Successful operative management of an asymptomatic chest lesion in hereditary multiple exostosis

Pelc, Hugh J.S.; Walker, J. C. F.; Davies, Geraint

doi:10.1097/bpb.0b013e32835875d2

Cited by 6 publications

(3 citation statements)

References 17 publications

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“…Alternatively, biological meshes derived from cadaveric human dermis, porcine intestinal submucosa, porcine dermis, bovine pericardium, and bovine dermis (SurgiMend ® ; Integra LifeSciences Corp., Plainsboro, NJ, United States) have been used with acceptable results[ 14 , 16 , 21 , 22 , 26 , 32 - 35 ]. For robust reconstruction of very large defects, metallic rib substitutes have been used in pediatric patients[ 19 , 36 , 37 ]. There have been no comparative studies of the different prosthetic materials, which should be a focus for future studies.…”

Section: Discussionmentioning

confidence: 99%

Chest wall osteochondroma resection with biologic acellular bovine dermal mesh reconstruction in pediatric hereditary multiple exostoses: A case report and review of literature

AlShehri¹

2023

World J Clin Cases

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BACKGROUND Hereditary multiple exostoses is a rare genetic disorder characterized by the growth of multiple osteochondromas affecting primarily long bones. Chest wall lesions may represent a challenge, particularly in pediatric patients. Pain is a common manifestation. However, life-threatening complications can result from direct involvement of adjacent structures. Surgical resection with appropriate reconstruction is often required. CASE SUMMARY A 5-year-old male who was diagnosed with hereditary multiple exostoses presented with significant pain from a large growing chest wall exostosis lesion. After appropriate preoperative investigations, he underwent surgical resection with reconstruction of his chest wall using a biologic bovine dermal matrix mesh. CONCLUSION Resection of chest wall lesions in children represents a challenge. Preoperative planning to determine the appropriate reconstruction strategy is essential.

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Section: Discussionmentioning

confidence: 99%

Chest wall osteochondroma resection with biologic acellular bovine dermal mesh reconstruction in pediatric hereditary multiple exostoses: A case report and review of literature

AlShehri¹

2023

World J Clin Cases

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“…A partial rib defect was reconstructed with a metal plate to stabilize the anterior chest wall[ 28 ]. In a 2-year-old girl, a Gore-Tex sheet was used to cover a 12 cm × 6 cm defect of the chest wall caused by rib resection[ 30 ].…”

Section: Discussionmentioning

confidence: 99%

Spontaneous pneumothorax in a 17-year-old male patient with multiple exostoses: A case report and review of the literature

Nakamura¹,

Asanuma²,

Shimamoto³

et al. 2021

WJO

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BACKGROUND Multiple exostoses generally develop in the first decade of life. They most frequently arise from the distal femur, proximal tibia, fibula, and proximal humerus. Costal exostoses are rare, contributing to 1%–2% of all exostoses in hereditary multiple exostoses (HME). They are usually asymptomatic, but a few cases have resulted in severe thoracic injuries. Pneumothorax caused by costal exostoses is rare, with only 13 previously reported cases. We report a new case of pneumothorax caused by costal exostoses. CASE SUMMARY A 17-year-old male with HME underwent surgery for removal of exostoses around his right knee. Four months following the operation, he felt chest pain when he was playing the trumpet; however, he did not stop playing for a week. He was referred to our hospital with a chief complaint of chest pain. The computed tomography (CT) scan revealed right pneumothorax and multiple exostoses in his right ribs. The CT scan also revealed visceral pleura thickness and damaged lung tissues facing the exostosis of the seventh rib. We diagnosed that exostosis of the seventh rib induced pneumothorax. Costal exostosis resection was performed by video-assisted thoracoscopic surgery (VATS) 2 wk after the onset. The patient’s postoperative course was uneventful, and there was no recurrence of pneumothorax for 2 years. CONCLUSION Costal exostoses causing thoracic injuries should be resected regardless of age. VATS must be considered in cases with apparently benign and relatively small exostoses or HME.

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“…Sci. 2020, 21, 2686 2 of 14 osteochondroma [5,7,8]. There are many risks associated with surgery and the development of a pharmacological therapy is a promising non-surgical alternative to potentially prevent the development and/or treat existing osteochondromas [5].…”

Section: Introductionmentioning

confidence: 99%

Understanding the Action of RARγ Agonists on Human Osteochondroma Explants

Garcia

Tian

Imamura‐Kawasawa

et al. 2020

IJMS

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Osteochondromas are cartilage-capped growths located proximate to the physis that can cause skeletal deformities, pain, limited motion, and neurovascular impingement. Previous studies have demonstrated retinoic acid receptor gamma (RARγ) agonists to inhibit ectopic endochondral ossification, therefore we hypothesize that RARγ agonists can target on established osteochondromas. The purpose of this study was to examine the action of RARγ agonist in human osteochondromas. Osteochondroma specimens were obtained during surgery, subjected to explant culture and were treated with RARγ agonists or vehicles. Gene expression analysis confirmed the up-regulation of RARγ target genes in the explants treated with NRX 204647 and Palovarotene and revealed strong inhibition of cartilage matrix and increased extracellular matrix proteases gene expression. In addition, immunohistochemical staining for the neoepitope of protease-cleaved aggrecan indicated that RARγ agonist treatment stimulated cartilage matrix degradation. Interestingly, cell survival studies demonstrated that RARγ agonist treatment stimulated cell death. Moreover, RNA sequencing analysis indicates changes in multiple molecular pathways due to RARγ agonists treatment, showing similarly to human growth plate chondrocytes. Together, these findings suggest that RARγ agonist may exert anti-tumor function on osteochondromas by inhibiting matrix synthesis, promoting cartilage matrix degradation and stimulating cell death.

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Successful operative management of an asymptomatic chest lesion in hereditary multiple exostosis

Cited by 6 publications

References 17 publications

Chest wall osteochondroma resection with biologic acellular bovine dermal mesh reconstruction in pediatric hereditary multiple exostoses: A case report and review of literature

Chest wall osteochondroma resection with biologic acellular bovine dermal mesh reconstruction in pediatric hereditary multiple exostoses: A case report and review of literature

Spontaneous pneumothorax in a 17-year-old male patient with multiple exostoses: A case report and review of the literature

Understanding the Action of RARγ Agonists on Human Osteochondroma Explants

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