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| CASE REPORT |
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| Year : 2022 | Volume
: 9
| Issue : 4 | Page : 142-143 |
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Early morning neuroparalytic syndrome – A diagnostic dilemma: A case report
Bandya Sahoo, Reshmi Mishra, Mukesh Kumar Jain, Jyoti Ranjan Behera, Amit Ranjan Rup, Arun Kumar Das, Sibabratta Patnaik
Department of Pediatric, KIMS, Patia, Bhubaneswar, Odisha, India
| Date of Submission | 22-Apr-2022 |
| Date of Decision | 27-May-2022 |
| Date of Acceptance | 28-May-2022 |
| Date of Web Publication | 20-Jul-2022 |
Correspondence Address:
Dr. Reshmi Mishra
Department of Pediatric, KIMS, Patia, Bhubaneswar - 751 024, Odisha
India
 Source of Support: None, Conflict of Interest: None
DOI: 10.4103/jpcc.jpcc_35_22
Snakebite is a commonly seen problem in tropical countries like India. Early morning neuroparalytic syndrome and cranial nerve palsies are the usual presentations. Locked-in syndrome (LIS) is a rare presentation. We present a 1-year and 6-month-old female toddler with acute onset of weakness in all four limbs, who developed LIS. The patient was given anti-snake venom on day 3 of admission and fully recovered after 3 weeks of ventilation. Snakebite should be suspected in any child presenting with early-onset neuroparalytic syndrome or LIS even if there is no history of snakebite or any bite mark.
Keywords: Locked-in syndrome, early morning neuroparalytic syndrome, snakebite
How to cite this article:
Sahoo B, Mishra R, Jain MK, Behera JR, Rup AR, Das AK, Patnaik S. Early morning neuroparalytic syndrome – A diagnostic dilemma: A case report. J Pediatr Crit Care 2022;9:142-3 |
How to cite this URL:
Sahoo B, Mishra R, Jain MK, Behera JR, Rup AR, Das AK, Patnaik S. Early morning neuroparalytic syndrome – A diagnostic dilemma: A case report. J Pediatr Crit Care [serial online] 2022 [cited 2023 Feb 8];9:142-3. Available from: http://www.jpcc.org.in/text.asp?2022/9/4/142/351516 |
| Introduction | |  |
Snakebite accounts for 1.2 million deaths, with nearly a quarter being under 15 years.[1] The annual incidence of snakebite in Odisha is between 25,000 and 60,000, with mortality between 400 and 900 annually.[2],[3] Children are more likely to have a severe systemic manifestation of envenomation due to their smaller body mass for venom distribution. Krait bite is very notorious as they are indoor and nighttime biters associated with minimal to no symptoms. The classical clinical constellation, “early morning neuroparalytic syndrome,” is usually seen with krait bite. Despite the high degree of suspicion, the diagnosis may be obscure.
| Case Report | | |
A 18-months female toddler presented to the emergency department with history of weakness of lower limbs and slurring of speech for 1 day which progressed to weakness of all four limbs 12 hours before admission. On admission, she was hemodynamically stable with Glasgow Coma Scale (GCS) 12/15; drooping of both eyelids was present. Neurological examination revealed quadriplegia with hypotonia, grade 0/5 in all limbs, hypotonia, and absent superficial and deep tendon reflexes with plantar unresponsive. Within a few hours of admission, GCS deteriorated to 3/15, requiring intubation and mechanical ventilation.
Doll's eye movements, corneal reflexes, and cough reflex were absent. Both pupils were dilated (8 mm) and not reacting to light. Respiratory system examination revealed scattered coarse crepitation at both left infra-axillary regions with vesicular breathing. Cardiovascular and abdominal examinations were clinically normal. On detailed history taking, it was found that the symptoms had started at 2 a.m. when the child was sleeping on the bed. She wake-up started crying and was unable to stand. Within 6 h, the child was unable to sit. The parents did not report any venomous bite.
Infectious etiology was ruled out with a normal complete blood count and C-reactive protein. Serum electrolytes, renal function test, liver function test, cerebrospinal fluid study, and MRI brain were normal. A nerve conduction study showed predominantly axonal motor polyneuropathy affecting both lower and upper limbs. Given the above, the diagnosis of Guillain–Barre syndrome was entertained, and intravenous immunoglobulin started. On day 2, the child had a generalized tonic–clonic convulsion. Electroencephalography (EEG) showed abnormal interictal EEG with epileptiform activity from the temporoparietal region. Although there was no history of snakebite and no bite marks could be detected, parents were interrogated due to strong clinical suspicion, and a snake was found in their house. The patient was given anti-snake venom (ASV) on day 3 of admission. On day 5 of admission, the child developed hypertension. She also had persistent tachycardia. She was managed with ventilator support, intravenous phenytoin, broad-spectrum antibiotics, and nifedipine. However, motor paralysis and low GCS persisted for 3 weeks after that child gradually started opening eyes, responding to parents with the neck movement.
| Discussion | | |
A common krait is nocturnally active and leads to a painless early morning bite. Therefore, in 60%–70% of cases, a bite occurs when the patient is asleep, and the bite site is undetectable in 17%.[4] It may present as early morning neuroparalysis (EMNP) with deep coma and absent brainstem reflexes. This patient had EMNP with absent brainstem reflexes, and there was no mark of snakebite over the body. Similar cases have been reported by Mali et al.[5] The risk of mortality is high in younger children with ptosis or cardiac arrest at admission; this can be reduced by timely recognition and early respiratory support.[6]
Elapid (common krait) venom contains alpha- and beta-bungarotoxins which act at the presynaptic and postsynaptic membrane of the motor end plate.[7] Krait venom neurotoxins lead to autonomic disturbance, including abdominal pain, vomiting, perspiration, mydriasis, tachycardia, and hypertension due to decreased cholinergic activities.[8] The patient in this study developed tachycardia, hypertension, and intermittent perspiration during the stay in the pediatric intensive care unit.
Locked-in syndrome (LIS) is a neurological syndrome in which the patient cannot communicate despite being conscious. Snakebite is one of the peripheral causes of LIS, where the elapid neurotoxin acts presynaptically by preventing the release of acetylcholine from the nerve terminals. Previous studies[9],[10] have demonstrated LIS in children, which lasted up to 1 week. The patient in this study started recovering after 3 weeks, although snake venom has been documented to remain in circulation for a maximum of 7 days.[11] Irreversible binding of the toxin to the presynaptic portion makes clinical recovery slow in krait envenomation as recovery occurs only with the formation of new neuromuscular junctions, which could be the cause in this case. Another reason for delayed recovery in this patient could be the delay in treatment with ASV. Unlike other patients where ASV was given within a few hours, this child received it on day 3 of snakebite due to a diagnostic dilemma.
Snakebite should be suspected in any child presenting with early-onset neuroparalytic syndrome or LIS even when there is no history of snakebite or any bite mark and hence promptly treated with ASV that delay in recovery can be avoided.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form, the parents have given their consent for the child's images and other clinical information to be reported in the journal. The parents understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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