Recurrent Belly Dancer’s Dyskinesia with Pregnancy
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Dear Editor,
Belly dancer’s dyskinesia (BDD) is a rare disorder characterized by visible, involuntary, slow, semicontinuous, undulating abdominal wall movements. BDD typically affects the abdominal muscles, but can also involve both the hemidiaphragm and perineal muscles. BDD in pregnancy is even rarer, as a literature review yielded only 3 reported cases [1].
A 40-year-old woman, during her 40th week of pregnancy, presented with a one-week history of writhing, involuntary abdominal movements that started gradually and involved the anterior abdominal muscles. The abdominal movements were semirhythmic, painless, rolling contractions that mostly started from the lower right side of the abdomen and moved upwards and to the left, which resulted in undulating movements of the umbilicus. Movements were slow, brief, and repetitive, and recurred throughout the day at different frequencies that varied from 1–2 per minute to 3–4 repetitions per hour. These movements did not affect the patient’s breathing pattern or depth of breath. There were no specific precipitating or alleviating factors, and the patient was unable to voluntarily suppress the contractions. Distraction techniques did not significantly alter the frequency of the movements, and the movements did not respond to changes in the patient’s position or to breathing exercises, such as breath-holding or counting routines. The movements also did not subside during sleep. The patient reported an episode of depression two years prior to the onset of her current symptoms, for which she had refused medical treatment. She denied any history of trauma, spinal or abdominal surgeries, or personal or family history of similar episodes. Her eight previous pregnancies all ended with normal vaginal births. Drug history was unremarkable. Neurological and gynecological examinations were normal, except for the abdominal movements. There were no signs of fetal distress. Basic laboratory tests showed iron deficiency anemia with normal folate and vitamin B12 levels. Serum electrolytes, kidney, and thyroid function tests were normal. A clinical diagnosis of BDD was made. A normal vaginal birth, 2 days later, resulted in a healthy baby. A postpartum MRI of the thoracic spine was unrevealing.
Upon discharge, the patient refused pharmacologic therapy for dyskinesia. At a 2-month follow-up visit, the patient reported persistence of her symptoms and requested better symptomatic control. Clonazepam 0.5 mg three times a day was prescribed. After four weeks of drug therapy, the patient reported near-complete resolution of her symptoms, with greater than an 80% reduction in the frequency of the abdominal muscle movement episodes. However, two months later, the patient complained of a relapse of her symptoms, where abnormal movements occurred at almost the same frequency as at the first presentation. She also reported missing her period. A serum-based pregnancy test showed that the patient was pregnant. The daily dose of clonazepam was gradually increased to 2 mg twice a day, which achieved a near-complete remission. Consent was obtained from the subject, and the case report was approved by the Ethics Committee of the King Abdullah University Hospital.
Iliceto et al. [2] theorized that a dysfunction of inhibitory spinal interneurons, or a structural reorganization of local neuronal circuits may be the underlying pathophysiology of BDD. A long list of potential causes has been postulated. Dyskinesia may be secondary to a structural lesion, such as a syringomyelia, myelitis, spinal cord trauma, vascular lesion or malignancy [3]. Dyskinesia also may be due to spinal segmental myoclonus [4]. Metabolic causes, drugs, or other functional etiologies have also been postulated. In our case, laboratory and radiology examinations failed to identify a definitive etiology.
Some previous manuscripts suggest that local compressive or hemodynamic changes in the thoracic cord or roots from a gravid uterus is a possible etiology in pregnancy [5]; however, this hypothesis may not explain the relapse that our patient experienced during the first few weeks of the following pregnancy as it was too early to be explained by a gravid uterus (less than 8 weeks pregnant).
The pattern of the presenting symptoms was less consistent with chorea gravidarum as it typically manifests with irregular, nonrhythmic, and jerky movements that usually involve the limbs. Our diagnosis of BDD was further supported by the lack of symptom resolution, shortly after, or within weeks of delivery. A further distinction between BDD and chorea gravidarum is the possibility of symptoms continuing during sleep in BDD [6].
The management of BDD is challenging. Current treatment paradigms remain based on anecdotal evidence. Clonazepam and levetiracetam have both been reported to be effective [7]. Our patient achieved a near-complete resolution of the dyskinesia with clonazepam. Increases to the dose were required to improve her recurrent symptoms after becoming pregnant again.
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Conflicts of Interest
The authors have no financial conflicts of interest.