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Osteochondritis dissecans of the medial humeral trochlea in a competitive tennis player: A rare case and surgical insight 一例网球选手肱骨内侧滑车夹层性骨软骨炎:一例罕见病例及外科观察
Pub Date : 2026-06-01 Epub Date: 2026-01-12 DOI: 10.1016/j.joscr.2025.12.001
Akira Kinoda , Kazuki Sato , Yuichi Yamada , Yoshihiro Maruko , Takuji Iwamoto , Noboru Matsumura , Taku Suzuki , Yasuhiro Kiyota , Masato Yoshida

Background

Osteochondritis dissecans of the medial humeral trochlea is exceptionally rare, with no prior reports in tennis players.

Case presentation

A 14-year-old elite female tennis athlete presented with 6 months of medial left elbow pain exacerbated by serving. Examination revealed tenderness over the humeroulnar joint and a mild extension deficit. Computed tomography identified a loose osteochondral fragment posteromedial to the trochlea. Fat-suppressed T2-weighted magnetic resonance imaging demonstrated a hypointense line between the parent bone and fragment, leading to a diagnosis of osteochondritis dissecans. Surgery exposed and reattached the fragment using two 2.5 mm bone pegs. Radiographic union was achieved within 4 months, allowing a gradual return to serving. Six months after the surgery, she resumed unrestricted play.

Conclusion

We performed bone-peg grafting for medial trochlear osteochondritis dissecans in a tennis player's elbow and achieved favorable outcomes.
背景:肱骨内侧滑车夹层性软骨炎极为罕见,在网球运动员中尚无报道。病例介绍一名14岁的优秀女网球运动员表现为6个月的左肘内侧疼痛,因发球而加剧。检查显示肱骨尺关节压痛和轻度伸展缺陷。计算机断层扫描发现滑车后内侧有一疏松的骨软骨碎片。脂肪抑制的t2加权磁共振成像显示母骨和碎片之间的低信号线,导致诊断为剥离性骨软骨炎。手术使用两个2.5毫米骨钉暴露并重新连接碎片。4个月内实现放射愈合,允许逐渐恢复服务。手术六个月后,她恢复了不受限制的比赛。结论采用骨钉植入术治疗1例网球运动员肘部内侧滑车夹层骨软骨炎,效果良好。
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引用次数: 0
A case of focused extracorporeal shock wave therapy in a person with plantar fasciitis and hemophilia A 聚焦体外冲击波治疗足底筋膜炎合并A型血友病1例
Pub Date : 2026-06-01 Epub Date: 2026-01-14 DOI: 10.1016/j.joscr.2026.01.002
Takeshi Seki , Toshinori Masaoka , Toshiyuki Tateiwa , Ryui Miyashita , Yushi Chikasawa , Tsunehito Ishida , Kagehiro Amano , Ei Kinai , Kengo Yamamoto

Background

People with hemophilia often experience chronic musculoskeletal pain, but invasive treatments are avoided due to bleeding risk. Extracorporeal shock wave therapy (ESWT) is widely used for refractory tendinopathies, yet its application in hemophilia is rarely reported.

Case presentation

A 28-year-old man with severe hemophilia A and bilateral plantar fasciitis unresponsive to conservative therapy underwent focused ESWT after factor VIII replacement. Pain markedly improved after the first session and remained lower at 12 months without hemorrhagic complications.

Conclusion

Low-to medium-energy ESWT for plantar fasciitis can be safely performed in people with hemophilia when adequate coagulation factor activity is maintained.
血友病患者经常经历慢性肌肉骨骼疼痛,但由于出血风险,侵入性治疗是避免的。体外冲击波疗法(ESWT)广泛应用于难治性肌腱病,但其在血友病中的应用鲜有报道。病例介绍:一名28岁男性,患有严重的A型血友病和双侧足底筋膜炎,对保守治疗无反应。疼痛在第一次治疗后明显改善,并在12个月时保持较低,无出血性并发症。结论血友病患者在维持足够凝血因子活性的情况下,低至中能量ESWT治疗足底筋膜炎是安全的。
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引用次数: 0
A rare case of spinal epidural lipomatosis secondary to pituitary Cushing disease presenting with progressive paraparesis 一个罕见的脊髓硬膜外脂肪瘤病继发于垂体库欣病,表现为进行性麻痹
Pub Date : 2026-06-01 Epub Date: 2026-02-13 DOI: 10.1016/j.joscr.2026.02.001
Masahiro Hirahata, Muneyoshi Fujita, Tomoaki Kitagawa, Hirotaka Kawano

Background

Spinal epidural lipomatosis (SEL) is a rare condition characterized by excessive epidural fat accumulation. Although most cases are secondary to long-term corticosteroid use, SEL due to Cushing disease (CD) is extremely uncommon.

Case presentation

A 62-year-old woman presented with progressive paraparesis due to SEL associated with thoracic kyphosis from osteoporotic vertebral fractures. Emergency decompression was performed, followed by endocrine evaluation that revealed pituitary CD. The patient regained full neurological function after surgery.

Conclusions

This case represents the third reported instance of SEL caused by pituitary CD requiring spinal surgery. Early recognition and prompt decompression are essential for recovery.
脊髓硬膜外脂肪沉积症(SEL)是一种罕见的疾病,其特征是过度的硬膜外脂肪堆积。虽然大多数病例继发于长期使用皮质类固醇,但库欣病(CD)引起的SEL极为罕见。病例介绍:一名62岁女性因骨质疏松性椎体骨折导致的SEL伴胸椎后凸而出现进行性麻痹。手术后进行了紧急减压,随后进行了内分泌评估,发现垂体CD。手术后患者恢复了完全的神经功能。结论该病例是第三例报道的由垂体CD引起的SEL,需要脊柱手术。早期发现并及时减压对恢复至关重要。
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引用次数: 0
Subcutaneous flexor pollicis longus tendon rupture caused by scapho-trapezio-trapezoidal osteoarthritis: A case report of wide-awake surgery 舟状-梯形-梯形骨关节炎致拇长屈肌腱断裂:全清醒手术一例
Pub Date : 2026-06-01 Epub Date: 2026-03-02 DOI: 10.1016/j.joscr.2026.02.004
Seishiro Matsumoto , Takuya Uemura , Yusuke Miyashima , Sadahiko Konishi , Hidetomi Terai

Background

Subcutaneous flexor pollicis longus (FPL) tendon rupture caused by scapho-trapezio-trapezoidal (STT) osteoarthritis is rare. Wide-awake surgery under local anesthesia has become widespread in tendon-related hand surgeries because surgeons can precisely evaluate a patient's voluntary active finger motion.

Case presentation

Herein, we present the first case of an 86-year-old woman with FPL tendon rupture secondary to asymptomatic STT osteoarthritis treated with wide-awake tendon grafting. Intraoperatively, the FPL tendon completely ruptured owing to friction from the osteophyte in the STT joint. After resection of the bony spurs, the ruptured FPL tendon was reconstructed with grafting of the palmaris longus tendon, and the optimal tension was determined during active thumb movement. The patient was able to actively flex the interphalangeal joint of the thumb 12 months postoperatively.

Conclusion

This case highlights that asymptomatic STT osteoarthritis can potentially cause FPL tendon rupture through bony spurs and that wide-awake surgery offers advantages for precise intraoperative evaluation and repair.
背景:由舟状-梯形-梯形(STT)骨关节炎引起的拇长屈肌(FPL)肌腱断裂是罕见的。局部麻醉下的完全清醒手术在与肌腱相关的手部手术中已经广泛应用,因为外科医生可以精确地评估患者的主动手指运动。病例介绍在此,我们报告了一例86岁的女性,她的FPL肌腱断裂继发于无症状的STT骨关节炎,采用宽醒肌腱移植治疗。术中,由于STT关节骨赘的摩擦,FPL肌腱完全断裂。切除骨刺后,用掌长肌腱移植重建断裂的FPL肌腱,并在拇指活动时确定最佳张力。患者术后12个月拇指间关节活动自如。结论本病例提示无症状STT骨关节炎可通过骨刺导致FPL肌腱断裂,全清醒手术可提供精确的术中评估和修复优势。
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引用次数: 0
Long-term outcomes of bone marrow blood transfusion combined with low-intensity pulsed ultrasound and external fixation in early-stage Preiser’s disease: A 10-year follow-up case report 骨髓输血联合低强度脉冲超声和外固定治疗早期Preiser病的长期疗效:10年随访病例报告
Pub Date : 2026-03-01 Epub Date: 2025-06-14 DOI: 10.1016/j.joscr.2025.05.003
Akira Ikumi , Takeshi Ogawa , Yuki Hara , Yuichi Yoshii , Naoyuki Ochiai , Masashi Yamazaki

Background

Preiser’s disease, or avascular necrosis of the scaphoid, is a relatively rare condition, and its optimal treatment remains uncertain, especially in the early stages.

Case presentation

We described a case of a 37-year-old woman with stage I Preiser’s disease, who underwent bone marrow blood transfusion combined with low-intensity pulsed ultrasound and external fixation. Over a 10-year follow-up period, her wrist function was preserved, and disease progression was prevented.

Conclusion

This case highlights the long-term outcomes of bone marrow blood transfusion and low-intensity pulsed ultrasound for early-stage Preiser’s disease. While the combination of these treatments appeared effective, further studies are needed to determine the individual contributions of each modality.
preiser病或舟状骨缺血性坏死是一种相对罕见的疾病,其最佳治疗方法仍不确定,特别是在早期阶段。我们报告了一例37岁的I期Preiser病女性患者,她接受了骨髓输血联合低强度脉冲超声和外固定。在10年的随访期间,她的手腕功能得以保留,疾病也没有进展。结论本病例强调了骨髓输血和低强度脉冲超声治疗早期Preiser病的远期疗效。虽然这些治疗的组合似乎是有效的,但需要进一步的研究来确定每种方式的个人贡献。
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引用次数: 0
Successful treatment of pediatric scaphoid nonunion with a vascularized bone graft: A report of four cases 带血管骨移植成功治疗儿童舟状骨不连:附4例报告
Pub Date : 2026-03-01 Epub Date: 2025-10-21 DOI: 10.1016/j.joscr.2025.10.001
Ryosuke Koshio , Hirotaka Akezuma , Ichiro Okano , Keikichi Kawasaki , Yoshifumi Kudo

Background

Scaphoid fractures are rare in children, and the occurrence of nonunion is even more uncommon. While vascularized bone grafting (VBG) has been shown to be effective in adult scaphoid nonunion, the commonly used distal radius VBG carries a risk of growth plate injury in pediatric patients. This study reports the outcomes of VBG from the second metacarpal base for scaphoid nonunion in teenagers with open epiphysial line.

Case presentation

Since 2001, we treated four pediatric patients (≤14 years) with scaphoid nonunion using second metacarpal VBG. Grafts were harvested from the second metacarpal base, preserving the dorsal metacarpal artery, and fixed with headless screws and K-wires. Bone union was achieved in all cases. Although one case showed incomplete correction of DISI deformity, all patients had favorable outcomes in terms of range of motion and Mayo Wrist Scores.

Conclusions

Second metacarpal VBG achieved bone union and good function in pediatric scaphoid nonunion. Nonetheless, the potential limitations in correcting DISI deformity should be considered.
背景:儿童舟状骨骨折是罕见的,不愈合的发生更是罕见。虽然血管化骨移植(VBG)已被证明对成人舟骨不愈合有效,但在儿童患者中,常用的桡骨远端VBG存在生长板损伤的风险。本研究报告了从第二掌骨基底行VBG治疗骨骺线开放的青少年舟状骨不连的结果。自2001年以来,我们使用第二掌骨VBG治疗了4例儿童(≤14岁)舟状骨不连。从第二掌骨基部取出移植物,保留掌骨背动脉,用无头螺钉和k针固定。所有病例均实现骨愈合。尽管一例DISI畸形矫正不完全,但所有患者在活动范围和Mayo腕关节评分方面均有良好的结果。结论第二掌骨VBG治疗小儿舟状骨不连,骨愈合良好,功能良好。尽管如此,在纠正DISI畸形时仍应考虑到潜在的局限性。
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引用次数: 0
Symptomatic os vesalianum pedis triggered by gouty tophus: A case report 痛风性痛风引起的症状性足疱1例
Pub Date : 2026-03-01 Epub Date: 2025-09-01 DOI: 10.1016/j.joscr.2025.08.001
Yoshimasa Taniguchi , Tomonobu Hagio , Tetsuro Ishimatsu , Yuki Sugino , Seiya Tomonaga , Ryo Fukagawa , Ichiro Yoshimura , Takuaki Yamamoto

Background

Os Vesalianum Pedis (OVP) is an accessory bone at the base of the fifth metatarsal. While rare, symptomatic cases requiring surgery may arise following sports injuries or trauma. We present a unique case of symptomatic OVP induced by gouty tophi.

Case report

A 25-year-old male with recurrent gout attacks had discontinued medication without supervision. One month before presentation, he experienced worsening foot pain that persisted despite resuming medication, prompting a visit to our department. Physical examination revealed swelling and tenderness in both lateral feet. Blood tests showed elevated uric acid and C-reactive protein. Imaging studies demonstrated osteolysis, marginal sclerosis, and erosion at the fifth metatarsal base. Surgical resection of the OVP relieved the pain completely.

Conclusion

This is the first reported case of OVP induced by gouty tophi. Prolonged hyperuricemia likely promoted tophus formation, impairing mobility between the fifth metatarsal and OVP, resulting in pain.
足泡骨(Vesalianum Pedis, OVP)是位于第五跖骨底部的副骨。虽然罕见,但在运动损伤或创伤后可能会出现需要手术的症状。我们提出一个独特的情况下,症状性OVP引起痛风痛风痛风。病例报告:一名25岁男性反复发作痛风,在没有监护的情况下停药。在就诊前一个月,他经历了不断恶化的足部疼痛,尽管恢复了药物治疗,但疼痛仍持续存在,促使他来我科就诊。体格检查发现双侧足肿胀和压痛。血液检查显示尿酸和c反应蛋白升高影像学检查显示第五跖骨基底骨溶解、边缘硬化和糜烂。手术切除OVP完全减轻了疼痛。结论本病例为首次报道的痛风痛风痛风致OVP病例。长时间的高尿酸血症可能促进了结石的形成,损害了第五跖骨和OVP之间的活动能力,导致疼痛。
{"title":"Symptomatic os vesalianum pedis triggered by gouty tophus: A case report","authors":"Yoshimasa Taniguchi ,&nbsp;Tomonobu Hagio ,&nbsp;Tetsuro Ishimatsu ,&nbsp;Yuki Sugino ,&nbsp;Seiya Tomonaga ,&nbsp;Ryo Fukagawa ,&nbsp;Ichiro Yoshimura ,&nbsp;Takuaki Yamamoto","doi":"10.1016/j.joscr.2025.08.001","DOIUrl":"10.1016/j.joscr.2025.08.001","url":null,"abstract":"<div><h3>Background</h3><div>Os Vesalianum Pedis (OVP) is an accessory bone at the base of the fifth metatarsal. While rare, symptomatic cases requiring surgery may arise following sports injuries or trauma. We present a unique case of symptomatic OVP induced by gouty tophi.</div></div><div><h3>Case report</h3><div>A 25-year-old male with recurrent gout attacks had discontinued medication without supervision. One month before presentation, he experienced worsening foot pain that persisted despite resuming medication, prompting a visit to our department. Physical examination revealed swelling and tenderness in both lateral feet. Blood tests showed elevated uric acid and C-reactive protein. Imaging studies demonstrated osteolysis, marginal sclerosis, and erosion at the fifth metatarsal base. Surgical resection of the OVP relieved the pain completely.</div></div><div><h3>Conclusion</h3><div>This is the first reported case of OVP induced by gouty tophi. Prolonged hyperuricemia likely promoted tophus formation, impairing mobility between the fifth metatarsal and OVP, resulting in pain.</div></div>","PeriodicalId":100743,"journal":{"name":"JOS Case Reports","volume":"5 1","pages":"Pages 16-20"},"PeriodicalIF":0.0,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147414349","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Epithelioid angiosarcoma arising within a schwannoma of the back: A case report 发生于背部神经鞘瘤的上皮样血管肉瘤1例报告
Pub Date : 2026-03-01 Epub Date: 2025-05-31 DOI: 10.1016/j.joscr.2025.05.002
Yuta Watanabe , Naofumi Asano , Tsuyoshi Mizuma , Satoshi Kamio , Sayaka Yamaguchi , Tomoaki Mori , Hajime Okita , Robert Nakayama , Masaya Nakamura

Background

Schwannomas are benign tumors that commonly occur in peripheral nerves, and their malignant transformation is extremely rare.

Case presentation

A 53-year-old man was aware of a mass in his right back for eight years. As the mass increased in size and became painful, he was referred to our hospital. Magnetic resonance imaging (MRI) revealed a 3 cm mass within the muscle layer. The tumor margins were regular, but it was heterogeneously contrasted. A needle biopsy suggested schwannoma, and we performed a resection. However, pathologically, the tumor was found to be composed of a benign schwannoma, an epithelioid angiosarcoma, and diagnosed an epithelioid angiosarcoma arising from a benign schwannoma. We performed an additional wide resection, and there has been no recurrence for four years after surgery.

Conclusions

We present a rare case of epithelioid angiosarcoma that developed within a schwannoma. If progressive symptoms are present, malignant transformation should be considered.
神经鞘瘤是一种常见于周围神经的良性肿瘤,其恶性转化极为罕见。一名53岁男子8年来一直意识到他的右背部有肿块。由于肿块越来越大,变得疼痛,他被转介到我们医院。磁共振成像(MRI)显示肌肉层内有一个3厘米的肿块。肿瘤边缘规则,但对比不均匀。穿刺活检显示是神经鞘瘤,我们进行了切除。然而,病理发现肿瘤由良性神经鞘瘤和上皮样血管肉瘤组成,诊断为良性神经鞘瘤引起的上皮样血管肉瘤。我们进行了额外的大面积切除,手术后四年没有复发。结论我们报告一例罕见的上皮样血管肉瘤发生在神经鞘瘤内。如果出现进展性症状,应考虑恶性转化。
{"title":"Epithelioid angiosarcoma arising within a schwannoma of the back: A case report","authors":"Yuta Watanabe ,&nbsp;Naofumi Asano ,&nbsp;Tsuyoshi Mizuma ,&nbsp;Satoshi Kamio ,&nbsp;Sayaka Yamaguchi ,&nbsp;Tomoaki Mori ,&nbsp;Hajime Okita ,&nbsp;Robert Nakayama ,&nbsp;Masaya Nakamura","doi":"10.1016/j.joscr.2025.05.002","DOIUrl":"10.1016/j.joscr.2025.05.002","url":null,"abstract":"<div><h3>Background</h3><div>Schwannomas are benign tumors that commonly occur in peripheral nerves, and their malignant transformation is extremely rare.</div></div><div><h3>Case presentation</h3><div>A 53-year-old man was aware of a mass in his right back for eight years. As the mass increased in size and became painful, he was referred to our hospital. Magnetic resonance imaging (MRI) revealed a 3 cm mass within the muscle layer. The tumor margins were regular, but it was heterogeneously contrasted. A needle biopsy suggested schwannoma, and we performed a resection. However, pathologically, the tumor was found to be composed of a benign schwannoma, an epithelioid angiosarcoma, and diagnosed an epithelioid angiosarcoma arising from a benign schwannoma. We performed an additional wide resection, and there has been no recurrence for four years after surgery.</div></div><div><h3>Conclusions</h3><div>We present a rare case of epithelioid angiosarcoma that developed within a schwannoma. If progressive symptoms are present, malignant transformation should be considered.</div></div>","PeriodicalId":100743,"journal":{"name":"JOS Case Reports","volume":"5 1","pages":"Pages 1-6"},"PeriodicalIF":0.0,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147414390","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Bilateral tibial stress fracture and stress reaction: A report of 14 cases 双侧胫骨应力性骨折及应力反应:附14例报告
Pub Date : 2026-03-01 Epub Date: 2025-07-24 DOI: 10.1016/j.joscr.2025.06.001
Hiroaki Nishimoto , Junji Ohnishi , Lars Engebretsen , Tetsuo Yamaguchi

Background

Several studies have reported that stress fractures (SFs) and stress reactions (SRs) rarely occur simultaneously in both tibiae.

Case presentation

This study of 14 athletes (11 males and 3 females) diagnosed with bilateral tibial SFs or SRs demonstrated that SFs and SRs occurred most frequently in July and September (21 % each). The mean age was 17.0 ± 4.0 and 15.6 ± 2.1 years in the SF and SR groups, respectively. SFs and SRs showed a high incidence in the mid-tibia, with minimal side-to-side difference.

Conclusions

Bilateral tibial SFs and SRs were more common in young male athletes, especially in summer. These injuries commonly occurred in the mid-tibia, with minimal side-to-side differences in fracture height. Anterior SFs were treated surgically, whereas posteromedial SFs and SRs were treated conservatively, allowing a return to competition.
一些研究报道了应力性骨折(SFs)和应力反应(SRs)很少同时发生在双胫骨。本研究对14名被诊断为双侧胫骨SFs或SRs的运动员(11名男性和3名女性)进行了研究,结果表明SFs和SRs最常发生在7月和9月(各占21%)。SF组和SR组的平均年龄分别为17.0±4.0岁和15.6±2.1岁。SFs和SRs在胫骨中部的发病率很高,两侧差异很小。结论青年男性运动员双侧胫骨SFs和SRs多发,以夏季运动员居多。这些损伤通常发生在胫骨中部,骨折高度的侧对侧差异极小。前路SFs采用手术治疗,而后内侧SFs和SRs采用保守治疗,允许恢复竞争。
{"title":"Bilateral tibial stress fracture and stress reaction: A report of 14 cases","authors":"Hiroaki Nishimoto ,&nbsp;Junji Ohnishi ,&nbsp;Lars Engebretsen ,&nbsp;Tetsuo Yamaguchi","doi":"10.1016/j.joscr.2025.06.001","DOIUrl":"10.1016/j.joscr.2025.06.001","url":null,"abstract":"<div><h3>Background</h3><div>Several studies have reported that stress fractures (SFs) and stress reactions (SRs) rarely occur simultaneously in both tibiae.</div></div><div><h3>Case presentation</h3><div>This study of 14 athletes (11 males and 3 females) diagnosed with bilateral tibial SFs or SRs demonstrated that SFs and SRs occurred most frequently in July and September (21 % each). The mean age was 17.0 ± 4.0 and 15.6 ± 2.1 years in the SF and SR groups, respectively. SFs and SRs showed a high incidence in the mid-tibia, with minimal side-to-side difference.</div></div><div><h3>Conclusions</h3><div>Bilateral tibial SFs and SRs were more common in young male athletes, especially in summer. These injuries commonly occurred in the mid-tibia, with minimal side-to-side differences in fracture height. Anterior SFs were treated surgically, whereas posteromedial SFs and SRs were treated conservatively, allowing a return to competition.</div></div>","PeriodicalId":100743,"journal":{"name":"JOS Case Reports","volume":"5 1","pages":"Pages 12-15"},"PeriodicalIF":0.0,"publicationDate":"2026-03-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"147414347","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
Kaposiform hemangioendothelioma diagnosed following elbow painful contracture in an adolescent patient: A case report 卡波西样血管内皮瘤诊断后肘部疼痛挛缩的青少年患者:1例报告
Pub Date : 2025-12-01 Epub Date: 2025-03-17 DOI: 10.1016/j.joscr.2025.03.001
Yoko Tanabe , Robert Nakayama , Kayoko Uno , Yutaka Kurebayashi , Yosuke Yamamoto , Satoshi Kamio , Sayaka Yamaguchi , Tomoaki Mori , Naofumi Asano , Yohei Masugi , Taiki Nozaki , Noriko Aramaki , Masaya Nakamura

Background

Kaposiform hemangioendothelioma is a rare, locally invasive vascular tumor of intermediate malignancy that most frequently involves the extremities. Most cases manifest in infancy and are associated with life-threatening consumptive coagulopathy, known as the Kasabach–Merritt phenomenon, acute pain, and functional limitations.

Case presentation

We present an extremely rare case of kaposiform hemangioendothelioma in a 15-year-old girl who had had no remarkable symptoms other than skin erythema until she developed severe pain and elbow contracture during adolescence. Although marginal tumor resection necessitated concurrent triceps tendon resection, her symptoms resolved without recurrence over a 2-year follow-up.

Conclusions

This case highlights that probably congenital kaposiform hemangioendothelioma may not cause significant symptoms until adolescence and the importance of resection for symptomatic resectable lesions.
背景:卡波西样血管内皮瘤是一种罕见的、局部侵袭性的中度恶性血管肿瘤,最常累及四肢。大多数病例表现在婴儿期,并伴有危及生命的消耗性凝血功能障碍,即Kasabach-Merritt现象,急性疼痛和功能限制。病例介绍:我们报告一例极为罕见的卡样血管内皮瘤病例,患者为15岁女孩,除皮肤红斑外无其他显著症状,直到青春期出现严重疼痛和肘部挛缩。虽然边缘肿瘤切除需要同时切除三头肌肌腱,但在2年的随访中,她的症状消失,无复发。结论本病例强调先天性卡斑样血管内皮瘤可能在青春期之前不会引起明显的症状,以及对有症状的可切除病变进行切除的重要性。
{"title":"Kaposiform hemangioendothelioma diagnosed following elbow painful contracture in an adolescent patient: A case report","authors":"Yoko Tanabe ,&nbsp;Robert Nakayama ,&nbsp;Kayoko Uno ,&nbsp;Yutaka Kurebayashi ,&nbsp;Yosuke Yamamoto ,&nbsp;Satoshi Kamio ,&nbsp;Sayaka Yamaguchi ,&nbsp;Tomoaki Mori ,&nbsp;Naofumi Asano ,&nbsp;Yohei Masugi ,&nbsp;Taiki Nozaki ,&nbsp;Noriko Aramaki ,&nbsp;Masaya Nakamura","doi":"10.1016/j.joscr.2025.03.001","DOIUrl":"10.1016/j.joscr.2025.03.001","url":null,"abstract":"<div><h3>Background</h3><div>Kaposiform hemangioendothelioma is a rare, locally invasive vascular tumor of intermediate malignancy that most frequently involves the extremities. Most cases manifest in infancy and are associated with life-threatening consumptive coagulopathy, known as the Kasabach–Merritt phenomenon, acute pain, and functional limitations.</div></div><div><h3>Case presentation</h3><div>We present an extremely rare case of kaposiform hemangioendothelioma in a 15-year-old girl who had had no remarkable symptoms other than skin erythema until she developed severe pain and elbow contracture during adolescence. Although marginal tumor resection necessitated concurrent triceps tendon resection, her symptoms resolved without recurrence over a 2-year follow-up.</div></div><div><h3>Conclusions</h3><div>This case highlights that probably congenital kaposiform hemangioendothelioma may not cause significant symptoms until adolescence and the importance of resection for symptomatic resectable lesions.</div></div>","PeriodicalId":100743,"journal":{"name":"JOS Case Reports","volume":"4 4","pages":"Pages 97-101"},"PeriodicalIF":0.0,"publicationDate":"2025-12-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"145610538","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
引用次数: 0
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JOS Case Reports
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