Necrotizing cutaneous zygomycosis by Saksenaea vasiformis in post-COVID-19 individual: A rare case report

S Prasanna; Mayuri Mahajan; Nikunja Kumar Das; Nikhil Mahajan

doi:10.4103/mjhs.mjhs_146_22

CASE REPORT

Year : 2023 | Volume : 11 | Issue : 2 | Page : 164-167

Necrotizing cutaneous zygomycosis by Saksenaea vasiformis in post-COVID-19 individual: A rare case report

S Prasanna¹, Mayuri Mahajan², Nikunja Kumar Das³, Nikhil Mahajan²
¹ Department of Microbiology, Shri Sathya Sai Medical College and Research Institute, Sri Balaji Vidyapeeth (Deemed to be University), Chengalpattu, Tamil Nadu, India
² Dr. Hedgewar Hospital and Research Centre, Akola, Maharashtra, India
³ Department of Microbiology, D. Y. Patil Medical College Hospital and Research Center, D. Y. Patil Vidyapeeth, Pune, Maharashtra, India

Date of Submission	10-Nov-2022
Date of Decision	17-Jan-2023
Date of Acceptance	30-Jan-2023
Date of Web Publication	18-Apr-2023

Correspondence Address:
S Prasanna
Department of Microbiology, Shri Sathya Sai Medical College and Research Institute, Sri Balaji Vidyapeeth (Deemed to be University), Chengalpattu - 603 108, Tamil Nadu
India

Source of Support: None, Conflict of Interest: None

DOI: 10.4103/mjhs.mjhs_146_22

Abstract

We report a rare case of cutaneous zygomycosis by Saksenaea vasiformis in post-COVID-19 individuals. A 55-year-old patient was COVID-19 positive and treated as per the protocols. Following recovery after 5 weeks, presents to the surgery outpatient department with complaints of slowly progressive cutaneous lesion developed into ulcerative lesion over the left lateral part of the abdomen, hip, and thigh. Based on histopathological and microbiological findings, he was diagnosed with as a case of cutaneous zygomycosis by S. vasiformis. Initially, conservative management with intravenous amphotericin B was given and followed by surgical debridement; later, the patient succumbed. In general, mucormycosis is associated with immunosuppression or debilitating diseases. The mode of entry for molds and spores of zygomycetes is through the respiratory tract through the nose and then reaches the sinuses, orbit, and intracranial structures. Hence, early clinical diagnosis, direct smears, and proper interventions lead to a good prognosis and reduced morbidity caused by zygomycosis. The therapeutic management of fungal infection is quite challenging; hence diagnosing zygomycosis at the earliest will be appropriate, especially in an immunocompromised state. The misusing of prophylactic drugs, steroids, and immunosuppressants for COVID-19 should be avoided and must be used as per protocol and guidelines.

Keywords: Cutaneous zygomycosis, immunosuppressed, post-COVID-19, Saksenaea vasiformis

How to cite this article:
Prasanna S, Mahajan M, Das NK, Mahajan N. Necrotizing cutaneous zygomycosis by Saksenaea vasiformis in post-COVID-19 individual: A rare case report. MRIMS J Health Sci 2023;11:164-7

How to cite this URL:
Prasanna S, Mahajan M, Das NK, Mahajan N. Necrotizing cutaneous zygomycosis by Saksenaea vasiformis in post-COVID-19 individual: A rare case report. MRIMS J Health Sci [serial online] 2023 [cited 2023 May 27];11:164-7. Available from: http://www.mrimsjournal.com/text.asp?2023/11/2/164/374273

Introduction

Zygomycetes are aseptate filamentous fungi that are ubiquitous and can cause infection by inhalation of spores or by cutaneous inoculation. These zygomycetes are classified into two orders, Mucorales and Entomophthorales. The Basidiobolus species and Conidiobolus species belong to the order Entomophthorales. Mucor, Absidia, Rhizopus, Rhizomucor, Apophysomyces, Saksenaea, Cunninghamella, Cokeromyces, and Syncephalastrum are the infection causing Mucorales. The most common cause of zygomycosis is Rhizopus, followed by Mucor, Absidia, and Rhizomucor in descending order.^[1],[2]

The severity of infection by zygomycetes ranges from mild cutaneous to invasive and disseminated forms. The different forms of zygomycosis are rhino-cerebral, pulmonary, abdominal, pelvic, cutaneous, subcutaneous, and disseminated. Among the invasive fungal infections, zygomycosis is the third most common, following aspergillosis and candidiasis. The immunocompromised hosts develop severe conditions compared to immunocompetent hosts.^[3]

Uniqueness

Usually, cutaneous forms of zygomycosis are ultimately recoverable diseases with proper diagnosis and treatment. It is a gradual and slowly progressive disease among immunocompetent hosts, whereas it may become fulminant and complicated into a necrotizing disease by hematogenous dissemination.^[4] Here, we are reporting a case of necrotizing cutaneous zygomycosis by Saksenaea vasiformis in post-COVID-19 individuals.

Case Report

A 55-year-old male presents to the surgery outpatient department with complaints of a slowly progressive cutaneous lesion that developed into an ulcerative lesion over the left lateral part of the abdomen, hip, thigh, and groin region for the past 2 months. T he patient is a known diabetic and was post-COVID-19 recovered 4 months before. He was treated as a moderate case as per WHO and ICMR guidelines with methylprednisolone 250 mg multivitamins, zinc, and heparin for 7 days. On examination, the patient had tenderness over the left lateral part of the abdomen, hip, thigh, and groin region with edema and redness [Figure 1] and [Figure 2]. The necrotic wound was cleaned, and debridement was done twice under anesthesia until clear wound surface and margin were obtained. The biopsy was taken and sent for potassium hydroxide (KOH), special stains, and bacterial and fungal culture. The patient received piperacillin–tazobactam 2 g intravenous (IV) bis in die (BD) and itraconazole 200 mg IV BD. The 40% KOH mount of pus and tissues showed aseptate hyaline hyphae [Figure 3]. The blood culture was positive for Escherichia coli and sensitive only to colistin. The patient was started with an injection of colistin 1 M IU as an IV infusion after stopping piperacillin–tazobactam. The pus and tissue were cultured in Sabouraud dextrose agar (SDA) plates and were incubated at 25°C–30°C. After 7 days of incubation in SDA showed grayish-white cotton woolly (cotton candy appearance) with lid lifting growth [Figure 4].

Figure 1: Left lateral view: wide margined necrotic ulcer with tenderness over the left lateral part of the abdomen, hip, thigh, and groin region with edema and redness

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Figure 2: Top view: wide margined necrotic ulcer with tenderness over the upper part of the thigh and groin region after debridement

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Figure 3: The direct microscopic examination of pus and tissues after treatment with 40% potassium hydroxide revealed the presence of aseptate hyaline hyphae (black arrow)

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Figure 4: After 7 days of incubation, growth in sabouraud dextrose agar was observed; the colony was greyish-white cotton-woolly (cotton candy appearance) raised with lid lifting growth

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Microscopy by lactophenol cotton blue (LPCB) mount of cottony growth showed flask-shaped sporangia with a distinct spherical venter, long neck, and dichotomous branching arranged in single or in pairs, and dark rhizoids. It has prominent and dome-shaped columellae. It had small and oblong sporangiospores that discharge through the neck following an apical mucilaginous plug [Figure 5]. The fungus was identified as S. vasiformis with the help of KOH, histopathology, cultural characteristics, and LPCB mount. The same fungus was isolated again from a repeat sample. Initially, conservative management with IV amphotericin B was given and followed by surgical debridement. On day 6 of admission, the patient was put on a ventilator, and 24 h later, the patient succumbed due to sepsis and respiratory failure. Consent from the patient has been taken for the publication of this information.

Figure 5: Microscopy by lactophenol cotton blue mount showed flask shaped sporangia branching dichotomously with a distinct spherical venter and longneck, arranged in single or in pairs and dark rhizoids with dome shaped columellae (red arrow)

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Discussion

The mucormycosis is classified and given nomenclature based on restriction to the anatomic sites than by mycologic classification. The various clinical forms are pulmonary, disseminated, cutaneous, nasal, rhino-orbital, paranasal, parasinus, and rhino-orbital-cerebral or rhino-cerebral mucormycosis.^[5] The first human infection with S. vasiformis was reported in 1976; however, this genus Saksenaea was described in 1953. A few cases have been reported to date. S. vasiformis is a rare and emerging pathogen that causes severe infections even among immunocompetent hosts. Disseminated forms of disease by S. vasiformis are rare compared to cutaneous and subcutaneous infections.^[6],[7] The family of Mucoraceae is ubiquitous soil saprophytic fungi commonly distributed in decaying matter, bread, soil, air, dust, and hospital environments. Most clinical isolates belong to the genus Rhizopus among the Mucoraceae.^[8] This group of organisms is more common in a temperate climate and is related to the use of air conditioners. The most common predisposing risk factors are diabetes mellitus, immunosuppressing drugs, cancer chemotherapy, iron overload, neutropenia, stem cell transplantation, diabetic ketoacidosis, and HIV/AIDS.^[9] The mode of spread is through the respiratory route and has an affinity toward the arteries and veins, and causes thrombosis and infarction.^[10] The intracranial involvement is followed by disease progression from the nose and sinuses through the cribriform plate, superior orbital fissure, ophthalmic vessels, carotid artery, and perineural route by vascular occlusion, thrombosis, and infarction.^[9],[10] The initiation of treatment is mainly based on an accurate clinical picture and diagnosis with the help of computer tomography and magnetic resonance imaging. The availability of positive direct smears with specific fungal stains such as periodic acid–Schiff and Gomori's methenamine silver will be supportive. It is impractical to wait for culture reports as that may lead to further disease progression.^[10]

S. vasiformis is a broad, irregularly branching aseptate fungus like other Mucorales. Like other zygomycetes, S. vasiformis proliferates on routine media and produces grayish-white colonies. However, in routine media, any identifying sporulating structures may not appear. To induce sporulation, nutrient-deficient media such as tap water agar must be used. The distinct sporulating structures will be visible under microscopy after 1 to 2 weeks. The pathognomonic feature of S. vasiformis is its distinctive funnel-shaped pigmented sporangia and apical sporangiospores.^[11]

Strength and limitations

The extent of involvement in mucormycosis cases and its management is based on the clinical presentation, histopathological, microbiological, and imaging findings. The spread of the disease further can be controlled by early diagnosis and active surgical debridement.^[12] The high-dose steroid therapy in COVID-19 leads to complications in the presence of underlying mucormycosis. The mainstay of treatment is a combination of amphotericin B and surgical debridement. Long-term treatment is required with amphotericin B because it is fungistatic and not fungicidal. Orbital exenteration or local debridement at the earliest ha s a crucial role in the treatment protocol apart from antifungal therapy for a good patient prognosis.^[13] The patient succumbed in our case because of the late presentation and quite complicated state with S. vasiformis and superadded infections and sepsis, and respiratory failure.

Conclusion

Early clinical diagnosis, direct smears, and proper interventions lead to a good prognosis and reduced morbidity caused by zygomycosis. The therapeutic management of fungal infection is quite challenging; hence diagnosing zygomycosis at the earliest will be appropriate, especially in an immunocompromised state. The highly invasive skin lesions associated with this group of fungi, such as S. vasiformis, are the most important. They are to be screened and diagnosed earlier from culture and staining methodologies. The sudden increase in incidence and prevalence of mucormycosis and other opportunistic infections must be researched well to prevent and manage post-COVID-19 patients. The misusing of prophylactic drugs, steroids, and immunosuppressants for COVID-19 should be avoided and must be used as per protocol and guidelines.

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 his name and initials will not be published and due efforts will be made to conceal his identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

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