BACKGROUND Giant cell arteritis (GCA) is the most common large-vessel vasculitis in individuals over age 50 years. Although it typically affects extracranial branches of the carotid artery, central nervous system involvement is rare and can manifest with ischemic stroke. Links between GCA and malignancy have been reported, especially hematologic cancers, but paraneoplastic GCA associated with solid tumors remains exceptional and poorly understood. CASE REPORT We describe a 59-year-old man presenting with temporal headaches, right-hand paresthesia, monocular visual loss, and gait instability. Imaging revealed left thalamic ischemia and bilateral vertebral artery stenosis without atherosclerosis. Examination and Doppler ultrasound supported a diagnosis of GCA with associated polymyalgia rheumatica (PMR), although a temporal artery biopsy was negative. Inflammatory markers were only moderately elevated, consistent with reports of GCA cases with ischemic complications. PET-CT incidentally identified a mediastinal mass, confirmed as small-cell pulmonary neuroendocrine carcinoma. Symptoms improved with corticosteroids, but tapering below 15 mg/day caused relapse, requiring methotrexate. Despite partial oncologic response, brain metastases appeared 8 months later. Follow-up vascular imaging showed regression of arterial stenoses under corticosteroids, arguing against atherosclerosis and supporting the inflammatory nature of the lesions. GCA manifestations remained corticosteroid-dependent throughout follow-up. CONCLUSIONS The simultaneity of GCA and lung neuroendocrine carcinoma, persistence of corticosteroid dependence, and lack of remission despite oncologic response strongly suggest a paraneoplastic mechanism. This case emphasizes the need for heightened suspicion of underlying cancer in patients with atypical, biopsy-negative, or treatment-resistant vasculitis. Recognition of paraneoplastic GCA may improve early cancer detection and influence management decisions at the intersection of rheumatology and oncology.

Glucocorticoid therapy is prescribed for a variety of inflammatory conditions and is associated with severe adverse effects. A glucocorticoid withdrawal syndrome (GWS) may occur after prolonged glucocorticoid treatment-with or without biochemical glucocorticoid-induced adrenal insufficiency (GIAI). Previously, GWS was not considered an entity, probably due to the overlap between symptoms of GWS and GIAI. The Addison's disease-specific quality of life questionnaire (AddiQoL-30) is a validated tool for quantifying symptoms of adrenal insufficiency resembling GWS. In the present study, we test the hypothesis that patients with a low AddiQoL-30 score and/or low cortisol response to a short Synacthen test (SST), after cessation of prednisolone treatment, may benefit from low-dose hydrocortisone therapy without increasing the risk of metabolic and cardiovascular disease during prolonged cortisol exposure. REPLACE is a multi-centre, double-blinded, placebo-controlled randomised controlled trial in patients with polymyalgia rheumatica or giant cell arteritis after cessation of prednisolone treatment. Criteria for randomisation are an AddiQoL-30 score ≤85 and/or plasma cortisol response to SST, 30-min p-cortisol >100 and <420 nmol/L. Patients will be randomised to oral hydrocortisone (10 mg two times a day) or placebo for 16 weeks. Baseline and follow-up examinations comprise AddiQoL-30 questionnaire, SST, blood samples, standardised blood pressure, physical function tests and assessment of bone quality and body composition. At baseline, two comparator groups include: (1) patients with a SST-stimulated cortisol ≥420 nmol/L and AddiQoL-30 score >85; and (2) patients with a SST-stimulated cortisol ≤100 nmol/L. The study is conducted in accordance with the Declaration of Helsinki, registered at the Clinical Trials Information System (CTIS: 2024-513822-53-00) and Clinicaltrials.gov (NCT05193396), and publications will be in accordance with the recommendations of the International Committee of Medical Journal Editors. The trial is monitored by local independent Good Clinical Practice units and overseen by the Danish Data Protection Agency (journal no. 21/27119), the Regional Committees on Health Research Ethics for Southern Denmark (project ID: S-20210076), the Danish Patient Safety Authority and the Danish Medicines Agency. NCT05193396.

Gastrointestinal basidiobolomycosis is a rare fungal infection caused by Basidiobolus ranarum, increasingly recognized as an intestinal pathogen in children. Its clinical features closely mimic malignancy or inflammatory bowel disease, leading to frequent diagnostic delays. Although endemic in regions such as Saudi Arabia, Iran, and Oman, sporadic cases occur worldwide. Amphotericin B-liposomal remains a cornerstone therapy for invasive fungal disease, but no standardized treatment protocol for pediatric gastrointestinal basidiobolomycosis has been established. We report a 6-year-old Asian girl, born preterm with very low birth weight, who presented with persistent abdominal pain without organomegaly, alternating diarrhea and constipation, fever, and anorexia. Initial ultrasonography revealed colonic wall thickening with aneurysmal dilation at the splenic flexure. Colon biopsy confirmed Basidiobolus ranarum. The patient received intravenous liposomal amphotericin B (90 mg daily), with partial radiologic improvement but persistent symptoms, prompting exploratory laparotomy. Adhesiolysis, left hemicolectomy, and resection of jejunal and colonic masses were performed. Histopathology demonstrated dense eosinophil-rich granulomatous inflammation with multinucleated giant cells and a solitary fungal hypha, without classic Splendore-Hoeppli phenomenon, likely due to prior antifungal therapy. Postoperatively, the patient was discharged on oral itraconazole for 28 days with adjunctive cotrimoxazole. At 1-year follow-up, she remained asymptomatic with no evidence of recurrence. This case highlights the diagnostic challenges of pediatric gastrointestinal basidiobolomycosis and demonstrates successful management with liposomal amphotericin B followed by itraconazole. It underscores the ongoing gap in establishing standardized treatment and supports further prospective studies to define optimal antifungal and surgical strategies for this rare entity.

Proteins secreted from a mouth stylet of sedentary plant-parasitic root-knot nematodes self-polymerize to form a unique feeding tube structure within host cells modified into giant feeding cells by the nematode. Feeding tubes have essential functions as they complex with the host endomembrane system for nutrient uptake to sustain parasitism. Despite their significance, they remain one of the least understood aspects of nematode parasitism of plants. Their small size and location within giant-cells deeply embedded within galls encasing adult females has prohibited studies to isolate and discern their molecular composition. Here, we developed a protocol for the isolation and semipurification of root-knot nematode feeding tubes from giant-cell cytoplasm of several host plant species to provide a unique view of these structures at the light and scanning electron microscopy level revealing previously undescribed features of their structure. Our methods allowed for the isolation and solubilization of sufficient quantities of enriched feeding tubes enabling a comparative proteome analysis across host species that identified proteins with an increased likelihood to function in feeding tube formation. A comparison across root-knot nematode species further narrowed candidates to a conserved class of secretory proteins that specifically localized within secretory granules of the dorsal gland of adult females and in feeding tubes formed within host cell cytoplasm to unequivocally demonstrate these proteins as core components of feeding tubes. Our finding gives scientists a look into the protein composition of feeding tubes opening the door to a better understanding of their structure and function in nematode parasitism.

Weeds, especially purple nutsedge, are not just alternative hosts but highly susceptible host that drive the persistence and spread of rice root-knot nematodes. This study provides a comprehensive assessment of the host status of eight plant species, including rice and common rice-associated weeds, to the root-knot nematode Meloidogyne graminicola. By integrating quantitative infection assays with confocal laser scanning microscopy, we combined whole-plant measurements of nematode development with cellular-level visualization of feeding-site structures to characterize host suitability more precisely. The results revealed a continuum of responses ranging from weakly supportive to highly susceptible hosts. Purple nutsedge (Cyperus rotundus) showed the highest susceptibility under controlled conditions, with a reproduction factor (Rf = 77.25) exceeding that of rice (Oryza sativa, Rf = 15.45) and jungle rice (Echinochloa colona, Rf = 19.81). Digitaria sanguinalis also supported considerable nematode multiplication (Rf = 10.92). Confocal imaging provided temporal snapshots of feeding-site formation, giant cell development and gall progression in C. rotundus, complementing the quantitative findings. Several species, including Glinus oppositifolius and Stellaria media, supported minimal development, indicating limited suitability as hosts. Overall, the study demonstrates that multiple weeds commonly present in rice ecosystems can sustain M. graminicola development to varying degrees under experimental conditions. These results highlight the importance of considering weed species when evaluating nematode population dynamics and designing integrated management strategies for rice-based agroecosystems.

Activin-A has dual roles in osteoclast formation and foreign body giant cell differentiation from human CD14+ monocytes.

Tenosynovial giant cell tumor (TGCT), formerly pigmented villonodular synovitis (PVNS), may extend into the popliteal fossa and mimic a Baker cyst, complicating diagnosis and operative planning. A 53-year-old former endurance runner presented with posterior knee pain, swelling, intermittent locking, and a palpable popliteal mass. Examination demonstrated effusion and limited range of motion; weightbearing radiographs showed mild osteoarthritic change with effusion, and magnetic resonance imaging (MRI) revealed synovial hypertrophy with low T2 signal suggestive of hemosiderin deposition. Arthroscopic synovectomy via 4 anterior and 2 posterior portals was performed; histology confirmed diffuse, nonmalignant TGCT. Given persistent posterior disease, a staged posterior open approach (lazy-S incision) enabled en bloc excision of the extra-articular component. Adjuvant radiosynoviorthesis (intra-articular yttrium-90) was administered. At 8-year follow-up, there was no clinical or radiographic evidence of disease. This case emphasizes MRI recognition of hemosiderin-related low T2 signal, the value of combining arthroscopic synovectomy with posterior open excision in diffuse disease with popliteal extension, and selective use of adjuvant radiosynoviorthesis for durability.

Maxillary sinus floor elevation is a well-established procedure for increasing bone volume in the posterior maxilla, yet the regenerative outcome depends strongly on the choice of grafting material. This clinical study compared a high-temperature sintered xenograft (Bio-Oss®) and a low-temperature processed xenograft (Ti-Oss®) with regard to their regenerative and immunological profiles. Eight patients underwent split-mouth sinus augmentation with both materials, and biopsies were retrieved at 6 months after implant placement. Histological, histomorphometrical, and immunohistochemical analyses were performed to assess bone formation, material resorption, and tissue compatibility. Immunohistochemistry was applied to evaluate the local immune response, focusing on macrophage polarization and multinucleated giant cell activity. Quantitative histomorphometry determined the relative areas of newly formed bone, residual graft material, and connective tissue. Histopathological and histomorphometrical analyses demonstrated comparable levels of new bone formation in both groups, confirming reliable osteoconduction. Immunohistochemical evaluation revealed tartrate-resistant acid phosphatase isoform 5a (TRAP5A) expression in multinucleated giant cells adherent to both materials. Interestingly, Bio-Oss® induced a higher proportion of anti-inflammatory (CD163+) macrophages, whereas Ti-Oss® triggered a significantly greater number of pro-inflammatory multinucleated giant cells (CD11c+). These findings indicate that although both xenogeneic substitutes (Bio-Oss® and Ti-Oss®) achieve bone regeneration, they elicit distinct immune responses, which may influence long-term remodeling and graft integration. Consideration of osteoimmunological properties is therefore essential when selecting biomaterials for clinical sinus augmentation.

Immunohistochemical analysis of immune checkpoint proteins (PD-1, PD-L1 and PD-L2) in giant cell granulomas of the jaws and giant cell tumor of bone.

Microenvironmental niches dictate divergent fibroblast fates in reversible versus progressive lung fibrosis.

Is there histological evidence supporting capsulectomy in patients with breast implant illness: A case-control study.

Angioleiomyoma is a benign, pericytic (perivascular) neoplasm that most frequently arises in the dermis or subcutis of the lower extremities. Extensive calcification is extremely uncommon in this condition. An 80-year-old woman presented with a 20-year history of a slowly growing, painless mass in the dorsomedial aspect of the left great toe. Physical examination revealed a 2.5-cm, firm, mobile, non-tender mass. Radiographs showed a well-demarcated, densely calcified mass. Computed tomography confirmed the presence of a calcified lesion without bone involvement. Magnetic resonance imaging (MRI) exhibited a well-defined mass with intermediate signal intensity on T1-weightwed sequences and high signal intensity on T2-weighted sequences. Peripheral low signal intensity areas correlating to the calcified portion of the mass were also observed on both T1- and T2-weighted sequences. Contrast-enhanced MRI demonstrated intense, relatively homogeneous enhancement in the non-calcified portion of the mass. The patient underwent an excisional biopsy of the lesion. Histologically, the lesion is composed of bundles of bland, well-differentiated smooth muscle cells with small slit-like vascular channels. In addition, calcium crystal deposits surrounded by aggregates of epithelioid histiocytes and multinucleated giant cells were observed. The patient had no evidence of local recurrence at the latest follow-up. This unique case provides valuable insights into the understanding and treatment of acral calcified angioleiomyoma. Knowledge of this peculiar neoplasm is important because it can mimic a variety of benign and malignant soft-tissue tumors.

Tenosynovial giant cell tumor (TGCT) is a locally aggressive neoplasm associated with limited range of motion (ROM), stiffness, joint damage, pain, and reduced physical functioning (PF). The MOTION Phase 3 trial (NCT05059262) was a randomized, placebo-controlled, double-blind study of vimseltinib among patients with TGCT. The objective of the current study was to define meaningful changes in clinical outcome assessments (COAs) measuring active ROM, PF, and stiffness using qualitative and quantitative data from patients in the MOTION trial. Embedded exit interviews with patients in MOTION were conducted to explore meaningful changes in Patient Global Impression of Change (PGIC) anchors, active ROM, Patient-Reported Outcomes Measurement Information System (PROMIS)-PF, and Worst Stiffness numeric rating scale (NRS). Anchor- and distribution-based analyses of the MOTION data, informed by the exit interviews, were used to define responder thresholds. In the MOTION trial, 96/123 patients (78%) completed an exit interview. Most considered "minimally improved" responses for each question (PGIC-PF: 67%; PGIC-ROM 73%) as meaningful. Responder estimates ranged from 1.45 to 4.9 (PROMIS-PF), from 6.0 to 14.8 (active ROM), and from - 2.3 to - 0.5 (Stiffness). The cumulative distribution function curves show a clear separation between treatment groups at a wide range of values around the proposed thresholds. The responder definitions were at least a 3-point improvement for PROMIS-PF, a 10% improvement for active ROM, and a 2-point improvement for the Worst Stiffness NRS. Qualitative interviews facilitate integrating the patient perspective in the selection of anchors and defining meaningful change.

ABSTRACT Background and Aims A retrospective analysis was conducted on the clinical, pathological, and imaging features of 471 cases of pilomatrixoma, aiming to enhance clinicians' understanding of pilomatrixoma. Methods A total of 471 cases of pilomatrixoma diagnosed and surgically treated in the Affiliated Hospital of Southwest Medical University from August 1999 to July 2024 were retrospectively analyzed. Some cases were also diagnosed through preoperative fine needle aspiration, as well as ultrasonography, computed tomography (CT), and magnetic resonance imaging (MRI). Results Of these 471 patients, 224 (47.6%) were male, and 247 (52.4%) were female. Their ages ranged from 8 months to 90 years, with an average age of 18.2 years. The highest incidence of pilomatrixoma was observed in patients aged between 0 and 10 years, accounting for 46.5% (219/471). The most common site of pilomatrixoma is the face, followed by the neck. The most common area of the face is the parotid gland region, accounting for 24.8% (61/246) of facial cases. Histology and cytopathology revealed that the tumor cells usually consisted of basaloid cells and eosinophilic shadow cells, and inflammatory cells, foreign body giant cells, and calcifications were seen. Additionally, the ultrasonography showed a mass with calcification, a strong echo band, and parenchyma without calcification. The pilomatrixoma should be considered when the tumor presents as soft‐tissue density with varying degrees of calcification, well‐defined oval or circular nodules on CT, and characteristic reticular and annular hypersignal on T2WI and T2WI + FS on MRI. Conclusion Pilomatrixoma is commonly found on the face and neck of adolescent females. Cytopathology, ultrasound, and imaging features can provide clinical clues, and histopathology can make the final diagnosis. Complete surgical resection is the preferred treatment.

Giant cell arteritis-polymyalgia rheumatica spectrum disease (GPSD): Relation with neoplasms and possible role as a paraneoplastic syndrome

Benign bone tumors such as chondroblastoma, giant cell tumors (GCT), and aneurysmal bone cysts (ABC) are rare but clinically significant lesions that frequently occur in the epiphyseal regions of long bones, particularly near load-bearing joints in children and young adults. These tumors compromise the structural integrity of bone, leading to an elevated risk of pathologic fracture. Traditional methods for estimating fracture risk rely on simple geometric thresholds and volumetric ratios, but they fail to account for patient-specific differences in bone geometry, material heterogeneity, and physiological loading conditions. As a result, risk is often misclassified, which may lead to either overtreatment or missed prevention opportunities. To address this limitation, this study presents a preliminary demonstration of computed tomography-based finite element analysis (CTFEA) as a novel alternative method (NAM); computational framework using patient-specific CTFEA to evaluate fracture risk in four patients with benign knee tumors. Clinical computed tomography (CT) imaging and motion capture-informed joint loading were used to develop anatomically accurate, mechanically calibrated models incorporating nonlinear bone behavior. CTFEA simulations focused on walking, jogging, and partial weight-bearing conditions, captured localized stress and strain distributions, and were benchmarked against clinical and volumetric assessment criteria. CTFEA outperformed traditional methods by revealing mechanical vulnerabilities, including in cases classified as low-risk clinically, through its ability to simulate individualized loading scenarios. These findings demonstrate the feasibility and potential of CTFEA as a noninvasive, patient-specific alternative to animal or oversimplified models, with direct implications for preoperative planning and fracture risk stratification in orthopedic surgery.

Pulmonary artery aneurysms (PAAs) are a rare pathology with potentially devastating consequences. In this case series, we describe two cases of clinically isolated pulmonary arteritis and concisely review the literature surrounding this new clinical entity. Two Caucasian women, aged 65 and 73, were referred to our cardiovascular surgery center with incidental PAAs, both growing to over 65 mm in main pulmonary artery diameter. Both participants underwent serial thoracic CT-angiograms to monitor the pulmonary artery aneurysms. Both pulmonary artery aneurysms showed an interval increase in diameter, so they were repaired surgically. The main pulmonary artery was excised in both cases and replaced with a synthetic graft. No pre- or postoperative corticosteroids were administered. Neither of our patients had systemic symptoms of giant cell arteritis or elevated inflammatory markers. In both cases, only the main pulmonary artery was replaced to minimize surgical complexity and risk; however, the left pulmonary artery of one patient began to further dilate 5 years postoperatively. Currently, no reintervention is planned. These cases represent mounting evidence for the existence of clinically isolated pulmonary arteritis leading to a pulmonary artery aneurysm. This entity is analogous to clinically isolated aortitis and may represent a subclinical smoldering vasculitis. These rare aneurysms can progress over time and require surgical intervention. In the absence of guidelines for pulmonary artery aneurysm intervention, the present cases will serve to guide further management of this rare disease.

Large vessel vasculitis without aortitis in giant cell arteritis: About 4 cases

The diagnostic utility of SATB2 immunohistochemistry as an adjunct for differentiating osteogenic from non-osteogenic bone tumors: A systematic review and Meta-analysis.

Context. Undifferentiated carcinomas with osteoclast-like giant cells of the pancreaticobiliary tract (UCOGCs) are rare but distinctive tumors with limited literature. Objective. To study the clinicopathologic characteristics of UCOGCs including morphology, immunohistochemistry (IHC), management, and survival outcomes. Design. Assessment of 12 patients of UCOGC found over 10 years from a tertiary care oncology center database. Results. The mean age at diagnosis was 54.8 years (35-69 years) with a striking men:women ratio of 1:1.5. Eleven of 12 tumors (91%) involved the pancreas, while 1 was in the gallbladder fossa (8%). The mean tumor size was 8.8 cm (range: 4.2-18 cm). Fifty-eight percent showed metastasis at presentation, most commonly to the liver (57%). On microscopy, epithelioid-predominant histology was seen in 91%; 5 of these had an additional spindle/sarcomatoid morphology, while 1 tumor showed pure sarcomatoid histology. By immunohistochemistry, keratins were expressed in the neoplastic mononuclear cells (6 of 9, 67%). In terms of management, 2 of 11 (18%) patients underwent surgery alone; 1 of 11 (9%) received neoadjuvant chemoradiation, followed by surgery and adjuvant chemotherapy; 2 of 11 (18%) underwent surgery, followed by adjuvant chemotherapy; 4 of 11 (36%) received chemotherapy alone; and 2 of 11 (18%) received palliative care. On follow-up, 9 of 12 patients succumbed within a year of diagnosis (75%). The 6-month overall survival (OS) was 44%, and the mean survival period was 5 months (range: 8 days-1 year). Conclusions. UCOGCs are rare, aggressive variants of carcinoma and ought to be classified separately. Owing to their unique etiopathogenetic mechanisms, further exploration and large studies are required to devise newer, more effective therapeutic regimes.