Prognosis is excellent; however, appropriate follow-up is an important part of the long-term management of this disease in order to prevent and control the possibility of local recurrence.Amyloidosis is a group of idiopathic clinical syndromes caused by the deposition of insoluble fibrillar proteins (amyloid) in the extracellular matrix of organs and tissues. These deposits disrupt the function of the target organ. Amyloidosis can manifest as a systemic disease or a single-organ involvement . Its etiology still remains unclear. Deposits of amyloid in the larynx are rare, accounting for between 0.2 and 1.2% of benign tumors of the larynx. In this retrospective study, we report the clinical aspects, diagnosis, treatment and follow-up of five female patients with localized laryngeal amyloidosis without systemic involvement. The patients were all treated successfully using microlaryngoscopy with CO Fibrils may deposit locally or may involve every organ systemically; the current classification distinguishes a systemic form and a localized one, in which the deposits affect a single organ without a systemic disease .Until today, at least 27 types of amyloidoses have been identified, based on the different proteins that form in vivo amyloid fibrils. According to this classification, a lettering system describes different forms: the amyloid protein is designated with the capital letter A, followed by a suffix that specifies the nature of the protein substance or the precursor protein name. For example, the abbreviation AL designates amyloidosis derived by the light chains of immunoglobulins ,4.The systemic variant generally implies a poor prognosis with a clinical picture that evolves more or less rapidly toward exitus. On the other hand, localized amyloidosis is a rare condition that can be observed in every organ and presents a favorable evolution in most cases .The first case of laryngeal amyloidosis (LA) reported in the literature was published by Burow and Neumann in 1875 and referred to as an occasional autopsy finding . The larA greater incidence in men has been reported, with a 3 : 1 male–female ratio, especially between the fifth and the sixth decade of life .The symptomatology of LA is nonspecific and is characterized in most cases by hoarseness, dysphonia, foreign body sensation, dysphagia or dyspnea.The “gold standard” for the diagnosis of amyloidosis is based upon histological examination. Histology of the specimen using routine hematoxylin and eosin staining shows amyloid as the eosinophilic extracellular infiltrate, sparing the overlying epithelium. Further staining with Congo red shows a characteristic apple green birefringence and dichroism under a polarizing microscope. Staining with methyl violet discloses metachromatic pink-violet staining.Regarding clinical objectivity, LA generally results in one or more areas of swelling, located in the submucosa, covered by apparently healthy mucosa. This clinical aspect, so common in laryngeal infections, leads to important problems of differential diagnosis between benign diseases  and malignant ones  ,6.The treatments available depend on the site and size, as well as on the general condition, age and personal preference of the patient; before starting any therapy, it is essential to have an unequivocal documentation of the type of amyloidosis .The low prevalence of this pathology poses in the first place many diagnostic difficulties, and there is still no clear unequivocal treatment and management modality.This study aims to report the surgical experiences related to our cases, discuss the indications of the techniques used and summarize our report in comparison to others in the literature.2Five patients with LA were treated and followed in the Otorhinolaryngology Unit of the “Federico II” University of Naples from 1982 to 2018 .The five subjects studied are all female, in the age range between 14 and 47 years.All patients underwent radiographic examination (CT in the first four cases and MRI in the fifth case) to define the localization and extension of laryngeal lesions.In collaboration with the pathologists and hematologists of our university, the simultaneous presence of systemic amyloidosis was excluded. The work-up involved blood tests, echocardiogram, evaluation of renal function, hepatic function and biopsy of periumbilical fat and bone marrow. The type of amyloid was defined by using the immunogold labeling technique using electron microscopy.The exeresis is performed under general anesthesia using direct laryngoscopy. We tried to implement the surgical excision en bloc with adjacent safe tissues. In the first 6 months, the patients underwent monthly laryngoscopic check-ups and surgical evaluation in order to remove fibrin deposits or avoid scar adhesions or to perform biopsies; subsequently, bimonthly endoscopic check-ups were performed for the remainder of the first year and for the second year, then six-monthly for 2 years and yearly from the fifth year .2.12 laser, this lesion was removed; the histological examination of the surgical specimen highlighted the presence of a circumscribed amyloidosis nucleus.A 14-year-old female patient was admitted to the hospital with dysphonia that had arisen about 1 year ago and gradually progressed. Laryngeal examination revealed the presence of a circumscribed mass, arising from the left false vocal cord. Using endoscopy, with the COAfter about a month, a microlaryngoscopy check was performed: some biopsies were taken, and the histological examination excluded the presence of new amyloidosis manifestations.There has been no evidence of recurrence during follow-up, with an optimal vocal function outcome.2.2A 21-year-old female patient was hospitalized for dysphonia with vocal fatigue that had arisen about 2 years ago, which had progressively worsened. Clinical objectivity demonstrated the presence of a mass involving the right false vocal cord and the adjacent aryepiglottic fold.2 laser, and the typical pattern of LA was identified histologically. Approximately 1 month after the operation, a mass growing in the right false vocal cord extending to the adjacent portion of the epiglottis was identified. With the appearance of dysphonia, a new endoscopic resection using the CO2 laser was performed and a well-circumscribed submucosal amyloid deposit, surrounded by granulation tissue, was documented by the pathologist.The lesions were removed using COAfter several years, the patient no longer showed signs of recurrence or functional damage.2.32 laser, the lesion was removed en bloc; the histological examination revealed the presence of submucosal deposits of amyloid, surrounded by healthy tissue.A 34-year-old female patient was admitted to the hospital with a 1-year history of dysphonia and stridor; laryngeal examination documented the presence of two “pads” covered with mucosa of normal appearance, located in the subglottic region and extending to the anterior commissure. Using endoscopy, with the COFibrin deposits were removed 1 month after first operation during an endoscopic check-up for dyspnea and no signs of recurrence were documented. An excellent functional result persists.2.42 laser, the lesions were removed. Histological examination highlighted the presence of extensive accumulations of amyloid substance even on the excision margins.A 47-year-old female patient was admitted to the hospital for dysphonia and dyspnea that had appeared about 2 years ago. With fiberoptic laryngoscopy, various submucosal tumefactions were observed, involving the supraglottic, glottic and subglottic regions, especially on the right. A biopsy was performed, and no tumor was found. Using endoscopy, with the CO2 laser treatment was performed in order to vaporize the edematous flap in the left arytenoid region and resect a synechia at the level of the anterior commissure 2 years after the first surgical procedures.New symptomatic and large amyloid lesions were removed three times from both the vocal folds and at the level of the epiglottic pedicle. Finally, endoscopic COThe patient remains with dysphonia and glottic insufficiency, but there was no evidence of recurrence.2.5A 38-year-old female patient sought our attention for the onset of insidious dysphonia for about 4 years. She underwent surgery of the vocal cords using direct microlaryngoscopy for dysphonia, in another institute; the histological examination showed the presence of an amyloid substance but was not supported by the systemic study.The fiberoptic laryngoscopy demonstrated a red subepithelial mass on the left false vocal cord extending to the laryngeal ventricle. The neoformation was removed using microlaryngoscopy by the cold steel technique and subjected to further histological examination, which confirmed the diagnosis of amyloidosis . SystemiAfter 4 months of the surgical operation, an endoscopic control showed an asymptomatic small formation in correspondence with the surgical excision. In order to identify its nature, this lesion was closely monitored. In the successive laryngeal control, this mass had highly reduced itself and then disappeared. To date, after 1 year of follow-up, no other signs of recurrence appeared, and the function of the vocal cord has been excellent .Ethical approval: This research related to human use complied with all the relevant national regulations and institutional policies and was conducted in accordance with the tenets of the Declaration of Helsinki and approved by the institutional review board or equivalent committees of the authors’ institutes.Informed consent: Informed consent was obtained from all individuals included in this study.3In the aforementioned five cases, there are different anatomical and clinical pictures with different surgical implications. All patients showed symptoms before undergoing surgical treatment. In cases 1, 2, 3 and 5, endoscopic treatment demonstrated its indisputable validity; in case 4, this surgical approach led to problems and needed further interventions. This depends on the extent of the amyloidosis lesions; external conservative interventions, on the other hand, would not have been less complex or invasive. In case 5, the use of the cold technique is related to the small extension: it allowed a more rapid patient outcome, short hospitalization time and the possibility of controls in the absence of postoperative fibrin.Amyloid lesions generally have indistinct margins that are not easy to identify during surgery, and therefore the need for surgical revisions cannot be excluded a priori. Narrow-band imaging endoscopy (NBI) procedure, tested in the last case, cannot help the surgical choice. Currently, the cases have all perfectly recovered from the pathological process and present excellent functional results.4The clinical and surgical experiences reported in the literature on LA consist of case reports and small case series. To date, several treatment options have been reported.In our cases, there is a clear prevalence among the female sex. This epidemiological observation, verified by appropriate methods, would perhaps open a new path in the discovery of the etiopathogenesis of the pathology, which is still unknown.Regarding the site, according to the literature, in our cases the supraglottic areas are the most frequently affected ones, in particular the false vocal cords  ,7,8.T1-weighted signal intensity and low T2-weighted signal intensity. This is an important point of differentiation because the muscle is a simple reference and tumors do not appear in this way [Imaging techniques such as computed tomography and magnetic resonance play a fundamental role in determining the extension of the lesion or characteristic infiltration signs. MRI could play a superior role for this purpose. The characteristic signal of amyloidosis reveals an intensity equal to that of the skeletal muscles: intermediate this way .The only treatment for symptomatic LA is surgical. The treatment goal is to offer the patient a long disease-free interval with functional larynx preservation. There are conflicting views on the type, modality and timing of surgery .The goal of localized LA treatment is to offer to the patient as long a disease-free interval as possible with voice preservation and normal breathing.Some authors support the need for an external approach to completely dominate the lesion and promote its radical excision; others are instead convinced advocates of endoscopic surgery ,10,11,122 laser or cold instruments). In our experience, the technique choice depends on the location and extent of the lesions. The use of endoscopic surgery is demonstrated herein, underlining the possibility of obtaining a definitive clinical recovery in the five cases studied. Benefit of this surgery are radical excision of the tumor with a reduced trauma, excellent postoperative functionality results, short hospitalization and low socio-health costs.There are multiple endoscopic options (suspended microlaryngoscopy and tumor removal with CORegarding follow-up, the literature is still unclear; therefore, the need for careful and regular follow-up is emphasized .In fact, although the prognosis of LA is generally good, the probability of recurrent or residual disease is significant and death due to respiratory failure has been reported .In our opinion, this probably depends on an imprecise clinical evaluation of the peripheral margins of the lesions . UnfortuFor our patients, we performed monthly check-ups for the first 6 months, bimonthly until the end of the second year, then six-monthly for two more years and yearly from the fifth year onwards .Therefore, a laryngological follow-up period of at least 10 years after the last surgery is recommended: a later progression remains possible.52 laser or cold steel technique.LA is a rare disease and remains a not completely understood pathology. LA is prevalent in the male sex and affects the supraglottic regions, in particular the false vocal cords and the ventricle. Concerning epidemiology, other studies may confirm our data: probably, systemic amyloidosis is a prerogative of the male sex, whereas the laryngeal form is more frequent in the female sex. The first-choice treatment is surgery; in our opinion, the main aim is to guarantee organ functionality: the options are local endoscopic procedures such as COA fundamental part of the care pathway should be an adequate follow-up. We recommend monthly endoscopic check-ups for the first 6 months, then bimonthly until the end of the second year, then six-monthly for 2 years and yearly from the fifth year. Long-term follow-up should always be performed.


A 60-year-old female with diabetes presented to the department of internal medicine with a 1-month history of headache. The patient had an Eastern Cooperative Oncology Group performance score of 1. Moderate tenderness was observed by soft palpation of the skull. Laboratory investigations revealed an elevated level of alkaline phosphatase (604 U/L) which was far above normal range (100-350 U/L). The serum level of hemoglobin, creatinine and calcium were within normal range. Plain radiography and computed tomography (CT) showed many punched-out osteolytic lesions in the skull . CranialPunched-out lesions are osteolytic lesions without sclerotic rim on X-ray examination which are considered as hallmarks of multiple myeloma. Punched-out lesions are created by the absence of reactive bone formation as a consequence of tumor factors that combine to activate osteoclasts and inhibit osteoblasts . TherefoCholangiocarcinoma is a heterogeneous group of tumor derived from bile ducts cells, and represents the second most frequent malignant liver cancer. Incidence is globally increasing with a rate of 2.1/100,000 person-years in Western countries. Patients with cholangiocarcinoma are known to commonly present locally advanced diseases .


Intussusception in adult patients is uncommon and appendiceal lead points are particularly rare.We present the case of a 42-year-old male with a history of ulcerative colitis, presenting with sudden onset abdominal pain and bloody diarrhoea. Endoscopy revealed grossly normal mucosa in the descending colon with a congested polypoid mass in the proximal transverse colon. Computed tomography revealed ileocecal intussusception at the hepatic flexure. A right hemicolectomy was performed, where a grossly dilated appendix was noted, resected and sent for histopathological evaluation. Results revealed low-grade appendiceal mucinous neoplasm. Post-operatively, the patient remained symptom free, however required reintroduction of biologic therapy due to relapse of his ulcerative colitis 12 weeks later.This case depicts a rare acute surgical presentation and reminds physicians and surgeons of the importance of ‘thinking outside the box’ in clinical practice. Ulcerative colitis (UC) is the most common form of inflammatory bowel disease (IBD), with an incidence of approximately 2 million in European populations . UC is a9/L and neutrophilia of 18.3 × 109/L. C-reactive protein levels were 46 mg/L and haemoglobin was 14.8 g/gL. A colonoscopy was performed showing proctitis with spontaneous bleeding involving the distal 3–4 cm of the rectum. On visualisation of the left colon, the mucosa appeared normal. Upon inspecting the proximal transverse colon, a globular intraluminal mass was present (Fig. A 42-year-old male presented to the emergency department with crampy abdominal pain and bloody diarrhoea lasting 2 days, on a background of an 18-year history of UC. He reported multiple episodes of loose stools and fresh bleeding per rectum with one episode of vomiting. Regular medications included oral mesalazine 3 g once daily since diagnosis and subcutaneous adalimumab 40 mg bi-monthly for the 6 months prior. He initially attributed his symptoms to a ‘flare’ of UC, as a consequence of increased alcohol consumption of 70 units per week for the preceding 3 weeks. He did however note that this episode of abdominal pain was the most severe experienced to date. On physical examination, he was apyrexic, and haemodynamically stable. His abdomen was soft and non-tender. Bowel sounds were auscultated and normal. Laboratory findings were significant for leucocytosis of 20.5 × 10This clinical case illustrates complex interwoven medical, oncological and surgical pathological processes propelling this patient’s emergency admission. This patient’s extensive history and bona fide diagnosis of UC raised suspicion for an exacerbation of his disease or perhaps a benign or malignant neoplasm, however this presentation was due to a rare form of intussusception secondary to appendiceal LAMN. This highlights the clinical importance of ‘thinking outside the box’ when such patients present to the emergency room with a short history of severe abdominal symptoms.Intussusception is an exceptionally rare finding in adult populations, typically mimicking other abdominal pathologies . In adulPASSION study, Sahami et al. postulate the appendix as a defective barrier which may alter the colonic microbiome, allowing aberrant interaction between bacteria penetrating the mucosa with innate immune cells. This potentiates abnormal immunological responses, which is thought to drive the pathophysiology of UC [Initially, our patient had a favourable clinical response to biological therapy prior to relapsing 12 weeks post-discharge from hospital. Recent hypotheses suggest that appendiceal tissue plays a role in the inflammatory and pathological processes driving symptomatic UC. In introducing the gy of UC . Rachmilgy of UC , 15. TheIntussusception is a particularly rare phenomenon in adult populations, with incidental LAMN as a clinical lead point even rarer. This clinical conundrum highlights how a number of rare clinical pearls may be amalgamated into an acute surgical presentation and the requirement to ‘think outside the box’ in clinical practice


Liposarcoma is a malignant adipose tissue tumor which mainly originates from the extremities and retroperitoneum. Primary pleural liposarcoma is very rare. Spindle cell lipoma is a rare benign adipose tissue tumor. A 66‐year‐old male was referred to our hospital for the evaluation of a mass‐like opacity visible on chest X‐ray. Computed tomography (CT) scan revealed a well‐defined soft tissue mass with internal low attenuations and adjacent multiple nodules in the upper lobe of the left lung, and surgical excision was subsequently performed. Histopathological findings revealed adipose tissue with lipoblasts and spindle cells and immunohistochemical staining (IHC) revealed the tumor cells were strongly positive for CDK4 and MDM2. Histopathological examination of the small lung nodules showed spindle cell proliferation and adipose tissue without positivity for MDM2. Here, we report a rare case of primary pleural liposarcoma combined adjacent spindle cell lipoma of the lung. Liposarcomas are soft tissue sarcomas that usually present in the extremities and retroperitoneum. Primary pleural liposarcoma is extremely rare and more common in males.A 66‐year‐old male was referred to our hospital for further evaluation of a lung mass, which was incidentally found on CT scan. He was an ex‐smoker and had no previous medical history. Physical examination showed no abnormalities in the chest and revealed stable vital signs. Chest X‐ray showed a mass‐like opacity in the lower zone of the left lung Fig . CT scanLiposarcoma accounts for about 20% of all mesenchymal tumors, is the most common soft tissue sarcoma in adults and is usually found in the retroperitoneum and the extremities.Primary pleural liposarcoma is extremely rare and the exact incidence is unknown. It is known to be more common in males and is primarily a disease of individuals of 50 years of age.MDM2, CDK4, and HMGA2.Liposarcomas originate from the primitive mesenchymal tissue residues rather than mature fat tissue. While the pleural cavity is formed from mesoderm, mesenchymal cells that are left in this process go through malignant transformation and bring about pleural liposarcoma.Spindle cell lipoma, which is composed of mature adipose tissue, collagen, and spindle cells is a rare benign tumor and commonly found in middle‐aged and older males.Intrathoracic spindle cell lipoma is usually diagnosed by histological examination. Microscopic appearance of spindle cell lipoma is characterized by the classic triad of mature adipocytes, ropey collagen bundles, and bland spindle cells. Spindle cell lipoma is strongly CD34 positive on immunohistochemical staining and the spindle cells may stain for S‐100.In conclusion, here we report a rare case of primary pleural liposarcoma combined spindle cell lipoma of the lung. This report might be helpful for the diagnosis of rare intrathoracic lipomatous tumors in the future.The authors declare that there are no conflicts of interest.


Supernumerary teeth are frequently reported in dental clinical practice; however, eruption in nasal cavities and paranasal sinuses is an extremely rare clinical entity.st, 1886 to December 31st, 2017. We report two cases with a history of recurrent nasal discharge and obstruction. In both cases, clinical and radiological findings confirmed the presence of an inverted supernumerary tooth erupted in the sinonasal cavities . We managed the cases with transnasal endoscopic approach. A survey of the English literature identified 69 documented cases with intranasal supernumerary teeth within January 1Inverted supernumerary teeth should be considered among the potential causes of unilateral nasal obstruction and rhinosinusitis and included in differential diagnoses among the fibro-osseous lesions of the sinonasal cavities. Although supernumerary teeth are frequently reported in dental clinical practice, eruption in sinonasal cavities is a rare clinical entity, occurring in only 0.1-1% of the general population  showed the presence of a calcified mass in contact with the floor of the right nasal fossa, approximately 1.3 × 0.5 cm in size . The masCase 2A 28-year-old lady was referred to the Department of Otorhinolaryngology of the Humanitas Clinical and Research Center with a clinical history of recurrent left nasal obstruction, discharge, cacosmia, and pain in the left maxillary region. The patient was frequently treated with the administration of systemic antibiotics and corticosteroids at other institutions. The patient did not report any clinical history of facial trauma or surgery of cleft lip or palate. Clinical examination with a 30° rigid endoscope identified polypoid tissue and purulent discharge coming from the left middle meatus. Noncontrast-enhanced CT showed the presence of an inverted tooth erupted in the left maxillary sinus  concomitThe macroscopic analysis showed a cuboidal dental element, and the diagnosis of the inverted supernumerary tooth was confirmed by histologic examination. The patient was discharged the day after the surgery. She reported complete relief of the symptoms during postoperative care and showed complete healing of the surgical field 2 months after the surgery.Supernumerary teeth are described as dental elements in excess of the normal formula. The prevalence of supernumerary teeth in the Caucasian population is within the range of 1-3% . If we oMore than 90% of supernumerary teeth occur between maxillary central incisors, and in that situation, they are called mesiodens ,5. Howev The first case reported in the present study was an inverted mesiodens. The second patient presented a rarer situation; according to the site, the tooth was an inverted supernumerary molar. The etiology remains unclear, especially in the absence of a clinical history of surgery, trauma, Gardner’s syndrome, or anatomic malformations, such as cleft lip and palate. Three theories have been postulated about this developmental glitch. The first and earliest theory of phylogenic reversion by Von proposed that supernumerary teeth could be the phylogenic appearances of our extinct ancestors since they possessed three maxillary incisors; however , this thThe second theory suggests that there could be a split in the tooth bud to create two different teeth . The thiFew authors have speculated that supernumerary teeth might originate from a third tooth bed arising from the dental lamina near the permanent teeth ,16. Howest, 1886 to December 31st, 2017. A survey of the English literature allowed the identification of 2,717 articles with the search term “supernumerary tooth” in PubMed  and EMBASE . A further selection of the search term “inverted supernumerary tooth” reduced the search to 59 papers. The final selection included 69 documented cases of an inverted intranasal tooth, 71 cases with the consideration of the 2 present cases, within January 1th century, There have been reported 71 cases, includind our series; only 4 cases dated back to before 1958. th and beginning of the 20th century (According to Kuroda et al. , Albins  century . Marshal century . Smith e century . In 1996 century ,20. Most century , 21-23.In normal dental clinical practice, a supernumerary tooth can be diagnosed as a cause of the misalignment of a tooth, dilacerations of a permanent tooth, and cyst formation, such as a dentigerous cyst. However, in the rare occurrence of an eruption in the sinonasal cavities, the prevailing symptoms often lead the patient to refer to an ear, nose, and throat department. The most common complains are nasal discharge, unilateral nasal obstruction, nasal bleeding, and facial pain ,17,23-25Clinically, both the cases of the present study showed unilateral nasal obstruction and persistent fetid rhinorrhea. Rare complications, including the abscess of the nasal septum, deformity of the nasal pyramid, oral-nasal fistula, and nasal septal perforation, were also reported ,9. The dWith the improvement of endoscopic surgery and refinement of functional techniques, the nasal pathway has become the natural way of a tooth extraction with prevalent sinonasal growth. In endoscopic surgery, it is possible to have a clear visualization of the insertion, avoidance of injury to surrounding mucosa, and precise dissection with the preservation of surrounding structures . FinallyInverted supernumerary teeth are rare clinical entities; however, they should be considered among the potential causes of unilateral rhinosinusitis and included in the differential diagnoses among the fibro-osseous lesions of the sinonasal cavities. In this regard, the endoscopic approach is nowadays the treatment of choice.


Primary female genital tract lymphomas are sporadic neoplasms, accounting for 0.2% to 1.1% of all cases of extranodal lymphoma. The most common genital localizations are the cervix, the uterine corpus and the ovary, while primary lymphomas of the fallopian tube are quite unusual. According to literature searching in PubMed, this is the first reported case of primary diffuse large B-cell lymphoma of the fallopian tube.A 52-year-old woman presented with a more than 2 months history of intermittent lower abdominal pain. The gynecological examination showed that the uterus, as big as 3 months of pregnancy, had weak activity and no tenderness. The uterine rectum lacuna was like a hard nodule of about 3 × 2 cm, and an irregular solid mass was fixed and inactive in the right adnexa.In accordance with Ann Arbor staging system, a stage IE primary diffuse large B-cell lymphoma of fallopian tube was diagnosed for this patient, based on the tumor pathology, the results of bone marrow biopsy and computed tomography (CT) scan.After gynecological/urinary ultrasound, blood test, pelvic contrast enhanced CT scan and CT angiography of iliac artery, exploratory laparoscopy and following hysterectomy with bilateral salpingo-oophorectomy were performed. After the surgery, the patient was treated with combined Rituximab and chemotherapy and got complete response (CR).After the operation and R-CHPOP, following up for more than 1 year so far, the patient has no tumor recurrence and is still in good condition.It is very difficult to diagnose the primary diffuse large B-cell lymphoma of fallopian tube, not only because of its rarity, but also because of its non-specific clinical manifestations. It easily be treated as late ovarian cancer by gynecologist. So the pathology diagnosis and surgeons’ decision is very important. Because lymphoma is pretty sensitive to chemotherapy and easy to get complete response, so we no need to do an operation like ovarian cancer and should put chemotherapy as a primary method for lymphomas of the female genital tract. The most common genital localizations are the cervix, the uterine corpus, the ovary and the vagina/vulva.,3 The primary lymphomas of the fallopian tube are quite unusual. According to literature searching in PubMed, we only found case reports about follicular lymphoma, T-cell lymphoma and marginal zone B-cell lymphoma happened in the fallopian tube, but no DLBCL reported. In this article, we present a case with a primary extranodal DLBCL of the fallopian tube in a middle-aged woman, who was diagnosed based on histopathological and immunohistochemical findings. This case was first treated at the Department of Gynecology, Affiliated Hospital of Jining Medical University. The patient provided signed informed consent, and Institutional Review Board approval was obtained (No.2019C002).Primary gynecologic tract lymphoma accounts for 0.2% to 1.1% of all cases of extra-nodal lymphoma.2A 52-year-old woman from China presented with a more than 2 months history of intermittent lower abdominal pain. She had no abdominal distension, fever or wasting, but accompanying with swelling in the right lower limb . The skin temperature of the right lower extremity is higher than that of the left. The gynecological examination showed that the vulva, vagina, and cervix was regular, while the uterus, as big as 3 months of pregnancy, had poor activity and no tenderness. The uterine rectum lacuna was like a hard nodule of about 3 × 2 cm. At the same time, Pelvic examination revealed an irregular solid mass that was fixed and inactive of the right adnexa. No abnormal signs were found in the left adnexa. There was no swelling of superficial lymph nodes. No obvious abnormality was found in the blood routine, blood coagulation routine and liver function. The patient's previous medical history, clinical examination and other laboratory tests did not indicate any related autoimmune/immunocompromising disease or any other serious illness. Tumor markers: CA125 99 U/ml, HE4 110.7 pmol/L, CEA 1.84 ng/ml, AFP 3.95 ng/ml, CA199 <2.00 U/ML. Gynecological ultrasound examination showed that the right adnexal area had a cystic solid mass, about 4.9 cm × 3.9 cm × 3.7 cm in size, with clear boundary, poor sound transmission of cystic part, uneven echo of solid part, and accessible blood flow signal. Urinary ultrasound: the upper segment of the right ureter was dilated, about 1.0 cm width in a relatively wider place, and the hypoechoic mass was found at the iliac blood vessel, about 8.2 cm × 7.2 cm × 7.1 cm in size, with unclear boundary, irregular shape, uneven internal echo, and abundant arterial blood flow signals could be detected. Plain scan and enhanced scan of pelvic spiral computed tomography (CT): soft tissue mass is seen in the right adnexal area, about 8.2 cm × 6.5 cm × 5.0 cm in size  is a very rare entity, comprising 0.2–1.1% of all cases of extra-nodal lymphoma. PLFGT most commonly can arise from the ovary, uterus, cervix, vagina and vulva. A lot of cases analysis studies of PLFGT confirmed the above conclusion.,6–8 However, there was no report on the primary NHL of fallopian tube.Primary non-Hodgkin's lymphoma (NHL) is mainly derived from lymph nodes or lymphangiocytes of extranodal organs. 4 cases of primary follicular lymphoma of the fallopian tube,11 and one case of primary T-cell lymphoma of bilateral fallopian tubes.Primary lymphoma of the fallopian tube is extremely rare and most cases of fallopian tube involvement reflect more extensive disease arising in the ovary, with spread to the fallopian tube. Only rare cases of primary tubal lymphoma have been reported. However, all cases reported so far have not been DLBCL. These include 3 cases of primary extranodal marginal zone B-cell lymphoma (MALT-type lymphoma) of the fallopian tube, Once the NHL has been diagnosed, it must be staged, as staging has proved to be 1 of the most important factors to predict survival. Clinical examinations and other investigations including CT scan, full blood count, renal and liver function test, bone marrow aspirate and so on must be performed to set the stage of the tumor, following the Ann Arbor staging classification for extranodal lymphomas. In this case, peripheral blood, bone marrow puncture and outside pelvic CT scan showed no obvious abnormality, no history of lymphoma. Before operation, pelvic CT and gynecological ultrasound showed the mass of adnexa. After bilateral adnexectomy of the whole uterus, pathology showed diffuse large B-cell lymphoma of the right fallopian tube . The tumor foci invaded the myometrium and the wall of the fallopian tube; no tumor was found in bilateral ovarian tissues. Therefore, it is considered that the primary tumor is in the fallopian tube.Here, we describe a new case of primary DLBCL arising in the fallopian tube, clinically presented with intermittent hypogastralgia, pelvic mass and compression symptoms. As NHL is rarely seen in female genital system, it can be easily confused with any other common gynecologic malignancy. Therefore, it's very difficult to do an early diagnosis in most patients and patients are often diagnosed by results obtained from intraoperative or postoperative pathological evaluations.,15 Our patient was treated with a 6-cycles Rituximab and chemotherapy and achieved a complete response. Following up for 1 year, the patient is still in good condition.Based on this staging classification, the involvement of a single extra-lymphatic organ or site is defined as stage IE, as our patient. Due to the rarity of the disease, there is no standardized treatment plan at present. It is generally considered that this disease is a kind of malignant tumor sensitive to radiotherapy and chemotherapy. CHOP  chemotherapy combined with rituximab was used in the initial and recurrent treatment of aggressive NHL, which improved the overall survival rate of DLBCL.In conclusion, it is very difficult to diagnose the primary diffuse large B-cell lymphoma of fallopian tube, not only because of its rarity, but also because of its non-specific clinical manifestations. This report firstly introduces a new rare case of diffuse large B-cell lymphoma in the fallopian tube, presenting the whole diagnosis and treatment process of the disease, followed up closely and reported continuously.Conceptualization: Ye Zhou, Linqing Yang, Yunfei WangData curation: Ye Zhou, Chao Zhang, Yingying GongInvestigation: Chao Zhang, Yunfei WangMethodology: Ye Zhou, Yunfei WangProject administration: Yunfei WangSupervision: Yunfei WangWriting – original draft: Ye ZhouWriting – review & editing: Linqing Yang, Yunfei Wang


Inflammatory fibroid polyps (IFPs) are rare, clinically benign tumors that can present as an intussusception causing intestinal obstruction.We present a rare case of a 23-year-old man who suffered from intussusception caused by two IFPs. The patient complained of aggravated squeezing epigastric pain that prompted him to visit the emergency department. Moderate abdominal distention with mild tenderness was noted without signs of peritonitis. Abdominal computed tomography confirmed the presence of intussusception in the left lower quadrant. During laparoscopic surgery, the intussuscepted segment of the jejunum was identified, after which laparoscopic small bowel resection and anastomosis were performed. The patient was subsequently discharged without postoperative complications.IFPs are rare, clinically benign tumors of the gastrointestinal tract originating from the submucosa. IFPs can present as an intussusception causing intestinal obstruction.Surgical resection is the optimal treatment method for IFPs accompanied by intussusception. Intussu2A 23-year-old man visited the emergency department with a 3-day history of diffuse abdominal pain. The patient complained of aggravated squeezing epigastric pain every 3 min. Furthermore, he suffered from constipation, nausea, and vomiting for the past 2 days. The patient had no history of fever, hematochezia, and weight loss and had an unremarkable drug, family, or surgical history.On physical examination, vital signs, including blood pressure, heart rate, body temperature, respiratory rate, and oxygen saturation at room air, were normal. Moderate abdominal distention with mild tenderness was noted without signs of peritonitis. The patient did not exhibit guarding or rebound tenderness. An erect abdominal X-ray showed a stepladder sign, suggesting mechanical obstruction. Laboratory examination results, including blood chemistry, routine blood tests, and tumor markers, were normal.The patient was first admitted to the internal medicine division of gastroenterology. A nasogastric tube was then immediately inserted for gastrointestinal decompression, through which more than 500 mL of a greenish, foul-smelling fluid was drained per day. Two days later, abdominal computed tomography (CT) confirmed the presence of intussusception in the left lower quadrant . No massMacroscopic findings of the resected specimen revealed two intraluminal polyps measuring 5 × 3 × 3 cm and 2.5 × 2.3 × 1.2 cm that originated from the same location. The larger one had a pedunculated stalk, whereas the small one had a sessile stalk . Cross s3Intussusception was first reported by Barbette in 1674, while Jonathan Hutchinson performed the first surgical intervention in 1871 . This coIFPs are known by various names, including Vanek’s tumor, polypoid myoendothelioma, inflammatory pseudotumor, and eosinophilic granuloma . They arIFPs are usually found incidentally during endoscopy, imaging investigations, and surgery. Clinical aspects depend largely on the tumor size and location. Even if there is no obstruction or intussusception symptoms, IFPs become apparent with gastrointestinal bleeding caused by superficial mucosal erosion or ulceration . Though Radiological findings can be helpful in diagnosing IFP-related intussusception. Generally, abdominal X-ray is the first diagnostic tool used to detect bowel obstruction. Ultrasonography has also been considered an effective tool in the diagnosis of intussusception, with imaging features characterized by a target or ‘donut sign’. Ultrasonography has a sensitivity of 98%–100% and a specificity of 88%–89% . AbdominAlthough surgical resection has been the primary treatment method for IFPs, those that occur in stomach and duodenum can be removed through endoscopy. IFPs are usually benign and neither recur nor metastasize. However, given that establishing a diagnosis of IFP through preoperative imaging alone is difficult, the possibility of malignancy should be considered. In our case, we performed wide resection of the intussuscepted segment including the two IFPs. Given the possibility for malignancy in the gross appearance, extensive small bowel and mesentery resection with margins of at least 50 mm from the adjacent tissue should be performed. Regarding the technical aspect, this operation was performed using the laparoscopic technique without a first assistant. Currently, minimally invasive surgical approaches to abdominal surgery have been gradually replacing conventional open surgery considering the several advantages they offer, including reduced postoperative pain and better cosmetic outcomes . Given tTo the best of our knowledge, this case, in which two IFPs originated from the same location in the jejunum, is very rare. In summary, IFPs are rare, clinically benign tumors that can present as an intussusception causing intestinal obstruction. Surgical resection is the optimal treatment method for IFPs accompanied by intussusception since these lesions do not metastasize and have no recurrence rate.There are no potential conflicts of interest relevant to this article.There are no sources of funding relevant to this article.The institutional review board of Dankook University Hospital approved the protocol .Written informed consent was obtained from the patient.Concept: K.D.W.Data collection: P.K.B.Data analysis: J.Y.S.Writing papers: P.K.B, K.D.W.Supervision: K.D.W.None.Dong-Wook Kim is the guarantor.Not commissioned, externally peer-reviewed.


Metastatic tumors to the oral cavity are uncommon, representing approximately 1% of all cases of oral malignant lesions even when a metastatic disease is present. The 53-year-old female is presented complaining of abdominal pain, weight loss, and a loose stool recurring not more than three times per day. A computed tomography (CT) scan of the abdomen showed a retroperitoneal mass expanding along the body of the pancreas. Colonoscopy and gastroscopy with a gastric mucosa biopsy showed a normal result. After laparoscopic surgery, the primary site of adenocarcinoma was not confirmed. The patient was referred to the Maxillofacial Surgery Clinic with pain, swelling, and occasional bleeding around the lower right second mollar. Immunohistochemicaly, the tumor cells were positive for Cytokeratin (CK) 19, Cytokeratin (CK) 7, and homebox protein (CDX-2), which are highly sensitive markers of pancreatobiliar cancer. Therefore, the patient was diagnosed with pancreatic carcinoma. This report describes a rare metastasis of malignant pancreatic tumor to the lower right gingiva and highlights the importance of immunohistochemical examination and how it helped identify both the origin and the nature of gingival neoplasm. Metastatic tumors to the oral cavity are uncommon, representing approximately 1% of all cases of oral malignant lesions ,3. The pThe 53-year-old female presented complaining with abdominal pain, weight loss, and loose stool recurring not more than three times per day. The patient had been healthy, with no history of chronic illness. Ultrasound of the abdomen in 2017 showed a hyperechoic pancreas with hypodensity of the pancreatic body. Laboratory data showed normal CA-19-9 serum levels at 32.9 . Computed tomography (CT) scan of the abdomen showed a large calcified retroperitoneal mass medially to the left adrenal gland. For the purpose of taking a tissue specimen for pathohistological diagnostics, explorative laparotomy was done. The large tumor mass infiltrated the abdominal aorta, inferior vena cava, truncus celiacus, and the body of the pancreas. The biopsy confirmed adenocarcinoma, primarily of the colon . After sPostoperative PET/CT scan of the abdomen showed a retroperitoneal mass expanding along the body of the pancreas, which included the left adrenal gland and truncus celiacus. Continuing, the scan showed a soft tissue mass in right upper lobe of the lungs. Both masses showed an increased uptake of the FDG. Colonoscopy and gastroscopy with a gastric mucosa biopsy showed a normal result. Revision of the biopsy was performed after laparotomy was done, and it was immunohistochemically confirmed that the primary lesion is originating either from the colon or pancreas. The patient started treatment with a total of 10 cycles of FOLFOX  chemotherapy regimen and received the last dose in May of 2018. After the 6th cycle of FOLFOX regimen therapy, PET/CT scan showed an expansive mass retroperitoneally with mild morphological and metabolical regression but with a still active malignant disease. A nodal mass in the upper right lobe of the lungs also showed metabolic and morphological regression. However, tumor marker CA 19-9 serum level was elevated to 350.6 U/mL in August 2018. In that time, the patient complained of abdominal pain, weight loss, loss of appetite, and pain in the right part of the oral cavity. She was referred in September of 2018 to the Maxillofacial Surgery Clinic for further examination of the lesion and for an incision biopsy. She presented with pain, swelling, and occasional bleeding around the lower right second molar. Physical examination showed a 2 × 1.5 cm nodule, an unclear margin with soft texture, and a partial mucosal ulceration . There wTo best of our knowledge this is a rare report of a metastasis of pancreatic adenocarcinoma to the mandibular gingiva. In this case, oral metastasis was not the first indication of pancreatic carcinoma, but histological examination helped confirm the diagnosis. Metastatic tumors within the oral cavity are very uncommon. The head and neck region is usually not included in the staging scans for abdominal malignancies, so some micrometastases may pass undetected. Therefore, the incidence of metastatic diseases that affect the oral cavity is still unknown. Signs and symptoms which are commonly reported, as in our case, include pain in the affected area, bleeding, swelling, and discomfort ,8. MetasPancreatic neoplasm is an aggressive malignancy with 20–30% of potentially curable cases, which are localized when diagnosed. The overall 5-year survival rate of pancreatic neoplasms is approximately 5% . MetastaThe diagnosis of a metastasis in the oral cavity poses a considerable challenge and the practitioner needs to be careful of the risk these lesions present, while the pathologist has to determine the original tumor based on immunohistochemistry. This helps to determine and to increase the chances of a successful differential diagnosis. Various technologies such as the VELscope fluorescence method are used today to help diagnose primary and metastatic lesions of the oral cavity ,21. Meta


Case Presentation. A 52-year-old woman was consulted by a surgeon for a derivative felt in her abdomen. The patient said she felt a growing derivative in the abdomen about a month ago. After consulting and testing, a sigmoid colon mesenteric cyst (13 cm × 11 cm × 10 cm) was found. Complete excision of the cyst within healthy tissues was performed through laparotomy. The surgery had no complications. The initial pathological answer was a simple mesothelial cyst . However, immunohistochemical tests were performed that showed that diagnosis was mesenteric cystic lymphangioma (ML). Cystic lymphangiomas that have a link to the mesentery have been described less than 200 times.  Both mesenteric cysts and cystic lymphangiomas are scarce and clinically and radiologically almost identical derivatives, but their histological structure is fundamentally different.  Final differential diagnosis between different cystic derivatives is possible only based on histopathological examinations. Mesenteric lymphangioma is most common at a very young age, but in rare cases, it also occurs in adults. All clinicians should increase their awareness of the disease. Peritoneal cysts were first described as early as 1507 by the Italian anatomist Benevenni when a postmortem evaluation was performed. The first successful removal of the peritoneal cyst was completed in 1880 [Both mesenteric cysts and cystic lymphangiomas are very rare and clinically and radiologically almost identical derivatives, but their histological structure is fundamentally different .The main difference demonstrating the need for differential diagnosis lies in the fact that lymphangiomas, although usually benign, have a greater probability of becoming aggressive. They can become invasive and proliferate in the surrounding tissues and organs .A 52-year-old woman was consulted by a surgeon for a derivative felt in her abdomen. The patient said she felt a growing derivative in the abdomen about a month ago.There was also slight abdominal ache and discomfort while sitting and cycling.During the initial examination, deformation of the abdominal wall is observed and a painless derivative is felt in the lower left quadrant of the abdomen . ExternaUltrasound examination revealed a cystic derivative with clear boundaries.However, ultrasound is not the first-choice research for assessing the intestine and mesentery derivatives, due to difficulty in assessing a clear relationship with the surrounding tissues and structures.Therefore, CT scan was performed.A cystic derivative (13 cm × 11 cm × 10 cm in size) was found in the left lower abdominal quadrant according to the CT data. The derivative has no radiological evidence of malignancy. There were no signs of overgrowth into other structures, as well as no clear contact with large blood vessels. Radiologically, the derivative was most similar to a mesenteric cyst. Mild hydronephrosis due to pressure in the left ureter is also observed .The patient underwent laparotomy, during which a movable, thin-walled, cystic derivative (about 12 cm in size) with transparent content in a sigmoid colon mesentery was found. Complete excision of the cyst within healthy tissues was performed, protecting the surrounding blood vessels Figures  and 4.The surgery had no complications.The patient was discharged on the fourth postoperative day with an uneventful postoperative process. The patient underwent two ultrasound examinations during the first year of postoperative treatment, one after three months and the other after six months. Both examinations were free of visible pathological changes. One year after the operation, abdominal and pelvic computed tomography was performed; no visible pathology was found.The pathological findings were a thin-layered cyst with a focal fusion of the inner monolayer of the epithelium.The conclusion was that this was a simple mesothelial cyst.However, when the pathologist suspected that the diagnosis might be inaccurate, additional immunohistochemical tests were performed: derivative lining endothelial cells D2-40(+++) (cytoplasmic response), 100%; CD34(+)(cytoplasmic response), 10%; Calretinin(-); WT1(-); CD10(-); panCK(AE1/AE3)(-); CK7(-); and estrogen receptors(-) Figures  and 6.Immunohistochemical tests showed that the proper diagnosis was mesenteric lymphangioma (ML).The prevalence of simple mesothelial cyst cases in literature is scarce. The total number of reported cases in the world is less than a thousand. The cases described range in size from a few centimeters to giant cysts reaching 5 liters .The Perrot classification, which is used to classify cystic derivatives, indicates that the simple mesothelial cyst (SMC) is a benign mesenteric mesothelial cyst. Perrot and coauthors proposed to classify cysts according to histopathological features and origin into six groups: lymphatic, mesothelial, intestinal, urogenital, dermoid cysts, and nonpancreatic pseudocystic origins .It is considered that mesothelial cysts are caused by incomplete fusion of mesothelial layers .The common mesothelioma cyst is usually asymptomatic or manifests itself with very nonspecific clinical signs: slight abdominal ache, nausea, etc. The presence or absence of symptoms depends mainly on the location of the cyst in the abdomen. Symptoms are caused by “external pressure” . It is not possible to determine the threshold beyond which the cyst's size already causes symptoms, as this is highly dependent on the localization in which the cyst is located and the anatomical characteristics of the patient. As a result, some people may begin to experience cysts' symptoms while it is just a few inches in size, for example, when it forms near the urethra and pressures it. On the contrary, others may not feel any symptoms until the cyst reaches a large volume if it starts in the “free abdominal area.”Pain is not a very common symptom, and if it does occur, the first thing to think about is intestinal strangulation .Meanwhile, with regard to cystic lymphangiomas, it should be noted that the disease is so rare that in all of the English literature, cystic lymphangiomas that have a link to the mesentery have been described less than 200 times . It is tLymphangiomas can occur in any part of the body, most commonly found in the head and neck, and sometimes in the abdominal wall area. The discovery of lymphangiomas in the mesentery is an infrequent event . As a peLymphangiomas are classified into simple, cavernous, and cystic .The etiology of the disease is not known, but it is thought to be related to disorders of lymphatic system formation. Such an embryological theory is supported, at least in part, by the fact that the vast majority of cases are detected as early as childhood , 14. AboThere are small lymphoid aggregates in the cyst wall that help distinguish between lymphangiomas and simple cysts of the mesentery.However lymphangiomas might be difficult to distinguish from hemangiomas, and therefore, final diagnosis can only be confirmed immunohistochemically .If cystic derivatives in the abdomen are suspected, it is recommended that the first-line examination starts with an ultrasound of the abdominal organs. This examination is inexpensive, harmless, and gives good results for initial assessment (screening) . The onlThe disease may mimic the cavernous masses caused by tuberculosis, which is relevant in countries with a high incidence of tuberculosis  .Computed tomography or magnetic resonance imaging is recommended for a more detailed evaluation of the derivative before surgery, its interfaces with surrounding organs, and blood vessels .Although isolated studies are showing that active monitoring is an appropriate choice for asymptomatic patients , howeverUndoubtedly, the question may arise about the goal of immediate radical removal of a derivative, which is usually benign, especially considering that spontaneous regression cases of resembling derivatives can also be found in the literature . On the Suggestions are based on the fact that benign derivatives, though exceptionally rare, can change their degree of malignancy. This change might occur not only through the natural course of the disease (mesenteric lymphangioma is at risk of transforming into a sarcoma under the influence of ionizing radiation) but also due to the human factor in histological diagnosis (like in the case we describe) .It should also be noted that there have been described cases where even a simple mesenteric cyst has been reclassified to a malignant cyst with aggressive spread within ten years after six surgical interventions (to reduce the volume) .Laparoscopic methods for the removal of such cysts can be found in the literature . Still, In some cases, when surgical treatment is too risky, cyst drainage is described as a method of treatment. However, 1/3 of the patient's cyst recurred quite quickly, not to mention that the cells were not affected in any way, and not all of the complications of malignancy described above were avoided , 26.Drainage on its own is not a cure but rather a bridge to surgical treatment or symptom relief to reduce cyst volume.Other minimally invasive treatments of lymphangiomas include sclerotherapy with doxycycline or alcohol –29.It must be emphasized that these are isolated cases and not standard clinical practice. To our knowledge, no extensive studies are currently evaluating these methods. Therefore, we cannot predict whether it will be generally used.The final differential diagnosis between different cystic derivatives is possible only based on histopathological examinations.Mesenteric lymphangioma is most common at a very young age, but in rare cases, it also occurs in adults.Mesenteric lymphangioma is usually nonmalignant but can sometimes lead to very severe consequences.Timely and radical surgery can completely cure the disease and help prevent malignancy.Clinicians should increase their awareness of the disease.


Metastatic disease is one of the few rare causes which can present with clinical and radiological features of acute appendicitis. In this article, we present a case of a 33-years-old man with known primary gastric malignancy undergoing adjuvant treatment, who presented with clinical peritonitis. Imaging findings revealed acute appendicitis and a sealed-off appendiceal perforation. Diagnosis of metastatic adenocarcinoma to the appendix was confirmed on histopathology. Sound knowledge among clinicians and radiologists regarding clinical presentation and radiological findings of acute abdomen in patients with known primary malignancy can aid in rapid diagnosis and management. Metastatic disease to the appendix is a rare entity, the first presentation of which is usually acute appendicitis. Contrast-enhanced computed tomography (CT) is the modality of choice and usually reveals signs of appendiceal inflammation with a dilated caliber of the appendix, luminal obliteration by fluid, peri-appendiceal inflammatory changes as well as free and loculated fluid collections -3.We report the case of a 33-year-old man presenting with acute appendicitis resulting in sealed off appendiceal perforation secondary to metastatic adenocarcinoma from primary gastric cancer which was subsequently confirmed upon histopathology.A 33-year-old man who had undergone partial gastrectomy for gastric adenocarcinoma was undergoing adjuvant chemotherapy. His disease was in remission with no evidence of residual disease or recurrence on surveillance post-operative scans and endoscopic examinations. He presented to the emergency room two months after the start of adjuvant treatment with complaints of severe diffuse abdominal pain having a severity of 10/10 on the numeric pain intensity scale. His body mass index (BMI) was 24.5 kg/m2. His vital signs revealed tachycardia with a pulse rate of 118 beats per minute, fever of 38 °C, blood pressure of 120/80 mmHg, respiratory rate of 19 per minute, and oxygen saturation of 96% on room air. General and systemic examination was positive for generalized distress and severe diffuse abdominal tenderness upon palpation. Initial lab investigations revealed markedly increased white cell count of 23000/microliter and significantly increased serum lactate level of 43 mg/dL.He was admitted to the high dependency unit and started on supportive therapy, antibiotics, and analgesia. An urgent CT scan of the abdomen was ordered to determine the cause of his abdominal pain.A contrast-enhanced CT of the abdomen was subsequently performed to evaluate the cause of abdominal tenderness. There was no evidence of local disease recurrence on the scan. However, the appendix was dilated in caliber measuring up to 10 mm. It had surrounding inflammatory changes with peri appendiceal localized fluid collection and fat stranding Figures . Upon a The patient underwent an emergency laparotomy and appendectomy. Per-operatively the appendix was mildly inflamed and a large volume of ascites was drained. The anastomotic site of prior gastric surgery was unremarkable. He was shifted to the intensive care unit and mechanically ventilated for two days. His post-operative course was on a continuous improving trend and he was subsequently shifted to the general ward and later discharged from the hospital in a stable, asymptomatic condition. The histopathological examination of appendectomy specimen subsequently revealed inflammatory changes of the appendix. There were foci of poorly differentiated carcinoma as well. The immunohistochemical analysis was positive for cytokeratin and mucicarmine. A final diagnosis of metastatic adenocarcinoma with adjacent soft tissue involvement was made.Metastatic disease to the appendix is sparsely reported in the literature with common primary sites usually being the gastrointestinal and urogenital tracts, breast, lung, and the gallbladder ,3-6. MetAcute appendicitis can rarely be caused by metastatic spread to the appendix from a known primary malignancy. In this article, we present a case of a patient with known primary gastric adenocarcinoma who presented with signs and symptoms of acute abdomen; he was found to have acute appendicitis with a sealed-off perforation on imaging and the histopathology of appendectomy specimen revealed metastatic disease. There can be a number of reasons resulting in the possible delay in workup and treatment of this condition e.g. low clinical suspicion of appendicitis as a cause of acute abdomen and other mimickers which are more common in oncology patients like intestinal obstruction or bowel ischemia. Improving knowledge among clinicians and radiologists regarding this rare entity can help in rapid diagnosis and treatment.


Pancreatitis is caused by a number of different etiologies, most commonly caused by gallstone induced, alcohol, and familial hypertriglyceridemia. Other less common causes include trauma, medications, and autoimmune conditions. Drug-induced pancreatitis (DIP) is only responsible for less than 2% of all cases but is a very important etiology that has been observed with increasing frequency in the acute setting. Here we present a case of recurrent pancreatitis with no other risk factors except for the initiation of quetiapine approximately six months prior to the first episode.  Associated with over 100,000 hospitalizations per year, pancreatitis remains the number one gastrointestinal reason for hospitalization in the United States . With thA 46-year-old Caucasian male with past medical history of noninsulin dependent Type II diabetes, hypertension, recurrent pancreatitis episodes, asthma, and gout presented to the ED after experiencing severe right upper quadrant pain radiating to his back that started today. The patient stated that he was standing on the side of the road with his motorcycle because he was fixing a certain part that had become loose when suddenly he started to experience severe right upper quadrant pain that made him pass out. He denies hitting his head and stated he was out for approximately two hours (he looked at his watch and noticed how much time had passed), when he awoke he went straight to the hospital. He described the pain as 10/10, burning in nature, associated with an episode of nonbloody, nonbilious vomiting. He denied having any diarrhea, chest pain, shortness of breath, or trauma of any kind. Upon admission, his vitals were within normal limits, blood glucose: 565 mg/dL, calcium: 11 mg/dL, aspartate aminotransferase (AST): 52 u/L, alanine transaminase (ALT): 64 u/L, bicarbonate: 12 mmol/L, anion gap: 18 mmol/L, B-hydroxybutyrate: 0.82 mmol/L, triglyceride: 1213 mg/dL, lipase: 374 u/L, urinalysis was significant for protein 4+, ketones 2+. A CT of the abdomen/pelvis without contrast was significant for “acute edematous pancreatitis centered on the pancreatic head and uncinate process along with pancreatic fluid collection (Figures Although very uncommon, the prevalence of DIP seems to be growing as more cases are being reported -5. The eQuetiapine-induced hypertriglyceridemia causing acute pancreatitis has been reported in only six published case reports . The mosWe have seen many different etiologies that can result in pancreatitis and though not as common, medications should never be overlooked. If no obvious cause is identified, it is wise to always think of any new medications a patient might have recently started taking. We present this case in the hopes that it will help make physicians and providers aware of DIP and lead to early discontinuation of the offending agent.


Filariasis is a major health problem in certain parts of India. Microfilaria detection on exfoliative and fluid cytology is extremely rare and it is even very rare to find microfilaria coexistent with neoplastic lesions.A 45-year-old nulliparous female, who presented with complaints of lower abdominal swelling and pain. Initially, a clinical diagnosis of cystadenoma was made. Ascitic fluid cytology of the patient showed 3-dimensional clusters of cells along with an occasional microfilaria. On histopathology, a diagnosis of Mucinous cystadenocarcinoma of ovary was made.W. bancrofti followed by Brugia species and it affects young adults. The adult form of the filaria lodge in lymph vessels, and due to lymphatic blockage in neoplasms they appear in tissue fluid or on surface material. It is the first case report of microfilaria being detected in ovarian cystic fluid cytology in a patient of Mucinous cystadenocarcinoma of ovary.Most common cause of lymphatic filariasis is Filaria is an incidental finding in most of the cases, hence, cytopathologist should be vigilant and careful screening of all the slides should be done, especially in a country like India, where it is highly endemic. India contributes around 40% of the total global disease burden and about 50% of people are at risk of infection ,2. LymphAlso, there are no reported cases of microfilaria in ovarian cyst fluid in patients with co-existing ovarian neoplasms. Adult filarial worm frequently lodge in lymph nodes and in lymphatic plexus of the male genitalia. However, filariasis of the female genital tract is extremely rare .Here, we present a very rare case of mucinous cystadenocarcinoma of ovary in which ovarian cyst fluid cytology of the same patient demonstrated microfilaria. This case is reported in line with the SCARE criteria .2A 45-year-old nulliparous female presented with lower abdominal pain and swelling to Gynaecology OPD. On per vaginal examination, a mass was felt in right fornix. Rest of the general physical and systemic examination were within normal limits.CT-scan of abdomen showed an heterogenous right adnexal mass measuring 20 × 20 cm along with mild ascites, hepatomegaly and hepatic hemangioma.Based on the examination and radiological findings, a clinical diagnosis of ovarian cystadenoma was made. Right ovarian cystectomy was performed and was sent to histopathology department.Also, received were peritoneal fluid and intraoperative ovarian cyst fluid for cytological evaluation.2.1Cyst fluid was haemorrhagic on gross examination and on microscopy three dimensional clusters of cells with smooth outline were visualized. These cells had large hyperchromatic nucleus with scant cytoplasm. An occasional microfilaria was identified in the haemorrhagic background .Fig. 1Ce2.22.2.1Outer surface of the ovarian mass was glistening grey white with solid, cystic and congested areas. Cut surface showed multiloculated cysts of varying sizes which were filled with mucoid material and serous fluid .Fig. 2GrMultiple sections studied from ovarian mass showed a tumor comprising of tumor cells arranged in clusters, glandular and in singly scattered pattern. Tumor cells have round to oval vesicular nucleus with moderate amount of eosinophilic cytoplasm and showed infiltration into ovarian stroma  & 4. Muc2.3W. bancrofti followed by Brugia species. It affects young adults. It is transmitted through the bite of Culex mosquito [Filariasis is a major public health issue in south east Asia including Indian subcontinent and tropical region. In India, the most common cause of lymphatic filariasis is mosquito . It is emosquito .Two hosts are present in the life cycle of nematodes, female Culex mosquito as intermediate host and humans as definitive host. Microfilariae are the infective agent. Larvae forms from the mosquito enter the lymphatics of humans and develop into adult worms. They reside in lymphatics and release microfilaria into the peripheral blood to be taken up by another mosquito .W. bancrofti has a sheath longer than the larval body and nuclei are of equal size, countable and the tail tip is free of nuclei. Brugia malayi shows secondary kinks with its larval form showing coalescing uncountable nuclei, extending into the tail tip. Microfilariae on MGG slides appears shorter and stouter, and, on Papanicolaou-stained slides they appear longer and slenderer. Adult worm of W. bancrofti resides in lymphatic vessels, and microfilariae is found in peripheral blood (Microfilaria of al blood .Table 1MThe adult form of the filaria lodge in lymph vessels, and due to lymphatic blockage in neoplasms they appear in tissue fluid or on surface material. Microfilaria can also present as dermatitis and skin nodules. Microfilaria has been reported as an incidental finding in neoplastic lesions .Pantola et al.  reportedSane and Patel reported a case where adult filarial worm was identified in cystic teratoma of ovary . Mali BNShubham S et al. ,21 reporFew publications are available reporting coexistence of microfilaria and malignant neoplasms/malignant effusion on cytosmear. Concentration of parasites in neoplasms occurs due to their rich vascular supply .To the best of our knowledge, it is the first case report of microfilaria being detected in ovarian cystic fluid cytology in a patient of Mucinous cystadenocarcinoma of ovary.2.4Filaria is an incidental finding in most of the cases, hence, cytopathologist should be vigilant and careful screening of all the slides should be done, especially in a country like India, where it is highly endemic. Also, the presence of unexplained acute and chronic inflammatory cell infiltrate in the exfoliative cytology specimen of neoplastic effusions requires a thorough search for helminths.None declared.No funding has been received.Exempted as ethical approval not required.Informed written consent was taken from the patient for the publication of this manuscript and any images associated with it.Study concept or design - Dr Sonam.Data collection - Dr Shakti.Data analysis or interpretation - Dr Sompal and Dr Namrata.Writing the paper - Dr Vyshnavi.Dr Sonam Kumar Pruthi.NA.Not commissioned, externally peer-reviewed.


When a gastrointestinal intussusception is found, an underlying lesion should be excluded. When a gastrointestinal intussusception is found, an underlying lesion should be excluded. A 46‐year‐old woman was referred to the emergency department with severe epigastric pain. Physical examination, electrocardiogram, chest X‐ray, and laboratory findings were normal. A contrast‐enhanced computed tomography (CT) Figure  was perfThe patient underwent gastric endoscopy and biopsy. An ulcerated submucosal mass at the gastric fundus Figure  was seenIntussusception or invagination of the proximal gastrointestinal tract is uncommon, and only few gastrogastric intussusceptions have been reported. Most intussusceptions have been reported in children, where 90% of cases are idiopathic. In adults however, in up to 90% of cases an underlying pathology could be found. Neoplasms account for 65% of all causative lesions in adults.None declared.JV: made diagnosis, obtained and edited images, and prepared and drafted the manuscript. NM: obtained images and reviewed the manuscript. FV and JDM: reviewed the manuscript.


Meningiomas are primary intracranial tumors derived from arachnoid cap cells or meningothelial cells and usually display dural attachment. However, a small proportion of meningiomas that arise from the Sylvian fissure do not manifest dural attachment. Sylvian fissure meningiomas are relatively rare and have differential characteristics as compared with typical meningiomas. Herein, we reported a special case of atypical meningioma in the Sylvian fissure, that showed non-enhancement after contract management. The patient was a 30-year-old woman with a 2-year history of seizures. Preoperative computerized tomography and magnetic resonance imaging scans showed a calcific, non-enhancing lesion in the right insula lobe. The lesion was excised surgically for seizure control. Intraoperatively, the tumor was observed to be closely adhered to the middle cerebral artery (MCA), resulting in mild arterial damage. A case of Sylvian fissure meningioma was ultimately diagnosed by histopathological examination of the resected specimens. Sylvian fissure meningiomas are closely associated with the MCA and exhibit unusual imaging characteristics. Preoperative misdiagnosis may have serious adverse consequences and may result in incorrect surgery. To improve awareness of Sylvian fissure meningiomas on the differential diagnosis of Sylvian fissure lesions among clinicians, we present this report and briefly summarize previously reported cases to describe the clinical, pathological, radiological, and surgical features. Generally, meningiomas originate from the dura mater; however, some cases originate from the Sylvian fissure and show non-dural attachment. These cases can be easily missed preoperatively or may be misdiagnosed. Atypical meningioma is a distinct meningioma subtype and accounts for a small proportion of meningiomas with poor prognosis . At presThe patient was a 30-year-old homemaker with a 2-year history of seizures before admission. She had previously been treated with a routine antiepileptic  for 3 months; however, the drug was weaned off because of its side effect (weight gain). Subsequently, she was prescribed lamotrigine (200 mg per day) and topiramate (75 mg per day) for seizure control, but continued to have occasional seizures, once or twice a month. She had no other neurological deficit or past medical history, and results of systemic examinations were normal. She had no family history of hereditary diseases. Electroencephalography performed after admission detected epileptiform activity in the right cerebral hemisphere. Computerized tomography (CT) demonstrated a calcific lesion in the right Sylvian fissure and posterior part of the insula . An MRI *20 mm) was observed. It had a relatively elastic consistency and the mass partially invading the brain parenchyma of the insula, was densely adhered to the branches of the medial cerebral artery (MCA), and had partial calcification. Intraoperatively, despite careful micro-dissection, the MCA perforators were mildly damaged. Ultimately, several surgical specimens of the excised tumor were sent for histopathological analysis. Postoperatively, the results of histopathological examinations revealed a World Health Organization (WHO) grade II meningioma of atypical type  showed complete total excision of the tumor . The patMeningiomas are the second most common primary central nervous system tumors (CNST), accounting for approximately one-third of the primary CNST cases , 34. TheThe mean onset age of typical meningiomas is about 55 years, and the incidence increases with age, with women having a higher morbidity than men . HoweverThe majority of common meningiomas are WHO grade I, with approximately 16.9% cases being atypical (WHO grade II) . HoweverThe main radiological characteristics of meningiomas generally include extra-axial occupation and dura mater attachment . Contrarily, radiological imaging features of Sylvian fissure meningiomas have differential characteristics, such as intra-axial mass without dural attachment. It is extremely difficult to differentiate Sylvian fissure meningioma from other intracranial masses, such as low-grade glioma, teratoma, metastasis, cavernous hemangioma, tuberculous granuloma, etc. The previously reported 38 cases of Sylvian fissure meningiomas frequently showed hypointense or isointense lesions on T1- and T2-weighted MRI, with homogeneous or heterogeneous enhancement after contrast administration. Some cases also showed calcification or edema on CT and MRI scans, but these features were non-specific. Interestingly, the tumor in our case presented obvious calcification with negligible enhancement on the preoperative contrast-enhanced MRI scan, which significantly complicated the preoperative diagnosis. Meningiomas without enhancement have rarely been reported previously. Kubota et al.  and ZhanIt is well-acknowledged that the progressive growth and enlargement of typical meningiomas can oppress or enclose the arteries. However, owing to the layer of arachnoid membrane between the blood vessels and tumors, it is relatively easy to intraoperatively discern and separate the meningiomas from the adjacent arteries. Because of the close relationship between the Sylvian fissure meningiomas with the MCA and its branches, blood vessels are more likely to be damaged during resection surgeries. In our case, a branch of the MCA was injured intraoperatively despite careful micro-dissection, and the patient had a mild cerebral infarction postoperatively. Fortunately, the patient recovered without any obvious neurological impairment after an adequate recovery period and remained seizure free for about a year and a half postoperatively. According to the previously reported cases, 22 (57.9%) patients achieved gross total resection in the first attempt and showed favorable outcomes with a relapse-free status  at follow-up even after several years. Of the 15 patients who underwent subtotal resection of tumor, three patients underwent a secondary surgery, and four patients accepted subsequent radiation or gamma-knife therapy. However, the residual tumors in all 15 patients showed no sign of further progression. Patients at risk of serious postoperative complications with gross total excision surgery can be recommended for radiation or gamma-knife therapy , 22, 25.Sylvian fissure meningiomas are rare, and a preoperative diagnosis is difficult without adequate knowledge of the case. Due to the special origin, Sylvian fissure meningiomas generally present non-dural attachment and have a close relationship with the MCA . Tumor oThe studies involving human participants were reviewed and approved by Institutional Review Board of the Second Affiliated Hospital of Zhejiang University School of Medicine [Hangzhou]. Written informed consent was obtained from the patient for the publication of this case report.CC and ZZhu: drafted the manuscript. XinG and XiaG: acquisition of data. HJ, ZZhe, and AS: analysis or interpretation of data. JZhu and JZha: study concept and design.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Radiation-induced cavernous malformation (RICM) is a rare sequela of stereotactic radiosurgery (SRS) treatment of intracranial tumors. To date, no study reported on RICM after SRS for meningiomas originating from the skull base. The relationship between locus of initial meningioma and RICM has not been studied.A 57-year-old woman presented with persistent headaches and blepharoptosis at initial episode. MRI disclosed a right parasellar lesion, diagnosed as a cavernous sinus meningioma (CSM). After receiving a single-fractionated SRS, headache relieved, but blepharoptosis did not significantly improve. Three years and three months later, she returned with headaches and dizziness. MRI showed an enlarged CSM. Moreover, a new mass-like lesion, suspected hemangioma, appeared in the nearby right temporal lobe. After surgical removal of the new lesion and the CSM, the patient’s neurological symptoms significantly improved. Pathology confirmed CSM and temporal RICM.We report the first rare case of RICM occurring after SRS for CSM. The RICM may be in the same region as the initial tumor. Surgical intervention was preferred for symptomatic RICM and initial meningioma. We recommend long-term regular followup MRIs for patients with meningioma after SRS treatment. Due to the proximity of CSMs to critical cranial nerve (CN) and vascular structures, complete resection using an endoscopic or transcranial skull base approach comes with high surgical risks. SRS is an effective treatment for CSMs after their resection or as an upfront treatment –3. WhileRadiation-induced cavernous malformation (RICM) is a rare sequela after radiotherapy for intracranial tumors. Increased initial intracranial tumors including glioma, ependymoma, medulloblastoma, and cavernoma , 12, havWe report a case of RICM after SRS treatment for a CSM. A 57-year-old female presented with a persistent headache and right blepharoptosis in December 2015. The headache was characterized as a persistent needle-like pain in the right orbital and cervico-occipital region. Other symptoms included visual ghosting and tearing. Cranial MRI revealed an abnormal signal in the right parasellar region approximately 1.4 × 1.9 × 1.9 cm in size with an unclear boundary. It was slightly hypointense on T1-weighted images (T1WI) and slightly hyperintense on T2-weighted images (T2WI). The MRI signal enhanced significantly after administration of contrast , and immunohistochemical staining showed the tumor cells to be positive for EMA, vimentin, with 2% positive for Ki-67 indices, and S100 and GFAP negative Fig. a, b, c. With a good long term tumor control rate and low morbidity, SRS offers a viable alternative for treating skull base meningioma , 9. ReviRICM is also a radiotherapy complication for intracranial tumors, especially meningiomas. The pathophysiological mechanisms for RICM’s development remain unclear. Two hypotheses have been proposed . First, RICM is quite rare. According to our literature review from 1994 to 2018, only six cases of meningiomas have been diagnosed with RICM after conventional radiotherapy or SRS (Table According to the literature review, the locations wherein RICMs developed after conventional whole-brain radiotherapy may be random, either in the same area as the initial meningioma or in a different area, with possibly multiple nidi simultaneously emerging. While RICM after SRS treatment may be single, the location of the initial meningiomas was not specified in the literature Table . None ofSymptoms disappeared after surgical removal of two RICMs in the patient with drug-resistant headache . In NagyThe median detection time of RICM after cranial irradiation is 8 to 12 years –29, and In conclusion, we report the first rare case of RICM occurring after SRS for CSM. The RICM may be in the same region as the initial tumor. Surgical intervention was preferred for symptomatic RICM and initial meningioma. We recommend long-term regular followup MRIs for patients with meningioma after SRS treatment.


Supernumerary penises is an extremely rare congenital anomaly which affects one in every 5–6 million live births. Affected patients may have only a rudimentary penis, supernumerary penile glances or complete duplication or triplication of penises. Some patients may have some other associated congenital anomalies.A 3-month-old child presented because of left side hydrocele. There were evidence of two supernumerary penises in the perineum, the first one was about 2 cm in length with a glans and was attached to the root of the penis, and the third one was about 1 cm and was below the scrotum. Hydrocelectomy was performed. The two supernumerary penises were extending to perineal region and were attached to original penis, both had corpora cavernosum and spongiosum with no urethra inside. Both supernumerary penises were excised and both corpora were sutured with a fine slowly absorbable suture material. The patient was discharged with no postoperative events and follow up was done for one years with no reported adverse events.Triphallia (three penises) is unreported condition in human until now. Patients with supernumerary penises have unique presentation and no cases are identical. The position of the penis may be ectopic or orthotopic. Treatment is difficult because it poses medical, ethical, and cosmetic aspects. A combined multidisciplinary team is required for the management and long term follow up is required. Excision or reconstruction of the duplicate penis is required depending on the corporal development and anatomy of the urethra. Duplication of the penis or diphallia is reported to affect one in every 5–6 million live births. The extent of this anomaly and other associated anomalies vary greatly from patient to another. Affected patients may have only a rudimentary penis, supernumerary penile glances or complete duplication or triplication of penises. Some patients may have some other associated congenital anomalies .This condition occurs during the embryonic development of the penis between the 3rd and the 6th weeks of gestation, or may occur during the process of ventral migration and fusion of the paired mesodermal anlagen at the 15th week of gestation. Strong environmental factors like drugs and infections may affect the fetal caudal mass of mesoderm during this period suggesting that epigenetic mechanism may play a role in pathogenesis of diphallia [Penile duplication is divided on the basis of the presence of one or two corpora cavernosa in each penis into bifid phallus and true diphallia respectively, the term pseudo-diphallia is used to describe a small rudimentary penis. Most cases have a single corpus cavernosum in each organ; i.e. bifid phallus ,4.The penile urethra may show a wide range of associated anomalies, some patients may have a completely functioning urethra with normally placed meatus while others may have complete absent urethra .Imaging particularly ultrasound is helpful to confirm the diagnosis by detecting the presence or absence of corpora cavernosum or corpora spongiosum and their number, it also may detect other associated anomalies, magnetic resonance imaging provide a better quality of images in delineating the anatomy which is helpful to make an appropriate surgical decision ,5.The treatment of this condition is difficult because it poses medical, ethical, and cosmetic aspects. A combined multidisciplinary team is usually required for the management and long term follow up is usually required ,6.To the best of our knowledge, this is the first reported case with three penises or triphallia as no similar case is present in literature in human beings. This will add to literature because this may change the classification of multiple penises and help the authors for standardizing the management planes according to individual cases.The work of this report case has been reported in line with the SCARE 2018 criteria .2A 3-month-old child, who is native Kurd from Duhok city, presented by his parents because of left sided scrotal swelling and 2 skin projections in the perineum.There was negative history for drug exposure during pregnancy. The family history for any relevant genetic abnormalities and psychosocial histories were also negative.2.1The general examination revealed no abnormal findings.Examination of the genitalia revealed evince of a left sided scrotal swelling which was trans-illuminating on applying light suggesting a hydrocele. There were evidence of two projections in the perineum, the first one was about 2 cm in length with glans and was attached to the root of the penis, and the third one was about 1 cm and was below the scrotum, the two projections were consistent with supernumerary penises 2.2Ultrasound examination of the abdomen, kidneys, ureters, and urinary bladder showed no other associated anomalies.2.3Surgery was performed under general anesthesia, during surgery the hydrocele sac  was ligated at the inguinal canal.The two supernumerary penises were extending up to perineal region and were attached to original penis, both had corpora cavernosum and spongiosum with no urethra inside. Both supernumerary penises were excised and both corpora were sutured with a fine slowly absorbable suture material, the skin was then sutured with fine interrupted suture material.The operation was performed by a consultant urologist who is expert in the field of uro-surgery and penile reconstruction.2.4The patient was discharged with no postoperative events and follow up was done for one years with no reported adverse events.The family were advised about regular visits especially at the time of puberty and before marriage.3The condition of supernumerary penises is an extremely rare congenital anomaly and each case has a unique presentation and no cases are identical. The position of the penis may be ectopic or orthotopic ,3.As the karyotype is normal 46XY) in most cases, this make it difficult to explain the exact cause for the duplication because the genital tubercle is not a paired structure during normal embryonic development. Many theories are present suggesting its pathogenesis, but there is still a great debate and none of them is convincing [6XY in moMany genetic alterations have been implicated in the development of supernumerary penises, some of these include genes which encode for the expression of androgen receptors and are linked to the development of the male external genitalia .There are numerous associated anomalies which are reported in such patients, these may include abnormalities of the meatus like hypospadius, or epispadius, anomalies of the scrotum like bifid scrotum, anomalies of the testes like ectopic or undescended testis, anomalies of the blabber or the lower gastrointestinal tract, skeletal or pelvic bone anomalies, or cardiac anomalies. When the infant has major associated congenital anomalies, it is associated with high death rates .Many clinical classifications are present for this conditions, some authors classify it into a true supernumerary penis or just a bifid penis, which is further subdivided into partial or complete. The true complete type refers to complete duplication in which each penis has two corpora cavernosa and one corpus spongiosum, the partial type include either smaller or rudimentary penis. The term bifid phallus is applied when each penis has only one corpus cavernosum. In other cases only the glans is duplicated ,4.The extent of erectile function in such cases varies greatly, one or both penises may achieve erection, and rarely simultaneous ejaculation is reported if the condition is left untreated until puberty, while in pseudo-diphallia this is not present due to rudimentary phallus .There is no standard managing plan for all cases and the choice of the treatment plan depends greatly on the presence or absence of type of other associated congenital anomalies and the preservation of the continence and the erectile function. The aim of surgical intervention should be good urinary continence and stream, proper erection and an adequate cosmetic appearance .As this case is the first reported case, the management was based on the published papers, as many similarities are present between the cases. The rationale behind our surgical approach was that one of the supernumerary penises was apparently normal with urethra and the other 2 were accessary penises which were lacking urethra.The surgical procedure is individualized for each patient as each case has a unique presentation and may be associated with many other congenital anomalies. Surgery usually include excision of the supernumerary non-communicating penis or penises, penile reconstruction by joining both corporal bodies of each penis is recommended in cases of true complete diphallia, and urethral reconstruction ,4.4Triphallia (three penises) is unreported condition in human until now. Patients with supernumerary penises have unique presentation and no cases are identical. The position of the penis may be ectopic or orthotopic. Treatment is difficult because it poses medical, ethical, and cosmetic aspects. A combined multidisciplinary team is required for the management and long term follow up is required. Excision or reconstruction of the duplicate penis is required depending on the corporal development and anatomy of the urethra.The author has no conflicts of interest to declare.None.Ethical approval has been exempted by my institution for reporting this case.An informed written consent was taken from the family for reporting the case and the accompanying images.Dr Shakir Saleem Jabali and Dr Ayad Ahmad Mohammed contributed to the concept of reporting the case and the patient data recording.Drafting the work, design, and revision done by Dr Ayad Ahmad Mohammed.Final approval of the work to be published was done by Dr Ayad Ahmad Mohammed and Dr Shakir Saleem Jabali.N/A.Dr Ayad Ahmad Mohammed is guarantor for the work.Not commissioned, externally peer-reviewed.The family was worried about the future sexual life of their child, and they were informed that long term follow up is required especially at the time of puberty and before marriage.


In 1991, Genta and Haggitt described four patients with segmental ischemic colitis caused by idiopathic myointimal hyperplasia in the small mesenteric veins (IMHMV). There are now 33 published cases of IMHMV in the literature; however, this condition is still sufficiently rare that it poses a diagnostic challenge to pathologists and clinicians and is often clinically or histologically confused with inflammatory bowel disease (IBD) or ischemic colitis. IMHMV is characterized by intimal smooth muscle hyperplasia resulting in thickened small and medium‐sized mesenteric veins . Clinically, it presents with symptoms that mimic IBD, such as bloody diarrhea, abdominal pain, and weight loss. Surgical resection appears to be curative. The present case describes a 63‐year‐old Vietnamese man with cardiovascular risk factors who was diagnosed with IMHMV after many months of severe symptoms. A review of the current literature follows the case report. Idiopathic myointimal hyperplasia of the mesenteric veins (IMHMV) is an inflammatory bowel disease‐mimic, characterised by intimal smooth muscle hyperplasia resulting in thickened small and medium‐sized mesenteric veins . We describe a 63‐year‐old Vietnamese man with cardiovascular risk factors who was diagnosed with IMHMV after many months of severe symptoms. A review of the current literature, including 33 published cases of IMHMV, follows the case report. A 63‐year‐old Vietnamese man was electively admitted to a tertiary hospital in Melbourne after undergoing an outpatient colonoscopy to investigate progressive bloody diarrhea. Symptoms over the 3 months preceding admission included acute‐onset watery diarrhea, up to 10 bowel motions per day, fecal incontinence, and significant weight loss. There was no associated pain, nausea, or infective symptoms. In the community, a trial of metronidazole and an antidiarrheal agent was ineffective. Past medical history included a basal ganglia hemorrhage with residual left‐sided weakness and hypercholesterolemia, and he took regular medications for secondary stroke prevention. He had no family history of bowel cancer or inflammatory bowel disease (IBD). Apart from antibiotic exposure he did not have infective risk factors such as recent overseas travel or sick contacts.2, having lost 14 % of his body weight in 4 months. Abdominal examination demonstrated a soft abdomen, with no tenderness or peritonism. Severe malnutrition was present with loss of muscle mass and subcutaneous fat. There was no lymphadenopathy or clinical signs of thyrotoxicosis. Per‐rectal examination was unremarkable. Laboratory investigations showed mild lymphocytopenia (1.1 × 109/L) but an otherwise unremarkable routine blood panel. A nutrition screen showed hypoalbuminemia . A septic screen was negative, including QuantiFERON‐TB Gold and human immunodeficiency virus serology. There were moderate numbers of leukocytes and erythrocytes on a fecal specimen but no cultured bacteria and parasites, and Clostridium difficile toxin was not detected. Fecal calprotectin was mildly elevated (96.5 μg/g). Abdominal computed topography (CT) indicated thickening of the distal descending colon, favoring an infective or inflammatory colitis  is not caused by arterial thromboembolism, venous thrombus or vasculitis.Of the 34 cases of IMHMV that occurred in men, usually in late middle age (median age at diagnosis 62 years).et al.The most common site of disease is the left colon, particularly rectosigmoid colon. Three cases were reported to occur in the small bowel,The radiological appearance of IMHMV on computed tomography (CT) is suggestive of colitis, with thick, edematous bowel and fat stranding.In three cases, the diagnosis of IMHMV was reached prior to surgical resection via endoscopy and histology.et al. have suggested that chronic mechanical stress from intermittent torsion on the veins of the rectosigmoid colon may play a role in the development of myointimal hyperplasia.The pathogenesis of the myointimal hyperplasia in mesenteric veins is unknown. Genta and Haggitt and Abu‐Alfa and colleagues both noted that the venous changes were similar to those seen in saphenous vein graft failing around 12 months following coronary artery bypass surgery.Another hypothesis is that IMHMV represents a burned out stage of enterocolic lymphocytic colitis as one caseAlthough this entity was first described in 1991, it is still not recognized and diagnosed by gastroenterologists and anatomical pathologists until resection of the affected colonic segment. IMHMV does share some histological features of ischemic colitis unrelated to IMHMV—surface epithelial degeneration, crypt withering, and lamina propria hemorrhage. However, a recent, very elegant study by Yantiss and colleagues highlighted the pathognomonic three small vessel changes seen in mucosal biopsies and resections, “arteriolization” of capillaries, subendothelial fibrin deposits, and fibrin thrombi, which were typical of ischemic colitis due to IMHMV.IMHMV is a rare but important diagnosis that leads to significant morbidity and mortality due to delayed diagnosis. Although the optimal medical treatment is unknown, prompt diagnosis and surgical resection is curative and can prevent prolonged symptoms, malnutrition, and complications such as bowel perforation. We aim to highlight the importance of IMHMV as a rare but increasingly recognized diagnosis of ischemic colitis without apparent arterial compromise.