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CORRESPONDENCE Table of Contents  
Ahead of print publication
Disseminated fusariosis with endophthalmitis in a patient with acute myeloid leukemia


1 Department of Dermatology, Chang Gung Memorial Hospital, Linkou Branch, Taoyuan, Taiwan
2 Department of Ophthalmology, Chang Gung Memorial Hospital, Linkou Branch, Taoyuan, Taiwan
3 Division of Hematology-Oncology, Chang Gung Memorial Hospital, Linkou Branch, Taoyuan, Taiwan

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Date of Submission02-Apr-2022
Date of Decision23-Jun-2022
Date of Acceptance18-Jul-2022
Date of Web Publication20-Oct-2022
 


How to cite this URL:
Hsu TH, Huang TE, Hung YT, Su YJ, Hung WK, Sun PL. Disseminated fusariosis with endophthalmitis in a patient with acute myeloid leukemia. Dermatol Sin [Epub ahead of print] [cited 2022 Nov 28]. Available from: https://www.dermsinica.org/preprintarticle.asp?id=359339




Dear Editor,

Invasive fungal infection is a severe disease in immunocompromised patients. Besides Aspergillus, Fusarium is another cause of infection, especially in patients with acute leukemia and in those after hematopoietic cell transplantation or chemotherapy.[1] The typical cutaneous presentation of invasive fusariosis is diffuse erythematous papulonodules with central necrosis.[1] Disseminated fusariosis (DF) of multiorgan involvement of the lungs, eyes, and kidneys can cause morbidity.[2] Herein, we report a patient with DF who presented with fungemia, disseminated skin lesions, and endophthalmitis caused by Fusarium petroliphilum.

A 52-year-old man with relapsed refractory acute myeloid leukemia (AML) was admitted to the hematology ward for intensive chemotherapy, with the FLAG regimen (fludarabine, high-dose cytarabine, and granulocyte colony-stimulating factor). Seven days after initiating chemotherapy, the patient developed grade IV febrile neutropenia (absolute neutrophil count, 25/μl) and disseminated erythematous papulonodules with central necrosis [Figure 1]a and [Figure 1]b. Despite empirical treatment with acyclovir and anidulafungin, the cutaneous eruption progressed with persistent fever. A skin biopsy was performed, and histopathological examination revealed papillary dermal hemorrhage, a perivascular lymphohistiocytic infiltrate with erythrocyte extravasation, and few fibrin thrombi [Figure 2]a and [Figure 2]b. Periodic acid-Schiff staining with diastase demonstrated the presence of fungal hyphae within and around small dermal vessels [Figure 2]c. Furthermore, the growth of Fusarium spp. was identified in both blood and tissue culture. The colonies exhibited a grayish color, fine velvety surface, and a dark red reverse color on potato dextrose agar [Figure 2]d. Macroconidia were rare, curved, and three celled [Figure 2]e. Microconidia were reniform, 0–1 septate, and grouped at the tip of the conidiophore [Figure 2]f. The isolates were identified as F. petroliphilum based on sequence alignment and phylogenetic analysis of the transcription elongation factor-1α and the internal transcribed spacers of ribosomal DNA. The final diagnosis was DF caused by F. petroliphilum. Combination therapy of intravenous liposomal amphotericin B (L-AMB) and voriconazole (VOR) was administered.
Figure 1: (a and b) Disseminated papulonodular lesions with central vesiculation and necrosis. (c) External eye photo in the first ophthalmic visit showed mild cornea edema, moderate ciliary injection, and AC exudation at 4 o'clock. (d) The photo was taken after the first emergent vitrectomy and AC irrigation. We can still see cornea edema and hypopyon in AC. The AC exudation and hypopyon were tightly attached to the cornea endothelium and cannot be removed without severely damaging the corneal endothelium in the surgery. AC: Anterior chamber.

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Figure 2: (a) Histopathology revealed hyperkeratosis, acanthosis, thickened dermal vessels, and perivascular lymphocytic infiltrates with erythrocyte extravasation. (H and E stain, 100x) (b) Septate hyphae growing through the vessel wall. (H and E stain, 400×) (scale bar, 20μm) (c) Fungal hyphae were highlighted within and around small dermal vessels by PAS+D stain. (PAS+D stain) (scale bar, 20μm) (d) F. petroliphilum (from skin biopsy culture) grown on potato dextrose agar for 7 days.) (e) Curved, fusiform macroconidia (arrow) and reniform microconidia. (arrowhead) (lactophenol cotton blue stain) (f) Clumps of broadly reniform microconidia at the tip of the conidiophore. (arrow) (scale bar, 20μm) F. petroliphilum: Fusarium petroliphilum. PAS: Periodic acid-Schiff

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During antifungal treatment, the patient complained of left eye redness with blurred vision. The initial ophthalmic examination found ciliary injection and anterior chamber (AC) exudation [Figure 1]c, but normal fundus appearance and relatively spared visual acuity. The findings were different from typical signs of endogenous fungal endophthalmitis (EFE) that started with exudation in the choroid or retina. Since AML is a possible cause of anterior uveitis, we treated the patient with topical prednisolone initially. However, in the following days, the patient demonstrated progressive deterioration with downward visual acuity, increasing hypopyon, similar to [Figure 1]d, and most importantly, progressive vitreous opacity. Since EFE was highly suspected, we arranged multiple times of intravitreal injection (IVI) with amphotericin B and VOR, but the response was inadequate. Thus, vitrectomy with AC irrigation was performed, and F. petroliphilum was identified from the vitreous specimen. Because the AC exudation was sticky and could not be removed completely in a single procedure, two more times of vitrectomy and multiple times of IVI were performed to save the patient from evisceration. After 12 weeks of intravenous treatment with L-AMB and VOR, DF had been controlled and the patient was subsequently discharged.

F. petroliphilum is a common pathogen of economic plants; it also causes opportunistic infection in humans. Similar to other Fusarium species, F. petroliphilum exhibits a high minimum inhibitory concentration to most antifungal agents.[3] A recent review suggested that VOR or L-AMB should be used as first-line therapy in invasive fusariosis, and combined treatment may be an effective alternative in critical patients.[4] We successfully treated the patient who had relapsed AML and prolonged myelosuppression status, with the combination therapy of VOR and L-AMB.

EFE usually starts from the structure with sufficient blood supply, such as the choroid or retina. EFE with only AC involvement is extremely rare in clinical practice. Although hypopyon is not frequently presented in noninfectious uveitis cases,[5] it has been reported as a presentation of leukemia.[6],[7] Considering the patient was already receiving antifungal treatment and that the fundus finding was different from typical EFE, we diagnosed the patient as anterior uveitis but eventually realized it was EFE in disguise.

In conclusion, we reported the case of DF caused by F. petroliphilum complicated with EFE. DF should be considered as a differential diagnosis when a typical cutaneous presentation is encountered in patients with hematological malignancies. Identification of the pathogen is crucial for the selection of antifungals. Aggressive ophthalmic consultation and intervention may prevent irreversible blindness and evisceration.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that name and initials will not be published, and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Acknowledgment

This manuscript was edited by Wallace Academic Editing.

Financial support and sponsorship

Nil.

Conflicts of interest

Prof. Pei-Lun Sun, an editorial board member at Dermatologica Sinica, had no role in the peer review process of or decision to publish this article. The other authors declared no conflicts of interest in writing this paper.



 
  References Top

1.
Nucci M, Barreiros G, Akiti T, Anaissie E, Nouér SA. Invasive fusariosis in patients with hematologic diseases. J Fungi (Basel) 2021;7:815.  Back to cited text no. 1
    
2.
Nucci F, Nouér SA, Capone D, Anaissie E, Nucci M. Fusariosis. Semin Respir Crit Care Med 2015;36:706-14.  Back to cited text no. 2
    
3.
Ersal T, Al-Hatmi AS, Cilo BD, Curfs-Breuker I, Meis JF, Özkalemkaş F, et al. Fatal disseminated infection with Fusarium petroliphilum. Mycopathologia 2015;179:119-24.  Back to cited text no. 3
    
4.
Ruhnke M, Cornely OA, Schmidt-Hieber M, Alakel N, Boell B, Buchheidt D, et al. Treatment of invasive fungal diseases in cancer patients-Revised 2019 Recommendations of the Infectious Diseases Working Party (AGIHO) of the German Society of Hematology and Oncology (DGHO). Mycoses 2020;63:653-82.  Back to cited text no. 4
    
5.
Zaidi AA, Ying GS, Daniel E, Gangaputra S, Rosenbaum JT, Suhler EB, et al. Hypopyon in patients with uveitis. Ophthalmology 2010;117:366-72.  Back to cited text no. 5
    
6.
Tyagi M, Govindhari V, Pappuru RR, Ambiya V. Bilateral hypopyon uveitis in chronic myeloid leukemia. Ocul Oncol Pathol 2017;4:12-5.  Back to cited text no. 6
    
7.
Sudharshan S, Kumari A, Biswas J. Bilateral hypopyon as the presenting feature of chronic myeloid leukemia. Ocul Immunol Inflamm 2008;16:244-6.  Back to cited text no. 7
    

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Correspondence Address:
Pei-Lun Sun,
No. 5, Fushin Street, Guishan Dist., Taoyuan 333
Taiwan
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Source of Support: None, Conflict of Interest: None

DOI: 10.4103/1027-8117.359339



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