Clinical application of bone turnover markers in treating osteoporotic vertebral compression fractures and their role in predicting fracture progression.

This study aimed to investigate whether changes in the bone turnover markers (BTMs) during teriparatide therapy for osteoporotic vertebral compression fractures could reflect therapeutic effects by analyzing the relationship between clinical and radiological features and BTMs. A total of 33 patients with 51 osteoporotic vertebral compression fracture segments were included. Plain radiographs and BTM levels were evaluated at the pretreatment and at 3 months after teriparatide treatment. Based on serial vertebral compression ratio analysis, the progression of fracture was defined as a vertebral compression ratio decrease of ≥10%, relative to the pretreatment values. All segments were divided into 2 groups: the "maintain" group with 32 (62.7%) segments and the "progression" group with 19 (37.3%) segments. After the teriparatide treatment, serum osteocalcin and serum C-terminal telopeptide of type I collagen levels (P = .028 and .008, respectively), and change amounts of them were significantly larger, increasing (P = .001) in the progression group. The vitamin D (25OH-D) levels were significantly lower (P = .038) in the progression group; however, the relative changes in the 25OH-D levels between the 2 groups, before and after the treatment, were not significantly different (P = .077). The parathyroid hormone (PTH) levels were reduced by the teriparatide treatment in both groups, while the decrease in PTH concentration after the treatment was significantly more pronounced in the progression group (P = .006). Significant increase in the osteocalcin and serum C-terminal telopeptide of type I collagen levels and a simultaneous decrease in the PTH levels during the teriparatide treatment suggest that clinicians should assume the progression of fracture.

Risks and benefits of timely screw removal after thoracolumbar spine fractures treated with non-fusion technique.

BACKGROUND: Percutaneous-short segment screw fixation (SSSF) without bone fusion has proven to be a safe and effective modality for thoracolumbar spine fractures (TLSFs). When fracture consolidation is confirmed, pedicle screws are no longer essential, but clear indications for screw removal following fracture consolidation have not been established. METHODS: In total, we enrolled 31 patients with TLSFs who underwent screw removal following treatment using percutaneous-SSSF without fusion. Plain radiographs, taken at different intervals, measured local kyphosis using Cobb' angle (CA), vertebra body height (VBH), and the segmental motion angle (SMA). A visual analogue scale (VAS) and the Oswestry disability index (ODI) were applied pre-screw removal and at the last follow-up. RESULTS: The overall mean CA deteriorated by 1.58° (p < 0.05) and the overall mean VBH decreased by 0.52 mm (p = 0.001). SMA preservation was achieved in 18 patients (58.1%) and kyphotic recurrence occurred in 4 patients (12.9%). SMA preservation was statistically significant in patients who underwent screw removal within 12 months following the primary operation (p = 0.002). Kyphotic recurrence occurred in patients with a CA ≥ 20° at injury (p < 0.001) with a median interval of 16.5 months after screw removal. No patients reported worsening pain or an increased ODI score after screw removal. CONCLUSION: Screw removal within 12 months can be recommended for restoration of SMA with improvement in clinical outcomes. Although, TLSFs with CA ≥ 20° at the time of injury can help to predict kyphotic recurrence after screw removal, the clinical outcomes are less relevant.

Outcomes of Patients Undergoing Anterior Screw Fixation for Odontoid Fracture and Analysis of the Predictive Factors for Surgical Failure.

OBJECTIVE: Anterior odontoid screw fixation (AOSF) is a safe and effective treatment for type II and rostral type III odontoid fracture. This study aimed to report the outcomes of the AOSF surgery and evaluate the potential risk factors of surgical failure. METHODS: We enrolled 63 patients who underwent AOSF. Follow-up computed tomography was performed 6 months after the surgery and once a year thereafter to evaluate the union. Clinical data including the age, sex, presenting symptoms, cause of injury, fracture gaps, dislocation position, degree of displacement, screw direction angle, and time interval from injury to operation were collected. RESULTS: Successful fusion was achieved in 55 patients (87.3%) and surgical failure occurred in 8 patients (12.7%). Variables such as age, sex, dislocation position, degree of displacement, screw direction angle, and time interval from injury to operation were not significantly associated with the surgical failure. However, surgical failure was statistically significantly associated with the fracture gap. The overall mean fracture gap at the time of injury was 1.29 mm (range, 0-3.11 mm), and the incidence of surgical failure was 8.3 times higher when the fracture gap at the time of injury was > 2 mm (p = 0.019). CONCLUSION: When performing AOSF in patients with type II or rostral shallow type III odontoid fractures, the displacement of the odontoid fracture fragment should be appropriately reduced to the aligning position before screw insertion and downward reduction should be achieved by perforation of the apical cortex of the odontoid during screw fixation, even if the surgery is delayed.

Single-Stage Posterior Circumferential Stabilization Using Double Small Cages for the Treatment of Thoracic and Lumbar Spine Fractures.

OBJECTIVE: Controversy remains regarding the optimal methods for resection of the vertebral body, reconstruction of the anterior column, and decompression of the spinal cord in patients who have severe vertebral body destruction of the thoracic or lumbar spine with associated neurologic impairment. We report an alternative technique for primary treatment and salvage involving single-stage corpectomy followed by reconstruction of the anterior column using double small mesh cages via the posterior-only approach. METHODS: Plain radiographs and computed tomography scans, taken at different intervals, were used to measure local kyphosis, segmental height, and fusion grade. Pain was evaluated using the visual analog scale (VAS), and neurologic symptoms were classified according to Frankel grade. RESULTS: The mean kyphotic deformity improved by 14.47 ± 9.06 degrees (P < 0.001), and the mean segmental height improved by 7.17 mm ± 6.11 mm (P < 0.001) after surgery. Fusion was achieved at 84% of patients, within a median interval of 12 months. Kyphotic recurrence was observed in 2 patients (11%), segmental height loss occurred in 1 patient (5%), and both kyphotic recurrence and segmental height loss occurred in 1 patient (5%). None of the patients reported worsening pain or neurologic symptoms after surgery, and there were no surgery-related complications such as neural injury, cerebrospinal fluid leakage, cage dislocation, surgical site infection, or cardiopulmonary complications. CONCLUSIONS: Single-stage corpectomy followed by reconstruction of the anterior column using double small mesh cages via the posterior-only approach is a reliable and less invasive single-stage treatment and salvage option in selected cases.

Is the Agricultural Work a Risk Factor for Koreans Elderly Spinal Sagittal Imbalance?

OBJECTIVE: A primary degenerative sagittal imbalance has been considered because of unique lifestyles such as the prolonged crouched posture during agricultural work and performing activities of daily living on the floor. Previous papers have reported that sagittal imbalance disease is often seen distinctly in the farming districts of "oriental" countries such as Korea and Japan. However, this finding was only evaluated with the use of X-ray, and other factors such as magnetic resonance imaging (MRI), muscle volume, compression fracture, and laboratory results were not considered. Thus, using these, we evaluate the agricultural work-associated factors for Korean elderly spinal sagittal imbalance. METHODS: We recruited 103 Korean participants who had a sagittal vertical axis (SVA) of >5 cm in this Korean Elderly Sagittal Imbalance Cohort Study. The following were evaluated : radiological parameters, MRI, compression fracture, vitamin D, parathyroid hormone, C-terminal telopeptide, osteocalcin, bone mineral density and muscle fatty change, muscle volume, and health-related quality of life from patients' survey. Moreover, in this survey, the farmers' annual working hours were investigated. Subsequently, we analyzed the associated factors for spinal sagittal imbalance depending on occupation. RESULTS: A total of 46 participants were farmers, and the others were housewives, sellers, and office workers. The farmer group had more SVA (141 vs. 99 mm, p=0.001) and pelvic tilt (31° vs. 24°, p=0.004) and lesser lumbar lordosis (20° vs. 30°, p=0.009) and thoracic kyphosis (24° vs. 33°, p=0.03) than non-farmer group. A significantly positive correlation was noted between the working hour and SVA in the farmer group (p=0.014). The visual analogue scale score for back pain (8.26 vs. 6.96, p=0.008) and Oswestry Disability Index (23.5 vs. 19.1, p=0.003) in the farmer group were higher than that in the non-farmer group, but the Short Form-36 score was not significantly different between the two groups. The Mini-Mental State Exam score was significantly lower in the farmer group than in the non-farmer group (24.85 vs. 26.98, p=0.002). CONCLUSION: The farmer group had more sagittal imbalance and back pain in proportion to the working hours even though the muscle and bone factors and general laboratory condition were not significantly different between the two groups. These results supported that the long hours spent in the crouched posture while performing agricultural work were a risk factor for severe sagittal imbalance.

Langerhans Cell Histiocytosis Involving Second Cervical Vertebra and the Hypothalamus and Pituitary in an Adult.

BACKGROUND: Adult-onset Langerhans cell histiocytosis (LCH) with simultaneous involvement of the high cervical spine and the hypothalamus is rare. CASE DESCRIPTION: We have reported a case of adult-onset LCH in the second cervical vertebra with bony destruction and subsequent diabetes insipidus due to simultaneous involvement of the hypothalamus and pituitary stalk. Magnetic resonance imaging of the hypothalamus and pituitary lesion and immunohistochemistry of the cervical lesion revealed LCH. Posterior fusion of the cervical spine (first, third, and fourth cervical vertebrae) was performed, followed by systemic chemotherapy. The cervical fusion was well maintained, and the patient achieved clinical remission. No new LCH lesion was found during the follow-up of >2 years. CONCLUSIONS: Patients with known LCH of the spine showing new symptoms of diabetes insipidus should be examined for infiltrating lesions of the pituitary stalk or hypothalamus. In cases of severe instability of the spine, surgical treatment should be performed. If multiple and systemic LCH lesions are found, systemic chemotherapy should be administered.

Primary Extraosseous Ewing Sarcoma of the Thoracic Spine Presenting as Chest Pain Mimicking Spinal Schwannoma.

BACKGROUND: Extraosseous Ewing sarcoma (EES) usually has a pseudocapsule and high vascularity, making it well circumscribed and focally dense with contrast enhancement on magnetic resonance imaging (MRI). Consequently, it is difficult to diagnose and distinguish from other spinal tumors, based on pretreatment radiologic findings alone. Here, we present a case of EES involving the thoracic spinal column, which was suspected to be spinal schwannoma through pretreatment radiologic findings. CASE DESCRIPTION: A 54-year-old woman was admitted to our hospital with upper back and left-sided chest pain. Contrast-enhanced MRI of the thoracolumbar spine showed a 17- × 12-mm-sized mass in the epidural region and left neural foramen at the T6-7 level. Our preliminary diagnosis was spinal schwannoma. The patient underwent T6 hemilaminectomy. Intraoperatively, the lesion appeared as gray-colored soft mass with high vascularity, which seemed to have originated from the left T6 nerve root. The tumor was excised with en bloc resection. Histopathologic examination of the lesion revealed classical Ewing sarcoma with high cellularity of small round cells. Immunohistochemistry revealed strong positivity for cluster of differentiation 99 and FLI-1. Intensity-modulated radiation therapy was performed. The patient did not receive chemotherapy. Five years after surgery, follow-up spinal MRI and positron emission tomography computed tomography scan revealed no recurrence of the tumor or new lesions. CONCLUSIONS: Clinicians should consider EES in the differential diagnosis of other neural foraminal spinal tumors, such as schwannoma. If clinicians are confident that EES has been removed completely and there are no other lesions, radiotherapy is sufficient and additional chemotherapy may not be necessary.

Thoracolumbar burst fractures in patients with neurological deficit: Anterior approach versus posterior percutaneous fixation with laminotomy.

BACKGROUND: Thoracolumbar burst fractures (TLBFs) are the most common spinal trauma; however, their appropriate management has not yet been determined. In this study, we aimed to compare the clinical and radiological results of percutaneous pedicle screw fixation (PPSF) following posterior decompression technique versus anterior corpectomy and fusion technique for the treatment of TLBFs. METHODS: A total of 46 patients (2002-2015) with TLBFs were included in this study. The inclusion criteria were a single-level Magerl type A3 burst fracture of the thoracolumbar junctional spine (T12-L2). The patients were divided into two groups; Group A (22 patients) underwent anterior corpectomy and fusion, and Group B (24 patients) underwent PPSF after posterior decompression. Anterior corpectomy and fusion surgery were performed in 22 cases before April 2009, and PPSF following posterior decompression technique was used in 24 cases since then. For radiological assessment, the kyphosis angle was measured preoperatively, early postoperatively, and at the last follow-up using the Cobb angle. Mean correction of the Cobb angle after surgery, and loss of correction between the immediate postoperative and final Cobb angle were calculated accordingly. All neurological deficits were identified in the initial evaluation and graded using the American Spinal Injury Association(ASIA) grading system. Perioperative parameters including operation time, amount of blood loss, and mean hospital stay were also evaluated. RESULTS: The patients comprised 17 males and 5 females in Group A and 13 males and 11 females in Group B. In terms of the involved levels, there were three cases of T12, twelve L1, and seven L2 in Group A and one case of T12, thirteen L1, and ten L2 in Group B. The mean follow-up duration was 44.9 months in Group A and 14.7 months in Group B. The kyphotic angle was significantly corrected after surgery by 6.4° in Group A (p = 0.001) and 9.2° in Group B (p < 0.001). Among patients with neurological deficit, 11 of 15 in Group A and 20 of 23 in Group B demonstrated improvement by at least one ASIA grade at the final observation. However, there was no significant difference in neurological improvement between the two groups (p = 0.13). Mean operation time was significantly shorter (p < 0.001) and mean blood loss was significantly less (p < 0.001) in Group B than in Group A. Mean hospital stay was also significantly shorter in Group B (p < 0.001). CONCLUSIONS: Spinal canal decompression through small laminectomy followed by PPSF in the treatment of TLBFs with neurological deficits offers excellent clinical and radiological improvement as well as biomechanical stability. Furthermore, this can be a safe and effective surgical option with the advantage of less invasiveness in the treatment of TLBFs.

Unexpected Intradural Lumbar Disk Herniation Found During Transforaminal Endoscopic Surgery.

BACKGROUND: Intradural disk herniation (IDH) is defined as the displacement of the intervertebral disk nucleus pulposus into the dural sac. In this lumbar lesion, the affected level differs from that of a traditional extradural herniation: 55% of cases occur at the level of L4-5, 16% at L3-4, and 10% at L5-S1. Upper lumbar IDH is extremely rare. We present a case of an IDH at the level of L2-3 that was diagnosed during endoscopic surgery. CASE DESCRIPTION: A 65-year-old male patient presented with severe radiating pain in the anterior right thigh that was accompanied by a tingling sensation in the right calf and difficulty in walking. Physical examination showed normal strength. Bladder and bowel function was normal, but mild hypesthesia of the L3 sensory dermatome was observed. Magnetic resonance imaging revealed a herniated disk at the level of L2-L3 that was compressing the right side of the dura. A percutaneous transforaminal endoscopic lumbar diskectomy was planned. After foraminoplasty, no ruptured disk fragments could be found. During dissection of the adhesion between the dura and protruded disk, the dura was torn. Interestingly, through this dural opening, multiple fragmented disk portions were visualized among the nerve rootlets. We removed some of the soft disk material; however, complete removal of the disk fragments was predicted to damage the rootlets, and we decided to convert to microscopic surgery. The disk fragments were successfully removed via durotomy under microscopic assistance. The incised dorsal dura was primarily sutured with continuous stitches, and the defect on the ventrolateral side of the dura was patched and sealed using a harvested inner ligamentum flavum and Gelfoam (Pfizer, New York, New York, USA). After the operation, the patient's symptoms improved. There was no cerebrospinal fluid leakage. CONCLUSIONS: If there is any preoperative clinical or radiologic suspicion of IDH, a microscopic surgical approach should be considered to be the first-line option, as this is a safe and effective method for achieving IDH removal and dura repair without a postoperative neurologic deficit. Even during endoscopic surgery, if the surgeon expects even minor complications, we suggest converting to open surgery. In addition, the adequate sealing of the dura may be sufficient to prevent cerebrospinal fluid leakage, without the need for dural suture and lumbar drainage.

Traumatic lumbar disc herniation mimicking epidural hematoma: A case report and literature review.

RATIONALE: We report a rare case of traumatic lumbar disc herniation mimicking epidural hematoma. PATIENT CONCERNS: A 39-year old man presented with acute bilateral leg and back pain, following a crushing injury caused by a crane collapse. DIAGNOSIS: A computed tomography scan revealed multiple compression fractures of the thoracolumbar spine, including a burst fracture of the L4 vertebral body. Magnetic resonance imaging (MRI) showed an epidural mass, extending longitudinally and causing dural sac compression behind the L3 vertebra. The mass had isosignal intensity on T1-weighted imaging and showed mixed high and low signals on T2-weighted imaging. On the basis of the patient's clinical history and imaging findings, our provisional diagnosis was an epidural hematoma following major trauma. INTERVENTIONS: An emergency L3 laminectomy was performed with percutaneous screw fixation of L3-5. Intraoperatively, 3 large herniated disc fragments were found at L3 with no evidence of hematoma. OUTCOMES: The patient recovered normal motor function after surgery. At the last follow-up, 3 years after surgery, there was no residual neurological deficit apart from intermittent lower back pain. LESSONS: In cases where MRI findings reveal an epidural lesion with a longitudinal shape, similar to an epidural hematoma, with mixed signal intensity on T2-weighted images and high peripheral signal intensity on T1-weighted images, traumatic disc herniation should be included in the differential diagnosis.

Pericranial Flap Anterior Skull Base Reconstruction With Gelfoam Intradural Compression.

Anterior cranial base reconstruction is occasionally necessary following severe trauma. Several methods for reconstruction have been described and some authors have described their experiences regarding the use of a pericranial flap for anterior skull base reconstruction after trauma. A 26-year-old woman was admitted to our department with multiple facial bone fractures identified using facial bone computed tomography. Plastic surgeons performed surgery under general anesthesia for the patient's nasal bone fractures. On the seventh day after admission, the patient's brain computed tomography showed an abscess in the frontal lobe. Antibiotic treatment was started, but the lesion deteriorated. Anterior skull base reconstruction was then performed using a pericranial flap with gelfoam compression. No complications, including leakage of cerebrospinal fluid, cerebral hemorrhage, necrosis of the pericranial flap, or frontal lobe herniation, were observed 1 year following surgery. In our case, the authors performed a simple and effective treatment with reconstruction using pericranial flap and gelfoam compression without complications. This technique is useful for reconstructing defects in the base of the frontal bone resulting from various causes, as well as for fracture of the anterior skull base following trauma.

Retropharyngeal Granulation: Delayed Complication of Anterior Cervical Diskectomy and Fusion in C2-3.

BACKGROUND: A 26-year-old man presented with acute quadriparesis owing to a traffic accident. A computed tomography scan revealed a hangman fracture and locking of the left facet joint at C2-3. Magnetic resonance imaging showed a high signal change of the spinal cord on T2-weighted image and hemorrhage in the C2-3 level. CASE DESCRIPTION: An emergency closed reduction after anterior cervical diskectomy and fusion were performed for spinal stability and decompression of the dural sac. Five months postoperatively, C1-2-3 posterior wiring using an iliac bone graft with the Brook method was performed due to nonunion of C2-3. After using the posterior cervical approach, the patient began complaining about the difficulty in swallowing. A 5 × 2 × 1-cm-sized posterior pharyngeal wall mass was detected on an endoscopic examination. Despite conservative management, the retropharyngeal mass progressed and dysphagia worsened, so the retropharyngeal wall granulation mass was resected by a laryngologist. Despite removal of the granulation mass, dysphagia and throat discomfort persisted for about 2 years. The plate and screws were removed considering their possible correlation with the granulation tissue. One month after plate removal, the retropharyngeal granulation tissue resolved almost spontaneously. At the last follow-up, the radiologic examination showed well-fused C1-2-3 with good alignment. The patient had no residual neurologic deficits or dysphagia. CONCLUSIONS: Retropharyngeal granulation can occur as a late complication associated with plate fixation after anterior cervical diskectomy and fusion. Recurrent retropharyngeal wall granulation caused by plate irritation might only be resolved after plate and screw removal.

Outcomes of Anterior Cervical Fusion Using Polyetheretherketone Cage with Demineralized Bone Matrix and Plate for Management of Subaxial Cervical Spine Injuries.

OBJECTIVE: We evaluated the usefulness of a polyetheretherketone (PEEK) cage filled with demineralized bone matrix (DBM) and plate fixation in anterior interbody fusions for subaxial cervical spine injuries. METHODS: A retrospective review of 98 patients (58 women, 40 men; mean age, 49.7 years; range, 17-78 years) who underwent single-level anterior cervical discectomy and fusion (ACDF) using a PEEK cage filled with DBM and plate fixation for subaxial cervical spine injuries from March 2005 to June 2018 was conducted. Bone fusion, interbody height (IBH), segmental lordosis, and adjacent segment degeneration (ASD) development were assessed with plain radiographs and computed tomography. Clinical outcomes were assessed using a visual analog scale (VAS) for pain and the Frankel grade for neurologic function. RESULTS: The mean follow-up period was 27.6 months (range, 6-142 months). Twenty-one patients (21.4%) had an improvement of at least one Frankel grade. The mean preoperative and final follow-up neck pain VAS scores were 8.3±0.9 and 2.6±1.5 (p<0.05). All patients showed solid fusion at the final follow-up. The mean preoperative and final Cobb's angles were -3.7±7.9° and 1.9±5.1° (p<0.05). The mean preoperative and final IBHs were 36.9±1.7 mm and 38.2±1.8 mm (p<0.05). Five patients (5%) showed ASD. CONCLUSION: ACDF using a PEEK cage filled with DBM and plate fixation yielded high fusion rates and satisfactory clinical outcomes without donor-site morbidity. This procedure is safe and effective for single-level subaxial cervical spine injuries.

An outbreak of toxoplasmosis in squirrel monkeys (Saimiri sciureus) in South Korea.

BACKGROUND: Toxoplasma gondii (T. gondii) is an intracellular protozoan parasite that can infect warm-blooded animals including humans. New World monkeys, such as squirrel monkeys, are more susceptible to T. gondii than Old World monkeys, often developing fatal disease. METHODS: In this study, seven of thirteen dead squirrel monkeys at Seoul Grand Park were tested to find the cause of sudden death. RESULTS: The main histopathological findings included interstitial pneumonia, necrotizing hepatitis, and splenitis. Periodic acid-Schiff staining of liver, spleen, and lung revealed cyst structures consistent with bradyzoites. Amplification of the B1 gene was detected in the liver or spleen of all monkeys. Additionally, a restriction fragment length polymorphism assay and phylogenetic analysis of the GRA6 amplicon revealed a consistent clustering with the type II strain of T. gondii. CONCLUSIONS: This study is the first report of T. gondii infection of squirrel monkeys in Korea, and the first report of type II T. gondii based on GRA6 analysis in Korea.

Traumatic pericarditis caused by a bamboo twig in captive waterbuck (Kobus ellipsiprymnus).

A 19-year-old captive male waterbuck (Kobus ellipsiprymnus) exhibited traumatic pericarditis at necropsy. The animal weighed 182 kg at necropsy and revealed no remarkable findings in external observation. Severe pericardial adhesions with fibrosis, hepato-diaphragmatic adhesions, straw-colored ascites and hepatosplenomegaly were observed upon examining the internal organs. Perforations made by a 12-cm-long sharp-ended bamboo twig were detected in the reticulum, diaphragm, pericardium, lung and liver. Trueperella pyogenes was identified in pericardial fluid. To our knowledge, this is the first documented case of traumatic reticulopericarditis caused by a sharp-ended bamboo twig in a captive waterbuck.

Induction of antibody and interferon-γ production in mice immunized with virus-like particles of swine hepatitis E virus.

Virus-like particles (VLPs) composed of the truncated capsid protein of swine hepatitis E virus (HEV) were developed and immune responses of mice immunized with the VLPs were evaluated. IgG titers specific for the capsid protein of swine HEV were significantly higher for all groups of mice immunized with the VLPs than those of the negative control mice. Splenocytes from mice immunized with the VLPs also produced significantly greater quantities of interferon (IFN)-γ than interleukin (IL)-4 and IL-10. These newly developed swine HEV VLPs have the capacity to induce antigen-specific antibody and IFN-γ production in immunized mice.

Collision tumor with inflammatory breast carcinoma and malignant phyllodes tumor: a case report and literature review.

There have been some reports of coincidental presentation of breast carcinoma and phyllodes tumor in the same breast. Most of the cases were carcinoma that arose from a phyllodes tumor with a histologically identified transitional area, and they behaved less aggressively than the usually encountered carcinoma. Collision tumors are rare clinical entities in which two histologically distinct tumor types show involvement at the same site. The occurrence of these tumors in the breast is extremely rare. Here, we report a case of 45-year-old woman who had both invasive ductal carcinoma as the finding of inflammatory carcinoma and a malignant phyllodes tumor in the same breast. There was no evidence of a transitional area between the phyllodes tumor and the invasive ductal carcinoma. To our knowledge, this is the first report of a collision tumor of inflammatory breast carcinoma coincident with a malignant phyllodes tumor in same breast.

Solitary Langerhans cell histocytosis of skull and spine in pediatric and adult patients.

PURPOSE: Langerhans cell histiocytosis (LCH) is a rare neoplasm and has heterogeneous clinical presentation and behavior. We analyzed solitary lytic lesions of the skull and spine in pediatric and adult patients. METHODS: Between 2001 and 2011, 42 patients underwent surgery for LCH. Skull and/or spine involvement were evident in 21 (63.6%) of the 33 pediatric patients and 8 (88.9%) of the 9 adults. The 21 pediatric patients showed the unifocal monosystemic lesions in 10, multifocal monosystemic in 4, and multisystemic in 7. The eight adults comprised seven unifocal lesions and one multifocal monosystemic lesion. Of these cases, we analyzed the clinical courses of solitary LCH of skull and spine in 10 pediatric patients and 7 adults. RESULTS: The median age was 10.1 years (range: 1.1-14.1) in pediatric patients and 34.6 years (range: 26.1-52.0) in adults. The median follow-up was 3.1 years (range: 0.6-9.5). Total excision was done in 15 patients and biopsy in 2. Postoperative adjuvant chemotherapy was done in four pediatric patients and one adult, and comprised mass with dural adhesion (N = 2), skull base lesion (N = 1), atlas mass (N = 1), and vertebral lesion with soft tissue extension (N = 1). During follow-up, recurrence occurred in one pediatric patient who had a skull LCH with a dural adhesion. The patient experienced central diabetes insipidus and scapular pain due to pituitary stalk and scapula involvement 1.3 and 2.4 years later, respectively. CONCLUSION: Even if the solitary lesions of skull and spine show a favorable clinical course, some patients could show aggressive behavior.