Hydatid disease is caused by infection of Echinococcus Granulosus. Usually Hydatid Cysts occur in the liver and lungs. Presenting hydatid cysts in bone without hepatic affectation is rare and occurs in 0.5–2% of cases. Hence, this rare case makes the diagnosis difficult for the clinicians and, as a result, misdiagnosis of sacral Echinococcosis is common.The authors report on a 47-year male with primary sacral hydatidosis and 34 years of recurrence. He was admitted with compressive neurological symptoms like tingling pain, numbness, sciatica and foot drop. He has undergone 8 operations and has been treated with Albendazole. He has developed a Sacro-cutaneous fistula.When assessing sciatica, low back pain or lower limb weakness the pelvic cavity should be examined for hidden disease that might explain the neurological symptoms.A missed diagnosis of osseous Hydatidosis could be devastating. Accordingly, the sacral Hydatid cyst must be included as a differential diagnosis for compressive neurological symptoms. In clinical practice, surgery remains the gold standard for treating osseous Hydatidosis. •The symptoms of osseous echinococcosis are non-specific and widely variable.•Sacral hydatid cyst it should be included in the differential diagnosis of any bone mass caused compressive neurological symptoms.•Treatment of hydatid cyst is primarily surgical, demanding total removal without rupture.•Posterior approach is preferred over anterior due to feasibility of the intervention and decrease of the recurrence rate. It mainly found in the liver (~70%) and the lungs ~20%), but it can be found in all the body, including the bone (~0.5–4%) of all cases of Echinococcosis described in the literature , cases w0%, but iThe infestation is prevalent in most parts of the world, especially in sheep farming and cattle farming areas of the Mediterranean Sea, Asia, North and East Africa, South America, Australia, and the Middle East.Spinal echinococcosis’ symptoms are mostly related to pressure exerted by the cyst itself on nerve roots, leading to neurological manifestations of peripheral sensitivity loss, radicular pain, paraparesis, paraplegia, sphincter disturbance or bladder dysfunction .2A 47-year male was referred to our surgical clinic in 2006 with back pain, numbness in left leg, perineum. Neurological examination revealed perianal hypoesthesia and gradual paraparesis in the left muscle group with no bladder disorders.Neurological examination showed a weakness of the lower extremities, bilateral hypotonia and moderate flexion contracture of the left ankle. The Babinski response was positive.Physical examination showed that his reflex test was normal. Due to his complaint, the patient does not have a normal gait, he depends more on his right leg and avoids prolonged pressure on the left one. His symptoms were initially misdiagnosed in 1985 as Ankylosing Spondylitis and treated with for 5 years accordingly.His diagnosis of Hydatid Cyst was first made in 1990 by Abdominopelvic Ultrasonography that showed an expansive lytic lesion affecting the left iliac wing and the sacrum with cortical bone destruction and was later confirmed by pelvic CT scan (computed tomography) that revealed multiple expansive osteolytic lesions containing trabeculae with cortical thinning located in the left sacral ala and spreading to the left iliac bone.  there was an expansible lesion with high signal intensity in T2-weighted images and low signal intensity in T1-weighted images, sacral destruction and replacement by a multiloculate cystic mass . ClinicaThe treatment regimen included both surgical decompression of the lesion site and medical prescription of Albendazole.The patient had his first surgical procedure in 1995 with year-long physiotherapy, the patient remained asymptomatic for four years. Then, two operations were done in 1999 and 2003 accompanied by the deterioration of mobility in the left lower limb as signs of drop foot have appeared. Five later operations dating in 2006, 2009, 2012, 2015 and 2018, were performed with an orthopedic surgeon due to change in intervention's initial site “posterior intervention”.Following the last operation in 2018, the Patient has developed a Sacro-cutaneous fistula, through which daughter cysts would spring up . After eParaparesis and perianal hypoesthesia were improved almost completely in later controls extending over 3 years. The cutaneous fistula allowed the cysts to exit continuously and prevented them from gathering.3Hydatid disease is a well-known condition that affects liver and lungs. And although it is rarely affects the bone, it should be included in the differential diagnosis of any bone mass caused compressive neurological symptoms .When the bone is affected by hydatid cysts, it doesn't form encapsulated cysts like in other organs . For thiAlthough hydatid disease of bone is rare, it should be included in the differential diagnosis of any bone mass discovered in the human body.In a systematic review of 721 cases of Osseous echinococcosis, 95% of cases required one intervention, although in other cases up to four interventions were performed. Notably, complete excision of the lesion was only accessible in 16% of cases. The two main primary procedures in Spinal Echinococcosis would be decompression of the affected spinal root and stabilization of column following resection [In order to decrease recurrence, intraoperative irrigation with scolicidal solutions is advised. This can be performed using hypertonic saline, cetrimide, silver nitrate, sodium hypochlorite or glycerin .Formaldehyde was avoided in the last 5 surgical procedures due to its irritation features on root nerves being exposed using the posterior approach. This method was preferred over anterior approach due to feasibility of the intervention and decrease of the recurrence rate.In our case, Diagnosis was delayed because the pelvic cavity was not studied when radiculopathy symptoms started and the patient did not interact with any of the animals in his history.Recurrence is referred to the spongy characteristic of the sacral bone as those small spaces would allow seeding of the daughter cysts and are not accessible through surgical intervention.We noticed eruption of hydatid cysts through the Sacro-cutaneous fistula after exertion, such as holding heavy objects. This can be interpreted by the existence of hydatid cysts in the iliopsoas muscle so that any contraction of the muscle would cause the cysts to come out. We noticed closure of the fistula and relief of the pain when the patient does not exert much effort.4Treatment of hydatid cyst is primarily surgical, demanding total removal without rupture.In areas where hydatid disease is an endemic, sacral hydatid cyst it should be included in the differential diagnosis of any bone mass caused compressive neurological symptoms for early and correct treatment in order to decrease recurrence. In this case report we identified a case of recurrent osseous hydatidosis that was initially misdiagnosed which in turn led to mistreatment.Not commissioned, externally peer reviewed.Authors declare that there is no conflict of interest.This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors.We have the patient’s approval; no more approvals are required. The work has not been published previously.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Ammar Niazi and Kusay Ayoub diagnosed and treated the patient, Abdelaziz Alibraheem and Yasser Hendawi searched the literature, Mohammad Karam Abouzied, Samer Rajab Basha and Shaban Suliman wrote the manuscript and Abdelaziz Alibraheem and Ahmad Al-Mouakeh critically revised the article. All authors approved the final version of the manuscript.Abdelaziz AlibraheemEmail: abdelazizalibraheem@gmail.com


Giant cavernous hemangioma involving the nose extending to the nasopharynx and oropharynx with complete obstruction of the airway is very unusual and is yet to be described in the literature. In the present case, we have described a giant cavernous hemangioma successfully managed with endoscopic excision.A 38-year-old male patient presented with recurrent nasal bleeding for 24 months and progressive obstruction of the right nasal cavity for 8 months. Diagnostic nasal endoscopy showed a greyish mass filling the whole of the right nasal cavity and contrast-enhanced CT scan of the nose and paranasal sinus revealed a large heterogeneous contrast enhanced lesion in the nasal cavity. The endoscopic biopsy was suggestive of cavernous hemangioma. Endoscopic excision was done and the patient has been followed up for the past 12 months without any recurrence of the disease.Cavernous hemangioma is an uncommon benign entity of the nose and paranasal sinus. Due to the nonspecific clinical and radiological pictures, it is often a challenge for the preoperative diagnosis. A high index of suspicion and complete understanding of the clinicopathological profile of the patient is vital as the major differential diagnosis is the sinonasal malignancy simulating with a similar clinical picture. Although hemangiomas are common vascular malformation in the head and neck, sinonasal hemangiomas are rarely encountered in common clinical practice . The majA 38-year-old male patient presented with recurrent nasal bleeding for 24 months and progressive obstruction of the right nasal cavity for 8 months. Anterior rhinoscopy did not reveal any significant abnormality except an organized clot and diagnostic nasal endoscopy showed a greyish mass filling the whole of the right nasal cavity extending posteriorly towards the nasopharynx .Contrast-enhanced CT scan of the nose and paranasal sinus revealed a large heterogeneous contrast enhanced lesion filling the whole of the right nasal cavity extending the nasopharynx hanging into the oropharynx . Routine hematological examinations, including the coagulation profile, were found to be normal. Keeping in mind the possibility of sinonasal malignancy, the endoscopic biopsy was taken which revealed multiple dilated vascular channels of varying caliber, containing blood and fibrin, lined by flattened endothelium in the sub-epithelial tissue (H& E 20X),confirmed to be a cavernous hemangioma . The histological feature was suggestive of cavernous hemangioma. CT angiography was advised which did not reveal any dominant feeding vessel of the tumor .After the informed written consent, the patient was planned for the endoscopic excision of the tumor under general anaesthesia. The tumor was completely excised through endoscopic modified Denker’s approach without any significant blood loss. There were no significant intraoperative or postoperative complications detected in the patient. The final histopathology was confirmed to be the same as that of the preoperative diagnosis. The patient has been in regular follow-up for the past 12 months with serial nasal endoscopy without the recurrence of symptoms.Although capillary hemangioma is very often encountered in the nose and paranasal sinuses, cavernous hemangiomas are very rare and only a hand full of cases have been described in the literature –9. CliniOn diagnostic nasal endoscopy (DNE), it usually presents a pedunculated or sessile greyish/reddish mass in the nose with the special predilection to the females in their second to the third decade of life -7. In contrast, our patient was a 38 year male presented with complete nasal obstruction with the history of intermittent epistaxis and on DNE, the mass was found to occlude the while of the choana. In contrast enhanced CT scan, it usually appears as a non-homogenous mass due to the presence of hemorrhage and necrosis in most of the cases. Like a malignant lesion, it can cause thinning and erosion of the bone and can compress the adjacent structure . In the Treatment is primarily surgical, ranging from complete excision to local resection sparing the vital structures and the approaches could be open, endoscopic or combined depending on the extent of the lesion ,12. OverCavernous hemangioma is an uncommon benign entity of the nose and paranasal sinus. Due to the nonspecific clinical and radiological pictures, it is often a challenge for preoperative diagnosis. A high index of suspicion and a complete understanding of the clinicopathological profile of the patient is vital as the major differential diagnosis is the sinonasal malignancy simulating with a similar clinical picture.


Extramedullary hematopoiesis (EMH) occurs in patients with hematologicdisorders, but rarely within the paranasal sinuses. We report a case of EMHin a 17-year-old male with sickle cell disease (SCD) who presented withoccipital pain and sinusitis. A computed tomography (CT) scan demonstratedheterogeneous opacification of the right maxillary sinus concerning forallergic fungal sinusitis or a fungal ball with bony erosion. He was takento the operating room for endoscopic biopsy and a limited endoscopic sinussurgery. Grossly, his maxillary sinus was filled with spiculated osseoustissue. Final pathology demonstrated active hematopoietic bone marrowfilling the sinus.We present a case report and literature review of sinonasal EMH.We identified 14 articles with 15 patients. EMH was typically associated withSCD or beta thalassemia. The average age of presentation was 30. There was amale sex predilection with a ratio of 11:15. The most common presentingsymptom was a headache and nasal obstruction (33% for both). The most commonfinding on CT was a soft tissue expansile mass (73%). The most commonlyaffected location was the maxillary sinus (60%).This case report serves as a reminder to consider EMH as an uncommon cause ofsinus opacification, particularly in patients with SCD or beta thalassemia.The expansion of hematopoietic tissue may be identified as a sinus mass onCT. By recognizing the potential manifestations of chronic anemia, anaccurate and timely diagnosis can be made. A common clinical manifestation of sicklecell disease (SCD) is chronic anemia. The body responds by increasing hematopoiesis.RBC production classically occurs in the bone marrow of the long bones, pelvis,spine, and sternum.1 With chronically elevated erythropoietin levels, organs such as the spleenand liver help augment the body’s RBC supply.2 These organs are areas of fetal erythropoiesis that do not typicallycontribute to physiologic RBC production in adults. Other, less commonly involvedorgans that have been documented as sites of extramedullary hematoposesis (EMH)include lymph nodes, paravertebral regions, intra-spinal canal, pre-sacral region,nasopharynx, and paranasal sinuses.3Approximately 1 in 600 African-Americans are homozygous for the sickle cell gene.4 This is more common in patients with SCD and thalassemias when erythroidmarrow activity is high. Paraosseous EMH may remain clinically silent until thereare enough cells to form a tumor-like mass associated with symptoms.There are 2 types of EMH, extraosseous and paraosseous. The extraosseous form occursin organs with multipotent stem cells, such as the spleen and liver, that canproduce hematopoietic foci remote from bone marrow. This is seen in conditions thatprevent effective production of RBCs within the marrow, such as myelofibrosis.Patients typically develop splenomegaly or hepatomegaly that may manifest as earlysatiety, bloating, pressure, or abdominal pain. In contrast to extraosseus EMH,paraosseous EMH occurs just outside of the bone due to the herniation of hyperactive marrow.Although it is rare to see EMH within the sinonasal cavity, based on our literaturereview we believe this is the 16th reported case . The preA 17-year-old African-American male with SCD presented to his primary care physicianwith a 1-month history of severe occipital head pain, left facial numbness, and lefteye droop. His SCD had previously caused several emergency department admissions forpain and sickle cell crisis. Magnetic resonance imaging (MRI) was ordered to ruleout a possible skull infarct secondary to a sickle cell crisis. The MRI image didnot show an infarction, but the patient’s left maxillary sinus was completelyopacified and enlarged compared to the contralateral side. He was referred tootolaryngology for further evaluation.The patient presented to otolaryngology clinic with sinusitis and occipital pain. Thephysical examination was normal, but nasal endoscopy demonstrated medialization ofthe uncinate and medial maxillary wall within the left nasal cavity. There were nopolyps or purulence noted on either side. A noncontrast computed tomography (CT) wasordered and revealed opacification and expansion of the left maxillary sinus thatoccluded the ostiomeatal unit consistent with a fungal ball . Due to The patient was observed postoperatively without complications and discharged home.He has not experienced any subsequent episodes of facial pressure or pain since thesurgery. Nevertheless, he continues to have frequent exchange transfusions andstruggles with pain management for his underlying SCD.Data for the case report were collected from the electronic medical health record. Aliterature review was performed by searching the following keywords in PubMed:extramedullary hematopoiesis and paranasal sinus. Fifteen articles were identified. AfterrEMH was typically associated with SCD or beta thalassemia, with an equal prevalenceof each among the cases reviewed. There was a male sex predilection with 11 of 15 ofthe cases occurring in male patients. The average age of presentation was 30, with aminimum age of 28 months and a maximum of 72 years. The median age was 18 years.Interestingly, the 3 oldest patients had acquired conditions rather than inheriteddefects. Two of the patients had myeloproliferative disorders and the other hadPaget’s disease.5Any sinus can theoretically be affected, but the most commonly affected location wasthe maxillary sinus. There were 9 cases with maxillary involvement, 6 with sphenoidinvolvement, and 4 with ethmoid involvement. Nasal obstruction and/or headache was apresenting symptom in 33% of patients. Recurrent epistaxis was the presentingsymptom in 20% of patients. One reported case of sinonasal EMH was incidentallyidentified on CT scan after a motor vehicle collision.517 On CT imaging, thehematopoietic tissue typically appears as a soft tissue mass that may demonstratecalcifications. Several cases involving the paranasal sinuses showed boneremodeling, bulging, and protrusion into other sinuses. These findings may resemblethe appearance of allergic fungal sinusitis on imaging, and thus many of thepre-operative diagnoses included fungal etiologies. However, in the few cases thatutilized MRIs, sinonasal EMH demonstrated signal intensity and enhancement similarto that of red bone marrow.1EMH can present as a sinus opacification in patient chronic anemia from diseases suchas thalassemias, SCD, and myeloproliferative disorders. After reviewing theliterature, common themes were identified in the detection and diagnosis of EMH.2,5–17 O6 In addition to surgery, 2 of 15 patients were treated with exchangetransfusions which serve to address the underlying anemia and decrease the demand onthe extramedullary marrow.7 Normalization of hematocrit levels suppresses the hematopoietic foci to theextent that it no longer produces symptoms. Even in the more serious cases, withseizures, proptosis, and vision loss as presenting symptoms, patients weresuccessfully managed with conservative management.No definitive therapeutic guidelines for paranasal sinus EMH exist. Twelve of 15cases proceeded with a biopsy to confirm the diagnosis and to exclude neoplasm.Several authors have suggested that the hematopoietic tissue should not be removed,as it serves as a vital contributor to the patient’s RBC reserve.Extramedullary hematopoiesis within paranasal sinuses is a rare diagnosis in apatient with chronic anemia. Imaging is frequently concerning for allergic fungalsinusitis. A biopsy is required to diagnose EMH and rule out an underlyingmalignancy. We recommend that EMH be included in the differential diagnosis of asoft tissue, expansile sinus mass presenting in patients with known hematologicconditions.


Chordoma is a low-grade notochordal tumor of the skull base, mobile spine and sacrum which behaves malignantly and confers a poor prognosis despite indolent growth patterns. These tumors often present late in the disease course, tend to encapsulate adjacent neurovascular anatomy, seed resection cavities, recur locally and respond poorly to radiotherapy and conventional chemotherapy, all of which make chordomas challenging to treat. Extent of surgical resection and adequacy of surgical margins are the most important prognostic factors and thus patients with chordoma should be cared for by a highly experienced, multi-disciplinary surgical team in a quaternary center. Ongoing research into the molecular pathophysiology of chordoma has led to the discovery of several pathways that may serve as potential targets for molecular therapy, including a multitude of receptor tyrosine kinases , downstream cascades , brachyury—a transcription factor expressed ubiquitously in chordoma but not in other tissues—and the fibroblast growth factor [FGF]/mitogen-activated protein kinase kinase [MEK]/extracellular signal-regulated kinase [ERK] pathway. In this review article, the pathophysiology, diagnosis and modern treatment paradigms of chordoma will be discussed with an emphasis on the ongoing research and advances in the field that may lead to improved outcomes for patients with this challenging disease. Chordoma is a notochordal tumor of the skull base, mobile spine and sacrum which remains a considerable treatment challenge despite over 150 years of surgical and chemoradiotherapeutic advances since the first description of the entity by Virchow in 1857 , inevitaExtent of tumor resection is the most crucial factor in the prognosis of chordoma, with wide, en bloc resection being the surgical gold standard. Treatment by an experienced surgeon in coordination with a multidisciplinary team is paramount to achieving the best patient outcomes ,16,17,18Chordomas are presumed to derive from undifferentiated, extradural, vestigial remnants of the notochord, an embryonic structure that coordinates cell fate and development. This presumption is based on studies showing that the sites at which chordomas originate correspond well to the location of embryological notochord cell rests ,26, as wThe notochord itself is a longitudinal, axial structure present centrally  within humans and all other members of the phylum Chordata during embryological development. It courses cranially from the sacrum within the boundaries of what will become the vertebral bodies, exiting this path only briefly—ventral to what will become the clivus—to contact pharyngeal endoderm before returning dorsally, where it terminates at the level of the dorsum sellae of the sphenoid bone. The notochord provides position and fate information to the developing embryo and serves a structural role, acting as a primitive axial skeleton during embryonic development. In some vertebrates, the notochord is present throughout life and functions as a support structure during locomotion and other tasks. In humans and other higher vertebrates, however, the notochord ossifies to form the vertebral bodies and completely regresses within the first few years of life. Following this process only a small remnant of the notochord remaining as the nucleus pulposis of intervertebral discs ,26,29.Chordomas do not, however, arise from the nucleus pulposis. In fact, chordomas seldom involve the intervertebral disc—but rather arise from aberrant vestiges of the notochord that failed to properly regress. Multiple notochordal vestiges have been described from which chordoma may arise, such as ecchordosis physalifora, a hamartomatous mass found dorsal to the clivus, which, according to a retrospective series by Mehnert et al., may be seen incidentally on magnetic resonance imaging (MRI) in 1.7% of patients . Other eThough BNCT and other ectopic notochordal remnants have been hypothesized to serve as the precursors for chordoma , the incChordomas may theoretically arise from any anatomical location along the length of the notochord’s former embryological course. Though chordomas were previously thought to have a predilection for the clivus and sacrum, recent epidemiological studies suggest that they occur with a similar incidence in the skull base, mobile spine and sacrum . The pre6p27) encodes brachyury, which, as mentioned previously, is a transcription factor required for notochord development from mesodermal elements. Brachyury is expressed transiently in embryonic notochord and normally silenced in post-developmental tissues but has been shown to be aberrantly re-expressed in chordoma and some evidence exists for its causative role in chordomagenesis. Knockdown of brachyury expression, for instance, leads to suppression of chordoma cell line growth in vitro [6p27 [The T gene /mitogen-activated protein kinase kinase (MEK)/extracellular signal-regulated kinase (ERK) pathway mediates expression and signaling of brachyury in chordomas and may also play a role in chordomagenesis. Fibroblast growth factor is a known regulator of brachyury expression in normal tissue and FGF2, FGF3, MEK and ERK have all been shown to be expressed in cultured chordoma cell lines. Exposing these cell lines to an FGFR inhibitor reduced MEK/ERK phosphorylation and brachyury expression, inducing apoptosis and restricting cell growth . FurtherReceptor tyrosine kinases (RTKs) are another area of interest in the pathogenesis of chordoma and other cancers. RTKs are transmembrane proteins that translate extracellular stimuli into intracellular signaling cascades responsible for cell growth, differentiation and proliferation. Dysfunctional signaling within an RTK cascade can thus lead to the aberrant behavior observed in tumor cells and oncogenic mutations have been documented in a number of different RTK families . With chThe incidence of chordoma is only 0.08 in 100,000 individuals . Males , mobile spine 32.8%) and skull base (32%) 2.8% and ,45 and a6p27), which encodes brachyury, as mentioned previously [Chordomas are typically sporadic but a number of suspected familial forms have been documented, with a variety of familial genetic mutations identified in these cases, including recurrent germ-line mutations in the T gene , chordomas appear as a midline, well-circumscribed and expansile soft-tissue mass with lytic destruction of surrounding bone . IntratuOn T1-weighted magnetic resonance images (MRI), chordomas exhibit intermediate to low-signal intensity . Small fConventional angiographic evaluation of chordomas is typically non-specific and abnormal vasculature is rare. Although intracranial chordomas and those originating in the cervical spine have a propensity to envelope major arteries, luminal narrowing is not frequently seen due to the soft consistency of most chordomas. MR angiography and venography may be useful when evaluating for encasement of important vessels but conventional angiography is reserved for cases in which significant encasement or displacement of major arteries is suspected or when vessel test occlusion and/or embolization is being considered in efforts to achieve a more radical surgical resection .Though the diagnosis of chordoma may be suspected based on patient presentation and radiographic imaging, diagnostic certainty requires histopathological evaluation. CT-guided needle biopsy and tissue diagnosis is often recommended prior to definitive surgical intervention, as the differential diagnosis for chordomas includes other entities for which treatment approaches may vary .Virchow was the first to characterize the histology of chordoma . He descFour subtypes of chordoma are currently recognized: conventional (which is the most common), chondroid , poorly differentiated and dedifferentiated or sarcomatoid chordoma  ,58,59,60Chordomas pose a considerable treatment challenge due to their midline location, predilection for involving critical neurovascular anatomy, indolent growth patterns, tendency to seed/recur and resistance to traditional chemoradiotherapeutic modalities. Advances in surgical techniques and approaches, image guidance, radiotherapy strategies and the emergence of promising targeted molecular therapies, however, are beginning to change the outlook of the disease. In order to achieve the best possible patient outcomes, it is crucial that patients with chordomas are treated by multidisciplinary teams in experienced quaternary centers, where a well-planned biopsy, appropriate staging, maximal safe surgical resection, molecular sequencing, modern medical therapy, radiotherapy and relevant clinical trials are all accessible and included in the decision-making process. Performance of even some invasive diagnostic procedures outside of an experienced center has been shown to negatively impact patient prognosis .Surgical resection is currently the mainstay of treatment for chordoma and extent of surgical resection is one of the most important prognostic factors for patients with this disease ,65,66,67Biopsy of suspected chordomas should be performed in an experienced center prior to surgical intervention, as the diagnosis may significantly alter the overall plan. Recommendations for surgical margins may vary based on the grade of tumor and the presence of metastases and in some cases, radiotherapy and/or palliative procedures may be recommended rather than aggressive resection. CT-guided needle biopsy is recommended rather than open biopsy for suspected chordomas of the mobile spine and sacrum, as seeding is likely to occur as a result of open biopsy, but when a needle is used, the tract may be resected at the time of definitive tumor resection.The preferred surgical approach for skull base chordomas and chordomas of the craniocervical junction depends on the precise location and extent of the tumor, with anterior, anterolateral and lateral approaches being favored. Often a combination of approaches is used and in many cases, several repeat resections are needed throughout the course of the disease. In a large series by Wang et al., 238 patients with skull base chordomas treated over a period of 10 years were reviewed and the tumor location, surgical approach and outcomes were described .Conventional and expanded endoscopic endonasal approaches (EEA) are favored by many surgeons for resection of chordomas and other skull base tumors due to the minimally invasive nature of the approach (a physiologic approach corridor which obviates the need for a skin incision) and the increasingly favorable outcomes reported in the literature with this technique ,71. ChorGiven the tendency for chordoma to seed and recur locally as a result of capsule violation, precise characterization of resection margins is paramount when discussing surgical approach and outcomes and the term “en bloc” is lacking in this regard. As such, the Enneking or the Weinstain-Boriani-Biagini classifications are the preferred means of characterizing spinal and sacral chordomas and providing recommendations regarding margins of resection ,73,74,75As with chordomas of the mobile spine, sacral chordomas are suited to classification according to the Enneking staging system and the recommendations for intralesional, marginal or wide resections margins translate well to sacral chordomas. Performing wide or marginal resections of sacral chordomas, however, is made challenging by the uniquely complicated surrounding anatomy, including nerves contributing to lower extremity function, sexual function and bladder/bowel function, bony attachments to the lumbar spine and pelvis, the iliac vessels, surrounding gluteal and piriformis musculature, as well as the retroperitoneal and pelvic viscera, which may be involved depending on the extent of the tumor. Multiple surgical specialties  will likely need to be involved and multiple approaches  may be required to achieve the desired margins without capsule violation.Total sacrectomy is frequently performed in two stages, with an anterior approach being utilized first to create the desired margins between normal anatomy and the anterior aspect of the tumor, ligate and mobilize vessels and nerve roots as needed and create partial anterior sacroiliac osteotomies. A rectus abdominis myocutaneous flap with vascular supply from the inferior epigastric vessels may also be harvested prior to abdominal closure and stowed within the abdominal cavity to be pulled-through for coverage and closure after the second, posterior stage of the surgery, during which the sacrum and involved tumor is removed. Some authors have demonstrated favorable outcomes with posterior-only approaches for en bloc sacrectomy , althougIn many cases, in order to achieve the desired margins, nerve roots will need to be sacrificed and the functional results of such a sacrifice will vary depending on the roots involved. Low sacral amputations often lead to sacrifice of the roots distal to S3, which tends to result in minimal deficit, with the exception of a variable reduction in perineal sensation and sexual function—though sphincter function is typically preserved . Mid-sacHigh sacral amputation often leads to impaired stability and advanced instrumentation techniques described in detail elsewhere may be required to reconstruct the pelvic ring and re-establish spinosacral and sacropelvic stability in order for patients to be safely mobilized ,80,81.Wound dehiscence, wound infections and CSF leak are the most commonly cited complication of sacrectomy for chordoma, with as many as 1 in 4 patients requiring further surgery as a result ,14,82. PAs with chordomas in the skull base and mobile spine, the adequacy of resection margins has the greatest influence on prognosis, overall survival and risk of local recurrence ,14,67,79By most accounts, conventional radiotherapy is ineffective as a stand-alone treatment or when coupled with intralesional resections for chordoma ,46. The One of the challenges with regard to radiotherapy for chordomas lies in the crucial—and relatively radiosensitive—neurovascular anatomy with which chordomas are often intimately involved. Image-guidance, stereotaxis, dosimetric planning and the use of hadrons  have improved upon our ability to deliver high doses of ionizing radiation to chordomas while sparing nearby anatomy. Hadron radiotherapy takes advantage of the Bragg peak effect, wherein the maximum dose of radiation delivered by heavy, charged particles like protons and carbon ions occurs at a precise depth , immediately before the particle comes to rest, with relatively small doses of radiation delivered throughout the rest of a particle’s course ,89. The Although proton beam therapy is the radiotherapy delivery method preferred by some quaternary centers for the treatment of chordoma, the stereotactic delivery of photon therapy has been shown to confer similar durability of local control with an acceptable toxicity profile  and no eBrachytherapy is another means by which radiation may be delivered to chordomas. Dural plaques  have been used by some authors as an adjuvant to external beam radiotherapy to improve dose delivery to dural margins with less risk of toxicity to the spinal cord in tumors with epidural extension ,93. ObjeFinally, carbon ion radiotherapy has been described in the recent literature as an effective treatment option for skull base chordoma with acceptable late toxicity and local control . Furtherwww.clinicaltrials.gov accessed on 31 January 2021).Chordoma is notoriously insensitive to traditional chemotherapeutic agents ,95 but oCDKN2A gene and its protein product, p16INK4a [www.clinicaltrials.gov accessed on 31 January 2021) (Cyclin D-dependent kinases (CDK4 and CDK6) are another target for chordoma molecular therapy. These oncoproteins regulate cell cycle progression through the G1 phase into the S phase and have been shown to be overexpressed in chordoma cell lines and tissue samples due to loss of the p16INK4a . Palbocip16INK4a ,100 effiry 2021) .www.clinicaltrials.gov accessed on 31 January 2021), as well as a phase I trial studying the safety and initial effectiveness of the antibody with or without stereotactic radiosurgery for chordoma and other advanced cancers .Immune checkpoint inhibitors are a potential line of therapy in chordomas and other cancers, as immune checkpoints have been demonstrated to impair the tumor-killing ability of T-lymphocytes. Anti-programmed cell death protein-1 (PD-1) and anti-programmed cell death ligand-1 (PD-L1) are immune checkpoints shown to be expressed in chordoma cell lines ,102. Avewww.clinicaltrials.gov accessed on 31 January 2021). A modified vaccinia Ankara (MVA) poxviral vaccine vector has also been developed that encodes human brachyury as well as a triad of costimulatory molecules  and is part of a Phase I clinical trial in patients with advanced cancers . Finally, an adenovirus serotype 5 (Ad5) vaccine encoding brachyury, carcinoembryonic antigen (CEA) and mucin-1, cell surface associated (MUC1) has been developed and is currently being tested in a Phase I clinical trial for patients with advanced cancers . Finally, several studies have discussed pharmacological inhibition of the chordoma oncogene, TBXT (transcription factor brachyury), via inhibition of H3K27-demethylases as a promising novel therapy altering gene networks necessary for tumor survival [Brachyury is another potential target for molecular therapy, as the protein is present semi-ubiquitously in chordomas and brachyury silencing has been shown to cause growth arrest and senescence in chordoma cell lines in vitro . Brachyusurvival ,109,110.Aggressive resection  is the most important prognostic indicator for patients with chordoma ,13,14,75Other reported prognostic factors for chordomas include pathology (with dedifferentiated chordomas having a poorer prognosis), history of prior resection (secondary patients referred due to recurrence after a previous resection have a poorer prognosis) and in sacral chordomas, the presence of muscle/sacroiliac joint involvement and higher location (above S3), all of which are reported to confer a poorer prognosis ,58,59,78Even among conventional chordomas, however, biologically distinct tumor types appear to be present, with some chordomas behaving more aggressively than others despite apparently conventional histology . AdditioChordoma continues to present a considerable treatment challenge due to a number of factors, including: (1) indolent growth rate and resultant tendency to present late in the disease course, (2) propensity for invading through tissue planes, encapsulating critical nearby anatomy and seeding after resection and (3) its relative insensitivity to conventional radiotherapy and chemotherapy.Extent of resection and surgical margins are currently the most important prognostic factors in chordoma and thus it is crucial that patients with chordoma are referred soon after presentation to an experienced quaternary care center, where every step of the diagnostic and therapeutic process can be controlled for the best patient outcomes. Biopsy and surgery should be carefully planned and executed by an experienced surgeon in association with a number of other surgical specialties so as to result in the widest margins and least likelihood of recurrence possible. Molecular sequencing, radiotherapy and clinical trials for promising molecular targeted agents should ideally all be available to patients and incorporated into the decision-making process.The role of radiotherapy in the treatment of chordoma is controversial. Evidence regarding the benefit of radiotherapy is contradictory at times and little consensus exists for its role in chordoma treatment. Part of this discrepancy in findings, however, may be related to bias and confounding, in part due to differences in execution and terminology between studies. Currently, some centers proceed with adjuvant radiotherapy only in cases of incomplete and intralesional resections, which may subject the outcomes to selection bias. Furthermore, the terminology utilized in many series regarding the degree of resection is imprecise and this further confuses the outcomes. In spinal and sacral chordomas, in particular, we recommend that the term “en bloc” be avoided in lieu of Enneking-appropriateness, as the latter provides more consistent information regarding tumor violation during resection and has been shown to correlate well with local recurrence and overall survival.Wide or marginal resections have the greatest impact on local recurrence and survival and should thus be pursued whenever possible but always with consideration of risk and in a manner that balances extent of resection with patient morbidity. Often our focus is primarily on overall survival and recurrence and yet, for many patients, quality of life is at least equal in value to quantity.Though the prognosis of patients with chordoma is still relatively poor, our understanding of the genetic and molecular basis for chordoma pathophysiology continues to blossom and this is likely to hold the greatest promise in terms of providing a true cure. Early clinical outcomes of agents targeting PDGFR, EGFR, c-Met and other elements of the chordoma molecular pathophysiological cascade have been modest but our understanding continues to evolve. At the present time, surgical resection is the preferred treatment for chordoma but in time, advances in targeted medical therapy and radiotherapy delivery may lead to these modalities playing a more prominent role in the treatment of chordoma, with aggressive surgery being reserved only for the most advanced cases.


In cancer care, tissue seeding after curative resections is a known potential complication, despite precautions taken during surgical treatment. We present an uncommon case of an abdominal wall metastasis along the tract of a surgical drain following gastrectomy for gastric adenocarcinoma. To our knowledge, this is the first case of such an occurrence in the setting of a negative staging peritoneal lavage. Aside from the rarity of such a recurrence, this instance highlights an opportunity to reevaluate best practices with regard to the extent of coverage of postoperative salvage radiotherapy. The oncologic patient provides many challenges and may require multiple catheters for drainage and at times infusion of nutrition or therapeutic agents. These foreign bodies should be scrutinized both clinically and radiographically, as they may create vulnerabilities in keeping malignant diseases contained and controlled. We provide a review of the literature with reasonable treatment options for the benefit of future patients. Cancer treatment is multidisciplinary and comprehensive with the ultimate goal of tumor eradication to maximize survival and minimize morbidity. Complications must be handled expediently to mitigate morbidity and improve prognosis. Metastatic seeding following resection is a rare but significant occurrence warranting further therapy. Spread patterns of gastric cancer rely primarily on histology, with generic adenocarcinoma mostly involving the liver (48%), followed by the peritoneum (32%) and lung 15%), and least often to the bone (12%) [5%, and lA 62 year-old female with no significant past medical history was found to have a normocytic anemia (Hgb 8.6) on routine bloodwork. The primary care provider's workup included upper endoscopy  which re18FDG) positron-emission tomography/computed tomography (PET/CT) showed concentrated uptake within only the gastric mass, with a standardized uptake value (SUV) of 10.4, but no evidence of distant metastasis. Initial carcinoembryonic antigen (CEA) level was 5.6 ng/mL .Staging endoscopic ultrasound  demonstrDiagnostic laparoscopy showed no peritoneal disease, and lavage washings were negative for malignant cells. A multidisciplinary approach concluded the best path towards lessening disease burden prior to a curative resection for a Siewert III gastroesophageal junction cancer . NeoadjuFollowing standard preoperative cardiac clearance, the patient underwent a robotic-assisted total gastrectomy and D2 lymphadenectomy. En bloc resection included 4 cm of the distal esophagus as well as total omentectomy. Just prior to this, a small lymph node in the splenic hilum was sent for frozen section, which was negative for malignancy. Before construction of the esophagojejunostomy, frozen sections of the resection margins returned, which were also clear of any tumor. A Jackson-Pratt (JP) drain was placed behind the anastomosis. Endoscopic testing was negative for any leaks. A feeding jejunostomy was also placed.Postoperative recovery was initially as expected, with routine fluoroscopic interrogation of the anastomosis showing no leak with either water-soluble or barium contrast and with normal esophageal motility. On day 4, an episode of fever, tachycardia, and hypertension occurred. Antipyretic and empiric antibiotics were started, and a CT was performed for further investigation . This reSurgical pathology showed both lymphovascular and perineural invasion, with tumor within 1 mm of the omental margin. Only 2 of 27 nodes were positive, without treatment effect of the primary tumor present, yielding a final stage of ypT4aN1. A regimen of salvage chemoradiation was initiated, which included capectabine, as well as 1.8 Gy fractions divided over 25 sessions, for a total dose of 45 Gy focused on the surgical bed .Two months later, a near-tripling in CEA (6.4 from 2.3) was concurrent with a new CT finding of a 1.1 cm hypervascular lesion embedded within the right abdominal wall subcutaneous fat  in closeα [Gastric cancer specifically is known to spread primarily via the peritoneum; the two primary paths being stomata-like orifices along the omentum and transvessel migration facilitated by hypoxic-induced factor-1α . Esophagα . These sα . Studiesα , 7.Peritoneal lavage has been established as an adjunct in staging, albeit of debated utility , 9. A paAs it has been established that tract seeding can arise with a variety of tissue types and through variable media, it is reasonable to undertake protective measures to prevent tract seeding. While surgical literature has described the importance of resecting open biopsy tracts, this appears to be specific to sarcomas and not necessarily applicable to needle biopsies . An arguThis report serves as both an example and warning that gastrointestinal cancers with anastomotic leaks can seed catheter tracts, even with a negative staging lavage of the peritoneum. Any catheter site must be scrutinized, as it will extend beyond the irradiated field. Tumor markers and imaging are helpful in diagnosing abdominal wall metastases.Informed consent was obtained and a copy is available upon request. The case was anonymized, with the exemption from IRB approval.


The authors report a case of mobile and painful nodules on the bilateral plantar surface of a female patient referred by the rheumatology service, where she was being followed-up for rheumatoid arthritis. A nodule excision was performed for differential diagnosis and symptom relief; the histopathological analysis was compatible with a rheumatoid nodule. Although rheumatoid nodules are a common manifestation of rheumatoid arthritis, exclusive plantar involvement is seldom described in the literature. A 60-year-old caucasian woman was referred to the dermatology outpatient clinic by the rheumatology service, where she was followed-up due to a 20-year history of rheumatoid arthritis. The patient reported a two-year history of painful nodules on the soles of her feet, with slow growth, which hindered walking . The ulta)Fibromab)Rheumatoid nodulec)Lipomad)CalcinosisRheumatoid arthritis is a systemic inflammatory disease, primarily articular, that affects about 0.8% of the world population.There are a variety of differential diagnoses, including subcutaneous granuloma annulare, calcinosis, lipoma, and gouty tophi; however, the history of rheumatoid arthritis, especially in a context of exacerbation of the articular condition, favors the clinical diagnosis of rheumatoid nodule. In doubtful cases, the definitive diagnosis is established by histopathological findings of a granulomatous process characterized by a focus of fibrinoid necrosis surrounded by a palisade of histiocytes.4After excision and confirmation of the diagnosis of rheumatoid nodule, the patient is currently being followed-up by the rheumatology team, with improvement of joint symptoms after the introduction of certolizumab, which inhibits tumor necrosis factor-α (TNF-α). As the joint symptoms were controlled, the patient presented an improvement in symptoms and a reduction in the dimensions of the plantar nodules.None declared.Diego Henrique Morais Silva: Conception and planning of the study; elaboration and writing of the manuscript.Isaura Azevedo Fasciani: Conception and planning of the study; critical review of the literature.Neusa Yuriko Sakai Valente: Conception and planning of the study; obtaining, analyzing, and interpreting the data; effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases.Bethânia Cabral Cavalli Swiczar: Effective participation in research orientation; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases.None declared.


Teratomas of the spinal cord are rare tumors, particularly in adults, but there is an increasing body of literature documenting both their diagnosis and successful treatment with surgical resection. However, to date, the literature has largely characterized spinal teratomas as single solitary lesions. Here, we report on an adult patient who presented with signs of progressive lower extremity weakness. Imaging demonstrated two simultaneous lesions of the spine, an extramedullary lesion in the upper thoracic region and an intramedullary lesion in the mid-thoracic region. Both lesions were resected and pathologically determined to be mature spinal teratomas. To our knowledge, this is only the second report of this tumor presenting simultaneously at more than one location in the spine and the first time it has presented both as an intra-axial and extra-axial lesion. Our results suggest that the presence of more than one simultaneous lesion does not necessarily increase the risk of a more aggressive immature pathology.  Teratomas of the spine are rare. Even in the pediatric population, where teratomas as a whole are reported to account for approximately 3% of all tumors, spinal teratomas are infrequently seen . In the A 73-year-old male with an extensive medical history including dementia, prior esophageal carcinoma, and diabetes with associated neuropathy presented with complaints of progressively worsening weakness of the legs over several months. The family also endorsed multiple episodes of bowel and bladder incontinence over the past three days. Physical examination revealed good strength in the upper extremities, with only trace movement in the right lower extremity and no movement in the left lower extremity. He had a T4 sensory level and poor rectal tone. An MRI of the entire spine was performed and showed a 0.8 × 0.8 × 1.5 cm non-enhancing cystic extramedullary lesion at T1 as well as a 0.8 × 1.0 × 1.4 cm heterogeneously enhancing cystic intramedullary lesion at T4-5  while being decompressed via nasogastric tube. Despite gradual improvements in his abdominal distention, the family requested removal of the TPN and palliative care measures only approximately two weeks after his surgery. He was subsequently discharged home with hospice care measures. PathologyMicroscopic sections of both lesions revealed fibrous and adipose tissue with cystic spaces. The rostral lesion was composed predominantly of squamous-lined cyst with mature glial tissue, focal cartilage, GI-type glandular elements, and ciliated cystic spaces. The larger, caudal intramedullary lesion demonstrated admixed nerve, disorganized smooth muscle, immature appearing cartilage, squamous-lined cysts, and glandular elements Figure . The diaSpinal teratomas are rare, with large spinal tumor case series citing a less than 0.5% incidence among all spinal tumors ; howeverThe presence of multiple lesions presenting in the same patient is exceedingly rare among the reported cases. Indeed, to our knowledge, this is only the second such report. Despite our initial suspicion that the presence of multiple simultaneous lesions would correlate with an immature phenotype, neither the previously reported multiple lesion patient  nor oursThe current consensus for the treatment of mature spinal teratomas is maximal safe resection; however, in the case of single solitary lesions, there is a treatment bias in that the diagnosis itself requires surgical intervention. It is not as clear that this is the optimal course of action in the setting of multiple lesions, particularly when not all lesions are symptomatic. With the presence of multiple lesions and an intraoperative frozen section diagnosis of teratoma, the concern is that the presence of multiple lesions suggests an immature pathology, which necessitates a more aggressive approach. However, as seen in our patient, this is not necessarily the case. Indeed, even with multiple spinal teratomas, it is still likely that all of them are mature. Thus, the risk of developing a new neurologic deficit from a complication after operating on an asymptomatic level, as well as the low likelihood of progression if an asymptomatic level is left untouched, could favor a more conservative approach. However, given the still relatively small numbers of both reported immature spinal teratomas and spinal teratomas presenting with multiple lesions, we would argue that a suspicion for an immature pathology should still be kept for patients with multiple lesions. In our patient, we felt we could safely resect the rostral asymptomatic lesion and thus give him the highest chance for a cure regardless of whether the final pathology was suggestive of either a mature or immature teratoma. However, given his postoperative complication, it is debatable whether a better approach would have been to minimize the operative time by avoiding this level unless pathology from the caudal symptomatic lesion was suggestive of an immature teratoma. In the very least, we feel that this would have been a reasonable approach. Our case suggests that while rare, multiple mature teratomas of the spine can occur and, indeed, can occur in the adult. As such, it should be included in the differential of multiple spinal lesions and planned for accordingly, with particular attention being paid to the fact that multiple lesions have not thus far demonstrated a higher risk of an immature pathology. While maximal safe resection remains the goal, in cases of multiple mature spinal lesions, particularly when not all are symptomatic, this must be weighed against the risk of worsened neurologic deficit and the slow rate of growth.


Lipomas are rare primary heart tumors and may involve the endocardium, myocardium, or pericardium. Signs and symptoms depend on the tumor location and size. The intrapericardial lipoma we report has massive dimensions and mimics a pericardial effusion. A 38-year-old male complained of dyspnea and precordial pain. On physical examination, heart sounds were diminished. The patient had received extensive medication for a clinically suspected pericardial effusion due to heart failure. A voluminous mass resembling fat within the pericardial sac was revealed by transesophageal echocardiography and a computed tomography scan. The tumor was removed successfully by a subxiphoid surgical approach. The diagnosis of a 635 gram intrapericardial lipoma was confirmed by pathological examination. After surgery, the patient recovered well and was completely asymptomatic at a follow-up at 90 days. No medications were being taken since. The diagnosis of a pericardial effusion should be secured by imaging exams to avoid unnecessary medications. Cardiac lipomas can be readily recognized by their typical features on radiologic imaging. The surgical pathology examination confirms the diagnosis and rules out malignancy criteria. Primary heart neoplasms are rare diseases, and their incidence rate is up to 0.4% in necropsy series , 2. ApprSigns and symptoms vary according to the size and localization of the tumor. Cardiac lipomas can cause thoracic pain, fatigue, dyspnea, syncope, arrhythmias, or even sudden death. Nevertheless, most cases are asymptomatic , 16, 17.A 38-year-old male has complained of progressive dyspnea and sporadic precordial pain related to moderate effort for 5 months. Physical examination showed normal peripheral perfusion, and no edema was detected. On auscultation, no murmurs were heard; however, heart sounds were diminished. Clinically, a pericardial effusion due to heart failure was suspected and treated with pharmacologic drugs. The patient received digoxin, aspirin, furosemide, spironolactone, and carvedilol for 8 weeks until imaging exams were performed.Transesophageal echocardiography revealed a 79% ventricular ejection fraction and a hypoechoic mass within the pericardium attached to the free wall of the right ventricle and atria. A computed tomography scan showed an expansive mass in the anterior region of the pericardium with a homogeneously low density similar to fat. The tumor had a regular shape and measured 14.0 × 10.0 × 16.0 cm. The cardiac lesion extended from the upper to the lower thoracic region, displaced the heart backward, and reduced the right cardiac chambers .The patient was submitted to a pericardial window via a subxiphoid surgical approach, in which a pedunculated epicardial tumor inserted in the right ventricle was resected. No cardiopulmonary bypass was needed. At gross examination, the tumor was split into two irregular fatty fragments measuring 22.5 × 9.0 × 4.0 cm and 21.0 × 15.0 × 2.0 cm and weighing 635 grams . The hisTen days after surgery, the patient was discharged from the hospital without any medications. At a follow-up at 90 days, the patient was completely asymptomatic.Most cardiac lipomas are asymptomatic. Therefore, the diagnosis of these benign neoplasms is frequently done on autopsies. Medical literature may underestimate their real prevalence . GeneralThe majority of cardiac lipomas is sessile or polypoid masses implanted in the subendocardium or epicardium. Only 25% of cardiac lipomas arise within the myocardium , 20. OftSymptoms occur when the mass compresses the cardiac chambers and causes hemodynamic changes. Sometimes, it may cause pericardial effusions . IntrapeChest radiography, transthoracic and transesophageal echocardiogram, tomography scan, and magnetic resonance imaging play an important role in identifying the lipoma location and relationship to the heart , 36–38. Imaging exams are important to evaluate the biological behavior of heart tumors. However, malignancy and the definitive diagnosis can be made by histopathological examination only . On grosAlthough lipomas are benign and slow-growing tumors, the surgical approach is to prevent heart compression and hemodynamic changes , 11. SurPericardial lipomas' signs and symptoms are variable. Indeed, most of the patients are asymptomatic. Conversely, surgical removal is necessary to prevent cardiac compression and to exclude malignancy through the histopathological examination.


Intraneural ganglion cysts are benign soft-tissue masses located in the epineurium of peripheral nerves. They originate from nearby joint connections via articular branches. Traumatic events seem to play a role in their pathogenesis as well. Clinical manifestations include pain over the area of the cyst, palpable tender mass, hypoesthesia, and muscle weakness depending on the affected nerve. Our case highlights an uncommon clinical manifestation of this entity with acute foot drop, as the primary symptom, without any previous traumatic event, enriching by this way the current diagnostic thinking process of clinical physicians. We report a case of a 42-year-old military officer who presented to our emergency department with acute foot drop that appeared during a march. Initially, the common peroneal palsy was misdiagnosed as L5-S1 disc herniation, but investigation with lumbar MRI scan led to rejection of our primary diagnosis. After performing EMG of the lower extremity and knee MRI, an intraneural ganglion cyst of the common peroneal nerve was diagnosed. Patient was treated with surgical decompression of the cyst, followed by ligation and complete resection of the articular branch, as well as disarticulation of the superior tibiofibular joint. At a twelve-month follow-up, the patient showed significant functional recovery. This is, to the best of our knowledge, the first case of intraneural ganglion cyst manifested with an acute complete foot drop without a clear prior traumatic event. We underline the need for a high index of suspicion when dealing with cases of acute peroneal palsy without any accompanying symptoms. Intraneural ganglion cysts represent rare benign cystic lesions formed within the epineurium of peripheral nerves, near joints. The most common site affected is the common peroneal nerve and its branches, while similar cysts of the ulnar, sciatic, and tibial nerves have also been reported , 2.Patients usually experience pain, numbness, hypoesthesia, and muscle weakness along the distribution of the affected nerve. Positive Tinel's sign and a palpable tender mass around the area of the ganglion cyst are also common. The onset of symptoms can be gradual or, less commonly, acute, usually exacerbated by a previous traumatic event .The exact etiology of this condition remains unknown. Spinner et al. proposed the unifying articular theory, which highlights the key role of the articular branch in the pathogenesis of an endoneural ganglion cyst . AccordiThis study was conducted in accordance with the ethical standards of the institutional review board of our hospital and with the 1964 Helsinki Declaration and its later amendments or comparable ethical standards .A 42-year-old male presented to the emergency department of our military hospital with reported mild low back pain, associated with numbness in his right leg below the knee, and ipsilateral complete foot drop that occurred during a military march. The onset of neurological symptoms was acute, without any previous symptoms, reaching immediately its full intensity and was perceived by the patient as dragging of the foot. The patient had no history of lumbar spine disease. No traumatic event was reported during the march.Physical examination of the right lower extremity showed negative Lasegue's sign and straight leg raise test and hypoesthesia in the lateral aspect of the leg and dorsal aspect of the foot. Foot dorsiflexion and eversion and large toe dorsiflexion were severely impaired . There were no clinical signs of muscle atrophy on the anterior and lateral compartments of the leg. Physical examination of the contralateral lower extremity was normal. Knee and lumbar X-rays were normal.Our initial therapeutic approach included rest, NSAIDs, painkillers, and corticosteroid intramuscular injections. The patient was admitted to our clinic under observation status, and lumbar spine MRI scan was ordered; however, results did not correspond to the patient's symptomatology . MoreoveFollowing negative lumbar spine MRI results, a second more thorough physical examination was conducted, which revealed positive Tinel's sign in the area of the fibular neck, shifting diagnostic thinking process towards peripheral neuropathy. Subsequent EMG and NCS showed decreased conduction velocity and amplitude in the common peroneal nerve around the area of the popliteal fossa. Furthermore, knee MRI showed a multilobulated lesion in the area around the fibular head, in close proximity to the common peroneal nerve .Surgical exploration under loop magnification was decided. The common peroneal nerve was identified and followed to its bifurcation. Common and deep peroneal nerves appeared oedematous . The artPostoperatively, the patient was treated with a foot drop polyethylene splint and physiotherapy. Histological examination confirmed our diagnosis of an endoneural ganglion cyst . At a 3-Intraneural ganglion cyst cases reported in the literature are rare. The most frequent site of occurrence is reported to be the common peroneal nerve, followed by the ulnar and tibial nerves . AlthougCommon symptomatology of an endoneural ganglion cyst of the common peroneal nerve includes pain over the fibular head, with or without swelling, positive Tinel's sign, and paresthesia over the lateral surface of the tibia and dorsum of the foot. Some patients may present with gradual or acute weakness of the muscles located in the anterior and lateral compartments of the leg. Muscle denervation and atrophy have also been described , 13–16. MRI and/or ultrasound serve as useful tools of the diagnostic process. On ultrasonography, an endoneural ganglion cyst appears as a large well-circumscribed hypoechogenic lesion , while oUltrasonography is less time consuming than MRI and may be of value when guided percutaneous aspiration is decided, but it fails to illustrate the relation of the cyst with the neighboring anatomical structures as well as the articular connection and, unless a specialized radiologist in musculoskeletal ultrasonography is available or MRI is contraindicated, intraneural ganglion cysts are best imaged with magnetic resonance .Standard treatment of intraneural ganglion cysts is surgical excision of the ganglion and nerve decompression. An alternative minimally invasive treatment is decompression by percutaneous aspiration of the ganglion under ultrasound guidance, with or without corticosteroid injection . Both thThis article demonstrates a rare case of an acute foot drop caused by an intraneural ganglion cyst. To the best of our knowledge, this is the first case of an intraneural ganglion cyst resulting in an acute foot drop without a prior traumatic event. Our hypothesis is that of an existing asymptomatic intraneural ganglion cyst that deteriorated past the asymptomatic threshold point during the walk, possibly via an indirect mechanism, as proposed by Spinner et al. . AlthougIntraneural ganglion cysts, although infrequent, are well established in the literature and should be considered in the differential diagnosis of peripheral mononeuropathy, in order to avoid diagnostic pitfalls. Early diagnosis and surgical treatment with open decompression and concurrent address of the articular branch are of paramount importance to obtain positive outcome and minimize recurrence risk.


Abdominal wall biloma is an uncommon entity. We herein report an extremely rare case of cholecystitis with abdominal wall biloma after percutaneous transhepatic gallbladder aspiration (PTGBA).A 69-year-old woman was diagnosed with acute cholecystitis, and PTGBA was performed on Day 1. PTGBA was performed a second time because of re-expansion of the gallbladder and an increased CRP level on Day 3. Computed tomography was performed on Day 9 because we suspected recurrence of cholecystitis. It revealed a well-circumscribed fluid collection between the abdominal wall or the diaphragm and the liver. Based on these intraoperative findings, we diagnosed her with cholecystitis with abdominal wall biloma. Cholecystectomy and drainage of the abdominal wall biloma were performed on Day 10. The postoperative course was uneventful, and she was discharged on Day 18.Early cholecystectomy is the gold-standard treatment for acute cholecystitis, but cholecystectomy is not performed in some cases. PTGBA is much more convenient, quicker, and less costly, but inappropriate aspiration during the second PTGBA session might have spread the infected bile to the abdominal wall through the PTGBA route.This case represents the first reported case of a biloma within the abdominal wall after PTGBA. To prevent this complication, we should aspirate gallbladder bile sufficiently during PTGBA. In addition, we should consider performing alternative therapy, such as percutaneous transhepatic gallbladder drainage or an operation, when we fail to appropriately aspirate. This case report has been reported in line with the SCARE checklist .2A 69-year-old woman was admitted to our hospital at night due to acute upper abdominal pain. She was known to suffer from hypertension. An abdominal examination showed tenderness in the right upper quadrant. Murphy’s sign was positive. Initial investigations showed an elevated white blood cell number 18400/μl) and C-reactive protein level . Renal and liver function tests and amylase and coagulation screens were normal, as were her chest and abdominal plain findings. Computed tomography (CT) revealed enlargement of the gallbladder with a thickened and enhanced wall. There was a gallstone in the gallbladder. There was no fluid collection around the gallbladder . We diag00/μl andPTGBA was carried out with a 21-gauge needle under sonographic guidance. Infected bile (80 mL) was aspirated, and the needle was removed immediately. Because of re-expansion of the gallbladder and a further increased CRP level (35 mg/dl), PTGBA was performed a second time on Day 3, and 45 mL of infected bile was aspirated. She began to recover temporarily, but her abdominal pain and high fever recurred on Day 9. CT revealed a large, well-circumscribed fluid collection between the right diaphragm and the liver .Fig. 2ImWe planned open cholecystectomy for cholecystitis, and the drainage of subcapsular biloma of the liver on Day 10. Regarding the intraoperative findings, there is no bile and infectious in peritoneal cavity. The gallbladder was tense and reddened, but no perforation or fistula with the abdominal wall was seen. The liver was normal, and no fistula was noted between the liver and the abdominal wall, nor was a subcapsular biloma of the liver detected. There was an abdominal wall biloma located between the parietal peritoneum and abdominal lateral muscle or diaphragm A-B. We p3Early cholecystectomy is the gold-standard treatment for acute cholecystitis ,4. In prA previous study also showed that PTGBA was safer than PTGBD . AlthougBilomas, which are encapsulated, extrahepatic collections of bile, are often located in the subcapsular liver and intra-abdominal space. Abdominal wall biloma is extremely rare. There have only been two case reports of abdominal wall biloma, but both were complications of cholecystectomy ,10. The In the present case, inadequate aspiration during the second PTGBA might have caused the spreading of infected bile to the abdominal wall through the route of PTGBA, as the amount of bile aspirated during the second PTGBA was less than that of the first, and the gallbladder was extremely tense on intraoperative findings. Intrahepatic biliary tract injury by PTGBA was denied by intraoperative cholangiography, and there was no fistula between the gallbladder, liver and abdominal wall.To prevent a biloma from developing within the abdominal wall after PTGBA, we should aspirate the gallbladder bile sufficiently during PTGBA. When we are unable to aspirate sufficiently during PTGBA, we should consider alternative therapy, such as PTGBD or emergency cholecystectomy.The pre-operative diagnosis of abdominal wall biloma is difficult. In the present case, CT revealed a well-circumscribed right subdiaphragmatic collection. We diagnosed her with subscapular biloma of the liver preoperatively because it sometimes occurs with acute cholecystitis  and ther4We presented our experience with an extremely rare case of cholecystitis with abdominal wall biloma after PTGBA. To prevent this complication, we should aspirate gallbladder bile sufficiently during PTGBA. In addition, we should consider alternative therapy, such as PTGBD or emergency cholecystectomy, when we fail to aspirate sufficiently during PTGBA.The authors report no declarations of interest.All authors declare no source of funding.The clinical case is exempt from ethical approval.Informed consent signed by a patient to publish the article.Kenichiro Toritani: wrote the paper, study concept or design, data collection, data analysis and interpretation, performed the surgery.Mitsutaka Sugita: contribute to interpretation, revision, data collection and data analysis.Akiko Shimizu: performed the surgery.Akira Watanabe: approved the paper.Hidetaka Andrew Ono: approved the paper.Hiroyuki Baba: performed the surgery.All authors have approved the final article for submission.Kenichiro Toritani.Not commissioned, externally peer-reviewed.


Patients with Meigs' syndrome and elevated serum CA-125 are not frequently reported. A 59-year-old woman and a 48-year-old woman sought help because of progressive shortness of breath caused by pleural effusion. The presence of a pelvic mass was noted in both the patients and was thought to be the cause of the effusion. Both patients had elevated serum CA-125, which raised the possibility of malignancy. After complete resection of the tumors, the pathologic reports confirmed a benign and a low-grade malignant ovarian neoplasia, respectively. We comment on the outcome and follow-up of these two cases and briefly review Meigs' syndrome. The clinical presentation of a pelvic mass, ascites and pleural effusion is often associated with ovarian carcinoma. Furthermore, if blood and/or pleural fluid CA-125 are elevated, it raises suspicion for malignancy [Case 12 of 90%. Otherwise, the vital signs were normal. On auscultation, there were absent breath sounds and dullness to percussion over the lower half of the right hemithorax. The abdomen was globular and a firm, smooth, and slightly tender mass was palpated from the right hypochondrium to the pelvic area. No signs of ascites were evident. The chest X-ray and a representative CT image are presented  with a known history of presumed uterine myoma three years before presentation but did not follow up with her physician. On this occasion, she presented to the emergency room (ER) complaining of progressively worsening dyspnea on exertion, dry cough, and discomfort over the right hemithorax elicited by deep inspiration. She denied weight loss or fever, but admitted to increased abdominal girth in the previous two years, neglecting to consult about it. On physical examination, she was slightly tachypneic with no accessory muscle use, room air SpO Figures , 2. SeruDuring the hospital admission, she reported increasing dyspnea due to accumulation of the pleural effusion that relieved after a therapeutic thoracentesis. A median laparotomy was performed. A 21-cm right adnexal solid mass with smooth and pearly appearance Figure  and anot2 was 98% with normal vital signs. She had stopped smoking. The patient refused to undergo a repeat chest X-ray.She was discharged two days after the surgery. On a follow-up visit three months later she reported no complaints, her lungs were clear on auscultation and the room air SpOCase 22 was 95%. Other vital signs were normal. Breath sounds were absent on the right hemithorax and an abdominal mass with clear borders was palpated without clear signs of ascites. The chest X-ray and a representative CT image are shown  benign fibroma (they account for up to 2% of resected ovarian neoplasms . Imaging3) and is occasionally bloody [Meigs' syndrome presents more often in the fifth decade of life, and the cause of ascitic fluid appears to be a generalized secretion by the tumor itself. Moreover, the stimulation of the peritoneum's mesothelial cells by the release of vascular endothelial growth factor (VEGF), IL-1, IL-6, IL-8, and TNF-α from the tumor is reported as another cause of exudation. Only large tumors are frequently associated with ascites, which may not be detectable on physical examination, and the amount of ascites does not influence the amount of pleural effusion that develops. The genesis of the pleural effusion is similar to that of patients with hepatic hydrothorax, through pores in the diaphragm, and the similar characteristics between peritoneal and pleural fluid and the rapid reaccumulation of pleural fluid after evacuation supports this hypothesis. However, some authors believe that the pleural fluid arises from the transdiaphragmatic transfer of ascitic fluid by the lymphatic vessels, which seem to be larger on the right side, potentially explaining the finding of a right pleural effusion seen in approximately 70% of patients, being left-sided in 10%, and bilateral in 20%. Based on the protein levels, the effusion is usually an exudate with a low white blood cell count (fewer than 1000/mmA CA-125 test is not accurate enough to use for ovarian cancer screening in general because many noncancerous conditions can increase the serum level, including normal conditions such as menstruation and pregnancy, and noncancerous conditions such as cirrhosis of the liver, uterine fibroids or endometriosis. Besides ovarian cancer, certain cancers may also cause an increased level of CA-125, including endometrial, peritoneal and fallopian tube cancers , while aThe diagnosis of Meigs' syndrome should be entertained in all women with a pelvic mass, ascites and pleural effusion that present with a negative fluid cytology, and it is confirmed by the finding of a benign ovarian tumor and the nonrecurrence of peritoneal and pleural effusion once the mass is removed. It is important to be aware that, while awaiting for the pathology report, a high serum and/or fluid CA-125 does not compulsorily indicate malignancy.


Among the various forms of colorectal carcinomas, primary signet ring cell carcinoma (SRCC) of rectum is infrequent. Primary SRCC with adenoma is even rarer. Due to its low morbidity and lack of obvious manifestations at early stages, it is difficult to make an early diagnosis and perform surgical intervention in time. Herein, we reported a case of primary SRCC with tubular adenoma of rectum and also performed a review of the literature of such cases, in hopes of expanding the general understanding regarding such cases.A 61-year-old male patient presented with rectal bleeding for 1 week.A neoplasm could be palpated through a rectal examination, with a size of 4.0 cm by 3.0 cm, at a distance of 5 cm from the anal edge. Magnetic resonance imaging examination and colonoscopies were performed to confirm the finding, and 4 tissue specimens were obtained for histopathologic biopsy. The result of biopsy was high-grade intraepithelial neoplasia with an adenoma component.Surgical resection was performed, and histopathologic and immunohistochemical staining examination of the resection confirmed the diagnosis of SRCC with tubular adenoma.The patient was discharged from hospital 12 days postsurgery, without any complications. Further chemotherapy and supportive treatments were suggested to him and will be followed at a local hospital.Primary rectal SRCC has a rather low morbidity. Furthermore, a rectal SRCC with adenoma which was presenting in this case is even more rare. Besides lack of clinical characters, delay of diagnosis and treatment frequently occur. So far, a surgical procedure has still been one of the most effective treatments. Considering of metastasis and the poor prognosis, early diagnosis, in-time radical resection, and a comprehensive followed treatment are recommended for a higher 5-year overall survival. The term of SRCC is a descriptive term, denoted by the histologic observation of cell nuclei being pushed to the periphery due to excess intracytoplasmic mucin.–5 SRCC was firstly reported in 1951 by Laufman and Saphir. The majority of SRCC cases are found in stomach, although some cases have also been reported in breast, lung, bladder, pancreas, and colon-rectum.Colorectal carcinoma (CRC) is one of the major causes of cancer mortality worldwide. As such, improvements that allow for more timely diagnosis and earlier surgical intervention may significantly improve the 5-year survival rates and overall outcomes.Since the clinical manifestations typically appear late, colorectal SRCC is frequently detected at an advanced stage. Furthermore, SRCC tends to be more aggressive than carcinomas of other histologic types, leading to poor prognosis for colorectal SRCC cases.However, because of lack of obvious clinical characteristics of rectal SRCC, and poor awareness by the doctors, misdiagnosis of such tumors may occur, and some patients may consequently miss the optimum opportunities for intervention. Here, we reported a case of rectal SRCC with rectal adenoma components and its clinical characteristics, and hope to raise appreciation for the possibility of such rarer cases during clinical diagnosis Fig. .2A written informed consent statement was obtained from the patient following approval from the Institutional Ethics Committee of the Second Hospital of Jilin University.A 61-year-old male patient presented with a complaint of rectal bleeding for more than 1 week, with no abdominal pain or any changes of bowel movement. He denied having any history of recent fever, chills, nausea, vomiting, or weight loss. The patient had a history of smoking for around 25 years, of 20 cigarettes per day. He denied any history of drinking, or other concomitant diseases. His father died from heart disease, and his mother died from stomach carcinoma.A physical examination showed us that his overall condition was relatively normal: blood pressure, 124/84 mm Hg; heart rate, 74 beats per minute; respiratory rate, 17 breaths per minute; oxygen saturation, 100% on room air; and body temperature, 36.6°C. Abdominal examination also had no significant abnormal findings. However, rectal examination (knee-chest position) found that a neoplasm with a smooth surface was located at 5 cm from anal edge. Its size was around 3.0 cm × 3.0 cm. The root of this neoplasm could be moved slightly by palpation. No dark red blood or pus remains were found on the glove when the rectal examination was finished.9 cells/L ; red blood cell count, 4.53 × 1012 cells/L ; platelet count, 208 × 109 cells/L . However, the patient did show slightly increased carcinoembryonic antigen (CEA) levels. A colonoscopy confirmed the presence of a tumor at about 5 cm from the anal edge, with a size of 4.0 cm × 3.0 cm, smooth surface, wide basement, and surrounded by congestive mucosa. Four tissue specimens were obtained from this tumor for pathologic biopsy. The result of this biopsy revealed that tubular adenoma cells were found, together with cells of high-grade intraepithelial neoplasia. A consequent nuclear magnetic resonance imaging (MRI) examination confirmed that tumor was in the rectal cavity, with the location as same as that described during colonoscopy, with no swollen lymph nodes being found around the rectum  for the rectal carcinoma. During the surgery, we found that tumor in the rectum at a distance of about 6 cm distance from anal edge, with no obvious evidence of invasion of the tumor toward the surrounding tissue. Laparoscopic total mesenteric excision was performed, and several enlarged suspected lymph nodes were found during surgery. No complications occurred during or postsurgery. That patient was discharged from hospital 12 days postsurgery.Subsequent pathologic examination of the resected surgical specimen supported the diagnosis of rectal SRCC with tubular adenoma. From the serial sectioning of the surgical specimen, the histology assessment showed scattered foci of signet ring carcinoma cells invading the lamina propria nearby the adenomatous lesion  Fig. . MeanwhiAfter his discharged from hospital, a 6-month follow-up was performed by telephone. He was then asked to perform a blood examination to monitor levels of CEA and CA-199. Both CEA and CA-199 fell in the normal range postsurgery. Neither a computed tomography scan performed 3 months postsurgery or an MRI performed 6 months postsurgery found evidence of potential recurrence. At the same time, the patient also underwent 6 cycles of XELOX chemotherapy. The patient did not report experiencing any serious adverse effects, such as neutropenia, neurotoxicity, or bone marrow suppression.3 Due to the low incidence, SRCC has only been evaluated sporadically in a limited numbers of cases. However, SRCC is almost always reported to be found at an advanced stage and with a poor prognosis, as compared with the more common forms of common adenocarcinoma (CAC) and mucinous adenocarcinoma (MAC).,5,9,10The SRCC is a rather rare subtype of CRC. According to previously published literature, tits incidence ranges from 0.1% to 0.9% among all forms of CRC.3.1; adenoma, typically reported as a combination of adenoma cells and signet ring cells, just as this case–15; atypical epithelium, which is associated with p53-positive intraepithelial signet ring cells; and a combination of these lesions.There has been considerable debate about the histologic origin of SRCC for decades. Four main theories have been proposed: CAC, due to the finding of transition areas between typical CAC and SRCC in many cases Meanwhile, an A:T transversion at the 3rd base position of K-ras codon 61 has also been reported. Additional biomarkers, such as Reg IV and claudin-18, have been reported to be more highly expressed in SRCC as compared to CAC, and both markers have been previously implicated to be involved in gastric cancer. HATH1, MUC2, and SOX2, identified as the key genes involved in controlling mucin secretion in the gastrointestinal tract, have also been reported to be more highly expressed in SRCC, consistent with histologic findings of excess mucin buildup.–20A distinct pattern of Kras mutations and lower Kras mutation frequency has been reported for colorectal SRCC cases as compared with that of CAC.3.2,10,21–24 To confirm this significance, we further analyzing the TNM staging data for cases of SRCC, CAC, and MAC from our hospital over the past 2 years , and larger studies may be necessary to clarify the true utility of surgical resection for such patients.Normally, as with the other subtypes of CRC, a timely surgical resection of colorectal SRCC has been considered to be the most effective treatment with the highest promise of overall survival.4Due to the low morbidity and lack of defining clinical characteristics, rectal SRCC is usually diagnosed at an advanced stage. However, SRCC may be considered as a possibility in cases of hematochezia of unknown cause. Since SRCC is typically more aggressive and has higher risks of metastasis, timely detection and surgical intervention, and comprehensive follow-up treatments are recommended to ensure better patient outcomes.The authors appreciate the contributions of all the surgeons, coworkers, and friends involved in this study and thank the editors and reviewers for their help with this manuscript.Yong-Ping Yang and Tong-Jun Liu conceived of this study, designed it. Yong-Ping Yang and Ling-Yun Yu drafted the manuscript. Yong-Ping Yang, Ling-Yun Yu, Jian Shi, and Jian-Nan Li participated in design of this study, acquired the data and analysis of data. Xin-Yu Wang acquired the data. Yong-Ping Yang and Tong-Jun Liu participated in manuscript preparation and critical revision. All the authors read and approved the manuscript.


To report a case of localized suprachoroidal hemorrhage presenting as a choroidal mass.A 66-year-old woman presented with sudden onset pain in the right eye, one week following uneventful cataract surgery. The best corrected visual acuity (BCVA) was 20/160 and fundus examination showed a brown elevated choroidal mass temporal to the fovea in the right eye with normal retina and retinal vessels over it. The differential diagnoses considered were choroidal granuloma, melanoma, choroidal hemangioma, posterior scleritis, and localized suprachoroidal hemorrhage (SCH). Fluorescein angiography (FA) and indocyanine green (ICG) angiography were unremarkable except for mild disc leakage; B-scan showed a choroidal mass with high surface reflectivity and low internal reflectivity, and optical coherence tomography (OCT)showed an elevation of retino-choroidal complex with hyporeflective mass in the outer choroid with choroidal folds suggestive of SCH. Her systemic evaluation showed raised erythrocyte sedimentation rate (ESR) and consolidation in the upper lobe of the right lung. The patient did not take any additional treatment for her eye and the lesion regressed and visual acuity improved to 20/30 in one month.Delayed spontaneous suprachoroidal hemorrhage can present as a choroidal mass. Multimodal imaging helps to differentiate it from other sight-threatening and life-threatening ocular diseases. A localized spontaneous SCH can cause diagnostic dilemma by presenting as a choroidal mass. Multimodal imaging including fluorescein angiography (FA), indocyanine green angiography (ICG), ultrasound B scan, and optical coherence tomography (OCT) help to differentiate various choroidal mass lesions like melanoma, choroidal granuloma, choroidal hemangioma, posterior scleritis, and suprachoroidal hemorrhage (SCH). The present case highlights the role of multimodal imaging in differentiating SCH from other choroidal masses.Spontaneous suprachoroidal hemorrhage (SCH) is a rare occurrence associated with intraocular surgeries. A few cases of SCH following Valsalva maneuver and delayed postoperative hemorrhage have been reported.A 66-year-old woman presented with a history of sudden onset of excruciating pain in her right eye. She had undergone phacoemulsification and posterior chamber intraocular lens (PCIOL) implantation in the right eye a week before. Her systemic history was otherwise normal except for osteoarthritis for which she occasionally used analgesics. On examination, her visual acuity was 20/160 and 20/60 in right and left eyes, respectively. Slit lamp examination showed few Descemet's folds and PCIOL in the right eye and cataract in the left eye. Intraocular pressure (IOP) was 14 and 16 mm Hg by applanation tonometry. Fundus showed a localized dome-shaped elevated choroidal mass temporal to the fovea with no orange pigment changes or subretinal fluid, with a diameter of about 5–6 disc diameters (DD), with overlying choroidal folds, and the presence of a few retinal hemorrhages in the temporal periphery in the right eye [Figure 1 (A)]. The retina of the left eye was normal. The patient was treated with topical steroids (prednisolone acetate eye drops 1% six times a day) and oral paracetamol 650 mg thrice a day. Keeping a differential diagnosis of melanoma, choroidal granuloma, choroidal hemangioma and posterior scleritis in mind, the patient was referred to the oncology clinic. The patient underwent multimodal imaging and systemic workup. FA showed normal fluorescence pattern, except for minimal disc leakage and overlying choroidal folds in the right eye [Figures 2 (A–C)]. ICG showed normal fluorescence pattern [Figures 2 (D–E)]. The fundus autofluorescence was normal in both eyes. Ultrasound B scan showed a well-defined mass with high surface reflectivity and low to medium internal reflectivity without sub-Tenon's fluid [Figure 1 (B)]. OCT showed an elevation of retinochoroidal complex over a hyporeflective space in the outer choroid with a smooth anterior surface, pushing the inner choroid, along with minimal subretinal fluid [Figures 1 ]. The absence of intrinsic circulation and a normal FA and ICG ruled out choroidal melanoma and hemangioma. Systemic workup showed a positive Mantoux test (24 mm), and the chest X-ray showed lung consolidation in the upper lobe. The differential diagnoses considered were of either SCH or choroidal granuloma. She was referred to an internist to start antitubercular therapy (ATT). The patient came for a review at one month and had not taken any treatment for her eye other than topical steroids and paracetamol tablet. The lesion had spontaneously regressed and visual acuity had improved to 20/30 with a normal OCT [Figure 2]. (early hypo and late hyper-fluorescence in FA and persistent hypo-fluorescence in ICG) were not seen in this patient, but the disc showed minimal leakage in the late phase.In retrospect, a choroidal mass with no changes on FA or ICG, which was dome shaped, optically lucent in OCT and B scan, showing high surface reflectivity with low internal reflectivity and a spontaneous resolution were suggestive of a localized SCH. The typical FA and ICG features of choroidal granuloma The important risk factors are old age, hypertension, Valsalva maneuver and use of anti-coagulants. The dome-shaped elevation in the fundus due to SCH is usually limited to the area of fibrous adhesions concurrent with vortex veins; however, massive SCH can extend posteriorly. As it closely mimics other life-threatening conditions like choroidal melanoma, metastasis, and granuloma, patients are usually investigated.,3Spontaneous suprachoroidal hemorrhage is a relatively rare differential diagnosis of a choroidal mass lesion, which accounts for 2% of pseudo melanomas. The patient not only presented with ocular pain, which is usually seen associated with posterior scleritis or granuloma, but also with features suggestive of SCH, which caused a diagnostic dilemma. The fundus lesion of a brown elevated mass mimicked melanoma, hemangioma, and granuloma, but the features of melanoma-like orange pigments were absent.Though cataract surgery was uneventful in our patient, it could have been the precipitating factor. [Table 1].Multimodal imaging helps to differentiate various choroidal mass lesions,6 The presence of choroidal folds over the lesion with background normal fluorescence in FA and choroidal folds over an optically empty elevated lesion in OCT is a feature of SCH.,7 A choroidal hemangioma would show high surface and internal reflectivity in B scan and bright hyperfluorescence on FFA. The absence of typical features of granuloma in FA and OCT ruled out granuloma, but the disc leakage in the late phase of FA along with a history of pain created a diagnostic dilemma with granuloma and posterior scleritis. The disc leakage in this case, could be explained by postoperative inflammation.The B scan showed acoustic hollowing, but no choroidal excavation. This feature of SCH has already been described.A cautious interpretation of lab tests like Mantoux and meticulous analysis of multimodal imaging in correlation with clinical features is required to make the correct diagnosis of cases such as this one. Patients may be closely observed for one or two months for spontaneous resolution in case of strong clinical suspicion of localized SCH, and invasive investigations and extensive systemic workup may be considered in cases of non-resolution.Even though localized SCH is a rare entity and regresses spontaneously, it is rarely thought of by ophthalmologists in the differential diagnosis of a choroidal mass.Nil.There are no conflicts of interest.