Solitary extraskeletal osteochondromas are rare benign lesions usually located close to a joint and are characterized by the absence of continuity with the adjacent bone. They are usually found in the hand and feet and are extremely rarely reported in the growing skeleton.In this paper, we describe a four-year-old boy who presented with a solitary calcified tumor in the posterior part of his ankle. We performed a detailed evaluation using plain X-rays, a CT scan, and an MRI, which revealed a well-demarcated calcified tumor that had the characteristics of an osteochondroma but without any continuity with the bones of the ankle joint. The lesion was treated surgically with the excision of a giant osteochondral lesion. Pathological examination revealed mature cartilage at the periphery with cancellous bone in the central part.Thus, we present the clinical and laboratory investigation of a solitary extraskeletal osteochondroma in the ankle of a four-year-old boy, which is an extremely rare case. Osteochondromas are common benign lesions that affect the growing skeleton. They are primarily located in the metaphyseal area of long bones. Finding isolated osteochondromas in the bones of the hand or the foot is extremely rare. These osteochondromas have a clear origin in the bone of the foot or the hand, wherein the osteochondroma communicates with the medullary area of the host bone -3.Extraskeletal osteochondromas are rare and arise from the synovium. They are adjacent to a joint and have no continuity with the bone marrow of the proximal bones. They usually present as small multiple osteochondromas in a joint. Although no clear pathogenesis for osteochondromas exists, it is commonly suggested that they arise from synovial hyperplasia -7. The vThis paper describes a case of a four-year-old boy who presented with a solid lump that was a calcified tumor in the posterior aspect of his ankle. Radiographic evaluation using plain X-rays, a CT scan, and an MRI confirmed the diagnosis of osteochondroma. However, no clear communication was observed with the adjacent bones. The lesion was surgically excised, revealing a loose giant osteochondroma next to the ankle joint with normal synovial tissue. Pathology specimen confirmed the diagnosis of extraskeletal solitary giant osteochondroma. In this paper, we will present the clinical and radiological examination and the pathologic diagnosis for solitary extraskeletal osteochondroma in the ankle of a four-year-old child that, to the best of our knowledge, has not been reported previously. The study was approved by the Ethical Committee of Interbalkan Medical Center (193/14-3-2023).The family of a four-year-old boy brought the child to our pediatric orthopedic department for evaluation. His mother had noticed a solid mass in the posterior aspect of the left ankle. The boy was in excellent general health and was participating in all his age-related activities. He was able to walk without any noticeable limp and was performing hip-hop activities that showed no difference between the movements of his right and left legs.On conducting palpation, we found a solid, round, well-demarked mass without any skin alterations in the posterior medial part of the boy’s right ankle joint. No other bone projections were found in his skeleton. Dorsiflexion and plantarflexion of the affected ankle were restricted by 20 degrees when compared with the unaffected ankle. However, the patient did not feel any pain when pressure was applied to the mass.An X-ray of the ankle showed a calcified mass in the posterior and medial parts of the ankle joint. The distal tibial epiphysis was of normal shape, and the talus and calcaneum were of normal contour. Calcification was equally distributed in the mass Figures , 2.An MRI scan of the ankle revealed a heterogeneous mass adjacent to the posterior margin of the talus, with areas of intermediate- and high-signal intensity on short tau inversion recovery (STIR) sequences. Inhomogeneous enhancement was observed after intravenous administration of a paramagnetic agent. We could not observe a clear continuity either with the posterior aspect of the tibial or fibular epiphysis or with the posterior aspect of the talus. The synovium was normal without any signs of thickened bursa adjacent to the calcified tumor Figures -5.To further evaluate the relationship of the lesion with the adjacent bones, we performed a CT scan. The scan revealed that the lesion was largely calcified. A single lesion was found with dimensions of 19.6 mm longitudinal and 13.7 mm transverse. A small projection was noted in the posterior aspect of the talus; however, it had no clear continuity with the calcified mass. Based on these findings, we diagnosed the type of osteochondroma to determine whether it was extraskeletal osteochondroma or Trevor's disease. The differential diagnosis included myositis ossificans or calcinosis. Atypical extraskeletal Ewing sarcoma or chondroblastoma were exceptions for the final diagnosis  or a thin base (pedunculated osteochondroma). The bone marrow of the lesion should follow the magnetic resonance (MR) signal of the parent bone’s bone marrow in all sequences. The cartilaginous cap is well identified in MRI scans with a high MR signal on T2-weighted sequences due to high water content -16.Solitary synovial osteochondromas have a distinct radiological appearance. An X-ray evaluation reveals the presence of multiple periarticular calcified parts that present as loose bodies without bone erosion . These aEvidence suggests that synovial osteochondromas evolve in three phases. The first phase is characterized by active synovial disease; the second phase involves the formation of loose bodies; and in the final phase, only osteochondral bodies without synovial disease can be found ,19. MilgSolitary osteochondromas adjacent to a joint must be distinguished from the asymmetric overgrowth of the epiphysis, known as the Trevor disease. This usually affects the distal tibial epiphysis. Continuity with the epiphysis is observed, and it usually presents as spurs with endochondral ossification. It is covered cartilage onward, and projections are present in the ankle joint. Trevor disease can be localized, which affects a single epiphysis with more than one epiphysis in the same limb, or generalized, affecting the entire limp ,10,20-23In our patient, we could not detect any communication between the calcified mass and the adjacent bones. Other diseases such as myositis ossificans circumscripta, tophus calcification, and extraskeletal osteosarcoma were unlikely as the radiological evaluation had the characteristics of osteochondroma; however, they were included in the differential diagnosis. Solitary extraskeletal osteochondromas are histologically similar to osteochondromas that have communication with the affected bone ,15.Surgical treatment is the treatment of choice for the removal of osteochondromas and appropriate pathological examination. We performed an open procedure since the mass was protruding in the posterior aspect of the ankle and was not intra-articular-located. Our patient presented a loose giant osteochondroma without any connections with the adjacent bones.As our patient is young, we are regularly following him up at six-month intervals to evaluate the possibility of a relapse.


Epithelial-myoepithelial carcinoma is a rare malignant neoplasm of salivary glands. It is specifically found in the major salivary glands. The cases that emerge from minor salivary glands are rarely described. Histologically, it commonly exhibits a characteristic biphasic pattern consisting of epithelial and myoepithelial components. The histopathological resemblance to other benign and malignant neoplasms that also display myoepithelial characteristics makes the differential diagnosis challenging. Each differential diagnosis requires a very different management approach.Considering the difficulties of anatomopathological diagnosis and the rarity of epithelial-myoepithelial carcinomas emerging from minor salivary glands, we report a rare epithelial-myoepithelial carcinoma case of minor salivary glands in a 58-year-old woman. She was referred for a palatal swelling, evolving for more than 35 years, and reported recent pain and nasal obstruction. The mucosal swelling was located in the left maxilla within the hard palate, of a 45-mm-long axis crossing the medial line and extending to the premaxilla, without cervical lymph node involvement. A computed tomography scan revealed a palatal lesion involving the left and the right maxilla. Furthermore, the superior alveolar process, both left and right maxillary sinuses, the nasal cavities, and the nasal septum were included in the lesion. The final diagnosis was difficult to confirm despite multiple biopsies and was determined only from the excised specimen.The diagnosis of this tumor was challenging due to the clinical and histological similarities with other salivary tumors. The aim of this case report is to shed light on the distinctive features of these tumors and explore optimal screening and related management strategies. Epithelial-myoepithelial carcinoma (EMC) is a rare malignant tumor of salivary glands. Its prevalence is estimated in recent studies from 0.4% to 1% of all salivary gland tumors . The meaThe histopathological resemblance of EMCs to other benign and malignant neoplasms that also display clear myoepithelial characteristics makes EMC differential diagnosis challenging . Indeed,Considering the challenges associated with anatomopathological diagnosis and the rarity of EMCs emerging from minor salivary glands, we report a rare clinical case of minor salivary gland EMC. We aim to shed light on the distinctive features of these tumors and explore optimal screening and related management strategies ,3,6.A 58-year-old woman was referred by a general dentist for a palatal swelling, slowly evolving and completely overlooked by the patient for more than 35 years. No prior biopsies or treatments have been provided. The patient reported occasional pain and nasal obstruction for three weeks. She was not undergoing any treatment for these complaints. She had high blood pressure treated with candesartan and indapamide, hypercholesterolemia treated with atorvastatin, and hypothyroidism treated with levothyroxine. The patient presented active smoking with 30 pack-years. Physical examination revealed a mucosal swelling of the left maxilla within the hard palate, of a 45-mm-long axis crossing the medial line and extending to the premaxilla. This palatal mucosa swelling had a purplish-red color with polypoid outgrowth. It was firm on palpation Figure . The exaCone beam computed tomography (CBCT) and craniofacial computed tomography (CT) scan revealed a 45-mm-long axis, isodense lesion of the bony palate involving the left and the right maxilla, the superior left alveolar process, the nasal cavities, the lower part of the nasal septum and both left and right maxillary sinuses, with erosion of their medial wall. The lesion is rather lateralized to the left but extensively crosses the midline Figure .T1 gadolinium craniofacial magnetic resonance imaging (MRI) showed a tumor taking up gadolinium heterogeneously. The lesion was located in the hard palate with posterior extension to the soft palate. It measured 56mm, 44mm, and 36mm in length, height, and width respectively. MRI showed also bone invasion in most of the hard palate, in the right and left nasal cavities, and in the inferior turbinates. The maxillary sinus was also involved with lysis of the uncinate process on the left  showed a hypermetabolic lesion in the palate without nodal uptake Figure .These scans did not reveal any suspicious lesions elsewhere. No suspected hypermetabolic focus or submandibular and cervical lymph node involvement was found.Two oral biopsies were undertaken but were not conclusive. The first biopsy revealed a salivary gland tumor, most likely derived from a minor salivary gland, but the specific type could not be determined with precision. The differential diagnosis included EMC or myoepithelial carcinoma. The second biopsy provided additional insights, revealing a malignant tumor originating from the accessory salivary glands. It showed a dual epithelial and predominant myoepithelial component, suggesting an EMC, with the possibility of an ACC not being definitively excluded. Based on these anatomopathological results, it was deemed necessary to establish the precise histological type of the tumor by examination of the surgical specimen. Both the biopsies and the surgical specimens were analyzed by two qualified pathologists.On the basis of the clinical, radiological, and histological findings, this tumor was diagnosed as stage IV, with a tumor-node-metastasis staging of T4N0M0. After a multidisciplinary meeting, it was decided to perform a total maxillectomy and a reconstruction with a free fibula flap. Following the resection of the tumor, the histological analysis provided a definitive diagnosis of EMC, verifying that it was indeed a malignant tumor originating from the minor salivary glands, characterized by the presence of both myoepithelial and epithelial components. The myoepithelial cells were predominantly observed and were surrounded by hyaline material. Additionally, local progression of carcinomatous structures towards tumor necrosis was observed Figure .Immunohistochemistry analysis revealed the following findings: P63 showed diffuse and intense expression in the myoepithelial component, SOX10 displayed heterogeneous expression, while PS100, Calponin, and smooth muscle actin exhibited heterogeneous and less intense expression. The expression of cytokeratins AE1/AE3, EMA, and C-Kit (CD117) was mainly restricted to the epithelial component. C-Myb was negative, and there was no overexpression of P16 (P16 -).Based on these clinical, radiological, and histological findings, the final diagnosis was confirmed as EMC. The patient has been followed up for a period of two years, after prosthetic rehabilitation, and no recurrence or metastasis has been observed. The patient will continue to benefit from a personalized long-term follow-up.EMC is a rare tumor whose diagnosis can be clinically and histologically challenging. The absence of specific symptoms may also lead to delayed diagnosis. In this case, our patient had a longstanding lesion that had been developing for several years in the hard palate. The patient had overlooked this lesion for more than 30 years and did not seek any medical advice. Therefore, no biopsies or treatments have been provided during the long asymptomatic phase of the disease. Nasal obstruction and the onset of pain led her to seek dental consultation at the oral surgery department. However, in a case reported by Palaniappan et al., the palatal EMC was only of one-month duration and was quickly growing and becoming painful, leading to earlier diagnosis. It measured 15 mm on diagnosis. Two other palatal EMC cases were reported by Angiero et al., the first one was an asymptomatic lesion detected incidentally during a routine dental checkup with a diameter of 15 mm, and the second was an intermittent painful mass of 35×20 mm ,10. In oHistologically, the differential diagnosis may be confusing because of the complexities in distinguishing EMC from ACC and PA as described above. Microscopic examination shows similar biphasic composition of epithelial and myoepithelial cells in PA and EMC, which poses difficulties in the differential diagnosis. The anatomopathological analysis relies on combined HE with multi-IHC staining: EMA, p63, calponin, and the Ki-67 labeling index calculation. Vimentin and calponin are very sensitive and specific myoepithelial markers for salivary tumor myoepithelial cells that facilitate confirmation of diagnosis [According to the WHO classification of 2022, the EMC is considered a low-grade tumor . In a reIn our case, the clinical aspect of the tumor was as an intraoral mass, like the two cases reported by Angiero et al. describing two palatal masses in a 58-year-old woman and an 83-year-old man. However, in the case reported by Palaniappan et al., the palatal EMC was presented as a partially ulcerated lesion. All these patients were free of recurrence at the follow-up appointment of 15, 13, and 12 months respectively. Our patient had been followed-up for the longest duration which is 24 months post-surgery ,10.As regards the management of EMCs originating from the minor salivary glands, the role of radiotherapy is not well defined. Adjuvant radiotherapy is recommended in major salivary gland tumors when the primary tumor is >4 cm in size or the surgical margins are positive . HoweverIt is important to make the correct histological diagnosis because this changes completely the tumor management. Indeed, the ACC which is the principal differential clinical and histological diagnosis of EMC is classified by the WHO classification as an intermediate to high-grade tumor. It carries a poorer prognosis than EMC and requires post-surgical radiotherapy in almost all stages (II-VI). Moreover, as the tumor recurrence rate is much higher, a closer follow-up is recommended. The survival at five years is more than 95 % for stage I low-grade tumors and less than 10 % for stage IV high-grade tumors .Even though the efficiency of chemotherapy is not well established, some authors have suggested that chemotherapy be associated with radiotherapy when the delivered doses are high (around 65 Gy) especially when safety margins are not sufficiently extended while excision ,12.This article reported a rare case of EMC in the maxilla treated by maxillectomy. The diagnosis of EMC remains difficult due to its rarity. There is no consensus on the optimal management of the minor salivary glands EMCs. Longer-term cohort studies would provide a better understanding of their evolution and their optimal management and avoid late diagnosis. Clear standardized recommendations should, therefore, be put in place for the care of these patients.


Appendiceal inversion is a rare entity that can potentially mimic serious pathology and provide diagnostic uncertainty. They are mostly diagnosed intraoperatively or during endoscopies and scans for other reasons. We report a case of an asymptomatic patient treated for colon cancer without previous history of appendectomy. We provide long-term follow-up and aim to review the relevant literature. Appendiceal inversion or intussusception is an uncommon finding affecting about 0.01% of adults . AlthougIatrogenic inversion is done during open surgeries for appendicitis or malrotation and historically in normal appendixes to theoretically avoid future peritoneal contamination in cases of inflammation . This isAppendiceal intussusception has been theorized to occur due to local irritation causing abnormal peristalsis . SeveralWe report an incidentally found inverted appendix in a patient with colon cancer that underwent prolonged endoscopic and radiological follow up for their primary malignancy.Our patient is an 83-year-old Middle Eastern male. He initially presented to our center in late 2003 with bleeding per rectum. Clinical examination was unremarkable and a colonoscopy showed a descending colonic tumor confirmed as moderately differentiated adenocarcinoma. His staging was free of distant disease, and he underwent a left hemicolectomy with a final pathology of T3N1. His postoperative course was smooth and he went on to finish his planned adjuvant chemotherapy.His medical history includes hypertension, controlled diabetes mellitus type 2 and benign prostatic hyperplasia. He is a long-time smoker. His surgical history other than his colonic resection includes two TURP procedures in 1989 and 1992 and a diaphragmatic hernia repair in his late teens. There is no history of an appendectomy in the past.Our patient had a follow-up regimen of colonoscopies and CT scans for his colon cancer. The first mention of an inverted appendix was on his first postoperative colonoscopy in February 2005. It was also subsequently identified in further colonoscopies in years 2007, 2012 , 2017, and 2023 .CT scans also picked up this anomaly as early as 2010 . FurtherThe inverted appendix is a relatively rare occurrence. It most commonly affects females in their fourth decade and found incidentally on colonoscopies and CT scans. While they are benign in nature, they can present with symptoms if associated with other pathologies such as endometriosis, adenomas or mucinous neoplasms .Endoscopically, it appears as a tubular elongated polypoid structure arising from the appendiceal orifice , 12. EndAppendiceal intussusception is classified according to the McSwain classification: Type 1: tip of the appendix invaginates into the appendiceal lumen. Type 2: extension of type 1 where there is more pronounced invagination beyond the tip of the appendix. Type 3: the appendix intussuscepts all the way to the appendiceal orifice at the caecum, which is the most common type. Type 4: retrograde inversion of the proximal appendix distally toward the tip. Type 5: complete invagination of the appendix through the orifice into the cecum . Our casThere are no guidelines on how inverted appendices are treated . There iIn our case, we showed that long-term follow-up for appendiceal inversion in an asymptomatic patient is reasonable and appeared to be safe and reassuring. We did not notice any change in endoscopic or radiological morphology that warranted removal. Awareness of this entity is important to avoid unnecessary investigative risks.


Pseudotumor deltoideus is a localized region of irregular cortical thickening at the deltoid insertion that causes a diagnostic quandary for radiologists due to its unusual radiological characteristics. It is benign in origin, with the potential to act as a tumor stimulator, and has a variety of anatomic variants. The lesions can be characterized on an X-ray by an area of lucency at or around the deltoid tuberosity and a cortical irregularity or nearby eccentric marrow abnormality on computed tomography (CT)/magnetic resonance imaging (MRI). The presence of cortical thickening and lucency at the deltoid insertion are unusual radiological findings that provide a diagnostic challenge. In this article, we provide cases of shoulder pain together with radiological imaging to make this previously underappreciated condition more understandable. Further evaluation with CT/MRI should be performed in all cases of shoulder pain with conventional radiographic findings of cortical thickening and intracortical lucency. The diagnosis of the condition is aided by the presence of elongated lucency on CT and T2 hyperintensity in the cortex of the proximal humerus. The clinical and imaging characteristics are important in the diagnosis of this condition. It must not be confused for infection or malignancy, and a biopsy must never be attempted. Morgan et al. first used the term “pseudotumor deltoideus” in 2001 after they published a study on individuals who had shoulder pain and observed similar radiological clues at the deltoid insertion [Case IA 54-year-old male presented with a complaint of chronic left shoulder pain with mild restriction of movements. There was no history of trauma, significant weightlifting, or sports participation. On examination, there was pain associated with abduction and internal rotation of the nondominant left shoulder. Imaging revealed irregular cortical thickening, intracortical lucencies, and hyperintense T2-weighted lesion at the site of the deltoid muscle insertion. Based on these clinical and radiological findings, the diagnosis of pseudotumor deltoideus was made, and conservative treatment was advised. The patient was started on nonsteroidal anti-inflammatory drugs (NSAIDs), as well as regular physiotherapy and hot fomentation, and was then called for a follow-up appointment. The severity of the shoulder pain had significantly decreased at the two-month follow-up evaluation, and the complete range of motion had been restored. The follow-up X-ray, however, revealed no change. Case IIA 39-year-old female presented to OPD with a six-week history of left shoulder pain and difficulty with overhead movement. On physical examination, there was a mild restriction of overhead abduction. She was right-hand dominant with no associated history of trauma. Imaging revealed similar irregular cortical thickening and intracortical lucent lesion. She was managed conservatively and showed significant symptom resolution.Case IIIA 27-year-old right-hand-dominant female presented with a long-standing history of right upper and mid-arm pain. There was no associated history of trauma or sports-related injury. The physical examination of the patient revealed pain with internal and external shoulder rotation. Computed tomography (CT) and magnetic resonance imaging (MRI) evaluation revealed an intracortical lucent lesion with T1 hypointense and T2 hyperintense signal changes suggestive of pseudotumor deltoideus. He was managed conservatively, and the follow-up showed a resolution of symptoms.When an individual with a history of shoulder pain is observed in conjunction with imaging features such as irregular cortical thickening and intracortical lucency in the proximal humerus, it can lead to an unneeded biopsy or investigations based on the assumption of a malignant disease -3. To adAlthough several theories have been proposed to explain this phenomenon, it is uncertain what really caused it. A few possible explanations include an anatomic variant, erosions associated with tendinitis, secondary changes at the deltoid insertion due to burned-out neoplasm, and avulsive cortical irregularities . SimilarChronic avulsion injury of the deltoid also can present with similar clinical and imaging characteristics, posing a dilemma for the radiologist. MRI is helpful with features such as prominent thickness and irregularity of the cortex, increased T2-weighted signal intensity in the tendon insertion, and edema in the adjacent soft tissue directing towards avulsive injury . The preNeoplasm should be excluded during the evaluation of bone abnormality. Due to their eccentric position, two benign neoplasms, fibrous cortical defects and non-ossifying fibromas, are given special consideration as differentials ,8. They In this article, cortical thickening and intracortical lucency were observed at the site of the deltoid insertion. The radiological imaging findings were consistent with both pseudotumor deltoideus and chronic avulsion injury of the deltoid. There was no bony fragment, and MRI revealed no edema in the soft tissue adjacent to the deltoid insertion. The diagnosis of pseudotumor deltoideus was made after considering the patient's age, symptoms, a lack of history of injury from sports or other strenuous activity, and imaging findings. The same was confirmed during a two-month follow-up following physiotherapy, NSAIDs, and conservative care that resulted in symptom remission. Few studies have found a link between arm dominance and the prevalence of pseudotumor deltoideus . In our Various studies such as those of Singh and Tanwar, Morgan et al., and Adiguzel et al. described similar radiological clues of pseudotumor deltoideus ,2,9. SymA diagnosis of pseudotumor deltoideus should be taken into consideration in patients with shoulder pain when imaging reveals cortical thickening of the proximal humerus at the site of the deltoid insertion accompanied with irregularity and intracortical lucency on CT and T2 hyperintensity on MRI without any surrounding soft tissue changes. Unwanted biopsies should be avoided, and MRI is useful in the differential diagnosis.


Giant gallstone with a secondary hepatic abscess is a very rare phenomenon. We recently treated a patient with a giant gallbladder (GB) stone of size 11.5 cm with a hepatic abscess who presented with features of an acute abdomen. This was subsequently managed with an open subtotal cholecystectomy and concomitant hepatic abscess drainage. To the best of our knowledge and after a thorough literature search, this is one of the largest reported gall bladder (GB) stones with wall perforation and hepatic abscess in the Asian subcontinent. Gallstones are very common in southeast Asia, including the Gangetic part of India . GallstoA 57-year-old female with a history of hypertension presented with complaints of fever. The fever persisted over 15 days, was of sudden onset, associated with chills and rigor, and had a maximum recorded temperature of 103°F, with no significant diurnal variation. She reported pain in the upper abdomen for one day, which was sudden in onset, localized to the right upper quadrant, radiating to the right scapular angle, dull aching, moderate to severe in intensity, aggravated by food intake, and relieved by intravenous (IV) analgesics. There were no other bladder or bowel disturbances, loss of weight, or loss of appetite. The patient reported a history of yellowish discoloration of her eyes six months prior, which was not associated with itching or clay-colored stool, which was resolved with medical remedies.2 98% on room air. She weighed 74 kg with a height of 164 cm and body mass index (BMI) of 27.5. On abdominal examination, there was tenderness and guarding over the upper abdomen. No organomegaly or free fluid was noted.On examination, the patient was hypotensive with tachycardia (104/min), temperature 102°F, and SpOAfter starting IV analgesics and antibiotics, she was evaluated with an ultrasound abdomen that showed a gallstone of size 11.5 cm, impacted in the gallbladder (GB) neck, with discontinuation of the lateral gallbladder (GB) wall facing the liver along with an adjacent collection of 7.6 × 7 cm in the GB fossa. A plain abdominal x-ray showed an oval radiopaque shadow of size 5 × 5 cm in the subhepatic region without any evidence of intestinal obstruction  imaging on the same day that showed the GB as being over-distended and thick-walled 5.3 mm), with large radiopaque calculus (11.5 × 6 cm), with a breach in the lateral wall of the GB towards the surface of the liver Figure , and wit mm, withWith the working diagnosis of GB perforation, the patient was initially managed conservatively, and subsequently, surgical intervention was planned because of worsening abdominal symptoms and signs. Given the complicated nature of the giant gallstone, an open approach was considered for the patient's safety and the predicted difficulty of laparoscopy. After a generous Kocher’s incision, intraoperatively 100 ml of ascitic fluid was drained, and the note was made of omental clumping near the subhepatic region. A bilio-purulent collection of approximately 200 ml was drained from the liver near the GB fossa. The GB was inflamed, thick, and large, approximately 15 cm × 12cm, with a frozen Calot's triangle. No significant enlarged lymph nodes were noted around the GB, CBD, or portal vein. The GB fundus was incised transversely, and a stone of size 11.5 cm was removed with difficulty Figure . The mucPostoperatively, the patient was hemodynamically stable and afebrile. Abdominal signs settled over the next postoperative day, with minimal serous drain output. The patient developed a low-output biliary fistula postoperatively, which was successfully managed with ERCP CBD stenting using a 10 Fr biliary pigtail stent . Drain output became nil a day after ERCP stenting. The subhepatic drain was removed subsequently, and the patient was discharged with advice for stent removal after six weeks.The histopathological examination of the resected GB part depicted features suggestive of xanthogranulomatous cholecystitis (XGC). The patient was advised for regular follow-up with interval imaging. Gallstones disease (GSD) is more common in females, especially during their fertile periods. This is possibly due to increased estrogen turnover, with secondary effects on cholesterol efflux into the bile and GB motility . In IndiThe etiopathogenesis of gallstones results from an irregularity in the typical relationship among the major constituents of bile, including cholesterol, phospholipids, and bile acids ,7,8. TheClinically, GSD can have a broad spectrum of presentations, ranging from asymptomatic cases to biliary colic, acute and chronic cholecystitis, GB perforation, choledocholithiasis, and acute pancreatitis ,8. ApproGB perforation might be documented in 2-11% of patients with acute calculus cholecystitis, resulting from ischemia and necrosis ,11. The Laparoscopic cholecystectomy has been the standard of care for GSD, which can be achieved in almost 96% of cases ,5,7. TheA giant gallstone presenting with gall bladder perforation and a hepatic abscess is a rare entity that needs early diagnosis and immediate surgical intervention. Although a gallbladder perforation is usually managed with a conservative approach comprising a tube cholecystostomy or percutaneous drainage followed by interval laparoscopic/open cholecystectomy, an immediate surgical intervention either laparoscopic or open, would be more promising in hemodynamically unstable patients. Although laparoscopy is the standard of care for gallbladder stones, an open approach with total or subtotal cholecystectomy would be more appropriate and safer for complicated giant gallstones presenting with a frozen Calot’s triangle and compromised hemodynamic status. Early surgical drainage and cholecystectomy are the cornerstones of management for such complicated case. This index case is the largest reported complicated gallstone in the Asian subcontinent and was managed by open subtotal cholecystectomy.


Proximal-type epithelioid sarcoma of the perineum is a rare soft-tissue malignancy, and only 55 cases have been reported in the English literature to date. This tumor has an indetectable early symptom and frequent recurrences. Here, we present the case of a 31-year-old man with proximal-type epithelioid sarcoma of the perineum who underwent wide excision. Further, we reviewed the current literature regarding differential diagnosis and management of this disease. Magnetic resonance imaging (MRI) revealed a well-circumscribed solid and homogeneous lump in the right perineum Figure 1Macroscopically, the resected oval-shaped specimen measured 8 × 10 × 5 cm and was enclosed by a rim of fatty tissue Figure 33Proximal-type ES is an uncommon but aggressive soft-tissue malignancy with a poor prognosis. However, deficient information is available in the literature regarding the clinical aspects and management of proximal-type ES of the perineum. Seeking immediate treatment is uncommon in these patients because the tumor is often painless. Therefore, a proximal-type ES located deep in the perineum may become quite large at the initial diagnosis. Plain radiography, MRI, CT, and PET can help estimate the volume of the soft tissue mass and determine its metastaticity. Moreover, histological diagnosis of the soft tissue mass is essential.Unlike most soft-tissue sarcomas that are classified according to the presumptive tissue of origin, such as liposarcoma, synovial sarcoma, or leiomyosarcoma, the histogenesis of ES remains unclear. Immunohistochemical staining, however, may help identify the presumptive tissue of origin in patients with ES. ES is typically positive for vimentin, CK, and EMA.Surgical excision with negative gross margins is the mainstay of therapy for patients with primary sarcoma. Generally, there is no evidence of distant metastasis at the initial diagnosis. For localized disease, adjuvant radiotherapy after wide surgical excision is necessary to reduce the risk of local recurrence. In a retrospective review of 1,170 patients with newly diagnosed soft tissue sarcoma during a 7.5-year period, the incidence of distant metastases at diagnosis was 10%, 83% of which were in the lungs . HematogOwing to the rarity of such tumors of the perineum, published treatment recommendations are unavailable. At present, total surgical resection combined with postoperative chemotherapy and radiotherapy is the major treatment modality for primary proximal-type ES. However, EZH2 inhibitors and immunotherapy may represent potential treatments for such tumors . New feaThe original contributions presented in the study are included in the article/supplementary material. Further inquiries can be directed to the corresponding author.Ethical review and approval was not required for the study on human participants in accordance with the local legislation and institutional requirements. The patients/participants provided their written informed consent to participate in this study.SX: analysis and interpretation of data, drafting the article or revising it critically for important intellectual content; WW: final approval of the version to be published; LM: acquisition of data. All authors contributed to the article and approved the submitted version.


We reported two cases of the central nervous system (CNS) multiple myeloma (MM) with unusual presentation of sixth nerve palsy. The first patient developed in the setting of newly diagnosed MM and the second patient in the relapsed refractory setting. One underwent surgery, and the other received radiation. Both patients received systemic chemotherapy and noted improvement. We also performed a comprehensive literature review of previously published cases of sixth nerve palsy from MM. This review highlights the importance of recognizing this presentation of CNS multiple myeloma to avoid delays in diagnosis and to get appropriate management in time. Multiple myeloma (MM) involving the central nervous system (CNS) has various manifestations along its spectrum. The most common presentations are bone lesions as a part of systemic disease, causing significant pain. On the contrary, localized plasmacytomas can arise from the cranium (intraosseous) or dura, meninges or parenchyma (extramedullary) with or without evidence of systemic disease .The clinical symptoms of CNS plasmacytomas depend on the part involved ranging from extremity weakness, change in mental status, gait and visual disturbances to cranial nerve palsies . The diaCase 1A 55-year-old Caucasian male presented with a one-month duration of double vision mainly worse on looking to the right side, numbness of the right lateral aspect of lips and gums, right-sided hearing loss and tinnitus. He was also evaluated by an otolaryngologist and found to have asymmetric sensorineural hearing loss (SNHL) on the audiogram. He was evaluated by an ophthalmologist and diagnosed with right sixth nerve palsy. Computerized tomography (CT) scan showed a 4.2 cm × 2 cm expansile mass in the right jugular foramen with erosive changes of the adjacent bones including the right clivus, occipital condyle, petrous apex, erosion of the carotid canal and posterior wall of the sphenoid sinus with the extension of the lesion into the sphenoid sinus. There was no evidence of extension to the middle ear or involvement of the inner ear structures. Magnetic resonance imaging (MRI) confirmed the same findings as CT Figures -1D. He uOn further workup, bone marrow biopsy showed 6% plasma cells on aspirate and 15%-20% on core biopsy. The minimal residual disease (MRD) test was positive at 2.83%, and flow cytometry showed CD138, CD56, and CD38 positive and negative for CD45, 19, and 20. Fluorescent in situ hybridization (FISH) multiple myeloma panel showed +1q21.Lab work showed a hemoglobin level of 12.6 g/dl, calcium 10.1 mg/dl, serum creatinine 1.1 mg/dl, M component in serum 0, Bence-Jones proteinuria at 272 mg in 24 hours urine with a free kappa light chain in the urine immunofixation, with kappa light chain level of 102.8 mg/dL, with a kappa lambda ratio of 121.8, with decreased IgA and IgG levels and normal IgM levels. He was found to have several bone lesions defined by the positron emission tomography (PET) scan and the MRI studies including the sternum, the right shoulder, the scapula, the left clavicle and the skull base with very likely extramedullary disease in the right pelvic soft tissue measuring up to 5 cm in size. He was diagnosed with kappa light chain multiple myeloma, and he received induction chemotherapy with daratumumab and carfilzomib, dexamethasone, thalidomide, cisplatin, adriamycin, cyclophosphamide and etoposide (KDT-PACE). His ocular symptoms have completely resolved, and he will be undergoing an autologous stem cell transplant (ASCT) shortly.Case 2A 69-year-old Caucasian male with a past medical history of long-standing multiple myeloma presented with double vision of two weeks duration. He was off of any therapy for his MM when he developed diplopia. Prior to that he was having MM for almost six years and was extensively treated with induction, tandem ASCT and consolidation therapies and received multiple lines of maintenance regimens. On exam, he was noted to have left sixth cranial nerve palsy with no other neurological findings. MRI of his brain revealed a 2.2 × 1.0 × 1.8 cm enhancing mass in the left petrous apex and posterior cavernous sinus. PET scan showed fluorodeoxyglucose (FDG) uptake in the same lesion with no other new lesions or extramedullary disease (EMD) Figures -3E. His Based on the above findings, he was started on high-dose steroids, dexamethasone 40 mg daily for four days. He also underwent radiation to the lesion for five weeks. Post-radiation, his symptoms completely resolved. Imaging also demonstrated a reduction in left clivus lesion size and resolution of diffusion restriction Figures -4D. FollWe reported two cases of multiple myeloma associated with sixth cranial nerve palsy. Case 1 developed in the setting of newly diagnosed MM, while case 2 developed in the relapsed refractory background. Case 1 was noted to have multiple cranial nerve palsies, but case 2 was noted to have isolated sixth nerve palsy. Both the patients' MRIs clearly showed the origin of the lesion. Case 1 underwent surgery for tumor removal, but case 2 received radiation. Diplopia is resolved in both cases post-operatively and post-radiotherapy, respectively. Both patients received systemic chemotherapy and are currently in remission with no recurrence of neurological symptoms.Intracranial plasmacytomas are rare plasma cell neoplasms constituting less than 1% of all intracranial tumors and can occur as part of the manifestation of MM or as a solitary (primary) lesion arising from the skull, meninges and the brain . PlasmacWe performed a PubMed/Embase/Web of Science-based comprehensive literature review for previously published cases of sixth cranial nerve palsy among MM patients until November 2022. The search terms included abducens nerve" OR "sixth cranial nerve" OR "sixth nerve" OR "6th nerve" OR "6th cranial nerve" (Topic) and palsy OR palsies OR paralysis (Topic) and "multiple myeloma" OR "plasma cell myeloma*" (Topic). We reviewed all results and found the following previous publications as outlined below in Table The sixth nerve palsy in intracranial plasmacytomas can occur in both newly diagnosed and RR backgrounds. The symptoms' duration varies from weeks to months prior to the final diagnosis. The delay in diagnosis might be due to the challenges faced especially in newly diagnosed settings due to the rarity of occurrence with a low index of suspicion and the multitude of pathologies that can cause similar presentations. From Table Out of many risk factors associated with the development of CNS MM, one study noted that chromosome 17p 13.1 deletion was associated with 89% of CNS MM patients which was not seen in both of our patients . HoweverTreatment of CNS plasmacytomas can be approached through three modalities: surgery, radiation and chemotherapy. Surgery is usually reserved for tumors causing significant mass effect. Sometimes it serves as a modality in reaching out for a diagnosis of plasmacytoma . But oncTo conclude, we presented two cases of unusual presentations of CNS MM as sixth nerve palsy. The lesion was localized in MRI in both patients. One patient underwent surgery followed by systemic chemotherapy, and another patient underwent radiation first followed by systemic chemotherapy. Both showed improvement in their symptoms. This case series highlights the CNS as an unusual site for MM to start or relapse. A high index of suspicion needs to be maintained. Treatment should be initiated promptly with surgery or radiation followed by systemic chemotherapy.


Dysphagia is a common issue observed among the elderly, which can arise from various etiologies such as motility disorders and chronic neurologic conditions. Radiologists play a crucial role in diagnosing the cause of dysphagia, as they can identify anatomical abnormalities that may lead to the condition. One such anomaly is the hemiazygos vein, which is the left side equivalent of the azygos vein and can cause dysphagia if it crosses over the esophagus. To our knowledge, there are only 2 other recorded cases of azygos aneurysm/dilation causing esophageal dysphagia. In this context, we present a case report of a 73-year-old female with a 1-month history of weight loss and dysphagia due to a prominent hemiazygos vein. The case highlights the importance of thorough radiological evaluation in identifying the underlying cause of dysphagia and ensuring timely and appropriate treatment. Dysphagia is a common disorder experienced by the elderly. Thirteen percent of those 65 years and older and 51% of elderly individuals in long-term care facilities are affected by dysphagia Venous abnormalities rarely produce a mass effect, due to their easy compressibility. We report a case of a 73-year-old female with a month-long history of weight loss and dysphagia in the setting of a prominent hemiazygos vein confirmed by imaging. To our understanding, this is only the second case of a prominent hemiazygos vein causing an esophageal abnormality.A 73-year-old female with a medical history of asthma, osteoporosis, prior herniated discs, diverticulosis, prediabetes mellitus, and a 40-pack-year smoking history presented to the emergency room (ER) with a 1-month history of difficulty swallowing both fluids and solids, along with a sore throat. Four days prior to admission, she experienced one episode of black stools and right-sided neck pain while eating chicken, and reported a choking sensation, dysphagia, and odynophagia. The patient also reported night sweats, occasional constipation, sharp chest pain with swallowing, and unintentional weight loss of 10-20 pounds within the 1-month period. She denied any shortness of breath, tongue swelling, nausea, or vomiting. The patient recalled a colonoscopy 2 years prior that was inconclusive due to poor visualization from rectal bleeding.2 saturation on room air. Physical examination revealed right posterior cervical lymphadenopathy, but the patient appeared otherwise comfortable, and cardiovascular, pulmonary, and abdominal examinations were unremarkable. Palpation did not reveal any thyromegaly, and the abdomen was flat, soft, and nontender throughout. No hepatosplenomegaly was appreciated, and a digital rectal examination was negative for bright red blood or melena. Laboratory studies conducted during admission, including a complete blood count, basic metabolic panel, and hepatic function testing, were unremarkable for anemia, electrolyte, renal, or hepatic abnormalities. Given the patient's weight loss, progressive dysphagia and risk factors, esophageal malignancy was strongly considered as a potential diagnosis at this time.Upon admission to the ER, the patient's vital signs were as follows: body mass index of 25, temperature of 98.0°F, blood pressure of 135/79 mmHg, heart rate of 68 bpm, and respiratory rate of 16 bpm with 98% OIn the ER, the patient underwent a computerized tomography (CT) scan of the neck due to symptoms of choking sensation, pain and swelling. The scan revealed mild thickening of the upper esophageal wall, and a 3.5 × 2.7 cm mass located at the level of the carina. These findings were concerning for a possible neoplasm .Fig. 1CTAfter being admitted to the hospital due to a possible neoplasm, the gastroenterology service was consulted on the patient's second day of stay. They recommended an esophagogastroduodenoscopy (EGD), which was performed the same day, revealing no evidence of luminal lesions but indicating a possible superficial gastric ulcer . The EGDAfter receiving negative results from the EGD, we consulted the pulmonology service on day 7 of her stay due to the presence of right posterior cervical lymphadenopathy that was identified during the initial physical exam. Our goal was to rule out any potential intrathoracic malignancies. Following their recommendation, we conducted a CT chest with contrast, which revealed a soft tissue mass measuring 1.36 × 1.77 × 1.96 cm located in the posterior esophageal wall at the T5-T7 vertebrae level  and 4.FiAfter discovering the presence of a mass in the posterior esophageal wall, we added gastrointestinal mesenchymal tumors such as leiomyoma, leiomyosarcoma, and gastrointestinal stromal tumor to our list of potential diagnoses due to the possibility of involvement of the submucosal and muscularis layers of the esophagus. To gain further insight, we ordered a CT abdomen and pelvis, which revealed scattered diverticulosis of the colon consistent with previous CT scan findings . ThickenOn the eighth day of hospitalization, the interventional radiologist recommended a positron emission tomography (PET) scan after reviewing the CT images mentioned above to conclusively rule out the possibility of a pathological mass, and also recommended an endoscopic ultrasound. Upon review with radiology, it was discovered that a mass next to the mid-thoracic esophagus, initially detected by the PET scan, was actually a prominent hemiazygos vein with no significant increase in metabolic activity .Fig. 6, only one mentions the dilation of its tributary vein, the hemiazygos vein The cause of hemiazygos vein dilation can be further classified into physiologic and pathologic etiologies. Physiologic causes include azygos continuation of the inferior vena cava (IVC), and congenital superior vena cava (SVC) or IVC interruptions. In contrast, pathologic causes of a dilated hemiazygos include congestive heart failure, constrictive pericarditis, SVC and IVC obstruction, pericardial effusion, portal hypertension, right ventricular strain, and tricuspid insufficiency To understand the potential cause of dysphagia in this patient due to hemiazygos vein dilation, it is essential to comprehend the anatomy of the azygos vein. The azygos vein originates from the confluence of the upper lumbar and right subcostal veins before entering the thorax As mentioned earlier, the majority of cases of hemiazygos vein dilation or AVA are often asymptomatic and are discovered incidentally. However, symptoms may arise when aneurysms are larger than 5 cm and compress surrounding structures such as the esophagus, bronchi, SVC, and pneumogastric nerve To better understand the presentation of hemiazygos vein dilation/AVA, we can look at other case reports. Currently, to our knowledge, only 2 other recorded cases of azygos aneurysm/dilation causing esophageal dysphagia exist. Morton et al. Although complications such as mass effect have been described as a result of AVA/hemiazygos vein dilation, there is no universally accepted management strategy. For asymptomatic patients, a conservative or surveillance approach is utilized in the majority of cases When elderly patients present with dysphagia, consideration of other multifactorial causes of dysphagia is important, in addition to more common etiologies. In cases of hemiazygos vein dilation, radiologists play a crucial role in detecting and diagnosing the condition using imaging such as CT, MRI, PET, and ultrasound. Once diagnosed, surgeons may be consulted to determine the appropriate management strategy, whether it be conservative observation or surgical intervention.Shivam Khatri: Case Writer and Literature Review. Salomon Chamay: Case Writer and Literature Review Steven Chacko: Case Writer and Literature Review. Shorabh Sharma: Editor.Informed consent was obtained from the patient before writing up this case study.


Primary dural Hodgkin lymphoma (PDHL) is an extremely rare subset of Hodgkin lymphoma (HL). Its existence is controversial, as Hodgkin lymphoma is not traditionally thought to arise from the central nervous system (CNS) or its meninges and only 0.02% of patients with Hodgkin lymphoma have any CNS involvement. We report a case of a 71‐year‐old Caucasian man who presented with progressive fatigue and sudden onset slurred speech, disorientation, and memory loss. Brain imaging identified a large extra‐axial right frontal mass, and he underwent urgent subtotal resection. Pathology and subsequent workup revealed Stage IAE classical Hodgkin lymphoma of the right frontal dura, with no extra‐cranial disease or leptomeningeal spread detected. The patient was subsequently treated with ABVD chemotherapy (completed 2.5 of 4 planned cycles) and 36 Gy in 20 fractions of consolidative involved‐site radiotherapy (ISRT). He has been followed for 5 years with no clinical or radiological signs of recurrence. This is the second confirmed case of intracranial PDHL reported in the literature, with the longest follow‐up for any case of PDHL. Rare case of a 71 year old man with intracranial primary dural Hodgkin lymphoma (PDHL). Patient underwent subtotal resection of the mass, followed by adjuvant chemotherapy and consolidative radiotherapy, with no recurrence at 5 years follow up. Non‐Hodgkin lymphoma (NHL) is more common than Hodgkin lymphoma (HL) and typically arises in lymph nodes anywhere in the body, whereas HL typically occurs in the upper body. Central nervous system (CNS) involvement for patients with Hodgkin lymphoma is rare, and more commonly a result of secondary disease processes. It is estimated that only 0.02% of patients with Hodgkin lymphoma have CNS involvement.On initial presentation, PDHL patients are often symptomatic due to tumor mass effects, commonly presenting with headaches, cranial nerve deficits, dysesthesias, vertigo, and/or confusion. Due to the lack of specific HL‐related symptoms, the initial diagnosis of PDHL can be challenging as it often mimics meningioma on imaging, and histopathological confirmation typically requires surgical intervention for tissue sampling or excision. Depending on the extent of CNS involvement, attempts at complete excision and subsequent therapeutic management decisions can be challenging.In this report, we describe the case of a 71‐year‐old Caucasian male with primary‐stage IAE Hodgkin lymphoma of the right frontal dura who was treated with subtotal resection, ABVD chemotherapy, and consolidative involved‐site radiotherapy (ISRT). He has been followed for 5 years with no signs of recurrence. This is the second case of intracranial PDHL reported in the English literature.2In October 2016, a 71‐year‐old Caucasian man presented to a community emergency department in Ontario with a likely episode of unwitnessed seizure, new‐onset slurred speech, disorientation, memory loss, and lightheadedness. He also had a one‐week history of profound progressive fatigue. He did not have a measured fever, drenching night sweats, or weight loss. His past medical history included dyslipidemia, hypertension, obstructive sleep apnea, chronic lower back pain, and a previous lacunar stroke from which he had full recovery of function. The patient was managed initially in the emergency department with low‐dose aspirin, dexamethasone, and phenytoin.CT head with contrast revealed a 5 × 3.5 cm extra‐axial mass with smoothly marginated borders, overlying the right frontal lobe in the anterior cranial fossa. There was a mass effect on the right frontal lobe with a moderate degree of surrounding vasogenic edema. The initial radiologic impression was likely meningioma based on the round appearance and wide base on the floor of the dura at the right frontal fossa, along with a homogenous pattern of enhancement. MRI brain completed the subsequent day confirmed the tumor, but raised suspicions of lymphoma, as the mass had reduced in size in response to dexamethasone Figure . The patMicroscopic examination showed a lymphoproliferative neoplasm characterized by extensive areas of fibrosis, collections of small lymphocytes, and scattered larger atypical Hodgkin cells with pale eosinophilic cytoplasm, vesicular nuclei, and prominent nucleoli Figure . The lat9, erythrocytes 5.13 × 10,9 thrombocytes 340 × 10,9 neutrophils 8.6 × 10,9 lymphocytes 2.1 × 10,9 monocytes 1.4 × 10,9 eosinophil 0.1 × 10,9 basophil 0. Serology was negative for Hepatitis B, C, and HIV. Cardiac left ventricular ejection fraction was 74% on echocardiogram. Bone marrow aspirate and biopsy were negative for lymphoma. Lumbar puncture did not identify any leptomeningeal involvement. A full‐body PET/CT scan showed no metabolic activity at the resection site and no extracranial sites of disease. MRI revealed the residual disease following sub‐total resection. The final diagnosis was Stage IAE classical Hodgkin lymphoma involving the right dura.Blood work showed leukocytes 12.3 × 10The case was reviewed at a multidisciplinary case conference and a recommendation was made to treat the patient with four cycles of adriamycin, bleomycin, vinblastine, and dacarbazine (ABVD) followed by ISRT. The patient started cycle IA on Nov 18, 2016. Unfortunately, his treatment was complicated by febrile neutropenia requiring hospital admission and IV antibiotics, which resulted in a dose reduction of cycles 1B and 2A. The patient completed cycle 3A of ABVD chemotherapy, but chemotherapy was then discontinued at the patient's request due to poor quality of life from intolerable side effects of general malaise and fatigue. A post‐chemotherapy MRI head revealed an improving cystic collection at the previous surgical site, however the thick lobulated enhancement of the dural margin had increased in size, to 4.5  × 1.4 cm, felt to represent residual tumor mixed with reactive dural enhancement  to the right frontal region of the brain. Radiation volumes were delineated using an involved site definition focusing on the right dura post‐operative tumor bed plus residual fibrosis/tumor tissue. Radiation was delivered via Linac‐based intensity modulated volumetric arc technique (IMRT‐VMAT), using two opposing arcs and 6 mV photons. He did not receive any dexamethasone during RT. There were no unanticipated or severe acute or subacute toxicities observed.Following the completion of radiation, the patient underwent a surveillance CT head every 3 months for the first year. He was discharged from routine follow‐up at the cancer center in Feb 2019 at his request. Subsequent clinical follow‐up care was provided by his primary care provider, and this consisted of yearly visits and blood work (including CBC and LDH). The patient continues to be stable neurologically and is followed by a neurologist for antiepileptic management, on an annual basis, with no breakthrough seizure activity in over a year. Follow‐up imaging with MRI head in Jan 2019 Figure  and CT h3The vast majority of PDL cases are categorized as NHL, so clinical information is scarce regarding PDHLOur patient presented with slurred speech, memory loss, and lightheadedness, due to tumor mass effects, in contrast to published cases of PDHL that presented with neuromuscular weakness due to intradural spinal masses.In the extracranial setting, Hodgkin lymphoma is very responsive to both chemotherapy and radiation. Since the ABVD chemotherapy regimen was first introduced in 1975, it has been the standard of care for first‐line treatment of classical Hodgkin lymphoma without CNS involvement.In this clinical case, following chemotherapy, ISRT, a modern approach to radiation, was employed successfully. Thirty–six Gy was chosen given that the patient did not complete the 4 planned cycles of chemotherapy, and there was also radiologic evidence of residual dural enhancement post‐chemotherapy Figure . The volDue to the favorable prognosis of early‐stage Hodgkin lymphoma, there must be a careful balance between providing sufficient therapy to eradicate the disease while also minimizing long‐term sequelae from treatment.To make an early diagnosis of PDHL or any PCNSL subtype, the disease must be limited to the CNS; this contrasts with secondary CNS involvement by systemic lymphoma. The international PCNSL Collaborative Group recommends screening for extra‐cranial involvement using a CT scan of the chest, abdomen, and pelvis, whole body fluorodeoxyglucose positron emission tomography (FDG‐PET), bone marrow biopsy, and testicular ultrasound . Recent studies have questioned the necessity of bone marrow biopsy in the presence of a negative PET scan. A retrospective study found that only 0.6% (2/352) of patients with presumed PCNSL with negative PET scans had bone marrow biopsy findings that impacted management.A limitation of the current landscape of PDHL case reports is the lack of long‐term follow‐up data, with a median follow‐up of 12 months  post‐adjuvant therapy after completion of chemotherapy or radiation. (Table 4In summary, we describe a case of primary HL of the intracranial dura. To our knowledge, there has only been one previous case reported in the literature. Due to its rarity, the natural history of the disease and optimal treatment strategies are not well understood. This case of PDHL with 5 years of follow‐up demonstrated a favorable outcome after partial surgical resection followed by ABVD chemotherapy and ISRT. More extensive experience with PDHL is needed to provide further insight into the prognosis and optimal management of this rare condition.Tharsan Kanagalingam: Investigation; writing – original draft; writing – review and editing. Vikram Velker: Conceptualization; funding acquisition; investigation; supervision; writing – review and editing. Shervin Pejhan: Writing – original draft. Qi Zhang: Supervision. Joy Mangel: Supervision; writing – review and editing. Sympascho Young: Supervision; writing – original draft; writing – review and editing.None.


Acromegaly caused by ectopic growth hormone-releasing hormone (GHRH)-secreting tumor is exceedingly rare. We report a case of acromegaly secondary to GHRH secretion by an incidentally diagnosed pulmonary neuroendocrine tumor (NET) and review 47 similar cases in literature. A 22-year-old male patient presented with symptoms of pituitary apoplexy. Magnetic resonance imaging (MRI) showed apoplexy of a pituitary adenoma. Routinely prior to surgery, a chest radiography was performed which revealed a mass in the left lung. During investigation, the patient was diagnosed with metastatic GHRH-secreting pulmonary NET. In retrospect, it was noted that the patient had pituitary hyperplasia 20 months prior to the MRI which showed the presence of a pituitary adenoma. The histological findings confirmed somatotroph hyperplasia adjacent to somatotropinoma. This case suggests that GHRH secretion can be associated with pituitary hyperplasia, which may be followed by pituitary adenoma formation. Acromegaly is a chronic disease due to excess growth hormone (GH) and insulin-like growth factor type I (IGF-I)   . GH-secThe clinical features of ectopic acromegaly are indistinguishable from those resulting from somatotropinoma   . HoweveWe report a case of a male patient who was diagnosed with acromegaly due to GHRH-secreting pulmonary NET and somatotropinoma, who presented with carcinoid syndrome, and review cases of GHRH-secreting pulmonary NETs published in literature.Instituto Estadual do Cérebro Paulo Niemeyer Ethics Committees involved in the study (CAAE 39632820.8.0000.8110). Patient's written consent was obtained for publication.This study was conducted in accordance with the ethical standards of the Helsinki Declaration, with the approval of the A 22-year-old male patient presented with sudden and severe headache after dental extraction. Brain computed tomography (CT) showed an isodense pituitary lesion, with no contrast enhancement. No visual disturbance was present. Sellar region magnetic resonance imaging (MRI) performed nine days after the headache revealed a 3.3 × 2.4 × 1.4 cm lesion suggestive of pituitary apoplexy (PA)   . The paOf note, the patient had a brain MRI performed 20 months before due to mental confusion and dizziness that showed slight enlargement of pituitary gland of 9.31 mm    . The paOn biochemical assessment, the patient had increased IGF-I levels of 823 ng/mL  and basal GH levels of 2.05 ng/mL, panhypopituitarism . Diluted prolactin was 2.20 ng/mL (2.00-15.20). The patient was diagnosed with acromegaly and submitted to transsphenoidal surgery with complete removal of the pituitary lesion. The post-operative course was uneventful.18Fluor-fluorodeoxyglucose positron emission tomography (18F-FDG PET) CT scan and 68Gallium DOTA-Tyr3octreotide (68Ga-DOTATOC) PET CT revealed discrete increase in glycolytic metabolism and significant increase of somatostatin receptor expression in the ILL, left lobe hilum, hepatic hypovascular lesions, spine, sacrum and pelvis, consistent with a well differentiated metastatic NET . At baseline, chromogranin A level was 17,906 ng/mL , serotonin 806.8 mcg/L (30-200) and urinary 5-hydroxyindolacetic acid 19.3 mg/24h (2.0-9.0). Serum GHRH concentration was not available for measurement.On pre-operative evaluation, a routine chest X-ray was performed that showed a 4 cm central lesion in inferior left pulmonary lobe (ILL). CT scan demonstrated a 4.5 × 4.0 × 3.5 cm lesion in ILL. tic NET   . A bronPathological examination included histological and immunohistochemical studies. For light microscopy, 3 μm sections of formalin-fixed and paraffin embedded tissue (FFPE) were stained with hematoxylin-eosin and reticulin. Avidin-biotin complex techniques were used to demonstrate the presence of pituitary hormones as described previously   . AntiseGHRH mRNA in the pulmonary NET (5053 mRNA copies) compared to six normal pulmonary tissues ( beta-actin ( ACTB ), hypoxanthine guanine phosphoribosyl transferase ( HPRT ) and glyceraldehyde 3-phosphate dehydrogenase ( GAPDH )] were also measured being ACTB the most stable control gene; therefore, ACTB was used to control for variations in the amount of RNA used in the RT reaction and to adjust the relative expression of each of the transcripts analyzed. GH was not expressed in the sample. The specific primers used in qPCR are detailed in RNA from the FFPE tissue was extracted from the pulmonary specimen, at a concentration of 13.6 ng/uL. Reverse transcription (RT) and quantitative polymerase chain reaction (qPCR) were performed as previously reported   , showintissues   . Three 177Lu dose every 6 weeks. After four cycles of PRRT, 18F-FDG PET scan revealed complete metabolic resolution of the lesions and reduction of the primary pulmonary NET in about 22% in axial view. Pegvisomant was titrated to maintain IGF-I levels in normal range. Nowadays the patient is on pegvisomant 40 mg/week and octreotide LAR 30 mg/month. After one year of therapy, normalization of serum IGF-I to 197 ng/mL (99-289) and urine 5-hydroyindoleacetic acid to 8.9 mg/24h (2.0-9.0) were observed. Serum chromogranin A and serotonin levels decreased significantly but did not reach normal values . Brain natriuretic peptide levels and echocardiography are normal.Levothyroxine, prednisone and testosterone were prescribed for hypopituitarism. Pegvisomant 70 mg/week and octreotide LAR 20 mg/month were started after ectopic acromegaly secondary to GHRH-producing metastatic tumor was diagnosed, while waiting for peptide receptor radionuclide therapy (PRRT). Octreotide LAR was titrated to 30mg and was given after MEN1 and cyclin dependent kinase inhibitor 1B (CDKN1B) sequencing were negative for pathogenic mutations.Multiple endocrine neoplasia type 1 (MEN1) and 4 (MEN4) were investigated. Peripheral blood samples were collected with EDTA as anticoagulant and DNA was extracted from leukocytes. Somatic mutations were not analyzed due to paucity of tumor samples. Gentra PureGene Blood Kit  was used to obtain genomic DNA. The eluted was resuspended in 100 μL of DNA Hydration Solution following the manufacturer's instructions. After extraction, PCR reactions were performed on Applied Biosystem ProFlex™ PCR System . Amplification and sequencing were performed with PCR/Sanger Sequencing Primer pairs . Products were sequenced in both directions on ABI 3130xl Genetic Analyzer (Applied Biosystems) and electropherogram-derived sequences were aligned by using Benchiling and BioEdit software   . GermliThe patient was initially diagnosed with pituitary adenoma (PA) and afterwards with pulmonary mass which turned out to be a GHRH-secreting pulmonary NET associated with somatotropinoma and pituitary hyperplasia. Because of extensive metastasis, primary pulmonary tumor was not resected and pegvisomant and octreotide LAR were started early, followed by PRRT. Compared to literature, our case is the only case in which PA and pituitary hyperplasia were observed in pathology, and pituitary hyperplasia was seen on MRI scans before pituitary adenoma formation.To our knowledge, only 47 cases have been described of GHRH-secreting pulmonary NET causing acromegaly  on chest CT, like our case, and were more common on the right side [19 of 28 cases (67.8%) in which side was described], which was not our case. NETs were diagnosed by histology with the presence of positive immunostain for chromogranin and GHRH in the pulmonary specimen as found in our case, or suspected with positive functional imaging with increased serum GHRH. All patients except for three were submitted to pulmonary resection, even in the presence of metastatic lesions   , 12 . DIn line with our case, 14 (33.3%) patients already had distant metastases at diagnosis. Most common sites were liver and/or bone present in 12 (75%) of these metastatic cases, similar to our case. Other sites included lymph nodes, breast, ovaries, heart, thyroid gland and even one case of metastasis to pituitary gland   .As NET can secrete bioactive amines such as serotonin, patients can present with carcinoid syndrome. This syndrome was uncommon in pulmonary NET (8% of patients) because of lower production of serotonin compared to other NET, but can be more frequently encountered in patients with liver metastases (15% of patients), as was our case   . A delaMEN1 mutations have been described in several different endocrine tumors, with 26 cases being of pulmonary NET . Patients had signs and symptoms of acromegaly  for median period of 8.5 years   - 26  prnull cell adenoma) . Eight (20%) cases were suspected of having pituitary tumor, but only five cases confirmed pituitary adenoma at histopathology examination (four somatotropinoma and one denoma)   , 23 . Adenoma)   . In othPituitary adenoma and hyperplasia can coexist as we have seen in our case. In some reports, adenomatous transformation from pituitary hyperplasia have been described   . In theBiermasz and cols.    describSimilarly, in gangliocytoma , somatotropinomas could be concomitantly seen   , 27 . MIn humans, a causative association between GHRH hyperstimulation and pituitary somatotroph adenoma formation has not been demonstrated. Theoretically, there may be neoplastic transformation preceded by pituitary hyperplasia. Ectopic GHRH excess usually results in pituitary hyperplasia alone    that haAIP mutation had transition zones from hyperplasia to adenoma. Our case, however, did not show a transition zone from hyperplasia to adenoma in the pathological analysis, rather it showed pituitary adenoma adjacent to pituitary hyperplasia.Interestingly, other cases of somatotropinomas can also be associated with pituitary hyperplasia, as Villa and cols.    reporteRegarding treatment, for acromegaly, pegvisomant is the drug with the highest efficacy and therefore was prescribed from the beginning for optimal and rapid IGF-I lowering   , 38 . FIn conclusion, very few cases of acromegaly secondary to GHRH-secreting pulmonary NET have been described, and even fewer of associated somatotropinoma. Despite its rarity, pituitary adenoma may coexist with pituitary hyperplasia in acromegaly due to ectopic origin. In the setting of acromegaly, ectopic GHRH or GH secretion must be thought of when pituitary scan shows pituitary hyperplasia or does not reveal a lesion suspicious of adenoma. Although generalizations cannot be made by case reports, we speculate, in our case, that the GHRH-producing pulmonary NET might have led to the development of pituitary hyperplasia and posteriorly to somatotropinoma formation that was manifested as PA.


Abdominal contouring procedures are frequently performed following massive weight loss after a Roux-en-Y gastric bypass. This last procedure can be associated with late complications like the development of internal hernias . The purpose of this article is to present a case of Petersen hernia after complete abdominoplasty.A 42-year-old female presented to the emergency department with acute abdominal pain three days after undergoing a complete abdominoplasty following massive weight loss post laparoscopic gastric bypass (Roux-en-Y). The patient was diagnosed with a mechanical small bowel obstruction, likely due to an internal hernia (Petersen hernia) and underwent surgical correction.Gastric bypass surgery may be associated with small bowel obstruction from internal herniation. A sudden rise in abdominal pressure, as occurs with abdominoplasty with rectus plication and subsequent use of compression garments, may result in strangulation and intestinal ischemia. A high degree of suspicion of intra-abdominal complications after abdominoplasty is essential, particularly in the setting of post-bariatric surgery. Abdominoplasty is one of the most common aesthetic procedures performed worldwide . AbdominThe purpose of this article is to present a rare case of intra-abdominal complications after abdominoplasty related to previous laparoscopic gastric bypass (Roux-en-Y) - Petersen internal hernia. The symptoms and signs, management, and the possible etiology of this disorder are discussed.A 42-year-old female presented to the emergency department with intermittent diffuse abdominal pain with radiation to the left flank, nausea, and vomiting within one day of evolution. No genitourinary symptoms were reported. No bowel movements were reported in the previous four days. Three days before, the patient underwent an abdominoplasty with rectus plication and the procedure was uneventful. She has a history of laparoscopic gastric bypass (Roux-en-Y), 2,5 years before, after which the patient complained of chronic abdominal discomfort related to food intake. Blood tests showed normal hemoglobin, without leukocytosis, with mild elevation of C-reactive protein . CT Scan showed gastric, duodenum, and jejunal distension; with parietal thickening, wall heterogeneity, and mucosal hypercaptation in the proximal jejunal loop; a stop point in the proximal jejunum and rotation of mesenteric vessels, suggesting an internal hernia Figure .A diagnosis of mechanical small bowel obstruction, likely due to an internal hernia (Petersen hernia) was made. The patient underwent a median laparotomy. Lysis of adhesions and mobilization of the great epiploon was performed. Retrieval of the proximal intestinal loops from the Petersen space was accomplished. About 30 cm of necrotic proximal jejunum in the biliopancreatic limb was removed and a lateral-lateral jejunojejunal anastomosis was carried out. Subsequently, the Petersen space was closed with Vicryl 3/0 and the abdominal wall closed as usual. She underwent a course of antibiotherapy (ciprofloxacin and metronidazol) and was discharged five days later with a resolution of the symptoms. A subsequent CT scan showed no recurrence of the internal hernia.The Petersen internal hernia Figure  used to Patients with a history of gastric bypass surgery have a high incidence of small bowel obstruction from internal herniation, which is associated with non-specific upper abdominal pain, nausea, and anorexia . The facAbdominal pain in the setting of an early post-operative course of abdominoplasty may have a broad differential diagnosis. The most evident cause may be the pain associated with abdominoplasty by itself. When it is associated with obstipation, a postoperative ileus may also be a frequent cause. If abdominal guarding, rigidity or rebound tenderness, intra-abdominal complications must be sought like a stitch through the bowel, internal hernias, adhesions, and volulus. Even common inflammatory causes of acute abdomen may be coincidently present like appendicitis or cholecystitis. An adequate evaluation by an experienced surgeon is essential to diagnose and treat these difficult cases.Although a rare case is described in this report, a high degree of suspicion of possible intra-abdominal complications after abdominoplasty is essential, particularly in the setting of post-bariatric surgery. The plastic surgeon should be aware of this gastric bypass complication when performing body contouring procedures.


A rare case of an unexpected iatrogenic splenic rupture during a cesarean section is reported. The trauma was recognized early and treated conservatively without delay; thus, further complications were avoided. A 28-year-old woman with a history of previous cesarean sections was submitted for an elective cesarean section. Intra-operatively, minor bleeding from the left abdomen was noted and eventually assigned to an inferior pole splenic trauma treated conservatively without splenectomy. Although unclear, the injury was considered iatrogenic, probably due to the abdominal pressure for fetal delivery and the anatomy of the splenocolic ligament. This case highlights the clinical suspicion that is required despite routine surgical procedures for the early diagnosis of an unexpected splenic rupture when minor bleeding occurs intra-operatively from the upper abdomen. Early recognition and prompt treatment are of paramount importance for the safety of the fetus and the patient. In general, hemorrhage in pregnancy is the leading cause of maternal and fetal morbidity and mortality worldwide, with medical, obstetrical, and surgical etiologies. Some authors consider that pregnancy per se is a risk factor for splenic rupture, explained by pregnancies’ hypervolemia and alterations of the abdominal organs . This poSplenic rupture is labeled as spontaneous when it is not associated with antecedent trauma, systemic disease, or gross pathology and the parenchyma, the vasculature, and the capsule are normal . RuptureIatrogenic splenic rupture injury is defined as any involuntary damage caused to the spleen during an operation or a medical intervention . IntraopA 28-year-old woman, gravida 3, para 1, was admitted for an elective cesarean section at 38+1 weeks of gestation. Her obstetrical history included a previous cesarean section and a pregnancy termination due to a fetal cardiac abnormality. The medical history was insignificant, without splenomegaly and a normal body mass index (24.4 kg/m2). Under general anesthesia, a Pfannenstiel incision was performed. After a low transverse uterine incision and rupture of the membranes, abnormal fetal presentation was noted, and standard maneuvers to establish vertex presentation followed. A healthy female infant weighing 3100gr was delivered on time with Apgar scores of 8 and 10 in the 1st and 5th minutes, respectively. Routine closure of the uterine wall was accomplished with minimal blood loss. Intra-operatively, during the final control of the adnexas, the appendix, and the abdominal cavity, barely underlined but continuous minor bleeding coming from the upper left abdomen was ascertained. The general surgeon was called, and after evaluation, he approached the left upper quadrant with a Kocher incision, and the bleeding site was revealed. A splenic trauma had occurred at the inferior pole of the spleen, at the point where the splenocolic ligament is attached, which was noted to be extremely thin. The conservative technique of packing, topical surgical hemostatic agents, and non-absorbable sutures was enough to control the bleeding, and splenectomy was prevented. The estimated blood loss from the splenic injury was approximately 80 ml. The patient remained hemodynamically stable during surgery, and the postoperative period was uneventful.No pathological findings were indicated by the computed tomography (CT) scan after the operation Figure , 2.The mechanism of rupture was discussed but remained unclear, attributed most probably to the abdominal pressure for fetal delivery and the anatomy of the splenocolic ligament. The patient was discharged on the fourth day in good condition, and follow-up after forty days was normal. The laboratory results concerning hemodynamic stability before and after the operation are listed in Table If the spleen suffers an unintentional injury or rupture in an elective or urgent operation or medical intervention, it is defined as an “iatrogenic splenic rupture". The exact incidence of iatrogenic splenic trauma is hard to define since many cases of intraoperative complications that are treated conservatively are not finally described. This leads to an underestimation of this medical condition, which constitutes an indication for up to 40% of all splenectomies .Overtraction of the ligaments attached to the spleen  is the main mechanism of splenic rupture. Anatomical variations of the splenic hilum and the splenic artery branches may also contribute to incidental injury. Another cause could be the mobilization of the left colonic flexure and the traction at any portion of the greater omentum due to its close attachments with the splenic capsule. A previous medical history of surgical procedures may contribute to the presence of adhesions and constitute an independent risk factor for injury .The physiological changes following pregnancy may predispose to splenic injury. At first, increased circulation of estrogen and progesterone may increase the size of the spleen and splenic vasculature. The spleen position may also alter or lead to increased mobility. In general, splenic rupture occurs more often in women with advanced maternal age, in multiparous women, in multiple gestations, and in the third trimester of pregnancy, although cases in the peripartum and postpartum periods have also been reported. In addition, previous abdominal operations  could be present ,2.Spontaneous peripartum splenic rupture has been reported in patients with hemolysis syndrome, elevated liver enzymes, a low platelet count, preeclampsia, and splenic ectopic pregnancy. In the same period, increased circulation of blood volume or administration of fluid and blood products could engorge the spleen, making the organ more susceptible to injury. The most frequent clinical symptom is pain on the left side spreading up to the left shoulder. Interestingly, spontaneous rupture is similar in vaginal and cesarean deliveries ,3.Splenic rupture occurring during a cesarean section is quite rare. Trauma could occur from the abdominal incision, the insertion of packs, maneuvers for the neonatal expulsion associated with forceful pulsing on the upper abdomen and the uterine fundus, the use of retractors and sharp instruments, or even while removing clots from the paracolic gutters ,3. If thHowever, Sakhel et al.  reportedPhysical examination and laboratory tests are crucial in establishing a diagnosis. Abdominal ultrasound is useful in the quick diagnosis of intraperitoneal fluid loss or splenic hematoma and could be performed in the emergency unit or the theater. Additionally, a CT scan could also be used .Splenic rupture is most common in the peripartum and postpartum periods and appears to be difficult to diagnose. Common entities like postpartum pain after a cesarean section, intra-abdominal uterine bleeding, uterine rupture, or injury of the viscus present with similar findings. If shock is established, the differential diagnosis could be amniotic fluid embolism, pulmonary embolism, cardiogenic shock, intravascular coagulopathy, or septic shock .The management of splenic rupture may be conservative in milder lesions  or aggressive, ending in total splenectomy  ,11.Splenic artery angiography followed by embolization has also been described with a success rate of 85%; however, its role in the final management is unclear . The theIntraoperative splenic rupture during gynecological or obstetrical procedures is extremely rare but may cause significant morbidity. Bladder, ureteral, and gastrointestinal injuries are more commonly described. The need to extend the length of the initial Pfannenstiel incision is always possible when a difficult cesarean section is performed. In cases of known fetal malpresentation, vacuum-assisted delivery could be performed to avoid excessive abdominal pressure, and the obstetrician should not hesitate to call the general surgeon when necessary . FurtherThis is believed to be the first report of iatrogenic splenic rupture during a cesarean section with maintenance of the spleen and a favorable outcome. This case highlights the clinical suspicion required when unexpected minor but continuous bleeding originating from the upper abdomen occurs, and this hemorrhage must not be ignored. Our patient recovered very well, and splenectomy was avoided because the bleeding was recognized promptly, she remained hemodynamically stable, and the injury was small.


Bouveret’s syndrome is a rare form of gallstone ileus described as a gastric outlet obstruction from a gallstone that travels from the gallbladder to the bowel through a bilioenteric fistula. Despite its rarity, the mortality rate of this condition is high. Endoscopic treatment is preferred over surgery due to the association with lower mortality rate. To date, there are limited data about the application of holmium:yttrium-aluminum-garnet (YAG) laser lithotripsy for fragmentation of gallstones in Bouveret's syndrome. We present the case of a 74-year-old man with multiple cardiac comorbidities who presented with periumbilical pain, decreased appetite, and vomiting. The patient had previously been admitted three months prior with acute cholecystitis, and a cholecystostomy tube was placed. He had leukocytosis and purulent discharge in his cholecystostomy bag. Computed tomography (CT) scan of the abdomen and pelvis showed a change in the position of a previously seen large gallstone from the neck of the gallbladder on the last admission, to the lumen of the duodenal bulb on this admission. This indicated the development of a cholecystoduodenal fistula, with the stone passing through this fistula into the duodenal bulb, causing the complete obstruction. Endoscopic treatment was recommended by the surgery team due to cardiac comorbidities and the significant friability of the tissue requiring laparotomy. Initial endoscopic evaluation showed complete obstruction of the duodenal bulb by a large smooth stone, not allowing passage of a guidewire beyond the stone. Therefore, holmium:YAG laser lithotripsy was used. After two sessions of laser therapy, four days apart, each breaking a pigmented and calcified stone, it eventually passed through the small bowel into the colon, relieving the obstruction. The patient had a favorable outcome and did not require surgery. This case report shows that holmium:YAG laser lithotripsy is capable of delivering favorable outcomes, as seen in a patient with a heavily calcified and pigmented stone, older age, and multiple comorbidities. Holmium:YAG laser could be considered for use with endoscopic equipment for future management of this condition, especially in patients who have medical comorbidities and heavily calcified gallstones. About 10% of the general adult population in the United States has gallstone disease. Of these, 20-30% develop biliary pain or other complications from gallstones such as cholecystitis, cholangitis, pancreatitis, and gallstone ileus. Gallstone ileus is a condition when gallstones dislodge from the biliary system into the bowel lumen and cause bowel obstruction [This article was previously presented as a meeting abstract at the American College of Gastroenterology (ACG) 2022 conference on October 24, 2022.A 74-year-old male patient presented to the emergency department with two episodes of vomiting and one day of decreased appetite. He also reported periumbilical pain and nausea but no heartburn, fever, chills, or jaundice. His past medical history was significant for multiple cardiac comorbidities, including cardiomyopathy (left ventricular ejection fraction of 35%), atrial fibrillation, and ventricular tachycardia. He had an automatic implantable cardioverter defibrillator in place.He had been admitted to our hospital three months prior with acute cholecystitis and elevated liver enzymes. Due to his cardiac comorbidities, he was treated with intravenous (IV) piperacillin and tazobactam 4.5 gm every six hours for five days, cholecystostomy tube, and endoscopic retrograde cholangiopancreatography (ERCP) with sphincterotomy. The liver enzymes improved, and he was discharged. Six weeks after discharge, a tube check had confirmed the correct placement of the tube and decompression of the gallbladder.During this presentation, his pain was different from the right upper abdominal pain of his prior admission. Physical examination on this admission revealed epigastric and periumbilical tenderness with no guarding or rebound tenderness. His cholecystostomy bag had purulent drainage. His tube drainage had reduced considerably in the previous day.Laboratory results were significant for leukocytosis. He had normal liver enzymes and serum lipase. Computed tomography (CT) scan of the abdomen and pelvis showed a change in the position of a previously seen large 3.3 cm gallstone. During the previous admission, it was in the neck of the gall bladder. At this time, it was occupying the lumen of the duodenal bulb. This indicated the development of a cholecystoduodenal fistula, with the stone passing through this fistula into the duodenal bulb, causing complete gastric outlet obstruction Figure . The patThe initial endoscopic evaluation showed complete obstruction of the duodenal bulb by a large smooth stone, not allowing passage of even a guidewire beyond the stone Figure . FollowiThe level of obstruction in gallstone ileus is most commonly at the terminal ileum (60-70%), which is the narrowest part of the bowel, followed by the proximal ileum (25%), and then other parts of the small bowel. In rare cases of gallstone ileus 1-3%), the level of stone impaction can be located more proximally in the duodenum and cause gastric outlet obstruction. This rare condition is called Bouveret’s syndrome %, the le. In our The prevalence of this disease is highest in elderly females with a median age of 74 and a female-to-male ratio of 1.9 . The diaHistory and physical examination alone are not adequate to make a diagnosis of Bouveret’s syndrome. Moreover, laboratory results are mostly non-specific. Imaging modalities such as abdominal plain film, ultrasound, and CT help identify the locations of stones and even the locations of fistulas. The most common initial imaging in the elderly who present with symptoms of bowel obstruction is the abdominal plain film. However, abdominal plain films are diagnostic in only 21% of Bouveret syndrome cases. Rigler’s triad, consisting of small bowel obstruction, pneumobilia, and ectopic gallstone, is a pathognomonic sign of gallstone ileus. However, this triad is detected in only 30-35% of patients by plain films. Hence, the abdominal plain film alone may not be enough to reach the diagnosis. An ultrasound of the abdomen may be useful if it shows ectopic gallstones and pneumobilia. However, ultrasound depends on the radiologist's skills and may not reveal the exact location of the stones. Moreover, pain on examination and excessive gas in the abdominal cavity are one of the limitations. CT abdomen is required in most cases and provides the best sensitivity (93%), specificity (100%), and accuracy (99%). CT abdomen can detect Rigler’s triad in up to 78% of cases [Even though the success rate of endoscopic treatment is less than 10%, endoscopy should be considered before surgery . This isOne of the techniques to crush gallstones into small fragments is mechanical lithotripsy. This is accomplished by using baskets, forceps, lithotripters, or snares to crush the stones into fragments, followed by endoscopic removal of stone fragments . In caseEven though there are multiple endoscopic modalities available, about 91% of the patients still have to undergo surgery . EndoscoIn summary, we presented the case of an elderly man with Bouveret’s syndrome who had experienced a favorable outcome after endoscopic treatment alone using holmium:YAG laser for gallstone fragmentation. Although holmium:YAG laser lithotripsy is not a common endoscopic modality in gastroenterology, this case report shows that holmium:YAG laser is a promising tool for Bouveret’s syndrome. Holmium:YAG laser lithotripsy is capable of delivering a favorable outcome in a complicated patient, as seen in this present case with a heavily calcified and pigmented stone, older age, and multiple comorbidities. Holmium:YAG laser lithotripsy could be considered for use with endoscopic equipment for the future management of this condition in patients who have a high risk for surgery and heavily calcifies gallstones.