Progressive Transformation of Germinal Centers in Submandibular Area: Case Report
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Abstract Progressive Transformation of Germinal Centers in Submandibular Area: Case Report Su Won Park, Soo Mi Jang, Dong Yul Kim, Jang Ho Son, Yeong Cheo l Cho, Iel Yong Sung Department of Oral and Maxillofacial Surgery, Ulsan University Hospital, College of Medicine, University of UlsanProgressive transformation of germinal centers (PTGC) is mostly a disease of young adults and it presents as unexplained, asymptomatic, localized or generalized lymphadenopathy that is usually persistent or recurrent over a period of many years. PTGCs are larger than regular germinal centers and they contain a variable proportion of small mantle zone lymphocytes and so they become progressively transformed and they may resul t in the loss of clear demarcation between them and the mantle zone. PTGC may resemble the nodules of nodular lymphocyt e predominant Hodgkin's disease (NLPHD) and it may be mistaken for NLPHD. Histological and immunohistochemical stu dies are helpful in differentiating these diseases. Because of the relatively frequent recurrences of PTGC, follow-up and r epeat biopsy are indicated. Although PTGC is not considered to be a premalignant condition, PTGC may occur prior to, concur rent with or following NLPHD. This emphasizes the need for ongoing follow-up and repeat biopsy. Although PTGC is repor ted in 3.5% to 10% of the cases of chronic nonspecific lymphadenopathy, oral & maxillofacial surgeons are not widely a ware of this condition and its clinical implications. Herein, we present a case of PTGC. A 24-year-old male without any histo ry of immunodeficiency or autoimmune disease was admitted to the Department of Oral & Maxillofacial surgery at Ulsan Univ ersity Hospital for evaluation of a right submandibular swellin g. He had another mass on the right thigh that was noticed about 1 year ago. The submandibular lesion was completely resected and biopsied. The histological findings and immunohistochemical stains (CD3, CD15, CD20, CD30, CD57, BCL-2, EMA) were consistent with PTGC. He was followed up without any other comp laints for 9 months.Key word:Progressive transformation of germinal centersKeywords:
Mantle zone
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Center (category theory)
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To discuss the diagnosis and prognosis of progressively transformed germinal center and emphasize the necessity of long term follow-up after treatment.Three patients were diagnosed as progressively transformed germinal center (PTGC) from 2010 to 2012. The clinical characteristics, histological features, differential diagnosis, prognosis were analyzed with review of literatures.Among 3 cases, 2 were found around parotid gland, one was in the neck. All patients were young male adults, who received excisional biopsy and regular histopathological examination. They were followed up for 8 months to 2.5 years, no relapse was found.PTGC occurs more commonly in young male adults. It always appears as an isolated and painless enlarged lymph node, which responses poorly to antibiotics. The final diagnosis is relied on histopathological and immunocytochemical examinations. Close follow-up is necessary, and attention should be paid to nodular lymphocyte predominant Hodgkin's disease although there is no clear evidence about their relationship of development. Supported by Science and Technology Plan Project of Shaoxing City(2011A23029).
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Hyaline
Etiology
Cervical lymph nodes
Presentation (obstetrics)
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Progressive transformation of germinal centers (PTGC) occurs focally in reactive lymph nodes, and has been reported with increased frequency in patients with nodular lymphocyte predominance Hodgkin's disease (NLPHD). It has been suggested that patients with lymph node biopsy samples showing PTGC are at increased risk for the development of NLPHD, and that Hodgkin's disease may evolve from PTGC. We report five young men (ages 14-24 years, mean 18) with prominent lymphadenopathy and florid PTGC, in whom careful examination and follow-up showed no progression to Hodgkin's disease. Three patients developed adenopathy that involved several node groups and two had localized adenopathy. Cervical (2), inguinal (2) and axillary (1) nodes ranging from 3 to 4 cm were excised. The number of progressively transformed germinal centers (PTGCs) ranged from 10 to 123 per specimen (mean 67); single sections contained nine to 29 PTGCs (mean 19). In three cases the nodal architecture was significantly distorted, suggestive of NLPHD, but Reed-Sternberg cells were absent. Follow-up is available for all patients (all untreated): three patients had persistent adenopathy 1 year 4 months to 10 years after diagnosis. Results of repeat biopsy in two patients (2 and 3 years after diagnosis) showed florid PTGC with no evidence of Hodgkin's disease. One of these patients had one subsequent biopsy 8 years after presentation; results showed only rare PTGCs. The fourth and fifth patients, who had presented with isolated adenopathy, were free of recurrent adenopathy at 2 and 5 years. These cases suggest a syndrome of lymphoid hyperplasia with florid PTGC in adolescent boys and young men. Although adenopathy can persist, there has been no progression to Hodgkin's disease. Recognition of this syndrome is important to avoid overdiagnosis of LPHD. Close follow-up of these patients will be necessary to evaluate the relationship of this disorder to NLPHD.
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To clarify the clinicopathological features of progressive transformation of germinal center (PTGC) unrelated to nodular lymphocyte predominant Hodgkin's lymphoma in Japanese patients, we reviewed 42 cases and compared the results with those of the United States and Germany. Our results were similar to theirs, with male predominance (M/F ratio, 3:1) and the presentation of a solitary asymptomatic enlarged lymph node in the head and neck area as the common features. However, in Japan, PTGC occurs more frequently in elderly patients. In this study, 12 (29%) of the patients with PTGC were aged 60 years or more. Thirteen patients (31%) with lymphadenopathy in the neck and head area had developed localized chronic inflammation (chronic sialoadenitis=4, chronic tonsillitis=3, infectious epidermal cyst=2) or an autoimmune disorder (hyperthyroidism=2 and bronchial asthma=2). None of the patients developed a malignant lymphoma during the follow-up period of 5 to 238 months (median 27 months). Histologically, in a single longitudinal section of the lymph node, the PTGC occupied up to 5% of the total follicles in 22 patients, 5-10% in 10, 10-20% in 7, and more than 20% in 3. In 5 (12%) patients, an association with prominent marginal zone hyperplasia was also noted. This study also indicates that nodal involvement by various low-grade B-cell lymphomas exhibiting marginal zone distribution patterns should be considered as a differential diagnosis of PTGC. Moreover, in Japan, PTGC is thought to be involved in the etiology of florid reactive follicular hyperplasia in elderly patients.
Follicular hyperplasia
Etiology
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Follicular lymphoma (FL) is the second most commonly reported B-cell lymphoma and accounts for 6% of all orbital lymphomas. FL presentation in the pediatric population is rare. This study reported an 18-year-old male with a pink elevated lesion in the conjunctiva of the left eye and normal ocular parameters. Following a complete excisional biopsy, the lesion was oval and smooth grossly and showed dense proliferation with a high nuclear-cytoplasmic ratio microscopically. Immunohistochemical studies revealed positive CD20 and bcl-6 while bcl-2 was negative, which is a common feature for pediatric FL in contrast to bcl-2 positivity found by immunohistochemistry in FL occurring in adult patient population. No signs of recurrence were observed over 2 years of follow-up. This study adds a new case to the existing few reports. Interestingly, all reported cases were males including the patient, mean duration of onset was 5 months, and mean duration of follow-up was 18.4 months. The authors emphasize that total surgical excision with close monitoring is reasonable management of such clinical entity.Follicular lymphoma (FL) is a rare entity in pediatric patients. Six cases have been reported with conjunctival FL in the literature (age between 6 and 21 years). Interestingly, all of them were male including the patient.
Follicular lymphoma
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This study is based on 38 patients, each of whom had a malignant lymphoma in which the gonad was the site of the main tumor mass at the time of diagnosis. Histiocytic lymphoma was the predominant type in the 27 male patients; in the 11 female patients, poorly differentiated lymphocytic lymphoma was the most frequent type (36%). All of the neoplasms in the males were diffuse, whereas two of the females had neoplasms that were nodular and diffuse. A striking feature was the high frequency of vascular invasion (41%) in the testicular lymphomas, which was reflected in a high incidence (86%) of noncontiguous lung involvement at autopsy, suggesting hematogenous spread. Clinically occult disease is probably responsible for the short interval between the discovery of a gonadal mass and the appearance of generalized disease. A poor prognosis may be expected if there is evidence of generalized disease within one year after diagnosis. Only 2 patients had disseminated disease after a year, whereas all those who died of disseminated disease manifested it within six months after diagnosis. Disease-free survival times in excess of 60 months in 3 patients who were treated only by orchiectomy or oophorectomy indicate that the gonads may be the primary site of a malignant lymphoma.
Orchiectomy
Occult
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Grading (engineering)
Histopathology
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