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Pediatric Oncall Journal

Unusual cutaneous and neurological complications in a child with Multisystem inflammatory syndrome 10/09/2021 00:00:00

Unusual cutaneous and neurological complications in a child with Multisystem inflammatory syndrome

Nisha R, Poovazhagi V, Seenivasan S, Karthick A R.
Department of Paediatric Intensive Care, Institute of Child Health, Madras Medical College, Chennai, India.

Poovazhagi Varadharajan, 19 & 20, Aandal Garden, Ramapuram, Chennai-600089.
4 years female child was admitted with fever and loose stools for 1 day. She was resuscitated with fluids and inotropes for hypotensive shock. Child developed cutaneous discolouration over dorsal aspect of left hand that progressed to gangrene. IgG covid antibody was positive. She was diagnosed as MIS-C and treated with immunoglobulin, methylprednisolone, aspirin, anticoagulants and antibiotics. Later, she developed cerebellar features. Neuroimaging revealed bilateral cerebellitis. She improved over the next one week and was discharged on oral drugs. Cutaneous manifestation with such severe intensity and cerebellitis as a neurological complication in MIS-C is very unusual.
MIS-C, Cerebellitis, Cutaneous gangrene.
Multisystem inflammatory syndrome in children associated with covid 19 (MIS-C) is not uncommon these days. The common cutaneous manifestations seen in MIS-C are maculopapular rashes, erythema, edema, and urticaria.1 Cutaneous involvement in the form of gangrene has been reported in adults. Though not fully understood, hypercoagulable state may be a reason for such manifestation.2 Early identification of MIS-C and prompt management with anticoagulants, steroids, and ivig results in good outcome.3
Case Report
A 4 years female child presented in the emergency department with history of fever since one day, loose stools, pain in abdomen, vomiting and lethargy. On clinical examination her GCS was 12/15, temperature of 100.7 F. She had tachycardia and tachypnea with feeble pulses. She was hypotensive with systolic blood pressure of 60 mm Hg. She was immediately resuscitated with adequate fluid therapy, inotropic support, and oxygen support. Initial diagnosis of septic shock was kept and stress dose of hydrocortisone was given. In PICU, she had one episode of posturing at 12 hours of admission and she continued to be in an altered state. At 14 hours of admission, she developed blister with discolouration on the dorsum of left hand which progressed to involve the index, middle, and ring fingers [Figure 1]. Doppler study revealed normal flow. She continued to be febrile and required inotropic support. Her laboratory parameters are summarised in Table 1. Fever work up including typhoid, leptospirosis, scrub typhus and dengue were negative. Blood and urine cultures were negative. CSF examination was normal CSF viral markers [HSV and JE enterovirus] were negative.
Her D-dimers were elevated [2.16 mcg/ml], troponinT was normal [0.82 ng/L], procalcitonin [72 ng/dl] and Interleukin 6 [22.25 pg/ml] were elevated. Echocardiogram revealed ejection fraction of 72% and fractional shortening was 41%. Coronaries were normal. IgG Covid antibody was positive [69.98 AU/ml]. Covid RT PCR was negative. She was diagnosed and was being treated as MIS-C.

Figure 1. Discolouration of the hand.
<b>Figure 1.</b> Discolouration of the hand.

Table 1. Summary of the laboratory parameters.
Day of stay 1 3 7 10 12 18
Total count cells/cumm 8800 14700 13800 10500 29000 21600
Polymorphs% 64 81 81 74 63 73
>Hb g% 12 11.5 10.3 9.4 9 11
>Platelet in lakhs/cumm 3.9 2.9 2.22 2.3 7.1 6.28
>ESR mm/hr     42 60 72 17
>Ferritin mg/dl     260 450    
>CRP mg/L 48   49   neg  
>Serum sodium mEq/L 127 134 135 139 131 137
>Prothrombin time sec 32   20.1   24.1 11.7
>ApTT sec 60   40   49.4 26.4
>INR 2.2   1.44   1.68 0.88
>Urea mg/dl 45 19 27 18 27 18
>Creatinine mg/dl 1.2 0.4 0.3 0.4 0.3 0.3
>SGOT IU/L 45 195 87   37  
>SGPT IU/L 14 109 89   50  
>CPK   848   505   130

Management and outcome:
She received Intravenous immunoglobulin [IVIG 2 gm/kg], intravenous methylprednisolone [MPS 30 mg/kg/day] for 3 days with injectable heparin. Inotropes were weaned off by day 3. On day 3, she developed bilateral ptosis, nystagmus, inability to sit and clumpsiness of hands. MRI brain showed features of cerebellitis. The acetylcholine receptor antibody level was done as a part of myasthenia workup which was normal [<0.11 nmol/l]. The cerebellar features decreased by day 6 of hospital stay and she started walking with minimal support. The intensity of discolouration of the hands reduced over the next 4 days in PICU and she had developed gangrene of the distal half of left index finger. Protein C and S levels were normal. Intravenous MPS was gradually tapered and shifted to oral steroids. She stayed in PICU for 21 days. On follow up she had normal echo, no cerebellar features, distal phalanx of index finger got autoamputated [Figure 2]. Her repeat MRI, MRA, MRV were normal. Nerve conduction velocity was normal. Oral steroids and aspirin were discontinued at 6 weeks of treatment.

Figure 2. Amputation of the index finger.
<b>Figure 2.</b> Amputation of the index finger.
Cutaneous manifestations of MIS-C are known, but there are not many reports available in paediatric population. Presentation like chlilblains has been predominantly reported in Covid infection1 Covid lesions presenting as acroischemia, cyanosis, bullae and gangrene has been described in literature.2 The transient cutaneous involvement leading to gangrene and autoamputation in covid is uncommon. Children presenting with skin lesions have been found to have very low positivity rate in nasopharyngeal swabs for covid RT-PCR.2 Whether these manifestations are due to coagulopathy or hypersensitive reactions need to be studied. Given the coagulopathic nature of covid-19, consideration of thrombolytics is a must in critically ill children with involvement of vascular surgeons.4 Encephalopathy, seizures, focal brain lesions including thalamic lesions, ADEM features, thrombotic manifestations, posterior reversible encephalopathy syndrome, cerebellitis and myelitis have been reported as neurological features in children with Covid 19.5,6 Though rare this is one such child with transient cerebellar features both clinically and radiologically with complete recovery. Immune-mediated parainfectious features have been described involving the brain, spine, cranial nerves, and nerve roots.5 However ptosis as a manifestation in MIS-C has not been reported so far in literature. Ptosis as a presentation in MIS-C is a new finding which needs further research.

Lessons learnt:

  • Early recognition and management of MIS-C is lifesaving.

  • Abnormal cutaneous gangrene along with elevated D-dimers need early initiation of anticoagulants.

  • Careful systemic examination on daily basis helps in diagnosing uncommon manifestations like cerebellitis in MIS-C.

Compliance with Ethical Standards
Funding None
Conflict of Interest None
  1. Michael Gottlieb, Brit Long, et al. Dermatologic manifestations and complications of COVID-19. The American Journal of Emergency Medicine. 2020;38(9):1715-1721.  [CrossRef]  [PubMed]  [PMC free article]
  2. Zhang Y, Cao W, Xiao M, et al. Clinical and coagulation characteristics in 7 patients with critical COVID-2019 pneumonia and acro- ischemia. cma.j.issn. 2020;41(4):302-307.
  3. Ouldali N, Toubiana J, Antona D, et al. Association of Intravenous Immunoglobulins Plus Methylprednisolone vs Immunoglobulins Alone With Course of Fever in Multisystem Inflammatory Syndrome in Children. JAMA.2021;325(9):855-864. doi:10.1001/jama.2021.0694  [CrossRef]  [PubMed]  [PMC free article]
  4. Patelis N, Bisdas T, Tsiachris D, et al. The role of vascular surgeons in the treatment of covid-19 associated pulmonary embolism. Journal of Vascular Surgery. 2020 Sep 1;72(3):1148.  [CrossRef]  [PubMed]  [PMC free article]
  5. Lindan CE, Mankad K, Ram D, et al. Neuroimaging manifestations in children with SARS-CoV-2 infection: a multinational, multicentre collaborative study. The Lancet Child & Adolescent Health. 2020;5(3): 167-177.  [CrossRef]
  6. Abel D, Shen MY, Abid Z, et al. Encephalopathy and bilateral thalamic lesions in a child with MIS-C associated with COVID-19. Neurology. 2020;95(16):745-748.  [CrossRef]  [PubMed]  [PMC free article]


Cite this article as:
R N, V P, S S, R K A. Unusual cutaneous and neurological complications in a child with Multisystem inflammatory syndrome. Pediatr Oncall J. 2023;20: 51-53. doi: 10.7199/ped.oncall.2023.14
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