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PubMed1998

The clinical behavior of localized and multicentric Castleman disease.

BACKGROUND: Castleman disease, an unusual condition of unknown cause consisting of a massive proliferation of lymphoid tissue, remains a clinicopathologic diagnosis. Three histologic variants (hyaline vascular, plasma-cell, and mixed) and two clinical types (localized and multicentric) of Castleman disease have been described. OBJECTIVE: To analyze the clinical features, management, and outcome of patients with Castleman disease. DESIGN: Case series. SETTING: University referral hospitals. PATIENTS: All patients with Castleman disease who were seen at Texas Medical Center, Houston, Texas, between 1977 and 1995. INTERVENTIONS: Surgical excision for localized disease; surgery, combination chemotherapy, or prednisone for multicentric disease. MEASUREMENTS: Patients were identified according to initial presentation as having localized or multicentric Castleman disease. Patients within each group were further subdivided according to whether they had hyaline vascular, plasma-cell, or mixed disease. RESULTS: Data from 15 patients were analyzed. All 7 patients with localized disease underwent surgical excision and remain free of disease. The 8 patients with multicentric disease were further subdivided according to initial treatment: Three patients who received combination chemotherapy are currently alive and free of disease; 2 patients treated with prednisone are alive but have needed intermittent maintenance therapy for disease reactivations; and 2 patients treated with surgery only have died, 1 of infectious complications and 1 of non-Hodgkin lymphoma. CONCLUSIONS: Localized and multicentric Castleman disease are different clinical disorders with overlapping histologic features. Localized disease can be cured with surgery, but complete remissions in patients with multicentric disease have been achieved only with chemotherapy or prednisone given at the time of diagnosis.

Adolescent↗
PubMedDate not supplied

Patterns of and risk factors for recurrence after liver resection for well-differentiated hepatocellular carcinoma: a special reference to multicentric carcinogenesis after operation.

BACKGROUND/AIMS: We studied the patterns of, and risk factors for, recurrence after resection of well-differentiated hepatocellular carcinoma to make a strategy for such carcinoma. METHODOLOGY: The subjects were 36 patients who underwent liver resection for well-differentiated hepatocellular carcinoma. Multicentric carcinogenesis after the operation (multicentric recurrence) was diagnosed when a recurrent tumor included a component of well-differentiated hepatocellular carcinoma. Tumor-free survival rates of patients with various risk factors were calculated and differences between groups were evaluated. For multivariate analysis, Cox's proportional hazard model was used. RESULTS: All recurrent tumors after operation were of multicentric origin. Univariate analysis showed a history of blood transfusion, high alpha-fetoprotein concentration (> 20 ng/ml), and low platelet count (< 10(5)/mm3) to be significant factors. By multivariate analysis, a history of blood transfusion and a low platelet count were independent risk factors. Nonanatomic resection was not a risk factor. CONCLUSIONS: In patients with well-differentiated hepatocellular carcinoma, especially with risk factor(s) for multicentric recurrence, not only anatomic but also nonanatomic resection (partial resection) are indicated for a primary tumor, because almost all recurrent tumors are of multicentric origin. After resection of well-differentiated hepatocellular carcinoma, careful monitoring for multicentric recurrence is important in patients with risk factors.

Adult↗
PubMed1994

Renal angiomyolipoma. DNA content and immunohistochemical study of classic and multicentric variants.

Angiomyolipomas (AMLs) are polymorphic renal tumors that are composed of mature tissues and frequently associated with tuberous sclerosis; AMLs have long been considered hamartomatous in nature. We report the routine histologic and immunohistochemical features and DNA content analysis of two fatal cases of renal giant multicentric AML with distant organ involvement, and we contrast the findings with those of four similarly studied cases of classic solitary AML. Severe nuclear pleomorphism, significant mitotic activity, and necrosis, which are all characteristics of multicentric AML, were not seen in the cases of classic AML. Quantitation of DNA by image analysis of Feulgen-stained slides from paraffin-embedded blocks revealed an aneuploid pattern in the two cases of multicentric AML and an aneuploid pattern in one of the four cases of classic AML. Tumors in the liver, spleen, and lungs in one of the cases of multicentric AML were diploid. Immunohistochemical analysis revealed positive staining reaction of vascular and adipose tissue components with HMB-45 antibody in three of the six cases of AML. We conclude that AMLs may occur in a sarcomatous, infiltrating multicentric form involving multiple organs, that aneuploidy may be seen in lesions of both the multicentric AML and classic AML variants, that AMLs may feature DNA ploidy heterogeneity in multiple-organ sites, that HMB-45 immunoreactivity may be encountered in AMLs without evidence of nevomelanocytic differentiation, and that continued study of AMLs is needed to clarify further the histogenesis, lineage, clonality, and malignant potential of these tumors.

Adult↗
PubMed1996

[Multicentricity in renal adenocarcinoma: its incidence and the therapeutic implications].

Unsuspected multicentricity in single Renal Adenocarcinoma (RAC) together with the prospects of incomplete neoplasia removal are the 2 major disadvantages for the acceptance of traditional renal surgery in birenal carriers of medium size, low grade single RAC. This paper is a retrospective review of our series of RAC patients who underwent radical nephrectomy between January 1986 and October 1994 with the following purpose: 1) To evaluate our incidence of unsuspected multicentricity. 2) To evaluate the characteristics of such multicentricity. 3) To evaluate, in the assumption that traditional surgery had been indicated for patients with small size RAC, in how many patients tumoral resection would have been incomplete due to existence of satellite tumoral nodes in the preserved renal parenchyma. Of 110 patients undergoing radical nephrectomy, 11.8% (13/110) had unsuspected multicentricity. In 10 patients there was multiple satellite nodes with size ranging from 0.3 to 3 cm. We conclude that there is not relationship between multicentricity and size or stage of primary tumour and that, by applying strict criteria (size, location, well defined tumoral wall and form of presentation) to select the patients who can be candidates to traditional renal surgery, the probability to perform it in patients with multicentric RAC would be considerably reduced.

Adult↗
PubMedDate not supplied

The prognosis of patients with hepatocellular carcinoma of multicentric origin.

BACKGROUND/AIMS: The characteristics of patients with multicentric hepatocellular carcinoma (HCC) differ from those of patients with intrahepatic metastatic HCC. However, there are few reports regarding the long-term results in patients with HCC of multicentric occurrence. The purpose of this study was to clarify the prognosis of a group of patients with multicentric HCCs. MATERIALS AND METHODS: We evaluated the outcome in 28 patients with multiple hepatocellular carcinoma (HCC) suggestive of multicentric occurrence, all of whom underwent hepatic resection. The disease-free survival, recurrence pattern and survival were analyzed in the two subgroups of 12 patients with single advanced HCC (AdHCC) nodule and an early HCC (eHCC) or early advanced HCC (eAdHCC) nodule [Group A] and 16 patients with two AdHCC lesions [Group B]. These data were also compared with those of 58 patients with solitary AdHCC [Group C]. RESULTS: Both the disease-free survival and survival periods for Groups A and C were significantly longer than those for Group B, while no significant difference was seen between Group A and C. No significant differences among the three groups were recognized in the incidence of various types of recurrence pattern. CONCLUSIONS: The long-term results in patients with multicentric HCC apparently vary depending on the pathological condition at the time of operation. Among patients with multicentric HCC, those with AdHCC combined with an eHCC or eAdHCC are likely to show improved prognosis as a result of hepatic resection.

Carcinoma, Hepatocellular↗
PubMed1989

Nonpalpable in situ ductal carcinoma of the breast. Predictors of multicentricity and microinvasion and implications for treatment.

Fifty breasts with nonpalpable ductal carcinoma in situ (DCIS) were examined for the presence of microinvasion, multicentricity, and number of involved ducts to see if the biopsy specimen could have predicted the findings in the remainder of the breast. When DCIS was an incidental finding, fewer ducts were involved and no evidence of either microinvasion or multicentricity was found. Solid and cribriform DCIS were rarely multicentric or microinvasive; micropapillary DCIS was often multicentric, rarely microinvasive; comedocarcinoma was more likely to be both microinvasive and multicentric. Ductal carcinoma in situ as an incidental finding may be treated by excision alone; papillary and micropapillary DCIS are best treated by therapy aimed at the entire breast, although axillary dissection may not be required. Therapy for comedocarcinomas should include the entire breast and the axillary nodes.

Adult↗
PubMed2000

Multicentricity in pharyngoesophageal tumors: argument for total pharyngolaryngoesophagectomy and gastric transposition.

BACKGROUND: Pharyngoesophageal tumors pose a challenge to surgical management, and there is controversy in the literature as to the best procedure to be used. Advantages and disadvantages are mentioned for total pharyngolaryngoesophagectomy and gastric transposition (PLE&GT), free jejunal transplants, and free forearm flaps. One of the arguments for PLE&GT is the persistence or subsequent occurrence of multiple primaries in a field cancerization region. Multiple tumors in the head and neck/esophagus/lung axis have been reported. However, despite extensive investigation, there is little information on specific multicentricity in patients treated with PLE&GT for pharyngolaryngoesophageal carcinomas. METHODS: A clinicopathological study was undertaken in 35 consecutive patients who underwent PLE&GT for pharyngoesophageal cancer to evaluate synchronicity, multicentricity, and metachronicity. Only in situ and invasive carcinomas were considered. The findings were compared with the reports in the literature. RESULTS: Thirty-eight tumors were diagnosed preoperatively, with the main indications for PLE&GT being tumors located in the esophagus or hypopharynx (32 patients) and larynx (three patients). After the surgical treatment, 21 patients had single primaries (60%) and 14 (40%) had 25 multiple primaries in addition to their main primaries (total of 60 tumors in the whole group). Synchronous, previous metachronous and subsequent metachronous carcinomas occurred in 26%, 17%, and 8.5% of the instances, respectively. Twenty of the 25 multicentric carcinomas were invasive (80%). Either the main primaries or the multicentric carcinomas were located in the esophagus or hypopharynx (91.5% and 60%, respectively). Other sites included the larynx, oropharynx, oral cavity, and lung. CONCLUSION: The incidence of multicentric tumors in patients with pharyngoesophageal carcinomas may favor total PLE&GT as the procedure of choice, because it includes all the condemned upper pharyngolaryngoesophageal mucosa.

Adult↗
PubMed1980

Multicentricity of non-palpable breast cancer.

One hundred eighty-nine biopsies have been performed for clinically occult mammary lesions, detectable by mammography but not clinically apparent. Fifty-two cancers were demonstrated within this group, including 26 invasive ductal cancers, seven micro-invasive ductal cancers, 14 non-invasive ductal cancers, and five lobular carcinomas in situ. All mastectomy specimens were examined for multicentric foci of breast cancer in quadrants other than that in which the primary lesion was located. Of the invasive ductal cancers, 40% were multicentric. Of the micro-invasive ductal cancers, 57.1% were multicentric. Of the non-invasive ductal cancers, 45.5% were multicentric. The one mastectomy specimen from a patient with lobular carcinoma in situ did not have evidence of residual disease. The overall incidence of multicentricity in the 43 specimens examined was 44.2%. These findings suggested that any therapeutic procedure for either invasive or non-invasive ductal carcinoma that does not include total mastectomy may leave behind foci of cancer, which are a threat to the patient.

Breast Neoplasms↗
PubMed1982

Duct carcinoma in situ. Relationship of extent of noninvasive disease to the frequency of occult invasion, multicentricity, lymph node metastases, and short-term treatment failures.

Fifty-three breasts resected for a biopsy diagnosis of duct carcinoma in situ were studied with a serial subgross and correlated radiographic method of examination designed to permit quantitation of the extent of the noninvasive lesion in the breast. Overall frequencies of occult invasion and multicentricity were 21 and 32%, respectively. Among 24 lesions 25 mm or larger in extent (average, 63; median, 56 mm) 11 showed occult foci of invasion, 13 had multicentric foci and six had nipple involvement. Among 29 lesions less than 25 mm in extent (average, 10; median, 8 mm) there were no instances of occult invasion, four were multicentric and two had nipple involvement (P = less than 0.05 for multicentricity and occult invasion). Twenty patients with lesions averaging 8 mm in extent are being followed after excision only in an experimental program. There have been three local recurrences at an average follow-up of 44 months. All recurrences occurred ipsilaterally, two were within the prior biopsy site. All patients with recurrence are free of disease following local resection in two and modified radical mastectomy in one. For lesions with associated microcalcifications, the distribution of the mammographic microcalcifications closely approximates the extent of disease as confirmed histologically. These findings suggest that an important predictive factor for the presence of occult invasion and multicentricity in the resected breast is the extent of the noninvasive lesion.

Adolescent↗
PubMed1988

Multicentric intraepithelial neoplasia involving the vulva. Clinical features and association with human papillomavirus and herpes simplex virus.

Sixteen of 46 patients (35%) with Grade 3 vulvar intraepithelial neoplasia (VIN 3) were found to have an additional site of lower genital tract squamous cell neoplasia, primarily in the cervix. The frequency of multicentricity decreased significantly with age. In addition, patients with multicentric disease (involving the vagina and/or cervix in addition to the vulva) had a significantly higher frequency of multifocal disease involving the vulva (involving more than one location on the vulva) and of recurrence than patients without multicentric disease. Human papillomavirus (HPV) DNA was detected by in situ hybridization in 81% of the women with multicentric squamous cell neoplasia. No significant difference was noticed between patients with multicentric and unicentric squamous cell neoplasia in the detection rate of papillomavirus antigen, HPV DNA, the various HPV types, herpes simplex virus Type 2 (HSV2)-related antigen, type-specific antibodies to HSV, and dual HPV and HSV2 infections. These findings suggest that HPV and HSV2, although strongly associated with VIN 3, do not influence the development pattern of squamous cell neoplasia, and that all patients with VIN 3, especially if they are younger than 50 years of age, should be evaluated periodically for additional centers of lower genital tract squamous cell neoplasia.

Adult↗
PubMed2001

Treatment of unicentric and multicentric Castleman disease and the role of radiotherapy.

BACKGROUND: Although surgery is considered standard therapy for unicentric Castleman disease, favorable responses to radiotherapy also have been documented. The authors undertook this study to analyze the clinical factors, treatment approaches, and outcomes of patients with unicentric or multicentric Castleman disease, and to report the outcomes of patients with unicentric Castleman disease treated with radiotherapy. METHODS: The authors reviewed the medical records of 22 patients who had received a histologic diagnosis of Castleman disease at the University of Texas M. D. Anderson Cancer Center between 1988 and 1999. One patient with a concurrent histopathologic diagnosis of nonsecretory multiple myeloma was excluded from the study. In all patients, the diagnosis of Castleman disease was based on the results of lymph node biopsies. Disease was categorized as being either unicentric or multicentric and further subdivided into hyaline vascular, plasma cell, or mixed variant histologic types. Clinical variables and outcomes were analyzed according to treatment, which consisted of surgery, chemotherapy, or radiotherapy. RESULTS: Records from 21 patients were analyzed: 12 had unicentric disease, and 9 had multicentric disease. The mean follow-up time for the entire series was 51 months (median, 40 months). Four patients with unicentric disease were treated with radiotherapy alone: 2 remain alive and symptom free, 2 died of causes unrelated to Castleman disease and had no evidence of disease at last follow-up. Eight patients with unicentric disease were treated with complete or partial surgical resection, and all are alive and asymptomatic. All nine patients with multicentric disease were treated with combination chemotherapy: five are alive with no evidence of disease, and four are alive with progressive disease. CONCLUSIONS: Surgery results in excellent rates of cure in patients with unicentric Castleman disease; radiotherapy can also achieve clinical response and cure in selected patients. Multicentric Castleman disease is a more aggressive clinical entity and is most effectively treated with combination chemotherapy, whereas the role of radiotherapy in its treatment remains unclear.

Adolescent↗
PubMed2002

Multicentric mammary carcinoma: evidence of monoclonal proliferation.

BACKGROUND: Both the widespread use of screening mammography and emphasis on breast conservation have raised many questions regarding the clinical and therapeutic management of multicentric mammary carcinoma (MMC). MMC has been postulated to be either a clonal proliferation of a single mammary carcinoma or multiple independent synchronous primary tumors in the same breast. The goal of the current study was to evaluate the histologic features and immunohistochemical profile of MMC. We also compared the clinical outcomes of the patients in the current study with stage-matched and treatment-matched groups of patients with unicentric mammary carcinoma. METHODS: The authors studied 32 patients with T1-T2, N0-1, M0 multicentric invasive mammary carcinomas diagnosed between 1983-1988 and treated at The University of Texas M. D. Anderson Cancer Center. The histologic features of each tumor (including tumor type, nuclear grade, presence of in situ carcinoma, pattern of in situ carcinoma, and lymphovascular invasion) were evaluated. The authors performed immunohistochemical analysis of estrogen receptor (ER), progesterone receptor (PR), HER-2/neu, and Ki-67 in 25 cases, including 14 from which > 1 tumor was available to perform comparative immunohistochemical analysis. The clinical parameters of each case were compared with those of the unicentric breast carcinoma controls. RESULTS: The median age of the patients with MMC was 45 years (range, 28-69 years). Twelve patients had a family history of breast carcinoma (37.5%). The maximum tumor dimension ranged from 0.2-3.2 cm in the index lesion (median, 2.0 cm) and 0.1-2.5 cm in the second lesion (median, 0.9 cm). Twelve patients were clinically classified as having Stage I disease and 20 patients were considered to have Stage II disease at the time of presentation. Follow-up data were available for all the patients and follow-up ranged from 4.5-16 years (median, 6 years). The disease-free survival was 84% at 5 years and 73% at 10 years in the MMC patients and 78% and 70%, respectively, in patients with unicentric breast carcinoma (P = 0.4368). Histologically, 24 of the multicentric tumors were found to be infiltrating ductal tumors and 8 were found to be infiltrating lobular carcinomas. The nipple was involved in 10 cases. The histology of the multicentric invasive tumor was nearly identical in 31 cases (97%). Approximately 72% of the cases had in situ carcinoma in both tumors and 44% had lymphovascular invasion. Comparative immunohistochemical analysis of separate tumors was equivalent with regard to ER, PR, and HER-2/neu. The quantitative immunohistochemical staining for the proliferative marker Ki-67 differed between tumors in two cases. CONCLUSIONS: The near-identical morphologic and immunohistochemical patterns in the MMC cases in the current study support the hypothesis that early-stage synchronous tumors are a clonal proliferation of a single mammary carcinoma. Furthermore, the results of the current study support evaluating prognostic markers in only one tumor per MMC patient. There was no appreciable difference in the disease-free survival of patients with unicentric and multicentric breast carcinoma.

Adult↗
PubMed1989

Sequence of centromere separation: characterization of multicentric chromosomes in a rat cell line.

The B1 cell line of rat cerebral endothelium origin exhibits several dicentric and multicentric chromosomes. These chromosomes, unlike multicentrics in mouse (Vig and Zinkowski 1986) do not show premature centromere separation. All centromeres deposit kinetochore proteins and appear to be functional. Even the centromeres which fail to migrate to the poles during anaphase and make side arm bridges bind to spindle microtubules. Some multicentric chromosomes show kinetochores spaced apart with intervening stretches of euchromatin while others are located adjacent to each other thus exhibiting tandem repeats and forming a "compound" kinetochore (Brinkeley et al. 1984). Also, unlike mouse multicentric chromosomes in which different pericentric regions and the centromeres replicate at different times, the rat chromosomes appear to replicate all pericentric and centric regions in a given multicentric simultaneously. The present studies indicate that centromeres in rat and mouse replicate during the last part of the S-phase and in continuation with the pericentric heterochromatin.

Animals↗
PubMed2003

Significance of multicentric cancer recurrence after potentially curative ablation of hepatocellular carcinoma: a longterm cohort study of 892 patients with viral cirrhosis.

BACKGROUND: Tumor recurrence is common after potentially curative ablation of hepatocellular carcinoma; therefore, the incidence of intrahepatic metastasis and multicentric carcinogenesis was evaluated. METHODS: A cohort of 892 patients with cirrhosis caused by either hepatitis B virus (n=246) or hepatitis C virus (n=646) was followed up without interferon administration. RESULTS: Hepatocellular carcinogenesis rates were 28.0% at the end of the fifth year, 49.2% at the tenth year, and 61.3% at the fifteenth year. Sex, hepatitis virus, alpha-fetoprotein, platelet count, age, and indocyanine green retention test value significantly affected the carcinogenesis rate. Of 372 patients with tumor development, 162 (43.5%) received surgical or radical locoregional therapy. On the assumption that a second carcinogenesis after sufficient ablation of well-differentiated cancer was attributable to multicentric carcinogenesis, recurrence rates after curative therapy in all the 162 patients, and the recurrence rates in 39 patients with solitary, small, and well-differentiated histology were analyzed: the rates were 58.2% and 30.9% at the third year, 75.9% and 53.4% at the fifth year, and 89.6% and 79.9% at the tenth year, respectively. The estimated incidence of multicentric recurrence was, therefore, 31% or higher at the third year and 53% or higher at the fifth year. The actual second carcinogenesis rates (multicentric recurrence) were approximately 1.6 times as high as those of the virtual second carcinogenesis rates calculated from statistical simulation. CONCLUSIONS: A longterm cohort study disclosed that multicentric carcinogenesis was the principal mechanism of recurrence after curative ablation of hepatocellular carcinoma and that the concept of "carcinogenic stage" should be introduced to improve the understanding of carcinogenic events in viral cirrhosis.

Adult↗
PubMed2003

Multicentric glioma: our experience in 25 patients and critical review of the literature.

Multicentric gliomas are interesting and well-recognised entities with a yet unknown rate of occurrence. Single cases or small series are reported in the literature accessible to us, and we think this is the first large series describing true multicentric gliomas. We reviewed 25 patients selected according to the criteria defined by Batzdorf and Malamud. Multicentricity was found in 2% of patients with malignant gliomas. Longer survival was observed in patients who underwent surgical excision of the multicentric lesions. Multicentric tumours are rare clinical entities. Our data suggest that they should be surgically removed whenever possible, and histopathologic examination of the lesions is always advisable if they are located in sites inaccessible to surgery. Stereotactic biopsy represents a safe and satisfactory method for achieving sure diagnosis.

Adult↗
PubMed2006

Management of multicentric lesions of the lower genital tract.

OBJECTIVES: To report management and outcome of multicentric lesions of the lower genital tract. To define risk factors of recurrence. STUDY DESIGN: Retrospective review of multicentric dysplasias treated in our colposcopic clinic between 1996 and 2003. Multicentric dysplasias included CIN with VAIN and/or VIN. After primary treatment, follow-up was colposcopic, cytologic and virologic. RESULTS: Forty-four patients presented multicentric lesions out of 998 patients referred for CIN (4.4%). The average age was 36.8 years. Immunologic disorders were present in 20.4%. Ninety-one percent had cervicovaginal or cervicovulvar lesions, only 9% had three sites of genital dysplasia. 53.3% of lesions were concomitant. 79.5% of CIN were high grade, 62.5% of VAIN low grade and 62.5% of VIN high grade. Therapeutic modalities were as follows: conization for CIN (70.4%), CO2 laser for VAIN (33.3%) and surgery for VIN (41.7%). Forty patients were followed and had at least one post-treatment cytologic control; 55% of them had residual disease. Out of the 23 patients with at least two negative controls after treatment, 43.5% presented recurrence. Risk of recurrence was not statistically bound to such parameters as tabagism, immunologic disorder, high grade lesions, non-surgical treatment, and persistence of HPV infection after treatment. CONCLUSION: Multicentric dysplasias are associated with high rate of residual lesion and recurrence. Management of these lesions require long term follow-up.

Adolescent↗
PubMed2005

Multistep and multicentric development of hepatocellular carcinoma: histological analysis of 980 resected nodules.

BACKGROUND/AIMS: Histological observations support the concept of multistep and multicentric development of hepatocellular carcinoma (HCC) in cases of chronic liver disease. However, the relationship between the incidence of such a modality of development of HCC and the type of background liver disease has not been fully investigated. METHODS: A total of 980 HCC nodules resected from 664 patients were analyzed. Multistep HCC was defined as well differentiated HCC containing the portal tracts (early HCC), or the presence of early HCC-like areas in the periphery of the nodule. In cases with multiple nodules, if the smaller nodule showed the features of multistep HCC, or if each nodule showed a distinct histology, the case was defined to have multicentric HCC. RESULTS: Of the 980 nodules, 369 (37.7%) met the criteria of multistep HCC. Of the 664 patients, 177 (26.7%) had multiple nodules that met the criteria of multicentric HCC. Both the incidences of multistep and multicentric HCC were significantly higher in HCV-Ab-positive cases than in HBs-Ag-positive cases (46.0 vs. 19.1%, P<0.001 and 34.1 vs. 16.5%, P=0.005, respectively). CONCLUSIONS: Multistep and multicentric HCC develops most frequently in patients with HCV infection.

Carcinoma, Hepatocellular↗
PubMed2000

[Is there a place for interferon-alpha in the treatment strategy of multicentric Castleman's disease?].

INTRODUCTION: Castleman's disease is an unusual condition of unknown cause, consisting of massive proliferation of lymphoid tissue. Two forms (localized and multicentric) have been described. Interleukin-6 (IL-6) is at the core of the disease, being responsible for most of the clinical and biological signs that may be observed. Despite the benignancy of this pre-lymphoma condition, its course is usually aggressive and of poor prognosis in regard to the multicentric form. No consensus regarding treatment has been defined. Available data on the multicentric form of the disease are to scarce to allow any conclusion about the treatment timing and type of chemotherapy best suited to this condition. We report the case of a patient in whom interferon alpha (IFN-alpha) was used as first line treatment. EXEGESIS: The case of a 52-year-old man with multicentric Castleman's disease combined with high IL-6, in whom, however, testing for human herpes virus-8 proved to be negative, is described. Interferon alpha (4.5 MU/m2 three times per week during 18 months) administered as first line treatment induced dramatic improvement in the patient's general condition and normalization of the tumoral syndrome. Moreover, biological parameters and IL-6 returned to normal. Two years after interferon disruption, complete remission is still present. CONCLUSION: On the basis of the present data and those of two previous observations, anti-IL-6 and anti-infective properties of IFN-alpha are discussed. Treatment of multicentric Castleman's disease is based on corticosteroids and drugs derived from those pertaining to treatment of malignant lymphomas. Our results indicate that IFN-alpha is truly directed against Castleman's disease and has less toxicity than drugs usually prescribed. This argues for early use of IFN-alpha in Castleman's disease, in association or not with corticosteroids.

Castleman Disease↗