Recovery from Atrophic Autoimmune Thyroiditis in a Child: Thyroid Stimulation-Blocking Antibody as a Prognostic Marker.

IF 2.6 3区 医学 Q3 ENDOCRINOLOGY & METABOLISM
Chieko Kusano, Naoaki Hori, Tomonobu Hasegawa, Satoshi Narumi
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Abstract

Introduction: Atrophic autoimmune thyroiditis (AAT) is a form of autoimmune hypothyroidism characterized by the absence of a goiter. Thyroid stimulation-blocking antibody (TSBAb) has been detected in a subset of pediatric AAT cases. Although the disappearance of TSBAb has been related with the recovery of thyroid function in adult AAT cases, similar outcomes have not been documented in pediatric cases.

Case presentation: A 2-year-old Japanese boy presented for evaluation of stunted growth from 1 year 10 months of age. Tests for congenital hypothyroidism were negative on newborn screening, and he had no significant medical history. However, he showed symptoms of hypothyroidism (inactiveness, hair loss, dry skin), and primary hypothyroidism was confirmed by blood test (serum TSH level, 818 mU/L; serum free T4 level, <0.40 ng/dL). The patient exhibited a unique antibody profile: positive for TSH receptor antibody (TRAb) and TSBAb and negative for anti-thyroglobulin antibody (TgAb) and anti-peroxidase antibody (TPOAb). He was treated with levothyroxine, after which his growth was normalized. During the 8-year follow-up, the patient's TSBAb levels decreased, allowing for the discontinuation of levothyroxine therapy.

Conclusion: We reported the case of a 2-year-old boy diagnosed with AAT who presented with a characteristic antibody profile, negative for TgAb and TPOAb, but positive for TRAb and TSBAb. During 8 years of follow-up, TSBAb seroconversion to negative was observed, leading to treatment discontinuation at age 10 years. This case suggests that monitoring of TSBAb after a diagnosis of AAT may be used to determine treatment discontinuation even in children.

萎缩性自身免疫性甲状腺炎在儿童中的恢复:甲状腺刺激阻断抗体作为预后标志物。
萎缩性自身免疫性甲状腺炎(AAT)是一种以无甲状腺肿为特征的自身免疫性甲状腺功能减退症。甲状腺刺激阻断抗体(TSBAb)已在儿科AAT病例的一个子集中检测到。虽然TSBAb的消失与成人AAT患者甲状腺功能的恢复有关,但在儿科病例中没有类似的结果。病例介绍:一名2岁的日本男孩因1岁至10个月的发育迟缓而被提出评估。新生儿筛查先天性甲状腺功能减退试验呈阴性,且无明显病史。然而,他表现出甲状腺功能减退的症状(不活跃、脱发、皮肤干燥),并通过血液检查证实原发性甲状腺功能减退(血清TSH水平818 mU/L;血清游离T4水平< 0.40 ng/dL)。患者表现出独特的抗体谱:TSH受体抗体(TRAb)和TSBAb阳性,抗甲状腺球蛋白抗体(TgAb)和抗过氧化物酶抗体(TPOAb)阴性。他接受了左甲状腺素治疗,之后他的生长恢复正常。在8年的随访期间,患者的TSBAb水平下降,允许停止左旋甲状腺素治疗。结论:我们报告了一名2岁男孩被诊断为AAT,他表现出典型的抗体谱,TgAb和TPOAb阴性,但TRAb和TSBAb阳性。经过8年的随访,观察到TSBAb血清转化为阴性,导致10岁时停止治疗。本病例提示,在诊断AAT后监测TSBAb可用于确定是否停止治疗,即使在儿童中也是如此。
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来源期刊
Hormone Research in Paediatrics
Hormone Research in Paediatrics ENDOCRINOLOGY & METABOLISM-PEDIATRICS
CiteScore
4.90
自引率
6.20%
发文量
88
审稿时长
4-8 weeks
期刊介绍: The mission of ''Hormone Research in Paediatrics'' is to improve the care of children with endocrine disorders by promoting basic and clinical knowledge. The journal facilitates the dissemination of information through original papers, mini reviews, clinical guidelines and papers on novel insights from clinical practice. Periodic editorials from outstanding paediatric endocrinologists address the main published novelties by critically reviewing the major strengths and weaknesses of the studies.
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