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Tiwari C, Borkar N, Hussain N, Khubchandani N. Solitary osteochondroma of the rib: An unusual chest wall tumor in the pediatric age group. J Cancer Res Ther 2023; 19:1423-1425. [PMID: 37787320 DOI: 10.4103/jcrt.jcrt_679_21] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 11/04/2022]
Abstract
Pediatric chest wall tumors are unusual and can arise from bone structures or from adjacent soft tissues. Osteochondroma is a benign cartilaginous tumor arising from the metaphysis of bone; however, it is more common in extremity rather than in membranous bone. Although benign, osteochondroma of the rib may lead to fatal complications such as pneumothorax, hemothorax, fractures, and pleural or pericardial effusion. Therefore, some form of surgical management becomes necessary to treat these lesions. We present a case of 7-year-old female child with solitary osteochondroma of the rib. The tumor was surgically excised and the child is asymptomatic on follow-up.
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Affiliation(s)
- Charu Tiwari
- Department of Paediatric Surgery, All India Institute of Medical Sciences, Raipur, Chhattisgarh, India
| | - Nitinkumar Borkar
- Department of Paediatric Surgery, All India Institute of Medical Sciences, Raipur, Chhattisgarh, India
| | - Nighat Hussain
- Department of Pathology, All India Institute of Medical Sciences, Raipur, Chhattisgarh, India
| | - Naveen Khubchandani
- Department of Paediatric Surgery, All India Institute of Medical Sciences, Raipur, Chhattisgarh, India
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Alshehri A. Chest wall osteochondroma resection with biologic acellular bovine dermal mesh reconstruction in pediatric hereditary multiple exostoses: A case report and review of literature. World J Clin Cases 2023; 11:4123-4132. [PMID: 37388792 PMCID: PMC10303597 DOI: 10.12998/wjcc.v11.i17.4123] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/22/2023] [Revised: 05/02/2023] [Accepted: 05/12/2023] [Indexed: 06/12/2023] Open
Abstract
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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Affiliation(s)
- Abdullah Alshehri
- Department of Surgery, College of Medicine, King Saud University, Riyadh 11623, Saudi Arabia
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Nakamura K, Asanuma K, Shimamoto A, Kaneda S, Yoshida K, Matsuyama Y, Hagi T, Nakamura T, Takao M, Sudo A. Spontaneous pneumothorax in a 17-year-old male patient with multiple exostoses: A case report and review of the literature. World J Orthop 2021; 12:945-953. [PMID: 34888155 PMCID: PMC8613679 DOI: 10.5312/wjo.v12.i11.945] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 06/27/2021] [Revised: 08/07/2021] [Accepted: 09/17/2021] [Indexed: 02/06/2023] Open
Abstract
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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Affiliation(s)
- Koichi Nakamura
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Kunihiro Asanuma
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Akira Shimamoto
- Department of Thoracic and Cardiovascular Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Shinji Kaneda
- Department of Thoracic and Cardiovascular Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Keisuke Yoshida
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Yumi Matsuyama
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Tomohito Hagi
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Tomoki Nakamura
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Motoshi Takao
- Department of Thoracic and Cardiovascular Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
| | - Akihiro Sudo
- Department of Orthopedic Surgery, Mie University Graduate School of Medicine, Tsu 514-8507, Mie Prefecture, Japan
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Garcia SA, Tian H, Imamura-Kawasawa Y, Fisher A, Cellini A, Codd C, Herzenberg JE, Abzug JM, Ng V, Iwamoto M, Enomoto-Iwamoto M. Understanding the Action of RARγ Agonists on Human Osteochondroma Explants. Int J Mol Sci 2020; 21:E2686. [PMID: 32294904 PMCID: PMC7215996 DOI: 10.3390/ijms21082686] [Citation(s) in RCA: 7] [Impact Index Per Article: 1.4] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Grants] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/08/2020] [Revised: 04/03/2020] [Accepted: 04/08/2020] [Indexed: 12/18/2022] Open
Abstract
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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Affiliation(s)
- Sonia A. Garcia
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Hongying Tian
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Yuka Imamura-Kawasawa
- Departments of Pharmacology and Biochemistry and Molecular Biology, Institute for Personalized Medicine, Pennsylvania State University College of Medicine, Hershey, PA 17033, USA;
| | - Aidan Fisher
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Ashley Cellini
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Casey Codd
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - John E. Herzenberg
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
- Pediatric Orthopaedics, Sinai Hospital, Baltimore, MD 21215, USA
| | - Joshua M. Abzug
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Vincent Ng
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Masahiro Iwamoto
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
| | - Motomi Enomoto-Iwamoto
- Department of Orthopaedics, University of Maryland School of Medicine, 20 Penn Street, HSFII, Baltimore, MD 21201, USA; (S.A.G.); (H.T.); (A.F.); (A.C.); (C.C.); (J.E.H.); (J.M.A.); (V.N.); (M.I.)
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Mazza D, Fabbri M, Calderaro C, Iorio C, Labianca L, Poggi C, Turturro F, Montanaro A, Ferretti A. Chest pain caused by multiple exostoses of the ribs: A case report and a review of literature. World J Orthop 2017; 8:436-440. [PMID: 28567348 PMCID: PMC5434351 DOI: 10.5312/wjo.v8.i5.436] [Citation(s) in RCA: 8] [Impact Index Per Article: 1.0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 10/10/2016] [Revised: 12/19/2016] [Accepted: 03/02/2017] [Indexed: 02/06/2023] Open
Abstract
The aim of this paper is to report an exceptional case of multiple internal exostoses of the ribs in a young patient affected by multiple hereditary exostoses (MHE) coming to our observation for chest pain as the only symptom of an intra-thoracic localization. A 16 years old patient with familiar history of MHE came to our observation complaining a left-sided chest pain. This pain had increased in the last months with no correlation to a traumatic event. The computed tomography (CT) scan revealed the presence of three exostoses located on the left third, fourth and sixth ribs, all protruding into the thoracic cavity, directly in contact with visceral pleura. Moreover, the apex of the one located on the sixth rib revealed to be only 12 mm away from pericardium. Patient underwent video-assisted thoracoscopy with an additional 4-cm mini toracotomy approach. At the last 1-year follow-up, patient was very satisfied and no signs of recurrence or major complication had occured. In conclusion, chest pain could be the only symptom of an intra-thoracic exostoses localization, possibly leading to serious complications. Thoracic localization in MHE must be suspected when patients complain chest pain. A chest CT scan is indicated to confirm exostoses and to clarify relationship with surrounding structures. Video-assisted thoracoscopic surgery can be considered a valuable option for exostoses removal, alone or in addiction to a mini-thoracotomy approach, in order to reduce thoracotomy morbidity.
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Maeda K, Watanabe T, Sato K, Takezoe T, Migita M, Takahashi M, Ohno M, Tahara K, Fuchimoto Y, Uchikawa S, Takayama S, Kanamori Y. Two cases of asymptomatic rib exostosis treated by prophylactic surgical excision. JOURNAL OF PEDIATRIC SURGERY CASE REPORTS 2017. [DOI: 10.1016/j.epsc.2017.03.004] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 02/08/2023] Open
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