모유 수유아에서 발생한 구루병 경련의 임상양상
Clinical Features of Seizures Related to Rickets in Breastfed Children
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목 적 : 모유 수유에 의한 비타민 D 결핍성 구루병에 관한 연구들이 활발히 진행되고 있는 반면에 이때 발생하는 경련에 관한 연구는 없다. 이에 저자들은 완전 모유 수유에 의해 비타민 D 결핍성 구루병이 있었던 환아에서 발생하는 경련의 임상양상 대하여 알아보고자 하였다. 방 법 : 2000년 1월부터 2010년 12월까지 인하대병원 소아청소년과 외래를 방문하였거나 입원하였던 환아 중 완전 모유수유를 하면서 구루병을 진단받았던 환아들을 대상으로 후향적으로 조사하였다. 경련군과 비경련군으로 나눈 후, 두 군간의 임상적 특성과 생화학적 검사 결과를 비교 분석하였고, 경련 발생의 상대 위험도를 평가하였으며, 경련군에서 발생한 경련의 임상 양상에 대해 조사하였다. 결 과 : 완전 모유수유를 하면서 구루병을 진단받은 17명의 환아중 9명(53.0%)에서 경련이 발생하였고, 8명(47.0%)에서는 경련이 없었다. 두 군의 진단당시 평균 연령은 각각 4.1±2.0개월, 9.3±2.7개월로 경련군에서 통계적으로 의미 있게 낮았다(P<0.0001). 혈액 검사에서 혈청 Ca 수치는 각각 5.7±1.0 mg/dL, 9.5±0.9 mg/dL으로 경련군에서 통계적으로 의미 있게 낮았다(P<0.0001). 혈청 25-OHD3 수치는 각각 5.7±0.8 IU/L, 15.3±4.2IU/L으로 경련군에서 통계적으로 의미 있게 낮았다(P<0.0001). 경련 발생의 상대 위험비는 저칼슘혈증이 있있을때가 8배였고, 혈중 25-OHD3 수치가 8 ng/mL 미만일 때가 17배였다. 경련은 5분 미만의 전신 발작 또는 1분 미만의 부분 발작 형태로 다회성으로 나타났으나, 향후 간질이 발생하거나 항경련제를 투여받거나 비정상적인 신경 발달을 보인 환아는 없었다. 결 론 : 완전 모유 수유아에 의한 비타민 D 결핍성 구루병 환아에서 발생하는 경련은 저칼슘혈증에 의해 발생하고, 저칼슘혈증은 혈중 25-OHD3 농도의 영향을 받는다. 또한, 체내에서 칼슘 요구량이 많은 급성장 시기의 모유 수유아에서는 경련이 다른 시기보다 쉽게 발생할 수 있다. 완전 모유 수유아에 의한 비타민 D 결핍성 구루병 환아에서 발생하는 경련은 다회성으로 발생했으나 예후는 양호했다. 추후, 보다 많은 환아들을 대상으로 하는 대규모 전향적 연구가 필요하다.
Abstract
Purpose : This study was aimed to evaluate the clinical features of seizures in breastfed children with vitamin D deficient rickets. Methods : Seventeen children, breastfed and diagnosed as vitamin D deficient rickets at Inha University Hospital from January 2000 to July 2010, were retrospectively investigated. Subjects were divided into two groups according to the presence/absence of seizures. Demographic and biochemical results were compared and statistically analyzed between the two groups, and the relative risk for seizure occurrence was estimated. Clinical features of seizures were also analyzed. Results : Out of the 17 subjects, nine patients (53.0%) had seizures, while eight patients (47.0%) did not. The mean age for the two groups were 4.1±2.0 months and 9.3±2.7 months, respectively, which was statistically different between the two groups (P<0.0001). Serum calcium (Ca) and 25-hydroxyvitamin D3 (25-OHD3) levels were significantly lower in the 'seizure' group (5.7±1.0 vs. 9.5±0.9 mg/dL, P<0.0001; 5.7±0.8 vs. 15.3±4.2 IU/L, P<0.0001). The relative risk for seizure occurrence was 8 times higher in hypocalcemia and 17 times higher in 25-OHD3< 8 ng/mL. Seizures occurred several times as generalized or focal types, but none of them developed epilepsy nor showed developmental abnormalities later on. Conclusion : Seizures in breastfed children with vitamin D deficiency rickets are mainly due to hypocalcemia, which is affected by 25-OHD3 levels. Seizures may also occur more frequently in children in the stages of rapid growth. Although seizures occurred multiple times, future outcomes were favorable. Further large-scaled prospective studies are required in the future.
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