Background and Clinical Significance: Local recurrence of extremity leiomyosarcoma after multiple operations and radiotherapy presents a major therapeutic challenge. Adequate oncologic clearance must be balanced against preservation of skeletal stability and reliable wound coverage within a scarred and poorly vascularized tissue bed; Case presentation: A 78-year-old woman presented with a painful recurrent leiomyosarcoma of the anterior proximal third of the right leg after three previous operations and adjuvant radiotherapy. Preoperative magnetic resonance imaging demonstrated a recurrent soft-tissue lesion extending to the anterior surface of the proximal tibia. Staging computed tomography of the chest, abdomen, and pelvis showed no distant metastatic disease before definitive surgery. A one-stage limb-sparing procedure was performed, including resection of the recurrent tumor bed and an approximately 7-cm anterior cortical lamella of the proximal tibia containing an area considered suspicious for neoplastic involvement. A locking tibial plate was applied prophylactically to reduce the risk of pathological fracture. The approximately 12 × 10-cm soft-tissue defect was covered with a muscle flap from the medial head of the gastrocnemius and a free skin graft harvested from the paraumbilical region. Gross pathological examination identified an 8-cm subcutaneous tumor formation. Histopathological examination demonstrated well-differentiated leiomyosarcoma; the spindle cells expressed vimentin, actin, and desmin. All examined resection lines were free of tumor infiltration. No pathogenic microorganism was isolated, and no early postoperative complication was documented. The patient mobilized with walking aids and protected weight bearing for 30 days. At approximately 16 months, the flap and skin graft provided stable coverage, no postoperative tibial fracture or clinically apparent implant-related complication had occurred, and the patient was independently ambulatory. Postoperative MRI demonstrated expected postoperative changes and a small indeterminate subcutaneous focus without diffusion restriction, requiring continued surveillance; Conclusions: This case illustrates the feasibility of combining oncologic resection, prophylactic tibial stabilization, and vascularized soft-tissue reconstruction in a previously irradiated extremity. The documented early functional and reconstructive outcome supports this individualized limb-sparing approach, although longer oncologic surveillance is required.
Background: Pemphigus is a rare group of autoimmune blistering diseases characterized by autoantibody-mediated loss of keratinocyte adhesion, resulting in intraepithelial blister formation involving the skin and mucous membranes. Pemphigus Vulgaris (PV) is the most common type of pemphigus, which usually presents in adulthood and has a prevalence rate of about 2.83 cases per million person years worldwide. The prevalence rate in children and adolescents is relatively uncommon; however, it accounts for about 1.4 to 3.7 percent of all cases of pemphigus vulgaris reported worldwide. The involvement of oral cavity is clinically significant since it can present prior to the disease or as its predominant feature. Hence, this narrative review seeks to review literature on pemphigus with involvement of oral cavity in children and adolescents. Objective: The objective of this study was to review the current literature regarding epidemiology, pathogenesis, clinical presentation, diagnosis, histopathological characteristics, treatment, and outcomes of pemphigus with oral involvement in children and adolescents. Materials and Methods: A literature search was conducted using the PubMed/MEDLINE, Scopus, and Cochrane Library databases to identify relevant studies published between 2015 and 2026. The search strategy included terms related to pemphigus, pediatric patients, and oral manifestations. Articles involving patients younger than 18 years of age with oral involvement were screened according to predefined inclusion criteria. Following database screening, duplicate removal, and full-text assessment, 25 studies comprising a total of 51 patients with documented oral or orofacial involvement were included in the final review. Results: Pemphigus vulgaris was the predominant subtype, with oral lesions representing the initial or sole manifestation in the majority of patients. The gingiva, mucosa, tongue, lip, and palate were the most common affected areas in the mouth. The lesions in these areas usually appeared as painful erosion, ulcers, and desquamative gingivitis. Histopathology with the presence of acantholysis in the suprabasal area and direct immunofluorescence was the most definitive test for diagnosis. Systemic corticosteroids were the mainstay of treatment in conjunction with steroid sparing agents in some cases, with good results in difficult cases using rituximab. Overall, most patients achieved partial or complete clinical remission following appropriate treatment, although relapses were occasionally reported. Conclusions: Pemphigus in children and adolescents is rarely encountered; however, it should be included as a differential diagnosis of erosive/ulcerative lesions. Early identification, diagnosis, and treatment through a collaborative approach from dental practitioners are critical. Further research is required at multiple centers to gain a better understanding of the disease and to develop evidence-based guidelines for diagnosis and treatment of pediatric patients.
Background: Oral cavity cancer is a clinically relevant subgroup of head and neck malignancies and is associated with substantial treatment burden and healthcare utilization. Hospital length of stay (LOS) is an important indicator of inpatient resource use and complexity of care, yet large multicenter data from Germany are limited. Methods: This retrospective multicenter analysis used anonymized inpatient administrative data from 49 German hospitals; eligible oral cavity cancer hospitalizations were contributed by 34 of these hospitals. Adult inpatient hospitalizations (≥18 years) with malignant neoplasms of the oral cavity, defined using ICD-10-GM codes C00–C06, recorded between January 1 2019 and 31 December 2024 were included. The primary outcome was hospital LOS in days. Multimorbidity was quantified using the van Walraven-weighted Elixhauser Comorbidity Score. Prolonged hospitalization was defined as LOS ≥ 7 days and LOS ≥ 14 days. Associations between demographic, clinical, and treatment-related variables and LOS were examined using multivariable Poisson regression models. Because overdispersion was present, a negative binomial mixed model was additionally fitted as a sensitivity analysis. To account for inter-hospital variability, hospital was included as a random intercept in all multivariable models. Associations with prolonged LOS were analyzed using multivariable logistic regression models. All analyses were performed at the hospitalization level. Results: A total of 3957 inpatient hospitalizations for oral cavity cancer were included. Mean age was 65.6 years, and 66.2% of hospitalizations involved male patients. The median LOS was 6 days (interquartile range [IQR] 3–13; mean 10.2 days, standard deviation 11.8). Overall, 49.4% of hospitalizations had an LOS ≥ 7 days and 23.5% had an LOS ≥ 14 days. Older age, particularly >80 years, and higher comorbidity burden were associated with longer LOS (adjusted Poisson rate ratio [RR] for age > 80 years 1.17, 95% CI 1.13–1.21; high comorbidity burden RR 1.58, 95% CI 1.54–1.63). Several treatment-related variables, including surgical procedures in the oral and facial region, lymphatic system operations, blood transfusions, and complex intensive care treatment, were associated with prolonged hospitalization (e.g., blood transfusion RR 1.80, 95% CI 1.76–1.85; complex intensive care RR 1.70, 95% CI 1.65–1.75). Chemotherapy-related hospitalizations were associated with shorter LOS. Conclusions: LOS varied substantially across inpatient hospitalizations for oral cavity cancer in Germany. Older age, higher comorbidity burden, and markers of more complex inpatient treatment were associated with extended hospital stay. These findings may help identify hospitalizations at increased risk of prolonged LOS and inform inpatient planning and resource allocation.
Background and Clinical Significance: Patent Blue V (PBV) is widely used for sentinel lymph node mapping but may rarely cause severe hypersensitivity reactions. These reactions can present atypically, with predominant cardiovascular collapse and minimal cutaneous manifestations, potentially delaying recognition and treatment. Case Presentation: We report the case of a 52-year-old woman who developed sudden and prolonged refractory hypotension shortly after PBV injection during melanoma surgery. The reaction initially occurred in the absence of bronchospasm or early significant cutaneous manifestations. Profound cardiovascular collapse preceded the delayed appearance of erythema and piloerection, making early recognition of perioperative anaphylaxis particularly challenging. Hemodynamic instability required aggressive fluid resuscitation and prolonged vasopressor support. Subsequent diagnostic work-up, including serum tryptase measurement and allergological evaluation, confirmed PBV-induced anaphylaxis. PBV-induced anaphylaxis may present with isolated cardiovascular collapse without typical clinical features. Conclusions: Early recognition and prompt hemodynamic support are essential to optimize outcomes in the perioperative setting.
Development NGOs like the Ethiopian Evangelical Church Mekane Yesus Development and Social Services Commission (EECMY-DASSC) carry a vital mission: translating social vision into meaningful, lasting community impact. However, in Ethiopia's increasingly unpredictable landscape, a rift often opens between visionary leadership and day-to-day operational execution. This study evaluates how project management standards interact with systemic bureaucracy and regional security challenges, aiming to identify key operational gaps and provide actionable pathways for organizational resilience. A triangulated mixed-methods design was employed to capture both broad operational trends and deep lived experiences. A census survey was administered to 828 project personnel, complemented by in-depth qualitative interviews with senior executive leaders. Quantitative survey data were processed using SPSS, while qualitative responses underwent thematic content analysis to extract key operational themes. The study revealed a "Polarized Maturity Model" within the organization. EECMY-DASSC demonstrates remarkable strength in relational disciplines, earning high performance ratings in stakeholder engagement and quality assurance (Mean: 4.13). Conversely, critical operational functions lag significantly behind, most notably Risk Management (Mean: 2.57) and Cost Management (Mean: 2.71). Externally, project continuity is continuously constrained by rigid governmental regulatory hurdles and ongoing regional instability. The findings point to a critical "Leadership-Management Gap." While organizational leaders exhibit strong commitment to their social mission, the absence of an adaptive, forward-looking management framework leaves community initiatives highly vulnerable to disruption. Technical tools alone are insufficient to guarantee project success; they must be paired with an adaptive leadership mindset capable of navigating real-world volatility. To bridge this gap, the study recommends establishing a Strategic Program Management Office (PMO) to unify oversight, institutionalizing a proactive culture of risk governance, and delivering practical adaptive leadership training to field and management personnel.
Background and Clinical Significance: Eczema herpeticum (EH), or Kaposi’s varicelliform eruption, is a dermatologic emergency characterized by the abrupt onset of painful monomorphic vesiculopustules with potential for rapid dissemination. Atopic dermatitis (AD) is the main predisposing condition due to skin barrier dysfunction and impaired antiviral immunity. Early recognition is essential because delayed treatment may result in avoidable complications, including ocular involvement and systemic disease. Current recommendations emphasize immediate systemic acyclovir based on clinical suspicion, without awaiting laboratory confirmation. Case Presentation: A 4-year-old boy with moderate AD presented with a 6-day history of fever, malaise, and a rapidly progressive vesiculopustular eruption involving both eczematous and previously unaffected skin. The patient had a recent AD flare, molluscum contagiosum, and had initially received oral amoxicillin-clavulanate for presumed bacterial superinfection without improvement. Physical examination revealed widespread painful monomorphic umbilicated vesiculopustules with hemorrhagic crusts and mild bilateral conjunctival injection. Oral acyclovir was initiated within one hour of evaluation. Laboratory investigations showed mild inflammatory abnormalities without renal or hepatic involvement. Because lesional PCR was unavailable, complementary blood-based investigations were performed; HSV-1 IgM serology and blood PCR provided additional retrospective findings compatible with HSV-1 infection, while Gram stain and bacterial cultures were negative. Fever resolved within 24 h, no new lesions developed after day 3, and complete re-epithelialization was achieved after a 10-day course of acyclovir. Conclusions: This case highlights the importance of bedside recognition of eczema herpeticum in children with atopic dermatitis, particularly when painful monomorphic vesiculopustules are accompanied by fever and rapid dissemination. Early initiation of systemic acyclovir based on clinical suspicion remains the cornerstone of management. While PCR from vesicular lesions is the preferred diagnostic test when available, laboratory confirmation should not delay treatment. This report also illustrates common real-world challenges, including initial misdiagnosis as bacterial infection and limited access to optimal virological testing.
Background and Clinical Significance: Solitary fibrous tumor (SFT), historically termed hemangiopericytoma (HPC), is a rare fibroblastic mesenchymal neoplasm with variable biological behavior. Pulmonary involvement is uncommon and may represent either a primary thoracic tumor or metastatic disease from an extrapulmonary site. Its clinical course ranges from indolent, surgically curable disease to aggressive malignancy with local recurrence and distant dissemination. In this retrospective case, confirmatory STAT6 immunohistochemistry was unavailable; therefore, the tumor is described as a hemangiopericytoma/solitary fibrous tumor spectrum neoplasm. Case Presentation: We report the case of a 33-year-old woman who presented with sudden-onset hemoptysis and was found to have two large, well-defined bilateral pulmonary masses. Initial clinical and radiological evaluation raised suspicion of primary pulmonary tumors or other benign lesions. Because both lesions were considered resectable, staged pulmonary resections were performed. Subsequent reassessment of the patient’s medical history revealed previous surgeries for a poorly documented recurrent thigh tumor, later confirmed to represent the primary malignant hemangiopericytoma/solitary fibrous tumor spectrum neoplasm. Despite staged pulmonary resections, systemic chemotherapy, and further oncologic management, the disease progressed rapidly, with cerebral, bilateral pulmonary, mediastinal, and subcutaneous metastases. The patient died within 18 months of the initial pulmonary diagnosis. Conclusions: This case highlights the diagnostic difficulty of metastatic pulmonary hemangiopericytoma, particularly when the primary soft tissue tumor is inadequately documented. It emphasizes the importance of detailed clinical history, retrieval of previous histopathological reports, and long-term surveillance in patients with soft tissue tumors, even when initially considered benign.
Background and Clinical Significance: Renal disease is the leading cause of secondary hypertension in children and adolescents. Among younger patients presenting with severe hypertension, renovascular and renal parenchymal disorders should be considered promptly; Case presentation: We describe the case of a 16-year-old male who presented with severe fatigue and was found to have resistant arterial hypertension (180/120 mmHg). His medical history was notable for blunt epigastric trauma sustained during football training approximately 6 months before presentation. Magnetic resonance imaging of the kidneys and retroperitoneum demonstrated a large right-sided perinephric hematoma compressing the kidney. Plasma renin activity and aldosterone levels were markedly elevated, establishing the diagnosis of Page kidney. Percutaneous drainage was performed by placement of a drainage catheter into the perinephric collection, resulting in evacuation of a substantial volume of liquefied hematoma. Following the procedure, arterial blood pressure gradually normalized, accompanied by resolution of the hormonal abnormalities; Conclusions: Page kidney is a rare but important cause of secondary hypertension resulting from activation of the renin-angiotensin-aldosterone system due to external renal compression and impaired intrarenal perfusion. Although its clinical presentation may be insidious, delayed recognition can lead to severe cardiovascular and renal complications. Management includes percutaneous drainage or surgical decortication of the affected kidney, together with antihypertensive treatment targeting the renin-angiotensin-aldosterone system. Early diagnosis and treatment are essential to optimize clinical outcomes and preserve renal function.
Background and Clinical Significance: Eosinophilic sialodochitis (ES), also known as sialodochitis fibrinosa, is a rare disorder characterized by recurrent salivary gland swelling caused by intraductal eosinophilic mucous plugs. Typical histopathological findings include eosinophils and Charcot-Leyden crystals within ductal secretions, and characteristic imaging findings include salivary duct dilatation and glandular swelling. Although rare cases associated with sialolithiasis or calcification have been reported, high-attenuation material within the salivary duct on computed tomography (CT) has not been clearly described in ES. Case Presentation: A 56-year-old woman with allergic rhinitis presented with recurrent swelling and pain in the right submandibular area. CT and ultrasonography revealed a large sialolith in the right submandibular gland and dilatation of Wharton's duct. She underwent right submandibular gland excision for presumed chronic obstructive submandibular sialadenitis with a sialolith. Soon after surgery, she developed recurrent swelling of the right floor of the mouth, and CT showed persistent high-attenuation material along Wharton's duct without residual sialolith. Ductal massage discharged a brownish gelatinous material. Histopathological examination revealed numerous eosinophils and Charcot-Leyden crystals in both the discharged mucous plug and decalcified sialolith, fulfilling Baer's diagnostic criteria for ES. Physical extraction and anti-allergic medications were insufficient, whereas ductal irrigation with saline and triamcinolone acetonide markedly reduced mucous plug discharge. Symptoms were controlled during 18 months of follow-up. Conclusions: Retained eosinophilic mucin in ES may appear as high-attenuation ductal material on CT and contribute to salivary stasis and sialolith formation.
Background and Clinical Significance: Tooth surface loss is the irreversible loss of dental hard tissue caused by non-carious processes. Restoration of affected teeth can be challenging, especially on the palatal surfaces of maxillary anterior teeth when severe palatal hard-tissue loss is present without marked clinical crown shortening. In such cases, clinicians must preserve the remaining enamel for predictable adhesive bonding while ensuring accurate seating, complete adaptation, and controlled cementation of thin indirect restorations. To address these challenges, this case report describes a novel preparation-free approach in which two temporary incisal seating lugs were incorporated into the design of indirect resin-based composite palatal veneers; Case Presentation: A 35-year-old male patient was referred by his general dentist for management of extensive erosive tooth surface loss. Clinical examination revealed generalized tooth surface loss, with a distinctive pattern of severe palatal hard-tissue loss affecting the maxillary anterior teeth without marked clinical crown shortening. Restorative treatment was planned at a 2 mm increase in occlusal vertical dimension. The maxillary anterior teeth were restored with preparation-free indirect resin-based composite palatal veneers, each incorporating two temporary incisal seating lugs to provide a positive seating stop during cementation; the lugs were removed after cementation. Other affected teeth were restored using directly placed resin-based composite restorations. At six months, the veneers remained clinically acceptable, with stable occlusion, maintained pulp sensibility, no postoperative sensitivity, and high patient satisfaction; Conclusions: This technique may facilitate restoration positioning during cementation and may represent a useful clinical alternative when seating preparation-free palatal veneers is challenging.
Ethiopian government policies have promoted irrigation expansion particularly for cereal crops as a means to boost agricultural growth, reduce production risk, alleviate rural poverty, and improve food security. The experiment was conducted in Oda Bultum District for two consecutive years to evaluate and determine the optimal irrigation water level and application under furrow irrigation for wheat, and to identify the economic water use efficiency and net benefits of deficit irrigation. Two furrow-irrigation methods (conventional and alternate) and four irrigation levels (55%, 70%, 85%, and 100% of ETc) were used. The experiment followed a randomized complete block design (RCBD) with eight treatments replicated three times. Both physical and chemical soil samples were taken and analyzed. Except for plant height, most wheat agronomic parameters differed significantly between treatments (P < 0.05). The highest grain yield occurred with conventional furrow irrigation at 100% ETc. Although 100% ETc with the conventional method produced the maximum yield, it had lower water productivity and a lower marginal rate of return, and it did not promote irrigation water savings. Therefore, for a balance of yield and water savings, conventional furrow irrigation at 70% ETc is recommended; for drought-prone and water-scarce areas, alternate furrow irrigation at 100% ETc is recommended as an option for small-scale farmers.
Background and Clinical Significance: Hemophagocytic lymphohistiocytosis (HLH) is a life-threatening hyperinflammatory syndrome characterized by uncontrolled activation of macrophages and cytotoxic lymphocytes. Secondary HLH is most commonly associated with infections, malignancies, and autoimmune disorders. Visceral leishmaniasis (VL) is an uncommon infectious trigger of HLH, and the considerable overlap in clinical manifestations frequently delays diagnosis. Early recognition of the infectious trigger is essential, because prompt targeted therapy may prevent the need for prolonged immunosuppressive treatment and substantially improve outcomes. Case Presentation: A 68-year-old livestock farmer from northern Greece presented with a one-month history of persistent fever, fatigue, and night sweats. Laboratory evaluation demonstrated pancytopenia, severe hyperferritinemia, hypertriglyceridemia, and elevated soluble interleukin-2 receptor (sCD25) levels. After extensive, relevant screening for other underlying etiologies, which proved to be negative, bone marrow examination revealed hemophagocytosis, while anti-Leishmania serology and polymerase chain reaction (PCR) analysis of bone marrow aspirate confirmed infection with Leishmania infantum. Treatment with liposomal amphotericin B along with intravenous dexamethasone resulted in rapid clinical and laboratory improvement. Serial ferritin and sCD25 measurements closely paralleled clinical recovery, supporting their potential usefulness as biomarkers of treatment response. The patient remained asymptomatic at one-month follow-up. Conclusions: Secondary HLH associated with VL is rare, but potentially fatal. Clinicians should maintain a high index of suspicion in patients presenting with prolonged fever, splenomegaly, cytopenia, and marked hyperferritinemia, particularly in endemic regions. Early diagnosis and prompt initiation of targeted therapy are associated with favorable outcomes.
Background and Clinical Significance: Ruptured common iliac artery aneurysms may mimic urological disease, particularly when retroperitoneal bleeding causes ureteral compression and secondary hydronephrosis. This case is clinically relevant because the first symptom was acute stabbing testicular pain, followed by lumbar discomfort and ultrasound findings suggestive of obstructive uropathy. Case Presentation: A 73-year-old man with arterial hypertension and active smoking presented to the Emergency Department with sudden left lumbar pain that initially radiated to, or was perceived in, the left testicle. The testicular pain resolved spontaneously, but persistent poorly localized lumbar pain continued. He was afebrile and initially hemodynamically stable. Point-of-care ultrasound showed left hydronephrosis, while the abdominal aorta appeared unremarkable. Because the patient looked clinically unwell and the abrupt, stabbing onset was not fully explained by uncomplicated renal colic, contrast-enhanced abdominal and pelvic computed tomography was performed. CT revealed a fissured saccular aneurysm of the left common iliac artery with active contrast extravasation and a large retroperitoneal hematoma compressing the ipsilateral ureter, thereby causing secondary hydronephrosis. The patient underwent urgent endovascular treatment with percutaneous transluminal angioplasty and Bentley stent. Post-revascularization CT showed no further contrast extravasation, and the patient was discharged after seven days without complications. Conclusions: In older patients with cardiovascular risk factors, sudden testicular, flank, or lumbar pain may warrant consideration of retroperitoneal vascular emergencies, even when initial ultrasound suggests a urological diagnosis. In this patient, discordance between the clinical presentation and the initial ultrasound findings prompted contrast-enhanced CT, which enabled diagnosis and timely vascular management.
Background and Clinical Significance: Pulmonary disease caused by nontuberculous mycobacteria (NTM) represents an important diagnostic challenge in tuberculosis-endemic settings because its clinical, radiological, and microbiological features may overlap with those of pulmonary tuberculosis (TB). Accurate distinction between these conditions is essential to avoid diagnostic delay and inappropriate treatment. Case Presentation: We present two cases that illustrate the heterogeneity of imaging patterns associated with NTM disease. The first case involved a 55-year-old woman with previously treated pulmonary tuberculosis who presented with chronic productive cough, recurrent mild hemoptysis, and progressive nodular-bronchiectatic and cavitary abnormalities. Mycobacterium avium was repeatedly isolated from independently collected respiratory specimens and identified using a line probe assay (LPA). The second case involved a 65-year-old man with severe chronic obstructive pulmonary disease (COPD), bronchiectasis, previous tuberculosis, and extensive bilateral fibrocavitary lung disease. Respiratory specimens were acid-fast bacilli-positive, whereas GeneXpert MTB/RIF repeatedly failed to detect the Mycobacterium tuberculosis complex. Repeated cultures identified Mycobacterium xenopi, including isolates from two sputum specimens and one bronchial aspirate. Treatment was subsequently adapted according to species identification and multidisciplinary assessment. These cases illustrate two major phenotypes of pulmonary NTM disease: nodular-bronchiectatic disease caused by M. avium and fibrocavitary disease caused by M. xenopi. They emphasize that persistent acid-fast bacilli (AFB) positivity with negative GeneXpert MTB/RIF results should prompt consideration of NTM alongside other differential diagnoses, followed by mycobacterial culture and species-level identification. Conclusions: The diagnosis of pulmonary NTM disease requires integration of clinical manifestations, radiological evolution, and repeated microbiological confirmation. Early species identification, multidisciplinary treatment selection, and close follow-up may reduce diagnostic delays and support appropriate individualized management.
Background and Clinical Significance: Skin-limited Langerhans cell histiocytosis (LCH) is a clinically heterogeneous disease, ranging from self-healing forms to fulminant multi-organ failure, the latter being more often described in infants, especially preterm neonates. The optimal therapy for cutaneous LCH remains controversial; the possibilities vary from a watchful waiting approach to systemic chemotherapy. Case Presentation: This case report describes an exceptionally rare and clinically challenging course of skin-limited LCH in a prematurely born infant treated at the Department of Oncology and Hematology, Children’s Hospital Zagreb, Croatia. At presentation, the patient exhibited several features suggestive of aggressive disease biology. However, therapeutic decision-making was complicated by extreme prematurity and young age, both of which significantly increased vulnerability to treatment-related toxicity. Following failure of topical therapy, systemic treatment was initiated according to the LCH-IV trial, primarily due to concerns regarding potential evolution into multisystem LCH. During treatment, the patient developed multiple life-threatening complications, namely severe infections (Staphylococcus aureus endocarditis, Pneumocystis jirovecii pneumonia, and Enterobacter cloacae sepsis), aggravated by secondary hypogammaglobulinemia, neutropenia, and iatrogenic adrenal insufficiency. Conclusions: The varied nature of cutaneous LCH underscores the necessity for a tailored treatment approach. When deciding on the treatment modality, clinicians should weigh the benefits of aggressive therapies, ensuring better disease control, against the potential for severe adverse effects, particularly in young, fragile infants with immature immunity.
Background/Objectives: The present investigation was designed and conducted with the aim to evaluate the statistical significance of the Comprehensive Facial Injury (CFI) score in relation to total surgical time (ST), length of hospital stay (LOS), and the presence of head injury in patients with fractures of the maxillofacial skeleton. Methods: The study includes 332 patients with maxillofacial fractures, a subset of whom also had associated head injuries. The CFI score was calculated for each patient. The Kolmogorov-Smirnov test was applied to assess the distribution of score values within the cohort. The Kruskal-Wallis H test was used to compare ST (minutes), LOS (days), and Glasgow Coma Scale (GCS) scores across different CFI score groups. Statistical significance was defined as p < 0.05. Results: The majority of the patients were male, at 77.1%, with 22.9% female. The mean age was 36.4 years, with a standard deviation of 19.3 years. Definitive surgical treatment was performed in 58.7% of patients, while the remaining 41.3% were managed conservatively (with closed reduction and external fixation) or received no surgical intervention. A statistically significant association was found between the CFI score and both ST and LOS (p < 0.001). Associated head injury was found in 33 patients or 9.9%. Furthermore, significant correlations were observed between CFI score values and Glasgow Coma Scale scores (p < 0.001) as well as the presence of head injury (p < 0.001). Conclusions: In conclusion, the CFI score provides a straightforward and comprehensive framework for assessing both operative duration and hospitalization length in patients with maxillofacial trauma. Moderate correlations between the CFI score and all investigated variables support its reliability in assessing trauma severity. The CFI score may therefore be useful in guiding treatment planning and developing standardized clinical protocols. However, its predictive capacity requires further validation in future studies.
Background and Clinical Significance: Peripheral veno-arterial extracorporeal membrane oxygenation (VA-ECMO) substantially alters aortic flow dynamics, generating catastrophic false-positive pathology on standard imaging. We report a case of ECMO-induced artifacts mimicking a Stanford type A aortic dissection (TAAD), which led to an unnecessary exploratory sternotomy. Case Presentation: A 67-year-old man underwent extracorporeal cardiopulmonary resuscitation (ECPR) for a shockable out-of-hospital cardiac arrest. Post-resuscitation chest computed tomography angiography (CTA) and preoperative transesophageal echocardiography (TEE) demonstrated a prominent flap-like structure in the ascending aorta, prompting emergency sternotomy. Intraoperative exploration revealed no intimal tear. Subsequent evaluation confirmed an acute anterior myocardial infarction, managed with coronary intervention. Following a dismal neurological prognosis due to hypoxic encephalopathy, VA-ECMO was palliatively withdrawn on day 9, and the patient expired on day 19. The interaction between retrograde ECMO flow and varying levels of intrinsic cardiac function dictates the topology of flow disturbances. Absent native flow creates contrast layering within the aortic root, whereas preserved native flow creates a volatile downstream watershed zone. Based on these distinct phenotypes, we propose a novel conceptual framework for tailor-made imaging strategies titrated to native flow strength—such as temporary ECMO flow reduction for preserved native output, or circuit contrast injections for profound cardiac depression. Conclusions: ECMO-related artifacts present substantial diagnostic pitfalls. Clinicians should adopt a context-aware approach, integrating multi-modality imaging with hemodynamic status to implement individualized, physiologically guided imaging protocols.
Background and Clinical Significance: The oocyte zona pellucida (ZP) is an extracellular glycoprotein matrix with essential roles in oogenesis, fertilization, and early embryonic development. Abnormal ZP morphology is associated with female infertility and adverse outcomes after assisted reproductive technology (ART), but its molecular basis remains incompletely understood. Case Presentation: We present three patients with female infertility, abnormal oocyte ZP morphology, and adverse embryological outcomes, including fertilization failure and early developmental arrest. Whole-genome sequencing identified an extremely rare heterozygous missense variant in the ZP4 gene (rs1254095560) in one patient, resulting in the p.Leu342Pro substitution at a conserved amino acid position within the functionally important ZP-C subdomain of the protein. In silico modeling showed that the p.Leu342Pro substitution may alter the spatial folding of the ZP4 protein. Conclusions: These results and the known role of ZP4 in the organization of the zona pellucida allow this variant to be considered a candidate genetic factor potentially associated with disruption of the zona pellucida structure. The absence of comparable, apparently pathogenic coding variants in ZP genes in two other patients with a similar zona pellucida phenotype suggests genetic heterogeneity of this phenotype.
Background and Clinical Significance: Iatrogenic atrophy following intralesional corticosteroid injections represents a rare but potentially disfiguring complication, particularly when administered in the facial region for the treatment of inflammatory acne. Unlike conventional post-acne atrophic scarring, corticosteroid-induced tissue loss involves both dermal and subcutaneous compartments, resulting in a clinically distinct presentation that poses significant therapeutic challenges, with no established consensus on optimal management. Case Presentation: We report the case of a 26-year-old Caucasian female (Fitzpatrick phototype III) presenting with severe iatrogenic facial atrophy of the left cheek, resulting from multiple intralesional corticosteroid injections performed by a previous physician for papulo-pustular acne. The condition had been clinically stable for approximately two years at first evaluation. The patient underwent a stepwise multimodal protocol delivered over approximately 18 months (November 2023 to April 2025), combining non-ablative fractional laser remodelling (LightScan—Eufoton), Autologous Regenerative Therapy (ART, Seffiller technique, Seffiline srl), hyperdiluted calcium hydroxylapatite (CaHA—Radiesse, Merz Aesthetics) biostimulation, and additional non-ablative fractional photothermolysis sessions, supported by a topical cosmeceutical protocol. Documented follow-up extended to December 2024, 13 months after the initiation of treatment at our centre. Clinician-assessed severity of the post-acne atrophic scarring component improved from Goodman & Baron Grade 3 to Grade 2. Global aesthetic improvement of the treated area was rated as +2 (“much improved”) on the GAIS, with a patient satisfaction score of 5/5. No validated grading instrument was applied to the subcutaneous volume deficit itself, for which no such instrument is currently available. Conclusions: This case documents the clinical management and favourable outcome of a patient with severe iatrogenic steroid-induced facial atrophy treated with a personalised multimodal protocol. While the observed improvement is encouraging, the single-case design does not permit conclusions regarding the efficacy, reproducibility, or generalisability of this approach. Future controlled studies are needed to determine the therapeutic value of this multimodal strategy for this rare condition.
Background and Clinical Significance: Implant rehabilitation of patients with acquired maxillofacial defects remains challenging, particularly following high-energy war-related trauma. Gunshot and mine blast injuries frequently result in extensive hard and soft tissue loss, often requiring complex reconstructive procedures. Although cortically anchored implants have been successfully used in patients with severe maxillary atrophy and selected traumatic defects, evidence supporting their use for the immediate rehabilitation of critical-size war-related maxillary defects remains limited. Cortically anchored single-piece implants used in conjunction with an immediate loading protocol may provide an alternative rehabilitation strategy for selected patients who decline, or are unsuitable for, conventional implants and bone-grafting procedures. Case Presentation: Two patients with critical-size maxillary defects (approximately 3 cm) resulting from gunshot and mine blast injuries are presented. Treatment consisted of extraction of non-restorable teeth, placement of cortically anchored single-piece implants, including tubero-pterygoid implants, followed by immediate loading with fixed hybrid metal-acrylic hybrid prostheses. Clinical and radiological evaluation was performed using panoramic radiography and cone-beam computed tomography. Conclusions: Successful implant-supported prosthetic rehabilitation was achieved in both patients. Cortically anchored implants engaging the basal bone of the maxilla provided stable support for immediately loaded fixed prostheses despite substantial hard and soft tissue loss. Functional and aesthetic outcomes were satisfactory. Immediate prosthetic rehabilitation was successfully completed in both patients. A 12-month clinical and radiographic follow-up was available for one patient and demonstrated stable implant function without biological or prosthetic complications. Long-term follow-up of the second patient was not available because of active military service. Cortically anchored implant-supported hybrid prostheses may represent a viable treatment option for selected patients with critical-size maxillary defects resulting from gunshot or mine blast injuries, enabling rapid restoration of oral function and facial aesthetics while avoiding extensive bone-grafting procedures.
