Infantile Cortical Hyperostosis: Report of a Case with Observations on Clinical Manifestations, Radiology, and Pathology with a Late Follow-Up of Eight Years

Abstract

Purpose. The purpose of our study was to investigate clinical manifestations, roentgen images, histopathological studies, and evolution of the disease in patient displaying infantile cortical hyperostosis. Methods. Roentgenograms were made to evaluate a neonatal patient presenting multiple soft-tissue swellings. The initial radiographs insinuated that the disease had been present for some time in utero. Bone puncture biopsy of the tibia for histopathological observation and diagnosis conclusions was performed. Results. The disease was demonstrated radiographically by massive cortical diaphyseal thickening and also extensive periosteal new bone formation surrounding several bones. Results in blood count were as follows: discrete anemia, moderate leukocytosis, and elevated sedimentation rate. Histological pattern of tissue removed from tibia showed lamellar cortical bones and hyperplasia. Biopsy studies disclosed no evidence of neoplasia as well as of bacterial infection. Comments. Clinical manifestations in a neonatal patient displaying infantile cortical hyperostosis have gradually decreased. Radiograph findings have demonstrated complete recovery of bones manifested by the disease. The pathologic findings are in accordance with previous microscopic examination summarized by the literature. Total patient cure, without sequels, could be demonstrated.

Polyostotic cortical hyperostosis in an 8‐week‐old cat with a 3‐year follow‐up

“…It is characterised by cortical hyperostosis, commonly involving the mandible and appendicular skeleton. The initial symptoms usually appear within the first 6 months of life, with a mean age at onset of 9 weeks (Caffey & Silverman 1945, Borochowitz et al 1991, Kamoun‐Goldrat & Le Merrer 2007, Shandilya et al 2013), and they are represented by soft tissue swelling, irritability and pain on palpation due to cortical thickening that typically involves diaphysis, mandible, ribs and clavicles (Shandilya et al 2013, Pinheiro et al 2016). The infantile condition is generally self‐limiting with spontaneous regression within 3 years of life (Newberg & Tampas 1981, Kamoun‐Goldrat & Le Merrer 2007, Navarre et al 2013, Nistala et al 2014).…”

Craniofacial disorders and dysplasias: Molecular, clinical, and management perspectives

“…The periosteal reaction presents as an “onion-peel” or “wave-like” pattern characterized by multiple layers of new bone formation parallel to the long axis of the bone. There may be associated soft tissue swelling around the affected bones due to inflammation ( Pinheiro et al, 2016 ).…”