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A Laymen's Guide to Chapter Nine of CHRONIC FATIGUE

SYNDROME A BIOLOGICAL: APPROACH by Cort

CHRONIC FATIGUE SYNDROME A BIOLOGICAL APPROACH

Edited by Englebienne Ph.D., Kenny DeMeirleir M.D,

Ph.D., CRC Press. Washington D.C. 2002

Chapter Nine: Current Advances in CFS Therapy

By Pascale DeBecker, Neil R. McGregor, De Suet and

Kenny DeMeirLeir

(This chapter is where the rubber meets the road for CFS

patients. Yes, the research is interesting and it is gratifying to

see so many aspects of CFS put together in such a coherent

manner, but does all this work show up in the doctors office?

Does it show up in increased quality of life for CFS sufferers? In

so many ways this is the only chapter that matters for most of us.

Everything else, as they say, is prelude.

For me this chapter was both encouraging and discouraging

-encouraging because of all the different treatments out there

that can at take at least a nibble at or maybe even take a good

bite out of some of the symptoms that dog us. It was

encouraging as well in that some of the findings in the book are

leading to new therapeutic approaches. I have not heard, for

instance, of amino acid supplementation with regard to CFS.

It was discouraging in that, aside from Ampligen, there

appears to be no answer to the problem of RNase L dysfunction.

Nor is there a mention of something designed to halt the rather

massive amount of proteolytic cleavage going on. Nor, except for

one reference, is there any discussion of regulators of calcium

homeostasis or retinoic acid, two treatment possibilities

mentioned in the text. Most of the treatments appear to attack the

many co-morbid diseases or problems that afflict CFS sufferers.

The breadth of these treatments is certainly gratifying.)

9.1 INTRODUCTION

The authors begin by lamenting the state of the art of clinical

trials in CFS. In short, given the heterogeneity of the patient

population and the complexity of the disease origin, a number of

factors that need to be taken into account (gradual vs. sudden

onset, disease severity, duration, etc.) are not, and on top of that,

the studies are too small, too short, too ill defined, etc.

Questions regarding study efficacy have precluded the

acceptance of some treatments that have demonstrated clinical

benefit. Treatments at this stage are mostly symptomatic; no

`consistently effective, readily available, safe and affordable

pharmacological therapies' have yet been identified for CFS.

Against this rather muddled treatment background, the

authors note the profound effects CFS has on patient

functioning. Less than 10% of CFS patient return to their

`premorbid levels of functioning'; i.e. get well. Few patients ever

resume an active lifestyle. There clearly is a great need for

effective treatments for this illness.

PHARMACOLOGICAL THERAPY

Drugs Treating the Brain and Nervous System

While there does not appear to be an association between

RNase L activity or fragmentation and mood changes/psychiatric

symptoms, cytokine alterations in CFS can lead to symptoms

involving mood. Altered cytokine levels can cause depression

and anxiety through changes in brain neurochemistry.

Antidepressant use, sometimes at far lower doses than

suggested for depression, is therapeutic for some CFS patients.

The ability of non-psychiatrically therapeutic doses of

antidepressants to confer clinical improvement in CFS patients

suggests that the symptom reduction seen is derived from other

that an antidepressant effect

Benzodiazepenes `may be' used in treatment of anxiety but

are not effective in treating sleep disorders. Modafinil has been

successfully used in narcolepsy and could have some benefit in

CFS.

Tricyclic depressants, in particular those able to regulate

sleep, have been beneficial for some CFS patients. Nortriptyline

reduced symptoms and depression in one double-blinded

study. Significant side effects can accrue from using first

generation TCA's.

The authors suggest sedating tricyclic antidepressants for

severe insomnia (amitriptyline) and neutral (desipramine) for

hypersomnia.

Despite encouraging results from two preliminary `case

series', a recent study found that fluoxetine, a serotonin reuptake

blocker, offered no benefits. The authors suggest this was

because CFS patients exhibit depressed levels of

catecholamine not serotonin precursors. (This view was

substantiated by a recent study that found high levels of

tryptophan, the serotonin precursor, and low levels of tyrosine,

the dopamine precursor).

Doxepin, another tricyclic antipressant, had a 70% success

rate in an uncontrolled study.

A tricyclic monamine oxidase inhibitor, moclobeimide, had

significant but modest effects in an open study. Because it

influences catecholamine levels the authors suggest that it

maybe increasing tyrosine availability. Positive benefits from

another study using a different catecholamine agent

(phenelzine) suggest that antidepressants influencing

catecholamine levels may have the greatest efficacy in CFS.

Serotonin is involved in sleep and temperature regulation, pain

sensitivity, cardiovascular response and mood. All of these are

commonly disturbed in CFS. Results from studies investigating

serotonin have, however, been mixed.

Galantiamine, an acetylcholinesterase inhibitor, showed

promising results in an initial study containing some

methodological concerns but had no effect in a double-blinded

placebo controlled study.

Drugs Treating the Endocrine System

Growth hormone is of particular interest in CFS because fatigue

is common in growth hormone deficiency. An important

`anabolic agent', GH is essential for muscle homeostasis.

Attempts to increase plasma volume in order to combat neurally

mediated hypotension (loss of blood pressure when standing)

using fluodrocortisone were no more successful than placebo in

two recent studies.

CFS patients display reduced DHEA and DHEA sulfate levels

and exhibit a blunted DHEA response to ACTH injections. DHEA

has significant immune effects; it promotes the Th1 response

and can correct disregulated cytokine production. A subset of

CFS patients experienced benefits to two preliminary studies of

DHEA. Larger, double-blinded studies are needed to assess

DHEA's efficacy in CFS.

Immunomodulating and Antiviral Drugs

Ampligen is a manufactured dsRNA product that enhances

antiviral activity and has immunomodulatory and even anti-tumor

properties. It is able to modulate 2-5OAS and 2-5A as well as

interferons and interleukins, and can modulate both branches of

the immune system. Studies have thus far determined that

Ampligen is effective against at least 12 viruses. Ampligen is

particularly effective at enhancing the functioning of NK cells –

whose activity is inhibited in CFS.

Ampligen administration to a cohort of severely ill CFS

patients resulted in near normal 2-5A/RNase L pathway activity

and improved exercise tolerance and reduced neurocognitive

symptoms. Ampligen is able to induce the 2-5OAS system in

CFS patients to produce bioactive 2-5A trimers rather than the

dimers predominantly produced Ampligen has successfully

passed phase II clinical trials and is now in phase III trials. Six

months of Ampligen therapy resulted in improved quality of life

scores, significantly increased activity and cognitive ability.

Scores in exercise tests increased 10%. (This drug is obviously

not a panacea.) The authors suggest that ampligen therapy may

be appropriate for more severely disabled CFS patients. (In a

recent paper (JCFS 2003) Dr. Englebienne indicated that while

Ampligen can normalize the 2-5OAS-RNase L system, it most

unfortunately also upregulates PKR activity.)

Immunoglobulins in general, and IgG in particular, appear to

commonly occur in CFS. Intravenous immunoglobulin therapy

might benefit CFS patients by increasing antibodies against viral

infections or by correcting `immunoregulatory disturbances'.

CFS patients received benefits from two of four

placebo-controlled studies. Adolescents in particular appeared

to benefit from immunoglobulins. The cost, limited efficacy,

tolerability, and transient nature of the benefits preclude the

authors from recommending immunoglobulin therapy.

An extract from T lymphocytes, transfer factor contains a wide

assortment of `immune modulators', including parts of T helper

and cytotoxic cells, interferons, IL-2, etc. No specific (?) benefits,

however, were found in a double-blinded, placebo-controlled

study.

The considerable improvements experienced by CFS

patients in a small study of Isoprinosine suggests that larger

double-blinded placebo controlled should be undertaken. The

authors indicated that about 1/3rd of their CFS patients

responded after 1.5 grams of isoprinosine a day for several

months.

Amantadine is an antiviral agent that elevates dopaminergic

activity by releasing norepinephrine and dopamine from central

nervous system stores. Amantadine effectively treated fatigue in

multiple sclerosis, (a disease that bears some similarities to

CFS. See chapter eight.) Despite promising anecdotal reports,

not only were no benefits derived from an Amantadine trial, but

side effects were common. The frequency of side effects, the

authors suggest, probably indicative of a disordered

catecholamine metabolism in CFS.

A extract of mushrooms, Lentian appears to impact a wide

variety of immune responses; it induces multiple cytokines,

inhibits Th2 dominance, and enhances cell mediated immune

responses. As all these activities would very likely be beneficial

to CFS patients, the authors suggest clinical trials should be

done.

A porcine liver extract, kutapression inhibits HHV6 replication

and blocks EBV infection. Despite its antiviral activities, a

double-blinded placebo controlled trial of acyclovir gave no more

benefit that placebo.

A subset of CFS patients with diminished NK cell functioning

showed benefits from IFN-a, an interferon with wide antiviral and

immunomodulatory properties. The authors note that while CFS

patients with known virally associated co-morbid diseases may

derive benefit from IFN-a therapy, IFN-therapy has in the past

caused many of the symptoms (fatigue, pain, mood change)

associated with CFS.

Antibiotics - Treatments of the opportunistic infections often

found in CFS may significantly effect patient `morbidity " Using a

very sensitive and specific PCR procedure, several groups have

found mycoplasma spp. (especially M. fermentans) in about

70% of CFS patients (compared, in one study, to 6% of healthy

controls.) Mycoplasma appear to be able to more readily invade

cells with low RNase L levels.

Besides activating B lymphocytes, mycoplasmas produce

proteases that degrade immunoglobulins. Mycoplasmas can

infect nerve and synovial cells and other cell types. They are very

effective at evading the immune system. Nicolson et. al. have

found that most patients require multiple cycles (2-6) of 6 week

courses of antibiotics to completely recover (@1-3 years! Then

again, few other doctors speak of `complete recovery'.)

Therapeutic regimes to correct nutritional and vitamen

deficiencies are recommended during antibiotic therapy.

Antibiotic therapy has also been beneficial, at least in the

short term, in combating another common bacterial infection in

CFS, toxin producing coagluase negative staphylococci.

Relapse unfortunately was common, and different therapeutic

regimes are being explored. A staphylococci toxoid vaccination

program resulted in significant improvements in pain, sleep,

concentration and memory.

OTHER IMMUNE AGENTS - Two antihistamine drugs, rantidime

and cimetidine, produced significant remissions in patients with

chronic EBV infections in short-term studies. These drugs

should be more thoroughly assessed with regard to CFS.

Various nonsteroidal anti-inflammatory agents such as NSAIDS,

paracetomol or orco-codamol may be useful in muscle and joint

pain. Amitriptyline was helpful in easing muscle pain, improving

sleep and reducing fatigue.

In an assessment of 29 medical and alternative therapies,

CFS patients ranked anti-allergy and anti-yeast diets very highly.

The authors suggest, however, that systemic anti-fungal agents

not be used because of possible side effects, unless a fungal

condition is present.

By inhibiting apoptotic enzymes or relaxing smooth muscles

calcium channel blockers potentially can effect a variety of

symptoms found in CFS. Their use in CFS, however, has thus

far been limited. Nimodipine and amlodipine have been used to

alleviate pain. Goldstein reccomends dihydropyridines.

NUTRITIONAL FACTORS

Vitamins - Folic acid deficiency in several symptoms commonly

found in CFS such as fatigue, neuromuscular problems and

reduced brain functioning. A subset of CFS patients appear to

be deficient in folic acid. A small double blinded study that the

authors feel may have been too short and may have used an

insufficient dose of folic acid showed no benefits.

Hydroxycobalamin injections have been widely used to

combat the low central nervous system levels of vitamin B-12

reported in CFS. B-12 successfully treated abnormally shaped

red blood cells that researchers believed caused oxygen deficits

at micro-circulatory levels. Because two studies have shown

elevated levels of neopterin, a nitric oxide indicator,

hydroxycobalamin may be useful in scavenging nitric oxide and

its oxidizing by-product peroxynitrite in CFS.

Riboflavin, thiamine and pyrodoxine levels may also be

reduced in CFS. Ten mgs. of NADH, a reduced form of niacin,

generated substantial benefits in a double-blinded, placebo

controlled study. These benefits may result not from resolving a

niacin deficiency but from establishing a more favorable

oxidation/reduction environment.

Vitamin C boosts immune responses, has anti-viral

properties, and at high doses, can enhance NK activity ten fold

(!). Kodama et. al. reported that vitamin C infusions `in

association with the enhancement of endogenous

glucocorticoids' resulted in `clinical control' (?) of CFS. Adding

DHEA resulted in greater benefits.

Minerals - Several studies have indicated reduced red blood cell

magnesium deficiencies in CFS. CFS patients experienced

increased energy, decreased pain and improved mood in one

placebo controlled study. CFS patients with significant muscle

pain or who also have fibromyalgia should incorporate malate, a

mitochondrial ATP enhancing substance, into their magnesium

regime. Malate/magnesium significantly reduced the sensitivity

of tender points in fibromyalgia patients.

Low sodium intake can cause fatigue, headache, poor sleep

and inability to concentrate. Conversely increased sodium

uptake has successfully increased blood pressure in two

studies of CFS sufferers experiencing neurally mediated

hypotension. The authors suggest that CFS patients increase

their sodium intake.

Both zinc and iron deficiencies can lead to fatigue and other

symptoms associated with CFS. Iron levels may be reduced as

well in CFS patients with multiple infections. (Many

microorganisms suck up iron stores in the body.) An iron

assessment of iron metabolism should be done to determine if

iron supplementation is needed. The authors caution that iron

overload can occur in CFS patients who have the

hemachromatosis gene.

Amino Acids - Individually tailored amino acid mixtures can

improve symptomology in CFS, particularly with regard to

neurocognitive symptoms and muscle pain. Increased cytokine

levels result in increased protein breakdown

The wide variety of amino acid patterns found necessitate that

individualized amino acid regimes be employed in CFS. Single

amino acid supplementation, in particular, should be avoided

because, due to the nature of the amino acid transport system, it

can cause multiple deficiencies.

Possibly because of the increased IFN-y produced,

tryptophan degradation is enhanced in infectious diseases. It is

not clear, however, that tryptophan supplementation enhances

immunity. CFS patients exhibited reduced tryptophan levels in

two of three studies. The authors once again recommend

against supplementation of one amino acid.

An important factor in muscle metabolism, L-carnitine

deficiency is marked by muscle pain, weakness and fatigue

following exercise. L. carnitine helps in fatty acid transport

across membranes and optimizes the intracellular `lipid mileu',

an environment involved in lymphocyte proliferation and cytokine

production. About one third of CFS patients appear to benefit

from L-carnitine supplementation.

Plasma concentrations of glutamine were significantly lower

in CFS patients in one study but not another. Gluatmine

supplementation offered no benefits, however, to CFS patients in

one study.

NUTRITIONAL FACTORS - Fatty acids are essential substrates

for many cellular processes including oxidative phosphorylation

in muscle mitochondria and neuronal metabolism. Fatty acid

levels can fall during viral infections and remain low afterwards.

The lipid alterations associated with viral infections were not

found in CFS patients, however. Instead, levels of n-6 fatty acids,

which are precursors of inflammatory substances such as

prostaglandins and leukotrienes, are increased in CFS. Levels

of n- 6 fatty acids are positively associated with symptom

expression which in turn is associated with increased

inflammatory activity and/or viral activity.

Viral activity has been associated with reduced EFA levels. A

double-blinded placebo controlled study indicated that EFA's

(80% EPO, 20% fish oil) reduced muscle pain, dizziness,

concentration and depression. 's study, however,

indicated that high EPA levels were positively correlated with

increased symptom expression. No benefits were seen in

another study. The authors suggest EFA supplementation is not

indicated in CFS unless a demonstrable defect is shown.

Food intolerances are common in CFS. Removing allergens

from the diet can result in the complete resolution of

gastrointestinal symptoms.

Significant improvement of CFS symptoms, exercise

tolerance and post exercise fatigue was found in one open study

using coenzyme Q10.

OTHER THERAPIES - Significant improvements in pain, sleep,

concentration and memory were seen in a placebo-controlled

study using a Staphylococcus Toxoid Vaccine

NON PHARMACOLOGICAL THERAPY - Cognitive Behavior

Therapy (CBT) attempts to alter behavior patterns that may either

exacerbate CFS or hamper recovery. Such programs are often

important facets of rehabilitation for those dealing with chronic

illnesses. They focus not on the disease but on the

psychological responses to disease, some of which can have

physiological effects. In CFS CBT has focused on activity levels,

relaxation and breathing patterns. While CBT is beneficial in

coping with the psychological effects of CFS it does not appear

to alter the organic basis of the disease.

Deconditioning has been proposed to account for exercise

induced fatigue in CFS. Several studies, however, have failed to

find evidence for deconditioning. A subset of CFS patients with

evidence of deconditioning, however, improved with graded

exercise treatments. Seventy percent of CFS patients in another

graded exercise program trial experienced improvements in

fatigue, disability and mood. Severely impaired CFS patients,

however, do not appear to be good candidates for graded

exercise programs.

CFS patients experienced significant improvements in

sleeping, anxiety, pain, depression, fatigue and somatic

symptoms in a randomized controlled trial of Transcutaneous

Electrical Stimulation (TENS).

FUTURE DEVELOPMENTS - The disease progression in CFS is

complex. Treating this disease involves managing the three

processes described in these chapters; (1) the disease etiology,

(2) the host response to the disease and (3) the co-morbid

diseases that accrue as a result of the disruptions caused by the

disease.

Therapeutic regimes are needed for each component of the

disease process. These would include using antivirals,

antibiotics, and antifungals to attack the disease etiology and

co-morbid pathogens; anti-inflammatories, apoptotic regulators

and supplements to manage host responses and pyschological

adjuncts, graded exercises and anti-depressants to combat the

outcome of the disease process.

The interventions may range from treatments designed to

reduce cytokine levels (drugs, EFA manipulation, etc.), or alter

ion channel activity, or manipulate 2-5A synthetase/RNase L

pathways, ABC transporters, or even double stranded RNA

aptamers.

Promising Treatments the Authors Believe Need More Study:

DHEA, amino acids, magnesium,

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