Warthin tumor (WT) is a common benign salivary tumor of the parotid gland. Clinically, it occurs in men in their fifth to seventh decades who typically smoke cigarettes. WTs have been reported with different head and neck neoplasms and other salivary gland tumors within the same or another salivary gland. Kimura disease (KD) is a rare chronic inflammatory disease with unknown etiology affecting young to middle-aged Asian men. KD presents as an asymptomatic nodule in the head and neck area, with regional lymphadenopathy and salivary gland involvement.A 64-year-old Arabic man presented with a 10-year history of an asymptomatic swelling of the left face. Computed tomography showed a well-defined, multicystic mass with heterogeneous enhancement. The resected mass was composed of two distinct components. There was a well-demarcated proliferation of papillary and cystic oncocytic epithelium with lymphoid stroma, consistent with WT. Some areas exhibited sclerotic fibrosis, with multiple lymphoid follicles showing folliculolysis, follicular hyperplasia, and eosinophilic infiltrate. The patient’s immunoglobulin E level serum was elevated, confirming a coexisting KD. The patient underwent a left superficial parotidectomy, with no recurrence at a 30-month follow-up.This report describes the first concurrent case of WT and KD in the parotid gland. Warthin tumor (WT) is the second most common benign salivary gland tumor, accounting for 12–37% of parotid gland neoplasms . Most caKimura disease (KD) is a rare chronic inflammatory disorder that occurs predominantly in young Asian males, with sporadic cases reported in other regions . It presCoexistence of two different entities in a patient is an uncommon phenomenon. However, the presence of other non-salivary gland neoplasms with the very common WT has been reported to be as high as 37%, with head and neck squamous cell carcinoma being the most common neoplasm . SynchroA 64-year-old Arabic man presented with a left painless swelling of the face of a 10-year duration. The patient reported a recent increase in the size of the swelling. His medical history was significant for hypertension and cigarette smoking. Head and neck examination revealed a firm, non-tender parotid mass, measuring 10 cm in the greatest dimension. There was limited mobility of the mass, and the overlying skin exhibited no changes. There were no lymph nodes or facial nerve abnormalities. The computed tomography scan showed a 7.1 × 6.2 × 5.5 cm well-defined mass in the left parotid with heterogeneous enhancement and multiple cystic spaces. The mass displaced the sternocleidomastoid muscle posteriorly and medically but showed no infiltration . A complete blood count with differential and serum IgE level were performed and showed a normal absolute eosinophil count of 0.41 × 10Benign salivary gland tumors account for 73–86% of all parotid neoplasms, and WT is the second most common tumor 3–35%) after pleomorphic adenoma (73–80%) –14. WT, % after pet al. [Synchronous salivary gland neoplasms in the parotid gland are uncommon and account for only 3.6% of all parotid gland neoplasms , 22. Theet al.  reportedet al. . The patet al. .et al. [et al. [KD is a rare immune-mediated, inflammatory disorder with unclear etiology. It was initially reported in the Chinese population in 1937 by Kimm et al.  and in a [et al. . It pres [et al. . The mai [et al. .LCH, Hodgkin lymphoma, and angiolymphoid hyperplasia with eosinophilia (ALHA) were considered in the differential diagnosis in the present case. Immunohistochemical staining of CD1a and S100 failed to detect Langerhans cells. The clinical presentation and the diffuse lymphocytic infiltrate histologically raised the possibility of Hodgkin lymphoma; however, no typical Reed–Sternberg cells were present in the sections examined. Lastly, ALHA was previously confused with KD and used interchangeably in the old literature. ALHA is currently considered a vascular neoplasm with known FOS and FOSB gene rearrangements . HistoloThis is a report of concurrent WT and KD, a rare and novel presentation of parotid tumors that occurred as a single nodule with a recent increase in size. Interestingly, the surgeon noted a firmer consistency than a typical WT during parotidectomy. Although the management is similar for both entities, it is necessary to identify both components, since KD may require additional treatment and follow-up. In addition, the relationship between WT and KD is unclear and warrants further investigation.


Melanotic schwannoma is a rare tumor when it occurs in the sacrum. Though it is mostly classified as benign, the prognosis is unpredictable due to the possibility of recurrence and metastasis. Here, we reported a case of intraosseous of sacrum with good results and reviewed the literature.A 33‐year‐old male patient complained of low back pain and was discovered to have an obstruction at S2. Following the necessary imaging diagnosis, we treated the patient with piecemeal excision in conjunction with extended curettage, and the frozen biopsy revealed that the tumor was melanotic schwannoma. The intraosseous portion of the lesion was curettaged using high‐speed drill to enlarge the edge of curettage, and piecemeal excision for lesion within the sacral canal. After surgery, the patient received total 56Gy radiotherapy and frequent follow‐up. After 15 months follow‐up, there was no evidence of recurrence, and the nerve function was normal.Melanotic schwannoma that occurs intraosseous of the sacrum is extremely rare and lacks typical clinical manifestations; however it can be identified through careful pathological and imaging diagnosis. Intralesional extended curettage combined with radiotherapy can achieve a good local control with a satisfactory clinical effect in this rare disease. We discussed our successful management of this challenging tumor with a focus on preoperative diagnosis, surgical approach, and histological consideration to provide guidance for future clinical management in melanotic schwannoma. Melanotic schwannoma (MS) is a rare variant of schwannoma with capability of melanogenesis, which was first identified by Millar in 1932, accounting for only 1% of schwannoma.A 33‐year‐old male patient presented to our clinic with 2 months of low back pain and a half‐month of right thigh numbness. The patient reported that both pain and numbness were activity related. When he presented, he did not have intermittent claudication. A CT scan was taken at an outside hospital, which revealed an obstruction at S2. The physical examination showed normal sensation and tenderness in his bilateral legs. At admission, the Lasègue test was conducted, and a negative result was obtained. Manual muscle testing is also employed and gastrocnemius muscular strength for both legs was scored at 5. Laboratory tests revealed that the patients' common blood cell count, C‐reactive protein level, erythrocyte sedimentation rate, and tumor markers were all within normal limits.The CT scan showed an osteolytic lesion with local ossification at S2 level; the osteolytic lesion was located at the posterior of the vertebral column, and the calcified portion nearly reached the anterior of the vertebral column Figure .A magnetic resonance imaging scan of the sacral spine  staining and immunological marker reexamination. Melanotic schwannoma was confirmed by specialized pathologists. Under the light microscopy, the tumor cells were spindle‐shaped, epithelioid, and arranged in fascicles, palisades, or nests under the light microscope. The nuclei were relatively uniform in size, and the nucleoli were not visible. Mitotic figures were uncommon, but abundance of melanin was observed. There was no obvious necrosis and only cytoplasm was found. Typical schwannoma tissue conformation can still be seen in some areas. The tumor cells were immunohistochemically positive for homatropine methylbromide HMB‐45), S‐100 protein, partially positive SOX‐10, and negative for epithelial membrane antigen. The labeling index for Ki‐67 was approximately 5%–10%  is a remarkable tool in assessing the biological and histological characteristics as well as malignant capacity of the tumors prior to surgery, and therefore, is validated in assisting the prediction of patient prognosis.The biopsy was a useful method for differentiating MSs from other primary tumors. However, there is a risk of complications such as infection, wound hemorrhage, nerve injury, and other related problems after the procedure. As a result, we did not plan the core needle biopsy for this patient, and instead performed an intralesional curettage surgery. During the surgery, we discovered that S2 nerve root was the source of the tumor, and the sacrum was infiltrated by it. The border was not as clear as it appeared on CT, and there appeared to be a black spot in cancellous bone, so we used high speed drill to conduct extended curettage to remove the intraosseous portion, and piecemeal excision for lesion within the sacral canal.The MSs are distinguished by the accumulation of melanin in neoplastic cells and the formation of the cytoplasmic melanosome. Most MSs are well‐defined or encapsulated in a round or oval shape with cut section being black, brown, or gray. They typically range in diameter from 0.5 to 26 cm, with the majority being larger than 5 cm. Histologically, MSs can be classified into two types: conventional melanotic schwannoma (CMS) and psammomatous melanotic schwannoma (PMS). CMSs are typically composed of spindle‐shaped and epithelioid cells arranged in bundles or interlaced fascicular pattern, with nuclear palisading. Verocay bodies are uncommon in CMS, but pigmented granules can be seen in cytoplasm of most tumor cells. The nucleus can sometimes be obscured by excessive pigmentation. For PMSs, there are some stratified calcified globules, most of which are focal lesions with a round or oval shape. In approximately 60% of PMSs cases, intracytoplasmic vacuoles can be seen within the tumor cell, mimicking mature adipose tissue if there are only few of them, and tumor reported in this case fall into PMS category.This case should be carefully differentiated from the following three types of tumors: pigmented neurofibroma (TPN), metastatic malignant melanoma (MMM), and soft tissue clear cell sarcoma (STCCS). TPN often occurs in the dermis or subcutaneous tissue of the head or neck, or lower extremity such as buttocks or calves. With an undefined margin, the malignancy typically spreads along connective tissue and fat lobular septa and can surround skin appendages, etc. Small‐ to medium‐sized spindle or oval‐shaped tumor cells are sparsely arranged. The interstitium is composed of uniform collagen fibers that proliferate to form carrot‐like collagen bundles. Pigment cells are displayed within the cells in a dendritic manner. Angiohyalinosis is uncommon in neurofibroma, and S‐100 positive reaction are also less commonly seen in neurofibroma in contrast to schwannoma. MMM is closely related to the skin as nearby skin is the most common site for its regional metastasis. Patients with this malignancy are typically middle‐aged, generally over 40 years old and with a history of malignant melanoma. The pathology of MMM is characterized by obvious pleomorphism and cytologic atypia as well as large eosinophilic nucleolus, nuclear groove, intranuclear inclusions, and prominent mitotic figures. Unlike TPN and MMM, STCCS are most commonly seen in the distal end of extremity, particularly the feet and ankles, followed by the knees, thighs, hands, and forearms, and, on rare occasions, the torso, head and neck, intestines, and kidneys. The location of STCCS is deep‐seated, often connecting with tendons and aponeuroses, and can invade throughout subcutaneous tissues. The tumor cells are densely arranged in bundles, nests, or sheets, separated by thin or dense fibrous connective tissue. Often polygonal, oval, or spindle‐shaped, they are prominent by clear cytoplasm, round or oval‐shaped nuclei and eosinophilic nucleoli. Mitotic figures were not common and multinucleated giant cells can be seen occasionally. Immunohistochemical staining for S‐100, HMB‐45, Melan‐A, and NSE‐7 showed positive results in STCCS cells. However, in this case, the specimen shows spindle cells arranged in sheets and short fascicles, with heavy deposition of melanin pigment that largely obscures the nuclear details of most cells. Moreover, S‐100 and SOX‐10 immunostains are strongly and diffusely positive. By combining the aforementioned characteristics, our case can be differentiated from TPN, MMM, and STCCS.et al.via posterior approach alone; unlike type I tumor, type II sacral tumors originate in the sacrum but metastasize towards the presacral and subcutaneous region, and consequently combined anterior–posterior approach is necessary; type III tumor is located in the presacral or retroperitoneal alone, which an anterior approach is appropriate. Though some authors reported satisfying results without local recurrence can be obtained via piecemeal surgery with sacral nerve roots being preserved,et al.via posterior approach. The patient was regularly followed up once every 3 months and has shown no signs of recurrence or metastasis up to this point.Surgery is the first‐line treatment option, and a complete resection of tumor with clear margins is recommended. In 2003, Klimo et al. reported in one case of retrocaval MS that the use of pembrolizumab can lead to significant improvement in pain control and tumor stability, they emphasized the necessity to assess its applicability to general MS.et al.et al. revealed a 35% local recurrence and a 44% metastasis rate in MS patients, indicating a significantly more aggressive nature. Since MS patients tend to display late local recurrence and metastases, long‐term follow‐up is necessary despite of the absence of malignant histological characteristics.Adjuvant therapy, such as chemotherapy or radiotherapy, is not well‐defined yet, but for cases with aggressive histology , adjuvant therapy is recommended.via CT and MRI are advisable, and a long‐term follow‐up and observation of the patient is also required before a more definitive statement can be made concerning the prognosis of this rare case.Melanotic schwannoma is a rare variant of schwannoma with aggressive and malignant nature. Due to its rarity, MS is difficult to diagnose before surgery, and it is necessary for intra‐operative histological analysis to guide the extent of resection. So far, very few cases of intraosseous sacral MS have been reported. In this case, intralesional resection combined with radiotherapy not only avoids unnecessary neurological deficits but also achieves a good local control with a satisfactory clinical effect, which provide an alternative surgical management for MS. From our point of view, early and regular follow‐up All authors contributed to the study conceptualization and design. Material preparation and data collections were performed by Xiaobo Yan, Nong Lin, and Xin Huang. Data integration was performed by Keyi Wang. Pathology diagnosis was conducted by Yanbiao Fu. The first draft of the manuscript was written by XBY and approved by Zhaoming Ye. Keyi Wang was responsible for review and editing. All authors commented on previous versions of the manuscript and all authors read and approved the final manuscript.The authors declare that they have no competing interests.This study was approved by the Human Research Ethics Committee of the Second Affliated hospital of Zhejiang University School of Medicine. 2022‐0031. The written informed consent to participate was obtained from the patient.


Positron emission tomography (PET) combined with a magnetic resonance (MR) scanner (PET/MR) with 18F-fluorodeoxyglucose (FDG) tracer is being used in quite a few nuclear medicine centers. The aim of this study is to illustrate two uncommon cases of primary inferior vena cava leiomyosarcoma which were formerly evaluated with anatomical images such as computed tomography and ultrasound. These techniques were inferior in the definition of the tumor and its characteristics. F-18 FDG PET/MR was essential and provided all the necessary information: its origin, local extension, anatomo-metabolic behavior, form of presentation, and distant metastasis in one single diagnostic technique. PET/MR accurately contributed to the diagnosis in a shortened period of time and, therefore, in the prognosis of this disease with greater benefits. Inferior cava vein leiomyosarcomas (LMS) are an unusual entity, rising from smooth muscle cells with low growth rates. These features show the low recognition of this pathology, mainly due to its infrequency. Our medical staff was motivated to perform more complex studies available in our region, in order to improve localization accuracy of the suspected neoplasm, proving a precise diagnosis of an uncommon disease, later confirmed by histological studies.Positron emission tomography (PET) combined with a magnetic resonance (MR) scanner (PET/MR) imaging plays a pivotal role in the abdominal evaluation and characterization of retroperitoneal tumor lesions and extension to neighboring organs. The shortage of literature and case reports promoted a bibliography discussion and the presentation of the cases.The first patient, a 50-year-old woman, starts with prolonged abdominal pain and distension. An abdominal ultrasound is performed which shows an expansive hypoechoic, heterogeneous mass in the retroperitoneum. Consequently, a non-contrast computed tomography (CT) was performed to characterize the lesion. It exhibited a hypodense mass which displaced neighboring organs with no clear diagnosis.A retroperitoneal neoplasm is suspected, specifically a sarcoma; thus, since our center has a PET/MR, it was executed Fig. . PET/MR Our second patient is a 64-year-old male with a history of drug addiction, weight loss and abdominal pain. Firstly, a CT scan showed moderate left pleural effusion, with a voluminous expansive lesion of 12.3 cm in contact with the upper pole of the right kidney, and with compressive effects on the liver parenchyma. After a suitable evaluation in which renal or adrenal origin was considered, a PET/MR scan was necessary to distinguish the tumor properly and its extension , in a full-body PET 3D TOF acquisition scan in Hybrid Resonator, PET/MR General Electric SIGNA 3 Tesla. Pathological studies with immunostaining showed high-grade spindle cell sarcoma in both patients. Consequently, they both commence chemotherapy treatment. The former patient additionally underwent radiation treatment with positive outcomes with regular follow-up imaging to monitor for recurrences. Sadly, the second patient died with no further image evaluation.Inferior cava vein (IVC) leiomyosarcomas (LMS) are an unusual entity, rising from smooth muscle cells of the media, although it is the most frequent primary tumor from IVC. In general, LMS of the IVC account for 5% of all vascular LMS. Vascular LMS tumors represent about 1–2% of all LMS, which accounts for 10–20% of all sarcomas in correlation with the low proportion of cases described in the literature. Sarcoma represents 1% of all adult malignancies , intraluminal or mixed patterns. They could also be divided into an anatomical classification formerly described by Mingoli Fig. . The firThe slow rate of sarcomas and the type of growth reflect the variety of symptoms and the late clinical presentation: from asymptomatic to vague abdominal pain, distension, palpable mass, obstructive syndrome, Budd–Chiari syndrome, edema, hypertension and tumor thrombosis  is extremely valuable  and the plus points of metabolic scan (PET), unified in a single examination, to provide a more accurate diagnostic approach.We consider that the positive outcomes of this review will motivate the use of PET/MR in vascular sarcomas neoplasm in more centers worldwide, which may contribute to a deeper understanding of this rare neoplasm.


A man in his mid-50s presented with heaviness in the right lower abdomen for the last three months. Ultrasonography (USG) showed an intra-abdominal mass in the right iliac fossa. Contrast-enhanced CT (CECT) revealed a well-defined circumscribed mass near the ileocaecal junction, with a hypodense center, surrounded by a hyperdense periphery and well-defined capsule. A lower midline laparotomy was performed. Intraoperatively, a white, firm, smooth ball-like mass was found, lying freely in the abdomen. Histopathology revealed fatty tissues surrounded by a calcified shell, which was further surrounded by proteinaceous material. Such peritoneal loose bodies (PLBs) are free masses in the abdomen, with variable sizes. These are asymptomatic, unless they compress any nearby viscera, as was in our case. The objective of this case report is to make surgeons and radiologists aware of this rare entity, which can be a source of confusion in a case of mass in the abdomen. A peritoneal loose body (PLB) is a rare incidental finding that may lead to diagnostic confusion and undue anxiety due to imaging resemblance to gastrointestinal mesenchymal tumors. This entity is benign and remains largely asymptomatic. Its size may vary, ranging from a few mm to more than 10 cm [A 55-year-old male who had been complaining of right lower abdominal heaviness for previous three months presented to the surgery outdoor. There were no specific systemic complaints. Clinical examination was normal and no lump could be palpated in the abdomen.Initial ultrasonography (USG) of the abdomen revealed an intra-abdominal hypoechoic rounded mass of size about 7 x 6 cm in the right iliac fossa. Contrast-enhanced CT (CECT) scan of the abdomen, performed a month back, showed a well-defined circumscribed mass of size about 7 x 7 x 6.1 cm near the ileocaecal junction, with a hypodense center, surrounded by a hyperdense periphery and a well-defined capsule  Leiomyoma uterus: On USG, uncomplicated leiomyomas are usually hypoechoic but can be isoechoic or even hyperechoic compared to normal myometrium. Calcification is seen as an echogenic focus with shadowing. Cystic areas of necrosis or degeneration may be seen.2) Leiomyoma recto-sigmoid (if lying in the pelvis): On CT scan, it is seen as smooth-surfaced mass with loss of fat planes with recto-sigmoid showing varying degrees of internal necrosis. Moreover, heterogeneous contrast enhancement and dystrophic calcification may also be seen.3) Gastrointestinal stromal tumor (GIST) of the intestine: Small primary tumors are frequently well marginated and, on unenhanced CT, are seen to have low soft tissue density. On enhanced CT, these tumors, when small, typically show homogeneous enhancement.4) Foreign body: On CT scan, they show up with irregular margins with adhesion to surrounding structures or scars from previous surgery.A PLB can present a difficult diagnostic problem for the surgeon, with confusing findings that may indicate malignancy. This case report aims to alert clinicians about this rare finding of PLBs, which are benign but could be easily confused with well-circumscribed tumors like leiomyoma or GIST. Preoperative diagnosis of these lesions is difficult and a high index of suspicion should be kept in any symptomatic patient with a mobile lesion in the abdomen or a calcified lesion in the pelvis on X-ray. No specific treatment is required in asymptomatic patients. However, if these entities become associated with complications like intestinal obstruction, if there is an abdominal mass of obscure origin, or when the diagnosis is in doubt, then exploration is required. If the size is large and is causing pressure symptoms, it is often removed to confirm the diagnosis. Laparoscopic removal is also feasible. It is also equally important to allay the anxiety of patients.


Myelolipoma is a rare, benign, non-secreting tumor and its pathophysiology is of metaplasia of the cells of the adrenal cortex into reticuloendothelial cells. Although they are often small and asymptomatic, some cases of giant adrenal myelolipoma cause symptoms such as chronic pain. Few cases of adrenal myelolipoma have been reported in the literature. We present a case of a large right adrenal myelolipoma in a 26-year-old female patient, who presented with an adrenal mass, and discuss the challenges of diagnosis and treatment. Adrenal myelolipoma is a rare benign, nonfunctional lesion that is composed of macroscopic fat and mature hematopoietic tissue, resembling bone marrow . A few cN: 0.00–0.10); norepinephrine, 0.28 ng/ml (N: 0.10–0.50). Furthermore, detailed 24-h urinalyses during hospitalization showed normal urinary catecholamine levels. The rest of the endocrine exploration was normal. The patient was taken up for exploratory laparotomy. Intraoperatively, a giant solid mass was found arising from the right adrenal gland and was adherent the upper pole of the kidney, duodenum and liver. The patient underwent excision of this adrenal mass. Macroscopically, the mass measured 12 × 8 × 10 cm and was round, well circumscribed and encapsulated. Histologically, the tumor was confirmed as a myelolipoma, and no malignant foci were detected  peripherally and −30 HU in the center . Laboratdetected . The patAdrenal myelolipoma is a rare benign tumor. Its pathophysiology is thought to be metaplasia of adrenal cortical cells into reticuloendothelial cells in response to infection, chronic stress or degeneration of the adrenal gland , 2. The In our case, the patient was symptomatic of pain and the dimensions of the adrenal myelolipoma were >4 cm. In addition, the patient presented clinical signs of a hormone-secreting tumor , although the hormonal exploration was normal. This patient’s clinical signs and symptoms along with the CT imaging warranted concern for malignancy such as liposarcoma. This was the main indication for surgical treatment.The treatment of myelolipoma is not clearly defined. Some studies suggest that patients with small lesions should not be treated . HoweverIn conclusion, adrenal myelolipoma is a rare entity and is mostly asymptomatic. The diagnostic strategy is the same as for adrenal incidentalomas. The CT scan allows the diagnosis in the majority of cases. The surgical removal of myelolipoma is indicated when it presents a size >7 cm, with compressive symptoms or presenting with bleeding complications.None declared.None.


The treatment choice in the management of odontogenic cysts in the mixed dentition period depends upon the size, location of the cyst,the bone integrity of the cystic wall, and its proximity to vital structures. Enucleation is indicated with smaller cysts, achieved by careful removal of acomplete cyst without rupturing the cystic lining. Marsupializationand decompression are the treatments of choice for larger cysts as it can help to preserve thetooth bud of the successor tooth and reduce morbidity. Marsupialization is achieved by opening and deroofing the cyst, and making the cystic lining continuous withthe oral cavity or surrounding structures by suturing the edges of the incised mucosa to the cystic wall. This helps in maintaining the patency of the cystic lesion.In the decompression, a cylindrical device (drain) is placed in the lesion, which maintains communication between oral cavity to cystic lesion. This decreases theintracystic pressure and results in bone formation. We present two cases of odontogenic cyst in children, where we used a modified decompression technique. We developeda modified surgical stent with the use of a Hawley’s appliance, which led to cystic decompression, and eventual eruption of the successor tooth. Notably, this modifiedtechnique also reduced both patient discomfort and the number of clinical visits, making it an effective treatment option. The unique design of the appliance also acted as a space maintainer for the eruption of successor tooth, which is very critical in mixed dentition for future prevention of space loss and eventual malocclusion.The advantage of our design was its easy removal and insertion with minimal discomfort. Enucleation, marsupialization, and decompression are the treatments of choice for the management of odontogenic cysts. Enucleation consists of the careful removal of a complete cyst without rupturing the cystic lining, particularly indicated with smaller cysts. With larger cysts, however, enucleation can lead to mandible fracture, devitalisation of the adjacent teeth, and amputation of the successor tooth buds suggesting marsupialization and decompression are the more viable options  . MarsupThe purpose of this article is to report two cases of odontogenic cyst in the mixed dentition period, treated with a modified decompressiontechnique , which iA 7-year-old boy reported to the Department of Pediatric Dentistry with the primary complaint of pain and swelling in the upper left canine region over the last month.The patient experienced moderate pain during mastication lasting for 20-30 minutes after the onset. Upon clinical examination, diffuse extra-oral swelling was observed inthe infraorbital region extending from the lower border of eyelid to the ala tragus line and from the corner of nose to the outer canthus level of the left eye. On palpation,the swelling was bony hard and tender. An intraoral examination revealed a cortical plate expansion from the maxillary left permanent incisor to primary first molar. Theroot piece of the deciduous left canine, grossly carious first and second left primary molars were noted. A panoramic radiograph revealed a well- defined periapicalradiolucency measuring about 1.5cm×2cm around the periapical region of primary left canine with the successor tooth bud placed at a higher level than the contralateralpermanent canine tooth bud . Fine neAn 11-year-old boy visited the Department of Pediatric Dentistry with primary chief complaint of asymptomatic swelling in the upper left canine region over a3-monthperiod. Upon clinical examination, diffuse extra oral swelling was observed in the left infraorbital region extending from the lower border of eyelid tothe ala tragus line and from the corner of the nose to the outer canthus level of the left eye. Upon palpation, the swelling was bony hard and an intraoral examinationrevealed a buccal and palatal cortical plate expansion from the maxillary left permanent lateral incisor to the left primary second molar. The teeth in this regiondid not exhibit any sign of dental injury. A panoramic radiograph revealed a well- defined unilocular radiolucency measuring 2cm×2cm, which extended from thelateral border of the left lateral incisor to the mesial border of the first left premolar tooth bud and surrounded the root of primary left maxillary canine.The succedaneous tooth germ of permanent canine was located higher compared to its counterpart on the right side .DecomprThe decision of utilizing modified surgical stent was based on positive outcome of case study 1 as well as parental reluctance for regular visits to hospital.As described in case study 1, Hawley’s appliance was constructed from an alginate impression of the upper arch. A nasotracheal tube (size-3) was passed through the hole ofthe plate and the extraction socket into the cystic lesion until resistance was felt. The tube was then attached to the Hawley’s appliance with cold cure acrylic.The panoramic radiograph confirmed its position and extent in situ. An acrylic stopper was prepared to seal the tube during food consumption. Patient’s parents were taughtto irrigate the lesion regularly through the tube with copious amount of Betadine solution. The length of the tube was frequently trimmed as the permanent canine eruptedover two-and-a-half-year period. This uneventful eruption of the successor canine in its proper location again confirmed the usefulness of our appliance in management ofodontogenic cysts in mixed dentition . A folloThe treatment choice in the management of odontogenic cysts in the mixed dentition period depends upon the size and location of the cyst, the bone integrity of the cystic wall and its proximity to vital structures. Masuda et al.   reporteIn our cases, there was a close proximity of the tooth bud to cystic lining. Enucleation would have damaged the underlying tooth bud. Hence, we preferred to undertake decompression. The surgical procedure in our patients included the extraction of the involved deciduous tooth, the collection of incised pathological tissue for microscopic examination, and the establishment of an access point through the extracted socket into the body of cystic lesion. In both described cases, good bone healing with a normal eruption of the displaced permanent tooth was subsequently observed. The main purpose behind marsupialization /decompression is to make an opening or create an adequate window on the outer wall of cyst through which the cystic content is evacuated. This leads to the gradual ossification and size reduction of the cyst to an extent that the cystic lining becomes continuous with the oral epithelium  , 7. ThHowever, the main drawback of decompression in children is patient compliance as it involves frequent clinical visits and prolonged treatment till the eruption of successor. Principally, the patency of the cystic lumen is maintained either by tightly packing sterilized gauze or by means of silicon drain or decompression tube, which requires frequent dressing changes and cystic cavity irrigation by clinician  - 12. DThe authors certify that they have obtained all appropriate patient consent forms. In the form, the patients have given their consent for their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.Modified surgical stent is an effective and conservative approach of marsupialisation for large odontogenic cysts in mixed dentition. This appliance reduces the frequent clinical visits, maintains space for permanent dentition in children. There are no conflicts of interest.


KRAS mutations. It has been estimated to account 4% of previously diagnosed PRN.Papillary Renal Neoplasm (PRN) with polarity inversion is a less common subtype of kidney cancer with an apparently recognizable morphology, distinct immunohistochemical profiles, and frequent This is a retrospective case report of two patients diagnosed with PRNRP. Two males were found to have kidney mass accidentally through imaging examination in clinic. Both of the patients had no obvious discomfort and abnormal test indicators. Subsequently, they underwent partial nephrectomy in our center by the same surgeon and followed up closely with an impressive clinical outcome. The pathology reports indicated that their pathological features were consistent with PRNRP. The HE staining showed a monolayer of papillary or tubular structures, with small nuclei away from the cytoplasmic top of the basement membrane. The immunohistochemical results were GATA3 (+), vimentin (-).Our case reports and literature review suggested that PRNRP should be separated from traditional PRN and partial nephrectomy is a robust modality for PRNRP. The morphological, immunohistochemical, and genetic information of the cases we presented would provide important material for PRNRP to become a distinct category with benign clinical outcome. Papillary renal neoplasm (PRN) is the second most common subtype of kidney cancer, which takes up 15%-20% of renal cell carcinoma (RCC) –5. In 20The patient is a 54-year-old male, who was admitted to the hospital due to the discovery of a left kidney tumor by ultrasound. The patient had no complains of clinical symptoms, no fever, low back pain, or hematuria. He was a heavy smoker (30 pack/year). Urine culture and urine cytology of that patents demonstrated normal results. The results of blood routine, liver and kidney function, coagulation test indexes also showed no obvious abnormality. Ultrasonography of the urinary system demonstrated mixed echoes in the lower pole of the left kidney, which measured 4×3 cm in size, with a clear boundary, thick septa. A 1.1×1.0 cm regular shape, medium-high echo was detected on the wall. Enhanced CT showed a slightly high-density nodule at the lower pole of the left kidney, with a diameter of about 2.5 cm and a CT value of 57 HU on plain scan with no obvious strengthening , the lesThe postoperative pathology revealed PRNRP . The tumor incision is grayish red with medium texture and fine papillary shape. Tumor was confined to the kidney, with no extension into the perinephric adipose tissue, renal sinus, renal pelvis, or Gerota fascia. Immunohistochemical phenotypes were as follow: GATA3 (+), Keratin 34βe12 , AE1/AE3 (+), Keratin 7 (+), EMA (+), vimentin (-), PAX-8(-), CD10(-), CD117(-), CA9(-), TFE3(-), RCC(-), P504(-). After one year of follow-up, the patient recovered well without disease progression or recurrence.The patient was a 35-year-old male, who admitted to the hospital due to physical examination of a right kidney mass for 1 week. The patient had no clinical manifestations, no fever, low back pain, or hematuria. The findings of laboratory tests, including complete blood count, liver and renal function tests, and urine analysis, were within normal range. The patient was previously healthy with no family history of tumor.Ultrasound of the urinary system demonstrated an anechoic, regular mass at the middle and lower part of the right kidney measuring 1.9×1.6×1.3 cm in size, with clear boundary. Enhanced CT showed a 1.6×1.3 cm round high-density nodule in the right kidney with a CT value of about 80HU on plain scan. No enhancement after intravenous administration of contrast was noted. The mass was classified as Bosniak type III , AE1/AE3(+), Keratin 7(+), EMA(+), vimentin(-), PAX-8(-), CD10(-), CD117(-), CA9(-), TFE3(-), RCC(-), P504(-). The patient did not report any specific discomfort and disease progression at his one year follow-up.Both the pathological manifestations of the tumors from the aforementioned two patients were consistent with typical PRNRP. The HE staining showed papillary or tubular structures, the surface was covered with a monolayer of eosinophils, the cytoplasm was granular, with small nuclei away from the cytoplasmic top of the basement membrane. The immunohistochemical phenotype was GATA3 (+), vimentin (-) Figure 2CDKN2A gene silencing, SETD2 gene mutation, TFE3 gene fusion, or increased expression of NRF2–antioxidant response element signaling pathway (in situ hybridization (FISH) analysis demonstrated a low level of chromosomal abnormalities  of PRN have KRAS gene mutations; no KRAS gene mutation was detected in chromophobe RCC  and reduce the activation of growth factor-dependent AKT (growth factor-dependent Akt) thus leads to a worse prognosis than patients with other mutations; whereas p. Gly12Asp mutation plays critical a role by increasing the activation of PI3K (phosphatidylinosi-tol 3-kinase) and MEK . In PRNRP, the common KRAS gene mutation sites are p.Gly12Val, p. Gly12Asp and p. Gly12Arg, accounting for 61.5%, 34.6% and 3.9%, respectively  for the publication of any potentially identifiable images or data included in this article.LZ: substantial contributions to acquisition of data and involved in drafting. ZY, YW, WX, PB, and XY: equal contributions to acquisition of data. XY: substantial contributions to data acquisition, involved in drafting. All authors contributed to the article and approved the submitted version.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.All claims expressed in this article are solely those of the authors and do not necessarily represent those of their affiliated organizations, or those of the publisher, the editors and the reviewers. Any product that may be evaluated in this article, or claim that may be made by its manufacturer, is not guaranteed or endorsed by the publisher.


Urocolpos is a urine collection in the vagina, possibly due to vesicovaginal reflux (VVR) or a fistula. Urocolpos is a rare phenomenon in adults, and it is commonly underrecognized by radiologists. The occurrence of urocolpos due to VVR after a Caesarean section in adult females is a rare phenomenon; failure to recognize it may lead to misdiagnosis as vesicovaginal fistula and often leads to unnecessary medical interventions. We report a 37-year-old female who complained of abdominal pain and intermittent daytime urinary incontinence for six months, starting after a Caesarean section for her third baby. An abdominal computed tomography scan revealed a large fluid-filled vagina on a full bladder without anatomical abnormality. A post-voiding scan showed a complete resolution. In conclusion, urocolpos is an underrecognized entity that rarely coexists with VVR. Post-voiding resolution and vaginal contrast reflux are important findings that can aid radiologists in identifying such cases. Urocolpos is a urine collection in the vagina, possibly due to vesicovaginal reflux (VVR) or a fistula in adults . It is cThe occurrence of hydrocolpos due to VVR after a Caesarean section in adult females is very rare and may be misdiagnosed as a vesicovaginal fistula ,5. VVR iPatient information: a 37-year-old married female presented with a complaint of abdominal pain for six months. The patient had a history of intermittent daytime urinary incontinence immediately after voiding with no specific relation to increased intrabdominal pressure , urgency, and UTI that started after the Caesarean section for her third baby. She denied any history of dysuria, vaginal discharge, or hematuria. The menstrual history was unremarkable, without a history of any chronic medical condition or malignancy. The patient denied any similar problem in a previous Caesarean section.Clinical findings: the patient was overweight : 29.3 kg/m2). Examination of the abdomen and external and internal genitalia was unremarkable.Diagnostic assessment: blood counts and renal function tests were within normal limits. Urinalysis revealed only a few pus cells (10 WBCs/HPF). Urine culture revealed Escherichia coli sensitive to ciprofloxacin, levofloxacin, and cefixime. Transabdominal ultrasonography (USS) revealed an anechoic mass-like lesion located posterior to the urinary bladder with an associated anterior displacement and posterior acoustic enhancement. These findings were suggestive of a fluid-filled vagina (urocolpos). The adjacent abdominal structures were of normal appearance. The mass was completely unvisualized in the post-void USS image, implying a VVR diagnosis. Further evaluation with an abdominopelvic CT scan revealed a distended fluid-filled vagina on full bladder imaging suggestive of urocolpos  cases, with a mean age of 47 years, Haouas et al. and Prashant et al. [Urocolpos is a urine accumulation in vagina that causes cystic dilatation and distension. Obstructive vaginal lesions and urogenital malformations are frequently associated with this condition. Obstructive lesions appear early in life, but VVR typically occurs in prepubertal girls with a prevalence rate of 12-15% and is anticipated to become nearly absent in middle age, which is rationally described by gradual bladder descent and anatomical modification of the vesicourethral angle ,6,7. Thet et al. ,8,9. Simt et al. ,10. Our The probable mechanism of pelvic floor dysfunction among obese women is due to the effect of hypoestrogenism. Additionally, obesity is associated with increased intra-abdominal pressure that may lead to pelvic floor muscle weakness . These pThe most common diagnostic radiologic modalities for VVR are the USS, CT scan, pelvic magnetic resonance imaging (MRI), and voiding cystourethrography (VCUG) . In the Obstructive urocolpos and vesicovaginal fistula are associated with anatomical abnormalities that require surgical intervention. Therefore, it should be differentiated from non-obstructive urocolpos that are managed typically through behavioral therapy ,10. VariUrocolpos is an underrecognized entity that rarely coexists with VVR. Post-voiding resolution and vaginal contrast reflux are important findings that can aid the radiologist in identifying such cases to limit unnecessary medical and radiologic investigations, which provoke patient anxiety.


Central Giant Cell Granuloma constitutes approximately 7% of benign tumors of the jaws. The aggressive form of CGCG clinically behaves like a classic semi-malignant neoplasm. In the literature, the suggested method of treatment of aggressive forms of CGCG is curettage or resection with the margin of 0.5 cm. Surgical treatment, especially in the developmental age, entails disturbances in the growth and differentiation of tissues and deforms and disturbs the functioning of the stomatognathic system. Alternative treatment methods of the CGCG presented in this article lead to the patient avoiding a mutilating procedure and improve their quality of life. The aim was to present alternative method of treatment of aggressive forms of Central Giant Cell Lesion of the jaws—injections of dexamethasone into the tumor mass through drilled bony canals. Here, we present the three cases of aggressive forms of CGCG of jaws treated with dexamethasone injections into the tumor mass. Two cases resulted in regression of the tumor, which was confirmed in histologic evaluation after remodeling surgery. Those two patients were uneventful and showed no signs of tumor recurrence at 8 and 9 years of thorough follow-up, respectively. The third patient was qualified for the mandible resection due to the enlargement of the lesion and destruction of the cortical bone. According to our observations, if the proper patient discipline, and thorough, careful clinical and radiological examinations are provided, the dexamethasone injections could be a recommended method of treatment of intraosseous giant cell granuloma. The indication is restricted to the cases with preserved bony borders despite deformation. Additionally, leaving vital teeth in the lesion is also possible. The endosseous giant cell lesion, Central Giant Cell Granuloma (CGCG), is a benign lesion developing as a bone tissue response to an injury, inflammation or as a reparative bone reaction ,2. CGCG The clinical features of the tumor vary. Two forms of CGCG can be distinguished. The non-aggressive form (60–80%) is characterized by slow growth, low severity of symptoms, no cortical bone destruction and root resorption, and low likelihood of recurrence ,11,12,13Therefore, blood test results should be considered in the differential diagnosis to rule out a brown tumor. An increased level of calcium, alkaline phosphatase and parathormone in the serum or a decreased level of phosphorus indicate hyperparathyroidism. Radiological imaging should be differentiated from single bone cysts, odontogenic cysts and granulomas (non-aggressive form), aneurysmal cyst, solid ameloblastoma and odontogenic myxoma (the aggressive form) ,27,28.According to the literature, the treatment of CGCG depends on the location, extent and aggressiveness of the lesion and patient’s age ,21,26. TFor this reason, alternative methods for CGCG treatment are being sought . However, isolated scientific reports of Body et al. in 1981  and TerrAnother pharmacological treatment method for non-aggressive forms of CGCG recommended by some authors include the use of calcitonin intranasally or by subcutaneous injection ,34,35,36Other authors have suggested the use of interferon alpha-2a in the treatment of CGCG. However, due to its significant side effects, it should only be used in cases where other methods have failed or as a complement to surgical treatment ,34,35.In the available literature, no reports of non-surgical treatment of CGCG aggressive forms in developmental age were detected.The Department of Maxillofacial Surgery treated a large number of CGCG cases between 2011 and 2022, with each case being treated individually.It seems important to develop a new strategy for treating the aggressive form of CGCG. Treatment of those lesions, according to current guidelines requiring resection surgery, involves significant trauma to the patient, the loss of many teeth, and the need for a complex reconstructive procedure, even a free fibula flap surgery. This, especially for young patients, during the growth period, can significantly complicate their facial development, including at the recipient site.The purpose of this study was to clinically, radiologically and histopathologically evaluate the efficacy of a novel method of intraoperative administration of dexamethasone through bony canals to the tumor mass as a sole and definitive treatment option in cases of extensive, aggressive giant cell tumors with long-term follow-up.The study included three patients with an extensive aggressive form of CGCG confirmed by histopathological examination. Patients were qualified for a nonsurgical treatment—dexamethasone injection into tumor mass—based on the history, clinical and radiological examinations.Three patients reported to our Maxillofacial Surgery Department between 2013 and 2017. In all cases, medical history did not include systemic burdens, genetic load, or an injury from the past, except hypothyroidism in one patient. All the patients did not present any abnormalities in a physical examination except lesions in jaws. In addition, laboratory tests, including the level of parathormone (PTH), calcium and phosphates were correct.Due to the large size of the tumors, young age, and good communication with the patients and their families guaranteeing the discipline of follow-up visits, surgery was abandoned and the patients were qualified for nonsurgical, pharmacological treatment. It was carried out by a series of dexamethasone  injections into the tumor mass after drilling of two openings in the jawbone at the top of the bone deformities that corresponded to the largest bone defects in the CBCT studies. All the patients remained under permanent control of the Maxillofacial Surgery Outpatient Department.Patient M.K., male, aged 14 reported due to a tumor of the body of the mandible diagnosed in February 2013. The patient was obese. Physical examination revealed facial asymmetry with the distention of the body of the mandible on the right side at teeth 41–47 with decrease in the depth of the oral vestibule and the elevation of the floor of the oral cavity in the areas of both sublingual folds . The conAn orthopantomographic radiograph showed a multi-cavity osteolytic defect with septa extending from tooth 33 to 47 region a. CT scaFrom April to June 2013, the patient was administered 12 mg/3 mL of Dexaven to the tumor mass every 7 days, and then for the next 3 months 8 mg/2 ml of Dexaven was administered every 14 days . The injCurrently, 9 years after the completion of therapy and mandibular body modeling surgery, clinically and radiologically, there are no signs of recurrence a,b.Patient A.S., female, aged 29 reported to our department due to a tumor of the hard palate. The lesion appeared for the first time in June 2012 during pregnancy, and at the time when pregnancy was lost, it subsided. It appeared again during the second pregnancy in October 2012. In January 2014, the resection of tooth 12 root was performed, resulting in a short-term reduction in the lesion. Physical examination revealed firm palatal distension in the midline. The CBCT showed a multi-cavity osteolytic defect in the maxilla in region of tooth 13 to 24 a,b.In July 2014, a tumor specimen was collected. Histopathological examination revealed the diagnosis of “Tumor gigantocellularis pigmentosus”. From September to December 2014, the patient was administered, through two bony canals into the tumor mass, 12 mg/3 mL of Dexaven every 7 days, followed by 8 mg/2 mL of Dexaven every 14 days for the next 3 months. Tumor structure and bone tissue recovery were assessed using control orthopantomographic radiographs. The patient underwent a follow-up CBCT scan of the mandible 4 months after the start of therapy, which showed normal progressive bone remodeling. In February 2015, the patient underwent modeling surgery of the deformed hard palate using piezo surgery with macroscopic evaluation of bone tissue and collection of material for histopathological examination .Macroscopically, normal cancellous bone was identified and a small focus of altered tissues on the palate in the midline. The material for histopathological examination was collected in three specimens. Histopathological examination revealed tiny fragments of connective tissue and bone fragments and tumor fragments composed of spindle cells and osteoblastic giant cells with visible deposits of hemosiderin, and no histological features of giant-cell tumor. Currently, after 8 years of therapy and hard palate modeling surgery, there are no clinical or radiological signs of recurrence .Patient M.K., female, aged 18 reported due to a tumor of the body of mandible which was detected in January 2017. The onset of complaints in the form of numbness of the lower lip was diagnosed at the dental office and subsequently treated neurologically without effects. In April 2017, there was swelling of the mandibular body—a orthopantomographic radiograph was collected, which showed an osteolytic lesion of the body of the mandible. A diagnostic biopsy was performed and showed “a lesion consisting of fields of fibroblasts with hemorrhages and numerous multinucleated giant cells and blood-filled spaces.” At this time, physical examination revealed asymmetry with swelling of the chin and mandibular body areas—excessively hard, painful, reddened skin over the tumor, bilateral hypoesthesia of the third branch of the trigeminal nerve, loosening of teeth 33–43, reddening of the adjacent mucosa and a bluish-purple tumor with elastic consistency. A diagnostic biopsy was performed that showed “a lesion consisting of fields of fibroblasts with hemorrhages and numerous multinucleated giant cells and blood-filled spaces.” In June 2017, a tumor specimen was collected for histopathological examination revealing the CGCG. From June to August 2017, the patient was administered a total of 84 mg/21 mL of Dexaven to the tumor mass through two bone canals in divided doses at 2-day intervals while being advised to undergo endodontic treatment of teeth 33–43 and 70 mg of alendronic acid orally once a week. Tumor hardness and structure were continuously monitored during follow-up examinations performed on injection days. In September 2017, due to the enlargement of the lesion and destruction of the cortical bone found on clinical examination and follow-up radiograph, as well as the onset of steroid acne, drug treatment was discontinued and surgical segmental resection of the mandibular body from the area of tooth 37 to the angle on the right side with implantation of a reconstructive plate was performed.The patient was then qualified for reconstructive surgery using a free fibula flap (FFF) collected from the left lower extremity with simultaneous placement of four dental implants; the surgery was performed in October 2017. Currently, 5 years after the end of therapy, there are no clinical of radiological signs of recurrence. The summary of basic data for the case series is presented in CGCG usually occurs in patients in the 2nd and 3rd decades of life, so the age of the patients presented in this article corresponds to the data presented. In the literature, aggressive forms of CGCG that model the surrounding soft tissues are detected more often in the mandible than in the maxilla.Based on clinical features, there are two forms of CGCG: a non-aggressive form, characterized by slow growth and scanty symptoms, and an aggressive form, characterized by rapid growth, pain, bone destruction, displacement of teeth or their embryos, resorption of tooth roots ,11. AccoWhat is missing in the available research is the evaluation of the dentition with the preservation of viable teeth and a final histopathological assessment of therapeutic success. We found no case reports in the literature in terms of the exact size of the tumor, the viability of the teeth and definitive verification by histopathological examination after nonoperative treatment. There is also no description of the technique of depositing drugs, including dexamethasone, into the calcified tumor mass. In the cases we described, tooth viability was preserved after treatment. Surgery was required to reshape the bone and collect material for histopathological verification. Leaving viable teeth in the tumor is questioned in the literature, but the authors of the study proved the correctness of leaving viable teeth provided regular examinations, which may suggest the possibility of such a procedure in patients with discipline.The third case presented seems to have been unsuccessful due to the lack of bone constraints on the tumor, and thus insufficient healthy bone tissue around the tumor, which is a source of osteoblastic cells necessary for the regeneration process. The surrounding bone was thin and the teeth were loosened. Although a follow-up orthopantomographic image showed the formation of subtle trabecular tissue inside the tumor, we decided to perform radical surgery.Most authors take the view that surgical removal is the only treatment option for large and aggressive forms of CGCG. This type of treatment is unfortunately associated with post-surgical facial deformities and loss of teeth or their embryos, and thus the need for reconstructive procedures such as autogenous bone grafting, which carries the risk of serious complications, especially in children ,23,38.Another point worth considering is that even after surgery, aggressive forms tend to recur in 37.5% of cases. In the clinical cases described in this article, due to the large size of the tumors, the immature age of the patients, and good communication to ensure the discipline of follow-up visits, surgery was abandoned in two of the three cases and the patients were qualified for series of dexamethasone injections into the tumor mass. Most of the literature allows pharmacological treatment only for lesions of minor severity and non-aggressive form.In two of the described cases of aggressive, large lesions, pharmacotherapy was fully successful, while in the third case, due to complication (occurrence of steroid acne) and failure to achieve full therapeutic effect, surgical treatment was performed. The likely mechanism of action of dexamethasone injected into the tumor mass is to inhibit the production of lysosomal proteases involved in bone resorption by giant cell tumors, as well as induce osteoclast apoptosis, thereby accelerating bone regeneration.There are no reports in the available literature on pulp vitality and root canal treatment of teeth embedded in tumor masses. In the two described cases of successful pharmacological treatment, endodontic treatment of teeth embedded in the tumor mass was abandoned due to the absence of tooth root resorption and normal pulp response to thermal vitality tests.Dexamethasone injections could be the recommended treatment for intraosseous giant cell tumors in cases with preserved bony borders despite the deformity, with teeth left in the lesion if their pulp is vital during treatment. Further investigation is needed in order to create qualification criteria and clinical recommendations.Therapeutic success depends on patient discipline and regular injections two to three times a week without interruption for at least three months with regular clinical and radiological follow-up and monitoring for complications.


Of all schwannomas, 24-45% arise from the head and neck area. Laryngeal schwannoma is an uncommon site. We report a case of a 34-year-old male who presented to the emergency department with worsening sudden onset inspiratory stridor. On direct laryngoscopy, he was noted to have a laryngeal mass, which caused a ball-and-valve effect on the laryngeal inlet, resulting in airway compromise. He was subsequently intubated and admitted to the intensive therapy unit. He underwent endoscopic removal of a bulky submucosal swelling in the supraglottic region using coblation. The post-operative histopathological assessment confirmed features of laryngeal schwannoma. A six-month post-operative follow-up showed no signs of recurrence. Due to the rarity of this condition, the evidence for the most efficient management of these cases is still not clear. Diagnosis is by histology assessment, although clinical evaluation should raise suspicion. Surgery remains the mainstay of treatment with a good overall prognosis. In current literature, the technique of surgical excision varies with different outcomes. We report a case that was managed with micro-laryngoscopy and coblation with good effect. Schwannoma is a benign neurogenic tumour that arises from the Schwann cell of the peripheral nervous system. Verocay, in 1908, was the first to describe the tumour . Due to A 34-year-old male presented to the emergency department with a two-hour history of worsening stridor and early respiratory failure. Medical management, including intravenous steroid therapy, adrenaline via a nebulizer , and high-flow oxygen via a non-rebreather mask, was commenced. After an hour, there was a poor clinical response to the medical therapy, and thus, the decision was taken to intubate the patient with a size 8 endotracheal tube via direct laryngoscopy. The anaesthetist noticed a large laryngeal mass causing a ball-and-valve effect on the laryngeal inlet. The patient had a grade 1 intubation view. The patient had an unremarkable history apart from several months of sensation of a foreign body in the throat. He denied any symptoms of dysphonia, dysphagia, or odynophagia prior to admission. He was a cigarette smoker but otherwise fit and well with no known allergies. Family history was negative for relevant diseases.He was admitted to the intensive therapy unit (ITU) after being successfully intubated and ventilated. The following day, an attempt to extubate the patient was unsuccessful. He became stridulous, and the decision was taken to re-intubate him. A size 8 percutaneous tracheostomy was then performed to wean the patient off the sedation and ventilation. He was admitted to ITU for eight days, where he was fed via a nasogastric tube.During his admission, flexible fibreoptic rhino-pharyngo-laryngoscopy revealed the presence of a bulky 60 x 40 mm swelling arising from the right arytenoid cartilage and post-cricoid space. The airway was compromised with the ball-and-valve effect, but both vocal cords were freely mobile. There were no palpable cervical lymph nodes. An endoscopic evaluation has been recorded Video .He underwent an MRI scan of the neck, which showed a lobulated, exophytic soft tissue mass occupying the posterior oropharynx/laryngopharynx. It measured approximately 53 mm in craniocaudal diameter and 31 x 20 mm in axial diameter . There was no evidence of malignancy.The patient's symptoms improved significantly, his swallow function returned to normal, and his breathing improved one week after the procedure. He was subsequently discharged home. Follow-up was arranged at six-month intervals for a duration of three years after the operation, and his symptoms completely subsided. Flexible fibreoptic rhino-pharyngo-laryngoscopy did not reveal any signs of recurrence of the disease.Laryngeal schwannomas  are encapsulated tumours that arise from benign neoplastic Schwann cells. Schwannoma is a grade I benign tumour, according to the World Health Organization classification of tumours, which describes it as a tumour that resembles normal tissue and is slow growing . It affeLaryngeal schwannoma was first described by Suchanek in 1925 . In WongThe clinical presentation depends on the mass size and location of the laryngeal schwannoma. If the mass is small, patients are usually asymptomatic. The severity of symptoms varies and can include non-specific symptoms such as globus sensation, dysphagia/odynophagia, dysphonia, or stridor. Dysphonia may be caused by a decreased mobility of the vocal cords, in the event that the schwannoma originates in them, or more frequently, through impairment of laryngeal mobility by the effect of the tumour mass, which may mimic cricoarytenoid joint fixation . These sEndoscopic evaluation of the larynx will often reveal a submucosal swelling that could contribute to airway compromise. Other imaging modalities, including CT and MRI, are paramount in the diagnosis and management of laryngeal schwannomas. They provide information on the size, location, and extent of the disease. They also have the ability to demonstrate the typical features of a benign disease and aid in surgical planning.Histopathological assessment is important to distinguish schwannoma from neurofibroma. This is often difficult as the neurofibroma tends to recur, with 10% undergoing malignant transformation. In Enzinger and Weiss's 'Soft Tissue Tumours', they suggest that the histological diagnosis of schwannoma can be made in the presence of three features: (1) clear capsule; (2) presence of Antoni A and B areas; and (3) a positive reaction for S-100 .The literature shows that the main differential diagnoses are benign laryngeal tumours  and non-neoplastic lesions . Malignant lesions should also be excluded. Neurofibromatosis is an important differential diagnosis that should be excluded through careful clinical examination. Clinical features of neurofibromatosis include cafe-au-lait spots, cutaneous nodules, and freckling of skin folds . These fSchwannomas are radioresistant and hence do not respond to radiotherapy . SurgicaIn Wong et al.'s systematic review, 53 patients with laryngeal schwannoma underwent surgical excision. Different techniques were observed, including open and transoral approaches . The oveSome authors suggest that tumours located in the hypopharynx may not be suitable for endoscopic removal, as poor exposure may result in more mucosal injury . HoweverSchwannoma within the larynx is a rare, slow-growing, benign tumour that can present with a variety of symptoms. The overall prognosis for laryngeal schwannoma is good. Schwannomas are radioresistant, and thus surgical excision remains the treatment of choice. We believe that according to the size, site, and extension of the laryngeal schwannoma lesion, the best surgical approach can be determined. Endoscopic coblation using the laryngeal wand is tried for the first time in the treatment of laryngeal schwannoma in this case, and it showed a safe non-invasive way for complete excision of this lesion. A long follow-up is required to ensure that there are no signs of recurrence. Due to the limited number of cases reported in the literature, there is no consensus regarding the best modality of treatment or duration of follow-up required post-operatively.


Granulomatous mastitis (GM) is a rare disease, particularly among men. Herein, we present a case of GM diagnosed in a 63‐year‐old male patient who showed reduction in the tumor size during 3 months of observation. The optimal treatment strategy for granulomatous mastitis in men is still controversial due to its rarity. However, observation may be a viable option in asymptomatic cases. He was diagnosed with follicular lymphoma 5 years ago and treated with six cycles of a combination of rituximab and bendamustine, followed by treatment with rituximab for 2 years as maintenance therapy. Recurrence of lymphoma was detected at the cervical and axillary lymph nodes after 2 years of an uneventful post‐therapeutic course. Lenalidomide and rituximab were administered as treatment for recurrence. A CT scan performed at the end of chemotherapy showed disappearance of all target lesions, but appearance of a new mass in his left breast. The patient was referred to our department for further evaluation. On physical examination, a mass measuring approximately 1 cm in diameter was palpable in the subareolar area of his left breast, while no swollen lymph nodes were detected in the ipsilateral axillary region. Laboratory tests including tumor marker analyses were all within normal levels . CT scan confirmed the presence of a lesion measuring 13.9 × 12.9 × 6.1 mm in the subareolar portion of the left breast Figure . UltrasoSince there was no suspicion of malignancy and the patient was asymptomatic, he was admitted for observation. Ultrasonography performed after 3 months revealed that the size of the tumor was 7.9 × 5.5 × 5.2 mm Figure , which w3GM is diagnosed histologically by confirming the presence of non‐necrotizing granuloma formation, without any evidence of microorganisms such as Mycobacterium or fungi.GM is rarely observed in males. To the best of our knowledge, there are only 16 reported cases of GM in male patients.One possible reason for rarity in males is the absence of mammary lobules, which are usually affected during this disease. It has been reported that estrogen stimulation causes the development of acini and lobules in male breastsThe optimal treatment strategy for GM remains controversial. Treatment approaches include observation, oral corticosteroid administration, and surgical excision. Incision and drainage may be a treatment option for bacterial infection cases. Oral corticosteroids have been used as the first‐line treatment in some studies and reported to be effective in decreasing the size of GM.Of the 11 reported male cases in which treatment was mentioned, four patients underwent surgical excision. Core needle biopsy was not attempted in these four cases, and the lesion was surgically excised in toto as an excisional biopsy. Of the remaining seven patients, four who received oral corticosteroid therapy were symptomatic, whereas two who underwent observation did not have any symptoms other than the breast mass. In one patient who was treated with bromocriptine for pituitary tumor, the breast tumor disappeared after the treatment. None of the patients experienced recurrence during their clinical course, but the follow‐up periods were not sufficient to determine the long‐term outcomes. In our case, since the patient was asymptomatic and diagnosis of GM was confirmed histologically, we decided to observe the lesion.In contrast to other reported cases, the tumor in our case appeared during the clinical course of follicular lymphoma treatment. Although not histologically proven, one possible explanation for the occurrence of GM in our case may be the autoimmune response toward the extranodal lesion of lymphoma in the breast.To summarize, we presented a rare case of GM in a male patient. Observation may be a viable option in the asymptomatic cases. However, further studies including a larger number of patients and longer observation period are needed for reliable long‐term outcomes.Shinya Yamamoto: Supervision. Kazutaka Narui: Supervision. Yoshie Fujiwara: Supervision. Shoko Adachi: Supervision. Mahato Sasamoto: Supervision. Masanori Oshi: Supervision. Akimitsu Yamada: Supervision. Eita Kumagai: Supervision. Masako Otani: Supervision. Itaru Endo: Supervision.The authors declare no conflicts of interest regarding the publication of this article.This study was conducted ethically in accordance with the World Medical Association Declaration of Helsinki.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.