Melatonin deficiency and fibrous dysplasia: might a relation exist?
Fibrous dysplasia of bone might be monostotic, polystotic, or occurs as a part of McCune-Albright syndrome and Jaffe-Lichtenstein syndrome. Activating mutations of GNAS1 gene was identified in patients with fibrous dysplasia. However, fibrous dysplasia might occur in the absence of these mutations and fibrous dysplastic tissue was produced in vitro by the effects of excess exogenous cAMP on human osteogenic cells. It was proved that the fibrous dysplastic tissue is deficient in bone sialoprotein. Melatonin deficiency might be hypothesized in syndromes associated with fibrous dysplasia or formation of fibrous dysplasia-like tissue. The receptor RZR/ROR is the nuclear receptor of melatonin and the human bone sialoprotein gene contains a RZR/ROR response element. It was supposed that binding of melatonin to its membrane receptors results in changes in the levels of activity of nuclear cAMP that lead to alteration of expression of bone sialoprotein. Also, melatonin deficiency might increase cAMP in bone through its effect on prostaglandins of the E group. Further, melatonin deficiency might explain precocious puberty in cases of McCune-Albright syndrome. We might hypothesize that melatonin deficiency might play a role in development of fibrous dysplasia in some cases.
Abdel-Wanis
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ME
Orthopaedic Department, Faculty of Medicine, Kanazawa University, Kanazawa, Japan. wanis307@yahoo.com
Tsuchiya
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D001846:Bone Development / Q000502:physiology; D000242:Cyclic AMP / Q000502:physiology; D005357:Fibrous Dysplasia of Bone / Q000209:etiology* / Q000235:genetics; D019205:GTP-Binding Protein alpha Subunits, Gs / Q000172:deficiency / Q000235:genetics; D006801:Humans; D058574:Integrin-Binding Sialoprotein; D008550:Melatonin / Q000172:deficiency* / Q000502:physiology; D008954:Models, Biological; D011956:Receptors, Cell Surface / Q000235:genetics / Q000378:metabolism; D018160:Receptors, Cytoplasmic and Nuclear / Q000235:genetics / Q000378:metabolism; D044094:Receptors, Melatonin; D015290:Second Messenger Systems / Q000502:physiology; D012795:Sialoglycoproteins / Q000235:genetics / Q000378:metabolism
C546871:IBSP protein, human; D058574:Integrin-Binding Sialoprotein; D011956:Receptors, Cell Surface; D018160:Receptors, Cytoplasmic and Nuclear; D044094:Receptors, Melatonin; D012795:Sialoglycoproteins; D000242:Cyclic AMP; D019205:GTP-Binding Protein alpha Subunits, Gs; D008550:Melatonin