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PubMed1993

Ocular adnexal lymphoproliferative lesions.

Ocular adnexal lymphoproliferative lesions consist of a spectrum of disease entities, including reactive lymphoid hyperplasia, atypical lymphoid hyperplasia, and lymphoma. No clinical or radiologic criteria facilitate a distinction among these lymphoproliferative lesions. The two hyperplastic processes may evolve to localized or systemic lymphoma. A similar pattern is evident in other mucosa-associated lymphoid tumors elsewhere in the body. Most ocular adnexal lymphomas are small lymphocytic non-Hodgkin's tumors with an indolent course; frequently, they remain localized to the ocular adnexa. In comparison, intermediate- and high-grade lymphomas are less common in the ocular adnexa but more aggressive. An approach to the diagnosis and treatment of these complex entities is suggested. Despite new pathologic classification schemes, immunophenotypic labeling, and molecular genetic analysis, the prognosis for patients with small-cell lymphoma in the ocular adnexa is difficult to predict.

Conjunctiva↗
PubMed1999

T-cell and T/natural killer-cell lymphomas involving ocular and ocular adnexal tissues: a clinicopathologic, immunohistochemical, and molecular study of seven cases.

OBJECTIVE: Lymphomas of the eye and its adnexa are frequently of B lineage. This study aims to characterize the clinical and histopathologic features of the rare non-B-cell non-Hodgkin lymphomas (NHL) of these locations. DESIGN: Retrospective, noncomparative case series. PARTICIPANTS: Seven cases of T- and T/NK-cell lymphomas involving the ocular and ocular adnexal tissues. METHODS: A morphologic, immunohistochemical, and molecular analysis (polymerase chain reaction) of each of the tumors was undertaken. The lesions were classified according to the Revised European-American Lymphoma (REAL) classification. The clinical and follow-up data were collected. RESULTS: The patients included four women and three men ranging in age from 32 to 88 years (mean, 63 years). The presenting ophthalmic symptoms varied from a small nodule on the upper eyelid and conjunctival swellings to dramatic loss of vision associated with gross protrusion of the globe. Five of the cases presented were secondary manifestations of a systemic lymphoma in ocular tissues; two cases represented primary disease. Three cases were "peripheral T-cell lymphomas (PTCL), unspecified" with positivity for CD3, CD8, and betaF1 and negativity for CD56. Two cases were CD3+, CD30+, and CD56- and were classified as "anaplastic large-cell lymphomas of T-cell type" (T-ALCL). The remaining two cases showed an immunophenotype of CD3+, CD56+, and betaF1- and proved to contain Epstein-Barr virus (EBV) by in situ hybridization, consistent with "T/natural killer (NK)-cell lymphoma of nasal type." Clonal T-cell populations were shown in all three of the PTCLs by Southern blot (n = 1) and polymerase chain reaction (n = 2) for the T-cell receptor gamma and beta genes in one case of ALCL but not in the T/NK-cell lymphomas. Five patients died within 2 years; only two patients (one primary PTCL and one cutaneous T-ALCL) were disease free at 4 and 5 years' follow-up, respectively. CONCLUSION: This study shows that a heterogeneous group of T-cell lymphomas can involve the eye and its adnexal tissue. Most T-cell neoplasms are secondary manifestations of systemic disease and carry a poor prognosis. These findings, in conjunction with published data on ocular B-NHL, also indicate that immunophenotypic differentiation between T- and B-NHL in these locations is of clinical importance.

Adult↗
PubMedDate not supplied

A biomicroscopic method for quantitation of ocular and adnexal toxicity.

A biomicroscopic method for evaluation of ocular and adnexal toxicity in the white rabbit has been developed. When compared to the Draize method, the biomicroscopic method demonstrates a greater spectrum of clinical signs allowing for better dose-response characteristics. This method was found to be both reproducible and easy to execute. The biomicroscopic method, by defining discrete anatomical entities, allows for a high degree of predictability as to severity of ocular and adnexal toxicity as well as the occurrence of permanent ocular injury.

Animals↗
PubMed1997

Gray scale sonography, subjective evaluation of the color Doppler image and measurement of blood flow velocity for distinguishing benign and malignant tumors of suspected adnexal origin.

OBJECTIVE: To compare the capacity of transvaginal Doppler ultrasound examination with that of gray scale sonography as an aid in distinguishing benign and malignant pelvic tumors of suspected adnexal origin. STUDY DESIGN: One hundred and fifty-one women scheduled for laparotomy or laparoscopic surgery because of a pelvic mass of suspected adnexal origin underwent ultrasound examination including color and spectral Doppler techniques within 8 days preceding the operation. Based on the gray scale ultrasound image, each tumor was classified as being either a unilocular cyst, a multilocular cyst, a unilocular cyst with solid parts, a multilocular cyst with solid parts or a solid tumor. Tumor vascularization was visualized with the color Doppler technique, each tumor being characterized by the color content of the scan, as rated subjectively on a visual analogue scale ('tumor color score'). Blood flow velocity waveforms were obtained by placing the Doppler gate over the colored area with the tumor. The blood flow velocity waveform with the highest time averaged maximum velocity was used to characterize the tumor . The results of gray scale imaging and Doppler ultrasound examination were compared with the histology of the specimen. RESULTS: Ultrasound morphology correctly identified all the malignant tumors, with a false-positive rate of 61%; none of the 49 unilocular or multilocular cysts without solid parts was malignant, whereas 24% (24/102) of the tumors with solid components were. Among multilocular cysts with solid parts, both tumor color scores and time average maximum blood flow velocities were significantly higher in malignant than in benign tumors, but among solid tumors there was complete overlap in Doppler results between the malignant and benign subgroups. Using Doppler examination to discriminate between benign and malignant multilocular cysts with solid parts and ultrasound morphology for differentiation of the remaining tumors, all the malignancies in the study were detected with a false-positive rate of 32 or 38% depending on which Doppler variable was used. CONCLUSION: The present technique of Doppler ultrasound examination is helpful only in the differential diagnosis of multilocular cysts with solid parts. Therefore, the degree to which Doppler examination can contribute to the differential diagnosis of pelvic tumors will depend on the proportion of multilocular cysts with solid parts in the population studied: the greater this proportion, the greater the potential of the Doppler examination to improve diagnostic accuracy.

Adnexa Uteri↗
PubMed1996

Molecular analysis of ocular adnexal lymphoid proliferations. Characteristics of immunoglobulin VH, D and J segment usage.

We investigated the molecular characteristics of six cases of ocular adnexal lymphoid proliferation. Although their clinical diagnoses varied, all of them were diagnosed as having malignant lymphomas by immunohistochemical examinations and molecular approaches. Proliferating cells showed the B cell phenotype, positive for L26/CD20 and negative for UCHL1/CD45RO, and genotypic study by Southern blotting revealed that all cases had gene rearrangements of the immunoglobulin heavy chain with or without those of the kappa (kappa) light chain. Epstein-Barr virus infection was not identified. Usage of variable domains of the immunoglobulin heavy chain was analyzed. Subgroup 3 of the VH gene was present relatively often, in 3 of 6 cases, perhaps due to its having the largest number of VH gene segments. On the other hand, an unexpectedly high frequency of the DLR family of the D gene segment was observed by sequencing the third complementarity determining region. These molecular characteristics in ocular adnexal lymphoid neoplasms might be related to the clinical course.

Adult↗
PubMed1996

Adnexal adenocarcinoma of the upper lip.

A case of poorly differentiated adenocarcinoma of adnexal origin in the upper lip of a man aged 75 is reported which on presentation and initial biopsy was thought to be a salivary neoplasm. He had been aware of the lesion for 10 years but had sought treatment because of recent increase in size of the tumour. He subsequently developed bilateral metastases in cervical nodes. The histopathologic features and relationship of the tumour to the orbicularis oris muscle were consistent with a poorly differentiated adnexal adenocarcinoma of sweat-gland origin. Although carcinomas of skin adnexae are rare they should be considered in the differential diagnosis of tumours in the orofacial region.

Adenocarcinoma↗
PubMed1994

Laparoscopic Management of Suspicious Adnexal Masses

After our initial experience with 757 patients, we have extended our indications for laparoscopy to suspicious adnexal masses. Twenty of 29 ovarian cancers surgically treated since December 1991 were first evaluated by laparoscopy. In all, 321 patients were evaluated by laparoscopy in 1992 and 1993, including 145 women with an ultrasonographically suspicious mass (45.7%). At laparoscopy, 52 masses were diagnosed as suspicious, including 20 cases of malignant ovarian tumors. Sixteen patients were treated by laparotomy (5%), including 11 women with cancer, 2 with teratomas diagnosed as suspicious, and 3 with benign masses that could not be treated endoscopically (0.9%). In cancer treated by laparotomy, staging, including the assessment of the upper abdomen, was begun laparoscopically. When a cancer was managed laparoscopically, an early second look laparoscopy was performed routinely. Since many laparotomies may be avoided, laparoscopic surgery is promising in the management of suspicious adnexal masses. However, from two cases of tumor dissemination, we conclude that this management should follow strict guidelines and needs to be further evaluated.

Journal Article↗
PubMed1994

Operative Laparoscopy for Adnexal Cystic Masses in Patients Under 40 Years of Age

From January 1990 through October 1993, 527 patients under 40 years were submitted to operative laparoscopy for the treatment of 544 adnexal cystic masses by stripping or vaporization/coagulation techniques in nine centers in Rome. The mean age of patients was 27.8 years, ranging from 13 to 39. The mean diameter of the cysts was 45.8 mm. ranging from 20 to 130. The procedure was completed by laparoscopy in 518 cases (95.2%) whereas 26 cases (4.8%) were converted to laparotomy. Conservative treatment was performed in all cases except 6 (1.1%), where ovariectomy or adnexectomy was performed. Histopathology revealed a borderline tumor in two; in all others the cyst was benign. It is concluded that operative laparoscopy is the procedure of choice for the treatment of carefully selected adnexal masses in patients of reproductive age.

Journal Article↗
PubMed1994

Pelviscopic Therapy of Adnexal Tumors

Minimally invasive surgery after careful presurgical screening represents an accepted method of treatment for adnexal masses. Between 1990 and 1994, 1032 adnexal tumors were treated by ovarian cystectomy, ovarectomy, adnexectomy, and salpingectomy via pelviscopy. Ten borderline cancers were managed by placing the ovaries into tissue collection bags. In four cases of a preoperatively undetected ovarian cancer, the surgical procedure was changed to laparotomy. Compared with 325 laparotomies performed for ovarian malignancy in the same period of time, the surgical advantages of pelviscopy become evident.

Journal Article↗
PubMed1997

Microcystic adnexal carcinoma.

Microcystic adnexal carcinoma (MAC) is a rare adnexal tumour which has only recently been recognized as a separate clinicopathological entity. It typically affects the face of the middle-aged and often requires extensive surgical excision, due to its locally invasive nature. Its clinical significance is that, despite being locally invasive, MAC is typified by a lack of metastatic spread. We present a case and review of the literature.

Carcinoma, Skin Appendage↗
PubMed2001

Clinical, histopathological, and immunogenetic analysis of ocular adnexal lymphoproliferative disorders: characterization of malt lymphoma and reactive lymphoid hyperplasia.

Malignant lymphomas and reactive lymphoid hyperplasia (RLH) in the ocular adnexa are sometimes difficult to differentiate morphologically and have often been categorized together as a lymphoproliferative disorder. Immunogenotypic characters of these diseases have not yet been well clarified. This study included 76 cases of ocular adnexal lymphoproliferative disorders. These consisted of 52 cases of malignant lymphoma (43 primary and 9 secondary), 22 of RLH, and 2 borderline cases. There were slightly more male than female subjects. Diagnoses were based on morphology and immunophenotypic characteristics. Clonalities were detected by means of polymerase chain reaction (PCR), and immunoglobulin heavy-chain variable region (VH) genes were sequenced in 10 cases of mucosa-associated lymphoid tissue (MALT) lymphoma. MALT lymphoma constituted 86% (37 cases) of the primary lymphomas. MALT lymphomas were more indolent, more rarely disseminated, and had a lower death rate than the other primary lymphomas. Two patients exhibited coexistence of MALT and diffuse large B-cell lymphoma. The average age of patients with RLH was 5.5 years younger than that of those with MALT lymphoma. One of the cases of RLH later progressed to malignant lymphoma. B-cell clonality was detected by PCR in 57%, 55%, and 0% of primary lymphomas, MALT lymphomas and RLHs, respectively. Sequencing of VH genes revealed that the VH3 family was the most commonly expressed germline VH family (70%) and that DP-63, DP-54 and DP-47 genes were frequently found in the MALT lymphomas examined. PCR analysis was useful for differentiation between MALT lymphoma and RLH. Sequence analysis of VH genes showed that an autoimmune mechanism may be involved in the lymphomagenesis of ocular adnexal MALT lymphoma.

Adult↗
PubMed2006

Imaging features of ocular adnexal lymphoproliferative disease.

PURPOSE: To evaluate the imaging characteristics of a cohort of patients with ocular adnexal lymphoproliferative disease (OALD). METHODS: A noncomparative retrospective review between 1992 and 1995 and prospective study from 1995 to 2005 of the clinical, imaging and treatment of 105 patients presenting to tertiary orbital referral centre presenting with OALD. RESULTS: One hundred and five patients (mean age 61 years, range 11-90 years) with equal gender distribution were included. Fifty-three were primary and 52 were secondary. Computed tomography (CT) usually showed a well-circumscribed lesion of greater than brain density, moulding to adjacent tissues with moderate enhancement. Aggressive histology was associated with bone destruction, while moulding was associated with indolent histology (P<0.005).MRI in OALD showed intermediate signal intensity on T1- and T2-weighted images and moderate enhancement with gadolinium. Gallium scanning sensitivity to detect ocular adnexal disease was 25 and 57% for systemic involvement. Positron emission tomography (PET) upstaged (71%) of patients with systemic lymphoproliferative involvement, having a higher sensitivity than CT in detecting distant disease (86 vs 72%). CONCLUSIONS: CT and/or MRI are essential in the evaluation of OALD and can be used to establish that an orbital lesion may be lymphoprolifetaive in nature. Further, these imaging modalities may predict the behaviour of the lymphoma in certain cases. Gallium scanning provides no additional information to CT and does not influence patient treatment. PET represents an important addition to the assessment of OALD with real impact on patient management.

Adolescent↗
PubMed2002

Differentiation between malignant and benign follicular adnexal tumours of the skin by DNA image cytometry.

BACKGROUND: We have demonstrated in previous studies that DNA image cytometry (DNA ICM) can be helpful in detecting malignancy in sebaceous tumours of the Muir-Torre syndrome and sweat gland tumours. However, little is known about DNA ICM in cutaneous adnexal tumours with follicular differentiation. OBJECTIVES: To study a larger series of benign and malignant follicular adnexal tumours with DNA ICM. METHODS: We studied 13 malignant follicular tumours (seven trichilemmal carcinomas, five malignant proliferating pilar tumours, one pilomatrix carcinoma) and 55 benign follicular tumours (four tumours of the follicular infundibulum, seven Winer's pores, eight trichilemmomas, two trichofolliculomas, 16 trichoepitheliomas, 13 pilomatrixomas, five trichoblastomas) by DNA ICM. All cases were clear-cut as malignant or benign, respectively, on histopathological criteria. The stemline interpretation according to Böcking et al. (DNA distribution in gastric cancer and dysplasia. In: Precancerous Conditions and Lesions of the Stomach, Zhang YC, Kawai K, eds. Berlin: Springer-Verlag, 1993: 103-20) was performed in all cases. In addition, 5[c]-exceeding events (5cEE) and the 2[c] deviation index (2cDI) were calculated, except in one histopathologically benign tumour, which revealed euploid polyploidization, as the analysis of 5cEE and 2cDI is not valid in that case. RESULTS: A 2cDI threshold of 0.24 proved to be the most reliable marker for the distinction between malignant and benign follicular tumours. On the basis of this feature, all malignant and benign tumours were correctly classified. A specificity of 100% was achieved by all three interpretation methods, but the sensitivity of 2cDI for the detection of malignant tumours was superior to the analysis of 5cEE (sensitivity 77%) and to the stemline interpretation (sensitivity 23%). CONCLUSIONS: DNA ICM may be helpful in distinguishing between malignant and benign follicular tumours.

Aneuploidy↗
PubMed2001

Ocular adnexal lymphoproliferative disease: a series of 73 cases.

BACKGROUND: This study involved 73 patients with lymphoproliferative lesions of the ocular adnexa. The lesions were categorized using the Revised European American Lymphoma classification of lymphoid tissues and analysed to determine the frequency and prognostic impact of tumour type, location, stage and patient's age and sex. METHODS: The clinical, histopathological, immunohistochemical and phenotypic analysis by flow cytometry and follow-up data were studied. RESULTS: The ocular adnexal lymphoproliferative lesions included 70 lymphomas and six reactive lymphoid hyperplasia. Seventy-nine per cent had stage IE disease, 4% stage II, 1.5% stage III and 15.5% stage IV. Five patients (7%) had a past history of systemic lymphoma. Major histological types were extranodal marginal zone lymphoma (MZL) in 44 (63%), follicular (FL) in 12 (17%), diffuse large B-cell (DLBCL) in eight (11%), mantle cell (MCL) in two (3%), B-cell chronic lymphocytic leukaemia (CLL)/small lymphocytic lymphoma in two (3%), peripheral T-cell lymphoma (PTCL) one (1.5%) and natural killer cell lymphoma (NKCL) in one (1.5%). Longest survival was seen in those with low-grade lymphomas (MZL and FL) and worst in PTCL and NKCL. Lymphoma-related mortality was 2% for MZL, 33% for FL, 38% for DLBCL, and 100% for MCL, PTCL and NKCL. Systemic lymphoma was present prior to, at presentation or at subsequent follow up in 26/68 (39%) of all lymphoma patients, 17% for MZL, 38% for DLBCL, 83% for FL, and 100% for MCL, CLL, PTCL and NKCL. CONCLUSION: The majority of ocular adnexal lymphomas were low-grade B-cell lymphomas (MZL). Multivariate analysis showed that the only significant independent predictors of all causes of mortality were the histological type of lymphoma and the stage of disease at presentation.

Adult↗
PubMed1998

Diagnosis of adnexal malignancies by using color Doppler energy imaging as a secondary test in persistent masses.

The purpose of this prospective study was to compare the accuracy of B-mode transvaginal ultrasonography alone and in combination with color Doppler energy (or power Doppler) imaging in differentiating benign from malignant adnexal masses. A total of 192 consecutive persistent adnexal masses (159 benign, 33 malignant) were studied before surgery by B-mode transvaginal ultrasonography with and without color Doppler energy. In addition, CA-125 plasma levels were determined and spectral Doppler analysis was performed. By color Doppler energy imaging, a mass was considered malignant when arterial flow was visualized in an echogenic portion of a mass defined as malignant by B-mode. Intratumoral arterial blood flow could be readily detected by color Doppler energy imaging in all malignant tumors and in 94% of the benign tumors. The combined use of transvaginal B-mode ultrasonography and color Doppler energy imaging has greater accuracy in the diagnosis of ovarian malignancies than transvaginal ultrasonography alone (value of kappa: 0.81 and 0.63, respectively), reducing the number of false-positive results. The use of spectral Doppler analysis was of limited diagnostic value, with a kappa value of 0.17 for the pulsatility index (< 1) and of 0.41 for the resistance index (< 0.4). Also, the association with CA-125 increased the number of false-negative results. In conclusion, the use of color Doppler energy imaging seems to be a useful secondary test when a mass is suspected to be malignant by B-mode ultrasonography.

Female↗
PubMed2001

Microcystic adnexal carcinoma: an uncommon tumor with debatable origin.

BACKGROUND: Microcystic adnexal carcinoma is an uncommon skin appendage neoplasm exhibiting both pilar and sweat duct differentiation. This tumor remains a subject of controversy as to its differentiation profile, histogenesis, and classification which is reflected in the nomenclature used to designate the neoplasm in question. Beyond this controversy the tumor remains a diagnostic challenge because of its rarity, the histologic mimicry it may display, and its banal cytologic appearance; it also poses a therapeutic challenge, as it is characterized by slow but aggressive and destructive local growth extending beyond clinical margins together with a high tendency for perineural invasion and recurrence. OBJECTIVE: We report two cases of this unusual tumor illustrating some of its characteristics. Our review emphasizes the divergent opinions concerning its differentiation profile and its origin. An organoid nevus as the origin of microcystic adnexal carcinoma in one of our patients is discussed in this context.

Adult↗
PubMed2003

Clinical course, risk factors, and treatment of microcystic adnexal carcinoma: a short series report.

BACKGROUND: Microcystic adnexal carcinoma (MAC) is a locally aggressive neoplasm of both eccrine and follicular differentiation; it commonly presents on the head and neck of white middle-aged to older patients. OBJECTIVE: To review the course, risk factors, and management of 10 patients with microcystic adnexal carcinoma (MAC) and to compare treatment outcome by either Mohs micrographic surgery or standard surgical excision. METHODS: Tumor registry databases were used to identify cases at Roswell Park Cancer Institute in Buffalo, New York. Fifteen cases that were coded as MAC were reviewed. Ten cases were subsequently confirmed to be MAC. From these cases, data were collected on patient demographics, tumor location and size, risk exposure, metastasis, treatment, and recurrence of tumors. RESULTS: The average age of the patients was 63.4, and females outnumbered males seven to three. Nine tumors were located on the head and neck area. Five patients had a history of radiation therapy, and two patients were sisters. The first-time procedure was Mohs micrographic surgery in four cases and surgical excision in six cases. Three patients in the surgical excision group needed a second procedure, two of whom thereafter underwent Mohs micrographic surgery. In the Mohs micrographic surgery group, none of the patients required a second procedure, and all of the patients were disease free at an average of 23.3 months. One recurrence was noted in the surgical excision group, and the average follow-up was 32 months. CONCLUSION: This report supports the use of Mohs micrographic surgery for tumor control with the least possible procedures. Risk factors appear to include radiation treatment, ultraviolet exposure, and possibly genetics.

Adult↗