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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 6  |  Page : 346-348

Isolated renal mucormycosis in a 3-month-old infant


1 Department of Neonatology, Kokilaben Dhirubhai Ambani Hospital and Research Institute, Mumbai, Maharashtra, India
2 Department of Radiology, Kokilaben Dhirubhai Ambani Hospital and Research Institute, Mumbai, Maharashtra, India
3 Department of Pediatric Surgery, Kokilaben Dhirubhai Ambani Hospital and Research Institute, Mumbai, Maharashtra, India
4 Department of Pediatrics, Kokilaben Dhirubhai Ambani Hospital and Research Institute, Mumbai, Maharashtra, India
5 Department of Neonatal and Pediatric Intensive Care, Kokilaben Dhirubhai Ambani Hospital and Research Institute, Mumbai, Maharashtra, India

Date of Submission11-Jul-2020
Date of Decision26-Aug-2020
Date of Acceptance03-Sep-2020
Date of Web Publication11-Nov-2020

Correspondence Address:
Dr. Neha Nabar
Department of Neonatology, Kokilaben Dhirubhai Ambani Hospital and Research Institute, Mumbai - 400 086, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JPCC.JPCC_111_20

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  Abstract 


We report a case of isolated renal mucormycosis associated with renal failure in a 3-month-old infant. We have treated this patient by pyelotomy with the removal of the fungal ball and intravenous amphotericin B deoxycholate and achieved complete recovery.

Keywords: Amphotericin B, fungal ball, infant, isolated renal mucormycosis


How to cite this article:
Nabar N, Kalgaonkar S, Sandlas G, Singhal T, Joshi P. Isolated renal mucormycosis in a 3-month-old infant. J Pediatr Crit Care 2020;7:346-8

How to cite this URL:
Nabar N, Kalgaonkar S, Sandlas G, Singhal T, Joshi P. Isolated renal mucormycosis in a 3-month-old infant. J Pediatr Crit Care [serial online] 2020 [cited 2020 Nov 30];7:346-8. Available from: http://www.jpcc.org.in/text.asp?2020/7/6/346/300575




  Introduction Top


Mucormycosis is an opportunistic angioinvasive infection caused by fungi Mucorales, predominantly acquired by inhalation of sporangiospores, occasionally by ingestion of contaminated food, or by traumatic inoculation in immunocompromised clinical situations.[1],[2] The involvement of kidneys in mucormycosis is usually seen as a part of multisystem involvement. Isolated renal mucormycosis (IRM) is uncommon and reported largely from India.[3] Mucormycosis can be classified as rhinocerebral (seen predominantly in diabetics), pulmonary (often seen in neutropenic cancer patients), gastrointestinal mucormycosis, and cutaneous mucormycosis (following trauma and surgery).[1] Mucormycosis in neonates and young infants has been described earlier localizing to the gastrointestinal tract.[3] We report here a case of IRM in a 3-month-old infant, the youngest so far, which to the best of our knowledge have not been reported earlier.


  Case Report Top


A 3-month-old boy weighing 2.9 kg presented with acute-onset loose stools for 3–4 days followed by anuria and generalized edema. Initial evaluation at the local hospital showed hemoglobin of 9.8 gm/dl, total leukocyte count (TLC) of 10,600/cumm, platelets of 5 lakh/cumm, hyponatremia (128 mEq/L), hyperkalemia (6.1 mEq/L), elevated C-reactive protein (CRP) (5.8 mg/L), raised serum creatinine (1.6 mg/dl), and plenty of pus cells in urine with a sterile urine culture. He was treated with intravenous (IV) fluids and IV piperacillin-tazobactam, meropenem, and fluconazole. He remained anuric was referred to our hospital on the next day for dialysis. Birth history revealed preterm vaginal delivery at 33 weeks, weighing 1.5 kg, and had 2 weeks of neonatal intensive care unit (NICU) stay in view of respiratory distress and culture-negative neonatal sepsis.

On admission, vitals were stable, but he had pallor and sclerema. Investigations revealed hemoglobin of 8 g/dl, raised TLC (30,020/cumm) with neutrophilic leukocytosis (91%), raised CRP (22.8 mg/L), high creatinine (1.27 mg/dl), blood urea nitrogen (42.8 mg/dl), hypokalemia (2.56 mEq/L), and 5–10 pus cells in urine though all cultures (blood and urine) that were sent on the day of admission were sterile. Ultrasonography (USG) kidney and bladder (KUB) showed mildly enlarged kidneys with thick echoes occupying the right pelvicalyceal system. Lumbar puncture, fundoscopy, and two-dimensional echocardiography were normal.

IV fluids were administered as per acute kidney injury protocol. Meropenem was continued, and ciprofloxacin started for suspected urosepsis. Injection fluconazole was added on day 2 for the suspected fungal ball on USG KUB. Urine output normalized by day 3 and creatinine normalized by day 7 of admission though the pelvic echoes persisted.

Computed tomography scan of the abdomen and pelvis [Figure 1] on day 20 revealed moderate hydronephrosis of the right kidney with slightly dense material in the collecting system and proximal ureter with delayed excretion of contrast. Serial ultrasounds did not show any resolution of the pelvicalyceal echoes.
Figure 1: Computed tomography – right moderate hydronephrosis with hydroureter with slightly dense material in the collecting system and proximal ureter

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Double J (DJ) stent placement was done to relieve obstruction, and Micturating Cysto-Urethrogram was done to rule out reflux. Postprocedure, urine culture demonstrated filamentous aseptate hyphae suggestive of Mucorales [Figure 2]. Amphotericin B deoxycholate (1.5 mg/kg/d) was started in place of fluconazole. However, in view of no USG resolution, on day 27, fungal ball removal [Figure 2] with renal biopsy was performed through pyelotomy by lumbotomy approach to avoid peritoneal spillage, and DJ stent removal was also done. Histopathology confirmed the presence of Mucor and no fungal invasion of renal parenchyma. After surgery, urine cultures were negative for fungi. Injection amphotericin deoxycholate was continued for 4 weeks with a good clinical response. On a 3-month follow-up, urine fungal cultures were sterile, ultrasound was normal, and the infant was asymptomatic with good weight gain. The patient's workup to rule out primary immunodeficiency could not be done.
Figure 2: Fungal ball image and Histopathology Examination (HPE) – aseptate, budding and branching hyphae resembling Mucor

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  Discussion Top


RM occurs as a part of disseminated infection in patients with risk factors such as diabetes, malignancies, transplant recipients, corticosteroid therapy, neutropenia, HIV infection, IV drug use, low birth weight, malnutrition, and nosocomial infection.[1] Cases of IRM in healthy immunocompetent hosts have been predominantly from India, featuring young adults from rural backgrounds presenting with fever, flank pain, and renal failure with only a handful of cases reported in immunocompetent children.[4],[5] The presentation is similar to adults with very high mortality; the youngest reported having been 18 months old.[4]

The earliest reporting of RM in children concluded that it should be suspected in patients presenting with unexplained acute renal failure.[6] The most common site of presentation of mucormycosis in neonates is gastrointestinal and cutaneous, especially in premature infants.[7] Fungal balls in the kidney are due to candida in neonates or less commonly Aspergillus.[8] Mucor fungal balls are rare.

Probable predisposing factors in our case could be prematurity, low birth weight, and a 2-week stay in NICU for sepsis. However, the exact underlying cause could not be established with certainty. The current standard management of mucormycosis involves a combined surgical and medical approach.[9] Usual treatment incorporates nephrectomy and amphotericin B; however, there have been cases managed with amphotericin B alone.[4] Our case was managed with amphotericin B and surgical excision of the mass and biopsy of adjoining renal tissue. While literature recommends liposomal amphotericin B, we used amphotericin B deoxycholate due to superior renal penetration as compared to liposomal amphotericin B.[10]


  Conclusion Top


In an infant with acute renal failure with a past history of prematurity and prolonged neonatal intensive care, the possibility of a fungal etiology should be considered. Nonresolving pyelonephritis and characteristic imaging features point toward renal mucormycosis, which requires appropriate antifungal therapy and surgical intervention if needed.

Declaration of patient consent

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

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Prakash H, Chakrabarti A. Global epidemiology of mucormycosis. J Fungi (Basel) 2019;5:26.  Back to cited text no. 1
    
2.
Prakash H, Ghosh AK, Rudramurthy SM, Singh P, Xess I, Savio J, et al. A prospective multicenter study on mucormycosis in India: Epidemiology, diagnosis, and treatment. Med Mycol 2019;57:395-402.  Back to cited text no. 2
    
3.
Sarin YK. Intestinal mucormycosis in a neonate: A case report and review. J Indian Assoc Pediatr Surg 2010;15:98-100.  Back to cited text no. 3
[PUBMED]  [Full text]  
4.
Nayagam LS, Vijayanand B, Balasubramanian S. Isolated renal mucormycosis in an immunocompetent child. Indian J Nephrol 2014;24:321-3.  Back to cited text no. 4
[PUBMED]  [Full text]  
5.
Saran S, Naranje K, Gurjar M, Bhadauria D, Kaul A, Poddar B. Isolated renal mucormycosis in immunocompetent children: A report of two cases. Indian J Crit Care Med 2017;21:457-9.  Back to cited text no. 5
[PUBMED]  [Full text]  
6.
Gupta KL, Joshi K, Sud K, Kohli HS, Jha V, Radotra BD, et al. Renal zygomycosis: An under-diagnosed cause of acute renal failure. Nephrol Dial Transplant 1999;14:2720-5.  Back to cited text no. 6
    
7.
Roilides E, Zaoutis TE, Walsh TJ. Invasive zygomycosis in neonates and children. Clin Microbiol Infect 2009;15(suppl 5):50-4.  Back to cited text no. 7
    
8.
Vázquez-Tsuji O, Campos-Rivera T, Ahumada-Mendoza H, Rondán-Zárate A, Martínez-Barbabosa I. Renal ultrasonography and detection of pseudomycelium in urine as means of diagnosis of renal fungus balls in neonates. Mycopathologia 2005;159:331-7.  Back to cited text no. 8
    
9.
9 Levy E, Bia MJ. Isolated renal mucormycosis: Case report and review. J Am Soc Nephrol 1995;5:2014-9.  Back to cited text no. 9
    
10.
Pappas PG, Kauffman CA, Andes DR, Clancy CJ, Marr KA, Ostrosky-Zeichner L, et al. Clinical practice guideline for the management of candidiasis: 2016 update by the infectious diseases society of America. Clin Infect Dis 2016;62:e1-50.  Back to cited text no. 10
    


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