A 14-year-old girl presented to our department with a 1-month history of persistent extension of left arm, clenching of both fists, and plantar flexion of the toes on both feet. The onset of symptoms was abrupt and symmetrical. She also complained of difficulty in swallowing and breathing, leading to reduced appetite and sleep. She reported another episode of neck extension 1 year ago, which resolved spontaneously in 10 days after taking calcium and vitamin supplements.
The patient's history indicated that she had experienced behavioral challenges at home and at school (ie, physical altercations) for several years since the sudden death of her father. Although she preferred to be alone and did not particularly get along with siblings, no psychosocial stressors or history of mental disorders in the family were identified. Her academic outcomes were normal, and she attended school regularly up until 1.5 years ago when she began experiencing frequent physical symptoms of lightheadedness and pain/uncomfortable sensations in throat. After leaving mainstream school due to these health issues, she continued her studies in an alternative learning situation until the onset of her most recent symptoms. The patient also had a history of having tuberculous meningitis 9 years earlier for which she remained under follow-up care and completed 2 years of treatment.
She was able to perform activities of daily living as her dominant arm (right) was not severely affected. Physical examination revealed extension of arms and flexed and radially deviated wrists with flexion of the fingers at metacarpophalangeal, proximal, and distal interphalangeal joints of all fingers of both hands (Figure 1). There was no joint tenderness, joint or tendon sheath swelling, or any other lesions. The patient was unable to do any active digital extension secondary to complaint of severe pain. Also, she aggressively stopped any attempt to reflexively straighten the fingers or wrists. There were no visible or palpable signs of any Dupuytren's contractures on either side or trauma to both hands. Peripheral sensibility and blood perfusion were normal. Neck examination did not reveal any abnormality. Her gait was normal. The patient was uncooperative throughout the examination. Neurological examination of both upper and lower limbs was normal and did not reveal any organic etiology.
Clenched fists from different angles.
In Mental Status Examination, she was well-oriented in time, place and person, normal speech, eye contact, cognition, and perception. She resisted talking about her past and her emotions and presented a cavalier attitude toward her problem (ie, “la belle indifference”). There was no evidence of formal thought disorder, psychosis, or depression. However, she exhibited manipulative behavior and blamed the staff and physician for not being able to help her.
In the absence of any organic explanation, the patient was admitted to the child and adolescent psychiatry unit with the provisional diagnosis of functional neurological symptoms disorder (clenched fist syndrome [CFS]). All of her laboratory results, including complete blood count, liver function tests, urea and electrolytes, serum calcium, serum ceruloplasmin as well as ultrasound of the abdomen and magnetic resonance imaging of the brain, were normal. No other tests were advised by the physicians, neurologists, or orthopedic professionals.
On day 2 postadmission, she developed persistent extension in the right arm with complaint of moderate to severe pain in both arms and the neck. A week after admission, inward flexion of both feet was observed as well as an episode of lightheadedness and fainting, which lasted half an hour. No features of true seizure were noted during the episode. Clinicians recommended that the patient receive aggressive physiotherapy, but she refused to cooperate. However, after being educated about the psychopathological nature of what she was experiencing, she reluctantly accepted wrist splints, which counteracted the wrist flexion to some degree. She was extremely resistant and at times uncooperative to the therapy and used abusive language toward her family and the therapist during treatment.
After consultation with the surgical team, it was determined that it was necessary to unclench the hands under anesthesia as part of the treatment along with casting of the left hand, which was more flexed and had macerations due to palm hygiene problems (Figure 2). During follow-up, the patient improved with aggressive hand physiotherapy, including stretching exercises, supported psychotherapy, and pharmacotherapy. Both hands returned to the neutral position after 1 month.
Position of hands after treatment for unclenching under general anesthesia.
Relapse was noted with a return of the hands in pathological positions after 2 weeks of improvement, although it was less severe and improved again within a few days. Her prognosis is poor because she either does not comprehend or refuses to accept the graveness of her illness, so there is a high likelihood of discontinuation of the therapy and treatment.
Psychopathological hand disorders, including CFS, represent conversion of emotional turmoil to physical symptoms in a symbolic manner.1 In CFS, there are flexion contractures of several digits or whole hand in severe cases, leading to fist presentation, usually with swelling and no correlation with handedness.2 In contrast, a similar condition was described by Frykman et al.3 as psycho-flexed hand, where there is involvement of the dominant hand, minimal swelling, and the patients' fists are not entirely clenched. Both descriptions are now essentially considered to be the same entity with CFS being the preferred term. Reports indicate CFS as a conversion disorder being unconsciously motivated and unconsciously produced.1,4,5
CFS is a rare clinical presentation, with review of literature showing less than 40 cases reported worldwide and very few in adolescents. It is relevant to ask whether any organic etiology could have caused the symptoms in this patient. All standard diagnostic tools used to rule out any organic etiology before a CFS diagnosis should be made and were conducted in this patient; no pathological findings were discovered. We also considered a few differential diagnoses. For clenched fists to be diagnosed as nonorganic disorder, we should exclude Dupuytren's contracture, rheumatic arthritis, progressive systemic sclerosis, collagen diseases, peripheral nerve injuries, late complications of Parkinson's disease, and complex regional pain syndrome among others.5,6 This patient had no known physical disease of her hands prior to clenching of her fists. Onset was abrupt and symmetrical. With her history, it is hard to conceive of any of the above differential diagnoses to start acutely and symmetrically. Furthermore, each of these diseases have their distinct clinical features, which were not present in this case. Therefore, we find it reasonable to consider a psychopathological mechanism as the most likely cause. The most important diagnostic maneuver is examination of the patient's hands under anesthesia, in which clinical deformity completely resolves, as was noted in this patient. The presentation of CFS and the psycho-flexed hand is compatible with conversion disorder or functional neurological symptom disorder according to the Diagnostic and Statistical Manual of Mental Disorders, fifth edition7 criteria.
CFS occurs in different age groups and various psychiatric comorbidities like depression, schizophrenia, and obsessive-compulsive disorders; borderline and dependent personality disorders have been found to predate the onset of CFS.1–3,8 This patient had no previous psychiatric diagnosis or assessment, although her history was suggestive of oppositional defiant disorder (ODD) and schizoid personality traits, which are not specifically represented in published literature.
It is important to try to understand the psychodynamics of the diagnosis. Children with conversion disorder are noted to have communication problems in family, family conflicts, as well as unresolved grief in significant proportion of the cases, which can lead to somatic symptoms and presentations as a main emotional outlet.1 Grief from the loss of a parent as well as anger played a significant role in our patient's presentation. In the literature, it was noted that adolescents with CFS had a “consistent theme of repressed anger. …The hand bound into a fist symbolically expresses anger…” and “angry, hostile persons who were suspicious and tended to act out their anger maladaptively.”2 This case resembles that of another 14-year-old adolescent who was noted as having “…rigid mechanisms of avoidance and denial as defenses against expressions of impulses” and “flat rejection that emotional factors could be contributing to her disability.”2,9 Patients with CFS has been characterized as sad, hostile, anxious, frustrated, and tenacious (or SHAFT syndrome).10 The most probable diagnostic formulation is that we were dealing with an adolescent who was angry and suffering from ODD accompanied by repeated conversion attacks as a symptom of more generalized psychopathology rooted deeply in her personality. Projective tests would have been helpful to determine repression of powerful emotions but could not be performed due to her refusal to cooperate.
Treatment of patients with CFS is often difficult and involves a multidisciplinary team approach with doctors, psychologists, and physiotherapists. A multimodal treatment program consists of unclenching the hand under anesthesia, intensive physiotherapy (using stretching, splinting, and casting), and psychotherapy (eg, cognitive-behavioral therapy, biofeedback, hypnosis).1,5,9,11 Direct queries or closed-ended questions including accusations of malingering/manipulation by family members or clinicians are contraindicated. Surgical treatment is not indicated. Apart from psychoeducation about the psychological nature of her presentation, our patient received physiotherapy and wrists splints. Coping strategies to deal with psychological distress were the focus of CBT. The hands and feet were brought back to the neutral positions after 1 month. This case illustrates the diagnostic, prognostic, and therapeutic implications of uncommon presentations of psychopathological hand disorders.
- Eldridge MP, Grunert BK, Matloub HS. Streamlined classification of psychopathological hand disorders: a literature review. Hand (NY). 2008;3(2):118–128. doi:10.1007/s11552-007-9072-0 [CrossRef] PMID:18780087
- Simmons BP, Vasile RG. The clenched fist syndrome. J Hand Surg Am. 1980;5(5):420–427. doi:10.1016/S0363-5023(80)80071-2 [CrossRef] PMID:7430578
- Frykman GK, Wood VE, Miller EB. The psycho-flexed hand. Clin Orthop Relat Res. 1983;(174):153–157. PMID:6831798
- Nissen T, Wynn R. Clenched fist syndrome: a case report. J Med Case Reports. 2018;12(1):168. doi:10.1186/s13256-018-1729-5 [CrossRef] PMID:29909775
- Petrella L, Belkheyar Z, Oberlin C. The psychoflexed hand: new perspectives in diagnosis, classification and treatment. Chir Main. 2013;32(4):245–250. doi:10.1016/j.main.2013.04.005 [CrossRef] PMID:23712089
- Cordivari C, Misra VP, Catania S, Lees AJ. Treatment of dystonic clenched fist with botulinum toxin. Mov Disord. 2001;16(5):907–913. doi:10.1002/mds.1186 [CrossRef] PMID:11746621
- American Psychiatric Association. Diagnostic and Statistical Manual of Mental Disorders. 5th ed. American Psychiatric Publishing; 2013.
- Basaran K, Datli A, Kurt M. An important diagnosis for the hand surgeon: “the clenched fist syndrome.”J Plast Reconstr Aesthet Surg. 2014;67(9):1304–1306. doi:10.1016/j.bjps.2014.04.027 [CrossRef] PMID:24857425
- Grunert BK, Sanger JR, Matloub HS, Yousif NJ. Classification system for factitious syndromes in the hand with implications for treatment. J Hand Surg Am. 1991;16(6):1027–1030. doi:10.1016/S0363-5023(10)80062-0 [CrossRef] PMID:1748746
- Graham WP III, Shearer AW, Mackay DR, Santo J, Stratis JP. SHAFT syndrome revisited. Ann Plast Surg. 1999;42(4):411–416. doi:10.1097/00000637-199904000-00011 [CrossRef] PMID:10213403
- Meiwandi A, Papadakis M. The clenched fist syndrome: case report of a clinical rarity of special interest for psychiatrists and hand surgeons. BMC Psychiatry. 2019;19(1):349. doi:10.1186/s12888-019-2348-4 [CrossRef] PMID:31703662