Infantile fibrosarcoma (IFS) is a rare nonrhabdomyosarcoma soft tissue tumor and accounts for less than 1% of childhood cancers. Forty per cent are present at birth and only 10% of IFS occurs in the abdomen. Our case of neonatal fibrosarcoma presented as a distal small bowel stenosis complicated with meconium peritonitis. The diagnosis was by histology of the surgical resection. The diagnosis of IFS is challenging as there are no specific features of IFS on imaging. Any unexpected solid lesion should raise the suspicion of complicated bowel tumoral lesion. If a neoplastic lesion is suspected extensive, surgery may be postponed until the final diagnosis is made. Infantile fibrosarcoma (IFS) is a very rare condition occurring in approximately five new cases per 1 million children.A child of unrelated parents was born at 37 gestation weeks (GW) by Caesarean section . At 30 GW, fetal ultrasound (US) showed peritoneal ascites with septations and an echogenic mass adjacent to a right dilated bowel loop with an echogenic content . AntenaThe prenatal investigation of the amniotic fluid was normal (karyotype and digestives enzymes) and cystic fibrosis was ruled out. At birth, the abdomen was distended. Contrast enema demonstrated a micro colon. The US revealed echogenic ascites with septations, a large epigastric dilated digestive loop with echogenic content, surrounded by a large, hyperechoic mass, with slight vascularization on the Doppler. At surgery, MP with a small bowel stenosis  was confirmed along with a soft tissue mass adjacent to the liver and the posterior peritoneum. Taken together, these observations suggested the presence of a meconium cyst. The mass was resected with difficulties related to the close relation with the liver, and anastomosis was performed. Due to extensive adhesiolysis, the resection was complicated by a haemorrhagic shock and required a cardiopulmonary resuscitation . Due to extensive adhesiolysis, the resection was complicated by a hemorrhagic shock, that was characterized by a hemoglobin level lower than 8 g/dL, and below normal coagulation factor levels  that barely reached 30% of the expected values. We performed a cardiopulmonary resuscitation that lasted 37 minutes and required the injection of 10 adrenaline doses and the transfusion of 100 mL/kg of packed red blood cells (PRCB), platelets, and 2 units of fresh frozen plasma (FFP). Despite medical treatment, including the administration of albumin and vasoactive drugs, the noncontrolled bleeding persisted, requiring a second surgery to place a perihepatic packing. It was removed 5 days later.The histopathologic examination revealed a dense cellular tumor in the ileal intestinal wall with spindle-shaped cells in clusters from submucosa to subserosa. The tumor cells had oval nuclei and blurred cytoplasmic limits with high mitotic activity and frequent apoptosis. They were arranged in interlacing fascicles following a characteristic herringbone pattern. The blood vessels were sheathed in the tumor and their walls were thickened. Moreover, inside the segment named “meconium cyst,” there was a lumen which was full of meconium and bordered by an intestine mucosa infiltrated with tumor cells meaning that the “cyst” was a part of the tumor .i-67. Cytogenetic examination with fluorescence in situ hybridization (FISH) analysis did not confirmed a rearrangement of the ETV6 region, and an translocation of genetic material between theETV6gene located on the short arm of chromosome 12 at position p13. and theNTRK3gene located on the long arm of chromosome 15 at position q25.3 to create the  fusion gene, (ETV6-NTRK3) fusion transcript was not detected using Reverse Transcriptase - Polymerase Chain Reaction (RT-PCR).Immunohistochemistry showed that tumor cells only expressed vimentin, to the exclusion of markers like desmin, catenin, and transmembrane phosphoglycoprotein protein encoded by the CD34 gene (cluster of differenciation 34 founded in hematopoietic cells) (CD34). Many nuclei expressed KBecause untreatable and fatal neurological sequelae were diagnosed, secondary to the hemorrhagic shock, palliative care was provided to the patient who died at 63 days of life.The interest of this case lies in its location and its diagnostic timeline.In 71% of the cases, IFS develops in the limbs. Nevertheless, several ectopic cases were observed, particularly in the trunkWhen IFS is located in the abdomen, differential diagnosis, such as all intra-abdominal tumors, any remainder malignant mesenchymal tumors or sarcomas,On imaging IFS have nonspecific features. On US, they usually appear as a heterogeneous, well-defined soft tissue single mass,In our case, the MP associated to a distal bowel atresia was misleading and the mesenteric lesion visualized on imaging was not taken into consideration at first, despite the Doppler showed a slightly increased vascularization but without preferential location and an heterogeneous mass which evoked a solid lesion. Fetal MRI was not helpful and postnatal MRI was not performed. Perhaps additional MRI should be performed when the diagnosis is ambiguous which was not done.Pathological analyses of IFS show a tumoral lesion with regular walls, made of layers of closely packed spindle-shaped cells arranged in bundles and fascicles, resulting in a herringbone pattern. The mitotic activity is high and the nucleus is hyperchromatic. The vascularization is rich, and it is possible to find hemorrhagic, necrotic, or inflamed areas.The ETV6-NTRK3 fusion transcript (FT) associated with the t translocation is a good biomarker for diagnosis, and allowed us to differentiate IFS from adult fibrosarcoma, infantile fibromatosis, and other tumors with spindle cells.The genetic tests did not reveal the presence of an ETV6-NTRK3 fusion transcript, which doesn't mean it was absent because this test has a 9% false negative rateThe main treatment of neonatal IFS is the surgery with resection with as wide margins as possible, except if there is a risk of mutilation, in which case chemotherapy is used first. IFS indeed is a chemosensitive tumor that can be treated by the association vincristine-actinomycin (VA), a combination that demonstrated its efficacy with a response rate of 71%.The global survival rate varies between 80 to 100%.In our case, no alternative therapeutic could be considered because we never thought about a differential tumoral diagnosis. During the surgery, as the tumoral diagnosis never mentioned, the surgeon thought removing a cyst, usual complication about meconial peritonitis; thus, the surgery is the only alternative and performing chemotherapy was not an option. Maybe we found in this example, a reason of the wait and see approach before 3 months, because of the dramatic outcomes of the surgery while the tumoral evolution is not aggressive. However, the surgery was required because of the bowel stenosis. After surgery, chemotherapy was not an option since the cerebral lesions were too importantCystic meconium peritonitis (CMP) is a result of in-utero bowel perforation. It will lead to secondary inflammation to the peritoneal cavity resulting in fibrosis, calcification, and cyst formation. Difficult surgery is expected as a result of the fibroadhesion and inflammation. Any unexpected solid lesion should raise the suspicion of complicated bowel tumoral lesion. If a neoplastic lesion is suspected extensive surgery may be postponed until the final diagnosis is made


Dirofilaria repens infection presenting as peripheral lung nodules and mimicking a metastatic focus from a previously diagnosed cutaneous melanoma. To avoid invasive investigations before arriving at the correct diagnosis, dirofilariasis should be included as a part of the diagnostic process in subjects with lung nodules who live in (or have traveled to) endemic regions.We describe a rare case of  A 55-year old Italian woman with a history of cutaneous melanoma was admitted to the Thoracic Surgery Unit of Policlinico Umberto I due to the appearance of pulmonary nodules on a positron emission tomography/computed tomography follow-up exam , B.cox1 gene fragment (about 650 bp) was amplified using filarioid-generic primers [Dirofilaria repens, deposited in GenBank (Accession Number DQ358814.1).Physical examination and serum chemistry were normal. She was afebrile and in good condition. Due to the high suspicion of lung metastasis from the previously diagnosed melanoma, she underwent wedge resection in the right lower lobe. Gross examination of the surgical specimen revealed a nodular lesion of soft consistency with areas of necrosis, and histology confirmed extensive necrosis surrounded by chronic inflammatory reaction. The main finding consisted of the presence of worms embedded in the necrotic material, showing a thick cuticle and internal organs and exhibiting morphological features of a filarioid parasite , B. For Dirofilaria nor any other parasite was found in the blood.The patient had been born and was living in an urban area of Central Italy (Civitavecchia). Questioned on her habits, she reported recent travels to Northeast Italy and every year, in August, to a rural area in Bosnia Herzegovina. During these periodic trips, she recalled being frequently bitten by mosquitoes. Furthermore, the patient’s dog was analyzed by a veterinarian, and neither The patient was not given anti-infective therapy. At 3-month follow-up, physical examination and blood analyses were normal.Dirofilaria immitis and D. repens. The first has a worldwide distribution, and it mainly causes benign pulmonary nodules. D. repens is limited to the Old World, causing subcutaneous nodules and intraocular infections [Dirofilariasis is a mosquito-borne parasitosis caused by fections .Dirofilaria spp. is being increasingly detected in subcutaneous and/or ocular tissues and in asymptomatic, abortive internal sites of infection, that is, pulmonary nodules, with the clinical implication that these lesions are initially misidentified as malignancies, thus requiring invasive investigations before the correct diagnosis, which is mainly made during histopathology [These parasites circulate naturally between mosquitoes and canids, with humans as accidental hosts. athology .D. repens human infection, as they are typically caused by D. immitis.In the present case, the diagnosis was made after surgical excision of a nodule suspected for being lung metastasis, occurring several years after the melanoma finding. As for anamnesis, the patient reported several travels to endemic areas, including Northeast Italy and Eastern Europe, with frequent mosquito bites. It is noteworthy to underline that pulmonary nodules represent an unusual manifestation of a Despite increasing reports of human dirofilariasis in Italy  and othe


A case report reviewing MRI in a patient diagnosed with carcinosarcoma and uterine didelphys following presentation with post-menopausal bleeding. Staging MRI images demonstrate the anatomical anomaly and pathological features in these co-existing conditions. This report presents an interesting case of carcinosarcoma in a uterine didelphys. This anatomical abnormality is an uncommon finding and is very rarely complicated by carcinosarcoma. There are very few cases reported to date of this unusual condition. Our case is of a 77-year-old female, para 9, who presented with post-menopausal bleeding for 1 month. She followed the pathway for endometrial cancer using Northern Ireland Cancer Network clinical guidelines. This female’s co-existing uterine anomaly and malignant pathology are outlined, staged and beautifully illustrated with dedicated pelvic MRI. The images captured by MRI are used in all aspects of the patients care and treatment planning, and show the benefit of this modality in multidisciplinary meetings guiding gynaecological-oncology surgeons, who must aware of the anatomical variants before embarking on definitive surgery. There are few cases reported to date of these conditions co-existing. The rare anomaly of didelphys uterus is estimated in 1/3000 females.3 The most common presentation is post-menopausal bleeding, as is the case with our patient.Risk factors for endometrial cancer include oestrogen replacement, obesity, polycystic ovarian syndrome, null parity, tamoxifen and diabetes mellitus.A 77-year-old female (para 9) presented with post-menopausal bleeding for 1 month. She has had three sets of twins by caesarean section.Past medical history included myocardial infarction, primary coronary intervention with stent insertion, hypertension and hypercholesterolemia.On examination, clinical obesity was noted and bimanual palpation did not reveal a pelvic mass. On speculum examination, two cervices were identified.4The differential diagnosis for post-menopausal bleeding includes endometrial atrophy, endometrial polyps, submucosal fibroids, endometrial hyperplasia, endometrial carcinoma (approx. 10%) and oestrogen withdrawal.Initially she underwent trans-vaginal ultrasound scan at gynaecology outpatient clinic, which was diagnostic for uterine didelphys, with two distinct endometrial cavities and separation of the uterine fundi. The left endometrium measured 2 mm  and the right endometrium measured 10 mm, raising the suspicion of endometrial pathology.The patient then proceeded to outpatient hysteroscopy which revealed stenosis of the left cervix. The right cervix and cavity showed a vascular suspicious appearing endometrium, which was subsequently biopsied. It was not possible to sample the left endometrium due to the cervical stenosis.Pathology report revealed fragments of carcinosarcoma , with automatic grading of Grade III endometrial carcinoma.Following this, the female was discussed at local gynaecological-oncology multidisciplinary meeting and after clinic review to discuss the diagnosis, she proceeded to staging MRI scan of the gynaecological pelvis.Care pathway for endometrial cancer was followed using Northern Ireland Cancer Network (NICaN) clinical guidelines—within 28 days patients should undergo ultrasound scan, hysteroscopy, patient consultation to discuss diagnosis before MRI and discussion at multidisciplinary team meeting with a decision to treat being made. This female had imaging using an Magnetom Aera 1.5T MRI scanner with sequence protocol as per NICaN imaging guidelines using Gadovist 7.5 ml (Bayer) .The MRI demonstrated abnormal widening of the right endometrial cavity with invasion into the myometrium beyond the junctional zone. The pathological signal extended from the uterine fundus to the internal os without cervical stromal involvement.No parametrial or adnexal extension shown on post-contrast sequences. No significant iliac or para-aortic lymphadenopathy identified..The left uterus, endometrial cavity and cervix had a normal MRI appearance for a post-menopausal patient. The International Federation of Gynaecology and Obstetrics (FIGO) MRI staging of the right uterus was Stage 1b ; node negative .5 provided by the regionally recommended sequences  and usually well tolerated method of imaging with excellent results and good pathological correlation in staging of gynaecological malignancies. This correlation between MRI and pathological FIGO staging is regularly audited in our department to maintain recommended standards.MRI is an essential part of the care pathway and management protocol for patients with uterine malignancy such as in this case of carcinoma.7 For example, without detail regarding the fundal wall and endometrial cavities, it can be difficult to distinguish between bicornuate and septate uterine anomalies.The radiologist is then able to fully participate and enhance each patient’s multidisciplinary meeting discussion to guide appropriate treatment. In our case of uterine didelphys and in another anatomical anomalies it is essential for the surgeon to be aware of the relevant anatomy before embarking on definitive surgery. MRI images as shown clearly demonstrate the variants in this patient. MRI is particularly useful in distinguishing between different uterine malformations and is in most cases superior in this regard in comparison to ultrasound and hysterosalpingography.i.e. 1b versus IIIa) is perhaps explained by the inability of MRI to assess early lymphovascular invasion. Diffusion-weighted imaging is evolving as a useful technique in MRI for assessing tumour invasion. It is not currently part of our NICaN imaging guidelines but should be considered for future inclusion, perhaps precluding the need for gadolinium.Safety issues, patient compliance, poor renal function and movement artefact including bowel peristalsis may limit MRI staging. Image interpretation is also subject to limitations in spatial resolution. In this case report the difference in MRI and pathological FIGO staging (8 MRI can provide information with regard to myometrial invasion, cervical stromal involvement, serosal breech, adnexal and lymph node involvement, all of which are necessary to accurately stage any grade of endometrial cancer.9MRI report, including FIGO staging, along with pathological grading help provide the clinician and the patient with information regarding the planned treatment, risk stratification and likely prognosis.10 than the high grade carcinosarcoma, which carries a poor prognosis of 40% three year survival.11 The prognosis is further reduced because of our patients medical co-morbidities she was unable to have the recommended pelvic node clearance at the time of pelvic surgery.In our case, this female had Grade III endometrial carcinoma, also referred to as endometrial carcinosarcoma or malignant mixed mullerian tumour. Endometrial carcinoma may also be graded as I or II, both of which carry a better prognosisMRI is the most useful imaging modality in staging uterine malignancies such as carcinosarcoma. In this case anatomy is also well outlined and MRI is an excellent imaging technique for defining Mullerian tract anomalies.Patient care and treatment should be guided by gynaecological-oncology multidisciplinary meeting and review of staging MRI images.It is essential for the surgeon to be aware of the relevant anatomy before embarking on definitive surgery. MRI images as shown clearly demonstrate the anatomical variants in this patient.


Primary vitreoretinal lymphoma (PVRL) is a rare ocular condition and its diagnosis remains a challenge. The clinical presentation is variable and it can masquerade as chronic intermediate or posterior uveitis. We report an unusual case of primary central nervous system lymphoma (PCNSL) presenting as migrating retinal lesions with unique shapes. The diagnostic challenges are described and the clinical features of intraocular lymphoma are reviewed.A 53 year-old gentleman presented with unilateral visual disturbance and a wide area of retinal whitening with sharp borders temporal to the macula, corresponding to hyper-reflective subretinal changes on optical coherence tomography (OCT). The lesion resolved spontaneously after 6 weeks but was replaced by multiple punctate sub-retinal and sub-retinal pigment epithelial lesions. The second eye was involved 4 months later and there were new areas of hyper-reflective changes in both eyes, which migrated nasally within a week, with no evidence of scarring in the previous sites. The diagnosis of diffuse B-cell lymphoma was made on brain biopsy when the patient subsequently presented with acute confusion and magnetic resonance imaging brain scan showed a frontal lesion. Following systemic chemotherapy, the sub-retinal changes resolved with complete restoration of retinal architecture and improvement in visual acuity.We report an unusual case of PVRL presenting as migrating retinal lesions with unique shapes. PVRL is a great imitator and a high index of clinical suspicion is required in unexplained ocular lesions to prevent a delay in diagnosis. Primary central nervous system lymphoma (PCNSL) is a rare malignancy that is confined to the brain, spinal cord, eyes, and leptomeninges. Most cases are diffuse large cell non-Hodgkin B-cell lymphoma . It reprDiagnosis of PVRL remains a challenge as it can mimic chronic posterior uveitis. Kimura et al.  and FardWe report a case of PVRL presenting with very unsual and migrating retinal lesions in the absence of vitritis.A 53- year old gentleman presented to the emergency eye service with a 1 week history of right visual disturbance with constant multi-coloured photopsia and reduced visual acuity. There was no significant past ocular or medical history. He was involved in a road traffic accident (RTA) 3 weeks earlier when his car was hit from the back but he sustained no injuries at that time. He was otherwise systemically well.The best-corrected visual acuity (BCVA) was 6/12 in the right and 6/6 in the left eye. There were no anterior chamber or vitreous cells and no vitreous haze. In the right eye a large white deep retinal lesion, with an opaque appearance, was identified in the temporal retina, extending into the macular area and transecting the fovea Fig. . The lesThe history of RTA led to consideration of commotio retinae, secondary to presumed whiplash injury from the car accident. Investigations for inflammatory and infective pathologies were negative including normal inflammatory markers, autoimmune screen, serum angiotensin enzyme, chest x-ray and syphilis testing.Six weeks later, the patient reported full resolution of symptoms, including photopsia but the right BCVA remained the same. The area of retinal whitening and corresponding hyper-reflective sub-retinal OCT changes had resolved. Partial restoration of the EZ in the nasal macular was noted compared to earlier OCT. Multiple small macular sub-retinal pigment epithelium (RPE) deposits had appeared, with corresponding hyper-autofluorescence on fundus autofluorescence (FAF). The left eye remained normal.The patient was lost to follow-up and re-presented 4 months later with recent onset blurred vision. The BCVA had deteriorated to 6/15 right and 6/19 left. There was no cellular activity in the vitreous but the posterior vitreous face appeared condensed and thickened. New retinal lesions were evident in the maculae of both eyes: in the right, a long, vertical lesion with ill-defined edges and different orientation, shape and margin appearance to the original lesion, was present; Fig.  in the lOne week later, significant evolution of the right lesion morphology was observed: the temporal margin appeared more defined with re-emergence of a bright linear edge; the lesion had increased in size; new sub-retinal deposits were identified Fig.  and the Prior to review, the patient developed acute confusion and impaired neurological function. Magnetic resonance imaging (MRI) of the brain, lumbar puncture and urgent brain biopsy confirmed the diagnosis of primary CNS diffuse large B-cell lymphoma.He underwent MATRix  chemotherapy and brain and orbital radiotherapy. During treatment the patient noticed a significant and improvement in his visual symptoms. His last recorded BCVA, at 12 months follow up, was 6/7.5 right and 6/6 left. Clinical examination and repeat imaging demonstrated resolution of the large retinal lesions and reduced number of sub-RPE lesions in the both eyes with complete restoration of the outer retinal architecture on OCT Fig.  to d.FigThe clinical and imaging findings, including spectral domain OCT, detailed in this case provide an illustrative example of atypical disease presentation and evolution in PVRL. In particular, the observation of both spontaneous resolution of the large subretinal lesion, on clinical and OCT examination, and subsequent lesion migration is also very unusual in PVRL. Furthermore, the retinal lesions had unique and particularly distinct morphological features that evolved over time.Retinal infiltration in PVRL may involve the intra-retinal, sub-retinal or sub-RPE space . LymphomSpontaneous regression of tumour in any malignancy is unusual. This phenomenon has been described in a very limited number of cases of intraocular lymphoma –13. The Prior to treatment, we observed an interesting pattern of rapid sub-retinal lesion migration. The lesion shifted in location and the observed edge corresponded with the margin of the hyper-reflective sub-retinal changes on OCT. Following migration, the previously involved retina appeared clinically normal and OCT showed resolution of sub-retinal hyper-reflective change. Following treatment, full resolution of sub-retinal changes was observed on clinical and OCT examination, with no evidence of recurrence. In other reported cases, sub-retinal fibrosis and RPE atrophy have been described following resolution of sub-retinal infiltrate but have not been detected in our case .We have demonstrated the utility of multi-modal imaging for assessing retinal disease in PVRL including monitoring disease behaviour and response to treatment. OCT imaging and FAF identified more extensive disease than evident on clinical examination, including sub-RPE lesions and disrupted EZ beyond clinically observed lesion margins Fig. . In conc


Hemorrhoid is a common anorectal disease. Hemorrhoids accompanied by endometriosis are unusual. As endometriosis in the rectum may mimic many other diseases, including cancer and inflammation, its diagnosis may be difficult, especially when it is combined with other diseases.Here, we present a rare case of a patient with hemorrhoids accompanied by endometriosis in the rectum. The endometriosis mass was detected by digital rectal examination and CT scan and confirmed by pathological examination. The mass was approximately 0.8 cm × 0.6 cm and located in the muscularis and submucosa of the rectum 8 cm from the anus.In this case, hemorrhoid is a common disease of rectum and anal canal. However, when it is complicated by another rare disease, the rare one can be easily neglected because of the existence of the common one, especially when the two diseases have similar lesions or symptoms. We suggest that strict physical examination, such as the digital rectal examination in the current case, is critical for correct disease diagnosis. Endometriosis mainly affects females of child-bearing age . EndometA 37-year-old female was referred to our hospital for anal mass prolapse accompanied by bloody stools. Her symptoms started 3 years ago and continued until the time at which she was examined for this report. The patient had no abdominal pain, diarrhea, or weight loss. Prolapsus of the anus and rectum was detected by digital rectal examination. According to these findings, the patient was diagnosed with hemorrhoids. During the digital rectal examination, a mass of approximately 1.5 cm × 1.5 cm under the rectum mucosa 8 cm from the anus was also detected.The tissue samples were examined by hematoxylin-eosin (HE) and immunohistochemistry staining as described previously . The priAbdomen computed tomography (CT) examination detected a mass in the wall of the rectum Fig. . The nodThe histopathological findings are shown in Fig. Immunostaining findings are shown in Fig. Endometriosis occurring in the digestive tract mostly involves the rectum and sigmoid colon . The peaHematochezia is a common symptom in hemorrhoids and can also occur in endometriosis of the rectum , 10, 11.Endometriosis of the rectum is relatively rare compared to hemorrhoids. When these diseases coexist, the rare disease is easily missed, especially when the common one causes obvious symptoms but the rare one does not. Another reason for the diagnostic challenge is that these diseases have some similarities in histopathological findings.


It has been recognized that the incidence of prostatic utricle in boys is increasing and is closely associated with diseases such as hypospadias. However, the clinical features of prostatic utricle with normal external genitalia have received little attention.Based on this, a series of 22 male children with prostatic utricles has been compiled by adding our 3 patients to 19 cases reported. All children enrolled had normal external genitalia. Clinical data of the case was reviewed.Urinary tract infection, purulent urethral discharge and pyuria were the most common presenting chief complaint (41%), irritative lower urinary tract symptoms were present in 17% of cases, obstructive lower urinary tract symptoms were noted in 14%. Urinary retention has been reported in 18% and epididymitis has been reported in 14%. Relatively rare clinical symptoms are abdominal pain, hematuria, and hematospermia. A case of calculus formation and a case of neoplasia within the prostatic utricle has been reported. A cystic mass found by digital rectal examination is the most common presenting sign. A utricular lesion posterior to the bladder was revealed by imaging examination. Unilateral renal agenesis was associated in 32% of reports. Non-surgical approach was chosen in 3 cases, transrectal ultrasonography guided aspiration has been reported in 1 case. Endoscopic techniques were used in 3 cases. Open excision was used in 11 cases. The laparoscopic excision was chosen in 3 cases and Robot-assisted laparoscopy was reported in 1 case. Symptoms and signs disappeared after treatment in all children, and no recurrence occurred during follow-up.Prostate utricles without external genital anomalies are rarely reported in children, and are easily missed and misdiagnosed, often accompanied by recurrent urinary tract infections, lower urinary tract symptoms, epididymitis, dysuria and other symptoms. Imaging studies can confirm the diagnosis. Symptomatic and large utricles should be actively treated. The treatment program should be based on the age, clinical symptoms, and size and location of the utricle. The prostatic utricle is an enlarged diverticulum in the posterior urethra, which result from incomplete degradation of the Müllerian ducts or decreased androgenic stimulation of the urogenital sinus . ProstatAll cases of prostatic utricles published in English from PubMed were reviewed, the closing date was June 30, 2018. All patients included in our study were children (age ≤ 18 years). Four cases were excluded due to the lack of required clinical data, and the remaining 19 cases No. 4–22) and our own 3 cases (No. 1–3) constituted the subjects of this study [–22 and oThe age of 22 patients with clinical symptoms ranged from 2 months to 18 years (mean age 5.8 years). Cases of the prostatic utricles have been reported in all races but are more common in white races . Irritative lower urinary tract symptoms  were noted in 6 patients (17%). Obstructive lower urinary tract symptoms  were noted in 3 patients (14%). Urinary retention has been reported in 4 patients (18%) and epididymitis has been reported in 3 patients (14%). Relatively rare clinical symptoms are abdominal pain, hematuria, and hematospermia. A case of calculus formation and a case of neoplasia within the prostatic utricle has been reported. In 7 patients (32%), a digital rectal examination revealed a utricular mass above the prostate and posterior to the bladder. Less commonly reported signs include pelvic or abdominal mass and enlarged scrotal swelling.All included cases have completely normal genitalia, unilateral renal agenesis was noted in 7 patients (32%). Less frequently patients have associated with congenital obstructive posterior urethral and vesicoureteral reflux.Ultrasound was the most commonly used imaging method Fig. , provingNon-surgical approach  was chosen in 3 cases, transcrectal ultrasound (TRUS) guided aspiration has been reported in 1 case. Endoscopic techniques (utricle catheterization and aspiration and endoscopic utricle orifice incision) were used in 3 cases. Open excision  was used in 11 cases. The laparoscopic excision was chosen in 3 cases and Robot-assisted laparoscopy was reported in 1 case. No recurrence or repeated symptoms of the prostatic utricles were reported after any treatment.In 15 cases of surgical excision, pathological findings were reported in 7 cases. Two of them were lined with squamous epithelium, one was lined with transitional epithelium and 1 was lined with flattened cuboidal epithelium. Two utricles were noted to have areas of squamous metaplasia. A rare clear cell adenocarcinoma was reported in a prostatic utricle.The prostatic utricle is a rudimentary structure in the posterior urethra of males. After careful embryological investigation, the researchers believe that the cranial portion of the prostatic utricle is derived from the Müllerian ducts and the caudal segment has a mixed origin from the Müllerian ducts and Wolffian ducts and the urogenital sinus . In maleThe differential diagnosis need to be considered include Müllerian duct cyst, bladder diverticulum, urachal cyst, or a seminal vesicle cyst . Among tThe diagnosis is suspected when there is clinical manifestation and the mass is felt on the digital rectal examination. A pelvic ultrasound, a transrectal ultrasound or a perineal ultrasound can show cavity filled with fluid and its relationship with adjacent anatomical structures such as the prostate and urethra . In VCUGAppropriate surgical procedures have been described to treat symptomatic and enlarged prostatic utricle. The first is endoscopic utricle orifice dilation, catheterization and aspiration, and resection of utricle roof. These methods have the advantage of less invasive, but also have a relatively high risk of recurrence . We treaThe presence of a prostatic utricle in the absence of hypospadias, cryptorchidism or pseudohermaphroditism/intersex disorders is a rare finding. A combination of clinical signs and findings on physical examination guide diagnosis. Imaging test and urethrocystoscope can not only diagnose the prostatic utricle but also investigate its anatomical relationship. In general, small, asymptomatic, accidentally diagnosed prostatic utricles can be followed up without treatment. Complicated and large utricles need to be cured by surgery.


Calcification of ligamentum flavum (CLF) is an important cause of spinal stenosis and spinal cord compression. CLF does not usually induce immediate quadriparesis. Here we describe a rare case of immediate quadriparesis due to a large calcified mass containing liquids in the ligamentum flavum, which was easily confused with gout crystals.A 74-year-old Asian male felt progressive bilateral arm and leg weakness. On the fourth day, acute quadriparesis occurred.Coronal and sagittal computerized tomography (CT) and magnetic resonance imaging (MRI) showed a large circular mass in the left posterior part of the cervical 3/4 spinal canal, protruding into the canal, and occupying one-half of the spinal canal.Emergency laminectomy was performed at C3/4 level. The huge cyst was excised and 1 ml of white viscous liquid flowed out.After operation, CT and MRI showed a full laminectomy of C3/4 and complete decompression of the cervical spinal cord. Hematoxylin-eosin (HE) staining showed that large amounts of calcium was deposited around cystic tissues. Five-year follow-up after laminectomy showed good recovery.This case of immediate quadriparesis, caused by a large calcified mass containing fluid, is very rare. It should be at the earliest stage of calcification. Laminectomy is an effective treatment. This calcification was deceptive and was easily confused with gout crystals. It can help to understand the exact pathophysiology of CLF. Chondrocalcinosis and calcium pyrophosphate dihydrate (CPPD) crystal deposition is probably the main pathological manifestation of calcification.Calcification of ligamentum flavum (CLF) is an important cause of spinal stenosis and spinal cord compression. As reported in several studies, it mostly occurs in Asian populations rather than in Caucasian populations.–5 We observed a 15 mm × 20 mm calcified cystic mass containing white fluid, which maybe the main cause of immediate quadriparesis. The liquid even confused the orthopedists and made them think it was gout crystals. This case study will help to understand the exact pathophysiology of CLF. The study was approved by the EC office of Guangzhou University of Chinese Medicine, and the rights of the subjects were protected. The patient known all rights and interests, and has signed the informed consent.This disease occurs mostly in the thoracic and lumbar spines, but rarely in the cervical spine. The most common symptoms include numbness, dysesthesia, and pain. All these symptoms are progressive and do not suddenly worsen. Immediate quadriparesis caused by CLF is even rarer. To our knowledge, there are no reports of the relationship between CLF and immediate quadriparesis. In this report, we present the case of a patient with immediate quadriparesis caused by CPPD and 5-year follow-up after laminectomy. This case is different from other reported cases of CLF.22.1The patient was a 74-year-old Asian male who came to our hospital due to left limb weakness and difficulty with balance while walking, without dizziness, headache, panic or chest tightness. During hospitalization at the neurology department, he felt progressive weakness in bilateral arm and leg. No significant improvement was observed after treatment with circulatory improvement drugs. Four days later, the patient felt aggravated limb weakness and numbness and was unable to walk. The patient then had an emergency transfer from the neurology department to the orthopedics department for treatment. We conducted a motor examination, which revealed that the muscle strength of the upper extremity had markedly decreased . Bilateral lower extremity strength had also decreased . Sensation to pinprick had also decreased on the neck. Bilateral tendon hyperreflexia, positively bilateral Babinski signs and bilateral clonus hyperreflexia were also seen. The patient had a history of lacunar infarction for 6 years, and hypertension for 6 years, and had been prescribed daily use of nifedipine drugs.2.2Plain radiographs showed straightening of cervical curvature, hyperosteogeny and narrowing of intervertebral spaces. Coronal and sagittal computerized tomography (CT) showed a circular high density mass in the left posterior part of the cervical 3/4 spinal canal, protruding into the canal, and occupying one-half of the spinal canal Fig. a, b. Urg2.3We urgently took the patient to the operating room, performed a laminectomy at C3/4 level with resection of the calcified mass from the ligamentum flavum. During the operation, after laminectomy, we observed a calcified ligamentum flavum tissue in the left posterior part of the dural sac, which was about 15 mm × 20 mm in size. The cyst was excised and 1 ml of white viscous liquid flowed out. The excised cyst was sent for pathological examination. Residual ligaments and calcified tissues are difficult to separate due to adhesion to the dural sac, therefore they were placed in floating treatment. Finally, the range of laminectomy was extended until the compressed dural sac was filled and restored.2.4The excised cyst was sent for pathological examination Fig. a. Hemato2.5After surgery, CT showed a full laminectomy of C3/4 (Fig. 3There are several special features of this case. The patient suffered from acute quadriparesis. Without timely decompression, quadriparesis would have been irreversible and even life-threatening. In addition, during the operation, we observed a 15 mm × 20 mm calcified cystic mass containing 1 ml of a white viscous liquid. This has never been reported before. Immediate quadriparesis was most likely caused by a sharp increase of this liquid.–7 CLF is the most common issue. Nanko reported of a 70-year-old woman with radiculomyelopathy at C5–6 and C6/7 level in 1976, bringing attention to the fact that CLF is a cause of disease for the first time. In 1983, Yoshinobu reported of 4 cases of calcification of the cervical ligamentum, and noticed that calcification might be induced by the degeneration or abnormal nutritional state of the ligamentum flavum. Wada reported of a 54 year old male with neck pain, who was preoperatively diagnosed with CLF. Intraoperatively it was found to be CPPD crystal deposition along the spinal dura mater. Various factors, including the aging process, chondrocytic metaplasia, endocrine imbalance, metabolic diseases, and mechanical stress, have been reported as major causes of the development of CLF.,11 Pathological change studies speculated that during the early stages, CPPD deposited in the ligamentum flavum, and then during the middle and late stages, CPPD is transferred to a stable final form of hydroxyapatite crystals.,11 In our case, immediate quadriparesis was caused by a large calcified mass, and the 15 mm × 20 mm calcified cystic mass contained 1 ml of a white viscous liquid. This white viscous liquid may be the earliest form of CPPD aggregation that had not yet been calcified.Spinal disease through CPPD crystal deposition has been repeatedly reported.,5,12 But the gold standard for diagnosis of spinal gout is still monosodium urate crystallization pathology. Jegapragasant declared that for patients with lower back pain or nerve compression symptoms, spinal gout should be highly suspected if the patient has a history of gout, especially multiple nodules, hyperuricemia, and characteristic imaging changes. As reported by Takashi, a 70-year-old Japanese woman, was affected by pseudogout of the cervical yellow ligament, leading to neck pain. C6 laminectomy and removal of the C5-C6 yellow ligament was able to achieve a satisfactory therapeutic effect. Cervical laminectomy is likely to result in kyphosis deformity. However, according to the 5 year observation, the cervical curvature of the patient had recovered well.In this case study, the rapidly progressive course and liquid released during surgery confused the orthopedists, and made them think it was gout crystals. However, histopathology did not support this suggestion. The uric acid level of the patient was normal and no arthritis was detected in other joints. Prior to surgery, it is difficult to distinguish calcification and gout. Recently, along with the development of dual-energy imaging technology, a new option of imaging examination of gout has been made available. Its work principle is that different energy CT values of human tissues can be processed by software to display characteristic color coding for the chemical composition of tissues. Dual-energy CT can show the location and quantity of gout deposits.In conclusion, this study demonstrates a case of immediate quadriparesis caused due the presence of a large calcified mass in the ligamentum flavum. The 5-year minimum follow-up period showed good results, proving that laminectomy is an effective treatment. This calcification was deceptive and was easily confused with gout crystals. This case study will help us to understand the exact pathophysiology of CLF.Conceptualization: Weiji Yu, Yafei Cao.Data curation: Kun Gao, Heng Li, Weidong Liu, Weiji Yu.Formal analysis: Kun Gao, Heng Li.Funding acquisition: Yafei Cao.Investigation: Kun Gao, Heng Li.Supervision: Weidong Liu, Weiji Yu.Writing – original draft: Kun Gao.Writing – review & editing: Kun Gao.Yafei Cao orcid: 0000-0001-9193-376X.


Primary adenocarcinoma of the appendix is a rare disease in clinical practice. Moreover, primary adenocarcinoma of the appendix in the pediatric age group is even rarer with very little cases being published. Here, we report a case of primary adenocarcinoma of the appendix with local invasion into adjacent organs in a child who was initially diagnosed as having an acute appendicitis.A 13-year-old girl presented with abdominal pain of 3-month duration. Imaging study showed a mass including the fecalith that occupied her pelvic and right lower abdominal cavity. Drainage of the abscess and appendectomy were performed by the preoperative diagnosis of an acute appendicitis with an appendiceal mass. Postoperative histopathological examinations revealed the appendiceal adenocarcinoma. She then received the whole mass resection, ileocecal resection with lymph node dissection. The masses were tightly adherent with infiltration into the sigmoid colon, uterus, and right ovary. These organs were all dissected, and subsequent sigmoid colostomy was performed. We preserved the left ovary for her fertility. The pathological findings demonstrated negative margins and no lymph node invasions, and final pathological stage was pT4(SI)N0M0, stage II. After the operation, she received the chemotherapy with 6 cycles of 5-fluorouracil (5-FU), leucovorin (LV), and oxaliplatin (mFOLFOX6) and subsequent 6 cycles of simplified LV and 5-FU (sLV5FU2). The patient is doing well till today on follow-up without progression of the disease 5 years after the operation.Primary adenocarcinoma of the appendix is exceedingly rare in children. In this report, we described one of the youngest primary adenocarcinomas of an appendix case ever reported. When encountering atypical cases of the appendicitis, we should consider the possibility of primary adenocarcinoma of the appendix as it has an extremely poor prognosis and is usually diagnosed in advanced stages. Appendiceal cancer is a rare malignancy with an incidence of around 0.1 in 1,000,000, making up only 0.5% of all gastrointestinal malignancies . This caIn the pediatric aged group, the incidence of colorectal cancer is rare compared with that in adults. Although early diagnosis and improved management have resulted in reduced mortality since 1980s , the treHere, we report a case of primary adenocarcinoma of the appendix (PAA) with local invasion into adjacent organs in a child who was initially diagnosed as having an acute appendicitis.A 13-year-old girl presented with abdominal pain and bloody stool. For 3 months, she had repeated appearance of the abdominal pain and spontaneous disappearance of the symptom. At the beginning, the symptom was not strong and she had not seen a doctor. Her abdominal pain gradually got worse, and she determined to come to our hospital after she often had bloody stool. Imaging study showed a mass including the fecalith that occupied her pelvic and right lower abdominal cavity Fig. . AlthougShe then received the whole mass resection, ileocecal resection with lymph node dissection. At the first operation, we speculate that we resected body of the appendix but left the most of the mass and the tip of the appendix. Therefore, the mass which we resected in the second operation was derived from the tip of the appendix. The masses were tightly adherent with infiltration into the sigmoid colon, uterus, and right ovary. These organs were all dissected, and subsequent sigmoid colostomy was performed Fig. . We presAfter the operation, she received the chemotherapy with 6 cycles of 5-fluorouracil (5-FU), leucovorin (LV), and oxaliplatin (mFOLFOX6) and subsequent 6 cycles of simplified LV and 5-FU (sLV5FU2). The patient is doing well till today on follow-up without progression of the disease 5 years after the operation. We performed the stoma closure operation 2 years after the tumor resection.Primary adenocarcinoma of the appendix (PAA) is an extremely rare disease in clinical practice . There aColorectal carcinoma in children, although rarely discovered, comprises approximately 1% of pediatric neoplasm . It is aIn addition, there is no consensus for the goal of the operation for PAA in children. Generally, functional preservation operation has often been emphasized in pediatric surgery. Therefore, it is not easy to decide the grade of the extension in the radical operation for colorectal cancer such as PAA in children as there is very little reports being published. Furthermore, we have no guideline about the postoperative treatment protocol such as the resume of the chemotherapy.We reviewed some recent literature which has been published since 2000 and found seven cases of the colorectal adenocarcinoma in children who is under 15 years old (Table Furthermore, Table In this report, we demonstrate a case of PAA with local invasion into adjacent organs in a child who was initially diagnosed as having an acute appendicitis. We performed radical operation and adjuvant chemotherapy. The patient is doing well without progression of the disease after 5 years. Although little has been understood about PAA, we believe our experience may provide new insights into the guideline of the standard treatment for PAA in children.Primary adenocarcinoma of the appendix is exceedingly rare in children. In this report, we described one of the youngest primary adenocarcinoma of appendix case ever reported. When encountering atypical cases of the appendicitis, we should consider the possibility of primary adenocarcinoma of the appendix as it has an extremely poor prognosis and is usually diagnosed in advanced stages.


Giant congenital melanocytic nevus is a very rare condition characterized by a large skin lesion and an increased risk of complications like neurocutaneous melanosis and malignant transformation. Reports of giant congenital melanocytic nevus are scarce in the sub-Saharan African literature and here we present a case of this disease in a Cameroonian adolescent.A 12-year-old Cameroonian girl from the "Baka" ethnic group, with no relevant family and medical histories presented with a progressively extensive brownish-black nodular hypertrophic skin lesion of approximately 45 cm, which she had had since she was 2-days old. The lesion covered her entire back giving an appearance of “turtle child”, which was highly suggestive of a giant congenital melanocytic nevus. She was booked in for a surgical intervention organized by a health campaign within her community. Meanwhile she was provided with psychological support and her family was counseled on warning signs of complications which would warrant an urgent consultation.Here we presented a case of giant congenital melanocytic nevus, apparently the first in the Cameroonian literature. In view of the potential severe complications as well as psychological trauma of this pathology, we draw clinicians’ attention to this extremely rare but real pathology in our country, for a timely diagnosis and management. Melanocytic nevi refer to tumor-like malformations of the skin or mucous membrane, due to benign proliferation of pigment-producing skin cells called melanocytes . They beA 12-year-old Cameroonian girl from the "Baka" ethnic group and residing in a remote area of the East region of Cameroon presented with a progressively extensive, pruritic, and painless pigmented skin lesion on her back, persistent since she was 2-days old. She was born through normal vaginal delivery at term from an uneventful pregnancy. Her past medical and family histories were unremarkable. On our initial physical examination, she had normal anthropometric characteristics for age, as well as normal vital parameters. Examination of her skin revealed a large, irregular, well-demarcated and unequally pigmented (bluish-brown to black) multinodular hypertrophic nevus occupying almost all her back , which can be present at birth or may develop after a few weeks of life , 2, 4, 7et al. . Kopf anet al.  proposedNRAS mutations and to a lesser extent BRAF mutations [NRAS gene, located on the short arm of chromosome one at position 13.2, provides instructions for making a protein called N-Ras that is involved primarily in regulating cell division [With respect to epidemiology, approximately 1% of live births presents with a CMN. The small form occurs in 1 per 100 live births, the medium form in 6 per 1000 live births, and the incidence of the giant form is globally estimated at 1 in 20,000 to 500,000 live births , 9. In gutations . The NRAdivision . A mutatdivision , 7. Furtdivision , 8.Classically, GCMN is an asymptomatic disease but some patients may complain of pruritus and xerosis as shown in the case above. The findings on physical examination are variable but affected persons usually present with brownish to black, flat or nodular, well-defined lesions, associated with hypertrichosis. The surface of the nevus may be papular, warty, cerebriform, or rough, as in our case , 4, 11. The repercussion of GCMN can be organic or not. In fact, it is well established that the disfiguring character of this pathology may have psychosocial implications with deleterious impacts on the self-esteem of affected individuals, especially females –6; this Although the diagnosis of GCMN is mainly clinical, histopathology study when available is needed to affirm the diagnosis and rule out malignant transformations. In this regard, the most common histological findings are hyperkeratosis and hyperplasia, elongation of epidermal ridges, and increased number of melanocytes .Finally, the therapeutic options for GCMN vary from surgical excision to laser therapy. Treatment should be individualized, taking into consideration the age of the patient, the size and location of the lesions, the risk of melanoma, the possibility of a NCM, and the presence of other congenital abnormalities or comorbidities. The prognosis depends on all of these factors , 14, 15.Here we presented the case of GCMN in a Cameroonian girl, apparently the first in the Cameroonian literature. Although a very rare pathology, its diagnosis is mainly clinical and has a pivotal role in its timely management geared at averting its associated psychological trauma and risk of complications such as NCM and malignancies. We therefore draw clinicians’ attention to this extremely rare but real pathology in our country, in order to ameliorate its prognosis.


En bloc extensive resection of the penis and prepuce, without penile retroversion or pexy to ventral abdomen associated with a permanent perineal urethrostomy, was performed. Histology of the mass revealed a squamous cell carcinoma of penis and prepuce. The surgical technique that was adopted is a modified version of that already described that allows a more proximal resection of the penile body and is a valid option for treating advanced SCC lesions involving the penis. Early postsurgical complications  were managed with a medical treatment and resolved within 5 to 12 days. Three years after surgery the horse is in good body condition and does not show any sign of recurrence or disorders related to the surgery.A 15-year-old gelding was referred for a florid, cauliflower-like ulcerated mass, enclosing penis and prepuce together with penile urethra showing a malodorous purulent and blood-stained discharge and larvae infestation. It may apilloma , 3. It capilloma , 4.Suggested treatments of small and not complicated SCC of penis and prepuce are cryotherapy , 6 and c en bloc resection with or without penile retroversion [For extensive SCC surgical excision is recommended. Segmental posthectomy (reefing) and partial phallectomy –12 are ioversion –15 or peoversion , 17. en bloc resection of penis and prepuce and superficial inguinal lymph nodes with penile retroversion in 4 horses affected by SCC with 100% survival rate in 1-year follow-up without recurrences [ en bloc resection of the penis for SCC had a positive long-term follow-up without recurrences; in one case urine scalding due to lateral deviation of the urinary flow was reported. Another retrospective study by Van den Top et al. [ en bloc resection of penis and prepuce was performed in 13 out of 77 horses affected by SCC with lymph node enlargement and extensive genital involvement. Eight horses were available for follow-up and only one suffered neoplastic recurrence within 18 months [In a long-term follow-up study, 9 cases of SCC infiltrating the penile body were described with a survival rate of 55% (5/9) after penile amputation and urethrostomy . Mair eturrences  in 25 geurrences , 5 geldiShort- and long-term complication associated with the surgical procedures consisted in cystitis, mild to severe haemorrhage –15, woun en bloc extensive resection of penis and prepuce without penile retroversion, together with a permanent perineal urethrostomy, after a 3-year follow-up period, adding information about long-term clinical outcome after surgical treatment.This case report describes the successful treatment of a penile SCC in a gelding withA 15-year-old Argentinian Warmblood gelding was referred for treatment of a long-standing penile lesion affected by myiasis. glans penis together with penile urethra ( muscae spp. larvae. Poor body condition score (BCS 4/9) and strangury were also present.At referral the main complaint was a florid, cauliflower-like ulcerated mass, measuring 10 cm in diameter that enclosed the urethra  with malμg/kg, palpation of the penis revealed a diffuse thickening of the penile shaft with multiple ulcerations involving the inner and outer laminae of the preputial fold. Due to the regional anatomical alterations related to the mass (phlegmon and oedema), inguinal lymph nodes were not palpable. For the same reason ultrasonographic examination of external genitalia and regional lymph nodes was not conclusive. Transrectal examination was unremarkable.Once penis prolapse was obtained by intravenous administration with acepromazine 30 Urine analysis revealed the presence of leukocytes and nitrites, consistent with urinary tract inflammation. As the clinical condition of the horse and the primary lesion were severe, suggesting complete excision in any case, fine needle aspiration or excisional biopsies were not attempted; therefore surgical removal was combined with harvesting material for histopathological examination. en bloc resection of the penis and prepuce was decided; the eventuality of a penile retroversion or a permanent perineal urethrostomy was considered according to the intraoperatory findings and the degree of infiltration of the penis body [Based on clinical findings differential diagnoses were SCC of the penis and prepuce and/or habronemiasis. Due to the infiltrating pattern and the diffusion of the disorder and to the presence of multiple nonhealing lesions, leading, respectively, to difficulty at micturition and local infection, annis body . Systemi en bloc resection, a fusiform 40 cm long skin incision starting at the umbilicus was made along the midline. It extended caudally encircling the preputial orifice. Blunt dissection of subcutaneous tissue was performed around the penis until the abdominal fascia was reached and the body of the penis was released from its anatomic and vascular connections. Haemorrhage was controlled with electrocauterization and ligation of the major vessels . The dissection plane was extended to both the external inguinal rings and the external pudendal arteries and vein were ligated. The superficial inguinal lymph nodes appeared enlarged and therefore were excised. At palpation the penile body was extensively thickened suggesting amputation as proximal as possible: so penile retroversion was excluded and perineal urethrostomy was performed. To this aim, a tourniquet was applied proximally to the shaft of the penis and an extensive resection was performed close to the ischiatic arch, dividing the suspensory ligament of penis. The proximal stump showed a normal macroscopic appearance and was sutured with double transfixing sutures (2 USP absorbable multifilament) placed through the penile body in a dorsoventral direction, to obtain adequate haemostasis. Once urethral lumen was clearly visible a urinary catheter was placed. The outer perimeter of the tunica albuginea was apposed in a simple interrupted pattern with a 2/0 USP monofilament absorbable suture.Surgery was performed under general anaesthesia with isoflurane with the horse in dorsal recumbence. For theFor permanent urethrostomy a 8 cm skin incision was created on the perineal raphe starting about 7-8 cm below the anus, ending at the level of the ischiatic arch. Blunt dissection of subcutis, penis retractor muscles, and bulbospongiosus muscles was achieved until the urethra was visualized. The penis retractor muscles were sutured to the subcutis in a simple continuous pattern, using a 0 USP monofilament absorbable material. Then the urethral wall was incised longitudinally for about 6 cm and the mucosa sutured to the skin in a simple interrupted pattern with a 2/0 USP monofilament nonabsorbable material.The subcutis of the abdominal wound was sutured with a 0 USP monofilament absorbable suture material in a simple continuous pattern and the skin was sutured with metallic staples. A multitubular drain was applied laterally to the midline to the abdominal wall suture at the scrotal region .After recovery from anaesthesia the horse showed a moderate haemorrhage from the urethrostomy, which was particularly evident at the end of urination, leading after 48 h to a marked reduction of PCV (15%) and TP . As the haemorrhage seemed to come from the corpus spongiosum penis and surgical haemostasis was not feasible, it was successfully managed by the administration of tranexamic acid (15 mg/kg i.v. q12 h) for 2 days. Oral support with vitamin B complex and folic and pantothenic acid was given for 10 days until PCV and TP reached, respectively, 30% and 6.8 g/dL.Strangury but not pollakiuria was observed 3 days after surgery.Three days after surgery, the horse showed a serosanguineous collection above the abdominal wound and a moderate purulent discharge from the skin incision that were managed with daily manual massage and local disinfection. Five days after surgery, the urinary catheter and the multitubular drain were removed without complications.On day 12 standing surgical revision of the urethrostomy was required to remove some necrotic urethral mucosa leading to a partial dehiscence of the wound. Habronema spp. infestation was found. The margins of the excised tissues were free of neoplastic cells, and the regional lymph nodes were inflamed but not affected by the neoplastic process.Histopathology of the lesions confirmed the presumptive diagnosis of SCC of the penis and prepuce invading the subcutaneous tissue, not the albuginea. Lymphocytic and neutrophilic infiltration of the corpus cavernosum suggested chronic balanitis; moderate subcutaneous eosinophilic infiltration was also detected but no evidence ofAfter skin staples removal 14 days after surgery, the horse was discharged. Urination was unremarkable from the perineal stoma. No urine scalding or subcutaneous infiltration could be observed. PCV and TP values were back to preoperatory values.A clinical follow-up performed 2 months after surgery revealed reduction of the urethrostomy to a diameter of about 1.5/2 cm. Anyhow urination was unaffected with a mare attitude and no urine scalding was detected.A 3-year follow-up showed no recurrence of the neoplasm with normal urination. Urethrostomy did not show any further stricture as well as no urine scalding . en bloc extensive resection of penis and prepuce without penile retroversion as previously described [In this case report the long-term (3 years) successful treatment of a SCC of the penis body and prepuce in a gelding is described. The surgical treatment of the disorder was achieved with anescribed  associat Habronema spp., Halicephalobus spp., or Draschia megastoma, and coital exanthema by EHV-3 [Differential diagnoses in presence of preputial masses, oedema, and discharge, difficulty at urinating, and phimosis are other neoplastic disorders  , 2 and nby EHV-3 , 20. Altby EHV-3  these weEn bloc resection of penis and prepuce is indicated in cases where external genitalia are extensively affected by the neoplasia. It allows removal of the penis as far proximal as possible until healthy tissue is recognized. Despite invasiveness and postoperatory complications, this technique allows a high rate of success for the cases described in the literature with long-term survival and few tumour recurrences [urrences , 13, 15.En bloc resection of penis and prepuce is usually associated with penile retroversion and suture of the penile stump to a subischial skin incision to create a new urinary meatus [y meatus . In the y meatus .According to Van Harreveld et al. an option is to transect the penis just distal to the site typical of a routine perineal urethrostomy. The corpus cavernosum penis is closed and secured to local fascia and subcutaneous tissue, and a permanent perineal urethrostomy is performed. To our knowledge, no published reports of the long-term outcome of this surgical technique in the horse exist; the authors remark that this technique has worked well and appears to avoid the creation of a flexure in the penis .Recently Wylie and Payne reported 15 cases of extensive penile disorders treated with a subischial urethrostomy and penile amputation with preputial ablation performed under general anaesthesia in dorsal recumbence. Eleven SCC, 2 melanomas, 1 chronic preputial discharge without neoplasia, and 1 paraphimosis secondary to sedation were included. They describe a median survival time of 25.1 months, with 64.3% (9/14 cases) surviving >18 months [In the case described here, to find a normal macroscopic aspect of penile shaft and be consistently sure of the absence of neoplastic tissue left, the penile amputation had to be made very close to the ischiatic arch. To remove all the affected tissue the penile body was excised extensively, more proximally than in the position described by Archer and Edwards ; for thiAlthough penile retroversion allows the surgeon to direct urinary outflow more caudally than does penile amputation without retroversion , no probIn order to reduce ascending infections from the external urinary meatus, the ventral retractor penis muscles were sutured to the subcutis at the urethrostomy site, hypothesizing that their contraction could act as an external urinary sphincter closing the external opening of the stoma at the end of urination, avoiding bacteria ingress, urine dripping, and secondary scalding. The suture of the muscles to the subcutis ensured also a secure fixation of the penis stump, avoiding subcutaneous infiltration of urine.The complications after surgery encountered in this case were not different from those already described, particularly haemorrhage from the corpus spongiosum at the end of urination, infection of the abdominal wound, and partial dehiscence of the urethral mucosa sutured to the perineum , 22, 23.Postoperative haemorrhage was managed with the administration of tranexamic acid until bleeding stopped. Although haemorrhage is a well described major complication associated with this surgery, the preemptive use of antifibrinolytic agents was not attempted because of the concern about a possible increase in the risk of thromboembolic complications, such as deep vein thrombosis and acute myocardial infarction, related to the use of these drugs in human medicine , 25. FurInfection of the abdominal wound was managed by local daily disinfection and use of antimicrobial without other sequelae.The dehiscence of a permanent urethrostomy can be due to the excessive manipulation of urethral tissue  or to exEven though stricture formation can be a complication in 1/3 of perineal urethrostomy cases during early postoperative period and after removal of the urinary catheter –26, we d en bloc extensive resection of the penis and prepuce without penile retroversion associated with a permanent urethrostomy were the removal of as much penile body as possible, minimizing recurrence and avoiding tension related to phallopexy, and the possibility to create a urinary meatus which is functional and cosmetically similar to the mare's.The advantages of the corpus cavernosum penis, which was affected by chronic balanitis, worsened by the presence of myiasis; the chronic balanitis probably contributed to the palpable thickening of the body of the penis, simulating neoplastic infiltration. Although macroscopically enlarged, inguinal lymph nodes did not contain neoplastic cell, but, consistently with literature [Postoperative histopathology revealed an infiltration of neoplastic cells in the subcutaneous tissue close to the albuginea, but not in theterature , only materature . So in oThe lack of a standardized approach to follow-up evaluation and the definition of clinical success limit an objective consideration about the efficacy of the treatment itself and its prognostic value. Since most of the surgical treatments for penile and preputial disorders are considered as salvage procedures, compromising reproductive and original anatomy, it is essential to the clinician to perform a detailed evaluation of each clinical case and the appropriate treatment. en bloc extensive resection of the penis and prepuce without penile retroversion and permanent urethrostomy allowed the successful treatment of a locally invasive SCC of penis and prepuce in a gelding without recurrence after a 3-year follow-up time. Although short- and long-term complications occurred, they were managed and did not compromise the clinical outcome.In this case report the


We reported a 67-year-old woman in whom large atrial thrombi were found by chance during discontinuation of therapeutic anticoagulation. The patient, with a history of mitral valve replacement surgery, had stopped anticoagulation for months because of intractable gastrointestinal bleeding, during which she was found to have 3 large thrombi in the atria on transesophageal echocardiography: left atrial free-floating ball-shaped thrombus, left atrial appendage thrombus, and right atrial appendage thrombus. One month following diagnosis, she still had the free-floating thrombus despite adequate anticoagulation. Free-floating ball-shaped thrombus is a rare finding observed on echocardiography in patients with mitral valve disease and an even rarer finding in case of appendage thrombi coexisting. Free-floating ball-shaped thrombus in the left atrium is a rare type of cardiac thrombus that may cause embolism, syncope, congestive heart failure, and sudden death –3. It waThe woman was referred to the department of cardiology with consultation for left atrial masses detected by chance with follow-up echocardiography and computed tomography . Eight yWith the subsequent evaluation by transesophageal echocardiography, using an x8-2t transducer on an EPIQ 9 ultrasound machine , 2 atrial thrombi occupying the left and right atrial appendage cavities, respectively, in addition to the free-floating round-shaped mass were found . The lefBased on her clinical history , situations (discontinued anticoagulation and coexistence of appendage thrombi), and previous reports on similar cases –11, the Free-floating ball-shaped thrombus in the left atrium is a rare finding among patients with mitral valve disease with or without a history of valve replacement surgery –8. This Nevertheless, the mechanism for free-floating ball-shaped thrombus formation that we and others proposed above seems to differ from the observation by Wright-Smith et al., who demonstrated that the cut section of the ball-shaped thrombus resected from a patient with mitral stenosis represented a laminated and onionskin appearance with central cavitation . Their fBecause of the intricate clinical situation, the patient could not receive either surgery or thrombolytic therapy, although prompt surgical removal of the thrombus is generally preferred . We restAlthough similar cases were abundantly described –11, to tThis is the first report in which ball-shaped free-floating thrombus and biatrial appendage thrombi were coincidentally imaged on echocardiography. In this case, however, surgical removal of the thrombus was given up because of serious clinical conditions of the patient.


Head and neck Synovial sarcoma (SS) accounts for 3–10% of all total body SS. It is rare to find it in the oral cavity, especially on the floor of the mouth.We present a 44-year-old Chinese male, who had been misdiagnosed as fibroadenoma, with a swelling on the right submandibular region for more than 3 months. The radiology examinations and the pathology results indicate the diagnosis of SS of the floor of the mouth.The patient only had a surgical operation, without radiotherapy and chemotherapy. At the first follow-up, the patient exhibited no clinical or radiographic complications, and the patient was asymptomatic on subsequent visits.Misdiagnosis results the delay of diagnosis and treatment of SS. Immunohistological analysis might be the most important tool to confirm the diagnosis of SS. Synovial sarcoma (SS) is a rare malignant neoplasm of unknown histological origin, accounting for 5.6% ~ 10% of all soft tissue sarcomas .. Most sThe tumor usually occurs in close association with tendon sheaths, bursae, and joint capsules, primarily in the para-articular regions of the extremities. Head and neck SS accounts for 3–10% of all total body SS, with high incidences in the hypopharynx, the postpharyngeal region, and the parapharyngeal space , 6. It’sA 45-year-old male reported that he had a swelling approximately 5.2 × 2.8 × 5.9 cm on the right submandibular region for more than 2 months and was admitted to the local hospital. At the local hospital, the diagnosis of fibroadenoma on the floor of mouth was made. An incisional biopsy was performed under guidance of ultrasound and was submitted for histopathologic examination, the examination revealed that expansion of the lymphatic vessels could be seen in the right sublingual. After an intra-oral excision in the floor of mouth of the right submandibular region (Inferior to the mylohyoid), the immunohistochemistry showed Vimentin was positive, Ki-67 percentage was about 35%, CD34, S-100, CK, P63 and LCA were negative. After discharged from that hospital, he felt that the swelling had grown again in the same area, even rapidly in the near week.One month after the first operation at local hospital, the patient was referred to our hospital for a growth nodule on the floor of the mouth, associated with pain, numbness, and dyspnea. Intraoral examination revealed a proliferative and ulcerated mass measuring approximately 6.0 × 1.0 cm in the right sublingual involving the right floor of the mouth extending from the alveolus of the left mandibular cuspid to the right mandibular 2nd molar teeth and extending anteriorly crossing the midline of the tongue. The mass was firm, with an unclear border, and the surface covered with black pseudomembranous Fig. . The immThe surgical treatment included primary tumor resection, cervical lymph node dissection, and anterolateral musculocutaneous flap reconstruction Figs. , 5, 6, 7Within 1 year after surgery, the patient was seen in our out-patient clinic every month to check the recovery and whether there was recurrence or lymph node metastasis. Six months later, the first radiographic postoperative follow-up showed no recurrence. One year after surgery, PET-CT examination was performed, and the patient had no recurrence and distant metastasis. Then the patient was followed up at six monthly intervals and is still alive with no evidence of recurrence after 46 months since undergoing curative intent surgery.Synovial sarcoma (SS) was first documented by Simon in 1865  and was SS usually occurs in the lower limbs and trunk, especially in the periarticular soft tissues . Head anFor tumors that have recurred multiple times or diameter > 5 cm, the growth rate tends to accelerate and may cause an emergency. In this case, the patient’s tumor rapidly increased with surface bleeding after admitted to our hospital. Moreover, he even suffered from severe dyspnea due to the tumor and clot blocking the airway. According to the postoperative tumor anatomy, the central part of the tumor showed liquefaction necrosis with some dark brown liquid Fig. , which mIn our case, the patient was previously diagnosed as fibroadenoma because of the rarity of SS and lack of typical clinical and imaging manifestations. Although Pantomography, CT, MRI, and PET-CT can be used as diagnostic tools, smaller SSs often show similar imaging features to benign tumors on CT and MR imaging , 17. DesSynovial sarcoma can be classified into three major histopathological subtypes: 1) monophasic SS containing uniform spindle cells or epithelial cells, 2) biphasic SS composed of epithelial cells arranged into glandular structures with spindle cells arranged into fascicles, 3) poorly differentiated SS characterized by the presence of spindle and/or round blue cells . In histThe other cause of misdiagnosis might be the previous immunohistochemistry lacked some key immune marker detection such as EMA, CD99, TLE-1, and Bcl-2, etc. So far, there is no single immunological marker specific to synovial sarcoma has been found. SS shows positive of TLE-1, AE1 / AE3, EMA, CK7, CK19, Vimentin, Bcl-2, CD99, and S-100 while generally shows negative express of CD34, CD31, actin (HHF-35) or Myoglobin , 21. SomFor some cases with atypical histological morphology or confusing results of immunohistochemistry, detection of SYT-SSX fusion gene by molecular biology or cytogenetic technology can be used to help diagnose with synovial sarcoma . It was The optimal approach to the treatment of SS is still unclear and there is no standard treatment protocol . It is rIn our case, chemotherapy was not performed. The importance of chemotherapy has not been widely acknowledged due to the lack of evidence that chemotherapy was associated with improved overall survival. According to the Yang’s report , after oThe prognosis of synovial sarcoma is often associated with tumor location, size, patients’ age, surgical procedure, degree of differentiation . GeneralIn this case, as the rarity SS tumor, the patient was misdiagnosed at the local hospital. Immunohistological analysis might be the most important tool to confirm the diagnosis of SS. Surgical treatment focus on primary tumor resection without chemotherapy might be considered according to the patient’s situation.


Juxta-articular myxoma (JAM) is a very rare myxoid tumor in pediatric age. JAM is frequently located at large joints, particularly in the knee, and most lesions present as palpable swelling sometimes associated with pain or tenderness. Only three cases of pediatric JAM have been reported in the literature to date. We describe a case of JAM in a 12-year-old boy, characterized by several unique aspects related to age, site of onset, size, and associated symptoms. The child was in good health and denied any recent trauma. Physical examination showed a right buttock lesion with a tight, but not painful, flexible consistency; superficial venous prominence or discoloration of the skin was absent. The range of motion of the hip was complete for both active and passive movements and free from pain. The patient had no lameness nor any painful symptoms in performing daily activities. Biochemical investigations including complete blood count, liver and kidney function, inflammatory markers, and electrolytes were normal. No bone involvement at right hip radiograph. Ultrasonography showed, in comparison with another done before admittance, a complex mass with solid and cystic components increased in size of about 5 cm in 3 days. At lower-limb contrast MRI, evidence of an oval lesion, measuring 10 × 15 × 8 cm, located between the gluteus medius and the gluteus maximus. The lesion was well-capsulated, with branches and septa inside, weak endostructural diffusion, and wall irregularities with no infiltrative phenomena into surrounding tissues , low-grade fibromyxoid sarcoma, myxoid liposarcoma, and ganglion cyst, of which characteristics are resumed in Table JAM is an unusual benign lesion occurring between third and fifth decade of life2 followed by the elbow and shoulder, rarely ankle and wrist, and less frequently hip. Causes of JAM remain controversial, although the most credible hypothesis is post-traumatic or postarthritis disease.10 In some cases, the pain was present even with no clinically detectable lesions, while in some others, swelling without pain was present.In adults, the most frequent JAM localization is the knee (88% of cases),2 and one in the wrist chondromatosis, lipoma arborescens, and synovial hemangioma.12Several findings of our patient are similar to the other reported pediatric cases as like as medical history negative for both inflammatory diseases and trauma. Our patient was completely asymptomatic: In one of the two previously reported pediatric cases, the tumor was found in the knee without evidence of pain, while in the other two cases, the children presented with a painful mass.11 or compression effect caused by surrounding tissues, which may have led us to underestimate the size based on the MRI scan.A peculiar observation in our case was the dimensional discrepancy between the preoperative imaging and the excised mass, 2 cm bigger than expected. These data may indicate a rapid lesion growth, as previously described,14 reported fine needle aspiration citology of pediatric soft-tissue lesions and neoplasms as a first-line investigation. Anyway, in his study, histopathology provided more accurate subtyping than fine needle aspiration in 8/25 cases, all benign diagnoses as myxoma and lipoblastoma. We believe that even incisional biopsy can be misleading, thereby, delaying a correct diagnosis, while to differentiate JAM from other malignant entities is mandatory, and this could guide to a more aggressive approach. Surely, this point requires a higher debate.In our case, as like as in previously reported 5-year-old boy case, biopsy was inefficacious because of collection of nonrepresentative tissue. Recently, Agrawal et al1 a minimum period of 2-year follow-up should be considered.Wide consensus about high recurrence of myxoid tumors in case of incomplete resection is found, and postoperative follow-up is important to detect relapse. As recurrence usually occurs within 18 months,Management of children with JAM requires a multidisciplinary approach and long-term postoperative follow-up. In case of hip tumefaction without pain, JAM could be considered for a diagnosis, and a malignant entity should be excluded. Adequate treatment requires surgical removal of the entire mass. Preoperative biopsies seem to be misleading for diagnosis in the pediatric age group.


Acute facial nerve palsy secondary to neuroendocrine adenoma of the middle ear (NAME) is a rare disorder. There is only one case report in the literature describing similar findings. A 50-year-old man initially presented to ENT clinic with a right-sided middle ear mass and normal facial nerve function. Over the next six days, he developed House-Brackmann grade II facial paralysis. He underwent urgent surgical exploration of the tympanic cavity and excision of the middle ear mass via a post-auricular approach. Histopathological and immunohistochemical analysis revealed NAME. Three weeks after the surgery, facial nerve function returned to normal. No recurrence was found at a 3-year follow-up. Acute onset facial palsy induced by NAME is an extremely rare disorder. For a patient already affected by hearing impairment resulted from middle ear mass, facial weakness can have a significant additional detrimental impact on their wellbeing. The early complete excision of tumor is recommended not only as a curative treatment but also restoration of facial function. Neuroendocrine adenoma of the middle ear (NAME) is an uncommon benign neoplasm of the middle ear, first reported by Hyams et al. and Derlacki et al. in 1976 . These tA 50-year-old man presented to our Ear, Nose, and Throat (ENT) clinic with a three-month history of right otalgia, aural fullness, and hearing loss. He had no other significant past medical or surgical history. On otoscopic examination, a rounded red mass was observed completely filling the right external auditory canal and obscuring the view of the tympanic membrane. The manipulation of the tumor with a Jobson-Horne probe revealed it as a soft, non-tender, and non-friable mass. The results of tuning fork tests showed that Rinne’s test was negative on the right side, and Weber’s test lateralized to the right side indicating right conductive deafness. Facial nerve function was bilaterally normal. Rest of the ENT and systemic examination indicated insignificant results. Pure tone audiometry confirmed conductive deafness with an air-bone gap of 40 db in the right ear .Based on our clinical assessment, the differential diagnoses included the benign and neoplastic lesions of the external and middle ear. Regarding the extent of the lesion, the performance of a high-resolution computed tomography (CT) scan of the temporal bone indicated the opacification of the right middle ear cavity and the external auditory canal EAC, . MoreoveSix days after his initial clinic visit, the patient developed House-Brackmann grade II right-sided facial palsy with complete eye closure on minimal effort as well as mild forehead and oral asymmetry. After the admission of the patient, the intravenous steroid was administered for him. Since the symptoms of pain and acute onset facial palsy are typical symptoms of malignancy, we performed an urgent exploration of the right ear under anesthetic. Intra-operatively, there was a red fleshy mass arising from the middle ear through a large tympanic membrane perforation and filling the right EAC. The biopsy of the lesion was performed and sent for frozen section. The examined sections revealed solid nests of cells composed of small rounded nuclei with evenly dispersed chromatin, inconspicuous nucleoli, and eosinophilic cytoplasm, and there was no invasion of the mastoid cells. All these findings raised the possibility of a benign neoplasm of the middle ear. We subsequently proceeded the treatment procedure with an exploration of the tympanic cavity via a post-auricular approach. The lesion was encasing the malleus and incus; however, there was no obvious bony erosion. In order to achieve the complete excision of the tumor, the malleus and incus were removed after disarticulation at the incudostapedial joint. Furthermore, the tumor did not involve the inner ear and mastoid. After the middle ear mass was completely removed, we noticed edema and hyperemia of the facial nerve from small bony dehiscence in the fallopian canal. In this regard, type III tympanoplasty was performed with temporalis fascia graft placed on the stapes head.The microscopic examination of the tumor indicated round to cuboidal cells with fairly deformed nuclei and absent nucleoli . Immunohistochemistry was positive for cytokeratins  and synaptophysin . Based on these findings, the lesion was diagnosed as NAME. The patient was discharged by a course of oral steroids and antibiotics the day after the surgery. The discussions held about the case in the Multi-Disciplinary Team meeting and it was decided to perform workup to rule out the metastatic potential of the tumor, which was negative. At his 3-week post-operative appointment, his facial nerve function returned to normal (House-Brackmann grade I). There was no evidence of recurrence at a 3-year follow-up.The neuroendocrine neoplasms of the ear are rare neoplasms accounting for less than 2% of all primary ear tumors ,9. AmongAccording to the literature, the etiology of NAME is unclear. Middle ear mucosa is derived from the endoderm; however, skin cells with neuroendocrine features are absent in the middle ear cavity. There are two theories explaining the development of such tumors in the middle ear First theory states that an undifferentiated pluripotent endodermal stem cell may still be present in the middle ear mucosal surface. Second describes that the stroma of the middle ear derived from mesoderm and neural crest and the tumor may originate from neural crest-derived stem cell. Regardless of the theory, a metaplastic transformation would be needed within the resident cells of the middle ear for dual population established within these tumors .The neuroendocrine adenoma of the middle ear classified under middle ear glandular neoplasms. Currently, the most widely used classification for middle ear glandular tumors is based on the detection of immunohistochemical markers and presence of metastases proposed by Saliba et al. in 2009 . AccordiThe incidence of the tumor is equal in both male and female patients. The NAME usually occurs in adult patients with the reported age ranges of 20-80 years and mean age of 45 years . HoweverThe otoscopic examination of NAME usually shows retrotympanic mass. Perforation of the tympanic membrane, with the extension of mass into the external auditory canal (EAC) is a rare finding with only a few cases reported in the literature ,15. Our Immunohistochemistry plays an important role in differentiating various middle ear glandular neoplasms, including NAME. Therefore, their obtained results from different therapeutic procedures should always be interpreted along with conventional histopathologic findings.de novo or up-regulated expressions of specific tumor antigens. The advantages of this method outweigh the traditionally used special enzyme staining methods since they detect only a limited number of proteins, enzymes, and tissue structures (The immunohistochemistry technique involves the application of monoclonal as well as polyclonal antibodies to find out the tissue distribution of an antigen of interest with regard to ructures . In NAMEructures . Our casSurgical excision is the treatment of choice. The extent of the procedure is determined by the size and involvement of adjacent structures . SmallerFacial palsy secondary to benign middle ear tumors is rare, and the diagnosis of these tumors is a challenging issue given their asymptomatic features. In addition to facial schwannoma, other benign tumors of the middle ear that can cause facial weakness include glomus tumor, adenoma, and haemangioma -27. LikeAcute onset facial palsy due to NAME is extremely rare. For a patient already affected by hearing impairment resulted from middle ear mass, facial weakness can have a significant additional detrimental impact on their wellbeing. We recommend the early complete excision of the tumor, not only as a curative treatment but also restoration of facial function. In this regard, further research is required to have a better understanding of the pathology, management,and long-term outcome of NAME.


Traumatic intracranial aneurysms are rare lesions that occur after blunt or primarily penetrating mechanisms. These are extremely fragile vessel injuries associated with significant morbidity and mortality, especially after rupture. Disease natural history, surveillance strategies, and management are based on small case series. Here we present a case of a 29-year-old male with a large epidural hematoma after blunt trauma, who underwent emergent surgical intervention. Three months postoperatively, he presented with unusual cerebral bleeding. Clinical suspicion prompted a conventional angiogram, which diagnosed a ruptured cortical traumatic intracranial aneurysm. The patient was urgently treated by surgical clipping with a good outcome. Traumatic intracranial aneurysms (TICA) are uncommon lesions traditionally associated with significant morbidity and mortality. They can arise indirectly from the stretching forces in blunt injury, or more commonly from direct injury from penetrating objects, iatrogenic sources, or secondary traumatic injury from adjacent anatomic structures such as the falx cerebri, clinoid process, or basilar skull. TICAs can be further classified histologically as true, false or pseudo, mixed, and dissecting. True TICAs result after an injury to the intima and media, with the adventitia remaining intact and forming the aneurysm wall . ComparaTICAs constitute 0.23% to 0.64% of all aneurysms, although their true incidence is probably under-diagnosed since most are asymptomatic until rupture . They ocThese lesions are complex and difficult to manage, largely due to the unknown natural history and anatomic fragility. Unfortunately, most patients present with hemorrhage, which is subsequently associated with a higher mortality ,5. The mSelect cases of adults with cortical TICAs after skull fracture, craniotomy, or external ventricular drain (EVD) placement have been described -9. Here A 29-year-old male with no significant past history presented with a Glasgow Coma Score (GCS) of 10 after falling out of a three-storeyed building onto his head. Imaging showed a 19-mm-thick left epidural hematoma with a 5-mm midline shift, as well as a comminuted left temporal bone fracture  was found originating from the cortical veins near the sylvian fissure. Hemostasis was achieved using bipolar cautery and Surgicel® . No aneurysms or unusual bleeding were noted. Immediate postoperative computed tomography showed a reduced mass effect with no atypical residual bleeding. He was discharged on postoperative day (POD) 17 to an in-patient rehabilitation unit with a GOS 3. Eventually, he was able to return home functionally independent and without neurologic deficits.Three months postoperatively, he presented after being found on the ground unresponsive at home. He had a GCS 9, and there were no external signs of trauma. Imaging showed an unusual pattern of subarachnoid hemorrhage and SDH in the left frontoparietal region without mass effect Figure .Given the uncertain history and atypical imaging, the patient underwent a four-vessel digital subtraction angiogram (DSA). Imaging showed distal left middle cerebral artery aneurysm Figure .This discovery substantially changed management. He promptly underwent a left craniotomy for aneurysm clipping. Stealth-guided imaging was used in designing the craniotomy. Intra-operatively, the dome appeared grossly composed of adventitia. The neck was dissected and a single clip was placed. Postprocedure angiogram demonstrated no residual filling. His recovery was uneventful, and he was discharged home on POD nine at his neurologic baseline with a GOS 5.Diagnosis of TICAs can be practically challenging. Some have proposed screening DSA for fractures in the carotid canal and those with penetrating injury entering the pterional region, going through the middle cerebral artery territory, and crossing the midline . We woulManagement suggestions for TICAs are primarily inferred from a small case series -5,10. BoIn our opinion, TICAs represent dangerous lesions that warrant prompt treatment upon discovery. Stealth-guided imaging is helpful to locate cortical aneurysms where precise exposure is important both in limiting manipulation and visualizing parent vessels. Delicate dissection through a scar or an edematous traumatic tissue is difficult and is done patiently with micro-instrumentation. TICA walls are extremely fragile, and the surgeon must be prepared for intra-operative rupture. Traditional clipping may not always be feasible if an aneurysm is deemed too delicate, has ruptured, or the neck is poorly defined. Alternatives include trapping, wrapping, or excision with or without parent vessel reconstruction. During the closure, any bleeding should be controlled with meticulous hemostasis under direct visualization.The precise etiology, natural history, and diagnostic histology, in this case, are unknown. No dedicated vascular imaging studies were obtained during the initial hospitalization, given the typical presentation, blunt injury, and the lack of unusual bleeding on pre- and post-op imaging as well as intra-operatively. Therefore, we cannot definitively state if the cause of the TICA was due to the initial significant blunt trauma, iatrogenic, or delayed friction from the craniectomy bone edges or protective helmet; and secondly, we cannot comment on the time of origin, development, and rupture. The lack of outward trauma with no history on re-presentation three months later made us most suspicious of spontaneous TICA rupture as the cause of bleeding; although bleeding from an unknown trauma, given his status post craniectomy could not be entirely ruled out. Lastly, after clipping, the aneurysm dome was not extirpated and sent for diagnostic histology due to lack of clinical benefit and unacceptable risk. However, pre-operative DSA was most consistent with a TICA. Thus, in our opinion, the most likely diagnosis and the subsequent management of a ruptured TICA was made based on clinical history without an obvious mechanism, bleeding pattern, and angiographic features.In conclusion, we present an uncommon case of a delayed rupture of a cortical TICA. The natural history of these lesions is unknown, and developing a comprehensive screening protocol is difficult. Clinicians should consider this disease entity with an unusual history or bleeding pattern since angiographic findings can dramatically change decision making. Treatment plans should be individualized and performed urgently. Craniotomy for clipping of a cortical TICA is feasible, even in post-surgical patients.