Carvalho, D

Freitas, P

Nogueira, C

Pereira, J

Rios, E

Souto, SB

Vinha, E

Portuguese

Institutional repository of Hospital de Braga

ÓRgÃO OfiCiAL DA sOCiEDADE PORTUgUEsA DE REUMATOLOgiA1CARTA AO EDiTOR1. Department of Endocrinology, Diabetes and Metabolism, Centro Hospitalar são João, Porto, Portugal; faculty of Medicine, University of Porto, Portugal2. Department of Endocrinology, Hospital de Braga, Braga, Portugal3. Department of Pathology, Centro Hospitalar são João, Porto,Portugal; faculty of Medicine, University of Porto, Portugal; institute of Molecular Pathology and immunology at the University of Porto, Portugal4. Department of neurosurgery, Centro Hospitalar são João,Porto, Portugalder treatment with alendronate and cholecalciferol.Glucocorticoids (GCs) cause impaired bone forma-tion due to direct effects both on osteoblasts and os-teoclasts1. GCs also inhibit vitamin D action, reducingcalcium absorption from the gastrointestinal tract andrenal tubular calcium reabsorption2. The effects of GCson gonadotropins, growth hormone and insulin-likegrowth factor-1 may also contribute to osteoporosis de-velopment, and catabolic effects of GCs on muscleenhance fracture risk.Fractures are found in approximately 30-67% of pa-tients with Cushing’s syndrome (CS)1. Trabecular boneis more severely affected than cortical bone in GCs in-duced osteoporosis, because of its higher turnover rateand its greater sensitivity to GCs action3,4. Therefore,the vertebral bodies and ribs are the typical sites of frac-tures in CS. The European Registry on Cushing’s syn-drome showed that, in patients with any form of CS,men had significantly more vertebral and rib frac-tures compared with women (52 vs.18% for vertebrae;p<0.001 and 34 vs. 23% for ribs; p<0.05)5. Male gen-der was found to be an important risk factor for lum-bar osteoporosis and fractures in CD6.Disease severity and duration seem to play a crucialrole in determining permanent damage to the spine. Inthis case, surgical treatment would be indicated but thepatient refused. Some authors reported that GCs indu-ced osteoporosis is reversible7, but recovery of boneloss is gradual and may continue for 10 years beforeBMD normalizes8. A recent study showed a significantincrease in BMD at 3 months after surgery in the lum-bar spine in patients cured of CS due to adrenocorticaladenoma7. Another study reported that alendronatemay induce a more rapid improvement in BMD thancortisol normalization alone, maybe by restoring thebalance between bone formation and resorption9. It isalso useful in patients with persistent postsurgical hy-percortisolism. Since there is a substantial reduction ofbone formation, treatment with an anabolic agent thatVertebral osteoporotic fractures with height loss secondary to Cushing’s diseaseNogueira C1, Souto SB2, Rios E3, Pereira J4, Vinha E1, Freitas P1, Carvalho D1To the editor,A 37-year-old man was referred to our department dueto osteoporosis complicated with vertebral and rib frac-tures and loss of six centimeters in height within theprevious year. He also had hypertension, dyslipidemia,and secondary infertility and he was treated with alen-dronate and cholecalciferol. Physical examination re-vealed a moon face, central obesity, proximal muscleweakness, easy bruising, and kyphosis. Laboratory in-vestigation showed ACTH dependent hypercortisolism[24-hour urinary free cortisol: 687.3µg/day (36–137);cortisol after a 1-mg overnight dexamethasone sup-pression test: 39.5µg/dL (<1.8); ACTH:118pg/mL (< 53)]. Total testosterone was low [1.93mg/mL (2.8--8.0)]. Since pituitary magnetic resonance imaging sho-wed no adenoma, he was submitted to bilateral inferiorpetrosal sinus sampling, which favored Cushing’s di-sease (CD). Dual-energy X-ray absorptiometry (DXA)revealed osteoporosis of the lumbar spine (T-score -3.8,Z-score -3.8) and of the left femur (T-score -3.6, Z-sco-re -2.7). A thoracic computed tomography showedmultiple vertebral compression fractures in the thora-cic spine and increased thoracic kyphosis (Figure 1).After cryopreservation of gametes, the patient wassubmit ted to transsphenoidal surgery and histologicalfindings confirmed the diagnosis. He refused surgicaltreatment of vertebral fractures. DXA performed fivemonths after surgery showed osteoporosis of the lum-bar spine (T-score -3.4, Z-score -3.6) and osteopenia ofthe left femur (T-score -2.0, Z-score -1.9). He is still un-ACTA REUMATOL PORT. OnLinE fiRsT ÓRgÃO OfiCiAL DA sOCiEDADE PORTUgUEsA DE REUMATOLOgiA2Vertebral osteoporotic fractures with height loss secondary to cushing’s diseasepromotes bone formation and reduces vertebral andnon-vertebral fractures has also been suggested1. Teri-paratide is indicated to increase bone mass in men withprimary or hypogonadal osteoporosis who are at highrisk for fracture10. Although its use has not been stu-died in endogenous CS it is beneficial in patients withexogenous GCs induced osteoporosis.In conclusion, this case illustrates the need to con-sider secondary causes of osteoporosis, namely Cus-hing’s syndrome and other endocrinopathies, in ayoung patient with osteoporotic fractures.CoRREspondEnCE toCláudia NogueiraServiço de Endocrinologia, Diabetes e MetabolismoCentro Hospitalar São JoãoAlameda Professor Hernâni Monteiro4200-319 Porto-PortugalREFEREnCEs 1. Kaltsas G, Makras P. Skeletal diseases in Cushing’s syndrome:osteoporosis versus arthropathy. Neuroendocrinology 2010;92 Suppl 1: 60-64.2. Mazziotti G, Angeli A, Bilezikian JP, Canalis E, Giustina A. Glu-cocorticoid-induced osteoporosis: an update. Trends Endocri-nol Metab 2006; 17: 144-149. 3. Sambrook PN. Corticosteroid osteoporosis: practical implica-tions of recent trials. J Bone Miner Res 2000; 15: 1645-1649. 4. Francucci CM, Pantanetti P, Garrapa GG, Massi F, Arnaldi G,Mantero F. Bone metabolism and mass in women with Cus-hing’s syndrome and adrenal incidentaloma. Clin Endocrinol(Oxf) 2002; 57: 587-593. 5. Valassi E, Santos A, Yaneva M, et al. The European Registry onCushing’s syndrome: 2-year experience. Baseline demographicand clinical characteristics. Eur J Endocrinol 2011; 165: 383--392. 6. Zilio M, Barbot M, Ceccato F, et al. Diagnosis and complica-tions of Cushing’s disease: gender-related differences. Clin En-docrinol (Oxf) 2014; 80: 403-410. 7. Kawamata A, Iihara M, Okamoto T, Obara T. Bone mineral den-sity before and after surgical cure of Cushing’s syndrome dueto adrenocortical adenoma: prospective study. World J Surg2008; 32: 890-896. 8. Mancini T, Doga M, Mazziotti G, Giustina A. Cushing’s syn-drome and bone. Pituitary 2004; 7: 249-252. 9. Di Somma C, Colao A, Pivonello R, et al. Effectiveness of chro-nic treatment with alendronate in the osteoporosis of Cushing’sdisease. Clin Endocrinol (Oxf) 1998; 48: 655-662. 10. Watts NB, Adler RA, Bilezikian JP, et al. Osteoporosis in men:an Endocrine Society clinical practice guideline. J Clin Endo-crinol Metab 2012; 97: 1802-1822. FIGURE 1. Reconstructed sagittal (1A) and coronal (1B) CT image shows numerous compression fracturesinvolving the thoracic and lumbar spine, osteopenia and increased thoracic kyphosisA B

Vertebral osteoporotic fractures with height loss secondary to Cushing’s disease

http://repositorio.hospitaldebraga.pt/bitstream/10400.23/670/1/992_vertebral_osteoporotic_fractures_with_height_loss_secondary_to_cushing_s_disease_file%20%281%29.pdf

Vertebral osteoporotic fractures with height loss secondary to Cushing’s disease

Abstract

Similar works

Full text

Available Versions

Institutional repository of Hospital de Braga