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BACKGROUNDMost melanomas identified in the stomach are metastatic. The primary gastric melanoma (PGM) is extremely rare. As such, clinical reports of PGM are scarce in the literature, lending to the challenge of diagnosis and treatment.CASE SUMMARYA 31-year-old woman presented with a 1-mo history of dysphagia but no symptoms of abdominal pain, abdominal distension, nausea, vomiting, hematemesis, or melena. The patient reported an unintentional weight loss of 6 kg within that time. History-taking revealed no previous medical conditions or surgical events. Abdominal computed tomography at a local hospital had suggested gastric tumor. Endoscopic examination in our hospital found a large, irregular, black mass. Subsequent laparoscopic exploration found the tumor on the side of the stomach fundus penetrating through the serosa, and enlarged lymph nodes (groups 1, 3, 7, and 9) fused into a mass, surrounding the peripheral artery and inseparable. Postoperative immunohistochemistry suggested gastric malignant melanoma. Positron emission tomography-computed tomography confirmed PGM. Treatment with programmed cell death protein 1 antagonist (toripalimab) plus chemotherapy (paclitaxel) was initiated but discontinued upon tumor bleeding. At the last telephone follow-up, the patient reported poor general condition but was alive.CONCLUSIONAlthough unresolved and ongoing, this rare case of PGM expands the overall knowledge about this rare tumor’s diagnosis and management.  相似文献   

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BACKGROUNDCerebral proliferative angiopathy (CPA) is a rare vascular disease characterized by the presence of diffuse vascular proliferation, progressive vascular hyperflow and vasodilation of multiple vessels in the normal brain parenchyma. Unlike cerebral arteriovenous malformations, CPA has a mixed appearance between that of lesions with cell proliferation and endothelial proliferation. To date, the pathogenesis of CPA is unclear, in which changes induced by cortical ischemia in the elastic layer of the blood supply artery and smooth muscle cells may be involved.CASE SUMMARYIn this article, we retrospectively analyzed a case of hemorrhagic transformation of ischemic CPA diagnosed by digital subtraction angiography and reviewed the related literature for further exploration of its pathogenesis, diagnosis and treatment.CONCLUSIONThe information in the present case report may facilitate further clinical research on this cerebrovascular disease.  相似文献   

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BACKGROUNDSmall cell carcinoma (SCC) is a malignant tumour that is frequently accompanied by extensive metastasis. Primary renal SCC has typical characteristics related to SCC and is extremely rare, with no uniform treatment standard. Clinical treatment is mainly based on the literature. Here we report the diagnosis and treatment of an interesting case of primary renal SCC.CASE SUMMARYWe report a tortuous course of treatment for a 68-year-old man. Four years before diagnosis, the patient developed continuous gross haematuria, during which he underwent several ureteral biopsies, ureteral stricture relief, and urine exfoliated cell examinations; however, SCC was not confirmed. One month before radical resection of the renal pelvic carcinoma, the severe haematuria recurred. Computed tomography revealed transitional cell carcinoma in the right kidney and right upper ureter. A preoperative examination exluded the possibility of a pulmonary origin of the tumour, and primary renal SCC was diagnosed. The postoperative pathology findings were suggestive of SCC. The patient was treated with combined chemotherapy but died of tumour progression at 7 mo postoperative.CONCLUSIONOur patient''s disease onset in the context of a succession of regular testing and the fact that it occurred so quickly with perirenal encroachment immediately after diagnosis reveals the cruel and unforgiving side of the disease. Furthermore, patients with poor comprehensive treatment results require new treatment regimens.  相似文献   

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BACKGROUNDPituitary metastasis is an uncommon manifestation of systemic malignant tumors. Moreover, hyperprolactinemia and overall hypopituitarism caused by metastatic spread leading to the initial symptoms are rare.CASE SUMMARYA 53-year-old male patient was admitted to our hospital with complaints of bilateral blurred vision, dizziness, polyuria, nocturia, severe fatigue and somnolence, decreased libido, and intermittent nausea and vomiting for more than 6 mo. During the last 7 d, the dizziness had worsened. Laboratory investigations revealed overall hypofunction of the pituitary gland, but the patient had an elevated serum prolactin level (703.35 mg/mL). Preoperative magnetic resonance imaging revealed a tumor in the sellar region, accompanied by intratumoral hemorrhage and calcification. Thus, transnasal subtotal resection of the lesion in the sellar region was performed. The histopathological and immunohistochemical examinations of the resected lesion revealed metastasis of lung adenocarcinoma to the pituitary gland. Oral hydrocortisone (30 mg/d) and levothyroxine (25 mg/d) were given both pre- and postoperatively. Post-operatively, the clinical symptoms were significantly improved. However, 4 mo following the surgery, the patient succumbed due to multiple organ failure.CONCLUSIONHyperprolactinemia is one of the markers of poor prognosis in patients with carcinoma that metastasizes to the pituitary gland. Exogenous hormone supplementation plays a positive role in relieving the symptoms of patients and improving quality of life.  相似文献   

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BACKGROUNDAlthough the imaging features of coronavirus disease 2019 (COVID-19) are starting to be well determined, what actually occurs within the bronchi is poorly known. Here, we report the processes and findings of bronchoscopy in a patient with COVID-19 accompanied by respiratory failure.CASE SUMMARYA 65-year-old male patient was admitted to the Hainan General Hospital on February 3, 2020 for fever and shortness of breath for 13 d that worsened for the last 2 d. The severe acute respiratory syndrome coronavirus 2 nucleic acid test was positive. Routine blood examination on February 28 showed a white blood cell count of 11.02 × 109/L, 86.9% of neutrophils, 6.4% of lymphocytes, absolute lymphocyte count of 0.71 × 109/L, procalcitonin of 2.260 ng/mL, and C-reactive protein of 142.61 mg/L. Oxygen saturation was 46% at baseline and turned to 94% after ventilation. The patient underwent video bronchoscopy. The tracheal cartilage ring was clear, and no deformity was found in the lumen. The trachea and bilateral bronchi were patent, while the mucosa was with slight hyperemia; no neoplasm or ulcer was found. Moderate amounts of white gelatinous secretions were found in the dorsal segment of the left inferior lobe, and the bronchial lumen was patent after sputum aspiration. The right inferior lobe was found with hyperemia and mucosal erosion, with white gelatinous secretion attachment. The patient’s condition did not improve after the application of therapeutic bronchoscopy.CONCLUSIONFor patients with COVID-19 and respiratory failure, bronchoscopy can be performed under mechanical ventilation to clarify the airway conditions. Protection should be worn during the process. Considering the risk of infection, it is not necessary to perform bronchoscopy in the mild to moderate COVID-19 patients.  相似文献   

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BACKGROUNDAngiomyolipoma (AML), the most common benign tumor of the kidney, is usually composed of dysmorphic blood vessels, smooth muscle, and mature adipose tissue. To our knowledge, AML with cystic degeneration has rarely been documented. Cystic degeneration, hemorrhage, and a lack of fat bring great challenges to the diagnosis.CASE SUMMARYA 60-year-old man with hypertension presented with a 5-year history of cystic mass in his left kidney. He fell 2 mo ago. A preoperative computed tomography (CT) scan showed a mixed-density cystic lesion without macroscopic fat density, the size of which had increased compared with before, probably due to hemorrhage caused by a trauma. Radical nephrectomy was performed. Histopathological studies revealed that the lesion mainly consisted of tortuous, ectatic, and thick-walled blood vessels, mature adipose tissue, and smooth muscle-like spindle cells arranged around the abnormal blood vessels. The tumor cells exhibited positivity for human melanoma black-45, Melan-A, smooth muscle actin, calponin, S-100, and neuron-specific enolase, rather than estrogen receptor, progesterone receptor, CD68, and cytokeratin. The Ki-67 labeling index was less than 5%. The final diagnosis was a fat-poor renal AML (RAML) with prominent cystic degeneration.CONCLUSIONWhen confronting a large renal cystic mass, RAML should be included in the differential diagnosis.  相似文献   

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Currently, clean intermittent self-catheterization (CISC) is the most prevalent method of bladder management in patients with spinal cord injury (SCI) at discharge from rehabilitation centers. However, half of the patients discontinue using CISC and change to other methods of bladder management several months postdischarge despite the fact that it the best way to prevent urinary tract complications. Few studies, however, report the long-term consequences of CISC. In this case, we present a woman in her early fifties who had sustained thoracic SCI and had continued using CISC for 27 years without developing any complications. The possible reasons for her success were absence of incontinence because of underactive and normal capacity bladder; normal upper-extremity functions and absence of marked spasticity of lower extremities that facilitated CISC technique; and absence of sociovocational problems, enabling her to keep proper intervals between catheterizations each day. This case indicates that CISC is useful for long-term bladder management in patients with SCI, even for 25 years or more. Long-term outcomes of CISC and factors leading to success need to be delineated in future studies with larger samples.  相似文献   

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BACKGROUNDAppendiceal mucocele is a rare disease that causes obstructive dilatation of the appendix due to the intraluminal accumulation of mucin. We report a case of endoscopic diagnosis and treatment of an appendiceal mucocele.CASE SUMMARYA 47-year-old man presented with a protrusion around the orifice of the appendix discovered by colonoscopy incidentally. He was admitted to our hospital for a routine checkup without any symptoms. Abdominal computed tomography showed a cystic mass approximately 3 cm in diameter with fat stranding. The preoperative diagnosis was non-neoplastic appendiceal mucocele, and endoscopic treatment was performed. The endoscopic findings and pathological results supported our preoperative diagnosis. The endoscopic treatment of appendiceal mucocele was feasible and effective, which was confirmed by repeated endoscopy and post-operative computed tomography after 7 mo. CONCLUSIONEndoscopic therapy provides a new method for the treatment of appendiceal mucocele.  相似文献   

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BACKGROUND Solitary respiratory papilloma is a rare epithelial tumor that can be categorizedinto multiple subtypes depending on tumor location,histological presentationand number.The glandular type is the rarest,with only 30 cases available withinthe field.Hence,information on its identification and treatment is limited.In thisreport,we discuss the diagnostic strategy and management of glandularpapilloma,along with a review of the literature.CASE SUMMARY We describe a male 44-year-old nonsmoker who presented with a persistentcough and recurrent pneumonia,which he had experienced for over 2 years.Asolitary pulmonary nodule with an endobronchial lesion was found via computedtomography of the chest.After a biopsy was obtained,no definite diagnosiscould be made.Glandular papilloma of the lung was confirmed via videoassistedthoracoscopic anatomic resection of the right lower lobe of the lung.Thepatient remained disease-free after 6 mo follow up.CONCLUSION Minimally invasive surgery is feasible for the surgical resection of endobronchialglandular papilloma.Although rare,glandular papilloma should be consideredin patients with infection or endobronchial lesions.  相似文献   

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BACKGROUNDKallmann syndrome (KS), also known as hypogonadotropic hypogonadism (HH) or olfactory-gonadal dysplasia, is a genetic condition in which the primary symptom is a failure to begin puberty or a failure to fully complete it. It occurs in both males and females and has the additional symptoms of hypogonadism and almost invariably infertility. The condition has a low prevalence that is estimated to be 1 in 4000 for male HH cases overall and 1:50000 for KS. It is three to five times more common in males than females. Whether this is a true sex imbalance or a reflection of how difficult KS/HH is to diagnose correctly in males vs females has yet to be fully established.CASE SUMMARYThis article reports a 26-year-old male presenting with delayed puberty. The synthetic decapeptide luteinizing hormone-releasing hormone stimulation test showed that the secretion levels of follicle-stimulating hormone and luteinizing hormone were delayed. The eigengenes commonly associated with idiopathic HH (IHH) were screened, and an X-linked recessive (KAL-1) mutation was found. His gonadotropin and testosterone levels increased significantly after pulsatile gonadotropin-releasing hormone (GnRH) subcutaneous therapy by pump. A relevant literature review on the recent advances in the diagnosis and treatment of KS and genetic counseling was conducted.CONCLUSIONKS is caused by a KAL-1 mutation that follows an X-linked recessive inheritance pattern. Pulsatile GnRH subcutaneous therapy by pump was effective in this patient.  相似文献   

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BACKGROUNDTreatment of synchronous multiple primary malignancies is quite often very challenging. Herein, we report on a rare case of synchronous multiple primary malignancies in the esophagus, stomach, and jejunum.CASE SUMMARYA 50-year-old man who was a heavy drinker and smoker with a poor diet, and had a family history of cancer sought treatment due to dysphagia lasting for 4 mo. He was finally diagnosed with lower esophageal squamous cell carcinoma (pT3N2M0, G2, stage IIIB), gastric angular adenocarcinoma (pT3N2M0, G2-G3, stage IIIA) with greater omental lymph node metastasis, and jejunal stromal tumor (high risk). The high-risk jejunal stromal tumor was found during surgery. In spite of radical resection and adjuvant chemotherapy, lymph node metastasis occurred 21 mo later. The patient responded poorly to additional chemotherapy and refused further examination and therapy. He died of widespread metastases 33 mo after surgery.CONCLUSIONThis case indicates a poor prognosis of synchronous multiple advanced primary malignancies and the importance of comprehensive assessment in the population at high risk for cancer.  相似文献   

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BACKGROUNDPrimary hypertension is a common clinical disease. Pheochromocytoma and paraganglioma is a rare cause of secondary hypertension. The diagnosis of the latter is still difficult, and the relationship between the two is not clear. The successful diagnosis of this case confirmed that standardized etiological investigation of secondary hypertension is necessary, contributes to the accurate diagnosis of rare diseases, and is conducive to the formulation or optimization of treatment plans. It shows an example of the coexistence of primary hypertension and secondary hypertension.CASE SUMMARYThe patient was a 54-year-old male and was hospitalized with high blood pressure for 4 years. The patient’s blood pressure was measured at 150/100 mmHg during physical examination 4 years ago and had no paroxysmal or persistent elevated blood pressure, no typical triad of headache, palpitation, and sweating, without postural hypotension. After taking nifedipine sustained release tablets intermittently, the blood pressure did not meet the standard. Physical examination revealed blood pressure of 180/120 mmHg. There was no abnormality in cardiopulmonary and abdominal examination. The results of blood and/or urinary catecholamines/metanephrine and normetanephrine before and after operation were normal. Fundus examination revealed retinal arteriosclerosis in both eyes. There was a history of paraganglioma diagnosed by pathology after retroperitoneal tumor resection, a family history of hypertension, and a history of passive smoking. The clinical diagnosis was subclinical paraganglioma, primary hypertension, and hypertensive fundus lesions. The patient’s blood pressure was regulated, blood lipid was reduced, and anti-inflammatory, and symptomatic support were given. After treatment, the blood pressure was stable and up to standard without discomfort symptoms.CONCLUSIONSubclinical paraganglioma and primary hypertension can coexist. The holistic thinking in clinical practice is helpful to the early diagnosis of rare diseases.  相似文献   

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BACKGROUNDGranular cell tumor (GCT) of the pancreas is a rare neurogenic tumor. The first case of pancreatic GCT was described in 1975, and up to now, only 7 cases have been reported.CASE SUMMARYA 53-year-old male had a pancreatic mass for 1 mo. He was not treated at the local hospital, but referred to Henan Tumor Hospital for surgery. Preoperative imaging revealed a 2.0 cm × 2.5 cm-sized mass located in the body of the pancreas. At the microscopic level, a large number of eosinophilic particles are present in the oval tumor cells. The immunohistochemistry of this tumor cell display CD56 (+), blood vessels CD34 (+), Ki-67 (+) < 10%, and S-100 (+).CONCLUSIONGCT of the pancreas should be recognized as a preoperative differential diagnosis of pancreatic tumors. Surgical resection of the tumor should be attempted; however, GCT of the pancreas has a certain rate of tumor metastasis and recurrence. Therefore, GCT of the pancreas requires regular and long-term follow-up.  相似文献   

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BACKGROUNDPrimary intramedullary melanocytoma is an exceedingly rare type of primary melanocytic tumor in the central nervous system. Unfortunately, primary intramedullary melanocytoma lacks specificity in clinical symptoms and imaging features and there is currently no standard strategy for diagnosis or treatment.CASE SUMMARYA 52-year-old male patient suffered from weakness and numbness involving the bilateral lower limbs for 18 mo, and defecation and erectile dysfunction for 6 mo. Furthermore, these symptoms started to worsen for the last 3 mo. Preoperative magnetic resonance imaging (MRI) revealed an intramedullary tumor located at the T9-T10 level. In subsequently surgery, the maximal safe resection extent approached to 98%. The lesion was confirmed to be melanocytoma by pathological examination. In addition, the possibility of original melanocytoma outside the spinal cord was excluded after the examination of the whole body. Therefore, a diagnosis of primary intramedullary melanocytoma was established. The patient refused to accept radiotherapy or Gamma Knife, but MRI examination on July 28, 2020 showed no sign of development. In addition, on April 10, 2021, the recent review showed that the disorder of defecation and lower limbs improved further but erectile dysfunction benefited a little from the surgery.CONCLUSIONAfter diagnosing intramedullary melanocytoma by postoperative pathology, the inspection of the whole body contributed to excluding the possibility of metastasis from other regions and further suggested a diagnosis of primary intramedullary melanocytoma. Complete resection, adjuvant radiation, and regular review are critical. In addition, maximal safe resection also benefits prognosis while the tumor is difficult to be resected totally.  相似文献   

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