In psychological patients like our case, somatically expressed symptoms which can imply another psychological syndrome should be dealt with cautiously. In psychological patients like our case, somatically expressed symptoms which can imply another psychological syndrome should be dealt with cautiously. Although it can be found at all ages, it is more common among adults at the age of between 40 and 70 years and children under 10 years without any gender preference. While their typical sizes are up to 10 cm, they can emerge in larger sizes. However, very large mesenteric cysts are rare presentations of such benign tumors.Ganglioneuromas (GNs) are benign tumors in sympathic system, originating from neural crest cells. They are composed of ganglionic cells, Shawn cells, and neurites. Generally, this tumor is found in posterior mediastinum and retroperitoneum. In addition, there are a few rare cases of its formation in the adrenal medulla. GN tumors can be found at all ages. However, the rare cases of GN tumor of the adrenal gland are more common in children and young adults. GNs are often hormonally silent and present no symptom; hence, the clinical diagnosis is difficult and virtually in all cases accidental.Both GNs in adrenal gland and giant MCs are rare phenomena. However, their occurrence in a schizophrenic patient presenting as a pseudocyesis syndrome makes the case more noteworthy. To the best of our knowledge, there has been no report on such case in the literature and this article is the first case report on this subject.2The patient was a 55‐year‐old man, who previously diagnosed with schizophrenia and had been hospitalized several times because of the disease and claimed that he was pregnant. Unfortunately, due to his mental disorder, this abnormal statement was neglected. Therefore, the schizophrenia treatment continued until abdominal pain, coffee‐ground vomit, and abdominal distension occurred. At this stage, the patient was referred to the department of general surgery. A written consent was obtained from the patient for publication of this case report and any accompanying images.Apparently, the patient was cachectic with bilateral temporalis muscle atrophy which was not consistent with his age. Additionally, it was observed that the abdomen was evenly huge and distended. In palpation, a giant firm mass was detected in all over the abdomen. However, no mass with a definite boundary was observed in the examination. In the epigastric region, there was a mild tenderness without rebound tenderness. During the examinations, bowel sound in each quadrant of the abdomen was auscultated, and it was slightly heard in the left upper quadrant. Due to the patient's psychiatric problems, the only source of patient history was his relatives. According to their statements, the patient had experienced several times of coffee‐ground vomiting. Additionally, the patient had small bowel obstruction symptoms with low defecation frequency since a long time ago. This condition had continued until 11 days ago when there was no defecation and he had only gas passing. Since the mass was observed in the abdomen, abdominopelvic ultrasound was requested, and the result showed a cystic hypoechoic mass that had spread all over the abdomen.Computed tomography scan (CT scan) depicted a 21 cm × 33 cm nonenhancement monolocular cyst in the right part of the abdomen , and in 33% of cases, it has been in the colon mesentery (mostly arose from the right colon).Although the exact etiology of these masses is unknown, it is believed that factors such as benign proliferation of ectopic lymphatics, abdominal trauma, lymphatic obstruction, and degeneration of some lymph nodes may be involved in forming MCs.There is no specific pathognomic symptom for MCs. Depending on the expansion and location of the tumor, there may be a spectrum of symptoms ranging from no symptoms and random diagnosis in radiographic investigation to symptoms including abdominal mass, vomiting, obstipation, and abdominal distension. Furthermore, it may express chronic signs such as anemia and weight loss.In the case of late intervention, MCs may lead to some complications such as rupture, volvulus, obstruction, and torsion. In these scenarios, an emergent surgery would be the best approach.Differential diagnosis of MCs are (a) cysts with lymphatic origin such as simple lymphatic cyst or lymphangioma, (b) cysts with mesothelial origin such as simple mesothelial cyst, and benign or malignant cystic mesothelioma, (c) enteric cysts such as enteric duplication cyst, (d) cysts with urogenital origin, (e) dermoid cysts such as mature cystic teratoma, and (f) nonpancreatic pseudocysts such as traumatic and infectious cysts.Computer tomography (CT) and ultrasound (US) can be effective tools in the diagnosis of abdominal masses. Magnetic resonance imaging (MRI) is also helpful in diagnosis. US is employed in order to differentiate cystic from solid masses and determine the origin of mass. CT is also a useful tool in differentiating simple cysts (monolocular cysts) from multilocular cysts. Moreover, the origin of the cyst and its relation with the surrounding organs can be displayed in CTs. MRIs are more accurate compared with CTs and can provide more details; however, in most cases, CTs are sufficient for making an accurate diagnosis.The best treatment option for these cysts is either complete resection of the mass with or without the mesenteric origin or, in some cases, segmental resection of the involved intestine. This operation lowers the risk of malignancy transformation and can be performed through either laparoscopic or open surgery. In open surgeries, 5‐8 days of postoperation hospitalization is required, while in laparoscopic surgery 1 or 2 days is enough. However, in the larger sizes of these masses, the possibility of the laparoscopic surgery is limited. Furthermore, when the patient is not a good candidate for surgery, marsupialization, internal drainage, and aspiration are considered as suitable alternatives. However, the possibility of recurrence, infection, and fistulization increases.The second tumor, GN, is a benign tumor originating from neural crest cells, and its incidence rates in retroperitoneum and posterior mediastinum are 32%‐52% and 39%‐43%, respectively. However, in some rare cases, they might be found in adrenal medulla.Ganglioneuromas present no pathognomonic symptoms and often are hormonally silent. They will cause no signs in patients, unless they are giant tumors and compress the surrounding organs or they are endocrinologically active. However, none of these symptoms are specific to GNs.In sonography, these tumors appear as homogenous and hypoechogenic lesions. In CT, they are revealed as solid homogenous and hypodense masses. In MRI, T1‐weighted images usually show a lobulated homogenous hypointense mass, and T2‐weighted images show a heterogeneous one. However, none of these preoperational investigations can lead to a definite diagnosis. In some cases, these tumors cause an increase in urinary or plasma catecholamine or an increase in the level of steroid hormones such as cortisol and aldosterone. Nevertheless, these signs are not specific in diagnosis and sometimes cause misdirection. In fact, a definite diagnosis can only be made through postoperation histopathological examinations.There is a little probability of malignancy in GNs; therefore, complete resection of the mass is required. It is not necessary to continue the treatment with adjuvant therapy; in other words, GNs have a good prognosis.In conclusion, computed tomography scan and magnetic resonance imaging are both employed in the diagnosis, and the results are verified with the pathology report. It is noted that both of these masses required to be excised through either open or laparoscopic surgery, depending on the size of the tumor.The probability of random formation of the two tumors of ganglioneuroma and lymphangioma in a schizophrenic patient is almost zero, and it seems that some kind of syndrome is the causative factor. This probability encouraged us to search for reports of a syndrome along with probable simultaneous occurrence of these two tumors in the literature. Eventually, the only conceivable hypothesis was the involvement of the Noonan syndrome in which ganglioneuroma or lymphangioma might be observed.This patient was a known case of schizophrenia for several years. On the other hand, pseudocyesis is reported as a rare syndrome in schizophrenic patients.In further investigations, a GMC was detected, whose extreme size was rare, considering its benign nature. Moreover, during radiological examinations a ganglioneuroma was observed in adrenal which is also known as an adrenal incidentaloma.4In conclusion, it is advised that in psychological patients, somatically expressed the symptoms which imply another psychological syndrome should be dealt with cautiously.There is no conflict of interest to declare.AN: contributed to study concept and design and critically revised the manuscript for important intellectual content. BB and AS: acquired data, performed histopathological examinations, and reported images. FJ: drafted the manuscript and supervised the study. All authors: read and approved the final manuscript.The present study is in compliance with ethical standards and standards of research involving humansAUTHOR: 'Ethical statement' has been changed as 'Ethical approval'. Check and confirm.Because the patient was not mentally in a proper condition to sign the informed consent, therefore, his legal guardians signed the form for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor‐in‐Chief of this journal.


Torsion is the most common gynecologic emergency of the adnexal mass occurring during pregnancy. We report the clinical data of a case of twin pregnancy with adnexal torsion after in vitro fertilization (IVF) and embryo transfer, in which the patient underwent surgery for adnexal detorsion and experienced preterm delivery. One child died as a neonate, and the other child was diagnosed with hematological disease, which, to our knowledge, has not been reported previously. We also performed a systematic literature review to increase knowledge of the need for prompt surgical intervention.The patient was a 32-year-old pregnant woman, who presented to our center with acute onset of stabbing and non-radiating continuous lower left abdominal pain after urination, of 6 hours duration.Physical examination revealed that the lower abdominal tenderness was worse on the left side, and there were no signs of peritonitis. Transvaginal ultrasonographic examination indicated a multiloculated left ovary measuring 12.1 × 7.1 cm with sparse blood flow. The size of the largest cyst was 7.2 × 6.6 cm, the right ovary appeared normal, and two live fetuses were seen.laparoscopy was performed 1.5 hours later, which revealed a cystic and multilocular left ovary with a black purplish surface and thin wall. The left ovary and left fallopian tube had undergone 720° torsion (3 rotations), and detorsion was performed laparoscopy.The left adnexa recovered to near normal appearance 20 minutes postoperatively. The patient was discharged from hospital 5 days postsurgery, without complications. Unfortunately, the patient delivered two preterm babies at 30 weeks of gestation.We should be alert to the possible risk of adnexal torsion in pregnant women after IVF. Adnexal torsion necessitates prompt surgical intervention, detorsion and preserving ovarian function are the main treatment methods. Furthermore, the possibility of recurrence, and pregnancy outcomes for the patient, as well as newborn health, should be considered. We report the clinical data of a case of twin pregnancy with adnexal torsion after IVF and embryo transfer, and review the related literature in detail.Adnexal torsion is a high-risk situation for patients, presenting as acute abdominal pain during pregnancy, and it is a serious complication of assisted reproductive therapy (ART). With an estimated incidence of 1 in 5000 natural pregnancies,2The patient was a 32-year-old pregnant woman, who presented to our center for acute onset of stabbing and nonradiating continuous lower left abdominal pain after urination, of 6 hours duration. She had no history of prior abdominal trauma or surgery. Her husband had a diagnosis of male factor infertility. After ovulation induction, she underwent IVF with the gonadotropin-releasing hormone (GnRH) antagonist protocol. Twelve days later, 11 oocytes and 3 embryos were obtained. On day 3, ultrasonography showed that the right ovary measured 4.7 × 3.5 cm, the left ovary measured 6.6 × 6.0 cm, and the maximum depth of the recto-uterine pouch free fluid was 3.9 × 2.3 cm. Two fresh embryos were transferred and this was followed by a 60 mg progesterone injection once a day, and oral dydrogesterone 10 mg twice a day. On day 30 after embryo transfer, transvaginal ultrasonography identified intrauterine twin pregnancy, and the left ovary measured 13.2 × 13.1 cm. The relatively large anechoic rim sizes were 7.1 × 5.8 cm, 7.6 × 5.6 cm, and 7.3 × 5.1 cm, and the posterior fornix fluid measured 5.2 × 2.6 cm. The outpatient doctor suggested the patient should avoid strenuous activities. On day 45 after embryo transfer, the size of the left ovary was 13.8 × 9.5 cm. Six hours before seeking treatment, the patient experienced sudden onset of severe progressive abdominal pain after urination, and pain on the left lower side was obvious. She could not obtain relief form the pain after changing position, and the pain was accompanied by nausea, vomiting. Physical examination revealed that the lower abdominal tenderness was worse on the left side, and there were no signs of peritonitis. Transvaginal ultrasonography indicated a multiloculated left ovary measuring 12.1 × 7.1 cm with sparse blood flow; the size of the largest cyst was 7.2 × 6.6 cm, the right ovary appeared normal, and the two live fetuses were seen. Laboratory blood examination revealed increased white blood cell count , neutrophil granulocyte percentage (92%), and cancer antigen 125 (CA125) concentration (196.9 U/ml). Laparoscopy was performed 1.5 hours later, which revealed that the left ovary was cystic and multilocular with a black purplish surface and thin wall. The left ovary and left fallopian tube had undergone 720° torsion (3 rotations), and detorsion was performed laparoscopy  of cases occur between the 10th and 17th weeks of gestation, compared with 36.7% (11/30) occurring during the 15th and 16th weeks of gestation. In addition, the incidence decreases to <5.9%, if torsion does not occur before the gestational age of 20 weeks. The right adnexa is most commonly involved,–8 and the mass diameter in adnexal torsion events is often 6 to 10 cm,9,10  treatment;2.enlarged, hyperstimulated ovaries;3.pregnancy; and4.adnexal torsion. Oelsner et al reported that the most frequent histology in ovarian cystectomy is a corpus luteum cyst (12/31); 58% (18/31) of removed cysts are functional. In 1946, Way reported the first conservative management of adnexal torsion. In the report, adnexal structures were detorsed in 15 cases, and patients underwent cystectomy. Recently, detorsion via laparoscopy has become nearly standard treatment.–18 Some reports indicate that adnexal torsion can be treated by laparoendoscopic single-site surgery or transvaginal ovarian cystectomy during pregnancy at 25 to 27 weeks.,20 Although recurrence of adnexal torsion with conservative management is rare, it can threaten ovarian function and fetal safety.A corpus luteum cyst is the second most frequent pathology associated with adnexal torsion, with 33.3% (6/18) of patients with this pathology developing torsion. suggested that detorsion should be performed as soon as clinical symptoms appear in cases of recurrence, and oophoropexy is strongly recommended to avoid recurrent torsion. Recurrent adnexal torsion can also be prevented by adnexal detorsion and shortening the utero-ovarian ligament.,21 Seven cases of ovarian torsion were reported by Hosny et al; 2/7 cases had OHSS, and 50% of the patients showed clinical symptoms and hemorrhagic cysts. Fortunately, there was no recurrence in these 2 patients, who underwent detorsion and oophoropexy. Based on the above cases and investigations, it can be concluded that detorsion and oophoropexy can be performed easily and quickly and this approach is more appropriate for ovarian torsion in pregnant women and OHSS patients.We performed a systematic literature review to identify case reports of adnexal torsion during pregnancy after IVF. We identified 38 cases; 2 of these cases developed adnexal torsion recurrence, and both underwent three operations. Djavadian et al–8,19,23  delivered healthy infants. To our knowledge, our case is the first report of a pregnant patient undergoing surgery for adnexal detorsion and experiencing preterm delivery with neonatal death in 1 baby and hematological disease in the other. With our patient, we carefully considered that anesthesia-related drugs for the fetus were able to be used during pregnancy, as for all pregnant women. Preterm birth is the most frequent cause of neonatal death, and is also the second largest direct cause of death in children younger than 5 years of age, worldwide. Furthermore, multiple factors imply that using ART increases the rate of preterm birth.According to the related literature, most cases suggest that surgical treatment is beneficial to treat adnexal torsionIn conclusion, adnexal torsion is a rare but acute complication that generally occurs in pregnant patients after IVF. With the increased use of ART, we should be alert to the possible risk of adnexal torsion in pregnant women after IVF, detorsion and preserving ovarian function were the main treatments. However, the following concerns must also be considered: ovarian function, preterm birth, neonatal death, and the health of the child. Therefore, large clinical studies are still required.https://en-author-services.edanzgroup.com/) for editing a draft of this manuscript.We grateful acknowledge the support and assistance provide by all staff in The First Hospital’ Reproductive Medicine Center and thank the editors and reviewers for their help with this manuscript. We thank Jane Charbonneau, DVM, from Edanz Group (Methodology: Yanhong Liu, Dongyun Jia.Supervision: Tian Tian.Original draft: Meiling Yu, Qi Xi.Writing – review and editing: Meiling Yu, Qi Xi.


We performed incisional biopsies of the bilateral mandibular lesions and resections of the bilateral maxillary lesions. Surgical and histopathological findings established a diagnosis of florid COD in the bilateral mandible and maxilla, simultaneously complicated by multiple SBCs in the bilateral mandible. The ADC value of cystic component is useful for the diagnosis, if the COD-associated SBC exhibits solitary, nonspecific, or rare imaging findings.A simple bone cyst (SBC) is an intrabone cavity without epithelial lining, which occasionally occur with fibrous-osseous lesions. The imaging finding of the scalloped border and conserving the lamina dura, periodontal ligament spaces, or follicular spaces are considered radiographically pathognomonic of SBC. SBC has been reported to occur with fibrous-osseous lesions, including cemento-osseous dysplasia (COD). Computed tomography (CT) imaging findings are sparse, and there are no reports of magnetic resonance imaging (MRI) findings, including apparent diffusion coefficient (ADC) value calculated by diffusion-weighted MRI for the florid COD-associated SBC. We report a case of a 39-year-old woman who was referred to our hospital because a panoramic radiograph of her right mandible showed asymptomatic radiolucency in the apical molar area. CT images showed a low-density lesion in the periapical areas of the right mandible, which accompanied a well-defined, high-density lesion, and a mixed low/high-density lesion with an unusual marked irregular border in the left mandible. The MRI showed cyst-like extremely hyperintense signals on fat-suppressed T2-weighted images at the right mandibular low-density and the left mandibular mixed low/high-density areas. ADC map showed high values (over 2.5 × 10 A simple bone cyst (SBC) is a pseudocyst, which is diagnosed based on the finding of an intrabone cavity without epithelial lining . SBC appThis case report was approved by our institutional ethics committee (No. 18651). All procedures followed were in accordance with the ethical standards of the responsible committee on human experimentation  and with the Helsinki Declaration of 1975, as revised in 2008. Informed consent was obtained from the patient for being included in the study.A 39-year-old woman had asymptomatic radiolucency of the right mandible on X-ray radiographic examination for caries treatment at a primary dental office. She was referred to our hospital for further radiographic examination and treatment of the lesion. Her medical history revealed postoperative uterine cervical cancer. She had no significant family history. Clinical findings of extra- and intraoral appearances were unremarkable.−3 mm2/s) at cystic areas in the bilateral mandible  of COD-associated SBCs occurred in women with a mean age in the 40s, and the majority 95.7%) of the cases occurred in the mandibular molar area [.7% of thWe have reported an interesting case of florid COD-associated multiple SBCs, which showed a rare CT finding of mixed low/high-density with the marked irregular border and high ADC values of cystic components on MRI. The ADC value of cystic component is useful for the diagnosis, if the COD-associated SBC exhibits solitary, nonspecific, or rare imaging findings.


Osteoid osteoma (OO) is a benign bone tumor that usually presents between 10 and 35 years of age. The metaphysis and diaphysis of the femur and tibia are the typical locations. The diagnosis is usually straightforward when images reveal a radiolucent nidus surrounded by reactive sclerosis. However, the diagnosis is more difficult when it occurs at atypical locations with nonspecific and misleading appearance on images. OO may mimic juvenile idiopathic arthritis (JIA), bone infection, or malignancy. We present a 14-year-old male with a 4-month history of left hip pain. His pain was worse with playing hockey and lacrosse and in the morning and sometimes woke him up at night. His examination was significant for pain with flexion and external rotation of the left hip and for mild limitation of full external rotation. Blood work revealed normal complete blood count, erythrocyte sedimentation rate, and C-reactive protein. Left hip X-ray was unremarkable. Left hip MR arthrogram showed marked edema of the medial and posterior walls of the left acetabulum. CT-guided biopsy of the left acetabulum showed unremarkable flow cytometry and chronic inflammatory component raising concern about chronic recurrent multifocal osteomyelitis (CRMO). Bone scan revealed focal increased uptake in the left acetabulum and no additional abnormality. Repeat MRI with intravenous contrast showed a left hip effusion, focal synovial enhancement in the medial left hip, and acetabula edema. The patient failed treatment for presumed JIA and CRMO with nonsteroidal anti-inflammatory drugs (NSAIDs), steroids, methotrexate, and adalimumab. CT scan of the left hip was performed for further evaluation of the bone and showed 11 × 6 mm low attenuation focus with subtle internal nidus in the posteromedial aspect of the acetabular rim, suggestive of intra-articular OO. Radiofrequency ablation was performed with no complications, and the left hip pain improved. The atypical location resulted in delay of diagnosis for 12 months after presentation. We highlight the diagnostic pitfalls observed in atypical OO locations and the difficulties this creates with making the diagnosis. OO mimicking JIA has previously been described. We submit CRMO as another differential diagnosis which may be mimicked and demonstrate the vital role of CT scan in the diagnosis. Osteoid osteoma (OO) is a benign tumor of bone, which was first described by Jaffe in 1935 . It typiThe typical clinical presentation of OO is local pain that is most severe at night and can be relieved by nonsteroidal anti-inflammatory drugs (NSAIDs) . DependiOO can be managed conservatively. Invasive treatment is considered when pain is very severe and refractory to medications. Options include open or arthroscopic surgical excision of the tumor, CT-guided radiofrequency ablation, or CT-guided laser photocoagulation . TreatinOO is frequently misdiagnosed when it occurs in atypical locations as it may mimic juvenile idiopathic arthritis (JIA), bone infection, or malignancy . Here, wA 14-year-old male patient presented to our rheumatology clinic with a 4-month history of left hip pain. The pain was more severe in the morning and sometimes woke him up at night and also increased with practicing sports (hockey and lacrosse). His pain is 1-2 of 10 when he is resting and 7-8 of 10 when he is playing sports. There was no history of trauma. There was no associated swelling or morning stiffness, and no pain in any other joints.The patient was initially evaluated in the orthopedics clinic. Initial blood work revealed normal complete blood picture (CBC), erythrocyte sedimentation rate (ESR), and C-reactive protein (CRP). Left hip X-ray was normal. Initial MR arthrogram of the left hip showed marked edema of the medial and posterior walls of the left acetabulum . The patWith persistence of pain, CT-guided biopsy of the left acetabulum was performed and revealed unremarkable flow cytometry, and histopathology showed cortical bone with rare foci of marrow elements and a mild chronic inflammatory component and no evidence of malignancy. The patient was referred to the rheumatology clinic for further evaluation. Initial examination showed pain with flexion and external rotation with mild limitation of full external rotation of the left hip. JIA and CRMO could not be ruled out given his clinical examination and the histopathology result. The patient was started on prednisone 50 mg (0.7 mg/kg) once daily , and naproxen dose was increased to 375 mg twice daily. Two months later, left hip pain continued, and he started to experience pain of the right hip, right metacarpophalangeal (MCPs), and left clavicle. He was started on weekly methotrexate in addition for presumed diagnosis of JIA and/or CRMO. Bone scan was unremarkable apart from focal increased uptake in the left acetabulum .After two months of methotrexate treatment, the patient reported improvement in the intensity and frequency of the left hip pain; however, he was still experiencing pain with certain positions. The pain of the right hip, left clavicle, and right MCPs resolved completely. Due to persistence of the left hip pain, adalimumab 40 mg subcutaneous every 2 weeks was added, with no significant improvement in pain. He reported increased need for NSAIDs . Repeat contrast-enhanced MRI showed left hip effusion that decreased from before, with medial focal synovial enhancement and edema of the acetabulum . These fOver half of OO lesions occur in the lower extremity long bones, with the proximal femur being the most common location. The acetabulum is a rare site accounting for only 1% of all OOs . DiagnosDespite the advent improvement in imaging, diagnosis of atypical location of OO remains a challenge. Radiographic features of hip IAOO may differ from extra-articular lesions. Unlike extra-articular locations, plain radiographs provide only subtle findings due to the absence of perilesional sclerosis or periosteal reaction . On MRI,Surgical treatment for acetabular OO is challenging because of limited surgeon experience with therapeutic procedures and the complex anatomical location with proximity to the sciatic nerve and triradiate cartilage. Although numerous surgical approaches to acetabular OO have been described, including open surgery, arthroscopy, and CT-guided approaches, optimal management for acetabular OO has not been established . CT-guidOOs in atypical locations are difficult to diagnose due to nonspecific presentation and misleading appearance on imaging (MRI and bone scan). Thorough history and clinical examination together with CT scan are vital for diagnosis.


Five patients received adjuvant radiotherapy and chemotherapy after surgery. One patient with CCAC in UD associated with URA was treated with radical IMRT and adjuvant chemotherapy. The eight patients were followed up for an average of 7.9 years; in seven cases, there was no evidence of disease recurrence, while one patient relapsed and died after 1.5 years of treatment. On the basis of these findings, locally advanced CCAC in UD associated with URA can be effectively treated with radical IMRT.Clear cell adenocarcinoma of the cervix (CCAC) with genitourinary malformations is rare. Here, we report a case of CCAC in uterus didelphys (UD) associated with unilateral renal agenesis (URA) that was treated with intensity-modulated radiotherapy (IMRT) and high-dose rate intracavitary brachytherapy (HDR-ICBT). We also retrospectively reviewed the medical records of CCAC cases with genitourinary malformations treated at the National Cancer Center/Cancer Hospital  between December 2006 and June 2017. Eight cases of this rare condition were identified by pathologic diagnosis. Seven patients received surgical treatment including radical hysterectomy ( Clear cell adenocarcinoma of the cervix (CCAC) is rare, accounting for 4–9% of cervical adenocarcinomas . CCAC haHere, we report a case of CCAC in UD associated with URA treated with radical intensity-modulated radiotherapy (IMRT) and high-dose rate intracavitary brachytherapy (HDR-ICBT) at the National Cancer Center/Cancer Hospital  between December 2016 and June 2017. The patient's medical records were retrospectively reviewed, and we also carried out a comprehensive review of seven cases with this rare joint condition reported in the literature –8. Cliniin utero diethylstilbestrol (DES) exposure. Left modified radical mastectomy was performed on April 29, 2014. Postoperative histopathology confirmed infiltrating ductal carcinoma of the breast. The clinical stage (pTNM) was T2N1M0. There was strong positive staining for HER2, but negative staining for ER and PR by immunohistochemistry.The patient was a 65-year-old female, gravida 4/para 3 with no abnormalities during her deliveries. The limited record of her medical history showed no Following the surgery, the patient was treated with a docetaxel/carboplatin/trastuzumab regimen for six cycles and achieved complete clinical remission after completion of therapy. No tamoxifen or aromatase treatment was used.The patient was transferred to our institution on October 12, 2016 (15 years after menopause), after experiencing genital bleeding for 1 month. Diagnostic curettage was performed along with histopathologic analysis of an endometrial polyp. Speculum examination revealed mild ulceration of the cervix (~2 cm), and pelvic and rectal examinations showed right parametrial extension, with the patient complaining of pain during pelvic examination. A single cervix was observed by colposcopy under anesthesia. Pathologic analysis of a cervical biopsy suggested CCA. There was strong positive staining for cervical markers  by immunohistochemistry. A computed tomography (CT) scan of the chest and abdomen revealed UD, left renal agenesis, and no evidence of lymph node metastasis or distant metastasis. Magnetic resonance imaging (MRI) showed bilateral uterine malformation and local fusion of bilateral cervix. Abnormal nodules 2.5 cm in diameter were observed in the right parametrial area, along with polyps 1.5 cm in diameter in the uterine cavity. The human papillomavirus (HPV) test was negative. Tumor makers such as SCC, CA125, and CA19-9 were within normal limits. The diagnosis was CCAC IIB .2) and carboplatin (area under the receiver operating characteristic curve four)] from December 9, 2016, to January 12, 2017, followed by whole-pelvic CT-based IMRT. The clinical target volume (CTV) comprised the cervix, parametrium, uterus, upper third of the vagina, and regional lymph nodes . The upper field border was at the level of the L4/L5 interspace, and the caudal field border was at the lower margin of the obturator foramen  delivered to point A. The doses were referred to an adapted point A, 2 cm lateral and superior to each cervical structure. ICBT was performed by placing a tandem 6 cm in length into one side of the uterine canal, with two fractions of 7 Gy delivered to both the right and left sides. Complete response was achieved with therapy.After nearly 3.2 years of follow-up, there were no clinical or radiologic signs (by MRI performed every 6 months) of local or locoregional relapse. Acute toxicity was mild and there has been no significant late toxicity since the completion of treatment, as evaluated according to the National Cancer Institute Common Terminology Criteria for Adverse Events (CTCAE) v4.03.UD, which has a reported incidence of 1:1,026 , is causAlthough the etiology of CCAC is unknown, the currently held view is that a history of intrauterine exposure to DES plays a role based on a report that intrauterine exposure to non-steroidal estrogens, particularly DES, predisposed young women to the development of CCA . The aveThe clear cells of the cervix are thought to arise from mesonephric and Müllerian ducts . Some reThe main clinical symptom of CCAC is irregular vaginal bleeding accompanied by abdominal discomfort . Pelvic In patients with adverse prognostic factors, surgery is followed by chemotherapy and/or radiotherapy. For locally advanced stage II CCAC, neoadjuvant chemotherapy can improve the success rate of surgical resection and enhance sensitivity to radiotherapy . One patIn conclusion, the treatment of CCAC with genitourinary malformations is clinically challenging because of the rarity of this condition. IMRT and HDR-ICBT can be used to treat cases of locally advanced CCAC in UD associated with URA.The raw data supporting the conclusions of this article will be made available by the authors, without undue reservation.The studies involving human participants were reviewed and approved by the Cancer Hospital, Chinese Academy of Medical Sciences. The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.CL and MH conceived the study and wrote the manuscript. NL, JA, XL, and SX collected and analyzed the data. LW provided expert clinical knowledge. All authors critically edited the manuscript for intellectual content.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Colostomy via the intraperitoneal route is often performed during laparoscopic Hartmann’s operation or abdominoperineal resection. Internal hernia of the small intestine often occurs after colostomy. This report shows a rare case of internal hernia of the stomach associated with sigmoid colostomy after laparoscopic abdominoperineal resection for rectal cancer.The patient was a 79-year-old woman with a sigmoid colostomy. Computed tomography scan showed a markedly distended stomach in the space between the lifted sigmoid colon and the lateral abdominal wall. Laparoscopy revealed that the body of the stomach had passed through a hernia orifice located between the lifted sigmoid colon and the left lateral abdominal wall. The dislocated stomach was restored to its normal position, and the lateral defect was closed with the lateral peritoneum and the lifted sigmoid colon laparoscopically.Internal hernia associated with colostomy can lead to not only obstruction of the small intestine, but also obstruction of the stomach. We reported a successful case of the suture repair for the internal hernia of the stomach associated with colostomy. With progressive developments in laparoscopic rectal surgery, anus-preserving procedures have become available for patients with rectal cancer . HoweverMajor complications associated with colostomy after HO or APR are stoma stenosis, retraction, prolapse, parastomal hernia, and intestinal obstruction . Major cWe herein present a rare case of internal hernia of the stomach associated with sigmoid colostomy.A 79-year-old woman has presented with advanced rectal cancer located on the dentate line. Computed tomography (CT) detected no metastasis. Laparoscopic APR for low rectal cancer was performed, and a sigmoid colostomy was created via the intraperitoneal route during laparoscopic APR. The histopathology of the resected tumor revealed a well-differentiated adenocarcinoma. The patient was therefore classified as T2N0M0 and Stage IIA Fig. . Six monA colostomy is created in patients with rectal malignancies. The postoperative complications associated with colostomy are stoma stenosis, retraction, prolapse, parastomal hernia, and intestinal obstruction. Intestinal obstruction has been reported to occur in 3.5 to 7.2% of cases after stoma creation –8. The mIn this case, a part of the stomach entered into the hernia orifice as the advanced part, and the pyloric part was pressed by the lifted colon. It led to the output obstruction of the stomach, further expanding the stomach worsening the hernia. In this case, we closed the hernia orifice by fixing the lifted sigmoid colon to the abdominal wall. As a result, this treatment was successful.Laparoscopic colorectal surgery has developed progressively in recent years, and it has many advantages compared with open surgery, including better cosmesis, lesser pain, earlier postoperative recovery, and fewer intraperitoneal adhesions. Intraperitoneal adhesions sometimes cause postoperative intestinal obstruction. Laparoscopic surgery results in fewer cases of postoperative intestinal obstruction compared with open surgery . Many caInternal hernia associated with colostomy can lead not only to obstruction of the small intestine, but also to obstruction of the stomach. The present case is the first report on internal hernia of the stomach associated with colostomy. During laparoscopic HO or APR, creation of colostomy via the extraperitoneal route should be considered to avoid IHAC.


Testicular tumors represent 1% to 1.5% of all tumors in men. Those derived from Leydig cells are rare and account for 1% of testicular tumors. Leydig tumor cells can produce steroid hormones such as estrogen, progesterone and testosterone. The amount and type of hormones secreted by these tumors may produce complicated clinical characteristics in these patients.Here, we report a patient with azoospermia, a testicular Leydig cell tumor (LCT), and elevated plasma testosterone levels. We describe the diagnostic and therapeutic experience of this case, and our follow-up of the patient's clinical indicators and fertility status.The patient was diagnosed with azoospermia and a testicular LCT.The patient underwent testicular tumor removal and long-term follow-up.After 4 months of follow-up, the patient's semen examination index significantly improved and his wife became naturally pregnant. At 4 months of gestation, the fetus was delivered because of a ruptured amniotic cavity. Twenty-six months after tumor removal, the patient's sex hormone levels had completely returned to normal and spermatogenic function had partially recovered, but there was no natural pregnancy with his partner.For LCTs, testis sparing surgery may provide a safe and feasible option to restore spermatogenic function, although longer-term follow-up is required. Drug assistance may be required to maintain spermatogenic function and achieve fertility, and further research is required. Testicular solid tumors are almost malignant. LCT is a rare, mostly benign tumor in adults, although 10% to 20% may be malignant. Therefore, the identification of LCT and other malignant testicular tumors is important for guiding clinical treatments.Testicular tumors account for 1% to 1.5% of all tumors in men, and Leydig cell tumors (LCTs) account for only 1% of total testicular tumors.,5,6 Children may exhibit precocious puberty with symptoms such feminization (10% of patients may display breast development), genital enlargement, appearance of pubic hair, and deepening voice. Adult patients with LCT are mainly treated for a testicular mass or infertility. About 30% have symptoms of feminization, of which male breast development is the most common, and decreased sexual desire, erectile dysfunction, infertility and testicular and prostate atrophy. The different clinical symptoms may be related to the following mechanisms:Approximately 20% of LCTs occur in children aged 6 to 10 years, and 80% occur in adults aged 20 to 60 years. The clinical characteristics and hormone levels associated with LCTs vary greatly.(1)steroidogenic tumor cells produce estradiol (E2), progesterone and testosterone (T), and the amount and type of secreted hormones may affect the clinical characteristics of patients;(2),5,6the activity of aromatase is low in childhood and high in adults. Different levels of aromatase activity will affect the conversion of T to E2 and cause different clinical features.,8 The present surgical methods mainly include radical orchiectomy and testis sparing surgery (TSS), but the use of these techniques remains controversial. In recent years, several studies have confirmed the safety and effectiveness of TSS for adult LCTs.,9–11 For patients with benign tumors, especially children, and for non-fertility, and bilateral testicular disease, TSS surgery is recommended according to rapid intraoperative pathological results. Most patients return to normal after surgery, but some cases require adjuvant medication for fertility. Therefore, we retrospectively analyzed a case of LCT exhibiting azoospermia and elevated plasma T levels and present here the diagnostic and therapeutic experience.In addition to the above clinical manifestations, the diagnosis of LCT should also be performed with auxiliary tests such as hormone levels, tumor markers, B-ultrasound, and magnetic resonance (MR). Some LCTs secrete T and/or E2, but tumor markers such as alpha-fetoprotein (AFP), human chorionic gonadotropin (hCG) and lactic acid dehydrogenase (LDH) are often negative. B-ultrasound and MR can diagnose the tumor but cannot determine if it is benign or malignant. Clear diagnosis before surgery is very difficult and diagnosis still largely depends on postoperative pathological examination. LCTs are not sensitive to radiotherapy and chemotherapy, so surgical resection is currently the only effective treatment.2The study protocol was approved by the Ethics Committee of the First Hospital of Jilin University (No. 2019-239). Written informed consent was obtained from all patients.The patient was a 27-year-old married man who came to the Reproductive Medicine Center of the First Clinical Hospital of Jilin University for infertility treatment. The patient had experienced a right testicular bulge accompanied by tenderness, but no loss of sexual desire and erectile dysfunction. Physical examination revealed male appearance, body hair and no male breast development. The penis had developed normally without foreskin phimosis. When standing, the right scrotum drooped and the testicle was heavy with an approximate volume of 20 mL. Tenderness was positive, and the light transmission test was negative. The left testicle was about 8 mL, and there were no varicocele or other abnormalities. The bilateral groin did not touch the enlarged lymph nodes. A routine semen test indicated azoospermia. Hormone measurement results were: follicle-stimulating hormone (FSH): 0.11 (1.50–12.40) mIU/mL, luteinizing hormone (LH): <0.10 (1.70–8.60) mIU/mL, E2: 47.8 (25.8–60.7) pg/mL, prolactin: 217.2 (86.0–324.0) μIU/mL, T: 28.72 (9.90–27.80) nmol/L, LDH: 162 (135–226) U/L, AFP: 0.64 (<7.0) ng/mL, hCG: 2.39 mIU/mL. Testicular ultrasound showed the left testicular size was 34 × 22 × 16 mm (8.5 mL), the right testicular size was 46 × 28 × 21 mm (19.2 mL), and the upper end of the testicle was found in the 24 × 15 mm mixed echo zone, where blood flow signals were observed. MR showed the testicular lesions size to be about 23 × 19 mm, and the short T2 signal was abnormal, the enhanced scan of the coronal lesion was markedly high, and the lesion was adjacent to the epididymis. The prostate capsule was intact and no obvious abnormalities were found in the seminal vesicles, bladder or rectum. No abnormal enlarged lymph nodes were found in the pelvis. No signs of destruction were seen in the bone. There were no detected abnormalities in chromosomes and azoospermia factor region of chromosome Y.The patient underwent TSS at the First Hospital of Jilin University on May 26, 2017. Pathological examination showed a benign stromal cell tumor with a volume of 30 × 25 × 18 mm (9.6 mL). Seminiferous tubules were found in the tumor, tumor atypia was not obvious, no necrosis or pathological mitosis was observed, the mitotic count was 1/10 high power fields, and no tumor infiltration was observed in the peripheral margin. Immunohistochemical results were: Calretinin (+), Ki-67 (+<1%), ASLL4 (−), Vimentin (+), CK-pan (−), a-inhibin (+).The patient's wife became naturally pregnant in the 4th month after TSS. The wife was induced at 4 months of pregnancy because of a ruptured amniotic cavity. In September 2018, the patient returned to our hospital after 3 months of infertility. The patient's semen analysis is shown in Table 3,5,6 In this case, the tumor had an androgen secreting function, and the clinical findings were azoospermia and infertility. After TSS, the postoperative hormone levels returned to normal and spermatogenic function was partially restored. This indicates that the normal secretion of gonadotropins plays an important role in maintaining spermatogenic function. This clinical endpoints was consistent with other case reports of LCTs. Androgen secretion by LCTs was not sufficient to maintain spermatogenesis, likely reflecting a significant androgen-induced decrease in gonadotropin secretion. Sperm production is a very complex physiological process with multiple factors required for male fertility. It is well established that FSH, LH and T must function in an orderly manner during spermatogenesis. High levels of intratesticular T are essential for sperm production. The hypothalamic secretion of gonadotropin-releasing hormone (GnRH) stimulates the pituitary secretion of FSH and LH. LH promotes the secretion of T from Leydig cells, and FSH acts in conjunction with T to promote spermatogenesis via the testicular support cells. Testosterone, E2, dihydrotestosterone, and inhibin B secreted by supporting cells can negatively regulate GnRH, FSH and LH. Elevated T associated with LCT may be aromatized to form E2. In the current case, the patient had elevated plasma T levels and circulating E2 was normal, but FSH and LH serum levels were inhibited, which led to spermatogenic disruption. Therefore, the fluctuation of the postoperative semen index may be related to unstable hormone levels; however, further detailed analysis of hormone levels in the postoperative period is required.LCTs secrete androgens, which can be converted to E2 in adults, and the increased androgen and E2 levels exert different clinical manifestations. reported that for 17 patients that underwent TSS, the pathology showed no marked signs of malignancy, except for a slight increase in mitotic figures in tumors with a diameter of 31 mm, and no recurrence or metastasis was observed after 12–192 months of follow-up. Nazareno et al reported that 29 of 37 patients with LCT underwent TSS. The median follow-up time was 4.6 years, and no tumor recurrence or metastasis was observed. Andreas et al compared the therapeutic effects of testicular radical resection and TSS (eight patients). The average tumor diameters for patients with radical resection and TSS were 12.9 mm and 8.6 mm, respectively, and the median follow-ups were 77 months and 42 months, respectively. The authors proposed that TSS for LCT was safe. Additionally, Giorgio and other investigators have reported no tumor recurrence after surgery for LCT.The main treatment for LCT is orchiectomy. The European Urological Association guidelines state that TSS is not recommended for unilateral testicular tumors, but TSS can be performed in certain special cases such as prepubertal or fertile patients. However, even if a postoperative pathological diagnosis indicates a benign tumor, regular follow-up is required. Gianluca et alThe follow-up treatment for these patients should include examination of fertility, monitoring hormone levels and semen indicators, pregnancy test of partner at 3 months, partner examination (if no pregnancy), and B-ultrasound analysis.4Hormone-releasing LCTs produce various clinical features. Azoospermia and the diagnosis of LCT have rarely been reported worldwide. In this article, the patient was diagnosed with azoospermia after a year of infertility with a normal sexual life. During diagnosis and treatment, a right testicular mass was found on B-ultrasound. After the testicular tumor was removed, sex hormone levels returned to normal, spermatogenic function was partially restored, and one initial pregnancy was achieved. Prior to the pathological diagnosis of LCT, the detection of plasma sex hormone levels and contrast-enhanced MR scans may facilitate the diagnosis of LCT from other testicular malignancies and guide the choice of surgical methods for patients with LCT. The main treatment for LCT is orchiectomy. For patients with small tumor volumes, prepubertal and bilateral testicular stromal tumors, or for other practical reasons, TSS may provide a safe and feasible treatment, and also restore some spermatogenic function. Drug assistance may be required to maintain spermatogenic function and achieve fertility, and further long-term research is required.Conceptualization: Yang Yu.Formal analysis: Qingkuo Kong.Resources: Yang Yu.Validation: Hongguo Zhang.Writing – original draft: Qingkuo Kong.Writing – review & editing: Tian Tian, Meiling Sun, Ruizhi Liu.


Spiradenocarcinoma, or malignant eccrine spiradenoma (MES), is a rare sweat gland tumor originating from eccrine sweat glands. To the best of our knowledge, only two cases of MES of the external auditory canal have been reported to date. Here, we report a third case of MES located in the external auditory canal. The iThe patient was a fifty-six-year-old man who initially complained of swelling in his left external ear canal. Over the previous year, he had gradually experienced severe left trigeminal neuralgia, which was occasionally accompanied by a sensation of ear fullness. On physical examination, the lesion was a solitary nodule, with a maximum diameter of 1.6 cm. The lesion was a tender, smooth, firm swelling nodule that was skin-colored with a wide base, and it had not ulcerated the left external auditory canal, which was covered with intact skin. The lesion occluded the ear canal and obstructed the view of the tympanic membrane. The patient’s hearing in this ear was slightly affected. There was no sign of regional or distant metastasis on examination before surgery. The remaining clinical imaging examinations, including head, neck and cranial nerve examinations, were normal. Routine laboratory test results, including those of liver function, kidney function, total electrolyte levels, coagulation function and complete blood counts, remained within normal limits. A high-resolution positron emission tomography (PET)-computed tomography (CT) scan of the whole body demonstrated the fluorodeoxyglucose (FDG)-avid mass described above (maximum standard uptake volume (SUVmax 2.6), while no involvement of the bony canal walls was identified Figure 12O2 after exposure to a graded ethanol sequence. Antigen retrieval was used to enhance CK-7, EMA, P63, Vimentin, ER, PR, Her-2, S-100, CD56, NSE, Syn, CgA and Ki-67 immunohistochemistry by placing the sections in citrate buffer (pH 6.0) under high pressure for 3 min. Then, the sections were washed and incubated for 1 h at room temperature. The sections were washed in phosphate-buffered saline and then incubated with secondary antibody for 20 min at room temperature. After washing, the sections were stained with DAB until the desired stain intensity developed; then, the sections were mounted before observation by light microscopy. Positive and negative controls were included for each immunohistochemical run.The surgical specimen was fixed in 10% formalin and embedded in conventional paraffin blocks. Tissue sections (2 µm thick) were obtained for standard immunohistochemical staining according to routine protocols. The sections were placed in absolute ethanol, followed by 95% ethanol for 2 min, 80% ethanol for 2 min, and distilled water for 5 min; then, the sections were rehydrated in distilled HSurgical pathologic findings led to the diagnosis of primary MES of the external auditory canal. Histological examination revealed that the tumor was located in the dermis and subcutis with no involvement of the epidermis and no residual tumor in the tumor-free margin. The benign area of tumor tissue showed typical ES histological characteristics, consisting of two types of cells: small cells with darkly staining nuclei surrounding larger cells with pale cytoplasm Table 1.de novo MES located in the external auditory canal. No relevant results were found in the patient’s routine health examination performed 1 year before the diagnosis. Whether it is de novo or arises from a benign ES, MES is associated with aggressive behavior, a high recurrence rate, and subsequent development of fatal metastases. Malignant transformation usually requires 20 to 30 years from the onset of the initial benign lesion, with an extremely variable range from 6 months to 70 years , PET-CT, chest X-ray, ultrasound, mammography and scintigraphy of the lymphatic drainage area, have been used to further define the size and extent of these primary neoplasms . In thisvs. 43%, death 8 vs. 43%). Similarly, as early as 1997, Tay et al. .The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Accounting for only 5% of all renal and urothelial tumors, upper tract urothelial carcinoma (UTUC) is a rare genitourinary malignancy. Although management guidelines for UTUC recommend radical nephroureterectomy (RNU) with resection of a bladder cuff as the ‘gold standard’ treatment, the solitary kidney status after this procedure may lead to higher rates of dialysis, cardiovascular morbidity, and overall mortality. In an effort to mitigate these attendant risks, ureteroscopy (URS) and laser photoablation represent a valid treatment option for these patients with high comorbidities and/or low-risk disease and willing to undergo an intensive surveillance program.Minimally-invasive endoscopic management of UTUC was first suggested for imperative cases as chronic kidney disease, solitary kidney, bilateral UTUC, and the good results obtained in terms of cancer control lead clinicians to offer this approach also to elective cases .The endoscopic treatment of upper tract UTUC coincided with the development and refinement of percutaneous renal surgery, ureteroscopy, and laparoscopy. These techniques can now be combined to provide histologic diagnosis of filling defects of the upper urinary tract, remove small to even large intraluminal lesions, or remove the distal ureter or the entire kidney and ureter with endoscopes alone.® (rivaroxaban), AAS, simvastatin and digoxin.We present the case of an 85-year-old male with asymptomatic gross hematuria for 1 month. The patient, despite advanced age, presented good performance status, and as comorbidities, presented cardiomyopathy, previous stroke (one year) and using XareltoIn the requested imaging tests, with better accuracy, the MRI revealed a lesion in the upper right ureter of a superficial and non-invasive character. Ureteroscopy then revealed a typical urothelial, pedunculated lesion of approximately 1cm, and was then resected by endoscopic resection with YAG-Holmium laser, and extraction of the specimen with Dormia's basket.The surgery was performed without any complications, with a total time of 30 minutes and the patient was discharged on the first postoperative day, with a double-j stent, and asymptomatic.Anatomopathological examination revealed superficial urothelial carcinoma (pTa) grade II/(high grade).


Fibrofolliculoma is a benign, perifollicular, connective tissue tumor, and it usually arises in the form of multiple lesions, but rarely as a solitary lesion. We report a case of solitary fibrofolliculoma on the eyelid.A 68-year-old female presented with an asymptomatic mass on the right upper eyelid. The lesion appeared as a flesh-colored, dome-shaped, smooth nodule being the size of 5 × 5 × 4 mm, with eyelashes protruding from the surface, and located on the upper lid margin. Shave excision was performed, and the diagnosis of fibrofolliculoma was confirmed finally through histological exam.Solitary fibrofolliculomas rarely arises on the eyelid. However, it should be suspected when a flesh-colored and doom-shaped lesion of the eyelid is encountered. The benign tumor on the lid margin can be removed by shave biopsy. Fibrofolliculoma usually is a clinically asymptomatic multiple connective tissue tumor, appearing perifollicular, skin-colored and located on the head or neck. Multiple fibrofolliculomas generally are inherited as an autosomal dominant trait and share clinical characteristics of Birt-Hogg-Dubé (BHD) syndrome, which is associated with multiple fibrofolliculomas, acrochordons, trichodiscomas, and internal neoplasms , 2.Fibrofolliculoma, rarely presents as a solitary lesion, since being firstly reported in 1984 . SolitarA 68-years-old Mongoloid woman presented with an asymptomatic, flesh-colored lesion on the right upper eyelid. The lesion had slowly increased in size over 5 years. No similar lesions were found on other parts of the body. Her medical and family histories were unremarkable, and she had experienced no triggering trauma.Upon ophthalmologic examination, the protruding lesion was found to be approximately 5 × 5 × 4 mm and located on the upper lid margin Fig. a. PalpatThe lesion was removed by shave excision under local anesthesia. The lesion was non-slidable, and it was adherent to the tarsal plate and its covered skin. Anterior lamella of the eyelid was resected with a trigonal wedge, with the removal of 1 mm of extra tissue from the margin of the lesion, and the thin layer of the tarsal plate. The anterior lamella defect of the upper eyelid was repaired using A-T flap. A gray line split was performed on the cut ends of skin defects, then, the skin defects were sutured directly. We checked the preauricular lymph nodes, and no lymphadenopathy was found. After the operation, the patient was compression bandaged for 24 h.Histologically examination of the lesion showed a well-defined tumor mass involving a hair follicle, and a proliferation of multiple thin strands of basaloid cells, extending from the central follicle into the surrounding fibrous stroma. The fibrous stroma presented a sharp contrast with the surrounding dermis. Hematoxylin-eosin stains contained mucin content in the stroma Fig. . The hisTwo weeks after the surgery, the patient had no particular complain. The right upper lid showed almost identical to that of the left Fig. c. DuringThe cases of solitary fibrofolliculoma are extremely rare, since being first reported in 1984 . Only eiHistologically, fibrofolliculoma has both an epithelial and a mesenchymal origin, showing distinctive and characteristic features with minor variation . The cenSince solitary fibrofolliculoma is extremely infrequent and definitively diagnosed only by histological results, it can be easily overlooked or clinically misdiagnosed. The 37-year female patient reported by Chang and coauthors was previously misdiagnosed as chalazion and received incision and curettage only. For years, her condition had not improved, recurring several times . FortunaSharing the characteristics of BHD syndrome, fibrofolliculoma is considered to be hamartomas, composed of both connective tissue and follicular epithelial components . Most fi2 laser, or erbium-doped YAG laser, might be a better choice for multiple fibrofolliculomas or recurrent lesions [Surgical excision is usually chosen for the skin fibrofolliculomas in the first operation for pathological diagnosis. The CO lesions , 13. Cur lesions , 15. Ben lesions , 17. AlsWe have presented a rare case of the flesh-colored and doom-shaped lesion arising on the eyelid and show the histologic features of fibrofolliculoma. Although rare, a diagnosis of solitary fibrofolliculoma may be considered when a similar lesion is observed. As the benign tumor on the lid margin, shave excision including a 1-mm margin of normal tissue should be considered. Our report highlights both the clinical and the histopathological features, which are important for appropriate treatment and prognosis prediction.


Spinal epidural hematoma without significant trauma is a rare condition with potentially severe outcome. This case report and systematic review of the literature illustrates the clinical presentation, risk factors, evaluation, treatment and outcomes of spinal epidural hematoma without significant trauma in children.We report one case of a 7-year-old girl who developed a neck pain after minor cervical sprain. MRI showed a right posterior epidural hematoma extending from C2/3 to T1. The hematoma was surgically evacuated, and the histopathology showed an arteriovenous malformation. Postoperative MRI showed complete evacuation of the hematoma and no residual vascular malformation. We report a second ASE with idiopathic spinal epidural hematoma of a 4½-year-old boy presenting with neck pain. MRI showed a right-sided latero-posterior subacute spinal epidural hematoma at C3–C5. Owing to the absence of any neurological deficit, the patient was treated conservatively. MRI at 3 months showed complete resolution of the hematoma.Spinal epidural hematoma without significant trauma in children is a rare condition. It may present with unspecific symptoms. Screening for bleeding diathesis is warranted and neuroradiologic follow-up is essential to rule out vascular malformation. Whereas most children have a favorable outcome, some do not recover, and neurological follow-up is required. Spinal epidural hematoma (SEH) without significant trauma is rare in children. In the literature, the term “spontaneous spinal epidural hematoma” was used to describe SEH without clear traumatic etiology. Since this term, however, covers cases of idiopathic SEH, bleeding due to coagulopathy or vascular malformations and hemorrhages after minor trauma, it was recommended to avoid the terminology . In the The clinical presentation of SEH without significant trauma in children is often nonspecific, including irritability, pain, torticollis, and neurological deficit –15. In sA 7-year-old girl without other health problems presented to the pediatric emergency department with a 10-h history of neck pain following a benign neck flexion-extension movement associated with a cracking noise. The past medical history was unremarkable. There was no a priori history of non-accidental trauma. Clinical examination revealed contractions of the para-spinal cervical muscles with no neurological deficit. Despite oral analgesia, the pain gradually increased. At the child’s second visit, no new clinical abnormalities were noted. Twenty-four hours after presentation, she developed progressive weakness of both arms and legs and paresthesia of the right hand. Bladder or sphincter dysfunction was not reported. Magnetic resonance imaging (MRI) of the spine showed hyper acute right posterior epidural hematoma extending from C2/3 to T1, leading to spinal cord compression , and the past medical history was unremarkable. He was discharged home with oral analgesia. Persistent pain, neck stiffness, and torticollis lead to a second visit 5 days later. Physical examination on admission showed an alert child with marked torticollis, pain-induced limitation of neck movements and tenderness on palpation of the neck muscles. Neurological examination was otherwise normal. Emergency computed tomography (CT) and MRI of the spine revealed a right-sided latero-posterior subacute spinal epidural hematoma at C3–C5, exerting a mass effect on the spinal cord see Fig. d-f. No vSEH without significant trauma in children is rare. Literature on this condition is scarce and restricted to case reports, case series and narrative reviews. In order to describe clinical manifestation, risk factors and etiology, treatment options, and outcomes after SEH without significant trauma in children we present two illustrative cases. In addition, we performed a systematic literature review using PubMed (from 1950), Cochrane Library (from 1994), National Institute for Health and Care Excellence Evidence (NICE evidence: from 1999), and the Excerpta Medica Database (Embase: from 1947). The date of the last search was 24 February 2017. The following search terms were used: “spontaneous spinal epidural hematoma”, “spinal epidural hematoma”, “spontaneous spinal epidural bleeding”, and “spinal epidural bleeding”. Additional publications were identified from reference lists of selected articles. We included original studies, case reports, and case series written in English, German, French, and Spanish. Congress abstracts were not included. Only articles reporting on patients aged younger than 18 years diagnosed with SEH without significant trauma were included. Articles reporting cases of traumatic spinal epidural hematoma were excluded. Extracted data included age, symptoms and clinical findings at presentation, sex, neuroimaging studies, location of SSEH, type and timing of treatment, risk factors and etiology, and neurological findings at clinical follow-up. Furthermore, the neurological findings described at presentation and follow-up were graded using the American Spinal Injury Association (ASIA) Impairment Scale according to the International Standards for Neurological Classification of Spinal Cord Injury , which iWe identified 153 cases of children with SSEH. The search results, including the stepwise elimination process, are illustrated in Fig. Of the 153 cases included in the final selection, 99 were boys  and 50 were girls  with a median age of 7 years (IQR [interquartile range] 20–156 months). The male to female ratio was 6.5:3.5. For 4/153 cases (2.6%), sex was not reported. Table SEH without significant trauma in children may manifest with local pain before any progressive neurological deficit due to spinal cord compression is evident. The classical triad is severe localized spinal pain, radicular pain, and sensorimotor deficit. In contrast to adults , manifesTrivial spine trauma  preceded the SEH in 20% of all cases. In 55% of the children with cervical SEH, minor spinal trauma preceded the bleeding. Vascular malformation  was found as the cause of SEH in 14%. Arteriovenous malformation was the most frequently reported type of vascular malformation. MRI is considered the gold standard for the diagnosis of SEH and the first choice in the diagnostic work-up. As MRI may not always rule out vascular malformations, a conventional spinal angiogram is essential in negative cases. The use of spinal angiograms was only reported in a few instances. Vascular malformations may be underdiagnosed in children with SEH without significant trauma, especially in cases with preceding minor trauma. In our first case, there was no evidence of vascular malformation in MRI and conventional angiography, but histopathological signs of an arteriovenous malformation were found in evacuated material. We assume that the vascular malformation was compacted due to the space occupying effect of the hematoma. Hence, the rupture of a pre-existing arteriovenous malformation triggered by minor trauma is the most likely diagnosis in case 1. Like Sivakumaran, King, Bodi, Chandler and Walsh  we recomBefore surgical decompression, factor replacement is the most important treatment for SEH due to coagulation disorders, depending on the severity of the disorder, to guarantee adequate hemostasis and to prevent hematoma progression and further neurological aggravation. We found that median treatment delay was shorter in patients with coagulation disorder (4 days compared to 10 days). We assume that the clinicians’ alertness to SEH in patients with bleeding diathesis is greater, leading to a prompter diagnosis. In general, SEH represents a surgical emergency. However, case 2 illustrates that not all cases with SEH without significant trauma in children require a surgical approach. In the literature review 36 of 151 patients did not undergo surgery, because of factor replacement for hemophilia (27 cases), rapid resolution of symptoms (4 cases), or no neurological deficit (5 cases). In cases (such as in our case 2) with long hematoma (less compressive) and in stable patients with minimal neurological deficit, or in cases with early resolution of the hematoma, conservative management may be considered , 33. TheTwo percent of the patients with SEH described in the literature died. We found that 65.5% of the surviving patients of children (aged under 18 years) with SEH without significant trauma recovered completely (see Table To conclude, literature regarding SEH without significant trauma in children is scarce and restricted to case reports and case series. There is a lack of higher-level evidence. Children with SEH without significant trauma often present with unspecific symptoms, such as irritability. This may delay diagnosis and timely initiation of treatment, especially in young children and infants. After a trivial cervical trauma and increasing local pain, clinicians can be falsely reassured and diagnose a trivial torticollis, like in our case. Any progressive cervico-thoracic pain, even after trivial traumas, should alert clinicians. The most common contributory factors are bleeding diathesis and vascular malformation. Therefore, coagulation studies are essential in every child with SEH without significant trauma. MRI is the neuroradiologic procedure of choice in these children. However, as MRI may not rule out vascular malformation, conventional angiography should be considered for this purpose.Additional file 1. Alphabetical list of authors of articles included in the review and summary of study design and findings.


Neuroblastoma is an embryonal malignancy that arises from neural crest cells. Adult adrenal neuroblastoma is a rare disease, and less than 100 cases were reported in the literature. Adult neuroblastoma commonly presents with abdominal  lump and pain. A 35-year-old male patient presented with a giant (20 cm × 17 cm × 12 cm) nonfunctional left adrenal mass. He underwent en-bloc surgical excision of the left adrenal gland along with the left kidney. Histopathological examination revealed adrenal neuroblastoma . We present here the surgical management of the rare adult adrenal neuroblastoma. Neuroblastoma (NB) is the most common extracranial solid malignancy in children that account for 15% of the cancer mortality . Less th2. Clinical examination revealed a palpable mass in the left hypochondrium extending 7 cm below the left costal margin that was firm, smooth, and nontender.A 35-year-old male with history of neurocysticercosis under medication presented to the surgery department with left upper abdominal pain and lump of one-week duration. The pain was acute in onset, dull aching type, intermittent, and severe enough to disturb daily activities. He was non-smoker, non-alcoholic, normotensive, and non-diabetic with no family history of malignancy. His body mass index was 25.3 kg/mμg/day, respectively. Serum cortisol and aldosterone were normal. Patient was managed with en-bloc left adrenalectomy, left nephrectomy, and paraortic lymph node dissection through left subcostal hockey stick incision under epidural-general anesthesia. The left renal vessels were completely encased, and abdominal aorta was partially encased by the lymph nodal mass. The surgery last for 210 minutes with blood loss of approximately 300 ml. The patient was discharged on postoperative day 7. Gross histopathology examination of the surgical specimen (The computed tomography with contrast (CECT) of the chest and abdomen-pelvis  showed aspecimen  revealedspecimen . Tumor cNeuroblastoma consists of a group of neuroblastic tumors  that originate from neural crest cells. It is exclusively the disease of childhood. The incidence rate is 60% before the age of one year drops to 10% at age of 14 years. The incidence rate for patients aged over 30 years is 0.2 cases per million inhabitants per year . Out of The clinical presentation of neuroblastoma depends on the tumor's location, local invasion, and metastasis. The head and neck region, central nervous system, chest cavity, and abdominal cavity are most common primary sites of adult neuroblastoma. About 50% of the abdominal neuroblastoma is adrenal in origin , 9, 10. Management of neuroblastoma requires a multimodality approach that includes surgery, radiotherapy, high-dose chemotherapy with stem cell rescue, and Iodine131-MIBG therapy , 9. A reGuidelines suggest laparoscopic adrenalectomy for malignant adrenal tumor with size less than 6 cm . Number There is lack of standard treatment guidelines in the adult population due to rarity of the disease. The Surveillance, Epidemiology, and End-Results registry (SEER) database had a total of 181 adult neuroblastomas from 1973 to 2010. In those cohort of 181 adult (18-60 years) neuroblastomas, L1 and L2 diseases had better progression-free survival (PFS) and overall survival (OS) with the combination of surgery and radiation than surgery alone , 9. In rAdult adrenal neuroblastoma of this size is the first of its kind in literature. Surgical management in L2 adrenal neuroblastoma is advised for local control of the disease. Minimal invasive surgery is a new standard for adrenal tumor. Multimodality treatment can offer long-term favorable oncological outcome in adult neuroblastoma. The treatment outcome is multifactorial. Larger and longer study series of the adult neuroblastoma can guide us through the challenges posed by the rarity of the disease.


We reported the case of a two-old-year boy with a painful acute hemorrhagic edema. This is a self-limited benign condition: usually, affected children are well appearing and this strongly support the diagnosis. In the opposite, in our case, we observed a painful presentation of the edema. Therefore, we demonstrated that rarely, this condition could have also a painful presentation.This case report helps clinician to know that also acute hemorrhagic edema could have a painful presentation, so we must considered it in the differential diagnosis with sepsis, sickle cell crisis and child abuse. We believe that these findings will be of interest to pediatricians. Purpura has a target-like appearance and involves face, ears and extremities [Acute hemorrhagic edema (AHE) is a rare cause of edema and purpuric lesions in children in first years of life  .Typicalremities . Usuallyremities . AHE sharemities . Both coremities . Evidencremities , but theA two-year-old boy presented at the emergency department for an extremely painful bilateral non-pitting edema of both hands Fig. . No feveDuring observation, both the edema and the purpuric lesions spread to arms, feet and legs Fig. .Fig. 3PWe made diagnosis of acute hemorrhagic edema. Our patient received treatment with ibuprofen. Lesions resolved in 72 h.AHE represents a challenge for the pediatrician at the emergency department. Usually, in contrast to striking cutaneous lesions and rapid progression, the overall child general condition is good  and charIn contrast, in our case, the edema was significantly painful and the child very disturbed, so much that he could not use his hands nor walked. Not recognizing the pain as a possible part of this clinical picture, may lead to a misdiagnosis of potentially serious pathologies, which need adequate treatment, such as sepsis, sickle cell crisis, autoimmune thrombocytopenia, coagulopathies or child abuse.We believe that knowing this unexpected and unusual presentation of the disease could be of interest to pediatricians: in AHE, in contrast to the dramatic cutaneous eruption, clinical conditions are usually optimal, but sometimes they could be badly. Awareness of this may avoid possible misdiagnosis.


Acute choledocholithiasis results when stones form in the gallbladder and then pass into the common bile duct, where they may become lodged and cause obstruction. To our knowledge, very few cases are reported in which multiple imaging techniques had failed to detect the presence of gallstones, as per current literature review. We report a case of a 73-year-old woman with nausea, vomiting, and jaundice who was found to have choledocholithiasis with negative imaging on abdominal ultrasound (US), CT, and magnetic resonance cholangiopancreatography (MRCP). The prevalence of cholelithiasis is approximately 20.5 million (6.3 million men and 14.2 million women) in the United States [A 73-year-old woman, with a history of hypertension and hypothyroidism, presented to the ED with a two-day history of nausea and vomiting, following meals. Associated symptoms included bloating, mild epigastric discomfort, anorexia, and choluria. The patient denied any diarrhea, constipation, sick contacts, recent travel, alcohol, tobacco, or illicit drug use.Physical examination was notable for tachycardia and icteric sclerae. Abdominal exam was unrevealing including negative Murphy’s sign and absent organomegaly. On admission, laboratory testing showed elevated white blood cells (WBC) 20TH/uL, total bilirubin 4.1 mg/dL, alkaline phosphatase (ALP) 147 u/L, aspartate transferase (AST) 233 u/L, and alanine transferase (ALT) 177 u/L. Lipase was within normal range. Hepatitis serologies were negative. Urinalysis was positive for urobilinogen.Abdominal ultrasound was obtained and was negative for gallbladder wall thickening, gallstones, or intrahepatic biliary duct dilatation but the common bile duct was reported as borderline dilated at 0.63 cm. Based on these results, the patient underwent abdominal CT which revealed minimal fat stranding adjacent to the gallbladder with no gallstones visualized. Due to the concerning nature of symptoms, MRCP was performed, revealing a thick-walled hydropic gallbladder with surrounding inflammation, without gallstones. As a result of the concerning clinical findings and high level of suspicion, an ERCP was obtained and revealed a filling defect and mild dilation of the entire common bile duct secondary to a gallstone obstruction Figure . BiliaryThe clinical and laboratory diagnosis of acute choledocholithiasis is difficult. Elderly patients are known to possibly lack the findings of Charcot’s Triad, which classically comprises abdominal pain, jaundice, and fever. Some studies suggest only 15.6% of younger and 18.8% of elderly patients as found to have the classic components of Charcot’s Triad . An exteOur patient was found to have acute choledocholithiasis in spite of false-negative imaging including abdominal ultrasound, abdominal CT, and MRCP. Based on these findings and a high clinical suspicion an ERCP was performed resulting in the removal of two stones with nonpurulent golden-brown bile noted after biliary sphincterotomy and balloon extraction.The MRCP provides extensive detail of the biliary tract and has a sensitivity of 81%-100% and a specificity of 92%-100% in detecting choledocholithiasis . UltrasoThis case demonstrates the need for increased awareness when analyzing and interpreting negative findings on the appropriate initial radiological investigations. The diagnosis of choledocholithiasis is confirmed with advanced imaging techniques, including MRCP and ERCP. Given the various diagnostic and therapeutic options available for managing choledocholithiasis, clinical interpretation and assessment are paramount. Difficult stone burden and difficult biliary anatomy may require advanced surgical, endoscopic, and percutaneous techniques to extract biliary stones. This case demonstrates the importance of maintaining a high-degree of clinical suspicion and utilizing noninvasive techniques when attempting to confirm the presence of choledocholithiasis.


Lipomas are common benign tumours which occur in up to 2% of the population. They are classified as giant when larger than 5 cm. Although they are usually asymptomatic, giant lipomas of the hand may cause compression of the underlying tissues.A 62-year-old Caucasian male presented to the Plastic and Reconstructive Surgery outpatient clinic with numbness and pain in his left hand. The numbness in his fingers pointed to compression of the median nerve, as well as the ulnar nerve. He presented with a rapidly progressive swelling in his left palm. An MRI scan of the hand was made, which showed a lipoma of approximately 8,5 cm in diameter. The swelling was surgically removed and sent for histopathological analysis, which confirmed the diagnosis of benign giant lipoma of the hand. Two weeks postoperatively, pain and numbness significantly decreased.Neural injury in carpal tunnel syndrome is related to the duration and degree of compression. A giant lipoma is considered malignant until proven otherwise since variants with high potential for metastasizing exist. Distinguishing between a benign tumour and a malignant lipoma is essential, since a more radical treatment plan might be required.Giant lipomas of the hand are a rare cause of carpal tunnel syndrome and a malignant variant should always be suspected. A preoperative MRI scan should be performed. Rapid en bloc excision is necessary in case of compression of the underlying tissues. Localization in distal extremities is uncommon, occurring in less than 1% of cases . A giantDirect compression of the median or ulnar nerve can result in symptoms such as paraesthesia, pain and loss of strength ,2. In caWhile most lipomas are benign, a rapidly growing and painful mass is more likely to be malignant . A giant2The work has been reported in line with the SCARE 2020 criteria . A 62-yeAn ultrasound showed two masses of similar density and architecture. The ulnar and radial swelling measured 27 × 14 mm and 35 × 15 mm, respectively. An MRI scan was performed which showed a lipoma of 4,8 × 1,8 × 8,6 cm volar to the flexor tendons . The tuml-shaped skin incision in the palmar crease. Secondly, the median nerve was identified and protected after dissection of the superficial component of the lipoma. Subsequently, we proceeded to dissect the deep palmar space and the component located in Guyon’s canal. The branch of the ulnar nerve in Guyon’s canal was spared.Due to the progression of symptoms, rapid excision of the mass was recommended. The carpal tunnel was opened through an The lipoma was removed completely  with no 3Neural injury is related to the duration and degree of compression . A giantA giant lipoma is considered malignant until proven otherwise , since vOther more aggressive, but less common, forms with high potential for metastasizing 17–30%) are the dedifferentiated liposarcoma (DDLS); the myxoid round cell liposarcoma (MRCL); and pleiomorphic liposarcoma (PMLS). These types have high recurrence rates ranging from 34% to 45%. Interestingly, the DDLS can arise de novo or be present in recurrence of WDLS. DDLS is more often found in the retroperitoneum 0% are th. For the4In case of a giant lipoma of the hand, a preoperative MRI scan is highly recommended, considering the risk of malignant potential. Furthermore, detailed visualization of the mass in relation to other viable structures is an asset ,4,8. We Nothing to declare.Nothing to declare.No ethical approval was necessary.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.All authors contributed to the conception and design. Jean-Bart Jaquet and Mariam Gabrian performed the surgery. All authors contributed to drafting, critical revision, and final approval of the article.Not Applicable.Jean-Bart Jaquet, Belle Tellier and Mariam Gabrian are all guarantors for the report.Not commissioned, externally peer-reviewed.


Schwannomas are tumors that originate from the myelin sheath of Schwann cells . These tAn 83-year-old female presented to our clinic for a mass over the right lower abdomen. She denied having symptoms such as abdominal pain, nausea, vomiting, or bowel changes. She had been diagnosed with this mass in 2015 by her primary care physician, but she failed to follow up. The patient's medical history was significant for hypertension and hysterectomy. There was no reported personal or family history of inflammatory bowel disease or gastrointestinal cancer. She reported no previous history of esophagogastroduodenoscopy or colonoscopy. On examination, patient was hemodynamically stable. Her abdomen was soft and nontender and had no gross palpable masses. Laboratory test results were within normal limits. A repeat CT of the abdomen and pelvis revealed a well-circumscribed mass anteriorly adjacent to the cecum and just deep to the intra-abdominal wall musculature, measuring 4.4 cm in transverse dimension and 4.0 cm in anteroposterior dimension, with low attenuation in the central region .There was no other abnormal pathology identified throughout the small and large intestines. CT-guided core needle biopsy revealed spindle cells under hematoxylin and eosin (H&E) sections Figures . ImmunohA robot-assisted laparoscopic right hemicolectomy with an intracorporeal anastomosis was the procedure planned for the suspected cecal mass. A 5 mm Optiview technique was used over the left upper quadrant at Palmer's point to access the intra-abdominal cavity. The abdomen was insufflated to 15 mmHg. Two additional robotic ports were placed inferior to the initial port under direct visualization. These ports were separated by a distance of 8-10 cm. The da Vinci Xi surgical system was docked. Lysis of adhesions was performed in the pelvic region. Upon further dissection, it was noted that the suspected cecal mass was actually a transverse colon mass being held down by adhesions over the right lower quadrant. The operative approach was changed due to the intraoperative findings. A midline hand-assisted port incision was made and the transverse colon was eviscerated. A segmental resection with 5 cm margins was performed, followed by an extracorporeal side-to-side stapled anastomosis. Lembert sutures were used to reinforce the staple line. The colon was placed back into the abdominal cavity. The fascia and skin were closed. The patient tolerated the procedure well without complications. She was discharged home on postoperative day one after appropriate pain control, food tolerance, and adequate ambulation.The operative specimen was grossly described as a discrete well-circumscribed encapsulated mass focally abutting the underlying mucosa. There was no involvement of the mucosal surface, and there was no extension through the intestinal wall.Primary gastrointestinal schwannomas are rare unless there is an association with neurofibromatosis . Although generally asymptomatic, these tumors may cause symptoms such as abdominal pain, rectal pain, hematochezia, and tenesmus . Both im


Vascular iris lesions are rare and can sometimes be associated to systemic vascular lesions. They usually cause spontaneous recurrent hyphema. The differential diagnosis should be considered primarily with iris rubeosis and with highly vascular tumors as iris melanoma. Generally, vascular lesions of hemangioma type are located in the iris without extension to iridocorneal angle. We present a case of a 77-year-old male with an iris vascular lesion suggestive of racemose hemangioma, who is asymptomatic, and with 360-degree iridocorneal angle affectation showing no lesions at any other location. There are a variety of tumors and iridian lesions that range from cystic tumors to even solid melanocytic and nonmelanocytic tumors. According to Shields et al., the nonmelanocytic lesions constitute 11% of iridian tumors, and of these, the vascular type constitutes 2% [A 77-year-old male was referred for a possible study of unilateral iris rubeosis in right eye (RE) found in a routine exploration previous to cataract surgery. The patient was asymptomatic and did not refer any previous ocular pathology except divergent strabismus since childhood with left eye amblyopia. The patient had a personal history of arterial hypertension with renal repercussion, metabolic syndrome, auricular fibrillation, prostatic syndrome, and moderate chronic obstructive pulmonary emphysema. In the exploration, he presented a visual acuity (VA)  of the RE of 0.5 and in the left eye (LE) of 0.15, as well as divergent strabismus. In a slit lamp, we observed radial iridian vascular engorgement at 5 to 7 o'clock location in RE. The vessels seem to originate from the iris stroma and then extended circumferentially . GonioscPeripheral tumor masses, such as choroidal melanoma, retinoblastoma, metastasis, or ciliary body lesions, were ruled out by fundus examination with indentation and maximum midriasis.Supra-aortic trunk exam with ecoDoppler showed no other causes of ocular ischemia. No carotid stenosis with hemodynamic significance was observed. In the differential diagnosis, entities such as sickle-cell anemia, Takayasu disease, giant cell arteritis, or carotid-cavernous fistula were also proposed, but additional tests were not requested since they have low probability in the patient's clinical context.An anterior segment optic coherence tomography was performed to refine the characterization of the lesion, and poorly defined intrastromal hyporeflective spaces were observed coinciding with the lumen of the vessels .For the differential diagnosis between neovascularization and tumor/vascular malformation, a fluorescein angiography (FA) was performed and normal contrast uptake was demonstrated without extravasation of the latter at the level of the iridian, iridocorneal angle, and retinal vessels , vasculopathies such as sickle-cell anemia or Eales disease, and tumors .FA plays a fundamental role in detecting the presence of neovascularization and areas of retinal ischemia. In iridian vascular malformations, FA is characterized by an early hyperfluorescence that may also be visible later and, unlike rubeosis, it will not produce contrast extravasation. FA is a very useful but invasive test, so new diagnostic methods such as optical coherence tomography angiography allow us to perform a more precise exploration of the iridian vasculature in a noninvasive way .In the literature, there have been no reported cases of iridian vascular lesions of the hemangioma type in which the vascular malformation extends 360 degrees through the iridocorneal angle. Shields et al.  describeIn general, the management of these lesions consists of observation without any specific treatment, since most of them are asymptomatic, as in this case. In cases where there has been spontaneous bleeding, improvement has been reported after treatment with argon laser photocoagulation applied directly over the vascular lesion . In caseVascular malformations of the iris are very rare and can sometimes be associated with vascular lesions at other locations; however, in our case, we describe the existence of a vascular malformation known as racemose hemangioma type iris, with 360-degree extension by the iridocorneal angle without any other lesions. These are asymptomatic lesions that require follow-up and a differential diagnosis, mainly with iris rubeosis and neovascular glaucoma. Tumors such as ciliary body melanoma, although rare, should be ruled out.


Plasma cell granuloma (PCG) is a rare non-neoplastic entity, with the precise etiology remaining unclear. Vertebra-affected spinal PCG has not been reported yet. This report presented a case with cross-segment spinal PCG in thoracolumbar region.A 32-year-old male patient presented to the authors’ hospital since his health check-up results showed osteolytic lesions in the thoracolumbar spine. He felt asymptomatic throughout the course. Radiological examination revealed destructive changes at T12 and L1 vertebrae. Whereas laboratory examination excluded malignant tumor. The results of routine incisional biopsy remained inconclusive, thereby necessitating complete excision of the lesions. Finally, the infiltration of plasma cells observed by pathological examination of the surgical specimen confirmed the diagnosis of PCG.To the authors’ knowledge, this was the first case of cross-segment spinal PCG with osteolytic property. The possibility of PCG should be considered for the diagnosis and differential diagnosis of an osteolytic lesion in the spine. Since the etiology of PCG is unknown, the disorder was confirmed based on excluded diagnosis. Surgical resection is recommended both for the definite diagnosis and treatment of spinal PCG. Plasma cell granuloma (PCG), firstly mentioned by Bahadori and Liebow, is a pseudotumor-like condition characterized by the polyclonal proliferation of plasma cells. The etiopathogenesis of PCG is still unknown. Although PCG is regarded as a generally benign and nonrecurring lesion, the outcome of those with local aggressiveness or recurrence may become complicated. PCG is commonly seen in lung and occasionally seen in esophagus, mouth, stomach, thyroid gland, intestine, kidney, lymph node, and skin. Additionally, PCG was ever found in jaw and temporal bone. However, vertebra involved PCG has not been reported yet. We here reported the first case of cross-segment PCG occurred in the thoracolumbar spine. This report was approved by the Ethics Committee of our hospital. Informed consent was obtained from the patient concerning the data submitted for publication.A 32-year-old man presented to the local hospital for health check-up and the computed tomography (CT) scan pitched upon osteolytic lesions in his thoracolumbar spine. Three days later, he was referred to our hospital for further management. Throughout the course, he had no pain or fever, with negative neuroradiological manifestation in his lower limbs. His past medical history was unremarkable. He had no history of keeping bird or travelling to the epidemic area in the past few years. Risk factors for tuberculosis and HIV exposure were not found. Physical examination showed no tenderness or obvious mass in the thoracolumbar level. No neurological dysfunction or lymphadenopathy was noted. The X-ray film observed a radiolucent lesion at T12 vertebra and enlargement of the right pedicle of L1  or pseudotumor and inflammatory fibrosarcoma, of which inflammatory pseudotumor was the most commonly called , 13, 14.. Christian Schneider et al. even recommended rituximab for PCG in cases which are refractory to corticosteroid treatment or inaccessible to complete surgical excision [. In addition, recurrence is also observed, especially in cases of subtotal excision, which emphasizes the need for more specific long-term therapies with fewer adverse effects [There is no consensus regarding therapeutic approaches for PCG. The available treatments include surgical excision, radiotherapy, administration of immunosuppressors (cyclosporine) and corticotherapy , 25–27. excision , 30. In  effects , 31.PCG is a rare non-neoplastic lesion characterized by the presence of polyclonal plasma cells. We presented the first case of PCG occurred in the thoracolumbar spine, which was diagnosed eventually by pathological examination of completely excised specimen. Surgical excision is preferred for the treatment of spinal PCG. Cases with surgical contraindication can choose glucocorticoids or radiation therapy. Since the etiology of the present case of PCG remains unclear, more studies are required to identify the mechanism of this disease so as to improve the clinical outcome.