Pituitary apoplexy (PA) is caused by a sudden increase in pressure in the pituitary region due to acute hemorrhage, infarction or necrosis. PA can also be caused by restricting blood supply to the nerve due to compression of the internal carotid artery. Acute third cranial nerve palsy (third CN) secondary to PA is a rare medical emergency caused by bleeding within a growing mass within the sella turcica. We presented two cases of PA with isolated third CN palsy treated with transsphenoidal pituitary decompression. PA is therefore an important differential diagnosis to consider in patients with isolated third nerve palsy. The prognosis for isolated third nerve palsy in PA appeared successful, with variable recovery from medical and surgical intervention. Pituitary apoplexy (PA) is a sudden neurological deficit caused by an expanding mass within the sella turcica, which may develop from pituitary hemorrhage, infarction or both, or from a pre-existing pituitary tumor such as an adenoma . As a reA 56-year-old hypertensive man presented to our emergency department with a chronic frontotemporal headache that had been worsening for 2 days and was associated with acute left ptosis without blurred vision or diplopia. There had been no loss of consciousness or seizures. On examination, his Glasgow coma scale (GCS) was 15/15. The right pupil was 3 mm light reactive, whereas the left was 4 mm non-responsive. Visual acuity and visual field circumference were normal in both eyes.Magnetic resonance imaging (MRI)  revealed a sellar and suprasellar lesion measuring 3 × 2.9 × 1.8 cm with patchy areas of high signal on T1-weighted images and signal void on T2-weighted images suggestive of hemorrhage. The suprasellar component of the lesion was displacing optic chiasma superiorly and the sellar component was causing sellar expansion with mass effect on bilateral cavernous sinuses (left>right); right internal carotid artery (ICA) had a contact angle of 90°, whereas left ICA had >90° .Preoperative hormone analysis showed normal levels of thyroid-stimulating hormone (TSH), growth hormone (GH), insulin-like growth factor (IGF), follicle-stimulating hormone (FSH), prolactin, free thyroxine, cortisol and luteinizing hormone (LH) levels.PA was diagnosed when radiographic findings showed acute pituitary hemorrhage and subsequent pituitary adenectomy was planned, but surgery was delayed up to 13 days because of non-critical coronary artery disease detected on coronary angiography. When his condition stabilized, he underwent surgery. The tumor was soft to firm and a safe maximal resection was performed. Hemostasis was achieved and sella packed with fat harvested from the subcutaneous fat of the abdomen. Postoperatively, he suffered transient diabetes insipidus, which was treated with desmopressin injections and resolved by day 5 postoperatively. He was then discharged on the 8th postoperative day. At the time of discharge, he had residual ptosis, which gradually improved over 2 weeks based on clinical assessment at the outpatient visit. He fully recovered from ptosis over a month. Postoperative imaging performed at 6 weeks showed a residual tumor measuring 1.9 × 1.5 × 1.1 cm with no significant mass effect on the cavernous sinus; therefore, he was referred to an oncologist for postoperative radiotherapy .A 50-year-old gentleman presented with painful ptosis of the left eye for 4 days with no signs of raised intracranial pressure, seizure or loss of consciousness. Clinically, GCS was 15/15. The right pupil was 2 mm reactive to light, whereas the left pupil was 4 mm sluggishly reactive, with no signs of meningism.MRI imaging showed a 2 × 1.6 × 1.6 cm sized sellar lesion arising from the pituitary fossa with an intrinsic high T1 signal in the periphery, extending superiorly in the suprasellar region and laterally, causing a mass effect on the left cavernous sinus. A signal void was seen in T2-weighted images suggestive of hemorrhage .A preoperative hormonal evaluation was performed, which showed normal levels of TSH, GH, IGF, FSH, prolactin, free thyroxine, cortisol and LH.The patient underwent transsphenoidal pituitary adenectomy 3 days after admission. Intraoperatively, soft tumors with hemorrhagic components were seen. Hemostasis was achieved and the sella was packed with fat. The patient was managed in the ICU for 2 days and discharged on the 8th postoperative day. The ptosis gradually improved within 2 days of surgery, and during the 2-week follow-up in the outpatient clinic, the ptosis recovered completely. Follow-up imaging showed no evidence of a residual lesion in sella turcica .PA is a rare medical emergency caused by a growing mass within the sella turcica, secondary to pituitary hemorrhage or infarction, or within a pre-existing pituitary adenoma . We repoAccording to recent epidemiological studies, it has an annual incidence of 0.17 per 100 000 people and a prevalence of ~6.2 cases per 100 000 people , 4, 6. IA third CN palsy as a presentation of PA is rarely present . It is cThe differential diagnosis of third CN palsy can be narrowed down by the presence or absence of accompanying pain and pupillomotor impairment. Compression lesions, particularly aneurysms, result in pupillary involvement, and isolated complete third nerve palsy with normal pupillary function characterizes ischemic lesions . CompresSeveral explanations have been proposed to explain why PA causes third nerve palsy. Because the oculomotor nerve lies horizontally in the same plane as the pituitary gland in the lateral wall of the cavernous sinus, it is often the first CN involved in the lateral spread of a pituitary tumor or hematoma . BecauseImaging of our patient showed a sellar and suprasellar mass with an acute hemorrhagic component and a mild mass effect on the left cavernous sinus. As a result of this, in our patient, there is a high probability of pressure on the third CN or impairment of the vascular supply of the nerve due to compression of the vasa nervorum from the ICA as the cause of the third CN palsy. To prevent acute adrenal insufficiency, patients with PA require intravenous steroids until surgery. The definitive treatment for PA is neurosurgical decompression via a transsphenoidal approach. Early decompression can partially or fully restore pituitary and third CN function .et al. [Patients who underwent surgery had better clinical improvement and endocrinological results since a viable tumor component could be removed from constrained areas such as the cavernous sinus . Howeveret al. , conservet al. .Tumor size regression is directly related to better ophthalmologic and endocrinologic outcomes , 18. EarOur patients underwent transsphenoidal neurosurgical decompression and pathological examination, confirming the diagnosis of PA. After the operation, the third CN palsy was completely resolved. The first patient underwent delayed decompression due to cardiac problems where recovery of third nerve function was delayed compared with the early decompression in the second patient. This finding may favor early decompression to restore the function of the third nerve palsy.PA is an essential differential diagnosis in patients with isolated third nerve palsy. The prognosis for isolated third nerve palsy in PA appeared successful with variable recovery with medical and surgical interventions.


Despite a normal D‐dimer level, abdominal contrast‐enhanced computed tomography revealed superior mesenteric artery dissection. Abdominal contrast‐enhanced computed tomography is mandatory when examining patients with sudden‐onset abdominal pain, even those with a normal D‐dimer level.A 51‐year‐old man with untreated hypertension developed sudden‐onset epigastric pain The D‐dimer level can be normal in patients with isolated superior mesenteric artery dissection. Abdominal contrast‐enhanced computed tomography is essential for patients with sudden‐onset abdominal pain, even those with a normal D‐dimer level.  A 51‐year‐old man with untreated hypertension awoke with epigastric pain, which disappeared after a few minutes. Three days later, he was transferred by ambulance to our hospital because sudden severe epigastric pain appeared when picking up a baseball. On admission, his blood pressure was 256/147 mmHg. He had epigastric tenderness and exhibited profuse cold sweating. Despite his normal D‐dimer level of 0.46 μg/ml, abdominal contrast‐enhanced computed tomography (CT) revealed superior mesenteric artery (SMA) dissection  is as low as 0.06%.SU involved in concept, literature search, and drafting of manuscript. SY involved in concept and drafting of manuscript. MT involved in concept and literature search. SI‐Y involved in concept and revision of manuscript.No specific grant was received for this work from any funding agency.The authors state that they have no conflict of interest.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Ovarian teratomas are most common germ cell neoplasms. Immature ovarian teratoma comprises less than 1% of all ovarian teratomas. It usually occurs in first two decades of life.We report a case of 4 years old female child presenting with pain and huge lump in lower abdomen. On abdominal ultrasonography, it revealed a solid-cystic pelvic lesion arising from left ovary. Magnetic resonance imaging (MRI) corroborated the ultrasonographic findings. She underwent laparotomy with right oophorectomy with excision of the mass. The histopathological examination of the excised mass confirmed it to be immature ovarian teratoma with yolk sac tumor. The patient had an uneventful recovery with no sign of tumor recurrence at a one and a half year follow-up.In spite of immature ovarian teratomas having aggressive behaviour and lethal outcome, a high degree of suspicion and timely management can translate into a very good eventual prognosis. •In ovarian mass, patient usually presents with non-specific complaints but a high degree of suspicion and imaging is very important for diagnostic work up.•Now-a-days tumor markers have also proved to be an important tool for screening the patients of ovarian mass.•Early diagnosis and management do reduce the risk of complications like torsion, rupture, hydroureteronephrosis and gastrointestinal obstruction.•It also reduces the probability of metastasis and hence improves event-free and overall survival of the patient. It is a tumor derived from an abnormal germ cell which undergoes meiotic division. On the basis of histology, ovarian teratomas of two types-mature and immature. The more common of these tumors is mature ovarian teratoma also known as dermoid cyst. Immature ovarian teratoma is uncommon comprising less than 1% of all ovarian teratomas . It cont2A four-year-old female child from a rural background of Haryana presented to Pediatric Surgery outpatient department with complaints of abdominal lump for previous one month and bleeding per vaginum off and on for 7 days. She had started having pain abdomen for last 4 days as well which was sort of continuous. There was no history of fever or any alteration in bladder or bowel habits. There was no relevant past medical or surgical history. On examination, the patient was afebrile, moderately nourished and stable hemodynamically. Abdominal examination revealed a hard, non-tender, unevenly surfaced lump of about 20*20 cm with indistinct margins present in and involving pelvic, bilateral iliac, umbilical, bilateral lumbar and epigastric quadrants of the abdomen . It seem3Overall, immature teratoma is most frequently found in sacrococcygeal region, which happens to be the most common site for all types of teratomas in childhood . Ovary aImmature ovarian teratomas are graded histologically based on amount and degree of cellular maturity: having grade 1 mature and grade 3 immature. For proper grading, multiple sections of primary lesion and wide sampling of peritoneal implants are done. Both primary lesion and implants should be graded according to most immature tissue present .There are some classical tumor markers for these germ-cell tumors. AFP is useful for diagnosing both yolk sac tumor and immature teratoma. And for discrimination between these two Kawai et al. study states that 94.4% of patients with yolk sac tumor exceeded an AFP level of 1000 ng/ml and in 95.2% cases of immature teratoma it was less than 1000 ng/ml. Other tumor marker used for immature teratoma is CA19-9 with a positivity rate of 57.1% [Treatment for immature ovarian teratoma is grade dependent; that is patients with stage 1 grade 1 immature teratoma are often treated with surgical resection of the mass due to favourable prognosis and very low risk of relapse. In a database study by Pashankar in 2016, out of 179 patients (98 children and 81 adults) he reported that 90 children were treated with surgery alone, and all adults received adjuvant chemotherapy . And gra4In ovarian mass, patient usually presents with non-specific complaints but a high degree of suspicion and imaging is very important for diagnostic work up. Now-a-days tumor markers have also proved to be an important tool for screening the patients of ovarian mass. Early diagnosis and management not only reduce the risk of complications like torsion, rupture, hydroureteronephrosis and gastrointestinal obstruction but also reduces the probability of metastasis and hence improves event-free and overall survival of the patient.The authors state that the work has been reported in line with the SCARE 2020 criteria .Not ApplicableNIL1. Pradeep Kajal: Operated upon the patient.2. Namita Bhutani: Conceptualised the article, did the final editing and submitted the article.3. Kirti Saini: Reviewed the literature and wrote the article.4. Preeti Kadian: Arranged the photographs and managed the clinical part.NOT APPLICABLE FOR CASE REPORTS.Written informed consent was obtained from the patient for publication of this case report and accompanying images.DR PRADEEP KAJAL.DR. NAMITA BHUTANI.Not commissioned, externally peer-reviewed.The authors declare that they have no competing interest.


Both small bowel intussusception in adults and small bowel hemangioma are rare benign mass lesions. Moreover, a secondary effect of intussusception caused by hemangioma is extremely rare.A 87-year-old female suffered from intussusception and intestinal obstruction caused by hemangioma located in the small bowel , reporting abdominal distention without nausea and vomiting.Emergency abdominal and pelvic computed tomography showed an intussusception with the evidence of associated small bowel obstruction. Histological analysis revealed as small intestinal hemangioma accompanied by mesenteric ulcer.The patient underwent segmental resection of intussusception of intestine instead of invalid conservative treatment.Although the postoperative pathological results were inconsistent with preoperative imaging examination, the old woman recovered well.The literature on intussusception of small intestine has described several possible causes including hemangioma, which more likely results in gastrointestinal bleeding or abdominal pain. Yet we experienced a rare case presenting as abdominal distention without nausea and vomiting, Therefore, preoperative diagnosis and localization of these lesions is of great importance. We recommend high resolution contrast-enhanced computed tomography and magnetic resonance imaging should be considered in diagnosis while capsule endoscopy is not available owing to the intestinal obstruction, as long as in facilitating surgical excision. Small bowel hemangioma maybe as 1 feature of Renda–Oslar–Weber Syndrome in accordance with the hemangioma of lung, liver and oral mucosa. We present a case with intussusception surrounded by polypoid mass-hemangioma of small bowel, which was recognized as showing “cuppy sign” on several clinical imagines.Although the incidence of small intestinal hemangioma is rare, ranging from 5% to 10%The ethics committee of Qingdao center hospital approved this study, and the written informed consent was obtained from the patient’s legal guardian.A 87-year-old female had a history of vague pain on upper abdomen for 10 days. She visited our hospital reporting abdominal distention without nausea and vomiting, she had last opened her bowels 1 day before the admission, however, there was no typical history of blood or mucus from the rectum. Emergency abdominal and pelvic computed tomography (CT) showed 1 segment of small intestine was inserted into another, formed the sign of “cuppy” and edema of related intestinal wall located in the part of ileum, suggestive of an intussusception with the evidence of associated small bowel obstruction , no abnormality was found from the rest laboratory examination.On admission, analysis of peripheral blood showed mild anemia  ranged from 0 to 4, which proved good tolerability for the patient. No adverse and other unanticipated events occurred in hospital. meanwhile, it can result in impaired peristalsis, obstruction, and even vascular compromise directly. Intussusception of small intestine is a rare disease in adult patients while primarily in children. Childhood intussusception is idiopathic in 95% of cases while adult intussusception is secondary to intraluminal lesion, has a specific pathological changes – in general, all kinds of malignant or benign neoplasm. For example, intussusception of the large intestine occur more frequently secondary to malignant lesions: adenocarcinoma and lymphoma. However, a secondary effect of intussusception caused by small bowel hemangioma is extremely rare. Few cases reported of small bowel intussusception were hemangioma in origin.The definition of intussusception is mentioned above Figs.  and 2: i: Capillary lymphangioma, cavernous lymphangioma, cystic lymphangioma (hygroma), and hemolymphangioma (a combination of hemangioma and lymphangioma). All kinds of the categories may result in occult or massive gastrointestinal (GI) bleeding, perforation, abdominal pain, obstruction and intussusception, According to the location of the overlapping organs, intestinal overlap presents a variety of nonspecific clinical symptoms, GI bleeding and abdominal pain are the most common, leading to anemia in most patients. The unspecific symptoms of the early clinical manifestations in small intestinal hemangioma due to the particularity and complexity of the small intestinal anatomy. Small intestinal hemangioma is usually occult in onset, most of the patients are in a progressive stage once they have the typical symptoms, as a result, it is difficult to characterize with laboratory testing, meanwhile, it is hard to detect lesions with multiple inspections because of the concealed location of jejunum.Intestinal hemangioma is not a true tumor but one of the congenital benign vascular malformation lesions, which are rarely found in adult patients. These benign malformations are classified into 4 categories Small bowel capsule endoscopy is a noninvasive imaging test and can be recommended when the source of bleeding remains unidentified after upper and lower endoscopy.–14 The entire small intestine lesions including intraluminal ulcer, submucous polypus, parasite and foreign matter can be observed clearly, while capsule endoscopy also has limitations, such as biopsy, inaccurate positioning, not suitable for large amount of bleeding or intestinal obstruction. When positive findings are obtained on capsule endoscopy and there is no evidence of obstruction, double balloon enteroscopy or push enteroscopy is recommended for further management. As are also recommended in the American College of Gastroenterlogy clinical guideline (edition 2015).Although early diagnosis for small bowel hemangioma is quite difficult. However, several imaging modalities including wireless capsule endoscopy, double balloon push-type enteroscopy or colonoscopy, multiphase computed tomography enterography, magnetic resonance enterography, double barium contrast gastrointestinal imaging are currently available options to investigate small bowel lesions. The MRI sign of “filling-defect” image developed from small bowel neoplasm through oral iodine contrast, a relatively low-density shadow in T1 image while high-density shadow in T2 image. In this case, although a typical intussusception CT sign of “cuppy” and intestinal obstruction suggested the space occupying lesion in small bowel, however, the indefinite diagnosis was made due to the absence of 3-dimensional reconstruction of the small intestine. Therefore, we suggested that radionuclide scanning and selective angiography should be performed in order to confirm the diagnosis and further therapeutic schedule when endoscopy is not available . CT virtual endoscopy has been used to diagnose small intestinal hemangioma in domestic and abroad, but it has not been widely carried out in large medical centers.Despite the capsule endoscopy, contrast-enhanced CT and magnetic resonance imaging (MRI) play very important roles in the diagnosis of the lesions.Once the disease is diagnosed, it is appropriate for patients to receive surgical therapy. Laparoscopic or laparotomy exploration should be enforced based on the pathogenetic condition in case the indefinite diagnosis was made, thorough excision of the lesions is the ideal treatment. For a single lesion or dense lesions confined to 1 segment of intestine, the therapeutic effect can be achieved by intestinal resection. It is necessary to prevent postoperative short bowel syndrome and nutritional absorption disorder when large-scale resection of multiple lesions is performed.In conclusion, we experienced a rare case with small bowel intussusception caused by hemangioma, which is a benign vascular malformation that can cause chronic GI bleeding and intestinal obstruction presenting as chronic abdominal pain and distention. Therefore, preoperative diagnosis and localization of these lesions is of great importance. We recommend high resolution contrast-enhanced CT and MRI should be considered in diagnosis while capsule endoscopy is not available owing to the intestinal obstruction, as long as in facilitating surgical excision. In addition, capsule endoscopy and further double balloon enteroscopy or push enteroscopy for the diagnosis and management of obscure GI bleeding are considered as the 1st-line procedure, also the 1st-line initial examination of small intestine.Data curation: Yangyang Yu, Laixiang Zhang.Formal analysis: Laixiang Zhang.Resources: Chengyu Shi.Supervision: Cheng Gao.Writing – original draft: Chengyu Shi.Writing – review & editing: Chengyu Shi.


Colonic malakoplakia is an uncommon granulomatous development of cells resulting from the impaired capacity of the mononuclear cells to eliminate the phagocytosed bacteria, and in rare cases it can also affect the gastrointestinal tract. We report the case of a 78-year-old female patient that was admitted to hospital by The Emergency Department with the diagnosis of bowel obstruction, confirmed by the clinical and paraclinical investigations. We decided to surgically manage the case for suspicious symptomatic colonic neoplasm. The histological examination of the surgical specimens revealed colonic malakoplakia, characterized by the presence of the aggregated granular histiocytes and Michaelis-Gutmann bodies. Through this paper, we want to raise awareness for Malakoplakia, which remains an extremely rare disease that may affect multiple organs, and because it does not present specific symptoms or clinical manifestations, the final diagnosis remains the histopathological study. The clinical conduct should be decided after taking into consideration all the aspects of this pathology along with the benefits and risks for the patient. Malakoplakia was described for the first time by Michaelis and Gutmann in 1902 [This pathology is more frequently encountered in the urinary tract—about 75% of the reported literature cases. The gastrointestinal tract is the second most common site for the lesions, particularly the descending colon, sigmoid and rectum ,3. MalakIn our days, knowing the etymology of this rare pathology, we might state that the clinical appearance varies along the gastrointestinal tract and it can be identified as flat lesions, mucosal erosions or plaques, ulcerations or even polyps of different sizes. In some cases, patients might present tumors with characteristics of malignancy ,6.Colonic Malakoplakia was defined in most patients as part of a preexisting pathology like the diverticular disease or the ulcerative colitis, or in rare, isolated cases associated with a rectal or sigmoid colon adenocarcinoma. It has a non-specific clinical presentation in close relationship with the involved organ or region and it might result in symptoms such as abdominal pain, diarrhea, gastrointestinal bleeding or even bowel obstruction mimicking a colonic neoplasia .In the pathogenesis of the malakoplakia are supposed to be involved the deficiency of guanosine monophosphate dehydrogenase and the deficiency of beta-glucuronidase, which modifies the microtubular and lysosomal function resulting in the incomplete elimination of bacteria from the macrophages. Current evidence indicates a connection with the activity of the macrophages.β-glucuronidase is a type of glucuronidase, a member of the glycosidase 2 family, which catalyzes the hydrolysis of β-D-glucuronic acid residues from the non-translational end of mucopolysaccharides [β-glucuronidase is involved in the conversion of the conjugated bilirubin to the unconjugated form for reabsorption. The human β-glucuronidase is homologous to the enzyme Escherichia coli β-galactosidase [The human charides . It is ltosidase .The guanosine monophosphate-dehydrogenase is a regulator of intracellular guanine nucleotides and it is therefore important in DNA and RNA synthesis. The synthesis of guanine nucleotides is essential for maintaining normal cell function and growth. It is also important for maintaining cell proliferation and immune responses. B and T lymphocytes depend on guanosine monophosphate dehydrogenase for normal activation and function ,7.The macroscopic aspect of the malakoplakia lesion is solid, soft, friable, yellow or brown-yellow, of different sizes and with different distribution patterns. From a histological point of view, there is a diffuse histiocytic infiltration with eosinophilic granular cytoplasm (von Hansemann cells), containing characteristic basophilic laminated cytoplasmic inclusions, called Michaelis-Gutmann bodies, which are specifically colored in PAS staining (Schiff Periodic Acid) ,9,10.Malakoplakia is caused by deficits in phagocytic or degradative functions of the macrophages, as a response to the infections with Gram-negative coliform bacteria , which may lead to chronic inflammatory status, followed by accumulation of intracellular iron and calcium deposits that create mineralization (Michaelis-Gutmann bodies) ,11.The clinicians must be aware of the presence of this unusual pathology along the gastrointestinal tract. When the lesion can be identified as a polyp, nodule, presents ulcerations or is associated with lymph node involvement, malakoplakia can be very easily mistaken as a malignancy. The literature contains cases in which the patients diagnosed with malakoplakia presented various associations between colorectal adenocarcinoma, adenomas, systemic diseases or without any link with another pathology .The diagnosis can be very challenging, and in the majority of the cases stated in the literature, the pathological report was unexpected. In this paper, we are illustrating a case of colonic malakoplakia that was mimicking a locally advanced colorectal neoplasm, without any association with a colorectal adenocarcinoma, an adenoma or a systemic disease, which makes it a rare occurrence.T.D., a 78-year-old patient admitted by emergency in the 2nd Clinical Surgery Department of “Sf. Apostol Andrei” Emergency Clinical County Hospital Galati, for diffuse abdominal pain, absence of the intestinal transit, nausea and vomiting, symptoms which have started three days before presentation to the hospital.The laboratory examinations did not capture any pathological changes: WBC 5.560/mmc, Hb 11.8 g/dl, Ht 37.7%, PLT 252,000/mmc, Glycemia 99 mg/dl, AST 15 U/L, ALT 15 U/L, creatinine 1.17 mg/dl and urea 40 mg/dl.The abdominal radiography revealed hydroaerial levels in the right side of the abdomen and hypogastrium.left liver lobe = 58 mm,right lobe of the liver = 144 mm, with slightly increased echogenicity and microgranular structure,PV  = 12 mm,CBP (main biliary duct) = 5 mm,non-dilated CBIH (intrahepatic biliary duct),folded gallbladder in lower 1/3, with micro calculus of 5–6 mm,homogeneous pancreas,homogeneous spleen with 81 mm long axis,RK (right kidney) of normal size, IP retained, calculi of 5–6 mm, no pyelocaliceal dilations,LK (left kidney) of normal size, IP retained, calculi of 5–6 mm, no pyelocaliceal dilations,the urinary bladder half-full, without changes,marked aerocolia on the colic frame, without free intraperitoneal fluid.The abdominal ultrasound showed:We also performed an abdominal and pelvic computed tomography enhanced with contrast agent, which described a marked distension with hydroaerial levels and parietal aerial inclusions (pneumatosis) involving the intestinal loops, cecum and ascending colon, up to the hepatic angle, where a 10 mm thickened wall area was highlighted.Given the symptoms of the patient and the paraclinical findings and established clinical diagnosis of bowel obstruction, we decided to manage this case by performing an emergency surgery.During surgery, after close inspection of the whole abdominal cavity, the team identified a stenosing mass located in the hepatic angle of the ascending colon and subsequent bowel obstruction. We decided that a right hemicolectomy with ileotransversoanastomosis was the best surgical approach for our patient and we performed it.The postoperative evolution of the patient was favorable and uneventful, with the improvement of the general state, decreasing need for painkillers, active mobilization and good digestive tolerance.The patient was discharged after 14 days, and was aware and cooperative and in good general state.The histopathological diagnostic of the postoperator specimen (4394) was colonic malakoplakia as illustrated in a significant architectural remodeling of the colonic mucosa and submucosa with dense cellular inflammatory infiltrate,the dense cellular inflammatory infiltrate-composed of epithelioid macrophages with wide eosinophilic cytoplasm, variable-sized nuclei, with occasional nucleoli,dispersed syncytia with the appearance of multinucleated giant cells, with chaotically disposed nuclei andamorphous cytoplasmatic inclusions of cellular detritus type, with sporadic intercalated lymphocytes.The microscopic examination revealed:Neither areas of necrosis, nor viable microorganisms were detected.An area of ulcerated mucosa was identified, with reactive changes at the level of the remaining marginal epithelium.Additionally, no invasive dysplastic or neoplastic stigmata were identified.Furthermore, the anatomopathological department recommended immunohistochemical tests for a precise and definitive diagnosis of our patient.macrophages (positive CD68)-illustrated in the Figure 4with rare dispersed T lymphocytes (positive CD3),rare B lymphocytes  especially in the lamina propria,rare plasmacytes ,as well as rare mast cells (positive tryptase).The result of the immunohistochemical tests confirmed that the inflammatory infiltrate was predominantly comprised of:Additionally, most macrophages were positive for CD204 (MSR1), a receptor that plays a role in phagocytosis .Alpha-SMA was positive in the smooth muscle cells, from the level of the muscularis mucosae and the own muscle, being negative in the inflammatory cells.Malakoplakia represents a rare type of granulomatous inflammatory pathology that can affect multiple sites in the human body. The most frequent localization is the genitourinary tract, the digestive system, but also the nervous system, the respiratory organs and even the bones.It is most frequently encountered in patients diagnosed with chronic pathologies, like the ones illustrated in The most distinctive histological feature for the diagnostic of malakoplakia is represented by the Michaelis-Gutmann bodies, which are concentric, basophilic, round or oval lamellae, with a diameter of 5 to 10 µm, included in the cytoplasm of the histocytes, known as von Hansemann cells. These inclusions are generally positive PAS and von Kossa, being composed of a glycolipid matrix, probably of a specific microorganism and are covered with a calcium and iron layer. The foamy histiocytes are mainly dispersed in the lamina propria of the mucosa, next to rare lymphocytes and occasionally to multinucleated giant cells. Therefore, the precise diagnosis of this pathology can only be stated after microscopic examination, in the usual hematoxylin eosin staining, as well as by using the mentioned special staining, or by performing immunohistochemical tests .The literature data is illustrated by case reports and some small case series that bring light regarding this rare disease, its forms of presentation, the features of the patients diagnosed with it, their symptoms and the type of medical conduct that could be applied.We performed a PubMed search of other reported cases of digestive tract malakoplakia, especially the ones involving the colon, in order to illustrate the features of this rare pathology. We excluded the pediatric cases and the articles that could not be processed (no text available). The results are shown in As summarized in The etiology of malakoplakia is still in debate, but the specialty literature mentions possible pathogenic status like infections which involve Gram-negative pathogens, in particular E. coli and an abnormal response of the immune system, most probably generated by inefficient lysosomal functions of the macrophages ,10.rectal bleeding,diarrhea,abdominal pain in different grades,fever,weight loss in the recent period before presenting to the hospital,night sweats andintestinal tract obstruction.The clinical manifestations of the colonic malakoplakia depend on the extent of the colonic lesions and the coexisting pathology of the patient. They are nonspecific and include:Sessile or polypoid masses isolated in the rectosigmoid colon; the lumen may be narrowed which may suggest malignant stenosis. The inflammatory process may be transmural, similar to the Crohn’s disease.Diffuse involvement of the entire colon either with serpiginous polypoid lesions or with diffuse ulcerations; variant often identified in patients with compromised immunity, including the patients with renal transplant, immunosuppressive drug therapy and hereditary immunodeficiency.A single mass or in association with colonic neoplasms or adenomatous polyps. The incidence of colon malignancy associated with intestinal malakoplakia can reach up to 35% ,11.Three types of pathologic findings are described by endoscopic examination:malignant tumors,miliary tuberculosis andCrohn’s disease.Microscopically, the malakoplakia may be similar to:the Whipple disease,ceroid-like colonic histiocytosis,Wolman disease,Chediak-Higashi syndrome,sarcoidosis, orother granulomatous diseases .Colonic malakoplakia can mimic other pathologies, both macroscopically and microscopically and it is very important that before deciding the right management for the patient, the medical team must also take into consideration the differential diagnosis. The differential diagnosis after macroscopic examination may include: antibiotic therapy using agents that interact directly with the macrophages, like the quinolones or trimethoprim-sulfamethoxazolediminution or even suspension of the doses prescribed for the immunosuppressive therapya good control of the associated immunosuppressive pathologyendoscopic resections for the cases with localized lesionssurgical resection for the cases that present multiple organ infiltration and or suspected malignancy ,38,39,40The therapeutic management of the Malakoplakia varies from the medical therapy that includes antibiotics and surgical approach with different surgical techniques associated in the relationship with the patient’s needs. At this moment, there is no therapy considered the gold standard for this rare disease. Therapeutic alternatives are:In conclusion, Malakoplakia is an uncommon and rare granulomatous disease that can compromise any organ or structure in the human body. It is mostly encountered in the urinary tract and the gastrointestinal system being the second most frequent site involved, particularly the descending colon, sigmoid and rectum.The Michaelis-Gutmann bodies are pathognomonic for this disease, but they may be missing in the third stage of the Malakoplakia, when there is a progressive fibrous tissue, which may lead to bowel obstruction. The cases described in the literature state that this pathology affects the patients that have chronic diseases and immunosuppressive conditions. Due to the fact that the symptoms are non-specific, malakoplakia is generally identified postoperatively, from the histopathological analysis of the surgical specimen, just like the case that we illustrated.A good knowledge of this uncommon pathology may lead, in the future, to a preoperative diagnosis, less invasive investigations and may also avoid unnecessary surgical management.


Sudden onset of severe headache is the most common presentation of a ruptured intracranial aneurysm. Similar symptoms can be caused by pituitary apoplexy, and radiological examination is needed to distinguish between the two. Development of infarction and/or haemorrhage of the hypophysis with concomitant unruptured cerebral aneurysm has been described. However, intratumoural aneurysm within a pituitary adenoma presenting with the ictus of both pathologies is extremely rare.A 64-year-old man presented with classic symptoms of pituitary apoplexy. His symptoms improved after a few days, and he was discharged. However, he subsequently developed further episodes of sudden and severe headache together with visual decline and ophthalmoplegia. Radiographs demonstrated an enlarging sellar mass with suspicion of a ruptured internal carotid artery aneurysm, within the apoplectic pituitary macroadenoma. Although an endovascular procedure was planned, the patient developed massive subarachnoid haemorrhage resulting in brain death.This case report describes an intratumoural aneurysm of the cavernous internal carotid artery as a potential cause or result of pituitary apoplexy. Despite its rarity, this possible life-threatening complication of pituitary apoplexy should be recognised for prompt diagnosis and early management. Pituitary apoplexy is characterised by infarction and/or haemorrhage of the pituitary gland or adenoma. Patients typically present with abrupt onset of severe headache, visual disturbance, and hypopituitarism, which may be life-threatening . RadiogrA 64-year-old man presented with a sudden onset of severe headache and vomiting. According to the report from an outside hospital, his initial neurological examination was only remarkable for bitemporal hemianopia. A non-contrast computed tomography (CT) scan of the brain showed a pituitary adenoma (PA) with haemorrhage Fig. A. MagnetBecause of the lack of equipment at the local facility, the patient was transferred to our hospital for a transsphenoidal procedure 7 days after the first onset of headache. Because of the peculiar enhancement noted in the contrasted CT scan from the referring hospital, an ICA aneurysm was indeed probable. Hence, CT angiography was performed shortly after his arrival at our hospital. This imaging examination demonstrated a 22- x 23- x 30-mm lobulated arterial-enhancing outpouching lesion within a 26- x 34- x 46-mm apoplectic PA , a pseudoaneurysm, or false aneurysm, is an abnormal outpouching or dilation of an artery that is only bounded by the outermost layer of the arterial wall (tunica adventitia). Because of the poor support of the pseudoaneurysm wall, it is unstable and thus poses a higher risk of rupture than a true aneurysm of comparable size. Additionally, there is a risk of growth and rupture due to the pulsatile inflow and outflow of blood through the neck of the pseudoaneurysm. Although rare, death may occur if an intracranial pseudoaneurysm remains untreated . The cauIn conclusion, we have herein described the development of an intratumoural pseudoaneurysm of the cavernous ICA as a potential cause or result of pituitary apoplexy. The possibility of a complicated aneurysm should be considered in apoplectic patients with recurring episodes of severe headache with a crescendo pattern and a mass effect to the surrounding structures.


Situs inversus is a scarce congenital anomaly. Situs inversus totalis (SIT) is a mirroring of the normal. Thoracic and abdominal viscera transposition is a characteristic feature of situs inversus. It is considered to be a premalignant condition. This uncommon genetic disorder is often identified incidentally during thoracic and abdominal imaging. The coexistence of SIT and gastric cancer is rare. Because this anomaly is known to have associated anatomical and vascular anomalies, due care is required to identify it preoperatively and during the surgical procedure. At centers with prior experience, consistent with oncological practices, open surgeries, laparoscopic surgeries, and robotic surgeries can be done.We present a patient with a stomach adenocarcinoma with SIT who underwent distal gastrectomy with gastrojejunostomy along with resection and anastomosis of the transverse colon and capecitabin-oxaliplatin chemotherapy. The postoperative course was favorable. To our knowledge, only 13 cases of diffuse-type gastric cancer in a patient with SIT have been reported in the English-language literature. Gastric adenocarcinoma (GAC) is the 15th most common cause of cancer mortality in males and the third most common cause of cancer death globally . BecauseSitus inversus is a rare condition with an incidence of one in 10,000 live births. The male gender is affected more commonly than females. Aristotle was the first to describe this condition in animals. By the start of the 16th century, Fabricius identified the first human case of transposition of the liver and spleen. After 50 years of this discovery, Riolan, principal of the University of France, described two patients. Marco Severino is credited with the identification of the first case of dextrocardia in the early 16th century . KüchenmAllen described the case of a 30-year-old male having SIT with malignancy of the gastric region in the prepyloric area who expired a few weeks postoperatively. Here, we present a case with situs inversus with gastric malignancy who underwent gastric resection by an open method. In the early 2000s, synchronous presentation of situs inversus and stomach malignancy managed laparoscopically was documented first. In 2012, a patient with the synchronous finding of situs inversus and stomach malignancy was operated on with robotic assistance for a partial gastrectomy procedure . In the The coexistence of SIT and gastric cancer is rare. As the condition is associated with anatomical and vascular anomalies, detailed preoperative imaging is important to embark on definitive surgery such as gastric resection.A 48-year-old female visited the outpatient department with a complaint of severe pain in the lower abdomen for 20 days which was progressive. A history of vomiting was also present for 15 days. A careful physical examination revealed evidence of dextrocardia. The physician advised upper gastrointestinal endoscopy (UGE) and contrast-enhanced computed tomography (CECT) of the abdomen, along with other supportive blood investigations. UGE showed evidence of friable proliferative growth at the pylorus occluding total lumen, suggestive of gastric outlet obstruction (Figure A physician referred the patient to the oncology department for further management. The treating surgeon discussed the case in the tumor board meeting, where it was decided that surgery would be followed by chemotherapy. Hence, the surgeon obtained consent from the patient and relatives after consultation. The patient underwent distal gastrectomy with gastrojejunostomy along with resection and anastomosis of a part of the transverse colon as the transverse colon was infiltrated by a pyloric mass Figures , 4. PostAlthough SIT can be a hereditary disorder, the reasons behind it are not known. The present case of stomach carcinoma with SIT was operated by the open approach posing anatomical difficulties as the primary organs were a mirror image of their standard placement. Extensive lymph node dissection was conducted in the opposite way. A similar anatomical challenge was encountered by Yoshimoto et al., though they operated on the patient laparoscopically .Cao et al. documented a patient with poorly differentiated adenocarcinoma with SIT at the gastroesophageal junction . The preConsidering difficulties in arriving at a diagnosis by histopathology, it is not always a gold standard investigation in a case of stomach carcinoma. Immunohistochemistry is needed as gastric leiomyoma and gastric schwannomas are important differential diagnoses, as observed by Mulita et al. in their study .The presence of primary gastric cancer in conjunction with situs inversus is a unique clinical scenario. Even though the simultaneous appearance of gastric mucinous adenocarcinoma and SIT is a rare anatomopathological entity, all clinicians should be familiar with making an appropriate and precise diagnosis that will lead to quick and right therapy. It has been frequently documented that operating in such cases can prove very challenging even for experienced physicians due to extensive anatomical variations. There is also extensive proof of treating such cases with surprisingly positive outcomes. A thorough preoperative screening with various imaging modalities should be included in the prerequisites for this condition to minimize morbidity and mortality and decrease the pathological burden on society, especially in developing countries. Numerous approaches can be successfully used to treat such patients, such as robotic surgeries, open surgeries, and laparoscopic surgeries.


Transcatheter arterial chemoembolization (TACE) is a widely adopted treatment for advanced stage hepatocellular carcinoma (HCC). Nevertheless, several complications may occur, such as hepatic artery injury, nontarget embolization, pulmonary embolism, hepatic abscess, biloma, biliary strictures, and hepatic failure. However, bronchobiliary fistula is rarely mentioned before.A 65-year-old man with HCC underwent the TACE procedure, and then he encountered fever, dyspnea, abdominal pain, and abundant yellowish purulent bronchorrhea.Bronchobiliary fistula was diagnosed based on the computed tomography (CT) scan of his chest, which revealed the right lower lobe of his lung was connected to a hepatic cystic lesion.Percutaneous transhepatic cystic drainage was performed, and we obtained yellowish bile, showing the same characteristics as the patient’s bronchorrhea.We kept drainage of his biloma and provided supportive care as the patient wished. Unfortunately, the patient passed away due to progressive right lower lobe pneumonia 2 weeks later.This case exhibits a typical CT scan image that was helpful for the diagnosis of post-TACE bronchobiliary fistula. Post-TACE bronchobiliary fistula formation hypothesis includes biliary tree injuries with subsequent biloma formation and diaphragmatic injuries. Moreover, the treatment of bronchobiliary fistula should be prompt to cease pneumonia progression. Therefore, we introduce this rare complication of post-TACE bronchobiliary fistula in hopes that future clinicians will keep earlier intervention in mind. Among these, hepatocellular carcinoma (HCC), which is highly related to the infection of hepatitis B virus and hepatitis C virus, is the most common type of liver cancer. Transcatheter arterial chemoembolization (TACE) has been widely used as a curative treatment for intermediate-stage HCC, which are unsuitable for resection. However, despite its significant anti-tumor effects, the TACE procedure may induce several complications, such as hepatic artery injury, nontarget embolization, pulmonary embolism, hepatic abscess, biliary strictures, hepatic failure, and intrahepatic biloma formation.,3 Previous research demonstrated that conservative percutaneous drainage could manage biloma and have a good prognosis in most cases. In this case report, we share a rare complication, bronchobiliary fistula related to TACE-induced biloma that failed with conservative drainage.Liver cancer is a worldwide disease and has been estimated to exceed 1 million cases by 2025.®) as a systemic treatment. Computed tomography (CT) scan 2 months after the last TACE demonstrated a cystic lesion with surrounding daughter cysts without ring enhancement developed in S7, where the previous HCC was located. Thus, biloma was highly suspected, but no symptoms and signs were noted at regular follow-up. A month later, he encountered sudden onset of dyspnea accompanied by fever and yellowish sputum for a day. As a result, he sought medical help at another hospital, where he was treated as liver abscess and pneumonia. Percutaneous abscess drainage was placed. The bacterial culture from drainage revealed Citrobacter koseri. Piperacillin/tazobactam and meropenem were prescribed for 5 days. However, there were no significant clinical improvements, except for his fever. Subsequently, the patient was transferred to our hospital.A 65-year-old man with chronic hepatitis C was diagnosed with hepatitis C-induced HCC. Complete staging revealed AJCC staging cT4N1M0, Barcelona clinic liver cancer stage C. He went through 6 cycles of TACE and received two courses of palliative radiation therapy for HCC in the S4 and S6 of the liver in the past 15 months. Besides, he took Sorafenib , and intrahepatic bile duct dilatation.,6 Among these, bilioptysis is the most common symptom in almost all these patients. The image finding, the fistulous tract connecting pleural effusion and biliary tracts, was rarely seen on image scans (magnetic resonance imaging or CT) in previous reports, namely 2 out of 11 patients. Other diagnostic interventions include percutaneous cholangiogram, magnetic resonance cholangiopancreatography, 99mTcMebrofeninscintigraphy, sputum examination for bilirubin, and contrast-enhanced CT study can also help.,8 In our case, yellowish purulent bronchorrhea (serous in consistency) gave us a high suspicion of bilioptysis. Furthermore, the visible diaphragmatic defect on the CT scan can also confirm the diagnosis. Besides, an air-fluid level in the bronchial cyst proved the connection to the bronchus. The before mentioned are markable features on CT that can support the diagnosis of BBF.In a review article, a patient diagnosed with BBF usually bases on clinical symptoms. Therefore, the damage to the intrahepatic bile ducts will lead to biloma formation.,9 The increased intracavitary pressure in biloma possibly drives bile into the chest. Furthermore, infection, thermal injury by radiofrequency ablation, and surgical damage to the diaphragm provide diaphragmatic injuries that contribute to the development of BBF.,10,11 As in our case, even though the TACE procedure did not directly injure the diaphragm, bacteria isolated from the biloma indicated the infection status and may contribute to diaphragmatic injuries that form BBF.Although the etiology of BBF formation after TACE remains unclear, some explanations exist. Iodized oil or other embolic materials used in the TACE procedure may cause ischemia in the peribiliary capillary plexus. However, in some cases with conditions such as ongoing bile leaks may require surgical fixation.,6,12,13 There is no official guideline for the treatment of bronchobiliary fistula. However, a systematic literature review suggests open surgery should be the first choice after interventional techniques have failed or when BBF is secondary to tumors and biliary obstruction. Returning to our case, the patient underwent the intervention of percutaneous drainage, but it failed to cease the deteriorating condition. Therefore, we consulted a chest surgeon for fistula ligation. Nevertheless, surgical intervention was postponed due to his progressive dyspnea.The successful management of BBF is mostly drainage and close monitoring.In conclusion, despite the slime possibility, the TACE procedure might induce the formation of biloma and may subsequently cause BBF in a patient with a diaphragmic injury. Its unique presentation of bilioptysis and the fistula track on CT scans can be a way to confirm the diagnosis of BBF. This case reminds us to be aware of the possibility of BBF formation after the TACE procedure, which can be a fatal complication. Moreover, patients may benefit from earlier diagnosis and earlier surgical intervention.Yuan-Chun Lo and Ping-Wen Hsu contributed to writing the manuscript. Fatt-Yang Chew contributed to image interpretation. Hung-Yao Chen was responsible for patient care and manuscript preparation. All authors read and approved the final manuscript.Conceptualization: Yuan-Chun Lo, Ping-Wen Hsu, Hung-Yao Chen.Data curation: Yuan-Chun Lo, Ping-Wen Hsu.Formal analysis: Yuan-Chun Lo.Project administration: Hung-Yao Chen.Software: Fatt-Yang Chew.Supervision: Hung-Yao Chen.Validation: Ping-Wen Hsu, Fatt-Yang Chew, Hung-Yao Chen.Writing – original draft: Yuan-Chun Lo, Ping-Wen Hsu.Writing – review & editing: Yuan-Chun Lo, Ping-Wen Hsu, Hung-Yao Chen.


Pancreatic endometriosis is extremely rare with only 14 cases reported in themedical literature and its diagnosis on radiological imaging poses a greatchallenge. We report a case of a 31-year-old female patient with recurrentadmissions for pancreatitis of unknown aetiology and no relevant previousmedical history. Sectional imaging showed a cystic lesion in the tail of thepancreas and the diagnosis of a post-pancreatitis pseudocyst or a less likelypre-malignant mucinous cystadenoma was considered.On post-robotic resection of the pancreatic cyst, the histopathology analysis waspositive for endometrial stroma. Pancreatic endometriosis although rare shouldbe considered as a differential diagnosis for cystic lesions especially inpatients who are known to have pelvic endometriosis. Nevertheless, the goldstandard for the definite diagnosis of pancreatic endometriosis remainshistopathological. It is a common disease and can be seen in almost 5 to 10% of females in thereproductive age group. It is also seen in approximately 5% of postmenopausal females especially inpatients on estrogen hormonal replacement therapy. Endometriosis has been linked with a genetic predisposition with findings ofestrogen dependence and progesterone resistance. It is often associated with an inflammatory reaction and resultant fibroticchanges at the site of the endometriotic deposits.Endometriosis is defined as the presence of endometrial glandular tissue outside theuterine cavity.A 31 years old female patient presented to the emergency departmentcomplaining of epigastric pain associated with nausea and vomiting. She has a pastmedical history of recurrent episodes of acute pancreatitis and was treatedpreviously in another healthcare facility where no definitive aetiology was found.No other significant past medical history was noted and no cyclical pattern of painwas observed. No previous surgical history was disclosed nor family history ofpancreatic diseases or neoplasms was given.On physical examination, the patients vital signs were normal. Chest was noted to beclear. Abdomen was soft except for mild tenderness in the epigastric region.Laboratory blood work-up showed elevated pancreatic enzymes indicative ofpancreatitis. The patient was admitted as a case of acute pancreatitis for furtherinvestigation and the treatment was initiated.A computed tomography (CT) scan of the abdomen and pelvis with IV contrast was donetwo days after admission and showed no sign of acute pancreatitis, no peripancreaticfatty striation or calcifications. However, a unilocular cystic lesion was seen atthe tail of the pancreas measuring approximately 3.6 × 3.3 cm indiameter. Slight atrophy of the distal tail beyond the cyst was notable. No evidenceof mesenteric or retroperitoneal lymphadenopathy was seen. Major vasculatureappeared intact. Gadolinium-enhanced magnetic resonance imaging (MRI) of the abdomen was performed andthe results again showed a cystic lesion at the tail of the pancreas which appearedhypointense on T1 FATSAT in comparison with the pancreatic parenchyma andhyperintense on T2 HASTE sequences. There was no restriction of the cystic lesion ondiffusion weighted imaging (DWI) and apparent diffusion coefficient (ADC). Minimalcontrast enhancement of the wall on post-contrast T1 FATSAT images was depicted. Nogradient echo sequences were obtained. These findings on diagnostic imaging in addition to the patients history of recurrentpancreatitis suggested a diagnosis of a pseudocyst as a complication ofpancreatitis. Nonetheless, a mucinous cystic neoplasm was also included as adifferential diagnosis considering the location of the cyst, age, and gender of thepatient with the recurrent episodes of pancreatitis possibly due to obstruction toupstream pancreatic duct by the cystic lesion.−1) levels and was negative formalignant cells. CEA levels above 200 ng ml−1 raisesuspicion for a mucinous neoplasm, while amylase levels above200 U l−1 raise suspicion of a post-pancreatitis pseudocyst.The patient underwent an upper esophagogastroduodenoscopy and ultrasound-guided fineneedle aspiration of the cystic fluid was done which showed elevated CEA (370 mcg/L)and amylase  and 81% negative predictive value (NPV), while T2dark spots has a 93% sensitivity, 45% specificity, 72% PPV and 81% NPV.On MRI, ovarian endometriomas have a characteristic homogeneous T1 hyperintensity anda low T2 signal intensity. There may be heterogeneity to the T2 hypointensity andthis is referred to as shading and is due to different stages of degradation of theblood products as a result of cyclical episodes of bleeding. Another more specificsign of ovarian endometriomas is called the T2 dark spot sign which appears asdiscrete markedly hypointense foci in the cyst on The endometrioma in our case had atypical imaging features with low T1 signal andhigh T2 signal and therefore was not considered as a differential diagnosis. Patients with pancreatic endometriosis usually present with epigastric painand may be admitted with acute pancreatitis or acute abdomen. The cyclical catamenial nature of symptoms and a past medical history ofendometriosis may aid in suggesting the diagnosis. In addition to the frequently changing morphological findings and signalalterations on sectional imaging may also be helpful.Pancreatic endometriosis was first described in the literature in 1984 and isextremely rare with only 14 cases reported up to the current date. Cystic lesions in the pancreas could be classified into benign, premalignantand malignant cysts. The differential diagnosis includes post-pancreatitis pseudocysts, mucinouscystic neoplasms, serous cystadenomas or cystadenocarcinomas, cystic pancreaticadenocarcinomas, cystic neuroendocrine tumors and pseudopapillary tumors as well asectopic tissue such as endometrial cysts.The wide use of sonography, computed tomography, magnetic resonance imaging andpositron emission tomography PET resulted in a rise in the incidental recognition ofpancreatic lesions in asymptomatic patients.Pancreatic pseudocysts typically form within 6 to 8 weeks after an episode of acutepancreatitis. The imaging features include a round fluid-filled collection and thickenhancing fibrotic wall with no septations, wall calcifications or intracystic solidcomponents. On cystic fluid analysis elevated amylase and lipase levels are commonlyencountered with normal CEA levels.Mucinous cystic neoplasms typically occur in middle-aged female patients and areconsidered premalignant. Approximately 90% are notable at the pancreatic tail andcan reach a size of 20 cm in diameter at the time of diagnosis. They aremultilocular and internally-septated with a smooth enhancing contour and wallcalcifications. These neoplasms are lined with mucinous epithelium with varyingdegrees of dysplasia resting on a layer of cellular ovarian-like stroma which mimicsendometrial stroma and is usually positive for inhibin, both oestrogen andprogesterone receptors but negative for CD 10 on immunochemistry tests which is acommon marker for endometrial stroma.Elevated cyst CEA levels are more common with mucinous cystic neoplasms thannon-mucinous lesions and amylase levels are typically normal. However, imaging is usually non-specific due to overlapping features withpremalignant pancreatic lesions especially with the more common mucinous cystic neoplasms. Pancreatic imaging may show cystic lesions of a variable size and complexitywith varying degrees of hemorrhagic components. Magnetic resonance imaging may depict bleeding which appears as hyperintensefoci on T1 sequences with/without fat saturation. In the absence of intralesionalhaemorrhage, hypointense T1 and T2 image signal is usually seen.Diagnostic imaging and laboratory tests are valuable tools in the initial assessmentof pancreatic endometriosis. Nevertheless, hemorrhagic components are extremely uncommon in pancreaticmucinous tumors. Pancreatic endometriomas may show elevated cyst CEA levels as well aspositive CD 10 immunohistochemical marker which is used to denote endometrial stroma. Laparoscopy and histopathological analysis remains the gold-standard for thedefinitive diagnosis of pancreatic endometriosis.Up to the current date no typical diagnostic imaging features of pancreaticendometriosis have been established as only a few cases have been reported in the literature. If preoperative suspicion of malignancy cannot be ruled out then distalpancreatic resection with or without splenectomy is recommended.Ultrasound-guided fine needle aspiration is not recommended if mucinous cysticneoplasms are highly suspected due to the risk of seeding which can causepseudomyxoma peritonii. Thorough diagnostic investigations including intraoperative frozen sectionhistopathology may avoid extensive surgical resection leading to complications likepancreatic insufficiency. Concomitant evaluation of the pelvic cavity is essential as isolatedextrapelvic endometriosis is rare.Definitive diagnosis and treatment of pancreatic endometriosis usually requires amultidisciplinary approach including pancreatic surgeons and gynecologists with thecareful surgical excision of all endometrial lesions.In conclusion, pancreatic endometriosis is extremely rare but should be considered asa differential diagnosis for cystic lesions especially in patients who are known tohave pelvic endometriosis. Nevertheless, the gold standard for the definitediagnosis of pancreatic endometriosis remains histopathological due to theoverlapping radiological imaging features with other pancreatic cystic lesions.Endometriosis is a fairly common disease occurring in up to 5–10% ofpremenopausal females and 5% of postmenopausal females.Pancreatic endometriosis is extremely rare and often mimics other pancreaticcystic lesions including pseudocysts, premalignant and malignant cysts.Clinical features, radiological imaging and laboratory work-up are essentialand may aid in suggesting the diagnosis. However, for a definitive diagnosissurgical resection and histopathology are the gold standard as theradiological image findings of these lesions overlap with other cysticlesions especially premalignant and malignant mucinous neoplasms.


Primary cardiac angiosarcoma is extremely rare, and its prognosis remains poor, with a mean life expectancy of only a few months. Here, we report a case of primary cardiac angiosarcoma.A 49-year-old Japanese woman with a month-long history of dyspnea was admitted to our hospital for pericardial effusion. Chest computed tomography and cardiac magnetic resonance imaging showed a mass in the right atrium. The patient underwent surgical resection of the tumor, and the pathological diagnosis was angiosarcoma. The patient received radiotherapy after surgery. Six months following surgery, she underwent chemotherapy following the diagnosis of lung metastasis. The patient died 18 months after the initial diagnosis.Cardiac angiosarcoma is rare and difficult to diagnose early because it is associated with few symptoms. Moreover, there are currently no established guidelines for the treatment of this disease because of its rarity and sparse descriptive literature Therefore, multidisciplinary therapies should be considered, including surgery, radiotherapy, and chemotherapy.There is no standard treatment for cardiac angiosarcoma, but surgical resection, chemotherapy, radiation therapy, or a combination of these therapies may be useful. •Pericardial fluid cytology has little value in diagnosing cardiac angiosarcomas.•Imaging studies are essential for the diagnosis of primary cardiac angiosarcomas.•Surgery and radiochemotherapy extended survival to up to 18 months. This case report is compliant with the SCARE Guidelines 2A 49-year-old woman with a 1-month history of dyspnea was admitted to our hospital. Her medical history included hypertension, hyperlipidemia, and uterine fibroids. Chest computed tomography (CT) and echocardiography revealed a large epicardial effusion A. The paPreoperative transesophageal echocardiography, performed under general anesthesia, showed a mass measuring 28 × 41 mm originating from the free wall of the right atrium. It was not adherent to the tricuspid valve or septum and was thus resectable. Upon pericardiotomy, the tumor was visualized in the right atrium, adherent to the pericardium. The tumor was highly vascularized and had rough borders . Cardiop3Primary cardiac tumors have an incidence of 0.001–0.03 % on autopsies, and are benign in approximately 75 % of cases. Among the remaining 25 % of tumors, sarcomas are the most common Imaging studies are essential for diagnosing these tumors. They aid in determining tumor size, location, and relationship to the pericardium or valves. These tumors appear hyperintense or isodense to the myocardium on T1-weighted or proton density-weighted MRI. On T2-weighted imaging, they appear hyperintense due to increased vascular flow Due to the rarity of cardiac angiosarcomas and the lack of literature, there are currently no established guidelines for its treatment. However, multidisciplinary therapies, including surgery, radiotherapy, and chemotherapy, are most commonly employed. If the tumor is localized, surgical resection is the mainstay treatment and is useful for the final diagnosis. Unfortunately, many patients are in the advanced stages of angiosarcoma by the time of diagnosis, and the tumors are typically too large to be amenable to resection. The median survival for those who underwent complete surgical excision was 17 months, whereas it was 6 months for those who could not achieve complete remission The use of adjuvant therapies, including chemotherapy and radiotherapy, is debatable. No randomized trials have been performed to provide evidence for these adjuvant therapies. Furthermore, since cardiac angiosarcomas are rare, most available treatment modalities are for extra-cardiac angiosarcomas. The traditional cytotoxic chemotherapy drugs for angiosarcoma are anthracycline, ifosfamide, and taxanes (paclitaxel and docetaxel) 4Herein, we present a case of primary cardiac angiosarcoma that was managed with surgical resection and chemoradiotherapy. Cardiac angiosarcomas are rare and difficult to diagnose in advance because they are associated with few symptoms. They have no standard treatment; however, surgical resection, chemotherapy, radiotherapy, or a combination of these therapies may be useful.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.This study was granted an exemption from full ethical review, since clinical pictures of the patient or any information that could directly identify the patient were not used.Data are available upon request.Written informed consent was obtained from the patient's family for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.A.K., T.T., and O.K. performed the surgery and patient care postoperatively. S.T. managed the patient for chemotherapy and radiotherapy. The first draft of the manuscript was written by A.K. All authors have read and approved the final manuscript.This case report is not “First in Man” study.Akihiro Kobayashi.Not commissioned, externally peer-reviewed.The authors declare that there is no conflict of interest regarding the publication of this article.


Congenital defects in the tentorium cerebelli are quite rare occurrences and are often too small and asymptomatic. This is a case report of a female patient aged 11 years, complaining of headache, vertigo, and vomiting. Her computed tomography (CT) images show transtentorial herniation of occipital gyri across a developmental defect involving the anterior free margin of the tentorium cerebelli. Similar cases have been reported in the past as "incidental" and "potentially symptomatic" findings, and in at least one case as a proven pathological findings. Our case is unique in terms of the asymmetric bilateral configuration and comparatively larger size of the defect. We have included a review of the existing medical literature in order to derive learning points for the betterment of our understanding of a rare entity that can have significant implications. The principal function of the tentorium cerebelli is to prevent direct contact between the cerebral and cerebellar cortices . It alsoOur patient is a girl aged 11 years, complaining of headache, intermittent vertigo, and some episodes of vomiting for approximately a month. The patient is otherwise normal in demeanor as appropriate for her age, with normal involvement at school, games, and social activities. She did not complain of any visual or auditory difficulties and does not need to wear spectacles or hearing aids. Her birth had been without complications, followed by a normal growth pattern. She had received vaccines according to national immunization schedules and is not under any regular medications at present. Bedside neurological examinations were normal, including gait, balance, cranial nerve functions, visual acuity, and visual fields on both sides. As such, computed tomography (CT) was advised to rule out whether her symptoms are being caused by cerebral edema or any underlying optic pathway lesions.The CT shows an asymmetric defect involving the anterior free margin of the tentorial notch, spanning both sides of the midline. The defect measures 25 mm on the left side and 8 mm on the right. It allows inferomedial herniation of the precuneus and cuneus of the left occipital lobe Figure , along wAlso, there is about an 8 mm rightward deviation of the adjacent falx cerebri Figure . FurtherWhile occipital lobe herniation is mostly considered asymptomatic and unrelated with clinical features, there is at least one case report  where thHerniation occurs across the defect due to the pressure difference, which allows the herniated part to occupy a cistern or to push against another normal structure ,6. Such In our patient, the defect has quite a significant size (2.5 cm) on the right side, which allows direct contact between the occipital and cerebellar cortices. However, we could not make a size-by-size comparison with previous case reports, since most of them did not mention any measurements. Sun et al.  only staIn addition to having a defect that is quite large, our patient is also unique in terms of the defect being bilateral and asymmetric, whereas almost all the other case reports have mentioned unilateral findings -4,6. We Similar to ours, the report by Thomaere et al.  describeIt is also interesting to note that the first patient report of a congenital tentorial defect was published only in 1976 ,8, whereFocal cortical herniation due to isolated tentorial hypoplasia is most often a benign entity with no complications. However, CT or MRI images thereof may sometimes mimic critical pathologies like space-occupying lesions or underlying intracranial hypertension, since the herniated parenchyma occupies a cistern or pushes against another normal structure. It is thus important to remember in this context that such a herniated portion is essentially normal brain parenchyma and would exhibit a normal appearance on CT and all sequences of MRI, whereas pathological tissue would differ from normal in one sequence or another. Radiologists and clinicians need to remind themselves of this small basic principle, so as to avoid sending the patient for additional imaging or invasive procedures.Also of importance is the fact that while most herniations are asymptomatic due to very small size, those that are slightly larger may have nerve tracts compressed within the herniated brain matter. This can produce infarct or mass-effect-like symptoms. Therefore, it may be a good practice to make the imaging evaluation complete by means of MR tractography by DTI sequences, which can detect whether any nerve tracts are trapped or compressed. The use of DTI and visual field assessment would be particularly important if a patient presents with relevant symptoms, such as blurred vision, diplopia, vertigo, or vomiting.


The iliac vein is an extremely rare site of metastasis for extraskeletal mesenchymal chondrosarcoma (ESMC). Involvement of the veins usually leads to an extremely dismal prognosis. Here, we report a 50‐year‐old patient with retroperitoneal mesenchymal chondrosarcoma and initial metastasis to the iliac bone, which further progressed to involve the iliac vein. In this study, we reviewed the major characteristics of ESMC and the previously reported cases, considering the rarity of these tumors. Extraskeletal mesenchymal chondrosarcoma, a rare malignant neoplasm, has high local and metastatic recurrence rate and poor survival. We report here a rare case of iliac vein metastasis in ESMC. Surgical removal of the lesion remains the “gold standard” of treatment for localized tumors. For unresectable, advanced lesions, palliative chemotherapy is recommended. Chondrosarcoma is the third most prevalent bone malignancy after myeloma and osteosarcoma.2A 50‐year‐old woman with previously diagnosed retroperitoneal mesenchymal chondrosarcoma was referred to our hospital with the chief complaint of progressive left leg pain and swelling and paresthesia. The diagnosis of ESMC was made elsewhere during workup evaluation for sudden onset lower extremity deep vein thrombosis (DVT) with MRI findings of multiple T1, hypointense tissue lesions in the sacrum, femur and pelvic bones involving the bilateral iliac and pubic bones, left ischium with extension into the right acetabulum, right femoral head and ipsilateral lesser trochanter as well as proximal shaft of the femur consistent with chondrosarcoma. Histologic examination of the lesion biopsy was in favor of ESMC. The patient received five cycles of doxorubicin + ifosfamide regimen and was advised for surgical removal of the lesion, which was unsuccessful due to poor treatment compliance after discharge. Six months after the termination of last chemotherapy session, she referred to another hospital due to worsening of low back pain. Pelvic and lumbosacral MRI with Gadolinium revealed previous lesions along with evidence of bony metastasis involving the L3, L4, S1 and S2 vertebrae. This time, the patient was ordered four cycles of combined chemotherapy regimen  on Ewing's Sarcoma‐like protocol. During hospitalization, the patient developed COVID‐19 pneumonia and chemotherapy was discontinued for 2 months. Two months after hospital discharge, she referred to our hospital with progressive lower limb paresthesia and MRI evidence of enlarging previous lesions. Electromyography (EMG) and nerve conduction velocity (NCV) were performed to evaluate the cause of paresthesia, which showed early phase peripheral polyneuropathy due to chemotherapy. Considering the previous chemotherapy‐related adverse effects and lack of clinical response to earlier chemotherapeutic drugs, the patient received single‐agent gemcitabine regimen . Clinical improvement was achieved as resolution of low back pain and paresthesia. However, four months later, she referred to our emergency department due to progressive constant low back pain unamenable to various analgesics. She also had limb edema, which started one week before her admission, and she lost her ability to walk on her side. On the physical examination, no ulceration or pigmentation was observed. No lymphadenopathy was found in the inguinal region. She has tender 4+ nonpitting edema on her left leg in conjunction with tactile tenderness of her lumbosacral region. Her right leg had limitations in mobility and movement. The ankle‐brachial index was 1.02 in the left leg and 1.04 in the right leg.The computed tomography (CT) images Figure  displaye3Extraskeletal mesenchymal chondrosarcoma (ESMC) comprises a rare type of sarcoma originating from soft tissues, more frequently the meninges of the cranial and spinal cord, extremities, orbit and the lower extremities, particularly the thighs. Rarely, this tumor may arise from the retroperitoneum, kidneys, pancreas and hand musculature.Retroperitoneal mesenchymal chondrosarcoma has been reported rarely in the literature. In a cross‐series study of Ghafoor et al.Extraskeletal mesenchymal chondrosarcoma displays a strong tendency to locally and distantly metastasize, which makes the clinical outcome extremely dismal, with a reported 10‐year survival rate of 7%–26%.Magnetic resonance imaging of ESMC often demonstrates equal or low signal intensity on T1W1 and heterogeneous hyperintense lesions on T2WI, as the intramural noncalcified and calcified regions of EMCS tend to have high and low intensity on T2WI, respectively, they are usually visible as areas of high signal intensity around areas with low signal intensity or the characteristic “salt and pepper” appearance.4The current study shows the first case of extraskeletal mesenchymal chondrosarcoma with metastatic involvement of the iliac vein. Extraskeletal mesenchymal chondrosarcoma represents a rare entity of highly aggressive tumors with a propensity to metastasize locally and distantly. Magnetic resonance imaging findings include hypointense lesions on T1W1 and heterogeneously hyperintense lesions on T2WI. Metastases are common and involvement of less commonly reported sites may also occur.Mahsa Masjedi Esfahani: Conceptualization; data curation; formal analysis; investigation; methodology; project administration; supervision; visualization. Seyed Mohammad Ali Mirazimi: Conceptualization; data curation; investigation; methodology; resources; software; writing – original draft; writing – review and editing. Javid Azadbakht: Data curation; formal analysis; resources; validation; visualization. Fatemeh Dashti: Conceptualization; data curation; formal analysis; investigation; methodology; resources; software; validation; writing – original draft; writing – review and editing.No funding or sponsorship was received for this study or publication of this article.None declared. This manuscript has not been submitted to, nor is under review at, another journal or other publishing venue. The authors have no affiliation with any organization with a direct or indirect financial interest in the subject matter discussed in the manuscript.Informed written consent was obtained from the patient for publication of this report and any accompanying images.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Ameloblastomas of jaws are benign odontogenic tumors of epithelial origin with four clinical variants: solid multicystic type, unicystic type, desmoplastic type, and extraosseous type. The incidence rate of ameloblastoma is 0.92 per million person-years. Unicystic ameloblastoma refers to those cystic lesions that show clinical and radiologic characteristics of an odontogenic cyst but shows a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumor proliferation on histological examination. Here is a unique case of unicystic ameloblastoma involving the mandible in a 70-year-old patient. The case was managed by segmental mandibulectomy and flap repair. Unicystic ameloblastoma accounts for only 13% of all known cases in scientific literature. Considering the rarity of the lesion, the purpose of presenting this report on a clinical case is to emphasize the importance of radiological evaluation and histopathological examination for the diagnosis of ameloblastoma. This report highlights the importance of radiological and pathological examination of any pathology in the jaw even if it seems innocuous.Ameloblastoma is a benign odontogenic tumour with an aggressive growth tendency and a high risk for malignant transformation and metastasis.A 70-year-old man visited our hospital with symptoms of painless swelling extending from the buccal mucosa of the submental region to the right angle of the mandible. The bulge first appeared intraorally 4 years ago, gradually growing in size and being accompanied by a thick creamy discharge. The patient started to consume tobacco and smoke cigarettes as a teenager. Besides, he has no insignificant past medical, family history, or dental injuries.After 4 years of tumour occurrence, the patient was admitted for a thorough examination and evaluation. On clinical examination, a solitary, hard, painless swelling measuring 4 x 4 cm extending from the submental area to the right-sided angle of the mandible was detected above the anterior edentulous alveolar ridge. The buccal vestibule was affected intraorally by its enlargement. The mass was fixed with the underlying structure but free from the overlying structure. There was no tenderness or regional lymphadenopathy. There was no local rise in temperature. The patient has poor oral hygiene and numbness of the lower lip on the right side. On the basis of history and examination, the provisional diagnosis was given as odontogenic tumour and the differential diagnosis was given as odontogenic cyst and bone tumour.The patient was explained about the procedure in detail and written informed consent was obtained. The patient was subjected to hematological and radiological examination. Radiographically in orthopantomogram, the lesion showed multilocular radiolucency with a mixed radiographic pattern of honeycomb appearance interspersed with a soap bubble pattern in the right mandible .Contrast-enhanced computed tomography (CECT) scan demonstrated a well-defined expansile mixed density cystic lesion in the body of the mandible measuring 3.6 x 3.9 cm in size. Additionally, mental foramen was not traceable on the right side. Enhancing areas were noted consistent with the solid portion of the tumour and the non-enhancing content inferiorly was consistent with the cystic portion of the tumour .Incisional biopsy was taken from the site of the lesion. On hematoxylin and eosin stained soft tissues, cystic lumens lined by ameloblastomatous flattened epithelial lining were found in diverse sites. The epithelium proliferated within the lining as well as into the connective tissue wall, forming ameloblastomatous islands. The cystic lining was formed from the basal layer of the palisading columnar epithelium with reverse polarity. The central portions had stellate reticulum cells, while the rest of the area showed hemorrhage. These features are suggestive of unicystic ameloblastoma of the mandible with bony erosion.Then the patient was referred to the Department of Oral and Maxillofacial Surgery where he underwent a major local excision with segmental mandibulectomy and flap repair. The bony repair was not done due to the advanced age of the patient. The patient's postoperative course was uneventful, and he was discharged after 14 days. The patient is under strict follow up and no recurrence has been observed so far.1 Broca published the first detailed description of ameloblastoma in the year 1868. There is no prelidiction for sex. The incidence is highest in the third and fourth decades of life, with the median age of 35.9 years at presentation, and an age range of 4 to 92 years.4 The mandible accounts for 80% of instances, the molars and ramus being the most prevalent sites. It presents clinically as a slow-growing, relatively painless expansile tumour that causes tooth mobility, dental displacement, and a deformed facial appearance if left untreated.1Ameloblastomas are benign odontogenic tumours that are slow-growing, persistent, and locally invasive. They have the ability to grow rapidly, resulting in bone deformities.7 On histological examination, the UA shows a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumour growth.7 In line with the findings reported by many case reports, the histology of this case also revealed soft tissues, cystic lumens lined by ameloblastomatous flattened epithelial lining which proliferated within the lining as well as into the connective tissue wall, forming ameloblastomatous islands.Unicystic ameloblastoma is a distinct variant of ameloblastoma that was first described by Robinson and Martinez in 1977. UA refers to those cystic lesions that show clinical and radiologic characteristics of an odontogenic cyst but shows a typical ameloblastomatous epithelium lining part of the cyst cavity, with or without luminal and/or mural tumour proliferation on histological examination.8 In our case, it is associated with the submental area to the right-sided angle of the mandible. It presented as painless swelling with facial asymmetry, dental impaction, tooth displacement, mobility, or tooth resorption.The lesion is most commonly observed on the crowns of mandibular third molar teeth, although it can also be present in the interradicular, periapical, and edentulous areas.9 Similarly, the radiograph of the lesion of this case also identified a well-defined expansile cystic lesion with a mixed radiographic pattern of honeycomb appearance interspersed with soap bubble pattern in the body of the mandible with enhancing thin septa, enhancing soft tissue components along with sclerosis and erosion of symphysis.Radiographically, UA may appear unilocular or multilocular with soap bubble or honeycomb appearance; buccal and lingual expansion of the cortex invariably accompanies ameloblastoma. Thinned and intact cortex shows egg shell appearance.10 In ameloblastoma, there has been a significant correlation between microvessel density (which depicts the vascularization of tumour tissue) and contrast enhancement characteristics in CT scans.11 As a result, contrast enhancement is one of the most essential characteristics of benign odontogenic tumours. Because the pattern of bone destruction in intraoral radiographs suggested a tumour rather than a cyst, we decided to use CECT to evaluate the enhancement pattern of the tumour.Ameloblastoma shows cystic areas of low attenuation and isoattenuating solid regions on computed tomography (CT). CECT scan shows an enhancement effect in the solid components.12 Radiotherapy should be considered in patients with positive margins in whom re-resection is not recommended and for those with incompletely resectable tumours. In our case, we performed local excision with segmental resection of the mandible with a safety margin of 1 cm to avoid recurrence and flap repair for reconstruction.Management of ameloblastoma can be done in three ways (1) conservative which includes enucleation and curettage, as well as the use of adjuvant therapies such as Carnoy's solution and cryotherapy, (2) marsupialization, and (3) radical surgery, which includes marginal or block resection  and immediate bone reconstruction. Iliac crest grafts or microvascular fibular flaps may be required for facial reconstruction procedures.3 For this reason, close and prolonged follow-up examinations should be performed. Conventional radiography and clinical examination may fail to detect recurrences early enough. CT is the most sensitive imaging technique for detecting ameloblastoma, and is to be recommended whenever possible.10 Likewise, the recurrence of the lesion in our case was detected by CECT and managed accordingly.For conventional ameloblastomas, recurrence rates of 50 to 90% have been observed following curettage and up to 15% after marginal or block resection.The case highlights the importance of early checkups, imaging, and biopsy for early diagnosis and management of ameloblastoma. Close and prolonged follow-up should be performed for detecting recurrences. Documentation of more such cases makes the pathogenesis of these lesions more insightful. Research is required to correlate the prognosis of these lesions.


Crohn’s disease (CD) is a chronic inflammatory bowel disease characterized by transmural inflammation occurring anywhere along the gastrointestinal tract. Intestinal malrotation is an embryological error resulting in an abnormal gut anatomy. Although these two conditions rarely present concurrently, it is important to identify their presence, which is challenging due to their nonspecific, overlapping symptoms. Here, we present two patients with concomitant CD and intestinal malrotation. Both patients’ conditions required surgical intervention, which was complicated due to their unique anatomy. Clinicians should be aware of the potential pit-falls that may occur due to the anomaly and thus require a full understanding of the anatomy. Accepted: 22 March 2022</p>Intestinal malrotation is a result of an error in the embryonic rotation of the gut. Normal intestinal anatomy develops as a result of 270° of counterclockwise rotation . This prCrohn’s disease (CD) has an incidence of 3–20 cases per 100 000. Its incidence has been steadily increasing, globally . This inThe combination of intestinal malrotation and CD is quite rare, with three cases previously described in the literature. Here, we present two cases of patients with previously diagnosed intestinal malrotation and concurrent CD who underwent surgery for CD complications at our institution and are presented in order to add to the global experience of this unusual condition.A 40-year-old male patient with a history of CD presented with increasing flares of abdominal pain and weight loss of 8 kg in the previous year. The patient has a known history of intestinal malrotation diagnosed at age 12 and a chronic small bowel obstruction. As a child he underwent a Ladd’s procedure.Magnetic resonance imaging (MRI) enterography conducted a year prior to the current admission demonstrated the entire colon was found to the left of the abdominal midline . MRI demDue to history of CD, malrotation and chronic small bowel obstruction the decision was made to resect the small intestine. Surgical exploration was performed to alleviate obstruction and for failure of response to steroids, biological treatment and exclusive enteral nutrition.The surgery commenced with laparoscopy under general anesthesia. Many adhesions from previous surgeries and malrotation were noted, with the colon found in the left side of the abdominal cavity and the small intestine on the right. Due to the degree of adhesions and malrotation, the procedure was converted to an open approach through a midline incision. The section of the ileum causing the obstruction was visualized in the distal ileum, which included severe stenosis, with nearly 8-cm dilated small bowel proximal to the stenosis .A Heineke–Mikulicz strictureplasty was performed on the proximal stricture. A length of 35 cm of the distal terminal ileum was resected and a side-to-side anastomosis to the proximal small bowel was performed.The immediate postoperative course was uneventful, and the patient was discharged 4 days following the surgery. The patient was readmitted 4 days after discharge with shortness of breath and fever. He was diagnosed with nosocomial pneumonia and was treated with piperacillin-tazobactam. Bowel function and alimentation returned to baseline.A 26-year-old male presented 6 months prior to his surgery with abdominal pain, fever, non-bloody diarrhea and 13 kg of weight loss. He was given a course of antibiotics and underwent a colonoscopy where ileo-colonic disease was discovered. He was diagnosed with CD and malrotation was incidentally discovered at this time.Two months prior to surgery and before initiating CD treatment, the patient was hospitalized. A computed tomography (CT) demonstrated a 30-cm region of marked thickening in the terminal ileum, an adjacent multilocular abscess measuring 4.3 × 3 × 2.5 cm and intestinal malrotation. The patient was managed conservatively with antibiotics and total parenteral nutrition (TPN). Drainage could not be performed due to the abscess location.A follow up colonoscopy performed 1 month after antibiotic treatment demonstrated cobblestone mucosa along with a severe stenosis in the terminal ileum and a small bowel fistula. In addition, an abdominal CT showed a 10-cm section of bowel wall thickening in the terminal ileum and remnants of the abscess . IleocecSurgical exploration via laparoscopy was performed. Intestinal malrotation was identified in addition to a 30-cm section of inflamed bowel in the terminal ileum. The laparoscopy was converted to open laparotomy as the blood vessels could not be correctly identified and the abscess was adhesing the bowel wall to the mesentery. Despite laparotomy, resection of the terminal ileum along with the cecum was difficult and the superior mesenteric vein (SMV) could not be identified. As a result of subsequent congestion of the small bowel, the proximal SMV was identified without continuation to the small bowel mesentery. It appeared that the congestion was due to dissection of the ileocolic veins disrupting venous drainage from the small bowel. The superior mesenteric artery was normal. The SMV root was then anastomosed to the inferior vena cava with a Gore-tex graft. Small bowel congestion improved after vascular reconstruction, but mild congestion persisted. The abdomen was closed and a second look surgery within 24 h was planned. The following day repeat laparotomy revealed a viable small and large intestine, and an end ileostomy was performed. The large bowel remnant was left closed within the abdomen.The immediate postoperative course included admission to the intensive care unit, where he remained intubated while undergoing intense postoperative monitoring due to the vascular aspect of the surgery. Postoperative treatment included 9 days of piperacillin-tazobactam on recommendation from the vascular team and 11 days of TPN. The patient was discharged 15 days postoperatively in good condition with long-term anticoagulation due to the Gore-tex graft.Intestinal malrotation and concurrent CD is exceedingly rare; with only three cases reported in the literature . The firIntestinal malrotation is a congenital anomaly involving malpositioning of the small bowel and colon as well as malfixation of the mesentery that frequently presents in the first months of life. Malrotation presents symptomatically in ~1 in 6000 patients, however, it is commonly asymptomatic and thus the true incidence is likely higher .Normal intestinal rotation begins in the fourth gestational week. The embryonic intestinal loop herniates into the umbilical cord and rotates counterclockwise 90°. In the eighth to tenth week the intestine returns to the abdominal cavity and continues to rotate another 180°, totaling 270° counterclockwise . Common The clinical presentation of malrotation can be nonspecific and difficult to diagnose, particularly with comorbid CD. Patients with malrotation can suffer from repeated acute bowel obstruction as well as less specific symptoms such as generalized abdominal cramping . Due to Malrotation itself is not a surgical indication, in cases such as our patients with CD, it can provide a surgical as well as endoscopic challenge. It is of note that both patients required conversion to open procedures and the second patient required vascular reconstruction, perhaps due to anatomical variation of the vascular anatomy secondary to the underlying malrotation. With these concomitant conditions, one should consider advanced imaging prior to surgical intervention. A potential option that has been shown to be efficacious in inflammatory bowel disease patients is virtual endoscopy, which can be used to create 3D models with MRI . It is iConcomitant CD and malrotation is a rare combination. Therefore, we cannot conclude that there is a mechanistic relationship or causality between them. Although proper understanding of the anatomical anomaly based on imaging is recommended, anomalies can still be challenging to operate and are possibly at higher risk for complications.No external funding was provided for this study.None declared.