Kimura's disease of parotid gland presenting as solitary parotid swelling

BACKGROUND: Kimura's disease is a chronic inflammatory disorder of unknown etiology commonly seen among orientals and characterized histologically by lymphatic follicles, vascular proliferation, and marked eosinophilic infiltration. It has a predilection for the head and neck region. The lesion is benign but can be mistaken to be a malignant lesion. METHODS: Between January 1987 and December 1999, eight cases of Kimura's disease were treated at the Department of Plastic Surgery, Hospital Kuala Lumpur. All the patients underwent surgical excision followed by initial high-dose steroid therapy and low-dose maintenance. RESULTS: All the patients are men, seven belong to the Malay race and one is Chinese. The patients' ages at diagnosis are 18 to 46 years. All were initially seen with swelling in the parotid gland, ranging from 1 year to 20 years' duration. Pruritus of the overlying skin was seen in seven patients. Treatment by surgical excision and steroid therapy has led to no recurrences. CONCLUSION: Kimura's disease of parotid gland is rare. It is a chronic inflammatory condition also called eosinophilic hyperplastic lymphogranuloma. Treatment by surgical excision and steroid therapy has led to no recurrences.     

Clinical and pathological study of Kimura's disease with renal involvement

OBJECTIVE: Our objective was to retrospectively analyze the clinical and histological features of 8 Chinese patients with Kimura's disease and renal involvement. METHODS: Eight male patients with Kimura's disease admitted to Jinling Hospital from 1998 to 2006 were included. The diagnoses were confirmed by lymphoid biopsy. Peripheral blood eosinophil, CD4+ and CD8+ T lymphoid cell count and total serum IgE level were examined. Renal biopsy, IL-4 expression cells and IgE-positive cell counts in renal tissue were performed. Follow-up data were recorded in detail. RESULTS: Six patients were observed with the onset symptoms of subcutaneous mass or enlarged lymph nodes, and the other 2 with edema. Constitutional symptoms included bronchial asthma, enteritis, eczema, neuritis and nephrotic syndrome. Renal biopsy revealed mesangial proliferation with or without IgA deposition under immunofluorescence (n=6) and membranous nephropathy (n=2). Tubulointerstitial infiltration of eosinophils was found in 6 patients. IL-4-positive cells in renal interstitium were detected in 5 cases. IgE-positive cells were negative in all cases. Fusion of the epithelial foot process was observed under electron microscopy in patients with mesangial proliferation. All patients were sensitive to treatment with systemic prednisone, but 4 patients had a renal or extrarenal relapse after the dose tapered. Three patients who presented with solitary masses and underwent lymphoidectomy or mass excision were free from renal and extrarenal relapse. All patients had normal renal function at last follow-up. CONCLUSIONS: Mesangial proliferation, eosinophilic infiltration and podocyte confusion are prominent histological features of this cohort of patients. Patients are sensitive to prednisone therapy but apt to relapse. Lymphoidectomy can be helpful to prevent relapse.     

Clinical study of eosinophilic cystitis (2). Clinical observation on 5 cases of eosinophilic cystitis

Clinical observations were performed on 5 cases of eosinophilic cystitis that had been diagnosed by our criteria. In all 5 cases some kind of allergic diseases was found. General urinalysis did not show any definite tendency, but eosinophils in urine were found in 4 cases. Cystoscopy revealed only chronic inflammation and submucosal hemorrhage in 4 cases, and the other case showed ulcerative changes. A positive to immediate skin reaction was seen in 2 of the 4 cases. IgE RAST was positive for mite, house dust and mugwort in 1 of these 2 cases. In this case immediate allergic reaction was suspected as part of the cause of the cystitis.     

Minimal-change nephrotic syndrome associated with subcutaneous eosinophilic lymphoid granuloma (Kimura's disease)

A 29-year-old Japanese male with a 19-year history of subcutaneous eosinophilic lymphoid granuloma (Kimura's disease) was referred to the Nephrology Service of the Nihon University Hospital for evaluation of edema and massive proteinuria. The renal biopsy disclosed minimal glomerular lesions. In this paper a case of nephrotic syndrome associated with eosinophilic lymphoid granuloma is reported.     

复合动脉重建术治疗下肢动脉缺血症：附21例报告

目的：探讨下肢动脉缺血症复合动脉重建术的治疗经验。方法：全组２１例通过多普勒血流仪检测和动脉造影，确定动脉病变范围。复合近远端动脉重建１９例，股－胫后动脉旁路转流２例。结果：术后病死率４８％，截肢率９５％。术后１个月，病人静息痛缓解，肢端溃疡愈合，踝臂指数平均值０９５。平均随访２４４个月，近端动脉重建通畅率１００％，远端动脉重建通畅率８２４％，平均踝臂指数值０８５。结论：复合动脉重建术治疗下肢动脉缺血闭塞症是安全而有效的。手术成功的关键在于重建动脉流入及流出道的通畅     

Carotid body tumor (chemodectoma). Clinicopathologic analysis of ninety cases

The clinicopathologic findings in ninety cases of carotid body tumors seen at the Mayo Clinic from 1931 through 1966 are compared with those in 500 reported cases in the literature. In the ninety cases reviewed here, there were sixty-two men and twenty-eight women.     

胃嗜酸性肉芽肿21例报告

目的探讨胃嗜酸性肉芽肿的诊断和治疗方法。方法对２１例嗜酸性肉芽肿的临床资料进行分析。结果钡餐检查２１例,诊断胃癌７例,胃溃疡１４例。胃镜检查１２例,诊断胃癌５例,胃溃疡７例。１１例外周血中嗜酸性细胞增多。４例行近全胃切除加Ｄ３淋巴结清扫,１７例胃次全切除术。１２例随诊５～１５年,９例健在。结论胃镜活检及术中快速病检可确诊,胃大部切除术是主要的治疗方法     

Kimura disease: a clinicopathologic study of 21 cases

Kimura disease is a rare form of chronic inflammatory disorder involving subcutaneous tissue, predominantly in the head and neck region and frequently associated with regional lymphadenopathy and/or salivary gland involvement. This condition has a predilection for males of Asian descent and may clinically simulate a neoplasm. Kimura disease is sometimes confused with angiolymphoid hyperplasia with eosinophilia, which occurs in the superficial skin of the head and neck region. Although sporadic cases have been reported in non-Asians, there is no large, comprehensive study of Kimura disease in the United States. We report 21 cases with nodal involvement that, histologically, are consistent with Kimura disease. There were 18 males and 3 females (male/female ratio 6:1), 8 to 64 years of age (mean, 32 years), and included 7 Caucasians, 6 Blacks, 6 Asians, 1 Hispanic, and 1 Arabic. Anatomic sites of involvement included posterior auricular (n = 10), cervical (n = 6), inguinal (n = 3), and epitrochlear (n = 2) lymph nodes, with two patients having associated salivary gland involvement. Most (n = 16) cases had peripheral blood eosinophilia. Consistent histologic features were follicular hyperplasia, eosinophilic infiltrates, and proliferation of postcapillary venules. Follow-up data on 18 patients revealed that 13 were alive without disease (3 had recurrence), mean follow-up, 10.9 years; 4 were alive with disease (2 had a recurrence), mean follow-up, 8.8 years; and 1 died with disease (12.7 years). Kimura disease has been described more often in Asians, but it does occur in non-Asians with a similar clinicopathologic presentation. It is a distinctive entity with no known etiology. Kimura disease has characteristic histologic features that are important to recognize and can be used to differentiate it from hypersensitivity and drug reactions and infections.     

Penile cancer Clinicopathologic study of 64 cases

The clinical records of 64 patients with penile cancer admitted to the urologic services at the Presbyterian and Francis Delafield Hospitals from 1947 through 1969 have been reviewed. The pertinent data are analyzed and included in this report. The relationship between cancer of the penis and phimosis, and age and race are discussed. The treatment of choice was partial or total penectomy followed by external radiotherapy. Radical dissection of the inguinal lymph nodes did not seem to improve results while it contributed to increased morbidity. Survival rates were evaluated according to the types and stages of penile cancer, as well as to the methods of treatment. The over-all five-year survival rate was 49 per cent. Autopsy studies in 9 patients who died of penile cancer revealed the inguinal and pelvic lymph nodes were involved in 8 patients.     

Gastroesophageal junction hyperplastic (inflammatory) polyps: a clinical and pathologic study of 46 cases

Hyperplastic (inflammatory) polyps (HPs) of the gastric corpus and antrum typically develop in association with chronic gastritis. However, little is known regarding the etiology, pathologic features, and natural history of HPs of the gastroesophageal junction (GEJ). We have noted, anecdotally, that GEJ HPs often occur in patients without gastric pathology. The aim of this study was to evaluate the clinical, pathologic, and outcome features of patients with HPs of the GEJ, and to compare the data with a control group of individuals with HPs in the gastric corpus or antrum. One hundred thirty-four consecutive polyps of the GEJ were identified by a 5-year search through the pathology files of a major tertiary-care hospital. Of these, 46 (36%) polyps from 46 patients met the pathologic criteria for HPs and formed the basis of this study. The 46 study patients, and their polyps, were evaluated for a wide variety of clinical, endoscopic, and pathologic features including outcome on follow-up endoscopy. The findings were compared with 46 HPs from 46 patients of the distal stomach (antrum or corpus) that were obtained randomly from the same 5-year period. Compared with patients with gastric antral or corpus HPs, patients with HPs of the GEJ were significantly younger in age (mean age, 55.9 y vs. 63.0 y; P=0.04). Pathologically, GEJ HPs showed a significantly higher rate of multilayered epithelium (P=0.06) and association with Barrett esophagus (BE) (P=0.0001) compared with distal gastric HPs. All BE-associated GEJ HPs were associated with either ultrashort (<1 cm) or short segment (1 to 3 cm) BE. All other pathologic variables, including intestinal metaplasia, were similar to those of distal gastric HPs. In a subanalysis, BE-associated GEJ HPs (33% of all GEJ HPs) showed a higher male to female ratio and a higher rate of intestinal metaplasia compared with all other HPs. Furthermore, none of the BE-associated GEJ HPs were associated with chronic active gastritis versus the non-BE-associated GEJ HPs, although this was not statistically significant. Only 1 HP (from the GEJ) from both the study and control groups was associated with a neoplasm (signet-ring cell carcinoma). On follow-up, 1 patient with a GEJ HP and 4 with distal gastric HPs developed recurrent HPs and none of the patients from either patient group developed dysplasia or carcinoma. In conclusion, unlike HPs of the gastric corpus or antrum, a significant proportion of HPs of the GEJ arise in association with BE and without gastric pathology. In patients with BE, the columnar-lined segment is often ultrashort, and thus, an HP may be the first clinical/endoscopic manifestation of that disorder.     

Primary adenocarcinoma of urinary bladder. Clinicopathologic study of 16 cases

The clinicopathologic and immunohistochemical features of 16 pure adenocarcinomas primary in the urinary bladder were reviewed. Only 3 patients were found to have disease confined to the urinary bladder. Of 13 cases with follow-up only 3 are free of disease. Histologically, the tumors were classified as signet ring cell (3), colloid (3), colonic type (5), clear cell (1), and not otherwise specified (NOS, 4). Immunohistochemically, all tumors but one colloid carcinoma were immunoreactive for cytokeratin and epithelial membrane antigen, and most tumors were likewise immunoreactive for carcinoembryonic antigen. Eight cases were immunoreactive for Leu M1 antigen. Prostate specific antigen, S-100 protein, and placental alkaline phosphatase were uniformly negative. No correlation between immunohistochemical profile and histologic type or clinical outcome was found. The utility of immunohistochemistry and other pathologic findings is reviewed.     

阴囊Paget病的临床病理研究

目的 探讨阴囊Ｐａｇｅｔ病的临床特点与影响预后的因素。方法 对１１例Ｐａｇｅｔ特病患者的临床病理资料进行回顾性研究。结果 阴囊Ｐａｇｅｔ病易累及毛囊和汗腺但不易发生转移。扩大的病灶切除术是原发及局部再发Ｐａｇｅｔ病治疗的首选方法。１０例患者平均随访３８．６个月,无死亡病例。     

Lhermitte-Duclos disease (diffuse hypertrophy of the cerebellum). Report of two cases

Summary Two cases of Lhermitte-Duclos disease or diffuse hypertrophy of the cerebellum are presented. This brings the total number of such cases reported in the literature to 42. Pathologically the disease is characterized by a circumscribed cerebellar lesion consisting of thickening of the cortex with closely packed dysplastic ganglion cells in the granular layer and with large myelinated axons in the molecular layer. Purkinje cells are missing and the central white matter is greatly reduced.The first patient, a man 39 years of age, had, in association to the cerebellar lesion, a megalencephalic brain (2320 g). He had suffered from epilepsy since he was 24-years-old and died with metastasizing colon carcinoma. An electron microscopic study of the cerebellar lesion disclosed perikarya containing large amounts of rough endoplasmic reticulum, cell processes filled with coated and dense core vesicles. In addition, there were numerous enlarged myelinated axons in the molecular layer.The second patient, a woman 74 years of age, had a small area of diffuse hypertrophy of the left cerebellar hemisphere measuring one centimeter in diameter.Specific symptoms were very scant in the first case and absent in the second one indicating a very slow evolution — if any — of the disease process. The etiology and pathogenesis remain unknown. However, the organoid structure of the lesion, the frequent association of megalencephaly and other congenital abnormalities, and the occurrence of familiar cases would favor the disease being basically a developmental disorder.Dedicated to Professor Dr. K. J. Zülch on the occasion of his 70th birthday.     

Clinicopathologic spectrum of the so-called calcifying odontogenic cysts: a study of 21 intraosseous cases with reconsideration of the terminology and classification

The so-called calcifying odontogenic cyst (COC) represents a heterogeneous group of lesions that exhibit a variety of clinicopathologic and behavioral features. Because of this diversity, there has been confusion and disagreement on the terminology and classification of these lesions. We reviewed the clinicopathologic features of 21 intraosseous cases that were previously diagnosed as COC or under related diagnostic terms. Based on the biologic behavior, the lesions of the present series were divided into three subgroups: cyst, benign tumor, and malignant tumor. Sixteen cases (nine men and seven women) proved to be unicystic lesions with (five cases) or without associated odontoma. The lining epithelium of the cystic lesions fulfilled the histologic criteria for COC proposed by the World Health Organization, and their overall clinicopathologic features were consistent with that of developmental odontogenic cysts. The age of patients from the cyst group peaked at the second decade. The maxilla was affected more often (69%) than the mandible, with a predilection for the canine-premolar region (62.5%). Thirteen patients with follow-up information revealed no recurrence following enucleation. The four cases in the benign tumor group had variable clinicopathologic features. Two cases were solid tumors consisting of ameloblastoma-like sheets of odontogenic epithelium that contained ghost cells/calcification foci and juxtaepithelial dentinoid. Both patients experienced multiple recurrences following conservative surgeries. The other two lesions contained typical areas of COC and other types of odontogenic tumors (one ameloblastoma and one odontogenic myxofibroma). All four lesions occurred in the mandible and were relatively large. In the present series one case identified as malignant tumor arose from a previously benign COC. The tumor shared some features of COC (ghost cell foci and dystrophic calcification) but also had prominent mitotic activity, nuclear and cytoplasmic pleomorphism, areas of tumor necrosis, and infiltrative/destructive growth. Recognizing the extreme diversity in clinicopathologic features and biologic behavior among the so-called COCs, we suggest that the term COC should be used to specifically designate the unicystic lesions with or without an associated odontoma, i.e., lesions of the cyst group, and other related lesions identified as benign tumor and malignant tumor should be termed and classified separately. A tentative scheme with respect to the terminology and classification for this group of disparately behaving lesions was herein proposed to reflect the likely difference of their nature.     

Pyloromyotomy, 720 cases (1953-1982). Retrospective study

From 1953 to 1982 720 babies underwent pyloromyotomy for infantile hypertrophic pyloric stenosis. In this retrospective report the results obtained are compared to the data published in the literature.