Republication of "Gelatinous Transformation of Bone Marrow in the Calcaneus, Diagnosed by Open Bone Biopsy in a Patient With Anorexia Nervosa: A Case Report".

Gelatinous transformation of bone marrow (GTBM) is a complication of various diseases, one of which is anorexia nervosa (AN). We describe a rare case of a 20-year-old man who presented to our clinic with a 3-month history of heel pain without trauma. At presentation, he was noted to have a low body mass index (BMI) of 16.2 kg/m2 and pancytopenia. On magnetic resonance imaging, the left calcaneus showed low intensity on T1-weighted and high intensity on T2-weighted images. Open biopsy was done because we suspected that the lesion was either a lymphoproliferative tumor or a trabecular-type bone metastatic tumor. However, tissue histology of bone samples showed atrophy of fat cells with deposition of gelatinous material and a decreased hematopoietic cell population. Therefore, we made a diagnosis of GTBM, most likely caused by AN. We started treatment with nutritional support, and 6 months later, the hematological parameters returned to normal and BMI improved to 19.4 kg/m2. He was able to return to work and had no left heel pain. This case indicates that foot and ankle surgeons need to be aware of this rare pathology, although it might be difficult to diagnose without biopsy. To our knowledge, very few descriptions of GTBM in the calcaneus have been reported to date.

Combination chemotherapy of doxorubicin and ifosfamide with proton beam therapy for myoepithelial carcinoma originating in the paraspinal region: A case report and literature review.

Soft tissue myoepithelial carcinoma (MEC) is an extremely rare mesenchymal tumor that has a poor prognosis unless complete surgical resection is achieved. The present study reported a case of a 38-year-old woman with a tumor in the left paraspinal region at L2 to L3 with vertebral destruction. MEC was diagnosed based on molecular pathological examination of a biopsy specimen. Because curative surgery was expected to be difficult, a combination of chemotherapy with doxorubicin and ifosfamide and proton beam therapy as local therapy was performed, resulting in long-term survival for at least 7.8 years. To the best of our knowledge, this is the first case of soft tissue MEC for which classical cytotoxic chemotherapy and proton beam therapy were effective. Although surgical resection with negative margins is the mainstay of treatment for MEC, adequate doxorubicin-based systemic therapy and high-dose radiation therapy may be a feasible alternative in patients with unresectable or advanced MEC. Future studies on the relationship between molecular pathological features, including biomarkers, and the selection of therapeutic agents are warranted.

An anterolateral dislocated monteggia lesion with plastic lateral bowing of the ulna associated with ipsilateral epiphyseal fracture of the distal radius: a case report with 4 years of follow-up.

We encountered a rare presentation of anterolateral dislocation of the radial head with plastic lateral bowing of the ulna associated with ipsilateral epiphyseal fracture of the distal radius in a child. The patient was treated surgically and reached skeletal maturity 4 years later with no functional or growth deficiency.

A Rare Case of Acute-Onset Spastic Quadriparesis Caused by a Chondroma of the Cervical Spine.

Chondromas are benign cartilaginous tumors that occur very rarely in the spine. Moreover, chondromas of the extraskeletal origin are also very rare. In this case report, we describe an extremely rare case of chondroma arising from the ligamentum flavum in the cervical spine. A 67-year-old man presented to our clinic with acute-onset spastic quadriparesis. We performed emergent magnetic resonance imaging and found an epidural mass in the right ligamentum flavum at C4-C5. The acute-onset presentation was suspicious for intraligamentous hematoma in the ligamentum flavum at this level. We performed emergency decompression surgery and en bloc removal of the epidural mass with the right C4 and C5 lamina. The lesion had the appearance of cartilaginous tissue rather than hematoma. Based on the histological investigation, the final diagnosis was intraligamentous chondroma of the cervical spine. The quadriparesis improved postoperatively, as did the results of manual muscle testing in the affected area, and he was able to resume walking independently with a cane. At the one-year follow-up, the manual muscle testing results were almost normal. Surgeons should keep in mind the possibility of benign tumors including chondroma of the cervical spine when a patient presents with acute-onset quadriparesis.

Intimal sarcoma arising from the common iliac artery presenting with artery occlusion : a case report and literature review.

We present a rare case of intimal sarcoma arising from the common iliac artery in an 82-year-old man who presented with intermittent claudication. He had undergone endovascular therapy with self-expanding stents to both iliac arteries that had occluded soon after placement. After salvage bypass grafting, a diagnosis of intimal sarcoma with angiosarcoma phenotype from the iliac artery was made. Further bypass graft surgery relieved symptoms temporarily. However, the tumor progressed and the left limb became ischemic. The chemotherapy of eribulin did not prevent tumor progression. The patient died of the disease 20 months after the first surgery. J. Med. Invest. 66 : 205-208, February, 2019.

Identification of abnormalities in the lumbar nerve tract using diffusion-weighted magnetic resonance neurography.

INTRODUCTION: Abnormalities of the lumbar nerve tract caused by congenital variations or high nerve root take-off angles are difficult to visualize. Diffusion-weighted magnetic resonance neurography (DW-MRN) has recently been introduced for imaging of the lumbosacral region. The aims of this study were to identify lumbar nerve tract abnormalities caused by congenital variation or a high nerve root take-off angle using DW-MRN and to assess the diagnostic value of this imaging modality. METHODS: A total of 573 magnetic resonance images from 575 patients (261 men, 314 women; mean age 58.5 years) with low back/leg pain were retrospectively analyzed. We classified congenital variations in the lumbar nerve roots using the Neidre and MacNab criteria and investigated nerve roots with a take-off angle of 60° or more. RESULTS AND DISCUSSION: Congenital variations were identified in 8 patients (9 nerve roots, 1.6%). The most commonly identified variation was in the sacral nerve root (n = 5) followed by the L4 (n = 3) and L5 (n = 1) nerve roots. All variations identified were on the left side. There were 3 cases of type 1a variation, 1 of type 1b, 1 of type 2, and 4 of type 3. In total, 210 (36.6%) of the magnetic resonance images showed high nerve root take-off angles at the intervertebral foramen that was caused by disk herniation, spondylolisthesis, or osteophytes with degeneration. Patients with high nerve root take-off angles were significantly older than those without (P < 0.05). These slides can be retrieved under Electronic Supplementary Material.

Symptomatic Os Intercuneiform: A Case Report.

We encountered a case of an accessory bone in the foot in the distal portion of the tarsal navicular and the proximal portion of the intermediate cuneiform, namely an os intercuneiform. The patient presented with a history of pain on the dorsal aspect of the left foot, with tenderness and swelling. Perioperative findings revealed a synfibrotic connection between the accessory bone and the navicular and intermediate cuneiform. After unsuccessful conservative treatment, the accessory bone was excised, leading to postoperative symptomatic relief.

Total calcanectomy for metastasis of renal cell carcinoma in the calcaneus: A case report.

We present a rare case of metastasis of renal cell carcinoma to the calcaneus in a 59-year-old man who presented with pain and inability to bear weight on the left foot 3 years after right nephrectomy for renal cell carcinoma. He successfully underwent en bloc resection of his right calcaneus with a limb salvage procedure, total calcanectomy without bony reconstruction. Histological findings identified the lesion as a metastasis originating from a renal cell carcinoma. Recent follow-up examination showed no recurrence. To the best of our knowledge, this is the first reported case to be treated with total calcanectomy for renal cell carcinoma metastasis.

Atypical Lipomatous Tumor/Well-Differentiated Liposarcoma Developed in a Patient with Progressive Muscular Dystrophy: A Case Report and Review of the Literature.

BACKGROUND: Atypical lipomatous tumor/well-differentiated liposarcoma (ALT/WDLS) is an intermediate or locally aggressive form of adipocytic soft tissue sarcoma. Muscular dystrophy (MD) is characterized by progressive muscle atrophy and its replacement by adipose and fibrous tissue. Recently, some authors have reported that MD genes are related to neoplastic formation, but there have been no detailed clinical reports of ALT associated with MD. CASE PRESENTATION: A 73-year-old woman with a diagnosis of limb-girdle MD visited our department for recurrence of a huge tumor in her left thigh. She had undergone resection of a lipoma at the same site more than 20 years earlier. Imaging studies revealed a lipomatous tumor in her left thigh. We performed marginal resection including the adjacent muscles. Histological diagnosis was atypical lipomatous tumor. The postoperative course was uneventful, with no recurrence at 36 months of follow-up. CONCLUSION: We encountered a huge atypical tumor in a patient with MD. This is the first detailed report to describe an association between ALT and MD. We hypothesize that degenerative changes occurring in adipose tissue during muscle atrophy can cause lipomatous neoplasms and moreover that the mutation of MD-related genes may lead to the proliferation of tumor cells or to malignancy.

Burned-out seminoma revealed by solitary rib bone metastasis.

Burned-out tumor is a rare phenomenon in which a testicular tumor regresses in the primary lesion and progresses in a metastatic lesion. We report the case of a 30-year-old male with burned-out seminoma revealed by open biopsy of solitary 10th rib bone metastasis. He underwent inguinal orchiectomy, which revealed hyalinization, indicating a spontaneously regressed testicular tumor. Chemotherapy for seminoma was administered in three cycles of bleomycin + etoposide + cisplatin therapy. The chemotherapy was effective, and wide resection of the rib was subsequently performed. No postoperative chemotherapy was performed, and there has been no evidence of recurrence for 3 years postoperatively.

Fibroma of tendon sheath on the medial side of the knee: a case report.

Fibroma of tendon sheath, which is a benign soft tissue tumor, primarily affects the finger, hand, or wrist. It rarely involves the knee and only a few cases appear in the literature. Here, we report a case of fibroma of tendon sheath on the medial side of the knee, in a previously hardly reported location, and provide detailed imaging and histological findings. A 54-year-old man presented with his right knee pain and a palpable mass that had developed 3 months earlier. Magnetic resonance imaging showed isointensity in the soft tissue tumor on T1-weighted images, variable intensity on T2-weighted images, and contrast enhancement. The specimen obtained by needle biopsy showed no histological findings of malignancy. Marginal resection was performed and the microscopic diagnosis was fibroma of tendon sheath. Since fibroma of tendon sheath is relatively rare, the radiological feature is not specific, and a rate of local recurrence following excision is high, careful diagnosis, surgical treatment and long-term follow-up are necessary. J. Med. Invest. 64: 173-176, February, 2017.

Radiographic changes of cervical destructive spondyloarthropathy in long-term hemodialysis patients: A 9-year longitudinal observational study.

Analyses of radiographic changes and clinical symptom of destructive spondyloarthropathy (DSA) on consecutive 42 patients managed with long-term hemodialysis were performed to elucidate radiographic changes of DSA and the factors that influence to the destructive changes. Patients underwent plain radiographs of the cervical spine with 9 years interval. Grading of radiological feature from lateral view was classified into grade 0 to grade 3. Clinical symptom was evaluated using modified Japanese Orthopaedic Association scoring system for cervical myelopathy (mJOA score). Destructive changes were observed in 3 patients at the first examination, and those were observed in 15 patients 9 years after the first examination. There is no statistically significant difference between the duration of hemodialysis and the grade. The mean age at the onset of hemodialysis, however, was significantly higher in patients of grade 2 and 3 than those of grade 1. Older patients with long-term hemodialysis had destructive changes. Destructive changes commonly observed in lower cervical spine. The average numbers of the involved disc level were 1.6 in grade 2 and 1.0 in grade 3. Clinical symptoms were varied in each grade and there was no statistically significant difference in total mJOA score among these grades. J. Med. Invest. 64: 68-73, February, 2017.

Diffusion tensor imaging and tractography of the sciatic nerve: assessment of fractional anisotropy and apparent diffusion coefficient values relative to the piriformis muscle, a preliminary study.

OBJECTIVE: Piriformis muscle syndrome (PMS) is underdiagnosed. To evaluate the potential of diffusion tensor imaging and diffusion tensor tractography as innovative tools for the diagnosis of PMS by functional assessment of the sciatic nerve, the aims of this study are to assess the reproducibility and to evaluate the changes in the parameters at levels proximal and distal to the piriformis. MATERIALS AND METHODS: Fractional anisotropy (FA) and the apparent diffusion coefficient (ADC) of the sciatic nerve at three levels were quantified twice each by two examiners using the fiber-tracking method. In the first part of the study, laterality and reproducibility were evaluated using intraclass correlation coefficients (ICC) in ten healthy volunteers. In the second part of the study, the healthy side and symptomatic side were assessed in ten consecutive patients with sciatica. There were three patients with no findings on lumbar magnetic resonance imaging (MRI). RESULTS: There was no laterality in either FA or ADC values in asymptomatic patients at any level. The mean intra-rater ICC was 0.90 and the mean inter-rater ICC was 0.87. FA was significantly lower and ADC significantly higher on the symptomatic side at each level in patients with sciatica. In the three sciatica patients with no findings on lumbar MRI, FA was significantly lower and ADC was significantly higher only at levels distal to the piriformis. These patients experienced full pain relief after ultrasound-guided injection of local anesthesia. CONCLUSIONS: Diffusion tensor imaging and diffusion tensor tractography might be innovative tools for the diagnosis of PMS.

Delayed Diagnosis of Primary Bone and Soft Tissue Tumors Initially Treated as Degenerative Spinal Disorders.

Background Symptoms of primary bone and soft tissue tumors located in the trunk mimic those of degenerative spinal disorders such as low back pain or pain or numbness of the legs. Due to their rarity, especially in sarcoma located in the trunk, diagnosis is sometimes delayed. Methods A retrospective review was performed of the records of 383 patients with primary bone and soft tissue tumors who visit our out patient clinic from 2011 to 2013 at a single institution. Patients with delayed diagnosis of primary bone and soft tissue tumors initially treated as degenerative spinal disorders for more than 2 months were identified. Results Of the 383 patients, 5 (1.3%) were initially diagnosed as having degenerative spinal disorders. All 5 patients had bone or soft tissue tumors in the lumbar spine or pelvis. Three patients had a malignant tumor (osteosarcoma, chondrosarcoma, or small round cell sarcoma) at the progressive stage, and 1 died of lung metastasis. Two patients had a benign tumor (aneurysmal bone cyst or simple bone cyst). Conclusions Our findings suggest that when low back pain and leg pain are refractory to conservative treatment, further radiological examination should be conducted. J. Med. Invest. 63: 274-277, August, 2016.

Complete spontaneous regression of a subpubic cartilaginous cyst: a case report.

Subpubic cartilaginous cyst was recently reported as a rare degenerative mass on the pubic symphysis. We report here a 59-year-old woman who presented with a vulvar mass that showed complete spontaneous regression 48 months after the initial visit. Treatment was only wearing brace. This is the first report of complete spontaneous regression of a subpubic cartilaginous cyst. In the case of small subpubic cyst, observation and follow-up alone may be sufficient. J. Med. Invest. 63: 319-322, August, 2016.

Arthroscopic excision of unstable os acromiale associated with impingement syndrome: a case report.

Os acromiale is a rare anatomical variant that is caused by failure of fusion of the acromial apophysis and is usually asymptomatic. We report a case of impingement syndrome of the left shoulder secondary to unstable os acromiale, which was initially overlooked and confirmed only during arthroscopic examination. Arthroscopic excision of the unstable fragment was successful without residual dysfunction of the deltoid muscle.

Pigmented villonodular synovitis of the knee joint in a 5-year-old girl treated with combined open and arthroscopic surgery: a case report.

Pigmented villonodular synovitis is an extremely rare disease in skeletally immature patients. Erosive destruction of the involved joint leads to early arthritis, and its high recurrence rate makes treatment challenging. Multiple surgical approaches exist, but it is unclear as to which among them achieves the lowest possible recurrence rate and morbidity. We report the case of a 5-year-old girl with left knee pain and swelling who was diagnosed with diffuse pigmented villonodular synovitis of the left knee based on MRI findings. Combined open and arthroscopic surgery was performed to completely remove the tumor. Postoperative histopathological examination confirmed the diagnosis of diffuse pigmented villonodular synovitis. The postoperative course was uneventful, with a gradual improvement in symptoms. There were no signs of recurrence on postoperative MRI performed at the 8-month follow-up, with neither knee pain nor limitation of range of motion. The favorable outcome in this case suggests that combined open and arthroscopic surgery may be an effective method for treating pigmented villonodular synovitis in skeletally immature patients.

Acceleration of skeletal age MR examination using compressed sensing.

PURPOSE: To examine the feasibility of accelerating magnetic resonance (MR) image acquisition for children using compressed sensing (CS). Skeletal age assessment using MRI sometimes suffers from motion artifacts because of the long scan time in children. Reducing image acquisition time may provide benefits by reducing motion artifacts, increasing efficiency of examination, and creating a stress-free environment. MATERIALS AND METHODS: Undersampling patterns for CS were optimized and CS-based examination with the acceleration factors of 3 (CS3, 55 seconds per scan) and 4 (CS4, 41 seconds per scan) was performed for 59 subjects (35 boys and 24 girls; mean age, 9.1 years; age range, 4.4-15.3 years) using a 0.3T scanner. The skeletal age was assessed by two raters (A and B). RESULTS: The interrater and intrarater reproducibility in skeletal age assessment was high (Pearson's r = 0.966 [CS3(A1) vs. CS3(A2)], 0.962 [CS4(A1) vs. CS4(A2)], 0.935 [CS3(A1) vs. CS3(B)], and 0.964 [CS4(A1) vs. CS4(B)]; P < 0.001). The errors in skeletal age assessed on the basis of CS-reconstructed images were similar to those assessed on the basis of fully Nyquist-sampled images. CONCLUSION: These results demonstrate the validity and reliability of skeletal age examination accelerated by CS-MRI. We conclude that the acceleration factor of 3 was optimal. J. Magn. Reson. Imaging 2016;44:204-211.

State-of-the-art ultrasonographic findings in lower extremity sports injuries.

Athletes sometimes experience overuse injuries. To diagnose these injuries, ultrasonography is often more useful than plain radiography, computed tomography (CT), or magnetic resonance imaging (MRI). Ultrasonography can show both bone and soft tissue from various angles as needed, providing great detail in many cases. In conditions such as osteochondrosis or enthesopathies such as Osgood-Schlatter disease, Sinding-Larsen-Johansson disease, bipartite patella, osteochondritis dissecans of the knee, painful accessory navicular,and jumper's knee, ultrasonography can reveal certain types of bony irregularities or neovascularization of the surrounding tissue. In patients of enthesopathy, ultrasonography can show the degenerative changes at the insertion of the tendon. Given its usefulness in treatment, ultrasonography is expected to become essential in the management of overuse injuries affecting the lower limb in athletes. J. Med. Invest. 62: 109-113, August, 2015.