Primary adenomatoid tumor of the adrenal gland: A case report and literature review.

RATIONALE: Adenomatoid tumors are rare benign tumors, mainly involving the reproductive tract, such as the epididymis in men and the uterus and fallopian tubes in women. However, a few cases can occur outside the reproductive tract. Herein, we report a rare case of a primary adenomatoid tumor of the adrenal gland. PATIENT CONCERNS: A 50-year-old man underwent ultrasound examination and was found to have a right adrenal mass without elevated blood pressure, weakness after fatigue, frequent nocturnal urination urgency, pain, or a history of hematuria. The patient's general health was normal. Computed tomography revealed a polycystic mixed-density lesion in the right adrenal region, approximately 7.3 × 4.5 cm in size. DIAGNOSES: Based on the clinical information, morphological features, and immunohistochemistry results, a pathological diagnosis of primary adenomatoid tumor of the adrenal gland was made. INTERVENTION: Excision of the right adrenal gland and tumor through the 11 ribs. OUTCOMES: The patient's postoperative course was uneventful. LESSONS: Preventing misdiagnosis adenomatoid tumors with other types of adrenal gland tumors or metastatic tumors is imperative. Morphological and immunohistochemical features can help diagnose primary adenomatoid tumors of the adrenal gland.

A Rare Pancreatic Tumor with Adenomatoid Tumor-like Findings.

A 74-year-old woman was referred to our hospital for the evaluation of slightly elevated tumor marker levels. Computed tomography revealed a well-demarcated tumor, approximately 15 mm in diameter, in the pancreatic tail. Endoscopic ultrasound-guided fine-needle aspiration findings suggested poorly differentiated cancer. The tumor was surgically resected, but postoperative pathologic confirmation was not possible. After one year without treatment and no recurrence, an evaluation by a specialized facility was requested for a definitive diagnosis. Adenomatoid tumor was deemed most likely based on the histopathology and immunostaining findings; however, a definitive diagnosis was difficult because of atypical findings. The patient was recurrence-free for 36 months at the last follow-up.

Adenomatoid tumors of ovary mimicking malignancy: report of 2 cases and literature review.

BACKGROUND: Adenomatoid tumors (ATs) are benign tumors originating from the mesothelium. ATs of the ovary are rare, and can easily be confused with malignancy due to the histomorphological diversity. Thus, it is difficult in histopathological and differential diagnosis, especially during intraoperative frozen pathological diagnosis, which directly affects the resection scope of surgery. CASE PRESENTATION: In this study, we reported two patients (58 and 41 year old) with ovarian ATs. AT of patient 1 occurred in both ovaries at different time points and she had been diagnosed with Hashimoto's thyroiditis. AT of patient 2 occurred in right ovary. Intraoperative frozen pathological diagnosis was performed in both cases and laparoscopic salpingo-oophorectomy was undergone on the lesion side according to benign freezing diagnostic result. Ovarian ATs, the final diagnoses of the 2 cases were concluded after histological, extensive immunohistochemical (IHC), histochemical, and fluorescence in situ hybridization analyses. CONCLUSIONS: Our results show that ovarian ATs may not be related to BAP1 or CDKN2A/p16 mutations. In addition, the case 1 suggests that ATs may be associated with immune dysregulation. When encountering such similar lessions, we recommend that a series of immunohistochemical, histochemical and molecular biological techniques should be used for diagnosis and differential diagnosis to avoid misdiagnosis. Improving understanding of the rare ovarian ATs which mimic malignancy is necessary to prevent overresection.

Leiomyo-adenomatoid Tumor of the Uterus: Finding the Rare in the Routine.

Leiomyo-adenomatoid tumour (LMAT) is a rare benign neoplasm and very few cases of LMAT of the uterus are documented in the literature. Uterine LMATs are usually detected incidentally during the histopathologic evaluation of routine myomectomy or hysterectomy specimens for leiomyomata. Thorough evaluation of the morphological features and a concise immunohistochemical panel allows for accurate classification of this benign neoplasm.

Leiomyoadenomatoid tumours of the epididymis: A new case report and review of the literature.

Benign tumours of the epididymis are rare, and the most common tumour types include adenomatoid tumours, representing more than half of all cases, and leiomyomas. Here, we reported a case of leiomyoadenomatoid tumours of the epididymis, a very rare, benign histological entity with only few cases described in the English literature, which have been reviewed and summarised. Clinically, the lesion presented as a solitary mass growing at the level of the tail of the right epididymis. After the intraoperative frozen section analysis revealed a benign adenomatoid lesion, the mass was enucleated with a conservative surgery sparing the testis. This case highlights the importance for both pathologists and urologists to be aware of these rare, but benign, tumours, to avoid misdiagnosis, especially in the setting of frozen intraoperative consultation, or primary radical surgical procedures, as radical orchiepididymectomy without frozen section consultation.

Adenomatoid Tumor of the Uterus: A Report of 6 Unusual Cases With Prominent Cysts Including 4 With Diffuse Myometrial Involvement, 4 With Uterine Serosal Involvement, and 2 Presenting in Curettage Specimens.

We evaluated the clinicopathologic features of 6 adenomatoid tumors of the uterus with unusual features. All the tumors differed grossly from the usual adenomatoid tumor, typically being ill-defined and occupying >50% of the myometrium, essentially replacing it in 4. The neoplasm extended to the endometrium in 2 cases and in one of these it formed an intracavitary mass; in both the tumor was first diagnosed in a curettage. In the other 4 cases, the adenomatoid tumor was discovered in a hysterectomy specimen performed for irregular vaginal bleeding (3 patients), and the finding of a pelvic mass on a computed tomography scan in a patient with right lower quadrant pain. The tumors extended to the uterine serosa in the form of small grape-like vesicles or cysts in 4 cases. All tumors contained the typical small often irregularly shaped spaces but also had prominent cysts. When cysts involved the serosa, the microscopic appearance mimicked that of peritoneal inclusion cysts. In one case with serosal involvement, a prominent papillary pattern was also present. The cysts were typically closely packed with minimal intervening stroma but were occasionally separated by conspicuous smooth muscle bundles. The stroma in one case was extensively hyalinized. Two tumors were focally infarcted. A striking, but minor, solid growth in which the tumor cells were arranged in tightly packed nests or interanastomosing cords and trabeculae was seen in 2 tumors. The unusual gross and microscopic features of these tumors can cause significant diagnostic difficulty and bring into the differential diagnosis entities that are usually not realistic considerations. The presentation of 2 tumors in a curettage specimen represents an unusual clinical aspect.

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.

Non-random adenomatoid tumours of the female genital system: A comparative clinicopathologic analysis of 14 cases.

The aim of this study was to evaluate adenomatoid tumours (AT) clinicopathologically in the female genital tract and compare the histomorphological features of ATs according to their uterine or tuba-ovarian location. Cases of AT were excised and collected from female genital tracts between the years of 2010-2017. Cases were evaluated depending on their clinical findings, localisation and pathological properties. There were 14 cases of AT. Ten cases were uterine, and 4 cases were adnexal tumours. The diagnostic ratio of uterine ATs was 64.3%, and of tuba-ovarian ATs was 21.4% (P > 0.05). The size of the largest tumour was 6 cm. Two of the uterine and one of the ovarian cases had a macrocyst; 2 uterine and one ovarian case had a microcyst; and 6 uterine had a combined microcystic/trabecular pattern. Uterine cases showed a higher number of smooth muscle component, signet-ring cells and infiltrative nature compared with other cases (P < 0.05). All uterine cases were infiltrative. Most of ATs of the female genital system were small in size and incidentally diagnosed in our cases but rarely detected as an adnexal mass forming lesion which mimics a malignancy. A comparative clinicopathologic analysis of these cases should be considered with the histomorphological and immunohistochemical features for an accurate differential diagnosis.

Uterine adenomatoid tumor associated with lymph node lesions: a case report.

We report a case of uterine adenomatoid tumor (AT) with regional lymph node involvement in a 49-year-old woman. Magnetic resonance imaging revealed an aggregated cystic mass in the posterior uterine wall with partial protrusion of the tumor outside the uterus, and cystic masses of same characteristics in the bilateral obturator and right common iliac lymph nodes. FDG PET/CT revealed no significant FDG uptake in the uterine and lymph node lesions. Taking possible lymph node metastasis into consideration, hysterectomy and lymph node biopsy were performed and it revealed AT of the uterus and the lymph nodes histopathologically.

Respiratory Epithelial Adenomatoid Hamartoma is Frequent in Olfactory Cleft After Nasalization.

OBJECTIVES: To assess the site and histopathology of polyps at the first revision surgery for recurrent nasal polyposis (NP) after radical ethmoidectomy (nasalization). STUDY DESIGN: Retrospective study. METHODS: Between January 2008 and December 2015, a total of 62 patients having undergone revision surgery for recurrent NP after nasalization were included. The site and histology of the recurrence of polyps were analyzed according to operative and pathological reports. RESULTS: Histology showed classical inflammatory nasal polyps (CINP) in 91% of nasal cavities at primary surgery versus respiratory epithelial adenomatoid hamartoma (REAH) or REAH associated to CINP in 54.8% at revision surgery (P < .0001). Polyps were principally observed in the ethmoidal complex in 70% of nasal cavities during primary surgery and in the olfactory clefts in 88.7% during revision surgery (P < .0001). The mean interval between nasalization and first revision surgery was 8.8 ± 4.4 years (0.4-21.7 years). This interval was significantly shorter for grade 3 polyps, polyps removed from both ethmoidal complex and olfactory cleft at primary surgery, association of CINP and REAH at primary surgery, and when primary surgery had preserved the middle turbinates. CONCLUSION: Polyp recurrences after nasalization were mainly observed in the olfactory clefts and can be different histological features: inflammatory polyps, respiratory epithelial adenomatoid hamartoma, or a combination of both. LEVEL OF EVIDENCE: 4 Laryngoscope, 130:2098-2104, 2020.

Synchronous occurrence of benign mesothelioma and adenomatoid tumor of uterus: A case report and review of literature.

INTRODUCTION: Synchronous occurrence of benign cystic mesothelioma and adenomatoid tumor of uterus (UAT) are very rare and few cases have been published in the English literature. They are easily misdiagnosed as malignant by clinicians, due to the lack of reports. PATIENT CONCERNS: A case of benign mesothelial combined with uterus adenomatoid tumor (UAT) in a 48-year-old Chinese woman was reported. Our patient presented with abdominal pain and surgery showed a large subserous mass (12.0 × 11.4 × 9.8 cm) combined with a small intramural solid nodule (2.0 × 1.0 × 1.0 cm), and multiple minute neoplastic growth on the ovary. DIAGNOSIS: Due to the patient's symptoms, pathological findings, she was diagnosed with synchronous occurrence of benign mesothelioma and UAT. INTERVENTIONS: We treated her with a total hysterectomy and bilateral adnexectomy. OUTCOMES: The patient is now in stable condition, without any signs of recurrence during 1 year of follow-up. LESSONS: Most mesotheliomas are malignant, synchronous occurrence of benign mesothelioma and UAT are extremely rare. And they are often misdiagnosed as malignancy by clinicians. Our case report can improve the awareness of the disease and avoid excessive treatment.

[Tunica albuginea adenomatoid tumor. A challenging case managed conservatively]. / Tumor adenomatoide da túnica albugínea. Um desafio no tratamento conservador.

The adenomatoid tumors are rare benign mesothelial lesions that in males affect mainly the epididymis. They account for over one third of paratesticular tumors, and the treatment of choice has been surgical excision. The origin in testicular parenchyma or the tunica albuginea is extremely rare. Clinical signs and imaging studies are often inconclusive in differentiate from a more common malignant intratesticular solid tumour, which can result in unnecessary orchiectomies. We present a case of adenomatoid tumor of tunica albuginea where clinical suspicion and intraoperative frozen section analysis led to a partial orchiectomy.

Testis Sparing Surgery for Benign Testicular Masses: Diagnostics and Therapeutic Approaches.

PURPOSE: Small benign testicular masses are often misinterpreted as germ cell tumors and immediate inguinal orchiectomy is performed. We analyzed the diagnostic and therapeutic workup of testicular masses to improve preoperative stratification algorithms. MATERIALS AND METHODS: We performed a retrospective, single center analysis of the records of 522 patients diagnosed with primary testicular masses of unknown malignant potential. RESULTS: A total of 28 patients (5%) showed a primary benign tumor after resection, including Leydig cell tumors in 9 (32%), epidermoid cysts in 9 (32%), adenomatoid tumors in 8 (29%) and Sertoli cell tumors in 2 (7%). The median volume of benign tumors was significantly less than that of malignant tumors (0.75 cm3, range 0.1 to 2.1 vs 15, range 4.5-39.9, p ≤0.001). At a cutoff of 2.8 cm3 tumor volume most accurately differentiated between benign and malignant disease, and it was a predictor of malignancy with 83% sensitivity and 89% specificity (OR 1.389, 95% CI 1.035-1.864, p = 0.029). Symptom duration in patients with benign tumors was significantly longer (365 days, range 25.5 to 365 vs 20, range 7 to 42, p ≤0.001). Also, tumor markers were unaltered in benign lesions. In patients with benign tumors significantly more fertility disorders or cryptorchidism were found (p ≤0.001) as well as a tendency toward lower testosterone (3.9 µg/l, range 0.9 to 4.9 vs 5.3, range 3.5 to 6.8, p = 0.084). Testis sparing surgery was performed in 22 of all patients (79%) with benign tumors. There was no case of relapse during followup. CONCLUSIONS: Nongerm cell tumors should be considered when small testicular masses have a volume of less than 2.8 cm3 and there are hormone disorders or normal tumor markers. Immediate orchiectomy should be avoided, favoring testis sparing surgery.

Bilateral seminomas coexisting with adenomatoid tumor: An unusual occurrence.

Coexistence of bilateral seminomas and adenomatoid tumor is rare. We encountered an interesting case of bilateral testicular seminomas along with a paratesticular nodule which was diagnosed as an adenomatoid tumor on histology. Although seminomas and adenomatoid tumor are frequent neoplasms, bilaterality and their coexistence have been rarely described and can pose diagnostic difficulties. Herein, we describe a case of a 53-year-old man who presented with bilateral testicular swellings which were diagnosed as bilateral seminomas with an adenomatoid tumor in the left paratesticular region on histopathology. The pathological findings of these coexistent tumors and the utility of immunohistochemistry in establishing a correct diagnosis in such scenarios are discussed.

Tumor odontogénico adenomatoideo: reporte de un caso / Adenomatoid odontogenic tumor: report of a case

El tumor odontogénico adenomatoide (TOA) es una lesión benigna,infrecuente, clasificada por la OMS dentro de los tumores odontogénicoscon participación del ectomesénquima que muestra una morfología histológica muy peculiar. Esta entidad patológica es de baja prevalencia, representa 0.1% de los tumores y quistes de losmaxilares con raras recidivas. Su frecuencia de aparición es más comúnen pacientes jóvenes, generalmente mujeres, de mayor aparición en maxilar superior, asintomático, asociado a dientes sin erupcionar(principalmente caninos) que plantea diagnósticos diferenciales entre otras lesiones de mayor agresividad como el quiste dentígero y el ameloblastoma. Se presenta el caso de una paciente de nueve años de edad con lesión tumoral en el sector del maxilar superior izquierdo de 40 días de evolución. Clínicamente hay ausencia del órgano dentario número 23. Se indica la realización de una radiografía panorámica, en la cual se observa la presencia del órgano dental 23 en el piso de órbita del maxilar superior izquierdo. Se procede a la remoción quirúrgica con diagnóstico presuntivo de quiste dentígero, se biopsia el total de la lesión, con diagnóstico definitivo por histopatología de TOA, con buena evolución clínica odontológica.

The adenomatoid odontogenic tumor (TOA) is a rare, uncommon,WHO-classified lesion in odontogenic tumors with ectomesenchyma,which shows a very peculiar histological morphology. This pathologicalentity is of low prevalence representing 0.1% of the tumors and cystsof the jaws with rare recurrences. Its frequency of appearance is morecommon in young patients, generally females, of greater presentationin the upper jaw, asymptomatic, associated with unruptured teeth(mainly canines) that presents diff erential diagnoses among other moreaggressive lesions such as dentigerous cyst and ameloblastoma. Wepresent the case of a nine-year-old patient with tumor lesion in the leftupper jaw of 40 days of evolution. Clinically there is absence of the tooth23. A panoramic radiograph is indicated, in which the presence of thetooth 23 is observed in the orbital fl oor of the upper left jaw. Surgicalremoval is performed with a presumptive diagnosis of dentigerouscyst; the total of the lesion was biopsied, with defi nitive diagnosis byhistopathology of TOA with good odontological clinical evolution.

Laparoscopic Excision of a Uterine Adenomatoid Tumor and a Coexisting Ovarian Teratoma: A Case Report and Literature Review.

Adenomatoid tumors (ATs) are rare, benign neoplasms occurring mainly in reproductive organs such as the uterus, ovaries, fallopian tubes, and testes. Uterine adenomatoid tumors (UATs) are generally incidentally diagnosed during histopathological examination of excisional biopsies performed for other indications, most commonly uterine leiomyomas. We herein present a 38-year-old woman who underwent laparoscopic excision of a uterine leiomyoma and a right ovarian teratoma. Microscopic examination of the excisional biopsy revealed that the enucleated uterine tumor was composed of proliferating glandular tissue covered with single-layered cells that were surrounded by proliferating smooth muscle cells, corresponding exactly to the features of UATs. The excised ovarian cyst was confirmed to be a typical mature cystic teratoma. According to these histopathological findings, the patient was finally diagnosed with a UAT and coexisting teratoma. No recurrence was detected up to 6 months after excision. To the best of our knowledge, this is the eighth case report on laparoscopically enucleated UATs. Although recurrence risk may be low in UATs, further case reports are necessary to elucidate the safety and validity of laparoscopic excision for UATs.

A caecal pseoudotumour with an incidental adenomatoid testicular tumour in a man with right undescended testis: a case report.

BACKGROUND: Inflammatory pseudotumour refers to a non-malignant tumour-like mass resulting from an inflammatory reaction that is composed of granulation tissue with leukocyte infiltration that commonly occurs in the paediatric or young adult population. These tumours occur more commonly in the lungs and the orbit but rarely does it affect the gastrointestinal tract. It poses a clinical diagnostic challenge since it is a benign condition than can mimic the malignant counterpart. Our case is a rare presentation of the caecal pseudotumour in the presence of a right undescended abdominal testis evaluated as a caecal tumour with a differential diagnosis of a testicular malignancy. CASE PRESENTATION: We report a 53-year-old male who presented with clinical signs suggestive of right colon tumour and undescended right testis. Intra-operatively, a caecal mass was found with no clearly discernable appendix and extensive adhesion of the right colon to the retroperitoneum, to the liver and gall bladder. A testis was found adherent to the posterior aspect of the caecum and terminal ileum. A right hemicolectomy was performed. Histopathology findings revealed an inflammatory mass with abundant fibroblast proliferation and chronic inflammatory cells infiltrate, involving bowel wall and periceacal adipose tissue; no malignant cells were identified. The testis had within it an adenomatoid tumour nodule. He had uneventful recovery and was discharged home 7 days post-operatively. At the moment, he is symptoms free. CONCLUSIONS: The occurrence of right colonic inflammatory pseudotumour and co-existent adenomatoid testicular tumour arising from a cryptorchid testis is very unusual. This would make one incline towards a malignant testicular lesion in the presence of cryptorchidism. Testicular adenomatoid tumour is a rare benign neoplasm, mostly affecting fully descended testis and usually does not warrant orchidectomy for purposes of preserving testicular function. On the other hand, surgical resection remains the only safe and curative treatment option available for inflammatory pseudotumours.

Adenomatoid tumor of the mediastinum.

Adenomatoid tumors are relatively uncommon benign tumors of mesothelial origin, usually occurring in the male or female genital tracts. Extragenital adenomatoid tumors are quite rare. Here, we report a very rare case of adenomatoid tumor of the mediastinum. A 67-year-old woman was admitted to our hospital with an abnormal shadow that appeared on a routine chest radiograph. After further radiological evaluation suggested a diagnosis of thymoma, surgical resection was performed, and histologic and immunohistochemical examination yielded a definitive diagnosis of adenomatoid tumor. The patient remained clinically free of disease at follow-up, 19 months later.