Acute Flaccid Myelitis of Unknown Etiology in California, 2012-2015

Haren, Keith Van; Ayscue, Patrick; Waubant, Emmanuelle; Clayton, Anna Marie; Sheriff, Heather; Yagi, Shigeo; Glenn-Finer, Rose; Padilla, Tasha; Strober, Jonathan B.; Aldrovandi, Grace M.; Wadford, Debra A.; Chiu, Charles Y.; Xia, Dongxiang; Harriman, Kathleen; Watt, James; Glaser, Carol

doi:10.1001/jama.2015.17275

Cited by 127 publications

(207 citation statements)

References 29 publications

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“…6–8 CSF glucose was normal in all except 3 reported CDC cases. 6–8 Oligoclonal bands and anti–aquaporin 4 antibodies were negative in tested patients. 6,7 …”

Section: Reviewmentioning

confidence: 84%

“…Altered mental status was absent in the CHCO cohort, but present in 22% of CDPH cases and 11% of CDC cases. 6,8 Bowel or bladder dysfunction was present in 18% of the PCH and 51% of the CDPH cohorts, but absent in the CHCO cohort. 6,10 …”

Section: Reviewmentioning

confidence: 95%

“…6 Most patients reported clinical improvement in their preceding illness before return of fever accompanied by headaches, stiff neck, or pain in the neck, back, or affected limb around the time of neurological deficit onset. 6,7 …”

Section: Reviewmentioning

confidence: 99%

“…7,10 In contrast, CDC surveillance reported numbness in affected limbs in 24% of patients and CDPH surveillance reported that 44% of patients had sensory deficits: 31% with pain/temperature deficits and 22% with fine touch/vibration deficits. 6,8 …”

Section: Reviewmentioning

confidence: 99%

“…Though not included in the case definition, cranial nerve dysfunction was noted commonly among AFM cases from CDPH and CDC surveillance. 6,8 In the CHCO and PCH clusters, 3 children had cranial nerve dysfunction without the associated limb weakness required to meet CDC AFM case definition. 7,10 Hypophonia, dysarthria, and dysphagia consistent with CN IX and X dysfunction; diplopia attributed to cranial nerve VI dysfunction; and facial weakness attributed to cranial nerve VII dysfunction were most common.…”

Section: Reviewmentioning

confidence: 99%

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Acute flaccid myelitis: A clinical review of US cases 2012–2015

Messacar

Schreiner

Haren

et al. 2016

Annals of Neurology

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This review highlights clinical features of the increasing cases of acute flaccid paralysis associated with anterior myelitis noted in the United States from 2012 to 2015. Acute flaccid myelitis refers to acute flaccid limb weakness with spinal cord gray matter lesions on imaging or evidence of spinal cord motor neuron injury on electrodiagnostic testing. Although some individuals demonstrated improvement in motor weakness and functional deficits, most have residual weakness a year or more after onset. Epidemiological evidence and biological plausibility support an association between enterovirus D68 and the recent increase in acute flaccid myelitis cases in the United States.

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“…6–8 CSF glucose was normal in all except 3 reported CDC cases. 6–8 Oligoclonal bands and anti–aquaporin 4 antibodies were negative in tested patients. 6,7 …”

Section: Reviewmentioning

confidence: 84%

Section: Reviewmentioning

confidence: 95%

Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

Section: Reviewmentioning

confidence: 99%

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Acute flaccid myelitis: A clinical review of US cases 2012–2015

Messacar

Schreiner

Haren

et al. 2016

Annals of Neurology

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Clinical Subpopulations in a Sample of North American Children Diagnosed With Acute Flaccid Myelitis, 2012-2016

et al. 2019

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IMPORTANCE Acute flaccid myelitis (AFM) is an emerging poliolike illness of children whose clinical spectrum and associated pathogens are only partially described. The case definition is intentionally encompassing for epidemiologic surveillance to capture all potential AFM cases. Defining a restrictive, homogenous subpopulation may aid our understanding of this emerging disease. OBJECTIVE To evaluate the extent to which the US Centers for Disease Control and Prevention (CDC) case definition of AFM incorporates possible alternative diagnoses and to assess the plausibility of a case definition that enriches the biological homogeneity of AFM for inclusion in research studies. DESIGN, SETTING, AND PARTICIPANTS Retrospective case analysis of children younger than 18 years diagnosed as having AFM between 2012 and 2016 using the CDC case definition. Group 1 included patients recruited from the United States and Canada based on the CDC case definition of AFM. Group 2 included patients referred to the Johns Hopkins Transverse Myelitis Center for evaluation of suspected AFM. Patients' records and imaging data were critically reviewed by 3 neurologists to identify those cases with definable alternative diagnoses, and the remaining patients were categorized as having restrictively defined AFM (rAFM). Clinical characteristics were compared between patients with rAFM (cases) and those with alternative diagnoses, and a case description distinguishing these AFM groups was identified. Interrater reliability of this description was confirmed for a subset of cases by a fourth neurologist. Data were analyzed between May 2017 and November 2018. MAIN OUTCOMES AND MEASURES Proportion of patients with possible alternative diagnosis. RESULTS Of the 45 patients who met the CDC AFM case definition and were included, the mean age was 6.1 years; 27 were boys (60%); and 37 were white (82%), 3 were Asian (7%), 1 was Hispanic (2%), and 4 were mixed race/ethnicity (9%). Of the included patients, 34 were classified as having rAFM, and 11 had alternate diagnoses (including transverse myelitis, other demyelinating syndromes, spinal cord stroke, Guillain-Barre syndrome, Chiari I myelopathy, and meningitis). Factors differing between groups were primarily asymmetry of weakness, lower motor neuron signs, preceding viral syndrome, symptoms evolving over hours to days, absence of sensory deficits, and magnetic resonance imaging findings. A case description was able to reliably define the rAFM group. CONCLUSIONS AND RELEVANCE We present an approach for defining a homogeneous research population that may more accurately reflect the pathogenesis of the prototypical poliomyelitis-like subgroup of AFM. The definition of rAFM forms a blueprint for inclusion criteria in future research efforts, but more work is required for refinement and external validation.

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A Parental Perspective on Strengthening Knowledge After Acute Flaccid Myelitis

2019

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We write as nurses, advocates, physicians, and most importantly as parents who are joined through our children's experience with acute flaccid myelitis (AFM) in the United States in the last decade. In 2012, neurologists in California reportedtotheCentersforDiseaseControlandPrevention (CDC)astrangeclusterofnon-polio-associatedAFMcases, characterized by virus-associated injury to the motor neurons in the spinal cord. 1 At the time, the CDC felt that these cases did not exceed the very low expected background rate of AFM associated with nonpolio viruses. Certainly, more than 60 years ago, parents feared the warm summer months because they carried the risk of infection by the polio enterovirus. But thanks to the 1953 discovery of a polio vaccine, poliomyelitis rates plummeted. The affected children aged. As a consequence, most physicians, nurses, and physical therapists trained in the United States have never seen a case of pediatric AFM.Against this backdrop of absent training in medical personnel and a sluggish response from the CDC, one of us (E.O.) created a private Facebook group (called Parents of Children With Polio Like Syndrome/Acute Flaccid Myelitis) for parents navigating this rare condition with little guidance.Wehavereadeverypost,sharedinformationgleaned from AFM specialists (many of our clinicians had little access to this expertise), celebrated incremental and enormous gains in some of our children, and grieved together as 2 children died of complications of their paralysis. We have watched in horror as the group expanded to include members from other continents and exceeded 500 members this year. Here, we would like to share 3 observations with readers who, as pediatricians, are at the forefront of detecting, reporting, and caring for new cases.First,wedreadlatesummer.Wegrievethosefinaldays whenourstill-normalchildrenclimbedtheirlastplayground structuresortooktheirlastrunsdowntheblockortheirlast independent breaths. We relive the trauma of the hospitalization, when at night we kissed our brave children, not knowing whether they would awaken stable, intubated, or unable to ever walk again. In addition, we fear the coming wave of new bewildered parents to join the site, who will spend sleepless months away from work and their other children,battlingfortheirchildren'srecovery.In2013,2015, and 2017, we were lulled by the relative quiescence in AFM cases and hoped that the risk had passed. Lulls were followed by spikes in incidence in 2014, 2016, and 2018. The risk has not passed, and the cause remains elusive.

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Acute Flaccid Myelitis of Unknown Etiology in California, 2012-2015

Cited by 127 publications

References 29 publications

Acute flaccid myelitis: A clinical review of US cases 2012–2015

Acute flaccid myelitis: A clinical review of US cases 2012–2015

Clinical Subpopulations in a Sample of North American Children Diagnosed With Acute Flaccid Myelitis, 2012-2016

A Parental Perspective on Strengthening Knowledge After Acute Flaccid Myelitis

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