Attacking Pain At
Its Source: New Perspectives on Opioids
Christoph Stein, Michael Sch”fer,
Halina MachelskaÝ - Nat Med 9(8):1003-1008, 2003.
The treatment of
severe pain with opioids has thus far been limited by their unwanted central
side effects. Recent research promises new approaches, including opioid
analgesics acting outside the central nervous system, targeting of opioid
peptide-containing immune cells to peripheral damaged tissue, and gene transfer
to enhance opioid production at sites of injury.
The first step
in pain treatment is usually pharmacologic, along with cognitive and behavioral
approaches in more chronic stages. Opioids are the most powerful drugs for
severe acute and chronic pain, but their widespread use is hampered by side
effects such as depression of breathing, nausea, clouding of consciousness,
constipation, addiction and tolerance[1]. A new generation of opioid
drugs is now emerging. This class selectively activates opioid receptors
outside the central nervous system, thus avoiding all centrally-mediated,
unwanted effects[2]. Endogenous ligands of these peripheral opioid
receptors are produced within skin and immunocytes[3]. This has led
to new directions of research, such as the selective targeting of opioid
peptide-containing cells to sites of painful injury, and the augmentation of
opioid synthesis by gene transfer.
Acute and
chronic pain are frequently associated with inflammation as a result of tissue
destruction, abnormal immune reactivity or nerve injury. Within damaged
peripheral tissue, primary sensory neurons transduce mechanical, chemical or
heat stimuli into action potentials. These sensory neurons have myelinated (Aδ)
or small-diameter unmyelinated axons (C-fibers, or 'nociceptors'). The latter
are particularly sensitive to the vanniloid receptor-1 ligand and the
neurotoxin capsaicin, and are considered the dominant fibers in clinical pain.
Fibers normally transferring innocuous touch (Aţ) can also contribute to pain
after nerve damage. After synaptic transmission and modulation within the
spinal cord, nociceptive signals reach supraspinal and cortical sites, where
they can be perceived as painful.
Three cDNAs and
their genes have been identified, encoding the µ-, δ- and κ-opioid
receptors (MOR, DOR and KOR, respectively)[1]. All three receptors
mediate pain inhibition and are found throughout the nervous system, in somatic
and visceral sensory neurons, spinal cord projection and interneurons, midbrain
and cortex. The locations of different opioid receptors on functionally
distinct cell types within local circuits of the brain and spinal cord can
result in additive or sometimes opposing interactions between receptor types
(for example, KOR can have antianalgesic actions)[1,4].
Recently, opioid
receptors have been identified on peripheral processes of sensory neurons (Fig.
1). The cell bodies of these neurons in dorsal root ganglia express all three
mRNAs and receptor proteins (reviewed in ref. 5). Opioid receptors are
intra-axonally transported into the neuronal processes[6-10] and are
detectable on peripheral sensory nerve terminals in animals[9,11-13]
and humans[14]. Colocalization studies confirmed the presence of
opioid receptors on C- and A-fibers[15], on vanniloid
receptor-1-positive visceral fibers[16] and on neurons expressing
isolectin B4, substance P or calcitonin gene-related peptide[13,17,18],
consistent with the nociceptor phenotype. Sympathetic neurons and immune cells
can also express opioid receptors but their functional role is unclear[8,19,20].
The binding characteristics of peripheral and central opioid receptors are
similar[8,21] but the molecular mass of peripheral and central

Figure 1. Opioid receptor transport and signaling in primary afferent
neurons. Opioid receptors and neuropeptides (such as substance P) are
synthesized in the dorsal root ganglion and transported along intra-axonal
microtubules into central and peripheral processes of the primary afferent
neuron. At the terminals, opioid receptors are incorporated into the neuronal
membrane and become functional receptors. Upon activation by exogenous or
endogenous opioids, opioid receptors couple to inhibitory G-proteins. This
leads to direct or indirect (through decrease of cyclic adenosine
monophosphate) suppression (-) of Ca2+ or Na+ currents,
and subsequent attenuation of substance P release. The permeability of the
perineurium is increased within inflamed tissue. OR, opioid receptor; sP,
substance P; EO, exogenous opioids; OP, endogenous opioid peptides; Gi/o,
inhibitory G proteins; cAMP, cyclic adenosine monophosphate.
There
is no dispute that peripheral, spinal and supraspinal opioid receptors mediate analgesic
effects. However, the relative contribution of each site to a given analgesic
effect after the systemic (intravenous or oral) administration of an opioid
agonist has not been examined thoroughly. Recent studies suggest that
systemically[22,23] and even centrally (intracerebroventricularly)
injected opioid agonists may act predominantly through peripheral opioid
receptors. This finding was attributed to the active transport of opioids from
the brain to the blood by the saturable P-glycoprotein pump within endothelial
cells of the blood-brain-barrier[24]. Finally, tissue damage
stimulates the expression of peripheral opioid receptors, which likely leads to
a concomitant increase of their function.
All three types
of opioid receptors inhibit high-voltage activated calcium currents in cultured
primary afferent neurons (reviewed in ref. 5). These effects are transduced by
G-proteins (Gi or Go or both)[5,18,25] and may
be relatively resistant to desensitization[26] or tolerance
development. Although it is well known that opioids induce membrane
hyperpolarization as a result of increased potassium currents in central
neurons, this has not yet been detected in dorsal root ganglion neurons[27].
Thus, the modulation of calcium channels seems to be the primary mechanism for
the inhibitory effects of opioids on peripheral sensory neurons. In addition,
opioids, through inhibition of adenylyl cyclase, suppress
tetrodotoxin-resistant sodium-selective and nonselective cation currents
stimulated by the inflammatory agent prostaglandin E2 (refs. 28,29).
Tetrodotoxin-resistant sodium channels are selectively expressed in
nociceptors, where they have an important role in impulse initiation and action
potential conductance. These channels mediate spontaneous activity in
sensitized nociceptors[30] and accumulate at the site of injury in
damaged nerves, leading to ectopic impulse generation[31]. The
latter observations may explain the notable efficacy of peripheral opioids in
inflammatory and neuropathic pain[10,20,32,33]. Consistent with
their effects on ion channels, opioids attenuate the excitability of peripheral
nociceptor terminals, the propagation of action potentials, the release of
excitatory proinflammatory neuropeptides (substance P, calcitonin gene-related
peptide) from peripheral sensory nerve endings, and the vasodilatation evoked
by stimulation of C-fibers (reviewed in ref. 5). All of these mechanisms result
in analgesia or anti-inflammatory actions (Fig. 1).
Peripheral
opioid analgesia is augmented under conditions of tissue injury such as
inflammation, neuropathy or bone damage (reviewed in refs. 5,32,34). One
underlying mechanism is an increased number of peripheral opioid receptors. In
neuronal cell cultures, MOR transcription is upregulated by the cytokine
interleukin (IL)-4 through the binding of STAT-6 transcription factors to the
MOR gene promoter[35]. This promoter also responds to activator
protein-1 (ref. 36). In dorsal root ganglia, the synthesis and expression of
opioid receptors can be increased by peripheral tissue inflammation[21,37].
Subsequently, the axonal transport of opioid receptors is greatly enhanced[8,9,38],
leading to their upregulation and enhanced agonist efficacy at peripheral nerve
terminals[19]. IL-1ţ can stimulate this axonal transport[38].
In addition, the specific milieu (low pH, prostanoid release) of inflamed
tissue may increase opioid agonist efficacy by enhanced G-protein coupling and
by increased neuronal cyclic adenosine monophosphate (cAMP) levels[21,28,39].
Inflammation also increases the number of sensory nerve terminals ('sprouting')
and disrupts the perineural barrier, thus facilitating the access of opioid
agonists to their receptors[40]. Clinical studies indicate that the
perineural application of opioid agonists along uninjured nerves (such as the
axillary plexus[41,42], intrapleural and intercostal nerves[43],
and mandibular nerve[44]) does not reliably produce analgesic
effects, supporting the notion that inflammation promotes accessibility and
efficient coupling of opioid receptors in primary afferent neurons. In fact, it
is unknown whether opioid receptor coupling normally occurs in axons or only in
nerve terminals. Finally, the secretion of endogenous opioid ligands within
inflamed tissue may produce additive or synergistic interactions at peripheral
opioid receptors.
An important
question is whether tolerance -- a loss of analgesic efficacy after recurring
application of agonists -- also develops at peripheral opioid receptors.
Peripheral tolerance has been observed in animal models using repeated opioid
pretreatment in the absence of persistent inflammation[45-48].
However, because the number, affinity and coupling efficacy of opioid receptors
are enhanced under inflammatory conditions, these studies do not permit
conclusions regarding tolerance in pathological situations. In other models,
peripheral opioid analgesia is resistant to the development of tolerance[49,50],
and clinical studies suggest a lack of cross-tolerance between peripheral
exogenous and endogenous opioids in synovial inflammation[14].
Clearly, more investigations will be needed to define the differences in
peripheral opioid receptor function between noninjured neurons, nerve damage
and persistent inflammation, and to elucidate underlying mechanisms such as
changes in receptor affinity, phosphorylation, G-protein coupling and
internalization[51]. From the clinician's viewpoint, the induction
of tolerance by opioid pretreatment in the absence of painful tissue injury is
not illustrative because patients usually do not consume opioids when they are
not in pain (except in the case of opioid abuse).
Three families
of opioid peptides are well characterized; the endorphins, enkephalins and
dynorphins. They bind to all three opioid receptors. Each family derives from a
distinct gene; their respective precursors are pro-opiomelanocortin (POMC),
proenkephalin and prodynorphin. Additional selective µ-ligands, the
endomorphins, have been isolated but their precursors are not yet known[1].
Immune cells are
the most extensively examined source of opioids interacting with peripheral
opioid receptors[3]. Transcripts and peptides derived from POMC and
proenkephalin, as well as the prohormone convertases PC-1/3 and PC-2, which are
necessary for post-translational processing, were detected in immune cells[52].
The expression of immune-derived opioids is stimulated by viruses, endotoxins,
cytokines, corticotropin-releasing hormone (CRH) and adrenergic agonists[52].
In conditions of painful inflammation, POMC mRNA, ţ-endorphin, met-enkephalin
and dynorphin-A are found in circulating cells and lymph nodes[53,54].
In the injured tissue, these peptides are upregulated in lymphocytes,
monocytes, macrophages and granulocytes[9,14,55-57].
Adhesion
molecules direct the migration of circulating leukocytes to injured tissue. The
initial step, rolling, is mediated by selectins on leukocytes (L-selectin) and
endothelia (P- and E-selectin). The rolling leukocytes are exposed to
tissue-derived chemokines that upregulate the avidity of integrins, which
mediate the firm adhesion of cells to endothelia by interacting with
immunoglobulin superfamily members such as intercellular adhesion molecule-1
(ICAM-1). Finally, the cells migrate through the vessel wall, directed by
platelet-endothelial cell adhesion molecule-1 (PECAM-1) and other
immunoglobulin ligands. Interruption of this cascade can block immunocyte
extravasation[58].
These events
also govern the homing of opioid-producing cells (Fig. 2). In inflamed tissue,
leukocytes containing ţ-endorphin coexpress L-selectin[59].
Opioid-producing cells, vascular P-selectin, ICAM-1 and PECAM-1 are
simultaneously upregulated[59,60]. Blocking the selectins or ICAM-1
reduces the number of opioid-producing cells[60,61]. Thus,
circulating opioid-producing immune cells home to damaged tissue to secrete the
opioids and then travel to the regional lymph nodes[53,54] (Fig. 2).

Figure 2. Migration of opioid-producing cells and opioid secretion within
inflamed tissue. P-selectin, ICAM-1 and PECAM-1 are upregulated on vascular
endothelium. L-selectin is coexpressed by immune cells producing opioid
peptides. L- and P-selectin mediate rolling of opioid-containing cells along
the vessel wall. ICAM-1 mediates their firm adhesion and diapedesis. Adhesion
molecules interact with their respective ligands. In response to stress or
releasing agents (such as CRH or IL-1), the cells secrete opioid peptides. CRH
and IL-1 elicit opioid peptide release by activating CRH receptors and IL-1 receptors,
respectively. Opioid peptides or exogenous opioids bind to opioid receptors on
primary afferent neurons, leading to analgesia (see Fig. 1). The immune cells,
depleted of opioids, then migrate to regional lymph nodes. OP, opioid peptides;
CRHR, CRH receptor; IL-1R, IL-1 receptor; EO, exogenous opioids.
As in the
pituitary, the release of opioids from immunocytes is regulated by IL-1ţ and
CRH (Fig. 2). This release is receptor-specific and calcium-dependent and is
mimicked by elevated extracellular potassium, consistent with a regulated
secretory pathway as in neurons and endocrine cells[53,54]. In
vivo, small, systemically inactive doses of CRH and cytokines elicit
opioid-mediated analgesia within injured tissues[3,62]. Furthermore,
environmental stress and endogenous CRH are triggers for local opioid release[55,63].
The potency of this intrinsic pain inhibition is proportional to the number of
opioid-producing immunocytes[56]. An effective central blockade of
pain decreases the migration of such cells to injured sites, indicating a
reduced need for peripheral 'pain-control' cells[64]. Both CRH- and
stress-induced analgesia can be extinguished by immunosuppression[3,11,62]
and by blocking the extravasation of opioid-containing leukocytes[60,61].
In patients undergoing knee surgery, opioid-producing lymphocytes and
macrophages accumulate in the inflamed synovium. Blockade of intra-articular
opioid receptors increases postoperative pain[55], apparently
without inducing cross-tolerance to locally administered morphine[14].
Other opioid
sources include the pituitary and adrenal glands, but neither seems to be
involved in peripheral pain inhibition[65]. Keratinocytes may
release opioids upon stimulation by endothelin-1 (ref. 66). Finally, sensory
neurons produce opioid peptides[57] and can be stimulated to
overexpress by gene transfer[67].
This basic research
has stimulated the development of new opioid ligands acting exclusively in the
periphery without central side effects[2]. A common approach is the
use of hydrophilic compounds with minimal capability to cross the blood-brain
barrier[68,69]. Penetration of the blood-brain barrier may not be
entirely precluded at high doses, however, and the polarizing residues may
interfere with the affinity at opioid receptors. Among the first compounds
tested were loperamide (originally known as an antidiarrheal drug)[70]
and asimadoline[68]. Peripheral restriction was also achieved with
newly developed arylacetamide[71,72] and peptidic KOR agonists[73]
by replacing 3,4-dichloro substituents on the phenyl acetyl group by
trifluoromethylphenyl acetyl in the former.
Although early
attempts to demonstrate peripheral opioid analgesia in normal tissue failed,
there was more success with models of pathological pain (Table 1; reviewed in ref. 32). In chronic inflammation of the
rat paw, local injection of low, systemically inactive doses of µ-,
- and
-agonists produces analgesia that is dose dependent,
stereospecific and reversible by selective antagonists, indicating the
activation of peripheral MOR, DOR and KOR[74]. Asimadoline, a
-opioid agonist, elicits immediate analgesia and delayed
proinflammatory effects in this model[68,75], whereas the peptidic
KOR agonists produce both peripheral analgesic and anti-inflammatory effects[73].
Possible underlying mechanisms of the latter include reduced release of
proinflammatory neuropeptides[76,77] or cytokines[78], or
diminished expression of adhesion molecules[79]. Potent
antinociception was also shown in models of nerve damage[10,20,33]
and visceral[80], thermal[46,48] and bone pain[81].
Thus, peripheral opioids are effective in a wide variety of animal models,
including visceral and neuropathic pain, which are often resistant to
conventional drug treatment in humans (Table 1).
Numerous
controlled studies have demonstrated significant analgesic effects following
local application of opioids at sites of injury (Table 1; reviewed in refs. 5,34). Pain relief can be measured
by reductions in subjective pain intensity scores, by extended time intervals
to the patient's first request for additional pain medication, by a decreased
number of patients asking for supplemental analgesic drugs, or by diminished
total consumption of supplemental analgesics.
Intra-articular
administration of the µ-agonist morphine is the best examined clinical
application[34]. After knee surgery, it reduces pain scores or
supplemental analgesic consumption (or both), in a dose-dependent fashion, by a
peripheral mechanism of action and without side effects[5,34].
Intra-articular morphine is active even in the presence of opioid-containing
inflammatory cells[14] and in chronic rheumatoid and osteoarthritis[82,83].
Its effect is similar to a standard intra-articular local anesthetic or steroid
injection and is surprisingly long lasting (up to 7 days)[83,84].
This may be the result of morphine's anti-inflammatory activity, as evidenced
by a reduced number of synovial inflammatory cells[83]. A limitation
of its use in chronic arthritis is that repeated injections carry a risk of
infection and cannot be easily applied to more than one joint.
Other controlled
clinical trials examined patients undergoing spinal fusion surgery, who
reported postoperative analgesia from a local morphine injection at the iliac
bone harvest site. Even one year later, the incidence of chronic donor site
pain was still significantly reduced[85]. In chronic inflammatory
tooth pain, the submucous injection of morphine resulted in long-lasting pain
relief after dental sugery[44]. Similar to the animal studies, the
analgesic effect was absent in patients without pre-existing inflammation[44].
In unilateral corneal abrasions, topical morphine attenuated pain on the
lesioned, but not the intact, side. Importantly, no detrimental effects on
wound healing were found[86]. In postoperative visceral pain,
locally injected opioids produced analgesia in the urinary bladder[87]
and after laparoscopic tubal ligation[88]. These findings may be of
growing importance with the increasing numbers of endoscopic procedures and
with the heightened awareness for the need to pre-empt the development of
chronic pain.
In contrast,
several studies have found no peripheral effects of opioids[41,42].
The majority of those trials examined the injection of agonists into the
noninflamed environment along nerve trunks. This suggests that intra-axonal
opioid receptors may be 'in transit' and may not be available as functional
receptors at the membrane. In addition, many negative studies were flawed by
methodological shortcomings such as lack of study sensitivity (very little pain
in the placebo group), small sample sizes, lack of tissue inflammation,
inadequately low doses or the superimposition of general or local anesthetic
effects[34,41,42,89].
New peripherally
restricted opioids have recently entered human trials. Asimadoline was not
successful in experimental pain[90] or in patients undergoing
arthroscopy[75]. This was attributed to its low potency and partial
ability to cross the blood-brain barrier in humans, resulting in central side
effects at higher doses. However, a second-generation
-agonist markedly reduced visceral pain in patients with chronic
pancreatitis without severe side effects[72].
These findings provide
new insights into intrinsic mechanisms of pain control, and suggest innovative
strategies for developing drugs and alternative approaches to pain treatment.
Immunocompromised patients (such as those with AIDS, cancer and diabetes)
frequently suffer from painful neuropathies that can be associated with intra-
and perineural inflammation, with reduced intraepidermal nerve fiber density
and low CD4+ lymphocyte counts[91]. Thus, it may be
useful to investigate intraepidermal opioid receptor density, opioid production
and release, and migration of opioid-containing immune cells in these patients.
The important role of selectins and ICAM-1 in the migration of
opioid-containing cells to injured tissue indicates that antiadhesion
strategies for the treatment of inflammatory diseases may in fact carry the
risk of exacerbating pain. It would be desirable to identify adhesion
molecules, chemokines or other stimulating factors that selectively attract
opioid-producing cells to damaged tissue. Augmenting the synthesis or secretion
of opioid peptides within injured tissue may be accomplished by growth factors,
stem-cell or gene therapy: in sensory neurons, the synthesis, transport and
peripheral release of enkephalins can be enhanced by herpesvirus-derived
vectors containing proenkephalin cDNA, leading to a decrease in chronic pain
and inflammation in polyarthritic rats[67]. Another study showed
successful transfection of rat skin with POMC cDNA delivered by a nonviral
gene-gun system, resulting in increased tissue levels of ţ-endorphin and a
tendency to decrease acute inflammatory pain[92].
In view of the
ongoing debate about the potential for abusing centrally acting opioids, it is
crucial to promote the development of new, peripherally selective opioid drugs.
Beyond the absence of central side effects, these compounds may offer
advantages such as anti-inflammatory effects, lack of tolerance, lack of
constipation (in the case of KOR agonists[72]), lack of
gastrointestinal, renal and thromboembolic complications associated with
nonsteroidal anti-inflammatory drugs, and efficacy in neuropathic pain. Locally
administered opioids are already used in clinical practice[34], but
new drugs applicable by systemic routes are needed. A prototype has been
successfully applied intravenously in patients with pain from chronic
pancreatitis[72], and an orally available, peripherally restricted
opioid antagonist was recently introduced for the treatment of postoperative
ileus[93]. Many efforts are currently being undertaken to develop peripherally
acting analgesics by aiming at individual excitatory receptors or channels on
sensory neurons[94]. The major advantage of targeting opioid
receptors is their mechanism of action: the inhibition of calcium (and possibly
sodium) ion channels renders the nociceptor unexcitable to the plethora of
stimulating molecules expressed in damaged tissue. Thus, peripherally acting
opioids can prevent and reverse the action of multiple excitatory agents
simultaneously, instead of blocking only a single noxious stimulus. Uncovering
mechanisms that can enhance the availability of endogenous opioids within
injured tissue, the signal transduction of peripheral opioid receptors and the
exclusion of novel opioid drugs from the central nervous system will open exciting
possibilities for pain research and therapy.

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