Updates on Somatoform Disorders (SFMD) in Parkinson's Disease and Dementia with Lewy Bodies and discussion of phenomenology

doi:10.1016/j.jns.2011.07.010

Journal of the Neurological Sciences

Volume 310, Issues 1–2, 15 November 2011, Pages 166-171

https://doi.org/10.1016/j.jns.2011.07.010 Get rights and content

Abstract

Somatoform Disorders (SFMD) were recently described in Parkinson Disease (PD) and Dementia with Lewy Bodies (DLB). The present paper updates the observations in our cohort of patients and further details clinical phenomenology.

Of 3178 patients consecutively referred to our Institutions from 1999, 1572 subjects had neurodegenerative diseases and 1718 psychiatric disorders. After 2–9 years of follow up, 488 patients were labelled as PD, 415 as Alzheimer Disease, 162 as DLB, 48 as Progressive Supranuclear Palsy, 48 as Multiple System Atrophy and 49 as Fronto-Temporal Dementia. The frequency of SFMD (DSM-IV-TR criteria) was determined in each diagnostic category by direct observation of SFMD symptoms, psychiatric interviews, SCL 90Rss, collection of previous general practitioners and hospital charts.

The frequency of SFMD was considerably higher in DLB (29 patients, 18%) and PD (37 patients, 7.5%) than in any other group (0–2%). The frequency of SFMD in psychiatric patients was 2%. SFMD in PD and DLB were characterised by motor and non-motor patterns and were often accompanied by catatonic signs consisting of posturing stereotypies and negativism (55%). SFMD symptoms preceded PD motor signs by 6 months–5 years in 92% of the 29 DLB and 37 PD patients and in 70% SFMD were recurrent at follow-up. In 93% of these patients, hypochondria was a preceding or concomitant background.

Introduction

Somatoform Disorders (SFMD) have been recently described in patients with Parkinson Disease (PD) and Lewy Body Dementia (LBD) in a cohort study comparing incidence in other neurodegenerative disorders including Alzheimer's disease (AD), Fronto Temporal Degeneration (FTD), Multiple System Atrophy (MSA), and Progressive Supranuclear Palsy (PSP) [1]. Concomitantly with this recent paper, SFMD or somatisation traits were recently quoted as a highly prevalent non motor psychiatric manifestation of parkinsonism and PD [2] with prevalences reaching 45% [3].

In order to fully clarify the features and relevance of SFMD in parkinsonism, our paper updates the previous study [1] with further observations obtained in the last three years after the study closure, and discusses phenomenology and possible implications.

Section snippets

SFMD definition

The definition of SFMD suggested by the Diagnostic and Statistical Manual of Mental Disorders, Text Revision, DSM-IV TR [4] relates to a general category including a broad spectrum of disorders, identified as somatisation disorder (DSM-IV TR cod 300.81), undifferentiated SFMD (cod 300.82), conversion disorder (cod 300.11), pain disorder (cod 307.89), body dysmorphic disorder and hypochondria (cod 300.7). The frequency of each of these specific disorders in the general population, varies from

Why should we look for SFMD in PD and DLB?

Focusing on somatic complaints in the presence of an organic illness might constitute an apparent contradiction, despite clinical practise evidences that overlapping somatisation complaints are common problems, e.g. pseudoseizures and pseudovertigo in presence of organic diseases [7]. The different psychiatric classification systems or textbooks do not exclude SFMD in presence of organic diseases, but obviously imply that proper efforts of categorization should be applied [8].

Before that recent

The SFMD assessment

Whereas the rational for the study rested apparently on solid grounds, the identification of proper methods to assess SFMD presence presented a challenge. It was necessary to discriminate all possible PD motor and non motor signs, including “inner tremor” and dystonic pain of neck, shoulders, back and limbs (regions where rigidity and bradykinesia are usually more marked) [18], central neuropatic pain and (because of the age of patients) arthralgic pain. Therefore, assessments of SFMD were

Population and study design

The prospective study was based on a cohort of 1572 new patients evaluated in our Movement Disorder and Memory Clinics from 1999 to 2009, serving a population of 1,300,000. The present report updates the previous study based on 978 patients evaluated from 1999 to 2006.

The prevalence of SFMD was evaluated as percentage of patients affected amongst the different diagnostic groups (PD, DLB, AD, MSA, PSP, FTD) presenting to our Movement Disorder and Memory Clinics in the described time span.

Only

Evaluations

Patients of each disease group (PD, DLB, AD, FTD, MSA, PSP) were evaluated according to consensus criteria and laboratory studies including MRI, SPECT-DAT scan, as reported in the original study [1].

Statistics

In the present paper we report only prevalence studies. In the original paper [1] statistical comparisons were based on matching different groups of patients in cross-sectional and follow-up evaluations.

Prevalence of SFMD in patients with neurodegenerative disorders

From 1999 to 2009, 1572 new patients were evaluated in our Movement Disorder and Memory Clinics. During a follow up of at least two years, 1210 patients were classified as affected by neurodegenerative disorders. 488 were diagnosed as having PD, 415 as AD, 162 as DLB, 48 as PSP, 48 MSA, 49 as FTD.

Table 1 reports the prevalence of SFMD in the different diagnostic groups resulting from direct observations, interviews, previous hospital charts, and reports cognition test scores. Rates of hospital

SFMD motor patterns in PD and DLB

1.
Unilateral or bilateral bent knee and tiptoeing gait: The clinical pattern resembles the bilateral bent knees and tiptoeing gait pattern or posture described as a manifestation of late PD [23]. When unilateral, this posture resembles spastic hemiparesis, although this posture is also rarely observed, with Babinski sign, after acute onset of central paresis.
It was observed in 12 PD and 9 DLB patients (in 10 patients unilaterally), in 4 PD as first SFMD symptom, in 4 during a recurrence. In 6 PD

SFMD non motor patterns

1.
Partial Cotard syndrome or dysmorphogenesis sensory delusions: consisting of complex delusions of progressing body deformation [24]. It was observed in 13 PD and 9 DLB patients, and had the following bizarre thought content:
- a.
  Delirium of penis shrinkage and of disappearance of penis into the abdomen [25]. It was described by one patient, concomitant with the onset of PD symptoms.
- b.
  Delusion of ingrowths of thoracic or abdominal mass identified as growing xyphoid process, epigastric mass, moving

Concomitant signs

The appearance of the main SFMD patterns described above was accompanied by several concomitant features, including mannerism (12 PD and 12 DLB patients), restlessness (6 PD, 5 DLB), verbal aggressiveness (10 PD, 8 DLB), verbosity and viscosity (10 PD, 10 DLB), treatment refusal (all). In 14 PD patients psychogenic tremor, psychological Romberg, variability of strength, wobbling gait, knee buckling were observed in follow-up visits. Globus pharyngis was observed in 16 PD and 10 DLB patients. In

Discussion

Our report describes and shows that SFMD symptoms can be identified in a relevant percentage of PD patients (7.5%) and in higher percentage of DLB patients (19%), increasing the original prevalence of, respectively, 7% and 12%.

SFMD observed in our PD and DLB patients included elements which are proper of conversion and somatisation disorders–hysteria, but also bizarre somatic delusions, and catatonic signs, thus suggesting that proper psychiatric categorizations for SFMD in PD and DLB should

Acknowledgement

The work has been financially supported by the Italian Ministry of Health, Grant Young Researchers 2007.

References (29)

R. Noyes et al.
Prevalence and correlates of illness worry in the general population
Psychosomatics
(2005)
M. Onofrj et al.
Cohort study on somatoform disorders in Parkinson disease and dementia with Lewy bodies
Neurology
(2010)
D.A. Gallagher et al.
What are the most important nonmotor symptoms in patients with Parkinson's disease and are we missing them?
Mov Disord
(2010)
C. Siri et al.
Psychiatric symptoms in Parkinson's disease assessed with the SCL-90R self-reported questionnaire
Neurol Sci
(2010)
American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders-Text Revised (DSM-IV-TR)
(2000)
W. Rief et al.
Somatization symptoms and hypochondriacal features in the general population
Psychosom Med
(2001)
H. Merskey
Conversion, dissociation, or doxomorphic disorder
B.L. Sadock et al.
Kaplan and Sadock's comprehensive textbook of psychiatry
(2009)
C.D. Ward
Neuropsychiatric interpretations of postencephalitic movement disorders
Mov Disord
(2003)
X. Li et al.
Impact of psychiatric disorders on Parkinson's disease : a nationwide follow-up study from Sweden
J Neurol
(2008)

A.E. Lang et al.

Psychogenic parkinsonism

Arch Neurol

(1995)

S.A. Factor et al.

Psychogenic movement disorders: frequency, clinical profile and characteristics

J Neurol Neurosurg Psychiatry

(1995)

J.A. Saint-Cyr et al.

Behavior and the basal ganglia

Adv Neurol

(1995)

H.L. Crimlisk et al.

Slater revisited: 6 year follow up study of patients with medically unexplained motor symptoms

BMJ

(1998)

Cited by (39)

Functional comorbidity in Parkinson disease: A window of opportunity
2024, Parkinsonism and Related Disorders
The Association Between Somatic Symptom Disorders and Neurocognitive Disorders: A Systematic Review
2023, American Journal of Geriatric Psychiatry
Citation Excerpt :
There were more females (63.2%) than males in these studies, with an average age of 60.9 years (11.9). Studies reporting manifestations of SSRD in subjects with a neurodegenerative disorder at baseline included 2 case-control studies22,25 and 2 prospective longitudinal studies,23,24 with the latter's representing 2 different publications of the same longitudinal cohort. In these studies, there were more females (58.8%) than males and the average age was 67.2 years (3.0).
Onset of neuropsychiatric symptoms in older adults may represent prodromal manifestations of neurodegenerative disorders. The association between the onset of somatic symptom and related disorders (SSRD) and the subsequent development of neurodegenerative disorders remains unclear. A critical review of studies describing the association between SSRD and neurodegenerative disorders, such as Alzheimer's disease, Parkinson's disease, Frontotemporal dementia, and Lewy body dementia was performed.
To critically review studies describing the association between SSRD and neurodegenerative disorders, such as Alzheimer's disease, Parkinson's disease, Frontotemporal dementia, and Lewy body dementia.
A systematic review of Web of Science Core databases was carried out from inception of databases up to May 2021 to identify observational studies pertaining to both SSRD and neurodegenerative disorders. Data was extracted and compiled regarding subjects enrolled, age at onset of the SSRD and at onset of the neurodegenerative disorders, and specific SSRD manifestations and underlying neuropathologies reported.
Thirteen articles were included. Of the 123 identified subjects with SSRD at baseline, 34.1% developed a neurodegenerative disorder, with 80.9% of these being a Lewy body spectrum disorder. The interval between onset of SSRD manifestations and subsequent development of a neurodegenerative disorder was less than 3 years for half of the cases. Of the 1,494 subjects with a neurodegenerative disorder at baseline retrieved, SSRD manifestations were reported in 33.4% of Lewy body spectrum disorders cases. Onset of SSRD manifestations antedated or was concomitant to the diagnosis of the Lewy body spectrum disorder in 65.6% of cases.
While limited, current evidence suggests a possible association between late-onset SSRD and the subsequent development of neurodegenerative disorders, notably Lewy body spectrum disorders.
Functional neurological disorder and somatic symptom disorder in Parkinson's disease
2022, Journal of the Neurological Sciences
Citation Excerpt :
FND-PD/DLB patients frequently denied the PD diagnosis (73%), more often complained about intolerable adverse reactions to the prescribed medications [50], as due to the nocebo effect. FND-PD patients often exhibit dramatic intolerance to minor therapeutic changes (i.e., changes of the drug brand while maintaining the same compound and regimen), psychogenic off states after the drug intake, refractoriness to therapeutic adjustments, and even self-prescription of dose increments or inappropriate treatments (like the use of Dopamine Agonists against the physician's advice due to psychotic symptoms or impulse control disorders) [4,37,44]. In later studies [44,51,52], the prevalence of FND in PD was found to be remarkably higher than in our original study, with figures ranging from 19 to 67%, compared to the 8–19% of our original cohort [4]).
The occurrence of Functional Neurological Disorder (FND) and Somatic Symptom Disorder (SSD) in PD was not commonly accepted until recently, despite some evidence that emerged in the pre and early L-Dopa era. More recently, the recognition of FND and SSD were noted to be relevant for the management of PD. FND and SSD appear early in the course of PD, often preceding motor symptoms, may interfere with treatment outcomes, often acquire psychotic features during progression, and are mixed with and often concealed by the progressive cognitive decline. We review the related features from the range of the available reports and discuss theoretical models conceived to explain the potential pathophysiological background of these disorders. Finally, we suggest that FND and SSD should be included among the non-motor symptoms of PD and be considered a prodromal feature in a subset of patients.
This article is part of the Special Issue "Parkinsonism across the spectrum of movement disorders and beyond" edited by Joseph Jankovic, Daniel D. Truong and Matteo Bologna.
Victor Parant (1848–1924) and the first report of psychosis in the course of Parkinson's disease with dementia
2021, Revue Neurologique
Until the beginning of the twentieth century, neurologists considered that mental disorders in the course of Parkinson's disease (PD) occurred in the terminal phases of the disease or were due to coincidental pathologies. Benjamin Ball (1834–1893), in 1881 and 1882, drew attention to the frequency of cognitive and depressive disorders in PD. In 1883, Victor Parant (1848–1924), referring to Ball's work, published the first detailed observation of a PD patient with dementia and psychotic symptoms. Parant was an alienist running a private clinic for mental diseases in Toulouse, France. One of his main interests was the question of the responsibility of the insane, and he was called upon as a forensic expert in several cases. In this context, Parant examined a man who had been suffering from PD for several years, and later developed concurrently severe cognitive impairment and psychotic disorders. The patient would meet modern criteria for PD-associated psychosis: he had multimodal hallucinations (visual, auditory and somatic), visual illusions, and paranoid delusions. He also reported unusual symptoms: supernumerary limbs and Alice in Wonderland syndrome. Parant forwarded the far-sighted hypothesis that cognitive and psychotic disorders were due to the extension of PD lesions within the brain. The unheralded work of Victor Parant should be recognized in the history of neuropsychiatry.
Functional movement disorder comorbidity in Parkinson's disease: Unraveling the web
2021, Parkinsonism and Related Disorders
Citation Excerpt :
When compared to a pure PD cohort (matched by age, sex and PD duration), patients with FMD comorbidity were found to have a higher incidence of depression, anxiety and previous psychiatric condition, PD family history, dyskinesias and daily levodopa dose, resource consumption [17], and to suffer greater cognitive impairment during follow-up [12]. The most frequent overlapping FMD in PD included gait disturbances and tremor [16,17], possibly followed by paresis, dystonia [13], and parkinsonism [18]. Speech disorders and myoclonus were less frequent [17].
Functional movement disorders are commonly seen in neurology services and may coexist with other neurological diseases. This combination is known as “functional overlay” and an increasing interest on this topic has emerged in the past decade as the field of functional neurological disorders has moved forward. Some neurological diseases may be more prone to develop “functional overlay” than others, and within the field of movement disorders, most studies have focused on patients with Parkinson's disease. This review comprehensively summarizes the current body of knowledge on this topic and provides an expert opinion to equip clinicians with a pragmatic approach to recognize functional movement disorders in patients with Parkinson's disease, to communicate the diagnosis and to become familiar with potential therapies in this complex clinical scenario. Potential underlying mechanisms and risk factors that may play a role in increasing the vulnerability of Parkinson's disease patients to develop functional movement disorder comorbidity are also discussed within the framework of modern neurobiological theories of brain functioning.
Invalidation of Parkinson's disease diagnosis after years of follow-up based on clinical, radiological and neurophysiological examination
2019, Journal of the Neurological Sciences
Diagnosis of Parkinson's disease (PD) is mainly based on clinical features. Accurate neurological examination is required but dopamine transporter (DaT) single photon emission computed tomography (SPECT) could be perfomed to support the diagnosis in ambiguous cases. The aim of this work is to describe the characteristics of patients with a prolonged PD misdiagnosis.
We collected data from 24 patients initially diagnosed with PD who had an atypical long-term evolution. We analyzed demographic and clinical characteristics and antiparkinsonian drugs medication. Brain MRI, DaT-SPECT and/or accelerometry/electromyography (EMG) recording were performed in a subgroup of patients. We analyzed the causes of erroneous PD diagnosis as well as the final diagnoses.
Mean age at PD diagnosis was 60.4 ± 14.8 years. Symptoms at onset were rest tremor (n = 19), gait instability (n = 7) and micrographia (n = 4). Mean duration before diagnosis correction was 8.4 ± 5.3 years. All patients were treated by antiparkinsonian drugs with a mean daily levodopa equivalent dose (LED) of 508.1 ± 528.4 mg. All 18 patients who underwent DaT-SPECT had a normal result. The most frequent final diagnoses were essential tremor (n = 11) and functional movement disorders (n = 9).
Cases that have been initially diagnosed as PD and then progress in an atypical long-duration fashion may have been misdiagnosed. Absence of genuine bradykinesia, non-sustained response to antiparkinsonian drugs, or absence of levodopa-related side effects should prompt the clinician to reappraise the diagnosis and to consider performing a DaT-SPECT.

View all citing articles on Scopus

View full text

Updates on Somatoform Disorders (SFMD) in Parkinson's Disease and Dementia with Lewy Bodies and discussion of phenomenology

Abstract

Introduction

Section snippets

SFMD definition

Why should we look for SFMD in PD and DLB?

The SFMD assessment

Population and study design

Evaluations

Statistics

Prevalence of SFMD in patients with neurodegenerative disorders

SFMD motor patterns in PD and DLB

SFMD non motor patterns

Concomitant signs

Discussion

Acknowledgement

Psychosomatics

Cohort study on somatoform disorders in Parkinson disease and dementia with Lewy bodies

Neurology

What are the most important nonmotor symptoms in patients with Parkinson's disease and are we missing them?

Mov Disord

Psychiatric symptoms in Parkinson's disease assessed with the SCL-90R self-reported questionnaire

Neurol Sci

American Psychiatric Association: Diagnostic and Statistical Manual of Mental Disorders-Text Revised (DSM-IV-TR)

Somatization symptoms and hypochondriacal features in the general population

Psychosom Med

Conversion, dissociation, or doxomorphic disorder

Kaplan and Sadock's comprehensive textbook of psychiatry

Neuropsychiatric interpretations of postencephalitic movement disorders

Mov Disord

Impact of psychiatric disorders on Parkinson's disease : a nationwide follow-up study from Sweden

J Neurol

Psychogenic parkinsonism

Arch Neurol

Psychogenic movement disorders: frequency, clinical profile and characteristics

J Neurol Neurosurg Psychiatry

Behavior and the basal ganglia

Adv Neurol

Slater revisited: 6 year follow up study of patients with medically unexplained motor symptoms

BMJ