Hybrid oncocytic/chromophobe tumor (HOCT) is defined as tumor composed of renal oncocytoma (RO) and chromophobe renal cell carcinoma (CHRCC). Sporadic HOCT is extremely rare, the preoperative diagnosis is difficult, and no guidelines for clinical therapy. We report a case who is the youngest male patient of sporadic HOCT in the world, review the previously reported cases, and share the clinical features, diagnosis, treatment, and prognosis of HOCT.A 30-year-old man was admitted with the complaints of incidental right renal tumor detected by abdominal ultrasound. He had no complaints of urological symptoms, abdominal pain, osphyalgia, and hematuria. Abdominal contrast-enhanced computed tomography revealed an 85 mm × 80 mm × 80 mm unilateral and solid renal mass, and no findings of metastases.The preoperative diagnosis was right renal tumor.Laparoscopic right radical nephrectomy was performed.Histopathology demonstrated a mixture of cells with the morphologic features of those seen in CHRCC and RO. The patient was final diagnosed as sporadic HOCT. After follow-up of 14 months, the patient had no complaints and evidence of disease recurrence.Sporadic HOCT is extremely rare. It is possible that core biopsy could improve diagnostic accuracy. Laparoscopic radical nephrectomy or nephron sparing surgery should be considered the clinical therapy of the sporadic HOCT patients. The clinical behavior of HOCT is still entirely uncertain and should be proved by studies with available long follow-up. Hybrid oncocytic/chromophobe tumor (HOCT) is defined as tumor composed of varying amounts of cells with features of RO and CHRCC. Usually, the numerous HOCT have been initially described in patients with Birt–Hogg–Dubé syndrome (BHDS) or with renal oncocytosis without BHDS.2Informed consent was signed by the patient for the publication of this report and related images.3A 30-year-old man was admitted with the complaints of incidental right renal tumor detected by abdominal ultrasound. His temperature and blood pressure was normal. He had no complaints of urological symptoms, abdominal pain, osphyalgia, and hematuria. No cutaneous lesion or history of spontaneous pneumothorax was noted. All blood and urine examinations were within normal limits. Abdominal contrast-enhanced computed tomography revealed a heterogenous mass of 85 mm in diameter in the upper pole of the right kidney, displaying central stellate hypodensity areas inside with punctate calcification, and no findings of metastases Fig. A–E. Lapa4,3RO in youth is an infrequent renal neoplasm and the one with the CHRCC component is extremely rare. HOCT have been described in patients with BHDS or with renal oncocytosis.The preoperative diagnosis of HOCT is very difficult. Clinically, sporadic HOCT is mostly unilateral and solitary, whereas HOCT in individuals with BHDS or oncocytosis are often bilateral and multiple. Patients with BHDS usually have fibrofolliculom, pulmonary lesions including bullae and spontaneous pneumothorax.HOCT possess the characteristics of CHRCC and RO simultaneously. In renal tumor biopsy, HOCT can only be found incidentally. It was reported that about 15% of RO diagnosed by renal tumor biopsy were actually HOCT, which raise the issue of a potential misleading diagnosis when HOCT is biopsied.,3 reported that they recognize HOCT, as a low malignant potential tumor managed similar to renal tumors of other type, should be first considered the clinical therapy by nephron-sparing surgery. If partial nephrectomy is not technically suitable, onetime radical nephrectomy should be performed. The sporadic HOCT usually pursue a favorable clinical course, as no recurrence and metastasis has been documented.,8,9 However, the follow-up should be no longer than 10 years.There are no guidelines for clinical therapy of the sporadic HOCT patients, because the researches for this tumor were mostly retrospective with relatively small sample size. The majority of urologists and pathologists5Sporadic HOCT is an extremely rare tumor. The preoperative diagnosis is difficult and easily misdiagnosed as RO. It is possible that core biopsy could improve diagnostic accuracy. Laparoscopic radical nephrectomy or nephron sparing surgery should be considered the clinical therapy of the sporadic HOCT patients. The clinical behavior of HOCT is still entirely uncertain and should be proved by studies with available long follow-up. Urologist and pathologist must pay more attention to this rare tumor.Supervision: Chaozhao Liang.Writing – original draft: Bingbing Hou.


Parathyroid autotransplantation is an increasingly common procedure given the increasing rate of hyperparathyroidism. However, post-autotransplantation imaging is not commonly performed and the imaging findings can mimic liposarcoma. Therefore, radiologists should be aware of the imaging characteristics of parathyroid autotransplantation. Here we discuss the CT and Tc99m-Sestamibi 4D-CT findings of parathyroid autotransplantation. We will also discuss the pathophysiology of liposarcoma and present the gross histological findings seen on pathology. Implantation of parathyroid tissue into the forearm after a total parathyroidectomy or thyroidectomy is well described -3. AlthoA 56-year-old male with a past medical history of end-stage renal disease on hemodialysis, congestive heart failure, groove pancreatitis, chronic atrial fibrillation, valvular heart disease, and obesity, who was status post four gland parathyroidectomy for hyperparathyroidism seven years prior presented to the hospital for chronic bilateral lower extremity nonhealing wounds which had been worsening. He was found to have a parathyroid hormone level of over 3,200 pg/mL  as well as a soft right forearm mass located at the site of previous autotransplantation. The mass was further characterized with a CT scan of his right forearm under the indication “distal forearm tumor”. The CT scan showed a fat containing mass at the lateral aspect of his forearm measuring 4.6 x .9 x 5 cm with soft tissue nodularity suspicious for an atypical lipomatous tumor or liposarcoma  level and concern that the mass on his right forearm may be a lipomatous tumor due to the CT scan interpretation, the decision was made to resect the mass. Upon resection, the operative report stated that the mass did have the appearance of possible lipomatous features given that it had a different characteristic than the surrounding subcutaneous fat. The surgical pathology report came back as nodules of hyperplastic parathyroid tissue involving fibroadipose tissue and skeletal muscle Figures , 6.A repeat Technetium-99m Sestamibi study performed three days later showed no uptake at 20- and 60-minute delayed imaging to suggest residual parathyroid tissue Figure . IntraopParathyroid tissue during a total parathyroidectomy can be preserved in situ, autotransplanted to the brachioradialis or sternocleidomastoid muscle, or allotransplanted from a donor . There iParathyroid autotransplantation is a common procedure done after parathyroidectomy in the setting of hyperparathyroidism. Lack of imaging of autotransplantation can be challenging in that its appearance can mimic a lipomatous malignancy such as liposarcoma, with the diagnosis necessitating biopsy for the final diagnosis. In the setting of hyperparathyroidism after parathyroidectomy, nuclear medicine studies such as Technetium-99m Sestamibi scan may be helpful in identifying the location of the transplanted parathyroid gland.


Laryngeal cysts are rare lesions that may occur at any mucosa-lined location within the larynx. Papillomas are also benign lesions of the larynx.We report a 34 year-old-patient with a laryngeal cyst incidentally found during screening endoscopy and presenting as a soft tissue mass on a computerized tomography scan. A papilloma concomitant with the cyst was detected intraoperatively.Concomitant tonsillar cyst and papilloma of the larynx.The lesion was completely resected with a bipolar radiofrequency plasma ablation (RFA) device.Pathologic examination showed a tonsillar cyst and papilloma in the larynx. Six months later, there has been no evidence of recurrence.To our knowledge, this is the first report of a concomitant tonsillar cyst and a papilloma in the larynx. Asymptomatic laryngeal cysts can be detected endoscopically. RFA is safe and effective for endoscopic management. Tonsillar cysts are characterized by a squamous epithelial lining with underlying follicular lymphoid tissue, resembling tonsillar crypts. Moreover, tonsillar cysts are always located in the valleculae, epiglottis, and pyriform fossa. Papillomas are common benign tumors of the larynx and pharynx and have been associated with viral infections. Although papillomas are benign, they may recur easily. To date, however, there has been no report in the English language literature of any individual patient with a concomitant tonsillar cyst and papilloma. This report describes a 34-year-old patient with a laryngeal cyst that was incidentally found during screening endoscopy, followed by intraoperative identification of a papilloma concomitant with the cyst. The lesion was resected successfully by simultaneous excision, with the pathology results confirming a tonsillar cyst and papilloma.Laryngeal cysts are rare lesions that may occur at any mucosa-lined location within the larynx. Laryngeal cysts have been reported to account for approximately 4.3% to 6% of all benign laryngeal lesions.2A 34-year-old man was referred with abdominal pain, abdominal distension, and loss of appetite for 6 months. At his first visit to the Department of Gastroenterology, he was evaluated by diagnostic electronic gastroscopy, which detected a mass in his throat. He had no history of dyspnea, dysphagia, odynophagia, foreign body feeling, or hoarseness. He subsequently visited the Department of Otolaryngology for throat discomfort after his last endoscopic examination. Physical examination showed that the patient was healthy-looking in appearance, with other characteristics, including his tonsils, thyroid gland, and lymph nodes, being unremarkable. Computed tomography (CT) imaging showed a mass bulging out of his pharyngolaryngeal cavity Fig.  and flexThe patient underwent endoscopic excision of the mass. Transoral fiberoptic intubation was performed under general anesthesia. A 2 × 2 cm mass was observed on the laryngeal surface and the right edge of the epiglottis, which appeared to be a cyst. Following suction to remove the cyst at its base, however, another mass was found to emanate from the underside of the cyst. This mass had the appearance of a papilloma, measuring about 1 × 1 cm Figs.  and 4. H3 Congenital laryngeal cysts, first described in 1881, may obstruct airways in neonates. In contrast to laryngoceles, which are air-filled dilatations of laryngeal saccules, laryngeal cysts are fluid-filled dilatations unconnected to the laryngeal ventricle. Although the symptoms of laryngoceles and laryngeal cysts are similar, including hoarseness, cough, stridor, and even dyspnea, depending on lesion size and location, these lesions can be differentiated by CT and magnetic resonance imaging (MRI). CT and MRI are also good for determining the location, size, extent, and anatomical relationships of these lesions.Laryngeal cysts are generally benign and have been reported to be more prevalent in women than in men. This classification, however, cannot determine the genesis of the lesions, and is limited to the resulting histologic findings. These cysts have also been classified into 3 types: epithelial, tonsillar, and oncocytic. The lesion in our patient can be classified as tonsillar type. Although tonsillar cysts have been reported to be related to lymphoepithelial cysts of the oral cavity, the origin of tonsillar cysts remains unclear. A third classification system has divided laryngeal cysts into 2 types, with Type 1 cysts confined entirely to the larynx, and Type II cysts extending beyond the larynx.Table Laryngeal cysts have been classified into 2 types: ductal and saccular. A patient with a laryngeal saccular cyst caused by recurrent respiratory papillomatosis has been described. Laryngoscopy of this patient showed a large cyst-like mass filling the right ventricle of the larynx, with further examination showing papillomas extending along the false vocal fold and into the ventricle on the side of the lesion, resulting in hoarseness, airway obstruction, and dysphagia. In contrast, our patient presented with a single papilloma originating from papillary hyperplasia of the cyst epithelium, and was asymptomatic.Papillomas are the most frequently occurring benign tumors of the larynx and have been associated with human papilloma virus (HPV). Histologically, papillomas are vascularized, consist of connective tissue covered by stratified squamous epithelium, and project outwardly in finger-like fronds. It is unclear whether transoral laser marsupialization or complete cyst removal provides better outcomes. Although marsupialization has been reported to be highly successful, the remaining base of the cyst can lead to cyst reaccumulation. Our preference was to completely remove the cyst. Therefore the lesion in our patient was completely excised with RFA, which has been proven safe and effective, with improved surgical precision, minimal bleeding, and better healing.The main treatment of laryngeal cysts is surgical removal, both by endoscopic management and external approaches. In contrast, laryngoceles may be treated conservatively. Endoscopic procedures are optimal for most endolaryngeal lesions, with the laryngofissure approach often used for lesions that have spread beyond the larynx.4To our knowledge, this is the first report describing a concomitant tonsillar cyst and papilloma in the larynx. The lesion was completely excised using a bipolar RFA device, which has been shown to be safe and effective. There has been no evidence of recurrence at follow-up.Writing – original draft: Qiang Liu.Writing – review & editing: Hong Huo.


Intestinal obstruction resulted from balloon migration is an extremely rare but serious late complication of the intragastric balloon (IGB). The aim of this study is to report a case of small bowel obstruction occurring in a middle age corpulent female following embedding of IGB. A 47-year-old obese female presented with abdominal pain, nausea, and vomiting for two days. She had a history of an endoscopically placed IGB nine months before presentation. Physical examination showed an obese woman with mild distress, and the right upper abdomen was tender. The plain abdominal radiograph showed gas shadow in the stomach and the duodenum, esophago-gastro-duodenoscopy showed an empty stomach and balloon migration from the stomach. Under general anesthesia, laparotomy was performed, a three-centimeter antimesenteric enterotomy was done and the balloon extracted from the proximal jejunum. Intestinal obstruction is an extremely rare complication of IGB. It should be managed by laparotomy and extraction of the balloon. •Intestinal obstruction resulted from intragastric balloon migration is an extremely rare complication.•It is serious complication and needs general anesthesia and laparotomy.•In this study, a case of small bowel obstruction occurring in a middle age female following inserting of intragastric ballon has been presented and discussed. Some studies demonstrated IGB superiority in comparison with diet regimen . Up to dIntestinal obstruction resulted from balloon migration is an extremely rare but serious late complication of IGB occurring in 0.8% of the cases . The aim22) presented to the emergency department with cramp-like abdominal pain, attacks of nausea and vomiting for two days. The pain was in the central region of the abdomen, not responding to analgesics but relieved by vomiting. The patient reported having an endoscopically placed IGB  nine months before the presentation. The total weight loss was 15 kg (BMI was 41 kg/m2 at placement). She had negative past medical, past surgical and drug history. Physical examination showed an obese woman with mild distress, the abdomen was soft and did not distend; there was tenderness in the upper abdomen without guarding or rebound. Her bowel sound was positive. Blood pressure; 130/80 mmHg, pulse rate; 78 bpm, respiratory rate 15 bpm, temperature 37.3 C, Hemoglobin; 13 g/dl, WBC 7 × 109/L. The plain abdominal radiograph showed gas shadow in the stomach and the duodenum with no air-fluid levels  37 kg/md levels , Esophagd levels . The patd levels  and the d levels , the ent3Therapy with IGB has been used for decades as a minimally invasive approach to treat obesity despite the inconsistent evidence regarding its efficacy ,2. In th2 with failed previous attempts at weight loss [The clinical use of an inflatable IGB was reported for the first time in 1982 in a series of five women . The bezght loss ,8. The lTherapy with IGB, regardless of the types of device, is associated with transient and minor postoperative discomfort (30%). Less commonly, it is associated with major complications such as gastric perforation 0.1%), gastric ulcers (0.5%) and spontaneous deflation 3–23%) with potentially subsequent small-bowel obstruction (0.8%) [%, gastri3% with pIntestinal obstruction subsequent to a spontaneous emptying of an IGB has been reported to occur within a few months of placement ,8. In thIf IGBs remain longer than the scheduled period, it may spontaneously deflate, migrate and result in bowel obstruction. Most patients have been managed by an operation; however, double-balloon enteroscopy appears a promising alternative to treat intestinal obstruction induced by partially deflated IGBs .The learning point in this study is that, in any patient after IGB insertion, close observation and awareness about the serious complications like intestinal obstruction is mandatory. Early diagnosis of rupture and migration may be treated by endoscopy but intestinal obstruction should be managed by laparotomy and extraction of the balloon from the bowel lumen.A written informed consent has been taken from the patient for the publication of this report.Approval has been taken from Kscien organization for scientific research, no. 10.No source to be stated.Ayad Ahmad Mohammed: The team leader, revising the draft with final approval of the manuscript.Sardar Hasan Arif: Revising the draft, reviewing the literature and follow up with final approval of the manuscript.Abdulwahid M. Salih: The surgeon who did the first operation. Final approval of the manuscript.Fahmi Hussein Kakamad: Writing the manuscript, reviewing the literature and final approval of the manuscript.There is no conflict to be declared.Researchregistry1412.Fahmi Hussein kakamad.


An otherwise healthy 20-year-old male presented with an exophytic, polypoid, yellowish lesion involving the dorsal surface of his tongue, which he reported being present since birth and unchanged. This was removed by surgical excision and diagnosed as a leiomyomatous hamartoma. Histological examination revealed a combination of fibrovascular connective tissue, conspicuous smooth-muscle bundles, adipose tissue, minor salivary gland tissue, blood vessels, lymphoid tissue, peripheral nerves, and normal skeletal muscle. This case is exceptional due to the patient's age, as until now, lingual leiomyomatous hamartomas have been reported almost exclusively in a paediatric population. To our knowledge, this is the eldest age at which a LLH has been reported in the literature. This underscores the need for clinicians to consider this rarely reported entity when considering the radiographic and clinical differential diagnoses for these lesions, both in the paediatric and adult populations. We also present a review of the literature regarding lingual leiomyomatous hamartomas. This element . Oral ha element . An oral element . They ar element . In most element , 3. Due  element .Here, we report an unusual case of a lingual leiomyomatous hamartoma (LLH) occurring in a healthy 20-year-old male, treated successfully by surgical excision. We also present the histological and immunohistochemical features as well as a review of the literature. This case is exceptional due to the patient's age, as until now, LLHs have been reported almost exclusively in a paediatric population. To our knowledge, this is the eldest age at which a LLH has been reported in the literature.A 20-year-old medically fit and healthy male presented for an assessment in preparation for orthognathic surgery. On examination, a 1.5 cm diameter exophytic midline tongue lesion  was noteThe MRI report described a 1.5 cm protuberant mass arising from the dorsal aspect of the tongue in the midline at the approximate junction of the oral component and base. The imaging suggested that this was in part fatty, probably arising from the submucosa, and is also seen to demonstrate very mild contrast enhancement. There appeared to be intact overlying mucosa and no apparent involvement of the intrinsic tongue muscles. The sublingual space and salivary glands appeared normal.Thereafter, the mass was surgically excised and submitted for histological examination including haematoxylin and eosin staining and immunohistochemistry.α-smooth-muscle actin demonstrated large concentrations of smooth-muscle bundles; however, S-100 was found only in peripheral nerve bundles intermingled with smooth-muscle fibers ; the solid leiomyoma is a true neoplasm of smooth-muscle origin, which is rare in the oral cavity .Clinically, an intraoral solid mass in or near the midline of the maxilla and the tongue, especially in children, should involve LLH as a differential diagnosis. Other lesions that have a similar clinical presentation such as a lipoma, granular cell tumor, choristoma, leiomyoma, fibrous epithelial polyp, and benign mesenchymoma should likewise be considered. As the appearance of LLH can be nonspecific, other lesions such as vascular or lymphatic lesions, mucus extravasation phenomena, reactive or traumatic lesions, benign or malignant neoplasm, and cystic lesions should not be neglected. Additionally, in a paediatric population, hamartomas should be differentiated from lingual thyroid thorough clinical examination, imaging, and histopathological examination.Given the nonneoplastic nature of hamartomas, complete removal of the tumor mass by wide surgical excision is the accepted treatment of choice. Recurrences have been reported where surgical excision is incomplete. Wushou et al.  studied This case is exceptional due to the patient's age, as until now, LLHs have been reported almost exclusively in a paediatric population. This underscores the need for clinicians to gain familiarity and consider this entity when considering the radiographic and clinical differential diagnoses for these lesions, both in the paediatric and adult populations.


Tonsillar lymphoma usually presents as unilateral or bilateral infiltration of diffuse large B‐cell lymphomas. We report a case of a 79‐year‐old man with near‐complete obstruction of the upper airways due to T‐cell prolymphocytic leukemia cells. Surgical resection was safely performed to reduce burden of disease. Tonsillar lymphoma usually presents as unilateral or bilateral infiltration of diffuse large B‐cell lymphomas. We report a case of a 79‐year‐old man with near‐complete obstruction of the upper airways due to T‐cell prolymphocytic leukemia cells. Surgical resection was safely performed to reduce burden of disease. A 79‐year‐old man was referred to our hospital because of persistent sore throat, dysphagia, and shortness of breath over the past two months. Physical examination of the oral cavity revealed bilateral tonsillar hypertrophy resulting in the near‐complete obstruction of the upper airway Figure A and disT‐PLL is an aggressive cancer characterized by the hyperproliferation of post‐thymic prolymphocytes. T‐PLL accounts for ~2% of mature lymphocytic leukemias in adults.Because of patients’ life‐threatening condition, age, and severe risk of alemtuzumab infusion‐associated reactions, we initially tried cytoreducing the disease with age‐adjusted chemotherapy based on cyclophosphamide, vincristine, and high‐dose prednisone followed by the administration of 400 mg venetoclax for a month.However, after an initial response, we observed a worsening of clinical conditions that prompted surgical excision that improved the burden of the oral symptoms. While T‐PLL cutaneous infiltration is frequently observed in this disease, this is the first report describing a bilateral tonsillar involvement.The authors declare that there is no conflict of interest regarding the publication of this article.All authors contributed to the care of this patient. SDS and FDL: completed the surgical procedure. All other authors critically revised the manuscript. IB and GR: were responsible for the preparation of the manuscript. IB: collected patient's consent.


Eosinophilic gastroenteritis (EGE) is a rare disease occurring commonly in patients with a history of allergies, asthma, food sensitivities, or parasite exposure. The condition has been postulated to occur when eosinophils accumulate at the intestinal epithelium resulting in inflammation, recruitment and degranulation of mast cells, and further propagation of eosinophil activation. While laboratory and microbiological data may point to the diagnosis, the condition is only confirmed via biopsy demonstrating the eosinophilic infiltration of the intestinal epithelium. Multiple medications have previously been implicated in causing EGE; however, this is the first documented case associated with emtricitabine/tenofovir. Eosinophilic gastroenteritis (EGE) is a rare disease that is challenging to diagnose based on clinical findings, especially in patients without a history of allergies, asthma, food sensitivities, or parasite exposure. We report a biopsy-confirmed case of EGE likely related to treatment with the combination of emtricitabine/tenofovir. Repeat administration of the drug resulted in abdominal symptoms and imaging findings consistent with colitis, which resolved following the cessation of therapy.A 54-year-old male with previously diagnosed human immunodeficiency virus (HIV) had recurrent presentations for colitis since initiating emtricitabine/tenofovir. In the past, he had self-discontinued this medication, resulting in the resolution of gastrointestinal (GI) complaints, but subsequent re-initiation of the medication led to a recurrence of symptoms and hospitalization. The abdominal computed tomography (CT) scan reported focal colitis in the descending colon, as seen in Figure The diagnosis of EGE is based on the presence of eosinophilic infiltration of the stomach, duodenum, esophagus, and colon without any known cause of eosinophilia . While pThis case should raise awareness that there are more undocumented medications that can cause EGE. We propose that EGE be considered in the differential diagnosis of patients taking emtricitabine/tenofovir who develop recurring gastrointestinal symptoms and have imaging findings consistent with colitis even if peripheral eosinophilia is not noted. Colonoscopy and biopsy may be warranted during the evaluation in order to definitively diagnose or rule out the disease.


Primary gastrointestinal lymphoma is the most common type of extra-nodal lymphoma, representing about 30%-50% of all extra-nodal involvement. The stomach is the most common site, with the colon and rectum accounting for a minority of occurrences. Primary colorectal lymphoma is uncommon, representing only 0.3% of all large intestinal malignancies and approximately 3% of gastrointestinal (GI) lymphomas, with the majority of these being B-cell non-Hodgkin lymphoma and diffuse large B-cell lymphoma (DLBCL) being the most common subtype. We present a case of an 85-year-old male who presented with symptoms suggestive of bowel obstruction, who, after further evaluation, was diagnosed with primary non-Hodgkin lymphoma of the colon, DLBCL subtype. The gastrointestinal(GI) tract is the most common site of primary extra-nodal lymphoma, with the vast majority being non-Hodgkin lymphomas (NHL). The colon is the primary site in around 17% of cases. Diffuse large B-cell lymphoma (DLBCL) of the colon is the most common subtype of extranodal non-Hodgkin's lymphoma, and while there is conflicting data regarding the most common part of the colon being involved, most studies report the cecum as the most common location. Due to the rarity of reported cases, there is no agreeable protocol on how to manage primary DLBCL of the colon. Currently, surgical resection is the cornerstone of treatment followed by the cyclophosphamide, doxorubicin, vincristine, and prednisone (CHOP) regimen-based chemotherapy and/or radiation therapy. The role of targeted therapy and immunotherapy is still being evaluated but may be a promising alternative to the present standard treatment. We present a case of primary DLBCL of the colon in an 85-year-old male patient followed by a literature review and discussion of the reported cases in the literature.An 85-year-old male with a history of atrial fibrillation presented to the hospital with acute shortness of breath and was subsequently admitted under the diagnosis of acute heart failure exacerbation. At admission, the patient was found to be anemic. His last colonoscopy was five years ago, with findings of lipoma in the ascending colon as well as sigmoid diverticulosis. At the time of admission, he had been more confused per his daughter. He was complaining of progressive fatigue and struggling with constipation, as he had been without bowel movement for several days prior to admission. He also had decreased appetite and lost around six pounds in the past four months. The patient subsequently had a chest X-ray that demonstrated multiple rounded pulmonary nodules throughout the lungs. The patient was clinically diagnosed with bowel obstruction. He subsequently underwent a computed tomography (CT) scan of the abdomen and pelvis, which showed a long apple-core lesion seen in the mid to distal sigmoid colon, which was about six cm in length with well-defined extension into the mesentery and discrete retroperitoneal lymphadenopathy. The patient then underwent a colonoscopy, which showed a near-complete obstructing sigmoid mass with ulceration. Multiple biopsies were taken from this sigmoid mass. The pathological evaluation was positive for atypical lymphoid cells Figure  that hadThe gastrointestinal (GI) tract is the most common site of primary extra-nodal lymphoma, with the vast majority being non-Hodgkin lymphomas. In recent years, there has been an increase in the incidence of primary GI non-Hodgkin lymphoma. A retrograde study  done in A retrograde study  that wasThe colon is a rare location for GI lymphoma, representing only 3% of all cases  and the DLBCL staging involves a CT or magnetic resonance imaging (MRI) scan, positron emission tomography (CT-PET) scan, and/or bone marrow biopsy in the case of advanced stages to determine bone marrow involvement. If neurological symptoms are present and central nervous system involvement is suspected, brain imaging and lumbar puncture might also be considered. Recent advances in gene expression profiling and immunohistochemical (IHC) analysis on tissue biopsy have allowed us to differentiate the subtypes of DLBCL and tailor treatment to each type. For example, patients with the activated B-cell (ABC) disease subtype are less likely to respond well to cyclophosphamide, doxorubicin, vincristine, and prednisone (CHOP)-based regimens than those with germinal center B-cell (GCB) disease . Corners In our review of the literature, we found multiple reported cases and case series, however, we only cited those that were crucial for the purposes of this report. We will give a brief discussion regarding the presenting epidemiology, presentation, and treatment options of these cases. The male to female ratio of the reported cases was 2.2:1, with 13 male patients and six female patients. The mean age was 68 years old. From the 19 cases, 18 mentioned the location of the lymphoma as following: sigmoid 44%, cecum 39%, and the ascending colon 17%. Presenting symptoms include abdominal pain, weight loss, diarrhea, constipation, lower GI bleeding, vesicocolic fistula, and altered mental status. The most common presenting symptoms were bowel obstruction and abdominal pain, present in 40% of 15 cases. Three patients had colonic perforation during admission, representing about 16% of the patient population. Sixty-eight percent of the patients had colectomy or hemicolectomy as part of their treatment with CHOP as the main chemotherapeutic regimen based upon the International Prognostic Index (IPI). The IPI is a common prognostic scoring system used for DLCBL, and it is also useful for primary DLCBL of the colon .Primary DLCBL of the colon is a rare entity representing only 1% of all colon malignancy, with the cecum being the most common location. Diagnosis is often established after a pathological evaluation of colonic biopsy or after colonic resection. Advances in gene expression profiling and immunohistochemical analysis (IHC) have allowed for tailored treatment for each type. Patients with the ABC disease subtype are less likely to respond well to CHOP-based regimens than those with GCB disease. Primary DLBCL of the colon remains an aggressive disease with a poor prognosis. Treatment options include surgical resection, chemotherapy, and radiation therapy. A bone marrow transplant is considered in select cases. Given the current era of rapid advancements in technology, targeted therapy and immunotherapy should also be considered as potential therapeutic options.


Arachnoid cysts (ACs) are congenital, extra-axial lesions containing fluid similar to the composition of cerebrospinal fluid. Usually found incidentally, these lesions are observed with serial imaging to document their growth patterns and stability, and are then followed conservatively until clinical symptoms develop. Surgical options for symptomatic arachnoid cysts include cyst aspiration, cyst evacuation with fenestration into the subarachnoid space, and shunt procedures including cysto-peritoneal and cysto-ventricular shunts. Intra-cystic hemorrhage and subdural hematoma are rare and more emergent sequelae of ACs that may require an emergent craniotomy. This case report further documents a rare cause of spontaneous subdural hematoma, as well as serves as a pivot point for further discussion into whether continued neuroimaging surveillance in patients with ACs would prove to be beneficial. Arachnoid cysts (ACs) are congenital, non-neoplastic, extra-axial, intra-arachnoid cystic lesions that contain fluid similar to the composition of cerebrospinal fluid -3. Most A 29-year-old Caucasian female with a past medical history of a migraine without aura, a left convexity arachnoid cyst Figures -1B, and Arachnoid cysts (ACs) are congenital, non-neoplastic, extra-axial, intra-arachnoid lesions that contain a fluid similar in composition to cerebrospinal fluid -3. They ACs are increasingly being recognized as a risk factor for the development of chronic subdural hematomas (CSDH) in children and young adults. A case series and review of the literature reported by Xuanxuan et al. described 14 cases of CSDH associated with ACs that required treatment with either burr hole drainage or conventional craniotomy . Six of Our patient presented with a worsening headache and right-sided weakness secondary to an AC that was complicated by an acute/subacute SDH with an intra-cystic hemorrhage component. Additionally, our patient’s arachnoid cyst was not in the middle cranial fossa, a location that seems to be more prone to AC-associated hemorrhage. The acute, atraumatic SDH our patient developed appears to represent an even more rare sequalae for ACs. Spontaneous intra-cystic hemorrhage and ASDH has been reported by Adin et al. in two patients at their institution . One patArachnoid cysts are typically incidental lesions that are found while the patient is obtaining neuroimaging for unrelated problems. They are benign, extra-axial lesions that usually do not result in neurologic deficits. We presented a unique case of a spontaneous acute SDH and intra-cystic hemorrhage associated with an AC in a 29-year-old female. AC-associated hemorrhage is more commonly found in the setting of recent trauma. Patients with a known AC need to be educated on the potential for both spontaneous or trauma-induced hemorrhage associated with their AC. Current imaging surveillance of these lesions is not standardized. Our institution typically repeats an MRI of the brain between six months to one year to document any growth of the AC. If the AC demonstrates stability, we do not employ any repeat imaging unless clinically indicated. Whether longer imaging surveillance would be beneficial to detect AC-associated hemorrhage and lead to earlier definitive treatment, is a question worth asking and would require studies to evaluate the cost-effectiveness and outcomes of longer imaging surveillance.


Fibroadenomas are common benign lesions of the breast that are usually found young patients. Giant fibroadenomas are uncommon benign lesions, defined as fibroadenomas of >5 cm in size, which are usually found in patients of less than 20 years of age.A 39-year-old premenopausal woman presented with a right breast tumor that had rapidly increased in size and which showed ulceration and bleeding. Needle biopsy showed mixed connective tissue and an epithelial tumor without a leaf-like pattern, but indeterminate. Total mastectomy and skin grafting were performed. Histopathology confirmed the diagnosis of giant fibroadenoma.In comparison to all previous reports on patients with giant fibroadenoma, this patient was relatively old and the etiology was unknown. Although rare, an appropriate therapeutic strategy should be decided according to the results of a histopathological examination.Early treatment could allow breast preserving surgery and patients should be recommended to undergo reexamination with awareness of progression. They feature polyclonal proliferation of both epithelial and stromal tissue, and are hyperplastic lesions rather than true neoplasms. Fibroadenomas usually present as hard, mobile, painless, obvious boundaries and easily accessible nodules . Giant fThe work has been reported in line with the SCARE criteria .2The patient was a 39-year-old woman who had become aware of a right breast mass 7 months previously. The mass had rapidly increased in size with the manifestation of ulceration and bleeding. She came to the hospital for emergency hemostasis. Her menstrual cycles were regular and she was not on any hormone therapy. A clinical examination showed a firm and mobile breast tumor on the outside of the right breast of approximately 20 cm in size, with a necrotic part in the lower quadrant of the breast, thickening of the surrounding skin, and capillary protrusion . The rigThe patient underwent total mastectomy and skin grafting, with skin taken from the lower abdomen under general anesthesia. The actual size of the tumor was 23.5 × 24.5 × 11 cm and the weight was 3.2 kg . The pat3While fibroadenomas are common benign lesions in breast tissue, giant fibroadenomas are very rare. Giant fibroadenomas are usually encountered in pregnant or lactating women. Their growth is associated with increases in estrogen, progesterone, and prolactin . Those fInitially, we suspected a malignant tumor, such as a malignant phyllodes tumor or breast cancer, due to the size and the increase in size over a short period with ulceration and bleeding. US is non-invasive modality and is useful in the diagnosis of lesions in breast tissue. On US, fibroadenomas appear as a well circumscribed round or oval solid mass, with weak internal echoes in a uniform distribution and intermediate acoustic attenuation ,6. SmithBoth fibroadenomas and phyllodes tumors consist of proliferating epithelial and stromal cells. The differential diagnosis between fibroadenomas and phyllodes tumors is clinically important due to differences in therapeutic strategies. Fibroadenomas can be followed up safely or simply enucleated, while phyllodes tumors must be treated surgically . FurtherIn our case, the preoperative CEA level was mildly elevated and decreased to the normal range after surgery. We could not find any malignant features in the resected specimen. CEA is a glycoprotein involved in cell adhesion, which is normally produced in gastrointestinal tissue during fetal development . The lev4Skin grafting or plastic surgery might be needed for patients who undergo mastectomy for giant fibroadenoma. An early diagnosis and treatment could prevent this situation and patients should be recommended to undergo reexamination with awareness of progression.No funding on this study.Approval to publish this case report was waived by the institution.Written informed consent for publication of this case report and accompanying images was obtained from the patient. A copy of the written consent is available for review by the Editor-in-Chief of this journal, on request.KY, SK, CS, KM, and NH performed the surgery and were responsible for the care of the patient. KA performed the pathological examination. MX, KY, and CS designed and drafted the manuscript. MM, KK, MT, KA and SE reviewed and revised the manuscript.This is not systematic review or meta-analysis. Also this is not randomised clinical trial.Kosho Yamanouchi MD, PhD.The authors declare that the work described has not been published previously, that it is not under consideration for publication elsewhere, that its publication has been approved by all authors and either tacitly or explicitly by the responsible authorities where the work was carried out, and that, if accepted, it will not be published elsewhere—including electronically in the same form in English or any other language—without the written consent of the copyright holder.Not commissioned externally peer reviewed.The authors report no conflict of interest.


Penoscrotal edema is typically caused by lymphatic obstruction, which can have both primary and secondary causes. Studies describing congenital penoscrotal edema are rare. Surgery can be divided into two types: The first approach involves extensive removal of diseased tissue and tissue reconstruction. The second approach is removal of the lesions and creating additional lymphatic vascular anastomoses.We present a case report of a 15-year-old patient with recurrent penoscrotal edema and swelling of both lower extremities. The literature were also reviewed to provide additional information. Physical examination revealed slow lymphatic reflux of the lower extremities and no obvious abnormalities in testicular morphology, bilaterally, or blood supply. Surgery was performed by excising the affected skin and subcutaneous tissue and the flaps was cut in the middle in Y shape to cover the penis and scrotum. Postoperative follow-up revealed wound integrity and patient satisfaction with the outcome.Excision and reconstructive surgery are the primary treatments for penoscrotal edema. The majority of reported patients undergoing excision and reconstruction achieved satisfactory reshaping and improved their life quality.The online version of this article (10.1186/s12894-019-0456-6) contains supplementary material, which is available to authorized users. Penoscrotal edema is typically caused by lymphatic obstruction, which can have both primary and secondary causes. Lymphatic malformations result from congenital causes (primary lymphedema). Acquired (secondary) chronic genital lymphedema can be caused by genital infection, tumors, lymphadenectomy, injury, or irradiation. Scrotal lymphadenopathy may be transient or persistent, and occurs at any age. For some patients, conservative treatment is sufficient and in others, the appearance of the edema or a patient’s quality of life requires surgical treatment to improve , 2.Studies describing congenital penoscrotal edema are rare –5. TreatSurgery can be divided into two types: The first approach involves extensive removal of diseased tissue and tissue reconstruction. The second approach is removal of the lesions and creating additional lymphatic vascular anastomoses. The former is the classic approach, which is also more extensive . Many teA 15-year-old boy presented to Fujian Medical University with giant scrotal elephantiasis and swelling of both lower extremities. The penoscrotal edema began fifteen years earlier, soon after his birth, and it resulted in bilateral lower extremity edema with the penis becoming buried by the scrotum. His scrotal size was massive, and for the past 5 years, the glans penis was not visible nor palpable  or caused by obstruction or disruption in the lymphatic vessels (secondary), which develops more frequently than primary lymphedema , 2, 11. Patients most often suffer from a sense of heaviness and fatigue. In later stages, the hyperkeratotic skin forms a vesicle filled with exudative lymph. The damaged skin barrier allows bacteria into the protein-rich lymphoid fluid in the vesicle, which frequently leads to cellulitis and postoperative complications such as abscesses, wound infections, and wound dehiscence.The main purpose of surgery is to reduce scrotal volume, reconstruct the scrotum, and repair the skin of the penis . A previCases of penoscrotal edema are rare, and some reported cases are summarized in Table Pathological results have not been reported in every previous study. However, the most significant microscopic features in previous reports were non-specific inflammation with connective tissue proliferation; hidradenitis suppurativa was found in Konety et al.’s study . The penExcision and reconstructive surgery are the primary treatments for penoscrotal edema. The majority of reported patients undergoing excision and reconstruction achieved satisfactory reshaping and improved their life quality to some degree within a 2–3-year follow-up, including patients who underwent microlymphaticovenous anastomosis. No recurrence was reported in the previous reports listed in our study. Our patient’s incisions healed in 2 weeks, with good final appearance and satisfactory erectile function by 3 months, postoperatively.Additional file 1:Images of the scan of lymphoscintigraphy (Transverse section). (JPG 1114 kb)Additional file 2:Images of the scan of lymphoscintigraphy (Transverse section). (JPG 1118 kb)Additional file 3:Images of the scan of lymphoscintigraphy . (JPG 1112 kb)Additional file 4:Images of the scan of lymphoscintigraphy . (JPG 896 kb)


Hepatic angiosarcoma (HA) accounts for 2% of primary liver tumors. Though rare, it is exceptionally deadly. The initial presentation of HA is nonspecific and no tumor markers have been associated with it. In general, liver function is maintained until later stages of the disease, often leading to diagnosis once the disease is already advanced or metastatic. In this report, we present the case of a 54-year-old male whose vague symptoms and non-diagnostic liver biopsy delayed the diagnosis of a rapidly progressing HA. Hepatic angiosarcoma (HA) is a particularly rare, non-cirrhotic, primary malignancy of the liver, accounting for 2% of liver cancers . HoweverDemographically, the malignancy has a male to female predominance of 3:1 with the majority of patients diagnosed in their sixth decade of life . In thisThe patient, a 54-year-old male with a past medical history significant for coronary artery disease, hypertension, and hyperlipidemia, initially presented with complaints of dyspnea on exertion for several weeks. The patient reported experiencing recent weight gain, increased abdominal girth, and lower extremity edema. An inpatient echocardiogram showed moderate pericardial effusion with possible markers for tamponade. He underwent a pericardial window computed tomography (CT) scan that showed two incidental hypoattenuating foci in the liver, the largest measuring 2.2 cm in diameter Figure . There wTwo months later, the patient returned to the hospital due to increasing abdominal pain. A CT scan of the abdomen and pelvis showed new lesions and nodules as well as evidence of hemoperitoneum presumed to be due to ruptured hepatic and splenic lesions. At the time, his laboratory findings showed: total bilirubin 3.7 mg/Dl, direct bilirubin 1.0 mg/Dl, AST 108 U/L, ALT 105 U/L, ALP 250 U/L, platelet count 29 Thou/uL, and lactic acid 4.6 mmol/L. A second liver biopsy was performed and pathology showed solid spindle cell proliferation. Immunohistochemical staining was positive for cluster of differentiation (CD)31, CD34, and Factor VIII, indicating likely HA (Figure The patient was subsequently started on a cycle of gemcitabine. A follow-up MRI of the abdomen and pelvis two weeks later showed a progression of metastatic disease within the liver, spleen, spine, lung bases, and pericardium, with many of the metastases demonstrating signal characteristics consistent with interval hemorrhage Figure . The larThe liver makes up only 5% of all primary angiosarcoma lesion sites . In caseRadiologically, HAs vary in their appearance. Grossly, the tumor may appear with one of four different growth patterns: multinodular, a single dominant mass, mixed patterns of a dominant mass with smaller nodules, or an infiltrating micronodular tumor . MetastaPrimary HA is diagnosed based on the results of a histopathological examination. Though historically, HA has been linked with environmental toxins, including thorotrast, arsenic, radiation, vinyl chloride, anabolic steroids, and exogenous estrogens, most cases of HA never yield a causative factor. It should be noted that there is no difference in gross or microscopic pathology between the toxin-related HAs with their 20- to 30-year latency periods and idiopathic HAs . In our Treatment for HA has yielded generally poor outcomes due to the late stage and profound metastasis at diagnosis. Better prognosis is seen in patients that present with a single tumor mass, small tumor size, lack of metastases, low-grade lesion, and negative surgical resection margins . For thoHA is a malignancy that combines a nonspecific presentation with a very rapid progression of illness and, ultimately, death. While imaging and biopsy are important instruments in disease diagnosis, their sensitivities are not absolute. Early clinical suspicion and surgical intervention remain paramount in reducing the risk of mortality in HA.


Intramural hematoma of the colon is a rare complication of colonoscopy. We present a case of a 78-year-old woman on warfarin who presented with hematochezia and hypotension due to intramural hematoma of the sigmoid colon after colonoscopy with polypectomy of small polyps in the right colon. Spontaneous hematomas of unknown etiology have also been described.4 Only a few cases after diagnostic lower GI endoscopy without or with polypectomy have been published.9 Antithrombotic therapy and a redundant sigmoid colon, which often results in a difficult endoscopic procedure, looping, and requirements for increased external abdominal pressure are the main risk factors for a colonic hematoma after colonoscopy.5 Patients most commonly present with abdominal pain and hematochezia.10 Diagnosis is confirmed with colonoscopy and abdominal contrast-enhanced computed tomography.10 Initially, conservative therapy is indicated, but when symptoms persist, surgical resection is required.10 A fatal clinical outcome is rare.9Intramural hematoma of the gastrointestinal (GI) tract is rare and is least common in the colon.2 was used for insufflation.A 78-year-old woman with a medical history of congestive heart failure, pacemaker implantation due to iatrogenic atrioventricular block after heart surgery, anticoagulant therapy with warfarin for a mechanical aortic and mitral valve, chronic kidney disease stage III, arterial hypertension, and with no history of abdominal surgery was admitted due to hematochezia that had started a few hours earlier. Three days previously, she had a colonoscopy for a further diagnostic workup of microcytic anemia. The endoscopic procedure was difficult due to a redundant sigmoid colon. As a result, increased external abdominal pressure was applied to reach the cecum. The procedure was prolonged; the total procedural time was 75 minutes. CODuring the withdrawal, 2 sessile polyps 8 and 3 mm in the cecum were resected with a cold snare. Histology revealed low-grade dysplasia adenomas. On physical examination, she appeared in mild distress. Digital rectal examination revealed hematochezia, and she was hypotensive. On examination, her abdomen was soft and non-tender. Hemoglobin was 7.9 g/dL, platelets were 255,000 μ/L, and the international normalized ratio was 1.05. Baseline hemoglobin was 9.6 g/dL. The patient received the last dose of low–molecular-weight heparin (LMWH) dalteparin 16 hours before diagnostic colonoscopy, which was reintroduced in the same evening after the procedure and continued in full dose until she was admitted to the hospital.On arrival, post-polypectomy bleeding was suspected and an emergent colonoscopy was performed Figure . She recDuring the hospitalization, the patient had several minor self-limiting episodes of hematochezia. After 9 days, we performed a control sigmoidoscopy Figure . Erythem11 The estimated incidence of GI hematomas in anticoagulated patients is 1 in 250,000.4 The main causes are trauma, anticoagulation therapy, and hematologic disorders. Spontaneous hematomas of unknown etiology have also been described in the literature.4 Only a few cases after diagnostic lower GI endoscopy without or with polypectomy have been published.9 The common feature in almost all described cases is a difficult and long colonoscopy due to a redundant colon.Hematomas have been reported to occur in all parts of the GI tract but are least common in the colon.7 Our patient presented with hematochezia 72 hours after the colonoscopy. Unlike in other described cases, abdominal pain was not present in our case. In most cases, the clinical picture improved after the correction of anemia with transfusions and hemodynamic resuscitation, which are the cornerstone of conservative management of intramural colon hematoma.6 Our patient was hospitalized for 2 weeks.We assume that sigmoid hematoma in our case was caused by the colonoscope shaft in combination with prolonged and increased external abdominal pressure. In cases of intramural hematoma after diagnostic colonoscopy without polypectomy, the patients noticed hematochezia 6 and 12 hours after the procedure, and in one case, hematoma was noticed during the same colonoscopy on withdrawal.2DS2-VASc score was at least 5) and a high risk of thrombosis if left without anticoagulant therapy. Surgical therapy is required when symptoms persist or the clinical picture deteriorates.9 The outcome of colon intramural hematoma is rarely fatal.9 A similarity between our case and those published in the literature is that more than half of the patients with intramural hematoma were taking antithrombotic agents.9The main reason for the prolonged hospitalization was several episodes of self-limiting hematochezia with the need for further transfusions and the need to monitor reintroduction of LMWH. In addition, our patient had a high risk of stroke and cardiovascular complications  and bridged with LMWH.Author contributions: A. Gavrić wrote and reviewed the manuscript. R. Dežman reviewed the manuscript and provided and commented CT images. S. Stefanović, J. Drnovšek, and B. Štabuc reviewed the manuscript. A. Gavrić is the article guarantor.Financial disclosure: None to report.Informed consent was obtained for this case report.


Echinococcus granulosus has an osseous involvement of about 0.5%–2.5% of all cases in humans. The location of hydatid cysts in the tibia is seldom described in the medical literature, and its diagnosis and treatment is challenging.Hydatid disease in humans caused by the parasitic tapeworm This paper presents a patient, with a long term, well tolerated, bilateral total knee arthroplasty (TKA), treated at our clinic, with a recent history of pain and oedema in her left upper leg. After achieving a, radiologically and histhopathologically, well documented, diagnosis of Echinoccocosis lesion in her left proximal tibia, a surgical intervention was planed, a wide resection of the cyst performed, and a specific anti-helmintic therapy was instituted. Four years later, she returns to our observation complaining of pain and unable to bear weight on her left knee, from which a pathologic fracture, adjacent to the tibial component, was diagnosed.After surgical debridement of the newly advanced hydatid cyst growth, the TKA was revised, and due to the tibial component failure and the femoral implant loosening, a semi-constrained total knee revision arthroplasty was executed. Functional outcome was excellent.Although challenges in treatment of musculoskeletal hydatid cysts (HC) lesions have been documented, data regarding the musculoskeletal HC lesions resembling tumor is scarce, and those resulting in a prosthetic failure have not been published. The authors intend to add data concerning the therapeutic approach to this entity. This parasite has 12 different types, though 4 types are known to cause disease in humans: garthrus . The strgarthrus . Clinicagarthrus  Althoughgarthrus .2Pseudomonas aeruginosa, and appropriate antibiotics where then administered according to the susceptibility test carried out. Pathology results revealed hydatid cyst of the tibia. Segmental resection was planned, and the surgical approach revealed a diaphyseal cyst adherent to the surrounding tissues, which were markedly oedematous, with multiple membranous whitish tissues in aggregation. Fluid was aspirated from the cyst, and the sample was sent for microbiology and serology tests. After curettage of the lesion and power-pulse lavage, povidone-iodine-alcohol solution was injected. Due to the fragility of the remaining tibial diaphysis, an external fixator was applied. Microscopy confirmed the diagnosis and revealed osseous tissue with hyaline and germinative membranes, lymphocytes, and monocytes.In 2010 a 75 year-old female patient presented at our clinic with a 2-year history of pain and recent emergence of a discharging sinus at her left upper leg. She had a history of bilateral gonarthrosis and underwent elective right and left knee total arthroplasty 5 years before. The procedures and the post-operative follow-up were uneventful. Her physical examination revealed slight swelling and tenderness with a mild seropurulent discharge on the antero-lateral aspect on her proximal left leg. There was no other systemic complaint. Her personal and family histories were unrevealing. There was no history of fever, trauma, previous tuberculosis or bone tumors. Lower limb x-rays were performed and the radiographic examination revealed a well demarcated cystic structure in her left tibia, 4 cm below the distal tibial component of the knee arthroplasty . A purulAlbendazole and praziquantel, antihelminthic drugs, at doses of 10 and 25 mg/kg, respectively, were started. The patient recovered uneventfully and was discharged shortly after the procedure. She was clinically and radiologically evaluated in the following months, revealing progressive bone growth, remodeling and consolidation, having the external fixator extracted 6 months after the initial procedure.The patient then remained exempt of pain, swelling or other complications, until May 2014, when suddenly she comes to our clinic, complaining of pain and unable to bear weight on her left knee.X-ray studies revealed a pathologic periprosthetic fracture, below the tibial component, resulting from an extension of the previously treated hydatic cyst . A treat3Periprosthetic fractures around the knee can be seen in femur, tibia and patella after total knee arthroplasty (TKA) . The treHowever, there have been, to our knowledge, no reported cases of tibial component failure following a fracture due to an Hydatid Cyst of the proximal tibia.The case presented demonstrates how Echinococcosis develops via exogenous vesiculation in bones, eventually leading to mechanical weakening and destruction of the bone trabeculae. The lesion was infiltrative in nature and progressed slowly, culminating in a pathologic fracture below the tibial component of the knee arthroplasty.We found that the pathological fracture was associated with accelerated wear and more extensive osteolysis. Both lead to poor prosthetic survival, as seen with the loosening of the femoral component.In the present patient, severe bone defects and lateral laxity due to huge cystic lesions compelled us to use a semi-constrained type prosthesis with a long stem. In this case, wide, radical surgical excision of the cyst required removal of most of the proximal metaphysis, and hence the implantation of this type of prosthesis, as seen in other published work . The bonLiterature supports a combined surgical and medical approach to cases of musculoskeletal infection with Echinococcus, although the specific duration of antihelminthic therapy is yet to be determined ,16. We bIn this case, the treatment provided the patient with relief of pain and stable daily life. However, the follow-up period is short; thus, a longer-term follow-up is needed to investigate the real clinical usefulness of the treatment, and moreover, to evaluate the possibility of recurrence of the disease with respect to its known high recidivism rate.4Locally aggressive, benign bone tumors require specific techniques to avoid recurrence, thus the experience from their treatment can be used as an analogy for the treatment of musculoskeletal hydatidosis. Wide resection is essential to achieve local control and reduce the recurrence rate, and the massive defect presents a significant challenge to the reconstructive orthopedic surgeon.Prompt diagnosis is of paramount importance for preventing destruction and complications and early treatment is essential if the affected bone is to be fully eradicated and the healthy tissue salvaged. Complete surgical excision is the treatment of choice for osseous hydatid disease.This case report supports the use of massive bone autograft as a valid reconstruction technique after extensive bone resection as a result of hydatidic disease.The authors declare that they have no conflict of interest.The authors declare there was no funding in collection, analysis, interpretation of data, writing and submitting this manuscript.The ethics committee of our institution  has approved the submission and publication of this article.A written and signed consent was obtained from the patient and a copy is available for review by the Editor-in-Chief of this journal on request”.All the authors have been involved in the creation of this manuscript.Dra Carmen Silva and Dr. Jorge Santos have clinically evaluated the patient, which was thereafter treated, both medically and surgically. Dr João Leiria also guided the strategy of treatment and leaded the revision procedure.Dr. Germano Nascimento, Dra Raquel Marques, Dr. João Francisco and Dr. Carlos Silva, made all the literature research, and draft of the manuscript.Registered in ClinicalTrials.gov Identifier: NCT03622346.Dr Germano Nascimento.Not commissioned, externally peer-reviewed.


Splenogonadal fusion is a rare and benign condition. Diagnosis is challenging for clinicians. Despite its indolence, diagnosis is often confirmed after orchidectomy. Surgery is mandatory, particularly to rule out the extremely rare association with malignancy.We report a case of splenogonadal fusion in a 38-year-old North African man presenting a palpable scrotal mass. We describe clinical aspects, pathogenic hypothesis, radiological features, as well as surgical management principles.Splenogonadal fusion is rarely suspected and diagnosed preoperatively. A diagnosis is made once an ectopic testicular mass is associated with cryptorchidism and suggestive radiological signs. A better knowledge of the clinical and radiological features of splenogonadal fusion provides an opportunity for conservative surgery. Splenogonadal fusion is a rare congenital defect, defined by the presence of ectopic splenic tissue in the scrotum. It is usually discovered in adolescents with a scrotal swelling. As imaging findings are not distinctive, histological diagnosis is often made after radical orchidectomy. We report a new case of splenogonadal fusion in an adult patient presenting a palpable scrotal mass. Pathogenic and clinical features, imaging findings, and surgical management will be described and discussed.Mycobacterium tuberculosis screening. A scrotal ultrasound showed a homogeneous testicular parenchyma, with a conserved vascularization on Doppler. An extratesticular mass was observed, attached to the upper pole of his testis. The mass was isoechoic to the testis parenchyma, and poorly vascularized Doppler , and lactate dehydrogenase (LDH) were in the normal ranges. There was no bacterial growth in urine analysis, including ler Fig. .Fig. 1ScHe underwent a radical inguinal orchiectomy. We first performed a high ligation of the spermatic cord. The operative specimen included the testis and the tunica vaginalis in one piece Fig. . The macHistological examination of the operative specimen confirmed the presence of regular splenic tissue in the suspect mass, without any signs of malignancy. The splenic proliferation had its proper and regular capsule, demarcating it from the testis. Testicular pulp, the albuginea and the tunica vaginalis had a preserved microscopic architecture Fig. . He was Splenogonadal fusion is a rare congenital anomaly, first described in 1883 by Boestrom .It occurs generally on the left side. Splenogonadal fusion is not exclusively observed in male patients. A few cases were reported in female patients. It is underdiagnosed in female patients, as ovaries are less accessible to physical examination .Two types, with an equal frequency, were described by Putschar and Manion : the conThe clinical presentation is not specific and diagnosis is often made on histological examination of the operative specimen. The discontinuous form usually presents as a hard scrotal nodule, mimicking a testicular tumor. In some cases, scrotal swelling is associated with specific splenic conditions, such as infectious mononucleosis or salmonellosis . A bowel99mTc) is a valuable option once splenogonadal fusion is suspected. Radioactive tracer fixation in the spleen and the suspect mass is similar, confirming the ectopic splenic origin of the testicular appended mass [Scrotal ultrasonography is not accurate enough preoperatively; as sonographic aspects are various, sensitivity and specificity are not high . Magnetided mass .Even if splenogonadal fusion is almost always a benign condition, surgery is still mandatory. In fact, histological examination of the operative specimen ensures formal diagnosis and rules out the infrequent association with testicular malignancies . The assEven if imaging affords a better prediction and diagnostic orientation, many patients still undergo unnecessary orchiectomies. Once operative aspects are known, a surgeon can opt for conservative treatment, saving the testis . In the A better knowledge of splenogonadal fusion may increase the preoperatively indicated and diagnosed cases, in order to reassure patients and adjust the therapeutic attitude. Surgery remains essential, as malignancy must be ruled out. More specific imaging features and extemporaneous histological examination enable avoidance of an unnecessary orchiectomy.


Pituitary abscess is a rare condition with nonspecific symptoms that can be delayed. Proper diagnosis needs to occur preoperatively so that the management can be set up accordingly. Accurate diagnosis is challenging because many differential diagnoses can exhibit the same magnetic resonance imaging features.We report two cases of pituitary abscess. The first patient was a 66-year-old Arab woman who underwent a surgical procedure for a pituitary macroadenoma and presented 3 months later with chronic headaches and panhypopituitarism. A pituitary abscess was found on the follow-up magnetic resonance imaging. The second patient was a 64-year-old Arab man with no medical history who presented with a chiasmal syndrome with headaches and panhypopituitarism. Brain magnetic resonance imaging showed a heterogeneous pituitary mass that turned out to be a pituitary abscess intraoperatively. These two patients were treated with hormone substitution, endoscopic transsphenoidal drainage, and antibiotherapy, with excellent outcomes.Pituitary abscess is a rare and serious condition. Preoperative diagnosis can be challenging because of the many existing differential diagnoses upon imaging. Magnetic resonance imaging is the mainstay technique of imaging due to its multimodal nature. These cases demonstrate the variable patterns of a pituitary abscess seen on magnetic resonance imaging and the potential difficulties in achieving an accurate diagnosis preoperatively due to many other conditions potentially exhibiting the same magnetic resonance imaging features. The pituitary abscess (PA) is a rare clinical entity, often with poor prognosis , accountThis report describes two cases of a rare condition and aims to serve an educational purpose for radiologists to increase their awareness of this condition, especially among patients who have risk factors.9/L (4.0–11.0 × 109/L).A 66-year-old Arab woman with diabetes mellitus of 4 years’ duration underwent transsphenoidal (TSS) resection of a nonfunctioning pituitary macroadenoma in May 2018. Her immediate postoperative course was free of events. Three months after the procedure (August 2018), she checked in to our institution with chronic headaches. Her physical examination showed bitemporal hemianopia. Relevant blood screening results were as follows: thyroid stimulating hormone ultra sensible (TSHus), 0.3 mIU/L ; free thyroxine (FT4), 0.7 ng/dl (1–2); adrenocorticotropic hormone (ACTH) (8:00 a.m.), 3 pg/ml (10–40); cortisol (8:00 a.m.), 6 μg/dl (10–20); follicle stimulating hormone (FSH), 20 IU/L ; luteinizing hormone (LH), 10 IU/L ; estradiol, 1.2 pg/ml ; growth hormone (GH), 2 ng/ml (< 10 [women]); prolactin, 0.7 ng/ml (2–20 [nonpregnant]); C-reactive protein (CRP), 2 mg/L (< 3.0); and white blood cells (WBC), 10.5 × 10Brain MRI showed a pituitary mass measuring 4 cm of the major axis, exhibiting low T1-weighted (T1w) signal, high T2-weighted (T2w) signal, and diffusion-weighted imaging (DWI) with rim enhancement after injection of gadolinium. The mass was responsible for a compression of the optical chiasma  leak in conjunction with a valsalva maneuver. Closure was achieved with Gelfoam packing  and Surgicel overlay . The purulent material grew a  PA Fig. .Fig. 2S9/L (4.0–11.0 × 109/L).A 64-year-old Arab man with no major medical history consulted in December 2017 for chronic headaches and progressive vision loss, evolving for 1 year, with neither fever nor asthenia. His physical examination was remarkable only for a chiasmal syndrome. He did not have diabetes insipidus. Relevant blood screening results were as follows: TSHus, 0.25 (0.5–5); FT4, 0.7 ng/dl (1–2); ACTH (8:00 a.m.), 5 pg/ml 10–40); cortisol, 3 μg/dl (10–20); FSH, 0.7 IU/L  and PA. These MRI findings were mostly in favor of a remodeled pituitary mass. That is what was shown on the final MRI report.Staphylococcus aureus, and histopathological examination showed no tumoral lesion. On the basis of an antibiotic sensitivity test, targeted antibiotherapy was administered for 3 weeks. The postoperative course was uneventful, with progressive improvement of the clinical state. Follow-up MRI at 3 months showed complete drainage of the abscess , accounting for about 70% of cases, and secondary PA arising within preexisting pituitary lesions, accounting for 30% [PA is a rare condition, often with a poor prognosis. Only about 200 cases have been described in the literature to date  since th for 30% . The pri for 30% , 8. PA c for 30% , 10, 11. for 30% , 13. PA  for 30% , 8.Many authors believe in the existence of an “abscess-sterile” entity or a “noninfectious PA” because the presentation does not always involve fever and high blood leukocyte count and because there is a high percentage of negative microorganism culture results. Although some authors believe that a sterile abscess is not a true abscess, but rather a liquefaction of necrosis of an infarcted pituitary gland or the contents of an atypical pituitary cyst, others maintain that a sterile culture may be due to an inadequate bacteriological technique in regard to fungus and anaerobic bacteria difficult to culture or to a broad use of antibiotic therapy initiated before or during surgery , 14.The clinical presentation of PA is usually misleading because there are no specific symptoms. The symptoms are predominantly represented by headaches, pituitary hypofunction, visual disturbances, and an increased intracranial pressure syndrome, whereas infectious syndrome is often discreet and inconstant , 8, 14. An accurate diagnosis has to be ensured preoperatively so that management can be set up accordingly. Multimodal MRI is the mainstay imaging technique to ensure a proper diagnosis. However, despite all the advances in MRI, accurate diagnosis is challenging because many other differential diagnoses can exhibit the same MRI features, thus leading to the high rate of misdiagnosed PAs .et al. [MRI scanning of PAs demonstrated an intrasellar/suprasellar (64.7%) or limited intrasellar 35.3%) cystic or partially cystic pituitary mass , 15–17. .3% cystiet al. case series, PA was misdiagnosed in one-third of the cases, which is one of the lowest rates of misdiagnosis of PAs in the literature [DWI is widely used to differentiate cerebral abscess from other necrotic masses . Brain aterature .In our patient 1, we did not have difficulties in achieving the diagnosis of PA, owing to the fact that classical MRI features were present. In addition, we were aided by the recent pituitary surgical procedure of the patient, because it represents a predisposing factor. However, in our patient 2, we could not achieve the proper preoperative diagnosis, because the patient had no relevant medical history, and his presentation on MRI was atypical. We associated these MRI characteristics with a pituitary adenoma with hemorrhagic necrosis and cystic degeneration. Accurate diagnosis was achieved intraoperatively. PAs can develop alongside or even complicating a local tumor, and close exploration after drainage and irrigation of the abscess showed no sign of tumoral mass in both of our patients.The diagnosis of PA is challenging, partly because of its rareness (leading to the physician’s lack of experience) and because many other pituitary conditions can exhibit the same MRI features. All these factors contribute to the relatively high rate of misdiagnosis of PAs. Radiologists have to be aware of these intricacies and react accordingly by considering PA as a possible diagnosis, mainly in the presence of underlying conditions favorable to the development of a PA, such as a history of pituitary surgical or radiation interventions, growing local infection or mass, diabetes mellitus, and immunodeficiency. The main differential diagnoses are pituitary adenoma (with necrosis or cystic degeneration), pituitary apoplexy, Rathke’s cleft cyst, cystic craniopharyngioma, and lymphocytic hypophysitis (mostly in children) .Regarding the treatment of PAs, although antibiotics alone could be successful , most auet al. [Prompt diagnosis and treatment of PAs yield a favorable prognosis. In the systematic review of the literature by Agyeis et al. , visual In conclusion, PA is a very rare disease of the sellar region that is difficult to correctly diagnose on the basis of clinical manifestations and MRI. Despite advances in MRI, preoperative diagnosis of PA is still challenging to radiologists. MRI of PAs usually reveals a cystic or partially cystic pituitary mass, sometimes accompanied by some special signs. Despite its rareness, a PA must be considered in the case of a pituitary mass associated with intracranial hypertension, pituitary hormone dysregulation, and an infectious syndrome (which is a less reliable sign). In addition, there are some underlying predisposing factors, such as a history of pituitary surgical or radiation interventions, local infection or growing local mass, and diabetes mellitus. The treatment is a combination of surgery, antibiotics, and hormone substitution when needed. These cases demonstrate the variable patterns of PA in MRI and the potential difficulties in achieving an accurate diagnosis preoperatively, because many other pituitary conditions can exhibit the same MRI features. However, MRI remains the mainstay technique of imaging because of its multimodality. By associating these two cases, we aim to show a typical MRI finding of PA (patient 1) and an atypical one (patient 2). Radiologists have to be aware of the variable MRI patterns of this condition and deal with the findings accordingly.


Phyllodes tumors are biphasic fibroepithelial tumors that account for fewer than 1% of mammary tumors. They usually appear in middle-aged women, with an average size of 4–7 cm and rapid growth.A 25-year-old woman sought care after excision of recurrent nodules in the right breast, with a diagnosis of borderline phyllodes tumor. She had no new lesions on physical examination. Imaging studies revealed an anechoic collection and nodular areas in a previous surgical site, correlated with pathology. Immuno-histochemical examination was positive for vimentin, calponin, Ki-67 and estrogen receptor. Systemic staging did not show metastases. Skin-reducing mastectomy was performed on the right side, followed by reconstruction and left reduction mammaplasty for symmetry. At 8-month follow-up, there was no relapse of the lesions on clinical and ultrasonographic examinations.Phyllodes tumors are rare neoplasms characterized by rapid growth, occurring in women between 35–55 years of age. They are classified as benign, borderline and malignant, according to histological parameters. There are reports of genetic mutations in TP53 associated with malignant phyllodes tumor. On immunohistochemistry, there is a greater tendency to malignancy in tumors with Ki-67 and estrogen receptor expression. Imaging methods may aid in diagnosis, which is only definitive after excision and histopathological analysis of the tumor. These tumors have high local recurrence rates and possibility of metastases, closely related to histology.Phyllodes tumors are a great challenge to the surgeon. Treatment is surgical, with wide excision; radiotherapy and chemotherapy are controversial. It is also known as cystosarcoma phyllodes, adenomatous myxoma and pseudosarcoma adenoma [Phyllodes tumors (PTs) account for fewer than 1% of mammary tumors [ adenoma . PTs are adenoma ,4. They  adenoma . Because adenoma .The objective of this study, reported according to SCARE criteria , is to d2A Caucasian female sought care at 22 years of age for evaluation of a mass characterized by progressive growth located laterally in the right breast, measuring 3.3 cm; she underwent surgical excision, with anatomic-pathological examination suggestive of FA. One year later, the patient had a palpable mass of 4.7 cm in the same location, and a lumpectomy was performed, with a repeated diagnosis of FA. At eight and 12 months thereafter, two new tumors, both 4.5 cm, were resected, and benign PTs were diagnosed with compromised resection margins. After another eight months, at the age of 25, three new lesions appeared; all nodules were excised, with borderline PT diagnosis, positive margins for the largest (3 cm) and benign PT for the other two.Over time, a 6-cm scarring lesion was presented, laterally in the right breast, consequent to the previous procedures, and thickening of the underlying tissue, without phlogosis . On palpAll lesions occurred in the right breast and were asymptomatic. Because of the history of frequent relapses, a right-sided skin-reducing mastectomy was chosen, followed by reconstruction. The intraoperative frozen section showed a scarring lesion underlying the surgical marking and another nodular hemorrhagic lesion in a super-medial quadrant with resection margins greater than 1.0 cm in relation to the adjacent tissues. On the left, reduction mammaplasty was chosen for symmetry, because of the patient's large breast volume , Fig. 3.3PTs are rare breast neoplasms characterized by rapid growth ,7,8. TheHistologically, PTs have leaf-like cellular lobulations and biphasic fibro-epithelial constitution, with predominance of stromal elements over epithelial cells. The World Health Organization classifies them as benign 60–75%), borderline 13–26%) and malignant (10–20%) [6% and ma%, borderUnlike breast cancer, which has a well-established association with mutations in the BRCA1 and BRCA2 genes, there remains insufficient evidence linking genetic variations to the development of PT. There are reports of mutations associated with malignant PT, as in the PT53 gene; this was absent in our patient, whose test showed only variants of uncertain significance in BRCA1 and BRCA2 .Diagnostic imaging methods have low accuracy . US and While malignant PTs have high potential for local recurrence and metastases, benign PTs tend to recur only locally . Local rTreatment of PT is surgical, regardless of subtype, ranging from wide local excision to mastectomy. Currently, there is a greater tendency towards conservative approaches, because PT is rarely multifocal. Recent guidelines recommend that PT > 3 cm be resected with free margins ≥1 cm, with no need for axillary staging; lumpectomy may be indicated for small tumors, because it allows preservation and mammary function ,17,20. SAdjuvant therapy has no proven efficacy in terms of disease-free survival, and was not provided in this case. Some studies, although controversial, suggest chemotherapy for metastatic PTs and radiotherapy for malignant PTs with positive margins, especially when local control is desired and surgical treatment is not possible. Furthermore, radiotherapy may be useful in recurrent PTs, because it appears to be associated with lower rates of local recurrence ,8,17,20.4PT of breast is a rare neoplasm, posing great challenges to the surgeon. It presents high rates of local recurrence and possibility of metastases. Histopathological analysis is considered the definitive diagnostic method. Treatment is surgical, with wide excision, whereas radiotherapy and chemotherapy are controversial.None to declare.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.Not applicable. Since this is a case report, not a research study, ethical approval has been exempt/not required.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.Bruno Bisognin Garlet: Conceptualization, Methodology, Investigation, Writing – Original Draft, Writing – Review & Editing, Visualization, Supervision, Project Administration.Luciano Zogbi: Conceptualization, Methodology, Writing – Review & Editing, Visualization.Juliana Piveta de Lima: Writing – Review & Editing, Visualization.Paulo Pereira de Souza Favalli: Resources.Frederico Diefenthaeler Krahe: Resources.Not applicable.Bruno Bisognin Garlet.Not commissioned, externally peer-reviewed.


Primary peritonitis without an identifiable intra-abdominal source is extremely rare in healthy individuals; it is commonly seen in cases of nephrotic syndrome, cirrhosis and end-stage liver disease, ascites, immunosuppression, and inflamed peritoneum due to pre-existing autoimmune and oncological conditions.Streptococcus pneumoniae. A 30-day follow-up was performed and she was asymptomatic without any sign of infection.We present the case of a 68-year-old Caucasian woman operated on due to acute abdomen with a provisional diagnosis of acute appendicitis. During the operation a small amount of free intra-abdominal fluid was found. Her uterus, ovaries, and fallopian tubes were macroscopically normal. Therefore, with the suspicion of appendicitis, appendectomy was performed. Her blood cultures were negative while peritoneal fluid was positive for capsulated form of Streptococcus pneumoniae commonly causes upper respiratory tract infection and cutaneous infections. It very rarely causes gastrointestinal infection and it is very rarely responsible for primary peritonitis and septic shock syndrome.Pneumococcal peritonitis has a rare occurrence and represents a clinical challenge because of its subtle and non-specific clinical findings. The interest in our case lays in the relatively rare diagnosis of primary peritonitis mimicking acute appendicitis. Enterococcus species, are rarely involved [Primary peritonitis (PP) is a diffuse infective inflammation of the peritoneal cavity in the absence of a localized source –3. PP isinvolved –10 and, 3/uL white blood cells (WBC). C-reactive protein (CRP) value was 5 mg/dl . A computed tomography (CT) scan  mass spectrometry . An antibiotic susceptibility test was performed using E-test method and interpreted using European Committee on Antimicrobial Susceptibility Testing (EUCAST) guidelines 2017 [A 68-year-old Caucasian woman presented to our emergency department complaining of acute onset of severe abdominal pain in the right lower quadrant that began approximately 48 hours earlier; she had a temperature of 39.1 °C and heart rate of 98/minute. She denied any recent fever, chills, hemoptysis, hematochezia, or change in bowel habits. She had no history of trauma or surgery; she did not take any regular medication; she did not use an intrauterine device (IUD) or other local contraceptive. She had normal sex activity with the same partner . No relevant history of infection in her family was reported. On her presentation to our emergency room, a physical examination revealed a localized peritonism in the right lower quadrant. At rectal examination, a normal sphincter tone was found with no palpable masses and normal stool. Other features were unremarkable. Laboratory values on admission showed an hemoglobin of 13.3 g/dL, 36.4% hematocrit, with 19.00 × 10nes 2017 . The orgS. pneumoniae is the most common cause of community-acquired pneumonia and the second most common cause of purulent meningitis, while intra-abdominal pneumococcal infection is rarely found [ly found –18.S. pneumoniae in the worldwide literature up to 9 September 2017. We analyzed all cases reported in the literature [et al. [We conducted a review of the literature by searching the PubMed database for all published series and case reports of PP due to terature –60. Pediterature –21, secoterature , and casterature –25 were terature , 56–60.  [et al. , that pr [et al. –64. With [et al. , 54. The [et al. . Hemsley [et al.  reportedOf interest, a peculiarity of this condition is the absence of mixed organisms. In fact, no case reported an association of multiple infective agents.There is no definite clinical pattern or features which might help in the diagnosis. Usually the clinical picture closely resembles that of appendicitis or secondary peritonitis with or without sepsis which, in most cases, is the presumptive diagnosis –60.Management of this condition is strictly linked to the diagnosis. While PP can be suspected in patients with comorbidities in the presence of a negative radiologic investigation, it is hardly recognizable in young and immunocompetent patients with no risk factors, like in our case. This confirms the fact that, based on an erroneous diagnosis, most of these patients are operated on despite negative results from imaging.S. pneumoniae have been identified worldwide, and the prevalence of these resistant strains is as high as 57% in some countries [non-responders and in patients with sepsis, exploratory laparotomy is a well-accepted treatment [The best diagnostic algorithm is, in our opinion, Westwood and Roberts’ . Antibioountries , 64. In reatment , 66. Thereatment . It remareatment . Laparotreatment , 70. BasPrimary pneumococcal peritonitis without pre-existing peritoneal disease is rare and represents a cultural and clinical challenge, especially for surgeons, because of its subtle and non-specific clinical findings. The interest in our case lays in the rare diagnosis of PP mimicking acute appendicitis in a healthy woman, without history of recent acute pneumonia or pelvic inflammatory disease (PID) and the isolation of pneumococcus in its capsulated form. This case report reinforces the need for an appropriate clinical algorithm in those patients without medical history and active comorbidities when the diagnosis of acute appendicitis is not clear at the time of operation, especially for female patients. No direct diagnosis is possible. The treatment of choice is the fast initiation of antibiotic therapy. Although surgical therapy is generally not required for the treatment of primary pneumococcal peritonitis, it may be necessary to exclude secondary peritonitis or in non-responders.


Schwannoma is usually benign, encapsulated spindle cell tumor which arises from schwann cells of nerve sheath, and is the most common of the neurogenic mediastinal tumors. Various imaging modalities can be applied to assess posterior mediastinal mass which is often found incidentally without symptom and frequently misdiagnosed for other benign conditions both clinically and radiologically in which clinicians should be aware of. Schwannoma is usually benign, encapsulated spindle cell tumor which arises from schwann cells of nerve sheath, and is the most common of the neurogenic mediastinal tumors. Various imaging modalities can be applied to assess posterior mediastinal mass which is often found incidentally without symptom and frequently misdiagnosed for other benign conditions both clinically and radiologically in which clinicians should be aware of. A large proportion of them are benign. Schwannomas are the most common type of neurogenic tumor observed in thorax which originated from the peripheral nerve sheath of Schwann cells and often located in the posterior mediastinum.2A 60‐year‐old woman with cough was referred to our hospital for general check‐up. The patient had no pathologic symptom and underlying disease. Chest X‐ray showed an incidental mediastinal mass in posterior cardia area Figure A. Transt3Schwannoma is the most common benign neurogenic tumor that originated from the peripheral nerve sheath of Schwann cells. Intrathoracic schwannoma is most frequently located in the posterior mediastinum.None declared.YJS: involved in literature search and the writing of the manuscript. SHS: investigated and supervised the overall work. SK, DKK, KHK, and DIK: discussed the results and advised about the case. Do‐Kyun K: performed the operation and provided surgical information. JYK: provided pathological information and images.


Brown tumor is a non-neoplastic fibro-cystic expansile bone lesion caused by parathyroid hormone excess. It has been commonly described in patients with primary hyperparathyroidism and secondary hyperparathyroidism due to chronic kidney disease. However, it is very rare to encounter a brown tumor in the setting of nutritional vitamin D deficiency. We describe the case of a 16-year-old girl who presented with brown tumor-like lytic lesion of femur caused by severe longstanding vitamin D deficiency. Treatment with elemental calcium and cholecalciferol resulted in correction of hyperparathyroid state, with the resultant disappearance of the bony lesion and remarkable symptomatic improvement. Unnecessary orthopaedic intervention may be avoided using a high index of suspicion and performing targeted investigations in such cases. Brown tumor, also called as osteitis fibrosa cystica is an area of localized bone resorption due to parathyroid hormone excess. Histologically, the lesion resembles a giant cell tumor and diagnosis can be missed if blood levels of calcium and parathyroid hormone are not measured. Brown tumors are regarded as a characteristic bony manifestation of primary hyperparathyroidism, however, they have also been described in hyperparathyroidism secondary to chronic kidney disease. Despite the high prevalence of vitamin D deficiency (VDD), there are only a few cases describing brown tumor secondary to hyperparathyroidism complicating longstanding VDD -5.A 16-year-old girl presented to us with complaints of progressively increasing pain in left thigh for two years. At the time of presentation, the pain was moderately severe in intensity, requiring analgesics on a regular basis. It used to worsen on ambulation, resulting in limitation of her activities of daily living. There was no history of any local swelling or redness. She denied history of local trauma preceding the onset of pain. There was no history to suggest malabsorption or use of anticonvulsant drugs or indigenous medications. She was born out of a non-consanguineous marriage with normal birth and developmental history, and none of the family members had history of bone disease. Nutritional history was notable in the form of only occasional intake of milk and dairy products. The family used to live in an overcrowded basement of a two-storeyed building where exposure to sunlight was inadequate. Besides, patient preferred to remain indoors, moving out briefly only during early morning and evening hours in her premorbid state. For these complaints, she was evaluated in an outside hospital and diagnosed to have an aneurysmal bone cyst of the left femur. She was advised to undergo surgical intervention for the same; however, the anxious family brought her to our center for a second opinion.Examination revealed a young, lean female with the height of 163 cm, a weight of 40 kg and BMI of 15 kg/m2. She had proximal myopathy involving bilateral lower limbs and walked with an antalgic gait with waddling towards the left side. There were no evident deformities involving the long bones or spine. Rest general and systemic examination were unremarkable.Laboratory investigations of the patient have been summarised in Table Total calcium was 7.9 mg/dl , inorganic phosphorous 2.8 mg/dl , alkaline phosphatase 1324 IU , blood urea, creatinine, total protein, albumin, and liver function tests were normal. Tests for complete blood counts, fasting plasma glucose and urine routine examination were unremarkable. Serum intact parathyroid hormone was 840 pg/ml , 25(OH)D3 <4 ng/ml , total T4 8.2 ug/dl , TSH 3.6 uU/ml . X-ray pelvis revealed an expansile lytic lesion involving the trochanteric region of left femur, pseudofractures of bilateral femoral neck along with the widened joint space and irregular margins at the pubic symphysis  60,000 IU sachet once a week for eight weeks followed by once a month maintenance therapy. She was subsequently followed up at three monthly intervals. At follow-up visits, the patient reported significant improvement in pain and, by six months, she had returned to her premorbid functional sate. Biochemical parameters Table  and radiAlthough tissue diagnosis was not available, the remarkable clinical and radiological improvement in association with correction of the hyperparathyroid state indicated that the lytic lesion was brown tumor related to parathormone excess.Brown tumors are unifocal or multifocal bone lesions, characterized by increased osteoclast activity and fibroblast proliferation, encountered in patients with uncontrolled hyperparathyroidism. The term brown tumor is actually a misnomer, since these lesions are non-neoplastic. Histologically, the lesion is similar to a giant cell tumor, comprising of multinucleated giant cells in a background of spindle cell proliferation with large amounts of hemosiderin which imparts brown colour to the lesion. Although these lesions can occur at any skeletal site, the most common sites are the pelvis, ribs, long bone, clavicle, and mandible -7. RadioBrown tumors have been described at a frequency of 3%-4% in primary hyperparathyroidism and 1%-2% in secondary hyperparathyroidism in most series . Brown tIn secondary hyperparathyroidism, the lesions have been described predominantly in the setting of end-stage renal disease with poorly controlled hyperparathyroid state -20. AmonDue to the reversible nature, excision of brown tumor is not recommended unless the lesion causes compressive symptoms, significant cosmetic disfigurement or fails to regress on follow up. In our patient, the lesion healed completely with correction of secondary hyperparathyroidism using calcium and vitamin D supplementation. Healing of the lesion concurrent with improvement of metabolic parameters confirmed the diagnosis of brown tumor in this case. To the best of our knowledge, this is the first case reporting brown tumor-like lytic lesion in a long bone due to secondary hyperparathyroidism complicating longstanding VDD.We have described the case of a teenage girl, who presented with an expansile lytic lesion in the left femur, which was initially misdiagnosed as an aneurysmal bone cyst. Basic investigative work-up revealed nutritional VDD with secondary hyperparathyroidism, the treatment of which resulted in complete disappearance of the lesion. A basic metabolic work-up should be performed in all such cases and a therapeutic trial of calcium and vitamin D, followed by reassessment at 2-3 months may be done in indeterminate cases. Suspecting and investigating for secondary hyperparathyroidism is extremely important since appropriate treatment of the underlying metabolic state may prevent an unnecessary orthopedic intervention.