Disc-coloration of an ochronotic cervical intervertebral disc in a patient with alkaptonuria: Case report and review of the literature.

OBJECTIVES: Alkaptonuria is a rare inborn disorder of phenylalanine and tyrosine metabolism. It is characterized by an accumulation of homogentisic acid and its oxidation products, possibly resulting into connective tissue damaging. "Ochronosis" is a main feature, which is characterized by tissue discoloration and even alkaptonuric arthropathy. Cervical spine involvement is exceptional and there is a paucity of reports on surgical interventions in these patients. We explored the literature concerning cervical spine involvement in patients with alkaptonuria. PATIENTS AND METHODS: We performed a review of the literature, in which patients with alkaptonuric degenerative changes of the cervical spine were examined. Articles were obtained from MEDLINE. Search terms included: "cervical", "alkaptonuria", "alkaptonuric changes" and "black disc". Additional studies were identified by checking reference lists. Furthermore, we present the case of a 46 year old patient with critical cervical spinal canal stenosis who underwent C6-C7 anterior cervical microdiscectomy and interbody fusion, in order to prevent myelopathic changes. CARE statement guidelines were followed. RESULTS: Peroperatively, we did not encounter any macroscopic abnormalities of the skin, muscles or ligaments. A black discoloration of the nucleus pulposus was observed. Peroperative and postoperative course was uneventful. CONCLUSION: Alkaptonuric degenerative abnormalities most commonly involve the lumbar spine, although the cervical spine can be affected in rare cases. Most frequently, the diagnosis of alkaptonuria can be made based on the clinical phenotype many years before symptoms secondary to ochronotic arthropathy develop. A retrospective diagnosis based on peroperative black discoloration of spinal structures has been described. A black discoloration of the intervertebral disc should encourage the neurosurgeon to further explore the possibility of alkaptonuria, even in the absence of a clear phenotype. Surgical results are mostly satisfactory. Further studies are required in order to better understand this pathology and its postoperative course.

Altered Circulating Immune Cell Distribution in Traumatic Spinal Cord Injury Patients in Relation to Clinical Parameters.

Following a spinal cord injury (SCI), an inflammatory immune reaction is triggered which results in advanced secondary tissue damage. The systemic post-SCI immune response is poorly understood. This study aimed to extensively analyse the circulating immune cell composition in traumatic SCI patients in relation to clinical parameters. High-dimensional flow cytometry was performed on peripheral blood mononuclear cells of 18 traumatic SCI patients and 18 healthy controls to determine immune cell subsets. SCI blood samples were collected at multiple time points in the (sub)acute (0 days to 3 weeks post-SCI, (s)aSCI) and chronic (6 to >18 weeks post-SCI, cSCI) disease phase. Total and CD4+ T cell frequencies were increased in cSCI patients. Both CD4+ T cells and B cells were shifted towards memory phenotypes in (s)aSCI patients and cSCI patients, respectively. Most profound changes were observed in the B cell compartment. Decreased immunoglobulin (Ig)G+ and increased IgM+ B cell frequencies reflected disease severity, as these correlated with American Spinal Injury Association (ASIA) impairment scale (AIS) scores. Post-SCI B cell responses consisted of an increased frequency of CD74+ cells and CD74 expression level within total B cells and B cell subsets. Findings from this study suggest that post-SCI inflammation is driven by memory immune cell subsets. The increased CD74 expression on post-SCI B cells could suggest the involvement of CD74-related pathways in neuroinflammation following SCI. In addition, the clinical and prognostic value of monitoring circulating IgM+ and IgG+ B cell levels in SCI patients should be further evaluated.

Generalization of fear of movement-related pain and avoidance behavior as predictors of work resumption after back surgery: a study protocol for a prospective study (WABS).

BACKGROUND: Previous studies indicated that about 20% of the individuals undergoing back surgery are unable to return to work 3 months to 1 year after surgery. The specific factors that predict individual trajectories in postoperative pain, recovery, and work resumption are largely unknown. The aim of this study is to identify modifiable predictors of work resumption after back surgery. METHODS: In this multisite, prospective, longitudinal study, 300 individuals with radicular pain undergoing a lumbar decompression will be followed until 1-year post-surgery. Prior to surgery, participants will perform a computer task to assess fear of movement-related pain, avoidance behavior, and their generalization to novel situations. Before and immediately after surgery, participants will additionally complete questionnaires to assess fear of movement-related pain, avoidance behavior, optimism, expectancies towards recovery and work resumption, and the duration and severity of the pain. Six weeks, 3 months, 6 months, and 12 months after surgery, they will again complete questionnaires to assess sustainable work resumption, pain severity, disability, and quality of life. The primary hypothesis is that (generalization of) fear of movement-related pain and avoidance behavior will negatively affect sustainable work resumption after back surgery. Second, we hypothesize that (generalization of) fear of movement-related pain and avoidance behavior, negative expectancies towards recovery and work resumption, longer pain duration, and more severe pain before the surgery will negatively affect work resumption, pain severity, disability, and quality of life after back surgery. In contrast, optimism and positive expectancies towards recovery and work resumption are expected to predict more favorable work resumption, better quality of life, and lower levels of pain severity and disability after back surgery. DISCUSSION: With the results of this research, we hope to contribute to the development of strategies for early identification of risk factors and appropriate guidance and interventions before and after back surgery. Trial registration The study was preregistered on ClinicalTrials.gov: NCT04747860 on February 9, 2021.

Cone-beam CT to assess bony fusion following anterior cervical interbody fusion.

PURPOSE: Assessment of bony fusion following anterior cervical interbody fusion (ACIF) is usually done by plain film or CT. We present the first clinical application of Cone-Beam CT (CBCT) to evaluate bony fusion after ACIF. METHODS: A 56-year-old man with disc herniation at C6-C7 underwent ACIF surgery using a compressed nanocrystalline hydroxyapatite interbody device (nanOss-C, Pioneer Surgical Marquette, MI, USA) and a nanocrystalline hydroxyapatite bone graft filler (nanOss Bioactive, Pioneer Surgical Marquette, MI, USA). Imaging follow-up was performed by CBCT (NewTom 5G, QR Srl, Verona, Italy) at 1 day, 6 weeks, 3 and 9 months post-operatively. Two independent assessors quantitatively measured the greyscale changes of the bone graft filler and qualitatively evaluated the bony fusion process. RESULTS: Quantitative analysis of the images showed a steadily increasing matrix density of the bone graft filler over the 9 months follow-up, suggesting increasing calcification. Qualitative evaluation demonstrated different stages of the bone fusion process within the disc space around the cage, at the interface between cage and endplates, and at the interface between bone graft filler and the endplates. CONCLUSIONS: CBCT provides high-resolution cross-sectional imaging of the cervical spine after ACIF. For the first time, in vivo evaluation of the bone graft filler within the centre of the circumferentially radiodense cage and detailed cross-sectional evaluation of bone fusion was achieved. Confirmation of these promising outlooks of CBCT in a large cohort of ACIF patients is needed with regard to routine clinical application and evaluation of different interbody devices.

Giant intracranial capillary hemangioma associated with enlarged head circumference in a newborn.

The authors describe the case of a patient with an intracranial capillary hemangioma, and they review the recent literature on intracranial capillary hemangiomas with special attention to their differential diagnosis and management. The only sign in this 7-week-old boy was head enlargement. There were no neurological deficits, and imaging revealed a large intracranial lesion in the right temporal fossa. The results of biopsy confirmed the diagnosis, and, after endovascular embolization, the entire lesion was resected. The incidence of intracranial capillary hemangioma is very low but may be underestimated. In the present case, the size of the tumor prompted surgical treatment. The natural behavior of extracranial capillary hemangiomas, however, suggests that a conservative approach with follow-up and steroid therapy may also be considered.