Neurol Sci DOI 10.1007/s10072-014-1841-2
LETTER TO THE EDITOR
Guillain2Barre´ syndrome following measles infection: case report and review of the literature Antonietta Filia • Giuseppe Lauria
Received: 4 April 2014 / Accepted: 16 May 2014 Ó Springer-Verlag Italia 2014
Dear Editor, We report a case of Guillain-Barre´ syndrome (GBS) occurring after measles infection and review the published literature on the subject. A previously healthy 48-year-old man developed fever, cough and constipation, followed by a maculopapular rash over the face and neck. Measles infection was diagnosed. Four days later, he developed back pain and paraesthesias in the hands and feet. Computed tomography, magnetic resonance imaging scans of the brain and a chest radiograph were normal. One day later, he was admitted to hospital for numbness in the hands and feet bilaterally, dyspnoea, perioral pain, headache and nausea. The patient was afebrile and unable to walk and stand upright. He denied prior neurological illnesses. Examination showed weakness of the upper and lower extremities, more pronounced proximally, and symmetrical hypoesthesia. Deep tendon reflexes were absent. Nerve conduction studies showed moderate increase of motor nerve distal latencies, slowing of motor nerve conduction velocity, desynchronization of compound motor action potential amplitudes and reduced amplitude of sensory nerve action potential in the upper limbs. Serum measles IgM antibodies were elevated. Cerebrospinal fluid examination disclosed albumin-cytological dissociation
A. Filia (&) Infectious Diseases Epidemiology Unit, National Centre for Epidemiology, Surveillance and Health Promotion, Istituto Superiore di Sanita`, Viale Regina Elena 299, 00161 Rome, Italy e-mail: [email protected] G. Lauria Carlo Besta Neurological Institute, IRCCS Foundation, Milan, Italy
and elevated measles IgM antibody titres. Isoelectric focusing showed a mirror pattern. No paraprotein was found. Antibody titres to cytomegalovirus, rubella, Epstein-Barr virus and herpes simplex viruses 1 and 2 were negative. Anti-ganglioside antibodies were not measured. GBS of the demyelinating type was diagnosed and treatment with intravenous immunoglobulin was given. Over the following three days, the patient developed cranial nerve palsies, tetraplegia, and respiratory insufficiency necessitating admission to the Intensive Care Unit and a tracheotomy. Three weeks later he experienced severe cardiovascular autonomic dysautonomia which resolved within five days. The patient then underwent plasma exchange and began to improve clinically. He was discharged seven weeks after admission, with residual left 7th cranial nerve palsy, severe weakness of foot dorsiflexion, mild proximal muscle weakness of all four extremities, and mild neuropathic pain in the lower limbs. Measles is a vaccine-preventable, acute viral illness which is well-known to cause neurological complications, the most frequent being post-infectious measles encephalitis (PIME). The pathogenetic mechanisms involved in measles neurological complications remain to be completely understood. Interestingly, PIME appears to be caused by an abnormal immune reaction against myelin basic protein and the pathogenesis of GBS also seems to involve an autoimmune-mediated process. Approximately two-thirds of patients with GBS report a preceding infection but the pathogen frequently remains unidentified. Campylobacter jejuni, cytomegalovirus, Epstein-Barr virus, Mycoplasma pneumoniae have been shown to be related to GBS and there is some evidence that varicella-zoster and influenza viruses and H. influenzae may also be triggers.
123
Neurol Sci Table 1 Published studies and case reports describing cases of Guillain-Barre´ syndrome (GBS) following measles infection Reference
Type of study
Number of GBS cases with measles as antecedent infection
Patient age (years)
Time interval between onset of rash and neuropathic symptoms
Lab confirmation of measles
Bourne and Scott [1] Miller et al. [2]
Case report Review of 911 cases with neurological complications of measles Literature review of 638 GBS cases associated with infections Case series
1 10
5.5 years 5-21 years (range)
NS NS
11
NS
7 days 9 days on average (in four cases the interval was only 4-5 days) NS
2
9 years, 5.5 years
5 days (in both cases)
Yes
1 1 1 2 1 1
40 years 19 years 39 years 3 years, 5 years 28 years 12 years
Asymptomatic 15 days 6 days 12 and 10 days 4 days 4 weeks
Yes Yes Yes NS Yes Yes
Leneman [3]
Lidin-Jandon and Strannegard [4] Dru¨eke et al. [5] Guerin et al. [6] Nakamura et al. [7] Murthy [8] Tomiyasu et al. [9] Loomba A and Maertens [10]
Case Case Case Case Case Case
report report report series of 15 cases of GBS report report
No (clinical cases)
NS not specified
A literature review was conducted for studies and case reports describing GBS cases temporally associated with measles. Six case reports, two case series and two reviews were found for a total of 31 cases, most of which reported during the 1950s and 1960s, before the widespread introduction of measles vaccine (Table 1) [1–10]. One review involved a series of 911 patients with neurological complications following measles: polyradiculitis was reported in 10 cases (1.1 %) [2]. In another review, 11 (1.7 %) of 638 GBS cases associated with infections were found to have had measles as an antecedent infection [3]. In a series of 15 GBS cases, two (13 %) occurred following measles [8]. Of note amongst the case reports, one GBS case occurred in association with optic neuritis and PIME [9]. In conclusion, clinical, neurophysiological, and laboratory findings in the reported case support a diagnosis of measles-associated GBS. Other triggers for GBS were ruled out. Underreporting of measles complications by clinicians may partly account for the rarity of cases in the literature. The decreased incidence of measles and its complications since widespread use of measles vaccine may also contribute. No association between GBS with measles vaccination has ever been demonstrated [11]. The possible relationship between measles infection and GBS should be further documented in future cases of GBS temporally associated with measles and evaluated by appropriate studies.
123
References 1. Bourne IB, Scott RB (1952) Guillain-Barre´ syndrome. Report of a case following measles. Arch Pediatr 69(1):1–5 2. Miller HG, Station JB, Gibbons JL (1956) Parainfectious encephalomyelitis and related syndrome. Quart J Med 25(100): 427–505 3. Leneman F (1966) The Guillain-Barre´ syndrome; definition etiology and review of 1,100 cases. Arch Intern Med 118(2):139–144 4. Lidin-Janson G, Strannegard O (1972) Two cases of GuillainBarre´ syndrome and encephalitis after measles. Br Med J 2(5813):572 5. Dru¨eke TB, Pujade-Lauraine E, Poisson M et al (1976) Measles virus and Guillain-Barre´ syndrome during long-term hemodialysis. Am J Med 60(3):444–446 6. Gue`rin JM, Raux M, Lamotte M (1981) Syndrome de GuillainBarre´ apre`s rougeole. Sem Hop Paris 57:1215–1216 7. Nakamura A, Masayuki M, Hashimoto T et al (1993) A case of acute polyradiculoneuropathy following measles infection. Rinsho Shinkeigaku 33(5):565–567 (Japanese) 8. Murthy JM (1994) Guillain-Barre syndrome following specific viral infections—an appraisal. J Assoc Physicians India 42(1): 27–29 9. Tomiyasu K, Ishiyama M, Kato K et al (2009) Bilateral retrobulbar optic neuritis, Guillain-Barre´ syndrome and asymptomatic central white matter lesions following adult measles infection. Intern Med 48(5):377–381 10. Loomba A, Maertens P (2011) A 12-year-old female with measles-induced Guillain-Barre´ syndrome. J Investig Med 59(2): 383–384 11. da Silveira CM, Salisbury DM, de Quadros CA (1997) Measles vaccination and Guillain-Barre´ syndrome. Lancet 349(9044): 14–16
LETTER TO THE EDITOR
Guillain2Barre´ syndrome following measles infection: case report and review of the literature Antonietta Filia • Giuseppe Lauria
Received: 4 April 2014 / Accepted: 16 May 2014 Ó Springer-Verlag Italia 2014
Dear Editor, We report a case of Guillain-Barre´ syndrome (GBS) occurring after measles infection and review the published literature on the subject. A previously healthy 48-year-old man developed fever, cough and constipation, followed by a maculopapular rash over the face and neck. Measles infection was diagnosed. Four days later, he developed back pain and paraesthesias in the hands and feet. Computed tomography, magnetic resonance imaging scans of the brain and a chest radiograph were normal. One day later, he was admitted to hospital for numbness in the hands and feet bilaterally, dyspnoea, perioral pain, headache and nausea. The patient was afebrile and unable to walk and stand upright. He denied prior neurological illnesses. Examination showed weakness of the upper and lower extremities, more pronounced proximally, and symmetrical hypoesthesia. Deep tendon reflexes were absent. Nerve conduction studies showed moderate increase of motor nerve distal latencies, slowing of motor nerve conduction velocity, desynchronization of compound motor action potential amplitudes and reduced amplitude of sensory nerve action potential in the upper limbs. Serum measles IgM antibodies were elevated. Cerebrospinal fluid examination disclosed albumin-cytological dissociation
A. Filia (&) Infectious Diseases Epidemiology Unit, National Centre for Epidemiology, Surveillance and Health Promotion, Istituto Superiore di Sanita`, Viale Regina Elena 299, 00161 Rome, Italy e-mail: [email protected] G. Lauria Carlo Besta Neurological Institute, IRCCS Foundation, Milan, Italy
and elevated measles IgM antibody titres. Isoelectric focusing showed a mirror pattern. No paraprotein was found. Antibody titres to cytomegalovirus, rubella, Epstein-Barr virus and herpes simplex viruses 1 and 2 were negative. Anti-ganglioside antibodies were not measured. GBS of the demyelinating type was diagnosed and treatment with intravenous immunoglobulin was given. Over the following three days, the patient developed cranial nerve palsies, tetraplegia, and respiratory insufficiency necessitating admission to the Intensive Care Unit and a tracheotomy. Three weeks later he experienced severe cardiovascular autonomic dysautonomia which resolved within five days. The patient then underwent plasma exchange and began to improve clinically. He was discharged seven weeks after admission, with residual left 7th cranial nerve palsy, severe weakness of foot dorsiflexion, mild proximal muscle weakness of all four extremities, and mild neuropathic pain in the lower limbs. Measles is a vaccine-preventable, acute viral illness which is well-known to cause neurological complications, the most frequent being post-infectious measles encephalitis (PIME). The pathogenetic mechanisms involved in measles neurological complications remain to be completely understood. Interestingly, PIME appears to be caused by an abnormal immune reaction against myelin basic protein and the pathogenesis of GBS also seems to involve an autoimmune-mediated process. Approximately two-thirds of patients with GBS report a preceding infection but the pathogen frequently remains unidentified. Campylobacter jejuni, cytomegalovirus, Epstein-Barr virus, Mycoplasma pneumoniae have been shown to be related to GBS and there is some evidence that varicella-zoster and influenza viruses and H. influenzae may also be triggers.
123
Neurol Sci Table 1 Published studies and case reports describing cases of Guillain-Barre´ syndrome (GBS) following measles infection Reference
Type of study
Number of GBS cases with measles as antecedent infection
Patient age (years)
Time interval between onset of rash and neuropathic symptoms
Lab confirmation of measles
Bourne and Scott [1] Miller et al. [2]
Case report Review of 911 cases with neurological complications of measles Literature review of 638 GBS cases associated with infections Case series
1 10
5.5 years 5-21 years (range)
NS NS
11
NS
7 days 9 days on average (in four cases the interval was only 4-5 days) NS
2
9 years, 5.5 years
5 days (in both cases)
Yes
1 1 1 2 1 1
40 years 19 years 39 years 3 years, 5 years 28 years 12 years
Asymptomatic 15 days 6 days 12 and 10 days 4 days 4 weeks
Yes Yes Yes NS Yes Yes
Leneman [3]
Lidin-Jandon and Strannegard [4] Dru¨eke et al. [5] Guerin et al. [6] Nakamura et al. [7] Murthy [8] Tomiyasu et al. [9] Loomba A and Maertens [10]
Case Case Case Case Case Case
report report report series of 15 cases of GBS report report
No (clinical cases)
NS not specified
A literature review was conducted for studies and case reports describing GBS cases temporally associated with measles. Six case reports, two case series and two reviews were found for a total of 31 cases, most of which reported during the 1950s and 1960s, before the widespread introduction of measles vaccine (Table 1) [1–10]. One review involved a series of 911 patients with neurological complications following measles: polyradiculitis was reported in 10 cases (1.1 %) [2]. In another review, 11 (1.7 %) of 638 GBS cases associated with infections were found to have had measles as an antecedent infection [3]. In a series of 15 GBS cases, two (13 %) occurred following measles [8]. Of note amongst the case reports, one GBS case occurred in association with optic neuritis and PIME [9]. In conclusion, clinical, neurophysiological, and laboratory findings in the reported case support a diagnosis of measles-associated GBS. Other triggers for GBS were ruled out. Underreporting of measles complications by clinicians may partly account for the rarity of cases in the literature. The decreased incidence of measles and its complications since widespread use of measles vaccine may also contribute. No association between GBS with measles vaccination has ever been demonstrated [11]. The possible relationship between measles infection and GBS should be further documented in future cases of GBS temporally associated with measles and evaluated by appropriate studies.
123
References 1. Bourne IB, Scott RB (1952) Guillain-Barre´ syndrome. Report of a case following measles. Arch Pediatr 69(1):1–5 2. Miller HG, Station JB, Gibbons JL (1956) Parainfectious encephalomyelitis and related syndrome. Quart J Med 25(100): 427–505 3. Leneman F (1966) The Guillain-Barre´ syndrome; definition etiology and review of 1,100 cases. Arch Intern Med 118(2):139–144 4. Lidin-Janson G, Strannegard O (1972) Two cases of GuillainBarre´ syndrome and encephalitis after measles. Br Med J 2(5813):572 5. Dru¨eke TB, Pujade-Lauraine E, Poisson M et al (1976) Measles virus and Guillain-Barre´ syndrome during long-term hemodialysis. Am J Med 60(3):444–446 6. Gue`rin JM, Raux M, Lamotte M (1981) Syndrome de GuillainBarre´ apre`s rougeole. Sem Hop Paris 57:1215–1216 7. Nakamura A, Masayuki M, Hashimoto T et al (1993) A case of acute polyradiculoneuropathy following measles infection. Rinsho Shinkeigaku 33(5):565–567 (Japanese) 8. Murthy JM (1994) Guillain-Barre syndrome following specific viral infections—an appraisal. J Assoc Physicians India 42(1): 27–29 9. Tomiyasu K, Ishiyama M, Kato K et al (2009) Bilateral retrobulbar optic neuritis, Guillain-Barre´ syndrome and asymptomatic central white matter lesions following adult measles infection. Intern Med 48(5):377–381 10. Loomba A, Maertens P (2011) A 12-year-old female with measles-induced Guillain-Barre´ syndrome. J Investig Med 59(2): 383–384 11. da Silveira CM, Salisbury DM, de Quadros CA (1997) Measles vaccination and Guillain-Barre´ syndrome. Lancet 349(9044): 14–16
