Literature DB >> 29383307

Colonic Angioectasia in an Adolescent Boy with Hoyeraal-Hreidarsson on Long-Term Anabolic Steroid Therapy.

Racha Khalaf1, Carmen Cuffari2.   

Abstract

Androgen therapy has proven efficacy in treating patients with bone marrow failure who are not candidates for bone marrow transplantation. Herein, we report on a case of colonic angioectasia secondary to oxymetholone use in an adolescent patient with Hoyeraal-Hreidarsson syndrome (HHS). A 13-year-old Caucasian male with HHS characterized by cerebellar hypoplasia, developmental delay, microcephaly, esophageal strictures and myelodysplasia presented with severe hematochezia from colonic angioectasia secondary to long-term oxymetholone therapy. These vascular lesions resolved spontaneously once this anabolic steroid was discontinued. While androgen therapy is often recommended for certain anemias and myelodysplastic syndromes, clinicians should be aware of the potential complication in developing these perceived uncommon colonic angioectasias. Moreover, pediatric gastroenterologists should familiarize themselves in identifying these vascular lesions by colonoscopy, especially among the high risk groups on long-term anabolic steroid therapy.

Entities:  

Keywords:  Angioectasia; Colon; Hoyeraal-Hreidarsson syndrome; Testosterone congeners

Year:  2018        PMID: 29383307      PMCID: PMC5788953          DOI: 10.5223/pghn.2018.21.1.68

Source DB:  PubMed          Journal:  Pediatr Gastroenterol Hepatol Nutr        ISSN: 2234-8840


  8 in total

Review 1.  Review of oxymetholone: a 17alpha-alkylated anabolic-androgenic steroid.

Authors:  A M Pavlatos; O Fultz; M J Monberg; A Vootkur
Journal:  Clin Ther       Date:  2001-06       Impact factor: 3.393

2.  Haematological recovery in dyskeratosis congenita patients treated with danazol.

Authors:  Amin Islam; Shafquat Rafiq; Mike Kirwan; Amanda Walne; Jamie Cavenagh; Tom Vulliamy; Inderjeet Dokal
Journal:  Br J Haematol       Date:  2013-06-19       Impact factor: 6.998

3.  What are the essential symptoms in the Hoyeraal-Hreidarsson syndrome?

Authors:  S Ohga; T Kai; K Honda; H Nakayama; T Inamitsu; K Ueda
Journal:  Eur J Pediatr       Date:  1997-01       Impact factor: 3.183

4.  Response to androgen therapy in patients with dyskeratosis congenita.

Authors:  Payal P Khincha; Ingrid M Wentzensen; Neelam Giri; Blanche P Alter; Sharon A Savage
Journal:  Br J Haematol       Date:  2014-02-12       Impact factor: 6.998

Review 5.  Pathophysiology and management of inherited bone marrow failure syndromes.

Authors:  Akiko Shimamura; Blanche P Alter
Journal:  Blood Rev       Date:  2010-04-24       Impact factor: 8.250

6.  Danazol-induced cystitis: an undescribed source of hematuria in patients with hereditary angioneurotic edema.

Authors:  G L Andriole; C Brickman; E E Lack; I A Sesterhenn; N Javadpour; W M Linehan; M M Frank
Journal:  J Urol       Date:  1986-01       Impact factor: 7.450

7.  Gastrointestinal involvement in a woman with dyskeratosis congenita.

Authors:  K E Brown; T E Kelly; B M Myers
Journal:  Dig Dis Sci       Date:  1993-01       Impact factor: 3.199

Review 8.  Unraveling the pathogenesis of Hoyeraal-Hreidarsson syndrome, a complex telomere biology disorder.

Authors:  Galina Glousker; Fabien Touzot; Patrick Revy; Yehuda Tzfati; Sharon A Savage
Journal:  Br J Haematol       Date:  2015-05-04       Impact factor: 6.998

  8 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.