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 » Introduction
 » Case Report
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Table of Contents    
CASE REPORT
Year : 2012  |  Volume : 60  |  Issue : 5  |  Page : 514-516

Combined involvement of muscle, nerve, and myoneural junction following Leptospira infection


1 Department of Neurology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India
2 Department of Microbiology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh, India

Date of Submission25-Aug-2012
Date of Decision25-Aug-2012
Date of Acceptance27-Sep-2012
Date of Web Publication03-Nov-2012

Correspondence Address:
Sunil Pradhan
Department of Neurology, Sanjay Gandhi Postgraduate Institute of Medical Sciences, Lucknow, Uttar Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0028-3886.103202

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 » Abstract 

Leptospirosis is a zoonosis prevalent worldwide and is endemic in many parts of India. In early leptospiremic as well as late immune phase of the disease kidney, liver, heart, and lungs are commonly involved. Neurological manifestations are rare but may occur during immune phase in the form of aseptic meningitis, meningoencephalitis, seizures, myelitis, polyradiculoneuritis, and myalgia. In this report, we describe a rare case of leptospirosis with combined involvement of nerve, muscle, and myoneural junction in generalized fashion along with pulmonary, renal, and hepatic dysfunction.


Keywords: Leptospira, myasthenia, myoneural junction, myopathy, myositis, neuropathy


How to cite this article:
Pradhan S, Tandon R, Kishore J. Combined involvement of muscle, nerve, and myoneural junction following Leptospira infection. Neurol India 2012;60:514-6

How to cite this URL:
Pradhan S, Tandon R, Kishore J. Combined involvement of muscle, nerve, and myoneural junction following Leptospira infection. Neurol India [serial online] 2012 [cited 2020 Jan 28];60:514-6. Available from: http://www.neurologyindia.com/text.asp?2012/60/5/514/103202



 » Introduction Top


Leptospirosis occurs when abraded skin or intact mucous membranes of eye, throat, and gut come in contact with water contaminated with the urine of reservoir animals such as rodents harboring Leptospira interrogans.[1] With improved clinical awareness and laboratory back-up, there is upsurge in the diagnosis of Leptospirosis in India. [2] Since the 1980s, cases of leptospirosis are being reported during the monsoon months from coastal states of peninsular India such as Gujarat, Maharashtra, Kartnataka, Kerala, Tamil Nadu, and Orissa, and from the Andaman Islands. [3],[4] Some sporadic cases have also been reported from northern parts of India including Uttar Pradesh. [5]

Leptospirae multiply in blood and tissues and after an incubation period of 7-10 days they bind to capillary endothelium resulting in vasculitis, which results in the involvement of kidney, liver, heart, and lungs. [6] The infection may take a subclinical form, a milder anicteric form, or a more florid multi-organ form, Weils's sysndrome. The initial Leptospiremic phase is often followed by immune phase. [6] Although mild neurological symptoms such as headache, vomiting, restlessness, confusion, delirium, hallucinations, fatigue, and myalgia may occur during early Leptospiremic phase of illness, most of the serious neurological complications occur during the late immune phase. These include aseptic meningitis, meningoencephalitis, subarachnoid hemorrhage, acute disseminated encephalomyelitis, seizures, myelitis, and polyradiculoneuropathy including Guillain-Barre syndrome and optic neuritis. [7] Severe form of combined central and peripheral involvement (meningo-myelo-encephalo-polyneuritis) has been described. [8] This report describes a patient of leptospirosis with unusual neurological complications, probably for the first time.


 » Case Report Top


A 25-year-old man presented with 4 months history of gradually worsening difficulty in swallowing, speaking, and limb weakness that occurred 20 days after an acute febrile illness of 10 days duration. He would drink liquids very slowly and carefully to avoid nasal regurgitation. Speech was of low volume with an appreciable diurnal variation with worsening by the evening. There was hypotonia in the limbs with motor weakness of 2-3/5 in the lower limbs and of 3-4/5 in the upper limbs, proximal muscles were weaker than the distal muscles. The weakness was asymmetrical and little more on the left side. There was no diurnal variation in the limb motor weakness. All the deep tendon reflexes were absent and plantar response was bilaterally flexor. There was no sensory deficit. He needed feeding tube for swallowing difficulty and mechanical ventilation for respiratory muscle weakness.

Blood biochemistry showed evidence of mild hepatic and renal dysfunction, which improved over a period of 2 months. Cerebrospinal fluid examination was essentially normal. Serum creatine kinase levels were normal but lactate dehydrogenase (LDH) levels were high (717 IU/L). Serology was negative for antibodies (ELISA) against dengue virus and human immunodeficiency virus (HIV). The patient's serum was tested in duplicate for Leptospira genus specific IgM antibodies against purified Leptospira Patoc 1 antigen using SCIMEDX LEPTOSPIRA Microwell ELISA kit (USA) and was found to be positive. Magnetic resonance imaging (MRI) of the dorsal and lumbar spine was normal.

Nerve conduction study revealed reduced amplitudes of compound muscle action potentials (CMAP) as well as sensory nerve action potentials (SNAP) with mild reduction in conduction velocities and no conduction block, suggestive mainly axonal polyneuropathy. Electromyography (EMG) showed myopathic motor unit potentials with increased proportion of small amplitude and short duration polyphasic potentials [Figure 1]a. Occasional fibrillation potentials were also observed. On slow repetitive nerve stimulation (3 Hz; 2 s), decremental response was observed over right nasalis (-40%) and right abductor pollicis brevis (-32%) muscles [Figure 1]b. On fast repetitive nerve stimulation (10-30 Hz; 5 s), no incremental response was observed. These findings were suggestive of postsynaptic myoneural junction defect.
Figure 1: Concentric needle electromyography of the patient showing small amplitude (100-500 μV), short duration (1-5 ms) triphasic and polyphasic motor unit potentials suggesting myopathic pattern (a) and decremental response (-40%) recorded over the nasalis muscle after 3 Hz supramaximal stimulation of the facial nerve (b)

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The patient's swallowing and respiratory dysfunction dramatically improved with oral pyridostigmine 60 mg four times a day. In view of persisting renal dysfunction a course of steroids with doxycycline and ceftriazone was given. Clinical, biochemical, and electrophysiological parameters normalized in 2 months and steroids were withdrawn in 3 months without any sign of recurrence in the next 6 months of follow-up.


 » Discussion Top


A 10-day-long fever 1 month prior to the present illness that was associated with pulmonary, hepatic, and renal involvement may represent Leptospiremic phase of infection. The diagnosis of leptospirosis was confirmed with IgM ELISA. The occurrence of neurological symptomatology 20 days after the subsidence of acute fever suggests second or the immune phase of the disease.

Myopathy is perhaps under reported and passed on simply as myalgia. One study reported 37% for myositis and 39% for cardiac muscle involvement. [9] Painful and tender proximal muscle weakness with high LDH levels and myopathic EMG suggested myopathy in our patient. Involvement of myoneural junction was perhaps the most conspicuous neurological complication in our patient. Review of English literature failed to show documentation of such neurological complication in Leptospirosis. In our patient, the disease began with difficulty in swallowing, low volume voice, and nasal intonation of speech; these symptoms had diurnal variation. Further, repetitive nerve stimulation test showed decremental response in facial as well as limb muscles suggesting postsynaptic receptor blockade. There was a dramatic improvement in these myasthenic symptoms with the use of pyridostigmine. Peripheral neuropathy predominantly involving lower limbs has been described as a late complication of leptospirosis. [10] It was difficult to diagnose neuropathy in this patient who already had underlying myopathy and myoneural junction defect. However, features suggestive of neuropathy included absent deep tendon reflexes and electrophysiological evidence of reduced CMAP amplitudes as well as sensory potentials and EMG showing fibrillation potentials.

This is a unique case of Leptospirosis with simultaneous occurrence of neuropathy, myopathy and postsynaptic neuromuscular junction disease, the transient form of the latter being reported here for the first time.

 
 » References Top

1.Faine S. Guidelines for the control of leptospirosis. WHO Offset Publ 1982;67:1-171.  Back to cited text no. 1
    
2.Sumathi G, Chinnari Pradeep KS, Shivakumar S. MSAT - a screening test for leptospirosis. Indian J Med Microbiol 1997;15:84.  Back to cited text no. 2
    
3.Singh J, Sokhey J. Epidemiology, presentation and control of leptospirosis. Proceedings of the third round table conference. Series - Leptospirosis. Ranbaxy Science Foundation (3); 1998. p. 17-31.  Back to cited text no. 3
    
4.Kamath SA, Joshi SR. Re-emerging of infections in urban India--focus leptospirosis. J Assoc Physicians India 2003;51:247-8.  Back to cited text no. 4
    
5.Manocha H, Ghoshal U, Singh SK, Kishore J, Ayyagari A. Frequency of leptospirosis in patients of acute febrile illness in Uttar Pradesh. J Assoc Physicians India 2004;52:623-5.  Back to cited text no. 5
    
6.Speelman P. Leptospirosis. In: Braunwald E, Fauci AS, Kasper DL, Hauser SL, Longo DL, Jameson JL, editors. Harrison's Principles of Internal Medicine. 16 th ed. New York: McGraw-Hill Medical Publishing Division; 2001. p. 2172-6.  Back to cited text no. 6
    
7.Bezerra HM, Ataíde Júnior L, Hinrichsen SL, Travassos FM, Travassos PT, Silva MJ, et al. Involvement of the nervous system in leptospirosis. I. Evaluation of neurologic aspects. Arq Neuropsiquiatr 1993;51:457-63.  Back to cited text no. 7
    
8.Lepur D, Himbele J, Klinar I, Vranjican Z, Barsic B. Anti-ganglioside antibodies-mediated leptospiral meningomyeloencephalopolyneuritis. Scand J Infect Dis 2007;39:472-5.  Back to cited text no. 8
    
9.Watt G, Padre LP, Tuazon M, Calubaquib C. Skeletal and cardiac muscle involvement in severe, late leptospirosis. J Infect Dis 1990;162:266-9.  Back to cited text no. 9
    
10.Mumford C, Dudley N, Terry H. Leptospirosis presenting as a flaccid paraplegia. Postgrad Med J 1990;66:218-20.  Back to cited text no. 10
    


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