Pilomatricomas are rare skin tumors related to hair follicles. They typically present in children, and the most common locations are head and neck. Pilomatricomas are usually painless; however, they can cause cosmetic problems. Treatment is decided on an individual basis and involves local excision. However, caution should be taken if the lesion is malignant, as resection with safe margins (0.5-1.0 cm) is determinant of the overall outcome. Radiotherapy is indicated in cases of residual tumor tissue or recurrence. The role of chemotherapy in pilomatricomas is still undetermined. We report a 55-year-old female with multiple lumps on her shoulder and back. The sonographic features of these lesions were typically consistent with pilomatricomas. The patient decided to opt for excision due to cosmetic reasons and the histopathology features were suggestive of pilomatricoma. A pilomatricoma, also known as “calcifying epithelioma of Malherbe,” is a rare skin tumor. Malherbe and Chenantias first described this tumor in 1880 . It usuaA 55-year-old female patient with no significant past medical history presented to our hospital with a painless swelling behind the right shoulder and upper back for two years. The swellings were stable in size; however, the patient documented recurrent infection of the lesions for which she took antibiotics. On examination, there were multiple subcutaneous bumps behind the right shoulder and upper back. The swellings were solid, well-defined, with no punctum or sinus, and the skin was intact with no signs of infection.Ultrasound soft tissue was done for further evaluation, which demonstrated a benign lesion in the subcutaneous region of the right posterior shoulder with a few calcifications and peripheral halo sign typically consistent with pilomatricoma Figures , 1B.Lesions with similar sonographic features were also seen in the lower cervical region and the left cheek Figures , 2B.The patient was counseled about the benign nature of the swelling; however, she opted for surgical excision due to cosmetic reasons. Histopathology confirmed the presence of encapsulated and circumscribed lesions having basaloid and ghost cells Figure .On follow-up visits, after three and six months, there was no reoccurrence of the lesions and the patient is healthy to date.Pilomatricomas are rare skin tumors and present as superficial firm nodules. They are typically painless and only rarely show overlying skin changes. Typical presentation includes a firm, reddish or bluish, moveable subcutaneous nodule usually having a lobulated surface similar to the exhibition in our patient ,4. Due tEpidemiological studies suggest a bimodal pattern of incidence . The firMultiple cellular and molecular pathways are thought to play a role in the development of pilomatricomas. Out of these, CTNNB1/Wnt/β-Catenin signaling pathway is the most widely demonstrated in the pathogenesis of pilomatricomas . AdditioHistologically, pilomatricomas are follicular neoplasms, with well-defined circumscribed cystic structures having soft borders . PilomatVarious radiological modalities have been implicated in diagnosing pilomatricoma. Out of these, ultrasound is the primary for its easy accessibility. Pilomatricomas appear as solid isoechoic tumors on ultrasound and a clear hypoechoic outline. These tumors are typically completely calcifying; however, some tumors show partial calcification. A computed tomography (CT) scan yields better images with more details of calcifications. On MRI, pilomatricoma appears as homogenous (66.7%) or inhomogeneous (33.3%), hypointense lesions on T1-weighted images with ring-like enhancement on post-contrast images. A few of the features of fat-suppressed T2-weighted images include reticular hyperintensity, peri-tumoral fat stranding, and circular target sign .Treatment is usually local excision for the sake of cosmetic reasons in case of benign lesions. However, caution should be taken if the lesion is malignant, as resection with safe margins (0.5-1.0 cm) is crucial to the overall outcome . Other mPilomatricomas are rare skin tumors related to hair follicles, mostly present in children. These tumors are primarily found in the head and neck and are mostly asymptomatic and have a reddish or bluish appearance. Typically, pilomatricomas are 5-10 mm in size, regular or irregular in shape, and can be tender on palpation. Ultrasound can help in the diagnosis and typically presents as an isoechoic lesion with peripheral halo sign. Management is usually conservative; however, some patients opt for excision due to cosmetic regions. Histopathology shows encapsulated and circumscribed lesions having basaloid and ghost cells with areas of calcification and keratinization. More research is needed to further understand the pathophysiology of pilomatricomas.


Ileal diverticula usually remain asymptomatic. If complicated, they may present as intra-abdominal or pelvic abscess. Abscess formation in the presacral space is very rare. The rupture of abscess and spread of pus to extra-pelvic sites through anatomical structures of the pelvis is extremely rare. It carries high mortality if not diagnosed on time.We report a case of 52-year-old woman presented with thigh phlegmon and septic condition. The CT scan revealed free air in the left leg and pelvic presacral fluid collection descending to extra-pelvic direction through the greater sciatic notch. Moreover, ileal fistula to presacral abscess was suspected. Multiple incisions and fasciotomies were urgently performed to treat thigh phlegmon. Subsequently, laparotomy was carried out and ileal fistula was excised. Histological examination of the surgical specimen demonstrated that the fistula to presacral abscess has formed due to perforated ileal diverticulum. 3 years after the surgery the patient remains healthy without recurrence.Rupture of presacral abscess to extra-pelvic site due to ileal diverticulum fistula is an extremely rare case, to our best knowledge, never reported in literature. Due to a rare occurrence and early septic complications if diagnosed late, this condition carries a high mortality rate.Intrapelvic pathology must be considered in patients with thigh phlegmon in order to prevent complications and associated mortality. Meckel’Pelvic abscess in peritoneal cavity is quite common complication of abdominal pathology . HoweverThough symptoms of the complicated diverticula and presacral abscess are variable and nonspecific, the extra-pelvic manifestation is very rare. It may follow several routes, including greater or lesser sciatic notch, obturator foramen, inguinal or femoral canal, presenting as an extra-pelvic abscess or phlegmon ,9. MisceIn this report, we present an extremely rare case of presacral abscess manifestation as thigh phlegmon due to the ileal diverticulum fistula. To our best knowledge, there are no reported cases of ileal diverticulum fistula presenting in such a fashion. This case is reported in consensus with SCARE 2020 guidelines .2A 52-year-old woman presented to the emergency room with intense pain and swelling of the left leg and lumbar area, accompanied by high fever. According to the patient, the pain occurred 6 days ago without traumatic events. After assessing the patient’s medical history it was known that 23 years ago she delivered a baby via Caesarean section. Unfortunately, an early postoperative period was complicated: laparotomy, hysterectomy and cholecystectomy were performed due to pelvic abscess formation and peritonitis. At a subsequent time the patient remained healthy until her current hospitalization.Clinical evaluation revealed swelling of the previously mentioned areas and painful palpation. There was significant redness in the lateral middle part of the left thigh. Initial blood workup showed elevated levels of serum C-reactive protein (518 mg/L) and procalcitonin (4.4 ng/mL). Due to a septic condition, the patient was admitted to the intensive care unit.For further evaluation, a computed tomography (CT) scan was performed. It revealed free air in the left leg  and pres3A thorough search of the databases did not reveal any similar reports about small bowel diverticula, accompanied by pelvic abscess and presenting with extra-abdominal symptoms. A handful of case reports were presenting a complicated large bowel diverticulitis occurring as necrotizing fasciitis in extra-abdominal sites.Ileal diverticula are mostly found incidentally during radiological investigations, laparotomy or autopsy ,4. In moPresacral abscesses are uncommon, mostly originate after rectal surgery ,11. In oAlthough CT scan showed fixated ileum to the abscess and the colonoscopy with terminal ileoscopy raised suspicion of ileal fistula, the diagnosis was doubtful since none similar cases have been reported before. It became clear after the second surgery when the fistulated ileal diverticulum was found, excised and verified histologically. Perhaps chronic inflammation in the old pelvic presacral cavity led to fistula formation of the ileal diverticulum, resulting in new abscess formation. Pathology report suggested the origin of the diverticulum to be more likely congenital. According to literature, the most common congenital anomaly is Meckel’s diverticulum. Usually it is located at a distal ileum approximately 60 cm from the ileocecal valve . In our Spread of intra-abdominal infection to extra-abdominal sites carries a high mortality rate if diagnosed late . Fortuna4The purpose of presenting this case is to raise awareness of extremely rare manifestations of complicated small intestine diverticula and pelvic abscesses. Although rupture of the presacral abscess to the thigh due to ileal diverticulum fistula is extremely rare, intrapelvic pathology has to be considered in patients with thigh phlegmon. An early diagnosis and appropriate interventions are crucial for reducing the incidence of further complications and the associated mortality.The authors declare that they have no conflicts of interest.Not applicable.Authors were complying with Klaipeda university hospital requirements. The data used are anonymous and patient rights are preserved.Each patient signs general form of informed consent while admitted to Klaipeda university hospital, and item number 5 states the following: “I am informed that Klaipeda university hospital is scientific and teaching institution, and I agree to participate in teaching process during my treatment here, and I agree that anonymous data concerning my health can be used for teaching and scientific purposes.” Therefore, this form enables us to collect and use the data.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.All authors contributed to the article conception and design. Material preparation, data collection and analysis were performed by Dainius Simcikas, Alisa Maksimova-Cesnaviciene and Mindaugas Gvazdaitis. The first draft of the manuscript was written by Dainius Simcikas and all authors commented on previous versions of the manuscript. All authors read and approved the final manuscript.Not applicable.Dainius Simcikas.Not commissioned, externally peer-reviewed.No data available.Not applicable.


Background: Enterobiasis or oxyuriasis from Enterobius vermicularis is an infection usually localized in the large bowel and cecum. Generally, the symptoms are characterized by anal itching, and intestinal or nervous disorders. Rarely, it is responsible for death. Methods: A forensic autopsy of a 52-year-old white male inmate who died 5 days after hospitalization was performed. Histological and toxicological analyses were also performed. Results: The death occurred by localization of Enterobius vermicularis in the duodenum and in the proximal ileum, with intestinal haemorrhage, inflammation, and peritonitis documented by histological examination. Conclusion: This is a common infectious disease, and can rarely occur with a fatal outcome, even in advanced populations. The lack of knowledge related to the rarity of death from enterobiasis disease can determine a dangerous concern. Gastrointestinal infections are a less common cause of sudden death compared to other conditions such as cardiovascular diseases, but they are equally important ,2. AmongWe report a case of death with forensic autopsy, in which EV in the duodenum and in the proximal ileum was found histologically. Such a death is rare, but this report shows that it can occur.Case History: A 52-year-old white male inmate with a history of non-steroidal anti-inflammatory drugs (NSAIDs) therapy and enalapril therapy for hypertension was admitted to the emergency room for repeated lipothymia in the absence of sweating, with hematemesis from the previous evening and melaena from three days before. The patient was hemodynamically unstable with acute anemia. The hemoglobin value upon admission was 6g/dL, while the procalcitonin in the blood was not evaluated. Therefore, a computed tomography (CT) scan of the abdomen was performed, which revealed a narrow lumen of the second portion of the duodenum; furthermore, the esophagus-gastro-duodenoscopy (EGDS) examination revealed multiple sub-centimeter lymph node formations in the stomach with normodistended walls due to insufflation, and fundus and gastric bodies occupied by food residues and clots; at the level of the first duodenum, there was an ulcerated lesion covered by a large clot. After a worsening of the condition, the patient was transferred to Intensive Care, was intubated and underwent therapy to restore hemodynamic balance. On the fifth day, the hemodynamics were unstable, and the anemia persisted. An emergency gastroscopy was performed in resuscitation, which revealed the absence of blood in the esophagus, stomach, and duodenum, and ulcerative lesion of the duodenal bulb with circumferential extension to the intestinal wall. Conditions precipitated due to common complications of hypovolemia. Hemorrhagic shock and peritonitis due to enterobiasis were assessed as causes of death. After 72 h, an autopsy was performed in accordance with the recommendations on the harmonization of forensic autopsy rules of the Committee of Ministers of the Council of Europe (1999) and according to the commonly accepted criteria for sudden cardiac death (SCD). Femoral blood was analyzed for alcohol (ethanol) and volatiles by head-space gas chromatography coupled with a flame ionization detector (GC/HS-FID). All post-mortem specimens were screened for the presence of the main different classes of drugs , using immunological or chromatographic methods as appropriate. A systematic toxicological analysis (STA) was performed by the LC-MS/MS system (API 3200 triple quadrupole ABI-SCIEX) in multiple reaction monitoring (MRM) mode.The autopsy showed some thin blood-stained fluid in the peritoneal cavity between the intestinal loops at the duodenum and the proximal ileum in the absence of macroscopic evident perforations A. We didThe toxicological analysis showed no evidence of drugs or alcohol.For each organ, sampling was carried out. After fixation in 10% neutral buffered formalin and paraffin embedding, sections for hematoxylin−eosin (E&E) staining for morphological diagnosis were performed. The histology of the duodenum and proximal ileum tract showed diffuse necrosis with multiple areas where trans-mural hemorrhage, granulocytic inflammation, and EV were detected . In partEnterobiasis or oxyuriasis is an intestinal parasitosis that mainly affects children, with predominantly anal itching and intestinal symptoms. After the ingestion of infective eggs, larvae hatch in the duodenum and descend into the crypts of the duodenum and the ileum where they develop and mutate into adult worms. In the beginning, larvae adhere to the mucosal surfaces of the small intestine, where they develop to sexually mature adult form and establish a chronic infection causing mucosal inflammation and ulceration. Then, the larvae can also penetrate the deeper layers of the intestinal wall where they establish chronic inflammation, destroying the tissue layers. Commonly, adult individuals settle in the colon from where pregnant females migrate to the perianal area at night, while the host is sleeping, to lay eggs. In fact, a diagnosis of enterobiasis in the living can be performed by applying a transparent adhesive tape in the perianal area immediately after waking up, followed by microscopic analysis. A reliable diagnosis of enterobiasis is also based on a correct intestinal sampling. Multiple samples of the intestinal wall must be taken at random in order to find the pinworms inside. In histological samples, the thick cuticles and internal organs of the worms can be easily recognizable within the intestinal wall, from the mucosal epithelium to the serosa, according to the transverse and longitudinal sections of paraffin inclusions .Sometimes, although very rarely, EV can also migrate into the other tracts, such as the urinary bladder, peritoneum, kidneys, liver, lungs, and other organs. Generally, it is considered that EV is a non-invasive parasite, which cannot penetrate a healthy intestinal mucous membrane. However, there is increasing evidence that EV infection may be the cause of ileal inflammation. By causing mucosal inflammation and ulceration, pinworms damage the integrity of the bowel mucosa and can then penetrate the deeper layers of the bowel wall or lead to intestinal perforation and hemorrhage ,12,13,14This case is interesting because it offers a reflection in the framing of an inmate with a history of drugs associated with repeated events of lipothymia, hematemesis, and melaena caused by intestinal parasitosis. Unfortunately, there was not enough time to diagnose the EV infections due to the rapid worsening of the patient’s condition. The instrumental investigations were able to identify the ulcerative complications that may be common to different diseases, such as the side effects of drug medications. A CT of the abdomen, EGDS, and gastroscopy showed the absence of blood in the oesophagus, stomach, and duodenum, along with ulcerative lesions of the duodenal bulb with circumferential extension to the intestinal wall. However, the diagnosis of EV infections is generally made on the identification of the eggs in the anal region or of the pinworm after intestinal biopsy. Therefore, intestinal parasitosis should be considered in the differential diagnosis of clinical syndromes like gastrointestinal haemorrhages.This case study presents the worst consequences of enterobiasis in a young adult. A large number of human pinworms were observed histologically in the duodenum and ileum wall, surrounded by inflammatory cell infiltration, bowel hemorrhage, and peritonitis. Therefore, a correct differential diagnosis between enterobiasis and gastrointestinal ulcer hemorrhages from other causes can be useful for correct treatment of patients.


Pilomyxoid astrocytoma (PMA) is a freshly described figure of low-grade neoplasms encountered in early childhood. Nevertheless, its precise classification by the World Health Organization (WHO) is still debatable. Making an exact diagnosis relies on histological and immunohistochemical pathognomonic features with specific radiological findings. PMA behaves aggressively with a shorter progression-free survival, and its management is unfortunately still arguable. We describe a rare case of PMA involving the suprasellar region who displays symptoms consistent with diencephalic syndrome. The diagnosis was made by magnetic resonance imaging (MRI) focused on the hypothalamic-pituitary axis, and the patient underwent a subtotal tumor resection combined with chemotherapy. Diagnosis of brain tumors should be kept in mind in young children with generalized and severe unexplained loss of subcutaneous fat with failure to thrive after ruling out classical causes. Pilomyxoid astrocytoma (PMA) is a rare entity usually described in the hypothalamic-chiasmatic area. A wide variability of diagnostic criteria may overrate the PMA's prevalence by all of the pilocytic neoplasms (PA). It generally concerns infants and very young children (3 months-2 years). Clinical presentation is not well defined. But, the clinicians should be aware of diencephalic syndrome as an unconventional cause of failure to thrive during early childhood which is directly related to neoplasms regarding central nervous system, especially in the hypothalamus and the optic chiasm region. A meticulous assessment of morphologic features and an imminent follow-up is essential because of the aggressivity of PMA and its fluctuating clinical behavior.We report a case of PMA exhibited by failure to thrive; which is a rare outlined association in the literature.An 11-month-old boy was admitted to our university hospital for further exploration of poor weight gain (<3rd percentile).The child was a full-term newborn of first-degree consanguineous parents with an up-to-date vaccination record. The family history was uneventful, and no genetic disorders were reported. Birth growth parameters were within normal ranges, as well as psychomotor development.At 6 months, the child's weight gain became slow despite increased caloric intake.The physical examination revealed pallid and dry skin, besides lack of subcutaneous fat. The patient was afebrile with stable vital signs. His weight was 5 kg with a weight for age value below the third percentile on the grow chart. His head circumference and his length were normal for age. He had a sunken anterior fontanel, a muscle atrophy , and lefLaboratory investigations embracing electrolyte levels, complete blood count, renal function panel, liver parameters, and hormonal assay were normal: FSH (follicle-stimulating hormone): 0.32 mUI/ml (1.5–14), LH (luteinizing hormone): 0.16 mUI/ml (1.2–10), testosterone: 0.078 ng/ml, 8 AM cortisol: 18.5 ug/dl (3.7–19.4), ultrasensitive TSH (ultrasensitive thyroid-stimulating hormone): 2.59 mUI/l (0.27–4.2), and FT4 (free thyroxine): 21.8 pmol/l (12–22). Levels of AFP  (0–7) and HCG (human chorionic gonadotropin) (0–7) were also within normal ranges: 1.2 mUI/l and 6.82 ng/ml, respectively. These hormone measurements were based on Chemiluminescence Microparticle Immunoassay (CMIA).Brain MRI performed upon admission exhibited a suprasellar mass with solid and noncalcified configuration measuring 59∗41∗40 mm in maximum dimensions, without neither peritumoral edema nor parenchymal infiltration. The tumor was isointense on T1-weighted MRI and hyperintense on T2-weighted MRI and homogeneously enhanced upon contrast administration. The lesion extended into the retrosellar region, optic chiasm, and right hypothalamus with a displacement of the anterior and the middle cerebral arteries. The lesion compressed mainly temporal lobes, cerebral peduncles, and the anterior surface of the pons .The patient underwent a right pterional craniotomy, besides a subtotal tumor resection (95%). Complete excision could not be made because of its anatomical relationships.After the surgery, the child's clinical course declined with blindness of the left eye and left-sided mild hemiparesis besides focal seizures prevented by levetiracetam twice a day.Hormonal assessment in the postoperative period showed a panhypopituitarism: TSHus: 0.013 mUI/l (0.64–6.27), FT4: 7.32 pmol/l (12.1–18.6), blood plasma cortisol level at 8 am < 0.2 ug/dl, and ACTH (adenocorticotropic hormone) level <5 pg/ml.Therefore, hormonal replacement medications have been immediately introduced including hydrocortisone (12.5 mg/day), levothyroxine (25 ug/day), and oral desmopressin (0.2 mg/day).Histological examination revealed monomorphous bipolar cells with a generous myxoid matrix and an angiocentric disposition of the tumor cells. Rosenthal fibers and eosinophilic granular bodies were not identified, with a very limited number of mitotic figures without any necrotic tissue. Immunochemistry showed oligodendrocyte lineage transcription factor 2 (oligo-2) and S-100 positively stained cells while they seemed negative for IDH1 and P53. The proliferation index of Ki-67 was about 8%.During a 6-month follow-up, the patient's clinical status was stable since the postoperative phase. MRI of the hypothalamic-pituitary axis showed a residual suprasellar mass with a sellar component compressing the optic chiasm, cerebral pedicles, and pons anterior surface, with the cystic component extending to third and lateral ventricles. Otherwise, a cystic lesion of nearly 17 mm was also noticed. Chemotherapy was suggested, instead of surgical reintervention based on vincristine and carboplatin. The patient died after 2 sessions due to medullary anaplasia.PMA was first singled out by Tihan et al. in 1999 [In 2007, WHO classification [In light of recent studies concerning the extensive histology and genetics at the crossroads between PMA and PA, it is suggested to put PMA between brackets until further behavior clarification.Regardless of thic academic classification, well-conceived cohort studies with considerable evidence showed indisputably that PMA tends to behave more aggressively than PA implying increased mortality rates and a poor disease-free survival .Up to date, PMA incidence statistics remain unknown, but they are rarely reported in the literature . HoweverThese tumors concern mostly young children generally between 3 months and 2 years. American literature mentioned an overall diagnosis mean age of 18 months for PAs . In our The clinical manifestations of PMA match with those of other pediatric brain tumors including developmental delay and altered level of consciousness, besides hydrocephalus and generalized weakness along with focal neurological symptoms. Endocrine dysfunction and visual impairment such as nystagmus have been described as a pathognomonic sign in 55% of cases .A rare presentation of hypothalamic/chiasmatic neoplasm is failure to thrive with generalized loss of subcutaneous fat also known as diencephalic syndrome. This syndrome was first described by Russel in 5 children with hypothalamic astrocytoma .In fact, failure to thrive was the presenting symptom in our case, without any defects suggesting chronic malnutrition, allying especially deep emaciation with nystagmus.The overall uncommoness of this syndrome in this situation causes a considerable time gap between symptom onset and diagnosis.Failure to thrive takes part of the most frequent troubles in paediatrics. It may be an outcome of diverse disorders such as nervous anorexia; absorption issues (Crohn's or celiac disease …); or any other chronic illness. Subsequently, an uncommon trouble should be suspected if dietary and behavioral interventions have been useless . In factIn rare cases, different types of tumors such as ependymomas, dysgerminomas, or gangliocytomas have been associated with diencephalic syndrome. But sometimes, the causative tumor is unclassified .β-lipotropin) lead to lipolysis increase and reduction of subcutaneous adipose tissue [Diencephalic syndrome is rarely described despite its morbidity, which may be responsible for a diagnosis delay for about 11 months from symptoms onset. The initial clinical feature is generally severe emaciation even if the caloric intake is correct . Other se tissue . Therefoe tissue .MRI imaging  is the gold standard in delineating the central nervous system and seems helpful to identify differential diagnosis and plan the therapeutic strategy. While PA is the most prevalent type of brain tumors in childhood with precise radiographic features, there are few reports describing imaging findings of PMA. It is commonly believed that they can occur anywhere along the neuraxis, but most of them tend to be located in the chiasmatic/hypothalamic region and almost always arise from the midline.Komotar et al.  analyzedOthers  reportedLiterature review  noticed Using proton MR spectroscopy  demonstrIn our presentation, the MRI showed a suprasellar solid mass measuring 59∗41∗40 mm isointense on T1-weighted MRI, hyperintense on T2-weighted MRI, and homogeneously enhanced after gadolinium injection, extended to the optic chiasm and right hypothalamus.Obviously, differential diagnosis between PA and PMA seems to be challenging on the ground of imaging findings alone. Histopathological study along with immunohistochemistry of these tumors remains the cornerstone of diagnosis approach and prognosis purposes as well.The origin of PMA's cell is still ambiguous compared with PA. The bulk of studies proposed an astrocytic origin whereas others lean towards the ependymal or radial glia source .This tumor is identified by monomorphously organized little bipolar cells with spindle-shaped nuclei and a considerable myxoid background. They usually spread from vessels in an angiocentric pattern (pseudorosettes).PMAs are commonly compact and noninfiltrative, but they can present infiltration and entrap typical brain structures. Rosenthal fibers and eosinophilic granular bodies are missing. Focal necrosis is almost never seen .Immunohistochemical features of PMA are practically close to those of PA. On the one hand, they are positive mainly for GFAP , S100 protein, vimentin, Olig2 (oligodendrocyte transcription factor), and S0X10 (SRY-related HMG-box). But, on the other hand, they are negative for neuronal markers  such as In our case, histopathology and immunochemistry describe classical features.The treatment of PMA is still a debatable challenge for every therapist. Moreover, there is no consensus for standard therapy. In fact, surgery or, particularly, gross total resection has an impact on diagnosis, tumor control, and relief of the mass effect over brain elements.Due to the toxicity of chemotherapy or radiotherapy for infants, adjuvant therapy may be instituted after tumor recurrence following initial gross total resection in case of partial resection with neurological impairment or high Ki-67 index.In general, chemotherapy is used regardless of the child's age. Nevertheless, radiotherapy is limited to children above 3–5 years of age .The discovery of mTOR and MAPK pathway alterations allowed revolutionary targeted therapy trials .In our case, the surgeon decided to proceed with subtotal tumor resection (95%). Complete excision could not be made because of its anatomical relationships, and chemotherapy was indicated after bringing out a residual mass with the cystic component in 6 months postoperative MRI. This adjuvant therapy was required based on our patient's neurological disorders  after the partial tumor excision.As PMA's clinical experience is not yet rich enough, precising the prognosis of this tumor is a hard labor. As a point of fact, PMA showed a tendancy of aggressivity in comparison with PA. Moreover, a higher rate of local recurrence and dissemination in the cerebrospinal fluid was observed among patients with PMA. Progression-free survival is even shorter than that of PA .Our patient's chemotherapy protocol was designed as follows: an induction period followed by maintenance chemotherapy, for a total of 10 cycles. The assigned chemotherapy medications were vincristine and carboplatin. Unfortunately, our patient died after 2 sessions of chemotherapy due to medullary anaplasia.According to numerous studies, there is no correlation between BRAF status and the clinical outcome. Nevertheless, others mentioned that KIAA1549-BRAF fusion may improve progression-free survival .Further studies are necessary to identify specific tumor's molecular/genetic abnormalities in order to underline effective therapy and elucidate the patient's outcome.The nonspecific clinical aspects of diencephalic syndrome and its fluctuating presentation deeply reflects the lack of understanding of its pathogenesis, and we should keep in mind the necessity of brain MRI within etiological investigations to make diagnosis as early as possible and plan an adequate therapy. We report a rare and neglected presentation of PMA revealed by diencephalic syndrome. That is why every clinician should think of brain neoplasm as a cause of unexplained failure to thrive in childhood.


Ameloblastoma is a rare tumor, benign but rapidly extensive and prone to recurrence. Its management remains difficult and its treatment relies mainly on surgery. For giant ameloblastoma or in people with an advanced stage the gesture remains very mutilating.This is a 22 year old patient, the onset of the symptomatology dates back to 2 years ago with the appearance of a mandibular swelling that increased in size with dental mobility. this motivated the patient to consult a CT scan and a panoramic radiograph as well as a biopsy that objectified an ameloblastoma. He benefited from a surgical excision with reconstruction.It is a rare tumor that mainly affects young people and especially males. It can be discovered by chance or generally in front of a mandibular swelling. Radiological examination is essential as well as biopsy to confirm the diagnosis. The treatment is surgical, which consists of an exeresis with safety margins. Reconstruction should be discussed especially for young healthy subjects.Although ameloblastoma remains a benign tumor, it is a tumor that evolves rapidly and recurs a lot, which is why it is necessary to take care of it quickly with a radical treatment and a regular follow-up with the patients. •Ameloblastoma is a rare benign but locally aggressive tumor that recurs frequently.•Ameloblastoma remains a benign tumor whose management must be rapid to avoid significant loss of substance because the choice of reconstruction remains difficult.•The maxillofacial surgeon must choose between different treatments with a regular follow-up of the patient. It is an epithelial neoplasm developed from dento-forming cells without a mesenchymal component  see Fig, Fig. 5Clinically ameloblastoma manifests as a jugal or symphyseal swelling all depends on the location. The diagnosis is made by panoramic radiography and CT scan. The confirmation is done by anatomopathological examination. Management must be rapid so that the procedure is as non-mutilating as possible. Regular monitoring is necessary to avoid any recurrence.L'a^ge médian de survenue est de 35 ans, les deux sexes sont également affectés, avec une prédominance chez la race noire pour certaines études. La majorité des améloblastomes sont polykystique et sont plus difficiles à éradiquer que les variétés mono-kystiques et périphériques .Ces ame ´loblastomes se divisent en ame ´loblasto-mes intra-osseux (centraux) et tissulaires (pe ´riphe ´riques). Il existe, par ailleurs, diffe ´rents types histologiques d'ame ´lo-blastomes Nous allons presenté un cas d'ameloblastome dont le diagnostic sest fait tardivement et donc la chirurgie a été mutilante d’ ou linteret d'une prise en charge rapide.This case report has been reported in line with the SCARE Criteria(2) .2The patient was 22 years old with no particular pathological history. The onset of the symptomatology dates back to 2 years ago with the appearance of a painful mandibular swelling of the mandible progressively increasing in volume with dental mobility, which motivated the patient to consult . On clinThe patient was seen at the consultation with good improvement and good healing. The anatomopathological examination confirmed the nature of a follicular and plexiform ameloblastoma without any sign of malignancy.3Ameloblastoma is a benign tumor of odontogenic origin, locally aggressive but slowly progressive. Its origin is the epithelium involved in tooth formation, namely the enamel organ, the epithelial remnants of Malassez, the wall of odontogenic cysts (dentigers) or also the basal epithelial cells of the oral mucosa .Ameloblastoma are rare and represent only 1% of all maxillary tumors and 11% of odontogenic tumors .The etiology is of unknown origin; so far, no potential risk factors have been identified.Ameloblastoma affects with predilection the male sex, it seems that the disease is more frequent in black subjects  as it isAmeloblastoma most often involves the mandibular more than 80%  the anguAmeloblastoma is characterized by a slow but rapidly extensive evolution which makes the management difficult and the surgical procedure very mutilating.The most frequent reason for consultation is facial swelling, whether mandibular or maxillary.The delay in consultation can be explained either by the slow evolution of the tumor, the negligence due to the fact that the tumors are painless and of slow evolution let think that the process will regress spontaneously, the low socio-economic level and the geographical distance of the patients .Most of the time, ameloblastoma are discovered by chance, by radiography made for other reasons.Ameloblastoma has a general histologic structure made up of a center formed by islands that have radiated epithelial cells with a loose texture with frequent formation of microcysts, and a periphery consisting of an epithelium whose cells have an inverted nuclear polarity and supra nuclear vacuoles .There are many histological types: follicular, uni-cystic, plexiform, peripheral, desmoplastic.The radiological examination is essential for the diagnosis, it allows us to orientate ourselves but only the anatomopathological examination allows to confirm it.Several types of images can be found at the radiological level:A unilocular cystic image that can be confusing.A multilocular image that translates into a honeycomb or soap bubble image.3.1In the event of a delay in diagnosis, do not hesitate to request a scan in addition to an orthopantomogram.On scan, it is usually an extensive tumor surrounded by a bony shell, with irregular boundaries with hypo dense cystic areas and solid iso dense areas with contrast of the bony component after injection .The 3D reconstruction gives us information on the limits of the tumor and its relationship with the lower dental canal.The IRM is indicated when the tumor arrives at the level of the soft parts.Imaging is used to guide the diagnosis and follow-up of ameloblastoma.Ameloblastoma recurs frequently, so a radical treatment is necessary. There are two types of treatment: conservative treatment by marsupialization, enucleation and curettage, and radical treatment which consists of resection of the bone tissue while respecting the margins of exeresis.The treatment is surgical, it is generally mutilating especially for advanced cases. The surgical treatment can be conservative or radical, the therapeutic decision must take into consideration several factors including the age of the patient, the anatomical location of the lesion, its extension, the radiological aspect, the evolutionary potential and the probabilitý of a regular follow-up of the patient .Even after apparently well-conducted surgical treatment and despité wide resections with pathologically healthy recuts, ameloblastoma is likely to recur probably because of micro tumor bone foci existing distant from the main tumor .Many authors advocate a wide resection at the outset with a safety margin in the periphery .4Ameloblastoma is a rare tumor, benign but locally very aggressive, its treatment is mainly surgical. The surgical gesture can be very mutilating especially when the diagnosis is made late, hence the interest of a rapid management with a radical surgical treatment and limits of wide exeresis to avoid as much as possible a recurrence.Proton beam irradiation would likely be advantageous for patients with maxillary ameloblastomas extending to the skull base to adequately treat the tumdor and reduce the dose to the central nervous system and visual system .


Reduction en mass (REM) is one of the rare complications of inguinal hernia reduction. Although REM can be detected on the basis of specific computed tomography (CT) scan findings, many radiologists are not familiar with its radiological appearance because of the scarcity of this complication, which may cause a delay in diagnosis.The patient reported in this article was a 50-year-old Persian man with a history of inguinal hernia, who had been referred with the periumbilical pain that radiated to the right lower quadrant and developed following hernia replacement by the patient himself. REM diagnosis was based on clinical examination and CT scan findings, and surgical treatment was performed by the Lichtenstein repair and mesh implantation.Although REM usually occurs after reduction with compression in the inguinal hernia, this unique case report highlighted the possibility of REM after self-reduction. Surgeons and radiologists should consider REM in patients with a history of inguinal hernia presenting with intestinal obstruction symptoms, even without any apparent signs of hernia in the physical examination. Reduction en mass (REM) is one of the rare complications of inguinal hernia reduction inside the abdomen. REM is defined as the placement of the herniated sac inside the abdominal wall while the contents of the sac remain incarcerated or strangulated. Commonly, this sac stays in the preperitoneal space .REM usually occurs after reduction with compression in the inguinal hernia. Although REM can be detected through specific computed tomography (CT) scan findings, many radiologists are not familiar with its radiological appearance because of the scarcity of this complication, which may delay the diagnosis . Here weA 50-year-old Persian man was admitted with periumbilical pain that radiated to the right lower quadrant from 12 hours earlier. The patient had a history of right inguinal hernia 3 years ago. Previously, he was hospitalized twice because of incarceration and was a candidate for surgery, but he had not consented to surgery. The patient also had a history of ischemic heart disease and right hip arthroplasty. Regarding habitual history, he consumed opium orally.A day prior to admission and following inguinal hernia reduction by himself, the patient developed constant pain and a consequent inability to pass gas or stool. On physical examination, periumbilical and right lower quadrant tenderness (RLQ) was revealed. There was no acidosis or leukocytosis in the blood tests. Since there was no evidence of the previous hernia on the physical examination and the patient presented intestinal obstruction symptoms, including bilious vomiting 2–3 times, the medical team decided to obtain a computed tomography (CT) scan of the abdomen and pelvis Fig. a. CT scaThe patient underwent laparotomy with a midline incision. Adherence to the wall of the hernia sac, and slight discoloration in a 5-cm-long intestinal loop approximately 50 cm before the ileocecal valve, which improved after about 10 minutes, were observed inside the inner ring of the inguinal canal  is reported to have the best performance for detecting groin hernias with 95% sensitivity and 96% specificity. However, because of the high cost of MRI, a CT scan with a sensitivity of 80% and a specificity of 65% and ultrasonography with a sensitivity of 86% and a specificity of 77% can be used instead to detect inguinal hernias [Prompt surgical treatment is essential because any delay will exacerbate the symptoms and cause inevitable complications . InteresThis unique case report highlights the importance of REM after self-reduction. Besides, this report stresses that a patient with a history of inguinal hernia and intestinal obstruction symptoms should be considered for REM, even without any apparent signs of hernia in the physical examination.


Staphylococcus aureus, but other bacterial isolates may be emerging. It is a novel disease for this age group and requires unique management. Only 32 cases of neonatal suppurative parotitis have been described in the English-language literature over the last 35 years.Acute suppurative parotitis is a rare finding in the neonate. It is commonly caused by We describe a case of a 14-day-old male who presented to the pediatric emergency department with a 24-hour history of swelling and redness of the right cheek. On examining him, purulent material was seen inside his oral cavity. He was subsequently hospitalized with a diagnosis of neonatal suppurative parotitis and received five days of parenteral antibiotics with improvement in swelling and redness. He was discharged home with oral antibiotics.Although neonatal suppurative parotitis is rare, it should be suspected in newborns presenting with an erythematous pre-auricular mass with or without any predisposing factors. We describe a rare case of acute suppurative parotitis in a neonate and review the published literature. Staphylococcus aureus, but other bacterial isolates may be emerging. It is a novel disease for this age group and requires unique management. We describe a case and present a literature review of neonatal parotitis based on a 14-day-old male who presented to the pediatric emergency department (ED) with swelling and redness of the right cheek and was diagnosed with acute suppurative parotitis.Acute suppurative parotitis is a rare finding in the neonate. It is commonly caused by A 14-day-old male presented to the ED with a 24-hour history of swelling and redness of the right cheek. He had been doing well since birth. His mother noticed that he was crying between his feeds and appeared fussier. The parents denied fever, any kind of rash, or contact with a sick person. The cheek swelling was associated with the skin redness. The baby was born at 38 weeks twin delivery, cesarean section, no complications at birth, and was gaining weight appropriately.In the ED, the patient was afebrile with normal vitals for his age. On exam, he had right facial redness and crying with palpation of the right cheek . The lefS. aureus. After 48 hours of IV antimicrobials rapid clinical improvement was noticed. After five days of IV antibiotics, he was transitioned to oral clindamycin for 10 days with a resolution. He was sent home and the parents were advised to follow up with his pediatrician.His laboratory tests showed a normal complete blood count. His comprehensive metabolic panel was also within normal limits except for total bilirubin and direct bilirubin levels . A respiWhat do we already know about this clinical entity?Neonatal suppurative parotitis is a rare condition. Diagnosis is made clinically with purulent material exuded from the Stensen duct being a pathognomonic sign.What makes this presentation of disease reportable?This patient presented with purulent fluid coming out of his mouth and redness of face. Use of point-of-care ultrasound (POCUS) with clinical findings, prompted early laboratory testing and antibiotics.What is the major learning point?Use of POCUS and cultures from purulent fluid help in diagnosis and need for full sepsis workup as suppurative parotitis can lead to bacteremia and meningitis.How might this improve emergency medicine practice?Neonatal suppurative parotitis is an uncommon infectious presentation without fever needing prompt recognition and management.Neonatal suppurative parotitis is an uncommon disease, with a prevalence of 3.8:10,000 of neonatal admissions in an Italian study by Speigel et al in 2004.The parotid gland is more frequently infected than the other salivary glands because of its exclusive serous secretion without the bacteriostatic properties of the mucoid component.6Staphylococcus aureus is the most common pathogen isolated. Less-frequent agents are other Gram-positive cocci , Gram-negative bacilli , and rarely anaerobic agents .S. aureus.The diagnosis is based on clinical findings. In most cases NSP was unilateral and the most prevalent sign at the time of admission was swelling with or without redness of the parotid region. Fever is found in less than half of the cases. Bacteremia is present in up to 90% of cases and there can be meningitis associated with 33%. Purulent material exuded from Stensen’s duct is a pathognomonic sign of NSP.9The differential diagnosis includes infectious and non-infectious etiologies. Infectious causes include lymphadenitis, cellulitis, soft-tissue abscess, osteomyelitis, buccinator muscle infection, and parotitis.5Ultrasound findings can confirm the diagnosis, as in our case. Ultrasound can also be useful to identify soft tissue abscesses requiring surgical intervention, or non-infectious etiologies.13S. aureus, other Gram-positives such as GBS, and Gram-negatives. Based on the possible anaerobic causes, metronidazole was added. Most authors recommend starting therapy with an association of an anti-staphylococcal agent and an effective antibiotic against Gram-negatives, with adjustments after the results from the cultures are available.The diagnosis of NSP in our patient was made based on clinical findings and with ultrasonography confirmation. After collecting the blood cultures, urine cultures, and CSF cultures, empiric IV antibiotic therapy with vancomycin and cefepime was immediately started. This covered the most common pathogens such as Most cases are managed conservatively, with prompt antibiotic therapy and adequate hydration essential for a good outcome. Surgical intervention is reserved for the rare cases with an inadequate response to medical therapy or for those with organized abscesses.14S. aureus is the most common pathogen isolated from infants with NSP. Exudate culture should be sent for testing. Most patients can be treated conservatively, provided that the empiric antibiotic treatment covers the causative agents according to the local sensitivity pattern and is started early. The prognosis of the disease is generally excellent.Although neonatal suppurative parotitis is rare, it should be suspected in newborns presenting with an erythematous pre-auricular mass with or without any predisposing factors.


Dear Editor.Focal scleral nodule (FSN), renamed by Fung et al. in 2020 , is charWe would like to describe 2 cases. Our first case was a 56-year-old woman who was found to have an abnormality in her right fundus. Her medical and family history was unremarkable. The patient’s best-corrected visual acuity (BCVA) was 20/20 OD. Funduscopic examination revealed an orange lesion of less than two-disc diameter on the inferonasal site of the macula Fig. . FluoresThe LSFG color map of mean blur rate (MBR) showed localized cooler color at the lesion  than at the macula , indicating blood flow was disrupted in the FSN site Fig. .Our second case was a 56-year-old woman who was referred to our clinic because of a yellowish-white lesion in her left macula. Past medical and family history was unremarkable. BCVA was 20/16 OS. The fundus and other findings were similar to those in Case 1 Fig. , but theOn LSFG, the lesion showed a cooler color Fig. .Since the yellowish-white lesion in this disease was once considered an inflammatory disease of the choroid, Hong et al. named it unifocal helicoid choroiditis in 1997 . However


A case of a purely eccrine nevus in an adolescent patient presenting with focal hyperhidrosis on an area comprising the left forearm and the dorsal aspect of the left hand is described. No clinically evident lesions were identifiable. Dermatopathologic findings were subtle, showing only a slight increase in the number of eccrine glands. Clinicopathological correlation was paramount to achieve the diagnosis. Hyperhidrosis is a skin disorder characterized by excessive sweating. It can affect at general, regional, or focal levels. It may have a primary origin, with excessive diaphoresis generally appearing in localized and symmetrical areas, such as axillae, palms, soles, or craniofacial region. Alternatively, it might be secondary to multiple causes, having more frequently a generalized involvement. Some series have quantified primary cases at more than 90%, with secondary cases being infrequent. Regarding the age of onset, most patients initiate excessive sweating between 14 and 25 years of age. However, given the full functionality of the eccrine glands from birth, it can be observed in infants and children [Uncommon causes of secondary focal hyperhidrosis include eccrine nevi. These are considered hamartomas, characterized by hyperplasia and/or hypertrophy of eccrine glands. Eccrine nevi usually affect the forearms  and present during childhood or adolescence . The anaThe diagnosis of this entity is based on clinical suspicion followed by pathological confirmation. In this manuscript, we present a case highlighting the importance of this clinicopathological correlation.A 16-year-old female patient with no relevant past medical history came to our clinic for evaluation of excessive sweating on an area comprising the left forearm and the dorsal aspect of the left hand. This focal hyperhidrosis started when the patient was four years old, disturbing her during the colder seasons , and worsened with stressful situations.Physical examination revealed no apparent cutaneous lesions a. The stA punch biopsy was performed. The biopsy showed a slightly increased density of eccrine glands within the dermis, with no other abnormalities .The patient was treated with multiple intradermal injections of botulinum toxin type A on the affected area, with a satisfactory response, remaining with anhidrosis seven months after follow-up.Eccrine nevi are rare lesions included in the group of organoid nevi, with few reported cases in the literature. Clinical features of eccrine nevi are variable, including their age of presentation, number, location, size, signs, and symptoms ,5.Most of the cases are only clinically evident when excessive sweating appears, as happened in our case. In some instances, it can be challenging to distinguish eccrine nevi from some forms of idiopathic localized unilateral hyperhidrosis. This hyperhidrosis is focal, on sharply demarcated areas of less than 10 × 10 cm in size, and occurs in short episodes frequently worsened by stress ,6. Howev-Angiomatous eccrine nevi (or sudoriparous angiomas) show an augmented number of capillaries .-Eccrine-pilar angiomatous hamartoma comprises hair follicles associated with the eccrine angiomatous complexes .-Mucinous eccrine nevi have abundant mucin deposits around the increased number of ducts of the eccrine glands ,12.Histopathologically, purely eccrine nevi—such as the depicted case—are considered infrequent . They prThere are multiple options to treat eccrine nevi. Conservative management is an option if they remain asymptomatic or with mild symptoms. Topical antiperspirants such as aluminum salts plug the eccrine sweat pores, leading to the degeneration of secretory cells. Anticholinergic drugs such as glycopyrrolate work by competitively blocking the activation of eccrine glands. These may yield positive therapeutic results but usually need repeated applications  and are A case of a purely eccrine nevus in an adolescent patient presenting with focal hyperhidrosis has been described. Dermatopathologic findings were subtle, and clinicopathological correlation was paramount to achieve the diagnosis.


Left displacement of the third gastric compartment (LDC3) in alpacas is an extremely rare condition and has not been reported thus far. Therefore, we describe the clinical diagnosis and treatment of LDC3 in an alpaca.Vicugna pacos) was presented to evaluate a 3-day history of abdominal distension causing loss of both appetite and thirst, along with oliguria and low to no defecation. Clinical examination, X-ray examination, surgical exploration, and determination of gastric pH (pH ~ 2.35) confirmed that LDC3 resulted in abdominal distension. The gastric wall of the displaced third gastric compartment was incised for the expulsion of pneumatosis, and a medical-grade silicone tube was inserted into the incision to remove the effusion by siphoning. Surgical treatment proved to effectively alleviate the abdominal distension caused by LDC3 without apparent side effects.A 2-year-old brown female alpaca (To our knowledge, this case is the first known report of LDC3 in an alpaca in China. A similar condition, left displaced abomasum, has previously been described in cattle and sheep. Since its initial identification in 1950, left displaced abomasum (LDA) has been increasingly observed in cattle . Such diVicugna pacos), a member of the South American Camelids (SACs) family, belongs to the suborder Tylopoda of the order Artiodactyla, whose gastric anatomy is adapted to the physiological process of rumination and the degradation of plant cell wall contents to a great extent [Alpaca  in alpacas is similar to the abomasum of cattle and sheep in functions such as the secretion of gastric juices and food digestion . Thus, iA 2-year-old, 58 kg brown female alpaca was presented to the Animal Hospital of Heilongjiang Bayi Agricultural University  to evaluate abdominal distension. For ornamental purposes, the alpaca was purchased into a sighting agricultural park of Daqing in Heilongjiang Province in China. The alpaca was fasted on the day of purchase but allowed free access to water. On Day 3, it was fed large quantities of carrots and a small amount of alfalfa green grass. During this period, the alpaca presented with unformed stool followed by loss of both appetite and thirst, along with oliguria and low to no defecation. The sick alpaca was treated in our hospital on Day 6.On admission, the alpaca exhibited overall malnutrition and abdominal distension Fig. . The recAbdominal X-ray showed an oval, tubular, notably demarcated, low-density shadow (suggestive of effusion and pneumatosis) in the middle portion of the left posterior abdomen Fig. A. The paSurgery was performed as previously described for LDA in cattle . BrieflyNext, a gastric wall incision of approximately 1 cm was made to release gas. A medical-grade silicone tube was inserted into the incision to reduce abdominal bloating and drain most of the contents appeared yellowish-brown and muddy in appearance, with a rancid odour Fig. B. The pHThe aetiology of LDA in cattle is multifactorial . AbomasaThe diagnosis of LDA in cattle is based on clinical signs and auscultation and percussion of the abdomen. As previously reported for LDA in cattle, the alpaca with LDC3 in this case report had a normal rectal temperature, heart rate and respiratory rate , 16. As In the case reported here, abdominal X-ray examination was a simple, non-invasive approach of confirming a clinical suspicion of LDC3 and monitoring the position of displaced C3. The normal radiographic appearance of C3 in alpacas has been described previously . ImagingIn the alpaca described here, surgical exploration supported a clinical suspicion of LDC3. Abdominal exploration revealed no anatomic abnormalities or adhesions and concomitant or concurrent disorders. The determination of gastric pH also supported a clinical suspicion of LDC3. In addition, hypochloremic and hypokalemic metabolic alkalosis is the most common metabolic change in cattle diagnosed with LDA. Blood gas and electrolyte analysis have been used to support a clinical suspicion of LDA in previous studies , 25. BasSurgery to correct abomasal displacements likely accounts for the majority of nonelective surgical procedures in cattle but is uncommon in small ruminants . MultiplOur case highlights the diagnosis and management of previously unreported LDC3 in an alpaca presenting with overall malnutrition and abdominal distension. Left displacement of the third gastric compartment should be considered in the differential diagnosis for SACs with abdominal distention. The combination of clinical examination, X-ray examination, surgical exploration, and gastric content pH detection is useful in the diagnosis of LDC3. Surgical correction is highly effective for treating LDC3 in alpacas. Importantly, this is the first case report of alpaca LDC3 in China, which aims to further improve the knowledge of LDC3 in a previously unreported species and to help veterinarians diagnose and treat this disease by borrowing the clinical approaches to LDA in cattle and sheep. Further research on the blood clinicopathological features and pathoanatomical structures of LDC3 in SACs is warranted to better understand the pathology and treatment of C3 in these species.


Lipoma is a benign mesenchymal tumor of soft tissue that occurs in almost all parts of the body where fat normally exists. Retroperitoneal lipomas are very infrequent condition with about 20 cases represented in the literature since 1980. They usually present as an abdominal mass or with pressure symptoms to adjacent organs.A 66-year-old, post-menopausal woman referred to Imam Hossein Medical Center due to abdominal pain. Abdominopelvic magnetic resonance imaging (MRI) revealed a large mass containing fat component without enhancement on the right side of the pelvis. Tumor markers were within normal ranges. The patient underwent laparotomy and a 12 cm retroperitoneal mass which was located on the iliopsoas muscle with extension into the inguinal canal was resected with pathology report of lipoma. There has been no recurrence after one year of follow-up since surgery.In the differential diagnosis of retroperitoneal pelvic mass at all ages, lipoma should be considered as a rare cause. The wide spectrum of retroperitoneal neoplastic and non-neoplastic soft tissue masses, reflects the complex anatomy of this region. Of the retroperitoneal tumors, 80% are malignant with liposarcoma representing the most common histological type (45%). However, retroperitoneal lipoma is a rare disease with only few cases of this condition reported in literature. Lipomas are encapsulated benign tumors of adipose tissue which can manifest at any fat containing body part . LipomasA 66-year-old multiparous woman, with a history of nine deliveries and post-menopausal for the last ten years with a pelvic mass was referred to Imam Hossein Hospital as a tertiary medical center in Tehran, Iran. The patient’s clinical symptoms including intermittent abdominal pain in the hypogastric region began about 3 weeks ago. Other associated symptoms included urinary frequency and urgency as well as stress incontinence for past two years. The patient underwent total hysterectomy and bilateral salpingo-oophorectomy about 8 years ago due to uterine prolapse. Her past history also included hypertension, hyperlipidemia, and well-controlled diabetes.In bimanual examination, left adnexal fullness was detected. On abdominal examination, we were unable to palpation of the mass due to the patient's obesity. Trans-abdominal ultrasonography showed an 84×103×112 mm heterogeneous mass with lobulated border on the right side of the pelvis extending to the right lower abdominal quadrant, with no vascular flow on color Doppler. Abdominopelvic magnetic resonance imaging (MRI) with and without contrast revealed a 150×77 mm large non-enhancing fat containing mass, suggestive of lipoma, located on the right side of pelvis extending along the anterior aspect of iliopsoas muscle . Cancer The patient underwent laparotomy with midline incision. In the abdomen, a 12 cm long retroperitoneal mass was observed on the right side of the pelvis. The mass was located on the iliopsoas muscle, and extended to the inside of the inguinal canal. The mass had a lipoid appearance and consistency and was completely resected without rupture of capsule . Lipoma Lipoma is a benign encapsulated tumor of adipose tissue, mostly manifested in fifth and sixth decades of life and has no gender predominance , 4. The Familial tendency is observed in some lipoma cases . RetropeThese soft and mobile masses are typically asymptomatic and grow slowly; but over time, the pelvic lipomas can reach considerable sizes and become symptomatic . A diverDistinguishing retroperitoneal lipomas from well-differentiated liposarcomas could prove difficult on imaging, and also biopsies may be confusing. Abdominal masses are frequently assessed by ultrasonography as the initial imaging modality. On ultrasound, lipoma appearance is of an oval hyperechoic mass with well-defined regular margins. Thin fibrous septa may be detected within the mass, and there is a lack of intra or perilesional vascularization on color Doppler examination . In presPercutaneous biopsy is controversial due to the risk of local spreading through the implantation of neoplastic cells along the route of the puncture, should it be a case of liposarcoma. Management of retroperitoneal lipomas is surgical resection and extreme caution to preserve the integrity of the capsule. Encapsulated presentation of the tumor presents a clear cleavage plane with surrounding structures, facilitating the surgical resection .In gross examination, lipomas are encapsulated tumors with multi-lobed appearance resulting from yellowish rosy colored tumor with interspersed whitish fibrous septa, similar to our patient. It is noteworthy that low grade liposarcomas (grade I) may have the same delimited appearance .Less thaIn conclusion, in the differential diagnosis of retroperitoneal pelvic mass at all ages, lipoma should be considered as a rare cause.


Lipofibromatosis is a rare, benign, soft tissue tumor that usually presents in children. Low incidence and lack of specificity in clinical presentation make its diagnosis difficult.This is a case report of a patient with a giant lipofibromatosis on the back that resembles an infantile hemangioma, which posed great difficulty in diagnosis due to atypical clinical manifestations. After the postoperative pathological and immunohistochemical examination and fluorescence in situ hybridization, the patient was finally diagnosed with lipofibromatosis.The incidence of fibromatosis was low. This case presents an atypical clinical manifestation since the tumor growth was on the back, and this can easily cause misdiagnosis. This case suggests that the diagnosis of lipofibromatosis depends on the pathology and fluorescence in situ hybridization. Lipofibromatosis is a rare, benign, soft tissue tumor that develops mainly in children and is congenital in approximately one-fifth of cases . It was 9/L, 3.98 × 1012/L, 112 g/L, and 436 × 109/L, respectively. On laboratory tests, no significant abnormality was found. A small portion of the tumor was taken for histopathological examination, and lipofibromatosis was suspected. Upon consulting at the pathology department of another hospital, fibrous hamartoma of infancy was suspected. The mass was subsequently excised completely under general anesthesia with preoperative blood preparation  fibromatosis, fibrous hamartoma of infancy (FHI), calcifying aponeurotic fibroma, lipoblastoma, lipoma, neurofibroma, and lymphatic malformation , 11. HerIHs are the most common benign tumors in infancy and childhood. They may appear as erythema shortly after birth and gradually increase in size. They can grow up to nine months of age and then gradually fade away . AccordiKHE on the body surface usually presents as a tough skin or subcutaneous mass with a purplish-red, nodular or plaque appearance, and poorly defined margins due to petechiae or dilated capillaries. KHE or TA can cause the Kasabach-Merritt phenomenon, a class of clinical manifestations associated with thrombocytopenia, microvascular hemolytic anemia, and consumptive coagulation dysfunction, in addition to vascular malformation. Confirmation of KHE diagnosis requires pathological evidence. KHE lesions are characterized by a large number of nodules of varying shapes and fuzzy borders infiltrating the surrounding area. The internal morphology of the nodules combines the features of both IH and Kaposi’s sarcoma. There were clusters of fusiform endothelial cells and highly tortuous, tangled microvessels, as well as a large number of poorly developed slit-like lumina filled with red blood cells. The thin-walled crescent-shaped vascular cavities and enlarged lymphatic cavities surrounding the tightly compressed lobules of the microvessels in TA lesions are also characteristic features of TA .FHI usually occurs in children under two years of age, and approximately 20% of cases present as congenital lesions. A painless subcutaneous mass involving the axillae, trunk, upper arms, and external genitalia usually develops . The chaLPF-NT is the most difficult to identify. Agaram et al. first reported LPF-NT in 2016 as a rare, soft tissue tumor that occurs mainly in the trunk and extremities of children and young adults. The tumor consists of CD34-positive spindle cells, similar to the infiltrative growth pattern of lipofibromatosis. The two are distinguished by positive S-100 protein expression and NTRK1 gene rearrangement in LPF-NT . Wang etIn conclusion, a case of lipofibromatosis with an atypical clinical presentation and confusing history, appearance, and location was reported. Thus, for such a large or rapidly enlarging lesion, depth and extent can be confirmed by imaging first, and the diagnosis can be made by biopsy. This also suggests that the diagnostic process of lipofibromatosis can be further improved, and easier and faster methods can be developed to help physicians detect and intervene early.


Beckwith‐Wiedemann syndrome is a complex multisystem disorder that requires collaboration of medical and dental teamfor its diagnosis and management. We present a dental overview and an update of the clinical and molecular diagnoses of Beckwith‐Wiedemann syndrome and its management with emphasis on macroglossia. A 1‐year‐old male infant diagnosed with a rare congenital disease Beckwith‐Wiedemann syndrome (BWS) since birth presented with macroglossia, hemihypertrophy, and mild bilateral nephromegaly. Surgical debulking of the enlarged tongue was performed. This report emphasized the significance of tongue enlargement in the clinical diagnosis of BWS and case management.Beckwith‐Wiedemann syndrome is a complex multisystem disorder that requires collaboration of medical and dental teamfor its diagnosis and management. We present a dental overview and an update of the clinical and molecular diagnoses of Beckwith‐Wiedemann syndrome and its management with emphasis on macroglossia. One of the target features to be addressed is macroglossia, since approximately 90% of patients diagnosed with BWS exhibit this feature.In this paper, a case of BWS is presented to create further awareness and highlight the clinical features of BWS mainly macroglossia and diagnosis guidelines. In addition, we highlighted the management of macroglossia, which is the most significant complications associated with BWS.2A 1‐year‐old male infant who had been diagnosed after birth by his pediatrician with BWS, with macroglossia as the first feature that led to the diagnosis, was referred to the Oral and Maxillofacial Department at the faculty of dentistry of King Abdul Aziz University. On clinical examination that was performed by the maxillofacial surgeon, the patient presented with macroglossia, which led to an inability to close his mouth, interference with occlusion, feeding and swallowing difficulties, and drooling Figure , and hemTo confirm the diagnosis clinically, ultrasonography showed mild hepatomegaly and mild bilateral nephromegaly with no other detectable abnormalities. Accordingly, liver and kidney functions were assessed including the alpha fetoprotein level for tumor screening, and all values were within the normal limit. His vital signs and blood sugar level were normal. No molecular confirmation was performed for this case so far. These findings support the clinical diagnosis of BWS according to the scoring system, including two cardinal features and one suggestive feature. Our patient's score was 5, ie, 2 points for macroglossia, 2 points for lateralized overgrowth, and 1 point for hepatomegaly and nephromegaly.Surgical debulking of the enlarged tongue was performed by oral and maxillofacial surgeons and was performed at age 1 year under general anesthesia because he had breathing and swallowing difficulties. The patient was placed in the supine position with nasal intubation, and a modified reduction glossectomy was performed using the stellate anterior wedge procedure. A specimen of the tongue was submitted to the Oral and Maxillofacial Pathology Department. Histopathology using hematoxylin and eosin staining revealed muscular hyperplasia Figure .At discharge and after 7 days of follow‐up, the patient was well. During the follow‐up at age 2 years (1 year after the surgery), difficulties in airway and speech were not observed Figure . In conc3BWS is considered the most common congenital overgrowth disorder. Patients with BWS have diverse clinical manifestations. With this dilemma, a scoring system can aid in the diagnosis of patients as in our case. A clinical score of ≥4 based on the diagnostic scheme, with at least two cardinal features, is sufficient to satisfy the clinical diagnosis of classical BWS without molecular confirmation, as in our patient.Macroglossia is one of the primary manifestations of BWS diagnosis, reported in nearly 90% of BWS cases.Aside from the functional difficulties caused by macroglossia, BWS highly predisposes affected children to cancerous and non‐cancerous tumors. Hepatoblastoma, a form of liver cancer, and Wilms tumor, a kidney cancer, can develop. The risk of severity of these tumors depends on clinical and molecular findings.In the present case, the patient obtained a score of 5, thus confirming the diagnosis of BWS. The tongue reduction procedure is the best treatment option that proved effective in reducing complications. However, some patients may require a second reduction glossectomy at an older age. Follow‐up and monitoring by a dental team is important to maintain function and restore the aesthetic appearance of the patient.None declared.All authors have made substantial contributions: SL: collected and interpreted data and wrote the manuscript. SA: obtained patient consent, collected data, treated the patient, and revised the manuscript. AY: revised the article and approved the final version for submission. NB: involved in the analysis and interpretation of data and critically revised the manuscript.Informed consent was obtained from the parent regarding the publication of images and data.


Mixed germ cell tumors are uncommon tumors that contain two or more types of malignant, primitive, or germ cell components. This study aims to report a rare case of extragonadal mixed germ cell tumor of the sacrococcygeal area.A 4-year-old female presented with lower back swelling for two weeks. It was associated with pain and fever. Investigations showed elevated Alpha-fetoprotein and normal beta HCG levels. Magnetic resonance imaging showed a large well defined heterogeneous mass at the midline of the lower coccygeal region, which displaced the rectum anteriorly without invasion. Surgical dissection of the mass with excision of the coccyx was performed. Afterward, the patient was referred to an oncology center for chemotherapy.The emergence of extragonadal germ cell tumors may be caused by a disruption in the migration of primordial germ cells along the urogenital ridge, which then undergo a malignant transformation as a consequence of their microenvironment. Another idea states that extragonadal germ cell tumors occur when germ cells that routinely migrate into the extragonadal region during embryogenesis undergo malignant transformation. •Germ cell tumors (GCT) are rare and diverse group of heterogenous tumors.•Young people's gonads (ovary or testes) are frequently affected.•Mixed GCTs are uncommon tumors that contain two or more types.•Mixed GCTs of the sacrococcygeal region are a rare occurrence. They arThis study aims to report a rare case of extragonadal mixed GCT of the sacrococcygeal area in line with SCARE 2020 guidelines .2Patient's information: A 4-year-old female presented to the pediatric department with lower back swelling for two week duration. She was born with a normal vaginal delivery at 38 weeks of gestation and was not admitted to the neonatal intensive care unit. She was breastfed for two years and had no significant past medical or surgical histories during her upbringing. She was born from a non-consanguineous marriage, and there is no family history of a similar disease.Clinical findings: The condition was associated with pain and fever. On digital rectal examination, a firm mass was palpable on the right upper anal sphincter.Diagnostic assessment: Alpha-fetoprotein (AFP) was elevated (>1000), while beta HCG was normal. Complete blood count (CBC), sodium (Na), potassium (K), chloride (Cl), and serum creatinine all were within the normal reference ranges. Magnetic resonance imaging (MRI) showed a large well defined heterogeneous mass at the midline of the lower coccygeal region measuring (55*47mm) and slightly extended to the right side. The mass was encasing the lower two coccygeal segments and possibly invading the right puborectalis muscle. The mass was displacing the rectum anteriorly without invasion.Therapeutic intervention: Surgical dissection of the mass under general anesthesia in a prone position was performed. Coccyx was excised, and the rectum was intact. The surgical specimen was sent for histopathological examination. The result showed a mixed germ cell tumor . The largest tumor diameter was 5.5 cm.Follow-up: The postoperative course was uneventful. The patient was referred to an oncology center, and she received four cycles of chemotherapy. The last MRI reported no obvious residual or recurrent mass.3Extragonadal GCTs are GCTs with histology linked with gonadal origin. Primary extragonadal GCTs are rare, and the actual incidence of this form of cancer is unclear. It is believed that they account for 3%–5% of all GCTs . Mature The clinical course is unpredictable, with a possibility of aggressive behavior and extensive metastases . SacrocoThe most frequently used initial imaging technique is sonography, which is generally followed by MRI . PrimaryThe treatment of mixed GCTs concentrates on the most malignant components. Complete surgical excision is the primary therapy for children with endodermal sinus tumors with adjuvant chemotherapy assessed on a specific instance basis . The plaMixed GCTs, which mostly consist of teratomas with other malignant components , have been observed to be more aggressive. More than half of affected individuals die within two years due to local invasion or distant metastases  . PatientIn conclusion, the occurrence of primary mixed GCT in the sacrococcygeal region is a rare condition. Proper initial surgery with proper staging biopsies, followed by combination chemotherapy, can significantly improve these patients' prognosis.The manuscript approved by ethical committee of the University of Sulaimani.No source to be stated.Abdulwahid M. Salh: major contribution of the idea, literature review, final approval of the manuscript.Wrya N. Sabr: Surgeon performing the operation, final approval of the manuscript.Fahmi H. Kakamad, Karzan M. Salih: literature review, writing the manuscript, final approval of the manuscript.Rawezh Q. Salih, Berwn A. Abdullah, Ahmed G. Hamasaeed: literature review, final approval of the manuscript.1.The study was not registered because it is a case report.Fahmi Hussein Kakamad is the Guarantor of this submission.Written informed consent was obtained from the family of 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 commissioned, externally peer reviewed.There is no conflict to be declared.


Blepharoptosis (ptosis) is among the most common disorders of the upper eyelid encountered in both optometric and ophthalmic practice. The unilateral or bilateral drooping of the upper eyelid that characterises ptosis can affect appearance and impair visual function, both of which can negatively impact quality of life. While there are several known forms of congenital ptosis, acquired ptosis  is the predominant form of the condition. This review summarises the prevalence, causes, identification, differential diagnosis, and treatment of acquired ptosis. Particular attention is paid to the differential diagnosis of acquired ptosis and emerging treatment options, including surgical and pharmacologic approaches. Literature cited in this review was identified via a broad search of the PUBMED online database for English-language, peer-reviewed publications including search terms such as “ptosis,” “epidemiology,” “etiology,” “eyelid,” “surgical,” “pharmacologic,” “Müller’s muscle,” “adrenergic,” “visual field,” and “quality of life.” Relevant primary and review articles were reviewed and cited when providing unique primary data or a current summary of fundamental concepts. Also included, when relevant, were primary or review articles not identified via PUBMED, but cited in publications retrieved via this literature search.Blepharoptosis, more commonly known as “ptosis,” is an abnormal drooping of the upper eyelid with the eye in primary gaze. This drooping can affect one or both eyes, and based on time of appearance, it is broadly classified as either congenital (present at or shortly following birth) or acquired (appearing later in life). Ptosis is broadly recognised as being among the most common disorders of the eyelid encountered in the clinic, however data from large population-based studies are limited. Estimates of ptosis prevalence are largely based on data from region-specific studies, which report rates between 4.7 and 13.5% in adult populations and support the widespread nature of the condition .–29.25–29Another known cause of ptosis is ocular surgery. A systematic literature review reported an 11.4% incidence of ptosis following ocular surgery, with the highest rate (13.4%) occurring among patients who underwent glaucoma surgery, followed by corneal (10.3%), strabismus (10.0%), cataract (9.4%), and mixed (6.5%) surgeries . PostsurPtosis following ocular surgery can be transient or persistent. Factors suspected of causing transient postsurgical ptosis include the occurrence of postsurgical oedema, haematoma, foreign body reaction, and use of neuromuscular blockade, while proposed mechanistic causes of more persistent postsurgical ptosis include the use of mitomycin C in glaucoma surgery, direct trauma to the tarsal plate, bridle suture use (with higher incidence occurring with a closed approach), and rigid eyelid speculum use, which can lead to levator aponeurosis dehiscence or detachment from the tarsal plate –35.Similarly, transient ptosis has been reported as an adverse event following periocular neurotoxin injection –38. A brrd cranial nerve palsy, CPEO, oculopharyngeal muscular dystrophy, Horner’s syndrome, and myasthenia gravis [Ptosis can also be secondary to a range of underlying neurological or muscular conditions, including 3a gravis , 16. TheAccurately identifying ptosis, as well as its underlying aetiology and severity, is essential to successful management. Thorough clinical examination and differential diagnosis is also needed in order to rule out similar conditions or most importantly, diagnose any serious underlying cause requiring more immediate medical intervention Table .Table 3Drd cranial (oculomotor) nerve palsy, myasthenia gravis, and CPEO. In a study of patients referred for ptosis surgery, 5.6% of cases had a neurogenic cause, and among these cases, the majority were due to serious underlying aetiologies . While myogenic causes  were likewise uncommon in the study population , 30% of the patients in this group had an underlying diagnosis of CPEO [The initial diagnostic step is a review of patient history to understand timing of ptosis onset, as a sudden appearance may signal serious underlying pathology. If patient history suggests that ptosis may be secondary to a more serious condition, subsequent evaluation can be conducted based on the observable clinical signs. The serious neurological or muscular conditions most commonly encountered in clinical practice include Horner’s syndrome, 3 of CPEO .rd cranial nerve palsy—which innervates, among other muscles, the levator palpebrae superioris—has a unilateral and variable presentation but is typically accompanied by diplopia and a “down and out” position of the affected eye due to partial or complete muscular paresis [rd cranial nerve palsy can be secondary to an acute event such as ischaemia, aneurysm, or trauma, or to compression of the nerve by an expanding mass. Because the 3rd cranial nerve delivers most of the parasympathetic fibres destined for the eye, dilation of the ipsilateral pupil can be observed in some cases. Pupillary involvement requires neuroimaging for the presence of an aneurysm or of a tumour that may be compressing the nerve. Lack of pupillary involvement often suggests a microvascular cause, such as diabetes mellitus [Horner’s syndrome, in its acquired form, is usually secondary to interruption of sympathetic innervation of the superior and inferior tarsal muscles due to trauma, certain tumours, or stroke. It is characterised not only by mild unilateral ptosis of the upper eyelid, but also the lower eyelid , ipsilateral pupillary miosis, facial anhidrosis, and a positive pupillary response (dilation) to topical phenylephrine (which can be used to differentiate between pre- and post-ganglionic Horner’s syndrome) or apraclonidine , 39, 40. paresis , 16, 40.mellitus , 16, 40.Ptosis can also be an early symptom of myasthenia gravis, a condition caused by autoantibody blocking or destruction of nicotinic acetylcholine receptors, and may be accompanied by external ophthalmoplegia , 16, 40.Evaluation of the periocular skin and soft tissues is essential to identifying or excluding ptosis secondary to a mass weighing down the upper eyelid , 16, 22.Also essential to the clinical workup is the exclusion of “pseudoptosis” conditions, which involve no pathology of the upper eyelid retractor muscles or levator aponeurosis, but instead are due to pathologies of other structures that indirectly affect eyelid position. Pseudoptosis can arise, for example, due to a range of mechanical , anatomical , or neurogenic  causes, or contralateral eyelid retraction (thyroid eye disease) , 16, 40.After appropriate examination of the ocular and periocular structures, assessment of upper eyelid function can be performed with a few simple measurements. The distance from the central pupillary light reflex to the central margin of the upper eyelid ) helps define the presence and severity of ptosis. In the normal eye, MRD-1 is typically 4–5 mm, and a decrease in this measure signals the presence of ptosis , 16. LesEyelid crease height, the distance from the upper eyelid crease to the eyelid margin, can likewise be informative. Normal eyelid crease height generally ranges from 7 to 8 mm in males and 9–10 mm in females, and an increase in this measure can indicate disinsertion of the levator aponeurosis . PalpebrVisual field testing is an important tool for measuring any functional deficits caused by ptosis , 11, 49.The standard of care for ptosis management is surgical intervention. Elevation of the upper eyelid for functional or cosmetic purposes can be successfully achieved with a variety of techniques targeting the upper eyelid retractor muscles and aponeurosis –58, and If there is dehiscence or disinsertion of the aponeurosis but levator muscle function remains good, levator muscle advancement (aponeurosis repair), using an anterior or posterior approach, can be performed. Levator resection is used in cases in which levator function is in the fair-to-good range (> 4 mm), with the amount of muscle resected dependent upon the degree of pre-surgical levator function , 44. If Surgical intervention has been demonstrated to improve elevation of the upper eyelid and superior visual field deficits, and these clinical improvements can accordingly improve patients’ performance of activities of daily living and HRQoL outcomes. As noted in Acquired ptosis overview, prevalence, and impacts above, patients who undergo ptosis surgery report improved ability to perform common visual tasks and activities of daily living, leading to an improved functional index , 13, 49.A retrospective analysis of 1519 patients who underwent ptosis surgery revealed that revision was required in 8.7% of cases, with a 6.8% revision rate in patients who underwent a posterior-approach procedure and a 9.5% revision rate in those who underwent an anterior-approach procedure . Over- aNon-surgical approaches to managing ptosis—and clinical evidence supporting any of these approaches—have been comparatively limited Table . The mos1/α1D, α2A, and α2C adrenergic receptor subtypes [Topical sympathomimetic agents, including phenylephrine, apraclonidine, brimonidine, and naphazoline, have ophthalmic applications outside of ptosis, but have also been evaluated for their effects on the ptotic upper eyelid based on their potential to activate Müller’s muscle, which has been shown to express the αsubtypes –19. A resubtypes . While tsubtypes , limitin2-adrenergic receptors and weaker agonist activity at α1-adrenergic receptors. Via its α1-adrenergic receptor-mediated effects, topical apraclonidine can temporarily reverse anisocoria due to Horner’s syndrome, thus supporting this diagnosis [1-adrenergic receptors [Apraclonidine demonstrates strong agonist activity at αiagnosis . A case iagnosis . A largeeceptors .1D-adrenergic receptor subtype in responsive eyelids, suggesting that the drug’s effect on the upper eyelid is mediated at least in part by agonism of α1D receptors [Apraclonidine’s effect in ptotic patients has also been evaluated in a number of small-scale studies. A retrospective case series examining 7 patients with ptosis following cosmetic botulinum toxin injection revealed a mild effect of apraclonidine 0.5% on ptosis, but only when used within 4–6 weeks of ptosis self-resolution, suggesting that apraclonidine response might help predict resolution time . A latereceptors .While studies of limited apraclonidine dosing in patients with ptosis have revealed no notable safety concerns, prospective studies of its chronic use for other ophthalmic applications  have reported ocular  and non-ocular (such as dry mouth and contact dermatitis) side effects that have led to discontinuation of use –71. In t2-adrenergic receptor agonist), but not brimonidine 0.2% or phenylephrine 0.12% [Other adrenergic agents have also been evaluated for potential applications to ptosis. A study of 20 healthy adult volunteers found significant elevation of the upper eyelid for up to 2 h following instillation of naphazoline 0.05%  has been reported. Oxymetazoline is a direct-acting α1- and α2-adrenergic receptor agonist [More recently, the efficacy and safety of an oxymetazoline 0.1% ophthalmic solution approved for the treatment of acquired ptosis (Upneeq agonist , 80. Lik agonist . Using t agonist . Importa agonist . No tachThe prevalence and wide-ranging clinical and functional implications of acquired ptosis make timely and accurate diagnosis and treatment important for eye care practitioners. Acquired ptosis is most often due to age-related changes in the upper eyelid retractor muscles , 25, butSurgery is an effective treatment option for ptosis, but non-surgical approaches have been extremely limited in both number and effectiveness. Because treatment via surgical intervention may be limited to a relatively small proportion of patients, finding ways to incorporate novel non-surgical therapeutic options into practice presents the potential to treat a wider range of patients. The evidence regarding a newly approved pharmacologic agent for the treatment of acquired ptosis  is thereWhile advances in ptosis treatment are encouraging, these remain only part of the clinical equation. To effectively treat ptosis, timely and accurate diagnosis is essential. In particular, comprehensive clinical examination and differential diagnosis are critical to understanding whether a patient’s ptosis is due to primary pathology of the upper eyelid retractor muscles—and can thus be effectively managed by surgical or pharmacological means targeting the upper eyelid—or whether the underlying cause is a more serious underlying neurological condition requiring different intervention. While in many cases ptosis might only be evaluated and treated when its onset is sudden or severity is high, examination of the upper eyelid for mild-to-moderate or progressive cases can be incorporated into the comprehensive eye exam with relative ease. Together with a focus on awareness and diagnosis, focused surgical or non-surgical treatment based on the clinical evidence offers the promise of improved ptosis treatment for more patients.


Oral mucosal melanoma (OMM) represents an extremely rare entity that is associated with a poor prognosis due to late diagnosis and early metastasis. Here, extensive surgical therapy is the therapy of choice. In contrary, for OMM in situ, the respective therapeutical recommendations are lacking. In this case report, treatment modalities of an OMM in situ of the palate, including the maxillary alveolar process, are reported. The tumor relapsed twice despite adequate surgical therapy and reconstruction. Therefore, irradiation was performed as an adjuvant therapy. At a follow-up of two years, the patient was free from recurrences. Primary oral mucosal melanoma makes up less than 1% of all malignant melanomas and far below 1% of all oral malignant tumors. The main localization is the hard palate and the gingiva of the upper jaw ,3. Oral A 46-year-old male patient presented due to a rapidly progressive, blackish, irregular macula of the oral mucosa, measuring approximately 2 cm × 1.5 cm .Eight years ago, a superficial, spreading, malignant melanoma of the left thigh was excised. There were no other relevant diagnoses or risk factors. Magnetic resonance imaging (MRI) confirmed a tumor-suspected mass in the upper jaw in regions 11 to 21 . MetastaThe histopathological examination of an incisional biopsy showed an oral mucosal melanoma (OMM) in situ. At that time, a complete excisional biopsy did not appear to be possible due to the tumor size, invasiveness, and the expected extension of the resection. After discussing the case at the interdisciplinary tumor board, the tumor was excised together with the surrounding bone and teeth 12, 11 and 21 that were in close contact with the lesion. The definitive histopathological examination showed subepidermal nests of cells with round oval nuclei, pigmentation, hyperchromasia, small fluctuations in nucleus size and prominent nucleoli. The described nests were positive for HMB45 and Melan-A with a proliferation index of 25% (Ki-67). No Melan-A/HMB45 gradient was detectable. Overall, the finding was consistent with OMM in situ, which had been excised with sufficient safety margins  .Three months later, another blackish change in the oral mucosa of approximately 2 × 2 mm in region 012 was noticeable .The mass, including the adjacent parts of the alveolar ridge and the palatine bone, was resected once again. Histopathologically, a R0-resected recurrence  of the previously known OMM in situ without bone infiltration was confirmed. After secondary granulation of the excision wound, two dental implants were inserted in regions 012 and 021 .Six months later, another recurrence occurred in the area of the hard palate , which wThe prosthetic restoration was carried out with a ceramic, fully veneered, implant-supported bridge .After consultation of the interdisciplinary tumor board and careful education of the patient, the informed decision of adjuvant irradiation was made. In order to prevent the tongue and buccal mucosa from receiving undesirable consequences of irradiation, a patient-specific device combining a tongue-depressing radiation stent and buccal  retractor was designed .Afterwards, the tumor bed was fractionally irradiated with a circular safety margin of 2 cm with a radiation dose of 57.5 gray. The patient presented at regular intervals for clinical and ultrasound follow-up examinations. After 2 years of follow-up, there was no evidence for tumor recurrence or secondary tumors .A bluish, brownish or black macular or nodular change in the oral mucous membrane in the sense of a melanotic OMM is usually impressive and easy to recognize. In contrast to this, up to 40% of OMMs are amelanotic and therefore show no typical pigmentation, which makes a clinical diagnosis significantly more difficult ,8,9. HowThe expansion of the surgical therapy of OMM is controversially discussed. In this context, an extended surgical resection of OMM may result in an increased disease-free survival . López eSurprisingly, there are several case reports of a melanoma in situ of the hard palate, some of which tend to recur even after years, despite adequate therapy . In contThis case represents a special surgical challenge since guidelines and recommendations concerning the surgical safety distance for OMM in situ are still missing. Therefore, the surgical extension as well as a potential radiation therapy are based on the local anatomical conditions, the assessment of the surgeon and the patient’s desire.