Ariceta Gema, Langman Craig B
Division of Pediatric Kidney Diseases, Hospital Cruces, Vizcaya, Spain.
Eur J Pediatr. 2007 Apr;166(4):303-9. doi: 10.1007/s00431-006-0357-z. Epub 2006 Dec 14.
Growth failure appears frequently in children with X-linked hypophosphatemic rickets (XLHR) due to hypophosphatemia, disease severity, body disproportion, and primary bone abnormality. Recombinant human growth hormone (rhGH) increases phosphate tubular reabsorption and phosphate level in blood and, thus, constitutes an attractive but controversial therapy in short children with XLHR, those efficacy was demonstrated in small uncontrolled series. Our aim was to report our experience regarding growth in XLHR. Twenty-seven children with XLHR--20 girls, seven boys--diagnosed at a median (md) of 1.46 years of age, (range 0.39-8.5 years), were studied at 10.12 years of age (1.58-18.56), md (range). All received oral treatment with phosphate and calcitriol. At the first visit, grouped Z-height was -1; (-4.58; 0.54) md (range). After 5 years' follow-up (0.92-15.6), Z-height was -0.91 (- 4.56; 0.17), not different from that at baseline (P = 0.465). In 16 children entirely controlled in our program upon presentation, a "catch up" phenomenon after the rickets had healed (P = 0.823) or throughout the long-term was not observed (P = 0.995). Eight patients had a Z-height </= -2SD at the last visit, and impaired linear growth was associated with age >2 years at diagnosis, male gender and non-adherence to treatment. Four children, all boys, received rhGH, and in two cases with sufficient follow up stature normalized. No rhGH side effects were observed, and phosphate and calcitriol doses remained stable. Linear growth failure appeared in a third of XLHR children. Efforts need to be made to reduce the age of diagnosis and to improve adherence to treatment. Treatment with rhGH should be considered early, after the rickets has been controlled, in those patients with impaired growth or delayed diagnosis.
由于低磷血症、疾病严重程度、身体比例失调和原发性骨异常,生长发育迟缓在X连锁低磷性佝偻病(XLHR)患儿中经常出现。重组人生长激素(rhGH)可增加肾小管对磷的重吸收和血液中的磷水平,因此,对于身材矮小的XLHR患儿来说,它是一种有吸引力但存在争议的治疗方法,其疗效已在小型非对照系列研究中得到证实。我们的目的是报告我们在XLHR患儿生长方面的经验。对27例XLHR患儿(20例女孩,7例男孩)进行了研究,这些患儿诊断时的中位年龄为1.46岁(范围0.39 - 8.5岁),研究时的年龄为10.12岁(1.58 - 18.56岁),为中位年龄(范围)。所有患儿均接受了磷酸盐和骨化三醇的口服治疗。初次就诊时,Z值身高为 -1;(-4.58;0.54),中位值(范围)。经过5年的随访(0.92 - 15.6),Z值身高为 -0.91(-4.56;0.17),与基线时无差异(P = 0.465)。在我们项目中就诊时完全得到控制的16例患儿中,未观察到佝偻病治愈后(P = 0.823)或长期的“追赶”现象(P = 0.995)。8例患者在最后一次就诊时Z值身高≤ -2SD,线性生长受损与诊断时年龄>2岁、男性性别及治疗依从性差有关。4例患儿,均为男孩,接受了rhGH治疗,其中2例随访充分,身高恢复正常。未观察到rhGH的副作用,磷酸盐和骨化三醇的剂量保持稳定。三分之一的XLHR患儿出现线性生长失败。需要努力降低诊断年龄并提高治疗依从性。对于生长受损或诊断延迟的患者,在佝偻病得到控制后,应尽早考虑使用rhGH治疗。