Concurrence of Marjolin's Ulcer in the Lower Limb in a Patient with Idiopathic Multicentric Castleman Disease: A Case Report.

Idiopathic multicentric Castleman disease (iMCD) is characterized by the benign proliferation of lymphoid cells in multiple regions. However, the co-occurrence of epithelial malignancy and idiopathic multicentric Castleman disease (iMCD) is rarely reported. Herein, we present a case of iMCD mimicking lymph nodal metastasis of Marjolin's ulcer in the lower extremity. A 53-year-old male presented with an unhealed chronic ulcer on the left lower leg and foot accompanied by an enlarged mass in the left inguinal region. Intralesional biopsy was performed, and pathological examination showed squamous cell carcinoma (SCC). Imaged studies revealed left calcaneus bone invasion, and lymph nodal metastasis was suspected by the cancer TNM staging of T4N2M0 pre-operatively. The patient received below-knee amputation and lymph node dissection; intraoperative histological examination showed no lymphatic nodal malignancy and diagnosed the patient as having iMCD with lymphadenopathy. The patient recovered uneventfully and was referred to a hematologist for further treatment.

Successful management of type IV hypersensitivity reactions to human insulin analogue with injecting mixtures of biphasic insulin aspart and dexamethasone.

Although hypersensitivity reaction to insulin was supposed to be less-frequent with current insulin analogue, case reports with different types of allergic reactions to insulin analogue were still reported. The most common form is type I hypersensitivity reaction with IgE-mediated. Besides, type III (IgG and IgM-mediated) and type IV (T-cell mediated delayed reaction) hypersensitivity reactions were also reported. Here we presented a long-standing type 2 diabetes with insulin requirements with hypersensitivity reactions to insulin actrapid, insulin aspart, insulin glargine, insulin detemir, and biphasic insulin aspart 30. Insulin desensitization was performed as initial management but failed as skin biopsy with immunohistochemical staining proved type IV hypersensitivity reaction. We continued with the next treatment approach using subcutaneous injection with the mixture of biphasic insulin aspart 30 and dexamethasone to alleviate allergy, and the result was successful with steroid-free biphasic insulin aspart 30 injection eight months later. Besides, the treatment effect had lasted after ten years even with switched type of insulin analogue from biphasic insulin aspart 30 to insulin glargine and insulin aspart. The case report demonstrated a good example of how clinicians deal with the rare but important questions of hypersensitivity reactions to insulin analogue.

Lingual leiomyomatous hamartoma with bifid tip and ankyloglossia in a patient without oral-facial-digital syndrome: a case report and literature review.

Here is a rare case of lingual leiomyomatous hamartoma (LLH) with bifid tongue tip and tongue-tie in a patient with non-oral-facial-digital syndrome (OFDS). A 29-year-old male consulted for a painless tumor over the midline of the tongue dorsum measuring 2 × 1.5 cm. The tumor was excised and the tongue-tie was corrected. Diagnosis of LLH was based on histo-pathologic and immuno-histochemical studies. The epidemiologic data and differential diagnosis of LLH, as well as related literature, are discussed. To date, only 14 cases of LLH have been reported in English literature. This may be the first reported case of LLH with bifid tip and ankyloglossia in a non-OFDS patient.

Primary small cell carcinoma of kidney after renal transplantation: a case report and literature review.

Extrapulmonary small cell carcinoma (EPSCC) is a rare neoplasm comprising 2.5% to 5% of small cell carcinomas (SCCs). Bladder SCC is the most common site of genitourinary tract. Primary renal SCC is extremely rare. We report a case of primary SCC of the kidney which is rarely reported in the urinary tract and presents an aggressive clinical picture. A 59-year-old female visited a urologic clinic with complaint of persistent left flank soreness 10 years after undergoing renal transplantation. Abdominal computed tomography showed a left renal pelvis tumor. After the patient received left nephroureterectomy with bladder cuff resection, her pathology results showed SCC. After surgery, she received adjuvant systemic chemotherapy, and her recovery has been uneventful as of 8 months. Primary renal SCC presents with an advanced tumor stage and a short median survival period, therefore early intervention and close follow-up are recommended.

An Optimized Melanin Depigmentation Method for Histopathological and Immunohistochemical Staining of Formalin-Fixed Paraffin-Embedded Tissues.

Introduction. The difficulty in diagnosis of severe melanocytic lesions is a problem to be overcome in pathological practice. Melanin bleaching is an effective approach to ameliorate melanin disturbances in severely pigmented lesions. Although various methods for improving melanin pigmentation in immunohistochemical staining have been reported, these depigmentation methods still need to be optimized and standardized. In this study, the coloring efficiency of 3,3'-diaminobenzidine (DAB) and alkaline phosphatase (AP) after melanin depigmentation was compared under the automatic immunohistochemical staining platform. Methods. The applicability of the optimized depigmentation method was validated in 10 formalin-fixed paraffin-embedded (FFPE) blocks of ocular melanoma tissues. Specimens were demelaninized with 10% hydrogen peroxide at 60°C for immunohistochemical staining (Melan-A and SOX10), and tissue chromogenic staining was performed with DAB and AP detection systems, respectively. Results. The optimized depigmentation method including immunohistochemistry (IHC) could be completed in 3 h, effectively preserving cell morphology and immunoreactivity. Among these, the color-rendering effect and contrast of AP are better than DAB. Conclusion. This optimized method can effectively remove melanin and improve the accuracy of IHC staining interpretation. AP staining has better visibility and readability without the interference of residual melanin. The comparison results showed that after melanin depigmentation, the immunohistochemical staining agent was replaced with red AP, which avoided the misjudgment caused by brown DAB when melanin depigmentation was incomplete. This improved method can be applied to future histopathological and immunohistochemical staining of melanin-deposited tissues.

In vivo 1H MRS for musculoskeletal lesion characterization: which factors affect diagnostic accuracy?

In vivo (1)H MRS is a noninvasive imaging technique for the identification of malignancy. Musculoskeletal lesions vary in their composition, causing field inhomogeneity and magnetic susceptibility effects which may be technical and diagnostic challenges for MRS. This study investigated the factors that affect diagnostic accuracy in the use of MRS for the characterization of musculoskeletal neoplasms. During a 7-year period, 210 consecutive patients with musculoskeletal lesions larger than 1.5 cm in diameter were examined. MRS of a single-voxel point-resolved spectroscopy sequence with TE = 135 ms was undertaken using a 1.5-T scanner. Lesions with a choline signal-to-noise ratio larger than 3.0 were considered to be malignant tumors. The diagnostic accuracy was calculated for all lesions and for subgroups on the basis of lesion type (bone and soft tissue), lesion composition (mixed and solid nonsclerotic), lesion size (≤4, >4-10 and >10 cm), MR scanner (MR scanner 1 and 2) and selected voxel size (≤3, >3-8 and >8 cm(3)). Multivariate logistic regressions were performed to estimate the associations between each factor and diagnostic accuracy. The diagnostic accuracy was 73.3% for all lesions. The accuracy was 54.4% for mixed lesions and 80.4% for solid nonsclerotic lesions (p < 0.001). The diagnostic accuracy was lower for larger lesions [86.8% for lesions of ≤4 cm, 71.6% for lesions of >4-10 cm (p = 0.04) and 63.6% for lesions of >10 cm (p = 0.007)]. There was no difference in diagnostic accuracy for bone versus soft-tissue lesions or as a function of MR scanner or voxel size. By the use of multivariate logistic regression, a solid nonsclerotic lesion was 3.15 times (95% confidence interval, 1.59-6.27) more likely than a mixed lesion to have a diagnosis (p = 0.001). MRS can be used to characterize musculoskeletal lesions, particularly solid nonsclerotic lesions.

A mucormycosis case in a cirrhotic patient successfully treated with posaconazole and review of published literature.

Mucormycosis is an invasive fungal infection associated with a high mortality rate, especially in immunocompromised hosts. Mucormycosis rarely occurs in cirrhotic patients. Here, we report a case of mucormycosis with underlying liver cirrhosis and diabetes mellitus. The patient suffered from maxillary sinusitis and osteomyelitis, and the infection was successfully treated with antifungal agents, surgical debridement, and hyperbaric oxygen therapy. The antifungal treatments used were liposomal amphotericin B, itraconazole, and posaconazole. Although our patient had liver cirrhosis (Child-Pugh classification B), no hepatic decompensation was developed during the treatment course of posaconazole. This is the first report of the safe and effective use of posaconazole for the treatment of mucormycosis in a cirrhotic patient.

IgG4-related disease mimicking renal pelvis tumor with peritoneal carcinomatosis.

IgG4-RD may rarely present as a retroperitoneal fibrosis, mimicking carcinomatosis. Clinicians should consider this disease when encountering an idiopathic peritoneal and retroperitoneal fibrosis with renal involvement and hydronephrosis.

Cocaine-related vasculitis causing scrotal gangrene.

OBJECTIVE: To describe a case of possible cocaine-related vasculitis resulting in gangrene of the scrotum and review the literature on cocaine-related vascular complications. CASE SUMMARY: A 22-year-old male presented with fever, painful swelling of the right scrotum, and a blackened ulcerated lesion on the right scrotum 3 hours after smoking crack cocaine. Blood and urine cultures and serologic tests were negative, and the D-dimer level was normal. Echocardiogram showed no evidence of vegetation and results of a chest X-ray were normal. Despite treatment for presumptive orchitis with intravenous levofloxacin 750 mg/day, the ulcerated lesion of the right scrotum progressed the next day. The patient received debridement of the necrotic tissue and pathology revealed some degenerated vessels with fibrinoid deposits and inflammatory infiltrates, suggestive of vasculitis. No further tenderness and swelling of the scrotum occurred and the patient was discharged after 10 days. When he returned for follow-up, the wound had healed completely. DISCUSSIONS: Cocaine-related vascular complications, including ischemic stroke, myocardial infarction, and peripheral occlusive disease, all of which have various possible mechanisms, are of clinical importance. Vasculitis is one of the mechanisms of vascular complications associated with cocaine use. According to the Naranjo probability scale, cocaine was the possible causative agent of the patient's vasculitis, which led to scrotum gangrene. To our knowledge, this is the first report in the literature of scrotum gangrene developing after crack cocaine was smoked. CONCLUSIONS: This case report describes a rare occurrence of gangrene of the scrotum associated with cocaine smoking, which is consistent with cocaine's potent vasoconstrictive activity. Cocaine abuse has the potential to cause clinically significant ischemic events anywhere in the body, independent of the method of administration.

Medullary thyroid carcinoma with poor differentiation and atypical radiographic pattern of metastasis.

A diagnosis of sporadic medullary thyroid carcinoma (MTC) is complicated. On first diagnosis it may present with distant metastasis. There has been inconsistency regarding metastatic MTC tissue expression of calcitonin, its tumor marker. Adding to the difficulty is the fact that the radiographic pattern of pulmonary metastasis from MTC may vary substantially among patients. Herein is reported the case of a 73-year-old man who presented with two ill-defined pulmonary opacities, clinically resembling primary lung carcinoma. MTC was diagnosed on histopathology of tissue obtained from a total thyroidectomy. The pulmonary biopsy specimens were confirmed to be MTC metastasis on positive immunohistochemical staining of chromogranin-A and synaptophysin, even though only a few cells were stained for calcitonin. To the authors' knowledge this is the first reported case of MTC presenting initially as complex pulmonary metastasis with weakened expression of calcitonin in the metastatic lesion.

Lingual carcinoma metastases presenting as spontaneous pneumothorax.

Spontaneous pneumothorax is a rare manifestation of primary lung cancer or metastasis. We report a 39-year-old man with well-differentiated squamous cell carcinoma of the tongue and cervical lymph node metastases. He developed lung metastases and spontaneous pneumothorax 22 months later after intra-arterial infusion chemotherapy. The patient was managed with partial lung resection under thoracotomy. The pneumothorax resolved completely after the operation. Histological examination demonstrated metastatic squamous cell carcinoma, which had led to a bronchopleural fistula with subsequent induction of pneumothorax. The patient recovered uneventfully and continued to receive adjuvant chemotherapy in the oncology surgery outpatient department. Unfortunately, the tumors of the tongue and cervical lymph nodes progressively enlarged despite treatment. Eventually, the patient died of respiratory failure 5 months later. In most of the previously reported cases, pulmonary metastases associated with spontaneous pneumothorax usually originate from osteogenic or soft-tissue sarcomas. Although rare, pulmonary metastasis should be considered in the etiology of spontaneous pneumothorax. Despite advanced disease, surgical treatment may be feasible.

Eccrine porocarcinoma of the auricle: a case report.

Eccrine porocarcinoma (EP) is a rare skin malignant lesion representing 0.005-0.01% of all cutaneous tumors. It is a tumor that most commonly present in elderly people aged over 60 years. Approximately 250 cases of EP have been reported since this disease was first described in 1963. However, only three cases occurring specifically on the ear (including the current case) have been documented in the literature to date. Based on the rarity of EP of the ear, we present this 78-year-old man with EP on the right ear lobule, which was diagnosed accidentally during the management of other unrelated problems. The etiology, diagnosis, treatment and prognosis of this disease are discussed, with a brief review of the literature in this report.

Pioglitazone retrieves hepatic antioxidant DNA repair in a mice model of high fat diet.

BACKGROUND: Pioglitazone was reported to improve hepatic steatosis and necroinflammation in human studies. To investigate whether the hepato-protective effect of pioglitazone was associated with an improvement of antioxidant defense mechanism, oxidative DNA damage and repair activity were determined in a high fat diet model. Male C57BL/6 mice were respectively fed with a 30% fat diet, the same diet with pioglitazone 100 mg/kg/day, or a chow diet as control for 8 weeks. Tissue oxidative stress was indicated by malondialdehyde concentration. Oxidative DNA damage was detected by immunohistochemical 8-oxoG staining. Enzymatic antioxidant defense was detected by the real-time PCR of superoxide dismutase (Sod1, Sod2) and DNA glycosylase (Ogg1, MutY). Oxidative DNA repair was detected by immunohistochemical staining and western blotting of OGG1 expression. RESULTS: Our results show that hepatic steatosis was induced by a high-fat diet and improved by adding pioglitazone. Malondialdehyde concentration and 8-oxoG staining were strongly increased in the high-fat diet group, but attenuated by pioglitazone. Gene expressions of antioxidant defense mechanism: Sod1, Sod2, Ogg1 and MutY significantly decreased in the high-fat diet group but reversed by pioglitazone co-administration. CONCLUSION: The attenuation of hepatic oxidative DNA damage by pioglitazone in a high-fat diet may be mediated by up-regulation of the antioxidant defense mechanism and oxidative DNA repair activity. The diminution of oxidative damage may explain the clinical benefit of pioglitazone treatment in patients with non-alcoholic fatty liver disease.

A single lesion showing features of pigmented eccrine poroma and poroid hidradenoma.

Poroid hidradenoma (PH) is a variant of poroma. This entity was defined by Abenoza and Ackerman in 1990. This neoplasm shows architectural characteristics of hidradenoma (tumor cells confined entirely within the dermis in both solid and cystic components) and cytologic characteristics of poroid neoplasm (poroid and cuticular cells, the latter showing ductal differentiation). We herein document a case of single poroid lesion with the features of both eccrine poroma and PH. The patient was a 55-year-old woman with a pigmented nodular lesion on her upper back for 7 years. The histopathologic features of the lesion were consistent with those of eccrine poroma and PH. Unlike most eccrine poromas, this case was pigmented, clinically and microscopically.

Duodenal metastasis of malignant pleural mesothelioma.

Metastatic malignant mesothelioma of the pleura is uncommon at the time of initial diagnosis. The gastrointestinal lumen is rarely found at autopsy in patients with widespread disease. Here, we describe an extremely rare case of isolated duodenal metastasis of sarcomatoid mesothelioma of the pleura in a 73-year-old man, without memory of any direct exposure to asbestos. The possibility of gastrointestinal tract metastasis should be considered in the presence of anemia or positive occult blood test in patients with malignant pleural mesothelioma.

Melanin Bleaching With Warm Hydrogen Peroxide and Integrated Immunohistochemical Analysis: An Automated Platform.

OBJECTIVE: Diagnosing melanocytic lesions is among the most challenging problems in the practice of pathology. The difficulty of physically masking melanin pigment and the similarity of its color to commonly used chromogens often complicate examination of the cytomorphology and immunohistochemical staining results for tumor cells. Melanin bleach can be very helpful for histopathological diagnosis of heavily pigmented melanocytic lesions. Although various depigmentation methods have been reported, no standardized methods have been developed. This study developed a fully automated platform that incorporates hydrogen peroxide-based melanin depigmentation in an automated immunohistochemical analysis. METHODS AND MATERIALS: The utility of the method was tested in 1 cell block of malignant melanoma cells in pleural effusion, 10 ocular melanoma tissue samples, and 10 cutaneous melanoma tissue samples. Our results demonstrated that the proposed method, which can be performed in only 3 hours, effectively preserves cell cytomorphology and immunoreactivity. RESULTS: The method is particularly effective for removing melanin pigment to facilitate histopathological examination of cytomorphology and for obtaining an unmasked tissue section for immunohistochemical analysis.

Hepatic steatosis and fibrosis in chronic hepatitis C in Taiwan.

Hepatitis C virus (HCV) infection has been associated with hepatic steatosis. However, the role of hepatic steatosis in the pathogenesis of HCV infection remains controversial. In our study, 425 consecutive HCV-viremic patients with biopsy-proven chronic hepatitis C (male, 264; mean age, 49.0 years) were enrolled. Scoring of hepatic steatosis was based on the method described by Kleiner and on histopathology performed using the Knodell and Scheuer systems. HCV RNA level and genotypes were determined at the time of biopsy. Hepatic steatosis was observed in 30.8% of patients, including 113 mild, 16 moderate, and 3 with severe hepatic steatosis. Patients with a body mass index (BMI) <23 kg/m(2) had a significantly lower rate (18.9%) of hepatic steatosis (P<0.001). Hepatic steatosis did not correlate with the hepatic necroinflammatory activity, but was related to hepatic fibrosis (P=0.035). Hepatic steatosis was also not associated with HCV RNA level, and the distribution was similar between patients with HCV genotype 1 and genotype 2 infection. According to multivariate analysis, BMI is the strongest risk factor associated with hepatic steatosis, followed by hepatic fibrosis and triglyceride level with odds ratios (95% confidence intervals) of 2.51 (1.49-4.23), 2.06 (1.14-3.70), and 1.02 (1.01-1.03), respectively. Hepatic steatosis was associated with being overweight, hepatic fibrosis, and triglyceride level in chronic hepatitis C.