Li-Ching Lee Reaches Out
Bringing the latest, greatest tools in autism diagnosis and treatment to the whole world.
On a torrentially rainy day, the Bloomberg School’s Li-Ching Lee and some colleagues paid a visit to Rangpur Community Medical College Hospital in northwest rural Bangladesh.
The researchers were there in Bangladesh to adapt standard autism diagnostic tools, which had been developed and used primarily in affluent, Western countries, for an autism epidemiologic study in the region of Gaibandha. Ethics rules for most American-led research require that anyone identified with a medical condition through a study is at least referred to treatment—that’s why the researchers wanted to tour the hospital nearest to the study site that offers care for developmental disorders.
As they approached the facility, hundreds of patients and family members milled around, outside, and in—along with goats, dogs and other animals—because there was no door. With limited electricity, the inside of the hospital was dark. The windows, which should have offered at least a little light, were covered with layers of dust.
“When resources are scarce, survival often is a priority over developmental disorders, like autism,” says Lee, PhD, ScM ’97, associate director for Global Autism at the Wendy Klag Center for Autism and Developmental Disabilities. “Saving lives from starvation and preventing children from dying of diarrhea are a higher priority than knowing what autism is.”
But for nearly a decade and a half, Lee’s work has revolved around trying to get people to know and care about autism spectrum disorder (ASD) in places where most people have not heard of it—those communities that are socioeconomically disadvantaged, which cover the vast majority of the planet—and on getting children who have ASD the help they need. “This is also a human rights issue when you know there are treatments available to improve long-term outcomes but they aren’t available to families who need them the most,” Lee says.
Lee is no stranger to poverty herself. Raised in rural Taiwan, she remembers picking fruits and vegetables growing wild on mountain hills for sustenance and wondering where her family’s next meal might come from.
That’s a major reason why, after finishing her Master of Science degree at the Bloomberg School (then called the Johns Hopkins School of Hygiene and Public Health) in 1997, Lee knew that she wanted to focus on helping disadvantaged populations. After wrapping up her PhD from the School of Public Health at University of North Carolina-Chapel Hill in 2003, she was quickly drawn to ASD research.
By the early 2000s, ASD had begun to attract significantly more attention than it had in the past in the U.S., U.K., Canada, Australia and other Western countries. However, in low- and middle-income countries (LMICs), fewer researchers focused on ASD, a disorder that is characterized by problems with social interactions and communication, and by a tendency to engage in restricted-interest and/or repetitive behaviors. As recently as 2015, 86.5 percent of ASD cases identified in epidemiological studies came from North America, Europe and Japan, but only 10 percent of the world’s children actually live in these areas, according to a study in Autism Research. The global autism research community has grown since Lee started her career in the field, but gains in research funding have been minimal and she remains one of the few researchers worldwide who specializes in this niche.
In affluent nations, there are typically a multitude of resources available to families with a diagnosed child—at least, for those willing (and able) to afford the costs. Estimates from a study published in 2014 by Harvard researchers, extracting data from the 2011 National Health Interview Survey, suggest that families in the U.S. who have a child with ASD pay an extra $17,000 per year for the child’s health care and education. That doesn’t include the cost of behavioral interventions, which aren’t typically covered by insurance and can tack on tens of thousands of dollars more.
“That’s outrageous for many families,” Lee says. But as extreme as these costs are for American families, she adds, they would be a cruel impossibility for families in LMICs, where many people struggle to afford a single meal every day.
Even diagnosing autism is a financial impracticality in these countries, Lee explains, since being able to definitively diagnose ASD requires standardized tools backed by scientific consensus. The two tools most commonly used, usually in combination, are the Autism Diagnostic Interview-Revised (ADI-R), a caregiver interview, and the Autism Diagnostic Observation Scheduled-2 (ADOS-2), an intensive, one-on-one assessment that a trained expert administers to a child. Both tests, considered gold standards in the field for gathering information to make an ASD diagnosis, are copyrighted by the same publisher. Changing the tools to make them more culturally relevant—or even just translating them into a different language—can cost hundreds of thousands of dollars.
Not only are the tools pricey, but the publisher imposes rules that make purchases from LMICs nearly impossible. Research training for enough local clinicians to administer these assessments, as well as maintaining quality in administering the tools and generating meaningful prevalence and research data, can cost millions of dollars.
The need for tools to diagnose and treat ASD is clear to Lee with each individual with ASD she meets in these low-resource settings.
Lee cites the example of one 32-year-old woman who had been misdiagnosed with schizophrenia her entire life because her doctors weren’t aware of ASD. When Lee met her in Asia, the woman was drugged with medicines that wouldn’t help and locked alone in an “observation room” because caregivers didn’t know how to handle her violent outbursts. In a different case, parents of a girl with ASD told Lee that they allowed and “encouraged” their daughter to play near a busy road in hopes that an accident would ease their burden. Another parent that Lee met was covered with bruises because her husband beat her every day, blaming her for their son’s challenging behaviors.
All these experiences are not unusual in low-resource settings around the globe. “Families who have a member with autism often feel desperate, hopeless and helpless, particularly when they don’t have knowledge and resources,” Lee says.
In 2003, soon after Lee joined the Bloomberg School as an assistant scientist, she and her colleagues started studying ASD in China, the country with the world’s largest population, where few resources have been devoted to helping those with developmental disorders or even to determining their prevalence.
With NIH funding, they began by adapting screening tools that are widely used as standard practice in high-income, English-speaking countries to Mandarin Chinese to cast a wide net for finding possible ASD cases. By 2007, they’d completed the years-long process of translating ADI-R and ADOS—tools that can help researchers form a more definitive diagnosis after a positive hit on initial screening tests—to Mandarin Chinese and validating these tests in Chinese populations. In 2007, they launched a similar study in Taiwan and completed it in 2011.
Building on that work, Lee took the next step: sharing interventions with families whose children had been diagnosed with ASD. In 2012, with support from Autism Speaks, Lee and her team launched a pilot project of parent-mediated intervention based on pivotal response therapy (PRT) for children who had been diagnosed with ASD in southern Taiwan.
PRT is a behavioral intervention most often applied in a “naturalistic” setting where a child’s daily life takes place (such as home), Lee explains, giving it advantages over other center-based interventions for low-resource settings. The team trains parents to implement behavioral intervention techniques such as knowing when to give attention and when to ignore certain behaviors. To help encourage positive behaviors, parents might reward the child with praise or incentives when such behaviors occur—like providing a ball after a child asks for it by name—or at least makes an attempt. For disruptive behaviors, such as tantrums, parents might turn away after making sure a child is safe or learn strategies to avoid triggers in the first place.
In 2013, Lee collaborated with Bloomberg School professors Parul Christian, DrPH ’96, MSc, MPH ’92, and Keith West, DrPH ’86, MPH ’79, in the Department of International Health to launch a similar ASD epidemiologic study led by Christian and funded by Autism Speaks in Gaibandha, Bangladesh, where the Bloomberg School has had a presence since 1999. Through a program called JiVitA, the School’s Center for Human Nutrition began work in Gaibandha to study the effects of micronutrient deficiencies in outcomes for mothers, infants and children. JiVitA has since expanded its scope to cover many other areas of maternal and child health, with about 800 members of both Johns Hopkins and local staff.
Piggybacking on this mission, the researchers began the arduous effort to adopt and adapt ADI-R and ADOS-2 to be used in the country. From Lee’s experience in China and Taiwan, they already had an idea of the challenges they might face in making these assessments culturally relevant and acceptable.
Consider a task where the ADOS-2 administrator asks a child to show how he or she brushes their teeth, notes Susan Risi, PhD, a clinical psychologist who specializes in autism diagnosis. In the American version, examiners might expect a child to show them how he stands at a sink, turns faucet handles, puts toothpaste on toothbrush, uses the toothbrush to brush his teeth and then rinses his mouth. However, says Risi, most villagers in Gaibandha have to trek to a community well to access water, if they’re lucky enough to have one around. Most of the children don’t even own a toothbrush, adds Lee.
“Even though the concept of cleaning their teeth isn’t totally unfamiliar, the context of it is different in this community,” Risi says. “The sink situation is not relevant where there are no sinks. The usual idea of brushing your teeth at a sink is unfamiliar, so a child might not be able to show us how they brush their teeth and thereby do poorly on that task.”
The ADOS-2 also has several pictures that aren’t culturally relevant or appropriate, Risi adds. For example, several tests involve explaining scenarios in pictures—some of which involve pigs, a symbol of uncleanliness that many people in majority-Muslim countries like Bangladesh don’t want to look at. One picture features people frolicking at a beach dressed in bikinis and other swimsuits, adds Lee, which would be very inappropriate for many in conservative cultures.
Another challenge in assessment, Lee says, is how to quantify cultural difference. For example, an oft-reported early sign for ASD is delayed speech. However, in some cultures, it’s considered a desirable feature for young girls to be quiet and reserved. “What’s a red flag in some cultures is a positive in others,” she says. “We definitely can’t discount the effect of cultural influences on observed behaviors.”
IMPLEMENTATION, THEN EVACUATION
With all this in mind, the JiVitA team embarked on its project, screening thousands of families in Gaibandha and using a trained local clinician, Hafizur Rahman, to administer the ADI-R and ADOS-2 to children whose screening tests suggested a higher probability of ASD. The team’s plan was mainly to adapt the screening and diagnostic tools in this population and to help ascertain the prevalence of ASD in this region. There was no grant money left to pursue intervention—at least, that’s what Jasmine Blake, MSPH ’16, was told when she met with Lee in November of 2014.
Blake, who had worked for almost a year as an applied behavioral analysis therapist with children with autism in Maryland and is now a medical student at the University of Maryland School of Medicine, was curious about Lee’s work in global autism and wanted to help however she could. “I thought it would be great to analyze data or do some statistics on the data from the Bangladesh epidemiologic study. I wasn’t really thinking that I’d be implementing anything,” she remembers.
But within weeks, Lee suggested that they both should apply for grants from different centers at Johns Hopkins—Blake for a student travel award from the Johns Hopkins Center for Global Health and Lee for a faculty award from the Bloomberg School’s Wendy Klag Center—for an intervention project in Gaibandha. After both grants were awarded in early 2015, Blake, along with Eric Rubenstein, ScM ’14, and Peng-Chou Tsai, MD, MPH ’13, headed west to learn about PRT strategies from experts at San Diego State University and the University of California, San Diego. Their goal: Develop a program based on the implementation study Lee’s team had performed for low-resource and under-served families in Taiwan.
Taking advantage of Rahman’s rapport with the Gaibandha community, Blake and the team decided to use a “train the trainer” approach to instruct Rahman in how to coach parents on implementing the behavioral intervention. By September 2015, all was going swimmingly—Rahman’s training was proceeding without a hitch, 10 families with a child with ASD or other developmental disabilities between the ages of 8 and 10 had been selected for the pilot intervention project, and plans were underway for an initial group education seminar with all the parents. Then in October, a series of murders of foreigners culminated in the killing of a Japanese aid worker in a nearby district. Blake and several Hopkins colleagues were asked to evacuate.
“I was crushed,” Blake remembers. “I did not expect to fall in love with Bangladesh as much as I did, and I was heartbroken about not being able to give help to families who desperately needed it.”
HELPING FAMILIES THRIVE
She and her colleagues weren’t sure whether the project would ever be able to restart. But in April 2016, the U.S. Department of State removed the strongest travel restrictions, making it possible for Blake and her colleagues to return. Back in Bangladesh, they worked double time to make sure they’d be able to carry out a modified version of the intervention with limited travels and shortened training time (due to safety concerns) before the grant funding ran out. When parents and other relatives of the 10 children showed up for training, there were many fathers in the group. This was a surprise, based on the culture’s women-centered model of childcare, and a testament to the strong desire these families had for much-needed help in raising a child with autism or developmental disabilities, Blake says.
After a few days, the same families came back for individualized training on how to handle their child’s most challenging behaviors—from violent outbursts to a lack of simple communication. The researchers weren’t sure how involved parents would be after the initial group session. But when Rahman asked parents if they’d been practicing the techniques, most said that they had, multiple times every day.
The effects, parents said, were life-changing: A mother whose child had terrible tantrums at the market learned how to avoid the busiest times that would trigger a sensory overload and to utilize a “first, then” schedule—a technique that provides a child with a sense of what’s coming next. This allowed the mother to shop in relative peace. Other parents, frustrated with their daughter’s lack of communication, learned to offer her rewards and patience while she made attempts to communicate, leading to a significant boost in the girl’s vocabulary.
“So many parents overwhelmingly responded that they wished they’d known these techniques sooner,” Blake says. “ASD presents at an early age. These parents have known their child was different for a long time, but they didn’t know what to do.”
Although the project was small, Lee says, it’s an indicator for what’s possible with more time and more resources, fueling her drive to help even more families around the world. No funding currently exists to scale up their efforts in Bangladesh or any other LMIC. But Lee hopes the value of intervention and the knowledge gained from these initial studies will spur foundations and government agencies to support ASD work.
Eventually, she says, she has no doubt that researchers will find the cause or causes of ASD. But in the meantime, being able to give families the tools they need to thrive amid an ASD diagnosis—particularly in places with few or no existing ASD resources—will be key to helping children with a diagnosis fit in and eventually contribute to their communities.
“Parents’ love for their children is not different because of where they live or how many resources they have,” Lee says. “People with autism should be able to benefit from the growing body of knowledge and available treatment for this condition, no matter where and who they are. That is a human right.”