Ectopic ovaries are a rare occurrence. A 33-year-old woman presented to our unit for evaluation of a 2-year history of sporadic abdominal pain that was becoming sharp and frequent. Computed tomography (CT) suggested a gastrointestinal tract mesenchymoma. An abdominal laparotomy was performed and the tumor was excised for pathologic evaluation. A rapid frozen section pathologic examination showed a solitary fibrous tumor (SFT). The final pathology report was an ectopic ovary with corpora lutea bleeding. Ectopic ovaries are benign and the present case is the first report involving an ectopic ovary mimicking a gastrointestinal stromal tumor (GIST). The patient recovered well after surgery. Maldevelopment of the genital tract can lead to ectopic ovaries and surgery is a good management choice. The present case provides a possible differential diagnosis for GISTs. Ectopic ovaries are rare embryologic abnormalities with an estimated prevalence between 1:29,000 and 1:93,000 gynecologic admissions . Herein,9/L; neutrophilic granulocytes, 76.8%; hemoglobin, 120 g/L; and platelet count, 322 × 109/L. Computed tomography (CT) revealed an intestinal stromal tumor  is also advised.The present case is an ectopic ovary mimicking a GIST. Maldevelopment of the genital tract can lead to an ectopic ovary and surgery is a good management choice. We have shared our clinical experience to help guide the management of similar cases and offer a differential diagnosis of GISTs.All datasets generated for this study are included in the manuscript and/or the supplementary files.Written informed consent was obtained from the patient for publication of this case report and the accompanying images.JP and SW: conceptualization. HW: data curation. ZF: investigation, validation, and writing of the original draft.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


A pulmonary nodule is a common incidental finding on chest imaging, which includes a wide variety of differential diagnosis. Pulmonary hyalinising granuloma is a rare disease aetiology of pulmonary nodule(s). We report a 74-year-old female who was referred to the respiratory clinic with incidental finding of a solitary pulmonary nodule on chest X-ray. CT confirmed the presence of a 1.2 cm solitary pulmonary nodule in the left upper lobe with no lymphadenopathy. The patient underwent wedge resection, and histopathological examination of the lesion confirmed pulmonary hyalinising granuloma. In most previously reported cases, patients had multiple lesions on chest radiography. Solitary pulmonary lesion is an uncommon presentation of this clinical entity and only a few cases have been reported in the literature. Pulmonary hyalinising granuloma (PHG) is a rare condition, presenting more often as multiple pulmonary nodules. It is characterized by fibrosing nodules, consisting of central whorled deposits of lamellar collagen. The nodules are benign in their clinical course but may slowly grow in size. Often, a biopsy is required to establish the diagnosis of PHG. Presentation as a solitary nodule is particularly rare.A 74-year-old female was referred to the respiratory clinic with incidental finding of a pulmonary nodule on chest X-ray. She did not have any other respiratory symptoms. She was a non-smoker throughout her life and had no exposure to asbestos or tuberculosis. Her past medical history was significant only for chronic lymphoedema and recent hip replacement.Subsequently , CT thorax was performed, which confirmed a 1.2 cm pulmonary nodule in the posterior segment of the left upper lobe. There was no cavitation or calcification and no discrete nodules were present in the remaining lung parenchyma. There was no mediastinal or hilar lymphadenopathy .On positron emission tomography scan, the pulmonary nodule was found to be non-avid . Her pulHistology showed abundant eosinophilic material with scattered giant cell reaction. No necrosis or epithelioid granulomatous inflammation was seen. Lamellar fibrosis was evident .Masson’s stain confirmed hyaline lamellar fibrosis whereas Congo red stain for amyloidosis was negative  and 5. TPHG is included in the differential diagnosis of diseases such as infectious granulomatous diseases , non-infectious granulomatous diseases , amyloidosis and rheumatoid lung involvement.The patient had nodule excision through wedge biopsy.The patient is stable after surgery and no complication has been reported on follow-up.1 The mean age of presentation is 45, with an age range of 15–77.2 There is no gender or racial predilection.2 It could present with vague symptoms. In 25% of cases it is asymptomatic and is discovered incidentally on routine chest radiographs.PHG was first described in 1977.2 Solitary pulmonary nodules are rare.4 Usually they grow slowly, or may not grow at all; spontaneous regression has also been reported. Positron emission tomography-CT may or may not reveal increased metabolic activity in PHG lesions, although it is useful in ruling out metastatic lesion.5In most cases, PHG presents as multiple, unilateral/bilateral pulmonary nodules with well-defined circumscribed borders. Lesion size ranges from a few millimetres to 15 cm, with an average size of 2 cm.9 However, our patient did not have any of the above associations, and clinically there was no evidence of any autoimmune disease. The aetiology of PHG remains unclear. It is postulated that it results from abnormally elevated immune response to one of the various infectious or autoimmune processes. Various associations of PHG have been reported in the literature, such as fungal or mycobacterial lung disease, rheumatoid arthritis, lupus-like anticoagulant, sclerosing mediastinitis, sarcoidosis, IgG4-related sclerosing disease, idiopathic thrombocytopenic purpura and lymphoproliferative disorders.10Definitive diagnosis of PHG may require a wide excisional biopsy. Histologically, it is described as a densely fibrotic nodule with patchy chronic inflammation around. The nodules are composed of thick, hyalinized bands of collagen that are arranged in whorls.11The prognosis of PHG is generally good with no known malignant potential. Solitary lesions tend to be stable. Some patients with multiple lesions may show progressive enlargement of nodules and worsening dyspnoea. There are some reports of recurrence after resection. There is no definitive treatment but successful resolutions of the multiple lesions have been reported after administration of glucocorticoids.Sometimes, it is difficult to differentiate radiologically the true nature of a pulmonary nodule and biopsy is required for definite diagnosis.Although solitary nodule PHG is rare, this case report demonstrates one such case and hence should be considered as differential diagnosis.We should actively look for any associated condition in a patient diagnosed with PHG.Written informed consent was obtained from the patient for the publication of this case report,including accompanying images.


The gastrointestinal tract is a common extranodal site for lymphomas. However,primary gastrointestinal lymphomas are rare. Diffuse large B-cell lymphomas(DLBCL) are the most commonly encountered type in the gastrointestinal tract.Most of the DLBCL are CD5 negative. CD5+ DLBCL is very rare and a poorprognostic subtype of lymphoma. We report a rare case of primary small bowelCD5+ DLBCL that evolved from being a localized low International PrognosticIndex–scored disease into an advanced and aggressive disease primarily dictatedby the presence of CD5 antigen positivity. CD5 expression in DLBCL is apoor prognostic marker. CD5 is usually expressed in chronic lymphocytic leukemia,mantle cell lymphoma (MCL), and less frequently in DLBCL. De novo CD5+ DLBCL is arare and poor prognostic subtype of lymphoma. They account of at least 10% of allDLBCLs.6DLBCL-expressing CD5 antigen seems to be a unique subgroup that is phenotypically,genotypically, and immunophenotypically different from the other CD5− DLBCLs. Thisrare immunophenotypic lymphoma has clinical and prognostic implications. We describea case of de novo CD5+ DLBCL involving small intestine, which differed from all theclinical characteristics previously studied for this subtype.Primary gastrointestinal (GI) lymphoma is a very rare entity, constituting only about1% to 4% of all GI malignancies.6This is a 50-year-old gentleman who presented with symptomatic anemia secondary tosevere iron deficiency. On imaging, he was found to have an exophytic soft tissuemass in the cecal tip adjacent to appendix. His colonoscopy was consistent with anear circumferential large polypoid mass at the ileocecal valve extending into theterminal ileum. Biopsy of the ileocecal mass  showed d1 On the other hand, primary GI lymphoma are very rare. It accounts for about1% to 4% of GI malignancies.The most common extranodal site for lymphoma is the GI tract.2 Primary GI lymphoma is usually of B-cell lineage, with certain histologicalsubtypes having a relative predilection sites. For instance, MALT (mucosa-associatedlymphoid tissue) lymphoma is common in stomach, whereas MCL in terminal ileum,jejunum and colon, and follicular lymphoma is usually found in the duodenum.4 Since 1961, Dawson’s criteria7 have been used to characterize primary GI lymphomas; these are negativeperipheral lymphadenopathy, negative mediastinal lymphadenopathy, normal white bloodcell counts, predominance of bowel lesion with only lymph nodes affected in theimmediate vicinity, and no liver or spleen involvement. Molecular studies have shownthat CD5 is a negative regulator of B-cell receptor signaling,8 modifies intracellular calcium, and modulates B-cell physiology by activatingvarious signaling pathways, including ERK1/2, PI3K, and calcineurin.9 CD5 also adds to B-cells survival advantage through stimulation of autocrineinterluekin-10 production.10 The mechanism leading to overexpression of CD5 in DLBCL remains unclear. Themajority of CD5+ DLBCL cases belong to the activated B-cell subtype ofDLBCL.12 Clinically, this molecular subtype of DLBCL follows anaggressive clinical course similar to T-cell lymphomas.13Stomach is the most common involved sites than intestine and ileocecal area.14 Different treatment approaches have been studied previously involving surgeryplus chemotherapy and chemotherapy alone for localized GI lymphomas. Early tumorstage, right-sided lesion, and DLBCL histological pattern seem to be the clinicalcharacteristics of optimal surgical candidates.15 For patients with localized disease (Lugano stage I/II), surgery pluschemotherapy yielded a lower relapse rate (15.3%) than did chemotherapy alone. The 3-year overall survival (OS) rate was 91%in the surgery plus chemotherapy group and 62% in the chemotherapy-alone group.16 By opting for the surgical approach, we reduced his risk of perforationduring therapy as well as his exposure to chemotherapy side effects by reducing thenumber of chemotherapy cycles.The role of surgical intervention in primary colon lymphoma was reviewed by Cai et al.5 analyzing the clinical characteristics of 63 cases of DLBCL by grouping themas CD5+, CD5− CD10+, and CD5− CD10−. As per the study, the median age at diagnosiswas 57 years, with slightly more preponderance in females. Sixty-two percent of thecases had advanced stages (III and IV), and almost half of the patients hadextranodal involvement.17 The extranodal sites included arm, nasal cavity, skin, spleen,stomach, testis, tonsil, thyroid, breast, and GI tract with highest number of casesin breast. Rare cases of involvement of liver has also been reported.18 Almost all of the patients did not show positivity for HIV or HTLV1. Tenpercent of the cases showed BCL2 rearrangements, while 25% of the cases had BCL6rearrangement. All cases had negative cyclin D1 by immunohistochemistry. As per thisstudy, the clinical characteristics of this subgroup of CD5+ DLBCL included femalepreponderance, elderly onset, advanced stage at presentation, and with frequentinvolvement of bone marrow.17Although CD5− DLBCL commonly involves GI tract, de novo CD5+ DLBCL involvement of GItract is very uncommon. An extensive case study was done by Harada et al19 These cases were compared with 384 CD5− DLBCL and 128 cyclin D1–positive MCLcases. As per this study, CD5+ DLBCL patients had a higher age distribution with amedian age of 66 years with a higher female preponderance. When compared withCD5-neagative DLBCL, the subgroup of patients with CD5 positivity showed aggressiveclinical features, >1 performance status, higher lactate dehydrogenase (LDH), Bsymptoms, advanced stage at diagnosis, higher IPI, and with the involvement of morethan one extranodal sites with bone marrow involvement being more frequent. Most ofthe CD5+ DLBCL subtypes showed centroblastic morphology and had immunophenotypiccharacteristics of CD5+, CD10−, CD19+, CD20+, CD23−, and cyclin D1−. The OS curvewas significantly (P = .0026) inferior compared with CD5+ DLBCL.19Kobayashi et al published another larger study involving 109 de novo CD5+ DLBCL casesprimarily studying their clinical characteristics.20 and advanced stage at diagnosis with more frequent central nervous system involvement.21 But our patient had low LDH, no B symptoms, good performance status, low IPIscore, and with localized stage at diagnosis thereby differing from the onesdescribed in the studies. Although rituximab-based chemotherapy has improved the OSin CD5+ DLBCL, it still remains much lower compared with CD5− DLBCL. Unfortunately,the rate of central nervous system involvement has not improved with the use ofrituximab.22As per these large studies CD5 expression in DLBCL is associated with elevated LDH, Bsymptoms, extranodal involvement, poor performance status, higher IPI,Even though we opted for the best treatment approach of upfront surgery followed byR-CHOP chemotherapy for the localized small bowel lymphoma to reduce the toxicityand improve the OS, the CD5 expression of the lymphoma dictated the prognosisirrespective of the mode of therapy. The disease showed an aggressive clinicalcourse and progressed through the R-CHOP chemotherapy, proving the fact that thesesubtypes of lymphomas are molecularly and clinically different from CD5− DLBCL.Whether more intensive chemotherapy like REPOCH or Hyper-CVAD would have benefittedfor these rare lymphomas is still a question of debate as there are no consensus inthe treatment regimens for these unique subtypes.Our case posed a diagnostic challenge by presenting as a rare and unique immunotypicsubtype of DLBCL in a unique location with poor prognostic features. Optimalmanagement in these cases are not clear. Our case was very unique in presentinginitially with good prognostic markers and later evolving into an aggressive variantwith poor prognostic features, likely due to CD5 expression. Whether highlyintensive regimens other than R-CHOP or even stem cell transplant would salvagethese patients remains to be studied yet.


Hibernoma is a rare benign lipomatous tumor showing differentiation of brown fatty tissue. To the author's best knowledge, there is no known case of malignant transformation or metastasis. Due to their slow, noninfiltrating growth hibernomas are often an incidental finding in the third or fourth decade of life. The vast majority are located in the thigh, neck, and periscapular region. A diagnostic workup includes ultrasound and contrast-enhanced MRI. Differential diagnosis is benign lipoma, well-differentiated liposarcoma, and rhabdomyoma. An incisional biopsy followed by marginal resection of the tumor is the standard of care, and recurrence after complete resection is not reported. The current paper presents diagnostic and intraoperative findings of a hibernoma of the upper arm and reviews similar reports in the current literature. Fat-containing tumors are the most common soft tissue tumors, their prevalence increasing with age . The disA 44-year-old patient presented in our clinic with ongoing shoulder and arm pain for 3 years. He had noticed a painless tumor with feeling of pressure in his upper right arm for some months. The patient was diagnosed with an impingement syndrome and supraspinatus tendon rupture and consequently attributed the swelling to a spontaneous rupture of the long head of the biceps tendon. There was no history of recent trauma or injuries. The patient was in good health without systemic signs of infection, weight loss, or fever and stated no prior medical history.Clinical examination showed a painless tumor on the right upper arm, starting lateral to the axillary fold expanding about 10 cm distally. On palpation, the tumor was elastic and firm with only little translational mobility. There was no sensory or motor dysfunction. The overlying skin showed no signs of infection or other noticeable changes.Conventional X-rays revealed a soft tissue tumor without calcification. Ultrasound could confirm the suspected tumor, with clear margins to the surrounding muscle and similarities to fatty tissue. Slightly increased perfusion was present at the margins of the lesion.mm  with an intramuscular location, deep to the fascia of the M. biceps brachii. The fascia was not penetrated, and the tumor showed clear margins to the displaced muscle tissue. Signals in T1w and T2w were hyperintense but slightly inferior to subcutaneous fat on T1w. Contrast enhancement was shown in the periphery of the lesion  that seem to have different prevalences depending on anatomic location, age, and gender. This variety in histological characteristics causes the aforementioned difficulty to distinguish hibernoma from lipoma and liposarcoma in imaging studies , 17, 18.Treatment is complete marginal resection , 7. The Removal should be advised to rule out the possibility of a malignant lesion with hibernoma-like differentiation, which could be missed in a small biopsy . AdditioHibernoma is a rare, benign, lipomatous soft tissue tumor. There is no known risk of malignant transformation or metastatic spread. Differentiation to malignant soft tissue tumors like low-grade liposarcoma can be difficult to impossible when based only on radiographic imaging, and an incisional biopsy is mandatory in most cases. Marginal resection is curative with no reported recurrences after complete resection.


Brown tumor is a bone lesion that arises in the setting of excess osteoclast activity in hyperparathyroidism. It consists of fibrous tissue, woven bone, and supporting vasculature, while contains no matrix. The characteristic of brown-colored lesion is a result of hemosiderin deposition into the osteolytic cysts. Two cases of young women aged 26 and 29 years old, respectively, are known with a history of end-stage renal disease (ESRD). Dialysis is performed two times/week over the last 7 years. Our patients presented with an intraoral mass of the hard palate since 12 months ago and decreased body height of 10 cm. The lesion causes difficulties in swallowing and talking. Laboratory workup showed elevated parathormone or PTH (3.391 pg/mL and >5.000 pg/mL). Neck ultrasound showed enlargement of the parathyroid glands. Supporting examination to diagnose brown tumor are neck ultrasound, CT of the neck, and parathyroid sestamibi scan. We performed parathyroidectomy. Pathology revealed hyperplasia of the parathyroid. The tumor regressed significantly within 2 weeks following the surgery, and we still observe tumor regression as well as reduction in PTH level. As clinicians, we should be alert to other possible causes of bony lesions. Clinical examination, laboratory finding, and imaging present important information to diagnose brown tumor. Secondary hyperparathyroidism is a complication of renal failure, developed from many factors such as calcitriol deficiency, vitamin D deficiency, and the increased retention of phosphorus , 2. ThesAnother complication of hyperparathyroidism is brown tumor, with incidence rate of 0.1%. Brown tumor is a giant cell granuloma which appears as a result of the imbalanced osteoclast activity and peritrabecular fibrosis . It consBrown tumor presents as a fixated growing mass with bony consistency. The tumor itself is usually asymptomatic but causes disability in chewing, talking, and breathing if it occurs in the craniofacial region . It may A 26-year-old female patient complained a growing mass in the hard palate since 12 months ago. This mass causes swallowing and closing mouth difficulty. She also complained about changes in the shape of her finger bones, mandible, and ribs. The decrease in height of 9 cm and weight loss of 10 kg were observed within 2 years. The patient experienced end stage of renal failure and has been treated with hemodialysis 2 times per week for seven years since 2009.The physical examination showed a mass in the palatum durum and mandible, measured 4 × 6 × 2 cm and 3 × 4 cm in size, respectively. The mass has rough surface, hard-solid consistency, with no tenderness. A mass in the right neck was measured 3 × 5 cm in size. The mass has smooth surface and solid consistency .The result of the palatum and mandible biopsy was compatible with ossifying fibroma or fibrous dysplasia. Neck ultrasonography showed a solid mass in the right thyroid, bilateral cystic nodule in the thyroid lobes, and small cystic nodule in the thyroid isthmus. Bone survey showed the diffused decreasing bone density, fractured right clavicle, multiple lytic lesions among the cranium, humerus, clavicle, iliac, and pubic bone . ParanasBased on the physical examination and laboratory findings, we concluded brown tumor as the diagnosis due to secondary hyperparathyroidism. The treatment for this patient was right parathyroidectomy and right isthmulobectomy with frozen section. The right isthmulobectomy was chosen because the patient has secondary diagnosis which was right nontoxic nodular enlargement of the thyroid gland. In addition, the technique was considered to be appropriate to access the parathyroid gland with the antecedent removal of the thyroid gland. Frozen section revealed hyperplasia of the right parathyroid. Three days following the surgery, PTH level was significantly decreased from the initial preoperative level (851.10 pg/mL). Result from definitive pathology anatomy was hyperplasia of the parathyroid gland.One month following the surgery, the patient felt regression among her hard palate and mandible mass and ease in swallowing. During follow-up, we performed additional evaluative examination. Neck 4D CT was done 2 times at the 2nd and 5th month of follow-up. The first one showed multiple lymphadenopathy around the neck and left supraclavicle, no residual mass in the right thyroid, no abnormality in the left thyroid, and no ectopic parathyroid gland was found. While the second Neck 4D CT found a single solid lesion in the left thyroid and 2 solid lesions in aortopulmonary window, suggestive of parathyroid nodes. At the 6th month of follow-up, sestamibi parathyroid scan showed no abnormality and no residual mass in the right thyroid lobe. PTH level evaluation performed in the 4th and 7th month showed an elevation to 909.7 pg/mL and 1.137 pg/mL, respectively.Patient complained pain in all her bones and started walking with a cane. Seven months following the first surgery, we decided to perform left parathyroidectomy and left lobectomy with frozen section and intraoperative PTH evaluation. Thirty minutes following parathyroidectomy, the PTH level decreased significantly to 76.95 pg/mL. Frozen section result revealed hyperplasia of the left parathyroid.Two weeks following the second surgery, the mass regressed significantly, and symptoms were much reduced.A 31-year-old female patient complained a mass in her maxilla and mandible. The mass was gradually increasing in size over the last 6 months. The mass caused talking disturbances, swallowing difficulty, dilating gap between upper teeth, and narrowing gap between lower teeth. She also complained hard masses in her fingers and the middle of chest. There was a decrease in height (15 cm in 8 years) and weight (19 kg in 8 years). The patient was on end stage of renal failure and has been treated with hemodialysis 2 times per week for eight years since 2008. The patient was planned to be performed renal transplantation. Comorbidities were reported among this patient, which are chronic heart failure, grade II hypertension, lung tuberculosis on therapy, and hepatitis C. The physical examination showed facial asymmetry with mass in the palatum durum, measured 4 × 3 cm in size . PalpatiX-ray of the maxilla and mandible showed diffused decreasing bone density and multiple lytic lesion, which suggest metabolic bone disease or renal osteodystrophy or hyperparathyroidism . BNO shoLaboratory examination showed the elevated ureum (202 mg/dL), creatinine (11.1 mg/dL), and normal calcium level (9.7 mg/dL). In November 2016, PTH level was >5000 pg/mL and phosphate level was 6.1 mg/dL.Physical examination and laboratory examination lead us to brown tumor as the diagnosis due to secondary hyperparathyroidism. The treatment for this patient was subtotal parathyroidectomy with frozen section and intraoperative PTH evaluation. Intraoperative parathyroid hormone level was evaluated following subtotal parathyroidectomy. The result decreased to normal range 10.37 pg/mL . Frozen section revealed hyperplasia of the parathyroid. Calcium level was evaluated every day for 7 days, and the result ranged from 5.0 mg/dL to 7.4 mg/dL. The parathyroid hormone on the 3rd month following the surgery was 4.31 pg/mL. Definitive pathology anatomy result revealed hyperplasia of the parathyroid with water clear cell parathyroid adenoma.The masses in the maxilla, mandible, and sternum were observed to be significantly regressed. This patient then underwent renal transplantation approximately 4 months following parathyroidectomy. One month following the renal transplantation, PTH level was 6 pg/mL.There are numerous options for hyperparathyroidism treatment such as dietary modification, pharmacological therapy , and parathyroidectomy . The indIt is very important to determine the extent of parathyroidectomy prior to surgery to prevent recurrent or persistent disease and avoidance of permanent hypoparathyroidism , 15. TheThe first choices for preoperative examination are combination of ultrasonography and technetium Tc99m sestamibi scan, with the sensitivity of 74%–95% for single gland disease. Whereas for multigland disease and multiple adenomata, the sensitivity of ultrasonography is 15%–35% and sestamibi scan is 30%–44% . 4D CT mThere are three methods of surgical treatment: subtotal parathyroidectomy, total parathyroidectomy with autotransplantation, and total parathyroidectomy without autotransplantation . SelectiIn the first case, we decided to perform right parathyroidectomy and right isthmulobectomy without intraoperative PTH examination due to limited facilities. The isthmulobectomy was chosen because the patient has secondary diagnosis which was nontoxic nodular enlargement of the thyroid gland. In addition, the technique was considered to be appropriate to access the parathyroid gland with the antecedent removal of the thyroid gland.In the 4th and 7th month of follow-up, it is found that the PTH level was still high, 909.7 pg/mL and 1.137 pg/mL, respectively. 4D CT revealed single solid lesion in the left thyroid and 2 solid lesions in the aortopulmonary window, suggestive of parathyroid nodes. Therefore, 7 months after the first operation, we performed excision of superior part of the left parathyroid and left lobectomy with intraoperative PTH examination, and the result was 76.95 pg/mL. In the second case, we performed subtotal parathyroidectomy with intraoperative PTH examination, and the result was 10.37 pg/mL. In the 3rd month of follow-up, PTH level was 4.31 pg/mL. This patient underwent renal transplantation approximately 7 months after subtotal parathyroidectomy. PTH level one month after follow-up was 6 pg/mL.Our delay in the PTH examination is due to the fact that this examination is not covered by the national health insurance. PTH examination is considerably expensive in Indonesia, which costs approximately 60 USD. Therefore, the PTH examination is not a routine test for this patient.Brown tumor is one of the escalating problems due to secondary hyperparathyroidism because of ESRD. The clinical presentation of secondary hyperparathyroidism should be integrally assessed to reveal the possibility of developing brown tumor in the future, especially for patients who present with mass in the maxilla and mandible. A significant increase in the level of PTH and decreased bone density lead to the diagnosis of brown tumor. 4D CT and sestamibi are suggested to be the preferable examination to reveal the parathyroid abnormalities. Surgery and intraoperative assessment of PTH are the suggested treatment of choice.


Hernia sacs are a common anatomic pathology specimen, which rarely contain malignancy. We present a case of rapidly growing pancreatic adenocarcinoma, which initially presented as metastasis to an umbilical hernia sac. The patient was a 55-year-old male with a two-year history of umbilical hernia. Two months prior to herniorrhaphy, the hernia became painful and the patient experienced nausea and weight loss. The gross examination did not reveal distinct lesions. Microscopically, the hernia sac was diffusely infiltrated by moderately differentiated adenocarcinoma, which was positive for CK7 and pancytokeratin and negative for TTF-1, CK20, PSA, and CDX2. Clinical laboratory tests found elevated levels of CA 19-9 and CEA. Computed tomography scan with intravenous contrast showed a 5 cm ill-defined and hypoattenuating mass involving the pancreatic tail and body, as well as numerous ill-defined lesions in the liver and peritoneal carcinomatosis. The patient had an earlier noncontrast computed tomography scan four months prior to the surgery, which did not detect any lesions in the abdomen. This case highlights the importance of intravenous contrast with computed tomography for the evaluation of pancreatic lesions and also emphasizes the importance of thorough histologic evaluation of hernia sacs for the detection of occult malignancy. Hernias are very common conditions encountered in medicine, and more than 20 million hernias are estimated to be repaired every year around the world. In the recent years, the number of midline abdominal wall hernia repairs has increased. The current relative order of the various hernia repair types is as follows in decreasing frequency: inguinal, umbilical, epigastric, incisional, paraumbilical, femoral, and rare forms, for example, spigelian . We repoThe patient is a 55-year-old man with past medical history significant for two-year history of umbilical hernia, diabetes mellitus type 2, hypertension, gout, chronic kidney disease with proteinuria, diverticulosis, obesity, and osteoarthritis. The patient presented to the clinic because of umbilical hernia pain, which developed over the two months. The pain localized to the periumbilical region and left lower back, and it was exacerbated with food intake and sometimes relieved by 5 mg hydrocodone tablet. He also reported nausea and fifteen pounds weight loss over the two months, which he attributed to decreased food intake. The physical examination showed a 1 cm tender and irreducible mass superior to the umbilicus. The patient underwent herniorrhaphy and the gross examination of the surgical specimen did not reveal any masses or lesions.The microscopic evaluation showed diffuse infiltration of the connective tissue by malignant cells with hyperchromatic nuclei, inconspicuous nucleoli, and abundant eosinophilic cytoplasm . There wThe laboratory findings showed elevated levels of CA 19-9  and CEA (14.2 ng/mL). The patient underwent a subsequent computed tomography scan with intravenous contrast, which showed a 5.0 × 2.7 cm ill-defined and hypoattenuating mass located in the pancreatic tail and body , with peAfter the diagnosis, the patient refused chemotherapy and decided to undergo palliative care. The patient had rapid progression of the disease and died within two months of the initial histologic diagnosis of malignancy.Malignant tumors of the umbilical region can be primary or secondary, constituting 17% and 83%, respectively, of all the malignant umbilical tumors. The presence of umbilical subcutaneous nodule known as “Sister Joseph's nodule” has been commonly associated with intra-abdominal malignancy. The most common reported sites of origin for Sister Joseph's nodule are stomach 25%), ovary (12%), colorectal region (10%), and pancreas (7%) [5%, ovaryIn a retrospective study of 145 patients with umbilical/paraumbilical hernia by Kenig et al., 23 patients (15.9%) were diagnosed with intra-abdominal malignancy. The logistic regression analysis demonstrated that the patient's age, preoperative symptoms, anemia, and weight loss were statistically significant risk factors that were associated with the presence of an intra-abdominal malignancy. The most common intra-abdominal malignancies are colorectal cancer , pancreatic cancer , malignant tumors of the adnexa , and kidney cancer  [There are recently published articles that question the utility of histologic evaluation of hernia sac specimens in the practice of cost-effective medicine , 6. WangBy radiology, the use of intravenous contrast with computed tomography can help aid in the early detection of pancreatic lesions. Given our patient's history of chronic kidney disease, he underwent a prior noncontrast computed tomography scan four months earlier, which did not detect the pancreatic lesion. After the histologic diagnosis, the computed tomography scan with intravenous contrast detected the 5 cm hypoattenuating mass. The standard modality for the detection of pancreatic cancer with >90% accuracy is multidetector computed tomography. The use of intravenous contrast is essential, because pancreatic adenocarcinomas are hypovascular and have lower attenuation than healthy pancreatic parenchyma . HoweverIn conclusion, the routine histologic evaluation of the hernia sacs is important in the diagnosis of occult malignancies, because lesions may not be grossly evident. In addition, the use of intravenous contrast with computed tomography can help aid in the early detection of pancreatic adenocarcinoma, because most lesions present as a hypoattenuating mass.


Biliary intraepithelial neoplasia (BilIN) is often distinguished by what it is not: the precancerous lesions are not mass-forming, are not the cause of bile duct obstruction, and are small enough (less than 5 mm long) to evade detection by the naked eye. Here, we describe an atypical case of BilIN resembling cholangiocarcinoma (CC) that was large enough to be identified by diagnostic imaging and presented with obstructive jaundice caused by a hematoma in the common bile duct (CBD).A 64-year-old man presented to our hospital with upper abdominal pain and anorexia. Initial laboratory examinations revealed increased total bilirubin and a computed tomography (CT) scan revealed a dilated CBD. Gastroenterologists performed an endoscopic sphincterotomy (EST), which revealed that the cause of obstructive jaundice was a hematoma in the CBD. Enhanced CT scan and magnetic resonance cholangiopancreatography (MRCP) performed after the hematoma was drained showed improved dilation of the CBD and an enhanced wall thickness of bile duct measuring 25 × 10 mm at the union of the cystic and common hepatic ducts. A cholangioscope detected an elevated tumor covered by sludge in the CBD, and we performed an extrahepatic bile duct resection and cholecystectomy. The postoperative course was uneventful and the pathological examination of the resected tumor revealed that although the ulcerated lesion had inflammatory granulation tissue, it did not contain the components of invasive carcinoma. Many consecutive intraepithelial micropapillary lesions spread around the ulcerated lesion, and the epithelial cells showed an increased nucleus-to-cytoplasm ratio, nuclear hyperchromasia, and architectural atypia. The pathological diagnosis was BilIN-1 to -2. Immunohistochemical staining showed that S100P was slightly expressed and MUC5AC was positive, while MUC1 was negative and p53 was not overexpressed.We experienced an atypical case of BilIN mimicking CC that presented with obstructive jaundice caused by a hematoma in the CBD. Our case suggested that the occurrence of BilIN can be triggered by factors other than inflammation, and can grow to a size large enough to be detected by image analyses. Cholangiocarcinoma (CC) is the second most common primary liver cancer and carries a high post-resection morbidity and mortality rate , 2. MostA 64-year-old man presented to our hospital with upper abdominal pain, jaundice, and anorexia. He had diabetes and was a social drinker but a lifetime non-smoker. Computed tomography (CT) scan revealed a dilated CBD, and acute cholangitis was suspected. The patient was referred to our hospital and admitted to the gastroenterology department for further investigation and treatment. Initial laboratory examinations revealed a white blood count (WBC) of 9770/μL, hemoglobin of 12.4 g/dl, increased C-reactive protein (CRP) of 5.47 mg/dl, total bilirubin of 7.75 mg/dl, AST/ALT of 176/281 IU/L, alkaline phosphatase of 815 IU/L, and ɤ-GTP of 132 IU/L. The serum tumor markers carcinoembryonic antigen (CEA) was within the normal range at 2.6 ng/ml and cancer antigen 19–9 (CA19–9) was elevated at 1162 U/ml. Both hepatitis B surface antigen (HBsAg) and antibodies to hepatitis C virus (anti-HCV) were negative. A plain CT scan on admission showed a high-density accumulation spreading throughout the CBD, and the entire CBD was dilated (Fig. BilIN has been defined by Zen et al. in 2005 as microscopic lesions of flat or low-papillary dysplastic epithelium , 14. BilIn our case, there were two atypical findings regarding BilIN. One is that it occurred in a patient with no history of a prior biliary disease or chronic inflammation. Although the mucosa around the tumor showed redness, the range was limited to the junction of the cystic and common hepatic ducts. The other atypical finding is the large tumor size of 25 × 10 mm. Although BilIN is usually unrecognizable by macroscopy, the tumor was clearly detected in CT and could be observed as an elevated mass in the cholangioscope. To the best of our knowledge, it was larger than any other case of BilIN without CC reported in the literature so far and was therefore very difficult to differentiate from CC. These two findings in our case suggest that the occurrence of BilIN can be triggered by factors other than inflammation and that the tumor can grow to a size large enough to be detected by image analyses without shifting to CC.The tumor was ulcerated and hemorrhaging in the CBD when the patient was referred to our hospital. In our case, obstructive jaundice caused by the hematoma in the CBD led to the detection of BilIN before progressing to CC. CC is not a major cause of hemobilia, which is present in only 3% of cases . It was It is important to detect precursor lesions early in cancer therapy, although the prognosis of BilIN without CC has not been clarified and a uniform treatment guideline is lacking. And unlike endoscopic resection for adenoma in the gastrointestinal tract, a minimally invasive treatment in the bile duct has yet to be developed. The only curative treatment for BilIN prior to malignant transformation is surgical resection . In surgMany molecular and genetic alternations have been found to occur and accumulate in BilIN during the multistep process of cholangiocarcinogenesis , 21. TheWe experienced an atypical case of BilIN mimicking CC that presented with obstructive jaundice caused by a hematoma in the CBD. Our case suggested that the occurrence of BilIN can be triggered by factors other than an inflammatory condition and can potentially grow to a size large enough to be detected by diagnostic imaging.


Benign metastasizing leiomyoma (BML) is a rarely found entity with few documented cases in the literature, usually occurring in women of reproductive age with a history of myomectomy or hysterectomy. The leiomyomas can metastasize to several organs, the lungs being the most commonly affected. We report a case of a 40-year-old female patient who presented at our institution with low back pain. She underwent a lumbar MR that revealed the presence of an expansive and compressive mass in the body of L4. This mass was biopsied, corresponding to a metastasizing leiomyoma with no malignant characteristics. Subsequently, a CT examination showed several soft tissue density round masses in both lungs, but the most striking feature was a 12 cm mass located in the left iliac crest. There was asymmetrical uterus enlargement, caused by the presence of several leiomyomas. Since the lesions were estrogen and progesterone positive, hormone suppression consisting of oophorectomy followed by anastrozole was the chosen treatment. No signs of progression were observed at the 6-month follow-up. This case is one of the very few that occurred in a woman with no previous uterine intervention, adding further evidence that surgery is not an essential condition for this entity to develop. BML is a rarely found condition that affects women with a history of uterine leiomyomas. Steiner first described BML in 1939 in a report of a patient who died from the effects of extensive pulmonary metastasis that were histologically identical to multiple leiomyomas concurrently present in the uterus [BML usually occurs in women of reproductive age that present, most of the times, with a previous history of myomectomy or hysterectomy . The leiA 40-year-old female patient presented at the Emergency Department of our institution with complaints of back pain for the last three days that started after moderate physical exertion. The patient referred no pain relief after taking anti-inflammatory drugs and denied respiratory symptoms such as chest pain, dyspnea, or cough. No weight loss, anorexia, or other symptoms were reported. There was no referral of previous surgeries or medications. A different, stronger anti-inflammatory drug was prescribed, but three days later she returned to the hospital, where a lumbar radiograph revealed the presence of a lytic lesion in the L4 vertebra. Due to the nonspecific appearance of the lesion, the patient underwent an MR of the lumbar spine. The exam revealed the presence of a heterogeneous, T1-hypointense mass in the body of L4, causing its partial destruction, and nerve root compression . A CT-guSubsequently, a contrast-enhanced CT was performed to evaluate if other organs were affected: there were several soft tissue density round masses in the thorax, the largest being located in the left lung, measuring 44 mm . There wThe clinical conduct included vertebral subtotal tumor removal, laminectomy, and pedicle screw fixation on L3-L5  to decomRegarding the low back pain, the patient referred a moderate improvement. Despite this, she needed to resort frequently to taking anti-inflammatory drugs after some physical exertion. The patient also reported a sensation of pressure in the pelvic region, especially in the lateral decubitus, most likely associated with the mass of the iliac bone.The causes of BML remain, in part, unexplained. On the one hand, BML is more frequent in women submitted to a previous myomectomy/hysterectomy, corresponding to the vast majority of the published cases to date . PeritonOn the other hand, the reports of BML occurring in women with an intact uterus imply that a previous uterine intervention is not an essential condition for the development of this entity. Therefore, there must be a cause, other than the mechanical spread of cells, that would explain the origin of these metastasizing leiomyomas. One hypothesis is that BML is related to metaplasia of the coelomic epithelium . AccordiThis case report is one of the very few that exhibits unusual clinical and radiological characteristics for BML: the fact that the patient was symptomatic at the time of diagnosis, findings in both lung and bone, and the absence of previous hysterectomy/myomectomy. By reviewing the current literature, there are, as far as we know, seven BML cases in women with an intact uterus. The most frequent (and unique) location of the metastasis of leiomyomas was the thorax –12. One Also noteworthy is the rarity of spinal BML, having only 10 cases been reported to date . DiagnosDifferential diagnosis plays a crucial role in BML. Malignancies, such as low-grade leiomyosarcoma, should be excluded through the evidence of lack of atypia or necrotic areas . It is nTreatment is not standardized due to the rarity of this entity. Several clinical approaches have already been adopted such as watchful waiting, surgical excision , oophoreIn this case report, we reviewed a rare case of BML developing in the bone and the lung, in a patient that had not been submitted to a previous hysterectomy. While histological studies were essential to confirm the diagnosis, imaging played a key role in localizing and monitoring of the lesions. Both the referring doctor and the radiologist should be alert to this diagnosis when a woman with a history of leiomyomas and multiple pulmonary nodules presents at a given context. In this case, this was not as easily assessable due to unique confounding factors. The treatment, in this case, was based on hormone production suppression, with no signs of progression at the follow-up. Given that BML may have several different clinical presentations and progression patterns, an individual approach should always be taken into account.


Intussusception in adult patient is rare. Gastroduodenal intussusception due to the gastrointestinal stromal tumors is infrequently described in the literature. Authors present a case of gastroduodenal intussusception due to the low-risk gastrointestinal stromal tumor of the lesser curvature of the gastric body with literature review.Sixty-two-year-old male was admitted to our hospital with symptoms of acute gastric outlet obstruction. Imaging studies confirmed a lesion of the gastric wall producing gastroduodenal intussusception with pylorus obstruction. Upon laparotomy a tumor mass of the lesser curvature of the gastric body that invaginated through the pylorus into the duodenum was found. Desinvagination and resection of the tumor with the adequate resection margins were performed. Histology reveled a low-risk gastrointestinal stromal tumor. Due to favorable outcome only observation was suggested by the multidisciplinary team.Gastroduodenal intussusception due to the gastrointestinal stromal tumor of the gastric wall is a rare event. Surgical resection is the treatment of choice. In selected cases laparosopic resection of the tumor can be performed. Intussusception rarely occurs among the adult patients. It accounts for 5% of all intussusception cases and in only 1% causes intestinal obstruction . Clinica9/L, normal range 4.0–10.0) and normal value of C-reactive protein . Tumor markers CEA and Ca 19–9 were within normal range.62-year-old Caucasian male presented to the emergency room with acutely worsening epigastric pain lasting for several days and black stool lasting for a week. Symptoms of vomiting, inappetence and weight loss that have been lasting for the past six months without doctor appointment was also reported in medical history. Patient had a history of diabetes mellitus on insulin therapy. Upon clinical examination abdomen was not distended, there was no signs of guarding or rebound tenderness. Laboratory data showed anemia , leukocytosis  and ultrasound of the abdomen on the outpatient bases only few days prior to admission to the hospital. EGD was technically demanding due to the poor passage of the endoscope through the stomach, duodenal bulbous and upper part of the duodenum. Inflation of the gastric body was not possible, therefore the visualization of the gastric wall was poor with no obvious intraluminal mass or hemorrhage detected. Additionally gastric peristalsis was described to be absent. Abdominal ultrasound showed tumor formation of the gastric body, measuring 7 × 5 cm, but no intussusception was described. CT scan revealed a 5.4 × 5.6 × 6.2 cm intraluminal tumor formation of the lesser curvature of the gastric body with well defined borders was described. Tumor mass caused invagination of the gastric cardia through the antrum and pylorus into the D2 part of the duodenum producing gastric outlet obstruction Figs. , 2. No dExplorative laparotomy was performed. Palpable gastric mass with impaction of the tumor through the pylorus into the duodenum without signs of disseminated disease in the abdomen. Was found Figs. , 4. Koch2. Proliferation marker Ki67 was less than 5%. Resection margins were free of disease. Tumor was pathologic staged as T3.Sample was send for pathohistological examination. Macroscopically specimen contained a well-defined 7.5 × 5.5 × 4 cm solid, grey mass with no necrosis Fig. , 7 and 8Patient case was presented on the multidisciplinary team meeting and due to low-risk GIST with favorable prognosis only observation is suggested.Authors report a case report of 62 years old patient with gastric outlet obstruction due to the gastroduodenal intussusception caused by a large, low risk GIST of the lesser curvature of the gastric body.Intussusception in adults is an infrequent condition, therefore when the patient presents with the symptoms of the upper gastrointestinal obstruction, other causes should be considered . GastroiReviewing the literature, patients with GIST and gastroduodenal intussusception most commonly presented with nonspecific symptoms of acute or intermittent abdominal pain with vomiting lasting from a day to several months Table  5–21]. –21. 5–21Standard histopathological examination of the specimen and H&E staining are not enough for the diagnosis of GIST. Immunohistochemical studies are the golden standard . GIST isIn case of gastroduodenal intususception, reduction of the invagination can be performed endoscopically. In described case, where a large GIST led to the intussusception and protrusion of the gastroesophageal junction through the pylorus, endoscopic reduction of the invagination was not feasible. Moreover, upon endoscopy, the stomach was not visible and the tumor was not recognized. Both imaging techniques, ultrasound and CT scan of the abdomen, respectively, described tumor formation of the gastric wall with CT scan describing the gastroduodenal intusussception. Desinvagination and desimpaction of the tumor was not possible until Kocher mobilisation of the duodenum and pancreatic head was performed.Surgery remains the treatment of choice for resectable GIST with no dissemination but also for oligometastatic disease with potentially resectable liver metastases. In many cases, especially in low-risk GIST, surgery represents the definitive treatment. A wide local resection with clear resection margins is recommended. In cases of invasion to the adjacent organs en-block resection should be performed. Systemic therapy may be indicated in patients with high-risk tumors and in patient with unresectable tumor or metastatic disease. Small lesions can be resected laparoscopically. Endoscopic resection of the GIST is not recommended . In the GIST of the gastric wall rarely produces gastroduodenal intussusception. As shown in our case patients with low-grade GIST have favorable prognosis and in most cases radical surgical resection is the only treatment needed. When GIST arise from the gastric wall, resection of the tumor with adequate resection margins should be performed. For small lesions where desinvagination can be achieved endoscopically, laparoscopic resection of the tumor can be considered. In cases of large, impacted lesions, open approach with desinvagination and tumor resection is advisable.


Incarceration of the gravid uterus is a rare obstetric disorder that contributes to pregnancy-related complications. To understand its clinical characteristics and managements, we have reviewed the etiology, risk factors, clinical characteristics and current treatments of an incarcerated gravid uterus based on 162 cases reported in the English language literature, including our patient.A 25-year-old primigravida, with a history of lymphatic tuberculosis, infertility due to blocked fallopian tubes and received in vitro fertilization. The patient presented with urine retention and lower abdominal pain in the early second trimester. Uterine incarceration was diagnosed based on pelvic examination and abdominal ultrasound. A Foley catheter was placed and manual reposition was successful. No episode of retention was experienced after the further enlargement of the uterus and its ascent. A healthy infant was delivered vaginally on 38th week of pregnancy.Uterine incarceration due to pelvic adhesions is rare and, because of it non-specific clinical presentations, is often misdiagnosed. Abdominal ultrasound is instrumental for the diagnosis because it can directly image the disturbed uterine and pelvic anatomy. There are limited treatment options for uterine incarceration, but definitive diagnosis allows procedures to treat and to reduce severe complications of uterine incarceration. Uterine retroversion is recognized as a normal variant and its prevalence is reported to be up to 15% of pregnancies in the first trimester [A 25-year-old patient, gravida 1, para 0, first presented to the regional hospital at 16 weeks of gestation and with chief complaints of vaginal bloody discharge for 6 days, unable to urinate, and mild lower abdominal pain. The patient was diagnosed with lymphatic tuberculosis at 17 years of age, but had no history of sexually transmitted diseases, pelvic inflammatory diseases, endometriosis, uterine leiomyomas, deep sacral concavity, surgery, or congenital uterine malformations, such as uterus didelphys. The patient experienced infertility for two years caused by bilateral obstruction of the fallopian tubes with uterine retroversion, which was detected by hystero-salpingo-graphy in the regional hospital. She eventually became pregnant by follicular aspiration and IVF-ET, and had been treated with daily intramuscular progesterone since the procedure. Subsequent ultrasound scans revealed normal pregnancy progression.At 15 weeks and 2 days of gestation, small amounts of bloody vaginal discharge persisted for one day, and an ultrasound showed that the placenta covered the internal os of the cervix. She was then admitted with a diagnosis of a low position of the placenta and continued receiving progesterone therapy. She experienced worsening lower abdominal pain and difficulty with urination on fifth day after admission and was transferred to our tertiary hospital at gestational age 16 weeks and 1 day.A repeated ultrasound scan in the tertiary hospital confirmed uterine retroversion with a fundus bending to the posterior fornix, which made the fundus into the lowest point of the uterus. The cervix was anteriorly transfixed behind the pubic symphysis and was barely above the fundus; fundal implantation of placenta was revealed Fig. . A fetusIncarceration of the gravid uterus refers to the entrapment of the uterus in the pelvic cavity behind the sacral promontory. It has been estimated to affect 1 in 3000 pregnancies . In addiWe identified 162 cases including our own for analysis , abdominal pain (35.80%), constipation (6.79%), vaginal bleeding (6.17%), pelvic pain (6.79%), back pain (4.94%), tenesmus (1.85%), perineal pain (0.62%), and large painful mass prolapsed outside the anus (0.62%). Fourteen patients (8.64%) are asymptomatic, but also have delivered viable infants in the end, indicating that asymptomatic patients may better outcomes of pregnancy compared to those with severely symptomatic. Clinical complications usually occur after twelve weeks of gestation and are mostly related to the pressure from anatomical structures adjacent to the entrapped uterus, including lower abdominal and pelvic pain, dysuria, urinary frequency, urinary retention, overflow incontinence, rectal pressure, and worsening constipation , 92. AmoDiagnosis of uterine incarceration remains difficult because its symptoms are often non-specific and absent in early pregnancy . It is wIn general, the features of pelvic examination can be described as follows. The cervix is anteriorly transfixed behind the pubic symphysis, making it difficult to expose. Additionally, sacculation of the posterior wall of the vagina and posterior fornix bulge may be observed, and the fundus is palpable within the curvature of the sacrum and could not be moved. Ultrasound examination could confirm incarceration. Abdominal sonography shows an advantage over transvaginal sonography in exhibiting the position of the cervix and its internal ostium and in determining the relationships between vagina, uterus and bladder, when the cervix is elongated and wedged behind the symphysis . MRI is No single treatment can be deemed more successful than the others for gravid uterus incarceration. Most obstetricians tend to replace the uterus in its natural position as soon as possible after diagnosis . AccordiIn 162 reviewed cases, cases of incarceration recognized in the first or second trimester of pregnancy account for 67.28% (109/162). Treatment of reposition was successfully attempted in 83 cases. After reposition, 68 patients successfully delivered infants , 98–101,In the present case, the patient achieved correction from incarceration, which was presumed to be related to pelvic adhesions at 16 weeks, by manual manipulation, allowing for an attempt at repositioning. However, it could not be neglected that this patient had a past medical history that involved a high correlation with pelvic adhesions, which could prevent the gravid uterus from normal enlargement and ascent due to possible refractory incarceration. If this patient did not experience relief from incarceration via manual reposition, surgery with laparotomy or laparoscopy might be considered based on the patient’s strong desire to deliver a healthy child.In conclusion, we report a case of gravid uterine incarceration with a history of lymphatic tuberculosis and IVF-ET. The particular risk factors, including a past history of infection and pregnancy by ART, made our case a relatively specific. As illustrated in the review of similar case reports, gravid uterine incarceration is a rare condition, but serious late gestational complications or poor obstetric outcomes may occur. Early diagnosis is the key to successful treatment. In view of the lack of specific signs or symptoms, additional physical and imaging examinations are critical to early diagnosis of this condition. Appropriate treatment measures that are tailored to the different gestation weeks may improve pregnancy outcomes.Additional file 1. Case timeline. Describe the past medical history and interventions, summaries from initial and follow-up visits, diagnostic testing, interventions and relevant dates. (DOCX 17 kb)Additional file 2. PRISMA flow diagram. Search terms sequentially applied to all English reports published until 2016 (when the search was conducted): “(“retroverted uterus“ OR “retroverted gravid uterus“) AND (“incarceration” OR “incarcerated uterus” OR “incarcerated gravid uterus”) AND ”. The bibliographies of relevant articles were also searched by hand to identify additional eligible studies. (DOC 33 kb)Additional file 3. Case reports of incarceration of the retroverted uterus that have been reported in the literature . There were 162 cases published, including the present case, based on all available articles which could be searched for in the PubMed database. Basic information of the patients and associated information from each case report worldwide were summarized for readers to obtain a general view on this condition, including possible risk factors, symptoms, diagnosis, treatment and outcomes. (DOCX 76 kb)Additional file 4. Maternal characteristics with incarceration of the gravid uterus. Based on the 162 case reports, we conducted statistical analysis and summarized the maternal characteristics in the table. (DOCX 19 kb)


Adrenocortical carcinomas (ACC) are rare with an incidence of 0.7-2 per million population per year and account for only 0.05%-2% of all malignant tumors. While majority of the functional ACC present as Cushing syndrome, recurrent hyperaldosteronism from metastatic ACC is exceedingly rare. We describe a 67-year old female presented with hypertensive urgency & hypokalemia as a result of hyperaldosteronism from an 8-cm right ACC. She underwent a radical right nephrectomy with adrenalectomy that normalized her blood pressure. However, a few years later she presented again with resistant hypertension from hyperaldosteronism, raising the suspicion of recurrence of ACC. A contrast-enhanced CT scan showed a normal left adrenal gland but revealed pulmonary metastases of ACC based on a lung biopsy. Chemotherapy was complicated with side effects leading to refusal of further chemotherapy, henceforth requiring high dose of spironolactone for blood pressure control. Despite curative surgery, metastatic functional ACC should be considered in patients presenting with secondary hypertension from recurrent hyperaldosteronism, due to its high recurrence rate. Besides standard cancer surveillance after a curative surgery, meticulous monitoring of blood pressure is a simple yet crucial way to detect cancer recurrence early. Adrenocortical carcinomas (ACC) are rare malignancy with an incidence of 0.7-2 per million population per year . While ~The patient is a 67-year-old Caucasian woman who presented with uncontrolled hypertension. Patient did not have major medical issues until her early 60s when she started noticing rise in blood pressure during her routine clinic visits. Physical examination was within the normal limits except her systolic blood pressure was consistently in the 200s. Laboratory evaluation was significant for hypokalemia of 2.7 meq/L, bicarbonate -24 meq/L and creatinine of 0.92 mg/dl. Further evaluation for possible etiology of secondary hypertension revealed high aldosterone-renin ratio. Computed tomography (CT) scan to evaluate hyperaldosteronism showed a large mass in right adrenal gland measuring 10 x 7.8 cm . FurtherSecondary hypertension from aldosterone secreting ACC is not only uncommon but may also be unfamiliar to the practicing clinicians. Amongst these odd etiologies of hyperaldosteronism, pulmonary metastasis of previously resected ACC is a rare entity. Deckers et al reported a case of hyperaldosteronism from peritoneal dissemination of the previously removed ACC . Calvo-RMineralocorticoid antagonists or adrenalectomy can be the option of treatment for classical causes of primary hyperaldosteronism, whereas the only potential lifesaving option for ACC is complete radical resection . It is iOur case highlights the importance of considering functional ACC in patients presenting with hyperaldosteronism. Clinicians should be cognizant of high rate of recurrence of ACC and incorporate blood pressure monitoring besides standard radiographic and laboratory surveillance after a curative surgery to detect recurrence of ACC early.The authors declare no competing interests.


A 51-year-old male with three weeks of increasing back pain, sciatalgia, and recent fever spike searched medical attention. Clinical examination highlights positive Lasègue and tender right fossa. Laboratory testing returns high C reactive protein and leucocytosis.Pelvic MRI reveals an ill-defined large enhancing overall low ADC mass Figure , C with Frozen sections on surgical tissue samples (anterior to the right anterior iliac crest) reveals connective tissue, fat, muscle, and inflamed tissue without malignancy. Formalin fixed and paraffin embedded HE-stained sections Figure  shows stThe match of radiology and pathology findings is consistent with pseudotumoral Actinomycosis. Treatment with Amoxicillin/clavulanic acid results in pain relief and clear clinical and biochemical improvement. After six weeks of treatment there is marked volume loss of the mass and better delineation of bladder and psoas muscle Figure . The pusActinomycosis is a rare, chronic, destructive, invasive gram-positive bacterial infection. It has been reported as odontogenic, in the head and neck (mandibular osteomyelitis) and thoracic region. Abdominal, pelvic actinomycosis is even more rare and mainly found in immunocompromised patients. In women, IUCD-associated pelvic actinomycosis has been reported. Our patient, however, had no medical history except for orchitis treated three months ago. Without clinical tell-tale signs, e.g. cutaneous sinus tracts, diagnosing actinomycosis can be a challenge. Aggressive pseudo-tumor presentation and hematogenous spread to the central nervous system and lungs is possible .Suggesting the diagnosis of actinomycosis early is vital in patient management: timely antibiotics do cure the patient. Besides clinical and laboratory findings of inflammation, imaging features can provide us with a few hints. Actinomycosis is highly aggressive, infiltrating tissues, crossing fatplanes, and can invade muscles, organs, bones, and joints. Small non-enhancing microabcesses with striking low ADC might be a clue to look for. During treatment, measuring ADC values allows for non-invasive monitoring of pus content in the micro-abcesses.


The most reported locations of GS are the temporal lobe and the frontal lobe, while only eight cases of GS originating from the posterior cranial fossa are reported in the literature.  Gliosarcoma (GS) represents a rare, high-grade (WHO Grade IV), central nervous system neoplasm, characterized by a very poor prognosis. Similar to other high-grade gliomas, GS affects mainly adults in the 5th week of gestation when patient presented with signs and symptoms of life-threatening raised intracranial pressure. Surgical excision was followed by early recurrence and eventually disease progression because the patient refused adjuvant treatment to save her fetus.  We report the first case occurring during pregnancy in a 33-year-old patient. Diagnosis was obtained on the 15 GS should be considered in the differential diagnosis of posterior cranial fossa tumors with radiological features of meningioma or glioblastoma, even in young patients. To this regard, sarcomas, solitary fibrous tumors, and even metastases should be considered, especially in light of the tendency of GS to give rise to extracranial localizations. Whenever an aggressive management with radical excision and adjuvant treatment is not safely achievable, disease progression is likely to be unavoidable. The 201in males . GS may in males . The mosin males , 11–17. 2P1A1) on the 15th week of gestation was admitted to our Emergency Department with a 3-week history of headache and unsteadiness followed by rapid worsening within 5 days characterized by projectile vomiting, confusion, and psychomotor agitation. A brain magnetic resonance imaging (MRI) scan showed tetraventricular hydrocephalus secondary to obstruction of the cerebrospinal fluid through the foramina of Luschka and Magendie caused by a homogenously enhancing cortical-subcortical lesion localized on the right cerebellar hemisphere, responsible for perilesional edema and characterized by evidence of dural infiltration. The latter, initially mistaken for dural tail, oriented toward the suspicion of posterior fossa meningioma  through the right Keen's point and gross-total removal of the lesion, which resembled an aggressive glioma rather than a meningioma. The general anesthesia was carefully tuned to avoid any impact on the fetus' wellbeing; the postoperative course was uneventful, the EVD was removed within 1 week, and the patient experienced a full recovery with unremarkable neurological status at the time of discharge from the hospital. The histology and the immunohistochemistry analysis surprisingly gave a final diagnosis of GS see ; this waWhile several cases of supratentorial primary GS have been reported in literature, only eight cases with a posterior cranial fossa localization have been described so far see . This deOn CT scan, GSs tend to appear as slightly hyperdense lesions with perifocal edema and marked homogeneous contrast enhancement, whereas on T1WI and T2WI MRI sequences, they usually show low and high signals, respectively , 18. OveThe in-depth immunohistochemical analysis conducted in our case expands on the pathological data previously described . Of noteThe prognosis of GSs is utterly dismal, and our case is a perfect example of how fast this tumor can progress if not adequately treated with adjuvant chemo/radiotherapy after its primary excision. Stereotactic Gamma Knife radiosurgery has been described following subtotal resections or in case of local recurrence despite an initial management with conventional radiotherapy –36.The most noticeable research projects aimed at improving outcomes of GS are currently attempting to focus on new drugs with an antiangiogenesis profile –40. ThesOne final note revolves around the correlation between pregnancy and brain tumors: a large retrospective case series of patients with gliomas produced by the French Glioma Study Group suggests that tumor growth accelerates in 80% of patients and grade may evolve during pregnancy, resulting in a significantly higher frequency of seizures which pose specific challenges to the treating team in terms of pharmacological choices . In factThe biologic explanation for tumor growth during pregnancy stems from the observation that multiple hormones and growth factors produced during fetal development also enhance the oncogenesis cascade: for instance, the production of angiogenic factors such as placental growth factor which correlates with gliomas has been widely established . WhereasOverall, the open questions regarding the challenges of treating pregnant women with new diagnosis of gliomas revolves around (a) the decision to discourage continuation of pregnancy and when to do so, (b) what monitoring plan is more appropriate for the mother and her fetus, and (c) which chemo- and radiation therapy protocol to suggest after pregnancy. Given the relevance of this clinical scenario, the most recent systematic review concluded that a multicenter individual patient level meta-analysis collecting granular information on clinical management and related outcomes is needed to provide scientific evidence for clinical decision-making in pregnant glioma patients .Although the posterior cranial fossa remains a rare localization for primary GS, this aggressive tumor should be considered in the differential diagnosis of meningiomas, gliomas, sarcomas, and isolated fibrous tumors, even in young patients. State-of-the-art surgical techniques such as the introduction into the surgical cavity of a probe for intraoperative ultrasound or a 30-degree angle endoscope assume particular relevance to maximize resection and minimize the risk of intraoperative blood loss and postoperative hematoma. While maximal safe resection is fundamental in the management of patients with GS, without adjuvant chemo/radiotherapy the risk of early recurrence/progression of the disease is remarkably high. The case presented here was particularly challenging given the understandable desire of our patient to carry on with her pregnancy. In fact, this situation obliged the patient, as well as each medical practitioner involved in her management, to face a series of ethical questions, where compassionate care had to take priority over clinical pragmatism.


Endometriosis is a relatively common condition in women of reproductive age. Malignant transformation of intestinal endometriosis is a very rare event. We report a case in which a patient with a history of endometriosis underwent surgery for malignant intestinal endometriosis.A 55-year-old woman complained of rectorrhagia and intermittent abdominal pain. A neoplasm was revealed by colonoscopy, CT scan and F18-FDG PET/CT of the recto-sigmoidal colon. The patient underwent a rectal anterior resection, hysterectomy and bilateral salpingo-oophorectomy for treatment. According to the histological and immunohistochemical presentation, the diagnosis of endometriosis-associated recto-sigmoid cancer was confirmed. The patient was treated with adjuvant chemotherapy for 6 months. During the follow-up appointment 22 months later, there was clinical and radiographic evidence of recurrence in the rectum. The patient received chemotherapy again and will receive another surgery after two more cycles of chemotherapy.We report a case of malignant intestinal endometriosis. Although there is no standard therapy for malignant intestinal endometriosis due to the rarity of this disease, surgery and adjuvant chemotherapy seemed to be rational. This case indicates that local recurrence may be a common situation after standard therapy. Endometriosis is a common proliferative disease among women. It occurs in 4–17% of women of reproductive age. It refers to the occurrence of the endometrial tissue growing outside of the uterus. Many organs can be involved in this process, including the fallopian tubes, ovaries, vagina, cervix and the Pouch of Douglas; extragonadal sites include the gastrointestinal tract, bladder, lungs, central nervous system, and even the skin , 3. The A 55-year-old woman was referred to the Peking Union Medical College Hospital in September 2015 for rectorrhagia and intermittent abdominal pain lasting 6 months. The patient had a past medical history of ovarian endometriosis and had undergone excision of bilateral ovarian chocolate cysts in 1988 when she was 30 years old. Histological examination showed benign bilateral endometriosis of the ovaries. The patient was not treated with any hormonal therapy following hysterectomy. She has a familial history of endometriosis comprising her mother, one sister and one aunt.Physical examination of the patient was unremarkable, except for the tenderness of the left lower quadrant of the abdomen.Serum CA125, CEA, and CA199 were within a normal range. A colonoscopy was performed Fig. . F18-FDGThe endoscopic biopsy specimens were fixed in 10% buffered formalin and embedded in paraffin. Four μm sections were stained with standard haematoxylin and eosin (H&E). Immunohistochemical stains were performed using the following antibodies: CK7, CDX-2, CA-125, Villin (Dako); CK20 (OriGene Technologies); PAX-8, WT-1(XiYa Biology); P16, ER, PR (VENTANA); P53 . The H&E slides and immunohistochemical stains were reviewed by two pathologists. The excised bowel was sent for histological analysis. The pathologic specimen consisted of an ulcerated mass with grey, solid, friable cut surface. The mass measured 3.5 × 2 × 1.2 cm and was attached to the wall of the colon. The mass involved the muscularis propria macroscopically. Microscopic sections revealed a high-grade serous carcinoma extending through all of the layers of the intestine Fig. . Tumour The patient received adjuvant chemotherapy consisting of 175 mg/m2 Taxol and AUC5 carboplatin for eight cycles. The patient experienced reversible chemotherapy-induced myelosuppression and gastrointestinal reactions during chemotherapy. The timeline of the process was shown in Table At 23-months, the patient had a follow-up appointment and reported difficult defecation at the 22nd month. The CT scan, PET-CT and colonoscopy showed local recurrence in the lower rectum, which was confirmed by pathological diagnosis. The patient has received Taxol and carboplatin chemotherapy again and will receive another surgery after two cycles of chemotherapy.Endometriosis is a common gynaecological disease, but malignant transformation of endometriosis is rare. The malignancies arising from ovarian endometriosis are most common, followed by those arising from foci in the pelvic peritoneum and the colorectal tract , 4, 5. IEndometriosis-associated intestinal cancer is rare, and it requires a clinical suspicion for diagnosis. In the case for patients with a medical history of endometriosis and / or suspected colorectal malignancy, immunohistochemical examinations are important for differential diagnosis. Currently, surgery, chemotherapy, radiotherapy and hormonal therapy are selected according to the particular situation of the tumour and the individual. The prognosis of these rare tumours may be improved with the application of targeted medication and immunotherapy.


Cardiac angiosarcoma is a rare malignant tumor, for which only surgery has been proven to be effective to date. Currently there are no reports as to whether a postoperative regimen of ifosfamide, epirubicin, and recombinant human endostatin is effective.The patient presented to us with chest pain and dyspnea.Enhanced computerized tomography (CT) and positron emission tomography–computerized tomography (PET-CT) suggested pericarditis and an atrial perforation, but malignancy was suspected, so the patient underwent an operation to resect the tumor and repair. Pathology of the tumor reseccted at operation showed the tumor to be an angiosarcoma.2 once every 3 weeks). However, 2 cycles later, pulmonary and hepatic metastases were found. Chemotherapy was then changed to ifosfamide, epirubicin  and recombinant human endostatin (7.5 mg/m2 days 1–14) in 3 weekly cycles.After the surgery, the patient was stared on a paclitaxel chemotherapy regimen (135 mg/mThree cycles later, follow-up showed that chemotherapy had delayed progression of the pulmonary metastases, but that the hepatic node was still growing. The patient has now survived 8 months post surgery and is still on follow-up.This case shows us that operation on late stage cardiac angiosarcomas can alleviate a patient's symptoms; postoperative paclitaxel monotherapy was insufficient and ifosfamide and epirubicin plus recombinant human endostatin has a limited effect on late stage cardiac angiosarcoma. Studies with a larger sample size are needed for verification of these findings. Others such as chemotherapy or radiotherapy were effective only in sporadic cases.–21 In this case, we aimed to discuss the effect of ifosfamide and epirubicin (IE) plus recombinant human endostatin (rh-endostatin) following palliative surgery.Cardiac angiosarcoma is the most common malignant cardiac tumor.2The patient is a 61-year-old man, complaining of chest pain and dyspnea persisting for over 1 month. Medical history is nonsignificant, except for having stage 3 hypertension for 3 years, which had been well controlled with Irbesartan and dihydrochlorothiazide. Before coming to our hospital, a TTE (transthoracic echocardiogram) at a local clinic showed a 1.4-cm pericardial effusion, and a computed tomography (CT) demonstrated bilateral inflammatory effusion of lung and plural effusion. After being admitted to the local hospital, pericardialcentasis and thoracocentasis were carried out for symptom relief and diagnostic purposes. The pericardial fluid revealed nucleated cell +, red cell ++++, Rivalta test positive, CA125 (cancer antigen 125) 2013.1 IU/mL, keratin 19 13.7 ng/mL, and squamous cell carcinoma 4.2 ng/m. Pleural fluid results were Rivalta test positive, total protein 30.8 g/L, lactate dehydrogenase 238 IU/L, glucose 8.13 mmol/L, and adenosine deaminase 14 IU/L. Positron emission tomography–computerized tomography (PET-CT) revealed: increased pulmonary metabolism of bilateral posterior segments of lower lobe, inflammatory effusion considered; upper lobe emphysema, pleural effusion, and a right lower lobe node, considered to be hyperplasia; increased metabolism of right epicardium, inflammation considered . After 2 cycles, a CT scan showed enlarged pulmonary nodes and hepatic metastases were found, the chemotherapy regimen was changed to IE  + recombinant human endostatin (7.5 mg/m2 days 1–14), in 3 weekly cycles. After 3 cycles, primary focus and lung metastases were stable without progression, but hepatic metastases were still enlarging. It is now 8 months postsurgery and the patient is still attending follow-up. The patient has provided informed consent for publication of the case.The ethical approval and consent was checked by ethical department of the 1st affiliated hospital of Zhejiang University. The surgery was ordered on the 2nd day of admission. A mediasternotomy and cardiopulmonary bypass through the femoral artery and vein were chosen. When the pericardial cavity was opened, abundant blood clots were seen Fig. . After c3 The most common presentation for angiosarcoma is one of chest tightness and pain, as in our patient. When the tumor infiltrates the cardiac wall, necrosis, perforation, and increased vascular permeability can cause pericardial effusion, even cardiac tamponade and its subsequent symptoms.,3 Another reason for symptom occurrence is tumor bulk. When the tumor grows to a certain size, it can cause blood flow obstruction, valvular stenosis, or insufficiency. Other symptoms include cardiac arrhythmia and pulmonary embolism. Usually when patients feel discomfort, the tumors are of a certain size and distal metastases may have already developed. The most common location of distal metastasis is lung, but liver, spleen, bone, and brain have also been reported.Primary cardiac tumors are rare with a prevalence of 0.02%, amongst which 25% are found to be malignant by pathology. Cardiac angiosarcomas are the most common histopathologic type originating from vascular or lymphatic endothelial cells, accounting for 30% of cardiac malignant tumors. Contrast studies are also helpful in differentiating thrombus, myxomas, and infiltrative growth. TTE and TEE are the most common diagnostic methods of cardiac disease, as they are able to show tumor flexibility, mobility, and attachment width dynamically. Before surgery or autopsy, some characteristics may indicate malignancy; right atrium mass; an enhanced CT or MRI demonstrating necrosis, and an abundant blood supply within the mass; a broad base or poorly defined attachment and myocardial invasion. PET-CT/MRI is a novel diagnostic technique, combining functional imaging with anatomic imaging, showing tissue metabolic activity of a certain part of body. It is helpful in mass differential diagnosis, surgical planning, cancer staging, and radiotherapy. In malignant masses, the maximum standardized uptake value (SUVmax) is higher than nonmalignant mass. In this case, the discrepancy between PET-CT and ultimate diagnosis in location of neoplasm came from the proximity of the 2 layers of pericardium and pitfall resolution of PET-CT. Definitive diagnosis of cardiac angiosarcoma depends on the tissue biopsy resected in surgery or fine-needle aspiration. Histopathologic characteristics are vasoformative features including cytoplasmic vacuoles containing red blood cells, hemophagocytosis and endothelial wrapping. Immunohistochemical staining of cardiac angiosarcomas is always CD34, CD31, FKI-1, and von Willebrand factor positive. When planning for surgery or other treatment strategies, it is necessary to take all these test results into consideration.As the symptoms are nonspecific, diagnosis of cardiac angiosarcoma needs advanced testing like magnetic resonance imaging (MRI), CT, ultrasonography, PET-CT, and autopsy. Cardiac MRI and CT scans can demonstrate tumor location, size, and local invasion. POT1 gene missense mutation in TP53-negative Li-Fraumeni-like family, numerical and structural changes in the chromosomes and the p53 gene. KDR, also called VEGFR-2, plays an important role in angiogenesis and vasculogenesis. Current therapies focusing on this target include fruquintinib, lenvatinib, pazopanib, regorafenib, vatalanib, and tivozanib. Only pazopanib has been applied to a case on a patient with metastatic cardiac angiosarcoma and resulted in the distal metastasis’ complete remission. No target drug has been found for the other mutations. For the gene mutations run on our patient, NRAS , EGFR , KRAS , BRAF (V600E mutation), ROS1 fusion, and EML4-ALK fusion were all negative.Due to the rarity of cardiac angiosarcomas, only a few gene mutations have been reported so far, these include KDR (G618R), MDM4,–17 When compared to other histopathology types, primary angiosarcoma has a shorter survival time (5 vs 17 months). Distal metastasis is an important predictor of poor prognosis, as the 3rd case series reported, with a survival time of just 5 months compared to 15 months in a localized patient, and the 4th case series which showed an overall survival of 6 months for angiosarcomas with distal metastases compared to 19.5 months for localized ones. So far, surgical resection is the only treatment method proved to reduce local tumor mass and alleviate obstruction symptoms such as peripheral edema, chest pain, and dyspnea. As the 3rd and 4th case reports revealed, surgical resection of a cardiac tumor mass can improve survival time from 6 to 17 months and 5 to 17 months, respectively. Though there is no statistical evidence, it is reasonable to believe that the prognosis of R0 resection  is better than R1  and R2 .Prognosis of primary cardiac sarcoma is especially poor for angiosarcomas. According to the 4 existing case series, survival time is 7, 5, 26.6, and 13 months. There are 3 cases reported of treating cardiac angiosarcoma with paclitaxel to good effect, from partial response to complete remission of primary tumor and distal metastasis but unfortunately it was not work in our patient.–21 Ifosfamide is a broad used chemotherapy drug in angiosarcoma, combining with doxorubicin achieved a well effect was reported and epirubicin, another anthracycline drug, always combined with ifosfamide as chemotherapy regimen for soft-tissue sarcoma. Endostatin is a wide spectrum tumor angiogenesis inhibitor, with slight side-effects. As angiosarcomas originate from endothelial cells, it is reasonable to think it may work in this patient, though there was no clinical trial or cases reported, and it turned out the effect is limited.Paclitaxel is a chemotherapy drug targeting tubulin which has been used effectively in angiosarcoma.Reviewing this case, it was a dilemma whether to choose surgery or not even when malignant cardiac tumor is suspected. According to the report, the operation undoubtedly can improve survival time, especially for R0 resection, but not in R1 or R2 resection because it can delay the chemotherapy or radiotherapy and cause tumor cell spreading through blood stream. But acute cardiac tamponade can result at any time once the tumor is of sufficient size, so even though circulation blockage was slight and distal metastasis were suspected, surgery was necessary for this patient. Furthermore, it we would not have been able to start the aggressive chemotherapy regimen without have positive tissue pathology.4Cardiac angiosarcoma is a rare but aggressive malignant tumor with poor prognosis. Early diagnosis and treatment can improve a patient's prognosis markedly; however, early metastasis are also common, so it is necessary to evaluate the stage of the cancer, local invasion and distal metastasis to decide on an appropriate therapeutic strategy. For cardiac angiosarcomas with distal metastases or those considered locally unresectable, surgery should still be considered as it has the potential to alleviate symptoms and possibly prevent cardiac tamponade. Postoperative paclitaxel monotherapy was ineffective and IE plus rh-endostatin has a limited effect on late stage cardiac angiosarcoma. Further clinical research of larger numbers of patients is needed.Lijun Jiang, surgeon of the 1st affiliated hospital of Zhejiang University, cardiac surgery doctor of Peking Union Medical College, visiting scholar of Stanford University School of Medicine.Conceptualization: Lijun Jiang.Data curation: Kexin Shi.Resources: Lijun Jiang.Supervision: Lijun Jiang.Writing – original draft: Xingjie Xu.Writing – review & editing: Xingjie Xu, Henry Davies.


Unilateral cervical chondrocutaneous branchial remnants (CCBRs) are rare, but when present, are typically located over the lateral aspect of the neck along the anterior margin of the sternocleidomastoid muscle. A CCBR in this location is called a choristoma. Here, we describe a choristoma in a 25-year-old female who disclosed a documented diagnosis of Meniere’s disease, and an expressed interest in bearing children within the immediate future. She presented with a unilateral swelling, located subcutaneously, midway over the anterior margin of the right sternocleidomastoid muscle. Due to her history, and the risk of possible radiological exposure to her fetus, an ultrasound-based examination of the neck and cardio-abdomino-pelvic organs was performed. Our clinical findings, details of the ultrasound results, and surgical data will be described. Cervical chondrocutaneous branchial remnants (CCBRs), being present both unilaterally or bilaterally, are rare lesions of embryonic origin. Chondrocutaneous branchial remnant is the universally accepted term for the presence of heterotopic cartilage subcutaneously in the cervical region . To dateCervical chondrocutaneous branchial remnants located in the cervical region, particularly along the anterior border of the sternocleidomastoid muscle, are known as choristomas. By definition, a choristoma is a benign growth consisting of normal tissue located at an abnormal location. They may present as cysts, sinuses, fistulae, or cartilaginous remnants . TheoretA 25-year-old woman temporarily living in Grenada visited a local otolaryngologist presenting with a past history of Meniere’s disease without treatment . On presentation, she expressed that the motivation behind the visit was to investigate the significance of her neck swelling. She had missed her period for two consecutive months. Routine examination showed a swelling over the right sternocleidomastoid muscle, midway along its anterior border Figure . The sweFamily history was unremarkable. No visible congenital anomalies such as aberrant implantation of the ears, auricular pits, auricular appendages, or fistulae were present. The swelling was about 0.75 cm in length x 0.5 cm in width. Palpation revealed a nontender structure in the subcutaneous plane unattached to the underlying tissue and mobile in every direction. The overlying skin was partially attached to the swelling, but it did not originate from within the skin (the skin above the swelling could be pinched up). No cervical lymph nodes draining the area were palpable. Examination of the left neck was unremarkable.Due to her anxiety with respect to conceiving, and the risk of teratogenic radiological exposure, an ultrasound-based examination of the neck and cardio-abdomino-pelvic organs was performed to identify if her CCBR had associated defects. Ultrasound showed a hypoechoic (likely cartilaginous) mass measuring 0.94 cm x 0.43 cm Figure . AbdominThe patient did not request surgical excision, as she was asymptomatic with the lesion for the duration of her lifetime. She was told, and reassured, that the ultrasound examination showed a benign cartilaginous mass. The ultrasound of her heart, abdomen, and pelvic organs also showed no abnormalities. This diagnosis allowed the patient to feel some comfort after what she assumed could have affected the health of her baby.Cartilaginous embryological remnants in the neck are rare, in contrast to the prevalence of accessory tragi, a fairly common finding compared to CCBRs . EmbryolDiagnosis of cartilaginous choristoma was made. Other possible reasons for a swelling in the neck in this presented case could be a thymic cyst, thyroglossal duct, branchial cleft cyst, pilomatricoma, or hamartoma Table . GoldenhHamartomas are described as excessive, focal over-growth of cells and tissues native to the organ in which it occurs . As embrA pilomatricoma is an abnormal swelling which is commonly located on the neck or head. However, on physical examination pilomatricomas are identified commonly by either or both the tent sign and the teeter-tot sign . The tenThe other differentials are possibilities, however, were overshadowed by the final diagnosis of CCBR due to all the facts presented within the case.Treatment is complete surgical removal as promptly as possible to get an exact histopathological diagnosis . If the A choristoma is a CCBR located specifically in the cervical region near the sternocleidomastoid muscle, particularly, a benign swelling that is firm on palpation and found subcutaneously. The choristoma in this case was present since birth on the right sternocleidomastoid muscle, had no size changes, was never infected, and not painful. This, with the assistance of ultrasound further confirming a hypoechoic cartilaginous mass, allowed the diagnosis of a choristoma to be made in this case. Other differential diagnoses were ruled out on further investigation.