Klinefelter Bone Microarchitecture Evolution with Testosterone Replacement Therapy

Piot, A.; Plotton, Ingrid; Boutroy, Stéphanie; Bacchetta, Justine; Ailloud, S.; Lejeune, Hervé; Chapurlat, Roland; Szulc, Paweł; Confavreux, Cyrille

doi:10.1007/s00223-022-00956-2

Cited by 10 publications

(9 citation statements)

References 44 publications

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“…This criterium can be seen as an arbitrary choice for starting TRT. It has been previously reported that starting TRT at testosterone levels below 200 ng/dL potentially yields more beneficial effects on BMD 14,28–30 …”

Section: Discussionmentioning

confidence: 99%

“…7 Various effects have been described, both positive and indifferent, on multiple clinical manifestations of KS. 10 As for the effect on bone, positive results were found after exclusive TRT, combination with vitamin D, early therapy start, or after focusing on men with overt androgen deficiency [11][12][13][14][15][16][17][18] ; however, in other studies TRT has proven ineffective in the prevention of low BMD. 8,[19][20][21][22] In some studies, osteoporosis/osteopenia could be resolved.…”

Section: Introductionmentioning

confidence: 99%

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The effect of testosterone treatment on bone mineral density in Klinefelter syndrome: A retrospective cohort study

et al. 2023

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BackgroundAlthough Klinefelter syndrome (KS) is the most frequent sex‐hormone disorder, there is ongoing uncertainty about the often associated sex‐hormone deficiency, its impact on common comorbidities, and therefore about prevention and treatment. In this study, we focus on bone loss, reported to occur in over 40% of KS patients, and the impact of testosterone replacement therapy (TRT) on this comorbidity.ObjectivesThis single‐center retrospective cohort study in a tertiary hospital compared the effect of treatment with TRT to no TRT on evolution of bone mineral density (BMD) in KS patients.MethodsAfter a medical chart review, a total of 52 KS subjects were included in this study. BMD was measured by dual‐energy X‐ray absorptiometry (DXA) and expressed as T‐scores.ResultsThe subjects were divided into three groups, according to TRT. In the subgroup that only started TRT after baseline measurements (mean age 31 years), we observed significant gain in BMD T‐score at the lumbar spine (0.58 ± 0.60, p = 0.003; mean gain of 0.62% areal BMD per year) and total femur T‐score (0.24 ± 0.39, p = 0.041; mean gain of 0.25% areal BMD per year) after a mean follow‐up period of 7.5 years. Compared to untreated subjects, a significant difference in evolution was demonstrated at the lumbar level (+0.58 ± 0.60 vs. −0.14 ± 0.42, p = 0.007). In untreated subjects with normal testosterone levels, a loss of BMD (−0.27 ± 0.37, p = 0.029; mean loss of 0.49% areal BMD per year) at the femoral neck was measured. This decline was equal to the predicted loss seen in the general male population.ConclusionTRT results in BMD gain in patients with KS with testosterone deficiency, mainly at the lumbar spine. However, this effect is limited (0.62% per year). Patients who were not treated with TRT because of sufficient endogenous testosterone levels, showed only the predicted age‐related bone loss during follow‐up. The need for TRT in maintaining bone health in KS should be evaluated on an individual basis according to the degree of sex steroid deficiency.

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Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

The effect of testosterone treatment on bone mineral density in Klinefelter syndrome: A retrospective cohort study

et al. 2023

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“…Lower BMD and altered bone microarchitecture has been reported in studies of adults with KS (26, 37,38,39,40,41,42). In a recent meta-analysis of 1141 patients with KS, a reduced BMD with an improvement after initiation of TRT was observed (43), but the relation between testosterone and bone mineral status in KS is not completely clear.…”

Section: Bone Mineral Contentmentioning

confidence: 97%

Reproductive hormones, bone mineral content, body composition, and testosterone therapy in boys and adolescents with Klinefelter syndrome

Krabbe

Petersen

Asserhøj

et al. 2023

Endocrine Connections

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Adult patients with Klinefelter syndrome (KS) are characterized by a highly variable phenotype including tall stature, obesity and hypergonadotropic hypogonadism as well as an increased risk of developing insulin resistance, metabolic syndrome, and osteoporosis. Most adults need testosterone replacement therapy (TRT), whereas the use of TRT during puberty has been debated. In this retrospective, observational study reproductive hormones and whole-body dual-energy X-ray absorptiometry (DXA) derived body composition and bone mineral content were standardized to age-related standard deviation scores (SDS) in 62 patients with KS aged 5.9 to 20.6 years. Serum concentrations of total testosterone and inhibin B were low, whereas LH and FSH were high in patients before TRT. Despite normal body mass index (BMI), body fat% and ratio between android fat% and gynoid fat% were significantly higher in the entire group irrespective of treatment status. In patients evaluated before and during TRT a tendency towards a more beneficial body composition with a significant reduction in ratio between android fat% and gynoid fat% during TRT was found. Bone mineral content (BMC) did not differ from the reference, but BMC corrected for bone area was significantly lower when compared to the reference. This study confirms that patients with KS have an unfavorable body composition and an impaired bone mineral status already during childhood and adolescence. Systematic studies are needed to evaluate whether TRT during puberty will improve these parameters.

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“…However, this is inferred from other conditions of male hypogonadism and from observational studies ( 21 , 23 , 24 , 25 ). Studies using high-resolution pQCT, a CT-based method that allows the study of the microarchitecture of bone, have shown that the microarchitecture is changed with reduced trabecular density at the tibia ( 26 ) and lower cortical BMD at the radius, but with an increase during TRT ( 27 ). In addition, another study demonstrated that in young adults with Klinefelter syndrome treated from adolescence, most but not all parameters determined with high-resolution pQCT improved ( 28 ).…”

Section: Klinefelter Syndrome Hypogonadism Bone Muscle and Fat Metabo...mentioning

confidence: 99%

New developments and future trajectories in supernumerary sex chromosome abnormalities: a summary of the 2022 3rd International Workshop on Klinefelter Syndrome, Trisomy X, and XYY

Gravholt

Ferlin

Gromoll

et al. 2023

Endocrine Connections

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The 3rd International Workshop on Klinefelter Syndrome, Trisomy X, 47, and XYY syndrome was held in Leiden, the Netherlands, on September 12-14, 2022. Here, we review new data presented at the workshop and discuss scientific and clinical trajectories. We focus on shortcomings in knowledge and therefore point out future areas for research. We focus on the genetics and genomics of supernumerary sex chromosome syndromes with new data being presented. Most knowledge center specifically on Klinefelter syndrome, where aspects on testosterone deficiency and the relation to bone, muscle and fat was discussed, as was infertility and the treatment thereof. Both Trisomy X, 47 and XYY syndrome are frequently affected by infertility. Transitioning of males with Klinefelter syndrome was addressed, as this seemingly simple process is in practice often difficult. It is now realized that neurocognitive changes are pervasive in all supernumerary sex chromosome syndromes, which was extensively discussed. New intervention projects were also described and exciting new data concerning this was presented. Advocacy organizations were present, describing the enormous burden carried by parents when having to explain their child’s specific syndrome to most professionals whenever in contact with health care and education systems. It was also pointed out that most countries do not have health care systems that diagnose patients with supernumerary sex chromosome syndromes, thus pinpointing a clear deficiency in the current genetic testing and care models. At the end of the workshop a roadmap towards the development of new international clinical care guidelines for Klinefelter syndrome was decided.

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Klinefelter Bone Microarchitecture Evolution with Testosterone Replacement Therapy

Cited by 10 publications

References 44 publications

The effect of testosterone treatment on bone mineral density in Klinefelter syndrome: A retrospective cohort study

The effect of testosterone treatment on bone mineral density in Klinefelter syndrome: A retrospective cohort study

Reproductive hormones, bone mineral content, body composition, and testosterone therapy in boys and adolescents with Klinefelter syndrome

New developments and future trajectories in supernumerary sex chromosome abnormalities: a summary of the 2022 3rd International Workshop on Klinefelter Syndrome, Trisomy X, and XYY

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