Discussion and conclusion
CFS is a very rare clinical entity, with less than 40 cases reported. It is a psychiatric syndrome without clear and well-established etiology that can be assigned to the group of conversion disorders. Therefore, it is to be differentiated from factitious disorders or malingering [1, 2]. Patients suffering from CFS develop flexion contractures of the hand and fingers with potentially devastating outcomes if left untreated. The term clenched fist syndrome was first introduced in 1980 by Simmons et al. [3], who described a case series of five patients of different age with flexion contractures of the entire hand. Particular affected fingers were the ulnar three digits. In 1983, Frykman et al. used the term psycho-flexed hand to describe a case series of five patients, who, similar to the clenched fist syndrome, presented with flexion contractures [9]. In this case series, though, the dominant hand was involved. The patients were all middle-aged and none of them had the entire hand clenched. While there are differences between both reported patient groups, it is possibly the same entity that varies in symptoms.
In the past, many authors described CFS as a subgroup of SHAFT-syndrome, which is a factitious disorder, leading affected patients to seek polysurgery and manipulate medical staff in order to satisfy their psychological needs. These patients tend to be sad, hostile, anxious, frustrating and tenacious, so the acronym SHAFT was built [6]. Many recent reports indicate that CFS is a conversion disorder that is unconsciously motivated and unconsciously produced [1, 4, 7, 8]. Therefore, it should not be grouped with the factitious disorders, which are unconsciously motivated and consciously produced, or with malingering which is consciously motivated and consciously produced [1, 2, 10].
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The DSM V criteria introduced the group of Functional Neurological Symptom Disorders as a subtype of conversion disorders, classified as F44.4 in the existing ICD 10 version. In the upcoming ICD 11 Classification these disorders are further categorized as Dissociative neurological symptom disorder, with movement disturbance (ICD Code: 6B60.8) [11]. In the literature these types of disorders are also often recognized as psychogenic movement disorders. This term emphasizes the psychogenic etiology [12]. Even though the terminology is not uniformly, the diagnostic criteria are very similar. In the upcoming ICD 11 “Dissociative neurological symptom disorders, with movement disturbance are characterized by symptoms such as chorea, myoclonus, tremor, dystonia, facial spasm, parkinsonism, or dyskinesia that are not consistent with a recognized disease of the nervous system, other mental and behavioural disorder, or other health condition and do not occur exclusively during another dissociative disorder” [11].
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Patients with CFS present with a variety of symptoms ranging from simple flexion deformities to contraction in the entire hand. Even minor trauma seems to trigger the disease in most cases. Bilateral involvement is also possible, but not the rule. Most often the disorder involves both interphalangeal joints. Additional psychiatric comorbidities are almost always present in patients with CFS and a thoroughly psychiatric assessment should be performed in the first consultation [3, 7-9, 13, 14]. Our patient showed an unexplainable flexion contracture of all digits at the level of the proximal and distal interphalangeal (PIP and DIP) joints of the right hand resembling previously described cases with no prior trauma. In addition, the patient had a positive psychiatric history, as she was suffering from obsessive compulsion disorder and depression. Both disorders have been closely linked to the clenched fist syndrome in the literature [1, 5, 14]. Mysophobia has not been increasingly represented in the published cases though [2, 12, 14].
In the physical examination, the majority of patients show flexion contractures in the interphalangeal and metacarpophalangeal (MCP) joint, resulting in clenching the hand into a fist. The ulnar-sided fingers are most commonly affected. Active extension is not possible and trying to passively extend the fingers without anesthesia is too painful to accomplish. In advanced cases, the contractures become permanent due to changes in the soft tissue, joints and tendons. Macerations and infections due to palm hygiene problems also occur in these cases [3, 7, 8, 13]. In our patient, all fingers including the thumb were affected at the level of metacarpal and interphalangeal joints. There was no joint tenderness, joint or tendon sheath swelling. Passive digital extension was painlessly possible in all fingers.
Radiography, laboratory tests, MRI and electromyography are in most cases normal, but comprise the standard diagnostic tools used to rule out any organic etiology before CFS diagnosis can be made. All of these tests were carried out in our patient and showed no pathological results. Differential diagnosis includes rheumatologic diseases, Dupuytren contracture, camptodactyly, complex regional pain syndrome, central neurological and peripheral nerve diseases. The fact that in our case all 19 hand surgeons being consulted, missed the diagnosis is not uncommon for CFS, since it is a relatively rare disease and most physicians are not aware of psychopathological hand disorders [1-3, 5, 6, 8, 9].
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The treatment of CFS consists of unclenching the hand under anesthesia, intensive physiotherapy and psychotherapy (e.g. CBT, biofeedback, hypnosis) [1, 2, 8, 15]. In prolonged cases, it is sometimes indicated to relieve the contractures surgically. Recent reports show promising results in patients that underwent successful psychotherapy. Surgery should only be performed in these patients and not on psychiatric unstable patients. Fixed chronic contractures that cannot be resolved through conservative treatment or intensive physiotherapy represent an indication for surgery. These contractures develop when the hand is held in a fixed clenched position for a long period of time [7, 8].
Our patient was educated about the psychopathological nature of her disorder and received splint and intensive physiotherapy. The goal of CBT was to convince the patient that the pathology of her hand could not be attributed to a clear organic etiology and to develop coping strategies [1, 15]. In our opinion, both therapy modalities (physiotherapy and psychotherapy) contributed to the improvement, as the patient could not see the necessity of physiotherapy without psychotherapy. The hand was brought back to the neutral position after 1 month.
Prognosis is described as rather poor in literature since most of the patients tend not to comprehend or neglect the graveness of their illness. Thus, treatment is not completed and drop outs are fairly common. The success of therapy correlates closely to the results of psychotherapy [1, 3, 4, 8].
We conclude that CFS should always be considered in patients with unexplainable flexion hand contractures, especially in the presence of a positive psychiatric history.
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