic agents like metformin and glipizide but her blood sugar was poorly controlled (HbA1c 10.6%). Diagnostic nasal endoscopy showed normal nasal cavity and nasopharynx. Ophthalmological consultation revealed loss of the vision in the right eye. Computed tomography (CT) scan of the nose and paranasal sinuses revealed normal nasal cavity but a mass involving right orbit and adjacent facial area (Fig. 2). A small piece of biopsy sample was taken from the facio-orbital mass (Fig. 3) which showed the picture of mucormycosis with some foci of nonseptate fungal hyphae with right angled hyphal branches (Fig. 4). She had undergone radical excision of the mass along with orbital exenteration followed by parenteral liposomal Amphotericin B (5 mg/kg). Patient follow-up showed no evidence of recurrence after six months of surgery. 3. Discussion Mucormycosis is caused by saprophytic fungi of many genera related to phycomycetes (zygomycetes) and order Mucorales [5]. The fungus has great affinity towards to arteries and adheres to the arterial wall. It grows along the internal elastic lamina of blood vessels causing thrombosis, ischemia and necrosis of the surrounding tissues. Mucorales are abundantly seen in soil, decaying vegetables, animal excreta and foodstuffs. They grow rapidly in humid environment and the sporangiospores are released and spread as airborne propagules. In India, the air borne spores are more during transition from summer to rainy season as it may be ideal for fungal growth [6]. Mucormycosis is a rare clinical entity and often affect immunocompromised patients.
S.K. Swain et al. / Journal de Mycologie Me´dicale 28 (2018) 538–541
Fig. 1. A mass (3 cm 3 cm) present at the right side facio-orbit region.
The organisms causing Mucormycosis is a fungus, primarily of the genus Rhizopus or Mucor. Mucormycosis may be of rhino-orbitalcerebral and pulmonary infections and these types of the diseases may lead to emergency situation to the patients [7]. In our case, the disease is affecting only orbit and nearby facial area without involving nose and sinuses. These are rapidly growing organisms, characterized by ribbon like hyphae with no or only few septae. The genera affecting human are Cunninghamella, Absidia (Lichtheimia), Mucor, Rhizomucor, Rhizopus and one the basis of geographical distribution, Saksenaea and Apophysomyces [8]. This aggressive and highly destructive fungal infections more often seen in immunocompromised hosts like patients with
539
hematological malignancies or those taking hematopoietic stem cells transplantation. Diabetic mellitus patients with ketoacidosis and transfusion/dyserythropoetic iron overloaded patients are also important risk groups. The most common predisposing factors associated with mucormycosis are uncontrolled diabetes mellitus especially with history of ketoacidosis [9]. Neutropenia, immunosuppressive therapy, acquired immune deficiency syndrome, malnutrition, dialysis, hematological malignancy and organ transplantation are often predisposing factors. Mucormycosis universally affects immunocompromised patients. The potent T-cell depleting agents used for immunosuppression as in organ transplantation are often leads to high risk for causing mucormycosis. Although mucormycosis is ubiquitous and rapidly spread, it seldom affects an immunologically competent patient. Therefore mucormycosis occurs among patients with serious underlying conditions like diabetic mellitus, leukemia, organ transplantations, acquired immunodeficiency syndrome, severe burn and immunosuppressive medications. Around 70 to 80% of mucormycosis patients have diabetes mellitus [10]. Radiological imaging will not establish but suggest the diagnosis of mucormycosis. CT or MRI are helpful for Facial and cerebral involvement of mucormycosis and determine loco-regional extension towards the orbit and brain and also identify the cerebral thrombosis [11]. Cavernous sinus involvement is best assessed by MRI [12]. Difficulties in diagnosis and followed by antifungal treatment with resistance to many commonly prescribed antifungal agents leads to high mortality in certain group of patients [13]. There is no such definitive diagnosis for the mucormycosis except histopathological confirmation by seeing the non-septate hyphae of the affected tissue [3]. Necrosis of the surrounding tissue prevents antifungal drugs like Amphotericin B to enter the site of infection [14]. Early debridement of the affected tissue gives better prognosis for successful treatment in mucormycosis [15]. Liposomal Amphotericin B treatment has better tissue penetration and considered as first line treatment [4]. However, monotherapy like surgery alone or Amphotericin B alone has limited efficacy whereas the combination therapy with
Fig. 2. CT scan (Fig 2a coronal cut and Fig 2b axial cut) picture showing mass at right facio-orbit area without involving nose and paranasal sinuses. a: coronal cut; b: axial cut.
540
S.K. Swain et al. / Journal de Mycologie Me´dicale 28 (2018) 538–541
period. Liposomal form better concentrates in macrophages including deep infected tissue with higher CNS penetration [19]. Posaconazole a new type of triazole is useful in failure cases with better efficacy in vitro and in vivo with good tolerance and very fewer side effects. Isavuconazole, a new type of extended spectrum of triazole which act against molds, yeast and dimorphic fungi and also approved for the treatment of invasive aspergillosis and Mucormycosis [20]. Total duration of antifungal drug treatment in mucormycosis varies with evolution, but at least advised for 12 weeks [9]. For prevention of the mucormycosis, the risk factors should be properly addressed. The mucormycosis is often seen in patients of uncontrolled diabetes mellitus which need a very good control for preventing this dreaded disease. 4. Conclusion
Fig. 3. Facio-orbital mass opened for taking biopsy.
liposomal Amphotericin B or Posaconazole and surgical debridement are recommended treatment [16]. Amphotericin B has potential toxicity on renal function and so its dose should be individually adjusted between 0.5 mg/kg/day to 1.0 mg/kg/day on the basis of body weight and renal functions of the patients. The total cumulative dosage of Amphotericin B is 2 to 4 g often advocated to the adult patient. Hyperbaric oxygen therapy and certain cytokines like GM-CSF and interferon gamma act as adjuvant treatment and these are under assessment. Hyperbaric oxygen therapy has fungi static effect and helps revascularization of the necrotic or ischemic tissue [17]. The survival rate of mucormycosis patients has been improved by new antifungal agents and combination therapy. Despite advancement in treatment modality and care, the overall mortality rate of mucormycosis patients remains high due to delay in diagnosis. A delayed diagnosis of mucormycosis of more than five days is associated with increased chance of death [18]. For successful outcome, correction of predisposing factors, timely diagnosis, surgical debridement and systemic anti fungal are needed in combination. The systemic Amphotericin B has its own side effects which need vigilant monitoring. Amphotericin B in classic desoxycholate form in the dose of 1–1.5 mg/kg/day or more preferably in liposomal form of Amphotericin B is highly useful for mucormycosis. The liposomal form has less nephrotoxicity and also allows higher doses in 5 to 15 mg/kg/day for prolonged
Fig. 4. Histopathology microphotograph showing broad non-septate hyphae (yellow arrow mark showed) with 908 branching (Eosin stain and 400 magnification).
Mucormycosis is a rare and dreaded fungal infection. It rapidly progressive disease with fatal outcome unless diagnosed early and treated promptly. Delayed diagnosis and treatment may lead to fatal complications and death. Treatment includes both medical and surgical. It needs an early and adequate treatment with liposomal Amphotericin B; surgical resection and control of risk factors can improve the prognosis. Prognosis depends on multiple factors and need early initiation of treatment. A multi-disciplinary approach often needed and consists of otorhinolaryngologists, ophthalmologists, neurologists and dentists for successful management of patients with mucormycosis. A rare location for mucormycosis like isolated facio-orbital area may be a site which should be kept in mind. Disclosure of interest The authors declare that they have no competing interest. References [1] Katragkou A, Walsh TJ, Roilides E. Why is mucormycosis more difficult to cure than more common mycoses? Clin Microbiol Infect 2014;20:74–81. [2] Bavikar P, Mehta V. Rhino-orbital-cerebral mucormycosis: a fatal complication of uncontrolled diabetes mellitus. Cureus 2017;9:e1841. [3] Viterbo S, Fasolis M, Garzino-Demo P, et al. Management and outcomes of three cases of rhino-cerebral mucormycosis. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2011;112:69–74. [4] Chamilos G, Lewis RE, Kontoyiannis DP. Delaying amphotericin B-based frontline therapy significantly increases mortality among patients with hematologic malignancy who have zygomycosis. Clin Infect Dis 2008;47:503–9. [5] Pinto ME, Manrique HA, Guevara X, et al. Hyperglycemic hyperosmolar state and rhino-orbital mucormycosis. Diabetes Res Clin Pract 2011;91:37–9. [6] Babu SV, Venkatesh U, Prasannaraj T, Shivaprakash KV, Prathima S. Sinonasal mucormucosis: a series of seven cases. Clin Rhinol 2012;5:25–7. [7] Kauffman CA, Malani AN. Zygomycosis: an emerging fungal infection with new options for management. Curr Infect Dis Rep 2007;9:435. [8] De Hoog GS, Guarro J, Gene J, Figueras MJ. Atlas of clinical fungi. Utrecht, The Netherlands: Centraalbureau voor Schimmelcultures; 2011. [9] Wali U, Balkhair A, Al-Mujaini A. Cerebro-rhino orbital mucormycosis: an update. J Infect Public Health 2012;5:116–26. [10] Hsiao YC, Tsan KW, Wang TY. Rhinocerebral mucormycosis in diabetes: a case report. J Intern Med Taiwan 2002;13:160–4. [11] Lmekki S, Zaki Z, El Alami MN. Mucormycose rhinoce´re´brale. Med Mal Infect 2012;42:171–3. [12] Mohammadi R, Nazeri M, Sayedayn SM, Ehteram H. A successful treatment of rhinocerebral mucormycosis due to Rhizopus oryzae. J Res Med Sci 2014;19:72–4. [13] Skiada A, Pagano L, Groll A, et al. Zygomycosis in Europe: analysis of 230 cases accrued by the registry of the European Confederation of Medical Mycology (ECMM) Working Group on Zygomycosis between 2005 and 2007. Clin Microbiol Infect 2011;17:1859–67. [14] Lanternier F, Sun HY, Ribaud P, et al. Mucormycosis in organ and stem cell transplant recipients. Clin Infect Dis 2012;54:1629–36. [15] Lai MC, Zhang W, Yang Z, et al. First case report of isolated penile mucormycosis in a liver transplantation recipient. Int J Infect Dis 2014;29:208–10. [16] Chakrabarti A, Das A, Sharma A, et al. Ten years’ experience in zygomycosis at a tertiary care centre in India. J Infect 2001;42:261–6.
S.K. Swain et al. / Journal de Mycologie Me´dicale 28 (2018) 538–541 [17] Almannai M, Imran H, Estrada B, Siddiqui AH. Successful treatment of rhinoorbital mucormycosis with posaconazole and hyperbaric oxygen therapy. Pediatr Hemato Oncol 2013;30:184–6. [18] Farojov R, Aydın O, Yılmaz C, Iakobadze Z, Do ganay L, Camlı D, et al. Rhinoorbita-maxillary mucormycosis after liver transplantation: a case report. Transplant Proc 2016;48:3210–3.
541
[19] Kim JG, Park HJ, Park JH, et al. Importance of immediate surgical intervention and antifungal treatment for rhinocerebral mucormycosis: a case report. J Korean Assoc Oral Maxillofac Surg 2013;39:246–50. [20] Graves B, Orla Morrissey C, Wei A, Coutsouvelis J, Ellis S, Pham A, et al. Isavuconazole as salvage therapy for mucormycosis. Med Mycol Case Rep 2016;11:36–9.