Background: Haglund’s syndrome (HS) is a painful condition that is caused by an exostosis of the posterior superior part of the calcaneus coupled with Achilles tendinopathy and retrocalcaneal bursitis. Both for the proper musculoskeletal assessment and for the differential diagnosis process of possible concurrent diseases deriving from other anatomical areas, the diagnosis of HS is still a challenge. Case Presentation: A 41-year-old male amateur runner was diagnosed and treated for low back pain and referred leg pain by his general practitioner. Due to ineffective results, he self-presented to a physical therapist (PT) with intense right heel pain, radiating up to the leg and to the lumbopelvic region. Results: The PT’s examination and interview relating to the sports activities led to the correct diagnosis and a proper orthopedic referral. At the one-year follow-up, the patient reported regular pain-free marathon running. Discussion: This case report highlights the central role of PTs working in direct access environments as primary care healthcare professionals for the management of musculoskeletal diseases, and their abilities in identifying patients with suspected pathologic conditions that may need referral for imaging, medical assessment or surgical intervention. Running is one of the most popular sports for adults  and imprA 41-year-old male amateur runner, industrial worker, presented to his GP complaining of deep and burning pain wrapping all his right lower limb, from the calcaneus to the lumbopelvic region , were not found during the examination of the back. Therefore, the PT decided to perform a thorough examination of the lower limb.The latter revealed a prominent tubercle on the posterior superior and slightly lateral aspect of the heel. This tubercle was red, swollen and highly painful to palpation. Functional and neuromuscular tests such as heel walk and walk on toes were both possible, but the heel walk exacerbated the familiar and intense pain at the right heel (NPRS 7/10). Moreover, bilateral heel rise tests were painful (NPRS 5/10) after 20 repetitions , and sinIn the first month after surgery, the PT’s program (3 sessions/week) was focused on restoring ankle and foot mobility, reducing pain and swelling, and walking retraining. Passive and active ankle joint mobilizations were performed to restore the arthrokinematic movements. Calf and lower back pain were successful treated with manual digital pressure of the myofascial pain points in the soleus, fibularis longus, and biceps femoris. Plantar flexors, knee flexors, gluteus medius and maximus muscles were strengthened with isometric exercises, and then with elastic resistance. The pain-free walking was achieved through balance exercises, aerobic reconditioning with an exercise bike, and neuromuscular exercises after one month from surgery.From the second postsurgical month, a progressive training of the lower limb strength and running was undertaken, with a scheduling of 2 sessions/week till the third month). The subject was evaluated on an instrumented treadmill  at a running pace of 6.5 min/km, and foot strike pattern was determined analyzing slow-motion video recording. The running benchmarks were analyzed using the OPTO-JUMP NEXT software . For a detailed description of the outcome measures see To the best of the authors’ knowledge, this is the first case report that compares the management of a common MSK disease like HS performed by a PT in DA, with the usual GP-led care. Despite the major clinical signs and symptoms of HS being clearly reported in the literature , and itsDifferential diagnosis is a key element of the PT’s clinical reasoning in DA settings, even if the patients have already been assessed or treated by other healthcare professionals. In fact, applying a thorough diagnostic process, PTs in DA are able to carry out a comprehensive clinical evaluation of different MSK afflictions and to screen doubtful conditions that could indicate an appropriate medical referral.HS is a common MSK condition that every trained healthcare professional should be able to recognize and properly manage.DA to a PT is a safe, cost-effective and time-saving practice for patients with MSK disorders.A thorough physical examination within the differential diagnosis process is mandatory in case of patients with MSK disorders.


Osteolytic lesions of the jaw are not uncommon. Such lesions usually arise from local pathologies, but some have systemic backgrounds. We describe a 12-year-old girl who presented with an asymptomatic left mandibular swelling. The bony swelling was corresponding to a radiolucent lesion in the left premolar/molar region. This lesion could have represented an inflammatory and developmental odontogenic jaw cyst, giant cell lesion, and odontogenic tumor. However, the workup investigations revealed secondary hyperparathyroidism due to vitamin D deficiency. A vitamin D replacement was initiated with a single I.M. injection of 300,000 I.U followed by 10,000 I.U orally, weekly. Six weeks later, her Vitamin D and parathyroid hormone were normalized, and she showed significant clinical and radiological improvement of the jaw lesion. At 18 months, follow-up the panoramic image revealed complete resolution of the radiolucency and stable normal parathyroid hormone and vitamin D levels. In conclusion, Jaw bone lesions can develop secondary to hyperparathyroidism due to vitamin D deficiency, and this should be ruled out before any surgical intervention. Treatment of such lesions lies in the correction of parathyroid excess with a careful and systematic approach. This may prevent unnecessary surgical intervention in such patients. Vitamin D deficiency is a health burden that has caused concerns worldwide over the past decades due to implications on bone biology, multiple sclerosis, diabetes, depression, and cardiovascular disease . DespiteRadiolucent lesions of the jaws are not uncommon, often detected in conventional radiography, and could be odontogenic or non-odontogenic . The proA 12-year-old female orthodontist was referred to Dr. Gaballah to assess and treat the radiolucent lesion in the mandibular left premolar-molar area. The lesion was identified during routine workups for orthodontic treatment to address anterior teeth' crowding . ClinicaPanoramic imaging showed a well-defined unilocular ovoid radiolucent lesion with a sclerotic rim extending from the distal aspect of the mandibular second premolar to the distal surface of the distal root of the mandibular first molar . There wVitamin D deficiency was treated as indicated by the endocrinologist before any surgery to correct hyperparathyroidism. As a result, the surgical biopsy was postponed. Treatment was begun correcting the hypovitaminosis with a single injection of Vitamin D3 300,000 I.U complemented by 10,000 I.U orally once a week. Six weeks post-intervention, a repeat vitamin D test showed a vitamin D level of 85 nmol/L with PTH lowered to 38 and serum calcium 2.4 mmol/L suggesting a substantial improvement in parathyroid function and calcium homeostasis. Parents also observed an increase in the patient's physical activity and cognitive performance. The same visit's panoramic follow-up showed some signs of bone formation in the affected region .The patient was kept on a maintenance dose of 10000 D3 I.U weekly. Based on progress in clinical, laboratory testing, and imaging of the jaw lesions , the parDespite the favorable latitude and abundance of sunlight in the Gulf region, the UAE population remains vitamin D deficient. Due to excessive heat and humidity in the relatively long summer season, increased sun avoidance, particularly in females, is noticed among the inhabitants –10. ContGiven the clinical and laboratory findings, the biopsy was not performed. We are conscious of the limitation that we were unable to characterize the lesion histopathologically. However, the significant clinical and radiological resolution of the lesion and correction of the secondary hyperparathyroidism bears evidence to the preliminary diagnosis of a brown tumor of secondary hyperparathyroidism caused by vitamin D deficiency. The biopsy was considered on the initial surgical plan. However, discovering the abnormal bone homeostasis and the improvement noted at the early stage of the Vitamin D replacement make the family deny that option. Keeping in mind, potential complications of surgical biopsy may include damage to surrounding tissue, excessive bleeding, bone fractures, and infection. However, in most cases, biopsy benefit generally overrides the risks in diagnosis and treatment planning. Brown tumor, also known as osteitis fibrosa cystica, is a benign, non-neoplastic, giant cell lesion that arises from excessive PTH levels due to primary or secondary hyperparathyroidism. They are preferentially found in the long bone, ribs, clavicle, pelvic girdle, and mandible. However, They can occur in any skeletal region in the body .In contrast, this manuscript is written a report documented a potential cause of a brown tumor of the left proximal femur in a 16-year-old female treated with calcium vitamin D supplementation . Complet


Epignathus is a rare, benign, congenital teratoma of the hard palate with an estimated incidence of 1 in 200,000 live births. Epignathus frequently leads to a high mortality (80–100%) due to airway obstruction in the neonatal period.We report a case of successful management of a rare oropharyngeal teratoma in a African newborn girl who was referred to our institute with a large protruding intraoral mass, combined with cleft palate, causing some respiratory compromise. The palatal mass was removed on the fifth day after birth, and a palatoplasty performed on day 30.Epignathus is a life-threatening condition at the time of delivery. Appropriate management begins with securing the airway, followed by complete resection of the tumor. Epignathus is a rare teratoma that arises from the oral cavity, most commonly from the palate. Congenital tumors of the oral cavity frequently affect normal development of adjacent structures, and tumors developing during morphogenesis of the palate are commonly associated with a cleft palate .In general, teratomas are uncommon malformations that arise in the three embryonic germ layers . They are felt to be the result of pluripotent cells that did not complete migration from allantois to the genital ridge during the fourth and fifth weeks of gestation .The physiologic impact of a teratoma is a result of its location and size. If located in the upper aerodigestive tract, they are high risk, frequently causing life-threatening complications from compromise of the airway. Because of this, they have a high mortality rate, and prenatal diagnosis may allow for better preparation to help optimize care at the time of birth –5.Some teratomas extend beyond the mouth, and these lesions can be associated with mid-face defects including cleft lip and palate. There are reports of palatal teratomas extending beyond the oral and nasal cavity and even extending into the cranial cavity with resultant destruction of brain tissue , 6.We have reported a case of congenital teratoma arising from the hard palate in a newborn girl. This report aims to show that diagnosis and successful surgical treatment of such a rare case are possible in a low-resource environment. It therefore contributes to the existing literature on management of giant epignathus.A 1-day-old female African newborn weighing 3500 g was referred for management of a large oral mass that protruded from the mouth and caused maxillary protrusion and flattening of the nose. The patient was having difficulty breathing . They can be solid or cystic and can be benign or malignant, but the majority are benign. dramnios –9.Teratomas occur in approximately 1 in 4000 births, and there is a female predilection. They are most commonly located in the sacrococcygeal area but can present in many areas of the body. Epignathus is very rare, occurring in only 1/200,000 live births. They account for only 2% of teratomas and almost always present in neonates. Most are benign, although malignancies have been reported . Althouget al., giant epignathus can lead to death during the neonatal period due to airway obstruction or can lead to malnutrition due to the inability of oral feeding. The newborn must therefore benefit from oxygenation and feeding by nasogastric tube while awaiting surgery. Complete surgical excision is the definitive treatment [As reported by Kumar reatment , 10.Dermoid, also called “hairy polyps”, containing epidermal and mesodermal elements are the most frequent;Teratoid, composed of ectodermal, mesodermal and endodermal elements, are very poorly differentiated;True teratoma, also contain the three embryonic germ layers, but differentiated in recognizable structures such as cartilage, hair, teeth, etc.Epignathus, are highly differentiated, rare, and have a high mortality rate due to their location [location .Teratomas are often classified into four types:et al. [Type 1 (skin and fatty tissue derived from two germ layers);Type 2 ; andType 3 (parasitic twins with well-differentiated organs and limbs).Yoshimura et al.  proposedHistopathologic examination in this case revealed an epignathic teratoma containing mostly epidermal tissue. It was a type I teratoma by the Yoshimura classification system.Oral teratomas can be associated with mechanical craniofacial abnormalities, the most common being cleft palate .When a tumor develops before the eighth week, it can interfere with the normal closure of the secondary palate in the midline. It can also hinder the confluence of the two halves of the nose or tongue, resulting in a bifid nose or tongue. It can also contribute to abnormal maxillofacial development with micrognathia and ogival palate . In our Epignathus is a rare oral cavity teratoma commonly presenting with symptoms requiring urgent management of respiratory distress.As in this case, large epignathus can cause some respiratory compromise, and it is commonly associated with cleft palate, but other anomalies may occur.Urgent treatment consists of airway management and nasogastric feeding tube. The definitive treatment requires complete resection of the mass and correction of associated anomalies.


A Meckel's diverticulum is a rare but known cause of an acute abdomen and can often be confused for acute appendicitis on physical examination. It is caused by an incomplete closure of the omphalomesenteric duct. It is present in 2% of the population and only 2% of those patients are symptomatic.This is the case of a sixty-four-year-old male presented to the surgical clinic at request of his primary care physician with concern for acute appendicitis. The patient had a CT A/P with IV contrast performed two days prior to his office visit for the same pain which was non-diagnostic. The patient was taken to the operating room and found to have Meckel's Diverticulitis which was managed by laparoscopic hand-assisted small bowel resection and anastomosis. The patient had an uncomplicated postoperative course. Pathology demonstrated ulcerated gastric mucosa and pancreatic tissue.Symptomatic Meckel's diverticulum is managed with small bowel resection versus diverticulectomy based on characteristics of the diverticulum. The most common type of ectopic tissue is gastric followed by pancreatic. It is rare to find both types of tissue together.This case describes an unusual case of a rare acute surgical pathology with non-diagnostic imaging and labs. This case also describes an exceedingly rare histopathology of a Meckel's Diverticulum with the presence of both ectopic gastric and pancreatic tissues. •Meckel's Diverticulum in adults commonly presents with inflammation, small bowel obstruction and perforation.•The recommendation for treatment of a symptomatic Meckel's diverticulum is surgical resection.•Meckel's Diverticulum contain ectopic tissue, most commonly gastric and pancreatic tissue but rarely together. It has been estimated to be present in two to 4% of the population . It was 2This case report presents a sixty-four-year-old Caucasian male with a three-month history of intermittent right upper quadrant and epigastric abdominal pain. The patient previously underwent an extensive negative work-up for biliary pathology in addition to multiple emergency room visits without any diagnoses. The patient went to his primary care provider two days prior to surgical evaluation where he was noted to have a temperature of 100.4 °F (38.0 °C) and computed tomography (CT) of the abdomen and pelvis with IV contrast was ordered due to concern for appendicitis. There was no acute pathology identified on this scan. The patient has a past medical history of coronary artery disease, hypertension, type 2 diabetes mellitus, obstructive sleep apnea, and GERD. Past surgical history was significant for a prior umbilical hernia repair with mesh. The patient had no known allergies, no significant family history and no significant social history. Due to concern for acute appendicitis the patient was referred to the surgical clinic for further evaluation.At the time of surgical evaluation, the patient reported a new, three-day onset of localized right lower quadrant abdominal pain. Review of systems revealed associated nausea and anorexia without vomiting, change in bowel habits, or rectal bleeding. On physical examination, the patient's vital signs were within normal limits. The patient appeared mildly uncomfortable but in no acute distress. His abdomen was obese with tenderness to palpation in the right lower quadrant as well as localized right lower quadrant percussion tenderness, rebound tenderness and a positive McBurney's point. Laboratory work-up was performed revealing a normal white blood cell count of 5.6 bil/L, electrolytes within normal limits, and a mildly elevated C-reactive protein level at 45 mg/L. The patient's CT scan had been performed at an outside facility and was unable to be personally reviewed but the report was available which reported no acute or abnormal intra-abdominal pathology.Based on the patient's physical exam and elevated CRP level decision was made to take the patient to the operating room for diagnostic laparoscopy with clinical concern for appendicitis. Other diagnoses in our different diagnosis were Meckel's Diverticulitis, inflammatory bowel disease, diverticulitis, perforated viscus, or an intra-abdominal malignancy. The patient received antibiotics and subcutaneous heparin preoperatively. The abdomen was sterilized using Chloroprep solution and was pneumoperitoneum was created using the Veress needle technique. Upon entry the omentum was noted to be in the right lower quadrant and inflamed in appearance. The appendix was identified and found to be normal in appearance. The rest of the abdomen was visually inspected with no immediate obvious pathology. Knowing the appendix was normally the decision was made to inspect the small intestine and remainder of abdomen to rule out other acute pathology. There was no evidence of inguinal or abdominal wall hernia. On inspecting the small intestine an inflammatory mass was identified with omentum tightly adherent to it located approximately 2 ft from the ileocecal valve. This mass was in the right lower quadrant of the abdomen. The omentum was carefully dissected free and the intestine closely inspected with an inflamed Meckel's diverticulum identified. The remaining small intestine was run with no further pathology identified. Based on a wide base and short length of the diverticulum the decision was made to perform a small bowel resection with primary anastomosis. The midline port was extended and a gel port was inserted. The Meckel's diverticulum was pulled through the gel port and an external bowel resection with primary stapled side to side anastomosis was performed.Intraoperative Imaging: The patient recovered well from surgery and was discharged three days postoperatively. Surgical pathology demonstrated benign small bowel with a Meckel's diverticulum with features of acute inflammation and rupture. Further pathological analysis demonstrated an ulcerated benign gastric-type mucosa as well as benign pancreatic tissue within the diverticulum, without evidence of malignancy. In office follow-up the patient was found to have an incisional wound infection of his midline incision which was treated with antibiotics and local wound care. A chronic draining wound developed and five months after his original surgery the wound was re-excised. He healed well afterwards with no further complication.Histological Evaluation: 3The presentation of a Meckel's diverticulum varies between the pediatric and adult populations. Pediatric populations most commonly present with gastrointestinal bleeding and obstruction . In factOnce a patient is diagnosed with symptomatic Meckel's diverticula, the standard of care is definitive surgical intervention. Surgical options are diverticulectomy, wedge resection, or segmental bowel resection. These procedures can be performed either open or laparoscopically depending on the surgeons comfort level. In pediatric surgery literature, the outcomes of laparoscopic vs open surgery have been found to be equivalent in regards to length of stay, postoperative complications, reoperations, and readmissions . KonstanBased on the “Rule of 2's” and in review of literature, approximately 2% of the population has Meckel's diverticulum and 2% of those patients will become symptomatic. Based on this, the majority of Meckel's diverticulum that are present are asymptomatic and surgeons will often encounter them while operating on patients for a different reason. The decision to resect an incidentally discovered diverticula has been debated with several studies reviewing the outcomes of incidentally resected diverticula. In a review by Zani et al., it was found that there was a statistically significant higher postoperative complication rate following resection of an incidentally identified Meckel's . In contThe omphalomesenteric duct is made up of pluripotent tissue that can mature into several different types of tissue in a MD. While the tissue is typically ileal tissue, ectopic tissues can develop within the diverticulum. The most common tissue is gastric tissue which is present in up to twelve to 26% of cases. The second most common type of ectopic tissue is pancreatic tissue, occurring in roughly two to 3% of patients. Other less common types of ectopic tissue are duodenal, colonic, endometrial, and hepatobiliary ,16. The This case report is also unusual in the patient's presentation. The patient had a several month history of right sided abdominal pain, initially worse in the right upper quadrant and epigastrium. The patient was evaluated for biliary pathology and reported a negative workup. Two days prior to presentation to the surgical clinic, the patient had developed worsening right lower quadrant abdominal pain and had a CT of the abdomen and pelvis that was performed with IV contrast that was negative for findings of appendicitis, diverticulitis, or intra-abdominal inflammation. Modern computed tomography imaging is able to detect a broad amount of intra-abdominal pathology so a patient with negative imaging and continued severe abdominal pain can present the physician with a diagnostic dilemma. In the setting of Meckel's diverticulitis, imaging findings will demonstrate a blind-ended tubular structure with wall thickening and enhancement with surrounding fat stranding. These findings can be seen in the case of acute appendicitis so the diagnosis of Meckel's diverticulitis on imaging requires the finding of a normal appendix . It is u4Meckel's Diverticulum is an uncommon presentation of right lower quadrant abdominal pain but should always be part of a physician's differential diagnosis. Physicians tend to rely more on radiographic imaging and laboratory findings and pay less attention to the physical examination to help elucidate a patient's abdominal pathology. This case is unique as the preoperative imaging was normal and raised no suspicion of the underlying acute surgical pathology. The main concerning feature in this patient was the physical examination prompting a diagnostic laparoscopy searching for appendicitis and instead finding acute Meckel's Diverticulitis. As stated by Dr. Charles Mayo in 1933, “Meckel's diverticulum is often suspected, always looked for and rarely found” [Not commissioned, externally peer-reviewed.Beaumont Farmington Hills Department of Medical Education.Not required for case report with no patient identifiers as there is minimal risk to the patient.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Not applicable.Marcos Rosado.Marcos Rosado: Primary author of case report, performed literature review.Thomas Serena: Contributing author and editor of case report.John Pui: Provided pathology slides and commentary, reviewed paper.John Parmely: Primary surgical attending, reviewed paper.None.


Introduction: Many ovarian or adnexal masses have an indeterminate appearance on ultrasound that can raise concerns about cancer. Although magnetic resonance imaging (MRI) has been reported to reliably distinguish between benign and malignant masses, studies evaluating the accuracy of MRI in community-based practice settings are lacking.Methods: Women who underwent MRI to further evaluate an ultrasound-detected adnexal mass in 2016–2017 within a large community-based health system were identified. MRI reports were classified as favoring malignancy, benign disease, or indeterminate, blinded to pathological outcome. With a minimum of 2 years of follow-up, all ovarian cancers and borderline tumors were identified, and the accuracy of MRI assessment was determined.Results: Among 338 women who had MRI to evaluate an adnexal mass, 144 (42.6%) subsequently underwent surgery. MRI favored malignancy in 7 (4.9%) cases, benign disease in 89 (62.2%) cases, and was indeterminate in 48 (33.6%) cases. Of the seven cases in which MRI favored malignancy, two cancers and five benign tumors were found. An additional 10 cases of cancer or borderline tumor were found among women who had MRI reports that were read as indeterminate (n = 6) or that favored benign disease (n = 4). The sensitivity, specificity, positive predictive value, and negative predictive value of an MRI favoring malignancy were 16.7%, 96.2%, 28.5%, and 92.7%, respectively.Discussion: In a large community-based setting, an MRI favoring malignancy was more likely to be associated with benign disease than cancer and identified only 16.7% of true malignant cases. The findings suggest that the ability of MRI to differentiate between benign and malignant adnexal masses in community-based practice settings is currently limited. However, as the ultrasound characteristics of benign and malignant masses overlap, many masses have an indeterminate appearance and can often raise concerns about ovarian cancer. Various strategies and algorithms have been suggested for differentiating between benign and malignant masses, but none have been widely adopted.However, these studies were almost exclusively done in referral-based settings in Europe in which MRI reports were read by a small number of expert radiologists, raising questions of the generalizability of the findings. Consequently, when faced with a woman with an indeterminate adnexal mass, many clinicians are uncertain whether they should obtain an MRI to further evaluate whether the mass represents ovarian cancer.Subjects were members of Kaiser Permanente Northern California (KPNC), a large integrated community-based health care organization that includes 19 medical centers and whose membership diversity reflects that of the surrounding community: 47% Caucasian, 7% African American, 22% Hispanic, 20% Asian or Pacific Islander, and 3% multiracial. The study was approved by the KPNC institutional review board, with a waiver of informed consent.18 In brief, the system classifies adnexal masses seen on ultrasound into four risk categories  based on ultrasound characteristics: category 0 masses comprise simple cysts and are considered benign; category 1 masses are cysts with thin septation or cysts with classic benign features for a hemorrhagical cyst, dermoid tumor, endometrioma, or hydrosalpinx, and are considered probably benign; category 2 masses are primarily cystic with minor solid components (must not be >1 cm with vascular flow) and are considered indeterminate; and category 3 masses have more significant solid components or are completely solid and are considered suspicious or potentially worrisome.A standardized reporting system for ultrasound-detected adnexal masses was implemented in the health system in 2015, enabling electronic identification of women identified as having an abnormal mass on ultrasound.The system additionally flags masses >10 cm in overall size with a designation of “X” to identify them as needing surgical consideration based on size. Patients were identified who had an abnormal adnexal mass reported on ultrasound in 2016–2017 followed by pelvic MRI within 6 months. Subjects who underwent MRI for reasons unrelated to the adnexal mass, or who had a diagnosis of ovarian cancer or metastatic cancer of any kind before MRI, were excluded.The study period was selected as the time period when the standardized ultrasound reporting system was in place to facilitate identification of patients, while providing at least 2 years of follow-up to assess cancer outcomes. Patient age, race/ethnicity, and body mass index (BMI) at the time of the initial ultrasound were extracted from electronic databases. Within the health care setting, pelvic MRI reports are read by a group of ∼490 radiologists.The MRI report was classified as favoring malignancy, favoring benign disease, or indeterminate by two independent reviewers (R.L. and E.S.), blinded to subsequent outcomes, based on the text of the report using the following rules: if the MRI stated that malignancy was suspected or favored or listed only malignant tumor types in a differential, the read was classified as favoring malignancy; if the report stated that benignity was suspected or favored, or listed only benign tumor types as possibilities, then the MRI read was classified as favoring benign disease; if the MRI did not indicate that either benignity or malignancy was more favored, or listed both malignant and benign tumor types in a differential, then the MRI read was considered to be indeterminate.All surgical procedures involving the adnexa, resulting pathology, and diagnoses of ovarian or fallopian tube cancer (referred to jointly in this report as “ovarian cancers”) occurring up to December 31, 2019, were identified using electronic databases and the institution's cancer registry, confirmed by manual review. For analysis, “malignancy” was defined as either ovarian cancer or borderline tumor diagnosis.The sensitivity, specificity, positive predictive value (PPV), and negative predictive value (NPV) of MRI for malignancy were determined when considering a “positive” MRI to be a report that favored malignancy, as well as when considering a “positive” MRI to be a report that either favored malignancy or was indeterminate. This was calculated for the cohort of women who had surgical pathology outcomes as well as for the entire cohort of women undergoing MRI, assuming benign disease for women who did not have surgery but remained clinically cancer free for at least 2 years.t-test was used for comparing age at initial ultrasound with a p value of 0.05 for significance.All univariate, bivariate, and multivariate analyses were performed using Statistical Analysis Systems (SAS) version 9.4. Comparisons for categorical variables such as ultrasound risk score of the adnexal mass and indication for ultrasound were performed using the chi-square or Fisher's exact test. The two-sided In 2016–2017, 338 women underwent MRI after ultrasound to further evaluate an adnexal mass. The median time interval between ultrasound and MRI was 2 weeks (interquartile range: 1–3.7 weeks). During the follow-up period , 144 (42.6%) of these women underwent surgery. Surgery was associated with larger masses, a lower BMI, and an abnormal CA 125 tumor marker level .Among the 144 women who had surgery, MRI favored malignancy in 7 (4.9%), benign disease in 89 (62.2%), and was indeterminate in 48 (33.6%) women. Ovarian cancer was found at surgery in 11 (7.7%) patients, borderline tumor in 1 (0.7%) patient, and benign disease in 131 (91.6%) patients. No additional cases of ovarian cancer were diagnosed outside of surgery. Of the 12 patients found to have a cancer or borderline tumor, MRI favored malignancy in 2 cases (16.7%), benign disease in 4 cases (33.3%), and was indeterminate in 6 cases (50%). Conversely, for the seven patients with an MRI favoring malignancy, cancer was found in two (28.6%) patients and benign disease in five (71.4%) patients. A flowchart of MRI results and outcomes is shown in When the MRI interpretation was compared with the standardized ultrasound report for the two cases wherein MRI correctly identified malignancy, the ultrasound was read as suspicious in one case and probably benign in one case. In the latter case, the ultrasound was read as probable hydrosalpinx and the patient was found to have a fallopian tube cancer. Of the nine additional cases of cancer in whom MRI was indeterminate or favored benign disease, the ultrasound was read as suspicious in eight cases and indeterminate in one case .In a large community-based setting in which MRI reports are read by a diverse group of radiologists, MRI did not accurately differentiate between benign and malignant adnexal masses. MRI favored malignancy in only 16.7% of malignant cases, and an MRI report favoring malignancy was more likely to be associated with benign disease.In our study, the overall prevalence of ovarian cancer in the population was low (12 cancers or borderline tumors among 338 women = 3.5%), which is the primary reason the NPV is high despite the relatively poor sensitivity and specificity. If only MRI reports that clearly favor malignancy are considered positive tests, the PPV was 28.5% and the NPV was 92.7%. The question of whether repeated MRI reports would improve accuracy is not addressed by this study but, in general, repeating tests for the same person reduces the error rate to the extent that the error is random (the likelihood of error is independent across consecutive tests).Although it is possible there is some element of random error in reading MRI, perhaps related to the reader, it seems likely that there are also specific patient or tumor factors that affect the performance of MRI. Three of the cancers were granulosa cell tumors that tend to be more solid. In these cases, MRI was read as indeterminate in two cases and favored benign in one case, suggesting that the differentiation between benign and malignant masses by MRI may be particularly challenging for predominantly solid tumors.The only case in which MRI performed better than ultrasound in identifying a cancer was that of a mass read as a large hydrosalpinx on ultrasound that was found to be a fallopian tube cancer. It is possible that there may be specific types of masses such as large hydrosalpinges that are difficult to fully characterize by ultrasound where MRI may provide an advantage. In any case, the main value of repeated tests arises from the comparison of the mass with itself over time, since stability argues strongly against malignancy regardless of imaging modality.10 In all the studies cited, MRI reports were interpreted by one or two expert readers to identify cancer in a tertiary care setting.11–17 Our findings raise questions about how generalizable these results are to community settings in the United States. The degree to which the discrepancy is attributable to differences in expertise versus different systems of reporting is unclear.To our knowledge, this is the first report to describe the accuracy of MRI for differentiating benign from malignant adnexal masses in a U.S. community-based practice setting. In a systematic review of the literature, six studies that evaluated the ability of MRI to differentiate between benign and malignant adnexal masses were identified, all of which were noted in the review to be subject to substantial potential bias.An interesting finding was that lower BMI was associated with surgical intervention. The reasons for this are unclear but suggest that perceived surgical risk played a role in determining clinical management.The main limitation of this study is that in this and nearly all practice settings, MRI interpretations are reported using an unstructured narrative format, which necessitated that the reports be secondarily classified in terms of whether malignancy or benign disease was favored. Although this was done by two independent reviewers blinded to outcome, it is possible that some reports, particularly those classified as indeterminate, were not intended to be interpreted in that way by the reading radiologist. However, even when indeterminate MRI reports were counted as being “positive” for cancer, the sensitivity remained relatively poor at 66.7%, whereas the PPV worsened to only 14.5%.19Furthermore, the need for clinicians to “interpret” MRI reports reflects what occurs in current clinical practice, since the narrative unstructured format of MRI reports results in considerable variability in reporting styles and terminology. It is possible that performance would improve if readers were required to assess and report malignancy risk using a standardized lexicon and scoring system in which readers were required to assess and report malignancy risk according to a standardized system.In addition, since MRI is not routinely obtained after ultrasound, there was undoubtedly selection bias for who underwent MRI toward women for whom there was some additional concern driving further evaluation. However, selection bias toward increased cancer risk among women who get MRI is appropriate from a clinical standpoint and would be expected to enhance, rather than hinder, the performance of MRI for detecting cancer.Study strengths include the diverse community-based nature of the population studied, which increases generalizability to other community-based practice settings. The closed integrated nature of the health system minimizes the likelihood that surgeries or cancer diagnoses were undetected. In addition, cancer diagnoses made at other institutions within California would have been captured by the institution's participation in the California Cancer Registry. Finally, unlike previous studies, we assessed the performance of MRI not only for women who underwent surgery but for the entire cohort of women undergoing MRI as well, since the PPV and NPV of MRI are most relevant to women when determining whether surgery is necessary.In summary, we found that MRI did not accurately differentiate between benign and malignant adnexal masses in a large community-based setting. Further research is needed to understand the factors affecting MRI performance and whether a standardized lexicon and risk stratification scoring system can improve performance in community-based settings.


Cerebrovascular diseases are a leading cause of disability and death worldwide. The definition of stroke etiology is mandatory to predict outcome and guide therapeutic decisions. The diagnosis of pathological processes involving intracranial arteries is especially challenging, and the visualization of intracranial arteries’ vessel walls is not possible with routine imaging techniques. Vessel wall magnetic resonance imaging (VW-MRI) uses high-resolution, multiparametric MRI sequences to directly visualize intracranial arteries walls and their pathological alterations, allowing a better characterization of their pathology. VW-MRI demonstrated a wide range of clinical applications in acute cerebrovascular disease. Above all, it can be of great utility in the differential diagnosis of atherosclerotic and non-atherosclerotic intracranial vasculopathies. Additionally, it can be useful in the risk stratification of intracranial atherosclerotic lesions and to assess the risk of rupture of intracranial aneurysms. Recent advances in MRI technology made it more available, but larger studies are still needed to maximize its use in daily clinical practice. Cerebrovascular diseases are a leading cause of disability and death worldwide. The definition of stroke etiology is mandatory to predict the prognosis and recurrence risk and guide therapeutic decisions ,2. The pVW-MRI has demonstrated its utility in a wide range of clinical situations, including differential diagnosis of intracranial vasculopathies, identification and evaluation of intracranial atherosclerotic disease and also risk stratification in patients with intracranial aneurysms ,8. When Therefore, VW-MRI has become an essential tool for clinicians in the field of cerebrovascular diseases, and continuous technological advances have made it more and more available and reliable for clinical applications.In this paper we will review the technical basis and the main clinical application of VW-MRI in cerebrovascular disease.VW-MRI requires sequences with multiple signal weightings, multiplanar 2D acquisitions or 3D acquisitions, high spatial resolution (HR) and suppression of luminal blood signal . AdditioMulti-contrast VW-MRI is needed for the differential diagnosis of vasculopathies and in the evaluation of plaque composition . TherefoThe use of 3D sequences with MPR reduces the overall MR examination time, avoiding multiple 2D acquisitions in different planes . On the In VW-MRI blood suppression techniques are needed to obtain black blood (BB) MR sequences with null signal within the vessel lumen and to avoid flow artifacts that may mimic vessel wall abnormalities. There are several methods to suppress the blood signal: spin echo sequences with a spatial pre-saturation band exploit the movement of blood spin ; double In VW-MRI the CSF suppression techniques are useful for a proper outer wall boundary evaluation and are particularly important when studying the peripheral branches of intracranial arteries . DelayedAtherosclerosis of cervical and intracranial arteries is a common cause of ischemic stroke ,23. The This section summarizes the role of VW-MRI for intra and extracranial vessels study.The use of conventional imaging for the recognition and phenotyping of the atheromatic plaque and stenosis quantification in intracranial arteries is hampered by the small vessel size .The VW-MRI features of the atheromatic plaque are the presence of focal, eccentric wall thickening, with hyperintensity on T2W sequences and wall enhancement on T1W postcontrast sequences . AlthougMoreover, VW-MRI improves the quantification of stenosis severity, with a similar performance when compared to digital subtraction angiography (DSA), which remains the gold standard technique ,33, and Plaque enhancement on T1W postcontrast sequences is a hallmark of symptomatic plaque and reflects intralesional inflammation and neovascularization . The enhSymptomatic plaques seem thicker than asymptomatic ones and have a higher degree of surface irregularity ,36. AnotA further marker of high-risk plaque is the IPH. Its VW-MRI features are mostly derived from extracranial studies and reflect the oxidative state of hemoglobin. Acute IPH is hyperintense on T1W sequences and it is defined as an intraplaque area of high signal, with intensity greater than 150% of the signal of adjacent muscles  or adjacAlthough the greater dimension of arteries in the extracranial district makes easier the recognition of atheromatic plaque and its features with traditional techniques, the role of VW-MRI is relevant likewise to explore plaque vulnerability .VW-MRI displays plaques’ surface and distinguishes three types of plaque: regular, irregular, and ulcerated (with cavities depth > 1 mm), which is strongly linked with risk of thromboembolism. Its sensitivity is similar to CTA , and it The main strength of VW-MRI in this context is the proper detection of different plaque components . It is tNeovascularization and inflammation are markers of plaque activity that correlate with the degree of enhancement .Most of the available evidence derives from anterior circulation imaging, whereas the role of VW-MRI in posterior circulation atherosclerosis remains poorly defined.To summarize, VW-MRI may provide additional diagnostic value in atheromatic plaque characterization and stratification of stroke risk in clinical practice, and it is a valuable diagnostic aid in patients with cryptogenic stroke and suspected intracranial atherosclerotic disease (ICAD). In Fakih et al. contribution, 7T VW-MRI allowed the etiology of stroke in 28/34 (82.4%) patients with cryptogenic stroke and clinically suspected intracranial vasculopathy to be defined, 89.3% of whom were diagnosed with ICAD .Central nervous system (CNS) vasculitis is a rare cause of stroke, affecting younger patients and also children . VasculiDiagnosis of CNS vasculitis is challenging, particularly in PACNS, and DSA and brain biopsy have a very low concordance, as DSA is positive in 33% of biopsy confirmed cases, and biopsy was positive in 8% of angiogram confirmed cases . MRI shoThe hallmark of CNS vasculitis on VW-MRI is concentric vivid vessel wall enhancement (VWE) of intracranial arteries, often multifocal, with variable associated luminal stenosis and wall thickening ,12. In aBeside PACNS, VW-MRI can depict inflammatory changes in a wide range of secondary vasculitis, including radiation-induced and those associated with infectious disease as the human immunodeficiency virus (HIV), syphilis and VZV ,52,53. PAnother application of VW-MRI is to target brain biopsy when considered essential for diagnosis. In this scenario it can increase sensitivity to 89% from a baseline performance as low as 36% detected in previous studies . Some auMain VW-MRI findings in CNS vasculitis compared and other intracranial vasculopathies are summarized in In giant cells arteritis (GCA), VW-MRI allows direct identification of the inflammatory changes in frontal and parietal branches of the superficial temporal artery and of the occipital artery. Presence of mural thickening (>0.6 mm) and significant mural enhancement confirm the diagnosis . Using 3In CNS vasculitis VW-MRI can also be used to follow-up patient during immunosuppressive therapy. In a prospective study, VWE regressed in seven of nine PACNS patients after treatment, at a median follow-up period of 8 months, with three of these who also showed regression of vessel stenosis . PersistIntracranial artery dissections (IAD) are an uncommon cause of ischemic stroke and subarachnoid hemorrhage (SAH). They frequently affect young patients without traditional vascular risk factors and in some patients can be the manifestation of connective tissue disease . IAD invThe main diagnostic radiological findings of IAD on luminal imaging include fusiform or irregular aneurysmal dilation at a non-branching site, long irregular stenosis associated with double lumen, rapid morphologic change on repeat imaging, focal stenosis and dilation (“string and pearl sign”) or arterial occlusion with recanalization at a non-branching site with fusiform or irregular aneurysmal dilation . DSA hasPathognomonic features of IAD are intramural hematoma, intimal flap and double lumen, and all can be identified using VW-MRI . In a reIntramural hematoma appears as an eccentric focal thickening of the arterial wall, with a crescent shape. The signal varies depending on the temporal stage. It becomes spontaneously hyperintense on T1-weighted sequences 48–72 h after onset for the presence of methemoglobin. And its recognition is higher in the late subacute stage of dissection than in acute and early subacute stages.  T2-weighAneurysmal dilatation of the outer wall can occur and should be evaluated measuring the diameter of the vessel at the perpendicular plane, at the level of the lesion.  Its evalDirect visualization of the intimal flap and/or double lumen is the most significant finding but the least frequently detectable. The intimal flap is visualized as a layer crossing the arterial lumen; it can be seen as a linear layer of low signal intensity in the context of a high signal hematoma in T1W pre-contrast sequences, as a linear enhancing layer on post-contrast T1 images or as a curvilinear hyperintensity in T2W sequences ,71.In post contrast T1 weighted sequences, focal enhancement of the arterial wall can be present in IAD, probably secondary to slow blood flow in the false lumen, inflammation, and enhancement of the vasa vasorum .The differential diagnosis of IAD presenting with focal stenosis or complete occlusion is sometimes difficult, particularly in middle cerebral artery (MCA) dissection, as intramural hematoma and intraplaque hemorrhage (IPH) may look similar. Therefore, integration with clinical information, particularly focusing on vascular risk factors and follow-up imagining are mandatory . In vertIAD are dynamic lesions and changes in appearance are present in more than 80% of cases in the first two weeks after dissection and can continue for months. Complete recanalization is seen in 20% and, overall, at 6-month recanalization is seen in 20–58% of patients, while 30–77% have no change in vessel lumen .RCVS is characterized by the multifocal vasoconstriction of intracranial arteries with consequent stenosis. The pathogenesis is thought to be a transient alteration of cerebral vasculature tone regulation. Clinically, thunderclap headache, often recurrent over one or two weeks, is the most common symptom; ischemic and hemorrhagic stroke are the main complications ,75. RCVSDSA is considered the gold standard to differentiate RCVS from CNS vasculitis and intracranial atherosclerosis and shows multifocal narrowing of intracranial arteries, that usually reverse in less than three months from onset. The dynamic nature of the stenosis is a cardinal feature for the diagnosis of the syndrome and can be appreciated even at close follow-up during the acute phase. Stenosis involves more frequently MCA, ACA and posterior cerebral artery (PCA); conversely, involvement of intracranial segment of the internal carotid artery is less frequent and usually point to another diagnosis as Moyamoya and intracranial atherosclerosis. The posterior circulation arteries are often involved and the abnormalities are more diffuse in RCVS then in other intracerebral arteriopathies . HoweverMRI can detect luminal stenosis with an 80% sensitivity compared to DSA and can contextually evaluate brain parenchyma for the main RCVS-associated complications that are important for therapeutic purposes. Associated imaging findings on MRI in RCVS patients are convexity SAH, posterior reversible encephalopathy syndrome (PRES)-like lesions, ischemic stroke, intraparenchymal hemorrhage and hyperintense cortical vessel on FLAIR images ,79.Even if DSA has a high sensitivity in detecting luminal stenosis in RCVS, the pattern and distribution of these lesions significantly overlap with vasculitis lesions (mainly PACNS), and differential diagnosis can be challenging, especially at presentation, when longitudinal follow-up imagining is not available. Moreover, misdiagnosis of RCVS with a vasculitic process can expose the patient to unnecessary and harmful immunosuppressive treatment that can precipitate the underlying disease ,81.In this context, VW-MRI can have an important role in discriminating between RCVS and its mimics .Intracranial VW-MRI findings in RCVS are multifocal stenosis with concentric iso- or hypo-intense wall thickening, often non-enhancing (≅55%), or less frequently with mild concentric enhancement (≅35%), that can rarely be more marked (≅10%) ,84. EnhaAt follow up, vessel wall enhancement and arterial wall thickening are expected to completely disappear in the majority of patients, but 10% of them can show persistent mild enhancement . o the aMoyamoya disease (MMD) is a progressive non-atherosclerotic, non-inflammatory occlusive disease, involving distal internal carotid (ICA) arteries and large intracranial arteries of anterior circulation. The consequent development of collaterals at the skull base is responsible for the “puff of smoke” angiographic appearance, that gives the name to the disease. Etiology is unknown and histological examination shows hyperplasia proliferation of smooth muscle cells, particularly involving the tunica media associated with an irregular elastic lamina ,87.Moyamoya syndrome (MMS) is characterized by similar progressive stenosis involving the same vascular territory, but secondary to an underlying disease; the differential diagnosis of the underlying cause includes sickle cell anemia, neurofibromatosis 1, radiation therapy, congenital syndromes, intracranial atherosclerotic disease and intracranial vasculitis. The differentiation between MMD and MMS is critical, because the treatment is different and some of the underlying disease have a specific and effective therapy .The diagnosis relies usually on luminal imaging, CTA or DSA. However, the pattern of luminal stenosis is very similar between MMD and MMS. VW-MRI can discriminate between different etiologies, directly depicting the pathologic process involving arterial walls ,90,91.In MMD, VW-MRI reveals ICA and middle cerebral arteries diameter shrinkage (ICA/basilar artery ratios < 1) with associated luminal stenosis. Vessel walls thickness can vary during the disease and can be normal or increased, with homogeneous T2 signal. Contrast enhancement can be either absent or mild, concentric, and homogeneous, involving distal internal carotid arteries and middle cerebral arteries; it can be bilateral. MMS related to vasculitic processes (V-MMS) presents with radiological findings of vasculitis, demonstrating vivid concentric enhancement of arterial wall, without positive remodeling or T2 signal inhomogeneity. Atherosclerotic MMS (A-MMS) can be diagnosed identifying the radiological characteristics of intracranial plaques as eccentric lesions with T2 heterogeneous signal and possible focal nonconcentric contrast enhancement, particularly in active symptomatic plaques ,92,93.In a retrospective study, there was a statistically significant improvement in overall diagnostic accuracy from 32% to 87%, respectively, from 37% to 90% in MMD, from 13% to 100% in vasculitic MMS and from 32% to 82% in atherosclerotic MMS .Aside differential diagnosis, vessel wall enhancement of steno-occlusive lesions in patient with MMD has been associated with intracranial hemorrhagic episodes  and incrIntracranial aneurysms are the leading cause of non-traumatic (SAH) . UnruptuInflammatory activity in the aneurysmal wall, neovascularization and atheromatic lesions inside the aneurysmal sac are key processes that leads to aneurysm wall fragility, and eventually to enlargement and rupture .VW-MRI may depict these processes and aneurysm wall enhancement (AWE) extent has been associated with the degree of histological inflammatory markers, as well as atherosclerosis, neovascularization, and macrophage infiltration ,99,100.VW-MRI may provide an added value in estimating the rupture risk and AWE is the most important VW-MRI finding associated subsequent SAH ,101. AWEIn several studies AWE was associated with UIA instability  and an iThe absence of AWE, integrated with known risk factors, may be useful to select patients with unruptured aneurysm who are suitable for a conservative approach. Specificity and positive predictive value were much lower, respectively 62.7% and 55.8%, and AWE should be interpreted only along with other known independent risk factors and patients’ characteristics to define an UIA at high risk of rupture before deciding for treatment .Another application of VW-MRI is the differentiation between intradural and extradural of unruptured paraclinoid aneurysms of ICA. This categorization is important for both prognostic information and therapeutic decisions, as intradural paraclinoid aneurysm have a significant risk of rupture with consequent SAH. In contrast, extradural paraclinoid UIA usually have a benign course and become symptomatic of mass effect on adjacent structures, but in case of rupture in the cavernous sinus, a carotid cavernous fistula may develop. In a retrospective study, VW-MRI was superior to DSA/MRA in the definition of the intra- or extradural location of paraclinoid ICA aneurysms, with a classification rate of 80% and 47%, respectively .In the context of a recent aneurysmal SAH, VW-MRI may provide additional information for the acute management. First it can help to identify the culprit aneurysm in presence of multiple lesions—in fact AWE is more common in recently ruptured and symptomatic aneurysm than in asymptomatic ones . Second,Lastly, in patients with nontraumatic SAH and normal DSA, which account for 15–20% of cases, VW-MRI revealed abnormal findings in 82.4% of cases, suggesting the utility of this technique in this setting. Abnormalities included intracranial artery dissections, blood blister-like aneurysms, ruptured fusiform aneurysms and focal nodular wall enhancement .However, despite the promising data, larger longitudinal studies must confirm these preliminary findings before they can routinely be used in clinical practice.Brain arteriovenous malformations (AVMs) consist of a network of abnormal blood vessels with a direct connection between arterial feeders and veins, without the interposition of capillaries. The main complication of AVMs is rupture, with consequent intracranial hemorrhage, and often seizures . ClinicaVW-MRI has been used to assess the rupture risk in UIA and to identify the culprit lesion in the presence of multiple IA in patients with SAH. The same approach has been proposed for AVMs. Particularly, VW-MRI can identify the site of the bleeding inside the AVM nidus showing VWE of the involved vascular structure and adjacent blood products; moreover, it can help to exclude a high-risk vascular structure as the source of bleeding. These data can be crucial if a targeted partial embolization of the AVM is planned to prevent rebleeding .However, this approach is based on a few case series ,115 and Overall, there is a scarcity of available data, and previous studies on the use of VW-MRI in AVMs have important limitations, and no strong conclusion can be drawn . More reIn VW-MRI misinterpretations of findings derive mainly from technically incomplete blood signal suppression and from conditions that may mask or mimic vessel wall enhancement.Incomplete blood signal suppression in VW-MRI sequences may mimic an eccentric or concentric vessel wall thickening. Artifacts due to incomplete blood signal suppression are more frequent where blood flow is reduced, recirculating or disturbed; in vessels that are curved and of large diameter , proximal or distal to a stenosis or in aneurysms ,120. AddSeveral factors may mimic a VW enhancement. The parenchymal enhancement of subacute ischemic stroke, due to the disruption of blood brain barrier, may appear as an inflammatory enhancement of a small parenchymal vessel. The use of diffusion weighted imaging (DWI) helps in depicting the area of ischemia . MoreoveThe central hypointensity of microhemorrhages with the surrounding enhancement due to the inflammatory response of the adjacent tissue ,123 may The enhancement of the vascular venous plexus surrounding the arteries at the skull base can be misinterpreted as vasculitis or atherosclerotic changes. A common site of this misinterpretation is the petrous segment of the internal carotid artery . SimilarLeptomeningeal enhancement can be misinterpreted as a perivascular inflammation. Multiple oblique planes of reconstruction of 3D VW-MRI may clarify the distribution of the enhancement and help to distinguish leptomeningeal from perivascular enhancement .Finally, false negatives may arise from the use of steroid therapy that can attenuate the degree of enhancement reducing the inflammatory activity . AdditioMajor pitfalls for the interpretation of VW-MRI are summarized in VW-MRI has become a valuable aid in investigating patients with acute cerebrovascular disease and shows a wide range of clinical applications. Recent advances in MRI technology and the diffusion of high magnetic field devices made VW-MRI widely available. Its value in the differential diagnosis of intracranial vasculopathy is already recognized, and VW-MRI may be included in the diagnostic workup of several patients, as it provides adjunctive information not obtainable with traditional luminal-based imaging techniques ,125. Fur


Background: Burning mouth syndrome (BMS) is a common condition of predominant oral pain without evident cause, that maxillofacial surgeons and otolaryngologists often refer to psychiatrists as somatic symptom disorder. In very rare cases, its typical burning symptom mimics those of other diseases in which serious fatal comorbidities may be missed. We encountered three rare cases of oral squamous cell carcinoma (OSCC) with the first symptom of burning tongue.Case Presentation: Case 1: A 68-year-old woman had burning pain on the left lingual margin for 8 years. Antidepressant treatment was not efficacious. Cytology and biopsy revealed OSCC. Case 2: A 70-year-old man had burning sensation and paralysis of the tongue for 6 months. Magnetic resonance imaging (MRI) revealed a 37 × 23-mm mass under the floor of the mouth and enlargement of lymph nodes on both sides. Case 3: A 90-year-old man had burning sensation of the tongue for 1 year. MRI revealed a 12 × 12-mm mass on the mandible with bone absorption.Conclusion: This case series suggests that psychiatrists must always be careful in regarding BMS as somatic symptom disorder and be cautious of the possibility of OSCC, especially in elderly patients. Burning mouth syndrome (BMS) is a common condition of chronic oral pain without any evident cause . PatientBoth SSD and BMS are the diagnosis of exclusion, thus thorough systemic and oral examination are required. But sometimes the assessment is truly challenging because in very rare cases, BMS's typical symptoms  might be similar to those of fatal diseases, such as oral cancer . Here, wA 68-year-old, divorced, a female office worker had burning and allodynia-like pain on the left lingual margin, which worsened and spread to the right side when she was talking and eating and with non-painful stimuli. She was referred to us by a family physician. The pain had started 8 years previously, and its severity fluctuated daily. No personal psychiatric history or family history of cancer was recorded. Her past medical history included Ménière's disease and cataract. At her first visit to our clinic, no abnormalities were found in the oral cavity, except leukoplakia on the lateral border of the left tongue . At thisA 70-year-old man with a burning sensation and paralysis on the right side of the tongue for ~6 months was referred to our clinic. A diagnosis of BMS was confirmed while he had visited a family physician, an otolaryngology clinic, a dental clinic, and the oral surgery of a general hospital, but none of which provided relief or satisfaction. He has a medical history of lumbar disk herniation but no formally diagnosed psychiatric disorder. He is a heavy smoker, a retired factory worker and currently lives with his wife and son. In our first examination, he complained about not only taste disturbance but also dysphasia, which was atypical for BMS. A brief pinprick test of the tongue revealed decreased mobility and sensory loss. By visual inspection, no abnormalities were detected other than an induration in the right side of the floor of the mouth . MagnetiA 90-year-old retired male accountant who suffered a burning sensation on the tip of his tongue for almost 1 year was referred to our clinic. Four months before the first visit, he was diagnosed with stomach cancer and lost 15 kg accordingly but refused to undergo surgery. For his oral symptoms, he visited the oral surgery and otolaryngology clinic of a general hospital where BMS was diagnosed. However, no specific treatment was performed and the pain persisted. He had no psychiatric history, but he and his son stated that his mood was mildly depressive, and his SDS score was 49, which was the upper normal limit. However, during our first examination, an induration was found under the midfloor of the mouth . MRI latAccording to the Diagnostic and Statistical Manual of Mental Disorders, 5th Edition , somatiFrom our observations, in Japanese clinical settings, oral surgeons and otolaryngologists may see many BMS patients in their busy daily practice, but barely spend enough time on each suspected case. Additionally, since most BMS patients showed no abnormal or any organic suspicion, if they were elderly patients with depressive symptoms or anxiety, primary care physicians may rapidly attribute it to psychogenic causes and refer to a psychiatrist as SSD. Clinicians, especially those who might not so familiar with this syndrome, can easily get into the habit of making so-called snapshot diagnoses . MochizuIn terms of OSCC, OSCC accounts for 90% of oral cancers and usually manifests as a growing mass with ulceration or red or white patches . There aOf the reported cases, patients initially received a diagnosis of BMS and after careful oral examination, were found to have OSCC. Even none of the above were referred to psychiatrists, we assume that in clinical situations, psychiatrists might encounter some referrals from other specialists with BMS diagnosis and regard them as somatic symptom disorder in their practice. Among these patients, there would be cases that do not respond very well with psychiatric therapeutics after several months. That clinical sign may imply a need to reconsider diagnosis, including the possibility of oral cancer. In such cases, multi-disciplinary collaboration among psychiatrists, oral surgeons, otolaryngologists, where responsibility is shared during treatment and follow-up, would be vital for success.The original contributions generated in the study are included in the article/The studies involving human participants were reviewed and approved by Faculty of Dentistry Tokyo Medical and Dental University approved this study (D2013-005). The patients/participants provided their written informed consent to participate in this study. Written informed consent was obtained from the individual(s) for the publication of any potentially identifiable images or data included in this article.TS, TT, CH, and MT have been involved in drafting the manuscript. YA, LM, YM, HS, NU, TI, HH, and AT revised the manuscript critically. All the authors have read and approved the final manuscript.The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.


Omental torsion, a rare cause of acute abdomen in children and adults, is difficult to correctly diagnose before surgery because it mimics the common causes of acute surgical abdomen. We present a case of greater omental torsion that was diagnosed by laparoscopy. A 37-year-old man presented with right lower abdominal pain and was suspected to have appendicitis. Blood tests revealed elevated C-reactive protein and white blood cell count, whereas computed tomography revealed a nodular mass and high-density lesions in the fat tissue. As the patient’s abdominal symptoms were severe and a clear diagnosis could not be made, we performed exploratory laparoscopy. Laparoscopy revealed omental torsion, and an omentectomy was performed. The patient’s pain had significantly reduced post-surgery, and post-operative recovery was uneventful. Thus, laparoscopic examination is useful for accurately diagnosing omental torsion and is less invasive than surgery. Torsion of the greater omentum is a rare acute abdominal condition that is difficult to distinguish from other acute abdominal pathologies. Additionally, omental torsion is difficult to accurately diagnose using blood tests and abdominal images, and abdominal pain often intensifies when the greater omentum is severely tortuous and obstructed. Exploratory laparoscopy is an optimal intervention, because it is less invasive than surgery and can be used to explore the cause of severe or worsening abdominal symptoms.Herein, we report a case of greater omental torsion diagnosed by laparoscopy in an acute abdomen and treated appropriately.A 37-year-old man with no history of surgery presented to our hospital with suspected appendicitis. The patient had right lower abdominal pain 2 days before admission, and the pain was so severe that the patient was unable to maintain a neutral spine position.3, respectively). Plain abdominal computed tomography (CT) showed a nodular mass and high-density lesions in the fat tissue near the hepatic flexure of the ascending colon  and white blood cell (WBC) count , which was not observed in the surgically treated group .2%, whic, 9 becauet al.  reportedet al. , 12. In Omental torsion often mimics other acute abdominal conditions; thus, it is difficult to diagnose. Laparoscopic examination is useful when diagnosis is doubtful or in cases with severe symptoms. Accordingly, we successfully treated a patient with omental torsion laparoscopically.Video_rjab237Click here for additional data file.


Parosteal lipomas are rare neoplasms comprising mature adipocytes situated in a proximity to bone. Although these tumors follow a benign course, the reactive osseous changes that may occur with such lesions might raise the suspicion of malignancy.Here we present a case of a 33-year-old male patient complaining of pain and swelling in the right anterior forearm without history of trauma.An magnetic resonance imaging of the region revealed a lobulated intramuscular fat intensity mass within the supinator muscle. Bony projection inseparable from the anterolateral radial diaphyseal cortex and periosteum was also seen. The radiological features suggested the diagnosis of parosteal lipoma.After the radiological diagnosis of a parosteal lipoma, the patient was offered a total surgical excision of the mass.The mass was removed successfully. Histopathology showed mature benign adipose tissue bordered by thin fibrous septa confirming the diagnosis of parosteal lipoma. Follow-up magnetic resonance imaging after 6 months did not reveal any signs of complications or recurrence.Distinction of the features of parosteal lipomas is needed to establish the accurate diagnosis, discriminate it from malignant lesions, predict potential neurovascular compromises, and follow up until a curative action is planned. Deep lipomas can be further classified into intermuscular, intramuscular, or parosteal. Parosteal lipomas are benign tumors mostly solitary, arising from mature adipose tissue near the periosteum of bones and they account for <0.3% of all lipomatous lesions.–6 Parosteal lipomas, as in other lipomas, are more frequent in the middle aged population (40–60 years’ old) and affect males and females equally.,8 Most patients with parosteal lipomas are asymptomatic, although they might complain from a slowly growing mass or swelling. However, pain, sensory disturbances and weakness may be encountered if neurovascular structures are compromised.–11 Although x-ray and computed tomography (CT) scans are considered useful in detecting and diagnosing parosteal lipomas, Magnetic resonance imaging (MRI) is the most preferable method in evaluating such tumors.,13 Parosteal lipomas are becoming of significant importance as half of patients having this type of tumors possess accompanying bony lesions that may be suspected to be malignant. In this article we present a rare case of parosteal lipoma of the proximal forearm accompanied with a literature review of similarly reported cases in the English-based literature. This case has been reported in line with the CARE criteria.Among tumors arising from soft tissues, lipomas are the most common benign form.2A 33-year-old male presented to our clinic, complaining of pain, and swelling in the right proximal anterior forearm (near the right elbow) that started after performing some sports. There was no history of trauma. Chronic or medical history, family history of chronic diseases, and drug history were all insignificant. On physical examination, a firm immobile swelling in the right anterior forearm was noticed. No skin discoloration, scars, or dilated veins were seen. Pain and tenderness at the site of the lesion were aggravated by wrist extension. Movement of the right elbow was within normal limits. No limitations in extension of the right wrist or fingers were detected. Radial pulses were palpated and bilaterally equal. An MRI was ordered to evaluate the detected mass and showed in Fig.  a large 33.1 was conducted using 3 different databases  via the key words and search terms “Parosteal Lipoma” OR “Periosteal lipoma.”A systematic thorough literature review following the Preferred Reporting Items for Systematic Reviews and Meta-Analysis guidelineTwo authors screened the obtained records from the selected databases independently by the title and abstract of the articles, then by full text. Records were included if they were case reports or case series reporting parosteal lipomas. Irrelevant studies or those written in languages other than English, or lacking full text, audits, letters to editor were all excluded. Different aspects of included cases were taken under consideration in the review such as, the publication year of the study, age, sex, symptoms of the lipoma, duration of symptoms, diagnostic methods used, location of the lipoma, measurements of the lipoma, presence of nerve compression, method of treatment, histopathological findings, and the follow-up period. Conflicts between authors in screening and data extraction were solved by the senior author of the study. Case reports were critically appraised by 8 questions concerning the demographics of the study, history description, clinical condition description, diagnostic tests or assessment methods, intervention of used procedure, post intervention clinical condition, and description of adverse events. Case series were critically appraised by 10 questions about the inclusion criteria and methods, reliability of condition measurement, validity of identification methods, demographics of the study, clarity of reported information, sufficiency of reported outcomes, and statistical analysis efficacy. In the quality assessment of both the case reports and case series, questions were answered by (yes), (no), or (unclear). Risk of bias for the study was determined based on the percentage of (yes) answered questions. A study was at high risk of bias if the percentage of (yes) answered questions was ≤49%, at moderate risk of bias if the percentage of (yes) answered questions was between 50% and 69%, and at low risk of bias if the percentage of (yes) answered questions was ≥70%.All included case reports and case series for data extraction were assessed for quality using the Joanna Briggs Institute critical appraisal tool for case reports and case series.4The search of parosteal lipoma cases in 3 different databases resulted in 433 records. A total of 226 duplicates were identified and removed. After title abstract screening and full text screening, 48 studies were finally included for data extraction Fig. .After screening obtained articles and assessing the methodological quality of the included studies using the Joanna Briggs Institute critical appraisal tool for case reports and case series by using the information provided by each article and referring to the authors of the studies in case of insufficient data, 3 studies were at high risk of bias (6.25%), 22 studies were at moderate risk of bias (45.83%), and 23 studies were at low risk of bias (47.91%). Figures. 6 and 7 demonstrate the risk of bias summary of included case reports and case series.Different features of included case reports and case series were extracted and are summarized in Table . The revThe forearm was noticed to be the most common location of parosteal lipomas in the reviewed studies as it appeared in 22 patients (34.38%), followed by the arm in 10 patients (15.63%). The presence of a parosteal lipoma in the clavicle, scapula, forehead, and the spine was extremely rare.Of the 32 patients with parosteal lipomas located in the arm or forearm, 9 patients had a PIN compression, nine had a radial nerve compression and 1 patient had a brachial plexus compression. Forty-three patients of the overall number did not report any nerve compression.Regarding the treatment of the parosteal lipoma patients of the review, 60 of them had a complete surgical resection of the tumor (93.75%), 2 patients were conservatively treated (3.125%) and 2 patients refused the surgery (3.125%).Histopathological examination was performed for fifty59 patients of those who underwent surgeries and confirmed the diagnosis of parosteal lipoma by observing mature fat cells without cellular atypia or lipoblasts.Follow-up periods of patients were not reported in twenty-four cases. The average follow-up period of the remaining studies was 19.44 months. None of the included studies mentioned any signs of tumor recurrence.5,18Lipomas are the most common benign mesenchymal tumors and are composed of adipose cells typically arising in soft tissues, affecting 1 of each 1000 persons and have a higher incidence to occur in males than in females. They can originate in any place where there are adipocytes. When lipomas are closely related to bones, they are called osseous lipomas which are divided according to their location of placement into intraosseous (within the bones) and Parosteal (on the surface of the bone).Lipomas of the bone are considered less frequent than other types of lipomas as they comprise <0.1% of all bone tumors. Until 1888, the term periosteal lipoma was sometimes used in the medical literature but, in his publication, entitled “A Parosteal lipoma, or congenital fatty tumor connected with the femur” in 1888, Power et al asserted the origin of those tumors and introduced the term “Parosteal lipoma” that is still being used until these days. The term periosteal was replaced by parosteal to emphasize the idea that this kind of tumors does not necessary originate from the bone itself but can be adjacent to it.The first publication of a parosteal lipoma case in the English based medical literature took place in 1866 by Smith et al in which it was described as a “Specimen of firm bilobed fatty tumor.”,8,23Parosteal lipomas are defined as benign rare tumors composed of mature adipose tissue closely related to the periosteum, usually asymptomatic, accounting for 0.3% of all lipomas and mostly affecting middle aged patients (older than 40 years) and are equally frequent in both females and males. The presence of such tumors in small bones such as the carpals is extremely rare.Parosteal lipomas usually develop in the diaphysis of long bones. The most common sites of parosteal lipomas in order are the femur, radius, tibia, and then the humerus. Depending on the size, location of the tumor, and the presence of adjacent neuromuscular or neurovascular structures, nerve compressions may develop thus causing deficits in sensory or motor functions and leading to muscle atrophy. Many cases, as in our case, reported the compression of the PIN due to parosteal lipomas of the forearm.,28A parosteal lipoma usually presents as a palpable, slowly growing, large, immobile mass. Reactive changes of the bone surrounding parosteal lipomas may also be seen. These reactions include cortical thickening, calcification, or sclerosis. Smooth scalloping of the cortex, or osseous bowing or projection, can be also visualized.On x-ray, a parosteal lipoma appears as a radiolucent fat containing mass that is well circumscribed with an intimate relation to the bone's periosteum. This typical characteristic finding may be difficult to appear if the mass is located in an area overlayed with osseous structures as in the pelvis. Using MRI, parosteal lipomas will be shown as lesions with signal intensity like that of subcutaneous fat. On some occasions, alongside bone projections, hyaline cartilages of intermediate (on T1 sequence MRI), or high (on T2 sequence MRI) signal intensities may be visualized. Fibrous tissues, may also be seen, they can be differentiated from cartilages by their low signal intensity appearance on T2 sequence MRIs even after contrast enhancement. Linear streaks of high signal intensity within muscles on all MRI pulse sequences representing fat can be an indicator to muscle atrophy due to nerve compression.On non-contrast CT scans, these lesions appear as well-demarcated masses of low density (−120 to −60 Hounsfield unites). CT and MRI are also useful in differentiating osseous projections in parosteal lipomas from osteochondromas because medullary continuities with adjacent bone are absent in parosteal lipomas.In planning for surgical excision, the relationship between osseous excrescence and lipomatous mass with adjacent muscles and underlaying bony cortex is better visualized on MRI due to its callability of multiplanar imaging. To avoid recurrence of the lipoma, wide and complete combined margin excision are suggested. Histopathological findings of mature adipocytes without cellular atypia can confirm the diagnosis of parosteal lipomas after the surgical excision.In this type of lipomas, the optimal method of management is complete surgical excision of the tumor, especially in cases wherein certain nerve compression by the mass is present, to reduce the chances of permanent nerve damage or muscle atrophy.6The clinical approach to parosteal lipomas requires collaborative efforts from physicians in multiple disciplines to diagnose accurately and treat promptly, as rapid restoration of patients’ quality of life remains the top priority in management. Distinction of the features of such lesions using MRI which is the criterion standard modality is vital, to establish diagnosis, discriminate it from malignant lesions, predict potential neurovascular compromises, and follow-up until a curative action is planned.We would like to thank our patient for consenting to the publication of this article.Conceptualization: Asma’a Al-Mnayyis, Mohammad Araydah.Data curation: Sarah Al Sharie, Mohammad Araydah, Muna Talafha, Fadi Haddad.Investigation: Asma’a Al-Mnayyis.Methodology: Sarah Al Sharie, Muna Talafha.Resources: Sarah Al Sharie.Supervision: Asma’a Al-Mnayyis.Validation: Asma’a Al-Mnayyis.Visualization: Asma’a Al-Mnayyis, Mohammad Araydah.Writing – original draft: Asma’a Al-Mnayyis, Sarah Al Sharie, Mohammad Araydah, Muna Talafha, Fadi Haddad.Writing – review & editing: Asma’a Al-Mnayyis, Sarah Al Sharie.


Desmoplastic fibroblastoma is an uncommon, benign, fibrous tumor exhibiting infiltrative growth. Most of these tumors are small, slow-growing, and develop as subcutaneous lesions in the extremities. Cases of desmoplastic fibroblastoma in the chest wall are quite rare, and the preoperative diagnosis of such cases remains challenging as these tumors can mimic the characteristics of desmoid-type fibromatosis, which often occurs in the chest wall. We aimed to describe a rare case of desmoplastic fibroblastoma exhibiting rapid growth in the chest wall of a patient that was successfully treated with marginal excision only by diagnostic imaging before surgery.A 79-year-old man was admitted to our hospital after experiencing right shoulder pain lasting for a few months. A 4 × 4 × 2 cm mass was incidentally detected at the right second rib two years prior. Chest computed tomography revealed a well-defined homogeneous mass with a muscle-like density along the right lateral chest wall, the size of which had increased to 12 × 10 × 4.5 cm in two years. Dynamic contrast-enhanced computed tomography revealed abundant vascularity at the periphery of the tumor. Magnetic resonance imaging revealed iso-intensity to muscle on T1-weighted images, slightly high intensity on T2-weighted images, and rim-like contrast enhancement at the periphery of the tumor, with uniform thickness on gadolinium-enhanced T1-weighted images with fat suppression. Rim-like contrast enhancement is an imaging feature that can distinguish cases of desmoplastic fibroblastoma from desmoid-type fibromatosis. We diagnosed the tumor as desmoplastic fibroblastoma by diagnostic imaging without tissue biopsy. Marginal excision with videoscopic assistance was performed through a small incision. The pathological diagnosis was desmoplastic fibroblastoma. The patient’s postoperative course was uneventful, and his shoulder pain was relieved after the surgery.Desmoplastic fibroblastoma in the chest wall is extremely rare, but should be considered in the differential diagnosis when desmoid-type fibromatosis is clinically suspected. Gadolinium-enhanced magnetic resonance imaging is helpful in confirming the differential diagnosis. Desmoplastic fibroblastoma is an uncommon, benign, fibrous, soft tissue tumor exhibiting infiltrative growth –4. TheseDesmoplastic fibroblastomas share some features with other infiltrative tumors with fibrous components –7; theseCases of desmoplastic fibroblastoma in the chest wall are quite rare, and it is important to differentiate these tumors from cases of extra-abdominal desmoid-type fibromatosis, which often occur in the chest wall –10. HereA 79-year-old man was admitted to our hospital after presenting with right shoulder pain that had continued for a few months. On admission, physical examination revealed winging of the right scapula. A 4 × 4 × 2 cm mass had been detected at the right second rib on chest computed tomography (CT) scans two years prior. A chest radiograph revealed soft tissue thickening in the right lateral upper chest wall Fig. a. Chest The patient refused a biopsy examination, preferring to undergo surgery through a minimal incision because of his shoulder pain. Marginal excision was performed through a small incision with the assistance of a thoracoscope . The tumor margin was marked on the skin surface with ink under ultrasound guidance. A 4 cm vertical utility incision was made along the middle axillary line, along with a 2 cm port incision at the triangle of auscultation (Fig. The resected specimen was an elastic, hard, disk-sharped mass, measuring 13 × 12 × 5 cm in size. The cut surface had a homogeneous, white-colored appearance, with a gray layer visible at the periphery of the tumor Fig. a, b. ImmAn extremely rare case of desmoplastic fibroblastoma was reported. Desmoplastic fibroblastoma should be considered in the differential diagnosis when desmoid-type fibromatosis in the chest wall is clinically suspected. Rim-like contrast enhancement at the periphery of the tumor with uniform thickness on gadolinium-enhanced T1-weighted MR images with fat suppression is the unique imaging feature in desmoplastic fibroblastoma that can distinguish it from desmoid-type fibromatosis preoperatively. However, when performing marginal excision of desmoplastic fibroblastoma in the chest wall only by diagnostic imaging before surgery, as in our case, it is necessary to consider the treatment strategy in case of desmoid-type fibromatosis as a result of pathological diagnosis.Desmoplastic fibroblastoma is a unique form of benign, fibrous, soft tissue tumor –4. TheseThe following fibrous tumors should be considered in the differential diagnosis of tumors occurring in the infrascapular region: desmoplastic fibroblastoma, desmoid-type fibromatosis, solitary fibrous tumor, and elastofibroma dorsi . Among tAn imaging feature that can distinguish desmoplastic fibroblastoma from desmoid-type fibromatosis is rim enhancement , 17, whiWe consider that rim enhancement can represent an area of infiltrative growth with abundant vascularity. A recent study reported the characteristics of the cells of a patient with a tumor that grew invasively into the surrounding tissue, which were distributed compactly at its periphery, forming a gray-colored layer . The insPatients diagnosed with desmoplastic fibroblastoma exhibit favorable surgical outcomes after marginal excision , 4, althAs an alternative to conventional open surgery, video-assisted surgery has been proposed for the resection of chest wall tumors, as it allows for a reduced incision size and less tissue trauma . In the When performing marginal excision only by preoperative imaging examinations, as in our case, it is necessary to consider the treatment strategy in case of desmoid-type fibromatosis as a result of pathological diagnosis. Marginal excision has insufficient surgical margin to reduce the local recurrence of desmoid-type fibromatosis –22. HoweThe histopathological features of desmoplastic fibroblastoma include the presence of bland stellate or spindle-shaped fibroblasts, as well as myofibroblasts undergoing infrequent mitosis that are embedded within an abundant and dense collagenous matrix, with low to moderate vascularity , 11, 18.Desmoplastic fibroblastoma in the chest wall is extremely rare, but should be considered in the differential diagnosis when desmoid-type fibromatosis is clinically suspected. Gadolinium-enhanced magnetic resonance imaging is helpful in confirming the differential diagnosis.


Preputial stone disease is the rarest type of urolithiasis. Adult males with severe phimosis and poor hygiene are mainly affected. A 90-year-old male sought treatment for steadily worsening urinary frequency, intermittency, incontinence, and pain at the tip of his penis of 3-days duration. Clinical examination revealed a palpable distended urinary bladder, a partial phimosis and a round, hard on palpation, and partly ulcerative lesion at the tip of the foreskin. A single, 1 cm in maximum diameter stone, was incidentally discovered beneath the prepuce and subsequently removed from the preputial sac. The patient refused further treatment with circumcision, and opted for conservative therapy of benign prostate hyperplasia. Personal hygiene remains the cornerstone in the prevention of the preputial calculi formation, while circumcision represents the mainstay of treatment for definite stone removal and elimination of the precipitating causes. Preputial stone disease (PSD) remains an exceptionally under-reported type of urolithiasis since its first presentation by Robert Clarke in 1794 . The priA 90-year-old male of low socio-economic status presented in the emergency department with acute urinary retention and a 3-day-long history of lower urinary tract symptoms (LUTS). More specifically, he complained of steadily worsening urinary frequency, intermittency, post-micturition dribbling, urge incontinence, and constant pain at the tip of his penis. His past medical history was significant for acute myocardial infarction, diabetes mellitus type 2, hypertension, dyslipidemia, and mild dementia.. Despite the stone’s firm embedment underneath the prepuce, the urethral meatus appeared intact hypothesizing that benign prostate hyperplasia could have triggered LUTS’s exacerbation. Moreover, no congenital urethral abnormalities could be detected.Clinical examination revealed a nontender, palpable distended urinary bladder. The foreskin appeared swollen and was painful on examination, and a round, about 1 cm in diameter, ulcerative lesion was noted on the left side of the tip of the foreskin. Intermittent, spontaneous leakage of small amounts of urine could be easily noticed through the tight preputial opening. We tried to retract the foreskin in order to identify the external urethral meatus and proceed with catheterization. After insertion of the index finger in the preputial cavity, tight adhesions were felt and a single, oval stone impacted beneath the foreskin was seen. A 1 cm in maximum diameter yellow-gray colored nonobstructing stone was eventually removed and the external urethral meatus was identified Subsequently, a 20-Fr Foley catheter was inserted and 1100 ml of urine was drained. Serum creatinine was mildly elevated  while blood chemistry and urinalysis were within normal limits. Abdominal ultrasonography showed mild bilateral dilatation of the upper urinary tract. Plain X-ray of the kidney, ureter, and bladder did not reveal any radiopaque shadows. During hospitalization, the patient was managed conservatively with hydration, antibiotics, and topical application of antifungal cream. The urine culture obtained during catheterization was negative. Following an uneventful recovery, he was discharged on the second-day post-admission and listed for circumcision. Nevertheless, he refused surgical treatment and opted only for the treatment of LUTS with alpha-blockers. There is a paucity of critical data in the existing body of literature pertinent to PSD . PreviouSimilarly, to our case, in 1997, Sonnex et al. described the presence of three preputial calculi consisting of smegma in a 24-year old patient with a partially retractable foreskin and coexistent balanoposthitis . The numApproaching the pathogenesis of PSD, we recognize three different types of preputial calculi: (a) those originating from inspissated smegma with lime salts, (b) calculi originating from precipitation of urinary salts because of urinary retention in the preputial cavity, which may be accompanied by an infection usually caused by urea-splitting bacteria, and (c) migratory calculi from the upper urinary tract to the preputial sac ,5,7. RegSmegma in the subpreputial space may act as a nidus promoter for stone formation while further inducing local inflammation, adhesion formation, and preputial stenosis with subsequent obstruction ,6.In our case, smegma solidification was possibly incriminated as the phimosis was not severe enough to cause urinary stasis, salt precipitation, or entrapment of a migratory stone. Moreover, this process could explain the reason behind local infection and adhesions that were encountered between the glans penis and inner prepuce in our patient. Besides, there was no indication of lithiasis elsewhere in the urinary tract, and the urine culture was sterile. Common presenting symptoms range from voiding difficulties, foul-smelling penile discharge, and chronic balanoposthitis to penile pain and acute urinary retention ,5,7,8,9.Physical examination usually suffices as calculi can be easily palpated in the preputial sac. At the same time, imaging modalities such as ultrasound and X-ray of the kidney, ureter, and bladder support diagnostically by unveiling lithiasis in the rest of the urinary tract ,4. Considering the above, dorsal slit incision or circumcision represent the surgical approaches for definitive treatment, thus permitting stone removal and elimination of the precipitating cause ,4,5.In conclusion, we emphasized the importance of personal hygiene in the prevention of preputial stone formation. Prompt recognition, circumcision, and weaning from causative agents are needed to approach this infrequent urological entity. Physicians should maintain a high index of clinical suspicion, thus performing a diligent genitourinary examination of the external male genitalia.