Literature DB >> 14594174

Growth hormone treatment in Aarskog syndrome: analysis of the KIGS (Pharmacia International Growth Database) data.

Feyza Darendeliler1, Pontus Larsson, Olcay Neyzi, Anthony D Price, Lars Hagenäs, Ilkka Sipilä, Ann Christin Lindgren, Barto Otten, Bert Bakker.   

Abstract

Aarskog syndrome is an X-linked disorder characterized by faciogenital dysplasia and short stature. The present study set out to determine the effect of growth hormone (GH) therapy in patients with Aarskog syndrome enrolled in KIGS--the Pharmacia International Growth Database. Twenty-one patients (20 males) were evaluated. Median age at start of treatment was 8.3 years (10-90th percentiles, 5.1-14.1 years) and median height SDS was -2.8 (10-90th percentiles, -2.1 to -3.7). The median dose of GH was 0.22 mg/kg/week (10-90th percentiles, 0.15-0.30 mg/kg/week) given at a median frequency of six (4-7) times per week. Prepubertal patients were followed longitudinally for 1 year (n = 13) or 3 years (n = 7). After 1 year, the median height SDS had improved from -2.8 to -2.3 in 13 patients. After 3 years, height SDS had improved significantly (p <0.05) to -1.8 (10-90th percentiles, -2.1 to -1.1) in the seven patients. No adverse events were noted. Although final height data for these patients are still awaited, the present results support the use of GH to promote growth in children with Aarskog syndrome.

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Year:  2003        PMID: 14594174     DOI: 10.1515/jpem.2003.16.8.1137

Source DB:  PubMed          Journal:  J Pediatr Endocrinol Metab        ISSN: 0334-018X            Impact factor:   1.634


  5 in total

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Authors:  Danielle Christine Maria van der Kaay; Anne Rochtus; Gerhard Binder; Ingo Kurth; Dirk Prawitt; Irène Netchine; Gudmundur Johannsson; Anita C S Hokken-Koelega; Miriam Elbracht; Thomas Eggermann
Journal:  Endocr Connect       Date:  2022-10-10       Impact factor: 3.221

Review 2.  Syndromic disorders with short stature.

Authors:  Zeynep Şıklar; Merih Berberoğlu
Journal:  J Clin Res Pediatr Endocrinol       Date:  2014

3.  FGD1 Variant Associated With Aarskog-Scott Syndrome.

Authors:  Yilin Zhu; Qingqing Chen; Haiyan Lin; Huifei Lu; Yangbin Qu; Qingfeng Yan; Chunlin Wang
Journal:  Front Pediatr       Date:  2022-07-14       Impact factor: 3.569

4.  Case Report: Aarskog-scott syndrome caused by FGD1 gene variation: A family study.

Authors:  Yijia Liang; Honglin Wu; Xiumei He; Xiyu He
Journal:  Front Genet       Date:  2022-08-16       Impact factor: 4.772

5.  A novel frameshift mutation in the FGD1 gene causing Aarskog-Scott syndrome patient with hypogonadism: a case report.

Authors:  Hongshuai Jia; Tiantian Ma; Ziqin Liu; Yuru Ouyang; Chunsheng Hao
Journal:  Transl Pediatr       Date:  2021-05
  5 in total

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