Adenomatoid tumor of the adrenal gland: unexpected guest.

Adenomatoid tumor (AT) is a benign growth that originates from mesothelial cells. Typically, it encompasses the uterus, fallopian tubes, and paratesticular area. One-third of all AT is in paratesticular area, and it accounts for 60% of all benign paratesticular tumors. ATs have been seldom reported as a case report in adrenal gland, liver, pleura, and mediastinal cavity. To date, English literature has only documented 46 instances of adrenal AT. We present the occurrence of a new clinically unexpected case of adrenal AT in a 37-year-old male patient. Computed tomography (CT scan) incidentally revealed the presence of a tumor. To the best of our knowledge, this is the first reported instance of primary adrenal gland AT reported in Saudi Arabia and the 47th instance worldwide in the English-based published literature. It is crucial to thoroughly investigate these tumors, utilizing techniques such as histopathological analysis and immunohistochemical staining to correctly diagnose AT.

MR Imaging Findings of Uterine Adenomatoid Tumors.

PURPOSE: Adenomatoid tumor is a rare benign genital tract neoplasm of mesothelial origin. Uterine adenomatoid tumors occur in the outer myometrium and may mimic leiomyomas. Because hormonal treatment is not applicable to adenomatoid tumors and laparoscopic enucleation is not easy as myomectomy, it is important to differentiate adenomatoid tumors from leiomyomas for the adequate treatment. The purpose of this study is to evaluate the MRI findings of adenomatoid tumor for the differentiation from leiomyoma. METHODS: MRI findings of surgically proven 10 uterine adenomatoid tumors in 9 women were retrospectively evaluated with correlation to histopathological findings. RESULTS: All 10 tumors appeared as solid myometrial masses and showed heterogeneous signal intensity with admixture of partially ill-defined slight high-intensity areas containing abundant tubular tumor cells and well-defined myoma-like low-intensity areas reflecting smooth muscle hypertrophy on T2WI including 4 lesions with peripheral ring-like high intensity. High-intensity areas on T2WI tended to show high intensity on diffusion-weighted imaging (DWI) with relatively high apparent diffusion coefficient (ADC), suggesting T2 shine-through effect due to abundant tubules. Intra-tumoral hemorrhage revealed on MRI was rare. Early intense contrast-enhanced areas on dynamic contrast-enhanced study were observed dominantly within the high-intensity areas but rarely within the low-intensity areas on T2WI. CONCLUSION: The outer myometrial mass with the admixture of well-defined low- and ill-defined high-intensity areas on T2WI may be suggestive of adenomatoid tumor. Peripheral ring-like high intensity on T2WI and DWI may also be suggestive. Dynamic contrast-enhanced MR study may be helpful for the differentiation from leiomyoma.

An incidental finding of large cell calcifying Sertoli cell tumor in an orchidectomy specimen for a separate adenomatoid tumor.

Introduction: Large cell calcifying Sertoli cell tumors are exceedingly rare. They are most commonly benign, but risks for malignancy include older age, larger size of tumor, and solitary tumors. To the author's knowledge, this is the first case reported of an incidental large cell calcifying Sertoli cell tumor when an orchidectomy was performed for a separate lesion. Case presentation: A 31-year-old male presented with a painless testicular lump. Ultrasound demonstrated an exophytic lesion in the superolateral aspect and calcifications were noted inferomedially and inferolaterally in the right testis. On histology from radical orchidectomy, the superolateral lesion was found to be an adenomatoid tumor, and the calcifications inferiorly represented a large cell calcifying Sertoli cell tumor. The background showed foci of germ cell neoplasia in situ but no evidence of invasive malignancy. Conclusion: Calcifications on ultrasound in isolation may represent large cell calcifying Sertoli Cell tumors.

Staghorn Calculus with Adenomatoid Tumor: A Case Report.

Testicular adenomatoid tumor is very rare. More understanding of the tumor and treatment is required for better outcomes. A 63-year-old man visited a urologist for staghorn calculus. During follow-up, he had recurrent left flank pain and intermittent hematuria. Computed tomography demonstrated left staghorn calculus, and then further assessment revealed left testicular swelling. Ultrasound showed epididymal mass. Percutaneous nephrolithotomy was deferred and we performed left radical orchiectomy. A pathologic examination revealed testicular adenomatoid tumor. This case highlights the importance of awareness of a very rare benign tumor in a patient with staghorn calculus.

Adenomatoid Tumor of Epididymis Associated with Tubular Ectasia of Rete Testis: Sonographic Evaluation.

Paratesticular tumors are a relatively rare group of mesenchymal tumors, accounting for 7%-10% of all intrascrotal tumors. Adenomatoid tumor of the epididymis is the most common epididymal tumor. It is difficult to distinguish intra- versus paratesticular tumors and to know the nature of the lesion (benign versus malignant) on clinical examination since they may have similar presentation; therefore, ultrasound examination of these lesions is required to demonstrate its extratesticular location and also to suggest its benign nature. Elastography can further help in characterizing the lesion by evaluating the stiffness of the tumor. Here, we present a case of adenomatoid tumor of the left epididymis with tubular ectasia of the rete testis.

Multiparametric Ultrasound Diagnostic Approach to Malignancy-Mimicking Adenomatoid Tumors of the Scrotum: Is Strain Elastography Enough?

Background: Paratesticular tumors (PTs) are very uncommon, accounting for almost 5% of intrascrotal tumors. Of these, adenomatoid tumors (ATs) represent about 30% and most frequently arise in the tail of the epididymis. Ultrasound (US) examination is the first-choice imaging method employed for the evaluation of the scrotum. Unfortunately, there are no specific US-imaging features useful for distinguishing an AT from a malignant lesion. To increase diagnostic accuracy and confidence, new sonographic techniques have incorporated real-time tissue elastography (RTE) under the assumption that malignant lesions are "harder" than benign lesions. Case report: In our paper, we describe a very rare case of a 60-year-old patient with a giant paratesticular mass mimicking malignancy when examined using RTE, i.e., it was stiffer than the surrounding tissue (a hard pattern), which, upon histologic examination, was identified as an AT. Discussion: Our case underscores that there is also a significant overlap between different types of scrotal lesions when RTE is used for examination. Thus, if a PT is found, the imaging approach should always be supplemented with more definitive diagnostic methods, such as FNAC or FNAB, which are the only diagnostic methods capable of leading to a certain diagnosis. Conclusions: Alongside underlining the importance of pre-operative imaging for making correct diagnoses and selecting the correct therapy, we wish to draw our readers' attention to this report in order to demonstrate the clinical implications of a giant AT presenting as stiff lesions when examined using SE.

Adenomatoid tumor in a young male case report.

Adenomatoid tumors are benign para-testicular tumors and account for about 30% of all paratesticular neoplasms in males. The most common presentation is scrotal swelling between the third and fifth decades. We reported an epididymal adenomatoid tumor in a 28-year-old patient.

Diffuse WT1 Positivity in Smooth Muscle Component Supports Dual Differentiation of Mesothelial Cells in the Histogenesis of Leiomyoadenomatoid Tumors-A Report of two Cases.

Leiomyoadenomatoid tumors of the epididymis are exceedingly rare biphasic tumors composed of an adenomatoid component in the form of gland-like structures lined by single flat or cuboidal cells admixed with smooth muscle. Radiological and gross findings cannot distinguish leiomyoadenomatoid tumors from the more common classic adenomatoid tumors or leiomyomas, and careful microscopic examination is critical in the identification of this esoteric variant. The histogenesis of this entity remains ambiguous. Common hypotheses include a collision tumor, a variant of an adenomatoid tumor with a smooth muscle component, or an adenomatoid tumor arising in the background of reactive smooth muscle hyperplasia. We present 2 cases of leiomyoadenomatoid tumors with diffuse nuclear WT1 positivity in both the adenomatoid and smooth muscle components, supporting the mesothelial origin of these tumors.

NR4A3 fusions characterize a distinctive peritoneal mesothelial neoplasm of uncertain biological potential with pure adenomatoid/microcystic morphology.

A focal adenomatoid-microcystic pattern is not uncommon in peritoneal mesothelioma, but tumors composed almost exclusively of this pattern are distinctly rare and have not been well characterized. A small subset of mesotheliomas (mostly in children and young adults) are characterized by gene fusions including EWSR1/FUS::ATF1, EWSR1::YY1, and NTRK and ALK rearrangements, and often have epithelioid morphology. Herein, we describe five peritoneal mesothelial neoplasms (identified via molecular screening of seven histologically similar tumors) that are pure adenomatoid/microcystic in morphology and unified by the presence of an NR4A3 fusion. Patients were three males and two females aged 31-70 years (median, 40 years). Three presented with multifocal/diffuse and two with a localized disease. The size of the individual lesions ranged from 1.5 to 8 cm (median, 4.7). The unifocal lesions originated in the small bowel mesentery and the mesosigmoid. Treatment included surgery, either alone (three) or combined with hyperthermic intraperitoneal chemotherapy (two), and neoadjuvant or adjuvant chemotherapy (one case each). At the last follow-up (6-13 months), all five patients were alive and disease-free. All tumors were morphologically similar, characterized by extensive sieve-like microcystic growth with bland-looking flattened cells lining variably sized microcystic spaces and lacked a conventional epithelioid or sarcomatoid component. Immunohistochemistry confirmed mesothelial differentiation, but most cases showed limited expression of D2-40 and calretinin. Targeted RNA sequencing revealed an NR4A3 fusion (fusion partners were EWSR1 in three cases and CITED2 and NIPBL in one case each). The nosology and behavior of this morphomolecularly defined novel peritoneal mesothelial neoplasm of uncertain biological potential and its distinction from adenomatoid variants of conventional mesothelioma merit further delineation as more cases become recognized.

Ultrasonography of a giant adenomatoid tumor of the tunica vaginalis: A case report and review of the literature.

Adenomatoid tumor is a kind of benign tumor, accounts for a low percentage of scrotal tumors. It usually locates in epididymis, less commonly, arises from the tunica albuginea, spermatic cord or tunica vaginalis of male's urogenital tract system. Here, we report a case of a 32-year-old male, which presented as a 6-month history of left scrotal swelling pain with a scrotal mass, and it had progressively enlarged over the last 1 month. The ultrasonography (US) described a giant well-defined hypoechoic mass in the left scrotum, a thick pedicle connected the mass and the left scrotal wall, and the pedicle had large blood vessels in it. Enhanced CT showed an irregular solid mass in the left scrotum, the left testicular artery was thickened to supply blood for the mass, and the radiologist mentioned it could not be excluded as a malignant lesion. The patient underwent left radical orchiectomy, the mass was diagnosed as an adenomatoid tumor of the tunica vaginalis by pathology. We review literature regarding adenomatoid tumors originating in the tunica vaginalis, summarize the ultrasonographic presentations, provide the idea of diagnosis and differential diagnosis to improve diagnostic accuracy and avoid unnecessary orchiectomy.

Adenomatoid mesothelioma arising from the diaphragm: a case report and review of the literature.

BACKGROUND: Adenomatoid mesothelioma is a rare subtype of malignant mesothelioma that can be confused with adenomatoid tumors, which are classified as benign. The clinical features and optimal management of adenomatoid mesothelioma have not been elucidated in the literature. In this report, we present an extremely rare case of adenomatoid mesothelioma that developed on the peritoneal surface of the diaphragm as well as a literature review of adenomatoid mesothelioma in the abdominal cavity. CASE PRESENTATION: The patient was a 61-year-old Japanese woman who had undergone resection of a malignant peripheral nerve sheath tumor of the hand 18 years prior. She was diagnosed with clinical stage I lung adenocarcinoma on follow-up chest radiography. Simultaneously, a 20-mm enhancing nodule with slow growth on the right diaphragm was detected on contrast-enhanced computed tomography. She presented no specific clinical symptoms. At this point, the lesion was suspected to be a hypervascular tumor of borderline malignancy, such as a solitary fibrous tumor. After a left upper lobectomy for lung adenocarcinoma, she was referred to our department, and laparoscopic tumor resection was performed. Adenomatoid tumors were also considered based on the histopathological and immunohistochemical analyses, but we made the final diagnosis of adenomatoid mesothelioma using the results of the genetic profile. The patient remains alive, with no recurrence noted 6 months after surgery. CONCLUSION: We encountered a valuable case of adenomatoid mesothelioma of peritoneal origin. There are some previously reported cases of adenomatoid mesothelioma and adenomatoid tumors that may need to be recategorized according to the current classification. It is important to accumulate and share new findings to clarify the clinicopathological characteristics and genetic status of adenomatoid mesothelioma.

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.

Adenomatoid tumor of the testis: A rare case report.

Adenomatoid tumors (AT) are benign tumors commonly found in paratesticular tissues. However, intratesticular AT are rare. Clinically and radiologically, the AT of the testis imitates the malignant neoplasia of the testis. Here, we present a case of the intratesticular AT.

Adenomatoid Tumor of the Adrenal Gland: Report of Two Cases and Review of the Literature.

Adenomatoid tumor (AT) is an uncommon benign neoplasm of mesothelial origin, usually occurring in the female and male genital tracts. Extragenital localization such as the adrenal gland is extremely rare. Until now, only 39 cases of adrenal AT have been reported in the English literature. Here we report two novel cases of adrenal AT that occurred in male patients aged 30 and 31 years. The tumors were discovered incidentally by computed tomography (CT). Macroscopically, the tumors were unilateral and solid, and the greatest dimension of the tumors was 3.5 and 8.0 cm, respectively. Histologically, the tumors consisted of angiomatoid, cystic, and solid patterns and infiltrated the adrenal cortical or medullary tissue. The tumor cells had low nuclear/cytoplasmic ratio, with no pathological mitosis or nuclear pleomorphism. Thread-like bridging strands and signet-ring-like cells could be seen. Immunohistochemically, the tumor cells were positive for epithelial markers (AE1/AE3, CK7) and mesothelial markers (D2-40, calretinin, and WT-1). The Ki-67 index was approximately 1 and 2%, respectively. The differential diagnosis of adrenal AT includes a variety of benign and malignant tumors. The patients had neither local recurrence nor distant metastasis at 21 and 8 months after removal of the tumor. In the literature review, we comprehensively summarized the clinical, morphological, immunohistochemical, and prognostic features of adrenal AT. Adrenal ATs are morphologically and immunophenotypically identical to those that occur in the genital tracts. Combining the histology with immunohistochemical profiles is very supportive in reaching the diagnosis of this benign tumor, helping to avoid misdiagnosis and overtreatment.

[Adenomatoid tumor of the adrenal gland: Clinicopathologic characteristics and differential diagnosis on two tumors of exceptional adrenal location]. / Tumeur adénomatoïde de la surrénale : aspects clinico-pathologiques et diagnostics différentiels de deux tumeurs de localisation surrénaliennes exceptionnelles.

Adenomatoid tumors are benign tumors from mesothelial origin, usually occurring in the genital tract. Extragenital locations, especially in the adrenal gland are extremely rare. Here we are reporting two cases of a 28-year-old and 50-year-old men with an adenomatoid tumor of the right adrenal gland. Usual morphological aspects join scattered and microcystic pattern with epithelioid or signet-ring cells. According to the morphological features, main differential diagnoses are adenocarcinoma metastasis, vascular tumors or mesotheliomas. Immunohistochemistry provides precious help to confirm the mesothelial origin thanks to positivity of epithelial markers (CK7, AE1-AE3, CK5/6) coupled to mesothelial markers (D2-40, Calretinin, WT1). On the other hand, there is no loss of BAP1 by immunohistochemistry and usually a surexpression of P16. Adrenal gland adenomatoid tumor is a benign tumor, which can be promoted by iatrogenous or constitutive immunodepression.

TRAF7 mutations and immunohistochemical study of uterine adenomatoid tumor compared with malignant mesothelioma.

Adenomatoid tumors (ATs) are benign mesothelial tumors with a good prognosis and usually occur in female and male genital tracts, including in the uterus. ATs are genetically defined by tumor necrosis factor receptor-associated factor (TRAF) 7 mutations, and a high number of AT cases show immunosuppression. On the other hand, malignant mesotheliomas (MMs) are malignant mesothelial tumors with a very poor prognosis. Genetic alterations in TRAF, methylthioadenosine phosphorylase(MTAP), and BRCA-associated nuclear protein 1 (BAP1) in ATs derived from the uterus and MMs of pleural or peritoneal origin were compared by gene sequence analysis or immunohistochemical approaches. Formalin-fixed paraffin-embedded tissues derived from patients were used for immunohistochemical staining of L1 cell adhesion molecule (L1CAM), BAP1, MTAP, and sialylated protein HEG homolog 1 (HEG1) in 51 uterine AT cases and 34 pleural or peritoneal MM cases and for next-generation sequencing of the TRAF7 gene in 44 AT cases and 21 MM cases. ATs had a significantly higher rate of L1CAM expression than MMs, whereas MMs had a significantly higher rate of loss of MTAP and BAP1 expression than ATs. There was no difference in the rate of HEG1 expression between the tumor types. Most of the ATs (37/44; 84%) had somatic mutations in TRAF7, but none of the MMs had somatic mutations in TRAF7 (0/21; 0%). In addition, a low number of AT cases were associated with a history of immunosuppression (9/51; 17.6%). TRAF7 mutation is one of the major factors distinguishing the development of AT from MM, and immunosuppression might not be associated with most AT cases.

Peritoneal well-differentiated papillary mesothelioma associated with infertility in a 37-year-old woman.

Well-differentiated papillary mesothelioma (WDPM) is an uncommon mesothelial tumor. The lesions may be single or multiple and usually behave in a benign or indolent fashion, sometimes persisting for many years. In the present case, a 37-year-old woman had experienced primary infertility for 12 years, and a diagnostic laparoscopy was performed. Approximately 200 mL of dark red, free fluid in the pelvis and more than 10 yellow-white nodules on the surface of the right round ligament, sacrum ligament, right fallopian tube, and both sides of the uterus were found. A lesionectomy was performed and immunohistochemical markers indicated WDPM with adenomatoid tumor. The patient was monitored by computed tomography and serum CA125 (cancer antigen 125) levels for 49 months with no recurrence. WDPM and adenomatoid tumor are both benign tumors of mesothelial origin. Because of the lack of effective radical treatment, regular follow-up is sufficient. However, the effects of estrogen and progesterone on WDPM and adenomatoid tumors during ovulation or pregnancy remains unclear. Although WDPM is not life threatening, a strategy to fulfill the fertility requirements of women with this condition is a new challenge for infertility doctors.

Paratesticular tumors. A clinicopathological study from a single tertiary hospital in North India.

OBJECTIVE: Paratesticular tumors (PTT) are rare and form a heterogenous group, ranging from benign to malignant high grade sarcomas. This study was undertaken to describe the clinicopathological spectrum of PTTs received over a 20-year period. METHODS: All primary and secondary PTTs diagnosed from 2000 to 2019 in the pathology department of a tertiary care hospital in North India were retrospectively reviewed. Gross, histopathological features and immunohistochemistry (IHC) findings were correlated with clinical details. RESULTS: A total of 169 intra-scrotal tumors were diagnosed during the study period, out of which there were 30 PTTs (in 27 patients) comprising 17.75%. Age range was 4 to 85 years (median 58 years). Benign PTTs were the commonest (n = 21, 70%), followed by metastasis to the paratesticular region (n = 6, 20%) and then primary malignant PTTs (n = 3, 10%). The commonest benign PTT was lipoma (n = 16, 76.19%), followed by adenomatoid tumor (n = 3, 14.28%) with one case each (4.76%) of cellular angiofibroma and hemangioma. Among primary malignant PTT, there were two cases of rhabdomyosarcoma, and one case of biphasic malignant mesothelioma. Metastatic tumors included four cases of prostatic adenocarcinoma, and one case each of pancreatic signet ring cell carcinoma and clear cell renal cell carcinoma. CONCLUSION: PTTs show a wide clinicopathological spectrum. Benign PTTs are commoner than malignant PTTs. Meticulous grossing and histopathological examination supplemented by IHC is essential for an accurate diagnosis of this heterogenous class of tumors, which influences the role of adjuvant therapy and patient prognosis.

Adenomatoid tumor of the skin: Differential diagnosis of an umbilical erythematous plaque.

Adenomatoid tumors are benign tumors of mesothelial origin that are usually encountered in the genital tract. Although they have been observed in other organs, the skin appears to be a very rare location, with only one case reported in the literature to our knowledge. We report a second case of an adenomatoid tumor, arising in the umbilicus of a 44-year-old woman. The patient presented with an 8-month-old erythematous and firm plaque under the umbilicus. A skin biopsy showed numerous microcystic spaces dissecting a fibrous stroma and lined by flattened to cuboidal cells with focal intraluminal papillary formation. This little-known diagnosis constitutes a diagnostic pitfall for dermatopathologists and dermatologists, and could be misdiagnosed as other benign or malignant entities. Through this case report, a practical approach and diagnostic keys have been devised to avoid misdiagnosis and overtreatment.

Leiomyoadenomatoid Tumor of Epididymis: A Variant of Adenomatoid Tumor.

Adenomatoid tumor is a rare tumor of mesothelial origin, usually arising in the epididymis. It is the most common paratesticular tumor of middle-aged men. A rare variant of adenomatoid tumor is leiomyoadenomatoid tumor which is characterized by prominent spindle cell myoblastic and myofibroblastic proliferation in the background of an adenomatoid tumor with tubular spaces lined by mesothelial cells. In some cases, the spindle cell component obscures the adenomatoid tumor component, complicating accurate diagnosis. Here, we report two cases of paratesticular leiomyoadenomatoid tumor in 28-year-old and 50-year-old patients. The tumors from both cases were centered in the epididymis and measured 1.0 cm and 3.0 cm, respectively. Both had similar morphology with myofibroblastic proliferation in one case and myoblastic (smooth muscle) proliferation in the other. Both cases followed a benign course without local recurrence or distant metastasis for 14 and 22 months postoperatively, respectively. We propose the use of the term "adenomyomatoid tumor" to describe a neoplasm exhibiting adenomatoid tumor admixed with either leiomyomatous or myofibroblastic proliferation.