Guillain–Barré Syndrome in India: Population-based validation of the Brighton criteria
Highlights
► We assess the sensitivity of case definitions for GBS in children in India. ► All cases had cerebrospinal fluid and nerve conduction studies. ► Diagnostic sensitivity was high: Brighton criteria levels 3 (86%), 2 (82%), and 1 (62%). ► Brighton Working Group case definitions are a plausible standard for capturing GBS. ► These case definitions may be valuable for diagnosis in other low income settings.
Introduction
Guillain–Barré Syndrome (GBS) is an acute, immune-mediated polyradiculoneuropathy and an important cause of acute flaccid paralysis (AFP) worldwide [1]. The number of cases of GBS increased following vaccination with the A76NJ 1976 influenza vaccine [2], [3], leading to heightened awareness of GBS. There is a need for a valid, reliable, practical, and global consensus definition of GBS at various levels of certainty especially when new vaccines are developed and provided to large populations [4]. Several case definitions of GBS existed prior to the 2009 H1N1 vaccination campaign including the widely employed Asbury–Cornblath case definitions, which involve ancillary diagnostic tests [5], [6]. More recently, the Brighton Collaboration developed case definitions of GBS with differing levels of diagnostic certainty [7]. These case definitions have undergone limited field testing, particularly in resource-poor settings.
GBS may be especially difficult to diagnose in resource-poor settings. Since the Brighton criteria include purely clinical case definitions as well as categories requiring further specialized testing, the need for additional resources to achieve diagnostic certainty for GBS could be challenging for large resource-poor populations.
In India, there is limited information available on the relative burden of GBS, particularly at a population level. Small case series of patients with GBS in India suggest that it may be an important cause of non-poliomyelitis AFP, including fatal AFP [8], [9], [10]. It is unknown whether the axonal subgroups, mainly acute motor axonal neuropathy (AMAN), are more common than the acute inflammatory demyelinating polyneuropathy (AIDP) form, as has been seen in other developing countries [11], [12], [13]. Based on nation-wide active surveillance data of AFP established by the global polio eradication initiative in India since 1997, we present a population-based cohort study of GBS in Indian children. We answer the following questions: (1) what are the epidemiological, clinical, laboratory, and electrophysiologic features of a subset of Indian children with GBS; and (2) what is the sensitivity of Brighton diagnostic criteria for GBS in children in India?
Section snippets
Epidemiological surveillance
The National Polio Surveillance Unit (NPSU) of India collects all cases of acute flaccid paralysis (AFP) in children <15 years (1997 to present) [14]. The system relies on weekly active surveillance in key health facilities (down to district hospitals) and passive reporting of AFP cases, immediate case investigations with collection of stool samples for virologic studies, and weekly active surveillance in key health facilities (down to district hospitals). AFP detection includes notification of
Results
Between January 2002 and December 2003, a total of 18,213 AFP cases were reported. Of these, 2587 inadequate cases were reviewed by the Expert Committee in India and 832 (32.2%) were categorized as GBS (Brighton Level 4) (Fig. 1). 745 case files (745/832, 89.5%) were available and reviewed in detail for this study. Of these, 27 cases were discarded because of alternative diagnoses, including diphtheria, paralytic rabies, and transverse myelitis. Of the 718 remaining files, there were 79 (11%)
Discussion
The diagnosis of GBS can be confirmed in low income countries using Brighton Working Group criteria with moderate to high sensitivity. The majority of clinically diagnosed GBS cases (86%) met the basic clinical definition of GBS (Brighton level 3) which requires neither CSF nor NCS analyses. There is little difference, and potentially no difference, between the sensitivity of Brighton levels 2 and 3 in this cohort (84% vs. 86%, p > 0.05).
Although a majority of cases met the level 3 diagnostic
Acknowledgements
The American Academy of Neurology (AAN) had no role in the design and conduct of the study; collection, management, analysis, or interpretation of the data; or preparation, review, or approval of the manuscript.
Conflict of interest statement: All authors declare no conflicts of interest. Funding: Dr. Mateen is supported by the 2010 Practice Research Fellowship Grant from the American Academy of Neurology. Dr. Cornblath has served as Consultant for Merck, Pfizer, Mitshubishi Pharma,
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