Clinical Manifestations and Diagnostic Considerations of C7-T1 Single-Level Myelopathy: A Case Series.

Cervical myelopathy is caused by compression of the cervical spinal cord for any reason. Cervical myelopathy most commonly affects the C5-6 level. However, C7-T1 single-level myelopathy is rare, and neurological findings may be atypical, making diagnosis difficult. We report three cases and discuss their clinical manifestations. Unlike other levels of cervical myelopathy, C7-T1 single-level myelopathy may present with gait disturbance without neurological deficits in the upper extremities. In addition, all three of our cases had different levels of spinal cord compression and locations of sensory deficits; at the C7-T1 level, the spinal cord compression may not correspond to the site of the sensory deficit. These features may help clinicians in the diagnosing of myelopathy.

Comparison of Postoperative Results With Prognostic Nutritional Index for Lumbar Disc Herniation.

INTRODUCTION: The prognostic nutritional index (PNI) is an immune-nutritional index simply provided by a blood test. We retrospectively compared the postoperative outcomes of patients with lumbar disc herniation divided into two groups according to the PNI. MATERIALS AND METHODS: Seventy-three patients who underwent surgery at our hospital were included in the study. All patients had herniation between one of the L3/4, L4/5, or L5/S intervertebral discs and underwent one posterior lumbar interbody fusion. These patients were divided into two groups: patients with a PNI of <50 (poorly nourished (PN) group) and patients with a PNI of ≥50 (well-nourished (WN) group). Evaluation items included patient background characteristics, operative time, blood loss, postoperative complications, and length of hospital stay. RESULTS: The results showed that the body mass index was significantly higher in the WN group than in the PN group (p=0.0221). The rates of collagen disease, steroid use, and postoperative complications were significantly higher (p=0.0475, p=0.0073, and p=0.0211, respectively) and the length of hospital stay was significantly longer (p=0.021) in the PN group than in the WN group. CONCLUSION: In conclusion, this study indicates that postoperative complications and the length of hospital stay are significantly worse in PN patients than in WN patients.

Factors Affecting the Second Complete Atypical Femoral Fracture after the First Atypical Fracture.

OBJECTIVES: Atypical femoral fracture (AFF) is an atypical low-energy subtrochanteric and diaphyseal femoral fracture. Even if bone fusion is achieved in patients with AFF, the risk of AFF in the contralateral femur must be considered. This study aimed to investigate the factors affecting complete AFF in the contralateral femur and conservatively treated incomplete AFF. SUBJECT AND METHODS: Radiographs of 111 femurs in 104 AFF cases were examined, and the femurs were classified as follows: 85 contralateral femurs with complete AFF; 18 contralateral femurs with incomplete AFF; 8 femurs with incomplete AFF without surgical treatment. Various patients' clinical data were collected, and we investigated the factors affecting the second complete AFF. RESULTS: Complete fractures occurred in 10 (9.7%) of 103 femurs without incomplete AFF at the first visit and in 3 (37.5%) of 8 femurs with incomplete AFF. The Kaplan-Meier curve revealed that lateral cortical bone thickening and thigh pain were associated with significantly poorer prognoses (p = 0.026 and p = 0.013, respectively). Multivariate analyses revealed that eldecalcitol usage after AFF onset (p = 0.0094) and previous use of bisphosphonate or denosumab (p = 0.0126) were protective factors for second complete AFF and that the presence of thigh pain (p = 0.0134) was a risk factor for second complete AFF. CONCLUSIONS: Eldecalcitol administration after bone union of first AFF may prevent AFF recurrence. In addition, painful incomplete AFF has a high risk of developing a complete fracture.

A Comparison of Hidden Blood Loss Between Multilevel Posterior Lumbar Interbody Fusion and Lateral Lumbar Interbody Fusion in Adult Spinal Deformity Patients.

STUDY DESIGN: Retrospective cohort study. OBJECTIVES: This study aimed to compare transfusion and blood loss volumes, particularly hidden blood loss volume, in adult spinal deformity patients undergoing lateral lumbar interbody fusion (LLIF) and posterior lumbar interbody fusion (PLIF). Corrective surgery for adults with spinal deformity patients can be performed via the traditional posterior approach (PLIF) or minimally invasive lateral approach (LLIF). LLIF is associated with longer or comparable operation times and lower intraoperative blood loss. However, hidden blood loss has not been compared between the two procedures. METHODS: We compared hidden blood loss and other blood loss volumes, transfusion volume, operation time, and radiographic surgical outcomes between LLIF patients (n = 71) and PLIF patients (n = 33) who underwent surgery in our institution from September 2011 to June 2018. All patients provided informed consent in accordance with the Declaration of Helsinki. Institutional ethics committee conducted this study approval was obtained. RESULTS: Transfusion volume and intraoperative and total blood loss volumes were significantly higher in the PLIF group. Operation time and HBL did not significantly differ. The hidden blood loss-to-intraoperative blood loss ratio was significantly higher in the LLIF group (113% vs 60%; P = .004). Radiographic measures of outcome were significantly better after surgery than before in both groups. CONCLUSIONS: Although intraoperative blood loss was less with LLIF than PLIF, HBL was similar between the approaches. Patients undergoing LLIF should be followed closely for postoperative anemia even if intraoperative blood loss is low.

Two Cases of Degenerative Cervical Spondylotic Myelopathy in Adults with Athetoid and Dystonic Cerebral Palsy.

BACKGROUND Patients with athetoid and dystonic cerebral palsy (ADCP) may develop degenerative changes in the cervical spine that can aggravate their neurological symptoms in adulthood. This report is of 2 cases of ADCP associated with degenerative cervical spondylotic myelopathy in a 39-year-old woman and a 52-year-old man, requiring different surgical treatments. CASE REPORT Case 1. The patient was a 39-year-old woman who had fallen down 7 years before surgery and had since been walking with a cane. Her gait disturbance had worsened in the 2 years prior to surgery, and numbness in her upper limbs appeared. In the year before surgery, spasticity and numbness in the lower limbs worsened, and fine motor impairment also appeared. Because of mild involuntary movements of the neck, cervical laminoplasty from C3 to C6 was performed, and her symptoms remained stable until the last follow-up 4.5 years after surgery. Case 2. The patient was a 52-year-old man who had fallen down 7 years before surgery, resulting in transient limb weakness. In the year before surgery, he had developed fine motor impairment. He subsequently developed gait disturbance and requested cervical surgery. Because of involuntary movements involving the neck and trunk, he underwent cervical posterior fusion from C2 to T1. Six months after surgery, the gait disturbance had improved. CONCLUSIONS This report describes 2 adults with a history of ADCP since birth and highlights that degenerative changes of the cervical spine can occur at a relatively early age in adulthood, requiring an individualized approach to management.

Acute respiratory distress syndrome relapsing in 10 months with an initial manifestation of polymyositis.

Autoimmune disorders are an important cause of acute respiratory distress syndrome (ARDS). We report a case of a patient with steroid-responsive ARDS that relapsed in 10 months with an initial manifestation of seronegative polymyositis. ARDS associated with polymyositis may develop earlier than myopathy and may relapse later.

A case of humidifier lung with a difficult differential diagnosis from COVID-19.

Acute respiratory illnesses that presented with diffuse ground-glass opacities (GGOs) on chest computed tomography (CT) scan suggest the diagnosis of coronavirus disease 2019 (COVID-19). However, many other diseases show similar CT findings, which often offer a difficult differential diagnosis. Here, we report a case of humidifier lung, a rare phenotype of hypersensitivity pneumonitis (HP), which mimicked COVID-19. A 71-year-old man was admitted because of dyspnea and diffuse GGOs found on chest CT scan. Although COVID-19 was initially suspected, his symptoms rapidly improved by the next day. A medical interview revealed that he had started using an ultrasonic humidifier 1 month ago. A high-resolution CT (HRCT) scan showed ill-defined centrilobular nodules and mosaic attenuation, which are typical of HP but atypical of COVID-19. The inhalation challenge test confirmed the diagnosis of humidifier lung. History-taking of humidifier use and a precise HRCT interpretation are helpful to differentiate it from COVID-19.

Early intervention of plasma exchange combined with intensive immunosuppressive treatment for anti-MDA-5 antibody-positive rapidly progressive interstitial pneumonia: Two case reports.

BACKGROUND: Anti-melanoma differentiation-associated gene 5 antibody (anti-MDA5 Ab) has to be reported to often cause rapidly progressive interstitial lung disease (RP-ILD) especially in East Asian countries. Even with the recommended rapid administration of immunosuppressive agents with high-dose corticosteroids, intravenous pulse cyclophosphamide, and calcineurin inhibitors, the prognosis of anti-MDA5 Ab-related RP-ILD is poor. Plasma exchange (PE) has been reported to be effective for steroid-refractory RP-ILD with anti-MDA5 Ab. However, the timing, frequency, and interval of PE for the treatment of RP-ILD with anti-MDA5 Ab have not yet been established. CASE PRESENTATION: We report two cases of RP-ILD with anti-MDA5 Ab treated by early intervention of PE combined with immunosuppressive treatment. Blood biomarkers including titers of anti-MDA5 Ab, serum KL-6 and ferritin were promptly decreased after each session of PE. Clinical symptoms, oxygenation and chest computed tomography abnormalities were completely improved after immunosuppressive treatment with PE. CONCLUSION: Early intervention of PE combined with immunosuppressive treatment may prevent the development to lethal severe respiratory failure in RP-ILD with anti-MDA5 Ab.

Evaluation of the Nature and Etiologies of Risk Factors for Diaphyseal Atypical Femoral Fractures.

OBJECTIVES: Differences in mechanisms of subtrochanteric and diaphyseal atypical femoral fractures (AFFs) are speculated in studies that analyzed differences in the patients' background. However, the etiologies of each type of AFF have not been studied in detail. This study aimed to investigate the nature and etiologies of the risk factors for diaphyseal AFFs. MATERIALS AND METHODS: Eighty consecutive Japanese patients with 91 diaphyseal AFFs (AFF group) and 110 age-matched women with osteoporosis (non-AFF control group) were included. Their clinical data were compared; factors affecting AFFs were investigated, and the etiologies of the risk factors for diaphyseal AFFs were examined. RESULTS: Multivariate analysis revealed that femoral serrated changes, bisphosphonate or denosumab usage, and lateral and anterior femoral curvatures were risk factors for diaphyseal AFFs (p < 0.0011, p = 0.0137, and p < 0.0001, respectively). Multivariate analyses revealed that serrated changes and low serum 25(OH)D levels affected the lateral curvature (p = 0.0088 and 0.0205, respectively), while serrated changes affected the anterior curvature (p = 0.0006), each significantly affected the femoral curvature. High serum calcium (Ca) levels, lateral femoral curvature, and anterior femoral curvature were predictors of serrated changes (p = 0.0146, 0.0002, and 0.0098, respectively). CONCLUSION: Risk factors for diaphyseal AFFs were bone resorption inhibitor usage, a strong femoral curvature, and serrated changes. Low serum 25(OH)D levels and serrated changes are risk factors for lateral curvature, while a high serum Ca level is a risk factor for serrated changes.

Glasgow prognostic score for prediction of chemotherapy-triggered acute exacerbation interstitial lung disease in patients with small cell lung cancer.

BACKGROUND: Predicting the incidence of chemotherapy-triggered acute exacerbation of interstitial lung disease (AE-ILD) in patients with lung cancer is important because AE-ILD confers a poor prognosis. The Glasgow prognostic score (GPS), which is an inflammation-based index composed of serum levels of C-reactive protein and albumin, predicts prognosis in patients with small cell lung cancer (SCLC) without ILD. In this study, we investigated AE-ILD and survival outcome based on the GPS in patients with ILD associated with SCLC who were receiving chemotherapy. METHODS: Medical records of patients who received platinum-based first-line chemotherapy between June 2010 and May 2019 were retrospectively reviewed to compare the incidence of AE-ILD and overall survival (OS) between GPS 0, 1, and 2. RESULTS: Among our cohort of 31 patients, six (19.3%) experienced chemotherapy-triggered AE-ILD. The AE-ILD incidence increased from 9.5% to 25.0% and 50.0% with increase in GPS of 0, 1, and 2, respectively. Univariate and multivariate analyses revealed remarkable associations between GPS 2 and both AE-ILD (odds ratio for GPS 2, 18.69; p = 0.046) and prognosis (hazard ratio of GPS 2, 13.52; p = 0.002). Furthermore, median OS in the GPS 0, 1, and 2 groups was 16.2, 9.8, and 7.1 months, respectively (p < 0.001). CONCLUSIONS: Our results suggest that GPS 2 is both a predictor of risk of chemotherapy-triggered AE-ILD and a prognostic indicator in patients with ILD associated with SCLC. We propose that GPS may be used as a guide to distinguish chemotherapy-tolerant patients from those at high risk of AE-ILD.

Glasgow Prognostic Score predicts chemotherapy-triggered acute exacerbation-interstitial lung disease in patients with non-small cell lung cancer.

BACKGROUND: Interstitial lung disease (ILD) in patients with non-small cell lung cancer (NSCLC) worsens the prognosis for overall survival (OS) due to chemotherapy-triggered acute exacerbation (AE)-ILD. The Glasgow Prognostic Score (GPS), which is based on serum C-reactive protein and albumin levels, has been suggested as a reliable prognostic tool for mortality in cancer patients, including NSCLC. In this study, we investigated whether GPS is a predictor for chemotherapy-triggered AE-ILD and the prognosis in patients with NSCLC and pre-existing ILD. METHODS: We conducted a retrospective review on 56 NSCLC and ILD patients at our hospital who received platinum agent-based treatment as first-line chemotherapy between June 2010 and May 2019. We categorized these patients according to their GPS (0-2) and compared the incidence of chemotherapy-triggered AE-ILD and OS. RESULTS: The GPS 0, 1, and 2 groups included 31, 16, and nine patients, respectively, out of 56. A total of 12 (21.4%) patients showed chemotherapy-triggered AE-ILD. The median OS was at 11.5 months (95% confidence interval: 8.0-15.1). The incidence of chemotherapy-triggered AE-ILD within the first year of chemotherapy in the GPS 0, 1, and 2 groups was three (9.6%), four (25.0%), and five (55.5%), and the median OS time was 16.9, 9.8 and 7.6 months, respectively. Univariate and multivariate analyses indicated that only GPS 2 could predict both chemotherapy-triggered AE-ILD and OS (P < 0.05). CONCLUSIONS: GPS assessment of patients with NSCLC and pre-existing ILD is a valuable prognostic tool for predicting chemotherapy-triggered AE-ILD and OS. KEY POINTS: SIGNIFICANT FINDINGS OF THE STUDY: We found that GPS 2 was an independent risk factor for chemotherapy-triggered AE-ILD and prognosis in patients with ILD associated with NSCLC. WHAT THIS STUDY ADDS: GPS may potentially enable the discrimination of patients tolerant of chemotherapy from those at an increased risk of AE-ILD and predict the prognosis in patients with NSCLC and ILD receiving chemotherapy.

Proximal junctional fracture and kyphosis after long spinopelvic corrective fixation for adult spinal deformity.

BACKGROUND: Proximal junctional fractures (PJFx) are the main cause for proximal junctional kyphosis (PJK), a complication of adult spinal deformity surgery. This study investigated risk factors for PJFx when performing long spinopelvic corrective fixation with lateral interbody fusion from T9 to ilium. METHODS: This was a retrospective study of 43 patients with a minimum follow-up of 2 years. Radiographic measurements including thoracic kyphosis (TK), sagittal vertical axis (SVA), T1 pelvic angle (TPA), proximal junctional angle (PJA), lumbar lordosis (LL), lower LL, and pelvic tilt were measured preoperatively, one-month postoperatively, and at final follow up. TK and LL were also measured in a fulcrum backward-bending position preoperatively. RESULTS: At final follow-up, PJK was found in 30/43 patients (69.8%); 20.9% of the cases had PJFx (9 patients). TPA (preoperative, and one-month postoperative) was significantly higher in the PJFx group than in the other groups. The differences in TPA, TK, and PJA between preoperative and one-month postoperative measurements in the PJFx group were significantly higher than those in the other groups. At final follow up, SVA was significantly higher in the PJFx group than in the "PJK without PJFx" group. TPA and TK were significantly higher in the PJFx group than in the other groups. PJA was significantly different between all groups. CONCLUSION: Preoperative large TPA was the only risk factor for PJFx. Preoperative flexibility of the thoracolumbar spine and overcorrection of sagittal deformity were not related to PJFx or PJK.

Evaluation of factors affecting the occurrence of second atypical fracture after bone union of the first atypical fracture.

PURPOSE: Teriparatide is sometimes used in the treatment of atypical femoral fracture (AFF). Even if bone union is achieved, orthopedic physicians must consider the risk of relapse. This study aimed to investigate the factors affecting AFF recurrence, and to determine the appropriate treatment for osteoporosis after bone union. METHODS: One hundred thirty-one consecutive AFFs in 113 Japanese patients were included. Eleven patients had AFF in the unaffected limb (9 patients) after the first AFF or re-fracture at the original fracture site (2 patients) after bone union of the first AFF was confirmed. We divided all patients into two groups: the second fracture group (22 AFFs in 11 patients) and non-second fracture group (109 AFFs in 102 patients). We compared clinical information between the 2 groups and investigated the factors affecting AFF recurrence using the Student t-, Welch t-, and chi-square tests. RESULTS: Although there was no significant difference in clinical characteristics between the 2 groups, multivariate analysis of factors associated with AFF recurrence identified short duration of treatment with teriparatide and active vitamin D3 (p = 0.0408 and 0.0366, respectively) as risk factors. Even in the analysis excluding subtrochanteric AFF, short periods of teriparatide and active vitamin D3 administration were observed as risk factors (p = 0.0484 and 0.0346, respectively). CONCLUSION: The administration of teriparatide for as long as possible after occurrence first AFF and the use of active vitamin D3 after completion of teriparatide therapy may be the most effective strategy to prevent the recurrence of AFF.

Diagnosis and Surgical Treatment of Thoracic Dorsal Arachnoid Web: A Report of Two Cases.

INTRODUCTION: An arachnoid web (AW) is a relatively rare disease and shows clinical symptoms and radiological findings similar to those of an arachnoid cyst (AC) or spinal cord herniation (SCH). Since the operative procedures for an AW are generally different from those intrathecal disorders, correct preoperative differential diagnosis is important. The purposes of this study were to report the usefulness of magnetic resonance imaging (MRI) and computed tomography (CT) myelography for diagnosing AW and to show the histological findings and clinical results. Case Description. Two patients, a 79-year-old man and a 43-year-old woman, are presented. The primary diagnoses were AC with ossification of the ligamentum flavum and epidural hematoma, respectively, in previous hospitals. They were finally diagnosed by the characteristic MRI and CT myelogram finding called the "scalpel sign." Histological findings showed epithelial cells and fibrous tissue derived from arachnoid tissues and microcalcifications. After surgery, the scalpel sign has vanished, and aggravation of their symptoms was prevented. CONCLUSION: An AW is refractory, but early detection by MRI and CT myelography and early treatment improve outcomes after surgery.

A case report of revision occipital-cervical fusion after atlanto-axial instrumentation failure for neurofibromatosis type I.

BACKGROUND: Neurofibromatosis type 1 is an autosomal dominant genetic disease with characteristic café-au-lait spots, neurofibroma, and dystrophic changes in the bones. However, complications involving atlanto-axial dislocation are rare. CASE PRESENTATION: We report a case of neurofibromatosis with atlanto-axial dislocation. The chief complaints were numbness of the upper limb and gait disturbance. We performed short fusion using the Brooks method. However, recurrence of the dislocation was found after 5 months recovery, and the patient underwent posterior fusion from the occipital bone to C4. Thereafter, she had a good postoperative course. CONCLUSIONS: Neurofibromatosis patients often exhibit a low bone mineral density because of dystrophic changes, and are prone to fragile bones. In the present case, the use of long fusion at the first surgery may have helped to form a strong fusion of fragile bone.

Effects of teriparatide on bone in autochthonous transgenic model mice for diabetes mellitus (Akita mice).

OBJECTIVES: The purpose of this study is to evaluate the effects of teriparatide (TPTD) on bone mineral density (BMD), bone strength, and bone quality in Akita mouse models of diabetes mellitus. METHODS: Twelve-week-old female Akita mice and control mice (C57/BL/6NCrSlc) were divided into 4 groups: control mice treated with vehicle (n = 7) or TPTD (n = 6); and Akita mice treated with vehicle (n = 6) or TPTD (n = 7). TPTD or vehicle was administered subcutaneously 3 times a week for 8 weeks. Blood glucose, serum sclerostin, total tibial BMD, femoral shaft bone strength, and bone quality using Fourier-transform infrared spectroscopy imaging were evaluated. RESULTS: No significant differences in serum sclerostin levels were evident among these groups after 8 weeks of treatment. TPTD significantly increased BMD in control mice (+12.7%, P = 0.02) and Akita mice (+29.2%, P = 0.001) compared with vehicle. Maximum load and stiffness were significantly higher in Akita mice treated with TPTD than in Akita mice treated with vehicle (+56.6%, P = 0.03 and + 90.5%, P = 0.02, respectively). On Fourier-transform infrared spectroscopy imaging, the mineral/matrix ratio was significantly lower in Akita mice treated with vehicle than in control mice (-12.2%, P = 0.02), and TPTD treatment significantly increased the mineral/matrix ratio (P = 0.003). CONCLUSIONS: TPTD thus improved BMD and bone strength in both control mice and Akita mice, with improvements in the mineral/matrix ratio among Akita mice.

Comparison of patients with diagnosed and suspected sacral insufficiency fractures.

BACKGROUND: Patients with sacral insufficiency fractures can have a range of symptoms, and because these fractures are difficult to detect using plain radiographs, it can be tough to make a definitive diagnosis of fracture early after injury. The aim of this study was to compare the diagnosis and treatment of patients with known sacral insufficiency fractures to those with suspected insufficiency fractures to clarify the features of sacral insufficiency fractures. METHODS: We compared patients with sacral insufficiency fractures (S group) to those with suspected insufficiency fractures (N group) using demographic data, symptoms, time to definitive diagnosis, radiological methods, and treatments. RESULTS: Patients in the S group were older than those in the N group (p = 0.0042) and showed less localized sacral pain (p = 0.0042). Almost all of the patients in the S group (74%) required magnetic resonance imaging for definitive diagnosis. CONCLUSIONS: Sacral insufficiency fractures should not be diagnosed based on the site of pain or using plain radiographs. Patient age and magnetic resonance imaging are more informative to obtain a definitive diagnosis of sacral insufficiency fractures.

Surgical Treatment for Severe Scoliosis in Patients with Reduced Cardiorespiratory Function after Surgery for Congenital Heart Disease: A Report of Two Cases.

PURPOSE: Congenital heart disease (CHD) is associated with an increased risk of scoliosis. The prognosis of scoliosis patients with CHD has improved because of advances in cardiac care. As a result, the frequency of surgery for scoliosis in this population has increased, although the risk of perioperative complications remains high. We treated two patients with CHD who underwent surgery for severe scoliosis. To avoid perioperative complications, we evaluated the preoperative cardiac status and anesthetic risks before posterior correction and fixation in both patients. METHODS: An expert anesthesiologist evaluated the anesthetic risk in each case, and an adequate reservoir of autologous blood was collected preoperatively. The patient in case 1 was at risk of significant blood loss and required extremely careful operative technique. The patient in case 2 had low cardiac output preoperatively. We therefore performed a thorough preoperative cardiac evaluation. Both patients were admitted to the intensive care unit postoperatively. RESULTS: Neither patient suffered serious complications, and both achieved favorable outcomes. CONCLUSIONS: Appropriate surgical technique and teamwork among experts are the keys to success in patients with severe scoliosis and CHD.