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 Table of Contents  
CASE REPORT
Year : 2020  |  Volume : 7  |  Issue : 6  |  Page : 343-345

Rhino-sinus mucormycosis with facial nerve palsy in a child with diabetic ketoacidosis


Department of Pediatrics, Bharati Vidyapeeth University Medical College, Pune, Maharashtra, India

Date of Submission22-Jun-2020
Date of Decision22-Jul-2020
Date of Acceptance30-Jul-2020
Date of Web Publication11-Nov-2020

Correspondence Address:
Dr. Rahul Jahagirdar
Bharati Vidyapeeth University Medical College, Dhankawadi, Pune - 411 043, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JPCC.JPCC_102_20

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  Abstract 


Poorly controlled type 1 diabetes mellitus predisposes the child to opportunistic infections. Mucormycosis is one such infection, Rhino-cerebral form being the most common. Maxillary sinusitis can lead to facial nerve palsy. We report here a 9-year-old boy with poorly controlled diabetes, who presented with left facial swelling and deviation of mouth to the left side in diabetic ketoacidosis. He was diagnosed to have rhinocerebral mucormycosis, maxillary sinusitis, and osteomyelitis with a complication of facial nerve palsy. The child recovered on treatment with IV antifungals and surgical debridement. This case is unique in presentation as fungal rhinocerebral sinusitis with facial nerve palsy highlighting the need of high index suspicion for fungal infection especially in underlying immunocompromised state like T1DM.

Keywords: Diabetic ketoacidosis, rhinocerebral mucormycosis, type 1 diabetes


How to cite this article:
Lohiya N, Jahagirdar R, Lohiya N. Rhino-sinus mucormycosis with facial nerve palsy in a child with diabetic ketoacidosis. J Pediatr Crit Care 2020;7:343-5

How to cite this URL:
Lohiya N, Jahagirdar R, Lohiya N. Rhino-sinus mucormycosis with facial nerve palsy in a child with diabetic ketoacidosis. J Pediatr Crit Care [serial online] 2020 [cited 2020 Nov 25];7:343-5. Available from: http://www.jpcc.org.in/text.asp?2020/7/6/343/300572




  Introduction Top


Pediatric Type 1 Diabetes Mellitus (T1DM) is a chronic systemic disorder due to insulin deficiency affecting the insulin-glucose metabolism.[1] Poorly controlled diabetes increases risk of infection and their severity.[2] It also predisposes them to opportunistic mycotic infection like mucormycosis which can be life-threatening.[3] Mucormycosis can manifest in syndromes such as pulmonary, rhino cerebral, gastrointestinal, cutaneous, central nervous system (CNS), disseminated, and miscellaneous (bones, joints, heart, and kidney). Most common among them is rhino-cerebral form. The involvement of cranial nerve in rhinocerebral mucormycosis suggests severe infection.[4] We report a 9-year-old boy known patient of T1DM who presented in Diabetic Ketoacidosis (DKA) and had mucormycosis with cranial nerve involvement.


  Case Report Top


A9-year-old boy known patient of T1DM since 6 years on subcutaneous (SC) multiple daily insulin injection was brought with complaints of fever and headache since 7 days, increased urine frequency and drooping of mouth to the left side with facial puffiness since 2 days. His blood sugars were noticed to be higher than usual since the onset of symptoms. There is no history of loss of consciousness, visual concerns, weakness in limb, or any other cranial nerve involvement. On examination, the child was lethargic with tachypnea (respiratory rate 26 per min) and acidotic breathing with bilateral pupils equally reacting to light. His heart rate was 110/min and blood pressure 110/78 mmHg. CNS examination was suggestive of the left lower motor neuron (LMN) facial nerve palsy. The child had tenderness over left maxillary region. Other systems were within normal limits. Blood gas analysis showed pH - 7.19, HCO3-9 mEq/L, urine ketones were large and blood glucose 531 mg/dL suggestive of moderate DKA. His weight was 19 kg (Z = −2.0), height 127 cm (Z = −0.74) and BMI 11.8 kg/m2 (Z = −2.5) suggestive of underweight. The child was shifted to intensive care unit and started on DKA management as per the International Society for Pediatric and Adolescent Diabetes 2018 protocol[5] with IV normal saline at 88 ml/hr. He was started on intravenous (IV) insulin at 1.5 IU/hour. Considering clinical picture he was initiated on IV Ceftriaxone (100 mg/kg/day). His investigations revealed TLC-9400/cmm (47% neutrophils, 33% lymphocytes, monocytes-14%, and eosinophils-6%), C-reactive protein (CRP) was 48 mg/L (normal < 10 mg/dL) HbA1c was 12.1% with sterile blood and urine culture. The child recovered from ketoacidosis after 18 h of fluids and insulin therapy. The child was shifted to oral feeding and SC insulin. However, his blood sugar continued to be in higher range (300–400 mg/dL) hence insulin dose was titrated (1.7 IU/kg/day) to control the sugars. His fever spikes persisted and CRP was still in the high (CRP - 35 mg/dL) despite 5 days of IV ceftriaxone treatment of 5 days hence antibiotics were escalated to IV Vancomycin (40 mg/kg/day) and IV meropenem was added for anaerobic coverage (60 mg/kg/day).

In view of the left LMN facial nerve palsy Magnetic Resonance Imaging brain was done which was suggestive of left acute rhino-sinusitis and infra-nuclear facial nerve palsy. Keeping this in mind nasal discharge was sent for culture to rule out invasive fungal rhinosinusitis. High-resolution Computed Tomography nose and peripheral nervous system [Figure 1] was done that showed mucosal collection in bilateral maxillary sinus and left anterior ethmoidal air cells suggestive of maxillary and ethmoidal sinusitis. Soft-tissue swelling in left premaxillary region and anterior left nasal bone suggesting changes of cellulitis, sclerosis with small lytic changes and cortical erosion in anterior wall of left maxillary sinus, left nasal bone, and frontal process of left maxilla suggesting osteomyelitis. Hence, the left endoscopic nasal debridement was performed. Histopathology examination of biopsy tissue was suggestive of invasive mucormycosis hyphae. The child was started on IV Amphotericin B liposomal complex (5 mg/kg/day) and treated for 28 days. IV vancomycin was stopped once the diagnosis of mucormycosis was made (after 5 days). Gradually his fever subsided, sugars became better [Figure 2] and insulin requirement came down (0.9 IU/kg/day). He was shifted to ward for completion of antifungal therapy. His facial paresis persisted but showed the improvement on follow-up.
Figure 1: High-resolution computed tomography Nose and peripheral nervous system showing mucosal collection in the left maxillary sinus suggestive of maxillary sinusitis

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Figure 2: Graph showing capillary blood sugar reading of the child. As shown there was an improvement in blood sugar after starting amphotericin B

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  Discussion Top


Pediatric patients with T1DM have immune system disorders (complement system deficiency, decreased inflammatory cytokine secretion, decreased T-lymphocyte response, decreased neutrophil function, and decrease of antioxidant system) and when associated with poorly controlled blood sugar increases the risk and severity of infections.[2] This predisposes them for higher risk of infections.

Frequency of rhino-orbital cerebral mucormycosis and facial nerve paralysis together is 11% in adults.[6] The proposed mechanism for facial nerve palsy is the probably the infection reaching the pterygopalatine fossa to inferior orbital fissure, orbital apex, and infratemporal fossa. The taste sensation in our patient was intact which is seen in patients with diabetes having facial nerve palsy due to the sparing of chorda tympani. This is different from facial nerve palsy due to viral or any other etiology.[7] The cause for vulnerability of distal facial nerve is not known. The diagnosis of mucormycosis is made by histopathological examination detecting pathognomonic feature of aseptate hyphae with right angled branching.[8] The treatment of mucormycosis is IV Amphotericin B (1–1.5 mg/kg/day) for 3–4 weeks and further continuation is required or not should be decided based on the clinical response.[9] Posaconazole is the alternative choice for treatment when resistant to amphotericin. However, early surgery in the form of debridement is needed and improves survival in fungal sinusitis. Hence, the management is multimodal with initiation of antifungal drugs and debridement.

A prospective multicenter study on mucormycosis in India has observed that uncontrolled diabetes is a common risk factor (56.8%) for the mucormycosis fungal infection highlighting higher incidence of mucormycosis in patients with poorly controlled diabetes.[10] In a study from north India, there were 4 children with Type 1 Diabetes and Rhino-Sinus mucormycosis reported to have a favorable outcome attributed to probable early onset of antifungal therapy.[11] However, our report is different as the child developed complication of facial nerve palsy highlighting the need of early intervention in the form of debridement. Another case report from south India reported a child with rhino-orbital cerebral mucormycosis involving maxillary sinus presented as DKA succumbed to the illness.[12] Our case also indicates that with early intervention it is possible to prevent such unfortunate event.

It also reinforces the need of optimum glycemic control. Such severe infections can be prevented with intense regular insulin therapy in the form of multi-dose injections and basal bolus regime. Carbohydrate counting should be taught to the patients. The patient education regarding sick day management is necessary to prevent DKA in children especially in poorly controlled diabetes.

Hence, it is important to suspect opportunistic infections like mucormycosis in a child with poorly controlled T1DM presenting with DKA. The presence of facial swelling should raise a suspicion of possible rhinocerebral mucormycosis. Facial nerve palsy is a late presentation of rhinocerebral mucormycosis warranting an immediate attention and prompt treatment with IV antifungals and debridement.

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.
Chiang JL, Maahs DM, Garvey KC, Hood KK, Laffel LM, Weinzimer SA, et al. Type 1 diabetes in children and adolescents: A position statement by the american diabetes association. Diabetes Care 2018;41:2026-44.  Back to cited text no. 1
    
2.
Calliari LE, Almeida FJ, Noronha RM. Infections in children with diabetes. J Pediatr (Rio J) 2020;96 Suppl 1:39-46.  Back to cited text no. 2
    
3.
Koc Z, Koc F, Yerdelen D, Ozdogu H. Rhino-orbital-cerebral mucormycosis with different cerebral involvements: Infarct, hemorrhage, and ophthalmoplegia. Int J Neurosci 2007;117:1677-90.  Back to cited text no. 3
    
4.
Davis RL, Robertson DM. Textbook of Neuropathology. 2nd ed. Baltimore: Williams & Wilkins; 1991. p. 761-3.  Back to cited text no. 4
    
5.
Wolfsdorf JI, Glaser N, Agus M, Fritsch M, Hanas R, Rewers A, et al. ISPAD Clinical practice consensus guidelines 2018: Diabetic ketoacidosis and the hyperglycemic hyperosmolar state. Pediatr Diabetes 2018;19 Suppl 27:155-77.  Back to cited text no. 5
    
6.
Ferguson BJ. Mucormycosis of the nose and paranasal sinuses. Otolaryngol Clin North Am 2000;33:349-65.  Back to cited text no. 6
    
7.
Pecket P, Schattner A. Concurrent Bell's palsy and diabetes mellitus: A diabetic mononeuropathy? J Neurol Neurosurg Psychiatry 1982;45:652-5.  Back to cited text no. 7
    
8.
Ferry AP, Abedi S. Diagnosis and management of rhino-orbitocerebral mucormycosis (phycomycosis). A report of 16 personally observed cases. Ophthalmology 1983;90:1096-104.  Back to cited text no. 8
    
9.
Yadav SP, Goel AK. Rhino-orbital mucormycosis–A case report. Int J Pediatr Otorhinolaryngol Extra. 2010;5:9-12.  Back to cited text no. 9
    
10.
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. 10
    
11.
Dayal D, Bakshi J, Jain P, Shivaprakash MR, Singhi S. Outcome of Rhino-Sinus mucormycosis in children with type 1 diabetes. Indian J Pediatr 2015;82:651-2.  Back to cited text no. 11
    
12.
Kumar MM, Poovazhagi V, Anbalagan S, Devasena N. Rhino-orbito-cerebral mucormycosis in a child with diabetic ketoacidosis. Indian J Crit Care Med 2014;18:334-5.  Back to cited text no. 12
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