Pampiniform plexus thrombosis is a very rare disease (only less than 25 published cases are available till date), and it is a diagnostic dilemma. The present case is an unusual condition of an elderly gentleman who was finally diagnosed as a case of spontaneous thrombosis of bilateral pampiniform plexus and was managed conservatively. Literature was reviewed to explore potential etiologies and therapeutic strategies.A 65-year-old afebrile gentleman, laborer (in brick industry), and non-smoker with no previous major health problems was admitted with swelling in the bilateral inguinal region. The swelling had started one and half months ago. He had developed severe pain over the swelling for last 1 day with tenderness and indurations. Neither he had history of previous surgeries, chronic cough, dysuria, prostatism, and trauma nor he presented any thrombogenic factors. There was no history of vomiting, abdominal pain, and obstipation. Physical examination revealed normotensive person with BMI of 22.5, was significant only for one tender, movable, and firm to hard 10 cm × 3 cm mass extending from the left deep inguinal ring up to the upper pole of the testis in the scrotum. Another 5 cm × 3 cm mass of similar characteristics was found extending from deep inguinal ring up to the root of the scrotum on right side. The testes and prostate were normal on palpation.On the contrary to preoperative USG, which clinched suspicion of incarcerated inguinal hernia, a thrombosed pampiniform plexus without any evidence of hernia sac was found on the left side during inguino-scrotal exploration. Wound was closed without doing any further procedure. Contralateral inguino-scrotal exploration was spared considering same nature of disease. Postoperative Doppler ultrasonography confirmed the diagnosis of bilateral thrombosed pampiniform plexus. MDCT of whole abdomen revealed no abnormality other than bilateral spermatic cord thrombosis. Blood thrombophilia screening came normal. The subject had an uneventful postoperative hospital course. With 2 years of follow-up, the gentleman is doing well, remaining asymptomatic and had returned to his usual life.Due to extreme rarity, spontaneous thrombosis of the pampiniform plexus may be a diagnostic dilemma and requires a high index of suspicion. Doppler ultrasound is the initial investigation of choice. In the absence of other concomitant disease, beginning the treatment conservatively instead of excising the thrombosed segment is more suitable. Spontaneous pampiniform plexus thrombosis is a diagnostic dilemma, and it is a very rare condition. Less than 25 cases of spontaneous thrombosis have been published in the literature till date . Acute iA 65-year-old afebrile gentleman, laborer (in brick industry) and non-smoker with no previous major health problems, was admitted for painful swelling in the bilateral inguino-scrotal region. The swelling had started one and half months ago in the bilateral inguinal region, and later, it gradually involved the upper part of the scrotum bilaterally. The swelling was small initially, but gradually attained presenting size in the last 4–5 days. The swelling did not reduce on lying down, but it used to get prominent in standing position. Initially, there was mild dragging and aching pain over the swelling, but the pain was increased and became severe since 1 day with tenderness and indurations. There was no history of vomiting, abdominal pain, dysuria, and obstipation. Neither he had history of previous surgery, chronic cough, prostatism, and trauma nor he presented any thrombogenic factors.Physical examination revealed normotensive person with BMI of 22.5, was significant only for one left sided elongated, tender, movable, and firm to hard 10 cm  × 3 cm  mass . It was extending from the left deep inguinal ring up to the upper pole of the testis in the scrotum. Local temperature over the swelling was raised with mild erythama. There was no visible or palpable cough impulse. “Get above the swelling” was not possible. As the swelling was irreducible, the deep ring occlusion test could not be performed. Dull note was found on percussion with no audible gurgling sound. Another 5 cm  × 3 cm  mass of similar character was found extending from the right deep inguinal ring up to the root of the scrotum. Both the testes were normally positioned in the scrotum, but the left one was mildly swollen. Prostate size and penile position were normal. Umbilicus was in normal position in scaphoid abdomen without any tenderness, visible peristalsis, or palpable mass.Contrary to pre-operative grayscale USG finding (hernia containing tubular loops), which clinched the suspicion of incarcerated inguinal hernia, on exploration of left inguino-scrotal region under spinal anesthesia, left spermatic cord was found to be thick, multi-lobulated, blackish-red colored, tubular mass of firm to hard consistency Fig. a, b. ThiPost-operative period was uneventful. The patient was put on anti-inflammatory drugs. Oral feeding was started from the next day, and early ambulation was encouraged. The swelling and pain started to get reduced gradually. There was no episode of shortness of breath or chest pain, tachycardia, and tachypnea in post-op period. BP was normal throughout the post-operative period. There was no development of calf tenderness. Post-operative ultrasonography with color Doppler study Fig. a, b confSpermatic vein thrombosis is an unexpected finding in the differential diagnosis of acutely painful inguino-scrotal region . Most ofIn the etiology of isolated spermatic vein thrombosis, there are many possible predisposing factors, such as trauma to the vascular endothelium, slow venous flow, and hypercoagulability . Kayes eIn the literature also, no report regarding recurrence was found after conservative management. There are no guidelines available for the management of this disease. Hashimoto and Vibeto reported that there is no need to excise the thrombosed plexus, as evidenced by the good results in their case . ConservIsolated spermatic vein thrombosis is a rare event and requires a high index of suspicion. Although present case is bilateral, spermatic vein thrombosis is almost always found at the left side. Doppler ultrasonographic examination is the procedure of choice in the diagnosis of the varicocele thrombosis with higher sensitivity and specificity. Exploratory surgical approach may be needed initially in the absence of Duplex study, to exclude an acutely infarcted testis or incarcerated hernia. But in the confirmed absence of other concomitant disease that necessitates urgent surgical intervention, beginning the treatment conservatively instead of excising the thrombosed segment is more suitable. Although conservative management, including watchful observation and NSAID without anti-coagulation, is acceptable for thrombosis out of external inguinal ring (pampiniform plexus), surgical excision and anticoagulation may prevent pulmonary embolism in deep-seated spermatic vein thrombus  and extending to the nearby renal vein. Surgeons should be aware of this rare clinical entity for prompt management of potential morbidity.


Various terms have been used to describe vascular lesions in the intestine, including angiodysplasia, arteriovenous malformation, and telangiectasia. Such lesions are common in adults and are typified by angiodysplasia, a type of arteriovenous malformation. In contrast, these lesions are rarely seen in the pediatric population. Angiodysplasia may cause gastrointestinal bleeding, which is sometimes an indication for treatment. Considering the high rate of recurrence after surgical treatment, conservative treatments are mainly chosen. We herein report an extremely rare case of a prolapsed colon due to an arteriovenous malformation successfully treated by resection in a 1-year-old girl. We also highlight the differences between pediatric and adult cases.A girl developed bloody stools at 7 months of age. She visited another hospital at 1 year of age because of continuing moderate hematochezia and recent onset of rectal prolapse. Colonoscopy showed a protruding lesion located 15 cm from the anal verge, suggesting a submucosal vascular abnormality. Contrast-enhanced computed tomography and magnetic resonance imaging at our hospital revealed the localized lesion with dilated blood vessels in part of the sigmoid colon; no other lesions were present in the gastrointestinal tract. Laparoscopic-assisted sigmoidectomy was performed. A subserosal vascular lesion was visualized and resected using end-to-end anastomosis. Pathologic examination of the 2.2 × 2.7-cm segment revealed several abnormally enlarged and ectatic blood vessels in the submucosa extending into the subserosa. The lesion was diagnosed as an arteriovenous malformation. The patient had a good clinical course without recurrence at the 2-year follow-up.An arteriovenous malformation in the sigmoid colon may rarely cause intussusception and prolapse of the colon. Complete resection is a radical and potentially effective treatment. Computed tomography and colonoscopy were useful for evaluation of the lesion in the present case. Vascular anomalies, which comprise vascular tumors and vascular malformations, can occur in all parts of the body and tend to more commonly affect the skin and soft tissue in children , 2. VariA previously healthy girl first developed bloody stools at 7 months of age. She then presented to another hospital for continuing moderate hematochezia and rectal prolapse at 1 year of age. She had no any cutaneous lesions or dysmorphosis indicating hereditary syndromes causing vascular lesion, such as Klippel-Trénaunay syndrome, Osler-Weber-Rendu syndrome, blue rubber bleb nevus syndrome, or Proteus syndrome. Colonoscopy performed at that hospital revealed a submucosal lesion with mucosal erosion in the sigmoid colon. Pathologic examination of a biopsy specimen revealed a hematoma. The patient was referred to our hospital for further investigation. A contrast enema revealed no abnormalities of the sigmoid colon or rectum. Colonoscopy showed a protruding lesion located 15 cm from the anal verge, suggesting a submucosal vascular abnormality such as a hemangioma. We planned a second look because the lesion was swollen and covered with erythematous and edematous mucosa, considered to be the result of the rectal prolapse and biopsy.Repeat colonoscopy performed 2 months later revealed that although the reddening and edema had resolved, the lesion had not changed in size Fig. a. ContraMagnetic resonance imaging (MRI) revealed bowel wall thickening in the same area and vascular engorgement with a flow-void appearance. The lesion was thought to be a focal and resectable vascular abnormality; therefore, laparoscopic-assisted sigmoidectomy was performed. An omega-shaped incision was made at the umbilicus, and a multichannel port  was inserted as a platform under direct vision. A 5-mm rigid endoscope was introduced into the EZ Access. A subserosal vascular lesion in the sigmoid colon was visualized Fig. . It had The lesion was extracted from the abdominal cavity through the umbilical incision and resected using end-to-end anastomosis. Pathologic examination of the 2.2 × 2.7-cm segment revealed several abnormally enlarged and ectatic blood vessels in the submucosa extending into the subserosa. Most of the dilated vessels had characteristics of veins, with thin walls and venous valves, while a few arteries had an elastic lamina and a tangled smooth muscle layer Fig. . The diaIn children, vascular anomalies such as infantile hemangiomas are more commonly seen in the skin and soft tissue, while they are rare in the GI tract , 2. VascIn addition, intestinal angiodysplasia in the adult population is usually thought to be an acquired degenerative vascular lesion related to aging and associated factors such as aortic stenosis, renal failure, or coagulopathy , 10, 11.The main symptom in the present case was hematochezia. An AVM might be an important cause of GI bleeding because a previous study showed that double-balloon or capsule endoscopy revealed an AVM in 6.3 to 18.1% of children with occult GI bleeding who underwent examinations by double-balloon endoscopy or capsule endoscopy , 14. AVMBecause type I lesion in adults are often present in multiple locations and often recur after resection, conservative treatments such as pharmacotherapy, intravascular embolization, and endoscopic argon plasma coagulation are often considered as the initial approach , 7. SeveBefore surgery, correct localization of the lesion is important for complete resection. For the detection of colonic angiodysplasia, CT angiography reportedly has sensitivity, specificity, and positive predictive values of 70%, 100%, and 100%, respectively . Our patIn summary, an AVM in the sigmoid colon may rarely cause intussusception and prolapse of the colon. Complete resection of an AVM in the GI tract is a radical and potentially effective treatment. Colonoscopy and CT were useful for evaluation of the lesion in the present case.


Case Description. We present a case of a 22-year-old Sri Lankan woman who presented with femur fractures in a background of childhood nephroblastoma and maxillary fibro-osseous tumor. The patient had biochemical parameters suggestive of primary hyperparathyroidism with a parathyroid mass. The histology following excision of the mass revealed a parathyroid adenoma. Based on the associated clinical manifestations, hyperparathyroidism jaw tumor syndrome was suspected, and genetic studies reported a positive CDC73 mutation with a whole-gene deletion of exon 1–17.  Primary hyperparathyroidism usually occurs secondary to parathyroid adenoma, multiglandular hyperplasia, or parathyroid carcinoma. The patients usually present with incidentally discovered high calcium level and systemic or skeletal manifestations. In young patients with primary hyperparathyroidism, familial syndromes including multiple endocrine neoplasia types 1, 2, and 4 and hyperparathyroidism jaw tumor syndrome should be considered.  Hyperparathyroidism jaw tumor syndrome is an important diagnosis to consider in a young patient presenting with classic clinical features due to the risk of malignancy, familial involvement, and need to monitor for progressive systemic manifestations. As this is a rare disease, it can often be missed due to low degree of suspicion and the ability of the jaw tumor to mimic a metastatic deposit. The most common presentation of primary hyperparathyroidism (PHPT) is asymptomatic hypercalcemia, which is incidentally detected in 80% of the patients . PHPT waThe skeletal manifestations are mainly due to reduced bone mineral density (BMD), which is more remarkable in the cortical bones such as forearm and hip than in the trabecular bones. The risk of hip fractures due to primary hyperparathyroidism has contrasting evidence. A large population-based cohort study carried out in Sweden with 19 years of follow-up revealed an increased risk of hip fractures in men but not in women . AnotherPHPT occurs mainly due to benign parathyroid adenoma in 80%, while multiglandular parathyroid hyperplasia contributing to 15–20% and parathyroid carcinoma to 0.5% . The majHPT-JT is an autosomal dominant syndrome with incomplete penetrance. The patients present with parathyroid tumors, ossifying fibromas of jaw, and a variety of renal and uterine tumors , 9. ThisWe report a case of a young female presented with recurrent low trauma femur fractures, with a significant past history of Wilms' tumor and maxillary tumor, who was ultimately diagnosed to have HPT-JT syndrome.A 22-year-old Sri Lankan woman was referred from the orthopedics unit to the endocrinology unit, National Hospital of Sri Lanka, for further evaluation of recurrent femur fractures. She first experienced a right sided subtrochanteric fracture following a low trauma injury at the age of 20 years , for whiShe had an interesting past medical history. She was diagnosed to have a left side nephroblastoma at the age of 8 years, for which she underwent left side radical nephrectomy followed by chemotherapy. Thereafter, at the age of 14 years, she developed a right sided facial swelling and was found to have a maxillary tumor which was described during the surgery as a well-circumscribed mass lesion in the left maxilla, eroding the anterior wall of maxilla and lateral nasal wall. She underwent partial maxillectomy for this, and histology revealed features suggestive of fibrous dysplasia.She did not have a history of chronic steroid use, hypogonadism, hyperthyroidism, rheumatoid arthritis, and exposure to cigarette smoking to suggest secondary causes for osteoporosis. She had neither any features of malabsorption nor inadequate exposure to sunlight to suggest a vitamin D deficiency causing these fractures. The histology of bone biopsies was negative for metastatic deposits.She did not have a family history of hyperparathyroidism, renal, or uterine tumors. There was no past history or family history suggestive of MEN 1. Her general and systemic examinations were normal apart from the healing fracture sites.Her investigation findings are summarized in Her investigations revealed high total calcium and urinary calcium excretion, with low serum phosphate level. She was found to have a vitamin D deficiency, which was corrected by vitamin D replacement. As high calcium and low phosphate were suggestive of a primary hyperparathyroidism, intact parathyroid hormone (PTH) was done after correcting vitamin D level, which was found to be very high at 1025 pg/ml (10–65 pg/ml), confirming the diagnosis of hyperparathyroidism.Z score of −5.6, suggestive of active chronic hyperparathyroidism, leading to bone resorption and reduced bone mineral density with preferential involvement of cortical bone and increased risk of fracture [Her left forearm dexa scan revealed osteoporosis with a fracture .Three-dimensional computed tomography (CT) of neck was carried out for localization, which revealed a 2.5 × 2.9 cm well-defined rounded mass with avid contrast enhancement posteroinferior to the left lobe of the thyroid gland, suggestive of a parathyroid mass . NuclearAt this point, due to the constellation of nephroblastoma, maxillary tumor, and primary hyperparathyroidism with a parathyroid mass, a possibility of hyperparathyroidism jaw tumor syndrome was suspected although a suggestive family history was not there. Therefore, the genetic studies were carried out via target region capture followed by next generation sequencing, using a specific indel detection tool, which revealed a positive CDC73 mutation with a large deletion of exon 1–17. This mutation was verified by reverse transcription polymerase chain reaction (RT-PCR), where exons 1, 10, and 17 were selected for verification. This confirmed the diagnosis of hyperparathyroidism jaw tumor syndrome . MEN 1 aThe patient underwent subtotal thyroidectomy with parathyroidectomy with removal of left lower parathyroid mass, which was a smooth, round, well-demarcated mass , and theImmediate postoperative PTH level was reduced by more than 50%–76 pg/ml, suggesting successful removal of the parathyroid mass. The histology revealed encapsulated parathyroid adenoma composed of a proliferation of polygonal cells separated by fibrovascular septa, with round to ovoid nuclei with dispersed chromatin and inconspicuous nucleoli and moderate eosinophilic cytoplasm. Mitoses were rare, and there were also cystic areas. There was no evidence of vascular invasion, necrosis, or invasion into soft tissues of thyroid gland. These features were compatible with a parathyroid adenoma.Currently, the patient is on calcium and calcitriol treatment and being followed up with serum calcium and phosphate levels with monitoring of urinary calcium excretion. Her imaging of the abdomen did not reveal a recurrence of nephroblastoma or uterine tumors. Biochemical screening of family members was negative, and genetic screening is underway.This case depicts a rare presentation of a rare disease. The typical initial presentation of individuals with HPT-JT is reported to be jaw and teeth deformities as a result of ossifying fibromas of mandible or maxilla . In contOssifying fibromas of the jaw, which this patient developed as the second manifestation of the disease at the age of 14 years, is a well-described manifestation of HPT-JT syndrome, which is shown to occur in 50% of these patients, usually in the adolescent age , 11. TheThe primary manifestation of hyperparathyroidism in this patient was bilateral low trauma femur fractures, which could have been contributed by the coexisting vitamin D deficiency. Vitamin D deficiency is known to be more prevalent among patients with primary hyperparathyroidism, due to multiple possible underlying mechanisms, such as increased conversion of 25-hydroxy vitamin D to 1,25-dihydroxy vitamin D and increased hepatic inactivation of 25-hydroxy vitamin D level . TherefoWith regard to the types of CDC73 mutations reported in the literature, the majority of CDC73 mutations are frameshift, nonsense, and missense variants, as well as small deletions and insertions –16. In fOur patient's atypical sequence of clinical manifestations with initial development being a nephroblastoma prompted the clinicians to suspect metastatic deposits at each of subsequent developments of maxillary tumors and femur fractures, deviating them from considering this familial syndrome for several years, which contributed to a delay in diagnosis of the condition.HPT-JT syndrome, although rare, is an important diagnosis to consider, especially in young patients presenting with classical constellation of clinical manifestations of parathyroid tumor, maxillary or mandibular tumors, and renal or uterine tumors, due to its risk of malignancy and importance of family screening. It needs close monitoring for new developments. The above case highlights an atypical presentation of this rare disease, which presented with nephroblastoma as the first clinical manifestations. Furthermore, this case is significant as the patient carries a rare type of mutation of a whole-gene deletion from exons 1–17 of CDC73 gene. To the best of our knowledge, this is the first genetically confirmed HPT-JT syndrome case reported from Sri Lanka.


IntroductionIntrauterine contraceptive devices (IUCD) are a commonly used, reversible, contraceptive method. Complications from insertion rarely include migration into the bladder. We report on two cases of intravesical migrated IUCD and present an algorithm for management based on recently published data.Materials and MethodsThe case records of two patients who underwent surgical procedures for migrated IUCD into the bladder were reviewed. A Pubmed search was performed to identify similar studies. A total of 25 papers met the criteria for inclusion.ResultsBoth cases were managed with laparotomy and partial cystectomy. A review of literature suggests recently reported cases of IUCD migration are rising, with most cases having been reported in the last decade. Bladder calculus developing over the migrated IUCD is the most common presentation. Most cases have been managed using endourological techniques. A small number of cases have required open vesicolithotomy or laparoscopic surgery. Rarely, laparotomy has been required. DiscussionIUCD migration into the bladder remains rare, however, recently the number of reported cases has risen. A thorough physical examination and radiological evaluation are warranted. Management is surgical in all cases. Most cases can be managed with endourological techniques. A treatment algorithm has been suggested in this paper based on recent data.ConclusionWith the rising use of contraception worldwide, the incidence of IUCD migration is possibly going to increase. Treating doctors need to be aware of the possible complications that may arise from a migrated IUCD, including bladder calculi. Intrauterine contraceptive devices (IUCD) are a popular method of contraception used by approximately 14.3% of women worldwide . MigratiIn this paper, we examine two cases of spontaneous IUCD migration into the bladder and then review the literature on the management of this rare complication.The case records of two patients who had bladder involvement of migrated IUCD were studied. For a literature review, a search of terms, ‘Intrauterine contraceptive device migration,’ ‘Urinary tract complications of intrauterine contraceptive devices,’ ‘Intrauterine contraceptive device migration in urinary bladder,’ ‘Intravesical intrauterine device migration,’ and ‘Urinary bladder intrauterine contraceptive device,’ were performed in Pubmed. Relevant studies in the English language were perused and studied and data collected.Case 1This is a case of a 40-year-old female patient who was referred with a lost IUCD. On presentation, there was no fever, no urinary symptoms, and no menstrual irregularities. A physical examination was normal. An X-ray revealed the IUCD (Copper-T) to be displaced outside the area of the uterus but within the pelvis. Ultrasonography (USG) performed confirmed that the uterine cavity was empty and showed an echogenic shadow suggestive of an IUCD on the right-side of the uterus. The kidneys, ureters, and bladder were normal. The patient was taken up for laparoscopic removal of the IUCD. During surgery, the IUCD was seen in the right parametrium, densely covered with adhesions. On mobilizing the IUCD, the IUCD was grasped but did not come out. A limb was found embedded in the right, lateral bladder wall. An urgent urological consult was called for. The procedure was converted into a laparotomy and the bladder mobilized on the right side. The IUCD limb was then isolated and the bladder wall marked with electrocautery, which was deepened using sharp dissection until the mucosa. The bladder wall and the IUCD were removed in total and the bladder wall repaired in three layers with absorbable sutures. A Foley catheter along with an intra-peritoneal drain was left in situ. The patient made an uneventful recovery.Case 2A 35-year-old, para 3 patient presented with dysuria, frequency, and urgency. On history, she revealed that she had had an IUCD insertion around a year prior to the presentation but had forgotten about it. At the time of presentation, she was amenorrhoeic for three months, and a pregnancy test was positive. A USG performed showed a bladder calculus of about 1 cm in size. She was taken up for cystoscopic removal of the calculus. However, the calculus was found to be adherent to the bladder wall, and on gentle traction, revealed a limb suggestive of an IUCD limb Figure . An elecThe details of the patients are noted in Table Studies which have described IUCD devices that have perforated into the bladder have been mentioned in Table IUCD is a widely accepted method of contraception. It is easily inserted, is reversible by removal, and causes few side effects . The comIUCD’s can perforate the uterus and then migrate into the pelvic or abdominal spaces. IUCD perforations have been divided into four types according to the anatomical spaces affected. The first compartment is the uterine cavity (type 1), the second is when the IUCD is confined to the myometrium (type 2) and the third compartment is when the peritoneal cavity is breached (type 3). When an IUCD penetrates the surrounding viscera, the perforation is type 4 . A uterine perforation may be primary or secondary. A primary perforation occurs at the time of insertion, whereas a secondary perforation occurs after a delay, probably due to pressure necrosis and inflammation of the uterine wall. , 28, 29IUCD migration may follow uterine perforation. It is a rare complication, occurring between 1.2 - 1.6 per 1,000 insertions  MechanisIUCD migration into the bladder is a rare complication and most commonly occurs between two and 10 years after implantation. In the first case of this series, the migration was detected three years after insertion, and in the second case, migration was detected after 12 months.After being in the bladder for a long time, encrustations form over the limbs of the IUCD which can then form a vesical calculus . Rarely,The initial approach to surgery in the first case was laparoscopic. However, due to dense adhesions between the IUCD and the surrounding tissue including the bladder, conversion to laparotomy was required. A partial cystectomy was needed in this patient. Shin et al demonstrated the use of laparoscopic approach alone to manage an embedded IUCD Sharma et al performed a cystoscopic retrieval of an intravesical IUCD . Sano etOf the twenty-six papers that have been cited in Table Based on the published data, an algorithm is suggested for the management of patients with migrated IUCD’s that may involve the urinary tract Figure .Most IUCD migrations occur at the time of insertion, and proper training of healthcare workers is imperative to prevent complications. Although rare, IUCD migration is a complication with high morbidity. IUCD migration into the bladder is a debilitating condition for the patient and warrants a multi-disciplinary approach with the use of imaging techniques and cystoscopy to locate the IUCD. Proper patient preparation is vital to a successful outcome. With an increasing number of women worldwide adopting some form of contraception, including IUCD, the incidence of migrated IUCD’s is going to rise in the future, and gynecologists, surgeons, and urologists need to be aware of this complication.


Esophageal atresia is one of the most common pediatric surgical conditions with a high mortality in developing countries. The esophagus and trachea have a complex embryologic development leading to a wide spectrum of esophageal atresia anomalies. Long upper esophageal pouch with distal tracheoesophageal fistula is one of these rare varieties. Only a handful of case reports exist on the subject thus far. Most reports are from low income countries.A neonate referred to our hospital after 2 weeks of life for excessive secretions, persistent respiratory distress and vomiting. Diagnosis of esophageal atresia was overlooked because nasogastric tube was advanced a considerable length. After repeat x-ray and esophagogram, we confirmed the diagnosis of type C esophageal atresia with a long upper esophageal pouch. Due to the dilemma in diagnosis, surgery was delayed after the 3rd week of life. Luckily the child survived even with extremely delayed intervention.Esophageal atresia can have an uncommon presentation which causes confusion in the diagnosis. Physicians need to be aware of its existence to avoid unnecessary delays in the management. Fortunately, this variant generally seems to have a good prognosis. There are 5 major types as described by Gross . Type C There is a very rare subtype of type-C EA first reported in 1960 45]. In this variant the level of the upper esophageal pouch is lower than usual, sometimes reaching as far down as the diaphragm 5. In thi. EsophagThere are only a handful of case reports entailing this rare phenomenon worldwide. We recently came across a similar case in our institution causing a diagnostic dilemma. The surgery was delayed to the 22nd day of life with a surprisingly good outcome. Herein we describe our encounter and discuss relevant literature on the subject. The work has been reported in line with the SCARE criteria .2A female neonate was referred to our center from the rural parts of Ethiopia. The mother had an uneventful antenatal follow-up and no abnormality was detected on ultrasound. The child was born term by spontaneous vaginal delivery. Subsequently she went home and was being breastfed when the mother noticed excessive salivation and vomiting after feeding.The mother took her to a local hospital where the child was admitted in the NICU for 2 weeks for the treatment of sepsis. She was kept NPO, put on intranasal oxygen, maintenance fluid and intravenous antibiotics. However, the respiratory distress and excessive salivation persisted. Referring physicians reported that feeding tube was able to pass down and the x-ray of the child was normal. They had attempted trophic feeding through the NG tube but the child persisted to have vomiting. Finally, she was referred to our center with a suspicion of Type E EA  on the 16th day of life.On presentation at our center, the child had excessive secretions coming out of the mouth and nose. She had some respiratory distress requiring intranasal oxygen but the other vital signs were stable. Auscultation of the chest revealed crepitations on the right lung, otherwise there was no cardiac murmur. An 8 French NG tube was able to pass more than 20 cm before it met resistance. X-ray taken showed tip of the feeding tube at T8 level. There was also consolidation of the right upper lobe and gas in the stomach .Fig. 1PlThereafter a barium swallow study was obtained which demonstrated a long dilated upper esophageal pouch reaching the lower chest . SurgeryThe child was operated onthe 22nd day of life by pediatric surgery fellows supervised by attendings. Bronchoscopy was not performed as it is not routinely practiced during EA surgery in our setting. The child was put in left lateral position after administration of general anesthesia. A right thoracotomy was performed through the 5th intercostal space and thoracic cavity entered through a muscle sparing approach. We found the upper esophageal pouch redundant, overlapping the distal fistula . After lPostoperatively the child didn’t require mechanical ventilation. She had developed postoperative pneumonia requiring intravenous antibiotics and hospital stay of 2 weeks. However, anastomosis healed well and she tolerated full oral feeds. She was finally discharged in excellent condition.3Delays diagnosis of tracheoesophageal fistula is a common problem in developing countries, mostly reported from India. It is associated with higher mortality due to malnutrition and pneumonitis . Thus faWhy the upper pouch is markedly elongated in this rare variant is unknown . Older rFrom our literature review we found only 9 previous reports describing this anomaly . Half ofIntraoperatively, there was no gap between the esophageal ends as long proximal esophagus overlapped with the distal one. As such, the surgery was described as being simple by most. After division of the fistula, different approaches were used to anastomose esophagus. These included side to side anastomosis and end to end anastomosis with or without resection of proximal redundant esophagus. Except for 2 children, all had favorable outcomes.In conclusion, physicians need to be aware of this rare variant of EA –TEF with an unusual presentation. Having a high index of suspicion will avoid delays in diagnosis and management. Overall, reported cases show a good survival. Our child was extremely fortunate as she survived her condition even with extremely delayed intervention.We declare that there is no conflict of interests.This study did not receive funding.Exemption from ethical approval was obtained from the department of surgery ethics and research committee at Addis Ababa University college of health sciences.Written informed consent was obtained from the patient's parent/guardian for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.SN and HW collected the data and drafted the manuscript. HGW was involved in the conception of the case report and supervised other authors. All authors read and approved the final manuscript.Registry not required as this is not a first-in-man case report.Dr. Samuel Negash.Division of Pediatric surgery.Addis Ababa University, Ethiopia.Not commissioned, externally peer-reviewed.


Peripheral artery mycotic aneurysms are rare occurrences. In this case, we review a 52-year-old lady with poorly controlled diabetes who developed a spontaneous left superficial artery mycotic aneurysm. She underwent excision and subsequent extra-anatomic bypass with a great saphenous vein graft. She had full functional recovery after a short period of rehabilitation. The term mycotic aneurysm, first coined by Osler in 1885, refers to localized, irreversible arterial dilatation due to destruction of the vessel wall by infection .In this case, we reviewed a 52-year-old lady with poorly controlled diabetes who developed a spontaneous left superficial femoral artery (SFA) mycotic aneurysm.A 52-year-old lady with hypertension, diabetes, and asthma presented with a one-week history of left groin pain. The pain started insidiously and was localized to the left groin. She was otherwise well and asymptomatic prior to this. There was no history of preceding trauma. Of note, she had a dental extraction procedure two weeks prior to this. She was only on inhaled corticosteroids for her asthma, without any systemic corticosteroids.On examination, she was found to have a tender left upper thigh mass that measured about 4 × 4 cm. Other than this, her lower limb neurovascular examination did not reveal any abnormalities. She did not have any features of distal emboli or splinter hemorrhages.9/L as well as a raised C-reactive protein of 247.7 mg/L. Her diabetic control was also poor with a glycosylated hemoglobin level of 15.4%. Initial blood cultures were also taken which did not reveal any microbes.Initial blood investigations revealed a total white cell count of 18.6 × 10She underwent an urgent computed tomographic (CT) angiogram of the lower limbs which revealed a thin wall eccentric saccular pseudoaneurysm arising from the proximal third of the left SFA measuring 2.0 × 2.6 × 3.0 cm with a gas containing intramuscular fluid collection seen. Mass effect from the collection and pseudoaneurysm was compressing the adjacent femoral vein causing central intraluminal filling defects, suspicious of deep venous thrombosis .A CT aortogram was done to rule out any other mycotic aneurysms. This revealed moderated intimal calcification of the infrarenal abdominal aorta and bilateral common iliac arteries. In addition to this, a wedge-shaped hypoenhancing area in the right renal inferior pole with adjacent fat stranding was noted, suggestive of possible pyelonephritis. An echocardiogram was also done, which did not reveal any vegetations.Urinalysis was performed but no microbes were detected. She was started on intravenous augmentin after the blood cultures were taken.She was planned for and underwent excision of the left SFA mycotic pseudoaneurysm with above knee femoropopliteal bypass with a superficial femoral vein thrombectomy. We made 2 separate incisions—a longitudinal groin incision to obtain proximal control and a longitudinal above knee incision to obtain distal control and access the aneurysm. Intraoperatively, she was found to have a 3 × 3 cm intramuscular pseudoaneurysm with a surrounding abscess cavity containing pus . During Extra-anatomic bypass with the great saphenous vein was to avoid the infected field and allow for partial closure of the wound without exposing the graft .Intraoperative blood cultures were taken from the pseudoaneurysm yielded pan-sensitive Escherichia Coli. Tissue cultures of the aneurysm also revealed the same organism.Postoperatively, she was monitored in the high dependency ward for a few days and then subsequently transferred to the general ward. Antibiotics were tailored based on the cultures and she completed a six-week course of intravenous cefazolin. Regular dressing change was performed for her wound initially, and this was then converted to negative pressure wound therapy. With this, her wound was healing well . FunctioMycotic pseudoaneurysms of the peripheral arteries are rare and can occur as a result of localized infection or systemic sepsis . With thIn a study of 57 patients with infected pseudoaneurysms [In a series of 26 patients by Brossier , they reThe aim of surgical management of infected pseudoaneurysms is similar to management of infected vascular grafts, such that all infected or necrotic tissue should be adequately debrided and resultant ischemia dealt with. In the case of our patient, given that she did not have existing peripheral vascular disease, ligation without revascularization would have resulted in a debilitating state for her. In an American study by Benjamin et al. , superfi


Gastric infarction is a rare condition often associated with high mortality due to a delay in diagnosis. The stomach which has a rich supply of blood is a rare site for such a condition. Gastric infarction has a long list of etiological factors. We report a case of a patient who was managed successfully following gastric infarction from gastric dilatation. An 18-year-old female student presented with a three-day history of abdominal pain associated with abdominal distension of two days. The abdomen was distended with generalized tenderness, rebound tenderness, and guarding. Bowel sounds were absent. Digital rectal examination was unremarkable, and a pregnancy test was negative. Biochemical tests were all normal. Intraoperatively, two litres of serosanguinous fluid was suctioned from the abdomen. About 300 mL of pus was suctioned from the pelvis. The gangrenous portion was resected, and repair was done in two layers using Conell and Lambert suture techniques. Acute gastric necrosis is a rare surgical condition that requires a high index of suspicion and prompts aggressive resuscitation and surgical intervention to obviate the high mortality rate associated with the condition. Acute gastric gangrene is rare due to the rich and anastomotic nature of blood supply to the stomach , 2. OnlySeveral possible causes have been assigned to gastric gangrene. Notable among these is herniation of the stomach through the diaphragm, gastric volvulus, acute necrotizing gastritis, complications after abdominal surgery, anorexia nervosa and bulimia, trauma, exposure to caustic materials, diabetes mellitus, and acute massive gastric dilatation from binge eating or pathological aerophagia –10. The An 18-year-old female student presented at the Accident and Emergency unit at the Komfo Anokye Teaching Hospital (KATH) with a three-day history of abdominal pain associated with abdominal distension of two days. The pain which was sudden in onset started four hours after a heavy evening meal. The pain was initially localized to the epigastrium. It had no known aggravating or relieving factors. The pain progressively worsened and became generalized the day after onset. It was associated with abdominal distension and absolute constipation. She was seen at a peripheral hospital and managed for a day with intravenous fluids, antibiotics, and subsequently referred for further management due to worsening of her condition. There was no history of fever, vomiting, had no history of peptic ulcer disease, and was not on NSAIDs or steroids. She had no history of alcohol use.2 was 96% on room air.On examination, she was conscious and alert, not pale, anicteric, well hydrated, and had a temperature of 37.8°C. Her pulse rate was 110 beats per minute, regular, and good volume. Extremities were warm, and blood pressure was 130/80 mmHg. Heart sounds were normal with no murmurs. Respiratory rate was 22 cycles per minute. The chest was clinically clear, and SPO3/UL), and neutrophils (90.8%). Biochemical tests were all normal , WBC . There were low levels of albumin (25.4 g/L), and the bun to creatinine ratio was above the reference range (36.7)  levels were high (83.4 e (36.7) .Figures The section of gastric showed transmural necrosis with congested blood vessels. There was no atypia .Acute gastric necrosis is a rare occurrence and mostly diagnosed intraoperatively. Because of its rarity, diagnosis is mostly delayed and virtually made only at laparotomy , 12. FinIn the event of massive gastric dilatation, a resultant pressure of about 14 mmHg is sufficient enough to cause venous insufficiency leading to ischaemia . IntragaCirculatory collapse has been a key feature of this condition in literature or several reported cases , 17, 18.The mortality associated with acute gastric necrosis ranges from 50 to 80 percent which underscores the severity or life-threatening nature of the condition . SignifiAcute gastric necrosis is a rare surgical condition that requires a high index of suspicion and prompts aggressive resuscitation and surgical intervention to obviate the high mortality rate associated with the condition.


Extraskeletal Ewing’s sarcoma is a rarity, with a renal primary in an adult, being even rarer. There is no consensus on the optimal imaging modality, as well as best therapeutic option, making them an enigma for clinicians.We report the case of a 34-year-old lady, a known case of invasive lobular carcinoma of the left breast , having completed treatment in 2017, wherein, on an ultrasound evaluation for left flank pain, was incidentally found to have a left renal mass. A CT scan corroborated with the ultrasound, with an additional Level 1, left renal vein thrombus. She underwent an open left radical nephrectomy with renal vein thrombectomy. Histopathology of the resected tumor revealed features of Ewing’s sarcoma of the kidney, confirmed by Fluorescent In Situ Hybridisation (FISH) and Immunohistochemistry (IHC).Primary renal Ewing’s sarcoma in an adult is a rare occurrence, with no characteristic imaging features, and no universally accepted guideline based management protocols. Akin with standard Ewings sarcoma treatment strategies, a margin negative- radical nephrectomy with adjuvant chemotherapy, seems the most apt treatment strategy. Ewing’s sarcoma constitutes a group of malignant round small cell tumours of primitive neuroectodermal origin with the most common sites being bone and soft tissues, particularly in children and young adults. Extraskeletal Ewing’s Sarcoma is a rarity, with a renal primary in an adult, being even more sparse.Literature suggests that Ewing’s sarcoma of the kidney (ESK) is aggressive and has a propensity for rapid clinical progression and metastasis. It is characterised by a balanced chromosomal translocation t11;22) which results in the production of the EWS/Friend leukaemia virus integration 1 (FLI-1) fusion gene. The treatment of choice remains radical nephrectomy followed by combination chemotherapy with or without adjuvant radiotherapy [1;22.A CT scan abdomen confirmed the ultrasound findings, with an additional Level 1, left renal vein thrombus . CT thorWith a working diagnosis of left renal cell carcinoma, she underwent an open left radical nephrectomy with left renal vein thrombectomy and retroperitoneal lymph node dissection.The procedure and post-operative course was uneventful, and she was discharged on day 5, without event.The post-operative histopathology report, however, confirmed the diagnosis of an Unifocal Ewing’s sarcoma of greatest dimension, 16 cm with lymphovascular (LVI) and perineural invasion (PNI) present. Resected margins were free of tumour. 2 of 9 perihilar lymph nodes were involved by tumour and Pre, para and interaortocaval lymph nodes were negative for malignancy. FISH was positive for Ewing sarcoma breakpoint region 1 (EWSR1) . On ImmuA bone scan done subsequently revealed no skeletal metastasis. The final histopathology report was discussed in our multidisciplinary tumour board meeting, and she was started on adjuvant chemotherapy, Vincristine + Adriamycin + Cyclophosphamide (VAC) regimen alternating with Ifosphamide and Etoposide (IE).Ewing’s sarcoma constitutes a group of malignant round small cell tumours of primitive neuroectodermal origin with the most common sites being bone and soft tissues, particularly in children and young adults. The average age at diagnosis is usually 30.4 years with a male to female ratio of 60:1.Primary renal Ewings sarcoma is a rarity, with clinical and radiological features mimicking other common renal tumours. An accurate diagnosis is usually possible post-operatively with a combination of FISH and immunochemistry . A charaRenal Ewing’s behaves more aggressively than at other sites. It has strong propensity for local recurrence, early metastasis to lymph nodes, lungs liver and bone . Due to In general, Ewing’s sarcoma treatment uses a multimodality approach with debulking surgery , chemotherapy with or without radiotherapy to the renal bed as the possible options. Even after multimodality treatment, prognosis remains poor with an overall 5-year disease free survival of 45–55% [Adjuvant radiotherapy can be offered in case of incomplete resection, positive resection margins, or recurrence, which was not so in our case.Follow-ups with laboratory and imaging tests are essential to assess recurrence and metastasis.Primary renal Ewing’s sarcoma is an aggressive tumour, particularly in adults, with a high propensity of metastases. With no specific imaging characteristics to delineate the same, a multimodality treatment approach mandating a margin negative- radical nephrectomy with adjuvant combination chemotherapy is recommended, in sync with the management of Ewing’s tumors at other body sites. Literature reports a poor overall prognosis, despite all forms of therapy, for this enigmatic condition.Click here for additional data file.


Sistrurus catenatus) was presented for evaluation of a subcutaneous mass affecting the ventral scales. The mass was diagnosed as a sarcoma via punch biopsy with no evidence of metastasis on diagnostic imaging. Surgical margins of 1-2 cm were planned to achieve complete excision of the neoplasm. A technique for tissue expansion was employed due to concerns regarding the ability to close the surgical site without excess tension or dehiscence. Two 27 mm diameter × 5 mm hygroscopic self-inflating tissue expanders were placed subcutaneously under the lateral scales adjacent to the mass. Maximum skin expansion occurred over a four-week period, and no direct negative effects were noted. Excision of the primary mass was performed routinely five weeks after implant placement. Primary closure of the defect was achieved with minimal tension by incorporating the expanded skin. While the surgery was successful with no evidence of metastasis, the snake died of sepsis two weeks postoperatively. This is the first report of the use of self-inflating hygroscopic tissue expanders to help close a surgical defect in a reptile.A 0.34 kg adult female Massasauga rattlesnake ( Reports of skin stretching and tissue expansion first appeared in the veterinary medical literature during the 1980s. The initial publications described the clinical utility of these methods in dogs and horses , methodsTissue expansion in both human and veterinary patients was historically performed via subcutaneous placement of a silicone balloon that was periodically expanded by the infusion of air or saline , 23. ThiSistrurus catenatus) presented in August 2017 for evaluation of a ventral scale mass. The snake was wild-caught as a juvenile and had an estimated hatch date of 2004. After capture, it was kept at a local wildlife nature center for six years and then donated to the North Carolina Zoo in 2011, where it had been housed since that time. The snake's medical history included hypopyon in the left eye noted in 2012 that resolved with antibiotic therapy and mild conspecific trauma in 2015 that healed without intervention.A 0.34 kg adult female Massasauga rattlesnake . For reference, the snake's ventral scale width and body height in the region of the mass measured 3.5 cm and 3.7 cm, respectively. The mass could be isolated from underlying internal organs and adjacent ribs using digital palpation. However, direct penetration into the coelomic cavity could not be ruled out based solely on palpation.Anesthesia for biopsy and staging was performed approximately three weeks later. The cranial half of the snake was manually restrained via a plastic tube using standard safety protocols for the handling of venomous snakes at the North Carolina Zoo. It was then lightly anesthetized with alfaxalone  3 mg/kg IV. Whole body radiographs including orthogonal views revealed a cutaneous ovoid soft-tissue mass containing an irregular mineral opacity. Metastases were not observed. Ultrasonographic exam showed that the mass had grown since the previous exam and was now 3.2 × 1.8 × 1.2 cm. The remainder of the coelomic ultrasound revealed multiple ovarian follicles and no discernable metastases. Biopsies were performed to further characterize the mass. An intradermal ring block was administered around the mass using lidocaine  2.2 mg/kg diluted to 0.2 mL with saline. The snake received meloxicam  0.2 mg/kg IM. A 2 mm punch biopsy was used to produce two biopsy samples. Touch impressions of the biopsies were obtained prior to fixation in formalin. Cytology was consistent with a soft tissue sarcoma.μL with 15% heterophils, 39% lymphocytes, and 46% azurophils. Results were comparable to five routine samples previously collected from this individual between 2011 and 2015, aside from a slight increase in percent azurophils . This was attributed to chronic inflammation associated with the mass. Packed cell volume (PCV) was 44%, and platelets appeared adequate on the blood smear. Plasma chemistry was performed using a point-of-care analyzer  and showed a hypercalcemia of >4 mmol/L (>16 mg/dL) that was consistent with active follicle production. Creatine kinase (CK) was also elevated at 1151 U/L. The CK elevation may have resulted from anesthetic injections or local muscle disruption due to the mass. The snake recovered routinely from the procedure and was returned to its normal enclosure. The biopsy sites appeared to be healing appropriately at recheck nine days later. Initial biopsy results reported an ulcerated spindle cell sarcoma with myxomatous stroma and a small amount of osteoid and mineralized material suggestive of osteosarcoma. No mitotic figures were noted, but neoplastic cells were observed infiltrating into the adjacent stroma.Blood was collected from the ventral tail vein and analyzed for complete blood count and plasma chemistry. Blood smear evaluation revealed a white cell estimate of 6970 cells/Based on the diagnosis of a sarcoma with no concurrent evidence of metastases and apparently good systemic health, surgical excision was planned. Currently, there are no studies critically evaluating the typical biological behavior, management strategies, or long-term outcomes for the treatment of sarcomas in reptiles . WithoutImplant placement was performed eight weeks after initial presentation. Anesthesia was induced with alfaxalone 5 mg/kg IV in the ventral tail vein, and anesthesia was maintained via isoflurane 0.25-1.0%. Two additional IV boluses of alfaxalone, 2.7 mg/kg each, and one dose of ketamine  5.5 mg/kg IV were needed to maintain a surgical plane of anesthesia. Apnea developed and the snake was intubated with a 2.1 mm endotracheal tube to provide intermittent positive pressure ventilation. Lidocaine was administered subcutaneously at the incision site for local anesthesia. Additional treatments included meloxicam 0.3 mg/kg IM, lactated ringer's solution (LRS) 22 mL/kg SQ, and butorphanol  1 mg/kg IM. Butorphanol was initially chosen by the primary clinician as part of a multimodal analgesic plan. This opioid is reported to provide pain control in snakes only at high doses  and is tIn mammals, the implants begin to expand 2-4 days after placement and then show a constant rate of expansion up to 14 days. However, at room temperature, expansion does not begin until 14 days and is complete at 28 days . Though Implant removal and tumor excision surgery was performed 36 days after implant placement. Anesthesia was induced and maintained as previously outlined. To access the implants, the skin was incised along the previous incision line. A combination of blunt and sharp dissection was used to disrupt a thin tissue membrane that had formed surrounding the implants. The implants were easily removed with gentle traction. The associated subcutaneous tissues and skin appeared vital with no overt fibrosis of the expanded skin and surrounding tissues . When laPostoperative care included meloxicam 0.3 mg/kg IM and LRS 20 mL/kg SQ daily for four doses. The remaining loose skin at the implant sites over the left lateral body wall was notably decreased by two days postoperatively. The surgical site appeared to be healing well at multiple veterinary checks during the next two weeks, and the snake's demeanor remained bright. However, it was found dead by keepers 14 days postoperatively. The animal had refused a meal several days prior, but otherwise had not shown clear premonitory symptoms.Gross postmortem examination revealed a mild amount of translucent coelomic fluid and adequate internal fat stores. The lungs and kidney contained numerous pinpoint to 0.02 cm diameter, discrete white masses, and a larger mass was identified within the cardiac ventricular lumen measuring 0.03 × 0.05 cm. The liver was irregular with multifocal areas of parenchymal collapse. The surgical site contained some bruising but otherwise appeared appropriate for this stage of healing.Metastatic neoplasia was suspected based on gross postmortem exam. However, histology revealed that the intracardiac heart mass corresponded to an area of severe, fibrinous endocarditis with mixed bacteria (vegetative endocarditis). The masses within the liver and lungs, as well as smaller foci in the kidneys, were revealed to be foci of necrosis and heterophilic inflammation with intralesional mixed bacteria. These foci were frequently associated with intravascular fibrin thrombi consistent with embolic sepsis. Histology of the surgical closure site showed marked inflammation and numerous reactive changes, but no overt neoplastic cells or bacteria were identified. The precise cause of the bacterial endocarditis with systemic emboli is unknown. Infection may have entered via the biopsy sites, implant or mass excision sites, or via the ulceration on the surface of the tumor itself. Alternatively, infection could have resulted from an entirely unrelated previous subclinical disease. Immunosuppression induced by the tumor, implant placement, or the stress of handling may have increased susceptibility to infection.The postoperative mortality provided an opportunity for histologic examination of the expanded skin two weeks after implant removal. The expanded donor site contained mild, subacute hemorrhage and edema with minimal inflammatory infiltrates.To the authors' knowledge, this is the first description of tissue expansion for preoperative surgical planning in a reptile and the first clinical use of STEs in a nondomestic animal.in vivo use in mammals, the manufacturer reports that implants held at room temperature typically begin expanding at 2 weeks [In the case reported here, the expansion period was slightly protracted, as might be expected for a reptile. In contrast to  2 weeks . TherefoThe manufacturer recommends that the implants are left in for two weeks in preparation for surgery, though both shorter (13 days) and longer periods (42 days) have been reported in dogs . ComplicThe success of tissue expansion via STEs in this patient is attributed in part to the innate compliance of ophidian skin, which has evolved to accommodate locomotion, macrophagy, and egg production. The mechanical properties of snake skin vary widely between locations on the body and among species , 38. ForThough this individual died of sepsis seven weeks after implant placement, the implants were considered an unlikely source of direct infection based on the healthy appearance of the implant site at removal, lack of clinical signs during the expansion period, and lack of inflammation in this area at time of death. Additionally, strict aseptic technique and perioperative antibiotics were administered. It is suspected that the tumor surface, biopsy sites, or excision sites represent the most likely sources of bacterial entry and subsequent endocarditis. The authors feel that STEs have strong potential as a viable tool for resurfacing surgical defects in snakes despite the two-week postoperative mortality in this case.Finally, this case represents the first report of presumed-extraskeletal osteosarcoma in a crotalid. Primary skeletal neoplasms such as fibrosarcoma and chondrosarcoma are prevalent in snakes , 43. HowThis paper highlights the utility of STEs for the excision of a locally invasive tumor in a rattlesnake. Specifically, preoperative tissue expansion permitted wide surgical margins and primary closure of the resulting defect. The side effects of STEs reported in domestic animals were minimal or not observed in this case. It is possible that stress and immunosuppression prompted by implant placement and postoperative care may have increased this animal's susceptibility to infection. Therefore, prognosis, feasibility of implant placement, patient demeanor, and immune status should be considered when choosing reptilian candidates for this novel technique.


Ameloblastoma is a benign locally invasive odontogenic tumor. It is the most common odontogenic tumor . Usually"Written informed consent was obtained from the patient guardians for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request”. It’s thRadiographically, ameloblastoma can appear as multilocular or unilocular radiolucency, most commonly it appears as a multiloculated radiolucency resulting in a honeycomb or as a soap bubble appearance. Roots of the teeth involved in the lesion may show varying degrees of resorption .The tumor is slow-growing and in its earliest stages, it might be asymptomatic and could be discovered incidentally. But when the tumor enlarges patients start to have symptoms like pain, facial deformity, and enlargement of the jaw-bones .This case report presents a case of a rare unicystic maxillary ameloblastoma in a 3.5-years old child. All work has been reported in line with the SCARE criteria .2A 3.5-year old girl, with no medical history, presented with swelling and pain on the left side of the face. Initially, two months back the swelling was asymptomatic, it was smaller in size, but it gradually enlarged to the present size and extended to the floor of the orbit, causing facial deformity and pain . There wOn extraoral examination, a diffused swelling over the left side of the face measures about 7 × 7 cm, involving the maxilla and the left eye. The skin overlying the swelling was stretched, the surface was smooth, and the skin color was normal. In palpation, it was hard and painful with no local rise in temperature. Intraoral examination revealed diffuse swelling in the left maxilla, extending from the midline to the posterior molars area and expanding laterally to involve the buccal vestibule and the cheek .Fig. 2PrComputed tomography revealed a very large well- defined radiolucent expansile unilocular lesion in the left maxilla with perforation to the floor of the orbit , Fig. 7.As the lesion was very extensive, the entire tumor has been removed using an extraoral incision (Weber Ferguson) , Fig. 9 3Ameloblastoma is a locally aggressive, epithelial odontogenic neoplasm, histologically, it resembles an enamel organ. It is non inductive which means that it fails to induce any formed calcified product such as enamel, dentin, or other materials .Ameloblastoma is classified into four types: multicystic, unicystic, peripheral, and desmoplastic. The most common form is the multicystic one. This case report discusses the unicystic type. The unicystic ameloblastoma was originally described by Robinson and Martinez in 1977. Unicystic ameloblastoma in comparison to multicystic ameloblastoma occurs mostly at a younger age with a range of 10.6–23.8 years [If a preliminary diagnosis of ameloblastoma is made, then CT imaging is highly recommended. CT imagining can demonstrate the anatomical extent of the tumor, therefore it can detect perforation of the outer cortex and invasion into surrounding soft tissues. Accordingly, if the soft tissue invasion is extensive then Magnetic Resonance Imaging (MRI) is recommended. In a post-surgical follow-up, CT examination is crucial .Incisional biopsies of large jaw cysts should be carefully examined for the presence of ameloblastomatous change, the enucleated specimens should be thoroughly sectioned to rule out mural or transmural extension. Unicystic ameloblastoma mostly is found as luminal and intraluminal forms .For Cystic ameloblastoma, treatment options can range from enucleation to resection, although they are more likely to recur if enucleated . In our 4This case of Unicystic Ameloblastoma was diagnosed carefully based on history and investigations. Ameloblastoma is more common in the mandible than in the maxilla, and in middle age than in children. Very rarely, we come across a case of ameloblastoma in the maxilla in a 3.5 years old child. Unicystic ameloblastoma is a tumor with a strong propensity for recurrence. Therefore, Adequate maxillectomy was performed with simultaneous reconstruction using buccal pad of fat with very acceptable results both functionally and esthetically.We wish to confirm that there are no known conflicts of interest associated with this publication and there has been no significant financial support for this work that could have influenced its outcome.No funding was received for this work.In accordance to declaration of Helsinki Patient guardians wrote informed consent for surgical intervention and for publication.“Written informed consent was obtained from the patient guardians for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request”.Dr Mohamed Touny: Writing the paper. First Surgery Assistant.Professor Mohamed El Sayed: Consultant in charge of the surgery.Primary surgeon. Postoperative follow up of the patient.Dr Nesreen Ibrahim: Data collection. Analysis of data. Patient follow up.Dr Zainab Al-Azzawi: Patient follow up. Data collection. Revision of manuscript.NA.Dr Mohamed Touny.Professor Mohamed El Sayed.Not commissioned, externally peer-reviewed.


Congenital pyloric atresia (CPA) is a rare gastrointestinal anomaly frequently associated with epidermolysis bullosa (EB). Although the complications of familial isolated CPA are minor, delays in diagnosis can increase the chances of morbidity.Three female infants born to a Japanese mother presented with CPA at birth. There was no consanguinity between the parents, and the spacing between pregnancies was 2 years in each case. All 3 siblings had a prenatal diagnosis of CPA owing to polyhydramnios and a dilated stomach, without dilatation of the rest of the gastrointestinal tract. All patients underwent reconstructive surgeries for establishing bowel continuity  soon after birth. Their postoperative courses were uneventful, and they grew up healthily, without any complications.Fetal ultrasonography is useful for diagnosing CPA prenatally. Successful prenatal diagnosis can lead to timely intervention after birth. Congenital pyloric atresia (CPA) is a rare gastrointestinal anomaly that accounts for less than 1% of all intestinal atresias . TherefoAnatomically, CPA can be classified into 3 subtypes: type A, pyloric membrane or web; type B, pylorus replaced by solid tissue; and type C, complete interruption with a gap between the stomach and duodenum , 5. AmonThe patients’ parents were Japanese, and there was no consanguinity between them. They had no family history of genetic disease. A summary of the 3 cases is shown in Table A healthy, 21-year-old G1P0 woman was referred to our hospital for preterm rupture of membranes at 32 weeks of gestation. An ultrasonography on admission revealed polyhydramnios ((amniotic fluid index [AFI]: 30 cm) and a remarkably dilated fetal stomach (66 × 31 mm)  Fig. a. PyloroThe second pregnancy, 2 years after Case 1, was complicated by threatened premature labor, and the mother was referred to our hospital at 31 weeks of gestation. Prenatal ultrasonography revealed polyhydramnios (AFI: 27 cm) and dilatation of the fetal stomach (50 × 27 mm). Although fetal magnetic resonance imaging (MRI) showed a dilated stomach, neither dilated intestines nor an associated anomaly was confirmed Fig. . A prenaLaparotomy confirmed type B CPA, and the atretic segment was 1 cm in length Fig. b. Bowel The third pregnancy, 2 years after Case 2, was again complicated by threatened premature labor. Prenatal ultrasonography showed polyhydramnios (AFI: 27 cm) and a dilated fetal stomach (45 × 16 mm) at 30 weeks of gestation. The third sibling, who was also female, was delivered by cesarean section at 36 weeks of gestation. Plain abdominal radiography and an upper gastrointestinal contrast study revealed complete obstruction of the pylorus, leading to the diagnosis of CPA.On laparotomy, type B CPA (atretic segment: 1 cm in length) was confirmed. In this case, gastroduodenostomy was performed in a side-to-side fashion without excision of the atretic segment. A nasojejunal tube was placed, and patency of the intestines was confirmed. Her postoperative course was uneventful. She achieved full oral intake 14 days postoperatively. She is now a healthy 6-year-old with normal growth and development.Congenital pyloric atresia (CPA) is an extremely rare gastrointestinal anomaly that accounts for less than 1% of all gastrointestinal atresias, with an incidence of 1 per 100,000 live births . In our Type B CPA is treated with either pyloroplasty (Heineke–Mikulicz or Finney) or gastroduodenostomy , with or without excision of the atretic segment , 5, 8. GITGA6, ITGB4, and PLEC) are known to cause EB with CPA, little is known about gene mutations in isolated CPA [CPA is frequently associated with other anomalies 43–66%), including epidermolysis bullosa (EB) 3–66%, in, 8. CPA ated CPA . In our Without prenatal diagnosis, accurate diagnosis after birth might be delayed because of non-bilious emesis. Complications resulting from delays in diagnosis (including aspiration pneumonia and gastric perforation) can be fatal, regardless of the excellent prognosis of isolated CPA , 9. TherWe herein report 3 cases of familial isolated CPA in siblings in which fetal ultrasonography was useful for diagnosing CPA prenatally. Prenatal diagnosis can lead to timely intervention after birth in such cases.


Nocardia paucivorans and provide a comprehensive review of intra-abdominal nocardiosis. The acquisition of abdominal nocardiosis is believed to occur via hematogenous spreading after pulmonary or percutaneous inoculation or possibly via direct abdominal inoculation. Cases of Nocardia peritonitis have been reported in patients on peritoneal dialysis. Accurate diagnosis of abdominal nocardiosis requires histological and/or microbiological examination of appropriate, radiologically or surgically obtained biopsy specimens. Malignancy may initially be suspected when the patient presents with an abdominal mass. Successful therapy usually includes either percutaneous or surgical abscess drainage plus prolonged combination antimicrobial therapy.Nocardiosis is primarily an opportunistic infection affecting immunosuppressed individuals, in whom it most commonly presents as pulmonary infection and sometimes cerebral abscesses. Isolated abdominal or retroperitoneal nocardiosis is rare. Here, we report the second case, to our knowledge, of isolated abdominal nocardiosis due to  Nocardia are gram-positive, aerobic, filamentous bacteria that most often cause pulmonary infection in immunosuppressed hosts, while isolated abdominal or retroperitoneal nocardiosis is rare [ is rare . In extr is rare ,3,4,5,6, is rare ,8.Nocardia paucivorans. We also provide a comprehensive review of the literature, summarizing the information on 39 previously published cases of intra-abdominal and retroperitoneal nocardiosis and 12 cases of peritoneal dialysis-related nocardia peritonitis [Nocardia paucivorans, a rare nocardial species.Here, we report the second case, to our knowledge, of isolated abdominal nocardiosis due to itonitis ,51,52,53An 81-year-old man with diffuse large B-cell lymphoma presented in January 2019 with abdominal pain and night sweats. In 2009, he initially presented with abdominal pain, weight loss, night sweats, and fever. Abdominal CT showed a mesenteric mass adjacent to the ileocecal area. Histopathological examination of a surgically obtained biopsy specimen showed the presence of CD20- and CD3-positive lymphoma cells, a perifocal lymphohistocytiary inflammatory infiltrate as well as necrotic lymphoma tissue. A bone marrow biopsy was free of lymphoma cells. Weekly rituximab was administered for 4 weeks, in combination with vincristin and bleomycin for the first 3 weeks. Rituximab plus cyclophosphamide, hydroxydaunorubicin, oncovin and prednisolone (R-CHOP) was administered for a total of 6 cycles until full remission was achieved in February 2010. After a successfully treated low-grade nodal relapse in 2017, the patient remained asymptomatic until February 2018 when persistent hypogammaglobulinemia was first noted and intravenous immune globulin was given at 4-week intervals. Since the last relapse in 2017, the patient had received no immunosuppressive therapy.3) were elevated. A chest X-ray showed no signs of pulmonary infection, and on CT scan of the head there were no signs of cerebral nocardiosis. Abdominal CT showed a heterogeneous mass in the right adrenal region  and the white blood cell count , ciprofloxacin and minocycline and resistant to amoxicillin/clavulanic acid. Nocardia paucivorans was identified by matrix-assisted laser desorption ionization time-of-flight mass spectrometry (MALDI-TOF MS). The mass spectra were acquired using the Microflex LT system and analyzed with the MALDI Biotyper Compass 4.1 software . The identification was confirmed by partial 16S rRNA gene sequencing as described previously [® MIC Test Strips  on Mueller Hinton agar +5% horse blood +20 mg/l ß-NAD , and the minimal inhibitory concentrations (MICs) were interpreted according to CLSI guidelines [Nocardia paucivorans in this patient could not be identified.A pigtail catheter was placed under CT-guidance for drainage of the abscess  and empi species . Cultureeviously . Antimicidelines . PCR andAntibiotic therapy was changed to intravenous trimethoprim-sulfamethoxazole and amikacin, and the patient clinically improved. The trough-level of amikacin was monitored and kept in the recommended range. However, the patient developed acute renal failure (eGFR decrease from 50 to 14 mL/min) and metabolic acidosis. This prompted the change of antimicrobial treatment to ceftriaxone and minocycline, and renal function recovered.Due to considerable residual abscess despite external drainage, surgical abscess drainage was performed and the patient further improved. He was discharged on oral minocycline and ciprofloxacin. On follow-up, six weeks postoperatively, he reported minimal abdominal pain, tolerated antibiotic therapy well, and blood inflammatory markers were normal. Antibiotic therapy was continued for a total of 6 months. On most recent follow-up, 6 months after antibiotics were discontinued, the patient was well, and abdominal ultrasound was unremarkable.Nocardia paucivorans. To identify patients in the published literature, several PubMed searches were performed. Search items included “nocardiosis”, “nocardia”, “paucivorans”, “abdominal”, “retroperitoneal”, “CAPD”, “peritonitis” and “infection”. Additional cases were identified through bibliographic review of retrieved publications. Abdominal nocardiosis was defined as clinical or radiological signs compatible with nocardiosis plus isolation of Nocardia species in at least one abdominal or retroperitoneal specimen regardless of the infection’s origin and extent of dissemination. Nocardia peritonitis was defined as clinical signs of peritonitis plus isolation of Nocardia species in ascites or peritoneal dialysate. N. paucivorans infection was defined as clinical or radiological signs compatible with nocardiosis plus isolation of N. paucivorans in an appropriate specimen, with clinical or radiological signs of infection. We limited our review to English and German language publications since 1966. The initial searches led to 93 publications being retrieved, of which 29 were excluded . Therefore, the present review includes 64 publications, including 49 cases of N. paucivorans infection, 39 cases of abdominal or retroperitoneal nocardiosis, 12 cases of nocardial peritonitis, plus our own patient with abdominal N. paucivorans infection.We reviewed the existing literature on abdominal nocardiosis and infections due to N. paucivorans. Third, we will concentrate on the characteristics of abdominal nocardiosis and Nocardia peritonitis.In this review of the literature, we will first discuss the presumed route of acquisition of nocardiosis based on previous case reports and reviews. Second, we will discuss previously reported infections with Nocardia are ubiquitously present in soil, water and decaying vegetation [Nocardia in immunosuppressed patients is followed by hematologic dissemination, eventually allowing infection at distant sites [Nocardia peritonitis in patients on peritoneal dialysis [getation . Infectigetation . In immugetation . With imgetation ,56,57. Pnt sites ,38. Addidialysis ,51,52,53N. paucivorans was first isolated in sputum in 2000 . To our In 18 of 49 cases 37%), no information was provided regarding the patients’ immune status. Of the remaining 31 cases, 11 patients (35%) were reported to be immunocompetent while 20 patients (65%) were overtly immunocompromised or had an underlying clinical condition. Thus, nocardiosis with N. paucivorans involves immunocompetent and immunocompromised patients, similar to infection with other Nocardia species [%, no infMicrobiologically documented lung involvement was reported in 29 of 49 cases 59%) of human N. paucivorans infection, while 19 cases 39%) involved the central nervous system (mainly brain abscesses). Additional cases include skin and muscle abscesses, bacteremia and endocarditis [9% involv% of humaThere are 39 cases of abdominal/retroperitoneal nocardiosis reported in the English and German language literature since 1966 ,41,42 T. In 16 oN. asteroides and N. farcinica (10 cases each) represent the most commonly isolated species. The route of Nocardia acquisition leading to abdominal or retroperitoneal infection remains unclear in most case reports. Some authors of published abdominal nocardiosis cases have speculated on the route of acquisition, which may have included inhalation of infected dust followed by hematogenous spread of Nocardia species , third-generation cephalosporins, extended-spectrum quinolones, carbapenemes, minocycline and linezolid [Nocardia species is a rare cause of peritonitis in patients on continuous ambulatory peritoneal dialysis (CAPD), with 12 published cases in the literature, which we summarize in Nocardia.Identifying and treating Nocardia peritonitis may be difficult for several reasons. First, the clinical presentation of Nocardia peritonitis typically is no different from ordinary bacterial peritonitis . Second,Nocardia species, cultures should be incubated for at least 10 days to avoid false-negative results [N. asteroides and N. farcinica are the most commonly isolated species in abdominal nocardiosis. N. paucivorans is a rare species leading to abdominal nocardiosis with only one previous case report, our patient thus representing the second published case. As in our patient, isolation of Nocardia species in the renal or adrenal region was the most common infection site amongst previous cases of abdominal or retroperitoneal nocardiosis. To date, there are no therapy guidelines for abdominal nocardiosis. In reported cases, successful therapy usually included either percutaneous or surgical abscess drainage plus prolonged combination antimicrobial therapy including TMP/SMX ranging from 3 to 12 months, as recommended for other forms of nocardiosis [Human nocardiosis is a rare opportunistic disease affecting primarily immunosuppressed adults. However, one third of patients have no immunocompromising condition ,7,63. Th results . N. asteardiosis . As demo


Being multiple and giant is even rarer.•The aim of this study is to report a case of unusual gastric outlet obstruction by two giant bezoars. Phytobezoar which is described as an undigested or incompletely digested food. It is an odd cause of gastric outlet obstruction (GOO). The aim of this study is to present and discuss a case of GOO caused by multiple giant bezoars.A 24-year-old female, presented with abdominal pain and vomiting (non-bilious) with negative past history. Examination and investigations revealed multiple giant bezoars requiring emergent surgical intervention. An exploratory laparotomy was conducted. Two giant bezoars were palpated in the stomach and removed through an anterior gastrotomy.Bezoars are regarded as rare benign causes of GOO. Bezoars can present with vomiting, nausea, and/or symptoms of GOO. Predisposing risk factors include delayed gastric emptying (as in case of diabetic mellitus) vagotomy, partial gastrectomy pyloroplasty, peptic ulcer disease, chronic gastritis, Crohn’s disease, and carcinoma of the gastrointestinal tract. The current case had phytobezoar without any known risk factor.GOO caused by phytobezoar can occur in patients without history of previous gastric surgery or diabetes mellitus. Urgent laparotomy may be indicated. Phytobezoar is described as an impacted indigested or incompletely digested vegetable and fruit fibers. Phytobezoar is an infrequent late complication of a previous gastrointestinal operation . Hypoaci1.1A 24-year-old female brought to the emergency department with abdominal pain and vomiting (non-bilious) for three day duration. The character of the pain was colicky in nature associated with nausea. She had no oral intake for two days because of pain. She reported history of mild dyspepsia, weight loss, and early satiety for which she used to take irregular anti-acid medications few months before presentation. The condition started to deteriorate in the last 3 days. There was negative past medical and past surgical history.1.2The patient was fully conscious, mildly dehydrated and have neither pallor nor jaundice. Heart rate was 96 beats/minute, regular with good volume, blood pressure was 100/70 mmHg and respiratory rate was 22 cycles/minutes. She had an evident uneven abdominal swelling with a centrally upturned umbilicus. Abdomen was soft on palpation, with a left hypochondrial intra-abdominal, firm, elliptical, smooth surface, mobile, not tender, not compressible, not pulsatile mass, extending to the epigastric region, measuring about 20 cm × 10 cm. Bowel sound was normal. Both rectum  and hernia orifices were empty.1.39 cells/L; erythrocyte sedimentation rate: 14 mm/hour; blood urea: 6.8 mmol/L; serum creatine: 113 μmol/L; serum potassium: 4.2 mmol/L; serum sodium: 138 mmol/L. Abdominal ultrasound displayed a large upper abdominal mass. Esophagogastroduodenoscopy (EGD) discovered two giant, tough, bezoars. The largest one extended from the gastric fundus to the pylorus, and took the shape of the stomach. A smaller one was round and gray. There was no evidence of gastric ulcer or gastritis. The endoscopy could not scope the duodenum.  conservative, in which the patients are prescribed prokinetic and enzymatic dissolvents like cellulose, papain, acetylcysteine and Coco-Cola or mechanical disruption such as endoscopic fragmentation, gastric lavage or extracorporeal lithotripsy. Small bezoar could be treated conservatively ,13. EndoThe recurrence rate of gastric bezoars has been reported to be around 14% . To prev3Phytobezoar can happen in patients without history of previous gastric surgery or diabetes mellitus, and should not be under estimated. Early diagnosis and treatment is very important to save the patient life and prevent recurrence. Although pharmacotherapy  and endoscopy are the excellent treatment options, surgery (laparoscopy and laparotomy) still play an important role in certain circumstances.No source to be stated.Approval is not necessary for case report in out locality.Consent has been taken from the patient and the family of the patient.Sahir Mahir: Surgeon performed the operation and follow up.Okba F. Ahmed, Shvan H. Mohammed, Fahmi H. Kakamad, and Rawezh Q. Salih: Writing the manuscript and follow up.Abdulwahid M. Salih, Drood C. Usf, Hemn A. Hassan and Fakher Abdulla: literature review, final approval of the manuscript.Not applicable.Fahmi Hussein kakamad.Not commissioned, externally peer-reviewed.There is no conflict to be declared.


Pituitary metastasis is rare, and it is the least common site of intracranial metastases. It is mostly asymptomatic but can present with diabetes insipidus, headache, ophthalmoplegia, visual disturbance and anterior pituitary dysfunction and in majority of cases patients known to have a primary malignancy.This study presents a 54-year-old male presented with a few months history of headache and drooping of both eyelids and after proper investigations and endoscopic resection of the sellar mass the diagnosis was confirmed to be pituitary metastasis of lung adenocarcinoma.Diagnosis and management of pituitary metastases are complex and depend on many factors. Endoscopic surgical resection and debulking alleviates symptoms and provides the definitive diagnosis which has great importance in cases with clinical presentation that does not strongly point to pituitary metastasis to avoid unnecessary radiotherapy and chemotherapy when possible. MetastaThis case report has been reported in line with the SCARE criteria .2A 54-year-old male known to have type II diabetes mellitus, hypertension, dyslipidemia and chronic kidney disease stage III. He presented complaining of on and off episodes of headache mainly at the frontal area for three months duration, which was accompanied with gradually progressing drooping of his left eyelid initially and then involving the right eyelid as well. It was not associated with visual impairment, seizures, fever, weakness or change in sensation and there is no history of trauma. His symptoms were not proceeded by an upper respiratory tract infection or a febrile illness. He also reported an occasional dry cough and he noticed some weight loss; however, it was not measured. The cough was not associated with chest pain, shortness of breath or hoarseness of voice. Upon examination, the patient was found to have bilateral ptosis, ophthalmoplegia and midway pupillary dilation, otherwise the rest of neurological examination was within normal. Brain CT scan and MRI with contrast was performed which showed a sellar and suprasellar enhancing lesion replacing the pituitary gland with extension along the pituitary stalk and invasion of the cavernous sinuses measuring 4.3 × 2.3 × 1.8 cm which was suggestive of an invasive pituitary macroadenoma or a pituitary metastasis. Figs. .Fig. 1MRWith these clinical information, the patient underwent endoscopic trans-sphenoidal sellar mass surgery. Frozen section showed the mass to be a metastatic carcinoma and tumor debulking was done. The final histopathological report was metastatic non-small cell carcinoma/Adenocarcinoma with primary lung origin. It was positive for pancytokeratin, TTF-1, Napsin A, and CK7. After the diagnosis, patient was sent for definitive management. Fig. 4A) 3Metastatic disease to the pituitary gland is considered a rare condition. Although, studies of autopsies have found the percentage of pituitary metastases to be as high as 27 % when including microscopic metastases and metastases to the surroundings of pituitary gland, most patients die without diagnosis ,8. The rIn an autopsy study conducted by Silverman et al. only 7 % of pituitary metastases were symptomatic . DiffereThe radiological modality of choice to evaluate a sellar mass is an MRI. Some of the features that suggest pituitary metastasis include being isotense on both T1 and T2, invasion of cavernous sinus, thickening of pituitary stalk, indentation at the diaphragma sellae giving a dumbbell shape, contrast enhancement and rapid growth in serial images. Most of these features are not specific making it difficult to differentiate pituitary metastasis from begin pituitary lesions ,8,11,12.Management of pituitary metastasis is complex and depends on many factors most importantly the extent and condition of the primary tumor. Endoscopic trans-sphenoidal excision and debulking procedures of pituitary metastases will not affect the survival but it will relieve improve symptoms namely headache and visual symptoms, quality of life and will provide a definitive diagnosis which is vital in cases where the clinical presentation is not suggestive of pituitary metastasis in order to prevent the unnecessary use of radiotherapy ,11,13,14In most cases the diagnosis of pituitary metastasis is based on radiological findings in the presence of primary tumor and it is not confirmed by histopathology . Moreove4Pituitary metastasis is a rare disease with no definitive features in clinical presentation and radiological images, making it difficult to diagnose. Its poor prognosis, especially when associated with wide spread malignancy, makes surgical resection and definitive diagnosis impractical in every case. However, endoscopic surgical resection and debulking procedure have an important role in alleviating symptoms, improving quality of life and confirming the diagnosis when clinical presentation does not strongly point to pituitary metastasis to avoid unnecessary radiotherapy and chemotherapy when possible.There is no conflict of interest regarding the publication of this paper.There was no need of financial support.Case reports does not require ethical approval in our institute.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on requestNot applicableDr. Ali Almomen, Dr. Aminah AlhashemNot commissioned, externally peer-reviewedAminah Alhashem: Data curation, Writing - review & editing. Mahmoud Taha: Supervision. Ali Almomen: Conceptualization, Supervision, Writing - review & editing.


With the advances of neonatology, the survival rate for “live-born periviable fetuses” weighing < 300 g, a subgroup of extremely low birth weight (BW) infants, has improved over the past 10 years. Meconium-related ileus (MRI) represents an early postnatal hazard, and, if medical evacuation fails, a surgical challenge in such immature babies. We report the interdisciplinary management of surgically treated MRI in a newborn with a BW of 273 g. According to the worldwide database held by the University of Iowa, he is registered as the tiniest male newborn in Europe. The boy was born in the 25th gestational week by cesarean section after a triplet pregnancy with twin–twin transfusion syndrome, him being the donor. He had a BW of 273 g, whereas his brothers had a BW of 740 g and 722 g. Cardiopulmonary stabilization and ventilation were successful. He developed MRI unresponsive to medical treatment. On day 14 of life, a minilaparotomy was performed in the right lower quadrant to externalize a loop of the distal ileum in a no-touch technique. Despite the small diameter of only 2 mm, a standard loop ileostomy could be fashioned. There were no intra- or postoperative abdominal complications. Bowel function and weight gain were adequate and the ileostomy was closed electively 5 months later at a body weight of 3.5 kg.In summary, minilaparotomy and loop ileostomy placement were effective to treat surgical MRI in Europe's tiniest male newborn. With the advances of neonatology, pediatric surgery reaches new frontiers as well. The term meconium-related ileus (MRI) was first introduced by Kubota et al in 1999Medical evacuation remains the first-line treatment.We report the interdisciplinary management of a newborn with a birth weight (BW) of only 273 g.https://webapps1.healthcare.uiowa.edu/TiniestBabies/getInfantList.aspx).The neonate was born in the 25th + 3 gestational week by cesarean section after a triplet pregnancy with twin–twin transfusion syndrome, him being the donor. He had a BW of 273 g and a body length of 26 cm, whereas his brothers had BWs of 740 g and 722 g. According to the database of the world's “Tiniest Babies,” held by the University of Iowa, he is registered as the tiniest male newborn in Europe (The infant showed respiratory distress syndrome grade 2 requiring immediate intubation and application of surfactant (200 mg/kg). Extubation could be performed on day 7. Gradually, he developed MRI unresponsive to medical treatment consisting of daily enemas with diluted Microlax. Additionally, the patient received two enemas with diluted water-soluble contrast media (Gastrografin). Abdominal X-rays on day 14 of life revealed distended meconium- and gas-filled small intestinal loops . AbdomiAlthough the survival rate of extremely low BW (ELBW) infants with a BW less than 500 g has significantly improved over the last 5 to 10 years,Obstruction of the gastrointestinal tract by tenacious meconium frequently leads to gastric residuals, a distended abdomen, and delayed food passage. Even in such extremely small babies, rapid evacuation using prophylactic enemas plays a key role to prevent MRI.If medical treatment fails and clinical deterioration develops, surgical decompression must be initiated despite the immaturity of the infantAbdominal surgery in ELBW infants, especially in babies weighing less than 500 g, carries an increased risk for higher postoperative morbidity and mortality versus very low BW infants.Generally, the outcome after surgery for MRI in ELBW infants has improved significantly from early mortality rates of 50% to 67% to present survival rate of near or at 100%.In detail, Kim et alIn our case, postoperative feeding followed the neonatal standards.In summary, we report a successful interdisciplinary management of MRI in Europe's tiniest male newborn (BW of 273 g). His favorable outcome following surgical MRI substantiates the debate that delicate, interdisciplinary management, precise timing, and minimal handling seem key factors of long-term survival with adequate quality of life in such extremely low BW babies.