Paroxysmal kinesigenic dyskinesia secondary to pseudohypoparathyroidism: a report of 2 cases and literature review
10.3760/cma.j.cn113694-20221228-00979
- VernacularTitle:假性甲状旁腺功能减退症导致发作性运动诱发性运动障碍2例并文献复习
- Author:
Qiulei CHEN
1
;
Shiwen WENG
;
Na WANG
;
Zhimei LI
;
Xiaoqiu SHAO
;
Qun WANG
;
Ruijuan LYU
Author Information
1. 首都医科大学附属北京天坛医院神经病学中心癫痫科，北京100070
- Keywords:
Pseudohypoparathyroidism;
Movement disorders;
Hypocalcemia;
Hyperphosphatemia;
Parathyroid hormone resistance
- From:
Chinese Journal of Neurology
2023;56(10):1119-1127
- CountryChina
- Language:Chinese
-
Abstract:
Objective:To explore the clinical characteristics of paroxysmal kinesigenic dyskinesia (PKD) secondary to pseudohypoparathyroidism (PHP), so as to improve the clinicians′ understanding of the disease.Methods:The clinical data of 2 cases of PKD secondary to PHP in Beijing Tiantan Hospital, Capital Medical University from June 2022 to October 2022 were summarized, and the related literature was reviewed to analyze the pathogenesis, clinical manifestations, laboratory examination and imaging characteristics of the disease, as well as its treatment and prognosis.Results:Ten cases of PKD secondary to PHP were previously reported. A total of 12 cases (including these 2 cases) were included in the study, of which 7 were males (7/12) and 5 were females (5/12). The onset age was 8-23 years. The clinical manifestations of the disease included paroxysmal limb torsion, limb stiffness, limb tremor, throwing movements, and dance like movements. There were 3 cases (3/12) who had seizure at the same time. Ten cases (10/12) were induced by exercise, while 2 cases (2/12) were not mentioned the inducing factor. Eleven cases (11/12) with paroxysmal symptoms lasting less than 2 minutes can be relieved automatically. Only 1 case had a family history. Laboratory examination of all patients showed low calcium, high phosphorus and significant elevation of parathyroid hormone. The head CT scans of 10 cases (10/12) showed multiple intracranial calcifications. There were 9 cases (9/12) alone received the supplementation of calcium and active vitamin D, while 3 cases (3/12) were additionally treated with antiepileptic drugs. All cases had good prognosis.Conclusions:PHP is a rare cause of secondary PKD. A small number of patients can have seizures at the same time. Most patients have a good prognosis after receiving calcium and active vitamin D supplementation.
