Osteochondrolipoma of the foot treated by surgical excision: a case report and literature review.

BACKGROUND: Osteochondromas, classified as a new benign subtype of lipomas and characterised by chondroid and osseous differentiation, are rare lesions that have been infrequently reported in previous literature. The maxillofacial region was reported as the most frequent localization, with infrequent occurrence in the lower limb. This paper represents the first documented case report of osteochondrolipoma in the foot. CASE PRESENTATION: A 51-year-old male patient presented with a chief complaint of right foot pain at the plantar aspect, accompanied by the observation of swelling between the first and the second metatarsal shafts. His complaint of pain and swelling started 10 and 4 years prior, respectively. Since their onset, both symptoms have progressed in nature. Imaging revealved a large mass exhibiting a nonhomogenous composition of fibrous tissue and bony structures. Surgical intervention through total excision was indicated. CONCLUSION: Osteochodrolipoma is a benign lesion that can affect the foot leading to decreased functionality of the foot due to the pain and swelling. Surgical excision is the recommended approach for this lesion, providing both symptomatic relief and confirmation of the diagnosis through histopathological examination.

Intraosseous myolipoma of the calcaneus.

A man in his 40s presented with an incidental finding of an osteolytic bone lesion. He sustained an ankle injury while inline skating, fracturing his lateral malleolus. Besides the fracture, radiographic imaging on the day of the injury incidentally revealed a well-defined solitary osteolytic lesion with a sclerotic rim within the right calcaneus. MRI showed an intraosseous, fat-containing lesion with focal contrast enhancement, assessed as an intraosseous lipoma with central necrosis. In the pathological analysis of a sample of the lesion an intraosseous myolipoma of the calcaneus was found-an unexpected and extraordinary finding. To prevent pathological fracturing, curettage and bone grafting were performed using autologous iliac crest bone in combination with allogenous bone chips.

Cutaneous Collagenous Vasculopathy: A Short Case Series and Literature Review.

ABSTRACT: Cutaneous collagenous vasculopathy (CCV) is a rare and idiopathic microangiopathy of superficial dermal blood vessels. There have been 75 cases described in the literature to date, not including the current report; however, given its clinical similarity to other primary telangiectasias, it is likely to be underreported and underdiagnosed. Here, we describe the clinical and histological features of 2 patients we newly diagnosed with CCV. Both generally fit the profile of prior cases and confirm previously described associations-they both are older White women, have rashes on their lower extremities, and have conditions and medications that are common among other reported cases. However, both are also somewhat atypical, as Patient 1 had symptomatic CCV and Patient 2 had a papular rash. As such, both cases broaden the spectrum of our current understanding of CCV. We also provide a comprehensive review of all published reports of CCV to date and uncover 2 previously unreported associations: one with visceral malignancy, and the other with hypothyroidism. Whether these association are coincidental is worth investigating in future studies.

A 50-Year-Old Man Presenting with Multiple Bone Lesions and a Diagnosis of Phosphaturic Mesenchymal Tumor of the Femur.

BACKGROUND Phosphaturic mesenchymal tumor (PMT) is an extremely rare mesenchymal neoplasm that is commonly seen in bone and soft tissue. It is associated with a paraneoplastic syndrome, oncogenic osteomalacia, due to tumor-induced urinary phosphate wasting. It is demonstrated to be predominantly mediated by fibroblast growth factor 23 (FGF23)/fibroblast growth factor receptor 1 (FGFR1) axis. Clinically, PMT usually presents as a solitary lesion in the bone. The diagnosis of PMT is challenging due to its non-specific clinical manifestation, radiologic findings, and morphological features. CASE REPORT We report the case of a 50-year-old man presenting with multiple lytic bone lesions and associated pathologic fracture of the right femur, clinically suspicious for multiple myeloma or other metastatic malignant process. Resection from the right femur showed a hypercellular lesion composed of oval-to-spindled cells infiltrating the native trabecular bone with admixed multinucleated giant cells. Immunohistochemical (IHC) staining and in situ hybridization (ISH) demonstrated the tumor cells were positive for SATB2, ERG, FGFR1, and FGF23 ISH. DNA and RNA next-generation sequencing showed marked increases in mRNA levels of FGF23 and FGFR1. The constellation of clinicoradiologic, histomorphologic, IHC, and molecular findings supported a diagnosis of primary benign PMT. CONCLUSIONS This case report discusses a patient with PMT presenting with multifocal lesions due to tumor-induced osteomalacia at initial presentation. We hope that this report will increase the awareness of clinician and pathologists of PMT as a differential diagnosis in patients presenting with multifocal lytic bone lesions. In turn, this will prevent misdiagnosis and overtreatment of a typically benign process.

Factors Influencing Venous Remodeling in the Development of Varicose Veins of the Lower Limbs.

One of the early symptoms of chronic venous disease (CVD) is varicose veins (VV) of the lower limbs. There are many etiological environmental factors influencing the development of chronic venous insufficiency (CVI), although genetic factors and family history of the disease play a key role. All these factors induce changes in the hemodynamic in the venous system of the lower limbs leading to blood stasis, hypoxia, inflammation, oxidative stress, proteolytic activity of matrix metalloproteinases (MMPs), changes in microcirculation and, consequently, the remodeling of the venous wall. The aim of this review is to present current knowledge on CVD, including the pathophysiology and mechanisms related to vein wall remodeling. Particular emphasis has been placed on describing the role of inflammation and oxidative stress and the involvement of extracellular hemoglobin as pathogenetic factors of VV. Additionally, active substances used in the treatment of VV were discussed.

Identifying risk factors and analyzing reconstructive outcomes in patients with lower-extremity soft-tissue sarcoma.

PURPOSE: Reconstructing defects after resecting soft-tissue sarcoma (STS) can be challenging. The aim of this retrospective study was to analyze the reconstructive outcomes and identify the potential risk factors in patients undergoing reconstruction after excision of lower-extremity STS. METHODS: Patients with lower-extremity STS were included. This database was compiled of patients from a single, large National Cancer Institute-accredited academic hospital. In total, 302 patients were included between January 2016 to January 2022. Univariate and multivariate analyses were performed to calculate odds ratios (ORs) for developing complications for each patient and surgical characteristic. RESULTS: The following factors were independent predictors of any complication: benign pulmonary disease (OR = 4.2; p = 0.02), preoperative radiotherapy (RT; OR = 2.5; p = 0.047), a tumor in the medial thigh (OR = 1.9; p = 0.03), body mass index (BMI) > 30 kg/m2 (OR = 1.05; p = 0.037), and full-thickness skin graft (OR = 5.4; p = 0.01). In the preoperative RT subgroup, reconstructing a defect via undermining and layered closure alone was an independent predictor of dehiscence (OR = 2.1; p = 0.02) and seroma (OR = 3.1; p = 0.02), whereas pedicled flaps (OR = 0.08; p = 0.001) and free flaps (OR = 0.05; p = 0.001) were independent protectors against any complication. CONCLUSION: Information derived from this analysis will assist with accurate preoperative patient counseling, which is crucial for informed decision-making and expectation management in lower-extremity STS. BMI and pulmonary function should be optimized to the extent possible to reduce postoperative complications. Patients treated preoperatively with RT should be reconstructed with a pedicled or free flap to optimize recovery.

Lymphangiogenesis and Lymphatic Zippering in Skin Associated with the Progression of Lymphedema.

Secondary lymphedema often develops after lymph node dissection or radiation therapy for cancer treatment, resulting in marked skin fibrosis and increased stiffness owing to insufficiency of the lymphatic system caused by abnormal structure and compromised function. However, little is known about the associated changes of the dermal lymphatic vessels. In this study, using the lower limb skin samples of patients with secondary lymphedema, classified as types 1-4 by lymphoscintigraphy, we first confirmed the presence of epidermal thickening and collagen accumulation in the dermis, closely associated with the progression of lymphedema. Three-dimensional characterization of lymphatic capillaries in skin revealed prominent lymphangiogenesis in types 1 and 2 lymphedema. In contrast, increased recruitment of smooth muscle cells accompanied by development of the basement membrane in lymphatic capillaries was observed in types 3 and 4 lymphedema. Remarkably, the junctions of dermal lymphatic capillaries were dramatically remodeled from a discontinuous button-like structure to a continuous zipper-like structure. This finding is consistent with previous findings in an infection-induced mouse model. Such junction tightening (zippering) could reduce fluid transport and cutaneous viral sequestration during the progression of lymphedema and might explain the aggravation of secondary lymphedema. These findings may be helpful in developing stage-dependent treatment of patients with lymphedema.

Primary Extranodal Nasal-Type Natural Killer/T-Cell Lymphoma of Lower Limb Muscles on 18 F-FDG PET/CT.

ABSTRACT: Extranodal nasal-type natural killer/T-cell lymphoma may arise anywhere outside the lymph nodes, predominantly including the nasal cavity, nasopharynx, oropharynx, upper respiratory tract, gastrointestinal tract, skin, testes, and salivary glands, whereas primary tumor arising in muscle is rarely documented. We report FDG PET/CT findings of primary extranodal nasal-type natural killer/T-cell lymphoma in the muscles of the right lower limb in an 82-year-old man.

A Rare Clinical Presentation With a Difficult Imaging Diagnosis of an Intra-articular Clear Cell Sarcoma of the Knee.

ABSTRACT: Clear cell sarcoma, a highly aggressive cell sarcoma with melanotic differentiation, typically occurs in the tendon and aponeuroses of the lower extremities and rarely develops in the intra-articular region. Herein, we present an extremely rare case of clear cell sarcoma originating from the intra-articular knee and suspected as benign tumors due to the benign-looking findings on MRI and PET/CT. The image results in our case were completely consistent with a tenosynovial giant cell tumor, resulting in the misdiagnosis. For differential diagnosis, especially malignant melanoma, histopathology, including IHC and FISH, was indispensable.

A new lymphography protocol and interpretation principles based on functional lymphatic anatomy in lower limb lymphedema.

Indirect lymphatic system imaging is essential for diagnosing lymphatic diseases. The basic methodology involves intradermal or subcutaneous injection of a contrast agent into the surrounding lymphatic capillary, and the flow of the contrast agent is identified using a detector. Many contrast agents that use near-infrared dye, including indocyanine green (ICG) fluorescent lymphography, are available. ICG is rapidly spreading as a convenient and safe lymphedema diagnostic method, because it does not involve radiation exposure, and the imaging equipment is more compact than other devices. The lymphatic system is a semi-open circulatory system with numerous lymphatic capillaries acting as blind ends. Anatomical information on the injection site and observation of specific lymphatic vessels and nodes is important. However, this anatomical information is lacking. Recent reports suggest that ICG fluorescent lymphography can be applied to cadavers in the same manner as living bodies. Furthermore, these reports have demonstrated the functional aspects of the capillary lymph vessel networks as well as their relationship with lymphatic vessels and lymph nodes. This review article describes the historical progression from the old to the new functional lymphatic anatomy and introduces a new functional lymphography technique for the lower limbs.

Effect of miR-206 on lower limb ischemia-reperfusion injury in rat and its mechanism.

Lower limb ischemia-reperfusion is a common pathological process during clinical surgery. Because lower limb ischemia-reperfusion usually aggravates ischemia-induced skeletal muscle tissue injury after lower limb ischemia-reperfusion, it also causes remote organ heart, intestine, liver, lung and other injuries, and there is no effective clinical treatment for lower limb ischemia-reperfusion injury, so it is urgent to study its injury mechanism. In this study, the rat model of lower limb ischemia-reperfusion was established by clamping the femoral artery with microarterial clips, and the wall destruction such as intimal injury, cell edema, collagen degeneration, neutrophil infiltration, and elastic fiberboard injury of the femoral artery wall was detected. The expression of inflammatory factors was detected by immunohistochemistry. miR-206 preconditioning was used to observe the expression of inflammatory factors, redox status and apoptosis in the vascular wall of rats after acute limb ischemia-reperfusion. Our findings suggest that vascular endothelial cell edema increases, wall thickening, neutrophil infiltration, and elastic fiber layer damage during IRI. Inflammatory factor expression was increased in femoral artery tissue, and miR-206 expression levels were significantly down-regulated. Further studies have found that miR-206 attenuates lower limb IRI by regulating the effects of phase inflammatory factors. In this study, we investigated the effect of miR-206 on inflammatory factors and its possible role in the development of lower limb IRI, providing new research ideas for the regulatory mechanism of lower limb IRI, and providing a certain theoretical basis for the treatment of lower limb ischemia-reperfusion injury after surgery or endovascular intervention.

Epidermal inclusion cyst embedded in benign fibrous histiocytic lesion with prominent epithelioid morphology.

Benign fibrous histiocytoma also known as dermatofibroma is one of the common mesenchymal neoplasms. It commonly develops in young adult with female predominance and predilection for the extremities, particularly lower extremities. Implantation of epidermis in the dermis or subcutaneous tissues may lead to the formation of epidermal inclusion cyst, which is the most common type of epithelial cyst. Development of epidermal inclusion cyst within a benign fibrous histiocytoma is a rare occurrence. This is a unique case of two unrelated lesions.

[A 50 year old man with progressive weakness in lower limbs].

Lambert-Eaton Myasthenic Syndrome (LEMS) is a rare neurological disorder caused by autoimmune antibodies attacking the presynaptic neuromuscular junction, in some cases caused by underlying cancer. The main clinical finding is fluctuating weakness of the extremities and a triad of symtoms can help physicians suspect the disease. A key to the diagnosis are the electrophysiological abnormalities seen in this group of diseases. Treatment with symtomatic and/or immunosuppressive therapy is important as well as a workup for possible malignancy. This article identifies the clinical features, diagnosis and treatment of this uncommon disease.

Giant muscle hydatid in lower extremity: a rare case with neurological symptoms as the first manifestation.

BACKGROUND: Human hydatid disease typically occurs in organs such as the liver and kidney. Primary solitary intramuscular hydatid disease, however, is rare. CASE PRESENTATION: We report a case of a giant muscle hydatid in the lower extremity, with neurological symptoms as the first manifestation. The symptoms specifically manifested as intermittent pain in the right lower extremity and numbness in the sole of the right foot. However, there were no obvious abnormalities detected in electromyography and lumbar MRI. Subsequent ultrasonography and calf MRI showed that the patient had cystic lesions in the calf. The patient was initially diagnosed with a muscle hydatid cyst. Treatment involved complete surgical excision of the lesion, and the diagnosis of a hydatid cyst was confirmed through macroscopic and microscopic histopathological examination after the mass was excised. The patient was given oral albendazole, and no recurrence was observed during the 12 months of follow-up. CONCLUSIONS: This case underscores the need to consider hydatid disease when diagnosing soft tissue masses in muscles, particularly in endemic areas. Patients may initially present with atypical symptoms like peripheral nerve issues.

Clinical Outcome in Patients With High-grade Soft-tissue Sarcoma Receiving Prosthetic Replacement After Tumor Resection of the Lower Extremities: Tokai Musculoskeletal Oncology Consortium Study.

AIM: This multicenter retrospective study aimed to clarify the surgical and oncological outcomes of patients with high-grade soft tissue sarcoma (STS) who underwent prosthetic replacement reconstruction after lower extremity tumor resection. PATIENTS AND METHODS: We retrospectively collected the data of 27 patients with high-grade STS. The mean follow-up duration after prosthetic replacement was 44.7 months. RESULTS: The mean age at surgery was 63 years. The mean tumor size was 16 cm. For reconstruction, proximal femur replacement was performed in 15 patients, distal femur replacement in six, and total femur replacement in six. The major complications were infections in nine patients and aseptic loosening in four. Nine patients developed local recurrence. The cause of revision surgery was infection in five patients, aseptic loosening in three, and metal allergy in one. The 5-year prosthetic survival rate was 51.1%. At the final follow-up, amputation was performed in five patients. The 5-year limb salvage rate was 76.8%. The mean functional score of the 25 patients who could be assessed was 16.0 (53%). Of the 27 patients, five were excluded from the survival analysis because they underwent prosthetic replacement for local recurrence. The 5-year overall survival rate in the remaining 22 patients was 45.3%. CONCLUSION: We identified a high rate of surgical complications and poor survival in patients with high-grade STS who underwent tumor resection and reconstruction using prosthetic replacement of the lower extremities, although limb salvage was achieved in 81.5% of the patients. Careful follow-up is needed for surgical complications and oncological events after surgery.

Primary cutaneous large B cell lymphoma masquerading as lupus vulgaris.

Primary cutaneous large B cell lymphoma, leg type is a rare and aggressive variant of cutaneous B cell lymphoma. It predominantly affects elderly women, with the lower limb being the most common site of presentation. The overall prognosis is poor, compared to other cutaneous B cell lymphomas. A 47-year-old man presented with a progressively enlarging nodule over the medial aspect of the left foot since 2 months. Clinical examination revealed a nodular plaque-like lesion with central ulceration that measured 7 × 7 cm, firm in consistency, and with ill-defined margins. The initial clinical diagnosis was lupus vulgaris. An incision biopsy was done, which on histopathology and immunohistochemistry revealed a rare diagnosis of primary cutaneous B cell lymphoma, leg type. The patient was started on chemotherapy; however, he succumbed to his illness about 1 year after the initial presentation. It is a rare type of cutaneous lymphoma, which may masquerade infectious disorders such as lupus vulgaris. A detailed histopathological and immunohistochemical analysis is essential for its correct diagnosis and management. Only a handful of cases of this rare condition are reported to date. This case has been reported in view of its rarity and unusual clinical presentation.

[Pretibial myxedema after Graves' disease: A differential diagnosis of lymphedema]. / Myxœdème prétibial pseudo-tumoral après une maladie de Basedow : un diagnostic différentiel du lymphœdème.

INTRODUCTION: Pretibial myxedema is a rare manifestation of Graves' disease, and pseudotumoral forms may be confused with lower limb lymphedema. OBSERVATIONS: We reported 3 cases of pretibial myxedema in 2 women and 1 man, aged 72, 66, and 49 years, treated for Graves' disease 3, 25 and 32 years previously. Two patients were active smokers. Lymphedema diagnosis of the lower limbs was suspected in the presence of bilateral pseudotumoral lesions of the feet, toes and ankles and the presence of a Stemmer's sign (skin thickening at the base of the 2nd toe, pathognomonic of lymphedema). Lymphoscintigraphy in one case was normal, not confirming lymphedema. CONCLUSION: Pretibial pseudotumoral myxedema is a differential diagnosis of lower limb lymphedema. This diagnosis is confirmed by questioning the patient about preexisting Graves' disease, the underlying etiology, to decide the appropriate treatment and to encourage cessation of smoking, which is a risk factor for pretibial myxedema.

Don't miss it: Extremity-located cyst hydatid may mimic soft tissue tumors.

OBJECTIVES: In this study, we present our experience in patients with hydatid cysts located intramuscularly. PATIENTS AND METHODS: Between May 2018 and May 2023, a total of 11 patients (3 males, 8 females; mean age: 29.1±13.6 years; range, 8 to 56 years) with intramuscular hydatid cysts were retrospectively analyzed. Demographic data, laboratory values, serological test results, location and size of the cyst, radiological imaging findings, and complications were recorded. RESULTS: The mean follow-up was 44.3±17.3 (range, 5 to 60) months. The mean mass size at the time of admission was 5.4±3.3 (range, 2 to 14) cm. Serologic tests were positive in the majority of cases (72.7%). Eosinophilia was negative in 72.7% patients. The rate of isolated muscle involvement was 81.8%. The rate of lower extremity involvement was 72.7%. The most common involvement was leg (36.4%), thigh (18.2%), and shoulder (18.2%). One patient developed compartment syndrome after cyst rupture during neoadjuvant antihelmintic therapy. There was no recurrence in any of the patients. CONCLUSION: Hydatid cysts should be considered in the differential diagnosis of slowly growing, deeply located, painless soft tissue masses, particularly in endemic areas. Although it is a rare complication, compartment syndrome may develop after spontaneous cyst rupture. Neoadjuvant antihelmintic chemotherapy can reduce complications. The combination of total surgical excision and chemotherapy yields successful results in the treatment of hydatid cysts located in the muscle.