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We reported an exceedingly rare adult case of intracardiac teratoma with a bicuspid aortic valve. A small mass was discovered in the right ventricle of a 37-year-old man by accident after he was referred to our hospital due to chronic chest distress and aggravated palpitation. We performed a surgical exploration with excision of the lesion after a repeat transthoracic echocardiography suggested a space-occupying lesion in the interventricular septum. A mature cystic teratoma was pathologically confirmed. During the half-year follow-up, no symptoms were reported. This case constitutes the first report of the smallest primary intracardiac teratoma in an adult with a bicuspid aortic valve.

Highlights • Ureterocele is rare especially with collecting system duplication in the nonorthotopic (extravesical) position in adult. • Ureterocele has a variety of clinical manifestations and complications. • Endoscopic treatment was performed to decompress hydroureteronephrosis. • In this case, laparoscopy heminephrectomy is needed due to dysfunctional uppers moiety. • Renal scintigraphy is a gold standard to assess function in the duplex kidney., Introduction Ureteroceles is a developmental anomaly with cystic dilation of the distal aspect of the ureter and are often associated with some urological anomaly such as a duplicated system or stenotic ureteric orifice. Presentation of case This study reports an ectopic ureterocele in duplication of collecting system associated with double ureters and ureteral ectopia in a woman aged 24 years with minor flank pain. Cystoscopy deroofing of the ureterocele performed and followed by secondary surgery laparoscopic heminephrectomy. Discussion Ureteroceles have various clinical manifestations and complications. Treatment for ureterocele depends on age, type of the ureterocele, obstruction to the draining system, and complications. No single method is sufficient for all cases, and management must be individualized. Endoscopic treatment has gradually broadened as a safe, minimally invasive, and effective procedure, but there is no consensus on its effectiveness for treating ectopic ureterocele. However, it is reported that 50–80% of cases after initial endoscopic treatment require secondary surgery. Conclusions Ureterocele is reported rarely in adults, especially with duplication of the collection system in the nonorthotopic (extravesical) position in women. Cystoscopy deroofing of the ureterocele can be performed to decompress the hydroureteronephrosis, and laparoscopic heminephrectomy can be performed due to dysfunctional uppers moiety. Long-term follow-up is required to monitor renal function, symptoms, and occurrence of vesicoureteric reflux.

An anomalous left anterior descending artery arising from the pulmonary artery (ALADAPA) is exceedingly rare. We present an adult case of ALADAPA that was repaired using a Dacron interposition graft.

Purpose A case is presented here of a persistent vitelline (omphalomesenteric) artery originating from the superior mesenteric artery. Results The artery gives off a branch to the intestinum still within the mesentery, runs freely through the abdominal cavity and reaches the umbilical region along the right medial umbilical fold. The lumen of the persistent vessel is open over the entire length, as can be seen in histological sections. Both umbilical arteries are still present and also have a lumen. Discussion Basic concepts for the normal and misguided embryonic development of these structures are shown. Finally, possible causes for the development of this malformation and its clinical implications are analyzed. Conclusion This malformation can be seen as an inhibition of the regression of the right vitelline artery. The knowledge of a persistent vitelline artery is important for surgeons, because this malformation can be a cause of intestinal obstruction.

Foreign bodies in the nasal cavity are rarely seen in adults, and most cases are traumatic in origin. Multiple foreign bodies in adults are particularly rare. Foreign bodies induce pain, bleeding, and infection but are easily removed in most cases. A patient presented with multiple toothpick foreign bodies in the nasal cavity, which were introduced intentionally through an oroantral fistula. We report an adult case with 37 toothpicks in the nasal cavity, which were removed successfully with a nasal endoscope. Oroantral fistula closure was achieved through oral surgery. Voluntary insertion of foreign bodies into the body by patients with mental illnesses is relatively difficult to identify. Evaluating and managing the psychiatric problems of such patients during treatment planning are necessary.

Background Sacrococcygeal teratomas are tumors originating from pluripotent embryonic germ cell layers located in the fetal coccyx. These tumors are highly vascular if they undergo malignant transformation. Typically, they are found in infants and children and occasionally can be diagnosed prenatally. Adult cases are very rare, and represent tumors present since birth with delayed detection. Case presentation We describe a case of a giant sacrococcygeal teratoma in a 25 years old female college student presenting with right gluteal swelling of 4 months’ duration. In addition to the huge disfiguring mass on the perineal area, she also had lower abdominal pain, urinary complaints, and difficulty with ambulation. Discussion Pre-operative impression was of a sacrococcygeal mass and histopathology following complete surgical excision revealed a sacrococcygeal teratoma. She recovered well after surgery with no radiologic evidence of recurrence at six months. Conclusion Although rare, sacrococcygeal teratoma should be considered as a differential diagnosis for female adults presenting with perineal and/or pelvic masses. Complete surgical excision remains the mainstay of treatment.

Atypical teratoid/rhabdoid tumor (AT/RT) is a highly malignant central nervous system neoplasm predominantly found in children under the age of 3 years, and is extremely rare in adults. There is no specific clinical presentations or radiological features in reported cases of AT/RT. Diagnosis of brain AT/RT is mainly dependent on the classical pathological characteristics. We report a rare case of AT/RT arising from the trigeminal nerve and leading to progressively multiple cranial nerve palsies in a 25-year-old male patient. Microsurgical resection of the tumor has been performed and confirmed the diagnosis by postoperative pathology. To our knowledge, this is the second case of adult-onset AT/RT originating from the trigeminal nerve.

Intussusception in adults is rarer than children and treatment in most cases in adults is surgical. Multiple Intussusceptions in our case with subacute intestinal obstruction and presence of abdominal lymphadenopathy with diffuse bowel wall thickening warrants non-surgical treatment. Presence of multiple intussusceptions is rare findings. We present a case of female aged 35 years presented with palpable abdominal lump associated with loss of weight and appetite. CECT abdomen showed abdominal lymphadenopathy and multisegmental small bowel thickening maximum upto 1.5 cm. Some segments of bowel also show aneurysmal dilatation associated with asymmetrical mural thickening. Telescoping of proximal bowel segment into adjacent distal bowel is seen in at least two places in ascending colon region (colocolic intussusceptions) and another in ileal loops (ileo-ileal). FNAC from the lymph nodes revealed non hodgkins lymphoma.

Congenital cystic adenomatoid malformation (CCAM) is a congenital pulmonary cystic disease that is mostly detected and diagnosed prenatally or during the neonatal period, while rarely being observed in adults. Here, we report an adult case of CCAM that was diagnosed following surgery for a recurrent pneumothorax. We further review 60 case reports on adult CCAM that have been previously published. The patient was a 29-year-old woman with a severe left pneumothorax. Her computed tomography scan showed the presence of multiple pulmonary cysts at the base of the left lower lobe. Since she had experienced a left pneumothorax twice previously, surgery was indicated. A wedge lung resection of the pulmonary cysts was performed thoracoscopically. The postoperative pathological diagnosis was type I CCAM. From the review, 7 adult CCAM patients (11.7%) out of 61, including the patient in the present case, presented with pneumothorax, while 21 patients (35%) presented with infection. Thirty-nine foci of CCAM (65%) were located in lower lobes. Moreover, malignancies were associated in 8 cases (13.3%). We propose that if multicystic lung lesions are found in pneumothorax patients, particularly in lower lobes, CCAM should be considered during the differential diagnosis, even in adults.

Vein of Galen aneurysmal malformation (VGAM) is the result of the direct communication between the arterial network and the median prosencephalic vein. It is a rare vascular congenital malformation representing less than 1% of intracranial abnormalities. This finding is very rare in adults, and it may or may not present symptoms during childhood. Most cases of VGAM can be detected in the fetus by ultrasonography. The referral of pregnant women with fetuses with this condition to centers where better facilities and resources for childbirth and immediate postpartum care are available has resulted in considerable improvement in the prognosis of newborns. Regarding treatment, the endovascular approach to VGAM includes arterial embolization and percutaneous transvenous techniques. The transvenous endovascular treatment was chosen in the case presented in this article.

Sickle cell crisis is an acute clinical condition, caused due to capillary occlusion by the deformed red blood cells, leading to vaso-occlusive status. Vaso-occlusion is an emergency condition requiring intensive care admission and carries a high mortality. Sickle cell crisis is usually managed with hydration, analgesics, and supportive care. Therapeutic red cell exchange transfusion is advised as an adjuvant, for the management of sickle cell crisis, and it is mainly practiced in the pediatric population. We report an adult case of sickle cell crisis managed with therapeutic red cell exchange transfusion in Intensive Care Unit and successful outcome in patient management.

International audience; In case of a nuclear reactor accident affecting a large population the internal contamination of the workers and the public can be assessed through in vivo measurements. Since iodine-131 is one of the most important releases and since it is of health concern, thyroid monitoring of this radionuclide is of special interest. Intercomparisons of thyroid in vivo measurements usually focus on the adult case. Here we report about a European intercomparison exercise for spectroscopic devices. Three sets of thyroid phantoms of unknown activity were circulated, representing the following ages 5 years-old, 10 years-old and the adult. Nineteen institutions from twelve countries in Europe took part in an intercomparison exercise. Twenty-nine measurement devices have been tested. In the adult case, 5 results were outside the acceptance criteria and 10 in the 10-years old case. In the 5 years-old case, the result was not reported for one measurement device and 11 results were outside the acceptance criteria. Devices that have been tested during this intercomparison now dispose of calibration coefficients for the three age groups. © 2019

We present a rare adult case of bilateral oval and round window atresia. Clinical and audiologic findings were suggestive of otosclerosis. High resolution CT Temporal bones showed unequivocal findings of bilateral oval and round window atresia. Atresia of these windows is a rare temporal bone anomaly. Presentation as an adult can confound the clinicians and warranting a closer look on the CT for atretic windows and subtle signs of otosclerosis in patients with conductive hearing loss.

Adult Hirschsprung's disease is a rare motor disorder of the gut that is frequently misdiagnosed as refractory constipation. We describe a 30-year-old patient with adult Hirschsprung's disease with a history of chronic constipation requiring daily enema. Barium enema and rectal biopsy showed short segment-type Hirschsprung's disease with a grossly distended sigmoid colon with fecal retention. Staged operations, including the Duhamel-GIA method as a definitive surgery, completely resolved the patient's symptoms. There were no complications by the end of a 7-year follow-up. Adult Hirschsprung's disease should be considered in the differential diagnosis of cases where adult patients present with chronic constipation. Despite technical difficulty of the modified Duhamel-GIA procedure due to chronic fibrous changes of the rectum, it is a feasible surgical option for treating cases of adult Hirschsprung's disease.

Case A 61-year-old man who was hospitalized with schizophrenia in a psychiatric hospital drank hot water estimated to be 90°C. Eight hours after injury, laryngopharynx edema gradually progressed, and his breathing deteriorated. Upon arrival at our emergency room, we secured his respiratory tract by nasal intubation under a bronchoscope. Outcome The edema gradually improved after peaking at hospital day 2, and he was extubated on hospital day 18. There were no apparent respiratory or esophageal problems, and he was discharged back to the psychiatric hospital on day 28. Conclusion These types of laryngopharynx burns caused by ingesting hot foods or drinks have been rarely reported for adults. In cases of adults, when the patient is in a special situation such as having a psychiatric disorder, it is necessary to assume that the laryngopharynx burns might be aggravated.

We herein describe an adult case of double-chambered right ventricle (DCRV) with symptomatic drug-intolerant paroxysmal atrial fibrillation (PAf). The woman was referred to undergo radiofrequency ablation (RFA), and mapping of the pulmonary veins (PVs) demonstrated that a spontaneous spike potential originating from the left inferior PV (LIPV) induced sustained Af in the second procedure. Accordingly, the LIPV was regarded as the arrhythmogenic PV. Since complete isolation of the PVs, the sinus rhythm has been maintained for at least two years. This is the first report to describe that RFA for drug-intolerant PAf was useful in a patient with DCRV.

Key Clinical Message We present a 57-year-old man with a 1.5-cm-diameter blood cyst in the left ventricle, which was incidentally detected by conventional diagnostic echocardiography before colon surgery. The cyst originated from the papillary muscle, and the pathological findings were compatible with a cardiac blood cyst.

Blood-filled cysts (BFC) within the heart are common findings at postmortem examinations of fetuses and infants. However, such cysts are very rare entities in adolescents and adults. We report here an adult case of BFC attached to the posterior leaflet of the tricuspid valve, demonstrating the importance of multimodal diagnostic imaging combining both echocardiography and magnetic resonance imaging.

Introduction: Congenital cholesteatoma is thought to be caused by inadequate folding of the epidermoid formation inside the middle ear cleft. During development of the middle ear mucosa, stratified squamous epithelium accumulates in the embryonic life. Its typical appearance is a “pearl” beneath the anterosuperior quadrant of the tympanic membrane. Presentation of case: We report 28 years-old case with congenital cholesteatoma in the posterosuperior quadrant of middle ear cavity. The main complaint was the hearing loss which had developed slowly over several years. Discussion: The case was surgically treated. Postoperative hearing result was satisfactory. Conclusion: Congenital cholesteatoma may occur in atypical locations and ages. Many authors prefer canal wall down tympanomastoidectomy. But it can also be treated successfully by intact canal wall tympanomastoidectomy with good hearing results. © 2015 The Authors. Published by Elsevier Ltd on behalf of Surgical Associates Ltd. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

This is the first reported case of an adult presenting with tethering symptoms, limb discrepancy on physical examination, a low-lying spinal cord, and duplicate filum terminale discovered intraoperatively. Intraoperative imaging and pathological analysis of a specimen confirmed the diagnosis of duplicate filum. This is the first reported adult case with duplication of the filum terminale. Release of both fila was necessary in this case to relieve the tethering symptoms.

Cavernous hemangiomas of the submandibular gland are rare. Signs and symptoms typically resemble those of sialolithiasis and chronic sialadenitis. If a lesion extends into the parapharyngeal space, otalgia and sore throat can result. Spontaneous regression is not a characteristic of cavernous hemangiomas. Surgical excision is a management option. We report the case of an adult with a submandibular gland cavernous hemangioma with parapharyngeal extension.

Chromosome 22q11.2 deletion syndrome (22q11.2 DS) has a very wide phenotypic spectrum that includes dysmorphic features, cardiac anomalies, and hypocalcemia arising from hypoparathyroidism. We herein describe an adult case of 22q11.2 DS with associated hypoparathyroidism and anomalies of the aortic arch. Because the patient had been diagnosed with primary hypoparathyroidism at another hospital, a diagnosis of 22q11.2 DS had been overlooked. A chest X-ray examination revealed widening of the mediastinum caused by a high-positioned right aortic arch, and we subsequently confirmed a diagnosis of 22q11.2 DS using fluorescence in situ hybridization. Because primary hypoparathyroidism is a rare disorder, physicians should be aware of the variable phenotypic features of 22q11.2 DS.

Background Intra-abdominal lymphangiomas are rare, benign tumors in adults. This report is the third documented case of a lymphangioma originating in the hepatoduodenal ligament that mimicked a simple liver cyst. Case presentation A 50-year-old woman was admitted with a cystic tumor in the right lobe of her liver and underwent laparoscopic excision of the cyst. Operative findings revealed that the cyst had developed in the hepatoduodenal ligament, not in the liver. A small part of the cystic wall remained on the dorsal surface of the hepatoduodenal ligament. Immunohistochemically, the tumor cells stained positive only for D2-40, leading to a diagnosis of lymphangioma (cystic type). Six months later, a cystic tumor recurred and was completely excised via laparotomy. No recurrence was observed after the second operation. Conclusions The incomplete excision of the cystic tumor led us to re-operate 6 months after the first operation for the local disease recurrence.

Multidirectional coronary artery fistulas (CAFs) are rare in patients with tetralogy of Fallot (TOF). We report an adult patient who underwent open-heart surgery for TOF 24 years before the discussed presentation. Coronary angiogram and cardiac computed tomography revealed multidirectional CAFs originating from the left main coronary artery and draining to the left atrium, pulmonary artery and subphrenic artery. The patient also showed additional congenital anomalies, such as persistent left superior vena cava and right-sided aortic arch.

Poststreptococcal reactive arthritis is a type of sterile arthritis which occurs after a betahemolytic streptococcal throat or skin infection. It was defined as scarlatinal arthritis in 1959. Poststreptococcal reactive arthritis is a kind of non-migratory arthritis that usually affects the knee and ankle joints. Although rare, small joints and the axial skeleton may also be involved. In this report, we present a patient with unilateral sacroiliac joint inflammation and adductor enthesopathy following an upper respiratory tract infection who was eventually diagnosed with poststreptococcal reactive arthritis.

Burkitt lymphoma is an uncommon type of non-Hodgkin lymphoma that often in - volves extranodal sites. The disease predominantly affects children and has an aggressive clinical course. We report the imaging findings of a 23-year-old woman with an atypical Burkitt lymphoma that involved both ovaries and kidneys. The patient has remained in complete remission three years after the initiation of treatment.

Rhabdomyosarcoma of the larynx is extremely rare in adults, as only 17 well-documented cases have been previously reported in the English-language literature. Of these, only 2 cases (both male) involved the alveolar subtype of rhabdomyosarcoma, and neither involved the true vocal folds. We report a case of alveolar rhabdomyosarcoma of the true vocal fold in 54-year-old woman. She was successfully treated with conservative surgery, radiation, and chemotherapy. Management of head and neck rhabdomyosarcoma has evolved from radical surgery to less morbid procedures supplemented with radiation and chemotherapy. However, because this tumor is so rare in the adult population, no adult-specific treatment regimen has emerged. Nevertheless, the success of treatment in the pediatric population supports its use in adults.

Atypical teratoid/rhabdoid tumours (AT/RT) are aggressive neoplasms of the central nervous system occurring mainly in the paediatric population. We reported a 32-year-old male patient who was admitted in emergency in unconscious state. The neoplasm was localised in the left frontal region and was totally excised. The histological and immunohistochemical report revealed AT/RT. This unusual presentation underlines the necessity of considering this devastating neoplasm in the differential diagnosis of malignant brain tumours of adults.

症例は21歳の男性で，腹痛，便通異常を主訴に来院した．大腸内視鏡検査で直腸の壁外性圧迫を認め，CT，MRIで膀胱直腸窩を中心に，径10 cmの境界明瞭な腫瘍を認めた．急速に腹痛，腹部膨満感が増悪し，超音波で多量の腹水を認め，腫瘍の破裂が疑われたため，緊急手術となった．術中所見では，骨盤内腫瘍の頂部が破裂し，同部からの出血による多量の血性腹水とムチン様内容物の流出を認めた．出血コントロールのために腫瘍の摘出が必要と判断し，腹会陰式直腸切断術を施行したが，腫瘍は膀胱から前立腺にかけて強固に癒着しており，骨盤内に腫瘍組織の一部が残存した．組織学的には胎児型横紋筋肉腫で，消化管・前立腺に浸潤を認めた．術後肺・肝転移が出現したため，放射線化学療法を施行し，画像上完全寛解となり，術後15か月生存中である．横紋筋肉腫破裂は検索しうる限りで自験例が5例目であり，きわめてまれな症例であるため，文献的考察を交え報告する．

Transverse divergent dislocation of the elbow, in which the radius and ulna dislocate in diverging directions, is an extremely rare injury (19 cases have been reported in the literature). Usually, this type of elbow dislocation occurs in children, and an adult case is extremely rare. In this report, we present a case of a transverse divergent dislocation of elbow in a 20-year-old woman, treated with conservative method and long-arm cast immobilization. At 9 years, she had no pain in the elbow, returned to full activities and had normal function. The X-ray of the elbow was normal, except for a small nonunited bony fragment adjacent to the coronoid process of the ulna.

Mumps is a common disease in childhood and has many complications, but laryngopharyngeal edema is extremely rare. I report herein on an adult case of mumps with laryngopharyngeal edema. An 18-year-old man, diagnosed as having mumps, came to our hospital complaining of dyspnea and swelling of the submandibular and subaural regions. Fiberscopic findings revealed marked edema of the vallecula and left arytenoid. The patient was admitted to our hospital and treated with intravenous steroids. His edema was soon improved with the treatment and he was discharged without undergoing a tracheostomy. The edema in the current case was caused by circulatory disturbance of the lymphatic flow due to swelling of the submandibular glands. This finding suggests that the airway should always be examined laryngoscopically in any case of mumps with combined swelling of the parotid and submandibular glands.

We report a rare case of an adult patient with single atrium without the characteristics of an endocardial cushion defect, associated with persistent left superior vena cava draining to the left side of the atrium. The patient was reasonably active and asymptomatic till the third decade of his life when the cardiac anomaly was discovered and surgically corrected.

This case report shows the successful treatment of Angle Class II maxillary protrusion using a mini-screw as orthodontic anchorage at the maxillary tubercle. A 28-year-old female had an overjet of +6.3 mm, and was missing the upper left first and right second molars. An orthodontic mini-screw was placed at the left maxillary tubercle as anchorage for the retraction of the incisor segment. This was followed by alignment of the dental arches with an edgewise appliance following extraction of the upper right first and lower bilateral second premolars. Good occlusion and facial aesthetics were achieved. The maxillary tubercle is a useful place for the application of a mini-screw to provide anchorage, if there is enough space for mini-screw placement.

症例は58歳の男性で上腹部痛を主訴に当科を受診し, 腸閉塞の診断で入院した. 腹部の手術の既往はなかった. イレウス管造影検査と腹部造影CT検査の結果から小腸腫瘍による腸閉塞と診断し, 手術を施行した. 開腹所見ではTreitz靱帯から約50cmの部位の空腸が腫瘍を先進部として腸重積をしており, 重積を解除した後に空腸部分切除術を施行した.腫瘍は大きさ35mmの粘膜下腫瘍で粘膜面は糜爛を形成しており, 漿膜面にも腫瘍が露出していた. 病理組織学的所見では細胞分裂像は認めなかった. 免疫染色ではc-kitが陽性で小腸gastrointestinal stromal tumor (GIST) と診断した. 現在術後再発を認めず1年間経過している.

Human herpesvirus-6 (HHV-6) is the main etiologic agent of exanthema subitum in young children. Central nervous system (CNS) infections in children due to HHV-6 have been described on many occasions. HHV-6 is also a common cause of infections in immunocompromised individuals. However, little is known concerning the impact of HHV-6 on the CNS in immunocompetent adults. We report the first case of relapsing HHV-6 encephalitis in a healthy 73-year-old female.

症例は69歳, 男性. 64歳時に胃癌で胃全摘出術を受けている. 腹部症状の訴えはないが, 胃癌術後の経過観察のため腹部CT検査を施行したところ, 回盲部にmultiple concentric ring signを認めたので腸重積を疑った. 大腸内視鏡検査の結果, 上行結腸癌または回腸癌による回盲部脱出と診断し開腹術を行ったが, 回腸癌を先進部とする腸重積と判明したので回盲部切除術を施行した. 病理組織所見では, リンパ節転移はなく, 深達度ss, 乳頭状腺癌であった.本症例は下血, 腹痛, 便通異常, イレウス症状等の通過障害を生じたことはなく, 重積, 整復を繰り返していたものと思われた. 術後4カ年無再発生存中である.回腸癌による腸重積症は稀な疾患であるが, さらに本症例は病理組織検査により胃癌と回腸癌による重複癌と診断され, 極めて稀な疾患であり報告する.

A 54-year-old woman was referred to our hospital for symptomatic sinus bradyarrhythmia with a sinus pause of 8 seconds. She was diagnosed with dextrocardia during childhood and discovered to have heterotaxy syndrome when she had an appendectomy during her teenager years. Chest and abdominal examinations by computed tomography showed multiple spleens located on the right side and abnormal drainages of the superior and inferior vena cava. Left isomerism was diagnosed by bilaterally bilobed lungs. Because of a patent bilateral superior vena cava, pacemaker leads were implanted using the right cephalic vein approach. Her fainting symptoms disappeared after pacemaker implantation.

Coarctation of the aorta is the fifth most common congenital cardiac anomaly encountered in adults. It is important for prognosis to diagnose and treat this anomaly early. An aneurysm might develop due to tunica media abnormalities in patients with coarctation of the aorta. We hereby present an adult case with a very rare combination of vascular anomalies including ascending aorta aneurysm, hypoplastic aortic arch, coarctation of the aorta and poststenotic aneurysm.

成人腸重積症は比較的稀な疾患で悪性腫瘍などの器質的疾患が原因であることが多い.今回腸重積症を合併した回腸悪性リンパ腫の1例を経験したので報告する.症例は46歳,男性.間歇的腹痛を主訴に来院し腹部超音波検査にて回盲部にmultiple concentric ring signを認めたため腸重積症と診断し精査をすすめたが器質的原因は不明であった.大腸内視鏡にて腸管の血流障害,虚血性変化を認めたため緊急手術を施行した.開腹所見は回腸上行結腸型の腸重積症で用手的に回腸を引き出すと,末端から約18cmの回腸に半周性の腫瘤を触知したため悪性腫瘍を疑い,可及的リンパ節郭清を含めた回腸部分切除施行した.病理組織所見は悪性リンパ腫diffuse large B cell lymphomaであり,回腸悪性リンパ腫が先進部となった腸重積症と判明した.術後補助療法としてCHOP療法施行中であるが現在再発徴候は認められない.

Langerhans cell histiocytosis is part of a larger group of syndromes described as histiocytoses. The disease may involve single or multiple systems including skin and nervous system. Here we report an adult case where Langerhans cell histiocytosis presented with diabetes insipidus and cutaneous ulcers.

症例は76歳の女性. 肺癌 (高分化腺癌, IB期) の手術既往がある. 開腹歴はない. 腹痛, 嘔吐を主訴に腸閉塞症の診断で入院し, 保存的に一時改善したが腸閉塞が再発したため手術を行った. 回腸末端から約150cmの回腸壁が瘢痕状に狭窄しており同部の小腸を切除した. 切除標本では腸間膜反対側に径20×15mmの粘膜下腫瘍を認めた. 病理組織学的所見では, 粘膜下層から漿膜の脂肪組織にかけて多数の膵組織を認め, ランゲルハンス島, 腺房細胞, 膵導管を有しておりHeinrich I型迷入膵と診断した. 迷入膵は胃, 十二指腸など上部消化管に好発し, 回腸迷入膵は比較的まれである. 自験例は, 開腹歴のない減圧処置に抵抗性の腸閉塞であり, さらに肺癌の小腸転移の可能性も考慮する必要があり, 示唆に富む症例であった.

The accurate clinical diagnosis of benign mixed tumors of the lacrimal gland is important for the proper therapeutic management. We present an adult case with a benign mixed tumor of the orbital lobe of the lacrimal gland, 8 years after periorbital blunt injury. The tumor lesion was diagnosed later in the persisting traumatic tumefaction region. Clinical examination, ultrasonography and MRI revealed a soft-tissue mass with high density and peripheral enhancement over the superior lateral portion of the right eye with expansion to and invasion of the orbital roof and lateral wall. Lateral orbitotomy was performed to resect the tumor. Histopathology disclosed a pleomorphic adenoma of the orbital lobe of the lacrimal gland. Pleomorphic adenomas of the lacrimal gland are seen rarely. The awareness of the clinical and diagnostic features of benign mixed tumors of the orbital lobe should help to avoid complications arising from an incisional biopsy or incomplete tumor resection.

We report one of the largest descriptions of the right atrium (RA) in an adult, in absence of Ebstein's anomaly, tricuspid stenosis and other common adult associations of RA enlargement, such as pulmonary hypertension secondary to chronic pulmonary disease or severe mitral valvular pathology and pulmonary embolism. The RA volume was estimated to be over 1400 ml and was notably disproportionate to that of the left atrium and either ventricle.

A 20-year-old man was admitted because of an abnormal shadow on chest X-ray film. Chest CT showed large cystic shadows and many small cystic shadows in the right lower lobe. He underwent right lower lobectomy of the lung under the diagnosis of cystic lung disease. Microscopically, cyst walls were covered by ciliated columnar cells and lacked cartilage. These findings supported the diagnosis of congenital cystic adenomatoid malformation of the lung (CCAM). CCAM presents respiratory distress mainly in neonates and presents pulmonary infectious signs in adults. It is rare for CCAM to be found by a chest X-ray film of medical check-up.