1.A case of vanishing mandible: Diagnosis and treatment considerations for Gorham-Stout disease of the mandible
Harroun Valdimir T. Wong ; Johanna Patricia A. Canal
Acta Medica Philippina 2025;59(5):75-81
Gorham-Stout disease is a rare osteolytic disorder with an unclear pathophysiology. It presents as lesions characterized by the loss of the bony matrix and the proliferation of malformed vasculature. At present, there are no gold-standard diagnostic evaluation protocols and it is diagnosed through a mixture of clinical, histopathologic, and radiographic findings. We report a case of a 19-year-old female with Gorham-Stout disease presenting with an 8-year progressive soft tissue mass in the mandible. Extensive osteolysis of the mandible with clustering of the mandibular dentition is noted on computed tomography (CT) imaging. Her case was discussed in a multidisciplinary conference and her treatment was radiotherapy followed by surgery ± reconstruction. We used a CT-based three-dimensional planning technique to give 40 Gy over 20 treatment sessions to the involved areas. Post treatment, a repeat CT was done at six weeks to reassess for disease progression or stabilization, followed by surgical excision. As of 31 October 2021, no evidence of recurrence is noted 48 months after treatment. Arriving at a definitive diagnosis with GorhamStout disease is challenging and a multidisciplinary team approach can help determine the treatment choice with best outcomes.
Osteolysis, Essential ; Gorham-stout Disease
2.A case of vanishing mandible: Diagnosis and treatment considerations for Gorham-Stout disease of the mandible
Harroun Valdimir T. Wong ; Johanna Patricia A. Canal
Acta Medica Philippina 2024;58(Early Access 2024):1-7
Gorham-Stout disease is a rare osteolytic disorder with an unclear pathophysiology. It presents as lesions characterized by the loss of the bony matrix and the proliferation of malformed vasculature. At present, there are no gold-standard diagnostic evaluation protocols and it is diagnosed through a mixture of clinical, histopathologic, and radiographic findings. We report a case of a 19-year-old female with Gorham-Stout disease presenting with an 8-year progressive soft tissue mass in the mandible. Extensive osteolysis of the mandible with clustering of the mandibular dentition is noted on computed tomography (CT) imaging. Her case was discussed in a multidisciplinary conference and her treatment was radiotherapy followed by surgery ± reconstruction. We used a CT-based three-dimensional planning technique to give 40 Gy over 20 treatment sessions to the involved areas. Post treatment, a repeat CT was done at six weeks to reassess for disease progression or stabilization, followed by surgical excision. As of 31 October 2021, no evidence of recurrence is noted 48 months after treatment. Arriving at a definitive diagnosis with GorhamStout disease is challenging and a multidisciplinary team approach can help determine the treatment choice with best outcomes.
Gorham-Stout disease
;
disappearing bone disease
;
osteolysis, essential
3.Acro-osteolysis in a Filipino male with Vinyl chloride exposure: A case report
Karl Babe G. Tagomata, MD ; Therese Eileen B. Lladoc-Natividad, MD
Acta Medica Philippina 2023;57(7):67-72
Occupational acro-osteolysis pertains to bone resorption of the distal phalanges of the hands and feet among workers with vinyl chloride exposure. We report the case of a Filipino man with osteolysis of the distal phalanges of the hands initially considered to have systemic sclerosis. The patient had gradual shortening of the fingers, thickening of the skin over the extremities, and hypopigmented patches over a span of more than 20 years. His lower extremities presented with non-pitting edema, skin thickening, and neuropathy, without shortening of the digits. Difficulty of ambulation was apparent due to the development of feet inversion. Radiographic findings of the hands and feet included resorption of distal phalanges, erosive and sclerotic changes, and narrowed joint spaces. Other conditions considered were Hansen’s disease, skeletal tuberculosis, and diabetic neuropathic arthropathy, which were eventually ruled out. The final diagnosis was occupational acro-osteolysis secondary to vinyl chloride exposure. The patient underwent serial total contact casting of the bilateral lower extremities to relieve bipedal edema and to reposition the feet. This case emphasizes the significance of investigating a patient’s occupational history and highlights a rare sequela of exposure to a commonly used chemical agent in the manufacture of polyvinyl chloride products.
bone resorption
;
vinyl chloride
;
Filipino
4.Osteoimmunology research in rheumatoid arthritis: From single-cell omics approach.
Nan HU ; Jing WANG ; Bomiao JU ; Yuanyuan LI ; Ping FAN ; Xinxin JIN ; Xiaomin KANG ; Shufang WU
Chinese Medical Journal 2023;136(14):1642-1652
Cellular immune responses as well as generalized and periarticular bone loss are the key pathogenic features of rheumatoid arthritis (RA). Under the pathological conditions of RA, dysregulated inflammation and immune processes tightly interact with skeletal system, resulting in pathological bone damage via inhibition of bone formation or induction of bone resorption. Single-cell omics technologies are revolutionary tools in the field of modern biological research.They enable the display of the state and function of cells in various environments from a single-cell resolution, thus making it conducive to identify the dysregulated molecular mechanisms of bone destruction in RA as well as the discovery of potential therapeutic targets and biomarkers. Here, we summarize the latest findings of single-cell omics technologies in osteoimmunology research in RA. These results suggest that single-cell omics have made significant contributions to transcriptomics and dynamics of specific cells involved in bone remodeling, providing a new direction for our understanding of cellular heterogeneity in the study of osteoimmunology in RA.
Humans
;
Osteoclasts/physiology*
;
Arthritis, Rheumatoid/pathology*
;
Inflammation/pathology*
;
Bone and Bones/pathology*
;
Bone Resorption/pathology*
5.Exercise regulates bone metabolism via microRNAs.
Yu YUAN ; Lin-Zhen RAO ; Shi-Hua ZHANG ; Yang XU ; Ting-Ting LI ; Jun ZOU ; Xi-Quan WENG
Acta Physiologica Sinica 2023;75(3):429-438
It has been well documented that exercise can improve bone metabolism, promote bone growth and development, and alleviate bone loss. MicroRNAs (miRNAs) are widely involved in the proliferation and differentiation of bone marrow mesenchymal stem cells, osteoblasts, osteoclasts and other bone tissue cells, and regulation of balance between bone formation and bone resorption by targeting osteogenic factors or bone resorption factors. Thus miRNAs play an important role in the regulation of bone metabolism. Recently, regulation of miRNAs are shown to be one of the ways by which exercise or mechanical stress promotes the positive balance of bone metabolism. Exercise induces changes of miRNAs expression in bone tissue and regulates the expression of related osteogenic factors or bone resorption factors, to further strengthen the osteogenic effect of exercise. This review summarizes relevant studies on the mechanism whereby exercise regulates bone metabolism via miRNAs, providing a theoretical basis for osteoporosis prevention and treatment with exercise.
Humans
;
MicroRNAs/metabolism*
;
Osteogenesis/genetics*
;
Cell Differentiation
;
Osteoblasts
;
Bone Resorption/metabolism*
6.Effect of dental follicles in minimally invasive open-eruption technique of labially impacted maxillary central incisors.
Jiayue HUANG ; Xian LIU ; Yan WANG ; Chongyun BAO
West China Journal of Stomatology 2023;41(2):197-202
OBJECTIVES:
To summarize the open-eruption technique of impacted anterior maxillary teeth, this study reports a technically improved operation on surgical exposure based on dental follicles and evaluates post-treatment periodontal health considering the effect of dental follicles.
METHODS:
Patients who underwent open-eruption technique with unilateral labially impacted maxillary central incisors were selected. The impacted teeth were assigned to the experimental group, and the contralateral unimpacted maxillary central incisors were assigned to the control group. In the surgical exposure, the new technique makes use of dental follicles to manage the soft tissue, so as to preserve soft tissue for better aesthetic results and healthier periodontal tissue. Tooth length, root length, alveolar bone loss, and alveolar bone thickness were recorded after the therapy.
RESULTS:
A total of 17 patients with unilateral maxillary central incisor impaction were successfully treated. The tooth length and root length of the two groups showed a statistically significant difference between the impacted and homonym teeth, with a shorter length in the impacted tooth (P<0.05). More labial alveolar bone loss was found in the experimental group compared with that in the control group (P<0.05). The outcomes of the cementoenamel junction width, pa- latal alveolar bone loss, and alveolar bone thickness did not indicate statistical significance between the experimental and control groups (P>0.05).
CONCLUSIONS
In the surgical exposure, the new technique uses dental follicles to manage the soft tissue and preserve it for better aesthetic results and healthier periodontal tissues.
Humans
;
Tooth, Impacted/surgery*
;
Incisor
;
Alveolar Bone Loss/diagnostic imaging*
;
Tooth Root
;
Dental Sac
;
Maxilla/surgery*
;
Esthetics, Dental
7.Clinical efficacy of simple taper retentive implants in immediate posterior dental implantation for 5-7 years.
Bihui REN ; Shuigen GUO ; Yehao XU ; Jieting DAI ; Hongwu WEI
West China Journal of Stomatology 2023;41(3):341-349
OBJECTIVES:
This study aimed to evaluate the long-term clinical efficacy of simple taper retentive implants in the posterior dental area after immediate implantation for 5-7 years.
METHODS:
Selected from January 2015 to December 2017 in the Fourth Affiliated Hospital of Nanchang University dental clinic line tooth area immediately after the implant prosthesis, a total of 38 patients, 53 implants, were deep into (bone under 2 mm or higher) and the upper structure was repaired. In addition, after the completion of tracking observation of 60-90 months, the implant surrounding bone health was recorded and analyzed.
RESULTS:
After 5-7 years of follow-up, 1 of the 53 implants failed to fall out, and the implant retention rate was 98.1%. The amount of bone resorption in the proximal and distal margins 5-7 years after implant restoration was (0.16±0.94) mm and (-0.01±1.29) mm, respectively, and the difference in bone height between the proximal and distal margins of the implant and the immediate post-restoration period was not statistically significant (P>0.05). No statistically significant differences were found in the effects of periodontitis, implant site inflammation, and smoking on peri-implant marginal bone resorption (P>0.05).
CONCLUSIONS
The single taper-retained implant broadens the indications for immediate implant placement in the posterior region, and its deep sub-osseous placement (≥2 mm below the bone) avoids to a certain extent the disturbance of the implant by external stimuli and the exposure of the cervical abutment of the implant, with the good long-term stability of the marginal bone around the implant.
Humans
;
Dental Implantation, Endosseous
;
Dental Implants
;
Immediate Dental Implant Loading
;
Follow-Up Studies
;
Dental Implants, Single-Tooth
;
Alveolar Bone Loss/surgery*
;
Treatment Outcome
;
Dental Prosthesis, Implant-Supported
;
Dental Restoration Failure
8.Research progress in biomechanics of Bristow-Latarjet procedure for anterior shoulder dislocation.
Shuhan ZHANG ; Min ZHANG ; Zhenxing SHAO ; Guoqing CUI
Chinese Journal of Reparative and Reconstructive Surgery 2023;37(5):518-525
OBJECTIVE:
To review the research progress of the biomechanical study of the Bristow-Latarjet procedure for anterior shoulder dislocation.
METHODS:
The related biomechanical literature of Bristow-Latarjet procedure for anterior shoulder dislocation was extensively reviewed and summarized.
RESULTS:
The current literature suggests that when performing Bristow-Latarjet procedure, care should be taken to fix the bone block edge flush with the glenoid in the sagittal plane in the direction where the rupture of the joint capsule occurs. If traditional screw fixation is used, a double-cortical screw fixation should be applied, while details such as screw material have less influence on the biomechanical characteristics. Cortical button fixation is slightly inferior to screws in terms of biomechanical performance. The most frequent site of postoperative bone resorption is the proximal-medial part of the bone block, and the cause of bone resorption at this site may be related to the stress shielding caused by the screw.
CONCLUSION
There is no detailed standardized guidance for bone block fixation. The optimal clinical treatment plan for different degrees of injury, the factors influencing postoperative bone healing and remodeling, and the postoperative osteoarticular surface pressure still need to be further clarified by high-quality biomechanical studies.
Humans
;
Shoulder Dislocation/surgery*
;
Shoulder Joint/surgery*
;
Biomechanical Phenomena
;
Joint Instability/surgery*
;
Bone Resorption
;
Arthroscopy/methods*
9.Research progress of bone graft resorption after Latarjet procedure for treatment of recurrent anterior shoulder dislocation.
Chinese Journal of Reparative and Reconstructive Surgery 2023;37(5):526-532
OBJECTIVE:
To review the research progress of bone graft resorption after Latarjet procedure for the treatment of recurrent anterior shoulder dislocation, and provide a guide for further research on bone graft resorption.
METHODS:
The relevant literature in recent years was extensively reviewed. The pathogenesis, classification, risk factors, clinical function impact, and management of bone graft resorption after Latarjet procedure for the treatment of recurrent anterior shoulder dislocation were summarized.
RESULTS:
Bone graft resorption is the common complication after Latarjet procedure for the treatment of recurrent anterior shoulder dislocation. Stress shielding and poor blood supply may contribute to the occurrence of bone graft resorption. The absence of significant preoperative glenoid bone loss, open procedure, earlier graft healing may to be the risk factors for bone graft resorption. Various assessment methods and classification systems are used to evaluate the region and severity of bone graft resorption. Partial resorption may be considered as a natural glenoid remodeling process after the surgery, but severe and complete resorption is proved to be one of the reasons for failed procedures and there is no effective measure to prevent it, except for accepting revision surgery.
CONCLUSION
The pathogenesis, risk factors, clinical function impact of bone graft resorption after Latarjet procedure for the treatment of recurrent anterior shoulder dislocation has not been fully elucidated and there is a lack of effective management strategies, so further clinical and basic researches are needed.
Humans
;
Shoulder Joint/surgery*
;
Shoulder Dislocation/surgery*
;
Joint Instability/surgery*
;
Bone Resorption/pathology*
;
Bone Transplantation
;
Recurrence
10.Regulatory function and mechanism of autophagy on osteoclast.
Jian-Sen MIAO ; Xiang-Yang WANG ; Hai-Ming JIN
China Journal of Orthopaedics and Traumatology 2023;36(4):357-363
Osteoclast (OC) is multinucleated, bone-resorbing cells originated from monocyte/macrophage lineage of cells, excessive production and abnormal activation of which could lead to many bone metabolic diseases, such as osteoporosis, osteoarthritis, etc. Autophagy, as a highly conserved catabolic process in eukaryotic cells, which plays an important role in maintaining cell homeostasis, stress damage repair, proliferation and differentiation. Recent studies have found that autophagy was also involved in the regulation of osteoclast generation and bone resorption. On the one hand, autophagy could be induced and activated by various factors in osteocalsts, such as nutrient deficiency, hypoxia, receptor activator of nuclear factor(NF)-κB ligand(RANKL), inflammatory factors, wear particles, microgravity environment, etc, different inducible factors, such as RANKL, inflammatory factors, wear particles, could interact with each other and work together. On the other hand, activated autophagy is involved in regulating various stages of osteoclast differentiation and maturation, autophagy could promote proliferation of osteoclasts, inhibiting apoptosis, and promoting differentiation, migration and bone resorption of osteoclast. The classical autophagy signaling pathway mediated by mammalian target of rapamycin complex 1(mTORC1) is currently a focus of research, and it could be regulated by upstream signalings such as phosphatidylinositol 3 kinase(PI-3K)/protein kinase B (PKB), AMP-activated protein kinase(AMPK). However, the paper found that mTORC1-mediated autophagy may play a bidirectional role in regulating differentiation and function of osteoclasts, and its underlying mechanism needs to be further ciarified. Integrin αvβ3 and Rab protein families are important targets for autophagy to play a role in osteoclast migration and bone resorption, respectively. In view of important role of osteoclast in the occurrence of various bone diseases, it is of great significance to elucidate the role of autophagy on osteoclast and its mechanism for the treatment of various bone diseases. The autophagy pathway could be used as a new therapeutic target for the treatment of clinical bone diseases such as osteoporosis.
Humans
;
Osteoclasts
;
Bone Resorption/metabolism*
;
Cell Differentiation
;
NF-kappa B/metabolism*
;
Autophagy
;
Osteoporosis
;
Mechanistic Target of Rapamycin Complex 1/metabolism*
;
RANK Ligand/metabolism*


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