Tumor Odontogénico Adenomatoide Quístico, presentación inusual. Reporte de dos casos / Cystic Adenomatoid Odontogenic Tumor, unusual presentation. Report of two cases

RESUMEN: El tumor odontogénico adenomatoide es un tumor benigno, poco común, que corresponde al 2-7% de los tumores odontogénicos. Se presenta con mayor prevalencia en la segunda década de vida y se ubica preferentemente en maxilar anterior. Es una lesión encapsulada, no agresiva, de crecimiento lento, que puede pasar desapercibida durante años. Deriva del epitelio odontogénico, y se caracteriza histológicamente por células epiteliales dispuestas en nidos, remolinos o cordones con escaso tejido conjuntivo. Puede presentar estructuras canaliculares un patrón ductiforme, con calcificaciones y presenta cápsula. El presente estudio reporta dos casos, el primero es una mujer de 30 años y el segundo el de un hombre de 36 años, ambos con un tumor odontogénico adenomatoide ubicado en zona maxilar anterior derecha, asintomático, sin asociación con diente incluido y de presentación quística a nivel imagenológico e histopatológico. Se discuten características clínicas, radiográficas, histopatológicas y terapéuticas de los casos.

ABSTRACT: Adenomatoid odontogenic tumor is a benign, rare tumor that accounts for 2-7% of odontogenic tumors. It appears with greater prevalence in the second decade of life and is located preferentially in the anterior maxilla. It is a slow-growing, non-aggressive, encapsulated lesion that can go unnoticed for years. It is derived from the odontogenic epithelium, and is characterized histologically by epithelial cells arranged in nests, eddies, or cords with little connective tissue. It may have canalicular structures and a ductiform pattern, with calcifications and it has a capsule. The present study reports two cases, the first is a 30-year-old woman and the second is a 36-year-old man, both with an adenomatoid odontogenic tumor located in the right anterior maxillary area, asymptomatic, without association with an included tooth and cystic presentation at the imaging and histopathological levels. The clinical, radiographic, histopathological and therapeutic characteristics of the cases are discussed.

Tumor adenomatoide de tubas uterinas, reporte de un caso y revisión de la literatura / Adenomatoid uterine tube tumor, case report and literature review

Resumen Los tumores adenomatoides del tracto genital son neoplasias poco frecuentes, de comportamiento benigno; debido a sus características histológicas se confunden fácilmente con tumores de estirpe vascular. Estudios recientes demuestran que los tumores adenomatoides del tracto genital femenino están genéticamente definidos por la mutación TRAF7 que activa la expresión aberrante de la vía NFkB. Presentamos el caso de una mujer de 27 años, asintomática, a quien se le realiza salpingectomía bilateral como método de anticoncepción definitivo; en el examen macroscópico de la tuba derecha se encuentra formación quística que por sus características microscópicas es diagnosticada como tumor adenomatoide de la tuba uterina. Finalmente se realiza una revisión de la literatura.

Abstract The adenomatoid tumors of the genital tract are rare neoplasms, with benign behavior; due to their histological characteristics, they are easily confused with tumors of vascular origin. Recent studies show that adenomatoid tumors of the female genital tract are genetically defined by the TRAF7 mutation that activates the aberrant expression of the NFkB pathway. We present the case of a 27-year-old woman, asymptomatic, who underwent bilateral salpingectomy as a method of definitive contraception; In the macroscopic examination of the right tuba, cystic formation is found which, due to its microscopic characteristics, is diagnosed as an adenomatoid tumor of the uterine tube. Finally, a review of the literature is made

Clinicopathological analysis of uterus and ovarian adenomatoid tumors / 中国现代医生

Objective To investigate the clinicopathological features, immunohistochemical expression and differential diagnosis of uterine and ovarian adenomatoid tumors. Methods 315 specimens of patients who underwent uterus and oophorectomy in our hospital from January 2016 to December 2017 were retrospectively analyzed, including 20 specimens of uterus and ovarian adenomatoid tumors. Their clinicopathological features and immunohistochemical expression were analyzed and related literature was reviewed. Results 20 cases of adenomatoid tumors were 6.34％ of surgical specimens, including 18 cases of uterine adenomatoid tumors and 2 cases of ovarian adenomatoid tumors. The average diameter of the tumor was (2.52±0.17) cm, and the tumor was located between the muscle wall, subserosal or uterine subserosal. Immunohistochemical staining showed CK (AE1/AE3), MC and calretinin were positive, CD34, ER, PR and Desmin were negative. No malignant lesions and recurrence occurred during follow-up after surgery. Conclusion Uterine and ovarian adenomatoid tumors are relatively rare, without specific clinical features, which is easy to be misdiagnosed and missed diagnosis. Correct use of immunohistochemistry can improve the detection rate of uterine and ovarian adenomatoid tumors.

Adenomatoid Tumor of Epididymis: A Rare Case Report

Adenomatoid tumors are rare benign neoplasms of the paratesticular region, most commonly occurring at the tail of the epididymis. We present a case of adenomatoid tumor in a 65 year old male, known case of prostatic adenocarcinoma, who presented in Urology OPD with painless swelling in testis. Bilateral orchidectomy was done with clinical suspicion of testicular carcinoma. On histopathology, diagnosis of adenomatoid tumor of epididymis was made. Due to its rarity, it is important for the physician and pathologist to be aware of this interesting entity in order to make a correct diagnosis.

Ultrasonic appearances and misdiagnosis analysis of intrascrotal adenomatoid tumors / 中国医学影像技术

Objective To investigate the sonographic features and to analyze misdiagnosis causes of intrascrotal adenomatoid tumors.Methods Preoperative sonographic data of 15 patients confirmed as intrascrotal adenomatoid tumor by surgery and pathology were retrospectively analyzed.Compared with pathological results,the sonographic features and the misdiagnosis causes were analyzed.Results Among 15 patients,the tumors located at the tail of epididymis in 8 patients,at the head of epididymis in 5 patients and within the testis in 2 patients.The lesions were misdiagnosed as inflammatory diseases in 3 patients,as cysts in 3 and as malignant tumors in 2 patients,while in the others were indeterminate.Solid tumors were detected with ultrasound in 12 patients,while cystica in 3 patients.Among 12 patients with solid tumors,lesions in 7 patients had clear boundary and regular shape,while the boundary in the rest 5 patients were less clear.The internal echo was homogeneous in 6 patients and inhomogeneous in the rest 6 patients with echoless and punctate hyperecho.Cystic mass was characterized by polycystic with isolation belt.Among 15 patients of intrascrotal adenomatoid tumor,detectable blood flow signals were found in 8 patients,while no obvious blood flow signals were found in the rest 7 patients.Conclusion Ultrasonic features of intrascrotal adenomatoid tumor were less characteristic.Combination of clinical information and other imaging findings might help to decrease misdiagnosis.

MRI findings of adenomatoid tumor of epididymis / 实用放射学杂志

Objective To investigate the MRI manifestations and diagnostic value of adenomatoid tumor (AT)of epididymis. Methods The MRI features of 5 patients with AT of epididymis were analyzed retrospectively,which were confirmed by histopathology,and some relevant medical literatures were reviewed.Results The epididymal tumor was manifested as moderate signal on T1 WI and T2 WI,similar to adjacent testicule,and as moderate signal with slightly high rim-enhancement on enhanced T1 WI in 4 cases.Secondary infarction and chronic granulomatous inflammation in 1 patient was found and presented unevenly moderate signal on T1 WI,slight hypointensity on T2 WI, hyperintensity on DWI and rim-enhancement on dynamic enhancement MRI.Few hydrocele was detected in all 5 patients.Conclusion AT of epididymis is a rare benign neoplasm with some characteristic MRI features which is helpful for the preoperative diagnosis.

Tumor adenomatoide pleural / Adenomatoid pleural tumor

Introduction: Adenomatoid tumors are rare benign neoplasms, pleural is a very rare location, reporting only 4 cases worldwide and none in our country. Case report: A 30 year-old female patient, that in the context of a preventive health examination, presents a mediastinal solid mass in the right hemithorax, in the thorax radiography. The lesion of 8 cm x 7 cm x 2.4 cm was surgically removed. Histology shows a lesion compatible with pleural adenomatoid tumor and inmunohistochemistry is positive for markers such as calretinin and cytokeratin 5/6. Discussion: The pleural adenomatoid tumor is an uncommon neoplasm with benign behavior and several differential diagnoses.

Introducción: Los tumores adenomatoides son neoplasias benignas poco frecuentes, cuya ubicación pleural es de muy baja frecuencia, reportándose sólo 4 casos a nivel mundial y ninguno nacional a la fecha. Las muestras parciales en biopsia intraoperaratoria, en ausencia de antecedentes clínico-radiológicos puede representar un desafío diagnóstico. Caso clínico: Paciente de sexo femenino, 30 años que en contexto de un examen preventivo de salud se pesquisa en la radiografía de tórax una masa sólida mediastínica en hemitórax derecho. Se extirpa lesión redondeada de 8 cm. La histología muestra lesión compatible con tumor adenomatoide pleural y la inmunohistoquímica positividad para marcadores Calretinina y Citoqueratina 5/6. Discusión: El tumor adenomatoide pleural en una entidad poco frecuente, de comportamiento benigno, con diversos diagnósticos diferenciales.

Ultrasonic appearances of intrascrotal adenomatoid tumor / 中华超声影像学杂志

Objective To explore the sonographic features of intrascrotal adenomatoid tumor.Methods The sonographic features of 21 cases with intrascrotal adenomatoid tumor confirmed by surgery and pathology were retrospectively analyzed.Results Twenty-one cases with intrascrotal adenomatoid tumor could be typed into 3 ultrasonic patterns:(1) The homogeneous echo type:15 cases,their ultrasonic features were homogeneous echo,including 5 cases with low echo,8 case with equal echo,and 2 case with high echo;5 cases with testicular albuginea adenomatous tumors were also homogeneous echo,2 cases of equal echo,2 cases of low echo and 1 case of high echo that was posterior echo attenuation slightly.(2) The edge halo type:4 cases,they showed the epididymis or testis sheath round or oval nodules,of which the middle was low echo and the edge is high echo halo ring.(3) The mixed echo type:2 cases,they showed cystic mixed echo of epididymis nodules,1 case with irregular cystic areas and poor sound transmission,while the other one with good sound transmission cystic areas and a small papillae on the wall.Conclusions Sonography is a valuable method for diagnosis of intrascrotal adenomatoid tumor.The edge halo type has sonographic features.

Leiomyoadenomatoid tumor of the uterus: Report of a rare entity and review of the literature.

Adenomatoid tumor occurs in the testicular spermatic cord and ejaculatory duct in males and in the fallopian tube and uterus in the females. These are usually small, benign lesions and are mostly incidental findings. Leiomyo-adenomatoid tumor (LMAT) is a variant of adenomatoid tumor, where in the smooth muscle component is predominant. Only nine cases of LMAT are reported so far in the English literature. We report one case and review the nine cases reported so far.

Clinicopathological analysis of Three cases with leiomyo-adenomatoid tumor of the uterus / 中国基层医药

Objective To investigate the clinicopathological diagnosis and differential diagnosis of leiomyoadenomatoid tumor of the uterus,to avoid clinical misdiagnosis and mistherapy.Methods The clinical histopathological and immunohistochemical features of 3 cases with leiomyo-adenomatoid tumor of the uterus were retrospectively evaluated,and the literatures were also reviewed.Results After hospitalization,before surgery,uterine smooth muscle tumor was diagnosed in 3 cases,adenomyomas were accompanied diagnosis in 2 cases.Through microscopic patterns of tumor and immunohistochemical staining showed,positive for MC and Calretinin,but negative for CD34,leiomyo-adenomatoid tumor of uterus was diagnosed,so gestrinone was stopped to use.Conclusion Pathological features and immunohistochemistry techniques are useful in establishing a correct diagnosis of leiomyo-adenomatoid tumor of the uterus,and avoiding misdiagnosis and mistherapy.

Concurrent hepatic adenomatoid tumor and hepatic hemangioma: a case report

A 45-year-old male with alleged asymptomatic hepatic hemangioma of 4 years duration had right upper-quadrant pain and was referred to a tertiary hospital. Computed tomography and magnetic resonance imaging scans revealed a hypervascular mass of about 7 cm containing intratumoral multilobulated cysts. A preoperative liver biopsy was performed, but this failed to provide a definitive diagnosis. The patient underwent a partial hepatectomy of segments IV and VIII. The histologic findings revealed multifocal proliferation of flattened or cuboidal epithelioid cells and a highly vascular edematous stroma. Immunohistochemistry findings demonstrated that the epithelioid tumor cells were positive for cytokeratin (AE1/AE3), vimentin, calretinin, and cytokeratin 5/6, and were focally positive for CD10, and negative for WT1 and CD34, all of which support their mesothelial origin. Immunohistochemistry for a mesothelial marker should be performed for determining the presence of an adenomatoid tumor when benign epithelioid cells are seen.

Adenomatoid tumors of female genital tracts: An analysis of 195 cases / 대한산부인과학회지

OBJECTIVE: Adenomatoid tumors of female genital tracts are benign lesions derived from mesothelium, occurring most commonly during the reproductive years. The aim of this study was to evaluate the overall incidence of adenomatoid tumors in Korean women and to analyze the clinical characteristics. METHODS: One hundred and ninety five patients with adenomatoid tumors were found in a retrospective medical records review of pathologic reports for 44,984 benign uterine diseases at Cheil General Hospital, from January 1995 to April 2009. RESULTS: The overall incidence rate was 0.42% of all benign uterine disease. Among them, 149 patients received hysterectomy, and 46 patients received uterine conservative surgery. Main symptoms of the patients were pain (25.1%), bleeding (30.2%), and palpable mass (18.5%). Most common associated pathologies were leiomyoma (46.6%), adenomyosis (25.1%) and endometriosis (13.1%). Most of the diagnosis was made postoperatively. Among 46 patients with conservative treatment, 13 patients showed successful pregnancy outcome. No recurrence occurred during the follow up period. CONCLUSION: Adenomatoid tumors are associated with fibroids and tend to mimic them clinically, making pre-operative diagnosis difficult. The recurrence is rare even after conservative operation. Our data about this benign neoplasm may be helpful for counseling patients after operations.

One case of adenomatoid tumor of uterus / 대한산부인과학회지

Adenomatoid tumor of uterus is described by Golden and Ash as a tumor that similar to adenoma but found obscured histogenesis. Adenomatoid tumor is a rare benign disease that originated from mesothelium and mostly occurred in male and rarely in female, and can be treated by simple excision of tumor because there is no recurrence or malignant change. We describe the clinical and histologic findings with references to other reported cases. We herein report a case of adenomatoid tumor of the uterus that gets an operation under clinical impression of myoma uteri.

Tumores adenomatoides uterinos: estudio anátomo-patológico e inmunohistocitoquímico / Adenomatoid tumor of the uterus: histological and immunohistochemical study

Antecedentes: Los tumores adenomatoides (TA) son poco frecuentes. Se encuentran principalmente en el aparato reproductor femenino y especialmente en el útero. No existen reportes a nivel nacional sobre estos. Objetivos: Describir las características histológicas e inmunohistoquímicas de los TA uterinos. Método: Estudio descriptivo de 32 muestras ingresadas al Servicio de Anatomía Patológica de Clínica Las Condes. Las muestras estudiadas fueron recopiladas entre noviembre de 1999 y noviembre de 2008. Resultados: El diagnóstico de TA fue realizado en 21 histerectomías y 11 miomectomías. En 14 casos se diagnosticaron como lesiones nodulares únicas (43,8 por ciento) y en 18 junto a leiomiomas (56,2 por ciento). El tamaño promedio de las lesiones únicas fue 2,6 cm, significativamente mayor que aquellas asociadas a leiomiomas. El patrón histológico predominante más frecuente correspondió al tipo angiomatoide (81,3 por ciento), seguido por los patrones adenoide (9,4 por ciento), sólido (6,3 por ciento) y mixto (3 por ciento), no se encontraron TA quísticos. El estudio inmunohistoquími-co fue positivo en el 100 por ciento de los casos para citoqueratina AE1/AE3, calrretinina, vimentina y D2-40. Fue negativo para CD31 y CEA. Sólo un 6,3 por ciento (2 casos) fue positivo para citoqueratina 5/6. Conclusiones: Los TA corresponden a tumores benignos de origen mesotelial. Generalmente su diagnóstico es un hallazgo. Al presentarse en el útero, se confunden generalmente con leiomiomas o se presentan en conjunto con estos. En base a lo anterior el tratamiento de estas lesiones debe ser conservador, bastando con la resección del tumor.

Background: The adenomatoid tumors (AT) are rare. They are found mainly in the female reproductive system and especially in the uterus. There is not national reporting on these. Objective: To describe the his-tological and immunohistochemical features of uterine AT. Method: Descriptive study of 32 patients admitted to the Pathology Department of Clinica Las Condes. The cases studied were collected between November 1999 and November 2008. Results: The diagnosis of AT was performed in 21 hysterectomies and 11 myomectomies. In 14 patients were diagnosed as nodular single lesions (43.8 percent) and in 18 cases associated with leiomyomas (56.2 percent), the average size of single lesions was 2.6 cm, significantly greaterthan those associated with leiomyomas. The predominant histologic type most often correspond to angiomatoid (81.3 percent), followed by adenoid patterns (9.4 percent), solid (6.3 percent) and mixed (3 percent), the cystic pattern was not observed. The immunohistochemical study of ST was positive in 100 percent for cytokeratin AE1/AE3, calrretinin, vimentin and D2-40. It was negative for CD31 and CEA. Only 6.3 percent (2 cases) were positive for cytokeratin 5/6. Conclusions: The AT is a benign tumor of mesothelial origin. Usually the diagnosis is a finding. In the uterus they are generally mistaken with leiomyomas or it is in associated with them. Based on the foregoing the treatment of AT should be conservative, only with resection.

Tumor odontogênico adenomatóide extrafolicular: uma apresentação infreqüente de um tumor raro / Extrafollicular adenomatoid odontogenic tumor: an unusual presentation of a rare tumor

O tumor odontogênico adenomatóide (TOA) é uma lesão benigna, não invasiva e de crescimento lento e progressivo. Existem três variantes clínicas, denominadas folicular, extrafolicular e periférica. Relatamos um caso de paciente do gênero feminino, com 35 anos, com TOA variante extrafolicular, localizado em maxila. Exames diagnósticos de imagem revelaram tumor localmente agressivo, com destruição óssea, sem invasão de tecidos adjacentes. A paciente foi tratada com enucleação cirúrgica conservadora. Encontra-se sem evidência de doença, após seguimento de cinco meses.

The adenomatoid odontogenic tumor (AOT) is a benign, non-invasive and low growth tumor. There are three clinical variants: follicular, extrafollicular and periferical. We report a case of a woman, 35 years-old with extrafollicular AOT, located in maxilla. Diagnostic image exams revealed locally aggressive tumor with bone destruction, without adjacent tissues invasion. The patient was treated with surgical enucleation. Nowadays, she has been with no evidence of disease after five-month follow-up.

A Diagnostically Challenging Case of an Infarcted Adenomatoid Tumor of the Epididymis

We describe a case of an infarcted adenomatoid tumor of the epididymis that was challenging to diagnose. A 20-year-old man presented with acute left scrotal pain. He was found to have a 2x1.5x1 cm tumor that was relatively well circumscribed on gross examination. There was a central necrotic area that exhibited gaping spaces and ghost outlines of epithelial cells. The periphery of the necrotic lesion showed focally viable adenomatoid tumor. The majority of the tissue adjacent to the necrosis consisted of granulation tissue, fibroblastic and myofibroblastic proliferation, and neutrophils. The fibroblasts and myofibroblasts showed plump nuclei, often with small nucleoli. No mitotic activity was present. The differential diagnosis for an infarcted adenomatoid tumor includes malignant mesothelioma, inflammatory myofibroblastic tumor, and inflammatory conditions. The key to diagnosing an infarcted adenomatoid tumor is to consider it in the differential diagnosis of any spindle cell tumor with necrosis occurring in the genital tract.

Adenomatoid Tumor of the Testis / 대한비뇨기과학회지

Adenomatoid tumors arising in the parenchyma of the testes are extremely rare neoplasms, and they uniformly behave in a benign fashion. The long reorted history of these tumors and the absence of distant metastasis suggest a benign nature. Thus, the treatment is surgical excision. We report here on a case of an adenomatoid tumor of the testis. Radical orchiectomy was performed, and this revealed an adenomatoid tumor of the parenchyma of the testis.

Adenomatoid Tumor of the Testis / 대한비뇨기과학회지

Adenomatoid tumors arising in the parenchyma of the testes are extremely rare neoplasms, and they uniformly behave in a benign fashion. The long reorted history of these tumors and the absence of distant metastasis suggest a benign nature. Thus, the treatment is surgical excision. We report here on a case of an adenomatoid tumor of the testis. Radical orchiectomy was performed, and this revealed an adenomatoid tumor of the parenchyma of the testis.

Adenomatoid Tumor of the Spermatic Cord / 대한비뇨기과학회지

Adenomatoid tumors are rare benign neoplasms thought to be of mesothelial origin. Although most reported cases developed from the epididymis, rare cases have been reported in the testicular tunica, spermatic cord and ejaculatory ducts. Because of the benign nature of this tumor, the treatment of choice is local excision. We report a rare case of adenomatoid tumor of the spermatic cord treated by local excision.

Adenomatoid Tumor between the Epididymis and the Testis / 대한남성과학회지

Adenomatoid tumor is the most common paratesticular tumor with an anatomic distribution limited to the epididymis and it rarely invades to the tunica vaginalis, spermatic cord and ejaculatory duct. Adenomatoid tumor is a benign neoplasm that is thought to be of a mesothelial origin. The treatment of choice for adenomatoid tumor is local excision because of its benign nature and the absence of distant metastasis. We report here on a rare case of adenomatoid tumor that was found between the epididymis and the testis, and it was treated by local excision of tumor.