From The Guardian, 14 May 2010 (Story by Sarah Boseley)
Kay Gilderdale helped her 31-year-old daughter to kill herself over the course of one long December night, crushing up sleeping pills and antidepressants when the morphine overdose she gave her to inject did not immediately work. It's almost incredible to think that a mother and daughter could be driven to such hellish extremes by a disease that is not fatal. Lynn Gilderdale had ME.
But Lynn's extraordinary and distressing story takes few people acquainted with ME (myalgic encephalopathy), also known as CFS (chronic fatigue syndrome), by surprise. If nothing else, it illustrates the despair that ME/CFS engenders. An estimated 250,000 people have the condition in this country, of whom 25,000 are children (Lynn became ill at 14). Most struggle to get a diagnosis, many are unhappy with the limited treatments available and all want to know what has caused them to be afflicted with this most miserable of illnesses, which saps their energy, wrecks their lives and leaves some like Lynn bedridden and tube-fed.
Last autumn, it suddenly looked as though they were going to get an answer. A paper was published in the highly regarded journal Science by the Whittemore Peterson Institute (WPI) in Reno, Nevada, a research establishment set up by Annette Whittemore, the wife of a millionaire who had made money in property. Their daughter, Andrea, developed ME/CFS when she was 11. Whittemore, searching for help for her daughter, met Daniel Peterson, a general practitioner who brought to light one of the earliest ME clusters more than 30 years ago, in Incline Village on the north shore of Lake Tahoe in Nevada, where he practised medicine. The two of them launched the WPI as a research centre dedicated to finding answers, and treatments for ME/CFS.
Peterson is one of many who believe the disease probably has a viral trigger. There is evidence that it can follow a viral infection, such as glandular fever. He put Andrea Whittemore on an experimental antiviral drug, which her mother has said has led to improvement.
Some cancers – cervical cancer is the best example – can also be triggered by a virus. In Incline Village, Peterson reported he had found a surprising number of rare cancers called mantle cell lymphoma, a form of non-Hodgkin's lymphoma, among his ME/CFS patients. That attracted the attention of a cancer researcher, Dr Judy Mikovits. She joined WPI and went to work to look for a viral trigger for the cancer cluster.
Mikovits was soon reporting that she had found high levels of viruses in the ME/CFS patients by comparison. And then, in the Science paper in October, came the revelation that rocked the ME/CFS community as well as scientists around the world. Mikovits, with Vincent Lombardi and other colleagues from the WPI, reported that they had found a recently discovered retrovirus called XMRV in the blood cells of 68 out of 101 ME/CFS patients they had tested.
The implications were huge. If the virus were proven to be the cause of ME/CFS, then treatment could not be far away. It would also, in the eyes of many of the angriest campaigners, put paid to arguments that ME/CFS is predominanantly a psychosocial disorder – a theory they abhor and which has led to extraordinary invective, denunciations and bitterness, especially on the internet.
Annette Whittemore was certainly convinced her institute had found the biological key to the disease that cut down her “beautiful daughter who has been ill for 20 years in spite of our best efforts”. Speaking to the CFS advisory committee of the US government's Department for Health and Human Sciences in late October, she said: “It ends the debate. CFS is not and never was a psychological disorder. Those who are ill have always known this. The physicians who take care of them have always known this and finally those who have attempted to keep patients from receiving medical care for this disease know this.”
She and Mikovits were delighted that suddenly scientists from all over the world were homing in on their research. Certainly they had aroused great excitement and curiosity. But there was also a major public health issue here. HIV is a retrovirus. A retrovirus can be passed from one person to another in blood and semen. If XMRV, first associated with prostate cancer, is truly linked to ME/CFS, then we need to know about it fast, to prevent it spreading.
Retrovirologists were instantly on the case, attempting to replicate the WPI's findings. Three teams, two in the UK and one in the Netherlands, have already reported their results. All of them have drawn a blank.
Many people within the ME/CFS community refuse to believe it. They say the scientists have not properly replicated the WPI work. It's the line the WPI takes too, adamant that it has made a breakthrough. Richard Simpson of Invest in ME, which runs a major annual conference at which Whittemore will be a speaker this year, says the negative studies were too rushed.
He points out that Science took six months to review the WPI paper before publishing and that the research had support from the National Cancer Institute and the Cleveland Clinic, one of America's top hospitals.
“These other studies are not replications. They haven't used the same methodological approach. In any area of medicine you have to go back to the original tests and do it in the same way.”
Simpson says he does not assume XMRV is the cause of the illness, but he and many others are deeply suspicious of what they believe are the attempts of scientists to dismiss once more the claims of ME/CFS to be considered a biomedical condition. They point out that it was classified as a neurological disease rather than a psychological illness by the World Health Organisation in 1969.
Listen to some – or read the internet – and you would think there is a massive organised conspiracy going on, led by the psychiatric community, but in conjunction with insurance companies and even government, to prove ME has no physical cause. (There are genuinely distressing stories about the failure of the Department for Work and Pensions to recognise that people with ME/CFS can be incapable of work, depriving them of sickness benefit.)
Nice, the National Institute for Healthcare and Clinical Excellence, has come under fire too for recognising only psychosocial treatments, in the shape of cognitive behaviour therapy and graded exercise programmes. They were taken to judicial review in 2009 by a group of people who claimed the experts who drew up their guidance were biased or had conflicts of interest. But Mr Justice Simon dismissed the case and took the unusual step of making a direct attack on those who brought it. “Unfounded as they were, the allegations were damaging to those against whom they were made and were such as may cause health professionals to hesitate before they involve themselves in this area of medicine,” he said in an afterword to his judgment.
Most of the internet vitriol is directed at psychiatrist Professor Simon Wessely from King's College London, who believes there may be viral triggers for the disease but who pioneered the psychosocial therapies, ran the studies on which Nice's guidance is based and started the first NHS treatment unit. One of the reasons why the first UK study to fail to find XMRV in patients was denounced by ME/CFS activists is that it was co-authored by Wessely.
The other UK study is authored by a collection of top scientists who have either never been involved with the ME/CFS community before or who have in the past enjoyed its approval. Among them is Jonathan Kerr from St George's, University of London, who has been looking for genetic clues to the disease and is actually collaborating on a different project with the WPI.
Their study was published in a deliberately low-key way in Retrovirology. The researchers studied 170 blood samples from two separate groups of patients. Kate Bishop, a leading retrovirologist from the National Institute for Medical Research, said they tried hard to find XMRV but failed.
“I feel very sorry in a way,” she says. “We were hoping it was true. One of the things patients don't seem to realise is that it is not in our interests to find a negative result either, but I do understand their frustration.”
She thinks there are few differences between the US and European patients, except possibly that the WPI cases all came from geographical clusters.
What the European labs do not want to do is to take samples from the WPI to test. One possibility they cannot discount is that the WPI samples are contaminated with the virus, although the institute insists this is not so. Nonetheless, the European labs would not want to risk contamination.
On the day of the publication of the Science paper, the WPI declared it had now found XMRV in 95% of blood samples from ME/CFS patients it had tested – an extraordinarily high result. “This finding clearly points to the retrovirus as a significant contributing factor in this illness,” said Mikovits in a statement on 8 October. But now the first euphoric blaze of publicity is over, the WPI appears to have put up the barricades. Its press officer said Mikovits was not giving interviews and asked for a list of questions. The questions went unanswered.
Meanwhile, the WPI has licensed the test it used to detect XMRV to a lab in Reno called Viral Immune Pathology Diagnostics (VIP Dx), charging $450 a time. Dr Vincent Lombardi, first author of the Science paper, was made director of operations for the licensing and development of the test. WPI points out that profits it makes are ploughed back into research, critics question the promotion of a test when scientists have not conclusively been able to show XMRV is a cause of ME/CFS and there is no treatment even if it is.
But patients are queuing up. Forty or 50 have been tested from the UK so far, and a substantial number have received a positive result. On an internet chat site, the comments are positive. “It must be a relief to finally know what has caused all this misery and stolen precious years,” says one contributor.
Many doctors and scientists who work in the ME/CFS area are not willing to talk about it. However, one clinician who treats patients – and who was part of the Nice guideline group – put his head above the parapet after Kay Gilderdale was acquitted at Lewes crown court of attempted murder. Alastair Santhouse, consultant in psychological medicine at the South London and Maudsley NHS Foundation Trust, was deeply concerned by much of the press coverage, which depicted ME/CFS as a terminal illness and wrote to say so in the British Medical Journal.
“It was being talked about in terms of the euthanasia/assisted suicide debate,” he said. “It is an awful illness – chronic, unpleasant and very isolating – but it is not a terminal illness. There are treatments available and they are not perfect but we as a profession should not be giving up on people.
“The patients I see in the clinic, generally speaking, are extremely relieved and pleased to find someone who is taking an interest in their case. They tend not to have any particular ideological position on the illness, but they just want to get better.”
Santhouse is offering cognitive behavioural therapy (CBT) and graded exercise therapy, in line with the Nice guidance. “They don't work for everyone. Doctors have enough humility to realise they are not the complete answer.” He doesn't pretend to know the cause but, he says, citing the historic treatments of foxglove for dropsy and quinine for malaria, “you don't always need to know the cause of something to be able to treat it”.
Interestingly, in this deeply traumatised and divided field, there is some consensus. Everybody agrees ME/CFS is a terrible illness that seriously damages the lives of entire families. Nobody claims to know the definitive cause. Everybody agrees more research is needed – although conspiracy theorists say biomedical research proposals are blocked by the Medical Research Council, while others say those put forward are just not of sufficiently high quality. And the patient groups agree that the inflammatory invective on the internet gets nobody anywhere. “From our point of view it is not productive,” says Simpson of Invest in ME. “You get extreme elements. But this is an illness. We just want to cure it.”
Charles Shepherd, medical adviser to the ME Association, says the real vitriol comes from a small number of people, but “it stifles debate in that people are not happy to express views if they think they are going to get shouted down or get abusive emails. It does scare decent people off from getting involved.”
Plenty of people have had – or are still having – very bad experiences with the medical profession, he says. Many GPs have no idea what to do. There are harrowing stories from people who are refused the employment and support allowance that has replaced incapacity benefit. Such things have radicalised people.
Sir Peter Spencer, former second Sea Lord and chief of defence procurement who has been chief executive of Action for ME for the last three years, agrees there is “far too much mud-slinging”, but adds that “the significant majority feel so washed-out that they don't have the energy for invective . . . the thing that has struck me most is how I admire their resilience under really difficult circumstances.”
Spencer, who still has all the drive of a senior naval officer, talks of the need for more specialist centres, especially for children, one of the issues taken up by the all-party parliamentary group on ME, which published its report into services in March. ME/CFS is the biggest single cause of long-term absenteeism among schoolchildren. Faster diagnosis and help for them is badly needed.
Whatever the final conclusion about XMRV, everybody hopes that the episode will generate more scientific research. But Simpson talks for the frustrated majority when he describes the case of one child on tube-feeding, whose consultant said she must have CBT and should be back in school within a week, and asks: “Why isn't science playing a part in trying to resolve this?”
He himself has two daughters with ME/CFS. It is not hard to imagine how tough that must be. “At the end of the day, it is about people's health,” he says. “My daughters are ill because of this. I can't see anyone in this country who is tackling the problem.”