STUDY DESIGN: A case report and review of previous literature are presented. OBJECTIVE: The objective of this manuscript was to report a case of pseudohypoparathyroidism (PHP) 1a simulating diffuse idiopathic skeletal hyperostosis and resulting in spinal cord compression, and discuss the pathogenesis of this disease. SUMMARY OF BACKGROUND DATA: Spinal cord compression due to PHP is not common, to the authors' knowledge, none of the previous reported cases simulated diffuse idiopathic skeletal hyperostosis. METHODS: The patient's history, clinical examination, imaging findings, and treatment were reported; and the pathogenesis was discussed. RESULTS: Characteristic findings were revealed from imaging studies, a multiplane reconstruction of the computed tomography images, and magnetic resonance imaging. The patient was treated by 2-stage posterior decompression on the basis of the images. A mild improvement was observed after the surgeries and the patient's neurology was not completely restored after 6 months. CONCLUSION: We reported a rare case of skeletal and ligamentous abnormality resulting in spinal cord compression associated with PHP. A multiplane reconstruction of the computed tomography images was very necessary for diagnosis and treatment of this case. The unfavorable neurologic restoration might be due to the severe injury of the spinal cord caused by diffuse ossification of the posterior longitudinal ligament and ligamentum flavum.
STUDY DESIGN: A case report and review of previous literature are presented. OBJECTIVE: The objective of this manuscript was to report a case of pseudohypoparathyroidism (PHP) 1a simulating diffuse idiopathic skeletal hyperostosis and resulting in spinal cord compression, and discuss the pathogenesis of this disease. SUMMARY OF BACKGROUND DATA: Spinal cord compression due to PHP is not common, to the authors' knowledge, none of the previous reported cases simulated diffuse idiopathic skeletal hyperostosis. METHODS: The patient's history, clinical examination, imaging findings, and treatment were reported; and the pathogenesis was discussed. RESULTS: Characteristic findings were revealed from imaging studies, a multiplane reconstruction of the computed tomography images, and magnetic resonance imaging. The patient was treated by 2-stage posterior decompression on the basis of the images. A mild improvement was observed after the surgeries and the patient's neurology was not completely restored after 6 months. CONCLUSION: We reported a rare case of skeletal and ligamentous abnormality resulting in spinal cord compression associated with PHP. A multiplane reconstruction of the computed tomography images was very necessary for diagnosis and treatment of this case. The unfavorable neurologic restoration might be due to the severe injury of the spinal cord caused by diffuse ossification of the posterior longitudinal ligament and ligamentum flavum.
Authors: Giovanna Mantovani; Murat Bastepe; David Monk; Luisa de Sanctis; Susanne Thiele; Alessia Usardi; S Faisal Ahmed; Roberto Bufo; Timothée Choplin; Gianpaolo De Filippo; Guillemette Devernois; Thomas Eggermann; Francesca M Elli; Kathleen Freson; Aurora García Ramirez; Emily L Germain-Lee; Lionel Groussin; Neveen Hamdy; Patrick Hanna; Olaf Hiort; Harald Jüppner; Peter Kamenický; Nina Knight; Marie-Laure Kottler; Elvire Le Norcy; Beatriz Lecumberri; Michael A Levine; Outi Mäkitie; Regina Martin; Gabriel Ángel Martos-Moreno; Masanori Minagawa; Philip Murray; Arrate Pereda; Robert Pignolo; Lars Rejnmark; Rebecca Rodado; Anya Rothenbuhler; Vrinda Saraff; Ashley H Shoemaker; Eileen M Shore; Caroline Silve; Serap Turan; Philip Woods; M Carola Zillikens; Guiomar Perez de Nanclares; Agnès Linglart Journal: Nat Rev Endocrinol Date: 2018-08 Impact factor: 43.330