Thoracic osteotomy for Gorham-Stout disease of the spine: a case report and literature review.

INTRODUCTION: Gorham-Stout syndrome is an aggressive, non-heritable skeletal disease characterized by osteolysis following minor trauma. The primary involvement of the spine is less common (10 %) and has been described in only about 20 cases; there is no consensus about the best way to treat this condition. PURPOSE OF THE STUDY: To report a case of Gorham-Stout syndrome involving the thoracic spine and to review the literature to suggest a post-operative treatment to prevent osteolysis. CASE REPORT: A thirty-year-old female patient was admitted to the unit in March 2013 for a pathologic T4 fracture. X-rays and CT scan revealed the onset of T4 osteolysis and an increase in thoracic kyphosis (the local kyphosis was up to 100°). We performed surgery by posterior approach, combining posterior fixation with screws and rods from T3 to T9, decompression and vertebral osteotomy of 65°. The immediate outcome of surgery was good and the patient returned home after 24 days. At 6 and 12 months of follow-up, the patient was walking normally with no neurological sequelae. CONCLUSION: We report a case of Gorham-Stout syndrome involving the thoracic spine that was successfully treated by interpedicular osteotomy associated with a 6 month follow-up. We suggest that this strategy can provide good results, because after fusion, the disease process remains stable. Because of the lack of cases reported, surgeons must be careful when using multiple treatments, because these treatments have many side effects. LEVEL OF EVIDENCE: Level IV case report.

Reference ranges for serum periostin in a population without asthma or chronic obstructive pulmonary disease.

BACKGROUND: The clinical utility of serum periostin as a type 2 biomarker in asthma is limited by lack of reference range values derived from a population without respiratory disease. OBJECTIVE: To derive age- and sex-related reference intervals for serum periostin from an adult population without asthma or COPD. METHODS: Serum periostin levels were measured in 480 individuals, comprising 60 female and 60 male adults in each of the 18- to 30-year, 31- to 45-year, 46- to 60-year and 61- to 75-year age groups. Key exclusion criteria included a doctor's diagnosis of asthma, chronic bronchitis or COPD, and a history of wheezing or use of respiratory inhalers in the last 12 months. The distribution of periostin and logarithm-transformed periostin levels was derived, and 90% confidence intervals for an individual prediction were calculated. RESULTS: The distribution of serum periostin was right skewed with a mean (SD) periostin of 51.2 (11.9) ng/mL, median (IQR) 50.1 (43.1 to 56.9) ng/mL and range 28.1 to 136.4 ng/mL. There was no association between logarithm periostin and age or sex, although levels were low in current smokers. The 90% confidence limits for periostin were 35.0 and 71.1 ng/mL. CONCLUSIONS AND CLINICAL RELEVANCE: Serum periostin levels in adults without asthma or COPD are similar to those in adults with asthma. Serum periostin measurements do not need to be adjusted to take account of a patient's age or sex, although levels are lower in current smokers. Reference values for serum periostin levels in adults without asthma or COPD are provided.

Reproducibility and repeatability of a new computerized software for sagittal spinopelvic and scoliosis curvature radiologic measurements: Keops(®).

PURPOSE: The purpose of this study was to evaluate the inter- and intra-observer variability of the computerized radiologic measurements using Keops(®) and to determine the bias between the software and the standard paper measurement. METHODS: Four individuals measured all frontal and sagittal variables on the 30 X-rays randomly selected on two occasions (test and retest conditions). The Bland-Altman plot was used to determine the degree of agreement between the measurement on paper X-ray and the measurement using Keops(®) for all reviewers and for the two measures; the intraclass correlation coefficient (ICC) was calculated for each pair of analyses to assess interobserver reproducibility among the four reviewers for the same patient using either paper X-ray or Keops(®) measurement and finally, concordance correlation coefficient (rc) was calculated to assess intraobserver repeatability among the same reviewer for one patient between the two measure using the same method (paper or Keops(®)). RESULTS: The mean difference calculated between the two methods was minimal at -0, 4° ± 3.41° [-7.1; 6.4] for frontal measurement and 0.1° ± 3.52° [-6.7; 6.8] for sagittal measurement. Keops(®) has a better interobserver reproducibility than paper measurement for determination of the sagittal pelvic parameter (ICC = 0.9960 vs. 0.9931; p = 0.0001). It has a better intraobserver repeatability than paper for determination of Cobbs angle (rc = 0.9872 vs. 0.9808; p < 0.0001) and for pelvic parameter (rc = 0.9981 vs. 0.9953; p < 0.0001). CONCLUSIONS: We conclude that Keops(®) has no bias compared to the traditionally paper measurement, and moreover, the repeatability and the reproducibility of measurements with this method is much better than with similar standard radiologic measures done manually in both frontal and sagittal plane and that the use of this software can be recommended for clinical application. LEVEL OF EVIDENCE: Diagnostic, level III.

Survey of practices in surgical management of trapeziometacarpal osteoarthritis in France in 2020.

The purpose of this study was to obtain an overview of French surgical practices for treating trapeziometacarpal osteoarthritis in 2020. An online survey was sent to 64 French hand surgeons: 32 authors of articles on carpometacarpal osteoarthritis of the thumb and 32 other surgeons randomly selected from the membership of the French Society of Hand Surgery (SFCM). The questions concerned demographic data, surgical practice, operative indications, choices for revision surgery, and eight clinical cases. The response rate was 56.2%. The most popular technique was trapeziometacarpal replacement (63.9%). During the previous 5 years, 31.6% of respondents had changed their practices, 69.2% of whom had adopted total joint replacement. Total trapeziectomy with ligamentoplasty and interposition was the second most frequent method. Most surgeons (77.8%) implemented medical treatment for 6 months to 1 year before resorting to surgery. In the clinical cases, agreement between surgeons was very low, with an overall inter-rater concordance coefficient of 0.182. Except for two cases (a young manual worker and a patient with a flattened trapezium) where no technique was significantly preferred, trapeziometacarpal replacement was chosen by a majority of respondents (p < 0.001). It was the most frequently performed surgical technique in France in 2020. However, there is no real consensus on choice of technique, which reflects the absence of guidelines.

Tiny but risky: The reasons why the Caspar pin distractor causes suffocating cervical hematoma-Two cases and a literature review.

Symptomatic cervical hematoma (CH) after cervical spine surgery through an anterolateral approach is a feared complication. In up to 60% of CH cases, no source of bleeding is detected during drainage. Bleeding from the pin holes of the Caspar distractor is a known complication, briefly mentioned in the patent, but harmfulness has never been thoroughly assessed. Our team experienced two consecutive postoperative acute CHs, in which the origin of active bleeding obviously came from pin holes, despite careful obturation. The aim of this study was to report these two cases and provide a comprehensive assessment of the dangers of Caspar pin distractors. The intrabody vascularization was well organized and there was a central pedicle arising from the center of the posterior wall. The pedicle penetrated deeply into the body and constituted Batson's channel posteriorly. Retrospectively, it was well-identified in both cases on preoperative imaging. Given the morphological features of the vertebral cervical bodies and Caspar pin, the pin may reach the center of the posterior wall where the pedicle arises. Comparison between vertebral body volumes and the volume of the screwable part of the pins revealed that the pin could occupy up to 7.3% of the total body if randomly inserted. However, pins are in fact inserted into a particular place that contains the pedicle. Epidural bleeding is variable and may be undetected before closure. This also depends on blood pressure variations and changes in the patient's position. Even though Caspar pins are tiny, the likelihood of intrabody vascularization damage appears to be significant. Caspar pins should not be used systematically. Pin hole obturation must be solid and deep. Alternative options such as an interbody distractor and a microscope for the discectomy should be considered.

Restricted access to innovative surgical technique related to a specific training, is it ethical? Example of the PIPAC procedure. A systematic review and an experts survey.

OBJECTIVE: Using the example of Pressurized Intra Peritoneal Aerosol Chemotherapy (PIPAC), we analyse the development model of this procedure and provide an ethical analysis of the involvement of the industry in a new development. SUMMARY BACKGROUND DATA: In the case of breakthrough innovation, medical training is essential for safe use of the new procedure. In some cases, pharmaceutical companies decide to organise this training. But when it becomes the only training opportunity to use the device, scientists and clinicians could be exposed to a conflict of interest? METHODS: We performed a literature review of PIPAC publications using the STROBE criteria. Then, we conducted interviews with an expert panel to analyse the ethical impact of involvement of the industry in the development of the PIPAC procedure. RESULTS: The number of publications has increased every year since the first publication in Germany, where the technology was developed in 2013. The scientific production was of good quality, with a mean STROBE score of 18.2 ± 2.4 out of 22 points. Ten of the 33 included studies declared a conflict of interest. From the interviews, the main axe concerning the implication of the industry was the training model. The company had decided that only trained and approval surgeon could perform the PIPAC procedure. All four interviewed practitioners agreed that it was initially a good way to implement the procedure safely, but later they felt uncomfortable about the control and validation by the industry. CONCLUSION: Based on the growing number of published papers from a growing number of international centres, the controlled training model is not limiting. However, the different levels of conflict of interest complicate transparency, and we postulated that this development model is limited to the beginning of the procedure diffusion. CLINICALTRIAL. GOV REGISTRATION: NCT04341337.

Magnetic resonance angiography: from anatomical knowledge modeling to vessel segmentation.

Magnetic resonance angiography (MRA) has become a common way to study cerebral vascular structures. Indeed, it enables to obtain information on flowing blood in a totally non-invasive and non-irradiant fashion. MRA exams are generally performed for three main applications: detection of vascular pathologies, neurosurgery planning, and vascular landmark detection for brain functional analysis. This large field of applications justifies the necessity to provide efficient vessel segmentation tools. Several methods have been proposed during the last fifteen years. However, the obtained results are still not fully satisfying. A solution to improve brain vessel segmentation from MRA data could consist in integrating high-level a priori knowledge in the segmentation process. A preliminary attempt to integrate such knowledge is proposed here. It is composed of two methods devoted to phase contrast MRA (PC MRA) data. The first method is a cerebral vascular atlas creation process, composed of three steps: knowledge extraction, registration, and data fusion. Knowledge extraction is performed using a vessel size determination algorithm based on skeletonization, while a topology preserving non-rigid registration method is used to fuse the information into the atlas. The second method is a segmentation process involving adaptive sets of gray-level hit-or-miss operators. It uses anatomical knowledge modeled by the cerebral vascular atlas to adapt the parameters of these operators (number, size, and orientation) to the searched vascular structures. These two methods have been tested by creating an atlas from a 18 MRA database, and by using it to segment 30 MRA images, comparing the results to those obtained from a region-growing segmentation method.

Absence of a food effect with a 145 mg nanoparticle fenofibrate tablet formulation.

OBJECTIVES: The present study was conducted to assess the effect of food on the bioavailability of fenofibric acid from a new tablet formulation containing fenofibrate nanoparticles. METHODS: In this 3-way crossover study, 45 subjects received in a random order one 145 mg fenofibrate tablet under high-fat fed (HFF), low-fat fed (LFF) or fasting (reference) conditions. Plasma concentrations of fenofibric acid were determined up to 120 hours post-dose. Comparisons were made between test (HFF and LFF) and reference conditions (fasting). RESULTS: Very close values of pharmacokinetic parameters were obtained following the three diffiferent regimens. The 90% confidence intervals (CI) for the ratio of geometric means of HIFF versus fasting condition were (1.018-1.088) for AUCinfinity, (1.020-1.090) for AUCt and (0.963-1.054) for Cmax with point estimate:s of 1.052, 1.054 and 1.007, respectively. The 90% CI for the geometric means of LFF versus fasting condition were (0.978-1.046) for AUGinfinity, (0.981-1.047) for AUCt and (0.964-1.055) for Cmax with point estimates of 1.012, 1.013 and 1.009, respectively. They all fall within the required limits for bioequivalence (0.80-1.25). A slightly prolonged tmax was observed following HFF conditions (4.3 +/- 1.9 hours, versus 3.6 +/- 1.2 hours and 2.3 +/- 0.7 hours under LFF and fasting conditions, respectively), without any effect on mean Cmax. CONCLUSION: The peak and overall exposures from the new 145 mg fenofibrate tablet were not affected by food. Therefore, this new fenofibrate tablet may be taken without regard to the timing of meals.

Change in acetabular version after lumbar pedicle subtraction osteotomy to correct post-operative flat back: EOS® measurements of 38 acetabula.

BACKGROUND: Abnormalities in acetabular orientation can promote the development of hip osteoarthritis, femoro-acetabular impingement, or even acetabular cup malposition. The objective of the present study was to determine whether pedicle substraction osteotomy (PSO) to correct sagittal spinal imbalance affected acetabular orientation. HYPOTHESIS: PSO performed to correct sagittal spinal imbalance affects acetabular orientation by changing the pelvic parameters. MATERIALS AND METHODS: This was a descriptive study in which two observers measured the acetabular parameters on both sides in 19 patients (38 acetabula) before and after PSO for post-operative flat-back syndrome. Mean time from PSO to post-operative measurements was 19months. Measurements were taken twice at a 2-week interval, on standing images obtained using the EOS(®) imaging system and sterEOS(®) software to obtain 3D reconstructions of synchronised 2D images. Acetabular anteversion and inclination were measured relative to the vertical plane. Mean pre-PSO and post-PSO values were compared using the paired t-test, and P values lower than 0.05 were considered significant. To assess inter-observer and intra-observer reproducibility, we computed the intra-class correlation coefficients (ICCs). RESULTS: The measurements showed significant acetabular retroversion after PSO, of 7.6° on the right and 6.5° on the left (P<0.001). Acetabular inclination diminished significantly, by 4.5° on the right and 2.5° on the left (P<0.01). Inclination of the anterior pelvic plane decreased by 8.4° (P<0.01). Pelvic incidence was unchanged, whereas sacral slope increased by 10.5° (P<0.001) and pelvic tilt decreased by 10.9° (P<0.001). The ICC was 0.98 for both inter-observer and intra-observer reproducibility. CONCLUSION: Changing the sagittal spinal alignment modifies both the pelvic and the acetabular parameters. PSO significantly increases sacral slope, thus inducing anterior pelvic tilt with significant acetabular retroversion. The measurements obtained using sterEOS(®) showed good inter-observer and intra-observer reproducibility. To our knowledge, this is the first study of changes in acetabular version after PSO.

How do fiber-supplemented formulas affect antroduodenal motility during enteral nutrition? A comparative study between mixed and insoluble fibers.

BACKGROUND: Fiber supplementation during enteral nutrition has been recommended, but the effect of soluble compared with insoluble fiber supplements on antroduodenal motility is unknown. OBJECTIVE: The objective of this study was to compare antroduodenal motor patterns in 8 healthy volunteers during and after gastric infusion of 3 different diets: a fiber-free diet, an insoluble-fiber diet, and a mixed-fiber diet (50% soluble fiber and 50% insoluble fiber). DESIGN: Manometric studies with the 3 different diets (2100 kJ) were performed in random order. Antroduodenal motility was monitored continuously for 6 h by using a pneumohydraulic system to calculate the number, amplitude, and duration of the pressure waves; the area under the curve (AUC); and the percentage of time occupied by motor activity before, during, and after each type of infusion. Variations in antral areas were measured by ultrasonography. RESULTS: The gastric motor response was significantly higher, whatever the diet, in the distal antral recording site than in the 2 more proximal sites. In the proximal but not the distal antrum, the number of waves, the AUC, and the percentage of time occupied by motor activity were higher (P: < 0.04) with the mixed-fiber than with the insoluble-fiber diet. No significant differences in variations of antral area were observed among the 3 diets. In the duodenum, motor variables were not significantly different among the 3 diets. CONCLUSIONS: A gastric infusion induced a greater motor response in the distal than in the proximal antrum. A mixed-fiber diet was associated with significantly greater proximal antral motility than was an insoluble-fiber diet. There was no significant difference among the 3 formulas in duodenal motor variables or in variations in antral area as measured by ultrasound.

Octreotide in refractory functional epigastric pain with nutritional impairment--an open study.

AIM: To test the therapeutic efficacy of octreotide administered subcutaneously for the relief of chronic refractory epigastric pain severe enough to provoke nutritional impairment. SUBJECTS AND METHODS: Seventeen patients were enrolled in an open trial. Epigastric pain had lasted from 1 to 8 years (median: 5 years), following anti-reflux surgery in eight patients. Median weight loss was 10% (range 10-15). The initial dose of octreotide was 50 microgram b.d, adjusted during the follow-up visits which were scheduled for months 1, 3, 6, 8, 10, 12 and every 3 months. At each visit, overall symptomatic improvement, frequency and intensity of symptoms were checked on a 10-cm visual analogic scale. RESULTS: At month 1, a progressive improvement of pain intensity was reported in 15 of the 17 patients, while octreotide was a therapeutic failure in two. In four out of 15, the daily dose of octreotide was increased to 100 microgram b.d. In these 15 patients, median follow-up was 7 months (3-27). The symptomatic benefit was maintained in each patient at month 3, with a median weight gain of 3.5 kg.2-5 An attempt to stop octreotide led to recurrence of symptoms in 2-3 days which were as intense as before the treatment. The 11 patients followed-up for at least 6 months reported persistent improvement of symptoms with octreotide and a median weight gain of 4 kg.3-7 Four patients were followed up for more 11-27 months: octreotide was withdrawn gradually in two who remained asymptomatic. Six of the 17 patients experienced minor side-effects, but none developed biliary sludge. CONCLUSIONS: This open study suggests that octreotide could be a promising alternative treatment when all others fail in refractory chronic functional epigastric pain severe enough to limit food intake and to induce nutritional impairment. These results must be tested by a placebo-controlled study.

Anatomy and embryology of the trigeminal nerve and its branches in the parasellar area.

The cavum trigeminale (Meckel's cave) anatomy is still poorly understood. Many different descriptions are found in the literature. In order to clarify the relationship of trigeminal ganglion and its branches with dura and arachnoid, we underwent an embryological and adult microanatomical and histological study. Serial sections of human embryos and fetuses were used. For adult study, microdissections and histological serial sections were performed. We found that dura and arachnoid stop at the trigeminal ganglion and do not extend the three branches of the trigeminal nerve. These three branches are embedded into separate peripheral sheaths. These results are important for clear understanding of the anatomy of the parasellar lodge (cavernous sinus) lateral wall.

The venous system of the lateral sellar compartment (cavernous sinus): an histological and embryological study.

The microanatomy of the lateral sellar or parasellar venous system (so-called cavernous sinus) is poorly understood and is still passionately debated. The exact nature of this venous structure is not yet clear whether it is a plexus or a sinus. In order to understand the anatomy of this area better, an embryological and adult microanatomical study was performed. Serial histological sections of human embryos and fetuses of well determined ages (6, 7, 8, 10, 11, 12, 14, 30 weeks) were used. The adult study was performed with microdissections and serial histological sections of laterosellar compartments. The analysis of this material confirms that the parasellar venous system is a true sinus, on the embryological and histological point of view. They differ from true veins. As other dural sinuses, it may contain different types of channels: from simple venous canal to complex venous plexus. The opinions of different authors are reviewed in the introduction and discussed with the results.

Comparative microanatomy of the lateral wall of the 'cavernous sinus' in humans and the olive baboon.

Despite many studies of the 'cavernous sinus' lateral wall, the anatomy of this area remains controversial. We performed a comparative microanatomical and histoarchitectural study in 14 humans and in 10 nonhuman primates (Papio cynocephalus anubis). Venous channels and cranial nerves were embedded in the 'interperiosteodural space'. The dura propria of the lateral wall could be removed without entering the venous compartment. The oculomotor and trochlear nerves were accompanied by an arachnoidal and dural sheath. The oculomotor nerve sheath stopped under the anterior clinoid process in baboons. The trigeminal ganglion was covered posteriorly with an arachnoid membrane and adhered firmly to the dura propria on lateral and anterior sections. The three branches of the trigeminal nerve had no arachnoid covering, except for arachnoid granulations in humans. In baboons, the oculomotor and trochlear nerves were thicker than in humans, while the ophthalmic nerve was thinner. The abducens nerve belonged to the lateral wall of the sinus in baboons and had no arachnoidal sheath except in the first millimeters of Dorello's canal. After leaving their arachnoidal and dural sheath, the intracavernous cranial nerves acquired a typical peripheral sheath. The venous channels in both species were true dural sinuses. Willis cords and adipose tissue were identified.

Anatomy and embryology of the lateral sellar compartment (cavernous sinus) medial wall.

In order to study the microanatomy of the lateral sellar compartment (cavernous sinus) medial wall, serial histological sections of human fetuses and adults, as well as dissections under operative microscope, were performed. The results were compared to high resolution Magnetic Resonance Images in human, to microdissections and to serial histological sections in adult nonhuman primates (Papio Cynocephalus anubis). We were able to show that the sellar compartment and both lateral sellar osteodural compartments are not separated from each other by a dural wall, but by a more or less dense, interrupted, fibrous tissue which derived from the mesenchyme surrounding the hypophysis, carotid artery, cranial nerves and venous channels. In the human fetus, the previous mentioned structures are located in a unique interperiosteodural space. Histoarchitecture of the superior and lateral wall dura-mater was different from the underlying mesenchyme derived connective tissue and was easily distinguished through histological examination. These findings correspond to MRI data. We conclude that there is no medial dural wall limiting the lateral sellar compartment (cavernous sinus), both parasellar and the hypophyseal compartment should be considered as a unique extradural space. The only dense connective tissue surrounding the pituitary gland is its own glandular capsule and the periosteum.

The perispinal spaces. Constitution, organization and relations with the cerebrospinal fluid (CSF).

In this study the structures of the three meningeal membranes which surround the spinal cord and delineate the perispinal spaces are described. The pia mater is composed of a deep layer and a superficial layer. This structural arrangement accounts for the relations of the pia mater with the spinal vessels and the cerebrospinal fluid (CSF). The morphology, structure and landmark value of the denticulate ligament are then defined. Following a brief description of the dura mater and the arachnoidea, the arrangement of the meningeal sleeves of the spinal nerves is detailed. Meningeal structures similar to the cranial arachnoidal granulations are described, and the question of CSF physiology is considered. The secretion and circulation of the CSF are briefly described, and the problem of spinal resorption is discussed. CSF resorption can be effected by ascending, lymphatic or venous routes.

[Arterial vasculature of the dura mater of the anterior cranial fossa]. / La vascularisation artérielle de la dure-mère de la fosse crânienne antérieure.

The arterial vascularization of the dura mater of the anterior cranial fossa belongs to the middle meningeal artery, branch of the external carotid artery, and to the ophthalmic artery issued from the internal carotid artery. This vascular diagram presents numerous variations, whose complexity may be explained by the vascular morphogenesis. The distribution of the dural arteries and the dural microvasculature are described.

[Vascular architecture of the caudate nucleus]. / Architecture vasculaire du noyau caudé.

The aim of this study is to describe the vascular characteristics of the nucleus caudatus (NC). This study is performed on 30 human brains by injecting the vascular system with gelatinous Indian ink. Examination of serial slices, 250 microns thickness, made translucent by the Spalteholz method, enabled us to follow the course and behavior of the vessels of the NC. The arteries of the NC belong to the group of the central arteries arising from the anterior and middle cerebral arteries or from the anterior choroidal arteries. Inside the different parts of the NC (head, body and tail) the central arteries and their ramifications give rise to a specific vascular network quite similar to this one of the putamen. This network consists of regular, polygonal, rectangular or square meshes. Each mesh is composed by the juxtaposition of vascular units formed by an arterial ring arranged around a principal vein which is surrounded by a capillary free space. The arterioles of the network of the NC are terminal or side branches of the central arteries. They are characteristic in their course by forming rope-like structures (coils) also described on the ramifications of the putaminal central arteries. Similarities between the vascular networks of the NC and the putamen are in relation with the histological structure, with the morphogenetic evolution, with the functional activity. The specificity and the clear delimitation of the vascular networks of the NC and of the putamen are particularly obvious in cerebral vascular diseases leading to infarcts strictly limited to these two nuclei.

[Ophthalmic artery, optic nerve and meninges: reciprocal relations]. / Artère ophtalmique, nerf optique et méninges: relations réciproques.

The origin of the ophthalmic artery exhibits several variants which can be explained by a vascular morphogenesis study. The particular relations of the ophthalmic artery with the optic nerve and the cranial meninges are examined using serial histological sections.