This is a case report of Basidiobolomycosis in a 65-year-old male patient from Jizan presenting with colonic perforation and concomitant liver involvement from February 2021 to July 2021. To control the infection, the patient underwent colonic resection and segmental liver resection, as well as three antifungal drugs. The treatment was successful, and the condition was completely resolved. Basidiobolomycosis is a rare granulomatous infection of the skin and subcutaneous tissue that can develop quickly and create a persistent infection of the subcutaneous tissue in immunocompetent individuals. It most usually affects infants under the age of one month and is rarely found in adults ,2.Even though gastrointestinal involvement is uncommon, it can affect the stomach, small intestine, colon, and liver. Basidiobolomycosis can cause symptoms that are similar to colonic or inflammatory bowel disease. Colonic bowel disease with concurrent liver involvement is uncommon, with only six cases reported. Colonic perforation caused by Basidiobolomycosis is extremely uncommon, with only two cases documented in the literature .In the current case, Basidiobolomycosis was diagnosed in a 65-year-old male who had colonic perforation and liver involvement. A comprehensive literature review was conducted from 1964 to July 2021 to evaluate previously reported cases and gather information on symptoms, diagnostic method, and treatment choices.A 65-year-old man from Jizan (southwestern Saudi Arabia) presented with a one-month history of lower abdominal pain, constipation, and weight loss. No significant medical or surgical history was recorded. Ethical approval (HA-02-J-008) was obtained from the Unit of Biomedical Ethics Research Committee at the Faculty of Medicine in King Abdulaziz University, Saudi Arabia.Abdominal examination revealed a palpable right-side-abdominal mass with mild tenderness. Laboratory tests revealed leukocytosis (white blood cell (WBC) count: 17,230/mcl), eosinophilia (2540/mcl (14.7%), microcytic hypochromic anemia (hemoglobin (Hb): 10.8 g/dL), and thrombocytosis . A computed tomography (CT) of the abdomen revealed circumferential intestinal wall thickening of the cecum and ascending colon, luminal constriction with enlargement of several mesenteric lymph nodes, and two lesions in the liver, segments five and seven, with peripheral enhancement and central hypodensity of 3 × 3 cm .Colonoscopy revealed an ulcerated, partially obstructed large mass in the proximal ascending colon. A biopsy revealed active eosinophilic colitis with necrotic debris. A liver biopsy of tissue from the mass was performed and showed an eosinophilic abscess. The patient was diagnosed as a case of cecal mass with the possibility of a cancer, colon with liver metastasis need to be rolled out.Escherichia coli, Candida albicans, and Basidiobolus spp. The patient was started on the antifungal medication voriconazole intravenously (200 mg BID) and was switched to an oral tablet (200 mg BID).The patient returned to the emergency room two weeks later with intense abdominal pain that lasted a few hours. On examination, the patient’s temperature was 38 °C, his pulse was 110 beats/min, and the abdomen was tender with rigidity and an absence of bowel sounds. Laboratory tests revealed the following: WBC count, 13,700/mcl; Hb, 10.4 g/dL; neutrophil count, 9210/mcl (89%); and eosinophil count, 0/mcl (0%). A CT scan of the abdomen showed cecal perforation with free air in the peritoneum and an increase in the size of the liver masses (3 × 4 cm) . EmergenTwo weeks later, the patient developed a fever and right-side abdominal pain. Laboratory tests revealed the following: WBC count, 17,230/mcl; neutrophil count, 11,350/mcl (65.9%); eosinophil count 2540/mcl (14.7%); and Hb, 10.8 g/dL. Percutaneous CT-guided drainage was used to drain the abdominal collections. The pleural effusion was aspirated, and percutaneous liver abscess drainage was attempted but failed due to the thick nature of the abscess. Another antifungal medication, itraconazole (200 mg PO. BID), was administered alongside voriconazole (200 mg IV BID) to control sepsis. The patient improved and was discharged from the hospital.Two months later, the patient presented with fever and right upper abdominal pain despite being treated with oral voriconazole (200 mg BID). Upon examination, there was tenderness in the right upper abdomen with low-grade fever. A CT scan of the abdomen showed a liver abscess in segments six and seven (3 × 4 cm and 4 × 2 cm respectively) . IntraveBasidiobolus ranarum (B. ranarum). It belongs to the family Zygomycetes. The Zygomycetes family includes two orders, Mucorales and Entomophthorales. Mucorales cause infection in immunocompromised patients such as diabetics and post-transplants. Entomophthorales, which include Basiodiobolus, cause infection in immunocompetent individuals [Basidiobolomycosis is a rare fungal infection caused by ividuals .Basiodiobolus spp are filamentous fungi that were first identified in 1886 in frogs . They arB. ranarum infection in a human (a subcutaneous infection) was reported in Indonesia [In 1956, the first case of Previous patients were mostly male, with ages ranging from one month to 80 years old . GastroiThe most common presenting symptoms of GIB are abdominal pain and fever; however, some reported cases presented with colonic obstruction , lower GLeukocytosis, eosinophilia, and anemia were present in most of the cases. Radiological investigation in most of the patients revealed either gastrointestinal wall thickening resembling inflammatory bowel disease, or a colonic mass that mimicked colonic cancer, with or without a concomitant liver mass.B. ranarum grows in the submucosa, so the histological examination of the biopsy commonly reveals inflammatory changes with eosinophilic infiltration and, in some cases, the presence of granulomas.Endoscopy and biopsy are inconclusive in nearly all reported cases. Therefore, a mucosal biopsy is usually performed, and All reported cases were diagnosed either by excisional biopsy of tissue from the mass or histological examination of the resected segment of the colon, which is considered the gold standard for diagnosis. Histological diagnosis usually reveals the diagnostic features of this fungal infection, namely the presence of fungal hyphae with Splendore–Hoeppli bodies, granuloma with abundant eosinophilia, and intensely radiating eosinophilic granular material. A few reported cases were diagnosed by polymerase chain reaction (PCR) and molecular methods such as DNA sequencing of samples obtained from equivocal tissue specimens.Treatment with antifungal therapy without surgical resection of the affected part has been successfully reported in a few cases . In thesGIB can be associated with a variety of signs and symptoms, and it can affect people of all ages. Most GIB cases presented with colonic involvement, few with liver involvement, and very rare cases presented with colonic perforation and concomitant liver involvement, as in this study. The diagnosis of GIB requires a high index of suspicion, and we recommend its inclusion in the differential diagnosis of abdominal masses associated with fever, weight loss, and eosinophilia, especially in endemic regions.


Penile schwannoma is a rare tumor. They commonly present as an asymptomatic, painless and slow growing mass. Other presentations include sexual dysfunction, most commonly dyspareunia, followed by erectile dysfunction, abnormal penile curvature or pain with ejaculation.A 26-year-old male presented atypically with painful nocturnal penile tumescence, along with multiple nodules over the dorsal penis. Excision of multiple penile tumors under general anaesthesia was performed and histopathologic examination revealed benign schwannoma.Our hypothesis is that the schwannoma lies along the axis of the dorsal penile nerve, and compression of this nerve occurs during his erection causing pain. However, there are limited presentations of painful erections in penile schwannomas, and we hope that future studies can help confirm this theory. Schwannomas are a form of peripheral nerve tumors made up of neoplastic Schwann cells that typically occur as solitary, encapsulated masses. They can occur throughout the body, but more commonly arise on the head, neck, or flexor surfaces of limbs , 2. The A 26-year-old man with a history of ankylosing spondylitis (Human Leukocyte Antigen B27 gene) and previous circumcision first presented with a one-year history of recurrent painful nocturnal erections. He had prior consultations with various urologists and did not respond to oral analgesia. The frequency of painful nocturnal erections increased from once per week, to thrice per week over the past year. Each episode of painful nocturnal tumescence lasted approximately 5 minutes and the patient was often awaken from sleep by the severe pain, which affected his sleep and quality of life. The patient mentioned that the nocturnal erections were stronger and more painful compared to when he was awake. There was no history of priapism, sexual transmitted disease, or genital trauma. There were no persons with known neurofibromatosis in his family. On examination, four lumps could be palpated over the dorsum of the stretched penis. Two were superficial nodules on the distal shaft, with one deep nodule each at the mid shaft and base of the penis. The nodules were 0.5 cm or less in diameter and firm in nature. The mid shaft nodule was tender on palpation and correlated with the site of painful nocturnal erections. The penis was otherwise unremarkable and there was neither penile curvature on erection nor any palpable lymph nodes in the femoral or inguinal areas. Nodules or café-au-lait spots were not present in the rest of the body.Over a five-year follow-up, patient developed worsening symptoms with the painful erections occurring twice every night from one episode a week. Physical examination and interval ultrasound imaging demonstrated an increase in the number of nodules from four to five with further growth of the existing nodules. Most noticeably, the right intracavernosal nodule increased from 4 mm to 7 mm in diameter. The patient decided for surgical excision of multiple penile nodules.Laboratory findings included normal blood cell counts, chemistries and urinalysis.Initial ultrasound penis showed multiple rounded heterogeneously echogenic nodules in the subcutaneous region of the dorsal penile shaft Fig. . The nod3Patient underwent excision of multiple penile tumors under general anaesthesia. A circumferential incision was made at the previous circumcision site. The penis was then degloved to its base and the layers dissected down to Buck’s fascia Fig. . There wThe Hematoxylin and Eosin stains and immunohistochemical studies were performed in accordance with local laboratory protocols. All specimens show similar morphology. The circumscribed and thinly encapsulated nodules Fig.  were madIn the follow up consultations over a year after surgery, there was no further nightly painful erections and patient was able to sleep well. On examination there is a small nodularity at mid shaft which is likely due to scar tissue formation. However, the patient did experience difficulty maintaining erection due to discomfort over the surgical site. This was managed well with Sildenafil 25 mg, with an improvement in International Index of Erectile Function  score frSchwannomas rarely present with penile pain. There have been postulations made regarding the correlation of symptoms to neuroanatomy. A literature review done by Huang et al. concluded that patients with penile root schwannomas are more prone to symptoms with discomfort or sexual dysfunction (4 of 6) compared with patients with penile shaft schwannoma 7 of 16) or glans schwannoma (2 of 7)  of 16 or. Pain maPenile schwannomas normally occur at the dorsal penile shaft. However, there have been documented cases where the tumor has infiltrated the glans and prepuce . In suchSchwannomas of the penis are usually benign, but four malignant variants have been reported in literature. No cases of benign penile schwannoma have been reported to be associated with hereditary diseases . SchwannThere is no strong evidence about the correlation between schwannoma and erectile dysfunction . This paSchwannomas of the penis are extremely rare and typically present as a solitary, asymptomatic, painless and slow-growing tumor. The rarity in this case is that our patient presented with painful nighttime erections. Based on the penile neuroanatomy, penile schwannomas at the mid shaft should originate from the dorsal nerve of the penis. Our hypothesis is that the schwannoma lies along the axis of the dorsal penile nerve and compression of this nerve occurs during his erection causing pain. However, there are limited presentations of painful erections in penile schwannomas, and we hope that future studies can help confirm this theory.


Colonic lipomatous polyps are often an incidental finding during colonoscopy. Generally, these types of polyps can cause gastrointestinal bleeding when they are larger than 4 cm in size. Some case reports have documented the occurrence of overlying adenomatous formations in the apical portion, as well as ulcerated mucosa. There is currently no standardized endoscopic removal technique for their treatment. In this report, we present a case of a large and ulcerated lipoma causing rectorrhagia, which was successfully treated with endoscopic en-bloc resection and endoloop placement. Colonic lipomas, which are recognized as non-epithelial benign tumors of mesenchymal origin, generally represent an incidental finding during colonoscopy. Their reported incidence varies from 0.2-4.4% . AlthougA 74-year-old male was admitted due to rectorrhagia. The patient had been undergoing regular therapy with an antiplatelet agent (clopidogrel 75 mg/day). On admission, his temperature was 37.2 °C, the blood pressure was 115/75 mmHg, the pulse rate was 85/minute, and his respiratory rate was 16/minute. Laboratory exams were remarkable for the presence of anemia only (hemoglobin level of 11.2 g/dl). Physical examination excluded signs of peritoneal reaction to palpation. At that point, it was decided to perform an urgent colonoscopy, which revealed both fresh and coagulated blood along all the colonic tracts and, in addition to the presence of diverticular orifices in the sigma, a large pedunculated submucosal lipoma of about 5 cm in the cecum, whose apex, after repeated washing, presented hematin and fibrin, related to ulcerated mucosa , using the setting of forced coag of 60 watts  Figure .At the end of the procedure, the retrieved specimen measured 3 cm Figure . The relColon lipomas are non-epithelial submucosal tumors and are predominantly located in the right colon and cecum in approximately 90% of the cases . EndoscoIn a systematic review of 88 articles regarding symptomatic large lipomas, 127 patients were analyzed in light of their clinical course . SeveralThis case highlights how endoloop could be used with excellent results to prevent both early and late bleeding related to endoscopic resection of large colic lipoma by expert endoscopists. Therapeutic digestive endoscopy has the advantage of being an easily repeatable procedure compared to surgery, and it allows for the treatment of most post-procedural complications quickly.


Denervation pseudohypertrophy is an uncommon cause of limb swelling, which may be overlooked. It is an important diagnosis to arrive at, as it instructs the search for an underlying cause which may itself require intervention. We present the case of a 32-year-old male rugby player with a 2-year history of left calf swelling and intermittent pain and tightness. He described a previous history of 2 left sided lumbar micro-discectomy surgeries. There was no tenderness or sensory deficit on examination. MRI of the left calf revealed muscular enlargement, with fat interspersed between the muscle fibers, in keeping with pseudohypertrophy. This has a number of causes, in this cause attributed to lumbar radiculopathy. This case highlights a rare but important cause of limb swelling which should be considered in the workup of a unilateral swollen limb. Muscle denervation pseudohypertrophy can be seen in a variety of conditions and settings, including secondary to peripheral nerve traumatic injury, peripheral neuropathic conditions , disorders of the anterior horn cells such as poliomyelitis, and due to radiculopathy A 32-year-old male rugby player presented with a 2-year history of swelling in the left calf. He described intermittent pain in the left calf with a sensation of tightness and occasional tingling in the left foot. Past medical and surgical history was significant for previous lumbar microdiscectomy.On examination there was asymmetry of the calves, the left being larger than the right. There was no tenderness on palpation. Pulses were present and normal. There was no sensory deficit.The patient was referred for compartment pressure testing, which was normal. An MRI scan of the calves was subsequently performed. This demonstrated asymmetric enlargement of the musculature of the left calf with evidence of fatty infiltration . The appUltrasound guided muscle biopsy of the left gastrocnemius muscle was then performed. Histopathological analysis demonstrated atrophy of skeletal muscle fibers, with extensive infiltration by mature adipose tissue. These findings, in concordance with the clinical history and the appearances at MRI were felt consistent with partial denervation pseudohypertrophy of the calf musculature reflecting incomplete neural injury due to degenerative lumbar disc disease and possibly also contributed to by post-operative scarring.Denervation pseudohypertrophy is an uncommon and therefore possibly overlooked cause of unilateral calf swelling. It is characterized by muscular enlargement due to the accumulation of fat between the muscle fibers. There is also muscular enlargement in true hypertrophy, however the enlarged muscle would demonstrate normal homogenous muscle signal intensity. Denervation pseudohypertrophy occurs due to partial denervation and has been described in association with a variety of conditions including diabetic neuropathy The diagnosis of muscular hypertrophy requires multidisciplinary input as outlined in our case, with good clinical history and examination, evaluation with MRI and histopathologic analysis all necessary. An accurate diagnosis is important as it instructs additional investigations as required to elucidate an underlying cause Deep infiltrating intramuscular lipoma is an important differential consideration There are no descriptions in the literature to our knowledge of successful interventions for denervation pseudohypertrophy secondary to lumbar radiculopathy. While the natural history of the condition remains poorly understand as it typically only comes to clinical attention when significant pseudohypertrophy has occurred and other causes of a swollen limb have been excluded, it is possible that the degree of neural injury required to cause this may be irreversible, or not completely reversible. The value of this diagnosis may lie in providing reassurance to patients and excluding the possibility of more sinister causesThe patient in our case underwent no specific interventions and was given reassurance and encouraged that the etiology of the limb swelling was not an aggressive one. He was referred back to see his spinal surgeon for an opinion regarding repeat surgery, however as he was functionally and symptomatically very well, and there was a possibility that the pseudohypertrophy would not be reversible following repeat surgical exploration, this was decided against.There are no financial disclosures.Consent provided by patient to Dr John Hynes.


Rhabdomyosarcoma is a highly aggressive cancer that is generally considered a disease of childhood. A vast majority of cases occur in those below the age of 20. Rhabdomyosarcoma can occur in any soft tissue in the body but is primarily found in the head, neck, orbit, genitourinary tract, genitals, and extremities. Prognosis is closely tied to the location of the primary tumor and the extent of metastatic spread. As with most sarcomas, rhabdomyosarcoma has a pattern of hematogenous spread which favors metastasis to the lungs. Other common areas include bone marrows, liver, breasts, and brain. One unusual pattern is the presence of diffuse bone marrow metastases in absence of significant soft tissue disease other than primary . Frequently in such cases, patients may have initial presentation similar to hematologic malignancy especially when the primary tumor is not evident. This pattern has been rarely described in the radiology literature. This pattern appears to be well documented in pathology literature. Even more rarely, in some cases, the primary tumor site may not be found after imaging and may remain undetermined even postmortem - only diagnosed by bone marrow aspiration. Awareness of this unique pattern is clearly important for radiologists, especially pediatric radiologists, as misdiagnosis can lead to delay in appropriate treatment that ultimately results in increased mortality. We present a case of rhabdomyosarcoma with this unique pattern of bone marrow metastases in which initial differential diagnosis favored a leukemic picture. This paper will go over the diagnostic techniques utilized throughout our patient’s disease course as well as treatment. Pediatric soft tissue sarcomas (STSs) are a rare group of tumors that originate from a primitive mesenchymal cell. They account for approximately 3%-4% of all childhood cancers in the United States at an estimated incidence of five per million. Rhabdomyosarcomas (RMSs) are the most common STS of childhood and represent approximately half of all STS in patients below 20 years of age . These cAn 11-year-old male presented in the emergency department (ED) for increasing levels of back discomfort after a fall a few days prior to presentation. Thoracic and lumbar spine films were obtained revealing loss of vertebral body height involving T12 concerning compression fracture  (approximately 70%) and ARMS (approximately 20%), however, other subtypes exist including pleomorphic (approximately 10%) and spindle/sclerosing (approximately 10%) . WhereasWhile local complications from primary sarcomas can cause significant morbidity, the most life-threatening complications are from the hematogenous dissemination. For most STS originating from the extremity, chest wall, or head and neck, the most common metastatic site is the lungs. Other sites include the BM, liver, breasts, and brain . ApproxiIn cases of RMS with BM metastases presenting with symmetrical diffuse BM involvement, patients can oftentimes be misdiagnosed with leukemia as initially thought in our case. This mimicry can be attributed to the nonspecific findings associated with small primary tumors that do not have overt clinical symptoms at their primary sites ,9-14. ThIn earlier studies, bone scan was the standard for finding osseous metastases. With recent studies though, FDG-PET has been shown to perform better at detecting BM disease during the initial diagnosis of RMS ,10,15. TIn conclusion, the prognosis for RMS with BM metastases is poor and has not substantially improved for the past two decades. While RMS BM metastases traditionally occur in metastatic diseases that have disseminated to multiple parts of the body, there have been reported cases in which BM metastases occur as the sole metastatic location. In some, the primary tumor is not found and can be misdiagnosed as acute leukemia. This concern for myeloproliferative disease can drive physicians to order initial imaging modalities such as MRI targeted towards marrow evaluation to examine the extent of disease. Though in retrospect, MRI may seem unnecessary, this is due to the initial confusing presentation. Once BM aspirate confirms the non-hematologic origin of the neoplasm, PET-CT should be used to detect the origin from an unknown primary. We acknowledge that the costly imaging modalities used in this case are not available worldwide and note this as a limitation in generalizability. This atypical presentation has been limited in radiology literature, however, through our case, we hope to raise awareness of this unique pattern for physicians across all specialties.


Congenital adrenal hyperplasia (CAH) refers to group of congenital diseases resulting from impaired adrenal steroidogenesis, and its most common cause is 21-hydroxylase deficiency. Testicular adrenal residual tumor (TART) is one of the major complications of CAH, possibly resulting from ectopic remnants of intra-testicular adrenal tissue which is stimulated by excessive secretion of adrenocorticotropic hormone (ACTH). This tumor can be misdiagnosed as Leydig cell tumor (LCT) in these people.The patient we are presenting is a 20-year-old man with a history of precocious puberty and a height below 3% of the population who underwent radical left testicular orchiectomy with a complaint of bilateral testicular mass, which is reported LCT in the pathology report. In preoperative imaging examinations, bilateral adrenal hyperplasia is observed. In hormonal examinations, the patient is diagnosed with CAH and has been treated with corticosteroids for one year.In patients who present with bilateral testicular mass, it is the best image by abdominopelvic CT scan before surgery to detect CAH. Congenital adrenal hyperplasia (CAH) refers to group of congenital diseases resulting from impaired adrenal steroidogenesis, and its most common cause is 21-hydroxylase deficiency. Testicular adrenal residual tumor (TART) is one of the major complications of CAH, possibly resulting from ectopic remnants of intra-testicular adrenal tissue which is stimulated by excessive secretion of adrenocorticotropic hormone (ACTH). This tumor can be misdiagnosed as Leydig cell tumor (LCT) in these people. Due to the possibility of histological misdiagnosis between TART and LCT, this case was reported to avoid unnecessary with TART diagnosis in mind in similar patients. The patient is a 20-year-old man who noticed a swollen testis about a year ago after falling off a motorcycle, which was painful but did not follow-up due to the lack of continued pain. A year later, he referred to a urologist again, who on examination, noticed swelling and a mass in both testes, and requested ultrasound and tumor markers for the patient. On ultrasound, a 26 × 21 × 26 mm hypoechoic mass containing echogenic foci suggesting calcification in the left testicle and another hypoechoic foci measuring 8 ×8 × 8 mm in the lower pole of the right testis and another small foci of 2 mm diameter was observed in the upper pole of the same testis, which primarily indicated a testicular tumor. In measuring tumor markers, BHCG, AFP and LDH markers were within the normal range. Due to the ultrasound and larger and painful left testicular mass, he became a candidate for left orchiectomy. In preoperation examinations, ultrasound was performed again and then computed tomography (CT) scan was requested for the patient. Complete abdominopelvic ultrasound before operation, reported bilateral adrenal hyperplasia was observed which eventually was recommended to perform CT scan. Examination of the testicles showed diffuse microlithiasis in both testes. At least two masses were seen in the left testis, the largest of which was 27 × 27 mm hypoechoic containing calcified foci with vascular flow. At least 4 hypoechoic masses with vascular flow were observed in the right testis, the largest of which was 5 × 3 mm. CT scan showed bilateral adrenal hyperplasia and two lymph nodes with maximum SAD of 7 mm in the para-aorta and aortocaval.The patient underwent a radical left orchiectomy and the pathology of removed testis reported consistently with sex cord stromal tumor in favor of Leydig cell tumor and in immunohistochemically (IHC) evaluation, calretinin was positive and CD99, WT1 and Melan A were weakly positive. After the operation, due to adrenal hyperplasia, hormonal tests were requested and the patient was referred to the endocrinology outpatient clinic. The patient has no history of hospitalization in infancy. He states that he was taller than his peers at the age of 7, but he has not increased in height compared to his peers since he was 10 years old. He did not mention the history of allergies to drugs and food and any sort of drug consumption. The parents were not related and his brother died at a young age (possibly a stroke). On examination of the patient, hair growth was sufficient and had a masculine pattern. He did not have gynecomastia on chest examination. 2. Due to the presence of bilateral adrenal hyperplasia in the imaging, the patient underwent laboratory tests due to suspicion of congenital adrenal hyperplasia. The laboratory tests showed high levels of aldosterone , ACTH , Renin activity  fasting ACTH , plasma renin activity , and 17-OH progesterone  and then due to the patient's age, adrenal hyperplasia on CT scan, history of precocious puberty and high levels of ACTH, the patient was diagnosed with CAH. Patient was then diagnosed with TART and was treated with corticosteroids for one year, which reduced the number of right testicular masses. The patient is still in the follow-up period.The patient did not have a history of hypertension and on examination, the patient's blood pressure was normal. The patient is 154 cm tall and weighs 60 kg. The patient's BMI was measured at 25.2kg/mCongenital adrenal hyperplasia is one of the most common autosomal recessive genetic disorders which caused by CYP21 (21-hydroxylase) deficiency in more than 90% of patients . With a misdiagnosis, the patient undergoes unnecessary orchiectomy, whereas TARTs typically respond to corticosteroid therapy and do not require surgery. Because of that, it is necessary to distinguish between these two. Correct diagnosis of TART from LCT is possible through thorough imaging, clinical, and histological examinations. While both LCT and TART occur in early adulthood , TART isIn addition, bilateral LCT and the concomitant presence of TART and LCT in patients with CAH have also been reported in studies , 16. TARHistologically, TARTs are similar to adrenal cortex tissue . Large pImaging is essential in the diagnosis and follow-up of TARTs. The first imaging is ultrasound which is a convenient and cost-effective method. Many of these lesions are hypoechoic and have a definite margin, but large lesions can be hyperechoic. On Doppler ultrasound, these lesions are usually hypervascular . MRI is In conclusion In this case, the patient presented with a painless bilateral testicular mass of unknown origin who underwent unilateral orchidectomy due to the malignant appearance on ultrasonography of the testes. In this case, it was difficult to differentiate TART from LCT, the main reason for that is the lack of CAH diagnosis in the patient's history. On the other hand, in the ultrasound, the masses were hypoechoic and despite the fact that the patient did not know that he has CAH, malignancy was at the top of our list of differential diagnoses. Another subject is the patient's pathology report, in which due to the similarity of the histological features of TART with LCT, in the absence of diagnostic suspicion of TART, is easily mistaken and leads to misdiagnosis. Immunohistochemical examination, especially the response of CD56, synaptophysin and CD10 can be helpful in the diagnosis of TART. According to the case, it is recommended that TART be part of our differential diagnosis in all people with CAH who present with testicular tumors, especially bilateral tumors. In cases where the patient presents without history of CAH and with bilateral tumor, it is best to perform an abdominal CT scan to examine the patient's adrenals before orchiectomy.


This report describes the clinical features of a granuloma within the layers of amniotic membranes (AM) along with its surgical management and outcome. An eight-year-old boy underwent an AM graft in the right eye for an acute chemical injury. As there was slow healing of the ocular surface, the eye was subjected to a simple limbal epithelial transplantation (SLET). Although the corneal surface was well epithelialized, a superior symblepharon was noted six weeks following SLET. Excision of the fibrotic tissue with an AM graft was carried out. Growth of a grayish-white fibrotic lesion in the visual axis of the cornea was noted eight months after the second AM graft. The optical coherence tomography line scan revealed the location of the lesion to be anterior to the retained SLET AM. This lesion progressively increased in size, so an excision biopsy was carried out. The lesion excised in toto with the AM, and an optically clear plane was noted. Histopathology of the tissue revealed the presence of myofibroblasts, which possibly originated from the AM fibroblasts. At the three-year follow-up period, there was no recurrence of the growth, and the final visual acuity was 20/40. This is a rare report of a granuloma case arising within the layers of an AM. The underlying etiopathogenesis could be due to the multiple AM grafts that the patient underwent. These repeated grafts can incite an immune response and lead to the formation of a granuloma. The special staining and the restoration of the corneal clarity with a stable ocular surface suggest the AM origin of the mass lesion. Removal of the layer of the AM with the granuloma has good outcomes, with no recurrence on long-term follow-up. The role of an amniotic membrane (AM) in a case of chemical injury is multimodal. In the acute phase, the AM helps the healing process through its anti-inflammatory and anti-fibrotic properties and by promoting surface re-epithelialization ,2. In thAn eight-year-old boy with acute chemical injury presented to the emergency clinic at our institute with a history of a fall of limestone in the right eye three days prior to presentation. There was redness, pain, and decreased vision since then. The visual acuity at the presentation was 20/320. On examination, there was significant lid edema, a superior conjunctival epithelial defect, and a total corneal epithelial defect Figure , Panel APostoperatively, the patient was started on hourly prednisolone acetate 1%, prophylactic antibiotics (moxifloxacin 0.5% four times/day), and carboxymethylcellulose 0.5% (eight times/day). Ten days after the initial presentation, a large persistent epithelial defect was noted under the AM. In view of the extensive involvement and the non-healing ocular surface, the patient was planned for a simple limbal epithelial transplantation (SLET). The surgical procedure was carried out as per the standard protocol [Postoperatively, the patient was started on a tapering dose of topical steroids  and prophylactic antibiotics (moxifloxacin 0.5% four times/day). The patient was followed up closely, and complete re-epithelialization of the corneal surface was noted within three weeks of the procedure. Six weeks following SLET, there was a formation of a superior symblepharon with fibrotic constriction, and to address this, excision of the fibrotic tissue with an AM graft was carried out. The AM was draped over the cornea and the superior bare scleral area and secured with fibrin glue. Following the surgery, the patient was given a tapering course of steroids with antibiotics similar to the regimen mentioned above.Six weeks after this procedure, the child presented with an unaided vision of 20/100. On slit-lamp examination, a 4.5 x 3 mm grayish-white lesion with ill-defined margins was noted in the subepithelial plane of the cornea in the visual axis. The surrounding cornea was uninvolved, and the SLET implants were intact. The rest of the eye was quiet  staining of the excised tissue provides an insight into its origin. The CK3 positivity and lack of CK19 staining indicate the corneal origin of the epithelium and the functioning of the SLET implants . During In the current case, the excision of the granuloma with the AM resulted in a large area of iatrogenically de-epithelialized cornea. This area healed over a course of two weeks, and the surface continued to be stable until the end of the follow-up period. This highlights the potential of the transplanted limbal stem cells to not only maintain the constant turnover of epithelial cells needed to sustain a normal corneal surface but also to re-epithelialize a damaged or denuded area. Also, these smaller islands of stem cells are able to cope with the increased demand of re-establishing a stable surface over a de-epithelialized area in a time frame similar to what was accomplished by the stem cells within a completely healthy limbus ,13.This case describes the clinical features and outcomes of an unusual case of a granuloma arising from an amniotic membrane. The patient had undergone repeated grafts of the AM, which is a potential risk factor for inciting an inflammatory response against the AM. The histopathology in the current case with mature myofibroblasts indicates the chronicity of the lesion. Although the exact origin of these cells is unclear, the special staining with positivity for vimentin and an intact collagen type III layer beneath the lesion suggests that the lesion could have originated from the AM. In our patient, there was no recurrence of the growth, and a stable, optically clear corneal surface was maintained with the limbal stem cell transplantation until the three-year follow-up period. This report thus highlights the need for awareness of such an entity in cases wherein multiple AM grafts have been carried out. By confirming the subepithelial location of the lesion with an OCT, a simple excision of the lesion can be performed, which is associated with good outcomes.


Uterine leiomyosarcoma is a rare malignant tumor that accounts for almost 2-5% of all uterine malignancies. It has the highest prevalence during pre and perimenopause. Further, it clinically resembles benign conditions like leiomyoma, and the diagnosis is confirmed with the histologic findings of the mass. Here, we present the case of a 70-year-old female who presented with lower abdominal pain for two years. Exploratory laparotomy with hysterectomy was performed, and the diagnosis of leiomyosarcoma was confirmed after histological examination of the resected tumor. Uterine leiomyosarcoma is a rare malignant tumor accounting for almost 2-5% of all uterine malignancies . The higA 70-year-old nulliparous woman presented to our hospital with complaints of lower abdominal pain in the hypogastric region, which was mild in nature and nonradiating for two years. She had her menopause 15 years back and did not have any known chronic illness. On examination, her vitals were stable; abdominal examination showed a fixed hard and nontender mass in the pelvic region of approximately 18-week size. On vaginal examination, her cervix appeared normal and the mass could not be felt separately.She was referred to our center. Ultrasonography (USG) of the abdomen and endometrial biopsy were performed at another facility before presenting to our hospital. Endometrial biopsy showed mucoid tissue mixed with inflammatory cells only. Tumor markers including cancer antigen 125  and carcinoembryonic antigen  were also tested earlier. A magnetic resonance imaging (MRI) of the abdomen and pelvis was advised, and exploratory laparotomy was planned because of the diagnostic dilemma. MRI showed a large heterogeneous signal lesion in the myometrium thinning the myometrial wall.As the diagnosis could not be confirmed using MRI, we performed an exploratory laparotomy with a total abdominal hysterectomy and bilateral pelvic lymph node sampling with omentectomy. Operative findings were of a uterus at 24-week size Figure , which wHistopathology examination of the mass Figures -5 showedThe viable area showed fascicles of tumor cells with indistinct cytoplasm. Nuclei were oval to elongated and had coarse nuclear chromatin. Nuclear pleomorphism was moderate, and mitosis was noted. These findings were consistent with leiomyosarcoma. Moreover, the omentum was unremarkable, and the right and left external iliac lymph nodes were free of tumor. The histological examination of the tumor mass confirmed the diagnosis in our case.Uterine leiomyosarcoma is a rare malignant tumor arising from the myometrium . It accoUterine leiomyosarcoma can present with vague clinical symptoms and imitate benign conditions such as leiomyoma . PatientThe pathological diagnosis of leiomyosarcoma depends upon the presence of necrosis, mitosis, and cytologic atypia in the proliferating muscle cells . AccordiHysterectomy is the preferred treatment for early-stage disease along with complete resection of the gross tumor . SystemiUterine leiomyosarcoma is a rare malignant tumor with vague symptoms that often mimic other benign uterine conditions. It is difficult to differentiate uterine leiomyoma from leiomyosarcoma before surgery as the diagnostic tools are not reliable. Thus, the diagnosis is confirmed by the histopathological examination of the tumor mass done after surgery. Hysterectomy is the preferred treatment for early-stage disease.


Hidradenocarcinomas are rare malignant sweat gland tumors that typically arise in the head and neck area. To the best of our knowledge, this is the only reported instance of hidradenocarcinoma of the abdominal wall as well as the first case arising from a region of prior trauma. A 72-year-old female presented with a left abdominal wall lesion, which she had first noticed after an injury to the area. Initially, the lesion remained stable in size, after which it became mildly pruritic, progressive in size, and expressive of a clear, non-odorous discharge. Imaging demonstrated a heterogeneous cystic density. Surgical pathology revealed a malignant dermal adnexal neoplasm composed of pleomorphic polygonal cells and focal intracytoplasmic lumina lined by eosinophilic cuticles, as well as areas of ductal differentiation, apocrine differentiation, and mucinous metaplasia. Surgical excision of the mass was performed, followed by adjuvant external beam radiotherapy (EBRT). The patient had no long-term toxicities or clinical evidence of local disease recurrence as of one year post-surgery and six months post-EBRT. Early diagnosis and treatment are essential to improving outcomes in patients with hidradenocarcinomas. Frequent follow-up is equally important, as these tumors have high recurrence rates. Hidradenocarcinomas are rare sweat gland malignancies, accounting for less than 0.0001% of tumors reported . Based oA 72-year-old female with a past medical history of hypertension, gastroesophageal reflux disease, and degenerative disc disease presented with a left abdominal wall lesion, which she had first noticed arising in an area of previous trauma from a motor vehicle collision three years prior to symptom onset. Initially, the lesion was described as asymptomatic with a cystic appearance and had remained stable in size and appearance for a prolonged period. However, three months prior to presentation, the lesion became mildly pruritic, progressive in size, and expressive of a clear, odorless discharge. The patient was referred to a general surgeon, at which point she was noted to have a left abdominal wall subcutaneous mass.Computed tomography of the abdomen and pelvis with intravenous contrast was performed, which revealed a heterogeneous cystic density in the left abdominal subcutaneous tissues measuring 4.7 x 2.5 x 2.8 cm with internal fat density, skin thickening, and retraction Figures -1C. The The patient underwent complete excision of the lesion. Upon pathological assessment, the specimen was grossly composed of a 7.5 x 4.5 cm ellipse excised to a depth of 4.7 cm, which contained a 2.6 x 2.7 x 2.0 cm firm mass with solid white components, as well as focal areas of hemorrhage, necrosis, and cavitation with a brown and bright yellow periphery. Microscopically, sections were composed of a malignant dermal adnexal neoplasm composed of pleomorphic polygonal cells with prominent nuclei, abundant clear to eosinophilic cytoplasms, and frequent mitoses. Focal intracytoplasmic lumina lined by eosinophilic cuticles were noted, as were areas of ductal differentiation, apocrine differentiation, and mucinous metaplasia. There was variable cystic degeneration and central necrosis. The surrounding stroma was sclerotic, exhibiting cancerization of sweat ducts and containing surrounding granulomatous response consistent with tumor rupture. On immunohistochemical stains, tumor cells were diffusely positive for epithelial membrane antigen (EMA), p63, and cytokeratin 7 (CK7), with patchy positivity noted for gross cystic disease fluid protein 15 (GCDFP-15), as well as focal expression of mammaglobin. Tumor cells were negative for expression of CD68, S100, CD34, cytokeratin 20 (CK20), and estrogen receptor (ER). These findings were consistent with a diagnosis of hidradenocarcinoma with apocrine differentiation. Surgical margins were clear but close (<0.1 cm) and there was no evidence of lymphovascular invasion.Three weeks following surgery, the patient underwent computed tomography of the chest, which revealed bilateral linear scarring versus atelectasis at the costophrenic angles with a 2-mm pulmonary nodule in the right upper lobe with scattered mediastinal lymph nodes in the prevascular and precarinal regions, which were <1 cm in short axis, not enlarged by size criteria. The study was otherwise unremarkable. The patient was discussed in a multidisciplinary conference, at which point positron emission tomography (PET) for completion of staging was recommended. F18-fluorodeoxyglucose PET was performed three months postoperatively and revealed the nonspecific mediastinal lymph nodes to have a maximum standardized uptake value (SUV) of 2.74, considered to be more likely reactive. The study was otherwise unremarkable.Given the aggressive histology and close margins, adjuvant external beam radiotherapy (EBRT) was recommended. She initiated EBRT 20 weeks postoperatively, with 60 Gy delivered in 30 fractions via a 3D conformal wedge-pair arrangement of right anterior oblique and left posterior oblique 10 MV photon beams with daily application of a 5-mm tissue-equivalent bolus Figures , 3A-3C. The patient was lost to follow-up for an extended period, but was contacted by telephone approximately 20 months following completion of EBRT, at which point she noted the skin of her abdomen had completely healed and had no complaints of palpable local recurrence or radiation toxicity.Hidradenocarcinomas are extremely rare sweat gland tumors that classically present as asymptomatic cutaneous lesions . They arThough hidradenocarcinomas may arise from an existing benign hidradenoma, they are much more likely to arise de novo ,5. Upon Growth of hidradenocarcinomas is variable, ranging from months to years before taking a more aggressive course including rapid enlargement and regional lymph node metastasis . DespiteThe optimal treatment for hidradenocarcinoma is unclear due to its rarity and conflicting reports of the effectiveness of adjuvant treatment . ContempGiven its poor prognosis, with five-year survival rates after surgery in the range of 30%, it is possible that an earlier diagnosis of hidradenocarcinoma may improve outcomes by allowing for adequate resection, appropriate adjuvant therapies, and prevention of metastatic disease . With raHidradenocarcinomas are rare sweat gland malignancies that typically arise in the head and neck region. Given the rarity of these tumors and conflicting reports in the literature, the optimal treatment for hidradenocarcinomas is unclear. In our patient, the hidradenocarcinoma occurred in the abdominal wall and was managed using surgical excision and EBRT. Although patient was lost to follow-up, a subsequent phone visit revealed complete healing of her abdominal skin without any recurrences or complications of radiation therapy. Early diagnosis and treatment is essential to improving outcomes in patients with hidradenocarcinomas. Frequent follow-up is equally important, as these tumors have high recurrence rates. Further studies must be conducted to determine the optimal treatment strategy for hidradenocarcinoma.


In this article, we review the multimodality imaging appearance of this material in situ longitudinally in the range of post-operative settings, in order to promote awareness of this entity when interpreting post-operative imaging. We present this as a pictorial review focusing primarily but not exclusively on the chest noting that the thoracic imaging appearance of Surgicel® is less well reported in the published literature. An understanding of this entity may help to minimize erroneous diagnosis of a postoperative complication leading to unnecessary interventions.Absorbable hemostatic agents such as Surgicel are hemostatic materials composed of an oxidized cellulose polymer used to control post-surgical bleeding and cause coagulation. This material is sometimes purposefully left  This serves as a hemostatic adjunct in the control of local hemorrhage, although the exact mechanism of action is uncertain. When this material saturates with blood, it swells into a brownish or black gelatinous mass, which aids in the formation of a clot , but becomes more concerning for infection with increasing time in the perioperative setting. usually within the first 10 days after surgery. On CT imaging, gas that escapes the gastrointestinal lumen into the mediastinum will typically collect near the surgical site, often in association with fluid, an imaging appearance that overlaps with that of Surgicel  immediately post-op, due to the presence of acute blood products, and becomes progressively lower in attenuation on CT to approach the range of complex fluid (20–40 HU) as these blood products evolve. This fluid is typically relatively homogeneous in appearance on CT and is often seen in association with a small amount of gas post-operatively, with lingering small locules of gas sometimes present up to 3 months following surgery .13 SurMagnetic resonance imaging: On MRI, Surgicel material is typically heterogeneous and hypointense to fluid on T2-weighted imaging, which differentiates it from abscess formation which is typically hyperintense to fluid on T2-weighted imaging (in situ post-operative Surgicel can be encircled by a rim that is hyperintense to muscle on non-intravenously enhanced T1-weighted imaging ( imaging . In addi imaging . There a In addition, Surgicel can induce the formation of granulomas, which can result in unnecessary surgical intervention. For example in the brain, granuloma formation associated with Surgical® has been reported to mimic tumor recurrence resulting in a subsequent surgical intervention to exclude tumor recurrence. As a result, Surgicel is typically removed from the surgical field whenever possible after hemostasis has been achieved.While Surgicel is frequently used safely in the operative setting, there have been some reports of complications secondary to use of this material. For example, the swelling and mass effect exerted by Surgicel has been reported to cause nerve compression potentially leading to paraplegia after thoracotomies.in situ to promote hemostasis in the post-operative setting. Due to similarities in imaging features of post-operative complications, Surgicel can be mistaken for potentially serious entities including post-surgical infection, anastomotic leaks and, in the brain, local recurrence of malignancy. Correlation with surgical operative notes, clinical data and knowledge of the expected imaging appearance of this material, and those of the entities with which it can be confused, can potentially prevent unnecessary intervention.In summary, Surgicel is commonly used in the surgical setting and is sometimes intentionally left


Pseudobulbar palsy (PBP) is characterized by supranuclear lesions in the corticobulbar pathway. Neoplasia, inflammatory, demyelinating, and stroke are possible etiologies of this disorder.We report an elderly female who presented with dysarthria. She was dysarthric with a hypernasal voice, no apraxia or aphasia was observed. Tongue movements were slow with limited amplitude. Her soft palate dropped bilaterally; gag reflex was present. Also, she reported swallowing difficulty and choking with her saliva. Bilateral vertical and horizontal gaze were intact to either voluntary or oculocephalic movements. A cranial CT scan was suggestive of artery of Percheron (AOP) infarction. Brain magnetic resonance imaging showed hypersignal on diffusion-weighted and T2-weighted images and hyposignal on apparent diffusion coefficient in both thalami. CT angiography scan revealed an AOP originating from the left posterior cerebral artery. The swallowing study with a videofluoroscopic demonstrated oral and pharyngeal phases with severe dysfunction.To the authors' knowledge, there are two cases of individuals with artery of Percheron infarction who developed PBP associated with other clinical syndromes. Still, isolated PBP following infarction of Percheron's artery was not reported. We hypothesized that the PBP may have occurred because of the existence of vascular territory variations in the perforating arteries that arise from the AOP. Pseudobulbar palsy (PBP) is characterized by supranuclear lesions in the corticobulbar pathway. Neoplasia, inflammatory, and demyelinating diseases are possible etiologies of this disorder. In this context, another common cause of PBP are multiple and recurrent strokes.There are only a few cases of bilateral paramedian thalamic infarction associated with acute pseudobulbar symptoms that have been reported in the literature. More specifically, to the authors' knowledge, there are two case reports of individuals with artery of Percheron (AOP) stroke who developed PBP associated with other clinical syndromes.Herein, we report a case of an adult female who developed isolated PBP secondary to infarction of AOP.A 70-year-old female presenting with a difficult and unclear articulation of speech with sudden onset was admitted to our hospital. The individual was previously healthy, retired, and her family history was negative for neurological diseases. Her vital signs  were normal and stable.On neurological examination, she was dysarthric with a hypernasal voice, no apraxia or aphasia was observed. Tongue movements were slow with limited amplitude and because of this it seemed that the tongue had difficulty moving, but when it was protruded no deviation occurred. Her soft palate dropped bilaterally; gag reflex was present. Also, she reported swallowing difficulty and choking with her saliva. Bilateral vertical and horizontal gaze were intact to either voluntary or oculocephalic movements. Palpebral oculogyric reflex was normal. Myosis, ptosis, nystagmus, and skew deviation were not present. On the National Institutes of Health Stroke Scale (NIHSS), she only scored in dysarthria, which was characterized by unintelligible slurring or out of proportion to dysphasia.Laboratory blood tests and urinalysis were within normal limits. A cranial computed tomography (CT) scan at the symptoms' onset  and two The thalamus is a complex structure formed by nuclei that integrate sensory, motor, and behavioral signals of the cerebral cortex with other pathways. Although there are significant variations and overlaps, this diencephalic large gray matter is supplied by four arteries: three derived from the vertebrobasilar system  and one derived from the posterior communicating artery (polar artery). The paramedian arteries are the most variable when we evaluate number, size, and territorial contribution to the thalamus. 5Percheron described three possible variations involving the paramedian thalamic-mesencephalic arterial supply. The most common variation is small perforating arteries arising from the first segment of the posterior cerebral artery (PCA). The second variant consists in the dependency of an isolated terminal branch nascent in the PCA for the bilateral blood supply of paramedian thalamus and rostral midbrain. Third, perforating arteries arising from a branch bridging the first segments of both PCA.6The second type, also known as the artery of Percheron (AOP), is considered a normal variant. The prevalence varies among different studies ranging from five to thirty percent in different populations. The classic clinical triad of symptoms in the infarction of the AOP is characterized by consciousness impairment, vertical gaze palsy, and cognitive or behavioral disturbances but a great variability of presentations was already described.7Only a few cases of patients with bilateral paramedian thalamic infarction who develop acute pseudobulbar palsy have been reported in the literature. We identified two cases after a review of the English-language published literature and we compared them with the present case .2,3,8 ToIn the cases of 9Any lesion in the corticobulbar tract can lead to the clinical manifestation of PBP characterized by dysarthria, dysphagia and nasal tuning speech. In this way, we hypothesized that the PBP may have occurred in the subjects of 7Karacostas et al and Lee et al proposed a different pathophysiological mechanism to this presentation.4


Strumal carcinoid is an unusual rare ovarian teratoma characterized by the presence of thyroid tissue with a carcinoid tumor. We report a case of a 60-year-old nulliparous woman, who presented with complaints of a decrease in appetite, urinary frequency, and left lower extremity edema. By ultrasound of the abdomen, a large multiloculated cystic lesion occupying almost the entire pelvis and measuring 24 x 14 x 20 cm with internal debris concerning either uterine or ovarian cystic carcinoma was seen. By MRI, it was confirmed to be an ovarian lesion. Labs revealed elevated cancer antigen 125 (CA125) of 105 U/ml and carcinoembryonic antigen (CEA) of 6.4 ng/ml. The patient underwent surgery and the intraoperative consultation confirmed teratoma with a neuroendocrine component. Grossly, it was a multicystic ovarian mass and on sectioning, it had partial solid and cystic areas with clear to mucoid fluid. Histopathology showed foci of ectopic thyroid tissue admixed with foci of well-differentiated neuroendocrine tumor, grade 1 (carcinoid) displaying insular and trabecular patterns consistent with the diagnosis of strumal carcinoid . Thyroid transcription factor-1 (TTF-1) and thyroglobulin immunostains highlighted ectopic thyroid tissue and synaptophysin highlighted neuroendocrine component. Strumal carcinoids are almost invariably benign and pathologic staging is not warranted. Treatment of strumal carcinoid is salpingo-oophorectomy. Strumal carcinoid is an ovarian teratoma accounting for 3% of all ovarian teratomas and is characterized by the presence of thyroid tissue with a carcinoid tumor. The carcinoid component is a well-differentiated neuroendocrine tumor with an excellent prognosis. Pre- and post-menopausal women have shown increased incidence. Symptoms range from a benign pelvic mass with mass-compression effects to carcinoid syndrome. The various histopathological patterns in the presentation have been a point of debate for the diagnosis. The occurrence of papillary thyroid carcinoma in the strumal carcinoid is a growing concern, hence patients should be diligently managed ,2.https://meridian.allenpress.com/aplm/article/145/9/e2/469635/Abstracts-and-Case-Studies-for-the-College-of)The abstract was previously presented as a meeting poster at the 2021 College of American Pathologists (CAP) Annual Scientific Meeting CAP21 held in Chicago on September 25, 2021, and also published in the Archives of Pathology & Laboratory Medicine, vol. 145, issue 9, September 2021.  presented with abdominal pain, loss of appetite, urinary frequency, and left lower extremity edema. Physical examination was pertinent for epigastric tenderness, palpable mass in the umbilical area, and the presence of an umbilical hernia. Abdominal ultrasound done at an outside facility revealed a large multiloculated cystic lesion occupying almost the entire pelvis measuring 24 x 14 x 20 cm containing internal debris concerning either uterine or ovarian cystic carcinoma. Magnetic resonance imaging (MRI) demonstrated a large complex cystic mass measuring approximately 29 x 14 x 21 cm with a solid enhancing internal component consistent with ovarian tumor (the imaging was done at an outside facility and only notes were available for the case report). Laboratory data were pertinent for cancer antigen 125 (CA125) of 105 U/ml (reference range <46 U/ml) and carcinoembryonic antigen (CEA) of 6.4 ng/ml (reference range < 2.5 ng/ml). The patient underwent a total abdominal hysterectomy with left salpingo-oophorectomy, omentectomy, pelvic aortic lymph node biopsy, umbilical hernia repair, and optimal peritoneal tumor debulking was performed. Intraoperative consultation on frozen confirmed teratoma with the neuroendocrine component. The gross examination of the left ovary (17 x 13 x 4 cm) revealed a partially solid (40%) and partially cystic mass (60%) containing clear, bloody, and mucoid fluid in different cysts. Histopathological examination of the left ovary showed foci of ectopic thyroid tissue with colloid-filled follicles admixed with foci of well-differentiated neuroendocrine tumor, grade 1 (carcinoid) consistent with the diagnosis of strumal carcinoid   the thyroid parenchyma displayed nuclear positivity by expression of thyroid transcription factor-1 (TTF-1) Figure . CytoplaStrumal carcinoid is the most common form of monodermal teratomas arising in the ovary. Nearly 3% of teratomas account for strumal carcinoid type which is an admixture of benign thyroid tissue and carcinoid. Strumal carcinoids can be pure and sometimes in association with mature teratomas, granulosa tumors, and mucinous cystadenomas of the ovary ,2. TheseThe histogenesis of its origin is being debated as it is mostly considered as a component of the teratoma as a germ cell origin. The carcinoid component is native to the ovary and less likely to be a metastatic component from the gastrointestinal tract -8. ThereThe primary strumal carcinoid of the ovary is mostly unilateral ,7. GrossThe differential diagnosis for strumal carcinoid can include granulosa cell tumor (microfollicular), benign Brenner tumor, metastatic carcinoid to the ovary, or metastatic thyroid carcinoma to the ovary ,8,12, anStrumal carcinoids are mostly confined to the ovary, are indolent, and resolve by surgical treatment by salpingo-oophorectomy ,8,11,12.Strumal carcinoids are rare but the most common variant of monodermal teratoma which can arise in the ovary. Benign thyroid tissue and well-differentiated neuroendocrine tumor are admixed with various architectural patterns and stromal hyalinization. Strumal carcinoids are unilateral benign tumors and pathologic staging is not warranted. Surgery is the common mode of treatment for these patients. It is important to identify these tumors, especially during intraoperative frozen sections to avoid extensive surgery and staging.


Intrathoracic schwannomas are rare and difficult to diagnose. However, they are the most common type of neurogenic tumor in the chest. Most patients are incidentally diagnosed or develop symptoms from mass effect, such as chest pain, dysphagia or dyspnea. Larger tumors have been resected using open approaches, while smaller ones are often excised with minimally invasive approaches.A 60-year-old woman with a prior Roux-en-Y gastric bypass and a history of dysphagia, decreased appetite, and weight loss was referred for evaluation. CT chest revealed an 8 cm soft tissue mass centered in the distal esophagus. Gastroscopy showed the tumor to be 8 cm as well, with 2 cm of normal esophagus prior to the gastric pouch. A right-sided video-assisted thoracoscopic (VATS) approach for enucleation was successfully completed with primary esophageal repair for an 8.0 × 5.5 × 6.5 cm schwannoma.Surgical resection for schwannomas is often indicated due to symptoms from mass effect . There are reports of VATS and robotic-assisted thoracic surgery approaches for small tumors. These techniques are appealing due to shorter length of stays and less post-operative pain. None have been described for lesions larger than 6 cm.Minimally invasive approaches such as VATS for large schwannomas are technically feasible and safe to perform without the need for a thoracotomy. •Intrathoracic schwannomas are the most common type of neurogenic tumor in the chest.•Schwannomas resected using video-assisted thoracoscopic surgery (VATS) are rare.•Smaller esophageal schwannomas (<6 cm) have previously been approached using VATS.•VATS can also be technically feasible and safe for larger lesions (8 cm). She was investigated with a chest x-ray  and echoOn gastroscopy, the tumor was identified starting at 30 cm extending to 38 cm . There wSurgical resection was planned by two experienced thoracic surgeons. The procedure started with right-sided VATS approach for enucleation of the tumor knowing that a thoracotomy may be needed, and possibly esophageal resection given the large size and location . As the The post-operative course was complicated by a small and contained leak that required no intervention. Pathologic evaluation of the specimen revealed a schwannoma (8.0 × 5.5 × 6.5 cm). A gastroscopy was performed 8 weeks postoperatively . This de3Esophageal schwannomas are rare neurogenic tumors This was a particularly challenging case given that the patient had a prior Roux-en-Y gastric bypass, limiting our surgical options. We offered a right-sided VATS with segmental resection. We did discuss that this option may not be feasible given the size of the mass and our ability to manipulate it. In that case, we would convert to a laparotomy and right-sided thoracotomy to complete an Ivor-Lewis esophagectomy. Given the size and location of the mass, our biggest challenge during the case was retracting the mass posteriorly. It became close to the spine, making our dissection around the inferior pulmonary ligament and the inferior pulmonary vein difficult. This required us to rotate the esophagus and complete the dissection from the contralateral side.4In this case report, we discuss a successful VATS resection approach for a large distal esophageal schwannoma invading the submucosa. We demonstrate that minimally invasive approaches for larger schwannomas are also feasible.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not commissioned, externally peer-reviewed.This study is exempt from ethical approval from our institution.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Dr. Sayf Gazala is the guarantor of this study.Not applicable.SG and CM performed the surgery. UK prepared the manuscript. SG, CM, NF reviewed and edited the manuscript for final submission.There are no conflicts of interest.


Lipomas are benign tumors composed of mature adipocytes. A 1-year-old-male baby was admitted to the hospital with a history of an abdominal mass, which had been incidentally detected by a local pediatrician. upon examining the child for a complaint of distention and chronic diarrhea. An ultrasound and computed tomography scan were performed, and findings revealed a fat mass, which was confirmed by laparotomy and microscopic results. At laparotomy, a soft yellow mass was found attached to the mesentery of the jejunum. The mass was enucleated without anastomosis, and the patient was followed up 4 months postoperatively with regular clinical examination and abdominal ultrasonography. This case was examined and reported in this study because lipomas are very rarely present in the mesentery of the intestine, especially at this early age. Lipomas may be found anywhere in a child’s body but rarely so in the mesentery of the intestine. To date, fewer than 50 cases of mesenteric lipoma have been reported in the literature published in English, with nearly a half occurring in children .Lipomas are usually asymptomatic until they grow large enough to cause symptoms, such as pain or constipation due to partial or complete obstruction or volvulus. Although, with relation to the all said cases, no malignant tumors are reported, the preferred treatment for lipoma is complete resection with or without bowel resection . LaparotA 1-year-old-male baby was presented to the hospital with a history of an abdominal mass. A local pediatrician in the outpatient clinic incidentally discovered it when he was examining the child for a complaint of distention and chronic diarrhea.The child was referred to the hospital and was admitted to the Pediatric Surgery Department. Physical examination indicated the mass was painless and mobile. The palpable mass was nontender and extended from the upper left quadrant to the umbilicus. Vital signs were normal, and the child was asymptomatic with a good appetite. He did not complain of vomiting, constipation or abdominal pain. His weight at admission was 9 kg. Laboratory tests were normal. Abdominal ultrasound showed a hyperechoic soft tissue lesion with a maximum diameter of ~10 cm, pushing the intestine to the right without obstruction of the intestinal lumen.Computed tomography (CT) of the abdomen revealed a homogeneous, well vascularized mass measuring 9.4 × 10.6 cm and encapsulated with thin septation in its medial part. The mass was located in the upper left quadrant and extended to the pelvis with an oval appearance . Other aLipoma is a fatty tissue composed of mature cells and it has a benign behavior. It is more common in adults aged 40–60 than in children, and it rarely occurs in children under 10 years of age . The lipUntil now, the causes of lipoma are not clear but some factors have been reported to be predisposing to the growth, such as diabetes mellitus, hypercholesterolemia, obesity, familial tendency, radiotherapy, chromosomal translocation and trauma , 6. In oLipomas are slow growing and they do not penetrate the surrounding organs, but some may cause symptoms when the tumor grows too large and compresses the adjacent organs , 7. The Ultrasound imaging can be used to distinguish lipomas from mesenteric cysts. Not to mention that it is better to perform an abdominal CT scan to distinguish lipoma from other echogenic tumors , 8. The Depending on its size, site and the presence of complications, complete excision of the lipoma alone or with the adjacent bowel is the performed treatment [10]. In our case, enucleation of the lipoma was performed without excision or injury to the adjacent intestine, and the appearance of the adjacent bowel and blood supply were normal.Lipoma is a benign mass that is very rarely found in the mesentery of the jejunum. Histopathology is the definitive diagnosis, and even though the mass is asymptomatic, complete excision is the preferred option.None declared.None.


Cystic partially differentiated nephroblastoma is a multilocular cystic variant of Wilms tumor that always presents in children. However, we encountered an elderly patient with cystic partially differentiated nephroblastoma. Therefore, we report it.A 74‐year‐old male presented with a left renal tumor detected with ultrasonography. Contrast‐enhanced computed tomography and magnetic resonance imaging revealed a 4 cm multilocular cystic tumor with septa, which suggested multilocular cystic renal cell carcinoma. Therefore, we performed a radical nephrectomy. The definitive diagnosis of cystic partially differentiated nephroblastoma was made with histopathological findings. After the surgical resection, no recurrence has occurred in the past 13 years.Cystic partially differentiated nephroblastoma can develop in adults, regardless of age. Furthermore, surgical resection can be used as an established treatment option in adult cystic partially differentiated nephroblastoma cases. CNcystic nephromaCPDNcystic partially differentiated nephroblastomaCTcontrast‐enhanced computed tomographyMCRCCmultilocular cystic renal cell carcinomaMRImagnetic resonance imagingWT1Wilms tumor 1Cystic partially differentiated nephroblastoma could develop in adults, regardless of age. Histopathological examination is needed for a definitive diagnosis since preoperative discrimination is difficult. Surgical resection can be used as an established treatment option in adult cases, as well as in children.CPDN is a rare kidney tumor classified as a multilocular cystic variant of Wilms tumor. Though CPDN primarily presents in children,r. Ultrasonography revealed an appearance of 4 cm multilocular cystic tumor with septa in the middle portion of left kidney (Fig. A 74‐year‐old male presented with asymptomatic left renal tumor. He underwent ultrasonography every year for postoperative surveillance of gastric canceney Fig. . Blood aney Fig. . MRI revney Fig. . Though ney Fig. . Histopaney Fig. . Immunopney Fig. . These rCPDN is a multicystic renal tumor classified as a rare variant of Wilms tumor with unique clinical characteristics.The differential diagnoses of renal multicystic tumors in adults are MCRCC, CN, and mixed epithelial and stromal tumors of the kidney.However, immunohistological criteria for the diagnosis of CPDN have not been defined yet. In the adult CPDN cases, only one case was positive for WT1 protein, which is positive in most cases of Wilms tumor.in children is surgical resection.The standard treatment for CPDN No recurrence occurred after surgery in at least three out of the five adult CPDN cases Table , suggestWe encountered the oldest adult patient with CPDN. CPDN can develop in adults, regardless of age. Since preoperative discrimination of CPDN from other multicystic tumors is difficult, histopathological examination is needed for a definitive diagnosis. Though immunohistological criteria for the diagnosis of CPDN has not been developed yet, immunohistological examination could contribute to confirm a diagnosis of CPDN. Surgical resection can treat CPDN in adults, as demonstrated in the present case where no recurrence was observed for 13 years after the operation. Complete resection should be accomplished to prevent recurrence.The authors declare no conflict of interest.Not applicable.Written informed consent was obtained.Not applicable.


Two patients came to the emergency department with sudden-onset abdominal pain. The first case was a 20-year-old male; a contrast-enhanced computed tomography (CT) scan revealed a 17 cm x 7 cm hematoma in the abdomen and left retroperitoneal space. Furthermore, the left kidney was not visualized by CT. With an emergent diagnostic laparotomy, the etiology of the bleeding was determined to be left atrophic-hydronephrotic kidney rupture. Thus, a left nephrectomy was performed. The second case was a 38-year-old male with a history of chronic hypertension; a CT scan revealed a 7 cm x 6 cm left perirenal hematoma. A left nephrectomy was performed due to hemodynamic instability on the second day of follow-up. A pathology specimen revealed a 1-cm renal cell carcinoma (RCC) in the kidney. In case of hemodynamic instability, spontaneous kidney rupture requires immediate surgical intervention; its causes include atrophic-hydronephrotic kidney and RCC. Spontaneous kidney rupture (SKR) is a rare urological emergency that can be lethal . It can Case 1A 20-year-old male patient with no history of trauma came to the emergency department with sudden onset abdominal pain. He reported a feeling of bursting and severe pain in the abdomen during weight training. The physical examination was compatible with acute abdomen findings. His intestinal sounds and rectal examination were normal. However, he had hypotension and tachycardia. The patient's urine analysis and serum creatinine level were normal. His hemoglobin level was 10.1 g/L (range 13-17.5 g/L). An abdominal contrast-enhanced computed tomography (CT) scan revealed a 17 cm x 7 cm hematoma containing active bleeding foci in the abdomen and left retroperitoneal space. Also, the left kidney was not visualized Figures , 1B. TheWhen the nephrectomy material was examined, fibrotic stenosis was observed at the ureteropelvic junction. In addition, the pathological analysis showed fibrosis in the ureteropelvic junction and chronic pyelonephritic changes in the renal parenchyma. The patient was stabilized after surgery and, on the third postoperative day, he was discharged.Case 2A 38-year-old male patient came to the emergency department with sudden-onset widespread abdominal pain. Physical examination of the patient, who had no history of trauma, was compatible with acute abdomen. He had chronic renal disease and hypertension in his medical history. However, the patient, who did not use any antihypertensive medication, had not had regular nephrology follow-ups. On admission, his blood pressure was 200/140, his creatinine level was 2.55 mg/dL (range 0.7-1.2 mg/dL), and his hemoglobin level was 8.5 g/L. An abdominal non-contrast CT scan revealed a 7 cm x 6 cm left perirenal hematoma extending towards the posteroinferior of the kidney; however, no pathological condition that would cause bleeding in the kidney could be detected on the CT scan Figures , 2B. He In addition, pathological analysis showed total glomerular sclerosis in the left kidney consistent with end-stage renal failure. Moreover, multiple cortical adenomas and cortical cysts were found in the kidney as well as a 1-cm papillary RCC in the upper pole. After his hemoglobin level stabilized after surgery, the patient was discharged with a serum creatinine level of 2.71 mg/dL on the fifth postoperative day.In this paper, we reported two SKR cases that presented with unusual symptoms. It is known that SKR can present with abdominal or flank pain, hematuria, or hypotensive shock similar to renal injuries due to trauma . The firAtrophic and hydronephrotic kidneys are enlarged enough to cover almost the entire abdomen in cases of severe hydronephrosis . BecauseSpontaneous rupture of the kidney is a rare entity that usually occurs in patients with RCC, angiomyolipoma, renal cysts, arteriovenous malformation, or vascular diseases such as periarteritis nodosa ,9. In thSKR is a rare urological emergency that threatens life when appropriate treatment is not done. However, these cases may present with unusual symptoms such as sudden onset abdominal pain like in our report. It should be kept in mind that SKR can be seen in patients with an atrophic-hydronephrotic kidney or chronic renal failure where RCC, cortical adenoma and cysts may coexist.


Giant cell tumor of soft tissue (GCT-ST), histologically resembling the GCT of the bone, is a rare tumor. The tumor has been categorized to have low malignancy. Few reports of local recurrence or distant metastasis and the use of chemotherapeutic agents for metastatic GCT-ST exist. Herein, we report the efficacy of pazopanib in a 78-year-old Japanese woman with GCT in the intrinsic back musculature with both post-operative local recurrence and lung metastasis. The patient visited the hospital with a three-month history of a palpable mass in the intrinsic back musculature. Following magnetic resonance imaging, the tumor predominantly exhibited slight hyperintensity on T2-weighted images and intense heterogeneous enhancement on contrast-enhanced T1-weighted images. A percutaneous needle biopsy was performed, and the pathological diagnosis was GCT-ST. The patient underwent surgery, and three months later she presented with not only local recurrence but also multiple lung metastases. The patient was immediately treated with pazopanib 400 mg once daily. One month after initiating treatment, a partial response in the pulmonary lesions was observed, and stable disease (SD) effects lasted for 11 months without severe adverse effects. Therefore, pazopanib treatment for metastatic malignant giant cell tumor of soft tissue achieved reasonable success. Giant cell tumor of soft tissue (GCT-ST) is a rare cancer that was first described by Salm and Sissons in 1972 as a distinct entity . This tuFurthermore, there are no previous reports about the effect of pazopanib treatment in elderly patients with metastatic GCT-ST. To the best of our knowledge, this is the first case report to present the efficacy of pazopanib in a patient with metastatic GCT-ST.A 78-year-old Japanese woman visited our department complaining of an enlarged mass localized in the intrinsic back musculature, which had been present for approximately three months. There was no tenderness or fever. Furthermore, no weight loss was noted during the initial visit, and her family history was unremarkable.A local CT scan revealed a 32 cm × 24 cm × 41 cm lesion superficially located in the intrinsic back musculature .Moreover, on T2-weighted MRI images, the tumor appeared as a heterogeneous mass, whose main component was solid, mixed with a small portion of varied-sized cysts .Following these observations, a core needle biopsy was performed, and the pathological diagnosis indicated a low-grade sarcoma. Based on this conclusion, wide resection of the tumor was performed. The resected specimen was approximately 45 mm × 25 mm × 40 mm in size, highly vascular, focally hemorrhagic, and adherent to fat tissue A.After careful separation, the tumor was macroscopically excised completely; however, histological examination revealed incomplete resection (R1 resection), many blood-filled cavities similar to those present in aneurysmal bone cysts, a mixture of mononuclear cells and multinucleated osteoclast-like giant cells with high mitotic activity (38 figures per 10 HPFs), and no notable nuclear atypia B,C. BaseBased on R1 resection, the additional wide resection was considered as the standard treatment. However, based on the strategy of demarcating surgical margins for bone and soft tissue sarcoma, it could be predicted that a large defect of the back would occur upon the removal of the tissues including vertebrae and ribs . It woulThe first follow-up was three months after the surgery, and an MRI scan  was perfUnfortunately, the tumor had recurred and was located at the surgical site. The pathological diagnosis indicated a low-grade GCT-ST after a second needle biopsy, and positron emission tomography–computed tomography (PET-CT) was conducted to investigate the metastasis A–D.®) regimen of 400 mg, once daily, was initiated to treat the metastatic GCT-ST. Regression of lung metastases was observed during the first month of treatment with pazopanib. Based on the Response Evaluation Criteria in Solid Tumors (RECIST) principles, treatment efficacy was evaluated as a partial response  and deep 30%) soft tissues ,3. Surgi0% soft t0% and dePositron emission tomography (PET)/CT with Fluorodeoxyglucose (FDG) is a crucial non-invasive diagnostic tool for the management of patients with soft tissue sarcomas . Previou®), a tyrosine kinase inhibitor that targets the vascular endothelial growth factor receptor, is a second-line treatment option for advanced and metastatic STS after treatment with anthracyclines fails [Pazopanib ) study reported a median overall survival (OS) of 11.2 months following treatment initiation (12.5 months in the pazopanib group). Nakamura et al. indicated that good performance status and being female were favorable prognostic factors for OS . In the Radiotherapy (RT) is an effective adjuvant therapy administered after surgery for adult STS to prevent local recurrence . AccordiWe report the case of a patient who presented with primary malignant GCT-ST located in the intrinsic back musculature with post-operative local recurrence and lung metastases. Post-operatively, a close follow-up is essential because of the malignant potential of the tumor, leading to not only local recurrence but also distant metastases. In the present case, pazopanib treatment was effective, as previously reported, and its efficacy lasted for 11 months. However, detailed data regarding the efficacy of pazopanib in the treatment of metastatic malignant GCT-ST are scarce, thus marking the significance of publishing such data. Our decision to choose pazopanib as first-line treatment for this patient may be controversial because the study by Grünwald et al. did not include patients with metastatic malignant GCT-ST. In general, evidence for this rare entity is very limited. Therefore, further studies are required to conclusively verify the efficacy of pazopanib in patients with metastatic malignant GCT-ST.


It should be kept in mind that non‐Hodgkin lymphoma may involve uterine cervix and a multidisciplinary approach should be adopted. It should be kept in mind that non‐Hodgkin lymphoma may involve uterine cervix and a multidisciplinary approach should be adopted.  When diagnosed, it can be confused with malignancies of the cervix uteri. A 40‐year‐old female patient who had presented with postcoital bleeding, dyspareunia, and vaginal discharge with a foul odor, preliminarily diagnosed with a cervical malignant mass, was diagnosed as non‐Hodgkin lymphoma with cervix uteri infiltration as a result of the pathological examination. PET‐CT revealed pathological involvement, and the medical oncology department administered chemotherapy. Cure was achieved in the patient with chemotherapy. In rare cases presenting with cervical uteri cancer, it should be considered that Non‐Hodgkin lymphoma may have systemic metastasis involving the cervix.Non‐Hodgkin lymphoma is one of the most common cancer types diagnosed in men and women in developed countries.Genital system involvement of non‐Hodgkin lymphoma can be seen in two ways. The first is localized lymphoma originating from the primary genital organ, and the second is genital organ involvement of systemic lymphoma, which is more common.Primary extranodal involvement of non‐Hodgkin lymphoma is rare. While the rate of primary extranodal disease is nearly 30% in Europe, it has been determined as 22%‐25% in America. Based on differences in geographical regions, this rate increases to nearly 50% in Middle Eastern and Asian countries.2After a mass was detected in the gynecological examination of the 40‐year‐old female patient who had presented with bleeding after sexual intercourse, dyspareunia, and watery vaginal discharge, and who had a history of 3 healthy normal vaginal births, a specimen for biopsy was obtained from the mass. The pathology was reported as cervical squamous type nonkeratinizing carcinoma, and the patient was referred to our tertiary hospital for advanced tests and treatment. The patient had no complaints other than postcoital bleeding, dyspareunia, and watery vaginal discharge with a foul odor. The gynecological examination revealed a normal vulva and vagina consistent with age; however, speculum examination revealed a massive lesion approximately 2 cm in size with irregular borders and fragile bleeding outside the transformation zone on the upper pole of the uterine cervix. The uterus was normal on ultrasound examination, and the ovaries and tubas had normal anatomical structure bilaterally. However, transvaginal ultrasonography revealed a normal uterine canal and a massive lesion of approximately 16 × 17 mm with irregular borders on the anterior surface of the uterine cervix that did not extend into the endocervical canal. A colposcopic examination was performed to obtain biopsy samples. The biopsies that had been carried out from the uterine cervical mass in another center also underwent the consultation of our hospital's pathology department. As a result of the pathological examination, CD 20‐positive high‐grade B‐cell lymphoma infiltration was determined. In the immunochemical examination of the preparations, the neoplastic cells were CD20, LCA and vimentin‐positive, and CD3, CEA, P63, CD56, and Pan‐CK negative  cells that affect the lymphoid, hematopoietic, and other extranodal systems, arising from precursor or mature cells.In the study conducted by the International Workshop Group (IWG) in 2007, after analyzing the response criteria, FDG‐PET became the standard method of evaluating the response in both Hodgkin and non‐Hodgkin lymphomas.In final words, unnecessary genital organ losses can be prevented with multidisciplinary efforts in systemic lymphomas that mimic malignancies of the genital system and involve the female genital system and cause gynecological complaints. Furthermore, most importantly, it is seen that the best results can be obtained by making the definitive diagnosis promptly and starting treatment.None declared.ÖB and MSB: wrote the manuscript. CK, CD, AB, and TS: contributed to clinical follow‐up. TS: revised the manuscript.None declared.Informed consent was obtained from the patient to publish the case.


Pseudomonas aeruginosa. Endoscopic sinus surgical mass excision was performed, and treatment with oral cefuroxime was administered. The patient recovered fully. A CB mucocele, though rare, should be considered in the differential diagnosis of an intranasal mass.Concha bullosa (CB) is a common sinonasal anatomic variant. The obstruction of a CB, though rare, might result in mucocele that may be misdiagnosed. In this report, we present a case of a 32-year-old female with a one-year history of unilateral nasal obstruction, headache, facial pain, foul nasal discharge, and hyposmia, initially misdiagnosed as a neoplasm. Computed tomography (CT) and magnetic resonance imaging (MRI) revealed a right middle CB infection with a mucocele. Laboratory cultures yielded  Concha bullosa (CB) is a unilateral or bilateral pneumatization of the nasal conchae and a common sinonasal anatomical variant with prevalence rates of 14%-53% . CB occuA 32-year-old Saudi female was referred to King Abdulaziz Medical City in Jeddah (KAMC-J) as a case of inverted papilloma for further evaluation. The patient complained of right-sided nasal obstruction for a duration of one year and headache, facial pain, nasal discharge, and hyposmia. Endoscopic examination identified a right-side nasal polyp; it appeared to arise from the lateral nasal wall, and it occupied the middle meats, while the left side was clear and patent. CT scan done in the referring hospital revealed a nasal mass extending from the lateral nasal wall to the maxillary sinus, displacing the lamina papyracea. CT of the sinuses was not done in KAMC-J because the patient was pregnant at that time.However, a biopsy of the right nasal mass was consistent with an inflammatory sinonasal polyp. After delivery, CT of the sinuses showed an expansile right nasal cavity mass with septal displacement to the left. Brain and neck magnetic resonance imaging (MRI) revealed a right maxillary sinus expansile mass with iso-to-high signal intensity in T1 and heterogeneous signal intensity in T2. The mass affected the adjacent structures, shifting the nasal septum to the left and the medial wall of the right maxillary sinus to the right, suggestive of a right middle CB infection with a mucocele  with septoplasty was scheduled. Preoperative CT of the sinuses revealed a mild interval reduction in the size of the right nasal cavity lesion . At the three-month post-operative follow-up visit, the patient had no symptoms, and nasal cavity examination revealed patent nasal passages with no synechiae.Histopathology reported CB with mucoid material, fibrin, and admixed bacterial morphology. The culture was positive for P. aeruginosa is made and treated by endoscopic surgery and antibiotics, followed by a full patient recovery. The patient’s presenting symptoms of right-sided nasal obstruction, headache, facial pain, nasal discharge, and hyposmia are the effects of an obstruction in the mucociliary transport system causing mucoceles of the paranasal sinuses or CB. This obstruction may occur from chronic rhinosinusitis, nasal polyps, facial trauma, or previous sinus surgery, resulting in the accumulation of secretions and an expanding mass [This study presents a case of chronic sinusitis due to maxillary sinus obstruction and polyps in an adult. After an initial misdiagnosis, a definitive diagnosis of a CB mucocele infected with ing mass , which iing mass .Mucoceles are most common in the frontoethmoidal region, maxillary sinus, sphenoid sinus, or posterior ethmoid in adults, and in 90% of cases, they are unilateral. However, mucoceles (or pyoceles) are mostly seen in the paranasal sinus, not in the CB . In the CB mucocele may present as a large nasal mass surrounded by a thin bone plate and covered with intact mucosa. CB mucoceles have more surrounding space than the mucoceles within maxillary, ethmoid, or frontal sinuses, which allows them to expand to large sizes before being detected. This expansion displaces the septum to the other side and obliterates the maxillary, ethmoid, and frontal sinus. Anterior rhinoscopy may reveal a large mass filling nasal cavity that is covered with smooth intact respiratory mucosa. The diagnosis is based mainly on the radiological characteristics of CT and MRI scans.Despite the presenting symptoms, the case was initially diagnosed as an inverted papilloma by CT; however, further evaluation by MRI reveals the final diagnosis of a CB mucocele. Because mucoceles may be misdiagnosed as tumors, this differential diagnosis should be considered in the assessment of an intranasal mass. The possibility of a polyp, papilloma, or tumor in a unilateral mass of the nasal cavity should be explored . A diagnP. aeruginosa. This is consistent with reports in two previous cases in Turkey in 2013 [Staphylococcus aureus [In this case report, the microbiological investigation of the infected CB reveals  in 2013 . Howevers aureus . A CB muThis case, an infected CB with a mucocele, is a rare condition that may occur without any predisposing factors. Therefore, a CB mucocele should be considered as a differential diagnosis of any slow-growing mass when a definitive diagnosis cannot be made without complementary imaging and endoscopic surgery, which are the main diagnostic and therapeutic management approaches in this case, respectively.