Hemifacial hyperplasia is a rare congenital malformation characterized by noticeable unilateral excess development of hard and soft tissues of the face. Asymmetry in Congenital Hemifacial Hyperplasia (CHH) is usually evident at birth and accentuated at the age of puberty. The affected side grows exponentially as compared to the unaffected side. Multiple tissue involvement has resulted due to etiological heterogeneity like heredity, chromosomal abnormalities, altered intrauterine environment, and endocrine dysfunctions. As this lesion is rarely seen in our routine clinical practice, we present a case of hemifacial hyperplasia with reported orofacial features that supplement existing clinical knowledge. This paper also adds knowledge to the readers regarding detailed investigation procedures which has complemented our diagnosis. Further emphasis has been placed on periodic approach to its diagnosis and multidisciplinary management following correct diagnosis.  Congenital Hemifacial Hyperplasia (CHH) is a rare developmental malformation exhibited by a unilateral enlargement of hard and soft tissues of the face. This asymmetrical overgrowth traditionally referred to as hypertrophy is more accurately termed as hyperplasia as this pathologic process involves abnormal proliferation of cells (hyperplasia) rather than growth of individual existing cells (hypertrophy) . SynonymCongenital Hemifacial Hyperplasia was first noted by Meckel JF in 1822 and, later, first case was reported by Wagner in 1839 .Hyperplasia presents as an isolated finding or in association with several syndromes like Beckwith Wiedemann syndrome, Proteus syndrome, Russell silver syndrome, and Sotos syndrome. Rowe  proposedWe present an interesting case of true congenital hemifacial hyperplasia (true CHH) in a 39-year-old male with unique orofacial manifestations, radiographic findings, and differential diagnosis with treatment in detail to supplement the existing knowledge.A thirty-nine-year-old male patient reported to the Department of Oral Medicine and Radiology with a chief complaint of asymptomatic swelling in his right cheek region since birth. The swelling gradually progressed to the present size and ceased to grow after 18 yrs of age. Family history was noncontributory. The patient was well oriented with stable vital signs.On extraoral examination, facial asymmetry with diffuse swelling was evident on the right side of the face measuring about 5 × 6 cm. The swelling extended superiorly to upper canthus of right eye, inferiorly up to 1 cm below the lower border of mandible, anteriorly until nasolabial fold, and posteriorly till the tragus of the ear Figures . Nose anOn intraoral examination, there was restricted mouth opening with a maximum interincisal distance measuring about 1 cm only. Enlarged right maxillary and mandibular alveolar arches, upper and lower labial mucosa, and buccal mucosa were observed. Dorsum surface of the tongue appeared engorged with polypoid excrescences (“multiple pebbly” appearance) representing enlargement of fungiform papillae . DistincOrthopantomogram (OPG)  revealedBased on the clinicopathological findings, a final diagnosis of Congenital Hemifacial Hyperplasia (CHH) was made. The patient refused to undergo extensive surgical procedures as the lesion was asymptomatic. Follow-up with this patient exhibited good prognosis and no evidence of malignant changes.Hemifacial hyperplasia (HFH) is a rare developmental anomaly exhibiting asymmetric facial growth by unilateral localized enlargement of all tissues in the affected area that is facial bones, teeth, and soft tissues . The degThe prevalence of hemifacial hyperplasia is approximately 1 in 86,000 live births . Women aVarious etiological factors like heredity, chromosomal abnormalities, altered intrauterine development, endocrine dysfunction, and vascular and lymphatic abnormalities remain implicated in this lesion. However, no single factor is directly related to the occurrence of this disorder .Of importance, Gesell suggested this condition due to the process of twinning mechanism. According to Noe and Bergman, mitochondrial damage overripened one-half of the fertilized egg resulting in excess generation of cells . RecentlLimited cases of hemifacial hyperplasia have been documented. These patients present with dental, skeletal, and soft tissues of the affected portion of the face . Dental  , zygoma, maxilla, and mandible is accentuated. Midline shift and altered occlusion with deviated occlusal plane are noticed. Accelerated jaw growth frequently results in exostoses with malocclusion [Skeletal development of cranial bonescclusion , 15. Sofcclusion , 17. AllRadiographically, this lesion presents with hard tissue and soft tissue enlargement on the affected side. These patients exhibit premature development and altered crown and root size/shape discrepancy along with a downward slant of the occlusal plane , 19. OurDifferential diagnosis of facial hemihyperplasia includes fibrous dysplasia, dyschondroplasia, congenital lymph edema, arteriovenous aneurysm, hemangioma, lymphangioma, neurofibromatosis, and malignant lesions (osteosarcoma and chondrosarcoma). These clinical conditions can be clearly differentiated from CHH based on specific radiographs and laboratory and clinical findings that supplemented our case diagnosis .Treatment ideally is not indicated for Congenital Hemifacial Hyperplasia (CHH) unless cosmetic considerations are involved. Procedures are deferred until physiological growth ceases. An integrated multidisciplinary approach in the field of dentistry is essential in providing an asymptomatic treatment. Treatment options may include soft tissue debulking of masticatory and subcutaneous tissues, preservation of neuromuscular functions, and reconstructive procedures  postoperatively. Till date, previous literature survey indicated no evidence of malignant transformation.A true case of Congenital Hemifacial Hyperplasia (CHH) is rare and unfamiliar to a clinician due to underreported number of cases in literature. This article presents a unique case report of a typical case of CHH with a detailed emphasis on diagnostic criteria  thus emphasizing to all the readers its importance. Treatment planning in such patients is tremendously difficult due to the involvement of multiple major surgeries for the removal of excessively grown hard and soft tissues. Further cases need to be brought into documentation so as to have a clear understanding regarding various aspects of this disease.


Colloid cysts are benign slow-growing cystic lesions located on the roof of the third ventricle that usually present with symptoms related to gradual rise of intracranial pressure. They mostly remain asymptomatic and sometimes grow progressively and cause diverse symptoms associated with increased intracranial pressure such as headache, diplopia, and sixth cranial nerve palsy. Here we report a 47-year-old female who presented to the emergency department with acute severe headache and nausea/vomiting. On MRI examination acute hydrocephaly due to hemorrhagic colloid cyst was detected. Acute hemorrhage in colloid cysts is extremely rare and may present with symptoms of acute increase in the intracranial pressure. Intracystic hemorrhage is very rarely reported as a complication of colloid cyst presenting with paroxysmal symptoms of acute hydrocephaly. Colloid cysts are benign embryologic remnants that are located on the roof of the third ventricle. These tumors grow slowly and may become symptomatic in third to fifth decades of life , 2. The The patient was a 47-year-old female who presented to the emergency department with acute headache and nausea/vomiting. She did not mention any previous history of severe headache or vomiting. On the neurologic examination papilledema was noted. There were no focal neurological deficits. She was dizzy but conscious and her Glasgow coma scale (GCS) score was 13. Computed tomography (CT) scan of the brain revealed enlargement of lateral ventricles and a well-circumscribed hyperdense mass on the roof of the third ventricle at the location of foramen of Monro . On MRI The patient underwent ventriculostomy followed by craniotomy and tumor resection and experienced an uneventful recovery. Histological examination confirmed the diagnosis of hemorrhagic colloid cyst.Colloid cysts are benign tumors of brain that rise from endodermal embryologic remnants and are classically located on the roof of the third ventricle , 2. The Approximately two-thirds of the cysts are hyperdense on computed tomography due to their high protein content; the remaining third appears isodense . On MRI


Osteoid osteoma is a benign bone tumour that mostly affects males in the second and third decade of their life. The lesion mainly occurs in long bones, usually in the femur and tibia, causing severe localized pain that is worse at night and responds to nonsteroidal anti-inflammatory drugs (NSAIDs). Diagnosis is usually made on the basis of history and radiographic findings. However, in more unusual locations as the hand and foot, diagnostic issues can arise. Treatment often includes complete removal of the tumor. We present a 22 year old male with osteoid osteoma involving the distal phalanx of the hallux. To our knowledge very few cases of great toe osteoid osteoma have been reported in the literature. A 10The characteristic pain pattern and clinical findings as swelling and deformity should raise a high suspicion for the diagnosis of osteoid osteoma (OO). Plain radiography is the initial diagnostic modality that can reveal the typical radiolucent nidus surrounded by an area of sclerotic bone. However, in more complex anatomic locations, bone scintigraphy has been used, where the lesion presents as a focal area of high uptake. Also, computerized tomography can provide better detail and aid in differential diagnosis. The use of MRI is somewhat controversial as it can obscure the differential diagnosis with more malignant lesions and lead to overtreatment.Treatment of OO typically consists of a trial of medical management with NSAIDs with a dramatic decrease in symptoms, which can lead to resolution of the disease , althoug2A 22 year old male patient was admitted in our department with right hallux pain for the past 18 months. There was no history of injury. The pain was progressive and worse at night. The pain was initially relieved by NSAIDs but in the last few months only partially and the patient was forced to limit activities due to the pain. There were no constitutional symptoms such as fever, weight loss or malaise.1). The patient was otherwise fit and well. Blood analysis was unremarkable. Radiographic workup included plain x-rays of the foot and a CT scan and revealed a small radiolucency with cortical sclerosis and a nidus formation in the medial side of the distal phalanx of the great toe .The distal phalanx of the right hallux was swollen and tender to palpation, with no obvious erythema or increase in temperature . Excision of the nidus and surrounding bone to macroscopically healthy borders was performed and histologic samples were sent for analysis . The skin was closed and the nail was used as a splint to preserve the preoperative hallux shape. Pathology confirmed the diagnosis of osteoid osteoma. The patient was immediately relieved from nocturnal pains and progressively completely relieved from hallux pain. Immediately postoperatively, the patient was mobilized with a wedge shoe. At two years follow up, there were no signs of recurrence or skin and nail problems and the patient was ambulating freely.The patient underwent operative excision of the mass through an antero-medial incision with atraumatic elevation of the nail bed and nail in one layer , as well as the keywords that were used. Abstracts of cases in English language were exclusively included for review. Abstracts were reviewed and full text articles were subsequently obtained. Thirteen cases of great toe osteoid osteoma were found in the literature (Table 2), making it a very rare or poorly reported entity.A systematic review of the PubMed Database was conducted. Search results are listed below  and tumours, both benign and malignant. Differential diagnosis should take into account conditions like osteomyelitis due to pseudomonas (mostly in diabetics), epidermoid inclusion cysts, chondrosarcoma, osteoblastoma, subungual squamous cell carcinoma, intracortical osteosarcoma and osteoid osteoma . DiagnosOsteoid osteoma is a well described rare benign tumour that mostly affects long bones. Localization of the tumor on the great toe of the foot, as described in our case, comprises a rare medical entity, as only few cases have been described according to literature. This fact illustrates the difficulties associated with the early diagnosis and treatment of the lesion. A combination of thorough medical history, radiographic examination and clinical suspicion should lead the clinician to the diagnosis. In our case, total resection of the lesion resulted in complete remission of the patient’s symptoms and no signs of recurrence at two years follow up.


Radiosurgery is being increasingly used post craniotomy for brain metastasis, instead of whole-brain radiation. We report a case of scalp metastasis following craniotomy and radiosurgery, along with a systematic review of the literature.Our patient was a 70-year-old male who presented with a scalp metastasis, two years after craniotomy and radiosurgery, for a solitary brain metastasis from esophageal carcinoma. Using Medline® , we performed a systematic review of the literature to identify cases of isolated scalp metastases following craniotomy for brain lesions.The scalp metastasis was in close proximity to the craniotomy site. Workup did not show any other site of active disease. Biopsy confirmed it to be a metastasis from esophageal carcinoma. The literature review did not yield any case of isolated scalp metastasis following craniotomy and whole-brain radiotherapy or radiosurgery. However, it yielded six cases of isolated scalp metastases following craniotomy for primary brain tumors.Isolated scalp metastasis has not been reported following craniotomy and whole-brain radiotherapy for brain metastases. Our patient likely had surgical seeding during craniotomy. These surgically implanted cells could not be ablated because the radiosurgery treatment volume does not cover the surgical tract. Further research is needed to identify risk factors for surgical seeding. For several decades, whole-brain radiation therapy (WBRT) remained the standard treatment for brain metastases. It was shown in a number of studies that surgery for solitary brain metastasis followed by WBRT resulted in improved overall survival compared to WBRT alone -4. The iAlthough still experimental, sometimes patients are treated with radiosurgery off study out of their preference. We report a case of scalp metastasis following craniotomy and radiosurgery, along with a literature review.Informed consent was obtained from the patient for this study.Our patient is a 70-year-old male. Initially, he presented in 2013 with progressive dysphagia. He was investigated with barium swallow and an esophago-gastroscopy. A tumor was identified at the gastroesophageal junction (GEJ); the biopsy showed moderately differentiated adenocarcinoma. He had further workup with computed tomography (CT)/positron emission tomography (PET) scan. Intense fluorine-18 (F-18) fluordeoxyglucose (FDG) uptake was noted at the GEJ and the proximal part of the lesser curvature of the stomach; a 7 mm lymph node in the gastro-hepatic ligament was mildly avid. The patient was staged as T2 N1 M0 and treated with neoadjuvant chemoradiation. Chemotherapy consisted of weekly carboplatin and paclitaxel. Radiation was delivered over five weeks at a dose of 4500 cGy/25 fractions to the tumor and regional lymph nodes. In July 2013, he underwent a total esophagectomy and proximal gastrectomy. The pathology revealed extensive fibrosis in the esophagus and stomach specimens consistent with post chemo-radiation changes. No viable or residual malignancy was identified. One out of nine perigastric lymph nodes was positive. The patient was treated with two cycles of adjuvant chemotherapy comprising of epirubicin, cisplatinum and 5-fluorouracil (ECF). He continued on surveillance with periodic gastroscopy.In December 2014, he presented with headaches. Magnetic resonance imaging (MRI) of the brain showed an enhancing cortically-based lesion with central necrosis in the left parieto-temporal region. It measured 4.1 x 3.7 cm and had surrounding vasogenic edema with mass effect on the lateral ventricle. He underwent a craniotomy and had an uneventful recovery. Pathology showed metastatic adenocarcinoma of esophageal origin. He received radiosurgery to the surgical cavity (1800 cGy single fraction) which was completed in February 2015  over the scalp lesion.Using Medline® , we performed a search for publications on scalp metastases following surgery. Our disease-specific medical subject heading (MeSH) terms were ‘brain’, ‘neoplasm’, ‘metastases’ and ‘scalp’, and our treatment specific MeSH term was ‘craniotomy’. To keep the search exhaustive, we did not apply any filters for publication date and article type. Two of the authors independently performed the search and selected publications that met the eligibility criteria. We identified additional publications from the reference list of the selected publications. Eligibility criterion was any report of isolated scalp metastasis from surgical seeding. We defined surgical seeding as any scalp metastasis close to the surgical tract where the surgical cavity did not have any recurrent disease. We excluded cases of direct extension of recurrent tumor into the surgical tract, and recurrent tumor with multiple sites of metastases.The scalp metastasis was not present prior to craniotomy and appeared in close proximity to the craniotomy site Figure . Biopsy The literature search identified 18 publications. A review of the reference list identified 15 additional publications. These reported a total of 27 cases of scalp metastases. Out of 27 cases, six met the eligibility criteria  in recent times, improvement in systemic therapy and aggressive treatment of oligometastases has considerably improved survival which was previously fairly low; b) brain metastasis was traditionally treated with whole-brain radiotherapy that might have sterilized the surgical tract; and c) surgical resection of brain metastasis recently became an established practice in select cases.The differential diagnosis of scalp lesion includes hypertrophied scar, second primary malignancy, new metastasis, and recurrence from surgical seeding. Pathology proved it to be a metastasis of esophageal origin. Thus, it was either a new metastasis or recurrence from surgical seeding. Given that isolated metastasis to the scalp are extremely rare and re-staging workup did not reveal any other site of active disease, we believe that it was likely the result of surgical seeding during craniotomy.Surgical seeding during craniotomy is uncommon. Very few cases have been reported which are mostly related to primary brain tumors. Avecillas-Chasin, et al. reported four cases of scalp metastases from recurrent meningiomas, along with the literature review . They coThe major limitation of our case report is that it is based on a single patient and thus the results are not generalizable to every case. Nevertheless, we included a systemic review which yielded six cases of isolated scalp metastasis likely from surgical seeding during craniotomy. This suggests that the risk of recurrence is not limited to the surgical cavity alone.In conclusion, the mechanism of scalp metastasis in our patient was likely surgical seeding during craniotomy. This occurred despite copious irrigation of the craniotomy site and the surgical tract. Since radiosurgery treatment volume did not include the surgical tract, it did not ablate surgically implanted cells. Further research is needed to identify risk factors for surgical seeding.


Juvenile ossifying fibroma (JOF) is a benign, bone-forming neoplasm occurring primarily in children and adolescents. JOF is an aggressive variant of ossifying fibroma of the jaw with a variable clinical behavior and a high tendency for recurrence. Early detection and prompt treatment are required to treat JOF successfully. This case report describes JOF in a 13-year-old girl presenting with a year-long, gradually progressive swelling on the right side of her face with typical clinical, radiological, and histopathological features. Ossifying fibroma is an uncommon, benign, bone-forming tumor distinguished from fibro-osseous lesions by the age of onset, clinical presentation, and aggressiveness . OssifyiA 13-year-old girl reported to the Department of Oral and Maxillofacial Surgery, Thai Moogambigai Dental College and Hospital, Chennai, India, with swelling over the right side of the face for the past year. She reported the swelling was gradually increasing in size over the past four months. The patient has a medical history of an epileptic attack, and she was diagnosed with tubular sclerosis of the brain in 2008, treated with medication since the diagnosis. Her last episode of epilepsy was in 2010 (seven years ago). Family history revealed her mother is also taking medication for epilepsy. The patient also reported a history of blurred vision in her right eye for the past year, and she was prescribed glasses for the high refractive index (amblyopia) in the right eye for correction.The extraoral examination revealed diffuse swelling on the right side of the face, extending from right alar region to the infraorbital margin superiorly, and extending laterally to the right cheek region Figure .The skinThe computed tomography scan of the right maxilla showed medial and lateral cortical expansion, with the lesion extending to the floor of the orbit Figure . The parWe performed an incisional biopsy that confirmed the diagnosis of JOF. We then planned a maxillary resection with skin grafting. The patient underwent general anesthesia, and we placed a Weber-Fergusson incision on the right side to approach the maxilla Figure . We reseHistopathology with hematoxylin and eosin staining showed irregular strands of highly cellular osteoids encasing plump and irregular osteocytes as well as multinucleated osteoclasts Figure . These fThe etiology of JOF is unknown. Suspected origins include traumatic, developmental, and odontogenic causes, and the periodontal ligament may also have a role in its etiology. Tumor suppressor gene HPRT2 has also been reported as a potential factor in JOF . There iIf the orbital bones or paranasal sinuses are involved, the patient may develop exophthalmos, bulbar displacement, nasal obstruction, and epistaxis . Our patRadiographically, lesions appear highly variable depending upon the stage of development and amount of calcification. JOF is characterized by three stages in radiographs. Stage I appears as a well-defined radiolucency with no evidence of internal calcification. Stage II  is characterized by flecks of radiopacities in the radiolucent area. Stage III  is a completely radiopaque mass . ErosionMicroscopically, JOF is characterized by the presence of trabeculae of immature woven bone, cementum-like tissue, or both in a fibrous stroma. PsJOF is characterized by a proliferation of benign spindle‑shaped fibroblastic cells with embedded mineralized structures, which may present with round to ovoid collections of bone with an osteoid rim. Multinucleated osteoclast‑like giant cells may be seen with occasional normal mitotic figures, but atypia is not a usual feature. Our patient had TrJOF, which characterized by a fibroblastic spindle cell stroma containing osteoid matrix, surrounded by osteoblasts and anastomosing trabeculae of immature woven bone, often intermixed with scattered clusters of multinucleated giant cells; mitoses may be present, but cystic degeneration is rare .JOF must be differentiated from malignant bone tumors other fibro-osseous lesions. The most prominent condition in the differential diagnosis is cemento-ossifying fibroma and fibrous dysplasia. The rapidity of growth, monostotic nature, and the well‑delineated radiographic margins differentiate JOF from fibrous dysplasia. Microscopically, cemento-ossifying fibroma contains cementicles , and whiThe recurrence rates after resection range from 30% to 56%. This may be due to the infiltrative nature of the tumor borders after an incomplete excision of the tumor, rather than any intrinsic biological properties of the tumor .JOF is a benign neoplasm of the bone that has a potential for excessive growth, bone destruction, and recurrence. The treatment of choice is surgical excision, especially when JOF is located in the maxilla, and requires radical surgery with healthy margins. Early detection, complete surgical excision of these lesions, and long-term follow-up evaluations are important in the clinical management of JOF cases, due to its aggressive nature and high recurrence rate. The prognosis of JOF is good unless it recurs locally.


A 32-year-old 22-week pregnant hypertensive woman with sporadic episodes of headaches, sweating, and facial flushing was diagnosed with pheochromocytoma through biochemical and imaging tests. Perioperative management included a multidisciplinary approach, symptom stabilization with α blockade followed by β blockade, and tumor resection by laparoscopic adrenalectomy at 24 weeks gestation. The diagnosis was confirmed by histopathological examination and immuno-histochemistry tests. The decision for surgical removal of the tumor was based on maternal symptoms, tumor size, gestational age, the possibility of doing a laparos-copy, and the expertise of the surgical team. Althougmination .A 32-year-old Caucasian woman at 22 weeks of gestation with singleton live fetus presented 5 weeks prior with sporadic episodes of headaches, facial flushing, and sweating. The obstetric ultrasound showed a healthy fetus. Her first pregnancy four years earlier was complicated by severe preeclampsia at 34 weeks gestation for which she was induced and delivered by emergency cesarean section. Since then, she was treated with methyldopa 500 mg/day, but her blood pressure was not monitored and she had no other complaints or complications. She was not on any regular medications, was not a smoker or an alcohol user, and there was no history of illicit drug use nor family history of multiple endocrine neoplasia (MEN). Due to palpitations, nine months earlier she underwent a cardiac stress test, which detected sustained supraventricular paroxysmal tachycardia, followed by an electrophysiology study that failed to reveal any disorder or dysfunction. The echocardiogram was also normal. Symptoms of head-aches, facial flushing, and sweating started at 17 weeks gestation. Routine laboratory tests re-vealed a fasting glucose of 92 mg/dL and serum creatinine of 0.5 mg/dL. Complete blood count, sodium, potassium, calcium, phosphorus, AST, ALT, total cholesterol and fractions, TSH, free T4, urinary sediment levels, serum aldosterone, calcitonin, parathyroid hormone (PTH), and cor-tisol were all normal. However, metanephrine and normetanephrine on 24 hours urine collection were 6873.2 μg/24 h  and 6299.2 μg/24 hours , respectively. Ultrasonography of the abdomen revealed a 10.1 × 9.5 cm mass in the right adrenal gland consistent with PCC . Blood pHistopathological examination and immu-nohistochemistry tests (positive chromogranin A and synaptophysin staining) confirmed the diagnosis of PCC.Five weeks after surgery, while remaining normotensive without antihypertensive drug therapy, the patient was admitted due to premature rupture of membranes, and one day later suffered placental abruption with severe bleeding. She underwent emergency cesarean section but the newborn, who was born alive, died prematurely within 48 hours.PCC in pregnancy can cause fatal hypertensive crisis that can be triggered by vaginal delivery, general anesthesia, the physical effects of the enlarging uterus, uterine contractions, or fetal movements , and maySymptoms may occur for the first time in pregnancy due to increased vascularity of the tumor and/or mechanical factors such as pressure from the enlarging uterus or fetal movements, which can stimulate catecholamine secretion , 8. ThesDetecting elevated levels of catecholamines and their metabolites in plasma and urine establishes the diagnosis of PCC. Once a diagnosis has been confirmed biochemically, ultrasonography and MRI are the preferred modalities of tumor localization for the safety of the fetus , 4, 5.The success of treatment will depend on the appropriate preoperative medical management, which should always be started with α-adrenergic blockade (phenoxybenzamine or prazosin) for adequate control of blood pressure . Pre-surTiming of tumor excision in pregnant women will depend on the gestational age at which di-agnosis is made, fetal development, and the severity of maternal symptoms. Surgery should be avoided in the first trimester — as organogenesis is incomplete and miscarriage is highly likely — as well as in the third trimester — because by then the enlarging uterus precludes adequate access and visualization of the abdomen . Thus, tPregnancy was previously a contraindication to laparoscopy because of concerns regarding the potential effect of carbon dioxide on the developing fetus and the safe entry into the abdominal cavity due to the displacement of anatomy by the gravid uterus. Additionally, increased abdominal tension by the pneumoperitoneum could reduce the vena cava and the uteroplacental blood flows , 8. NeveImportantly, perioperative management includes treatment of both mother and fetus . In the PCC is a rare but important cause of hypertension in pregnancy. It should be included and investigated as a differential diagnosis in cases of labile hypertension in pregnant women with or without associated symptoms, because inadequate treatment considerably increases maternal and fetal mortality. Management of PCC during pregnancy should be discussed on a case by case basis by a multidisciplinary team. Surgical resection is recommended during the second trimester of pregnancy. Considering its benefits and the expertise of the surgical team, laparoscopic surgery is the favored technique.


An epithelial trophoblastic tumor (ETT) is a kind of rare trophoblastic tumor that may have the correlation with a prior gestational event. Especially, the one that appears in the lung is extremely rare.Here, we present a 24-year-old female patient with the chief complain of vaginal bleeding for more than 1 month, who was found to have a large mass (7.5 × 4.5 cm) in the right lower lobe, and it was finally confirmed as lung ETT by postoperative pathology after a successful radical resection of the pulmonary lobe.As the reason of an extreme rare occurrence of the ETT, doctors can easily misdiagnose the disease. When the patient was found to have a mass with irregular vaginal bleeding and a high level of beta-human chorionic gonadotropin, we need to consider ETT. Currently, surgery is still the most effective method. Less than 100 surgically treated cases have been reported in the English literature, and the ETT which appears in the lungs as an original lesion is even more rarely. ETT is a kind of trophoblastic tumor, which is different from a gestational trophoblastic tumor or a placental site trophoblastic tumor (PSTT).Epithelial trophoblastic tumor (ETT) is a rare trophoblastic tumor originating from chorionic-type intermediate trophoblasts.2A 24-year-old woman who had 4 histories of abortion had vaginal bleeding for more than 1 month. She was not eager to have a baby during the previous 4 abortions, so she did not get any drug treatment during and after those abortions. The level of serum beta-human chorionic gonadotropin (β-HCG) was 9075.4 mIU/mL. Ultrasound revealed an inhomogeneous mass (1.6 × 1.2 cm) close to the inner side of the right ovary, and a fluid sonolucent area (2.6 × 2.6 cm) can be seen in the pelvic cavity. Ectopic pregnancy was considered and the woman underwent laparoscopic salpingectomy on the right side with uterine curettage in a local hospital. After the operation, serum β-HCG level in patients had no significant decline. Then the patient came to our hospital, her physical examination was normal except for vaginal bleeding and fresh scars in abdomen, and computed tomography (CT) scanning revealed a lesion with partial necrosis in the right lower lobe of about 8.2 × 4.3 × 5.8 cm in size . Here we present a young female patient with 4 abortion history with the chief complain of vaginal bleeding for 1 month, who was misdiagnosed with an ectopic pregnancy at a local hospital and was finally proved to have primary lung ETT. Pathophysiologically, the cells of ETT are from chorionic-type intermediate trophoblast cell. Histologically, ETT is composed of nests of uniform mononucleated chorionic-type intermediate trophoblastic cells with eosinophilic or clear cytoplasm. As was seen in our case, positive p63 and β-HCG expression can help to distinguish ETT from PSTT and cervical squamous cell carcinoma.ETT is a very rare tumor, which was first described by Shih and Kurman in 1998. As far as our knowledge, less than 100 surgically treated cases have been reported in the English literature. The onset age of ETT ranges from 15 to 48 years. The uterus is the most common primary site of ETT (40%), followed by the cervix (31%).Because of the low incidence of ETT, clinical understanding of ETT is insufficient. Such disease can be easily misdiagnosed as choriocarcinoma, ectopic pregnancy, or primary lung cancer. Choriocarcinoma is a type of cancer germ cell containing trophoblast cells that can make β-HCG. Its feature is the identification of intimately related syncytiotrophoblasts and cytotrophoblasts without any formation of definite placental type villi. Lung cancer like anaplastic carcinoma and small cell carcinoma can independently produce luteinizing hormone and β-HCG, which may cause painful bilateral or unilateral mammary gland hyperplasia. Generally, the main symptom of lung cancer is cough, hemoptysis, fever, and chest pain. X-ray or CT examinations are used to detect lung cancer and make definite diagnosis by histopathological examination. Ectopic pregnancy is a complication of pregnancy in which the embryo attaches outside the uterus. Dynamic monitoring of transvaginal ultrasonography and β-HCG is important for the detection of ectopic pregnancy.Our patient was diagnosed with ectopic pregnancy in the local hospital, and she underwent a laparoscopy there. After surgery, vaginal bleeding and high level of β-HCG still existed. For this kind of patient, especially having the following characteristics: woman of reproductive age, history of pregnancy or abortion, elevated levels of β-HCG, irregular vaginal bleeding, and pulmonary mass, the diagnosis of pulmonary ETT should be considered. the retrospective study from the literature shows that ETT seems to be resistant to combination chemotherapy regimens. Surgery is still the preferred treatment for the ETT at present. In this case, after complete resection of the tumor, β-HCG decreased rapidly to normal after pulmonary surgery, and the vaginal bleeding disappeared. So the patient did not receive postoperative chemotherapy. The patient recovered uneventfully after surgery and 1-year follow-up showed no evidence of recurrence.Surgery and chemotherapy are the main treatment for a tumor. Though most of chemotherapy regimens, for example, EMA-CO, EMA-EP, or EMA, have been successfully used in the treatment of gestational trophoblastic neoplasia,4An extrauterine epithelioid trophoblastic tumor of lung is extremely rare. Such disease can be easily misdiagnosed as choriocarcinoma, ectopic pregnancy, or primary lung cancer. For this kind of patient, especially having the following characteristics: woman of reproductive age, history of pregnancy or abortion, elevated levels of β-HCG, irregular vaginal bleeding, and pulmonary mass, the diagnosis of pulmonary ETT should be considered.


Acute abdominal pain is a common surgical presentation, and most often, the first line of consideration is to rule out acute appendicitis; this is more so when the patient is an adolescent or within younger age group. In most cases, other differentials are considered before omental torsion. Omental torsion is a cause of acute abdominal pain and sometimes mimics acute appendicitis in its presentation. We present a case of a 14-yr-old boy who presented with acute abdomen with symptoms mimicking acute appendicitis. Laparoscopy revealed torsion of the omentum. Omentectomy and appendicectomy were done, and the child discharged four days after following a remarkable recovery. Omental torsion can simply be defined as twisting of the omentum on itself especially on its long axis. Frequently, this is in a clockwise direction. In this situation, venous return is compromised; thus, the distal omentum becomes congested and oedematous. As the torsion progresses, arterial occlusion leads to acute hemorrhagic infarction, and eventual necrosis of the omentum occurs . TorsionA 14-year-old boy was admitted to the Emergency Department with a three-day history of severe right lower abdominal pain, anorexia, and fever. His past medical history is unremarkable apart from undescended testes that descended spontaneously at the age of 4. On physical examination, his pulse rate was 96/min, blood pressure was 132/62 mmHg, respiratory rate was 18/min, peripheral oxygen saturation was 99%, and his temperature was 37.4°C. Abdominal examination revealed guarding and tenderness on the right iliac fossa region. Lung and heart examination did not reveal any abnormality. He was given analgesia, and the blood was sent for analysis.The laboratory report showed a normal blood analysis except the C-reactive protein which was 41. His urinalysis was normal. Abdominal ultrasonography showed fat stranding in the right iliac fossa. Appendix itself could not be visualised.He was observed, and after six hours, his abdominal pain persisted with increasing intensity. A decision was made to perform laparoscopy. At laparoscopy, the greater omentum was flimsily adhered to the right lower side of the abdominal wall, and there was a hemorrhagic area on the central part of the omentum with patches of pseudomembranes; the corresponding distal part  appeared necrotic, and small serosanguinous fluid was present in the pelvis. Figures 1An omentectomy was carried out using a harmonic. Due to the congestion of the appendix, a decision was made to remove the appendix at the same time. Postoperatively, he made a remarkable recovery and was discharged on the fourth day.Histology report confirmed omental torsion . Appendix showed early acute appendicitis (congested vessels with no obvious exudate).Torsion of the omentum is a rare cause of acute abdominal pain and may occur at any age. It is difficult to diagnose clinically in the preoperative setting. Accurate preoperative diagnosis was reported in the range of 0.6–4.8% . TorsionClinically, it presents as an acute abdomen with sudden onset pain that is localised in the right iliac fossa. Fever, nausea, and vomiting are infrequent presentations. Sixty-six percent (66%) of presentations resemble appendicitis, and twenty-two percent (22%) resemble cholecystitis .Pathogenesis of primary omental torsion is not fully established but considered to be wide-ranged. Predisposing factors and precipitating factors have been reported. Predisposing factors include anatomic variations, obesity, and the arrangement of omental blood vessels , 6. PrecPreoperative diagnosis of primary omental torsion can be challenging. Basson and Jones analysed 223 cases of primary torsion and revealed that only one patient had been correctly diagnosed preoperatively . SometimThe current investigation tool and therapeutic management of choice is laparoscopy proceeding to laparotomy, identifying and removing the infarcted section of the omentum. Normal appendix, gallbladder, and fluid in the pelvic cavity make the diagnosis of omental torsion likely.Omental torsion should be considered if preoperatively diagnosed acute appendicitis and Meckel's diverticulum were not found and if the gallbladder and ovaries reveal no disease. Secondly, the presence of serosanguinous fluid in the peritoneal cavity mandates inspection of the omentum to exclude torsion . Awarene


Medulloepithelioma is a rare primitive neuroectodermal tumor of the central nervous system, usually developing in childhood. Due to its rarity, the optimal management is still unknown. The prognosis is poor, especially when resection is incomplete. Adjuvant radiochemotherapy is often indicated.We report a rare case of infratentorial medulloepithelioma in a 3 year old girl. She presented symptoms of increased intracranial pressure. On examination, she had coordination problems, ptosis and exotropia of the right eye. Magnetic resonance imaging demonstrated a large cerebellar vermix tumor. Immuno-histochemistry revealed a diffuse positivity for Vimentin and focal positivity for the epithelial membrane antigen, but Glial Fibrillary Acidic Protein and Synaptophysin were negative, the MIB-1 antibody was very high. She received postoperative craniospinal irradiation and died 7 months later.We describe the features  of our case and confront it to literature data. Medulloepithelioma of the central Nervous System (CNS) is an uncommon primitive neuroectodermal tumor (PNET), characterized by a highly malignant behavior . It is aA 3 year-old girl was admitted to the emergency department with intracranial hypertension picture without fever of 30 days duration. On examination, she had coordination problems, ptosis and exotropia of the right eye. Her vitals were stable. Brain magnetic resonance imaging (MRI) showed a large cerebellar vermis lesion compressing the 4th ventricle with surrounding edema and triventricular hydrocephalus, the mass demonstrated a heterogeneous low signal on T2-weighted Fig. . A subtoThe medulloepithelioma of the central nervous system is a rare tumor representing 1.5% of primary tumors of the CNS , with a Medulloepitheliomas are mostly supratentorial lesions commonly found in the periventricular location . They arThe clinical picture is nonspecific and is based on the seat of the lesion. Most often, it is a syndrome of intracranial hypertension with or without a neurological deficit. The mean duration of symptoms was 6 weeks .Appearances on MRI are very variable, they are typically iso to hypointense with grey matter on T1 weighted imaging and iso to hypointense on T2 weighted imaging, 95% of tumours will show contrast enhancement, particularly of the solid elements [The diagnosis of medulloepithelioma can be confusing with other primitive neuroectodermal tumors (PNET), such as medulloblastoma, neuroblast, the ependymoblastoma, teratoma or some glial tumors . The preThe prognosis of these tumors is generally poor due to their relative radioresistance and their inexorable tendency to recurrence and spread to intra-and extra-axial . The meaTable Necessity to look for alternative molecular markers and directed treatment similar to INI-1 in atypical teratoid rhabdoid tumors is emerging. In recent times, hTERT oncogene amplification has been demonstrated in a proportion of embryonal tumors, particularly medulloblastomas, as well as, medulloepitheliomas , a correThe medulloepithelioma is the most undifferentiated and malignant of PNET. It affects young children and can sit anywhere in the CNS, usually in periventricular. Clinical and imaging are not specific. Only histology with immunohistochemistry confirms the diagnosis by showing the typical appearance, with often positive immunoreactivity to vimentin. The prognosis may be better if we can make a complete surgical resection and radiotherapy.Written informed consent was obtained from the parents of patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.Inappropriate.


Anomalous papillary muscle insertion directly into the surface of the mitral valve leaflet is rare, especially in a subject without apparent evidence of hypertrophic cardiomyopathy. We present a case of this isolated congenital malformation producing two hemodynamic sequelae of dynamic left ventricular outflow tract obstruction and severe mitral regurgitation. Congenital malformations of the papillary muscles, including their direct insertion into the mitral valve leaflet, are uncommon and associated primarily with hypertrophic cardiomyopathy. Herein, we present a rare case of isolated anomalous papillary muscle insertion into the anterior mitral valve leaflet, causing both dynamic left ventricular outflow tract obstruction and severe mitral regurgitation, in a patient without clear-cut evidence of hypertrophic cardiomyopathy. We discuss potential mechanisms for the hemodynamics observed and underscore the importance of recognizing this clinical entity, as it may alter the surgical treatment.A 59-year-old healthy male sought an outpatient medical evaluation following two episodes of unwitnessed syncope that had occurred two weeks prior. He described the most dramatic event occurring after waking up from sleep in the mid-morning hours to urinate, when he was found by his family lying on the ground unconscious with a minor frontal scalp contusion. He denied any symptoms prior to the fall. His medical history was unremarkable for any atherosclerotic risk factors or rheumatic fever as a child. His vital signs were normal but physical examination was notable for a 2/6 crescendo systolic murmur at the right upper sternal border and a holosystolic murmur at the apex. An electrocardiogram showed normal sinus rhythm with no signs of left ventricular hypertrophy and normal ST segments and T waves. An event monitor did not reveal any evidence of arrhythmia.A transthoracic echocardiogram demonstrated normal left ventricular (LV) cavity dimensions and ejection fraction but color Doppler revealed significant turbulence across the LV outflow tract (LVOT). Pulse-wave Doppler of the LVOT revealed a late-peaking “dagger-shaped” systolic jet with a peak velocity of 3.94 m/s and gradient of 62 mmHg that, respectively, increased to 4.56 m/s and 83 mmHg with Valsalva maneuver Figures . There wThe final impression following diagnostic testing was that this subject had developed symptomatic, dynamic LVOT obstruction and asymptomatic, chronic severe mitral regurgitation, which were felt to be best treated with cardiothoracic surgery. The operation was performed via a median sternotomy with cardiopulmonary bypass and cardioplegic arrest. After opening the left atrium, the mitral valve leaflets appeared thickened and myxomatous, but notably, the anterolateral papillary muscle was anteriorly displaced and inserted directly into the A1 scallop of the anterior mitral leaflet with minimal chordae tendineae attachments Figures . FurtherCongenital malformations of the LV papillary muscles, including their direct insertion into various portions of the anterior or posterior mitral valve leaflets, are rare. Our greatest understanding of anomalous papillary muscle insertion (APMI) comes from its identification in subjects with hypertrophic cardiomyopathy (HCM). The largest two case series that included a total of 23 HCM subjects with APMI established its prevalence between 4.5 and 13% . In subjTo our knowledge, this is the first case report to describe APMI in a non-HCM subject leading to two hemodynamic consequences, significant dynamic LVOT obstruction causing syncope and severe mitral valve regurgitation (MR). Dynamic LVOT obstruction that is characteristic of HCM is due to a thickened LV septal wall and anterior mitral valve leaflet SAM that results in mitral-septal contact during systole. Increasingly, primary abnormalities of the mitral valve apparatus in HCM, including abnormal leaflet coaptation, leaflet elongation, chordal slack, and papillary muscle displacement, have been recognized as promoting SAM and generating obstruction , 15. EveThe mechanism of coexistent severe MR appears to be twofold. In HCM subjects, MR is typically posteriorly directed. In the present case, the echocardiographic appearance and histology of the mitral valve leaflets were consistent with myxoid degeneration, and so it is likely that the redundancy of the anterior mitral valve leaflet led to even more compromised coaptation surface. Secondly, the tethering effect of the hypertrophied anterior papillary muscle causing ventricular displacement of the anterior mitral leaflet may have been contributory, as described previously . The rolFor the typical HCM patient with drug-refractory symptoms, surgery with septal myectomy is the first-line therapeutic approach , 21. If The preoperative diagnosis of APMI can be elusive, especially if one does not have knowledge of this congenital variant. In our case, this anomaly was diagnosed by direct intraoperative visualization but only retrospectively fully characterized when reviewing preoperative echocardiographic imaging. This concern has been raised even when examining HCM subjects; as in one series, 90% of necropsy HCM specimens were retrospectively characterized by echocardiography once the diagnosis was elucidated at autopsy . In anot


Reconstructive surgery following an internal hemipelvectomy for a malignant pelvic tumor is difficult due to the structural complexity of the pelvis and the massive extension of the tumor. While high complication rates have been encountered in various types of reconstructive surgery, resection without reconstruction reportedly involved fewer complications. However, this method often results in limb shortening with resultant instability during walking. We reported herein leg lengthening performed to correct lower limb shortening after an internal hemipelvectomy, which improved ambulatory stability and overall QOL. An 18-year-old male patient came to our hospital to correct a lower limb discrepancy resulting from a left internal hemipelvectomy. His left pelvis and proximal femur had been resected, and the femur remained without an acetabular roof. His left lower limb was about 8 centimeters shorter. The left tibia was lengthened 8 centimeters with an external fixator. After the lengthening, the patient was able to walk without support and his gait remarkably improved. Additionally he no longer required placing a wallet in his back pocket as a pad as a means of raising the left side of his torso while sitting. Leg lengthening was a useful method of improving ambulation after an internal hemipelvectomy. The treatment of pelvic tumors involving the acetabulum in particular is difficult due to the anatomical complexity of the pelvis and the massive extension of the tumor . AblatioResection without reconstruction, that is, the “flail hip,” is an alternative form of pelvic tumor treatment , 12 but The purpose of this paper is to assess bone lengthening as a method of correcting leg length discrepancy and improving ambulation following an internal hemipelvectomy, especially in cases involving a nearly complete resection of the half pelvis and the ipsilateral proximal femur.A 13-year-old boy received the diagnosis of clear cell chondrosarcoma in his left ilium and underwent a left internal hemipelvectomy. The whole left ilium, most of the left ischium, part of superior pubis ramus, and the left femoral head and neck were resected. The total iliacus and the psoas major muscle were resected while the tendon of the psoas muscle was left intact. About half of the left gluteus maximus and medius muscles were left and resutured to the abdomen or back muscles after bone resection, preserving the extension and abduction functions of the left hip joint. The left adductor muscles were left intact.At 18 years old, the patient came to our hospital to correct the discrepancy in the length of his legs. A hip radiograph showed evidence of surgical resection of the left pelvis, femoral head, and neck . A radioBefore the operation to lengthen the limb, we gauged the desired effect using a shoe-lift to simulate changes in limb length. After several trials, an 8-centimeter shoe-lift raised the pelvis sufficiently to achieve evenness in the height and position of the hips. Then, the left tibia was lengthened using an unilateral external fixator . The osteotomy was performed at the proximal tibia, and three half pins 6 mm in diameter were inserted above and below the osteotomy site . The patThe patient was able to walk short distances without a crutch after fixator removal. One year later, he was able to walk without any assistance. Five years after fixator removal, he was able to walk 4-5 kilometers continuously without any apparent gait abnormality and could stand on the left leg steadily . The radThe annual incidence of bone malignancies is about 4–7 cases/million children under 15 years of age , 17. NeaWhile nonreconstruction following an hemipelvectomy can prevent complications such as those that occur when reconstruction is performed, some gait impairment will result from the discrepancy in limb length and the resultant instability of the lower limb.As a solution to this problem, Eilber et al.  reportedIn the present case, the discrepancy was approximately 8 centimeters although some variation resulted from the difference in loading on the affected and healthy limb. The 8 cm shoe-lift was effective, but in addition to being uncomfortably heavy, this height also made the patient prone to tripping over and too heavy to walk. Nonetheless the surgical lengthening was generally very favorable, allowing the patient not only to walk without any ambulatory aids but also to sit on a chair without using a wallet to bolster his torso.Another concern following the procedure was the possibility of the tip of the residual femur penetrating overlying soft tissue by loading during ambulation. In a study of the course of 98 untreated cases of congenital hip dislocation (CHD) among patients aged 17–65 involving a total of 132 hips, Kono  reportedTo assess the functional efficacy of flail hips, Takami et al.  reviewedThe present report features only one case of surgical intervention to treat the shortening of a lower limb due to an hemipelvectomy. The result was nonetheless very favorable from both the clinical perspective and patient satisfaction. We consider this method as a viable option for the treatment of impaired ambulation due to an hemipelvectomy.Lengthening the shortened limb was a useful method of improving ambulation following an internal hemipelvectomy.Five years after fixator removal, the patient walked without apparent gait abnormality, and could stand on the lengthened leg steadily without any support.


We report a rare case of a juxta-adrenal schwannoma that could not be discriminated from an adrenal tumor before surgical resection and was complicated by bilateral hyperaldosteronism. To the best of our knowledge, this is first case in which both a juxta-adrenal schwannoma and hyperaldosteronism co-existed.A 69-year-old male treated for hypertension was found to have a left supra-renal mass (5.8 × 5.2 cm) by abdominal computed tomography. His laboratory data showed that his plasma aldosterone concentration (PAC) was within the normal range, but his plasma renin activity (PRA) was reduced, resulting in an increased aldosterone/renin ratio (ARR). Load tests of captopril or furosemide in the standing position demonstrated autonomous aldosterone secretion and renin suppression. Adrenal venous sampling (AVS) with ACTH stimulation indicated bilateral hypersecretion of aldosterone. A left supra-renal tumor was resected because of the possibility of malignancy and was found to be a benign schwannoma arising from the juxta-adrenal region together with an adrenal gland. The dissected left adrenal gland was morphologically hyperplastic in the zona glomerulosa, but was immunohistochemically negative for CYP11B2 . Multiple CYP11B2-positive adrenocortical micronodules were detected in the adrenal gland, indicating micronodular hyperplasia. Although bilateral aldosteronism was indicated by AVS before the operation, the PRA, PAC and ARR values were within their respective reference ranges after resection of the unilateral tumor, suggesting that the slight increase in hormone secretion from the remaining right-sided lesion could not be detected after resection.A clinical and morphologic diagnosis of juxta-adrenal schwannoma is difficult, particularly in a case of hyperaldosteronism, as shown in this case. These data suggest the complexity and difficulty diagnosing adrenal incidentaloma. A schwannoma is a benign, encapsulated and slowly progressive tumor originating from the nerve sheath. It occurs predominantly in the head and neck region as well as in the flexor surfaces of the upper and lower extremities . RetropeWe treated a 69-year-old male patient with a juxta-adrenal schwannoma that could not be diagnosed preoperatively.Our patient is a 69-year-old Japanese man. At the age of 55 years, he underwent a medical assessment for occasional left abdominal pain. A left suprarenal mass was discovered by abdominal computed tomography (CT), and he was referred to our hospital. The tumor was 4 cm in size at that time, and no hormonal production had been detected (plasma renin activity (PRA), 0.5 ng/mL/h; plasma aldosterone concentration (PAC), 105 pg/mL; aldosterone/renin ratio (ARR), 210; the load test of furosemide in the standing position ; normal catecholamine and cortisol levels). Consequently, a diagnosis of a non-functional adrenal tumor was determined. The patient did not want to undergo surgery and was followed without surgical excision. The follow-up at our hospital continued for 5 years, and the tumor size (4.5 × 3.8 cm) did not appear to be increasing . His blood biochemistry data, including potassium, are within the normal range. His endocrinological data are also within the normal range, including the PAC (25.5 pg/mL), PRA (0.5 ng/mL/h) and ARR (51.0).A schwannoma is usually a benign tumor emerging from a peripheral nerve sheath. A schwannoma in the adrenal gland is very rare , 9 and tIn this case, the co-occurrence of a schwannoma and aldosterone-producing micronodules is considered to be incidental, but the possibility that humoral factors secreted from the schwannoma may have affected aldosterone secretion cannot be fully excluded. No case of a schwannoma and concurrent bilateral autonomous aldosterone-producing lesions in the same patient has been reported in the literature, including schwannomas arising from places other than the retroperitoneum. However, the etiologies of these non-neoplastic autonomous aldosterone-producing lesions in the adrenal gland remain unknown, and further investigations are required for clarification.After the operation, the patient had normal blood pressure with the administration of an antihypertensive drug (Amlodipine besylate 2.5 mg/day). His PRA, PAC and ARR values were within the reference ranges even though bilateral aldosteronism was indicated by AVS before the operation, possibly because slight changes in hormone secretion from the remaining right-sided lesion cannot be detected in the peripheral blood. A careful follow-up of the hormone levels and a pathological examination of a right-sided adrenal lesion, if present, are needed to clarify this supposition. Yamazaki et al. reported that all bilateral cases of aldosteronism have similar pathological features in both the right and left adrenals (18 out of 18 cases) . TherefoPreoperative diagnosis of a juxta-adrenal schwannoma is difficult, especially in a case with hyperaldosteronism. To the best of our knowledge, this is first case in which both a juxta-adrenal schwannoma and hyperaldosteronism co-existed, demonstrating the complexity and difficulty of the diagnosis of adrenal incidentaloma.


Calcinosis cutis is a condition of accumulation of calcium salts within the dermis leading to the formation of a calcified mass. This complication has been reported in acne vulgaris and other systemic metabolic disorders. This paper presents a rare case of calcinosis cutis in a 14-year-old male which was found at a routine orthodontic assessment. Acne vulgaris (AV) is a benign dermatological condition resulting from overproduction of sebum by the skin's sebaceous glands. The condition commonly occurs in adolescents and young adults. It primarily affects the face, upper back, and chest, producing two types of lesions, inflammatory lesions and comedones. Physical sequelae of the condition such as scar formation and hyperpigmentation are common, but rarer sequelae such as cutaneous calcifications are infrequently reported in the literature , 2.Calcinosis cutis occurs as a result of the deposition of calcium salts in the dermis and subcutaneous tissues. This complication has not only been reported in AV and systemic metabolic disorders such as hyperparathyroidism, infection, or connective tissue disorders but can also occur following trauma to an area of the skin. Calcinosis cutis can be subdivided into 4 groups based on aetiology: dystrophic, metastatic, iatrogenic, and idiopathic . The dysThis paper presents a case report of dystrophic calcinosis cutis in a 14-year-old male, resulting from inflammatory facial acne.A Caucasian male aged 14 years attending an orthodontic clinic for a routine assessment presented with an incidental radiographic finding. A well-defined calcified mass was visible at the apex of the maxillary right permanent canine on the orthopantomogram (OPG) radiograph .A lateral cephalometric radiograph showed that this mass was significantly superior to the dentition in the antral inferior nasal region . The masThis calcified mass had not been present on the OPG radiograph taken previously, at age 10 years .From the age of 12, it was reported that the patient suffered with facial AV. He attended a consultant dermatologist who treated the condition successfully. The calcified mass was previously assessed at dermatological review. Despite the fact that it was a solitary lesion, it was diagnosed as dystrophic calcinosis cutis. The dermatologist advised no intervention as there was a significant risk of facial scarring if surgically removed. The mass was asymptomatic and presented little risk of infection. The patient was placed on annual review with his dermatologist.Although the mass was palpable and mobile, the patient's dermis was intact and acne-free. The mass was not visible clinically on extraoral examination. Facial profile and contour were normal. Orthodontically, he presented with a Class III incisor relationship on a Class III skeletal base with crowding in the maxillary arch. Both the patient and his parents were given the option of orthognathic surgery to treat his malocclusion and underlying significant skeletal relationship discrepancy. After extensive discussions and reviews with a maxillofacial surgeon, they opted for orthodontic treatment only, namely, alignment of the maxillary arch teeth on a non-extraction basis, followed by permanent retention. The orthodontic treatment was uneventful and successful.Propionibacterium acnes. AV begins with the release of androgens, which in turn leads to the increased production of sebum in the sebaceous glands and intrafollicular hyperkeratosis [Acne vulgaris is a dermatological disease caused by changes in the hair follicle and its associated sebaceous gland, jointly called the pilosebaceous unit. AV affects approximately 80% of the population between 12 and 25 years of age. It does not display race or gender prevalence differences . Three feratosis . The resComedones are subdivided into blackheads and whiteheads. A blackhead is a comedone which is open to the skin surface allowing the contents to escape. The black colour is due to melanin pigmentation. The whitehead is a closed comedone, which does not allow its contents to escape.P. acnes leads to infection and the formation of acne lesions including papules, pustules, nodules, and/or cysts [Inflammatory lesions arise if the walls of a closed comedone rupture. Lipoid tissue is expressed into the surrounding dermis. This sets up a foreign body inflammatory reaction and coupled with or cysts .Healing of the AV inflammatory lesions can occur via two processes. Firstly, healing by fibrous tissue can lead to scar formation, while secondly, the epidermis portion of the remaining comedone walls sends out sheaths of epithelium to encapsulate any inflammatory material. The encapsulated mass can become thickened by the evaporation or absorption of fluid. This thickened mass, coupled with necrotic tissues, which are produced as part of healing, provides an ideal environment for the formation of calcifications known as calcinosis cutis –6.Calcinosis cutis is characterized by abnormal deposits of calcium salts in the dermis and/or hypodermis. It often presents as multiple hard pale plaques, nodules, or papules; however, it can present as a singular lesion also , 5. BaseDystrophic calcinosis cutis occurs in areas of tissue damage secondary to infection, inflammatory processes, connective tissue diseases, or cutaneous neoplasms . It is tDystrophic cutaneous calcification secondary to long-term acne was first reported in 1928 by Hopkins  who founDiagnosis of dystrophic calcinosis cutis is based on serological investigations, suitable imaging, and biopsy if required , 2, 4. HDue to its radiopaque nature, dystrophic calcinosis cutis can be visualized on plain film radiographs; however, other useful modalities which could have been used include cone beam computed tomography (CBCT) and ultrasound . These iIn cases of diffuse calcinosis cutis, such as metastatic or iatrogenic, medical management is required to correct the systemic imbalance. This includes the use of warfarin, bisphosphonates, minocycline, ceftriaxone, diltiazem, aluminium hydroxide, probenecid, intralesional steroids, and intravenous immunoglobulin . For casDystrophic calcinosis cutis can occur as a sequela of long-term AV or alongside underlying metabolic disorders. Dentists should be aware of this phenomenon, as it can occur in the facial region and may present as an incidental finding on routine dental radiographs, both intra- and extraoral.


Pelvic actinomycosis is an uncommon, slowly progressing granulomatous infection that has been associated with the presence of intrauterine devices. Due to its unspecific clinical and radiologic findings, it can mimic pelvic or intra-abdominal malignancy leading to mutilating surgery of high morbidity. Rarely, diagnosis is made preoperatively and in most cases surgical intervention is necessary. The patient in our case is a 42-year-old female with an IUD for 15 years diagnosed with pelvic actinomycosis. Patient was uniquely diagnosed preoperatively through paracentesis and treated conservatively with prolonged antibiotic therapy and without any type of surgical intervention. Follow-up at 1 year showed almost complete radiologic resolution of the inflammatory mass, nutritional recovery, and absence of symptoms. Pelvic actinomycosis can be successfully diagnosed and treated medically without surgical interventions. Actinomyces israelii, an anaerobic gram positive bacillus that is part of the normal flora of the oropharynx, gastrointestinal tract, and female genital tract [Pelvic actinomycosis is a rare indolent disease caused most frequently byal tract –5. The ial tract , 3, 6. Ial tract . Pelvic al tract . A definA 42-year-old Caucasian female, gravida 3 para 1 , was admitted with a 2-month history of diffuse abdominal pain, decreased appetite, nausea, vomiting, abdominal distention, weakness, and a twenty-pound unintentional weight loss. Her history was significant for a twenty-eight-pack-year tobacco use and a copper IUD placed approximately fifteen years earlier.On admission, the patient was found to be cachectic, tachycardic, and tachypneic but afebrile. Her abdomen was moderately distended, tender to palpation throughout but without acute peritoneal signs. It was diffusely tympanic but dull over bilateral lower quadrants, with hypoactive bowel sounds. External genitalia and vagina were normal. Cervix was parous in appearance with two strings visualized from the cervical os; IUD was removed and sent for microbiologic studies. Uterus was difficult to palpate secondary to pain and adnexal tenderness. Adnexa were diffusely tender with bilateral fullness. Rectal exam showed normal tone and normal rectovaginal septum with fullness palpated anteriorly.The patient's initial laboratory evaluation showed marked leukocytosis , anemia (Hgb: 10.9), hypoalbuminemia , and mildly elevated CA 125 (54). AST/ALT, CEA, and CA 19-9 were normal. No infectious disease was found per evaluation via urine and blood cultures, gonorrhea, chlamydia, vaginitis panel , tuberculin test, HIV, or hepatitis B and hepatitis C.CT of the abdomen demonstrated marked bowel distention and a complex cystic mass within the left pelvis with enhancing septations measuring 8.2 × 6.7 × 9.8 cm, retroperitoneal lymphadenopathy, and small ascites with peritoneal enhancement. Large distended bowel loops surrounded the heterogeneous lesion peripherally . Actinomyces  were started for coverage of pelvic abscess of unknown etiology with noted improvement in leukocytosis after 72 hours. Nasogastric tube was placed for upper GI decompression and control of emesis for suspected partial small bowel obstruction. Total parenteral nutrition was also started to improve her malnourished state. Gynecologic oncology was consulted and concluded that the likelihood of carcinomatosis was very low based on the patient's age, absence of omental caking or peritoneal implants on CT, and only mildly elevated CA-125. In the presence of a neglected IUD for 15 years with low suspicion for malignancy, actinomycosis was a likely diagnosis. CT-guided diagnostic paracentesis was performed after bowel decompression was achieved with NG tube. Purulent peritoneal fluid was obtained which was negative for malignancy, yet positive forinomyces . Copper inomyces .One week after initiation of antibiotic therapy, repeat CT of abdomen and pelvis showed stable to slightly improved fluid collections compared to prior exams. As final laboratory diagnosis was achieved, Vancomycin and Meropenem were discontinued after 8 and 11 days of treatment, respectively.The patient was started on Ampicillin-Sulbactam 3 g IV every 6 hours, which helped control her leukocytosis. On day five of the Ampicillin-Sulbactam, AST and ALT values were 209 and 148, respectively, and patient was diagnosed with transaminitis secondary to the Ampicillin-Sulbactam. The Ampicillin-Sulbactam was discontinued, which improved the transaminitis, and the patient was started on Penicillin 3 million units IV every 4 hours, which was continued for a total of 4 weeks.After 3 weeks of in-hospital management, patient was tolerating regular diet with regular bowel movements and improved pain control. Patient was discharged home with close outpatient follow-up arranged. At her outpatient follow-up four weeks later, she was transitioned to Penicillin 500 mg PO four times daily. Patient continued Penicillin orally for 1 year, during which she was gaining the strength and weight back with assistance of nutrition and occupational therapy professionals.Repeat CT of abdomen and pelvis 1 year after initiation of treatment demonstrated complete resolution of bowel distention, ascites, and adenopathy; the left adnexal mass had more than 50% size reduction, measuring 3.9 × 3.2 cm . The patPelvic actinomycosis has a characteristic invasion pattern that causes an intense inflammatory reaction with subsequent risk of intestinal obstructions, strictures, and even mass effect leading to ureteral obstruction. Acute surgical intervention is usually performed due to very unspecific clinical presentation with concern for malignancy or acute abdomen. This approach frequently leads to extensive removal of reproductive or gastrointestinal organs with greatly increased morbidity. If actinomycosis is highly suspected or diagnosed preoperatively, delayed surgical intervention after prolonged antibiotic therapy and metabolic support is the preferred approach.Diagnosing pelvic actinomycosis before surgical intervention is extremely uncommon. Although this chronic infection can manifest as tuboovarian abscesses, in many cases it mimics pelvic or intra-abdominal malignancy, cervix carcinoma, or colorectal cancer, leading to surgical intervention in almost all cases . To our In our case, diagnosis was successfully achieved through CT-guided paracentesis instead of the commonly performed surgical approach. The patient was then treated exclusively with noninvasive medical management. We found only six other reports of preoperative diagnosis of pelvic actinomycosis that support exclusive medical management in the absence of a surgical abdomen –10. As l


Factitious ulcers are characterized by self-inflicted lesions with multifactorial origin. These lesions are frequently found in head, neck, and hands. This report shows a 6-year-old boy diagnosed with factitious oral ulcers that occurred after the self-biting of buccal vestibule and nail-scratching of gingival tissue. Clinically, a significant swelling was observed, hard on palpation, located at the right lower third of the face, next to the posterior area of the mandible. In the intraoral examination, ulcers at different healing stages were noted on the swelling area. During the anamnesis, the father reported a change in his familial structure that triggers psychological stress, providing the clues to the presumptive diagnosis of factitious oral ulcers. We prescribed the topical use of Gingilone® three times a day to control the local pain and inflammation. At 7-day follow-up, we noticed the reduction of extraoral swelling and the initial healing of the ulcers. The presumptive diagnosis was confirmed at 30-day follow-up, with the lasting remission of oral lesions. The treatments of factitious oral ulcers should be individually tailored for each patient, focused on a multidisciplinary approach, including psychotherapy and periodic clinical control. To the best of our knowledge, gaps of evidence lead to the lack of standardized clinical protocols on this issue. The factitious ulcers are characterized by self-inflicted lesions related to multifactorial origin, such as accidents and/or chronic habits . Such lePeople more susceptible to develop factitious ulcer include individuals with syndromes, insensitivity to congenital pain, intellectual disability, autism, or schizophrenia, besides children from unstructured families, homeless people, drug addicts, and sexual abuse victims , 4, 5. T morsicatio buccarum [The factitious ulcers in nonsyndromic individuals are either intentional  or unintentional (organic) . The intbuccarum . These hbuccarum , 7.The prevalence of factitious ulcers among nonsyndromic individuals seems to be higher among children, teenagers, and young people 17 to 38%) compared to adults (4%) . Also, f% compareThis report shows a singular clinical case of factitious oral ulcers in a 6-year-old boy without systemic disease.A 6-year-old boy accompanied by his father came to the Clinics of Pediatric Dentistry and Stomatology of the Bauru School of Dentistry, University of São Paulo (FOB-USP). The father previously signed the free consent form, authorizing the treatment and the publication of this case. During the anamnesis, the father reported that they first sought a doctor suspecting of parotiditis that was not confirmed. The doctor referred the child to the dentist. Then, the father reported his concern with an abnormal tumescence that appeared in the right side of the boy's face two days ago. This lesion was associated with pain on palpation. Other relevant clinical findings were the allergy to insect bites and tonsillectomy performed eight months ago. The boy was not under medical treatment or medication use.The extraoral clinical examination revealed that the patient had a significant swelling, hard on palpation, in the right lower third of the face, next to the posterior area of the mandible's body . In the During the session, the boy was very quiet and cooperative, demonstrating his shy behavior. In the anamnesis, the father reported two significant facts: (1) a recent change in his familial structure, when his daughter left home after her marriage; (2) the very close relationship between his children. At that moment, we suggested the hypothesis of self-harm for attention, which led to the presumptive diagnosis of factitious oral ulcers. The lesions were probably produced by self-biting of buccal vestibule and nail-scratching of gingival tissue. These hypotheses were supported by additional reports, describing the boy's habit of introducing his fingers into the mouth, in the same side where the ulcers were observed.We established the treatment for pain and inflammation control, by the topical application of Gingilone ointment  on the ulcers, in the regime of three times a day for one week. After that, the patient was advised to return periodically until a conclusive diagnosis.At 7-day follow-up appointment, we observed the swelling regression during the extraoral examination. The intraoral examination revealed the initial healing of the ulcers. At 30-day follow-up appointment, we observed the total remission of the swelling and ulcers on the right side of the mandible Figures  and 6. TAt this moment, the patient is being periodically monitored to observe the possible recurrence of lesions. We recommended that the parents seek specialized psychological treatment to help the boy with the new family routine.Self-harm is the aggressive manifestation of the individual with psychological and emotional disturbances . It resuThis present clinical case of factitious oral ulcers was linked directly to the boy's emotional stress caused by a change in his family dynamics. The comprehensive anamnesis provided useful information reported by the father and was of major importance to determine the lesion etiology . StewartThe literature reports the presence of factitious ulcers on oral mucosa and face . Most ofThe clinical treatment of individuals with factitious ulcers is problematic because of the behavior of patients, normally very quiet and shy, reluctant in accepting the self-harm as the cause of the lesion itself , 11. In In clinical failures, the use of mouthguards could be an alternative to restrict the deleterious habits, by the introduction of a customized mouthguard with lingual and labial barriers . This apIn this present case, intraoral devices were not necessary to prevent self-harm. We achieved a satisfactory result by a clinical control of lesions with minimal intervention. The instruction of parents and the use of an anti-inflammatory ointment were enough to promote the healing of ulcers and the prevention of their recurrence. Besides, we emphasized the mandatory importance of oral hygiene to prevent secondary infections.A comprehensive anamnesis associated with clinical examinations is important to the correct diagnosis and proper treatment of factitious oral ulcers. The early diagnosis of lesions and the recognition of their risk factors enable a more conservative clinical approach and the maintenance of the anatomical and physiological health of oral and perioral structures. Due to the lack of available literature on this topic, the establishment of a standardized treatment protocol is difficult. To achieve successful outcomes, the treatments of factitious oral ulcers should be individually tailored for each patient, with focus on a multidisciplinary approach, including psychotherapy and periodic clinical control of possible recurrences.


The management of appendicitis with appendicectomy is very common in surgical practice. A recognised complication of appendicitis and appendicectomy is the formation of an enterocutaneous fistula. We present the case of a seventy-five-year-old woman who presented with an appendicocutaneous fistula on the background of an open appendicectomy performed sixty years prior to presentation. Appendicectomy is one of the most common surgical procedures performed in general surgical practice. Appendicitis and appendicectomy can be associated with a number of complications. Complications associated with appendicitis increase significantly when the appendix is perforated or gangrenous with periappendicitis . A rare A seventy-five-year-old female was referred by her general practitioner for management of a discharging sinus at the site of an open appendicectomy scar. The discharging sinus was associated with abdominal pain in the right iliac fossa. The patient reported that the pain commenced a month prior to presentation with the evolution of an erythematous area surrounding the scar that slowly evolved to a discharging sinus. Sixty years prior to presentation the patient had an open appendicectomy for management of clinical appendicitis. Unfortunately, due to the timing of presentation no record of that presentation or procedure existed for review. The patient reported no abdominal issues in her history after appendicectomy prior to this event.The patient's past medical history included ischaemic heart disease for which she had cardiac stents and a cerebral artery aneurysm that had been clipped approximately 21 years prior to presentation. Regular colonoscopies were unremarkable and there was no documented or pathological history to suggest any inflammatory bowel disease. Her regular medications included aspirin, atorvastatin, and metoprolol, and she was known to be allergic to penicillin and cephalosporins.Examination of her abdomen revealed a retracted appendicectomy scar with surrounding erythema and a small sinus discharging faeco-purulent material. Her abdomen was soft and nondistended with tenderness to palpation in the right lower quadrant with associated guarding. Biochemical investigations were unremarkable. Computed tomography (CT) of her abdomen and pelvis was performed and suggested chronic tethering of the lateral wall of the ascending colon to the adjacent abdominal wall with herniation of part of the lateral wall of the ascending colon through a defect in the oblique muscles with fistulation from this hernia to the skin surface .The initial treatment goal was to aim for a “controlled fistula.” A controlled fistula refers to an enterocutaneous fistula without evidence of sepsis or localised infection . The patThe patient was admitted for a definitive surgical procedure several weeks after her initial presentation. The aims of the procedure included refunctionalisation of the entire bowel, resection of the fistula and any associated bowel, and secure abdominal wall closure. The procedure commenced with en bloc resection of the appendicectomy scar with careful resection around the fistula tract while tracking its course into the intraperitoneal space. The fistula was found to be intimately related to the distal caecum that was adherent to the anterior abdominal wall. The specimen was liberated from the caecum using a linear stapler and was sent for histopathological analysis. The specimen consisted of a piece of fibrofatty tissue measuring 85 × 55 × 30 mm with an overlying ellipse of skin measuring 73 × 20 mm with an attached portion of intestine measuring 12 mm in length and 35 mm in diameter. On the skin an old scar measuring 55 mm was identified, and serial sections identified a sinus filled with faecolith measuring 3 mm in diameter. Sections of the specimen were examined microscopically that showed skin with underlying adipose tissue and a portion of a stump appendix showing mucosa with underlying submucosa and muscle layer. Surrounding tissue showed fibrosis, vascular proliferation, and foreign body type giant cell reaction. There was no evidence of malignancy. Overall the features favoured a stump appendix with a fistula tract.The patient was admitted to a surgical ward for postoperative management. This was uncomplicated and she was discharged home five days after procedure. To date the patient has had no complications.The formation of a postappendicectomy fistula is rare, but significant as the associated morbidities can be devastating. Major aetiological factors that contribute to postappendicectomy fistula formation include leakage from the appendiceal stump, neoplasm of the appendix and/or caecum, infective bowel conditions , inflammatory bowel disease , and distal obstruction , 6. The The relation of postappendicectomy complications to surgical technique, purse-string suture versus simple ligation of the stump, is not well established . PerhapsThe management of a fistula may be conservative or surgical. Nonsurgical options for a fistula include vacuum assisted closure (VAC), fistuloscopy with fibrin glue injection, and the use of monoclonal antibody pharmacotherapy in patients with Crohn's disease . SurgicaOur case represents a latent, rare complication of stump appendicitis after appendicectomy. From the evidence available to us, we believe that the aetiology of our patient's fistula was secondary to stump appendicitis in a partially herniated caecum.


Background. Adrenal collision tumors (ACTs), in which distinct tumors coexist without intermingling in the same adrenal gland, are rare and their actual prevalence is unknown. ACTs commonly consist of adrenal cortical adenoma, pheochromocytoma, or metastatic malignant tumor. Case Report. A 32-year-old woman who had been experiencing gastric discomfort for one month was referred to our hospital with abnormal imaging findings. The physical examination and the laboratory data including endocrine studies were unremarkable. Abdomen computed tomography (CT) and magnetic resonance imaging (MRI) showed two adjacent masses in the left suprarenal fossa, and a laparoscopic left adrenalectomy was done. Histological and immunohistochemical (IHC) examinations revealed two distinct tumors: a pigmented adrenal cortical oncocytoma (ACO) and a ganglioneuroma, respectively. Conclusion. Both tumors are rare in the adrenal gland and exist as ACTs only exceptionally rarely. This is the first reported case of coexisting oncocytoma and ganglioneuroma in the same adrenal gland to our knowledge. Most primary adrenal neoplasms are from either cortical cells or medullary chromaffin cells . AdrenalA 32-year-old Korean woman who had been experiencing gastric discomfort for one month was referred to Korea University Guro Hospital with abnormal imaging findings. Abdominal computed tomography (CT) revealed a 4 cm low-density mass and another 1.8 cm enhancing mass in the left suprarenal fossa, both of which were considered to have originated from the adrenal gland or retroperitoneal soft tissue . She hadLaboratory data showed normal values for complete blood cell counts and chemistry panel results. Endocrine tests were also done, and the results are summarized in An additional magnetic resonance imaging (MRI) scan showed two abutting masses on the left adrenal gland . T2 imagGrossly, two adjacent but uncombined well-demarcated oval-shaped masses were confined within the yellowish adrenal cortex . The smaThe larger mass (4.5 × 3.5 × 3.0 cm) was whitish to light yellowish in color, and the consistency was firm . MicroscThe postoperative course was uneventful. After one year, physical examination and follow-up CT also showed no remarkable findings.A collision tumor is said to occur when two or more histologically distinct tumors coexist without admixture at the interface . Since aThe pathogenesis of ACTs is unknown, but several possible theories have been suggested , 11, 12.ACOs are usually clinically silent , 13. In The prevalence of adrenal ganglioneuromas has been reported to range from 5 to 9.4% in a series of adrenalectomies . Owing tACTs, including oncocytomas and ganglioneuromas, have rarely been reported. Only one case showed an oncocytoma with an aldosterone-producing adenoma . GanglioIn conclusion, both oncocytoma and ganglioneuroma are unusual components of ACTs, and, to the best of our knowledge, the coexistence of these two tumors is unprecedented. Furthermore, a pigmented oncocytoma has not yet been reported in the adrenal gland, and it is also a very rare condition in other sites , 17. The


Pneumoperitoneum is not always associated with hollow viscus perforation. Such condition is called non-surgical or spontaneous pneumoperitoneum. Intrathoracic causes remain the most frequently reported mechanism inducing this potentially life threatening complication. This clinical condition is associated with therapeutic dilemma. We report a case of a massive isolated pneumoperitoneum causing acute abdominal hypertension syndrome, in a 75 year female, which occurred after difficult airway management and mechanical ventilation. Emergent laparotomy yielded to full recovery. The recognition of such cases for whom surgical management can be avoided is primordial to avoid unnecessary laparotomy and its associated morbidity particularly in the critically ill. Pneumoperitoneum (PP) commonly indicates a perforated hollow viscus that requires prompt surgical exploration and intervention . However2). She had no surgical past history and she experienced no other recent complaints especially gastrointestinal. On first examination by the resuscitation team, the patient was in a comatose state with GCS at 3/15. She was afebrile. She had blood pressure at 90/40mmHg and heart rate at 140bpm. She had no dyspnea. White blood cells count was 7500/mm3 and CRP, 8mg/L. Airway management has been performed by a young trainee after a rapid sequence induction using hypnomidate (20mg) and suxamethonium (100mg). Mallampati classification [A 75-year-old female was admitted in the medical ICU for a sudden onset severe coma related to acute ischemic stroke complicating persistent atrial fibrillation after a short course in the cardiology department. She was obese BMI, 36kg/m6kg/m2. SNon-surgical pneumoperitoneum (NSP) is a commonly described entity but not always recognized by physicians. Its prevalence is estimated at 5 to 15% . Most caThe diagnostic approach of NSP is usually facilitated by the chronological relationship between the procedure likely to cause and its occurrence. Indeed, in the present case, the PP onset in the hour following the airway management and mechanical ventilation startup was very suggestive . HoweverAlthough laparotomy is warranted in the case of surgical PP, both as diagnostic and therapeutic procedure, it remains questionable in the case of NSP. Mularski conducted a literature review which included 196 cases of NSP, only 45 of them underwent laparotomy without evidence of perforated viscus . The recNon-surgical pneumoperitoneum is an uncommon entity. Intrathoracic causes especially during ventilatory support are the most frequently reported. Such cases are difficult to manage and create a major surgical dilemma. Surgeons and intensivists should be aware of the possible causes of NSP and should play the decisive role in preventing needless laparotomy in these patients. In cases of massive or tension PP and worsening respiratory and/or hemodynamic condition, percutaneous peritoneal cavity drainage could improve cardiopulmonary parameters and should be tried as a first line treatment.


One rare complication of appendectomy is a retained appendicolith, which can become a focal point for infection presenting hours to years after surgery. We present a case in which a 50-year-old male presented to the emergency department with a small bowel obstruction one week post appendectomy. A diagnostic laparoscopy was performed, and a necrotic appendiceal specimen containing a staple line across the base as well as an appendicolith was removed. It is crucial to include rare surgical complications in our differentials, alongside the more common pathologies when approaching and treating patients with abdominal pain. There is a myriad of post-operative complications from laparoscopic surgery that we as emergency physicians may be prone to dismiss due to the prevalence and less-invasive nature of the procedure. The prevalence of laparoscopic surgery, however, should force us to remain keenly alert and aware of complications in both the acute and chronic state. It is imperative for us to keep the possibility of not only adhesions, but persistent infection of remnants , as well as the rarer occurrence of retained surgical specimens, in our differentials for our abdominal pain patients. In some cases these pathologies may not present until decades later.1Clostridium difficile colitis, and hernia repair, presented to the emergency department (ED) with a chief complaint of abdominal pain, nausea and diarrhea. The patient was one-week status post appendectomy performed at an out-of-state hospital. Patient was readmitted to the same hospital hours after the surgery with bloating and abdominal pain, was kept overnight and discharged the next day. Since the surgery, the patient had been experiencing worsening abdominal pain, distention, and nausea, with one bout of emesis a few days prior to presentation in our ED. On the day of presentation, the patient experienced three episodes of water diarrhea, night sweats without fever, and intermittent passage of flatus. An outpatient acute abdominal series performed the morning of presentation demonstrated numerous dilated small bowel loops with air-fluid levels suggestive of small bowel obstruction with no free air  of the abdomen and pelvis with intravenous (IV) contrast was performed for further investigation of a suspected intra-abdominal abscess in addition to a small bowel obstruction. Imaging demonstrated abnormal wall thickening along the cecum with dense fluid within the cecum concerning for hemorrhage, as well as a mildly distended small bowel loop in the left anterior abdomen. There was a surgical staple line adjacent to the inferior portion of the loop. Contiguous with the staple line was a small tubular density filled with air and a single calcification .Based upon these findings, there was concern for retained appendix in addition to small bowel obstruction and intra-abdominal abscess. Antibiotic coverage with IV piperacillin/tazobactam was started and the patient was admitted to general surgery. Diagnostic laparoscopy was performed that day, and in the operating room inflammation of the parietal peritoneum was noted as was a large purulent pocket located below the patient’s largest prior periumbilical port site. Additionally, a necrotic appendix was visualized and removed. Further examination demonstrated that the appendix specimen contained a staple line across the base of the appendix containing an appendicolith . The patAppendicitis is a common surgical emergency with over 270,000 appendectomies performed in the United States each year.4What do we already know about this clinical entity?Appendicitis is a common surgical emergency with over 270,000 appendectomies performed in the U.S, each year. Two rare complications of appendectomy are stump appendicitis and retained appendicoliths.What makes this presentation of disease reportable?Over the past 40 years, there have been 30 reported cases in the literature of retained appendicoliths causing intra-abdominal abscesses.What is the major learning point?Retained appendicoliths can have unusual presentations and present with complications days to years after surgery. We present a rare presentation of small bowel obstruction caused by an intra-abdominal abscess that developed from a retained appendicolith.How might this improve emergency medicine practice?This case serves as an important reminder to include these rare surgical complications in our differentials, alongside the more common pathologies, when approaching and treating patients with abdominal pain.Two rare complications of appendectomy are stump appendicitis and retained appendicoliths. Patients may present hours to years after surgery. Stump appendicitis is caused by a retained appendicular stump. A 60-year review of medical literature found a total of 57 reported cases of stump appendicitis.An appendicolith is a collection of fecal debris and calcium salts residing in the appendix that can lead to acute appendicitis. With increased use of CT, they are becoming a more common incidental finding. An extraluminal fecalith, however, is a focal point for infection and should cause concern for appendiceal perforation. Over the past 40 years, there have been 30 reported cases in the literature of retained fecaliths causing intra-abdominal abscesses.9As emergency physicians, it is crucial for us to include these rare surgical complications in our differentials, alongside the more common pathologies when approaching and treating our patients with abdominal pain.