Benign Transient Hyperphosphatasemia of Infancy and Childhood

소아기 일과성 고포스파타아제혈증 (Hyperphosphatasemia)에 대한 임상고찰

  • Kwon, Jung Hyun (Department of Pediatrics, College of Medicine, Ewha Womans University) ;
  • Park, Ji Hye (Department of Pediatrics, College of Medicine, Ewha Womans University) ;
  • Kim, Hae Soon (Department of Pediatrics, College of Medicine, Ewha Womans University) ;
  • Lee, Seung Joo (Department of Pediatrics, College of Medicine, Ewha Womans University) ;
  • Lee, Mi Ae (Department of Laboratory Medicine, College of Medicine, Ewha Womans University)
  • 권정현 (이화여자대학교 의과대학 소아과학교실) ;
  • 박지혜 (이화여자대학교 의과대학 소아과학교실) ;
  • 김혜순 (이화여자대학교 의과대학 소아과학교실) ;
  • 이승주 (이화여자대학교 의과대학 소아과학교실) ;
  • 이미애 (이화여자대학교 의과대학 진단검사의학교실)
  • Received : 2004.09.03
  • Accepted : 2004.10.12
  • Published : 2005.03.15

Abstract

Purpose : The aim of study is to investigate the clinical manifestations and the etiology of markedly-elevated serum alkaline phosphatase(ALP) levels in children without accompanying liver or bone disease. Methods : The serum ALP and other biochemical laboratory data of 4,989 patients attending the pediatric department of Ewha Womans University Mokdong Hospital between January 2002 to December 2002 were studied. The subjects' ages ranged from 4 months to 14 years. Among them, serum ALP levels were markedly elevated over 1,000 IU/L in 114 children. Among those 114 children, 97 children without liver or bone disease were reviewed retrospectively. Results : Of the 114 children with serum ALP activity of over 1,000 IU/L, 97(85.8 percent) children had neither liver or bone disease. The average ALP activity level was $1,539{\pm}948IU/L$. The male : female ratio was 1 : 0.7. Forty-four children(45.3 percent) were between 4 months to 1-year-old, 19 children(19.3 percent) were between 1- to 2-year-old. We observed a seasonal clustering of cases in during October with 10 cases(10.2 percent) and in during December with 11 cases(11.3 percent). The most common clinical presentation was respiratory tract infection in 25 cases(26.9 percent), and acute gastroenteritis in 17 cases(17.5 percent). Forty-four cases had follow-up ALP activity level, 1 month later and the activity decreased under 1,000 IU/L in 37 cases(84 percent). Conclusion : Hyperphosphatasemia is a benign and transient phenomenon. If there are no clinical and laboratory abnormalities of liver and bone, we suggest monitoring monitor the high serum ALP level 1 month later. If decreased, it seems that it is not necessary for specific examinations to exclude other diseases of malignant condition.

목 적 : 본 연구에서는 간이나 골격계 질환 없이 알카라인 포스파타아제만 증가된 환아들의 임상적 특징을 알아보고 원인과 의의를 알아보고자 하였다. 방 법 : 2002년 1월부터 2002년 12월까지 1년 동안 본원에서 알카라인 포스파타아제를 포함한 생화학 검사를 시행한 4개월부터 14세까지의 소아 4,989명에서 알카라인 포스파타아제가 1,000 IU/L 이상으로 측정된 환자 114명 중 골격계나 간질환이 없었던 환아를 대상으로 하여 의무기록을 이용하여 후향적인 조사를 하였다. 결 과 : 알카라인 포스파타아제가 1,000 IU/L 이상으로 측정된 114명 중 97명(85.8%)이 골격계나 간질환 없이 알카라인 포스파타아제가 상승하였으며 남녀 성비는 1 : 0.7로 남아가 많았다. 평균 알카라인 포스파타아제는 $1,539{\pm}948IU/L$이었으며, 연령 분포는 4개월에서 1세 미만이 44명(45.3%), 1세에서 2세가 19명(19.3%)으로 가장 많았으며, 월별 분포는 9월에서 12월까지 비교적 발병률이 높았다. 동반 질환은 기관지염 및 폐렴, 천식, 크룹 등의 호흡기계 질환이 25명(26.9%), 급성 장염이 17명(17.5%)으로 가장 많았다. 1개월 후 알카라인 포스파타아제가 추적검사가 시행되었던 44명 중 37명(84%)에서 1,000 IU/L 이하로 떨어졌다. 결 론 : 소아기에 알카라인 포스파타아제 상승하는 것은 골격이나 간의 질환이 없는 경우 비특이적 지표로 생각된다. 일과성 고포스파타아제혈증은 양성적이고 일시적인 현상으로써 임상적으로 간이나 골격계의 이상이 없으며, 1-2개월 후에 다시 검사하여 알카라인 포스파타아제가 감소하는 경우에는, 악성 종양이나 기타 질환을 배제하기 위하여 불필요한 특수 검사들을 시행할 필요가 없을 것으로 보인다.

Keywords

References

  1. Bach U. Behavior of serum alkaline phosphatase in prematurity, rickets and spasmophilia. Z Kinderheilkd 1954;74:593-609 https://doi.org/10.1007/BF00436256
  2. Kim SK, Park YW, Lee CG. A case of transient hyperphosphatasemia. J Korean Pediatr Soc 2000;43:856-60
  3. Oh JG, Choi DH, Oh KE, Cho MK, Sohn C. 6 Cases of transient hyperphosphatasemia of infancy and early childhood. Korean J Pediatra 2000;43:105-10
  4. Choi MH, Lee SW, Chung WS. A study of transient hyperphosphatasemia of infancy and childhood. Korean J Clin Pathol 1996;16:888-96
  5. Tolaymat N, de Melo MC. Benign transient hyperphosphatasemia of infancy and childhood. South Med J 2000;93:1162-4
  6. Nishino H, Horrii Y, Tanaka T, Yamagami T, Matsukura T, Kagamimori S. Follow-up study on effects of height velocity and puberty onset on biochemical markers of bone turnover. Nippon Koshu Eisei Zasshi 1999;46:47-60
  7. Tobiume H, Kanzaki S, Hida S, Ono T, Moriwake T, Yamauchi S, et al. Serum bone alkaline phosphatase isoenzyme levels in normal children and children with growth hormone(GH) deficiency : a potential marker for bone formation and response to GH therapy. J Clin Endocrinol Metab 1997;82:2056-61 https://doi.org/10.1210/jc.82.7.2056
  8. Kraut JR, Metrick M, Maxwell NR, Kaplan MM. Isoenzyme studies in transient hyperphosphatasemia of infancy, ten new cases and a review of the literature. Am J Dis Child 1985;139:736-40
  9. Griffiths J, Black J. Separation and identification of alkaline phosphatase isoenzymes and isoforms in the serum of healthy persons by isoelectric focusing. Clin Chem 1987;33:2171-7
  10. Stein P, Rosalki SB, Foo AY, Hjelm M. Transient hyperphosphatasemia of infancy and early childhood : clinical and biochemical features of 21 cases and literature review. Clin Chem 1987;33:313-8
  11. Carroll AJ, Coakley JC. Transient hyperphosphatasemia : an important condition to recognize. J Paediatr Child Health 2001;37:359-62 https://doi.org/10.1046/j.1440-1754.2001.00686.x
  12. Crofton PM. What is the cause of benign transient hyperphosphatasemia? a Study of 35 Cases. Clin Chem 1988;34:335-40
  13. Behulova D, Bzduch V, Holesova D, Vasilenkova A, Ponec J. Transient hyperphosphatasemia of infancy and childhood: study of 194 cases. Clin Chem 2000;46:1868-9
  14. Suzuki M, Okazaki T, Nagai T, Toro K, Setonyi P. Viral infection of infants and children with benign transient hyperphosphatasemia. FEMS Immunol Med Microbiol 2002;33:215-8 https://doi.org/10.1111/j.1574-695X.2002.tb00593.x
  15. Posen S, Lee C, Vines R, Kilham H, Latham S, Keefe JF. Transient hyperphosphatasemia of infancy : an insufficiently recognized syndrome. Clin Chem 1977;23:292-4
  16. Rosalki SB, Foo AY. Transient hyperphosphatsemia of infancy : four new cases and a suggested etiology. Clin Chem 1980;26:1109-10
  17. Nathan E. Transient hyperphosphatsemia of infancy. Acta Paediatr Scand 1980;69:235-8 https://doi.org/10.1111/j.1651-2227.1980.tb07067.x
  18. Steinherz PG, Steinherz LJ, Nisselbaum JS, Murphy ML. Transient, marked, unexplained elevation of serum alkaline phosphatase. JAMA 1984;252:3289-92 https://doi.org/10.1001/jama.252.23.3289
  19. Holt PA, Steel AE, Armstrong AM. Transient hyperphosphatasemia of infancy following rotavirus infection. J Infect 1984;9:283-5 https://doi.org/10.1016/S0163-4453(84)90640-6
  20. Okazaki T, Suzuki M, Nagai T. Recognition of benign transient hyperphosphatasemia. South Med J 2003;96:108-9