A Rare Subglottic Pleomorphic Adenoma: Magnetic Resonance Findings.

No previous study has published magnetic resonance imaging (MRI) findings for a subglottic pleomorphic adenoma. Here, we describe the case of a 62-year-old man with a subglottic pleomorphic adenoma. Endoscopic findings revealed a smooth-surfaced tumor arising from the subglottic posterior wall. MRI revealed the lesion as an isointense region on T1-weighted images, which was homogeneously enhanced. This lesion showed a heterogeneously hyperintense region on T2-weighted images. Diffusion-weighted imaging (DWI) showed slightly high intensity in the same area, with a normal or only slightly high apparent diffusion coefficient (ADC). Laryngomicrosurgery was performed for transoral excision of the subglottic tumor, resulting in a postsurgical diagnosis of pleomorphic adenoma.

Tracheal stenosis caused by unnoticed foreign bodies.

We describe an extremely rare case of tracheal stenosis caused by unnoticed microscopic fiber-like foreign bodies. A 66-year-old woman complained of dyspnea with inspiratory stridor. Magnifying electroendoscopy and computed tomography revealed stenosis involving the entire circumference of the tracheal lumen. Tracheotomy and biopsy were performed. Histologically, the lesion showed chronic inflammation with a deposition of fiber-like foreign bodies. The patient had no history of trauma or inhalation injury, but had undergone intratracheal intubation on 4 occasions. The lesion was incised using semiconductor laser photoresection, and the postoperative course was good. To the best of our knowledge, this represents the first report in the English literature of tracheal stenosis caused by unnoticed foreign bodies. The origin of these fiber-like foreign bodies remains unclear but might be related to chronic inflammation resulting from intratracheal intubations.

Urinary retention in a female patient caused by a urethral venous thrombus with massive edema.

Female urinary retention is rarely caused by an unknown condition. In such a case, common causes including neurogenic, anatomical, traumatic and iatrogenic voiding dysfunction are excluded. Both the radiological and histological findings specific to the condition remain unclear. We herein report a 75-year-old woman in whom a large venous mass and massive edema at the proximal urethra caused urinary outlet obstruction based on magnetic resonance images and histological findings. Ultrasonography and cystoscopy showed a urethral mass lesion protruding into the bladder neck. Fat-suppressed T2-weighted and gadolinium-enhanced T1-weighted magnetic resonance images suggested thrombosed veins with massive edema under the urethral mucosa. Transurethral resection of the mass lesion was carried out, and histopathological analysis proved the presence of thrombosed large veins. The patient regained normal urination, and the mass lesion did not recur. Thrombosed venous mass with edema at the proximal urethra might be an important cause of atypical urinary retention in women.

Pancreatic metastasis of leiomyoma found 27 years after uterine fibroid surgery.

A 59-year-old female was referred to our hospital for pancreatic tumor. She had undergone resection of uterine fibroids 27 years ago, and leiomyoma in the left thigh 8 years ago. Tumor markers were normal. A well-defined hypoechoic tumor measuring 26 mm × 22 mm was detected at the pancreatic tail using ultrasound. Inside the tumor, a small cystic structure was observed. High-sensitivity Doppler imaging revealed faint signals indicating blood flow from the periphery to the interior. Contrast-enhanced ultrasonography in the arterial phase revealed slow, heterogenous enhancement from the periphery to the center of the mass that occurred later than that in the pancreatic parenchyma. The tumor then exhibited homogeneous enhancement except for the periphery, with subsequent washout. Endoscopic ultrasound-fine-needle aspiration was performed. Histopathological examination diagnosed a leiomyoma. The left thigh tumor resected 8 years ago had been diagnosed as benign metastatic leiomyoma (BML). We considered that the current pancreatic tumor was also BML clinically. We report this case because pancreatic metastasis from uterine fibroids is extremely rare. Uterine fibroids are benign tumors, but there are rare cases of metastasis to various organs, which should be considered when performing ultrasound.

Primary Thyroid Lymphoma: Clinical Factors Predicting the Possibility of Diffuse Large B-Cell Lymphoma.

Aims: Among primary thyroid lymphomas (PTLs), diffuse large B-cell lymphoma (DLBCL) has a poorer prognosis than other indolent lymphomas such as mucosa-associated lymphoid tissue (MALT) or follicular lymphoma (FL). However, the clinical differences between DLBCL and indolent lymphoma remain unclear. Therefore, this retrospective study on PTL was aimed at investigating the clinical differences between DLBCL and indolent lymphomas and identifying the factors differentiating DLBCL from indolent lymphomas. Materials and Methods: Medical records of 28 patients diagnosed with PTL and treated at our institution between 2005 and 2022 were retrospectively analyzed. Data on the following clinical variables were extracted: sex, age, symptoms (pain and dysphagia), ultrasonographic appearance patterns, the presence of airway stenosis on computed tomography and laryngeal endoscopy, blood test results, disease stage, and pathological diagnosis. Results: In all, 13 patients were histologically diagnosed with DLBCL, 12 with MALT lymphoma, and 3 with FL. Significant differences in disease-specific survival rates were evident between the DLBCL and indolent lymphoma groups (68.2 vs 100%, P = .043). High lactate dehydrogenase levels (>230 U/mL) and airway stenosis were observed only in patients with DLBCL. Multivariate analysis identified that the presence of a linear echoic strand pattern and the absence of an echoic nodular pattern on ultrasound were independently associated with DLBCL (P = .0497 and .012, respectively). Conclusion: DLBCL can cause airway stenosis. The linear echogenic strand pattern and the absence of a nodular pattern should be recognized as predictive factors of DLBCL.

Application of Lip Biopsy for the Histological Diagnosis of Immunoglobulin G4-Related Disease.

We describe the case of a 51-year-old woman with immunoglobulin G4-related disease (IgG4-RD) diagnosed using lip biopsy. She reported having bilateral submandibular nodules for a month. Magnetic resonance imaging showed diffuse swelling in the bilateral submandibular glands (SMGs), suggesting inflammatory changes. Laboratory data revealed an elevated level of serum IgG4. Fine needle aspiration cytology of the SMG showed a considerable number of lymphocytes with degeneration but did not demonstrate specific findings for a definitive diagnosis of IgG4-RD. Lip biopsy was performed, and a biopsy specimen from the labial salivary gland showed abundant lymphoplasmacytic infiltration with a large number of IgG4-positive cells. The patient was diagnosed with IgG4-RD based on histological and laboratory findings. Findings of further examinations revealed that the patient had autoimmune pancreatitis, confirming our diagnosis. Four months after prednisolone administration, improvement of the submandibular and pancreatic lesions was observed. One year after the initial presentation, the serum IgG4 level was normalized. In cases of IgG4-RD with salivary gland involvement, lip biopsy might be one of the options for the histological diagnosis of IgG4-RD.

Methotrexate-Associated Lymphoproliferative Disorders Mimicking Granulomatosis With Polyangiitis: A Radiological Diagnostic Challenge.

Methotrexate-associated lymphoproliferative disorders (MTX-LPD) frequently involve the extranodal organs throughout the body. Among the extranodal occurrences of MTX-LPD, pulmonary involvement is most frequent. In contrast, there are only a few reports of MTX-LPD in the nasal cavity or paranasal sinuses. Moreover, there are no previous reports of MTX-LPD mimicking granulomatosis with polyangiitis (GPA) in imaging examinations. We describe a case of a 53-year-old woman with MTX-LPD mimicking GPA in the nasal cavity and lungs. She complained of left nasal obstruction and discharge, general fatigue, and continual fever for 2 months. The patient had been diagnosed with rheumatoid arthritis and received methotrexate (MTX) for over 10 years. Contrast-enhanced computed tomography revealed unenhanced masses in the nasal cavity and multiple masses with cavitary changes in the bilateral lungs, suggesting GPA. However, histological examination of the nasal lesion and a history of MTX treatment indicated a diffuse large B-cell lymphoma type MTX-LPD. Two weeks after MTX withdrawal, prominent improvements in both lesions were observed. Complete regression of the nasal lesion was observed 3 months after discontinuation of MTX. Thus, MTX-LPD may mimic GPA in imaging examinations.

The fine-needle aspiration cytology and clinical findings of Kikuchi-Fujimoto disease in pediatric patients: a retrospective clinical study.

BACKGROUND: Histological evaluation of lymph node is crucial for the definitive diagnosis of Kikuchi-Fujimoto disease (KFD). However, lymph node biopsy under local anesthesia is often difficult in pediatric patients. OBJECTIVES: We evaluated cytological findings for pediatric patients with prolonged cervical lymphadenitis clinically suggestive of KFD and investigated the clinical characteristics of patients diagnosed with KFD by fine-needle aspiration cytology (FNAC). METHODS: This retrospective clinical study included 58 Japanese pediatric patients with cervical lymphadenitis who underwent FNAC. RESULTS: Cytological diagnosis was KFD for 22 and suspicion of KFD for 11 patients. The remaining 25 patients were diagnosed with non-specific lymphadenitis (NSL). Tenderness was independently associated with a higher frequency of both KFD in narrow and broad senses, compared with NSL (p = .009; p = .038). The percentage of patients who underwent FNAC within 28 days from symptom onset tended to be higher among patients with KFD in a narrow sense than those with NSL (p = .052). CONCLUSION: This study indicated that the period from symptom onset to FNAC (<28 days) and the symptom of tenderness were associated with the cytological diagnosis of KFD.

Supraglottic subepithelial benign mass lesions: Focus on clinical features of sialolipoma-like lesion.

OBJECTIVES: Sialolipoma has been classified as a benign soft tissue lesion in the 2017 World Health Organization classification of head and neck tumors. To our knowledge, only one case of laryngeal sialolipoma has been reported in the English literature. We conducted a retrospective study to identify clinical characteristics of supraglottic sialolipoma-like lesion and differentiate it from other supraglottic subepithelial masses. METHODS: Medical records of 16 patients with supraglottic subepithelial benign mass lesions who underwent histological evaluation between 2003 and 2019 were retrospectively analyzed. Sialolipoma-like lesion was defined as a local finding of a well-circumscribed gross mass with pathological presence of salivary gland-like parenchymal lobules with evenly interspersed adipose tissue. RESULTS: Eight patients showed histological positivity for sialolipoma-like lesion, 3 for amyloidosis, 2 for hemangioma, and 1 each for cyst, lymphoid hyperplasia, and chondrometaplasia. Sialolipoma-like lesion tended to be predominant among men; those affected had a mean age of 52.8 (range, 39-74) years. By contrast, among patients with amyloidosis, the ratio of men to women was 1:2 (100% vs. 33%; p = 0.055). Fiberscopic examination of all patients with sialolipoma-like lesions identified well-circumscribed, yellowish masses, closely resembling local amyloidosis findings. Sialolipoma-like lesion was associated with a significantly higher body-mass index (BMI; 27.4 ± 2.8 kg/m2) than amyloidosis (21.6 ± 1.4 kg/m2; p = 0.014). The transoral approach was used for lesion resection in all patients with sialolipoma-like lesion. No patient experienced postoperative recurrence. CONCLUSION: Laryngeal sialolipoma-like lesion might be more prevalent than was previously reported, and histological examination is important to differentiate it from amyloidosis. Supraglottic sialolipoma-like lesion must be differentially diagnosed in patients with high BMI presenting with well-circumscribed, yellowish supraglottic masses.

Uterine leiomyosarcoma arising in leiomyoma: clinicopathological study of four cases and literature review.

In this study we examined four cases of leiomyosarcoma (LMS) arising in leiomyoma (LM). The patients ranged from 40 to 64 years old. Symptoms were unspecific and preoperative diagnosis of LMS had not been made. Size of the tumors ranged from 4 cm to 20.5 cm. All patients were in stage I and no patients developed recurrent disease. Histologically, the LMS component showed moderate to severe cytological atypia and an increased mitotic count in all cases. Tumor cell necrosis in the LMS was seen in three cases. Associated LM component was cellular in one case. The LMS component showed p53 expression in three of four cases and p16 expression was seen in two cases. The LM component lacked p53 expression in all cases and demonstrated p16 expression in two cases, including one case of cellular LM. In all cases, Ki-67-positive cells were less than 5% in the LM component and more than 25-30% in the LMS component. This study reveals that: (i) malignant transformation can occur even in relatively small LM; (ii) patients with LMS with an LM component have a favorable prognosis; and (iii) immunohistochemical examination of p53, p16 and Ki-67 is useful for identification of malignant focus.

Ovarian clear cell carcinoma arising in a mucinous cystadenoma.

Here we report a rare case of ovarian clear cell carcinoma (CC) arising in a mucinous cystadenoma. A 59-year-old woman presented with a dull pain at the left inguinal area. She was found to have a left ovarian cyst measuring 4.7 cm and underwent a salpingo-oophorectomy. The unilocular cyst contained thick mucin, lacking any solid components. Histopathologically, most of the cyst wall was lined with a single layer of tall columnar cells with basophilic mucin. Within limited areas, small glands consisting of clear cytoplasm aggregated and intermingled with or partially transitioned to the mucinous glands. The clear cell components had adenofibroma-like features devoid of significant nuclear atypia or stromal invasion, including distinctive foci of CC with a hobnail appearance and hyperchromatic nuclei surrounded by desmoplastic or hyalinized stroma. Immunohistochemically, both mucinous and clear cells were positive for estrogen receptor. Hepatocyte nuclear factor-1beta and laminin were positive in all of the clear cell components, in contrast to a consistent lack of staining in the mucinous epithelium. An eleven-month follow-up revealed no recurrence or metastasis. This case was considered as an early manifestation of ovarian CC arising in a mucinous cystadenoma in the absence of demonstrable endometriosis.

A vulvar fibroepithelial stromal polyp appearing in infancy.

We present here a first case of vulvar fibroepithelial stromal polyp (vFSP) appearing in infancy, including previously undescribed papule as an early form of vFSP. A 7-month-old girl presented with a small (adzuki-sized), soft, erythematous papule in the right labium majus. The size of the lesion increased up to 2.3 cm in the following 7 months and exhibited cerebriform or frond-like configuration. The surgically removed polyp was characterized by the proliferation of bland spindle cells with tapered cytoplasm, and the diagnosis of vFSP was established. Twenty-one months after the first surgery, a recurrent polyp 2.0 cm in size was resected. No further recurrence has been observed in the 17 months since the second surgery.

The efficacy of OK-432 sclerotherapy on thyroglossal duct cyst and the influence on a subsequent surgical procedure.

Background: Although there are studies regarding the efficacy of OK-432 sclerotherapy on thyroglossal duct cyst (TDC), its effects on surgical procedure following this therapy have not been properly described. Objectives: The present study aimed to delineate the prognostic factors of OK-432 sclerotherapy in patients with TDC and investigate its influence on subsequent surgical procedure and the histological characteristics in patients with poor response to OK-432 sclerotherapy. Material and methods: We conducted a retrospective analysis of the medical records of 20 TDC patients treated with OK-432 sclerotherapy. Results: Of the 20 patients, OK-432 sclerotherapy was effective in 5 patients (25.0%). OK-432 showed a lower effective rate in multilocular cysts (9.1%) than in unilocular cysts (44.4%), although not significantly. Five cases were treated with surgery following OK-432 sclerotherapy. There was no significant difference in the operating time and the amount of bleeding between patients with and without OK-432 sclerotherapy. From the results of the histological examination of the cyst wall, two cases had stratified squamous epithelium and two cases showed the absence of lymphocyte infiltration. Conclusion and significance: OK-432 sclerotherapy is an acceptable initial treatment for TDC, especially in unilocular cysts, because of lack of influence on surgical procedure.

Aberrant expression of an "intestinal marker" Cdx2 in pyloric gland adenoma of the gallbladder.

The aim of this study was to survey Cdx2 expression in pyloric gland adenoma (PGA) of the gallbladder. We reviewed 29 PGA cases, ten (34.4%) and seven (24.1%) of which showed intestinal metaplasia (IM) and squamous morule (SM), respectively. The immunostaining for Cdx2, beta-catenin, MUC5AC, MUC2, MUC6, and M-GGMC-1 was performed and scored (0 = negative, 1+ = <10%, 2+ = 10% to <30%, 3+ = 30% to <50%, 4+ = 50% to <70%, 5+ = 70-100%). Although its scores were relatively low (1+ or 2+), Cdx2 was frequently expressed in 27 cases (93.1%). Not only goblet and/or Paneth cells were positive but also non-IM cells in PGAs, as opposed to the lack of staining in the background mucosa. Cdx2 scores were not correlated with those of IM (p = 0.485) and MUC2 (p = 0.868). Of note, Cdx2 was positive in foci of SM in all seven cases, and there was a significant difference in Cdx2 scores between PGAs with and without SM. Furthermore, the p value of scores between Cdx2 and beta-catenin was 0.051, and both mean labeling indices (LIs) were correlated (r = 0.736). With Cdx2, higher morular LIs than glandular LIs were observed (p = 0.001). Finally, we concluded that aberrant Cdx2 expression in PGAs is closely associated with nuclear beta-catenin expression and SM in contrast with IM.

Mesonephric adenocarcinoma of the uterine corpus: a case report and review of the literature.

Mesonephric adenocarcinoma (MA) is a rare tumor of the female genital tract, mainly in the cervix and vagina, which is usually associated with mesonephric remnants or mesonephric hyperplasia. In the uterus corpus, MA is as rare as mesonephric structures, and only a few cases have been previously reported. Here, we report a rare case of MA of the uterine corpus. A 73-year-old woman presented with multiple nodules in the bilateral lung. Abdominal computed tomography scan confirmed a uterine tumor measuring 8.6 cm. All tumor markers, including CA125, were within normal limits. A total hysterectomy and bilateral salpingo-oophorectomy were performed. The tumor was confined to the myometrium and showed strong resemblance to cervical MA despite the absence of mesonephric hyperplasia or remnants. The most striking pattern consisted of large sheets of small round tubules, often with densely eosinophilic secretions in the lumen. In addition, the ductal pattern simulating endometrioid adenocarcinoma was also noted. A mixture of tubular and ductal patterns, most predominantly seen, formed more complex tubules and cribriform structures. Other elements consisted of a retiform pattern, serous adenocarcinoma-like papillary budding, and glomeruloid morphology. Immunohistochemically, the tumor cells were positive not only for cytokeratin, epithelial membrane antigen, and vimentin but also for CD10 and calretinin. No staining was identified for estrogen receptor, progesterone receptor, or androgen receptor. Adjuvant chemotherapy was begun for the patient, who is alive with disease 28 months later. We review the previously published cases of MA and discuss the principal differential diagnosis of MA in the uterine corpus.

CD8+, CD56+ (natural killer-like) T-cell lymphoma involving the small intestine with no evidence of enteropathy: clinicopathology and molecular study of five Japanese patients.

The present study reports five CD8+, CD56+ (natural killer (NK)-like) T-cell lymphomas involving the small intestine without evidence of enteropathy, from Japan. Three were intestinal T-cell lymphoma. The site of origin of the other two was not definitive. Four of five patients underwent emergency operation because of intestinal perforation. The small intestines of these patients had multiple ulcerative lesions with or without demarcated tumors. Histologically, the lymphoma cells were monomorphic or slightly pleomorphic and displayed epitheliotropism of varying degrees. Lymphoma cells of all patients shared the common phenotype: CD3+, CD4-, CD5-, CD8+, CD56+, CD57-, T-cell intracellular antigen-1+, granzyme B+. In contrast to nasal/nasal type NK-cell lymphomas, they had clonal rearrangement of T-cell receptor(TCR) genes and were negative for EBV-encoded RNA. Immunohistochemistry and genetics suggested that three cases were of alpha beta T-cell origin and two cases were of gamma delta T-cell origin. There was no evidence of enteropathy in any patient. The cases followed a clinically aggressive course with a frequent involvement of lung. According to the classification based on the recent genetic studies of European enteropathy-type intestinal T-cell lymphoma (ETL), the present cases could be classified as type 2 ETL.

Ovarian stromal cells as a source of cancer-associated fibroblasts in human epithelial ovarian cancer: A histopathological study.

Fibroblasts are a major component of cancer tissue and known to contribute to cancer progression. However, it remains unknown whether they are derived from local fibroblasts or of other origin. This study was designed to identify the contribution of local stromal cells to cancer stroma in human epithelial ovarian cancer. Seventy-six cases of surgically resected primary ovarian carcinoma (48 cases confined to the ovaries and 28 cases with distant metastases) and 17 cases of secondary ovarian tumor (e.g. colon cancer metastasized to the ovary) were enrolled in this study. The tissues were immunostained for forkhead box protein L2 (FOXL2), a transcription factor crucial for ovarian development and function, and markers for cancer-associated fibroblasts (CAFs) and inflammatory cells. Under normal condition, FOXL2 expression was restricted to ovarian stromal cells and some other types of cells in female genital tracts and never found in other sites of the body. FOXL2-positive cells were found in all primary and secondary tumors in the ovary, and were the dominant stromal cells in most cases. In contrast, only a few FOXL2-positive cells were found in peritoneal seeding sites of four serous carcinoma cases, and all the other tumors at extraovarian sites had no FOXL2-positive cells. FOXL2-positive cells in the ovarian lesion variably expressed CAFs markers, such as alpha-smooth muscle actin and fibroblast activating protein, as determined by double immunostaining. Background inflammation, but not histological subtype or origin of the neoplasm seemed to correlate with the proportion of FOXL2-positive cells. These results suggest that ovarian stromal cells are the main source of cancer stroma in the ovary but do not seem to move to distant sites via circulation together with tumor cells. Our results also support the hypothesis that cancer-associated fibroblasts may originate locally, which was previously demonstrated using animal models.

Sclerosing hemangioma with florid endobronchial and endobronchiolar growth.

Sclerosing hemangioma (SH) with endobronchial growth (SH-EG) is an extremely unusual form of SH. A case of SH-EG in a 47-year-old female is described. She suffered from a productive cough for 4 months. A chest CT scan revealed a well-circumscribed, parenchymal mass with endobronchial lesions continuously extending to the right main bronchus. Right upper sleeve lobectomy was carried out. The tumor was 4.8 x 3.5 cm in size, tan-brown, and elastically soft. It was located in the pulmonary parenchyma of the right upper lobe and continuously extended to the right upper bronchus. Furthermore, the main tumor also spread into segmental bronchi and peripheral bronchioles. Microscopically, the tumor consisted of round to cuboidal cells with papillary and solid patterns, partly showing sclerosis and hemorrhage. For 2.5 years after surgery, the patient has been well without recurrence or metastasis. Florid intrabronchial extension as seen in this case has never been documented in SH. To form an endobronchial component, it seems to be crucial that the parenchymal SH is located adjacent to the bronchus and involves it, followed by the destruction of the bronchial cartilage.