Incarcerated gravid uterus (IGU) is a rare condition that occurs when a retropositioned gravid uterus becomes entrapped within the pelvic cavity. Most patients present around the 17th week of pregnancy with symptoms such as pelvic fullness, urinary incontinence, abdominal pain, constipation, and vaginal bleeding. Rarely, patients are asymptomatic throughout pregnancy, leaving IGU undiagnosed and untreated. Here, we present an asymptomatic 26-year-old female who presented at 30 weeks of gestation with severe intrauterine growth retardation (IUGR) on serial obstetric ultrasounds. Further evaluation with ultrasound and MRI revealed an incarcerated uterus. This was complicated by severe fetal IUGR, abnormal biophysical profile, and oligohydramnios. This case highlights the importance of early diagnosis and treatment of IGU in order to prevent complications associated with the condition. Clinicians should be aware that, although uncommon, patients with IGU may be asymptomatic and that diagnosis should depend primarily on imaging findings rather than symptoms. Incarcerated gravid uterus (IGU) is a rare potential obstetric emergency that occurs when a retropositioned uterus becomes entrapped in the pelvis during pregnancy. A retropositioned uterus is considered a normal variant in the first trimester; about 15% of females have a retroflexed uterus before pregnancy, and 11% of females develop a retroflexed uterus during the first trimester of pregnancy . PatientA 26-year-old G2 P0010 (at time of presentation) female with an estimated gestational age (EGA) of 30 weeks presented with IUGR and oligohydramnios detected on serial obstetric ultrasounds. The patient has a history of a known retroverted uterus and a prior spontaneous abortion at 10 weeks of gestation. A transabdominal pelvic ultrasound in the first trimester showed the retroverted uterus and the intrauterine fetus  MRI of the abdomen and pelvis without IV contrast was performed. The uterus was significantly retropositioned, with the inverted fundus entrapped in the pelvis and embedded in the sacrum below the promontory. The vagina and cervix were markedly elongated, with the stretched cervix coursing anteriorly and to the left of the uterus. The cervical internal os was proximal to the urinary bladder, which was compressed anterior to the uterus. The fetal head was in the fundus in a complete breech presentation. The findings were compatible with an incarcerated gravid uterus Figures -6B.The patient subsequently underwent a targeted growth ultrasound that included a fetal biophysical profile with a non-stress test (NST) and an umbilical artery Doppler. The umbilical artery systolic velocity/diastolic velocity ratio (SDR), pulsatility index (PI), and resistive index (RI) were all elevated at 94%, 91%, and 86%, respectively Figure . The amnThe clinical decision was made to perform an unscheduled, non-urgent cesarean section for nonreassuring fetal status in the setting of a known incarcerated uterus. The patient received two doses of betamethasone injections prior to delivery. A cesarean section was performed at an EGA of 31 weeks and one day under spinal anesthesia. To avoid injuring the distorted bladder, cervix, and vagina, a vertical midline incision was made. Through electrocautery and stretching, the fascia was incised in the midline and extended cephalocaudally. The rectus muscle was divided bluntly in the midline. The peritoneum was entered bluntly, taking care to avoid injuring the viscera. The uterus was grasped, and the fundus was gently lifted from the pelvis and reoriented into its normal anatomical position. A classical uterine incision was performed. The bladder blade was inserted. The premature infant was delivered atraumatically and was handed off to an awaiting neonatal intensive care unit (NICU) nurse. The birth weight was 2 lbs, 14 oz. The newborn complications included respiratory distress and NICU admission. There were no immediate postoperative maternal complications. Both the mother and the neonate were later discharged from the hospital in satisfactory condition. The patient was advised to have a preconception consultation with maternal-fetal medicine to manage any future pregnancies. Due to her classical cesarean section, she would require a cesarean delivery for all future pregnancies as a vaginal birth would increase the risk of uterine rupture. The cesarean section would likely be planned for 36-37 weeks of gestation.IGU is a rare condition that occurs when the gravid uterus is entrapped in the pelvic cavity between the sacral promontory and pubic symphysis past 16 weeks of gestation. Risk factors for the condition include a retroverted or retroflexed uterus, endometriosis, leiomyomas, uterine anomalies, pelvic adhesions, previous abdominopelvic surgery, a deep sacral concavity with an overlying sacral promontory, and an incarcerated uterus in a previous pregnancy .Most patients with IGU present in the second trimester with symptoms due to compression of structures around the incarcerated uterus, such as urinary manifestations, abdominal pain, constipation, and vaginal bleeding. However, IGU may rarely be asymptomatic and thus may not be detected during regular prenatal visits due to a lack of suspicion. In such cases, IGU may not be diagnosed until the third trimester, when further evaluation is performed due to the presence of fetal complications such as IUGR, which was the case for our patient. Thus, early diagnosis should be based primarily on imaging findings rather than clinical symptoms. On transvaginal ultrasound, the cervix is elongated, anteriorly displaced, and stretches upward, superior to the bladder and pubic symphysis. This may make the cervix difficult to identify on ultrasound. Thus, difficulty identifying the cervix on ultrasound, especially in patients with predisposing factors, should raise suspicion for IGU and prompt further investigation. Of note, due to the abnormal configuration of the cervix, it may be confused as an empty uterus on ultrasound; this may result in misdiagnosing IGU as an ectopic pregnancy. A retroverted or retroflexed uterus may also be visualized. Importantly, an MRI is essential to confirm the diagnoses as it provides excellent visualization of the anatomic relationships in the pelvis, which is necessary for diagnosis. The uterus will be retropositioned, with the fundus embedded in the hollow of the sacrum. The cervix will be elongated and displaced anteriorly relative to the uterus. The bladder will be displaced superior to the pubic symphysis and compressed anterior to the uterus. Pelvic exam findings are also useful for diagnosis. On a pelvic exam, the cervix is difficult to expose due to its anterior and superior displacement against the pubic symphysis. Sacculation of the posterior wall of the vagina and the posterior fornix bulge may also be observed. The uterine fundus may be immobile and palpated within the curvature of the sacrum.The complications of IGU can be devastating. Serious maternal complications include bladder rupture, renal failure, uterine ischemia, uterine rupture, premature labor and delivery, and injury of the bladder, cervix, and vagina during delivery. Serious fetal complications include miscarriage in the first trimester, fetal death, and IUGR .The initial management of IGU includes attempting to reduce the incarcerated uterus through urinary bladder drainage, followed by chest-knee maneuvers. If this fails, manual uterine reduction may be attempted. Manual reduction poses serious risks, such as placental abruption, rupture of membranes, intrauterine fetal death, and preterm delivery. Manual reduction is only recommended before 20 weeks of gestation, as the risk of preterm birth is higher when performed beyond this point. In cases where patients are diagnosed past 20 weeks of gestation or if manual reduction fails, careful follow-up until a planned cesarean section is the preferred route of management. A cesarean section is necessary because the incarcerated uterus may impair the descent of the fetus. A vertical supraumbilical approach should be taken in order to avoid injuring the bladder, cervix, and vagina . At any Of note, there is a lack of general consensus regarding the optimal management of IGU due to the rarity of the condition. Considering the reasonable probability that IGU may resolve spontaneously and that manual reduction poses a risk of serious complications, some authors question if manual reduction is justified over expectant management, especially in patients without severe symptoms due to incarceration . In our Despite being exceedingly rare, it is important for clinicians to be aware of the possibility that patients with IGU will remain asymptomatic throughout pregnancy. Thus, imaging and pelvic exam findings should be relied on for early diagnoses of the condition. This allows for timely treatment and prevention of the potentially devastating complications of IGU.


Fibroadenoma (FA) in ectopic male breast tissue is extremely rare. The ectopic breast tissue (EBT) is mostly appearing along the milk line; however, uncommon locations are also seen, such as the present case.The authors report a 19-year-old male presenting with intestinal obstruction. The patient underwent laparoscopic surgery, and an excisional biopsy of the lesion was done. The histopathologic result confirms FA arising from EBT. This case is reported for its rarity. It indicates that FA should always be considered when there is an intra-abdominal suspicious mass.EBT presenting as FA is also reported in the face, posterior neck, chest, middle back, buttocks, vulvar, and thighs. In this case, the authors present a EBT presented as a FA in the intra-abdomen of a young male patient, causing intestinal obstruction. FA in the male breast is rare; however, benign breast parenchyma showing FA in the intra-abdomen of a male patient is extremely rare.When a tumor is palpated in the milk line, the existence of FA should be taken into consideration. FA of the male EBT in the intra-abdomen is extremely rare. However, a close follow-up of the patient is strongly recommended, as the carcinoma arising from FA has a very poor prognosis. HighlightsEctopic breast tissue (EBT) presenting as fibroadenoma (FA) is a very rare entity; it may be milk like.This case presents EBT presenting as FA in an adult male, which is extremely rare. Clinically patient presented with bowel obstruction and associated lower abdominal pain.The ultrasonography reported a mass lesion in the intra-abdomen. Thus, a biopsy is taken from the mass lesion that was diagnosed as FA.It should be taken into consideration that FA can be raised from EBT in a male patient in the milk line.Polymastia is a term for two or more than two breasts or ectopic breast tissue (EBT), which is a developmental anomaly and a rare entityA 19-year-old male referred to Al-Hayat Hospital presented with lower abdominal pain and discomfort for a year with repeated bowel obstruction reported in clinical data; clinically suspicious for tuberculosis and mesenteric lymphadenopathy. On physical examination, there was lower abdominal tenderness. The ultrasonography is reports descending testis, right side hydrocele, ascites, mild splenomegaly, and a few enlarged para-aortic lymph nodes with thick gut loops like structure a computed tomography (CT) scan is suggested. Meanwhile, the peritoneal tab is done and the fluid is sent to the Department of Cytopathology Human Medical Laboratory. Cytology was reported as negative for malignancy and mixed acute and chronic inflammation only. A CT of the abdomen and pelvis with contrast is done; the CT scan suggests a subacute small bowel obstruction due to an underlying infective stricture in the distal ileal loops. The patient gone under laparoscopic surgery congenital band released and biopsy is taken for histopathology examination. Gross examination revealed three pale-brown tissue fragments, the largest of which measures 4×3.5×2.2 cm. The external surface was irregular and lobulated. On cut sectioning, it revealed a gray-white cut surface. No calcification or necrosis was noted. The microscopic examination showed benign parenchymal tissue of the breast that is well-defined, consisting of large, compact mammary glands surrounded by ductal epithelium and myoepithelial cells covered with hyperplastic and fibrotic stroma with no evidence of granuloma or malignancy. The histopathological report was ‘benign breast parenchyma showing fibroadenoma’. The patient was healthy with no evidence of local recurrence after 1 year of follow-up : NA.Dr Sayed Hamid Mousavi and Jawad Amini.Not commissioned, externally peer-reviewed.Zekrullah Baset, Cytopathologist, Human Medical Laboratories, Kabul, AfghanistanNA.


A gonadal tumor was diagnosed in the first months of life in a patient with genital ambiguity, a 45,X/46,XY karyotype, and mixed gonadal dysgenesis. Gonadal biopsies at the age of 3 months revealed dysgenetic testes and a gonadoblastoma on the right testis. Even though gonadal tumors are rare in childhood, this case indicates that prophylactic removal of dysgenetic gonads should be performed as early as possible, especially when the female sex is assigned to a patient with a Y-chromosome sequence. Disorders of sex development (DSD) are a group of congenital conditions in which the chromosomal, gonadal, or anatomical sex is atypical. These conditions include disorders of gonadal development, comprising various types of gonadal dysgenesis (GD) (sis (GD) .A GD may be either complete (CGD), with bilateral streak and female internal and external genitalia in XX or XY individuals; partial (PGD), characterized by genital ambiguity and two dysgenetic testes or a dysgenetic testis and a streak gonad in 46,XY subjects; or mixed (MGD), when a 45,X/46,XY karyotype or its variants are found in a patient whose gonadal and genital phenotype is similar to that of PGD .In MGD, the presence of a 45,X cell line is associated with features of Turner syndrome, including short stature, cardiac and renal malformations, and thyroid disease, among others; thus, MGD and PGD have different prognoses ,4.The presence of Y-chromosome sequences in patients with GD increases their risk of developing gonadal tumors ,6. In paIn patients with MGD reared as girls, bilateral gonadectomy should be performed in childhood or upon diagnosis because of the risk of malignancy and insufficient gonadal function. For those reared as boys, removal of streak gonads and strikingly dysgenetic testes should also be done early; testes with lesser degrees of dysgenesis may be preserved in the scrotum and closely followed up for early detection of tumor development -11.The aim of this report is to describe a patient with MGD who developed a gonadoblastoma within the first months of life. This case can add knowledge to support the decision about early prophylactic gonadectomy in this group of patients.A 43-day-old infant without sex assignment was referred to us due to genital ambiguity. This infant was born at term by cesarean section and without neonatal complications, with a birth weight of 2,580 g and a length of 45 cm. The infant – the only child of healthy, unrelated parents – also had a healthy, 17-month-old maternal half-brother.The physical examination revealed a healthy infant with normal growth and without dysmorphic features, except for those of the external genitalia. A 3.5-cm phallus with chordee and penoscrotal hypospadias was present, and both gonads were palpable in the inguinal region. The features of the external genitalia were compatible with Prader grade III/IV , and theThe infant's karyotype was 45,X[7]/46,XY[43] and, on pelvic ultrasound, uterus and vagina were visualized. Basal levels of gonadotropins , total testosterone (2.51 ng/mL), androstenedione (8.76 mg/dL), estradiol (22 pg/mL), and 17-hydroxyprogesterone (216.6 ng/dL) were normal for the reference values at minipuberty . Abdominal ultrasound showed no urinary tract malformations, and echocardiography was also normal.Bilateral inguinotomy and biopsies of both gonads were performed at the age of 3 months to evaluate the degree of GD or the presence of ovarian tissue. At the same time, laparoscopy – which is the gold standard for the evaluation of internal genitalia  – confirBased on the diagnosis of MGD, the anatomy of the internal and external genitalia, and the wish of the parents, the female gender was assigned to the infant, and bilateral gonadectomy was performed. The histological evaluation showed signs of bilateral testicular dysgenesis . The right gonad had two neoplastic lesions: a premalignant one , within a region of undifferentiated gonadal tissue, and a gonadoblastoma  and 3.When the infant was 12 months old, a feminizing genitoplasty was successfully performed. She is currently 4 years old, well-adapted to the female social gender, and has a normal growth and neurological development.The finding of a 45,X/46,XY karyotype in patients with genital ambiguity and a female internal genitalia opens up the possibility for two differential diagnoses: MGD or ovotesticular DSD . The absEven though in the past patients with bilateral testicular dysgenesis have been histologically classified as having PGD ,5, the cin situ in a 2-year-old boy with ambiguous genitalia, a 45,X/46,XY karyotype, and a dysgenetic right inguinal testis .In conclusion, this rare case with an early neoplastic transformation confirms the need to carry out prophylactic gonadectomy in MGD patients as early as possible, particularly when the female gender is assigned to the infant.


Spontaneous hemorrhage of gastro-omental hemangioma is a rare disease. The treatment strategy for this disease changes as it develops. In the acute stage, surgery is the first priority, among which laparoscopic treatment should be the most frequently considered option in large general hospitals. Due to the rarity of this disease, such cases have rarely been reported.We present the first report of two eldely cases with gastro-omental hemangioma with hemorrhage by laparoscopic treatment. Both cases were initially admitted with upper abdominal pain, and abdominal computed tomography (CT) scan revealed masses alongside the greater curvature of the stomach. Laparoscopic surgery was conducted immediately in both patients. The two cases recovered well after surgery, and no obvious abnormalities were observed in the follow-up period.Gastro-omental hemangioma rupture remains an uncommon cause of intraperitoneal hemorrhage. Timely diagnosis and surgery are paramount for treatment. Medical institutions with the correct technology and equipment should perform laparoscopic treatment to minimize surgical trauma and promote rapid recovery of patients with abdominal apoplexy. Spontaneous hemorrhage of gastro-omental hemangioma is not common, and it may occur without any recent history of abdominal trauma, surgery, anticoagulant therapy, or systemic vascular disease. Clinical diagnosis should be made with abdominal B-ultrasound, abdominal CT, abdominal puncture, and other auxiliary examinations. The treatment methods generally depend on the disease development and the patient's conditions. In the onset or chronic phase, clinical observation and medical treatment are routine options, while in the advanced or acute phase, surgical treatment is preferred , 2. ReceA 61-year-old female was admitted to our hospital on September 16, 2018 due to persistent dull pain in the middle and upper abdomen for > 2 months, with aggravation for 1 day. The pain was relieved after taking an oral anodyne (specific name and dosage unknown) prescribed by a local physician, and no further examination or treatment was given. She denied any history of hypertension, diabetes, hepatitis, tuberculosis, heart disease, abdominal trauma, or operation. Physical examination showed the patient had acute facial features and pallid eyelid conjunctiva, and her skin was clammy and less elastic, with a body temperature (T) of 36.5°C, pulse frequency (P) of 80 beats/min, respiration frequency (R) of 20 beats/min and blood pressure (BP) of 126/80 mmHg. Abdominal moving dullness was positive, and non-coagulated blood was extracted by diagnostic abdomen puncture.3 , red blood cell count (RBC) of 3,100/mm3 , platelet (PLT) of 130/mm3 , Hematocrit (HCT) of 0.283 , and a hemoglobin (HGB) concentration of 91 g/dl . No abnormality was found on coagulation function examination. The above results indicated that the patient had an intraperitoneal hemorrhage combined with moderate anemia.Routine blood examination revealed the following: white blood cell count (WBC) of 9,800/mmAbdominal enhanced computed tomography (CT) revealed an effusion in the abdomen and pelvis, suspicious blood accumulation around the liver and spleen, and a slightly patchy high-density shadow on the side of the great curvature of the gastric body, about 9.8 × 6.9 × 6.5 cm in size . EmergenAn 81-year-old male was admitted on December 13, 2020 due to upper abdominal pain for 20 h. His medical history revealed hypertension for approximately 10 years, with a maximum blood pressure of 180/110 mmHg. He was receiving Nifedipine to control blood pressure. The patient had no history of hepatitis, tuberculosis, heart disease, diabetes, trauma, surgery, or blood transfusion.3, RBC 3,200/mm3 , PLT 91/mm3, HCT 0.311 , HGB: 107 g/L , and a normal coagulation index. The above results confirmed that the patient had an intraperitoneal hemorrhage combined with moderate anemia.Physical examination indicated acute facial features, clammy and weakened elastic skin, with T 36.7°C, P 90 beats/min, R 25 beats/min and BP 138/90 mmHg. His abdomen was flat, and the middle and upper abdomen had a moderate tenderness and rebound pain, without muscle tension; however, abdominal moving dullness was strongly positive, and non-coagulated blood was obtained by diagnostic abdominal puncture. Routine blood examination showed the following: WBC 9,800/mmAbdominal enhanced CT scan revealed the following: 1. Obvious local lumen stenosis was observed at the beginning of the Celiac axis, with a few calcifications on the wall. This was considered as atherosclerosis. Calcified plaques had formed in the abdominal aorta, bilateral common iliac artery, bilateral internal iliac artery, and right external iliac artery, partially accompanied by mural thrombosis . 2. HemaIntraperitoneal bleeding can have many causes, including trauma, iatrogenic or ruptured aneurysm, gynecological disease, malignant tumors, and inflammatory or autoimmune processes, but spontaneous or idiopathic bleeding is rare. Abdominal apoplexy is a type of acute abdomen usually caused by unknown factors, and is accompanied by intra-abdominal vascular spontaneous rupture and retroperitoneal hemorrhage. The pathogenesis of abdominal apoplexy is still unclear; however, a few reports have reported possible causes as follows: 1. Abdominal organ vascular malformations, visceral tumors, abnormal coagulation function, liver cirrhosis, hypertension, arteriosclerosis, rheumatism, immune system diseases, and so on. It has also been documented that visceral vascular rupture can lead to abdominal bleeding. Kumari J et al.  reportedPrevious reports have indicated that patients with abdominal apoplexy should undergo auxiliary examination, including blood routine examination, abdominal B-ultrasound, abdominal x-ray, abdominal CT, computed tomography angiography (CTA), angiography, intervention, etc –7. SurgeIn this article, we reported two cases of gastro-omental angioma with spontaneous rupture and bleeding. As shown in In this article, although the vital signs and hemodynamics were relatively stable in both cases, we chose to perform operation as both patients were advanced in age, and their tolerance to bleeding was poor. Moreover, abdominal B-ultrasound and CT examination both confirmed heavy intra-abdominal bleeding, and the bleeding sites were undefined. In fact, if the patient's conditions are permitted, further preoperative examinations, including CTA, MRI or upper GI Endoscopy, ect, should be conducted to identify the source of bleeding for these types of cases. In the follow-up  no obvious abnormalities were reported, indicating a good prognostic outcome.Taken together, this is the first report of two eldely cases with gastro-omental hemangioma hemorrhage treated with laparoscopy. Clinicians should consider the possibility of spontaneous rupture of gastro-omental hemangioma when diagnosing patients with high-risk factors for abdominal apoplexy. Furthermore, laparoscopic exploration should be conducted in medical institutions with the relevant experience, as this can greatly reduce the surgical trauma to the patients, while aiding in a quick recovery. Although laparoscopic treatment of abdominal apoplexy achieved good therapeutic effect in the two cases reported herein, in patients where laparoscopic surgery is difficult, the procedure could be converted to open laparotomy or exploratory laparotomy.


Littoral cell angiomas are uncommon primary splenic haemangiomas with rare malignant potential. We report a case of a 76-year-old male with an incidental solitary littoral cell angioma found within an accessory spleen. We provide an overview of the literature of littoral cell angiomas and highlight the diagnostic challenge and treatment of this important differential for general surgeons caring for patients with splenic masses. This is the first case to describe primary resection of a littoral cell angioma with splenic preservation. Littoral cell angioma’s (LCA) are rare primary splenic haemangiomas, with only several hundred cases described in literature to date . These t2), and a left adrenal lesion. No splenic lesions were identified. History was significant only for intermittent episodes of self-limiting pain in the left upper quadrant. Subsequent investigation with quad-phase adrenal CT confirmed an adrenal adenoma but was unable to further characterize the left upper quadrant tumour. Tumour markers  were within normal limits.An otherwise well 76-year-old male presented after a high-speed motor vehicle accident. Contrast-enhanced computed tomography (CT) chest abdomen pelvis revealed an incidental solid left upper quadrant mass anterior to the spleen , displaying a more prominent central area of decreased attenuation and peripheral cystic changes  and CD68 (histiocyte marker). Histopathology revealed interspersed nucleated red cells with occasional megakaryocyte-like cells and haemosiderin macrophages, indicative of extramedullary haemopoiesis . The speet al. suggests that fine-needle aspiration (FNA) may aid in diagnosis [LCAs remain a diagnostic challenge, and largely rely on tissue diagnosis. Radiological imaging studies such as magnetic resonance imaging and CT have difficulty differentiating LCAs from other splenic tumours such as lymphoma, angiosarcomas and tumour metastasis , 5. For iagnosis . This isiagnosis . Howeveriagnosis , 6–8.et al. identified 13.8% of LCAs were associated with other malignancies, most commonly lymphoma [et al. also highlights that risk of splenectomy may outweigh the risk of malignancy [Literature estimates that only 7-24% of LCAs present as solitary singular lesions, with most cases presenting with multifocal splenic nodules , 9. Howelignancy . Howeverlignancy . Our casA review of literature has revealed only one documented case of an LCA excision without full splenectomy, in an 18-year-old female undergoing a partial splenectomy in 2012 . In thisLittoral cell angiomas are a rare but important differential for left upper quadrant masses. Current recommended treatment is aimed towards surgical excision with splenectomy. This report encompasses several key features of LCA management and is the first to report a successful spleen-preserving littoral cell angioma excision.


We describe the case of a 69-year-old female who thought she had "food poisoning", which prompted her visit to the Emergency Department for evaluation of her vomiting and abdominal discomfort. Contrasted computed tomography imaging with contrast of the abdomen subsequently revealed the diagnosis of gastric volvulus, and the patient was promptly taken for surgical intervention. Gastric volvulus is rare and presents with a nonspecific history, exam, and laboratory findings. In the acute care setting, it is important to maintain a high clinical suspicion for this diagnosis, as timely imaging and intervention are crucial to decreasing patient morbidity and mortality. For a patient with gastric volvulus, or rotation of the stomach along one of its axes, early diagnosis and treatment are crucial to preventing mortality . GastricA 69-year-old female with a history of laparoscopic appendectomy 15 years ago but no other medical history presented to the emergency department (ED) with nausea and vomiting. The patient reported multiple episodes of emesis since symptom onset the evening before presentation. She denied hematemesis, diarrhea, and fever. The patient denied any significant abdominal pain, only reporting that the episodes of straining during emesis led to her abdominal muscles feeling constantly sore.On a physical exam, she was alert and oriented but appeared to be in moderate distress. She was afebrile at 97.6° F, her heart rate was 97 beats per minute, her oxygen saturation on pulse oximetry was 96%, and her blood pressure was 189/115 mm/Hg. Her cardiopulmonary exam was unremarkable, and she had normal distal peripheral perfusion. Her abdomen was soft and non-distended, with mild generalized tenderness to palpation but no rebound or guarding.Laboratory testing revealed mild hyperglycemia at 207 mg/dL and leukocytosis at 20.86x103 with 93% neutrophils. Otherwise, electrolytes were within normal limits. Considering the patient's age and nonspecific presentation, CT imaging of the abdomen and pelvis with contrast was obtained to further evaluate her generalized abdominal tenderness. CT  tube was placed with an immediate output of 500 cc of gastric contents. Less than three hours after the CT scan was obtained, the patient was promptly taken to the operating room for esophagogastroduodenoscopy (EGD). The EGD revealed gastric volvulus at the pylorus/duodenum in the intrathoracic portion of the hernia, with patchy mucosal necrosis observed. She underwent endoscopic gastric decompression and was admitted after this procedure, with plans for subsequent hiatal hernia repair during her hospital stay. Five days after her initial ED presentation and EGD, she underwent laparoscopic hiatal hernia repair and gastropexy. She was discharged home after a seven-day hospitalization without any significant postoperative complications. At the time of discharge, her pain was controlled, and she was tolerating a regular diet with the return of normal bowel function.This case describes a patient with a rare condition that can easily be misdiagnosed. Maintaining a high level of suspicion and obtaining prompt imaging is crucial to identifying the presence of gastric volvulus, as there are no specific history, or exam findings or diagnostic lab values. Although, previously barium swallow was the gold standard for diagnosis of gastric volvulus, CT scans are most accurate at diagnosing acute gastric volvulus . GastricRegarding the ED provider’s role in the diagnosis and management of gastric volvulus, a few things should be considered. Due to the nonspecific symptoms and lab values, a high index of suspicion must be present, specifically in patients who are greater than 50 years old, given the increased risk of gastric volvulus in this age group ,8. CT imAlthough definitive treatment in patients with acute gastric volvulus is surgical intervention, the emergency department providers’ decisions to order a CT scan in the older adult patient presenting with acute, non-specific abdominal symptoms could lead to the correct diagnosis of gastric volvulus -9. FurthIt is important that emergency physicians are aware of this rare diagnosis and consider it when evaluating patients with relevant histories and exam findings, as timely diagnosis and management are important for reducing morbidity and mortality.


To document a case of spontaneous regeneration of the mandibular body following subtotal mandibulectomy in a juvenile dog.A 3-month-old male intact French bulldog was presented with papillary oral squamous cell carcinoma located at the dorsal aspect of the molar region of the left mandible. Initial biopsy of the mass was performed by the primary care veterinarian. Complete clinical staging revealed no signs of metastasis. Computed tomographic images of the head showed minimal contrast enhancement of the mass with no signs of periosteal or bone involvement. Subtotal mandibulectomy was performed. Histopathology indicated complete excision of the tumor. The patient returned 8-weeks later for follow up and cleft palate surgical repair, at which time bone was noted in the mandibulectomy area on palpation. Repeat computed tomography of the head revealed complete regeneration of the left mandibular body from the level of the ramus to the mandibular symphysis. No treatment for malocclusion was necessary due to the reformation of a functional mandible.The present case demonstrates that spontaneous regeneration of the mandibular body is possible following subtotal mandibulectomy in immature dogs. Subtotal mandibulectomy is a radical procedure that can lead to long term complications including mandibular drift, malocclusion, and oral pain. This case report provides evidence that these sequelae may be mitigated or eliminated in young patients undergoing this procedure. En bloc excision, when possible, is considered the gold standard treatment for canine papillary oral squamous cell carcinoma (pOSCC)  . Dependi (pOSCC) –9. These (pOSCC) .In this case, the long-term consequences of a young puppy undergoing subtotal mandibulectomy were uncertain given the low prevalence of oral cancer in this age group. Other reports of pOSCC in young dogs only describe tumor recurrence rates in the follow-up period, but do not include clinical outcomes related to occlusion and mastication function –5. To thA 3-month-old male intact French bulldog was presented for evaluation and treatment of a pOSCC located at the dorsal aspect of the caudal left mandibular body. The mass had been noted incidentally by the referring veterinarian 2 weeks prior while assessing a congenital palatal defect. Initial biopsy of the mass resulted in a diagnosis of a pOSCC. Routine bloodwork performed prior to referral revealed a mild regenerative anemia (HCT 34%) but was otherwise unremarkable. On presentation, a conscious oral examination revealed an ulcerated, broad-based soft tissue mass located distal to the left deciduous mandibular third premolar tooth . A midliThe dog returned 2 days later for a left extended subtotal mandibulectomy to remove the mass. The extended subtotal mandibulectomy was elected with the goal of achieving clean margins while also preserving the anatomy of the ipsilateral TMJ . The samThe patient recovered from anesthesia and was hospitalized postoperatively on a constant rate infusion of fentanyl (3 μg/kg/h), carprofen , and ampicillin/sulbactam . The patient tolerated the feeding tube well and appeared comfortable. The dog was discharged to the care of its owners the following day with carprofen , gabapentin , and amoxicillin/clavulanic acid . The owners were instructed to feed the patient specific volumes through the feeding tube and prevent any access to chews or toys for 4 weeks.en bloc section of mandible reported complete surgical excision of the tumor. The patient returned 18 days after surgery, at which time a conscious oral exam revealed no evidence of mandibular drift or traumatic malocclusion. The patient returned again 8 weeks after the initial surgery for cleft palate repair, at which time firm tissue at the area of the mandibulectomy site was palpated on conscious oral examination. The same anesthetic protocol was utilized, and CT scan of the head was performed, revealing new mandibular bone formation from the ostectomy site to the level of the mandibular symphysis (Histopathology of the ymphysis . No manden bloc excision of benign and malignant mandibular neoplasia, gunshot wounds, and other trauma in pediatric human patients (This is the first report of spontaneous regeneration of the mandible following oral oncological surgery in a dog. Unlike older dogs that undergo partial mandibulectomy for tumor removal, this patient spontaneously regenerated the mandibular body within 8 weeks of the initial surgery. In humans, young patients who undergo mandibulectomy have been reported to regenerate new bone from progenitor cells within the remaining periosteum . For exapatients . The degpatients . Appositpatients . PeriostEarly detection of the oral tumor likely played an important role in achieving long term remission surgery. A subtotal mandibulectomy preserved adjacent periosteum, which may not have been possible if the tumor size and local invasion had made more extensive excision necessary. Anatomy such as the dentition and the neurovascular structures of the mandibular canal of the regenerated mandible cannot reform due to the loss of the necessary progenitor cells present during embryologic development. Nonetheless, regeneration of the body of the left mandible resulted in a functional lower jaw with no mandibular drift. Serial oral examinations following regrowth confirmed no signs of traumatizing malocclusion, and the patient continued to prehend food and water with ease.Reconstructive surgery following segmental or partial mandibulectomies in dogs has been described to help address the functional disturbances associated with these surgeries, such as traumatic malocclusion , 8, 16. The 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 animal study because the case report is a description of a clinical case. Written informed consent was obtained from the owners for the participation of their animal in this study.AW: Writing – original draft. SP: Writing – review & editing. NF: Writing – review & editing.


Angiomyolipoma is a benign mesenchymal neoplasm of a wide histological heterogeneity belonging to the PEComa “family.” The liver, after the kidney, is their second most frequent location. However, inflammatory hepatic AMLs constitute a rare entity, with only fourteen documented cases until 2020. These neoplasms can overlap morphological features of IgG4-related diseases, being of great diagnostic relevance to demonstrating myomelanocytic-lineage differentiation of the neoplastic cells. Case presentation: we present a new case of an inflammatory hepatic AML resembling an IgG4-related disease in a 35-year-old woman with a subcapsular 5 cm mass confined to segment VII of the right hepatic lobe. Although having reduced its size along the tumor’s natural evolution, complete tumor resection was decided due to its hypermetabolic behavior  assessed by PET-CT scan. Finally, the patient underwent a right hepatectomy due to spontaneous rupture and bleeding of the lesion during the intervention. All the diagnostic and therapeutic procedures occurred in the last months of the COVID-19 pandemic. Conclusions: This review aims to describe inflammatory hepatic AML histological and immunohistochemical features. We further sought to establish a clinicopathological contextualization of this tumoral subtype. First described in 1976 . Given iInflammatory hepatic AML, according to the existing literature, most frequently affects women with an average age of 46,5 years. Commonly found in the left hepatic lobe, inflammatory AML’s average size is 6,65 cm. Abdominal pain and discomfort are the typical clinical manifestations, and no association with tuberous sclerosis has been described , 4. ImagAlthough the exceptionally aggressive course of this disease has been reported, there are no examples of malignant inflammatory hepatic AML –5. SurgiThe case of a 35-year-old woman with a manifest lack of appetite, weight loss, asthenia, general malaise, and digestive discomfort is presented. The blood test revealed normocytic and normochromic anemia, thrombocytosis, and slight hypereosinophilia. The serological analysis showed positive IgM for Rickettsia coronii and indeterminate IgM levels for Mycoplasma pneumoniae. Treatment with doxycycline, 100 mg twice per day, was given. The evolution was a clinical improvement but without a complete resolution of the symptomatology. Ultrasonographical studies of the abdominal cavity detected a hypoechogenic lesion resembling a hepatic abscess. It affected the posterior aspect of the right hepatic lobe, measuring 8,4 cm x 6 cm. Contrast-enhanced CT scan describes this lesion as a solid image of 8 cm with peripheral hypervascularization with the radiological orientation of a hepatic adenoma. Finally, contrast-enhanced dynamic MRI defined it as a well-demarcated lesion of 7,8 cm (AP) x 9,3 cm (RL) x 6,7 cm (CC) with portal vessel deviation, T1-hypointense signal and intermediate signal in T2 compared to the normal hepatic parenchyma. A perfusion decrease was also observed, giving the diagnostic impression of a hepatic adenoma, so clinical follow-up was strongly recommended. However, the PET-CT scan evidenced a hypermetabolic behavior of SUVmax = 12,6, establishing the need to exclude a malignant lesion  and the same vaguely “Reed-Sternbergoid” cells with a mixed myomelanocytic phenotype. In this area, cells were alone or grouped in small aggregates Fig. c and d.Additionally, thickened walls were spotted at the periphery, with no evidence of fatty tissue in the tumor sample. Prominent lymphoplasmacytic infiltrate surrounding vessels was evidenced. Some signs of luminal obliteration at the fibrosclerotic areas were also seen Fig. c. SporadLow mitotic activity was observed (0–1 mitosis per 50 HPF). The inflammatory infiltrate globally accounted for more than 50% of the lesion. No necrosis nor lymphovascular invasion phenomena occurred. Even though tumor borders were expansive, resection margins were free of neoplasm.AML is a rare mesenchymal neoplasm of a usually benign biological behavior composed of a heterogeneous mix of blood vessels, smooth muscle /myoid cells (epithelioid and fusiform), and mature adipose tissue. It commonly develops in the kidney, followed by the liver in frequency –11. RadiInflammatory AML of the liver, described by Kojima M. in 2004 , is a paNo relation with tuberous sclerosis is reported in all cases published to date. There is no evidence of disease persistence after six months post-treatment, agreeing with 12 case descriptions that refer to prognosis and that include clinical follow-up data (ranging from 2 to 108 months).The currently depicted case displays a combination of diagnostic morphological features of hepatic inflammatory AML and histological characteristics in common with IgG4-RD, agreeing with the case described by Agaimy and Märkl . The tumIgG4-RD constitutes a fibroinflammatory systemic process with a substantial plasma cell infiltration affecting different organs . It is associated with fibrosclerosis and obliterative phlebitis. Elevated IgG4 serum levels are found in up to 50% of the cases –21. LiveA histological feature of interest for is the proportion of lymphoplasmacytic inflammation, more than 50% of the tumor , 13, 14.We believe the similarity, possible overlap, or concomitance of inflammatory AML with IgG4-RD must be considered. It would be appropriate to perform a thorough immunohistochemical evaluation of myomelanocytic markers in liver lesions cataloged as IgG4-RD to rule out the presence of AML and serum IgG4 determinations.In a purely speculative sense, a diagnostic dilemma arises from the striking variation in tumor size in a few months on radiological follow-up controls . The initial doxycycline treatment with an immunoregulatory effect without corticosteroids or other immunosuppressors (despite a prior diagnostic approach of IgG4-RD by CNB) does not fully explain the size reduction. This gives added value to the uneven distribution of the lymphoplasmacytic inflammatory component of AML that we present together with the alternating pattern of sclerotic fibrosis with areas of higher cell density and with pseudovascular/sinusoidal pattern. It is fair to believe that inflammatory AML of the liver may behave as hypermetabolic (elevated SUV by PET-CT scan) and dynamic lesion depending on the variable tumor-related inflammatory response. The heterogeneous histology and macroscopic appearance of the neoplasm may respond to the process of volume variation. This dimensional heterogeneity has not been observed by the other reports in the literature from the radiologist’s point of view –11, 13. In our case, remission was confirmed six months after the surgical procedure. Significantly, serum IgG4 levels reached normal parameters (78.8 mg / dL) compared to the first values (245.7 mg/dL). This data would support the hypothesis that those cases of inflammatory AML with histology similar to IgG4-RD may be associated with high levels of IgG4. Although having occurred during the COVID-19 pandemic, no delays in diagnosis or treatment were observed. Additionally, no SARS-COV-2 infection was detected during the whole process by RT-PCR.Getting familiar with this entity is relevant for an optimal differential diagnosis in hepatic tumors coursing with inflammation-rich histology, such as the inflammatory myofibroblastic tumor, the dendritic follicular cell tumor, and IgG4-RD. Well-vascularized tumors like hemangiomas should also be considered. Also, neoplasms of epithelioid-fusiform morphology, including hepatic adenoma, focal nodal hyperplasia, hepatocellular carcinoma, gastrointestinal stromal tumors, and metastases  . The demTo conclude, pathologists should identify this rare subtype of inflammatory AML of the liver , in which the percentage of inflammatory infiltrate is above 50%. Adipose cellular elements are sometimes absent, and the presence of vessels can be misinterpreted as non-neoplastic, which causes AML to go unnoticed. As a result, performing a targeted immunohistochemical analysis should include myomelanocytic differentiation markers to typify the neoplastic cell. Its similarity, possible overlap, or concomitance with IgG4-RD must be taken into account, being necessary to gather more documented cases to define a significant association between both entities. Finally, in our case, the variation of tumor size could be justified by a tumoral inflammatory response of variable course.


Cholesterol granuloma is a histopathological finding characterized by a mass of connective tissue and granulation tissue. It is primarily observed in the middle ear, mastoid process, or paranasal sinuses, with rare occurrences in the dental odontogenic region. A dentigerous cyst encloses the crown of an unerupted tooth by expanding its follicle and attaches to the neck of the tooth. Here, we report a 63-year-old female who presented to the dental clinic complaining of an ill-fitted denture. A panoramic radiograph showed a well-defined radiolucent lesion in the upper left maxillary sinus with an impacted third molar. Computed tomography revealed loss of the anterior and lateral sinus walls. The cyst was enucleated surgically. The final diagnosis was confirmed by histopathological examination, which revealed focal areas of cholesterol clefts in the cystic wall of the dentigerous cyst. Cholesterol granuloma (CG) is a histopathological entity with a cystic appearance . It is iDentigerous cysts are odontogenic cysts that develop from a tooth follicle around the crown of an impacted or unerupted tooth. It is the most common developmental cyst, accounting for up to 20% of all cysts of the jaws lined by epithelium . When inVery few cases of CG of the jaw associated with the wall of dentigerous cysts have been reported to date ,6,7. HerA 63-year-old female patient was referred to the Department of Oral and Maxillofacial Surgery with swelling on the left side of the face and a chief complaint of a recent ill-fitted denture. The patient reported that the problem started after she noticed a slowly growing bulge in her upper jaw three months before. Upon intra-oral examination, a swelling ranging from firm consistency to eggshell cracking was noticed at the left upper posterior maxillary side and occluding the buccal vestibule. There was no fistula, and the swelling was asymptomatic with no signs of local infection. A panoramic radiograph revealed an impacted upper left third molar into the left maxillary sinus with bone resorption and complete pneumatization of the sinus inferiorly until the alveolar crest level Figure . Multi-sThe lesion was surgically approached through the anterior maxillary wall under general anesthesia using the Caldwell-Luc approach. Complete enucleation of the lesion with the wisdom tooth was performed. The opened cystic mass measured 4.5 × 3.5 × 1.5 cm with a gray smooth inner lining. Microscopic examination revealed a cyst wall focally lined by pseudostratified columnar and stratified non-keratinizing squamous epithelium Figure  with areHere, we present a rare case of CG in the wall of a dentigerous cyst associated with an impacted molar and involving the maxillary sinus. To the best of our knowledge, there are 12 published articles, and a comprehensive review of the scientific literature on this topic was conducted, encompassing relevant publications from 1984 to the present -13. FurtIt seems that CG could be formed in any type of odontogenic cyst as a reaction to cholesterol crystals ,15. CholThe patient’s main complaint was a recent ill-fitted denture due to intra-oral swelling. Despite the invasion of this mass into the maxillary sinus, the patient did not report any history of rhinorrhea, sinusitis, facial pain, or nasal obstruction, which are the usual nonspecific symptoms similar to cases reported with invasion into the maxillary sinus .This opens the door to a dilemma regarding the pathogenesis of CG. The crystal clear etiology of CG has not yet been established; however, in these cases associated with the maxillary sinus, it could be explained by inadequate drainage or hemorrhage in the sinus following trauma with the consequences of hemolysis in the sinus, as cholesterol may come from the cell membrane of the erythrocytes. Along with impaired drainage, this cholesterol could be precipitated as crystals, which later evokes an inflammatory response as a foreign body with subsequent migration of leukocytes and macrophages and the formation of giant cells ,18.Yamazaki et al.  introducIn the present case, the lesion was part of an odontogenic cyst that involved the maxillary sinus. Both of these factors could be attributed to etiological factors. Almada et al.  pointed The potential conditions to consider include odontogenic keratocyst, mandibular hemangioma, aneurysmal bone cyst, mandibular AVM , central giant cell granuloma, Ewing sarcoma, juvenile ossifying fibroma, osteosarcoma, or Brown's tumor.There is no specific radiographic presentation for CG, and it could occur in both jaws with or without odontogenic cysts ,4, as thCG found in the wall of a dentigerous cyst associated with an impacted molar and involving the maxillary sinus is not considered a distinct clinical condition but rather a nonspecific histopathological reaction to cholesterol crystals. As its clinical and radiographic characteristics are nonspecific, the findings of this study emphasize the significance of considering CG in the differential diagnosis of odontogenic cysts and tumors. Histopathological analysis is essential for a correct final diagnosis of CG.Recognition of the true nature of the entity as a dentigerous cyst obviated the need for a different line of surgical treatment such as hemi-maxillectomy. We agree with Baldini et al., who performed systematic surgical enucleation as soon as the diagnosis of such lesions was made in order to avoid any complications related to its growth. We need more studies on CG cases in the oral cavity to learn more about their unique characteristics and better understand this rare condition.


We present the case of a pregnant 32-year-old woman who presented with a giant fetal facial tumour at 22 weeks. The mass, initially 4 × 3.5 × 3 cm in size, was largely cystic with a small solid component. It subsequently increased to 9 × 9 × 10 cm. Significant compression effects on the fetal orbit, temple and infratemporal fossa, with potential compression of the optic nerve, were noted on ultrasound and MRI. The cyst required drainage twice in the pregnancy: firstly to reduce the compression effects and secondly to facilitate caesarean delivery. Postnatally, the baby had significant compression and displacement of the craniofacial skeleton from the mass effect. Postnatal histology revealed a diagnosis of a teratoma. This case highlights the complexities and challenges surrounding the diagnosis and management of a giant fetal facial tumour. We present the complex case of a giant fetal facial tumour diagnosed at 22 weeks. There is a paucity of published data on both the antenatal and postnatal management of these large lesions. This case highlights the challenges of managing such cases, from antenatal diagnosis to delivery and the postnatal surgical management of such tumours. This case proved the importance of significant multidisciplinary team (MDT) involvement to ensure a good clinical outcome. The MDT included paediatric radiology, paediatric otorhinolaryngology, fetal medicine, neonatology and paediatric neurosurgery. It also adds to the body of literature on these unusual cases. Written consent has been obtained for the publication of this report with images.We present the case of a pregnant 32-year-old woman who presented in her second pregnancy. She had a history of one previous uncomplicated pregnancy with a term vaginal delivery and a birth weight of 2900 g. There was no relevant past medical history.A 22-week anatomy scan revealed a mass in the left temporal region measuring 4 × 3.5 × 3 cm Fig. a. FurtheThe patient was closely monitored with regular ultrasound scans. By 28 + 3 weeks, the cystic mass began to increase in size to 8.5 × 7.7 × 7.5 cm with the solid vascular solid component measuring 2.3 × 2.4 cm. A single vascular supply to this solid component was confirmed with colour Doppler. By 30 + 2 weeks, the tumour measured 9 × 9 × 10 cm and was significantly larger than the fetal head  and MRI imaging and genetic assessment as necessary. The use of ultrasound is still the modality of choice for imaging the fetus due to its speed, cost and availability. Due to the possibility for haemodynamic instability in teratomas , regularIn this particular case, two MRI scans were performed to ensure a patent airway at delivery. The two most common types of congenital masses that obstruct the airway are the lymphatic malformation and the teratoma . It was The survival rate of patients who presented with congenital fetal head and neck tumours is 35.35% . EXIT prThis case highlights the challenges of diagnosing and managing rare and complex fetal facial tumours. The management of this particular case was complicated by the cystic mass persistently refilling almost immediately post drainage. Careful management and regular ultrasound scans were necessary to monitor the size of the mass and its compression effects on surrounding structures. MRI was a very useful modality to assess whether the EXIT procedure was necessary and also to aid in our understanding of the tumour’s boundaries and therefore guiding our care. Delivery of a child with a large facial tumour has been rarely reported in the literature and requires MDT management. A further challenge in this case is the paucity of data available on these rare fetal facial tumours. A good clinical outcome was obtained following multidisciplinary antenatal care and early referral to neurosurgical and ENT teams in a large tertiary paediatric hospital.


Medicinal leech therapy (MLT) is used in various medical disciplines, among which are reconstructive surgery and microsurgery. Medicinal leech therapy is also often adopted by alternative and traditional medicine, aiming to treat various common medical symptoms, such as fever and arthritis.Congenital umbilical hernia is a rather common physical finding in the pediatric population, where every third Caucasian newborn, roughly, is being diagnosed with the condition, and even more so among the African population. Fortunately, most cases resolve spontaneously in the first years of life. Toddlers whose hernia does not close typically require umbilical hernia repair.This article describes the case of a five-year-old girl with an asymptomatic congenital umbilical hernia who was admitted to the ER due to an omental eventration that occurred following the placement of a leech on her umbilicus in her parents’ attempt to treat a febrile episode. She subsequently underwent an urgent umbilical exploration and a repair of her umbilical hernia.The main known risks of leeching are bacterial infection, anemia, prolonged bleeding, and, less frequently, pruritus, allergies, marked edema, and cellulitis. This article presents yet another possible complication that, to the best of our knowledge, has not been documented before in the literature.Several old-school therapies transcended over time into medical disciplines. Given that "traditional" practices often take place within households and communities, it is of crucial importance to point out potential complications, both rare and common, that can be caused by those practices in order to reduce the risk of severe, undesired outcomes. Indeed, the growing interface between traditional, alternative therapies and modern, conventional medicine urges better parental guidance and improved education regarding potentially harmful and unauthorized interventions. Medicinal leech therapy (MLT) or hirudotherapy is used in various medical disciplines, among which are reconstructive surgery and microsurgeries. It is often adopted by alternative and traditional medicine, aiming to treat various common medical symptoms, including, among others, fever and arthritis. As such, it is commonly classified as one of the old-school folk remedies applied in certain communities worldwide.An ancient therapeutic technique, MLT exploits the saliva secreted by blood-sucking leeches during their feeding process. Leeches secrete more than 20 identified bioactive substances, such as hirudin, antistasin, and saratin, that have analgesic, anti-inflammatory, anticoagulant, and platelet inhibitory effects, as well as antimicrobial and thrombin regulatory functions . In lighIn 2004, the United States Food and Drug Administration (FDA) approved the use of MLT for certain ailments. While officially limited to these fields only, the use of leeches for medicinal purposes, also known as "leeching", remains a popular therapeutic practice as a "home remedy" by traditional and alternative therapists . This kiCongenital umbilical hernia is a very common physical finding in the pediatric population, affecting up to 30% of all White newborns and with an estimated prevalence as high as 85% among newborns of African descent. Fortunately, numbers decrease thereafter due to spontaneous gradual closure, with 2% to 20% at one year of age and less than 4% at four years of age [In this case study, we report a case of a child who presented with omental eventration following the placement of a leech on the child's umbilicus in an attempt by her parents to treat a febrile episode. The child was diagnosed with an umbilical incarcerated hernia and omental eventration and, subsequently, underwent an urgent umbilical exploration with a repair of the umbilical hernia. To the best of our knowledge, this is the first case of its kind to be documented in literature.A previously healthy five-year-old girl was admitted to our emergency department following a two-day fever that was attributed to otitis media by her general practitioner. Her parents chose to treat her fever with homeopathic remedies, along with the placement of a leech on her umbilicus. Indeed, they later revealed that such treatment is used frequently in their family. However, when they removed the leech, they were surprised to find visible tissue protruding out of her umbilicus Figure , which lThe patient’s vital signs upon admission were as follows: blood pressure of 94/49 mmHg, heart rate of 138 bpm, temperature of 38.2 degrees Celsius, and 100% oxygen saturation in room air. Her physical examination revealed mild erythema of the left ear and an omental eventration in the center of the umbilicus, with signs of neither bleeding nor infection  of 16 mg/L. The patient was diagnosed with incarcerated umbilical hernia and omental eventration and was taken to an urgent umbilical exploration following the administration of broad-spectrum antibiotics, in accordance with our infectiologist’s recommendations.The surgery began with regular umbilical exploration , following an incision below the umbilicus that revealed an umbilical hernia of roughly 4 mm defect, containing omentum that protruded through a 3 mm bite-hall in the umbilicus itself Figure .The omentum was tied, ligated, and then sent for bacteriology (with negative results reported). The umbilical hernia repair took place using absorbable sutures, after which the defect was closed. As for the umbilicus, it was closed by central stitches using absorbable sutures. The patient was discharged the following day with no other complaints or postoperative complications.Current knowledge of common complications caused by leeching includes infections, anemia, and blood loss , with inThis case is a unique description of a new complication, not previously documented, of leech therapy: an eventration of the omentum through a previously asymptomatic umbilical hernia, occurring as a result of a leech-bite, following an attempt to resolve a febrile episode in a five-year-old girl by placing the leech on her umbilicus.Such a case of communication between the abdominal cavity and the outer atmosphere imposes a significant risk for serious complications, such as ileus, infections, sepsis, and, in severe cases, even death. Clearly, formal medical indications for leech therapy do not include the use of leeches as antipyretic agents, yet some people and certain communities do practice it, perhaps even on a regular basis. Such "therapy" can result, as described above, in an unwanted deep penetration of the leech that might, in turn, cause an eventration of the umbilical hernia sac along with its contents.Several old-school therapies transcended over time into medical disciplines. One of them, MLT, is being used in various medical procedures. The case reported hereinabove relates to the interface between such old-school therapies  and modern, conventional medicine. Given that "traditional" practices often take place within households and communities, it is of crucial importance to point out potential complications that can be caused by those practices in order to reduce the risk of severe, undesired outcomes. Leeching complications, such as the one described, underline the substantial importance of thorough parental guidance and professional communal education regarding potentially harmful and unauthorized interventions, such as the ones exploiting leeches, and in particular, those that include placing them on the umbilicus.


We present a case series of two rare cases of angioleiomyoma of the hand, an infrequent type of benign tumor. In the first case, a 41-year-old female presented with a left thumb mass that had increased in size over two years. Imaging studies revealed a destructive lesion involving the first webspace with infiltration of the first metacarpal, and the mass was initially suspected to be a sarcoma. However, a percutaneous image-guided fine-needle aspiration (US-FNA) and ultrasound-guided core-needle biopsy (USG-CNB) of the left-hand mass confirmed the diagnosis of angioleiomyoma. The mass was surgically excised, and the final diagnosis was consistent with the earlier USG-CNB. In the second case, a 63-year-old man with end-stage renal disease presented for consultation regarding dialysis access creation. During the examination, a large, soft, mobile mass adjacent to the wrist on the medial aspect of his hand was identified. This was presumed to be a lipoma. However, the histopathology report revealed a benign angioleiomyoma measuring 3.2 cm, which had been completely excised during the surgery. This case report highlights the importance of considering angioleiomyoma in the differential diagnosis of soft tissue masses and the utility of US-FNA and USG-CNB in diagnosing these tumors. Angioleiomyoma is a frequently painful, benign, solitary, subcutaneous, or deep dermal tumor made up of mature smooth muscle bundles surrounding and intersecting blood vessels. It is a rare tumor that is more common in middle-aged women; it tends to present in the lower extremities, and it rarely appears in the hand or fingers. While these tumors are typically smaller than 2 cm, they can grow as large as 6 cm ,2.The diagnosis of angioleiomyoma in the hand is often initially overlooked and can be difficult to distinguish from other types of tumors. However, it is essential to consider angioleiomyoma as a potential diagnosis, particularly when patients present with a painless mass in the hand or upper extremity. The treatment for angioleiomyoma is typically marginal excision, and the prognosis is excellent as these tumors are benign.Case oneThe patient was a 41-year-old right-handed woman who visited the clinic with a two-year history of a left thumb mass. She initially suspected it was a ganglion cyst due to its small size. However, a year ago, a surgeon attempted to remove the mass but aborted the procedure due to the mass's extent, and no pathology was sent. Since then, the mass had grown significantly in size. No personal or family history of cancer existed except for hypertension, obesity, and hyperlipidemia. The patient did not experience any numbness, tingling, or pain during the visit.During the examination, a 6x6 cm irregular mass was observed on the dorsal aspect of the left thumb, overlying the first metacarpal. The mass extended circumferentially around the base of the thumb and into the thenar eminence. However, the patient was able to actively flex, extend, and oppose the thumb to her small finger. There was no tenderness upon palpation of the mass, and the thumb exhibited good perfusion.The patient presented with an outside X-ray and magnetic resonance imaging (MRI) of her left hand. The X-ray revealed a destructive lesion involving the first webspace with infiltration of the first metacarpal. Associated soft tissue swelling was noted in the area of the mass, along with internal calcifications within the soft tissue mass circumferentially around the first metacarpal. The MRI showed a soft tissue mass in the thenar eminence of the hand that measured 6.3 x 4.3 x 5.5 cm. The mass disrupted the cortex of the thumb metacarpal. An associated infiltrative bony lesion measured 2.5 cm and was located in the bone marrow. The soft tissues between the extensor pollicis longus tendon and the first metacarpal were infiltrated, and the lesion invaded the interosseous musculature, the first metacarpal, and the flexor and abductor pollicis tendons.Based on the imaging studies, the mass was initially suspected to be a sarcoma, and surgical intervention, potentially including amputation of the thumb and index finger rays, was considered. A computed tomography (CT) scan of the chest and a computed tomography angiography (CTA) scan of the left hand were ordered, along with lab work. The CT scan of the left hand showed a lobulated mass measuring 4.0 x 6.0 x 5.5 cm in greatest anterior-posterior (AP), transverse, and craniocaudal dimensions, respectively, at the dorsal aspect of the first metacarpal, extending into the webspace and adjacent to the first metacarpal. This mass was predominantly located in the soft tissues; however, there was an osseous extension into the dorsal cortex of the mid-first metacarpal. The mass involved the first dorsal interosseous muscle and presented with ring and arc calcifications Figure . The CTAThe patient underwent a percutaneous image-guided fine-needle aspiration (US-FNA) and an ultrasound-guided core-needle biopsy (USG-CNB) of the left-hand mass. The US-FNA reported rare benign spindle cells, a bloody background, with few neutrophils and lymphocytes, and no evidence of malignancy. The final diagnosis from the USG-CNB was angioleiomyoma, which was also confirmed by a second opinion. Tumor cells were positive for smooth muscle myosin heavy chain (SMMHC) and negative for S100 and SOX10. The endothelial cells of blood vessels were positive for CD34 Figures , 3.Afterward, the mass was surgically excised at a different medical facility. The excised surgical specimen included one segment of skin with attached subcutaneous tissue measuring 6.7 x 4.6 x 4 cm, as well as one separate segment of bony tissue measuring 5 cm in the largest dimension and another separate segment of gray-white tissue measuring 2.5 cm in the largest dimension. Upon sectioning, the tissue was found to contain a fleshy tumor with some calcification. The final diagnosis of angioleiomyoma was confirmed, which was consistent with the earlier USG-CNB.Case twoA 63-year-old man with hypertension and end-stage renal disease presented for consultation for dialysis access creation. He was mapped to have a very suitable cephalic vein in the wrist. A large, soft, mobile mass adjacent to the wrist on the medial aspect of his hand was identified during the examination. After discussion with the patient, a decision was made to resect the mass, which was presumed to be a lipoma, at the same time the arteriovenous (AV) fistula was being created. The patient consented to both procedures.The patient was taken to the operating room, and a 2 cm incision was made over the medial aspect of the thumb. The subcutaneous tissue was dissected, and the mass was circumferentially dissected, gently retracted with an Allis clamp, and freed from its attachments. The surgery was completed without complications, and the mass was sent to pathology for identification. The radiocephalic fistula was created with a robust thrill in the cephalic vein in the arm.The histopathology report revealed a benign angioleiomyoma measuring 3.2 cm, which had been completely excised. Microscopic examination revealed a well-circumscribed tumor composed of spindle-shaped cells with interspersed various-sized blood vessels with different wall thicknesses. No significant increase in mitoses or necrosis was seen. The tumor cells were positive for smooth muscle actin (SMA), consistent with angioleiomyoma Figures , 5.Angioleiomyomas are slow-growing, painful tumors commonly occurring in the lower extremities and are more common in women. These tumors are often misdiagnosed and can be mistaken for a ganglion cyst, inclusion cyst, lipoma, fibroma, synovial sarcoma, other low-grade soft tissue sarcomas, haemangioma, and neurogenic tumor. Histopathology is necessary to establish the correct diagnosis. Angioleiomyoma is a benign tumor of smooth muscle origin characterized by a proliferation of well-differentiated smooth muscle cells with abundant vascularization -3.Angioleiomyomas in the hand are rare, and they are even more uncommon in the fingers. On physical examination, they appear as a subcutaneous oval or round mass with a maximum diameter ranging from 0.7 cm to 6.0 cm. Imaging findings are usually nonspecific, and on CT, angioleiomyomas may appear as a well-defined and otherwise unspecific cutaneous or subcutaneous soft tissue density mass, sometimes with calcifications. The diagnosis requires an excisional biopsy with confirmatory immunohistochemical evaluation -6.On microscopic examination, angioleiomyomas are characterized by a well-circumscribed, non-encapsulated mass of smooth muscle cells arranged in fascicles or bundles. Blood vessels, which are the defining feature of angioleiomyomas, are often seen within the tumor mass and may appear dilated and surrounded by smooth muscle cells. Immunohistochemical evaluation of angioleiomyoma typically shows positive staining for SMMHC, SMA, desmin, and h-caldesmon, which are markers of smooth muscle cells. In addition, angioleiomyomas often show positive staining for CD34, a marker of endothelial cells commonly expressed in the blood vessels within the tumor ,5,6. TheGanglion cysts are common, benign lesions often occurring on the hands and wrists. Histopathologic examination typically reveals a cystic structure filled with clear or mucinous fluid, lined by synovial cells, and surrounded by fibrous tissue. Lipomas are the most common soft tissue tumors; they are typically slow-growing, painless, and well-circumscribed. Histologically, lipomas are composed of mature adipocytes and fibrous tissue. Synovial sarcomas are malignant tumors that often occur in the extremities and consist of spindle-shaped cells arranged in a biphasic pattern with glandular or epithelial differentiation areas. Low-grade soft tissue sarcomas, such as leiomyosarcoma and myxoid liposarcoma, are malignant tumors that can be difficult to distinguish from benign tumors on imaging and gross examination. Histopathologically, both are spindle cell tumors with distinct morphologies. Immunohistochemistry may show positive staining for specific markers, such as SMA for leiomyosarcoma and S100 for myxoid liposarcoma .The mechanism of development of angioleiomyomas remains unclear. While classified as vascular hamartomas, these tumors' etiology has also been linked to mechanical and hormonal causes . We presIn conclusion, our case series highlights the importance of thorough evaluation and histopathological examination in diagnosing hand tumors. Although rare, angioleiomyoma should be considered in the differential diagnosis of painless, subcutaneous, well-circumscribed tumors of the hand and finger. A histopathological analysis is necessary for an accurate diagnosis and appropriate management. The treatment involves complete surgical excision, usually curative, with minimal recurrence.


MRI remains the best tool in the diagnosis of this disease entity however preoperative diagnosis remains a difficult task. A high degree of suspicion is raised when intraoperative findings and preoperative image description become incompatible.Lumbar disc herniation into the dural space is a rare phenomenon of lumbar disc degeneration with an unclear remaining pathogenesis. Intraoperative ultrasonography and histopathological examination of resected specimen help in the diagnosis of intradural disc herniation. Prompt surgery is recommended due to the high incidence of cauda equina syndrome. Histopathological examination showing disc tissue. Increased severity was described for the last 10 days with weakness in his left lower limb, and history of urinary incontinence (overflow) since the last 2 days. The personal history showed no diagnosis of hypertension or diabetes mellitus. No relevant familial or psychosocial history was reported. Performing the straight leg raising test, both legs were found to be at around 60 degrees of possible elevation. The extensor hallucis longus (EHL) and flexor hallucis longus (FHL) force test showed a strength of 3/5 on both sides. Sensory examination showed perineal and anterolateral right foot hypoesthesia. Performed MRI of the lumbo sacral spine revealed diffuse bulging of the disc with a left postero central and sub articular extrusion at L5‐S1 level, as well as thecal compression, and impingement of the left traversing S1 nerve root Figure . A presuOn examination, the Oswestry Disability Index (ODI) was found to be 92%.The patient underwent total laminectomy at the level of L5‐S1. The disc was successfully removed from both sides of the shoulder end of the nerve root. Additionally, foraminotomy was bilaterally performed. Intraoperatively only minimal disc fragments were found over the epidural space. The dorsal dura mater showed tension and bulging at L5‐S1 level Figure . SubsequPost operatively, the patient's left lower back pain and left leg radiculopathy along with the left lower limb weakness and numbness improved significantly. In addition, the patient also showed immediate cessation of urinary incontinence. The follow up at 1 and 3 months of surgery showed, significant improvement in the patient's symptoms with an ODI score of 10 and 6, respectively. The improvement is mirrored in the patient's abiltity to cope with most daily activities after the procedure. No further treatment is indicated apart from advice on lifting, sitting and exercise.3Intradural disc herniation (IDH) is a rare phenomenon, which accounts for only 0.25%–0.35%Males are more often prone to the entity of IDH than females. Average age of onset is between 50 and 60 years. Symptoms of IDH may vary depending on the location of the herniated disc, with cervical IDH patients presenting clinical findings of the Brown Sequard Syndrome.The pathogenesis of IDH remains unclear. Few theories have been stated. Several reports suggest that certain factors such as reduced epidural space, resulting from congenital or iatrogenic narrowing of the spinal canal or adhesions between annulus fibrosus, the posterior longitudinal ligament (PLL) and the ventral dura matter may be responsible for IDH.A preoperative assumption and diagnosis of IDH is difficult. Lesions on imaging might remain unseen or can easily be mistaken for other findings. Definite diagnosis is made only during surgery and not via imaging alone. In this case, the MRI did not indicate the presence of intradural disc fragments, therefore, diagnosis was made intraoperatively.Intraoperative ultrasonography is helpful in detecting foreign bodies in the dura mater.The dissection of the dural sheath in the anterolateral portions usually is a difficult step during surgery. Adhesions between the ventral dural sheath and the PLL are extremely resistant, unable to be separated using a sharpless dissection.4A high degree of suspicion of dural disc herniation is raised when intraoperative findings and preoperative description of image are incompatible. Intraoperative ultrasonography and histopathological examination of resected specimen additionally help in the diagnosis of dural disc herniation. Prompt surgery is recommended because of the high incidence of cauda equina syndrome.Utsav Bhattarai: Conceptualization; writing – original draft; writing – review and editing. Pritam Gurung: Supervision. Janam Shrestha: Writing – review and editing. Sudan Dhakal: Writing – review and editing. Samir Acharya: Writing – review and editing. Basant Pant: Supervision.None.None of the authors have potential conflicts of interest to be disclosed.Ethical approval of the case report is not needed by the local ethical guideline.Written informed consent was obtained from the patient to publish this report in accordance with the journal's patient consent policy.


Trichotillomania is a psychiatric disorder characterized by recurring urges to pulling out hairs, eyelashes, or down in other parts of the body. Trichophagia, which is the urge to ingesting the pulled-out hairs, can cause Rapunzel syndrome, an unusual disorder where gastric trichobezoars can be found in the small intestine. Trichobezoars, amorphous masses composed of undigested food formed by hairs, can obstruct the gastrointestinal tract up to simulating symptoms typical of bowel obstruction. Rapunzel syndrome, named after Grimm’s tale, may cause death, especially in the pediatric population, being it seldom over the age of 6; moreover, developing countries and environmental and familiar issues are listed as uncertain risk factors. The present case report deals with the death of a 4-year-old female occurred after lunch and following a series of vomit events; while no traumatic or pathological findings were revealed at the external examination, the autopsy revealed three large trichobezoars localized in the stomach and the small intestine. Despite death was due to gastrointestinal obstruction for multiple trichobezoars and collateral bronchoaspiration of dietary material, histological findings were totally non-specific, meaning that it is sometimes difficult to conclude that death is related to the primary pathological condition. Trichophagia is described as the urge to repeatedly ingesting hair, generally being one’s own or, anecdotally, other people’s hair, animal fur, hairs found on combs, eyelashes found on tweezers, etc. –3.Hair ingestion, as much as other not edible fibers, can cause complications known as trichobezoars, which are undigested hair masses localized in the gastrointestinal tract, frequently found in children and adolescents .Trichobezoars can obstruct the physiological transit of food bolus, thus causing typical symptoms of bowel obstruction, but they can also result in an atypical gastrointestinal syndrome . This meDiagnosis is frequently difficult to make since the syndrome and its manifestations are sporadic . RapunzeHerein, we present the case of a 4-year-old female, whose death occurred after lunch and following a series of vomit events. The autopsy revealed three large trichobezoars localized in the stomach and in the small intestine; the external examination did not reveal any sign of suspected negligence by the parents.., no imaging techniques were implemented) and thus discharged. Blood test showed neutrophil leukocytosis . During the short hospitalization, a psychological examination was performed, which resulted in highlighting a symbiotic relationship between the mother and the child, an extremely apprehensive behavior by the mother, who appeared extremely anxious; the neuropsychiatrist noted that when the mother did not intervene in the conversation, the child seemed to be more responsive and less whiny. Further psychological examinations were suggested, in order to prevent any kind of developmental delay, but not performed due to the patient’s scarce compliance. After being discharged, issues in eating and drinking became rarer, though associated with nausea; however, since the symptoms were blurred, patient parents did not seek other medical advice. Two months later, after eating pasta, the child vomited and lost consciousness. Her mother called for emergency services; however, the little girl was already showing clear signs of cyanosis, advanced life support protocols were useless, and she died in a few minutes. A forensic autopsy was performed; the scarce circumstantial evidence was the following: (i) the patient was an only child, and (ii) parents noticed that, sometimes, their daughter ingested her own hair, but, underestimating the behavior, decided for having it cut more frequently.A 4-year-old child was admitted because of vomiting and abdominal pain lasting for 2 days. The mother reported that her daughter had been having issues both in eating and in drinking; the patient appeared dehydrated. According to the mother, it was the first and only episode of these symptoms. The physical examination resulted negative; no pain was reported in abdomen palpation. During the following 5 days, she was clinically diagnosed with metabolic acidosis  resulted damaged. The girl had fair, 5-cm-long hair, without alopecia areas; eyelashes resulted normally constituted. The body presented some signs of needle injections in the right arm and in the left leg, ECG electrodes, and visible marks of cardiac massage.Anus appeared patulous; however, when inspecting it together with genitalia no traumatic signs were found, similarly to other body parts.The autopsy was performed almost 2 days after the death (46 h); after a 24-h-long period of observation, as required by local law requirements, the body was stored in the morgue for 22 h at an ambient temperature of −10 °C.The neurocranium, the meninges, and the brain had no macroscopic alterations. The pericardial sac showed hemorrhagic infiltration on its surface due to resuscitation attempts. The heart weight was 85 g and measured 7 × 6 × 4 cm in its diameters, without any sign of malformation or other pathological conditions. The lungs had an increased texture.The abdominal cavity was filled with about 600 ml of brown free fluid Fig. . AbdominAt the inspection and palpation, the gastrointestinal system, the stomach, and the intestine were hard, as in fecal findings. After opening the stomach, a solid, large, brown mass, composed of entangled hairs arose: the trichobezoar was covered by a brown fluid and occupied almost the whole gastric lumen. It weighed 307 g and measured 15 × 11 × 5 cm Fig. .Fig. 2GaOther two similar masses were found opening the intestinal loops: the one at the ileo-jejunum section weighed 72 g and measured 9 × 8 × 2 cm, while the one at the ascending and transverse colon weighed 46 g and measured 6 × 4 × 1.5 cm.After cutting the trichobezoar found in the stomach along its short axis, it showed a sort of internal nucleus composed of amorphous, friable, white material, 7.5 cm in diameter, and an external layer of partially digested and entangled hairs. The other two trichobezoars, once dissected, similarly presented a wall formed by hairs and some focal, whitish spots of amorphous material in the center Fig. .Fig. 3PaAt the microscopic examination, the lungs showed an intense and widespread congestion; dietary material was found in the alveolar spaces and in the bronchial lumen. The pancreas revealed an interstitial inflammatory lymphocytic infiltration consistent with chronic pancreatitis extending to the duodenal wall as chronic duodenitis. The stomach and the small intestine showed chronic lymphocytic inflammation of the mucosa and the submucosa Fig. a, b. MicTrichobezoars’ microscopic examination confirmed the dietary nature of the nucleus, specifying it was made of undigested vegetables mixed to hairs Fig. c, d.At the microscopic examination, the lungs showed an intense and widespread congestion; dietary material was found in the bronchial lumen Fig. .Fig. 5a As mentioned above, this case is about the death of a 4-year-old girl, whose autopsy found a voluminous trichobezoar in the stomach and two smaller, though still remarkable, ones in the small intestine; leading to the diagnosis of Rapunzel syndrome.In general, bezoars are masses formed by undigested or partially digested material, which grow in the gastrointestinal tract thus causing obstruction. The incidence rate of gastrointestinal obstruction events due to bezoars ranges from 0.4 to 4% . When beWomen under the age of 20 are the reference population of trichophagia and frequently they have been already diagnosed with other psychiatric disorders, such as anxiety, depression, or ADHD ; trichopSeveral case reports are no longer available, or they report unverifiable data, whilst it is well documented the case of an almost 4-year-old girl, affected by fetal alcohol syndrome, who died because of bronchopneumonia and malnutrition due to a 210 g trichobezoar in the stomach: social services were alerted for suspicion of neglect . AnotherIn our case, the child had been showing gastro-intestinal symptoms for 2 months, in association with nausea and dysphagia/anorexia. Trichophagia was pointed out by the parents, who decided to cut their daughter’s hair more frequently, without seeking any other medical and/or neuropsychiatric evaluation. The family’s socio-economic status was also not known; however, it was said it belongs to the middle class. Since the autopsy was requested by the district attorney, no talking with the parents was allowed, as it would be expected in any other diagnostic process: the scarce information was retrieved from the investigation dossier. The psychiatric evaluation performed during the short hospitalization occurred 2 months prior to the death was deemed inconclusive. Although parental abuse was the main suspicion of the district attorney, the external examination did not reveal any compatible sign; the size of the trichobezoars, though, does imply a chronic hair ingestion which was not confirmed. At the external examination, hair was measured (about 5 cm), which was coherent with the depositions given to the judiciary police.From a clinical point of view, even if Rapunzel syndrome may cause intestinal perforation and consequently death , it entaAs far as the autopsic findings are concerned, neither signs of bowel ischemia nor acute peritonitis were found, given that death occurred almost immediately because of the trichobezoars size; however, the lymphocytic inflammatory infiltration was intense and widespread not only in the gastrointestinal tract but also in the pancreas and in the myocardium. Ultimately, in the absence of other histological findings, the sum of the autopsic findings resulted in declaring the death related to the obstruction due to the three trichobezoars.The bronchoaspiration identified during the histological examination was related to the vomiting that preceded the death, as testified by the mother. In fact, vomiting is frequently described as a symptom for the presence of trichobezoars , althougThe conformation of the trichobezoar found in the stomach was intriguing, given its internal core, amorphous and composed of undigested dietary material, and its cortex formed by hairs. A similar structure was observed in the aforementioned two trichobezoars discovered in the small intestine. It is possible that hairs, once eaten, aggregated around a core of undigested food, such as vegetables which remain longer in the stomach. The other two formations may have grown autonomously in the small intestine or be the detached tails of the gastric trichobezoar, as other Authors suggested .In our case, we hypothesized a combined gastric and duodenal obstruction: since no signs of peritonitis were found, nor intestinal perforation, and given the death speed, our theory was that an endoluminal hypertension occurred first, and an abdominal one later, which compromised the venous and the inferior vena cava return, finally producing a compartmental syndrome. Moreover, in our hypothesis, another mechanism contributed to the physiology of the death, which was a water-electrolyte imbalance; upon this, the episode of vomit right before the death could be another stress factor.Therefore, our case can be described as an extremely rare death in a young person due to trichobezoars in Rapunzel syndrome. In fact, whereas the autopsic findings are incontrovertible, the intermittent and feeble symptomatology could not clinically prefigure the presence of the undigested hair mixed to food in the gastrointestinal tract. Moreover, the importance of a careful evaluation of non-specific symptoms in the pediatric population is here underlined since no imaging was performed during the brief hospitalization, despite the probable presence of the trichobezoars in the gastrointestinal tract even at that time.Given the results of our examination and the literature findings, Rapunzel syndrome is a clinical condition which is hard to diagnose in the pediatric population. At autopsy, findings suggest that large trichobezoars can rapidly cause death without manifesting any typical symptom even for a long time. Histological findings can be non-specific as well—as in this case, there were neither gross signs of acute peritonitis nor intestinal perforation—meaning that death, beyond being quick, is also difficult to relate to the primary disorder.Rapunzel syndrome is a complication of trichophagia that can be lethal in children.Symptoms of Rapunzel syndrome can be non-specific and vague, causing death as the first sign.Rapunzel syndrome may occur in unexpected environmental contexts, even in the middle and high classes.Actual cause of death can be unclear even at the histological examination.


Hydatid disease is a worldwide public health problem, especially in endemic countries, caused by the larval stage of Echinococcus granulosis. The pancreatic location of this disease is exceptional, representing only 1% of all possible locations, making this a widely misdiagnosed entity. We report a case of a 42-year-old man with a history of alcoholism and recurring abdominal pain, who presented to the emergency department with acute pancreatitis revealing a hydatid pancreatic cyst mimicking as a pseudocyst of the pancreas. The diagnosis was established using computed tomography and magnetic resonance imaging. Pancreatic hydatid cysts are an exceptional form of hydatid disease, and isolated involvement has rarely been reported We describe a case of a 42-year-old man admitted to the emergency department with typical pancreatitis symptoms, for which the initial CT scan reasonably evoked the differential diagnosis of a pseudocyst given the history of alcoholism and recurring abdominal pain, and the absence of typical radiological hydatid cyst characteristics. However, a subsequent MR imaging confirmed the diagnosis of a hydatid pancreatic cyst. This case report highlights the importance of considering this diagnosis in case of cystic lesions of the pancreas, especially in endemic areas.A 42-year-old male, with a history of alcoholism and recurring abdominal pain, originating from an endemic area of hydatid disease in North Africa, was admitted to the emergency department, for an acute onset of severe central epigastric pain radiating through the back and exacerbated in supine position, associated to nausea for 3 days. Moreover, he did not have any significant prior medical history. Clinical examination revealed a palpable mass occupying the left hypochondrium and the epigastrum with firm consistency. A standard lab report was ordered revealing elevated lipasemia of more than 20 times the normal range, a moderate cytolysis, high white blood count and CRP levels, with a mildly altered renal function . HydatidHydatidosis is secondary to the development of the larval form of Echinococcus granulosus in humans. Hydatid cysts can touch any organ or structure of the body. However, the pancreatic location has a prevalence of less than 1% Isolated hydatid cyst of the pancreas is a very rare entity. Our intention in presenting this case report is to highlight the possibility of a pancreatic location of a hydatid infection masquerading as a pseudocyst or a cystic neoplasm. In patients from endemic areas a pancreatic hydatid cyst should always be considered as a differential diagnosis.Hydatid cyst of the pancreas is an extremely rare and widely misdiagnosed entity, especially in case of an isolated cystic lesion simulating a pseudocyst or cystic neoplasm. Hydatid cyst should be considered as one of the differential diagnosis in pancreatic cystic lesions, as well as other organs, especially in patients originating from endemic countries. The management of this entity includes surgery, percutaneous techniques (PAIR) and antiparasitic drug therapy.Written informed consent for publication of this case study was obtained from the patient.