Cohort study on somatoform disorders in
Parkinson disease and dementia with
Marco Onofrj, MD
Laura Bonanni, MD,
PhD
Lamberto Manzoli, MD
Astrid Thomas, MD,
PhD
Address correspondence and
reprint requests to Prof. Marco
Onofrj, Department of
Neuroscience and Imaging,
University “G. d’Annunzio” of
Chieti-Pescara, Italy, Aging
Research Center, Ce.S.I.,
“G. d’Annunzio” University
Foundation, Chieti-Pescara, Italy,
Via dei Vestini, 66013 Chieti, Italy
onofrj@unich.it
Supplemental data at
www.neurology.org
1598
Lewy bodies
ABSTRACT
Objective: To assess somatoform disorder (SFMD) prevalence and impact in Parkinson disease
(PD) and dementia with Lewy bodies (DLB).
Methods: SFMD were assessed by direct observation of symptoms in the year coincident (⫾6
months) with definite diagnosis of PD, DLB, Alzheimer disease, multiple system atrophy, progres-
sive supranuclear palsy, or frontotemporal dementia, and by interviews with patients, caregivers,
and general practitioners, and reviews of prior hospital admissions, in a cohort of 942 patients
with neurodegenerative disorders. Matched groups of patients with PD and patients with DLB
without vs with SFMD were selected for comparisons and followed up over 4 years.
Results: The frequency of SFMD was higher in DLB (15 patients, 12%) and PD (29 patients, 7%)
than in other neurodegenerative diseases (0%–3%). SFMD consisted of conversion motor or
sensory disorders, often accompanied by delusional thought content; in one patient catatonic
symptoms were observed concomitantly with PD diagnosis. Evidence of SFMD symptoms, pre-
ceding diagnosis by 6 months–10 years, was obtained in 28 patients with PD and all patients with
DLB. A total of 22 patients with PD and all patients with DLB could be followed for 4 years. SFMD
symptoms recurred during follow-up, with catatonic signs developing in 9 patients with PD and 8
patients with DLB. Most baseline demographic and clinical features did not differ between sub-
jects with or without SFMD. Decline of cognitive function was greater in PD-SFMD patients than
in those without SFMD (p ⬍ 0.01); it was comparable to that observed in DLB.
Conclusions: The frequency of somatoform disorder (SFMD) (with catatonic signs) in Parkinson
disease and dementia with Lewy bodies suggests that SFMD are part of the spectrum of Lewy
body diseases. Neurology® 2010;74:1598 –1606
GLOSSARY
AD ⫽ Alzheimer disease; ANOVA ⫽ analysis of variance; BDI ⫽ Beck Depression Inventory; CAF ⫽ Clinician Assessment of
Fluctuations; DLB ⫽ dementia with Lewy bodies; DRS-2 ⫽ Dementia Rating Scale–2; DSM-IV ⫽ Diagnostic and Statistical Man-
ual of Mental Disorders, 4th edition; FAB ⫽ Frontal Assessment Battery; FTD ⫽ frontotemporal dementia; GP ⫽ general practitio-
ner; H&Y ⫽ Hoehn & Yahr; MMSE ⫽ Mini-Mental State Examination; MSA ⫽ multiple system atrophy; NPI ⫽ Neuropsychiatric
Inventory; PD ⫽ Parkinson disease; PDD ⫽ Parkinson disease with dementia; PSP ⫽ progressive supranuclear palsy; SCL-90R ⫽
Symptom Checklist-90-R; SFMD ⫽ somatoform disorder; UPDRS ⫽ Unified Parkinson’s Disease Rating Scale.
Recent literature on the psychiatric manifestations of Parkinson disease (PD)1 identified de-
pression and impulse control disorders as mental disorders preceding or occurring concomi-
tantly with parkinsonism and its treatment. Yet, “other mental symptoms” need to be
“recognized as sources of disability for patients with PD”2: a recent survey on patients hospital-
ized for PD3 found that prior hospitalizations for mental disorders other than depression
equally increased the risk of PD occurrence. The present study hypothesized that somatoform
disorders (SFMD) might be part of the presentation of PD and dementia with Lewy Bodies
(DLB).
SFMD is the general category grouping, on the basis of medically unexplained symptoms,
somatization and conversion disorders (once defined as hysteria), pain, and hypochondria,
while delusional disorders somatic type identifies a bordering category.4 Several reasons an-
From the Department of Neurology (M.O., L.B., A.T.), University G. d’Annunzio of Chieti-Pescara, Italy, and Aging Research Center, Ce.S.I., “Gabriele
d’Annunzio” University Foundation; and Section of Epidemiology and Public Health (L.M.), University G. d’Annunzio of Chieti-Pescara, Italy.
Disclosure: Author disclosures are provided at the end of the article.
Copyright © 2010 by AAN Enterprises, Inc.
chored our interest on SFMD in PD: SFMD
might precede the onset of neurodegenerative
disorders,5,6 conversion type SFMD were de-
scribed anecdotally in patients who developed
PD7–9, alexithymia (inability to distinguish
feelings from bodily sensation of emotional
arousal) is increased in PD,10 and, finally, the
remarkable intensity of response to placebo in
PD11,12 evidences that there is suggestibility in
PD and suggestibility was historically defined
as the hallmark of hysteria-SFMD.9,13 We
therefore designed a cohort study based on di-
rect observation of SFMD symptoms and
semistructured interviews, investigating hos-
pital and general practitioners’ (GP) reports,
describing 1) cohort prevalence of SFMD in
different neurodegenerative diseases; 2) pre-
sentation and phenomenology of SFMD
symptoms; and 3) comparisons between pa-
tients with PD and patients with DLB with
and without SFMD, at onset and during dis-
ease progression.
METHODS This prospective study was designed following
recommendation of the STROBE statement.14 It was based on a
cohort of 1,360 new patients evaluated from 1999 to 2008 in
our movement disorder and memory outpatient clinics in Cen-
tral Italy, Adriatic Coast, serving a population of 1,300,000. Re-
cruitment began in 2001 and ended in 2004 for PD and DLB,
and in 2006 for the other neurodegenerative diseases. Two cases,
initially considered incidental SFMD, were observed in 1999. As
a comparator prevalence estimate from the general population of
the same area referral system, we accessed patients with SFMD
from our psychiatry clinic, following 1,718 patients from 1999
to 2006.
Categorization of patients into SFMD and non-SFMD
groups was based on direct observation of symptoms in the year
(6 months before/after) coincident with the diagnosis of neuro-
degenerative diseases. As a second assessment, all patients under-
went semi-structured interviews by a rater blinded to neurologic
diagnoses. Interviews, based on DSM-IV-TR manual,4 investi-
gated somatic complaints with examples and a checklist pre-
sented to patients and caregivers, focusing on SFMD traits, such
as dependency, mannerism, viscosity, adoption of a sick role, and
histrionic dramatic descriptions of disease (e-Methods on the
Neurology® Web site at www.neurology.org). Evaluations of past
SFMD symptoms included information from prior hospital records
and reports from each patient’s GPs over the past 4 –20 years. In-
sight, somatic type delusional disorders were specifically investigated
as part of the Neuropsychiatric Inventory (NPI) assessment.15
The PD diagnosis was made according to the UK Parkin-
son’s Disease Society Brain Bank criteria.16 Parkinsonism was
evaluated with Unified Parkinson’s Disease Rating Scale
(UPDRS)17 and Hoehn & Yahr (H&Y) scale. Nonmotor symp-
toms of PD (olfactory and visual disorders, constipation, inner
tremor, pain)2 were analyzed as possible confounding factors.
Diagnosis of probable DLB was based on Consensus Criteria.18
Probable Alzheimer disease (AD) diagnosis was based on Na-
tional Institute of Neurological and Communicative Disorders
and Stroke–Alzheimer’s Disease and Related Disorders Associa-
tion criteria.19 Progressive supranuclear palsy (PSP) diagnosis was
based on National Institute of Neurological Disorders–Society
for Progressive Supranuclear Palsy criteria.20 Multiple system at-
rophy (MSA) diagnosis was based on Consensus Statement.21
Frontotemporal dementia (FTD) diagnosis was based on stan-
dardized clinical criteria.22 Neuroimaging and further diagnostic
evaluations are reported in e-Methods. All patients with PD,
DLB, MSA, or PSP underwent L-Dopa challenges.23 There were
no treatment restrictions for inclusion in the study.
All patients underwent Mini-Mental State Examination
(MMSE),24 Frontal Assessment Battery (FAB),25 NPI,15 and the
SFMD Semi-Structured Interview, and were followed for 2 years
in order to confirm diagnosis.
Two comparison studies were performed between patients
with PD and patients with DLB with and without SFMD.
The first compared 29 PD-SFMD and 15 DLB-SFMD pa-
tients at initial diagnosis with 87 patients with PD and 45 pa-
tients with DLB without SFMD, randomly selected and
matched for age (within 3 years) and disease duration
(6 months). These patients were also evaluated with the Clini-
cian Assessment of Fluctuations (CAF),26 the Beck Depression
Inventory (BDI),27 the SCL-90R, somatoform disorder sub-
item,28 and polysomnography for REM sleep behavior disorder.
All patients with PD with impulse control disorders or with in-
congruent neuroimaging studies or definite genetic mutations
were excluded from possible SFMD evaluation and from inclu-
sion in the matched PD group.
The second comparison was a 4-year follow-up assessment of
motor and cognitive outcomes. A total of 66 patients with PD
and 30 patients with DLB without SFMD, from the initial com-
parative group, were matched with SFMD patients completing
follow-up, for educational level (⫾2 years) and (MMSE) score
(⫾1 point), at onset of study. The 4-year follow-up study in-
cluded MMSE, Dementia Rating Scale–2 (DRS-2),29 and
UPDRS total assessments.
Institutionalization rates (admission to long-term care facil-
ity or need for full-time caregiver employment) were assessed in
the patients with PD and patients with DLB at 9 years after
study enrollment. Figure e-1 shows study design.
Standard protocol approvals, registrations, and patient
consents. The study received approval by our local ethical com-
mittee, was carried out according to the declaration of Helsinki
and subsequent revisions,30 and after complete description of the
study to the subjects or caregivers, written informed consent was
obtained. A written informed consent to disclose was obtained
by videorecorded patients. No patient names are spoken in the
videos.
Statistics. Normality distribution of variables was assessed with
Shapiro-Wilk test expressing means and standard deviations for
normally distributed continuous variables; medians and inter-
quartile ranges for other continuous variables; and percentages
for categorical variables. Differences in continuous test scores
across groups were evaluated using one-way analysis of variance
(ANOVA) with Bonferroni correction and Fisher exact test for
categorical variables. Wilcoxon matched-pairs signed-ranks test
assessed within-group variations in tests’ median score between
time 0 and 2– 4 years. Mean scale score changes over time be-
tween and among groups was assessed with 2-way ANOVA and
confirmed with linear regression (age, education as predefined
potential confounders). Time to institutionalization was evalu-
ated by log-rank test, represented by Kaplan-Meier estimates.
Neurology 74 May 18, 2010 1599
 Table 1 Comparison of the prevalence of somatoform disorders and catatonia in PD-SFMD and patients
with DLB, AD, PD, PSP, MSA, and FTDa

DLB AD PD PSP MSA FTD

(n ⴝ 124) (n ⴝ 303) (n ⴝ 412) (n ⴝ 40) (n ⴝ 36) (n ⴝ 28)

Age, y, mean (SD) 70.2 (8.3) 72.0 (10.6) 63.9 (12.5) 65.1 (2.1) 64.8 (3.2) 66.0 (3.0)

MMSE, mean (SD) 22.8 (2.5) 23.5 (3.0) 27.2 (1.6) 25.2 (1.3) 26.6 (1.4) 22.0 (1.5)

FAB, mean (SD) 14.6 (2.5) 15.5 (2.8) 17.0 (3.0) 11.0 (1.3) 16.8 (1.2) 11.7 (1.8)

NPI, mean (SD) 20.5 (8.2) 11.8 (5.6) 5.0 (3.8) 6.1 (4.4) 5.5 (2.7) 14.4 (5.4)

Presence of SFMD, n (%) 15 (12.0) 3 (1.0) 29 (7.0) 1 (2.5) 0 0

SFMD interview positive, n (%) 28 (22) 7 (2.3) 40 (9.7) 1 (2.5) 0 0

No. (%) of hospitalizations 4.0 (2.6) 3.8 (2.1) 1.8 (1.6) 4.5 (2.1) 4.2 (3.0) 3.2 (2.2)

No. of hospital discharges with 32/16 8/4 36/29 6/1 0/0 0/0
definite SFMD diagnosis/no. of
patients

Abbreviations: AD ⫽ Alzheimer disease; DLB ⫽ dementia with Lewy bodies; FAB ⫽ Frontal Assessment Battery; FTD ⫽
frontotemporal dementia; MMSE ⫽ Mini-Mental State Examination; MSA ⫽ multiple system atrophy; NPI ⫽ Neuropsychiat-
ric Inventory; PD ⫽ Parkinson disease; PSP ⫽ progressive supranuclear palsy; SFMD ⫽ somatoform disorder.
a
Presence of SFMD indicates actual observation of SFMD symptoms in n or % of patients. Positive interview indicates that
semistructured interview with patients and caregivers reported several aspects of histrionic health concern, medically
unexplained symptoms, confirmed by general practitioner. Number of hospitalization indicates hospital admissions for any
clinical reason. Data are reported as mean (SD) unless otherwise stated.

Constant incidence ratios in follow-up were checked by Nelson-
Aalen cumulative hazard estimates.
Analyses were made using Stata, version 10.0 (Stata Corp.,
College Station, TX).
RESULTS Prevalence of SFMD in different neurode-
generative diseases. Of 1,360 new patients evaluated
from 1999 to 2008, 42 patients were classified as
affected by neurodegenerative disorders with a
follow-up of at least 2 years: PD or genetic parkin-
sonisms (n ⫽ 412); AD (n ⫽ 303); DLB (n ⫽ 124);
PSP (n ⫽ 40); MSA (n ⫽ 36); FTD (n ⫽ 28).
SFMD symptoms were observed in the year coinci-
dent with diagnosis in 48 patients. Most of these sub-
jects had PD (n ⫽ 29, 7%) or DLB (n ⫽ 15, 12%).
Three had AD (1%), 1 had PSP (2.5%), and none
had MSA or FTD (p ⬍ 0.001 for comparisons be-
tween PD or DLB with any other group). The propor-
tion of patients hospitalized for SFMD symptoms, the
number of hospital admissions related to SFMD symp-
toms, and the overall prevalence of hypochondria or
SFMD prior to the primary neurologic diagnosis result-
ing from interviews were highest in the patients with
PD and patients with DLB (table 1). In 26 of the 29
patients with PD and 13 patients with DLB presenting
with SFMD symptoms, the semi-structured interviews
and GP reports evidenced coexisting hypochondria and
SFMD features. Interviews evidenced SFMD traits in
28 more patients (table 1), but these patients were not
enrolled in the study because direct observations or
prior hospital records sufficient to document SFMD
were not obtained.
In the reference population of patients from Psy-
chiatry, 25/1,728 (1.5%) received the diagnosis of
SFMD; none of these presented neurodegenerative
diseases during a 2-year follow-up (5 developed bipo-
lar disorders, 3 delusional disorders, 1 committed
suicide).Their age range was 39 ⫾ 5 years at the time
of SFMD diagnosis, and female predominance was
76% (19/6), highly different ( p ⬍ 0.01) from any
neurodegenerative disease group.
Temporal presentation and phenomenology. The re-
ferral diagnosis was PD in 21/29 patients; overlap-
ping SFMD symptoms were coexistent or emerged
within the first 6 months. In 5 patients, the reason
for referral was SFMD but PD signs became evi-
dent within 1–5 months. Three patients were orig-
inally classified as incidental PD-SFMD, but
developed PD signs in the context of SFMD in
3–5 and 6 years.
In 28/29 PD-SFMD patients, SFMD symptoms
could be documented at earlier times through inter-
view and hospital records. In 21, records docu-
mented SFMD symptoms preceding PD by 5–10
years. In 8 patients, hospital referrals diagnosed disor-
ders bordering on SFMD: fibromyalgia (3), causalgia
(2), temporomandibular joint pain (1), pseudoseizures
(1), and dissociative amnesias (1). In 26, interviews evi-
denced history of hypochondriasis 10 –25 years before
PD diagnosis.
Follow-up data were available on 22/29 patients.
In 20/22, recurrent SFMD symptoms were docu-
mented in the 1–2 years follow-up, and in 18/22 in
the 2– 4 years follow-up. SFMD symptom categories
fluctuated and all patients presented, in different
time periods, different motor, sensory, or pain symp-
toms. All patients had further SFMD symptoms be-
fore or after PD diagnosis.

1600 Neurology 74 May 18, 2010

 chogenic parkinsonian signs (distractible tremor),
Table 2 SFMD phenomenology in PDa
followed by olfactory hallucinations, were ob-
Coincident served, for 160 days, as incidental SFMD in 1 pa-
No. of with In Prior to
patients diagnosis follow-up diagnosis tient 5 years before the PD diagnosis. Videos 1–3
show paretic or postural symptoms and catatonic
SFMD symptoms PD DLB PD DLB PD DLB PD DLB
signs (see appendix e-1: video legends).
Total 29 15 29 15 22 15 29 15 Sensory conversion consisted of transient and re-
Hypochondriasis, % 90 87 90 87 90 87 90 87 curring symptoms categorized as simple anesthesia,
Motor conversion, % complex delusions of progressive body deformation
Paresis 76 60 28 33 48 7 31 40 (dysmorphogenesis or partial Cotard syndrome,31 in-
Abnormal postures 38 47 24 20 21 27 0 7 cluding ingrowth of body mass, penis shrinkage, mi-
Globus pharyngis 52 47 0 7 52 47 7 7
grating allergies), and recurrent multifocal pain with
gastrointestinal symptoms. All could be distin-
Psychogenic parkinsonism 3 0 0 0 0 0 3 0
guished from nonmotor PD signs because of con-
Catatonic signs 31 53 3 7 31 53 0 13
comitant thought content abnormalities or histrionic
Sensory conversion, %
representation of discomfort (retching, burping, glo-
Anesthesia 17 53 10 7 7 27 7 27
bus pharyngis, bizarre avoidance or manipulation be-
Body deformation delusions 45 20 17 7 10 7 42 7 haviors, insistent demand for invasive medical
Multilocalized pain with 100 73 17 27 69 27 93 53 procedures). Motor or sensory conversion symptoms
gastrointestinal symptoms
were never reduced by dopaminergic treatments.
Abbreviations: DLB ⫽ dementia with Lewy bodies; PD ⫽ Parkinson disease; SFMD ⫽ so-
matoform disorder. Comparative studies. Comparisons at initial diagnosis
a
Further details of conversion symptoms are reported in Results. Prevalences of SFMD of PD or DLB between patients with or without
symptoms in PD are calculated based on the number of patients with PD at onset of the SFMD matched for age and disease duration showed
comparative studies. Note that catatonic signs were reported in 2 patients before DLB
diagnosis.
no differences for patients with DLB and only minor
differences for patients with PD. University educa-
The same patterns of recurrent and shifting SFMD tion was significantly more frequent in PD-SFMD
symptoms during follow-up and in prior hospital patients. Gender, neuropsychological test scores, and
records were observed in the 15 DLB-SFMD patients. L-dopa equivalent doses were similar32 (table 3).
Hypochondriasis was reported in 13 patients. SCL-90R somatic disorder subitem and NPI
The mean number of SFMD recurrences during scores were higher in PD-SFMD patients compared
the 4-year follow-up was 3.4 ⫾ 1.6 in PD-SFMD to patients with PD without SFMD ( p ⬍ 0.05), but
and 1.5 ⫾ 1.0 in DLB-SFMD ( p ⬍ 0.001). Dura- no differences were observed in other SCL-90R sub-
tion of symptoms ranged from 7 days (globus phar- items. Depression was equally present in patients
yngis) to 180 days (body deformation). Table e-1 with and without SFMD as shown by BDI scores.
and table e-2 categorize SFMD symptoms in each The comparison with all newly diagnosed patients
PD and DLB patient as observed concomitantly with with PD (n ⫽ 236 without impulse control disorders
neurologic diagnosis, during follow-up, and as re- or genetic mutations) showed that PD-SFMD pa-
ported by interviews and hospital records. tients were older (70.6 ⫾ 5.5 vs 63.9 ⫾ 9.5, p ⬍
Table 2 reports categories and prevalences of 0.05). No feature distinguished the presence from
SFMD symptoms. Motor conversion symptoms in absence of SFMD in patients with DLB. MMSE,
PD and DLB consisted of histrionic representation DRS-2, and FAB scores, frequency of RBD and
of spastic paresis (stiff leg or bent knee with tiptoe- visual hallucinations, and treatment doses were all
ing), globus pharyngis, or exaggeration of late pos- statistically different ( p ⬍ 0.05– 0.01) in the com-
tural symptoms of PD, incongruent with disease parison DLB vs PD groups (with and without
duration or with specific phenomenology (e.g., SFMD) at baseline. Comparisons during and at the
lateral head tilt). Immobility and postural stereo- end of the 4-year follow-up between patients
typies typical of catatonia were observed in 9 pa- matched for age, disease duration, educational level,
tients with PD during recurrences (31%), in 1 and MMSE score at initial diagnosis showed instead
were observed concomitantly with PD onset, but relevant statistical differences (table 4).
hospital records documented prior SFMD symp- Rate and slope of MMSE and DRS-2 changes
toms 4 times in 6 years before PD diagnosis. Cata- through time separated PD-SFMD and both DLB
tonic signs were observed in 8 DLB-SFMD groups from PD without SFMD ( p ⬍ 0.001 for ev-
patients (53%): in 2 incidentally, in 1 2 years be- ery comparison) at each evaluation ( p ⬍ 0.001). Re-
fore diagnosis, in 1 coincidentally with diagnosis, gression analysis showed that MMSE and DRS-2 of
and in 5 during the first 2 years of follow-up. Psy- PD-SFMD and DLB patients with or without

Neurology 74 May 18, 2010 1601

 Table 3 Demographic, clinical, neuropsychological, and neuroimaging data at admission to the study:
comparisons of SFMD patients with age and disease duration–matched patients with PD and
patients with DLBa

PD-SFMD PD DLB-SFMD DLB

Variables (n ⴝ 29) (n ⴝ 87) (n ⴝ 15) (n ⴝ 45)

Age, y 70.6 (5.5) 70.1 (5.3) 72.0 (3.4) 73.5 (4.7)

Male, % 48 51 60 52

Educational level, y 12.9 (6.8) 12.3 (3.4) 12.1 (3.0) 12.2 (4.0)

University education, % of patients 68 29 32 28

Mini-Mental State Examination 27.1 (1.3) 27.2 (1.7) 23.3 (1.3) 23.1 (1.2)

Neuropsychiatric Inventory 9.3 (1.9) 3.7 (2.7) 11.1 (2.2) 11.2 (3.5)

Frontal Assessment Battery 16.8 (1.2) 17.3 (1.0) 14.5 (1.2) 14.5 (1.8)

Clinician Assessment of Fluctuation 0.0 (0.0) 0.0 (0.0) 4.7 (1.9) 4.6 (2.3)

REM sleep behavior disorders, % 38 24 60 67

Visual hallucinations, % 0 0 13 20

Somatization subscale of the Symptom Checklist 90R 32.2 (3.8) 22.3 (8.0) 22.0 (9.6) 22.5 (8.0)

Beck Depression Inventory 29.5 (8.7) 29.6 (7.6) 28.9 (5.5) 28.7 (7.0)

Unified Parkinson’s Disease Rating Scale–subscale III

BT 10.2 (2.9) 10.7 (2.9) 8.4 (2.6) 8.6 (2.2)

AT 6.0 (2.5) 7.1 (3.4) 6.0 (2.1) 5.8 (2.4)

Hoehn & Yahr staging 1.2 (0.3) 1.1 (0.2) 1.8 (0.4) 1.8 (0.3)

Total equivalent L-dopa dose 424 (44) 419 (50) 106.7 (40.6) 111.5 (22.0)

SPECT DAT scan, %

M 45 67 40 29

B 55 33 60 71

Abbreviations: AT ⫽ after dopaminomimetic treatment, during SFMD; B ⫽ bilateral posterior putaminal hypocaptation;
BT ⫽ before dopaminomimetic treatment; DLB ⫽ dementia with Lewy bodies; M ⫽ unilateral posterior putaminal hypocap-
tation; PD ⫽ Parkinson disease; SFMD ⫽ somatoform disorder.
a
Data are reported as mean (SD) unless otherwise stated. Equivalent L-dopa doses are dose administered at the end of the
year following PD or DLB diagnosis; 100 mg L-dopa ⫽ 1 mg pergolide, pramipexole, 4 mg ropinirole, 3 mg cabergoline.

SFMD could be described by a linear fit from base-
line to 4 years follow-up, thus showing that progres-
sion of cognitive decline (figure 1) was similar in
DLB and PD-SFMD patients and different from PD
without SFMD. Controlling for age and education,
the PD-SFMD, as compared to PD only patients,
showed a significantly lower reduction in DRS-2
score over time (regression coefficient for PD only
patients ⫽ ⫺8.16; 95% confidence interval ⫺7.55
to ⫺8.76; p ⬍ 0.001), confirming the large differ-
ence observed in the univariate analysis.
Progressive worsening of NPI scores was observed
in all groups, but significant differences distin-
guished PD-SFMD from PD without SFMD and
both DLB groups, throughout follow-up. Further as-
sessments evidenced the progression of cognitive and
behavioral decline in SFMD patients. Cognitive fluc-
tuations appeared in 9 PD-SFMD patients and, at 4
years, CAF score separated PD-SFMD from PD
group ( p ⬍ 0.001, table 4), while at baseline no dif-
ferences had been observed between the 2 groups.
VH were only present in DLB at baseline ( p ⬍
0.001), while 13 PD-SFMD patients had VH at the
end of study. Only 2 patients with PD without
SFMD reported VH at follow-up (table 4, p ⬍
0.001). At the end of the 9-year follow-up, all
PD-SFMD and DLB patients were institutionalized,
whereas none of the patients with PD required insti-
tutional care.
DISCUSSION Previous descriptions of SFMD7–9
and catatonic signs33,34 in PD are rare, and to our
knowledge, there are no reports presented on DLB.
This report shows that SFMD was identified in 7%
of patients with PD and 12% of patients with DLB
at the time of primary neurologic diagnosis and can
recur during follow-up. SFMD symptoms were more
frequent in PD and DLB than in AD, FTD, MSA,
and PSP, and more frequent than in our psychiatric
population. The rates in PD and DLB were higher
than rates reported for conversion or somatization
disorders in the general population.4
The design of the study, with patients included
only if actual SFMD signs were observed, with verifi-
cation through interviews (assessing psychiatric
traits), with hospital and GP reports, and with

1602 Neurology 74 May 18, 2010

 Table 4 Demographic, clinical, and neuropsychological data at admission to the study and after 2 and 4
years of follow-up in SFMD patients and patients with PD or DLB matched for education and
MMSE score at baselinea

PD-SFMD PD DLB-SFMD DLB

Variables (n ⴝ 22) (n ⴝ 66) (n ⴝ 15) (n ⴝ 30)

Age, y

Admission 70.7 (6.1) 70.0 (5.2) 72.0 (3.4) 73.2 (4.6)

Male, % 50 50 63 43

Mini Mental State Examination

Admission 26.8 (1.2) 27.2 (1.6) 23.3 (1.3) 23.0 (1.2)

2y 23.9 (1.5) 26.8 (1.5) 17.5 (1.4) 17.3 (2.2)

4y 17.9 (1.9) 26.4 (1.4) 12.1 (1.5) 12.0 (2.4)

Dementia Rating Scale–2

Admission 121.2 (2.8) 132.7 (2.8) 104.4 (13.9) 104.3 (14.7)

2y 106.4 (4.5) 128.3 (3.0) 88.7 (11.1) 88.6 (15.5)

4y 88.1 (10.5) 123.2 (3.2) 78.3 (10.2) 78.4 (12.4)

Neuropsychiatric Inventory

Admission 9.2 (2.0) 3.6 (2.7) 11.1 (2.2) 11.3 (3.4)

2y 13.6 (1.8) 4.0 (1.9) 19.7 (1.7) 18.9 (4.9)

4y 19.8 (3.5) 4.7 (1.9) 27.2 (3.0) 26.7 (3.6)

Frontal Assessment Battery

Admission 16.5 (0.4) 17.4 (0.9) 14.5 (1.2) 14.5 (1.9)

2y 14.4 (0.6) 16.6 (1.4) 11.5 (1.1) 11.6 (2.1)

4y 11.4 (0.6) 14.9 (2.1) 9.9 (1.0) 9.8 (1.8)

Clinician Assessment of Fluctuation

Admission 0.0 (0.0) 0.0 (0.0) 4.7 (1.9) 4.6 (2.2)

2y 0.2 (0.4) 0.1 (0.2) 5.9 (1.2) 6.2 (2.2)

4y 1.0 (1.2) 0.2 (0.6) 7.1 (1.6) 7.2 (2.4)

REM sleep behavior disorder, %

Admission 41 23 60 67

2y 68 42 80 87

4y 86 62 87 90

Visual hallucinations, %

Admission 0 0 13 20

2y 9 2 40 43

4y 59 3 93 93

Unified Parkinson’s Disease Rating Scale–subscale III

Admission 10.4 (3.1) 10.8 (2.9) 8.4 (2.6) 8.6 (2.1)

2y 22.8 (3.2) 23.6 (3.1) 10.9 (2.5) 11.1 (2.4)

4y 35.4 (3.6) 34.7 (3.8) 13.9 (2.6) 13.7 (2.9)

Hoehn & Yahr staging

Admission 1.2 (0.3) 1.1 (0.2) 1.8 (0.4) 1.8 (0.4)

2y 1.8 (0.3) 1.8 (0.3) 1.9 (0.3) 2.1 (0.2)

4y 2.2 (0.4) 2.1 (0.3) 2.0 (0.1) 2.4 (0.3)

Equivalent L-dopa dose, mg/day

Admission 390 (80) 407 (160) 106 (42) 113 (37)

2y 578 (50) 604 (62) 253 (62) 286 (58)

4y 780 (140) 857 (104) 310 (58) 336 (50)

Abbreviations: DLB ⫽ dementia with Lewy bodies; MMSE ⫽ Mini-Mental State Examination; PD ⫽ Parkinson disease;
SFMD ⫽ somatoform disorder.
a
Data are reported as mean (SD) unless otherwise stated.

Neurology 74 May 18, 2010 1603

 Figure 1 Comparison of Dementia Rating Scale–2 (DRS-2) slopes of progression in the 4 groups of patients
of the comparative study
The progression of cognitive decline in Parkinson disease–somatoform disorder (PD-SFMD) and in dementia with Lewy bodies
(DLB) was similar, as evidenced by similar decrements from the 2-year to the 4-year assessments in PD-SFMD and from the
admission to the 2-year assessments in DLB.
follow-up evaluations, allowed us to assess the consis-
tency of SFMD patterns during or before neurologic
disease course, and showed features of SFMD in PD
and DLB which were different from features re-
ported in DSM-IV-TR for conversion or somatiza-
tion disorders.4
Educational levels, gender, and age of patients
with PD and patients with DLB were different from
our psychiatric population and from literature.4 The
SFMD phenomenology was also partly different be-
cause SFMD in PD and DLB tended to recur, and to
shift among motor, sensory, and pain syndromes.
Symptoms were often characterized by delusional
qualities (body deformation, requests for invasive
medical procedures), and motor catatonic signs were
often observed (table 2, table e-1, table e-2). There-
fore, the general category of SFMD, rather than
DSM-IV-TR subcategories, was purposely used to
encompass variant presentations.
We acknowledge that we may have underesti-
mated the prevalence of SFMD, as PD-DLB patients
whose interviews were not matched by observation of
SFMD symptoms were excluded: recently high som-
atization SCL-90R scores were found in 40% of pa-
tients with PD,35 suggesting higher prevalence than
in our study. Nevertheless, obtaining consistency of
SFMD patterns was our intention. The main conclu-
sion of our study highlights not just the prevalence
but also the relevance and impact of SFMD on dis-
ease progression. We showed that identification of
1604 Neurology 74 May 18, 2010
SFMD in patients with PD has prognostic relevance:
SFMD had higher prevalence in patients with DLB
than in patients with PD, and while no differences
were observed initially between patients with PD
with or without SFMD, all PD-SFMD patients were
eventually diagnosed with PD with dementia
(PDD); slopes of progression of DRS-2, FAB, and
MMSE were similar in PD-SFMD and in DLB pa-
tients; and cognitive fluctuations and visual halluci-
nations appeared in DLB and PD-SFMD. All
findings suggest that SFMD in patients with PD is a
predictor of the development of dementia of the
DLB type.
The relationship of SFMD with dementia in
DLB and PD has practical implications.
First, in PD, identification of SFMD might be use-
ful to expand selection criteria for inclusion/exclusion of
patients in therapeutic procedures (e.g., transplant, elec-
trode implants) as inclusion of patients who predictably
develop dementia would interfere with efficacy/safety
evaluations. Second, SFMD with variable patterns, in-
cluding conversion, or somatic delusion and catatonic
signs might be useful in the diagnosis of DLB or PDD,
or might be added to definitions of clinical features in
DLB and PDD.18
Thus, the main message of our study is that com-
plex somatization patterns can be observed and
should be searched in PD and DLB, as their identifi-
cation can predict cognitive outcome, or support di-
agnoses. Recent neurobiological evidence suggests
that hysteria (conversion-somatization SFMD4) and
catatonia, both characterized by overactivity of or-
bitofrontal and anterior cingulate gyrus,36 –38 might
both represent the expression of an archetypal sham
death reflex (playing possum) present in animals as a
life-saving behavior39,40 and could thus be considered
a continuum. Indeed, our study shows that hysteria-
catatonia symptoms might appear as a continuum in
PDD and DLB, and suggests thus that investigating
their mechanisms in these neurodegenerative diseases
might help clarify their features in other conditions.
AUTHOR CONTRIBUTIONS
Statistical analysis was conducted by Dr. Lamberto Manzoli.
ACKNOWLEDGMENT
The authors thank Prof. C.G. Goetz for editing the manuscript and ad-
dressing the presentation and Prof. P. Calabresi, A. Antonini, A. Pado-
vani, P. Barone, G. Wenning, F. Stocchi, S. Jonas, and Dr. P. Tiraboschi,
F. Gambi, and G. Sepede for reading and discussing the manuscript.
DISCLOSURE
Dr. Onofrj served on scientific advisory boards for GlaxoSmithKline, No-
vartis, Lundbeck, Inc., Eisai Inc., Valeant Pharmaceuticals International,
Medtronic, Inc., and Newron; and receives research support from the
Italian Ministry of Health and Italian Ministry of Education. Dr. Bo-
nanni has received research support from the Italian Ministry of Health.
Dr. Manzoli has served as a consultant for Pfizer Inc. and has received
research support from the Italian Ministry of Health. Dr. Thomas serves
on a scientific advisory board for UCB; has received funding for travel
from GlaxoSmithKline; and has received honoraria from Boehringer In-
gelheim and GlaxoSmithKline.
Received October 6, 2009. Accepted in final form February 3, 2010.
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