Microcystic adnexal carcinoma (MAC), a malignant transformation of adnexal structures, constitutes a rare locally aggressive malignancy of skin. Generally, the disease is associated with local invasion and distant metastases are extremely rare. We presented a case of MAC with distant metastasis after adequate wide excision.A 65-year-old man presented with a scar-like lesion at his left hand’s middle finger. As pathologic result from incisional biopsy confirmed MAC, the patient was then treated with ray amputation. Eight years later, there was a presence of scar changes. There was a palpable node at medial epicondyle and presence of enlarged lymph nodes at axilla by imaging with Computed tomography (CT) scan. Following left hand amputation and node dissection at elbow and axillary region, pathologic examination confirmed recurrence and metastasis of MAC. He went through adjuvant radiation with a complete response. At present, two years after surgery, he remains in complete remission.The presence of a recurrent MAC with distant lymph node metastasis after primary surgery is rare. The metastatic route as presented with the positive nodes at elbow and axilla could be explained the spreading via lymphatic system.MAC arising at a hand may require wider excision in order to achieve best result. Although this malignancy is a slowly progressive disease, distant metastasis should always be concerned, especially when there are suspicious presentations. CT scan will render more details and make more precise diagnosis in the suspicious situation. Microcystic adnexal carcinoma (MAC), a subtype of MCANs and generally believed to derive from eccrine glands in origin, is extremely rare . As the This work is compliant with the SCARE checklist, and also, has been reported in line with the SCARE criteria .2A 65-year old man presented to our hospital eight years ago with a scar-like lesion at his left hand’s middle finger, specifically around proximal interphalangeal joint, he had noticed the symptoms for about 6 months prior to visit to the hospital. There was no previous history of burn or traumatic event. And also, no inflammation was noted. The lesion caused his affected joint to a limited range of motion and contracture deformity. As pathologic result from incisional biopsy confirmed MAC, the patient was then treated with ray amputation, a removal of the entire digit and greater part of its metacarpal bone. Pathologic report confirmed a complete tumor excision with free margin. Namely, there was no malignant cells seen within 2 mm or more from the tissue edge. Following a six-month interval surveillance, he had no symptoms until 8 years later when there was a presence of scar changes. The scar at the amputated stump had become harder and more contracted into the proximal part A. At rou3MAC was first introduced in 1982 as a rare locally aggressive malignancy of skin . The preThe patient had a primary lesion arising at his hand, while MAC mostly affects adnexal structures particularly eccrine glands in the head and neck regions . TherefoThe patient also had had lymph node enlargement at his elbow during the examination. Consequently, although we could not palpate any nodules at the axillary region, he was then scheduled for chest CT scan including axilla to evaluate whether this part was actually negative for malignancy. Fortunately, we could detect the clinically negative nodes by this imaging technique. As a result, we suggested that CT scan plays an important role in such a suspicious situation. Wide local excision (WLE) is the most commonly performed treatment in MAC. But, due to an aggressive infiltrative nature of the disease, WLE was reportedly associated with high rate of positive surgical margin that require re-operation and local recurrence rate as high as 60% in some literature . A plausAs the circumstance was a recurrence of the malignancy, only amputation might be inadequate. Although the postoperative chemotherapy had shown less benefit , adjuvan4MAC arising at a hand, a structure more complex than the head and neck region, may require wider excision or some specific techniques in order to achieve the best result. Although the malignancy is an insidious and slowly progressive disease, distant metastasis should be kept in mind if there are some clinically suspicious conditions. Finally, CT scan will help render more details and make more precise diagnosis in a suspicious situation.This work received no funding.The consent form and information sheet using in the process of obtaining a consent were approved by IRB at our institution.The patient has been informed prior to the conduction of this manuscript and informed consent has also been obtained. A copy of the written consent is available for review by the editor-in-chief of the journal on request.Kobkool Chakrapan Na Ayudhya and Vichack Chakrapan Na Ayudhya collected data and wrote manuscript.Panat Tipsuwannakul, Sarun Thongvitokomarn, Vorapatu Tangsirapat, Panutchaya Kongon, Juthamas Thananon and Sirirat Sookpotarom contributed to conceptualization.Paiboon Sookpotarom contributed to conceptualization, data curation, supervision and editing of the manuscript.Paisarn Vejchapipat finally edited this manuscript.NA.Paiboon Sookpotarom.Not commissioned, externally peer-reviewed.The authors declare that there is no conflict of interest regarding the publication of this article.


Adenocarcinomas account for approximately 40% of small bowel cancers. They are typically mucosal lesions with distinctive features on endoscopy. We describe a rare case of duodenal adenocarcinoma presenting as a subepithelial lesion which posed a diagnostic challenge. An 85-year-old male patient presented for investigation of iron deficiency anaemia. Initial upper endoscopy found a subepithelial duodenal lesion with central depression but otherwise normal appearing mucosa. Superficial biopsies of the duodenal lesion were unremarkable. Subsequent antegrade single balloon enteroscopy revealed active bleeding from the lesion which was refractory to endoscopic treatment. A complete local excision of the lesion via laparotomy was necessary to achieve haemostasis. Histopathology from the lesion showed a moderately differentiated duodenal adenocarcinoma with invasion into the submucosa but no evidence of lymphovascular spread. Duodenal adenocarcinomas are rare gastrointestinal tumours associated with a poor prognosis. This case report outlines a rare presentation of duodenal adenocarcinoma and highlights the importance of judicious assessment of lesions found on endoscopy. Advances in endoscopic diagnostic modalities could facilitate early diagnosis and improve therapeutic outcomes. Adenocarcinomas account for 40% of cancers in the small intestine and the most common site of involvement is the duodenum . They arAn 85-year-old male patient was referred for investigation of iron deficiency anaemia. His comorbidities included ischemic heart disease and localized prostate cancer with previous transurethral resection (TURP) and external beam radiotherapy (EBRT). Gastroscopy found the presence of two Forrest grade III antral ulcers which were biopsied. Histopathology from the gastric antral biopsies showed moderate chronic gastritis and focal intestinal metaplasia. His colonoscopy was unremarkable. CT abdomen pelvis with oral and intravenous contrast did not find a source of bleeding, evidence bowel obstruction or mural thickening suspicious for malignancy. He subsequently underwent capsule endoscopy for investigation of occult gastrointestinal bleeding which found a non-bleeding lesion in the duodenum. Antegrade single balloon enteroscopy again identified the 1.5 cm polypoid, subepithelial lesion in the lateral wall of the second part of the duodenum, distal to the major papilla. The lesion had central depression and minor ulceration but otherwise normal appearing mucosa on endoscopy . Initialth edition, Stage I, T1bN0M0)  has demonstrated utility in the assessment of subepithelial lesions found on endoscopy. It provides a minimally invasive method of acquiring tumour characteristics such as size, layer of infiltration, and the presence of local lymphadenopathy which holds prognostic value and guides the use of biopsy techniques. EUS itself can be combined with fine needle aspiration (EUS-FNA) and fine needle biopsy (EUS-FNB) techniques for the sampling of lesions arising from the submucosa and muscularis propria . EUS-FNBThe treatment approach for duodenal adenocarcinoma differs from that of other subepithelial lesions in the small bowel. The American Society of Gastrointestinal Endoscopy (ASGE) outlines an approach for the management of subepithelial lesions, which takes into account histological features, size, layers involved, distal spread, symptomatology, and other patient factors . EndoscoAlthough rare, there have been previous reports of duodenal adenocarcinoma presenting as a subepithelial lesion. Kojima et al. reported this presentation in a 63-year-old female patient undergoing investigation for epigastric pain . The lesThis case report describes an unusual presentation of duodenal adenocarcinoma as a subepithelial lesion with initially unremarkable superficial biopsies. The diagnosis was only made retrospectively following complete excision of the lesion. The failure of initial biopsies to successfully diagnose the underlying lesion exposes the limitations of current techniques in the sampling of subepithelial lesions not accessible via endoscopic ultrasound. Improvements in endoscopic modalities for the assessment of subepithelial lesions could facilitate early diagnosis and improve therapeutic outcomes.


Calcifying odontogenic cyst (COC) is a benign, locally aggressive, slow-growing lesion. Its occurrence constitutes about 2% of all odontogenic cysts. The most frequent sign is painless, slow growing swelling. Radiographically it appears as a well-defined unilocular radiolucency. The microscopical features of lesion showed well-delineated cystic proliferation of odontogenic epithelium with ghost cells and fibrous connective tissue wall. In the present study, 42 years old man who has COC associated with the impacted third molar treated with enucleation is reported after 6 months follow-up. COC is a developmental cyst with odontogenic origin, and constitutes 2% of all odontogenic tumors [Calcifying odontogenic cyst (COC) was defined by Pindborg in 1962 as an analog to calcifying epithelioma in the skin [c tumors . It may c tumors . Radiogrc tumors ,6. It afc tumors . It oftec tumors . It is dA 42 years old, systemically healthy male visited the Department of Oral and Maxillofacial Surgery, School of Dentistry, University of Abant Izzet Baysal, Bolu, Turkey for a routine check-up. His panoramic radiogram revealed an irregularly bordered radiolucent lesion with approximately 2cm diameter around the impacted right mandibular wisdom tooth . A cone et al. classified the lesion as cyst and neoplasm and proposed the term dentinogenic ghost cell tumor for the neoplastic variant [et al. reported that the least common pathology associated with impacted teeth is calcifying odontogenic cyst [et al. reported a case of COC with recurrence that showed transformation to giant cell odontogenic carcinoma [From the time it has been described, COC has led to many controversies regarding its nature, terminology and classification. The main reason for these controversies is the presence of two different forms as cystic and neoplastic lesions. Pretorius  variant . The pre variant . There i variant . Histolo variant . Mortazanic cyst . It has arcinoma . This poOral and maxillofacial surgeons should be careful about the radiolucencies associated with impacted third molars.The authors declare no competing interests.


Giant vulvar condyloma is usually associated with the HPV subtypes 6 and 11 and is characterized by excessive growth of verrucous lesions on the genitals and/or perianal region. It may be observed in sexually inactive as well as sexually active women. Immunosuppression plays an important role in the development of the disease. We report two cases of giant vulvar condyloma together with the review of the literature. One case was a 21-year old sexually inactive woman with a history of Type 1 Diabetes. Second case was a 20-year-old sexually active woman with a rapidly progressing disease and cervical dysplasia. Both cases were operated; all the condylomatous structures were resected with preservation of the anatomy and clitoral innervation and blood flow. Skin and subcuticular dehiscence was the only complication encountered in the first case. Main treatment of giant vulvar condyloma is surgical resection with maintenance of the vulvar anatomy. Preservation of especially the clitoral innervation as much as possible is very important. Genital human papilloma virus (HPV) infections are transmitted primarily through sexual contact, with a lifetime risk of 50-80% . The higHere we present two cases with giant vulvar condylomas and their management with a review of the literature.th day, a 2-3 cm wound dehiscence involving the skin and subcuticular fatty tissue developed on the perineal area near the anal orifice and was treated with secondary healing using sitz baths, antibiotics, and topical creams [A 22-year-old sexually inactive woman presented with a rapidly growing mass in the vulva and perianal region. The lesions appeared 3 months before presentation and developed rapidly. The patient had no sexual activity. She was a regular smoker and the medical history included Type 1 diabetes mellitus for 15 years. The serologic screening for Hepatitis B (HBsAg), Hepatitis C (Anti-HCV), Human Immunodeficiency Virus (Anti-HIV), and syphilis (VDRL) was negative. Patient had no history of sexual diseases. On physical examination, a giant mass thought of as a giant condyloma, extending from the mons pubis till the anal mucosal lining and distorting the labial and clitoral anatomy was observed . Biopsy l creams . Patholol creams . No recuA 20-year old sexually active woman presented with a giant vulvar mass involving the vulva and the anal area. Lesions began to develop 5 months before presentation and developed rapidly. Medical history was unremarkable. Serologic screening for Hepatitis B (HBsAg), Hepatitis C (Anti-HCV), Human Immunodeficiency Virus (Anti-HIV), and syphilis (VDRL) was negative. Patient had no history of sexually transmitted diseases. Physical examination of the patient revealed condylomatous masses extending from the lower perineum to the intergluteal folds . Biopsy Buschke-Loewenstein tumor is a sexually transmitted disease caused by HPV subtypes 6 and 11, with benign histological features, but with excessive local growth and high recurrence rates . They maRachman and Hasan reported a case of a 42-year-old woman presenting with giant vulvar condyloma diagnosed with systemic lupus erythematosus (SLE) one year ago and treated with surgical excision . The patCostaPinto et al. reported a 33-year-old patient diagnosed with SLE during pregnancy presenting with giant vulvar condyloma after delivery . The patPetrini et al. reported a case of a 16-year-old patient with alcohol consumption and illicit drug usage . The autMain treatment of giant vulvar condylomas is surgical excision. Apart from the aesthetic appearance of giant vulvar condylomas and the difficulty of surgical treatment, it is even more important to note that vulvar verrucous carcinoma should be included in the differential diagnosis of giant vulvar condylomas. Zerkan et al. reported a case of giant vulvar condyloma, which was resistant to conservative treatment and operated five times. The definitive pathologic examination revealed superficial vulvar carcinoma . Liu et In conclusion, giant vulvar condylomas, which are caused by low-risk HPV types and treated with surgery, should be distinguished from verrucous carcinoma. Local aggressively growing vulvar condylomata, are seen more often in women with immunosuppression and should be treated as soon as they are diagnosed.


Majority of these are malignant.•Differentiation between a vascular – leiomyoma and a leiomyosarcoma is chiefly upon histopathological examination. Imaging alone is non-contributory.•The rarity of the tumour makes predicting the prognosis a challenging prospect.•Renal Leiomyomas do not show aggressive behaviour and excellent prognosis is predicted post nephrectomy.•An eight-year long charting of progress of leiomyoma in our case has demonstrated the non-malignant potential of such tumours. Vascular leiomyoma originating in the renal vein is a rare entity. Our case allowed us charting of progression of renal vein leiomyoma for 8-years’ duration. Apart from intraluminous growth causing displacement of viscera, the leiomyoma did not show any aggressive behaviour. Eventually, removal of the tumour through nephrectomy provided complete symptomatic relief to our patient.A 75-year old female presented with an eight-year long history of severe pain in right flank. CT imaging revealed a mass arising from the right renal vein and a few hepatic tumours. The differentials included leiomyosarcoma, in view of hepatic lesions. Excision of the tumour was performed with radical nephrectomy. Immunohistopathological examination confirmed it to be a case of leiomyoma. Three months post-op, the patient reported complete pain relief and no adverse events, and there were no signs of recurrence.Vascular leiomyomas do not show any aggressive behaviour. However, their growth tends to displace the normal viscera, causing pressure effects that may give rise to intractable pain. Vascular obstruction affects GFR and renal function. In our patient, despite a long period of growth, the tumour did not show any malignant changes. Tumour excision through radical nephrectomy resulted in symptomatic improvement in our patient. Since imaging alone does not distinguish a leiomyoma from its malignant counterpart with certainty, surgical excision may be warranted in all the cases. Vascula2A 75-year old homemaker presented to us in November 2018 with severe pain in right flank of eight years duration. On examination, she was moderately obese ; the general physical exam was essentially normal and abdominal physical exam did not reveal any abnormality. She reported a history of diabetes mellitus and hypertension.After a gradual onset, 8 years ago, the intensity of her pain stayed 2–3 on a 10-point scale for the next 4–5 years. The pain had no aggravating factor and was relieved with meals. It was dull aching in quality and non-radiating. It did not interfere with her daily activities and rarely woke her up from sleep at night. The pain episodes always occurred at fixed time of the day. She had 1–2 such episodes per day on an average. A complaint of persistent bilateral pedal oedema unamenable to limb elevation and tight stockings was also elicited. Imaging done at that time had suggested of a renal tumour and a biopsy was scheduled. The patient declined any further treatment from then on. She sought pain relief from Homeopathic medication.Six years ago, she underwent open cholecystectomy for symptomatic cholelithiasis. However, a month of pain-free period post cholecystectomy was followed by a return of pain of same intensity and character , Fig. 4.For the last couple of years, patient reported worsening of pain. She rated the pain intensity at 10/10. The pain evolved more constant frequency and began radiating to her back.During her visit in November 2018, she had declined further imaging and had opted for continuation of palliative care instead.Later, a CECT done in April 2019 showed a 6.3 × 4.1 × 4.7 cm dense space occupying lesion in right paracaval retroperitoneal region. This was a significant increase in the dimensions of the tumour from the last imaging done a year ago that had measured it at 3.12 × 4.2 × 3.6 cm. The growth was splaying the renal vein and had advanced through inferior vena cava’s (IVC) lumen, causing its narrowing. There were prominent soft tissue densities in lymph nodes in peripancreatic retroperitoneal region. There were 2 focal hepatic mass lesions measuring 3 × 3.6 × 3.2 cm and 1.5 × 3 × 2.5 cm in subcapsular location. Note was also made on presence of multiple uterine leiomyomata. A tentative diagnosis of metastatic leiomyosarcoma of the right kidney was made. By this time, she still had significant residual pain despite medication with maximal doses of Dicyclomine, NSAIDs and opioids. At this visit, the patient agreed to a palliative surgical resection of the tumour.During surgery, a large hypervascular lobulated lesion was discovered medial to the right kidney, inseparably adherent to the right renal hilum, encasing and splaying the right renal vein with intraluminal extension into the infrahepatic IVC. Extensive colonic and omental adhesions required careful dissection. Further up, the growth was adherent to the 2nd–3rd part of duodenum and the uncinate process of the head of pancreas, without any infiltration. The growth was encroaching upon abdominal aorta at the level of right renal hilum, which needed to be carefully dissected out. An en-bloc right nephrectomy was performed to remove the renal lesion. The growth in the IVC required a 10 cm excision of its lateral wall and reconstruction. The post-op course was uneventful and the patient made a full recovery from the surgery.The report on the histopathological sample of the resected growth , Fig. 6 Three months after the surgery, the patient reported near complete absence of the debilitating pain. She experienced 1–2 episodes of pain rated at 3–4/10 in intensity occurring at fixed time of the day, associated with an onset of urge to defecate and completely relieved by defecation or when the urge to defecate died down. A repeat CT scan of abdomen done in October, 2019, did not reveal any evidence of disease recurrence.3Leiomyomas are benign mesenchymal tumours that commonly arise in smooth muscle–lined hollow organs. Histologically, leiomyomas may be classified as either simple, epithelioid, or with a prominent vascular component (vascular leiomyoma or angiomyoma). Vascular leiomyomas are further classified as capillary angioleiomyomas, cavernous type or venous angioleiomyomas . Among aThe Leiomyomas tend to grow from their origin in the wall of the vein, displacing the nearby structures, rather than infiltrating them. But they can grow along the vein wall and spread within its lumen in a downstream direction which can lead to thrombosis. Hence, the recommended procedure is excision of the mass and ligation of the feeder vessels ,8.Upon extensive search of literature, only five cases of leiomyoma of renal vein were found to have been reported . The dis4We reported a rare case of renal vein leiomyoma that clinically presented with flank pain of long duration. Eventually, tumour excision, along with nephrectomy, provided symptomatic relief to the patient. There were also no signs of tumour recurrence, just as reported in the literature. The diagnostic challenges and the pre-op clinical progression of the disease makes our case unique.This report did not receive any kind of funding or sponsorship from any external agency.The hospital ethics committee verified that the patient’s treatment did not involve any experimental procedure and all standard of care procedures were followed. This case report manuscript was approved by the ethics committee. The approval code was DHE401.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 upon request.All non-essential details have been omitted to maintain patient’s privacy and to ensure anonymity.All doctors were directly in charge of medical and surgical management of the patient.N M Gupta, MS: Case chief surgeon, case report concept, manuscript review.Kuldeep Dhawan, MS: Case surgeon, diagnostic work up and plan of surgery, case report design, supervisor.Nakul Bansal, MBBS: Case assistant surgeon, diagnostic work up and case analysis, post-op follow ups, literature and reference search, data collection and data interpretation, original manuscript preparation and preparation of revised drafts.Simran Dhawan, MBBS student: reference search and manuscript revisions.All authors have read and approved the final manuscript for submissionNot required as this was neither a research study, nor a first-in-man study.Dr Kuldeep Dhawan.Not commissioned, externally peer-reviewed.The authors have no conflict of interests to declare.


Congenital pyriform sinus fistula (CPSF) is a branchial abnormality originating from the third or fourth branchial pouch and is an important cause of anterior cervical abscess in children. Here we present a case of neck abscess in a newborn that was diagnosed as CPSF.A male infant with a birth weight of 3660 g was admitted to hospital 25 minutes after birth after discovery of a cystic mass with extensive skin swelling in the left side of the neck. B-mode ultrasonography of the left neck showed an anterior cervical cystic mass of indeterminate nature.Congenital pyriform sinus fistula.The neck abscess was incised and drained under general anesthesia. Examination under suspension laryngoscopy revealed a pyriform sinus fistula. Laser cauterization was performed simultaneously. The wound was dressed and anti-inflammatory treatment was provided.The neck wound healed uneventfully. After 3 months, the fistula was confirmed to be closed by laryngoscopy under general anesthesia. No recurrence was detected during 9 months of follow-up.CPSF should be strongly suspected in a patient with an unexplained neck abscess or recurrent acute suppurative thyroiditis, especially on the left side. The possibility of this disease should be kept in mind in a patient with an acute suppurative thyroid inflammatory response, especially on the left side and with repeated episodes, and timely examination is needed for a definitive diagnosis.,4 Several imaging investigations, including barium swallow X-ray, computed tomography (CT), magnetic resonance imaging (MRI), suspension laryngoscopy, and esophagoscopy, are generally used to confirm the diagnosis.Patients with congenital pyriform sinus fistula (CPSF) generally present with recurrent neck swelling, pain, and/or a fistulous orifice with purulent discharge in the anterior or lateral neck region. CPSFs tend to be misdiagnosed as acute suppurative thyroiditis, neck cellulitis, neck abscess, or another disorder, such as a thyroglossal cyst or second branchial cleft.2A male neonate was admitted to our Department of Pediatrics directly from the delivery room 25 minutes after birth following discovery of a cystic mass with extensive skin swelling in the left side of the neck Fig. . He had A chest X-ray suggested an increase in lung markings on both sides. No abnormality was found on color Doppler ultrasonography of the head. Echocardiography revealed a left-to-right  shunt, an atrial septal defect, and a patent ductus arteriosus. A B-mode ultrasonographic examination of the left side of the neck revealed an anterior cervical cystic mass of indeterminate nature. Routine blood, urine, stool, electrolyte, and liver and kidney function tests were unremarkable and myocardial enzyme levels were normal. Blood culture showed no bacterial growth. A B-mode ultrasonogram obtained at 39 weeks’ gestation had revealed a 2.8 × 2.6-cm anechoic mass in the left side of the neck with the esophagus and trachea shifted to the right. A further B-mode ultrasonogram obtained one day after birth revealed a 5.1 × 3.8 × 3.1-cm cystic mass extending from the posterior pharynx and left side of the trachea to the front of the ascending aorta beneath, with no obvious displacement of the trachea but forward and outward movement of the jugular vein. The mass was observed to be regular in shape with a visible capsule and a clear boundary. No obvious blood flow signals were found in the mass on color Doppler flow imaging. On the 6th day after birth, B-mode ultrasonography showed a 6.6 × 5.0 × 3.9-cm cystic mass in the deep cervical fascial space on the left side; the mass had a clear boundary, an irregular shape, and a thick cystic wall, with dense spot-like echoes and gas-like echoes in the cystic cavity but no substantial space-occupying echoes. The upper lateral margin of the mass was adjacent to the left submandibular and parotid glands, and the lower margin extended to the thymus gland; it was adjacent to the left carotid sheath on the deep side with no wrapping and adjacent to the esophagus and left thyroid lobe on the medial side. A portion of the mass extended through the posterior part of the trachea to the inside of the right carotid sheath. Color Doppler flow imaging revealed spot-like blood flow signals in the wall of the cyst but no obvious blood flow signals in the mass. Axial contrast-enhanced CT scan image showing an abscess cavity with an irregularly enhanced cystic wall (Fig. 2 laser at a frequency of 2 W in continuous output mode. An area of basal mucosa in the pyriform sinus with a radius of 10 mm was burned, taking the internal fistula as the center (Fig. The neck abscess was incised and drained under general anesthesia. Approximately 200 ml of foul-smelling pus was drained intraoperatively Fig. . Examinater Fig. .After surgery, the wound was dressed and anti-inflammatory treatment was provided. The neck wound healed uneventfully thereafter. Three months later, the fistula was confirmed to be closed by suspension laryngoscopy under general anesthesia Fig. . No recu3,7 A CPSF can be clinically classified as a sinus, that is, a blind-ending tract that opens to either the pyriform fossa or the skin, a cyst, which is not open to either, or a fistula, which is a tract connecting the pyriform fossa and skin. A sinus CPSF is most commonly encountered, although a fistula may develop after iatrogenic or spontaneous rupture of a cervical abscess. At least 80% of pyriform sinus fistulae occur in preschool children and the incidence is similar in both sexes. There is a general lack of awareness of this disease among non-specialist clinicians, which often leads to misdiagnosis and inappropriate treatment.CPSF is a branchial abnormality originating from the third or fourth branchial pouch and is an important cause of anterior cervical abscess in children. The lesion mainly occurs on the left side and may be associated with asymmetric development of the original great artery and bilateral branchial apparatus or a C-cell migration disorder. However, because of the abundant blood supply and thick fibrous capsule in thyroid tissue, these lesions rarely become infected under normal conditions. Accordingly, the possibility of this disease should be considered in a patient with an acute suppurative thyroid inflammatory response, especially if it is on the left side and recurrent. Prompt investigation is needed to make a definitive diagnosis and to exclude other causes of a neck mass, such as a thyroglossal cyst or a second branchial cleft.Patients with a pyriform sinus fistula usually show recurrent cervical swelling, pain, and an external opening with purulent discharge at the anterior border of the middle or lower third of the sternocleidomastoid muscle after an upper respiratory tract infection, often with symptoms of acute suppurative thyroiditis.,12 Both MRI and CT can identify a tract between the internal orifice and the neck mass or infection. MRI can detect soft tissue inflammation in the affected area. CT is considered to be more effective and requires a shorter scanning time than MRI.,13 CT typically identifies inflammation and/or an abscess near the lesser horn of the thyroid cartilage, with air bubbles forming in the lesion or along the putative tract or may reveal a loss of high density in the thyroid gland. Further confirmation of an internal orifice in the pyriform fossa by suspension laryngoscopy or esophagoscopy is the gold standard for diagnosis of CPSF.The diagnosis relies not only on clinical presentation but also on radiological findings. A barium swallow X-ray can reveal a sinus tract in the pyriform fossa, but the sensitivity of this method is only 50% to 80%.The external component of a pyriform sinus fistula is often absent and the internal fistula is concealed, which increases the likelihood of delayed diagnosis and treatment and leads to a relatively high rate of missed diagnosis and inappropriate treatment. Suspicion for CPSF should be high in a patient with an unexplained neck abscess or recurrent acute suppurative thyroiditis, especially if it occurs on the left side. Surgical treatment, such as endoscopic cauterization or open surgery, is required as soon as the diagnosis is made.A multicenter, prospective, randomized controlled study that includes long-term clinical follow-up is needed to investigate the advantages of surgical methods. Improvement in our understanding of this condition should lead to more effective methods of diagnosis and treatment. In summary, CPSF should be considered in neonates with a congenital cystic mass in the neck and in older pediatric patients with this presentation, even if there is no history of recurrent neck infection. In this case, a congenital cystic mass in the neck was found in the fetus and diagnosed as CPSF after birth, as opposed to repeated neck infections previously reported.4The need for approval to publish this report was waived by the Institutional Review Board of Shenzhen Children's Hospital. However, written informed consent was obtained from the patient's parents for publication of the details of the patient's case and the accompanying images.Data curation: Saihong Han.Investigation: Shuling Huang.Project administration: Yishu Teng.Resources: Shuling Huang.Supervision: Shuling Huang, Zhixiong Xian.Visualization: Guowei Chen, Lan Li.Writing – original draft: Yishu Teng.Writing – review & editing: Yishu Teng.


Background: Small bowel cancers, specifically duodenal cancer, occur at very low rates but require aggressive surgical resection when diagnosed. An even rarer finding is the presence of intestinal malrotation.Case Presentation: We present the unique case of a patient with both duodenal cancer and partial intestinal malrotation undergoing pancreaticoduodenectomy. We discuss the challenges faced and techniques used to successfully perform a surgical resection in this circumstance.Conclusion: Understanding of intestinal malrotation and review of the imaging is crucial in preparing for a resection of a duodenal tumor in a patient with this condition. A review of cases at the Massachusetts General Hospital found only 82 confirmed diagnoses of IM in adults for a 17-year period.3 In adults, IM is often asymptomatic and found incidentally. We present a unique case of a 76-year-old male who presented with both duodenal cancer and incidental small bowel malrotation with associated radiological studies, pathology report, and a review of literature.Small bowel neoplasms are a rare form of cancer in the United States, with ∼10,470 estimated new cases and 1450 estimated deaths per year.The patient is a 76-year-old Caucasian male who initially presented with melanotic stools and anemia requiring multiple blood transfusions. He had a medical history of hypertension, hyperlipidemia, chronic kidney disease, and a myocardial infarction that resulted in a cardiac stent placement. During his workup for anemia, he underwent an esophagogastroduodenoscopy (EGD) and colonoscopy. His colonoscopy revealed no worrisome lesions in the colon. EGD revealed a large malignant appearing mass 4–5 cm in size just adjacent to the ampulla, which was biopsied. Pathology analysis returned as invasive moderately differentiated adenocarcinoma. He had a computed tomography to further delineate the mass and to assist with operative planning, which showed thickening of the duodenum as well as a partial malrotation at the third and fourth portions of the duodenum such that the ligament of Treitz failed to cross the midline . LaboratThe patient was taken to the operating room for an open pancreaticoduodenectomy. Upon exploration, the duodenum and head of the pancreas revealed a palpable mass at the level of the second part of the duodenum and extending close to the first part of the duodenum and further caudal. It was necessary to proceed with a classic Whipple procedure since a pylorus-preserving pancreaticoduodenectomy would have compromised the duodenal margin. There was no evidence of carcinomatosis, malignant lesions in the liver, or omental implants. On exploration, various structures were abnormal. The duodenal C loop was vertical, and there was an ectopically located ligament of Treitz; it was found on the right side and inferior to its typical location. In addition, the presence of malrotation in the setting of significant mesenteric adipose tissue warranted that a longer portion of his jejunum be resected to yield sufficient mobility of the retained proximal jejunum to allow for a tension-free anastomosis of the jejunum to the pancreas remnant and biliary tree. The patient had normal positioning of the mesenteric arteries, which facilitated normal dissection of the pancreas around the superior mesenteric vein (SMV) and superior mesenteric artery (SMA). However, the presence of considerable mesenteric fat and bowel malrotation provided significant, although overcomable, challenges to our operation.The pancreaticojejunostomy, hepaticojejunostomy, and gastrojejunostomy anastomoses were completed without incident. The specimen was sent off to pathology analysis and the frozen section analysis revealed a duodenal adenocarcinoma, with all resection margins being negative for malignancy.Postoperatively, the patient experienced transient delirium and developed an International Study Group for Pancreatic Fistula (ISGPF) grade B pancreatic leak that was drained percutaneously. He was discharged to a rehabilitation facility on postoperative day 13.The pancreaticoduodenectomy specimen contained a 3.2 × 2.5 × 2 cm exophytic tan pink to red firm granular mass within the duodenum, ∼13 cm from the proximal gastric resection margin. The mass was adjacent to a normal ampulla of Vater. Upon sectioning, the mass grossly appeared to extend into the muscularis propria but did not involve the underlying pancreatic parenchyma. There was no dilation of the pancreatic duct. The mass was a moderately differentiated adenocarcinoma of the duodenum with focal invasion of the subserosal fatty tissues. A microscopic neuroendocrine tumor was also identified within the duodenal wall. It was well differentiated, low grade, and 2 mm in greatest dimension. All 12 regional lymph nodes recovered were negative for metastatic disease. All resection margins were also negative for neoplasm. The final staging of the duodenal adenocarcinoma was T3N0M0.A Pan-Cancer 42-Gene mutation panel of the specimen was performed. Analysis showed a G12V mutation of the KRAS gene, as well as a T>G nucleotide change in codon 560-3 of the TP53 gene. Both of these changes were classified as pathogenic. No further mutations were identified in the other 40 genes in the panel.5 Small bowel cancer occurring in the setting of IM is a rare combination and can pose unique surgical challenges as the anatomical variations can often be located in the pancreatic region. Rittenhouse et al. performed a retrospective review of the incidence of gastrointestinal malrotation discovered incidentally on preoperative imaging before hepatobiliary surgery. They found an incidence of 3 out of 1220 cases, or 0.2%.6 In planning for these cases, the surgeon must be prepared to deviate from the classic surgical techniques utilized to ensure a safe resection.Cancer of the small bowel is a rare entity and accounts for only about 3.3% of gastrointestinal tract neoplasms. IM is a congenital anomaly that occurs in 1 in 500 live births and very rarely presents in adults.6 In our case, we were required to divide the proximal jejunum 60 cm below the ligament of Treitz to create a tension-free anastomosis. This is a much longer proximal limb than is typically required. Mateo et al. discuss the need to modify the approach to pancreaticoduodenectomy secondary to the presence of Ladds' bands and the need to release these peritoneal extensions while exercising caution in avoiding the vascular arcades of the small bowel.7For example, in two cases reported in Rittenhouse et al., the jejunal limb had to be brought up for the pancreaticojejunostomy and hepaticojejunostomy in a paracolic manner, as opposed to the standard retrocolic manner.8 Although not the case in our patient, Mateo et al. demonstrated significant vascular variation of the SMV, SMA, and celiac trunk in three patients with IM undergoing pancreaticoduodenectomy.7 These vascular structures deliver most of the blood supply to the pancreas and midgut and are the centerpieces for dissection during pancreaticoduodenectomy. The presence of abnormalities in these critical vessels must be closely scrutinized on preoperative imaging preoperatively to prevent unintentional injury or ligation of the vessels.It is also imperative to have an understanding of the vascular variants that may be present in a patient with malrotation. The presence of the SMV found to the left of the SMA, known as the SMV rotation sign, may be indicative of IM in the adult patient.9–11 As the ligament of Treitz is an important landmark for identification of the mesenteric vein during pancreaticoduodenectomy, absence or ectopic placement of this structure carries with it the possibility of intraoperative injury or postoperative complications. In our case, the ligament of Treitz was displaced from the normal location and somewhat inferior. We worked carefully along the plane between the uncinate process of the pancreas and the transverse mesocolon to identify and dissect the SMV.Finally, the ligament of Treitz can often be found in an abnormal position or may be missing altogether in patients with IM.We present a rare case of a patient with adenocarcinoma of the duodenum and IM treated with pancreaticoduodenectomy. Close review of imaging and understanding of IM helped us prepare for the challenging but ultimately successful resection of the tumor.


This report presents a root-treated right maxillary first molar with recurrent abscess formation and a radiolucent periradicular lesion surrounding the distobuccal root of the right maxillary second molar. The patient underwent surgical retreatment, employing CEM root-end filling, which resulted in no relief from sign/symptoms. In the cone-beam computed tomography (CBCT), the relationship of the lesion with the mesio-buccal root of the second maxillary molar was detected. Despite the latest tooth showed positive responses to pulp sensibility tests, endodontic therapy was planned for it. During treatment, it became clear that the mesiobuccal canal pulp was necrotic, although vital pulp tissues were present in two other root canals. Following treatment, full recovery from all discomforts was obtained and the lesion healed after 18 months. This case showed that a more complicated evaluation such as CBCT should be used for diagnosis of perpetuated lesions. Furthermore, it might be probable that root canals of vital teeth become necrotic due to involvement in the adjacent apical lesion, a phenomenon known as anachoresis. As the dental pulp could not be directly inspected before endodontic treatment, indirect evaluation of the pulp status  Apical periodontitis is the sequel of the dynamic encounter between an endodontic infection and the host’s defense, which results in inflammation and destruction of periradicular tissues . If apicFor correct diagnosis and verification the origin of periradicular lesions, assessment of the health status of the pulp in involved teeth is necessary . Pulp seRadiographic evaluations play a crucial role in diagnosis of periradicular lesions. The extent of the lesion, number of roots and root canals, detection of the involved root or roots, and whether a lesion around one root has a communication pathway to another root, should be investigated through the radiographic examinations . The limThis report presents a case of apical periodontitis assumed to be related to a failed conventional root canal therapy of a maxillary first molar, which perpetuated even after surgical retreatment. Finally, it was proved to be attributed to the adjacent partially-vital maxillary second molar, which had given normal positive responses in pulp sensibility tests.A 51-year-old male with no history of systemic disease was admitted to the endodontic department of a private dental clinic. He was suffering from recurrent abscess formation in the right maxillary buccal vestibule near the right upper first molar. Radiographic examination  revealedAfter a 0.12% chlorhexidine mouth rinse, under local anesthesia with 2% lidocaine plus 1:80000 epinephrine , a full mucoperiosteal flap was retracted. The lesion was curetted and the specimen was sent for histopathological examination. Root-end resection, root-end preparation, and root-end filling with calcium-enriched mixture (CEM) cement  was performed and the flap was repositioned . HistopaAfter preparing the access cavity under isolation, it was observed that the mesial root canal had no bleeding/vital pulp. It was necrotic despite the existence of vital pulp in the two other root canals. Cleaning and shaping using full-strength sodium hypochlorite was performed; in the same session, root canals were obturated with gutta-percha  using Roth 808 endodontic sealer . Subsequently, the coronal restoration with amalgam was carried out . One weeIn this report, a persistent apical periodontitis is presented, attributing to a root-treated right upper first molar and to its adjacent second molar. The ultimate goal in an endodontic diagnostic procedure would be estimation of the histological status of the pulp and periradicular tissues through the radiographic and clinical data collected . ConsideAs the clinicians cannot inspect pulp tissue directly before starting endodontic treatment, indirect evaluation of the pulp status via clinical tests is necessary . The valThe spatial relationship between the lesion and the mesiobuccal root of the second molar which assumed to be vital was observed in the CBCT. In some anatomical regions, such as posterior maxilla, interpretation of two-dimensional periapical radiographs would be more difficult. Existence of various anatomical structures, such as maxillary sinus and zygomatic buttress, proximity of maxillary molars’ roots and their overlaps contribute to those difficulties . CBCT scAnother point is how the right upper second molar became necrotic, even though no caries, no defect in the restoration, no periodontal disease, and no physical trauma had occurred recently. There are rare cases in the reviewed literature that the periapical infectious lesion of a tooth spread to the root canals of the adjacent teeth and devitalized them. Extension of an apical periodontitis, associated to a mandibular central incisor, to the roots of contiguous teeth has been reported, which the contiguous teeth underwent necrosis. The process was attributed to the proliferation and retrograde entrance of microorganisms from the spreading infectious lesion to the unfilled root canals of the adjacent teeth . AnotherNowadays several calcium silicate based cements have been introduced in dentistry with the aim of different worthy applications in clinical practices like pulp capping, root-end filling, reparative material for perforations and periodontal defects. They can be set in contact with body fluids particularly blood, and can endure dislodging forces while positioning or masticating . MineralWhen a periradicular lesion is persistent in spite of good quality endodontic treatment, more complicated/expensive and less available para-clinical tests _such as cone-beam computed tomography_ should be considered.There might be a rare possibility of retrograde passage of bacteria from an apical infectious lesion to the adjacent teeth.


A mesenteric chylous cyst is defined as a cyst occurring in the mesentery of the gastrointestinal tract anywhere from the duodenum to the rectum and is diagnosed most often during the fifth decade of life.In our case report, we describe a case of 38-year-old Greek woman who presented at our Emergency Department complaining of abdominal pain without any other symptoms. Her medical and family histories were clear and she had never had any abdominal interventions. During an imaging examination with ultrasound of her abdomen, an anechoic lesion in her upper left abdomen was revealed. In a further investigation with computed tomography, a well-defined hypodense cystic 7.08 × 6.05 cm mass with mild enhancement was noted. The mass was excised by open laparotomy within healthy borders and the specimen was sent for pathological examination. The histopathological findings were found to be most consistent with a simple lymphatic (chylous) cyst of the mesentery. A review of the literature considering this rare entity was also performed to evaluate our treatment strategy and the result was analyzed.Chylous cysts represent a diagnostic challenge and they should be considered when a physician encounters an intraabdominal mass. Physical examination and imaging do not always provide a diagnosis and surgical management should be advised due to the potential complications that may develop. Mesenteric cysts represent a rare pathologic entity. Their existence was first reported in 1507 by Benevieni, an Italian anatomist, when he performed an autopsy on an 8-year-old boy and the first effective surgical excision was performed in 1880 by Tillaux . MesenteWe report a case of 38-year-old Greek woman who presented to our Emergency Department complaining of abdominal pain during the last week without any other symptoms. Her clinical history was clear and she had not noticed the occurrence of the same symptoms before. A clinical examination revealed only focal tenderness in the left part of her abdomen. Laboratory results were within normal limits. During an ultrasound examination of her abdomen, an anechoic lesion in her upper left abdomen was revealed. In a further investigation with computed tomography (CT), a well-defined hypodense cystic 7.08 × 6.05 cm mass with mild enhancement was noted Fig. . SurgicaAlthough chylous cysts are usually asymptomatic, however, they may give rise to symptoms due to compression of adjacent structures, stretching of the mesentery by rapid expansion, infection, or rupture with hemorrhage . There aThe specific diagnosis of these lesions is difficult prior to surgery as there are no pathognomonic symptoms or imaging findings. Abdominal radiographs are usually non-diagnostic. Ultrasound, which is often used in the initial evaluation of a suspected abdominal mass, may show a well-defined fluid-filled cystic structure adjacent to bowel loops –10. A flIn cases of a large chylous cyst, especially in symptomatic cysts, surgical excision is advised to prevent a potential malignant transformation as well as the development of complications. The preferred technique entails open or even laparoscopic enucleation of the mesenteric cyst; that is, the atraumatic separation of the cyst from the surrounding leaves of mesentery –13. WhenIn our case, after initial abdomen exploration, a mass sized 7.08 × 6.05 cm arising from small bowel mesentery was revealed. Our target was the complete excision of the cyst within healthy borders. In case of diverticulitis, open laparotomy approach is the treatment of choice. In this case, laparoscopy was not considered because enucleation of the cyst could not be performed safely due to adhesions of the cyst wall to surrounding mesenteric tissue. The total operative time was 105 minutes and our patient’s postoperative period was uneventful. She was discharged on the fifth postoperative day.Histopathological examination of the surgical specimen may reveal a unilocular or multilocular cyst, containing a viscous fluid with chylomicrons, cholesterol crystals, and triglycerides (chyle), surrounded by a single layer of flattened mesothelial immunoreactivity cells with cytokeratins and lining a fibrous wall with lymphocytes , 10, 12.The findings of our systematic review including location, gender, size, and publication date are shown in Table Chylous cysts represent a diagnostic challenge and they should be considered when a physician encounters an intraabdominal mass. Physical examination and imaging do not always provide a diagnosis and surgical management should be advised due to the potential complications that may develop.


Schwannomas are common, well-encapsulated benign tumors of the peripheral nerves. They rarely emerge from the median nerve in the forearm. Here we report a case of an unusual schwannoma measuring 3 × 4 × 3 cm originating from the median nerve in the proximal forearm of a 49-year-old man. The mass was painless, but Tinel’s sign was positive. Ultrasonography showed a solid, hypoechoic mass with central cystic areas in the flexor muscle group in the left forearm. Magnetic resonance imaging with contrast enhancement in T1 sequences revealed that it originated from the median nerve. Surgical resection was performed by separating the nerve fibers from the tumor without any complications. Histological examination confirmed it as a schwannoma. Schwannomas, which develop from Schwann cells, are the most common benign tumors of the peripheral nerves , accountThe clinical symptoms and radiological images of schwannomas can be similar to the other soft tissue tumors. Intraoperative findings and microscopic analyses with immunohistochemistry features make it easy to differentiate schwannomas from other tumors. Histologically, schwannomas have two components, namely highly cellular Antoni A and loosely cellular Antoni B patterns, and show uniformly intense immunostaining for S-100 protein .Here, we report a rare schwannoma in a 49-year-old male patient and present the clinical, radiological, and histological findings.A 49-year-old man presented with painless swelling in his left arm. A round, hard, mobile mass was detected at the volar aspect of his left proximal forearm on physical examination. Tinel’s sign was positive. No neurological deficits were detected. The patient had no comorbidities and no remarkable medical or surgical history. His general condition was optimal. Ultrasonography (USG) showed a solid, hypoechoic mass with central cystic areas located in the flexor muscles group in the left forearm. Magnetic resonance imaging (MRI) revealed a huge, well-encapsulated mass originating from the median nerve on contrast-enhanced T1 sequences Figure , 1B.USG-guided biopsy under local anesthesia confirmed the diagnosis of a schwannoma. The patient underwent surgery under local anesthesia for excision of the tumor. A tumor-centered incision was made at the midline of the ventral face of the left mid forearm for adequate exposure of both the proximal and distal sides of the tumor on the median nerve. A round, yellow-pink tumor was enveloped in an eccentric true capsule between the brachioradialis and flexor carpi radialis muscles under the skin. Following the incision of the tumor capsule, the nerve fibers entering and exiting the tumor were isolated. The encapsulated 3 × 4 × 3 cm sized mass of median nerve at the proximal forearm was completely resected in one piece without nerve injury Figure .Tinel’s sign of the median nerve was completely resolved in the early postoperative period. The pathological diagnosis was determined to be a schwannoma Figure .Immunohistochemical analysis of the tumor showed strong, diffuse expression of S100 protein Figure .The Ki-67 [DAKO (MIB-1)] index was 2%-3%, and smooth muscle actin (SMA) [SCYTEK (1A4)] was negative.Schwannomas, also known as neurilemmoma, are common, soft tissue tumors of the extremities that can be categorized as proximal i.e., those on the brachial plexus and upper arm, or distal i.e., those on the forearm and hand . SchwannSchwannomas may be found incidentally as painless masses or can exhibit neurological symptoms due to the compression of the surrounding structures. Symptomatic tumors can present with neurogenic pain in the extremities, local swelling, paresthesia, and motor weakness . A positAccurate diagnosis is the first step for the treatment of schwannomas. USG can identify the localization and origin of the nerve tumor and show its relationship with non-involved nerve fibers . MRI alsMicrosurgical resection is recommended for the treatment of all symptomatic schwannomas at first diagnosis and for asymptomatic cases with MRI evidence of increasing tumor size ,9. NeuroNeurofibroma and malignant peripheral nerve sheath tumors are differential diagnoses of Schwann cell neoplasms. Pathological and immunophenotypic features are useful for this differentiation . The typSchwannomas originating from the median nerve in the proximal forearm are rare. USG and MRI features are very useful for preoperative diagnosis. Pathological and immunophenotypic features are helpful for differential diagnosis of the schwannomas. Complete enucleation of large schwannomas is possible with adequate microsurgical techniques without any complication.


Tracheal head and neck squamous cell cancer recurrence without metastases may be related to physical displacement of cancer cells. They then developed recurrences matching the primary cancer along the entire tracheal length but without other distant metastases, suggesting possible novel mechanisms of spread.Head and neck squamous cell cancer (HNSCC) has a propensity to recur in the neck or locally, and are routinely managed with salvage surgery. If surgery is not possible, systemic therapy approaches including chemotherapy and immune therapy are used but are not curative. Herein, we present the cases of two distinct patients with HNSCC who were treated using standard approaches yet presented with extremely focal recurrences that were confluent across the lumen of the trachea but not at any other point.After successful surgery and chemoradiation for their primary lesion, both patients developed cancer recurrence along the entire tracheal length without distant metastases. Established protocols for treatment of this type of recurrence may consider this as distant hematogenous metastases; however, novel definitive chemoradiation therapeutic regimens were used with curative results possible. Isolated endotracheal recurrence remains so rare that only five such cases have been reported over the last 2 decades.1.12) and radiation therapy . Initially, he tolerated it well, but later developed pain and significant weight loss.A 56‐year‐old nonsmoking man presented with a left anterior ethmoidal sinus mass. Computed tomography (CT) scan found a T3N1M0 p16‐negative poorly differentiated papillary squamous cell carcinoma. Fludeoxyglucose positron emission tomography (FDG‐PET) scan identified a left nasal cavity mass with osseous destruction and asymmetric left level 2 adenopathy but no disease outside the neck. The tumor was a highly vascular, friable abnormal mass that invaded the lamina papyracea. It extended into the posterior frontal recess and lateral aspect of middle turbinate superiorly and could not be fully resected. After partial endoscopic resection, the patient underwent chemoradiation with 6 weeks cisplatin  for his extensive tracheal recurrences. PET imaging performed 5 months after completion of his last proton therapy showed no lung metastases and no FDG avid areas in the trachea. The patient remains without evidence of disease on bronchoscopic and radiographic evaluation 12 months after completion of all chemo and proton therapy  during the peri‐operative period following the resection of the patient's primary lesion. Another possible explanation is that this was isolated hematogenous spread sustained over many months. After each tracheal squamous cancer procedure, he was left with no measurable disease and declined systemic chemotherapy. He was offered intensity‐modulated radiation to the trachea which he declined. He did accept curative intent proton therapy (cGyRBE 5400/6400) with concurrent weekly paclitaxel  and concurrent radiation therapy , which he completed despite significant nausea and weight loss. Post‐treatment PET scan demonstrated subtotal resolution of the soft tissue in the suprahyoid of the neck and cervical nodes. However, 9 months post‐treatment, a PET imaging showed FDG avid soft tissue thickening in the posterior wall of the subglottic larynx and in a long segment of the anterior tracheal wall extending from the thoracic inlet to the carina (Figure Following partial resection, he was treated with cetuximab for 6 weeks (loading dose 400 mg/mThe patient underwent salvage surgery remove his pedunculated papillomatous laryngeal and tracheal lesions. These masses appeared malignant on histology, but did not demonstrate invasion of the basement membrane, which is typical of this variant. The recurrences in the anterior and posterior trachea were classified on biopsy as diffuse, persistent, unresectable papillary squamous cell carcinomas—which were nearly identical to the primary tonsillar lesion.Follow‐up imaging demonstrated continuing spread from the confluent crescentic lesions on the anterior wall of the trachea. There were no lung lesions noted or disease outside the trachea. A year after completion of radiation to the oropharynx and involved neck, he was given 69.96 Gy of radiation to the larynx and trachea. He was offered cisplatin concurrently but his performance status declined to the point that he was unable to tolerate more than 2 attenuated doses and completed the remainder of his treatment while hospitalized. Several months after his radiation, he developed pulmonary nodules consistent with pulmonary metastases.2Isolated endobronchial head and neck metastases with no lung parenchymal involvement were originally described as rare, occurring in 1/16 patients with various cancers.Isolated endobronchial HNSCC metastases are unique because they may represent novel patterns of spread and require creative, curative treatment approaches. Endobronchial metastases from other primary sites such as breast or colon primaries are presumed to spread hematogenously, and systemic therapy is reasonable. HNSCC creeping along the length of the trachea has an ambiguous mechanism of disease spread from the primary site. Possible mechanisms include inhalation or instrumentation during the peri‐operative period resulting in mechanical movement of viable cells into the trachea, where they became embedded in the mucosa and continued to grow. This mechanism of spread is analogous to implantation metastases of the skin from biopsy needles or surgical instruments. “Aerial” metastases to the lung from inhalation of laryngeal papillomas were proposed as early as 1932, which presents a similar mechanism to this presentation, in which the recurrences extend across large fields of the mucosal surface from the thoracic inlet to the carina.While cryotherapy ablation temporarily managed discrete recurrent lesions in one of the patients described, it was not an adequate curative treatment option. For our patients with tracheal spread of the cancer, chemotherapy or immune therapy would have been offered for both patients as noncurative, alternate standard treatment. For the first patient, proton therapy was delivered with curative intent along the extent of the trachea with concurrent chemotherapy and has resulted in durable disease control without the addition of any subsequent systemic therapy or visible recurrence. Proton therapy was proposed to reduce the toxicity of radiation to adjoining normal tissue,These two cases highlight a rare type of atypical HNSCC regional recurrence but defy easy explanation or categorization. Cancer primary site, p16 status, initial treatments offered as well as subsequent outcome all differed significantly, with no obvious common connection. Alternate approaches to directly ablative or radiation‐based approaches include an initial trial of systemic therapy, though currently poor response rates and the noncurative nature of these treatments make them less attractive to patients.3Isolated endotracheal recurrences of HNSCC can occur in both p16 positive and p16 negative cancers arising from various primary sites and should be managed by a multidisciplinary team taking into account the potential for cure with directly ablative and tracheal radiation approaches. Mechanical manipulation of tumors may have contributed to the subsequent development of endotracheal metastases, though unique cancer biology may have contributed. We recommend extra care to limit mechanical manipulation of tumors during office procedures or in the operating room. Patients with isolated endotracheal recurrences can be cured with radiation therapy, including proton therapy approaches.None.Mr Benjamin Aaron Bleiberg: contributed to concept for manuscript, majority of writing, and critical review of manuscript. Dr Muhanned Abu‐Hijleh: involved in critical review and editing of manuscript, generation of clinical findings in patient via direct care, and final review of manuscript. Dr William Moore: involved in critical review and editing of manuscript, generation of clinical findings in patient via direct care, and final review of manuscript. Dr Saad A. Khan: contributed to concept for manuscript, critical review and editing of manuscript, generation of clinical findings in patient via direct care, and approval of final draft of manuscript.


Some advanced thoracic malignancy cannot be resected safely by using of conventionalventilation, so some sort of cardiopulmonary support is needed for hemodynamic and ventilationmanagement of the patient. Using extracorporeal membrane oxygenation (ECMO) in comparingwith cardiopulmonary bypass has some advantages. Three patients with huge thoracic tumorswith different ages experienced major surgery in our center by using ECMO in order to facemajor complications mainly due to the size of mass to achieve better hemostatic stabilities, lowerbleeding, and injuries to main airways and secure oxygenation. This is the first case series in Iran,as our best knowledge that explains cases of huge chest mass which were operated perfectly byusing ECMO and short ICU stay and interestingly no major complications. Excision of huge thoracic mass is mainly difficult by using conventional mechanical ventilation and needs some type of cardiopulmonary support. By the current report, we are introducing three cases of thoracic masses that were actually difficult to manage without ECMO. We used ECMO via femoral artery and vein at the beginning of surgery. After successful resection of tumor, flow of ECMO was reduced gradually till it reached 30% of cardiac output and in the presence of hemodynamic stability and ideal oxygen saturation, ECMO discontinued but cannulas remained for about 30 minutes and finally after continuing stability of patient were removed.More than 40 years have been elapsed from the first ideas and versions of extracorporeal membrane oxygenation (ECMO) which was designed as a cardiopulmonary support in adults with multiple indications from respiratory failure management to cardiopulmonary transplantation. Severe respiratory failure with hypoxemia or hypercapnia which responses to mechanical ventilation insufficiently is the main indication for ECMO appliance. One of the best devices ever introduced for the medical approach in these patients is ECMO which affects O2 and CO2 exchange simultaneously.A 59-year-old woman was referred with the history of exertional dyspnea. She also had left-sided chest pain and BIPAP ventilation device dependence. Primary studies found a huge mass in left hemi thorax . The CT-A 45-year-old man with the 2 years history of exertional dyspnea, dry cough and a huge mass in right hemi thorax was referred to our center. The mentioned mass had a fatty density at CT scan while shifted the heart to the left . CT guidA 13-year-old boy with a one-month history of chronic cough in addition to dyspnea referred by oncology service. Vital signs and O2 saturation were rather normal except the respiratory rate of 26/minute. CT scan was done that showed a huge mass with compression effects on the heart . CT guid4 No need to full heparinization and suction as well as better biocompatibility, ECMO has been more attractive for surgeons in these situations.5 ECMO system is heparin-coated and intraoperative anticoagulation is done by only 70 IU/kg body weight heparin for the patient. Furthermore, ECMO has a closed circulatory system with no contact with a suction system in surrounding tissues which contain tumor and normal cell debris. A clean site of operation is always important to keep surgeon very accurate to have a precise dissection, especially in limited spaces like the chest that is truly provided by ECMO. Although Hoechter et al explained that ECMO had no advantages compared with CPB in case of lung transplantation and intraoperative blood transfer rate through their meta-analysis, they disclose that ECMO could reduce ICU stay and was superior in terms of 3-month and one-year survive concluding that ECMO has benefits in its intraoperative use, especially in short-term outcome.6 This is while Sharma et al. have more positive concepts of using ECMO in lung transplantation.7 In terms of morbidity and mortality, Lang et al presented their experience with ECMO on nine patients among them one developed the only ECMO-related problem in their report, lymphatic fistula at the groin, which needed surgical revision. They also reported one case of perioperative death.Resection of advanced thoracic malignancy is always challenging for surgeons that usually originate from the complexity of airway or vascular resection. Although hemodynamic stability is another major concern due to compressive effect of these masses or prolonged surgical manipulation. This case series explains advantages of ECMO in surgeries through which huge thoracic masses were excised when compression effects and the risk of local injuries to airways or great vessels are the main concern. In our patients there was marked compressive effect of tumor on heart and large thoracic vessels so hemodynamic instability especially at the time of lateral decubitus positioning was the main concern. Also according to this large dimensions one hemithorax was totally occluded and tumor was protruded to opposite side so patients had some degree of respiratory problems and with considering these problems using conventional ventilation was inappropriate for resection. Resection of advanced thoracic malignancies or large masses had been always a challenge when conventional ventilation techniques were used which was almost resolved by cardiopulmonary bypass (CPB) that provides oxygenation and hemodynamic stability at the same time. However, despite its perfect effectiveness, more needed blood products following full heparinization of the patient, the risk of tumor cell spilling into the suction system and potential immunosuppressive effects of the device downgrade the value of CPB, especially in cases with huge mediastinal mass resection.This is the first report in Iran, as far as our best knowledge, that explain cases of huge thoracic masses which were operated perfectly using ECMO with shorter ICU stay for patients and interestingly no major complications.Informed consent was obtained from the participants for publication of this case report.All authors declare no competing financial interests exist.


We report a 70-year-old woman diagnosed with unicentric Castleman’s disease with a contrast well-enhanced retroperitoneal tumor of 25 mm in diameter that located between the aorta and inferior vena cava. The imaging finding did not suggest any specific features, and no other lesion was detected. Laboratory examinations indicating malignant lymphoma such as soluble interleukin-2 were all negative. We resected the retroperitoneal tumor laparoscopically, and histopathological examination revealed hyaline vascular type Castleman’s disease. Although complete resection of hyaline vascular type unicentric Castleman’s disease results in a good prognosis, a late relapse has been reported and long-term follow-up is warranted. Importantly, these subtypes are involved in different therapeutic strategies and prognosis.13 We present a surgically treated unicentric HV CD occurring between the aorta and inferior vena cava (IVC).Castleman’s disease (CD) is a rare lymphoproliferative disorder. The etiology remains unclear; however, several theories have been proposed recently. The previous report suggested the association with human herpesvirus 8 (HHV-8) and human immunodeficiency virus (HIV) infection.123I-metaiodobenzylguanidine (MIBG) scintigraphy was also negative. As excisional biopsy and treatment, we resected the tumor laparoscopically. Histopathologic examination showed lymph follicles with expanding mantle zone. Intra-follicular stroma showed a marked proliferation of hyalinized vessels. The vessels penetrated germinal center to form a “lollipop” follicle, and follicular dendritic cells and small lymphocytes form concentric rings . HIV status test was also negative. Computed tomography (CT) scan showed a contrast well-enhanced retroperitoneal tumor of 25 mm in diameter that located between the aorta and IVC . Our ini4 Although viral infection and chronic low-grade inflammation are involved in the onset of CD, the exact pathogenesis is still unclear. Histopathological evaluation is necessary for the definitive diagnosis because there are no disease-specific features in symptoms, a serum marker, and imaging.1 CD has three histologic subtypes: HV, PC, and mixed type.1,2CD is a rare atypical lymphoproliferative disorder that was first described in 1954.3 It occurs in various parts of the body: the chest (24%), neck (20%), abdomen (18%), retroperitoneum (14%), axillary region (13%), groin area (6%), and pelvic region (5%), respectively.1 Multicentric Castleman’s disease (MCD) is a systemic disorder that had similar histologic features to UCD, but involving multiple sites.3 In UCD, the HV type is dominant accounting for 78%, while the PC type accounts for 75% in MCD.1CDs are categorized as unicentric and multicentric disease according to the anatomical distribution. UCD is a localized and single mass without evidence of other clinical or radiological adenopathy.13 The surgical approach for MCD does not necessarily improve its prognosis.13 The role of surgery is limited to obtain tissue for diagnosis and to relieve symptoms such as compression of vessels and airways, massive organomegaly, or bowel obstruction.1 Systemic therapy with steroids or anti-IL-6 monoclonal antibody has been used for MCD. However, the previous report suggests that the 3-year disease-free survival rate was 45.7% for MCD-PC patients.2These subtypes are directly related to the therapeutic approach and prognosis.1 However, local recurrence has been reported to occur even 14 years after complete resection for UCD-HV; therefore, long-term follow-up is needed as it is in the present case.5On the other hand, a surgical approach is a gold standard for the treatment of UCD. UCD has a favorable prognosis when achieved with complete surgical removal, and the cure rate is reported to be more than 90%.We reported a case of UCD. Since the imaging finding does not suggest any specific features, histopathologic evaluation is necessary for the definite diagnosis of CD. In general, complete resection of UCD-HV has a better prognosis; however, late relapse has also been reported, indicating a need for long-term follow-up.


Colonoscopy is considered as one of the most common performed procedures for both diagnostic and therapeutic purposes. Serious complication after colonoscopy could occurs like bleeding or perforation. Majority of the perforations are intraperitoneal, while extraperitoneal perforations are very rare and it may cause pneumoretroperitoneum, pneumomediastinum, pneumothorax and subcutaneous emphysema.A case report was described of 80-year-old female who underwent a diagnostic colonoscopy for recurrent abdominal pain who presented with severe abdominal pain and tenderness with CT findings of pneumoperitoneum, pneumoretroperitoneum and subcutaneous emphysema. The patient successfully treated surgically with laparoscopic Hartman procedure.Colonic perforation is rare complication of colonoscopy that can manifest intraperitoneal, extraperitoneal or a combination of both. Patient risk factors include advance age, female sex, diverticulosis, previous abdominal surgery and pre-existing colonic strictures. Technical risk factors includes excessive shearing forces during endoscopic insertion, dilation, biopsy and using electrocautery. Majority of the intraperitoneal perforation warrant a surgical intervention whereas isolated extraperitoneal perforation may be managed conservatively.Combined intraperitoneal and extraperitoneal colonic perforation are rare presentations following diagnostic colonoscopy and often difficult to diagnose based on the clinical manifestation only. Understanding the manifestation of extraperitoneal perforation will help to properly identify the condition and preventing morbidity and mortality in these patients. The freFurthermore, the majority of the CP are IPP, while RPP are extremely rare . An IPP 2An 80-year old Hispanic American female patient presented for the evaluation of recurrent abdominal pain, and excessive bloating after every meal, without any significant past history of weight loss and change in bowel habits. At initial presentation, her vital signs were within normal range. CT abdomen was insignificant. At this point, she was scheduled for the esophagogastroduodenoscopy (EGD), which showed no significant abnormality. However, her symptoms persisted, and one week later she was scheduled for colonoscopy by a senior gastroenterologist for the evaluation of lower gastrointestinal tract. She was vitally stable, and colonoscopy was passed through the anal canal under the direct visualization. Shortly after the introduction of the scope, significant diverticulosis and kinking were seen the level of the sigmoid colon. Every effort was made to pass, the scope safely beyond that level, but it was not successful; the procedure caused significant discomfort requiring higher dose of propofol and midazolam and the decision made not to proceed further. The scope was withdrawn, and the patient returned to the recovery room. Two hours following colonoscopy, the patient complained of severe LLQ abdominal pain, but she was vitally stable. Additionally, her abdomen was soft; however, significant tenderness in the LLQ of abdomen and subcutaneous crepitus in the right upper thigh was noticed. She was immediately rushed for CT abdomen which revealed massive pneumoperitoneum, pneumo-retroperitoneum, and subcutaneous emphysema , Fig. 3.At this point, the decision was made to take her back to the operating room (OR). She received proper preoperative intravenous fluids and broad-spectrum antibiotics. Distal sigmoid colon perforation with feculent peritonitis was seen, and laparoscopic Hartman procedure was performed. The postoperative period was uneventful, and her condition improved gradually. Her stoma was functioning very well, and diet advanced gradually. She was discharged home with home health care and scheduled for postoperative follow up in the clinic.3Since 1970, colonoscopy has been widely adopted by the physicians across the world as the primary diagnostic tool for patients with suspected bowel lesions ,3. Due tAlthough colonoscopy is considered an extremely safe procedure, serious complications can occur following colonoscopy, and iatrogenic CP being the most lethal ones . AdditioInterestingly, in a prospective analysis of 13,580 colonoscopies, Wexner et al. proposed that the complication rates following colonoscopies are associated neither with the experience level nor with the number of the colonoscopies carried out annually ,13. HoweAlthough CP can occur at any intestinal locations, the most commonly affected regions are sigmoid colon followed by the caecum ,16. In aAdvances in radiological imaging have also permitted better delineation and improved sensitivity in detecting CP following colonoscopy. Cho et al. in a study of perforations caused by diagnostic endoscopy and therapeutic colonoscopic interventions reported that abdominal roentgenogram (AR) is a cost-effective and useful method in detecting subdiaphragmatic free air (SFA), with a positive predictive value of 92% . HoweverTreatment of CP has historically been either conservative management or surgical intervention depending on the type of CP. Tiwari et al. reported that 75% of isolated RPP were treated conservatively, while 60% with combined IPP and RPP need surgical intervention . The rem4Combined IPP and RPP are rare presentations following diagnostic colonoscopy and often difficult to diagnose based on the clinical manifestation only. Clinically deteriorating patients following colonoscopy should alert the physicians to possible CP. CT abdomen provides excellent diagnostic information in CP patients, which can be managed either conservatively or surgically depending on the location and type of the CP.Nothing to disclose.No sponsor. Study was conducted at Brandon regional hospital.The study has been exempted by our institution.Written informed consent was obtained from the patient for publication of this case report and accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.Sahned Jaafar, MD was resident involved during the case and also writing the manuscript.Suy sen Hung Fong, MD was resident involved during the case and also writing the manuscript.Abdul Waheed, MD were involved in writing the manuscript and editing the draft of the manuscript.Subhasis Misra, MD, MS, FACS edited the manuscript and supervised the manuscript development.Keyur Chavda, MD was the senior surgeon; and also the guarantor of the study; who performed the case and approved the final version of the manuscript.N/A.Keyur Chavda, MD.Not commissioned, externally peer-reviewed.


Lipomas are very common benign tumors of mature fatty tissue that can occur in any part of the body. However, lipomas of the parietal peritoneum are extremely rare.A 36-year-old man presented with urinary frequency for 6 months. On computerized tomography of the abdomen and pelvis, a well-defined fatty mass measuring 20 × 11 × 6.5 cm in size, was found in the lower abdominal cavity. We performed a laparoscopic parietal-peritoneum-preserving excision of the mass. The patient was discharged without complications on post-operative day 6.To our knowledge, a laparoscopic excision with preservation of the parietal peritoneum for a giant parietal peritoneal lipoma has never been reported. Herein, we report a case of a giant lipoma of the parietal peritoneum successfully managed by laparoscopy. Lipomas are very common benign tumors of mature fatty tissue that can occur in any part of the body. There are a few reports of giant lipoma of mesentery or omentum –4. Howev3, and the weight was 942 g. The pathological diagnosis was reported benign lipoma with clear resection margin. The patient was discharged without complications on post-operative day 6.A 36-year-old man presented with urinary frequency for 6 months. He had no significant urologic abnormality and no palpable abdominal mass on physical examination. He denied abdominal pain, vomiting, anorexia, or bowel disturbances. There were no specific laboratory abnormalities. The abdomen and pelvis computed tomography scans showed a 20 × 11 cm, well-defined, fatty mass in the abdominal cavity. A mass was located between the abdominal wall muscles and the peritoneum and compressed bladder Fig.. We perfIn 2006, the first case of a lipoma of the parietal peritoneum was reported by Barut et al . Since tIn conclusion, this case highlights the fact that a giant lipoma of the parietal peritoneum can be an unusual cause of urinary disturbances. Laparoscopic procedures are feasible for the excision of a huge lipomas of the parietal peritoneum, and concomitant preservation of the peritoneum is useful for reducing postoperative pain.


The purpose of this paper was to report the five-year success of Biodentine™ partial pulpotomy in a young permanent molar, with signs and symptoms indicative of irreversible pulpitis and periapical lesion, in a nine-year-old girl. Preoperative clinical examination revealed a large carious lesion of the left mandibular permanent first molar. The patient reported pain on percussion. The tooth responded positively to the electric pulp test and had lingering pain after cold testing. A periapical radiograph showed a deep carious lesion and periapical lesion. Based on the clinical and radiographical examination, the tooth had signs and symptoms indicative of irreversible pulpitis and periapical lesion. During caries removal, pulp exposure occurred, and 2-3 mm in depth of pulp tissue at the exposure site was removed. Haemorrhage was controlled within four minutes with 2.5% sodium hypochlorite-moistened cotton pellets. Biodentine™ was then applied as both a pulp dressing and a temporary restoration. At the following visit, composite resin was placed over the Biodentine™ as a final restoration. During a five-year follow-up, the tooth was asymptomatic, had positive responses to sensibility tests, and had no discolouration. Follow-up radiographs showed a dentine bridge and periapical healing. Partial pulpotomy, one form of vital pulp therapy, has traditionally been indicated only in a cariously or traumatically exposed vital tooth with a clinical diagnosis of normal pulp or reversible pulpitis . When a Mineral trioxide aggregate (MTA) has been recommended as the gold standard for vital pulp therapy; however, it has some disadvantages, such as long setting time, poor handling properties, high cost, and the potential for tooth discolouration . BiodentBiodentine™ presents high biocompatibility with human dental pulp cells . The pulThe purpose of this paper was to report the long-term success of partial pulpotomy using Biodentine™ in a young permanent molar, with signs and symptoms indicative of irreversible pulpitis and periapical lesion.A nine-year-old healthy Thai girl presented to the Pediatric Dentistry Clinic, Faculty of Dentistry, Chiang Mai University, because of pain in the lower left quadrant upon eating and upon drinking cold liquids. Clinical examination revealed a large carious lesion involving the occlusal and buccal surfaces of the left mandibular permanent first molar . The patA local anaesthetic was administered, and a rubber dam was placed. During caries removal, there was a carious pulp exposure, approximately 2.5 mm in diameter. The exposed pulp appeared vital, judged by its appearance, bright red colour, and bleeding overflowing from the exposure site. Then, approximately 2-3 mm in depth of pulp tissue at the exposure site was removed with a sterilized long-shank, high-speed, round carbide bur and irrigated with 2.5% sodium hypochlorite (NaOCl). The bleeding was controlled with 2.5% NaOCl-moistened cotton pellets within four minutes . BiodentTwo days later, the patient returned for a definitive restoration. The outer layer of Biodentine™ was removed leaving 2 mm of space for final restoration with resin composite. A two-step total-etch adhesive  was applied, according to the manufacturer's instructions, over the Biodentine™ and the surrounding dentine wall. Finally, the cavity was filled with resin composite  and light cured for 40 seconds for each surface, followed by the finishing and polishing procedures . The treAlthough contrary to the traditional recommendation , partialWhen there is a pulp exposure, the appearance of the pulp tissue, the colour of the bleeding, and haemorrhage control at the exposure site are also clinical criteria commonly used to judge the vitality of the pulp. In this case report, the exposed pulp appeared vital, judged by its resilient texture, bright red colour, and bleeding overflowing from the exposure site. After pulp amputation, the bleeding was controlled within four minutes, indicating that the remaining pulp tissue was healthy and thus could be preserved. Some authors agree that pulpal bleeding can be used as a clinical indicator of pulpal inflammation , 17; howMoreover, there is still controversy regarding the type of irrigating solution and haemostatic agent most suitable for vital pulp therapy. NaOCl has been one of the most commonly used disinfectants for root canal treatment for many years; consequently, clinicians have arbitrarily adopted the use of NaOCl for vital pulp therapy , 19, 20.β1 (TGF-β1), nerve growth factor (NGF), and glial cell line-derived growth factor (GDNF) in dentine, contributing to the induction of tertiary dentinogenesis [Similar to our case report, previous studies have reported successful outcomes of vital pulp therapy, using bioactive endodontic cement, in teeth diagnosed with irreversible pulpitis and periapical lesion , 24. Theogenesis . De Rossogenesis .Compared to MTA, Biodentine™ has a shorter setting time, better physical and mechanical properties, and easier handling . The seaIn conclusion, Biodentine™ might be a suitable biomaterial in partial pulpotomy of young permanent molars, with signs and symptoms indicative of irreversible pulpitis and periapical lesion. However, further clinical studies with longer follow-up periods and larger sample sizes are recommended.


PDH in pediatric age group has rarely been reported in literature with only five cases of age up to 10 years were reported. PDH is a rare cause of intestinal obstruction, which may lead to subsequent strangulation and perforation of the bowel.Paraduodenal Hernia (PDH) is the most common variant of internal hernia and occurs most often in males during their 4We reported a 1.5 year-old male child presented with intestinal obstruction. The patient experienced abdominal pain, vomiting and irritability. Abdominal x-ray showed distal intestinal obstruction which was discovered to be a result of left PDH incidentally during the surgery. In addition, we performed a literature search using PubMed to identify the published cases of PDH. We also compared our case with the characteristics of all reported PDHs in toddlers and children up to 10 years of age in a concise table.Despite its congenital origin, PDH has been reported in childhood age group in very rare occasions rendering the accurate incidence of PDH in infancy and childhood unknown. PDHs can be asymptomatic or can present most commonly with recurrent upper abdominal pain. Diagnosis is quite difficult in the absence of symptoms but could be achieved using a computed tomography (CT-scan) in non-emergency symptomatic patients. Surgical repair is mandatory to avoid potential complications.As PDH can lead to major and life threatening complications, it must remain in our minds as a possible cause of intestinal obstruction. PDH couPDH accounts for 0.2–0.9% of all intestinal obstruction cases and frequently patients remain asymptomatic until a complete obstruction of the small bowel develops. PDH often presents with vague symptoms of abdominal pain, and/or vomiting . PDH is 2A 1.5-year-old male patient presented to our hospital with a three day history of persistent non-bilious vomiting, associated with paroxysmal irritability, hypo-activity and decreased oral intake. His last bowel movement was three days prior to admission. The child was born full term via normal vaginal delivery following uneventful pregnancy and had no other past medical or surgical history.On examination, the patient was hypoactive but alert. Abdomen was soft and non-distended with no palpable masses and intact hernaal orifices. Initial investigations revealed normal electrolytes, complete blood count, urine analysis, and urine culture. Abdominal x-ray showed air-fluid levels a. An abdWe managed the left paraduodenal hernia by reducing the sac contents into the abdomen cavity without bowel resection a, follow3Paraduodenal hernia, also called paramesocolic or mesocolic hernia, is a rare anatomic anomaly and accounts for 53% of all internal hernia which is defined as a protrusion of an internal organ through an opening in intraperitoneal recess. PDH is of congenital origin and formed by the herniation of small bowel through a congenital defect. This congenital defect is a potential space behind the mesocolon, formed as a consequence of fusion failure of the mesocolon with the parietal peritoneum .In general, internal hernia is three times more common in males than in females. Despite its congenital origin, PDH usually presents in patients between their fourth and sixth decades of life, in contrast to our patient who presented at the age of 1.5 years . An accuin utero herniation of the small intestine through the left paraduodenal fossa of Landzert. The fossa of Landzert is located lateral to the fourth part of the duodenum, posterior to the inferior mesenteric vein (IMV) and the ascending branch of the ascending left colic artery (LCA), directly beneath the posterior parietal peritoneum. Most authors believe that LPDH result from the malrotation of the midgut, by invagination of the small bowel into the avascular segment of the left mesocolon that therefore fails to fuse with the posterior parietal peritoneum [Congenital PDH is divided into three types, left PDH which accounts for 75% of the cases, right PDH accounting for the remaining 25% and the extremely rare transverse PDH . The lefritoneum . Still, ritoneum .Between the fifth and the twelfth week of normal gestation of the human embryo, the primitive midgut undergoes a counterclockwise rotation of 270° around the axis of the superior mesenteric artery (SMA). Usually after this process, fusion of the duodenum, mesentery and mesocolon with the peritoneum of the posterior wall takes place. The Right PDH develops when the pre-arterial limb aborts its counterclockwise rotation at 180° and the post-arterial segment continues to develop and rotates normally. As a consequence the small bowel will become trapped behind and to the right of the SMA and behind the ascending mesocolon which will fail to fuse with the peritoneum of the posterior wall . The extAlmost half of the PDH patients are asymptomatic and the other half can present with recurrent upper abdominal pain (43%) , symptomNonspecific findings can be found during physical examination, including abdominal distension and tenderness . In addiMany of the PDH are diagnosed intraoperatively, at autopsy or during radiological investigation for unrevealed complain. Therefore, the pre-operative diagnosis of asymptomatic PDH resembles a diagnostic challenge. Whenever symptoms develop, diagnosis made easier for clinician . Plain xOnce PDH is diagnosed, surgical repair is mandatory to avoid the increased risk of incarceration or strangulation which accounts for the 20–50% mortality rate . Surgica4PDH is challenging in diagnosis due to its rarity and non-specific presentation. It causes intestinal obstruction leading to serious consequences which can be fatal. Therefore, a high index of suspicion and early intervention are essential.No funding or grant support.The study is exempt from ethnical approval in our institution.The patient consent was obtained by the infant’s father. And the father accepted the final edition of the article.Study concept or design: Radwan Abukarsh, Ihsan Ghazzawi, Shareef Hassan.Data collection and data analysis: Sadi A. Abukhalaf, Aya Mustafa, Mohammad N. Elqadi.Writing the paper: Sadi A. Abukhalaf, Ahmad Al Hammouri, Khalil N.M Abuzaina, Nathan M. Novotny.This is a case report study. No need for registration.Dr. Sadi A. Abukhalaf.Not commissioned, externally peer-reviewed.The following authors have no financial disclosures: Sadi A. Abukhalaf, Aya Mustafa, Mohammad N. Elqadi, Ahmad Al Hammouri, Khalil N.M Abuzaina, Radwan Abukarsh, Ihsan Ghazzawi, Shareef Hassan, Nathan M. Novotny.