Progressive Pain Research Articles

Arginine Vasopressin Deficiency Heralding Rosai-Dorfman Disease With Neurological Manifestations

Abstract Rosai-Dorfman disease (RDD) is a rare heterogeneous disorder of non–Langerhans cell histiocytosis. The patient is a 24-year-old woman who presented with a 1-month history of polydipsia, polyuria, and 25-lb (11.3-kg) weight loss over 6 months and was found to have significantly elevated 24-hour urine volume (8.4 L). Prior to completion of the work-up for her presenting symptoms, she returned with a new complaint of disabling back pain and bilateral lower-extremity numbness with weakness refractory to conservative treatment. Magnetic resonance imaging (MRI) showed a prominent T2 to T4 stenosis from a soft tissue mass. Due to progressive pain and accelerating neurological symptoms, she was admitted for surgical debulking and biopsy. In the 10 hours she was unable to drink fluids surrounding her procedure, her serum sodium climbed to 160 mmol/L (reference interval, 137-145 mEq/L; [137-145 mmol/L]). Urine testing and desmopressin challenge revealed arginine vasopressin deficiency (AVP-D), formerly known as central diabetes insipidus. Pituitary MRI showed a mildly enlarged pituitary gland with loss of normal posterior pituitary signal supporting the diagnosis. Epidural mass pathology showed predominant histiocytes indicating RDD. This case highlights the diverse clinical manifestations of RDD and is an unusual instance of RDD linked with AVP-D and neurological involvement.

A new portable negative pressure wound therapy device: a prospective study investigating clinical outcomes.

Closed surgical incision sites at high risk of complications, and with exudate or leakage, are increasingly being managed with closed incision negative pressure wound therapy (ciNPWT) to reduce tissue stress and increase the force necessary to disrupt the incision. This study was undertaken to investigate the performance and safety of a canister-based, single-use NPWT (suNPWT) system when used on closed surgical incision sites. The investigation was designed as a prospective, open, non-comparative, multicentre study aimed at confirming the safety and performance attributes of the suNPWT system when applied to low-to-moderately exuding closed surgical incisions. The primary performance measure was the wound remaining closed from baseline to the last follow-up visit on day 14. Secondary performance measures included: wound and periwound condition; wear time of the system; product consumption; adherence to therapy; and patients' pain progress. Details of adverse events were also collected. Some 35 patients were recruited. The closed surgical incisions responded well to treatment with the tested suNPWT system. All wounds remained closed throughout the investigation. Consistent with other studies of ciNPWT reporting low infection rates, the current study observed either no or low exudation in 90.4% of wounds at the final visit, together with absence of surgical site infection. Pain severity levels were low, both at dressing change and during delivery of negative pressure. No serious adverse device events were reported. In this study, the suNPWT system supported the healing of closed surgical incisions with no safety concerns relating to its use.

Targeted therapy: P2X3 receptor silencing in bone cancer pain relief

AbstractBone cancer pain remains a significant clinical challenge, often refractory to conventional treatments. The upregulation of the P2X3 receptor in the dorsal root ganglia has been implicated in the pathogenesis of bone cancer pain. This study aimed to elucidate the role of the P2X3 receptor in this context and assess the therapeutic potential of receptor silencing. Utilizing a rat model with Walker 256 cells to simulate bone cancer pain, researchers conducted molecular analyses, including semi‐quantitative RT‐PCR and Western Blot, to investigate P2X3 receptor expression in the dorsal root ganglia. Results demonstrated a marked increase in P2X3 receptor levels in the dorsal root ganglia of the bone cancer pain model. Targeted silencing of the P2X3 receptor using specific shRNA delivered via lentiviral vectors significantly reduced pain sensitivity, underscoring the receptor's potential as a valuable therapeutic target. In addition, a comprehensive gene expression analysis leveraging the GEO data set GSE249443 was performed to explore the underlying biological pathways linked to bone cancer pain. This analysis provided insights into the intricate interplay between bone cancer pain and associated biological processes, offering a deeper understanding of the mechanisms involved in pain modulation and progression. In conclusion, this research identifies the P2X3 receptor as a critical molecular target for mitigating bone cancer pain. The selective silencing of the P2X3 receptor emerges as a promising and innovative therapeutic strategy, presenting novel avenues for managing bone cancer pain and potentially revolutionizing treatment approaches in this challenging domain.

Acute carpal tunnel syndrome after atraumatic rupture of the flexor tendons: a case report

Acute carpal tunnel syndrome (ACTS) is a rare entity, mainly associated with traumatic causes, although patients with predisposing factors such as taking anticoagulants or tendinopathies may debut with an atraumatic ACTS. This case study presents the case of a 77-year-old male patient, anticoagulated, who began with rapidly progressive pain and swelling in the right wrist, radiating towards the medial territory, with no history of traumatic triggering event. Examination showed paraesthesia in the volar aspect of the first to third fingers of the hand and increased pain on passive mobilisation of the fingers. Given the progressive evolution of the condition and the lack of response to both physical and pharmacological measures, the carpal tunnel was opened surgically. During surgery, a significant haematoma was observed secondary to the rupture of the deep flexor tendons of the fourth and fifth fingers inside the canal. Following release of the carpal tunnel, complete recovery of the symptomatology occurred. In conclusion, a high degree of clinical suspicion is necessary in a patient with an examination suggestive of ACTS. The treatment of choice is urgent surgical decompression of the carpal tunnel. Early intervention allows resolution of symptoms and reduces complications.

Understanding the role of machine learning in predicting progression of osteoarthritis.

Machine learning (ML), a branch of artificial intelligence that uses algorithms to learn from data and make predictions, offers a pathway towards more personalized and tailored surgical treatments. This approach is particularly relevant to prevalent joint diseases such as osteoarthritis (OA). In contrast to end-stage disease, where joint arthroplasty provides excellent results, early stages of OA currently lack effective therapies to halt or reverse progression. Accurate prediction of OA progression is crucial if timely interventions are to be developed, to enhance patient care and optimize the design of clinical trials. A systematic review was conducted in accordance with PRISMA guidelines. We searched MEDLINE and Embase on 5 May 2024 for studies utilizing ML to predict OA progression. Titles and abstracts were independently screened, followed by full-text reviews for studies that met the eligibility criteria. Key information was extracted and synthesized for analysis, including types of data (such as clinical, radiological, or biochemical), definitions of OA progression, ML algorithms, validation methods, and outcome measures. Out of 1,160 studies initially identified, 39 were included. Most studies (85%) were published between 2020 and 2024, with 82% using publicly available datasets, primarily the Osteoarthritis Initiative. ML methods were predominantly supervised, with significant variability in the definitions of OA progression: most studies focused on structural changes (59%), while fewer addressed pain progression or both. Deep learning was used in 44% of studies, while automated ML was used in 5%. There was a lack of standardization in evaluation metrics and limited external validation. Interpretability was explored in 54% of studies, primarily using SHapley Additive exPlanations. Our systematic review demonstrates the feasibility of ML models in predicting OA progression, but also uncovers critical limitations that currently restrict their clinical applicability. Future priorities should include diversifying data sources, standardizing outcome measures, enforcing rigorous validation, and integrating more sophisticated algorithms. This paradigm shift from predictive modelling to actionable clinical tools has the potential to transform patient care and disease management in orthopaedic practice.

Effectiveness of Maitland Mobilizations Along with Kinesio Taping on Reducing Pain and Disability in Subjects with Periarthritis Shoulder

Background: Frozen shoulder is a condition in which insidious and progressive pain and loss of active and passive mobility are manifested in the glenohumeral joint, presumably due to capsular contracture. Diabetes mellitus patients are more affected. Various methods were effective in treating frozen shoulder. The present study was done in frozen shoulder patients to determine the additional effect of Kinesio taping and Maitland mobilization in managing frozen shoulder. Frozen shoulder tends to develop in three phases, coming on slowly. Each stage may last several months. Method: The present study included 30 subjects with frozen shoulder. The individuals were randomly divided into 2 groups. GROUP A-experimental group, which received Maitland mobilization and Kinesiotaping treatment, while GROUP B is the control group that received only Maitland mobilization. Both groups first received hot moist packs for 20 minutes and an ultrasound for 5 minutes. Exercises were advised. Subjects received 4 weeks of intervention for 3 days/week. Before and after the intervention, SPADI outcome measures were measured. Result: There was a significant difference showing improvement in the mean of shoulder pain and disability index scale SPADI before and within the groups with p<0.0001. There was a statistical difference when post-intervention means were compared between the groups. Conclusion: Maitland mobilization with Kinesiotaping reduces pain and disability in subjects with peri-arthritis shoulder. KEYWORDS: Frozen shoulder, Maitland mobilization, Kinesiotaping, Shoulder pain and disability index scale (SPADI).

Cervical Myeloradiculopathy With Bilateral Foraminal Stenosis Treated by Unilateral Biportal Endoscopic Decompression with Alternating Hemilaminectomy and Bilateral foraminotomy: A Case Report and Technical Note

Cervical myeloradiculopathy is a debilitating condition characterized by progressive spinal cord dysfunction commonly caused by degenerative changes in the cervical spine. It typically exhibits an insidious onset and progressive functional decline, along with multiple exiting nerve root entrapments bilaterally in some patients. Traditional surgical interventions, such as discectomy and fusion, are associated with some complications and limitations. Unilateral biportal endoscopic (UBE) decompression can have significant advantages over traditional techniques when cervical central and bilateral foraminal decompression is required. In this case report and technical note, we describe our experience with this novel surgical technique for the treatment of cervical myelopathy. A 67-year-old patient presented with progressive neck pain; bilateral upper extremity radiculopathy; loss of power and sensation in the C5, C6, and C7 nerve root distribution; gait imbalance, and spasticity. Magnetic resonance imaging revealed multilevel cervical spondylosis with central canal and bilateral foraminal stenosis. The patient underwent UBE central and bilateral foraminal decompression. At a 9-month follow-up visit, he was able to walk unassisted, his reflexes had returned to normal, no spasticity was observed, and he had good grip strength in both hands. This is a novel technique of UBE decompression with bilateral foraminotomy at the C4–5, C5–6, and C6–7 levels and laminectomy named “the floating tip technique.” The procedure involved bilateral 3-level lamino-foraminotomy and hemilaminotomy on both C5 and C6. A hemilamina was removed on the opposite sides, and flavectomy was performed. Postoperative computed tomography findings showed an intact, complete muscular mass with an intact central spinous process. Therefore, this method was labelled the “floating tip technique.”

Ulnar Compression Neuropathy Secondary to Ganglion Cyst at the Elbow Joint: A Case Report

Ulnar neuropathy is a common peripheral neuropathy, with cubital tunnel compression being the second most frequent upper extremity compression syndrome. Ganglion cysts, though rare, can contribute to ulnar nerve compression in the cubital tunnel. Here, we present a case of a 62-year-old farmer with longstanding diabetes and dyslipidemia who experienced progressive pain and numbness in his medial fingers, exacerbated by elbow flexion. Initially misdiagnosed and treated for Hansen’s disease. Clinico-radiological evaluation revealed a multiloculated cystic lesion in the posteromedial aspect of his left elbow, compressing the ulnar nerve. Surgical excision of the ganglion cyst alleviated symptoms, though residual motor deficits persisted postoperatively. This case underscores the importance of considering ganglion cysts in the differential diagnosis of cubital tunnel syndrome, highlighting the diagnostic challenges and therapeutic considerations in managing this rare etiology.

Progressive pseudorheumatoid dysplasia involving a novel CCN6 mutation: a case report

ObjectiveThis study aims to report a case of progressive pseudorheumatoid dysplasia (PPRD) with two kinds of cellular communication network factor 6 (CCN6) gene mutation. In this paper, the clinical profile and the process of diagnosis were analyzed, and the related literature was reviewed. A 15-year-old boy, who developed progressive ankle and hip joint pain and enlargement with spine involvement, was diagnosed with PPRD. The erythrocyte sedimentation rate and C-reactive protein (CRP) were in the normal range; rheumatoid factor and anti-cyclic citrullinated peptide antibody (ACPA) were all negative. Human leukocyte antigen 27 (HLA-B27) was also negative. Cene study discovered two kinds of mutations in CCN6 gene: c. 802T>C and c.624dup. Radiographic studies revealed spine platyspondyly and shaped beaked, osteoporosis, and bilateral symmetric bony enlargements of the interphalangeal joints. Hip shows bilateral acetabulum and femoral head bone marrow edema, which revealed hip arthritis. Gene detection, laboratory examination, and typical radiographic features are helpful for the diagnosis of PPRD. This is the first report of c. 802T>C and c.624dup mutations in patients with PPRD in our country.

Meningeal Dissemination and Drop Metastasis From Glioma Presenting With Non-Epileptic Myoclonus and Minipolymyoclonus

We describe the case of a 36-year-old woman with a past medical history of low grade right frontal lobe glioma and focal epilepsy presenting with subacute, progressive, multifocal myoclonus and neck and back pain. Unlike her typical seizures, the myoclonus exhibited a distinct semiology, involving both positive and negative muscle jerks affecting multiple limb muscles while sparing the face. In addition, neurological examination revealed low-amplitude, arrhythmic movements of the hands and fingers, resembling minipolymyoclonus. There were no other neurological exam findings, including mental status changes, extrapyramidal signs or signs of myelopathy. Brain and spine MRI indicated leptomeningeal and spinal “drop” enhancing lesions, suggesting likely malignant evolution of the glioma. EEG ruled out a cortical origin of the myoclonus. Pharmacological trials with benzodiazepines and other antiepileptic medications were ineffective. The patient’s myoclonus was most likely spinal segmental in origin from meningeal spread of glioma. The spinal roots or anterior horns of the spinal cord may have represented a focus of hyperexcitability responsible for generating minipolymyoclonus. Our case expands the etiological spectrum of non-epileptic myoclonus and minipolymyoclonus to encompass meningeal carcinomatosis and drop metastases from glioma. These cases may occur even without overt signs of myelopathy. Recognizing such presentations holds significance due to the poor prognosis associated with meningeal spread of glioma and the limited response of non-epileptic myoclonus to symptomatic treatments.

CTRP9 attenuates peripheral nerve injury-induced mechanical allodynia and thermal hyperalgesia through regulating spinal microglial polarization and neuroinflammation mediated by AdipoR1 in male mice

Peripheral nerve injury triggers rapid microglial activation, promoting M1 polarization within the spinal cord, which exacerbates the progression of neuropathic pain. C1q/TNF-related protein 9 (CTRP9), an adiponectin homolog, is known to suppress macrophage activation and exhibit anti-inflammatory properties through the activation of adiponectin receptor 1 (AdipoR1) in various disease contexts. Nevertheless, the involvement of CTRP9 in microglial polarization in the context of neuropathic pain is still unclear. Our study aimed to how CTRP9 influences spinal microglial polarization, neuroinflammation, and pain hypersensitivity, as well as the underlying mechanism, using a neuropathic pain model in male mice with spared nerve injury (SNI) of sciatic nerve. Our findings revealed SNI elevated the spinal CTRP9 and AdipoR1 levels in microglia. Furthermore, intrathecal administration of recombinant CTRP9 (rCTRP9) substantially weakened mechanical hypersensitivity and heat-related pain response triggered by SNI. On the other hand, rCTRP9 mediated a phenotypic switch in microglia, from the pro-inflammatory M1 state to the anti-inflammatory M2 state, by influencing the spinal AMPK/NF-κB mechanism in SNI mice. Additionally, treatment with AdipoR1 siRNA or an AMPK-specific antagonist both reversed the effects of CTRP9 on the phenotypic switching of spinal microglia and pain hypersensitivity. Collectively, these results indicate that CTRP9 ameliorates mechanical hypersensitivity and heat-related pain response, shifted the balance of microglia towards the anti-inflammatory M2 state, and suppresses neuroinflammatory responses by modulating the AMPK/NF-κB pathway, mediated by AdipoR1 activation, in mice with SNI.Graphical

Phenotypic Variability in Camurati–Engelmann Disease: A Case Report of a Family with the c.653G>A Pathogenic Variant in the TGFB1 Gene

Camurati–Engelmann Disease (CED), or Progressive Diaphyseal Dysplasia, is a rare autosomal dominant disorder caused by heterozygous mutations in the TGFB1 Gene, essential for bone regeneration. This study examines the genotype–phenotype relationship in a family diagnosed with CED, specifically focusing on a missense variant (c.653G>A, p.Arg218Cys). The family comprised a mother and her two children, all of whom were found to carry the same disease-causing variant. The second child exhibited severe symptoms by age six, including progressive weakness and joint pain, leading to wheelchair dependency. The mother displayed milder symptoms with preserved independence. The firstborn son, initially asymptomatic, developed gait abnormalities and pain during adolescence. Clinical evaluations revealed characteristic hyperostosis of long bones, with significant variability in symptom onset and severity among family members, potentially indicative of genetic anticipation. This case underscores the importance of genetic testing and interdisciplinary management in CED, as traditional treatments, including corticosteroids and NSAIDs, often yield limited efficacy and notable side effects. Our findings contribute to the understanding of CED’s pathophysiology and highlight the necessity for tailored therapeutic approaches. The identification of the common TGFB1 variant in this family reinforces the critical role of TGFB1 in bone metabolism and suggests avenues for further research into targeted therapies. Such reports enhance awareness and provide valuable insights for healthcare professionals managing rare genetic disorders.

Beneficial Effects of Ginger Root Extract on Pain Behaviors, Inflammation, and Mitochondrial Function in the Colon and Different Brain Regions of Male and Female Neuropathic Rats: A Gut-Brain Axis Study.

Neuroinflammation and mitochondrial dysfunction have been implicated in the progression of neuropathic pain (NP) but can be mitigated by supplementation with gingerol-enriched ginger (GEG). However, the exact benefits of GEG for each sex in treating neuroinflammation and mitochondrial homeostasis in different brain regions and the colon remain to be determined. Evaluate the effects of GEG on emotional/affective pain and spontaneous pain behaviors, neuroinflammation, as well as mitochondria homeostasis in the amygdala, frontal cortex, hippocampus, and colon of male and female rats in the spinal nerve ligation (SNL) NP model. One hundred rats (fifty males and fifty females) were randomly assigned to five groups: sham + vehicle, SNL + vehicle, and SNL with three different GEG doses (200, 400, and 600 mg/kg BW) for 5 weeks. A rat grimace scale and vocalizations were used to assess spontaneous and emotional/affective pain behaviors, respectively. mRNA gene and protein expression levels for tight junction protein, neuroinflammation, mitochondria homeostasis, and oxidative stress were measured in the amygdala, frontal cortex, hippocampus, and colon using qRT-PCR and Western blot (colon). GEG supplementation mitigated spontaneous pain in both male and female rats with NP while decreasing emotional/affective responses only in male NP rats. GEG supplementation increased intestinal integrity (claudin 3) and suppressed neuroinflammation [glial activation (GFAP, CD11b, IBA1) and inflammation (TNFα, NFκB, IL1β)] in the selected brain regions and colon of male and female NP rats. GEG supplementation improved mitochondrial homeostasis [increased biogenesis (TFAM, PGC1α), increased fission (FIS, DRP1), decreased fusion (MFN2, MFN1) and mitophagy (PINK1), and increased Complex III] in the selected brain regions and colon in both sexes. Some GEG dose-response effects in gene expression were observed in NP rats of both sexes. GEG supplementation decreased emotional/affective pain behaviors of males and females via improving gut integrity, suppressing neuroinflammation, and improving mitochondrial homeostasis in the amygdala, frontal cortex, hippocampus, and colon in both male and female SNL rats in an NP model, implicating the gut-brain axis in NP. Sex differences observed in the vocalizations assay may suggest different mechanisms of evoked NP responses in females.

Neurosarcoidosis Masquerading as Spinal Stenosis.

A 65-year-old woman was admitted to the neurology department with a suspected demyelinating disease due to complaints of progressive pain and weakness in both upper and lower limbs, as well as urinary incontinence. MRI of the spine revealed complex disc osteophyte with compression of the spinal cord in the cervical and lumbar spine at several vertebral levels, and localized enhancement in the cervical spine at the site of maximal spinal canal stenosis. During her hospitalization, the patient underwent extensive evaluation to rule out any systematic inflammatory diseases, infections, and malignancy. Chest CT revealed bilateral mediastinal lymphadenopathy. Transbronchial mediastinal lymph node biopsy showed numerous non-necrotizing granulomas without evidence of malignancy. After a thorough and careful exclusion of a demyelinating, infectious, and paraneoplastic myelopathies, and based on clinical, radiographic, and pathological findings, the patient was diagnosed with both neurosarcoidosis and spondylotic myelopathy. She was then treated for neurosarcoidosis, including glucocorticosteroids, azathioprine, and a biosimilar of the anti-TNF alpha agent infliximab, resulting in both clinical and radiographic improvement. Intramedullary spinal neurosarcoidosis is very rare and may present with clinical features of spondylotic myelopathy, with typical imaging findings occurring only in areas of spinal canal stenosis.

Pseudomyogenic Hemangioendothelioma: A Case Report of a Rare Tumor with Unusual Presentation

Abstract Introduction/Objective Pseudomyogenic hemangioendothelioma (PHE) is an extremely rare neoplasm recently categorized by the World Health Organization (WHO) as an intermediate-grade vascular soft tissue tumor affecting young adults. While most documented cases exhibit local aggressiveness, we present a case with unusual presentation characterized by multiple pelvic and femoral lytic bone lesions associated with documented liver metastases. Methods/Case Report A mid-forties female presented with progressive left hip pain persisting for 2 weeks. Radiological assessments showed multiple lytic lesions in the left superior pubic ramus, right iliac wing, and left greater trochanter with associated pathologic fractures and soft tissue involvement. Additionally, multiple indeterminate hepatic and splenic hypodensities were noted. Histologic examination of the bone and liver biopsies revealed a neoplasm composed of spindled and epithelioid cells with hyperchromatic nuclei and eosinophilic cytoplasm. The neoplastic cells are arranged haphazardly and in short fascicles within a fibromyxoid stroma or sinusoidal spaces (in the liver) associated with scattered acute and chronic inflammation. Frequent mitoses are noted; however, no necrosis was identified. Tumor cells were positive for CD31, ERG, FOSB with equivocal staining for pankeratin and CK8/18. The top differential consideration was epithelioid hemangioendothelioma (EHE); however, sarcoma fusion next-generation sequencing panel exhibited features of a vascular neoplasm with WWTR1-FOSB, favoring PHE. Results (if a Case Study enter NA) NA Conclusion Pseudomyogenic hemangioendothelioma is a rare tumor that is prone for local recurrence and usually affect young adults (mean age: 30 years). Metastases are uncommon. To the best of our knowledge, we report a case of PHE with unusual presentation of early metastases to liver and possibly the spleen and highlights the complexity of PHE diagnosis.

Case Report : Undifferentiated Synovial Sarcoma Presenting as a Large Thoracic Mass: A Rare and Challenging Diagnosis

Background: Undifferentiated synovial sarcoma (USS) is a rare and aggressive subtype of soft tissue sarcoma. Its presentation as a large thoracic mass poses significant diagnostic and therapeutic challenges.  Case Presentation: A 34-year-old male presented with a three-month history of progressive chest pain and hemoptysis. Imaging studies revealed a large, cystic thoracic mass with pleural effusion. Initial serological testing suggested hydatid cyst, but subsequent testing revealed negative Echinococcus (Hydatid) IgG antibodies.  Diagnostic and Therapeutic Interventions: Repeat fine-needle aspiration cytology (FNAC) was deferred due to revised ultrasound findings indicating a solid fibrocystic vascular lesion. Bronchial artery embolization (BAE) was performed to control life- threatening hemorrhage. Thoracotomy and en-bloc mass excision were subsequently undertaken.  Histopathological Diagnosis: Histopathological examination of the resected specimen revealed undifferentiated synovial sarcoma and spindle cell sarcoma.  Clinical Implications: This case highlights the importance of correlating imaging and serological findings, managing vascular complications, and the efficacy of BAE in controlling hemorrhage. The significance of thoracotomy and mass excision in achieving diagnostic clarity and therapeutic success is underscored.  Conclusion: Undifferentiated synovial sarcoma presenting as a large thoracic mass poses significant diagnostic and therapeutic challenges. A multidisciplinary approach, incorporating imaging, serological testing, and surgical intervention, is crucial for achieving optimal patient outcomes.

Congenital Appendiceal Atresia: An Extremely Rare Disorder Presenting with Acute Abdomen

Abstract Introduction/Objective Congenital malformations of the appendix are extremely rare and difficult to diagnose preoperatively. One of those anomalies include congenital appendiceal atresia (CAA) which mimics acute appendicitis and is usually ignored in the differentials of acute abdomen. Herein, we present a case study of congenital appendiceal atresia associated with periappendicitis. Methods/Case Report A 62-year-old female with past history of diverticulitis presented with complaints of crampy, diffuse and progressive lower abdominal pain and nausea for two days. She denied fevers, chills or change in bowel movements. Physical examination revealed vitally stable patient with soft, non-distended and tender abdomen in bilateral lower quadrants, worse on the right side and showing positive rebound test. Her complete blood count and chemistry were within normal limits. A computerized tomography arterial portography (CTAP) scan with contrast demonstrated inflammatory stranding around the appendix without abscess, concerning for acute appendicitis. She then underwent laparoscopic appendectomy. Grossly, the appendix consisted of a cylindrical portion of yellow-pink fatty tissue covered by peritoneum measuring 6.2 x 3.3 x 0.5 cm. The peritoneal surface displayed patchy fibrinous exudates. Sectioning revealed yellow adipose tissue with no identifiable appendix. Microscopically, the entire specimen demonstrated a poorly formed, mainly fatty appendix without a discernible lumen or submucosal lymphoid tissue. There were scattered sparsely distributed non-continuous bundles of smooth muscle (muscularis propria) at the periphery, which were highlighted by desmin and trichrome stains. This atretic appendix, mesoappendix and peritoneal covering were acutely inflamed. The final diagnosis was poorly formed, acutely inflamed fatty appendix consistent with congenital appendiceal atresia with accompanying acute periappendicitis. Results (if a Case Study enter NA) NA Conclusion This case would add to the very limited existing literature for congenital appendiceal anomalies and highlight the importance of thorough surgical intraoperative abdominal exploration and submission of the entire specimen for microscopic examination in order to render a definitive diagnosis.

Intravascular Nodular Fasciitis Mimicking Thrombotic Complications in a 16-year-old male with May-Thurner Syndrome

Abstract Introduction/Objective Nodular fasciitis is a rare benign myofibroblastic proliferation commonly mistaken for malignant tumors due to its rapid growth and clinical presentation. May-Thurner Syndrome (MTS), characterized by compression of the left common iliac vein by the right common iliac artery, predisposes individuals to deep vein thrombosis (DVT). However, here we present a unique case of intravascular nodular fasciitis presenting as thrombotic complications in the context of MTS in a 16-year-old male. Methods/Case Report A 16-year-old male presented with progressive left hip and leg pain accompanied by recent swelling. Ultrasound revealed occlusive thrombi within the left side external iliac, common superficial femoral, and great saphenous veins, along with nonocclusive thrombi in left deep femoral vein. Magnetic resonance venography confirmed MTS and extensive thrombosis. Thrombectomy was done with fragments sent to pathology. Microscopic examination showed cellular areas with loose myxoid composition, proliferating spindle, myofibroblastic/fibroblastic, and stellate cells arranged in irregular bundles. Additional microscopic findings included mild cellular atypia, mitoses, focal macrophage aggregates, occasional multinucleate forms, foci of hemorrhage, fibrin exudate, ingrowing fibroblasts, and increased vascularization. Immunohistochemical stains demonstrated positivity for SMA, CD31, ERG, and CD34, highlighting prominent vascularization, while desmin, myogenin, myoglobin, STAT6, beta-catenin, S100, and AE1/AE3 were negative. Differential diagnosis included intravascular fasciitis, and possibly organizing thrombus with myxoid stroma. USP6 gene rearrangement (FISH) study was positive, confirming the diagnosis of nodular fasciitis. Results (if a Case Study enter NA) N/A Conclusion This case emphasizes the significance of maintaining a broad differential diagnosis when assessing specimens sent as thrombectomies. Recognizing intravascular nodular fasciitis as a potential presentation mimicking thrombosis is essential for accurate diagnosis and management.

Sonographic Verification and Clinical Significance of the Features of Surrounding Structures in Knee Osteoarthritis

Background: Osteoarthritis (OA) of the knee joint is one of the most dangerous diseases, the most significant manifestation of which is chronic pain syndrome. There is a low correlation between the radiographic stage of OA and pain progression. First of all, this is a variety of pain syndromes, among which is the pathology of periarticular structures (AS). To date, the contribution of this type to the clinical picture of OA has been sufficiently studied, primarily due to the fact that the list and frequency of these lesions have not yet been described.Aim: to describe the basis of the main periarticular structures in patients with different radiographic stages of knee OA according to ultrasound data.Materials and methods: The observational study has currently included 88 patients who had an outpatient appointment with a rheumatologist for knee osteoarthritis between 2021 and 2023. The study assessed 110 knee joints using clinical and ultrasound techniques.Results: The most common periarticular ultrasound changes, regardless of radiographic stage, were pes anserine tendinopathy (57.3 %), Baker’s cyst (45.5 %), fibrosis of the severe Hoffa body (40 %) and ligamentopathy of the medial collateral ligament (36). ,4). %). A significant correlation was found between the number of changes according to ultrasound and the radiological stage (ρ=0.45 [95 % CI: 0.28, 0.59], p <0.001) as well as between the VAS and the definition of identified ultrasound changes (ρ= 0.29 [95 % CI: 0.11, p=0.002); In addition, it was shown that late stages of OA are associated with a greater content of changes (p <0.001).Conclusion: Changes in periarticular structures are present in most patients with knee osteoarthritis; their number correlates with the VAS value and radiographic stage of OA. Details of these changes, their clinical significance and pathogenetic contribution to the progression of knee OA require further study.

Enhanced joint pain in diabetic patients with knee osteoarthritis is associated with increased synovitis, synovial immune cell infiltration, and erythrocyte extravasation.

Diabetes mellitus (DM) is an important risk factor for the development of osteoarthritis (OA), increasing OA progression and OA pain. To gain insight into the underlying mechanisms of how DM exacerbates OA processes and OA pain, this study analyzed histological differences of synovial tissues from non-DM and DM patients with OA and correlated these differences with knee pain severity. Synovial tissue was obtained from 12 non-DM and 10 DM patients with advanced knee OA who underwent total knee arthroplasty. Synovial inflammation was assessed using the Synovitis score developed by Krenn. The Knee Injury and Osteoarthritis Outcome Score (KOOS) was used to assess knee pain intensity and disability in OA patients. The number of mast cells, macrophages, nerve fibers, capillaries, larger vessels and erythrocyte extravasation were analyzed microscopically in histological and immunostained synovial sections from non-DM and DM patients. Association analyses were performed to determine associations between OA knee pain and synovial changes affected by DM. Synovial tissue from OA patients with DM had a higher synovitis score, more erythrocyte extravasation, and contained higher numbers of mast cells and macrophages compared to non-DM patients. The number of capillaries and vessels in the lining/sublining layer of the synovial tissue was reduced in DM patients. OA patients with DM had more severe knee pain compared to non-DM patients. The KOOS pain score was associated with the synovitis score, the number of tissue macrophages, and the number of mast cells in the synovial tissue (adjusted for age, sex, and BMI). In addition, the erythrocyte extravasation score was associated with the KOOS pain score and with the synovitis score. The study suggests that increased OA progression and pain severity in patients with DM result from more pronounced synovitis and synovial vascular leakage and increased infiltration of macrophages and mast cells.