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Research report Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid Rena Li 1 , Rif S. El-Mallakh * Mood Disorders Research Program, Department of Psychiatry and Behavioral Sciences, University of Louisville School of Medicine, Louisville, KY 40292, USA Received 2 May 2002; accepted 30 December 2002 Abstract Background: While the pathogenesis of manic-depressive, or bipolar, illness is unknown, an excess of intracellular sodium and calcium concentrations is thought to contribute to the development of the illness. Previous work has demonstrated a reduced adaptive response of the sodium pump to ethacrynic acid in lymphocytes obtained from bipolar subjects compared to psychiatrically normal controls. Methods: To further examine this phenomenon, we investigated several aspects of sodium pump response (transcription, translation, activity, and intracellular ion concentration) in lymphoblastoid cell lines derived from bipolar subjects and matched normal controls. Cells were treated with ethacrynic acid 100 AM for 3 days. Results: Normal control-derived cells exhibited an upregulation of sodium pump mRNA synthesis, protein expression, pump-specific binding and activity, and were able to maintain a normal intracellular sodium concentration. Cells derived from bipolar individuals did not alter sodium pump parameters in any way, and consequently, had a higher intracellular sodium concentration. Limitations: While bipolar lymphoblasts were from an inbred Old Order Amish population, the normal controls were from an outbred population. Conclusions: The results suggest that bipolar illness is associated with an abnormality in cellular sodium homeostatic regulation. D 2003 Elsevier B.V. All rights reserved. Keywords: Bipolar disorder; Ethacrynic acid; Na,K-ATPase; Sodium; Sodium pump Manic-depressive, or bipolar, illness is a relatively common condition afflicting 1% of the world’s pop- ulation (Goodwin and Jamison, 1990). Several mod- els have been presented. These span neurotransmitter, G protein (Avissar and Schreiber, 1989), second messenger (Warsh and Li, 1996; Manji et al., 1999; Emamghoreishi et al., 2000), and ionic regulation abnormalities (Dubovsky and Franks, 1983; El-Mal- lakh and Wyatt, 1995). The latter is particularly promising since it may underlie a common mechanism of mood stabilization (El-Mallakh and Huff, 2001) and has led to an interesting animal model of mania (Ruktanonchai et al., 1998; Decker et al., 2000). Both mania and bipolar depression have been asso- ciated with increased intracellular sodium (Na) con- centrations (Coppen et al., 1966; Shaw, 1966), and a decrease in erythrocyte sodium and potassium-activat- ed adenosine triphosphatase (Na,K-ATPase or Na pump) activity (Looney and El-Mallakh, 1997). Tem- poral gray matter of post mortem tissue obtained from 0165-0327/$ - see front matter D 2003 Elsevier B.V. All rights reserved. doi:10.1016/S0165-0327(03)00044-2 * Corresponding author. Tel.: +1-502-852-1124; fax: +1-502- 852-1115. E-mail address: [email protected] (R.S. El-Mallakh). 1 Present address: Sun Health Institute, Sun City, AZ, USA. www.elsevier.com/locate/jad Journal of Affective Disorders 80 (2004) 11– 17

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Page 1: Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid

www.elsevier.com/locate/jad

Journal of Affective Disorders 80 (2004) 11–17

Research report

Differential response of bipolar and normal control lymphoblastoid

cell sodium pump to ethacrynic acid

Rena Li1, Rif S. El-Mallakh*

Mood Disorders Research Program, Department of Psychiatry and Behavioral Sciences, University of Louisville School of Medicine,

Louisville, KY 40292, USA

Received 2 May 2002; accepted 30 December 2002

Abstract

Background: While the pathogenesis of manic-depressive, or bipolar, illness is unknown, an excess of intracellular sodium and

calcium concentrations is thought to contribute to the development of the illness. Previous work has demonstrated a reduced

adaptive response of the sodium pump to ethacrynic acid in lymphocytes obtained from bipolar subjects compared to

psychiatrically normal controls.Methods: To further examine this phenomenon, we investigated several aspects of sodium pump

response (transcription, translation, activity, and intracellular ion concentration) in lymphoblastoid cell lines derived from bipolar

subjects and matched normal controls. Cells were treated with ethacrynic acid 100 AM for 3 days. Results: Normal control-derived

cells exhibited an upregulation of sodium pump mRNA synthesis, protein expression, pump-specific binding and activity, and

were able to maintain a normal intracellular sodium concentration. Cells derived from bipolar individuals did not alter sodium

pump parameters in any way, and consequently, had a higher intracellular sodium concentration. Limitations: While bipolar

lymphoblasts were from an inbred Old Order Amish population, the normal controls were from an outbred population.

Conclusions: The results suggest that bipolar illness is associated with an abnormality in cellular sodium homeostatic regulation.

D 2003 Elsevier B.V. All rights reserved.

Keywords: Bipolar disorder; Ethacrynic acid; Na,K-ATPase; Sodium; Sodium pump

Manic-depressive, or bipolar, illness is a relatively

common condition afflicting 1% of the world’s pop-

ulation (Goodwin and Jamison, 1990). Several mod-

els have been presented. These span neurotransmitter,

G protein (Avissar and Schreiber, 1989), second

messenger (Warsh and Li, 1996; Manji et al., 1999;

Emamghoreishi et al., 2000), and ionic regulation

0165-0327/$ - see front matter D 2003 Elsevier B.V. All rights reserved.

doi:10.1016/S0165-0327(03)00044-2

* Corresponding author. Tel.: +1-502-852-1124; fax: +1-502-

852-1115.

E-mail address: [email protected]

(R.S. El-Mallakh).1 Present address: Sun Health Institute, Sun City, AZ, USA.

abnormalities (Dubovsky and Franks, 1983; El-Mal-

lakh and Wyatt, 1995). The latter is particularly

promising since it may underlie a common mechanism

of mood stabilization (El-Mallakh and Huff, 2001) and

has led to an interesting animal model of mania

(Ruktanonchai et al., 1998; Decker et al., 2000).

Both mania and bipolar depression have been asso-

ciated with increased intracellular sodium (Na) con-

centrations (Coppen et al., 1966; Shaw, 1966), and a

decrease in erythrocyte sodium and potassium-activat-

ed adenosine triphosphatase (Na,K-ATPase or Na

pump) activity (Looney and El-Mallakh, 1997). Tem-

poral gray matter of post mortem tissue obtained from

Page 2: Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid

Table 1

Lymphoblastoid cell lines used in the studies were matched for age

and gender

Patients Controls

Cell line Age Sex DOI Cell line Age Sex

GM05933A 25 F 1 GM06859A 25 F

GM05977A 26 M ? GM06160 25 M

GM05987B 30 F ? GM05392A 27 F

GM05989B 25 M ? GM07752 17 M

GM05999A 22 F 6 GM07544 22 F

GM07783 25 F 1 GM05377 24 F

GM08825 46 F ? GM05380 32 F

GM08852 34 F ? GM06862 34 F

GM09138 48 M ? GM05398A 44 M

GM09193A 54 M ? GM11500 45 M

GM09732 25 M 6 GM06875 26 M

GM10336A 21 F 6 GM07524 22 F

GM11051B 35 F 17 GM05423 32 F

GM11194 24 F 2 GM06051 26 F

GM11204 53 F 24 GM11120 58 F

DOI, duration of illness in years; F, female; M, male.

R. Li, R.S. El-Mallakh / Journal of Affective Disorders 80 (2004) 11–1712

bipolar subjects expresses less of the a2 isoform of the

Na pump than psychiatrically normal controls (Rose et

al., 1998). It has been purported that a primary increase

of intracellular Na can bring about activation of second

messengers without the presence of a neurotransmitter

signal (El-Mallakh and Li, 1993). Consequently, a

primary Na,K-ATPase abnormality may unify several

pathophysiological hypotheses.

Abnormalities in Na pump regulation in bipolar

subjects have previously been reported by two groups.

In 1981, Naylor and Smith reported that, unlike cells

from normal controls, lymphocytes obtained from

bipolar subjects failed to upregulate Na pump expres-

sion in response to treatment with 100 AM ethacrynic

acid for 3 days. In 1991, Wood et al. replicated Naylor

and Smith’s finding in unmedicated bipolar subjects.

The current study was performed to determine if

lymphoblastoid cell lines might respond in a similar

manner to lymphocytes, and to attempt to replicate and

expand previous reports.

1. Materials and methods

1.1. Lymphoblastoid cell cultures

Lymphoblastoid cell lines were obtained from the

National Institute of General Medical Science’s Hu-

man Genetic Mutant Cell Repository at the Corriell

Institute for Medical Research, Camden, NJ. Bipolar

subjects had a type I illness (as defined by Research

Diagnostic Criteria (Feighner et al., 1972)) and were

members of the Old Order Amish community (Ege-

land et al., 1987; Kelsoe et al., 1989). Control subjects

were screened for medical and psychiatric disease,

matched for age and gender, but were from a hetero-

geneous, outbred community (Table 1). Cells were

grown in RPMI-1640 medium with 15% fetal bovine

serum (FBS), 1% glutamine, at 37 jC and 5% CO2.

All cells were seeded at 0.2 million cells/ml in 75-ml

flasks and split every 2–4 days.

1.2. Cell treatment

Ethacrynic acid (Sigma, St. Louis, MO) was added

at final concentrations of 10�7, 10�6, or 10�5M for 72 h.

Cells were harvested after drug treatment and spun at

250�g for 3 min. The pellets were used for all assays.

1.3. Ouabain binding assay

Lymphoblast cell pellets were resuspended at 2

million cells/ml in K-free Krebs incubation solution

(pH 7.4) which contained 140 mMNaCl, 5.4 mMKCl,

1.2 mM MgSO4, 0.3 mM KH2PO4, 2.8 mM CaCl2,

12.0 mM HCl, 13.7 mM Tris base, 11.1 mM glucose,

and 1% FBS. Cells were incubated with a final con-

centration of 5, 25, 50, 75, or 100 nM [3H]ouabain

(DuPond NEN, Boston, MA) for 6 h at 37 jC. Theincubation was stopped by cooling in ice bath and rapid

vacuum filtration through Whatman GF/B filters

(Brandel, Gaithersburg, MD) followed by three, 5-ml

washes with ice-cold Kreb’s wash solution (same as the

incubation solution minus the glucose and FBS, pH

�7.4). The radioactivity on the filters was counted by a

Multipurpose scintillation counter (Beckman model

LS6500, Arlington Heights, IL). To obtain specific

binding, non-specific binding, determined in the pres-

ence of 10�4 M non-radioisotopic ouabain (Sigma),

were subtracted from the total binding.

1.4. 86Rb uptake assay (Na pump activity assay)

Lymphoblast cell pellets were resuspended in sa-

line solution at 2 million cells/ml. One hundred ml of

5 mM Ringer solution (123 mM NaCl, 1.3 mM CaCl2,

Page 3: Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid

R. Li, R.S. El-Mallakh / Journal of Affective Disorders 80 (2004) 11–17 13

1.2 mM MgSO4, 18.5 mM Na2HPO4, 100 mM KCl,

0.8 mg/ml sucrose, pH 7.4), and 100 ml of 1 ml

ouabain (Sigma) were added and incubated at 37 jCfor 30 min before adding 86Rb (DuPont NEN) at 2

ACi/ml in a total volume of 1 ml. The cell suspension

was then incubated at 37 jC for 1 h and pellets were

collected after spinning at 2700�g for 1 min. The

pellet was washed three times with ice-cold K-free

Ringer solution. Cell pellets were then mixed with

500 ml of 5% trichloroacetic acid solution (Fisher

Scientific, Fair Lawn, NJ), and spun at 5000�g for 15

min. Two hundred ml of the supernatant were added

to 5 ml Econo-Safe scintillation fluid (Research Prod-

ucts International, Mount Prospect, IL), and radioac-

tivity was measured by the Multi-purpose scintillation

counter.

1.5. Intracellular Na, K, and calcium measurements

Lymphoblast cell pellets were washed with isoton-

ic (0.1 M) MgCl2 solution, pH 7.4 (Sigma), three

times and lysed overnight with 30% nitric acid (Fish-

er). A flame atomic absorption spectrophotometer 372

(Perkin-Elmer) was used to measure Na, K, and

calcium (Ca) concentrations. Lamp current, wave-

length, and slit settings are shown in Table 2. Distilled

deionized water was used as background on all

analyzed samples. Data were calculated based on

daily standard curves and equalized per mg protein.

The concentrations of the standards were 10�5 M,

5�10�5 M, and 10�6 M.

1.6. Western blot

Lymphoblast cell pellets were homogenized with a

Microson Ultrasonic Cell Disrupter XL (Heat Sys-

tems, Farmingdale, NY). The protein was measured

by Lowry Assay using a Protein Assay Kit (Bio-Rad

Table 2

Settings for ion measurement on flame atomic absorption

spectrophotometer

Ion Lamp Wavelength Slit

current (k) setting

(V) (nm)

K+ 12 766.5 2.0

Na+ 12 589.0 0.7

Ca2+ 15 422.7 0.7

Laboratories, Hercules, CA). A sample of 15 or 30 mg

of protein was mixed with sample buffer and loaded

into 8% SDS gel (Bio-Rad). The sample buffer

contained 50 mM Tris–HCl (pH 6.8), 5% glycerol,

0.1% SDS, 0.3% mercaptoethanol, and 0.02% bro-

mophenol. The Na pump protein was separated elec-

trophoretically at 60 V for 3 h in running buffer which

contained 25 mM Tris, 192 mM glycine, and 0.1%

SDS. Protein was then transferred overnight from gel

to PVDF nitrocellulose membrane (Millipore, Bed-

ford, MA) at 40 V in a transfer buffer which contained

25 mM Tris, 192 mM glycine, and 0.5% SDS. The

membrane was blotted at 4 jC overnight with 5%

milk (Nestle Food Company, Glendale, CA), and

probed with mouse anti-Na,K-ATPase a1 monoanti-

body (UBI, Lake Placid, NY) for 1 h in 5% milk in

TBS with 0.1% Tween-20 (Sigma). The membrane

was washed with 0.1% Tween-20 in TBS three times

before blotted with horseradish peroxidase-conjugated

goat anti-mouse IgG secondary antibody (Rockland,

Gilbertsville, PA) for 1 h. The membrane was again

washed three times with 0.1% Tween-20 in TBS and

soaked in ECL developing solution mixture (Amer-

sham, Arlington Heights, IL) for 1 min before expo-

sure to Kodak XAR-5 imaging film (Eastman Kodak,

Rochester, NY). The product of the chemilumines-

cence reaction (a horseradish peroxidase system,

ECL, Amersham) was quantified with the Personal

Densitometer SI (Molecular Dynamics, Sunnyvale,

CA). The actual amount of protein loaded was quan-

tified by staining the membrane with Coomassie Blue

R-250/50% methanol–10% acetic acid for 1 min,

which was then destained by immersion in 80%

methanol–10% acetic acid for 3 min, followed by

10 min in 50% methanol–10% acetic acid (Sigma).

The resulting stain was photocopied and the image

quantified on the Personal Densitometer SI. The

number was used for the denominator in all reported

data.

1.7. RNase protection assay

Total RNA (10 ng) was dissolved in the hybrid-

ization buffer (40 mM Pipes, pH 6.4, 80% deionized

formamide, 0.4 M NaCl, 2 mM EDTA) and then

added to the radiolabelled antisense RNA probe

(1�10�6 cpm). (The a1 isoform oligoprobe was

designed using GenBank data with the following

Page 4: Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid

Fig. 1. Effect of 3 days of treatment with ethacrynic acid 10�5 M on several measures of Na,K-ATPase expression and function in

lymphoblastoid cells from bipolar and psychiatrically normal individuals. (A) [3H]Ouabain binding is lower in cells derived from bipolar

subjects (93.8F8.22% (S.E.M.) of baseline) than psychiatrically normal individuals (130.9F13.76% of baseline, P=0.03, F=5.2). (B)

Immunoblot density measures of the a1 subunit increases in cells of normal controls (149.8F17.01% of baseline) but does not change in

bipolar-derived cells (92.8F8.67% of baseline, P=0.009, F=8.36). (C) The mRNA signal of the a1 subunit as measured by RNase protection

increases in response to ethacrynic acid in normal cells (157.2F32.02% of baseline), but drops in bipolar-derived cells (72.7F18.78% of

baseline, P=0.046, F=5.18). (D) Na,K-ATPase activity as measured by 86Rb-uptake increases in normal control-derived cells (123.2F12.41% of

baseline) but decreased in bipolar-derived cells (80.5F4.14% of baseline, P=0.0034, F=11.61). (E) Ethacrynic acid treatment at 10�5 M

increases intracellular Na concentration in bipolar-derived lymphoblasts (128.3F18.12% of baseline) but not in cells derived from

psychiatrically normal controls (92.7F4.82% of baseline, P=0.04, F=4.93).

R. Li, R.S. El-Mallakh / Journal of Affective Disorders 80 (2004) 11–1714

Page 5: Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid

R. Li, R.S. El-Mallakh / Journal of Affective Disorders 80 (2004) 11–17 15

sequence: 5V-CTCAGCTGCACGAGCAGTGT-

TAATCCCCGGCTCAAGTCTGTTCCATA-3V). The

antisense RNA probe was transcribed with T7 RNA

polymerase in the presence of [32P]UTP. The hybrid-

ization mixture was incubated at 45 jC overnight

(>14 h), and then treated with RNase A (40 mg/ml)

and RNase T1 (70 U/ml) at 37 jC for 30 min. The

samples were treated with proteinase K (100 mg/ml)

at 37 jC for 15 min. and then electrophoresed on a 5%

polyacrylamide denaturing gel (8 M urea) at 150 V for

3 h. Gels were dried on Whatmann filter paper (What-

mann, Maidstone, UK) at 80 jC for 3 h and placed on

intensifying screen and exposed to Kodak XAR-5

film (Eastman Kodak) for 24 h at �70 jC.

2. Results

On all measures of Na pump expression or activity

there was a significant difference between bipolar-

and normal control-derived lymphoblasts. Specifical-

ly, cells from control populations responded to 3-day

treatment with ethacrynic acid by increasing Na,K-

ATPase synthesis and activity, while cells from bipo-

lar individuals did not respond (Fig. 1).

[3H]Ouabain binding is a measure of the number of

active Na pump units since ouabain binds only to

enzymes that are actively transporting ions. Ethacrynic

acid, at 100 AM (10�5 M) for 72 h, induced an increase

of 130% above baseline in normal cells. Cell derived

from bipolar subjects failed to show this response, and

remained unchanged at 94.5% (Fig. 1A) (P<0.05).

The affinity of ouabain binding was non-significantly

decreased in bipolar subjects (Kd=4.13F0.67 (S.E.M.)

at baseline to 3.94F0.67 nM after ethacrynic acid

treatment), but not in normal controls (4.94F1.05 to

5.0F1.57 nM). However, the number of ouabain

binding sites was significantly increased in cells from

normal individuals (Bmax increased from 33.5F2.1 to

46.8F1.99, P=0.001, after ethacrynic acid 10�5 M for

72 h), while cells from bipolar subjects showed no

change (36.4F4.99 to 33.6F6.15).

Similarly, the Western blot technique quantifies the

total amount of Na,K-ATPase present in the cells,

including inactive pumps. Ethacrynic acid induced an

increase of a1 isoform protein expression to 151.% in

normal cells (Fig. 1B). Cells from bipolar individuals

did not respond (92.8%) (Fig. 1B). The a2 and a3

isoforms are not expressed in lymphoblasts.

The difference in response to ethacrynic acid

appears to have its origins in the regulation of tran-

scription. In pilot experiments with RNase protection

technique that quantifies a1 isoform mRNA synthe-

sis, treatment with ethacrynic acid increased the

quantity of mRNA in cells from normal individuals

(153.3%) but decreased the quantity in bipolar-de-

rived cell lines (74.8%) (Fig. 1C).

Changes in Na pump activity as measured by 86Rb-

uptake parallel the changes noted in pump expression.

In normal cells ethacrynic acid induced a significant

increase in 86Rb-uptake (156.8%) while a small de-

crease was found in cells from bipolar individuals

(72%) (Fig. 1D).

Changes in intracellular Na concentration ([Na]in)

are consistent with changes seen in pump expression

and activity. Cells from normal individuals showed

essentially no change in [Na]in (92% of baseline) (Fig.

1E). Cells from bipolar individuals showed an in-

crease of [Na]in to 128% of baseline (Fig. 1E). Neither

intracellular K (bipolar 107%, normal control 106% of

baseline), nor total intracellular Ca (bipolar 90.6%,

normal control 90.1% of baseline) were altered.

3. Discussion

Lymphoblastoid cells derived from bipolar subjects

lack the ability to respond to ionic stress induced by

ethacrynic acid. This abnormality is evident at various

levels. First, there is a significant increase in ouabain

radioligand binding in cells from normal individuals

without any observed changes in cells derived from

bipolar individuals (Fig. 1A). Kinetic data indicate

that there is a significant increase in the number of

ouabain binding sites in cells from normal individuals

after ethacrynic acid treatment, while cells from

bipolar subjects show a slight reduction in Bmax.

Second, Na pump activity increased in normal cells

as reflected by 86Rb uptake but not in cells derived

from bipolar subjects (Rb replaces K in K-free sol-

utions) (Fig. 1D). There was no significant difference

in Na pump activity between these two groups prior to

ethacrynic acid treatment. Third, there was a signifi-

cant increase of [Na]in in cells from bipolar patients

after ethacrynic acid stimulation, but there was no

Page 6: Differential response of bipolar and normal control lymphoblastoid cell sodium pump to ethacrynic acid

R. Li, R.S. El-Mallakh / Journal of Affective Disorders 80 (2004) 11–1716

change in cells derived from normal individuals (Fig.

1E). Fourth, quantification of protein expression of Na

pump a1 isoform with Western technique indicated

that ethacrynic acid increases protein expression of a1

in cells derived from normal individuals, but not in

cells from bipolar individuals (Fig. 1B). Finally,

mRNA quantification by RNase protection indicates

transcription in normal cells but not in cells derived

from bipolar subjects (Fig. 1C).

The changes seen in the lymphoblastoid cell lines

studied herein reflect those reported previously in

fresh lymphocytes obtained from bipolar patients

and matched normal controls (Naylor and Smith,

1981; Wood et al., 1991). These earlier studies exam-

ined ouabain-binding and Na pump activity. The

current study has extended these observations to the

mRNA level and associated physiological changes in

Na concentration.

These data suggest that individuals with bipolar

illness may be unable to appropriately regulate Na

pump expression and activity in response to etha-

crynic acid challenge. Ethacrynic acid has several

actions on the Na pump. First, ethacrynic acid inhibits

the Na pump by blockade of phosphorylation and

prevention of ADP–ATP exchange. Second, etha-

crynic acid can decrease, slightly, the apparent affinity

for K by stabilizing the phosphorylated intermediate

in the pump exchange cycle. Both Rb-uptake and

ouabain binding are decreased by ethacrynic acid

treatment in the 10�4 to 10�6 M range. Sodium pump

inhibition results in an increase of intracellular Na

concentration. Normal cells respond to these changes

by increasing Na pump expression and activity. Cells

from Old Order Amish bipolar subjects did not exhibit

these responses and showed an increase of intracellu-

lar Na concentrations.

Mood state-associated increases of intracellular Na

concentrations have been reported in both mania and

bipolar depression (Coppen et al., 1966; Shaw, 1966).

Elevations of intracellular Na could bring about ele-

vations of free ionic Ca (Alexander et al., 1986;

Reddy et al., 1992), and could activate second mes-

senger systems in the absence of a first messenger

signal (El-Mallakh and Li, 1993). Higher intracellular

Na concentrations may also reduce the potential

difference across the membrane, resulting in more

easily stimulated neurons, a reduced number of neu-

rotransmitter quantal release with each action poten-

tial, and a slower clearance of free ionic Ca at the

presynaptic terminal (El-Mallakh and Wyatt, 1995;

El-Mallakh and Jaziri, 1990). If the increase in intra-

cellular Na concentration is related to a decrease of

Na,K-ATPase activity, recovery of the neuronal mem-

brane potential may be impaired and indices such as

the H-reflex recovery would be prolonged (El-Mal-

lakh et al., 1996). These events have been previously

purported to be central to the pathogenesis of patho-

logical mood states in bipolar illness (El-Mallakh and

Wyatt, 1995). The observations of the current study

support the idea that an abnormality in regulation of

the Na,K-ATPase may be a characteristic of bipolar-

derived tissues.

However, there are limitations to the current study

that prevent encompassing conclusions. The age of

the subjects is relatively young (31.3 years for

controls, 32.9 years for bipolar individuals). While

normal controls were carefully screened to rule out

psychiatric disease, they are not beyond the age of

risk for developing a mood disturbance. Additionally,

control subjects were not from the Old Order Amish

community (with the exception of two Amish con-

trols). Since this community had been established by

a small number of individuals, the difference in Na

pump response may be a reflection of a founder

effect, rather than a disease effect. Finally, the

utilization of a unique, inbred bipolar population

dictates that caution be exercised in generalization

of the data. These weaknesses are less striking when

one considers that similar observations have been

previously noted in lymphocytes of general commu-

nity-based bipolar samples (Naylor and Smith, 1981;

Wood et al., 1991).

In summary, the current study demonstrates a

divergent and anomalous Na pump response in live

leukocytes possessing the genetic heritage of bipolar

individuals. This abnormality may be central to the

pathogenesis of the disease states in this condition. At

the very least, these studies offer clues to the cellular

abnormalities that underlie bipolar illness.

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