Growth Hormone Therapy in Two Patients with Hereditary Multiple Exostosis

Galasso, Cinzia; Scirè, Giuseppe; M, Sanna; Carnazza, S; Bonaiuto, Francesco; Boscherini, B

doi:10.1177/000992289603501208

Cited by 4 publications

(5 citation statements)

References 6 publications

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“…Most individuals have at least one or multiple operative procedures (Bovée 2008). Growth hormone treatment has been performed in three cases of multiple osteochondroma (Galasso et al 1996, Martinez et al 2008.…”

Section: Introductionmentioning

confidence: 99%

Clinical and molecular studies of EXT1/EXT2 in Bulgaria

Stancheva-Ivanova

Wuyts

Hul

et al. 2011

J of Inher Metab Disea

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EXT1/EXT2-CDG (Multiple cartilagineous exostoses, hereditary multiple osteochondroma (MO); OMIM 133700/133701) are common defects of O-xylosylglycan glycosylation. The diagnostic criteria are at least two osteochondromas of the juxta-epiphyseal region of long bones with in the majority of cases a positive family history and/or mutation in one of the EXT genes. The authors report data on clinical symptoms and complications of 23 patients (from 16 families), discussing the family history, age of diagnosis, new clinical and molecular data. Fifteen mutations and large deletions, of which nine are new, were detected in the EXT1 and EXT2 gene by sequence analysis, FISH and MLPA analysis.

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Section: Introductionmentioning

confidence: 99%

Clinical and molecular studies of EXT1/EXT2 in Bulgaria

Stancheva-Ivanova

Wuyts

Hul

et al. 2011

J of Inher Metab Disea

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“…12 However, the results of most studies suggest that GHT is not associated with an increased likelihood of disease relapse. 13 There is no report of OS developing following GHT, 14 however, two-thirds and one-half of Harper et al's 6 and our patients respectively received GHT when OS was identified. Nevertheless, this did not reach statistical significance.…”

Section: Discussionmentioning

confidence: 56%

Osteochondroma after pediatric hematopoietic stem cell transplantation: report of eight cases

Bordigoni

Clément

et al. 2002

Bone Marrow Transplant

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Summary:Eight children developed osteochondroma (OS) at a mean of 88 months after hematopoietic stem cell transplantation (HSCT). The mean age at HSCT was 56 months (12-84). This represents a cumulative incidence of 20% among patients less than 18 years of age transplanted from 1981 to 1997. These eight patients underwent allogeneic (n = 2) or autologous (n = 6) transplantation for either acute leukemia (n = 6) or neuroblastoma (n = 2) after a conditioning regimen including TBI (n = 7) or a combination of Bu and CY. OS was multiple in seven patients and solitary in one. Eight lesions were resected and all were benign. Four children received growth hormone before diagnosis of OS, but there was no clinical, radiological or histological difference between those who did not. Univariate analysis showed an increased rate associated only with autologous HSCT, with a 31.7% probability of a new OS at 12 years after HSCT. Osteochondroma should be added to the other adverse effects of HSCT in children.

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“…GH replacement therapy was discontinued because of a poor linear growth response and a moderate increase in size of the exostoses. However, the diagnosis of GH deficiency was not confirmed by a third test [ 22 ]. Both the authors concluded that a careful follow-up in these patients is mandatory, even if no evidence of adverse effects on disease course was proven in their two patients.…”

Section: Discussionmentioning

confidence: 99%

Hereditary multiple exostoses and solitary osteochondroma associated with growth hormone deficiency: to treat or not to treat?

et al. 2015

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BackgroundOsteochondroma generally occurs as a single lesion and it is not a heritable disease. When two or more osteochondroma are present, this condition represents a genetic disorder named hereditary multiple exostoses (HME). Growth hormone deficiency (GHD) has rarely been found in HME patients and a few data about growth therapy (GH) therapy effects in development/growth of solitary or multiple exostoses have been reported.Case presentationWe describe the clinical features of 2 patients (one with osteochondroma and one with HME) evaluated before and after GH therapy. In the first patient, the single osteochondroma was noticed after the start of treatment; the other patient showed no evidence of significant increase in size or number of lesions related to GH therapy.ConclusionIt is necessary to investigate GH secretion in patients with osteochondroma or HME and short stature because they could benefit from GH replacement therapy. Moreover, careful clinical and imaging follow-up of exostoses is mandatory.

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Growth Hormone Therapy in Two Patients with Hereditary Multiple Exostosis

Cited by 4 publications

References 6 publications

Clinical and molecular studies of EXT1/EXT2 in Bulgaria

Clinical and molecular studies of EXT1/EXT2 in Bulgaria

Osteochondroma after pediatric hematopoietic stem cell transplantation: report of eight cases

Hereditary multiple exostoses and solitary osteochondroma associated with growth hormone deficiency: to treat or not to treat?

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