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Table of Contents    
LETTER TO EDITOR
Year : 2022  |  Volume : 70  |  Issue : 2  |  Page : 810-811

An Unusual Patient with Acute Multiple Cranial Palsy, Tongue Fasciculations, and Proximal Weakness


Department of Neurology, King George's Medical University, Lucknow, Uttar Pradesh, India

Date of Submission19-Sep-2018
Date of Decision14-Mar-2019
Date of Acceptance17-Mar-2019
Date of Web Publication3-May-2022

Correspondence Address:
Dr. Ravindra K Garg
Department of Neurology, King George's Medical University, Lucknow - 226 003, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.344674

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How to cite this article:
Garg RK, Gupta S, Rizvi I, Malhotra HS, Kumar N, Uniyal R. An Unusual Patient with Acute Multiple Cranial Palsy, Tongue Fasciculations, and Proximal Weakness. Neurol India 2022;70:810-1

How to cite this URL:
Garg RK, Gupta S, Rizvi I, Malhotra HS, Kumar N, Uniyal R. An Unusual Patient with Acute Multiple Cranial Palsy, Tongue Fasciculations, and Proximal Weakness. Neurol India [serial online] 2022 [cited 2022 May 22];70:810-1. Available from: https://www.neurologyindia.com/text.asp?2022/70/2/810/344674


Sir,

Botulism is an infrequently encountered but potentially serious neuromuscular disorder. Botulism often presents with generalized neuroparalytic syndrome. Cranial musculature is dominantly involved. Some cases of botulism land in to respiratory paralysis and may need ventilatory support. Botulism is caused by bacteria Clostridium botulinum. Foodborne botulism is the commonest type of botulism. Foodborne botulism occurs following ingestion of contaminated food containing preformed botulinum toxin.[1],[2] We are reporting an unusual case of foodborne botulism with some unusual clinical manifestations.

An 18-year-old man came with history of acute-onset abdominal pain, nausea, and vomiting 2 days ago. Patient had dinner in a marriage party and symptoms appeared at midnight. Next morning, patient developed dry mouth, drooping of eyelids, diplopia, hoarseness of voice, and difficulty in swallowing and nasal regurgitation of liquids. On second day, patient developed acute-onset symmetrical quadriparesis. These symptoms progressed further in next 18--20 h and patient was unable to move his limbs and unable to speak. He also had mild respiratory distress. On examination (on day 3rd), patient was afebrile and had pulse rate of 88/min and blood pressure was 118/72 mm of Hg without any postural drop. Respiratory rate was 24/min. On neurological examination, patient was fully alert and oriented. Signs of meningeal irritation were absent. Patient had bilateral ptosis, bilateral complete external ophthalmoplegia, and bilateral lower motor neurone facial palsy. Pupils were dilated and were sluggishly reacting to light [Figure 1]. Gag reflex was sluggish. There was generalized tongue fasciculations [Video 1]. Patient was unable to stand from squatting position. Power was decreased in all four limbs (4/5 on MRC scale). Bulk, nutrition, tone, and all deep tendon reflexes were normal. Sensory examination was normal. Plantar reflexes on both sides were flexor. All his blood biochemical parameters were normal. Cerebrospinal fluid examination was normal. Nerve conduction studies and repetitive nerve stimulation were normal. Magnetic resonance imaging of brain was normal. Patient was subjected to neostigmine test that was also normal. Patient was provided only supportive management. Patient condition rapidly improved within 48 h. Tongue fasciculations had markedly diminished [Video 2]. At discharge from hospital, patient was able to stand from squatting position without support. He had subtle ptosis.
Figure 1: An 18-year-old man was diagnosed with botulism. He shows bilateral ptosis and bilateral absent nasolabial folds

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Possible differential diagnosis in this patient considered were Guillain--Barré syndrome, myasthenia gravis, Miller--Fisher syndrome, brainstem stroke, and snake bite. However, in view of symmetric, descending paralysis, ptosis, bilateral complete ophthalmoplegia, bilateral facial palsy, swallowing difficulty, dry mouth and heralding gastrointestinal symptoms, we made the diagnosis of foodborne botulism. Normal cerebrospinal fluid, normal electrophysiology, and normal neuroimaging further supported the diagnosis of foodborne botulism.[3],[4]

In fact, the confirmed diagnosis of botulism requires identification of Clostridium botulinum toxins or spore in vomitus, serum, or stool by the mouse inoculation assay method. We did not perform these tests.

Our patient had unusual diffuse tongue fasciculations. Tongue fasciculations disappeared after patient had recovered. Tongue fasciculations are brief spontaneous contraction of a small number of muscle fibers resulting flickering of movement of tongue. Tongue fasciculations are a characteristic manifestation of anterior horn cell diseases, particularly, amyotrophic lateral sclerosis. In neuromuscular junctional disorders, myasthenia gravis is associated with tongue atrophy and fasciculations.[5],[6] Organophosphates inhibit acetylcholinesterase enzyme, which results in the accumulation of acetylcholine at peripheral nicotinic and muscarinic receptors. Fasciculations, in organophosphate poisoning result from increased activity of nicotinic receptors at the level of neuromuscular junction.[7] Among snakes of Elapidae group, Cobra venom contains postsynaptic neurotoxins, while krait venom contains both postsynaptic and presynaptic toxins. A rapidly progressive descending paralysis is the characteristic feature of elapid snake bite in India. Bilateral ptosis and complete external ophthalmoplegia are other hallmark features. Some of these patients fail to protrude their tongue and may have speaking or swallowing difficulties.[8]

In botulism, the defect lies at the presynaptic region of neuromuscular junction. The botulinum toxin hampers the release of acetylcholine at cholinergic nerve terminals. So, it is difficult to explain the reasons for tongue fasciculations in our case. A compilation of clinical data from 332 botulism cases, authors could not find tongue fasciculation even in single case.[4] After extensive search of literature, we came with a single report describing generalized fasciculations in botulism. Possible reason of fasciculation in botulism is either denervation of lingual muscles or unstable neuromuscular junction function.[9]

In conclusion, this was an unusual case of foodborne botulism as patient had diffuse tongue fasciculations.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Chatham-Stephens K, Fleck-Derderian S, Johnson SD, Sobel J, Rao AK, Meaney-Delman D. Clinical features of foodborne and wound botulism: A systematic review of the literature, 1932-2015. Clin Infect Dis 2017;66(Suppl 1):S11-6.  Back to cited text no. 1
    
2.
Sobel J. Botulism. Clin Infect Dis 2005;41:1167-73.  Back to cited text no. 2
    
3.
Adams DZ, King A, Kaide C. Cranial neuropathies and neuromuscular weakness: A case of mistaken identity. Clin Pract Cases Emerg Med 2017;1:238-41.  Back to cited text no. 3
    
4.
Rao AK, Lin NH, Jackson KA, Mody RK, Griffin PM. Clinical characteristics and ancillary test results among patients with Botulism-United States, 2002-2015. Clin Infect Dis 2017;66(Suppl 1):S4-10.  Back to cited text no. 4
    
5.
Burch J, Warren-Gash C, Ingham V, Patel M, Bennett D, Chaudhuri KR. Myasthenia gravis--A rare presentation with tongue atrophy and fasciculation. Age Ageing 2006;35:87-8.  Back to cited text no. 5
    
6.
Krishnan M, Balasubramaniyam N. Reversible tongue atrophy in acetylcholine receptor positive bulbar onset myasthenia gravis. J Neuropsychiatry Clin Neurosci 2014;26:E56.  Back to cited text no. 6
    
7.
Chhabria BA, Bhalla A. Tongue fasciculations in organophosphate poisoning. N Engl J Med 2016;375:e47.  Back to cited text no. 7
    
8.
Alirol E, Sharma SK, Bawaskar HS, Kuch U, Chappuis F. Snake bite in South Asia: A review. PLoS Negl Trop Dis 2010;4:e603.  Back to cited text no. 8
    
9.
Arora A, Sharma CM, Kumawat B, Khandelwal D. Rare presentation of botulism with generalized fasciculations. Int J Appl Basic Med Res 2014;4:56-8.  Back to cited text no. 9
    


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