Chalazion-mimicked eyelid angiosarcoma in a young Asian with good prognosis: a case report.

BACKGROUND: Angiosarcoma is an extremely rare malignant tumor. So far, only about 42 cases of angiosarcoma involving the eyelids have been reported. Eyelid angiosarcoma occurs more frequently in elderly Caucasian males and is prone to misdiagnosis. We present a case report in a young Asian male patient with eyelid angiosarcoma that was misdiagnosed as a chalazion. CASE PRESENTATION: A 46-year-old South Korean male with no underlying disease had a right lower lid mass. The lesion was initially misdiagnosed as a chalazion at a local clinic, but a diagnosis of eyelid angiosarcoma was made after the first biopsy trial. PET-CT was performed to ensure that there was no metastasis in the whole body. Surgical excision with enough surgical margin was used alone for treatment and reconstruction was performed with a tarsoconjunctival advancement flap (modified Hughes procedure), which helped ensure good cosmesis. No recurrence was observed 4 years and 5 months after the surgery. CONCLUSIONS: The current study presents the first case of chalazion-mimicked eyelid angiosarcoma in a young Asian male aged under 50 years. This case shows that even if a benign eyelid disease is suspected in a young patient, an incisional biopsy must be performed to confirm whether the lesion is malignant. Since the prognosis is good for the case of eyelid angiosarcoma, if there is no clear evidence of distal metastasis, surgical resection should be performed with an enough safety margin.

Concomitant tuberculosis of the iliotibial band mimicking a soft tissue tumor and tuberculous trochanteric bursitis: a case report emphasizing on magnetic resonance imaging findings.

The risk of tuberculosis (TB) increases in immunocompromised patients. Multiple myeloma is considered a risk factor for TB and myeloma patients with TB have a higher mortality rate than those without TB. Herein, we report a case of concomitant TB of the iliotibial band mimicking a soft tissue tumor and tuberculous trochanteric bursitis in a patient with multiple myeloma. In this article, the characteristic magnetic resonance imaging (MRI) findings were low T2 signals in the cystic fluid lesion, a dark T2 signal rim, and peripheral rim enhancement. These results could help differentiate TB of the iliotibial band and trochanteric bursitis from other pathologies. If the abovementioned findings were observed in immunocompromised patients, extrapulmonary TB may be expected even if chest radiographs are normal.

Ambient light robust gamut mapping for optical see-through displays.

An optical see-through (OST) display is affected more severely by ambient light than any other type of displays when placed in an outdoor environment with bright illuminance because of its transparency and thus, its inherent color distortion can worsen. It is hard to directly apply existing gamut mapping methods to an OST display because of its morphological gamut characteristic and the effect of ambient light. In this paper, we propose a new robust gamut mapping method which works against bright ambient light. The process is divided into two steps: lightness mapping (LM) and chroma reproduction. LM aligns the lightness level of sRGB gamut with OST gamut and partitions the region of OST gamut based on the relative size of the sRGB gamut and its lightness value. The second step (chroma reproduction) determines an appropriate chroma reproduction method (gamut compression or extension) and a proper direction for gamut mapping based on the characteristics of each region in order to minimize the effects of ambient light. The quality of color reproduction is qualitatively and quantitatively evaluated based on accurate measurements of the displayed colors. It has been experimentally confirmed that the proposed gamut mapping method can reduce color distortion more than the existing parametric gamut mapping algorithms.

Identification of Differentially Expressed Genes Associated with Extracellular Matrix Degradation and Inflammatory Regulation in Calcific Tendinopathy Using RNA Sequencing.

Calcific tendinopathy (CT), developed due to calcium hydroxyapatite deposition in the rotator cuff tendon, mostly affects women in their 40 s and 50 s and causes severe shoulder pain. However, the molecular basis of its pathogenesis and appropriate treatment methods are largely unknown. In this study, we identified 202 differentially expressed genes (DEGs) between calcific and adjacent normal tendon tissues of rotator cuff using RNA sequencing-based transcriptome analysis. The DEGs were highly enriched in extracellular matrix (ECM) degradation and inflammation-related processes. Further, matrix metalloproteinase 9 (MMP9) and matrix metalloproteinase 13 (MMP13), two of the enzymes associated with ECM degradation, were found to be highly upregulated 25.85- and 19.40-fold, respectively, in the calcific tendon tissues compared to the adjacent normal tendon tissues. Histopathological analyses indicated collagen degradation and macrophage infiltration at the sites of calcific deposit in the rotator cuff tendon. Our study acts as a foundation that may help in better understanding of the pathogenesis associated with CT, and thus in better management of the disease.

Alternative splicing induces cytoplasmic localization of RBFOX2 protein in calcific tendinopathy.

BACKGROUND: Calcific tendinopathy (CT) is characterized by deposits of calcium, most commonly found in the shoulder tendons. The exact cause and pathogenesis of CT are not fully understood. This study analyzed the expression pattern of RNA-binding protein fox-1 homolog 2 (RBFOX2), a crucial splicing regulator in tissue differentiation. METHODS: Normal and calcific tendons were compared for RBFOX2 mRNA level using quantitative reverse-transcription polymerase chain reaction. Intracellular localization of RBFOX2 protein was investigated using immunofluorescence microscopy. Normal and calcific tendon cDNAs were used to clone RBFOX2. Sequencing analysis identified coding sequences of the RBFOX2 isoform. RESULTS: The intracellular localization of RBFOX2 protein differed with disease status, with RBFOX2 localized in the cytoplasm in calcific tendons and the nucleus in normal tendons. Analysis of the RBFOX2 protein-coding sequence showed that exon 10, responsible for nuclear localization, was absent in calcific tendons. Splicing of RBFOX2 target genes CHD2 and MBNL1 was significantly affected by cytoplasmic localization of RBFOX2 in calcific tendons. DISCUSSION: Given the function of RBFOX2 as a splicing regulator in the nucleus, cytoplasmic localization of RBFOX2 protein in calcific tendons may have affected overall splicing events and altered gene expression. These results provide insights for comprehension of CT pathogenesis.

Autophagy-mediated upregulation of cytoplasmic claudin 1 stimulates the degradation of SQSTM1/p62 under starvation.

Claudin 1, a major tight junction protein, is highly expressed in various types of tumors such as thyroid, breast, and colorectal cancers. Moreover, claudin 1 is frequently found in the cytoplasm in various types of tumor cells. However, the cytoplasmic function of claudin 1 in tumors still remains largely unknown. Here, we investigated the novel function of cytoplasmic claudin 1 in autophagy. The mRNA expression level of claudin 1 was higher in several types of tumors than in normal tissues. Furthermore, colon tumor tissues showed increased autophagy compared with the adjacent normal tissues. Both endogenous and exogenous claudin 1 showed a cytoplasmic punctate staining pattern and were co-stained with the lysosome-associated membrane protein 1 (LAMP1). Importantly, autophagy-induced conditions, including starvation, increased the protein stability of claudin 1. Moreover, the increased level of claudin 1 stimulated autophagy by decreasing the level of the autophagy substrate, sequestosome1/p62 (SQSTM1), under autophagy-inducing conditions; activation of AMP-activated protein kinase (AMPK) and inhibition of mammalian target of rapamycin (mTOR). Taken together, we demonstrate that the novel function of cytoplasmic claudin 1 is related to autophagy. This study is the first to show a cytoplasmic function of claudin 1 as an autophagy regulator and provides the evidence that claudin 1-mediated autophagy regulation is an integral part of the mechanism by which claudin 1 regulates cancer progression.

Regeneration of thyroid follicles from primordial cells in a murine thyroidectomized model.

The functional unit of the thyroid gland, the thyroid follicle, dynamically responds to various stimuli to maintain thyroid hormone homeostasis. However, thyroid follicles in the adult human thyroid gland have a very limited regenerative capacity following partial resection of the thyroid gland. To gain insight into follicle regeneration in the adult thyroid gland, we observed the regeneration processes of murine thyroid follicles after partial resection of the lower third of the thyroid gland in 10-week-old male C57BL/6 mice. Based on sequential observation of the partially resected thyroid lobe, we found primitive follicles forming in the area corresponding to the central zone of the intact lateral thyroid lobe. The primitive thyroid follicles were multiciliated and had coarsely vacuolated cytoplasm and large vesicular nuclei. Consistently, these primitive follicular cells did not express the differentiation markers paired box gene-8 and thyroid transcription factor-1 (clone SPT24), but were positive for forkhead box protein A2 and leucine-rich repeat-containing G-protein-coupled receptor 4/GPR48. Follicles newly generated from the primitive follicles had clear or vacuolar cytoplasm with dense, darkly stained nuclei. At day 21 after partial thyroidectomy, the tall cuboidal follicular epithelial cells had clear or vacuolar cytoplasm, and the intraluminal colloid displayed pale staining. Smaller activated follicles were found in the central zone of the lateral lobe, whereas larger mature follicles were located in the peripheral zone. Based on these observations, we propose that the follicle regeneration process in the partially resected adult murine thyroid gland associated with the appearance of primitive follicular cells may be a platform for the budding of differentiated follicles in mice.

Rotator cuff bridging repair using acellular dermal matrix in large to massive rotator cuff tears: histologic and clinical analysis.

BACKGROUND: This study investigated the efficacy of the bridging repair using an acellular dermal matrix (ADM) and an ADM with stem cells in rabbits. Also investigated were clinical outcomes of ADM bridging repair for large to massive rotator cuff tears. MATERIALS AND METHODS: ADM, with and without stem cells, was used to cover a 5- × 5-mm-sized cuff defect in 17 rabbits, and biomechanical, histologic, and immunohistochemical analyses were conducted. Also evaluated were 24 patients with large to massive rotator cuff tears after ADM bridging repair. RESULTS: In the biomechanical test, the normal rotator cuff, cuff with ADM plus stem cells, and cuff with ADM in the rabbit model showed a maximum load (N) of 287.3, 217.5, and 170.3 and ultimate tensile strength (N/mm2) of 11.1, 8.0, and 5.2, respectively. Histologically, the cuff tendons with the ADM or ADM plus stem cells showed characteristically mature tendons as time passed. In the clinical study, the mean American Shoulder and Elbow Surgeons score improved from preoperative 50 to postoperative 83, the University of California Los Angeles Shoulder Rating Scale from 17 to 30, and the Simple Shoulder Test from 4 to 8, respectively. No further fatty deteriorations or muscle atrophy were observed on follow-up magnetic resonance imaging. A retear was found in 5 of 24 patients (21%). CONCLUSIONS: Bridging repair with ADM or stem cells in the rabbit model showed cellular infiltration into the graft and some evidence of neotendon formation. Clinically, ADM repair was a safe alternative that did not show any further fatty deterioration nor muscle atrophy in large to massive rotator cuff tears.

A rare case of primary malignant small cell carcinoma combined with urothelial cell carcinoma in the ureter.

BACKGROUND: Extrapulmonary small cell carcinomas have been reported in a variety of organs, and their incidence in the genitourinary tract is second only to that in the gastrointestinal tract. To date, however, only a few cases of small cell carcinoma of the ureter have been reported. Because the extreme rarity of this type of carcinoma, its clinical behaviour, diagnostic methods, and effective treatment modalities have not yet been determined. CASE PRESENTATION: A 59-year-old man presented with a 1-month history of painless gross haematuria. Urine cytopathology revealed a urothelial carcinoma and computed tomography revealed left hydronephroureterosis with a distal ureteral stone and a mildly enhanced fungating mass just below the stone-impacted site. The preoperative TNM stage was T2N0M0. The patient underwent simultaneous diagnostic ureterorenoscopy and left laparoscopic nephroureterectomy with bladder cuff resection. Gross examination showed a 3.5 × 3.0 × 0.8 cm white, partly yellow mass in the left distal ureter. Light microscopy showed a small cell carcinoma, overlaid on a urothelial carcinoma in situ, invading the ureter and external lateral resection margins. The small cell carcinoma was diffusely positive for neuron-specific enolase, and exhibited focal positivity for CD 56, synaptophysin, chromogranin and cytokeratin 20. The patient was treated with adjuvant chemotherapy, consisting of cisplatin and etoposide, and radiation therapy, and has been well, without evidence of tumour recurrence or metastasis in the 10 months after surgery. CONCLUSION: Small cell carcinoma of the ureter is rare. Although its clinical behaviour and diagnostic modalities have not been determined and it has yet to be diagnosed immunohistopathologically, multimodality treatment including surgery, chemotherapy and radiotherapy may improve patient survival.

Soft tissue pseudotumors of the hand and wrist mimicking malignancy: Two case reports.

RATIONALE: Most soft-tissue lesions in the hand and wrist have a benign etiology, and malignant tumors, such as soft-tissue sarcomas, are rare. Mimickers of soft tissue tumors in the hand and wrist are more common than true neoplastic lesions; however, soft tissue pseudotumors that mimic malignancy are very rare. PATIENT CONCERNS: This study describes 2 patients with soft tissue pseudotumors of the hand and wrist. Both patients presented with rapidly growing soft-tissue masses. Magnetic resonance imaging (MRI) revealed ill-defined margins and an aggressive appearance in both cases, leading to a strong suspicion of malignant soft tissue tumors. DIAGNOSIS: Both patients underwent incisional biopsies, and the final diagnoses were inflammation due to IgG4-related disease in the first case and chronic granulomatous inflammation in the second case. INTERVENTIONS: The first patient was administered oral steroids, while the second patient was treated with anti-inflammatory drugs. OUTCOMES: Both patients showed a reduction in swelling of the hand and wrist. LESSONS: Although the imaging approach for pseudotumorous lesions is similar to that for true soft tissue tumors, the management of these lesions is different. Biopsies should only be performed when the diagnosis is unclear.

Optimizing the photon ratio of red, green, and blue LEDs for lettuce seedlings: a mixture design approach.

BACKGROUND: Light control technology has been developed and studied for decades in controlled environment agriculture (CEA) for successful crop production. The effects of the light spectrum on plant growth can vary because plants have spectral specific responses, and mixed light elicits interactive combination effects. Response surface methodology (RSM) can be utilized with the design of experiments to optimize a response influenced by multiple inputs with limited data. In this study, we aimed to identify the optimal photon ratio in combination of red (R), green (G), and blue (B) light-emitting diodes (LEDs) for growing lettuce seedlings using RSM and a seedling-indicating parameter by performing a similarity analysis of response surfaces that elucidated the response tendency of different factors, such as light quality. RESULTS: The highest shoot fresh weight was obtained from the R treatment (red LED 100%) at the end of the seedling stage. However, the RGB141 (photon ratio of R:G:B = 1:4:1) treatment during the seedling stage resulted in the highest shoot fresh weight at the final harvest. The value of the leaf area multiplied by the leaf chlorophyll concentration (SPAD) was selected as the seedling-indicating parameter. The optimal RGB photon ratio that maximized this parameter was R:G:B = 30.6:44.0:25.4, and this ratio was verified by conducting identical cultivation experiments. During the first 6 days after transplanting, SPAD gradually increased in R-treated seedlings, while the optimal treatment maintained the value at a higher constant level, which supported our result of shoot fresh weight at harvest. CONCLUSIONS: Thus, we confirmed that the mixture design method allowed us to optimize the combined RGB photon ratios for the seedling stage in order to maximize the growth index of mature lettuce plants and to select an appropriate seedling-indicating parameter that represents the final harvest results to benefit crop production in CEA.

Actinomycosis Confined to the Nasal Septum.

Actinomycosis is common in the head and neck region but rarely occurs in the nasal septum. A 75-year-old male patient with an edentulous maxilla, hypertension, and diabetes developed actinomycosis confined to the nasal septum and showed mucosal necrosis and septal bony sequestration. The patient underwent surgery and medication therapy; this case was reported using endoscopic photographs and radiographs and a literature review was conducted to provide further context and understanding of the condition of the patient.

Deep Dichromatic Guided Learning for Illuminant Estimation.

A new dichromatic illuminant estimation method using a deep neural network is proposed. Previous methods based on the dichromatic reflection model commonly suffer from inaccurate separation of specularity, thus being limited in their use in a real-world. Recent deep neural network-based methods have shown a significant improvement in the estimation of the illuminant color. However, why they succeed or fail is not explainable easily, because most of them estimate the illuminant color at the network output directly. To tackle these problems, the proposed architecture is designed to learn dichromatic planes and their confidences using a deep neural network with novel losses function. The illuminant color is estimated by a weighted least mean square of these planes. The proposed dichromatic guided learning not only achieves compelling results among state-of-the-art color constancy methods in standard real-world benchmark evaluations, but also provides a map to include color and regional contributions for illuminant estimation, which allow for an in-depth analysis of success and failure cases of illuminant estimation.

Ghost-Free Deep High-Dynamic-Range Imaging Using Focus Pixels for Complex Motion Scenes.

Multi-exposure image fusion inevitably causes ghost artifacts owing to inaccurate image registration. In this study, we propose a deep learning technique for the seamless fusion of multi-exposed low dynamic range (LDR) images using a focus-pixel sensor. For auto-focusing in mobile cameras, a focus-pixel sensor originally provides left (L) and right (R) luminance images simultaneously with a full-resolution RGB image. These L/R images are less saturated than the RGB images because they are summed up to be a normal pixel value in the RGB image of the focus pixel sensor. These two features of the focus pixel image, namely, relatively short exposure and perfect alignment are utilized in this study to provide fusion cues for high dynamic range (HDR) imaging. To minimize fusion artifacts, luminance and chrominance fusions are performed separately in two sub-nets. In a luminance recovery network, two heterogeneous images, the focus pixel image and the corresponding overexposed LDR image, are first fused by joint learning to produce an HDR luminance image. Subsequently, a chrominance network fuses the color components of the misaligned underexposed LDR input to obtain a 3-channel HDR image. Existing deep-neural-network-based HDR fusion methods fuse misaligned multi-exposed inputs directly. They suffer from visual artifacts that are observed mostly in saturated regions because pixel values are clipped out. Meanwhile, the proposed method reconstructs missing luminance with aligned unsaturated focus pixel image first, and thus, the luma-recovered image provides the cues for accurate color fusion. The experimental results show that the proposed method not only accurately restores fine details in saturated areas, but also produce ghost-free high-quality HDR images without pre-alignment.

Deep Dichromatic Model Estimation Under AC Light Sources.

The dichromatic reflection model has been popularly exploited for computer vison tasks, such as color constancy and highlight removal. However, dichromatic model estimation is an severely ill-posed problem. Thus, several assumptions have been commonly made to estimate the dichromatic model, such as white-light (highlight removal) and the existence of highlight regions (color constancy). In this paper, we propose a spatio-temporal deep network to estimate the dichromatic parameters under AC light sources. The minute illumination variations can be captured with high-speed camera. The proposed network is composed of two sub-network branches. From high-speed video frames, each branch generates chromaticity and coefficient matrices, which correspond to the dichromatic image model. These two separate branches are jointly learned by spatio-temporal regularization. As far as we know, this is the first work that aims to estimate all dichromatic parameters in computer vision. To validate the model estimation accuracy, it is applied to color constancy and highlight removal. Both experimental results show that the dichromatic model can be estimated accurately via the proposed deep network.

Distinct Somatic Alteration Features Identified by Gene Panel Sequencing in Korean Triple-Negative Breast Cancer with High Ki67 Expression.

This study aimed to clarify the genetic difference between Korean triple-negative breast cancer (TNBC) and other breast cancer (BC) subtypes. TNBC was defined as the absence of hormonal receptors and human epidermal growth factor receptor 2 (HER2) amplification. DNA panel of the Ion Torrent Oncomine Comprehensive Assay (OCA) v3 was performed to identify somatic alteration in 48 specimens. In a total of 102 alterations (37 nonsense, 35 missense, 8 frameshift and 22 amplifications), 30 nucleotide alterations (24 nonsense, 1 missense, and 5 frameshift) were newly identified. The eight most commonly altered genes were PIK3CA, TP53, ERBB2, BRCA2, FANCD2, AKT1, BRCA1, and FANCA. TNBC had significantly lower mutation frequency in PIK3CA (TNBC vs. hormone receptor-positive and HER2-negative BC [HRPBC], p = 0.009), but higher mutation frequency in TP53 (TNBC vs. HRPBC, p = 0.036; TNBC vs. hormone receptor-positive and HER2- positive BC [HHPBC], p = 0.004). TNBC showed frequently higher Ki-67 expression than any positive BC (p = 0.004) due to HRPBC (p < 0.001). TNBC with high Ki-67/unmutated PIK3CA/mutated TP53 appears at a younger age (52.2 ± 7.6 years), compared to other subtypes (63.7 ± 11.0 years). TNBC with high Ki-67/unmutated PIK3CA/mutated TP53 may be related to relatively early onset BCThese findings demonstrate the genomic heterogeneity between TNBC and other BC subtypes and could present a new approach for molecular targeted therapy in TNBC patients.

Comparison of Ki-67 Labeling Index Patterns of Diffuse Large B-Cell Lymphomas and Burkitt Lymphomas Using Image Analysis: A Multicenter Study.

Diffuse large B-cell lymphoma (DLBCL) is the most common high-grade B-cell lymphoma found in Korea; it manifests with a variety of cellular morphologies and a high proliferation index. It is difficult to differentiate between DLBCL and Burkitt lymphoma (BL) based on immunohistochemistry, histology, and Epstein-Barr virus infection status owing to the overlap in findings. In this study, we performed comparative morphometric analysis to understand the proportional difference in Ki-67 staining between DLBCL and BL. We analyzed Ki-67-stained slides of 103 DLBCLs and 29 BLs that were pathologically confirmed using a three-tier classification system (negative, 1+, 2+, and 3+) to compare Ki-67 expression between BL and activated B-cell and germinal center B-cell subtypes of DLBCL and DLBCL with high proliferation indices (>90% of 2+ and 3+ cells). Patients with DLBCL were older than those with BL (62.1 versus 51.0 years). The number and proportion of negative cells (passenger and true negative cells) were significantly lower in BLs than those in DLBCLs (337.4, 5.9% versus 690.3, 12.4%). The number and proportion of 3+ cells were significantly higher in BLs than those in DLBCLs (5213.6, 96.3% versus 3132.4, 62.0%). BLs and DLBCLs with a high proliferation index showed similar results as those between BLs and overall DLBCLs. We were able to differentiate BLs and DLBCLs with 98.1% sensitivity and 100.0% specificity using an optimal cut-off of 97.9% of 2+/3+ Ki-67-positive cells. Thus, the Ki-67 labeling index may be a good differential biomarker for DLBCLs and BLs.

Eupatilin Ameliorates Cerulein-Induced Pancreatitis Via Inhibition of the Protein Kinase D1 Signaling Pathway In Vitro.

OBJECTIVE: The aim of this study was to investigate the effects of eupatilin on protein kinase D1 (PKD1) and nuclear factor kappa B (NF-κB) signaling pathways in cerulein-induced in vitro pancreatitis. METHODS: We used collagenase digestion to isolate pancreatic acinar cells from male C57BL/6 mice. In vitro acute pancreatitis was induced by treatment with a supramaximal dose of cerulein. Eupatilin was pretreated before stimulation with cerulein. RESULTS: Eupatilin significantly reduced cerulein-induced amylase release in pancreatic acini. Eupatilin treatment downregulated cerulein-induced expression of interleukin (IL)-1ß, IL-6, and CC chemokine ligands 2 and 5, but it upregulated expression of IL-4 and IL-10. We demonstrated that eupatilin pretreatment attenuated cerulein-induced necrosis in isolated pancreatic acinar cells. This effect of eupatilin was confirmed by lactic dehydrogenase assay, fluorescence-activated cell sorting analysis, and cytopathologic analysis. Eupatilin inhibited cerulein-induced activation of PKD1/NF-κB and the nuclear translocation of NF-κB. CONCLUSIONS: Our data demonstrated that eupatilin is a potential therapeutic candidate for the treatment of pancreatitis through its ability to reduce cellular necrosis and inflammatory responses by inhibition of the PKD1/NF-κB signaling pathway.

Noise-Robust Iterative Back-Projection.

Noisy image super-resolution (SR) is a significant challenging process due to the smoothness caused by denoising. Iterative back-projection (IBP) can be helpful in further enhancing the reconstructed SR image, but there is no clean reference image available. This paper proposes a novel back-projection algorithm for noisy image SR. Its main goal is to pursuit the consistency between LR and SR images. We aim to estimate the clean reconstruction error to be back-projected, using the noisy and denoised reconstruction errors. We formulate a new cost function on the principal component analysis (PCA) transform domain to estimate the clean reconstruction error. In the data term of the cost function, noisy and denoised reconstruction errors are combined in a region-adaptive manner using texture probability. In addition, the sparsity constraint is incorporated into the regularization term, based on the Laplacian characteristics of the reconstruction error. Finally, we propose an eigenvector estimation method to minimize the effect of noise. The experimental results demonstrate that the proposed method can perform back-projection in a more noise-robust manner than the conventional IBP, and harmoniously work with any other SR methods as a post-processing.

A rare case of multiple and huge simple bone cysts in Wilson's disease.

A simple bone cyst is a tumor-like lesion that is not a true neoplasm. It usually presents as a solitary lesion within the proximal humerus and proximal femur. The etiology of simple bone cysts is still to be elucidated. We describe our experience in the successful surgical management of a rare case of two, large-sized, simple bone cysts involving the entire left humerus and right femoral head, respectively, in a 34-year-old male patient with Wilson's disease and concomitant hepatic cirrhosis. We theorized that Wilson's disease could be the cause of the delay or blockage of the natural healing process of the simple long-bone cysts, leading to their abnormal growth and development. To our knowledge, such a combined occurrence of multiple, simple long-bone cysts and Wilson's disease has not been reported in the literature to date.