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https://doi.org/10.15644/asc53/3/9

Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia

Vanja Vučićević Boras ; Department of Oral Medicine School of Dental Medicine University of Zagreb and Department of Oral Diseases University Hospital Centre Zagreb, Zagreb, Croatia
Martin Jurlina ; Department of Otolaryngology, Head and Neck Surgery University Hospital Centre Zagreb, Zagreb, Croatia
Vlaho Brailo ; Department of Oral Medicine School of Dental Medicine University of Zagreb and Department of Oral Diseases University Hospital Centre Zagreb, Zagreb, Croatia
Katarina Đurić Vuković ; Department of Otolaryngology, Head and Neck Surgery University Hospital Centre Zagreb, Zagreb, Croatia
Pavle Rončević ; Department of Haematology University Hospital Centre Zagreb, Zagreb, Croatia
Sandra Bašić Kinda ; Department of Haematology University Hospital Centre Zagreb, Zagreb, Croatia
Danica Vidović Juras ; Department of Oral Medicine School of Dental Medicine University of Zagreb and Department of Oral Diseases University Hospital Centre Zagreb, Zagreb, Croatia
Dragana Gabrić ; Department of Oral Surgery School of Dental Medicine University of Zagreb and Department of Oral Surgery University Hospital Centre Zagreb, Zagreb, Croatia

Puni tekst: engleski, pdf (228 KB) str. 274-277 preuzimanja: 1.841* citiraj
APA 6th Edition
Vučićević Boras, V., Jurlina, M., Brailo, V., Đurić Vuković, K., Rončević, P., Bašić Kinda, S., ... Gabrić, D. (2019). Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia. Acta stomatologica Croatica, 53 (3), 274-277. https://doi.org/10.15644/asc53/3/9
MLA 8th Edition
Vučićević Boras, Vanja, et al. "Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia." Acta stomatologica Croatica, vol. 53, br. 3, 2019, str. 274-277. https://doi.org/10.15644/asc53/3/9. Citirano 23.09.2020.
Chicago 17th Edition
Vučićević Boras, Vanja, Martin Jurlina, Vlaho Brailo, Katarina Đurić Vuković, Pavle Rončević, Sandra Bašić Kinda, Danica Vidović Juras i Dragana Gabrić. "Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia." Acta stomatologica Croatica 53, br. 3 (2019): 274-277. https://doi.org/10.15644/asc53/3/9
Harvard
Vučićević Boras, V., et al. (2019). 'Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia', Acta stomatologica Croatica, 53(3), str. 274-277. https://doi.org/10.15644/asc53/3/9
Vancouver
Vučićević Boras V, Jurlina M, Brailo V, Đurić Vuković K, Rončević P, Bašić Kinda S i sur. Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia. Acta stomatologica Croatica [Internet]. 2019 [pristupljeno 23.09.2020.];53(3):274-277. https://doi.org/10.15644/asc53/3/9
IEEE
V. Vučićević Boras, et al., "Oral Mucormycosis and Aspergillosis in the Patient with Acute Leukemia", Acta stomatologica Croatica, vol.53, br. 3, str. 274-277, 2019. [Online]. https://doi.org/10.15644/asc53/3/9
Puni tekst: hrvatski, pdf (228 KB) str. 274-277 preuzimanja: 87* citiraj
APA 6th Edition
Vučićević Boras, V., Jurlina, M., Brailo, V., Đurić Vuković, K., Rončević, P., Bašić Kinda, S., ... Gabrić, D. (2019). Oralna mukormikoza i aspergiloza kod bolesnika s akutnom leukemijom. Acta stomatologica Croatica, 53 (3), 274-277. https://doi.org/10.15644/asc53/3/9
MLA 8th Edition
Vučićević Boras, Vanja, et al. "Oralna mukormikoza i aspergiloza kod bolesnika s akutnom leukemijom." Acta stomatologica Croatica, vol. 53, br. 3, 2019, str. 274-277. https://doi.org/10.15644/asc53/3/9. Citirano 23.09.2020.
Chicago 17th Edition
Vučićević Boras, Vanja, Martin Jurlina, Vlaho Brailo, Katarina Đurić Vuković, Pavle Rončević, Sandra Bašić Kinda, Danica Vidović Juras i Dragana Gabrić. "Oralna mukormikoza i aspergiloza kod bolesnika s akutnom leukemijom." Acta stomatologica Croatica 53, br. 3 (2019): 274-277. https://doi.org/10.15644/asc53/3/9
Harvard
Vučićević Boras, V., et al. (2019). 'Oralna mukormikoza i aspergiloza kod bolesnika s akutnom leukemijom', Acta stomatologica Croatica, 53(3), str. 274-277. https://doi.org/10.15644/asc53/3/9
Vancouver
Vučićević Boras V, Jurlina M, Brailo V, Đurić Vuković K, Rončević P, Bašić Kinda S i sur. Oralna mukormikoza i aspergiloza kod bolesnika s akutnom leukemijom. Acta stomatologica Croatica [Internet]. 2019 [pristupljeno 23.09.2020.];53(3):274-277. https://doi.org/10.15644/asc53/3/9
IEEE
V. Vučićević Boras, et al., "Oralna mukormikoza i aspergiloza kod bolesnika s akutnom leukemijom", Acta stomatologica Croatica, vol.53, br. 3, str. 274-277, 2019. [Online]. https://doi.org/10.15644/asc53/3/9

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Sažetak
A 54-year-old male patient with acute lymphoblastic leukemia was referred to the Department of Oral Medicine. He had a primary refractory disease and was treated according to HOVON71 and HAM protocol. Sixteen days after the start of the HAM protocol the patient developed palatal dark red/brownish lesion and maxillary vestibular exophytic lesion. Biopsy specimens from oral lesions were taken and microbiologic evaluation confirmed the presence of Aspergillus fumigatus and Rhizopus genus. The treatment of the patient consisted of the inferior maxillectomy and intravenous posaconazole and amphotericine B for the following 28 days. Since the coinfection with Aspergillus and Rhizopus is extremely rarely seen in the oral cavity, a diagnostic and therapeutic dilemma easily presents itself.

Ključne riječi
Mouth Diseases; Mucormycosis; Aspergillosis; Immunocompromised Host; Leukemia

Hrčak ID: 225689

URI
https://hrcak.srce.hr/225689

▼ Article Information



Introduction

Invasive fungal infections are the major cause of infection-related mortality in hematopoietic stem cells recipients. Although mucormycosis and aspergillosis are the most frequent fungal infections, coinfection in the same host occurs rarely (1). Furthermore, they can be frequently fatal in immunocompromised patients. Treatment options usually combine medical and surgical approaches, often including extended necrosectomies. Nevertheless, the prognosis of generalized fungal infections is very poor (2).

There have been several case reports describing combined aspergillosis and mucormycosis in various parts of the body, usually with a fatal outcome (3). However, one case report depicts a patient with relapsed acute myeloid leukemia with a combined Aspergillus and Mucorales infection (lungs, brain, spleen and bone) who has been a long time survivor (4). The aforementioned coinfection rarely occurs in healthy individuals. The case of a 22- year- old, otherwise healthy US Marine who sustained extensive soft tissue injuries was published (5). Additionally, Pozo-Laderas et al. (6) published a case of a 17- year- old immunocompetent male who developed this coinfection 11 days after a motorcycle accident.

The coinfection of mucormycosis and aspergillosis should be considered in immunosuppressed patients in order to establish early management that will lead to the improved prognosis of the patient (1).

Case report

A 54-year-old male patient was referred to the Department of Oral Medicine due to the dark red/brownish lesions on the left side of the palate (Figure 1) and vestibular exophytic, later ulcerative, lesion in the area of the teeth 25-27 (Figure 2). The lesion was well demarcated from the surrounding tissue, asymmetric and without hemorrhage.

Figure 1 Palatal mucormycosis and aspergillosis: the dark red/brownish lesions on the left side of the palate
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Figure 2 Vestibular mucormycosis and aspergillosis: the vestibular ulcerative lesion in the area of the teeth 25-27
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He was diagnosed in December 2017 with acute lymphoblastic leukemia (Ph negative and pre-B). He had primary refractory disease and was treated according to protocols HOVON71 (7) and HAM (high dose of cytosine arabinoside and mitoxantrone). During HAM protocol induced aplasia, the patient developed neutropenic colitis and Aspergillus niger was identified in his stool samples. At the time of the oral examination, the patient was treated with intravenous levofloxacin, acyclovir, metronidazole, collistine, meropenem and tigecycline.

Oral lesions appeared sixteen days after the start of the HAM protocol. The patient noticed a swelling of his left cheek. The abovementioned oral lesions were identified upon examination. The orthopantomograph (Figure 3) showed a radiopaque lesion within the left maxillary sinus. Teeth 23, 26 and 27 were avital and tooth 24 was vital. Increased loosening of these teeth was noticed. A severe form of periodontal disease was present. The patient had no pain in that area, except sometimes on palpation. No evident pathological periapical pathology within teeth could be noticed. A CT finding showed a non-homogenous bony structure within the maxillary alveolar ridge.

Figure 3 Orthopantomograph showed a radiopaque lesion within the left maxillary sinus
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Incisional biopsy of a vestibular lesion was taken. Histopathology showed tissue necrosis together with adipose tissue abundant with fungal hyphae and spores. Microbiological evaluation confirmed the presence of Aspergillus fumigatus and Rhizopus genus.

Intravenous posaconazole and amphotericine B were given for the following 28 days and inferior maxillectomy was performed. The maxillary defect was to be reconstructed after the completion of hematological treatment. Meanwhile, the patient was given an obturator. Due to poor general condition of the patient, further treatment of leukemia by the transplantation of alogenous stem cells was not possible. Therefore, the treatment with blinatumomab was initiated. Unfortunately, in June 2018, the patient died due to severe hemorrhagic shock and cardiopulmonary arrest.

Since the coinfection with Aspergillus and Rhizopus extremely rarely occurs in the oral cavity, it leads easily to the diagnostic and therapeutic dilemma.

Discussion

Acute invasive fungal infections in the paranasal sinuses and surrounding tissues are progressive and carry a high death rate in an immunocompromised patient (8). Gode et al. (8) described 37 patients with acute invasive fungal rhinosinusitis and reported that the palatal involvement was significantly associated with death rate.

There are only a few case reports about a concomitant aspergillosis and zygomicosis infection in the orofacial region (1, 2, 9).

Torres-Damas et al. (1) reported a case of a 78-year-old male patient with type 2 diabetes and ketoacidosis who presented with a swelling of the right side of his face, right facial paralysis, ptosis and a necrotic ulcer on the right palate due to the Aspergillus fumigatus and mucormycosis. Chermetz et al. (2) reported the case of a 17-year-old girl with combined aspergillosis and mucormycosis of the right side of her face with frontal maxillary area and upper airway involvement after the treatment of a recurrent glioma.

Maiorano et al. (9) reported a case of aspergillosis and mucormycosis in the patient with stage-IV Castleman disease who presented with a palatal ulceration that progressively involved the palatal mucosa and bone, the paranasal sinuses and the orbit.

Although invasive fungal infections are the major cause of infection-related mortality in hematopoietic stem cells recipients, in this particular patient invasive fungal infection occurred prior to the hematopoietic stem cell transplantation, during the period of post-chemotherapy aplasia.

Differential diagnosis of such cases includes malignancy and because of that, biopsy is mandatory. Due to high frequency of fungal infections in immunocompromised patients, microbiological testing and identification of a causative organism should be performed.

Treatment of these lesions includes surgical resection of the affected tissues as well as intravenous antifungals. Posaconazole or Amphotericin BS is the drug of choice. Caspofungine can be added in resistant cases. Nevertheless, the duration of the treatment is long (median 180 days) and the outcome is unpredictable, favorable in only 40-60% of the cases.

This case report highlights the importance of considering the coinfection with Aspergillus and Rhizopus genera in the orofacial area in patients with leukemia.

References

1 

Torres-Damas W, Yumpo-Cárdenas D, Mota-Anaya E. Coinfection of rhinocerebral mucormycosis and sinus aspergillosis. Rev Peru Med Exp Salud Publica. 2015;32(4):813–7. DOI: http://dx.doi.org/10.17843/rpmesp.2015.324.1777 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/26732934

2 

Chermetz M, Gobbo M, Rupel K, Ottaviani G, Tirelli G, Bussani R, et al. Combined Orofacial Aspergillosis and Mucormycosis: Fatal Complication of a Recurrent Paediatric Glioma-Case Report and Review of Literature. Mycopathologia. 2016;181(9-10):723–33. DOI: http://dx.doi.org/10.1007/s11046-016-0021-8 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/27350324

3 

Mölle M, Blaschke-Hellmessen R, Schuler U, Nowak R, Ehninger G. Disseminated aspergillosis and mucormycosis. A case report. Mycoses. 1996;39 Suppl 1:59–64. PubMed: http://www.ncbi.nlm.nih.gov/pubmed/8767273

4 

Davoudi S, Anderlini P, Fuller GN, Kontoyiannis DP. A long-term survivor of disseminated Aspergillus and mucorales infection: an instructive case. Mycopathologia. 2014;178(5-6):465–70. DOI: http://dx.doi.org/10.1007/s11046-014-9785-x PubMed: http://www.ncbi.nlm.nih.gov/pubmed/25086667

5 

Radowsky JS, Strawn AA, Sherwood J, Braden A, Liston W. Invasive mucormycosis and aspergillosis in a healthy 22-year-old battle casualty: case report. Surg Infect (Larchmt). 2011;12(5):397–400. DOI: http://dx.doi.org/10.1089/sur.2010.065 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/22004440

6 

Pozo-Laderas JC, Pontes-Moreno A, Robles-Arista JC, Bautista-Rodriguez MD, Candau-Alvarez A, Caro-Cuenca MT, et al. Mixed invasive fungal infection due to Rhizomucor pusillus and Aspergillus niger in an immunocompetent patient. Rev Iberoam Micol. 2015;32(1):46–50. DOI: http://dx.doi.org/10.1016/j.riam.2013.03.002 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/23583263

7 

Daenen S, van der Holt B, Dekker A, Willemze R, Rijneveld A, Biemond B, et al. Intensive chemotherapy to improve outcome in patients with acute lymphoblastic leukemia over the age of 40: a phase II study for efficacy and feasibility by HOVON. Leukemia. 2012;26:1726–9. DOI: http://dx.doi.org/10.1038/leu.2012.53 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/22358264

8 

Gode S, Turhal G, Ozturk K, Aysel A, Midilli R, Karci B. Acute invasive fungal rhinosinusitis: Survival analysis and the prognostic indicators. Am J Rhinol Allergy. 2015;29(6):e164–9. DOI: http://dx.doi.org/10.2500/ajra.2015.29.4245 PubMed: http://www.ncbi.nlm.nih.gov/pubmed/26637563

9 

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(1):28–33. DOI: http://dx.doi.org/10.1007/s00428-004-1126-x PubMed: http://www.ncbi.nlm.nih.gov/pubmed/15480762


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