Paroxysmal rhythmic pelvic movements: an unusual paediatric presentation of Chiari I malformation with syringomyelia
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LETTER TO THE EDITOR
Paroxysmal rhythmic pelvic movements: an unusual paediatric presentation of Chiari I malformation with syringomyelia Stefano Sotgiu 1
&
C. Scoppola 1 & A. Carta 1 & G. Pisuttu 1 & F. Salis 1
Received: 25 June 2020 / Accepted: 24 September 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020
Dear Editor: Chiari I malformation (CIM) is a common paediatric condition with variable and age-dependent presentation. Several children are diagnosed incidentally while others present with typical symptoms including Chiari headache, visual and balance disturbances, irritability in younger and syringomyeliarelated symptoms in older children [1]. Other children are being progressively recognized to present atypical CIM outset, including paroxysmal asymmetric motor and sensory symptoms, cerebellar fits, bulbar dysfunction, VI cranial nerve palsy and sleep apnoea [1], or unilateral facial hyperhidrosis in those with cervical syringomyelia [2]. Despite posterior fossa, surgical decompression is often successful; mechanistic correlation of CIM with unusual symptoms remains unclear. Involuntary pelvic muscle contractions are not directly linked to CIM, as yet. Here, we describe this unique presentation in a young girl and its possible pathophysiological phenomenology. After a few months of sporadic symptoms which prompted some paediatric evaluations, a 6-year-old girl presented with increasing paroxysmal episodes (at zenith 3 times/day, 5 min duration) of left head sweating, right body-side paresthesias, tingling sensations in her lesser pelvis and stereotyped, rhythmic pelvic movements resembling a coital behaviour. Paroxysms mostly appeared after head hyperflexion while playing videogames in sitting or supine position and were nor subjected to distractibility and suppressibility. Consciousness was always preserved. Differential diagnoses included neurological conditions such as photosensitive epileptic seizures, spinal myoclonus, dyskinetic disorders, restless leg syndrome, rhythmic
* Stefano Sotgiu [email protected] 1
Unit of Child Neuropsychiatry, University Hospital of Sassari, Sassari, Italy
movement disorders, affective or anxiety disorders, posttraumatic stress disorder, infratentorial tumours and pheochromocytoma. Gestation, family and psychologic history were normal, so did neurological examination, electrocardiogram, abdominal ultrasound, routine blood/urine, thyroid and infectious tests. Neither history of myelopathy, spinal anaesthesia or trauma nor of therapeutic or accidental usage of pain relievers or dopamine-receptor blockers were detected. Hyperkinetic movements were neither myocloniclike jerks nor dystonic-like sustained contractions, nor they were hypnic or exercise-induced. Diagnostic criteria for restless legs syndrome [3] were not met with no evidence of parasomnias, sleepwalking, nightmares, head and body rolling and banging. An extended EEG covering motor paroxysms showed no evidence of epileptic seizures. There are no separation anxiety, domestic violence, bedtime ri
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