Kosin Medical Journal

Case reports

Heterotopic ovarian hilus cells of the salpinx: a case report and review of the literature: Bomi Kim; Received April 20, 2023 Accepted August 6, 2023 Published online September 12, 2023; DOI: https://doi.org/10.7180/kmj.23.120 [Epub ahead of print]

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Abstract PDF Supplementary Material PubReader ePub Crossref - TDM: Ovarian hilus cells (OHCs), a counterpart of testicular Leydig cells, are usually found in the ovarian poles and produce androstenedione. Their origin remains a matter of debate, although OHCs are assumed to come from the adrenogenital primordium. OHCs are rarely observed around the poles of the ovary, including the mesoovarium, stroma (perisalpinx) of the salpinx, and the wall of paratubal cysts. Their clinical and pathological characteristics are not well-known because of their rarity. Herein, we present a case of ectopic OHCs in a 48-year-old woman. The patient underwent total hysterectomy and bilateral salpingectomy for vaginal bleeding due to multiple leiomyomas. We incidentally found OHCs in the stroma of the infundibulum of the salpinx, just beneath the tubal epithelium. Their size was less than 1 mm, and they were composed of large cells with central round nuclei and abundant clear or granular cytoplasm. OHCs share morphological and immunohistochemical profiles with ectopic adrenal glands, and the differential diagnosis is sometimes difficult. They do not exhibit microscopic encapsulation or the normal adrenal cortex zonation pattern. The patient was discharged and did not show any abnormal findings during 19 months of follow-up. Analyzing the characteristics of testicular Leydig cells will help understand how OHCs develop and why heterotopic OHCs occur in and around the salpinges.

Primary gastric leiomyosarcoma: a case report and literature review: Yedaun Lee; Received April 10, 2023 Accepted July 5, 2023 Published online August 18, 2023; DOI: https://doi.org/10.7180/kmj.23.118 [Epub ahead of print]

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Abstract PDF PubReader ePub Crossref - TDM: After separating gastrointestinal (GI) stromal tumors from true smooth muscle tumors of the GI tract, leiomyosarcoma (LMS) of the GI tract has become a rare tumor. Gastric LMS is extremely rare and accounts for 0.1% of all cases of LMS in the GI tract. There are few English-language reports of gastric LMS describing radiologic findings. Here, we report a case of gastric LMS and review the recent literature focusing on radiologic findings. An 80-year-old female patient was referred for evaluation of a gastric mass accompanied by severe anemia. The physical examination revealed no specific findings except for an anemic conjunctiva. Laboratory data showed a low hemoglobin level of 5.1 g/dL. Endoscopy revealed a huge subepithelial mass in the posterior wall of the gastric body. Contrast-enhanced computed tomographic images showed an intraluminal protruding enhancing mass with an internal stalk appearance in the gastric body. There was no internal necrosis or calcification. The patient underwent subtotal gastrectomy and was diagnosed with primary gastric LMS. The diagnosis of gastric LMS is challenging due to its rarity. Our case report suggests that the presence of an internal stalk or spouting appearance can help prompt the radiologist to consider gastric LMS in the differential diagnosis.

Squamous cell carcinoma of the pancreas with a pancreatic intraductal papillary mucinous neoplasm: a case report: Nam Kyung Lee; Received April 27, 2023 Accepted July 5, 2023 Published online August 17, 2023; DOI: https://doi.org/10.7180/kmj.23.123 [Epub ahead of print]

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Abstract PDF PubReader ePub Crossref - TDM: Squamous cell carcinoma (SCC) of the pancreas is very rare. No reports have described SCC accompanied by intraductal papillary mucinous neoplasm (IPMN) of the pancreas. This report presents the first known case of SCC with IPMN of the pancreas in a 71-year-old man, with a focus on radiologic findings. Here, the imaging features of SCC with IPMN of the pancreas were similar to those of IPMN of the pancreas with high-risk stigmata features.

Scrub typhus infection in a kidney transplant recipient: a case report: Dongyeon Lee, Joohee Jeon, Jae Sung Ahn, Chung Hee Baek; Kosin Med J. 2023;38(3):224-228. Published online June 9, 2023; DOI: https://doi.org/10.7180/kmj.23.114

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Abstract PDF PubReader ePub Crossref - TDM: Scrub typhus is a febrile disease that is endemic to Asia and the Pacific region. Its clinical manifestations include fever, myalgia, lymphadenopathy, and a characteristic eschar. The main manifestations of this disease are difficult to differentiate from those of other febrile illnesses; thus, a careful clinical examination and a high index of suspicion are crucial for an early diagnosis. Our case involved a 55-year-old female renal transplant recipient who presented with fever and sore throat in November. Her clinical symptoms did not improve after oral amoxicillin/clavulanate administration for 7 days, after which proteinuria and acute kidney injury were identified. After hospitalization, an eschar was found and immunoglobulin M antibodies against Orientia tsutsugamushi were detected by indirect immunofluorescence. She received oral doxycycline for 7 days and showed improvement in renal function and proteinuria. This is the first case report of scrub typhus infection in a kidney transplant patient in Korea. It is meaningful to report that the renal abnormalities associated with scrub typhus improved in a renal transplant patient through treatment of the disease. This case highlights the importance of examining the social history and symptoms of patients suspected of having scrub typhus in endemic areas. Early diagnosis and treatment are crucial in kidney transplant patients to preserve graft function and prevent fatal complications.

Scrub typhus with complications of acute myocarditis and cardiac tamponade in metropolitan areas: two case reports: Ki-Woon Kang, Wonho Kim; Kosin Med J. 2023;38(3):210-214. Published online June 9, 2023; DOI: https://doi.org/10.7180/kmj.23.111

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Abstract PDF PubReader ePub Crossref - TDM: Scrub typhus is known as one of the most common seasonal infections in endemic rural areas, but life-threatening cardiac complications in cases of scrub typhus are very infrequent. In addition, scrub typhus infection has been rarely reported among workers assembling pallets using manufactured wood in metropolitan areas. Herein, we present two cases involving myocarditis and cardiac tamponade as complications of scrub typhus. One patient died and the other patient survived. These cases indicate that scrub typhus infection could be an environmental hazard in metropolitan areas, especially in locations with poor hygiene, and highlight the need for timely diagnosis and proper management of severe scrub typhus infections. Therefore, we present these two informative fatal cases of scrub typhus infection presenting with myocarditis and cardiac tamponade as an environmental hazard in metropolitan areas.

Central diabetes insipidus following COVID-19 mRNA vaccination: a case report: Min-Young Kim, Jong Ryeal Hahm, Jaehoon Jung, Jung Hwa Jung, Kyoung Young Kim, Hosu Kim, Jong Ha Baek, Hwa Seon Shin, Kee Ryeon Kang, Soo Kyoung Kim; Kosin Med J. 2023;38(3):219-223. Published online June 9, 2023; DOI: https://doi.org/10.7180/kmj.23.105

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Abstract PDF PubReader ePub Crossref - TDM: The coronavirus disease 2019 (COVID-19) has been a major public health emergency worldwide. Vaccines were rapidly developed and approved to prevent the spread of viral infection. However, various side effects of the COVID-19 messenger RNA (mRNA) vaccines have been reported after their commercialization. A 24-year-old man visited our emergency department with polyuria and polydipsia that occurred after he received a COVID-19 mRNA vaccine 10 days beforehand. The initial laboratory findings showed very low urine osmolality with hyperosmolar hypernatremia. Based on these findings, diabetes insipidus was suspected, and sella magnetic resonance imaging showed an enlarged pituitary gland and the absence of posterior pituitary higher intensity. After 12 hours of using oral desmopressin acetate, urine volume decreased, and after 5 days of administration, serum electrolyte and serum osmolality improved. This case report of diabetes insipidus occurring after vaccination with the BNT162b2 mRNA COVID-19 vaccine is presented as a reminder that close monitoring is necessary for patients with polyuria and polydipsia after vaccination.

A case report of successfully treated metachronous gastrointestinal stromal tumor and colon cancer: Young Jin Heo, Ji Young Lee; Kosin Med J. 2023;38(1):60-65. Published online March 8, 2023; DOI: https://doi.org/10.7180/kmj.22.138

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Abstract PDF PubReader ePub Crossref - TDM: The diagnosis of gastrointestinal stromal tumor (GIST) has become relatively common in recent years, but little is known about its association with other malignancies. We present a rare case of successfully treated metachronous GIST and colon cancer with concurrent FOLFOX (5-fluorouracil, leucovorin, and oxaliplatin) chemotherapy and imatinib. A 63-year-old man presented with abdominal pain that had started 2 weeks ago, and endoscopic ultrasonography showed masses that were compatible with GIST on the duodenum. He underwent Whipple surgery. One year after the GIST diagnosis, two liver masses were found on abdominal computed tomography images taken for surveillance. A liver biopsy showed metastatic adenocarcinoma, not GIST. Colonoscopy was then performed to identify the primary site of the metastatic adenocarcinoma in the liver, and sigmoid colon cancer was found. He received 12 cycles of adjuvant FOLFOX concurrently with adjuvant imatinib. There were no serious adverse events of grade 3 or higher from either imatinib or chemotherapy. He has completed adjuvant imatinib and FOLFOX chemotherapy and there is no evidence of disease recurrence. When a synchronous or metachronous tumor is found in a GIST patient, the clinician should keep in mind the possibility of another primary tumor of different histopathology, as well as GIST recurrence.

Bochdalek hernia presenting gastrointestinal symptoms in late childhood: a case report: Sang Seok Jeong; Kosin Med J. 2023;38(1):56-59. Published online February 21, 2023; DOI: https://doi.org/10.7180/kmj.22.134

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Abstract PDF PubReader ePub Crossref - TDM: Most cases of congenital diaphragmatic hernia (CDH) can be diagnosed based on symptoms of severe respiratory failure during the neonatal period or fetal ultrasonography. However, some rare cases are diagnosed in late childhood or adolescence. In this case report, I describe an 11-year-old male patient diagnosed with late-onset CDH presenting with acute abdominal pain. The patient had recently experienced anorexia, nausea, and vomiting after eating. However, he reported no abdominal pain or past history of trauma. The abdomen was generally convex. All laboratory data were within normal limits. A chest X-ray revealed elevation of the left diaphragm. Chest computed tomography showed a defect in the left diaphragm. Based on the above radiologic findings, emergency surgery was performed after the diagnosis of diaphragmatic hernia. A surgical incision was performed in the left subcostal area. Finally, late-presenting Bochdalek hernia was diagnosed. The operation was completed and no specific findings on chest X-ray were found after surgery. The patient was discharged on the fourth day after surgery. In conclusion, CDH in late childhood or adolescence is rare and has various clinical manifestations. To avoid complications such as strangulation and bowel perforation, emergency surgery may be required. Thus, it is necessary to suspect CDH in children with recurrent gastrointestinal or respiratory symptoms, based on which an accurate diagnosis can be made and successful surgical treatment can be performed.

Dermoscopic features of an unusual case of targetoid hemosiderotic nevus: Sun Mun Jeong, Jang Hwan Jung, Do Ik Kwon, Seol Hwa Seong, Ji Yun Jang, Jong Bin Park, Min Soo Jang; Kosin Med J. 2023;38(3):215-218. Published online February 15, 2023; DOI: https://doi.org/10.7180/kmj.22.119

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Abstract PDF PubReader ePub Crossref - TDM: Targetoid hemosiderotic nevus (THN) is a rare variant of melanocytic nevus, characterized by a sudden development of a targetoid ecchymotic halo around a pre-existing nevus. THN clinically raises concern for malignant transformation due to its abrupt change in color and size. THN should be distinguished from other diseases showing a peripheral halo, including targetoid hemosiderotic hemangioma, halo nevus, and Meyerson nevus. Dermoscopy can help clinicians to differentiate THN from these diseases. The typical dermoscopic features of THN are known to be divided into two distinctive areas: the central melanocytic area and the peripheral ecchymotic area. In our case, dermoscopy revealed a novel bull’s eye pattern composed of a central area with characteristic features of benign melanocytic nevus, an intermediated white circular ring, and a peripheral milky red area. When a sudden change occurs in a pre-existing nodule showing targetoid features, dermoscopy should be considered before conducting a biopsy or surgical intervention.

Traumatic neuroma of the right posterior hepatic duct with an anatomic variation masquerading as malignancy: a case report: Jae Ryong Shim, Tae Beom Lee, Byung Hyun Choi, Je Ho Ryu, Jung Hee Lee, Kwangho Yang; Kosin Med J. 2023;38(1):66-71. Published online January 18, 2023; DOI: https://doi.org/10.7180/kmj.22.135

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Abstract PDF PubReader ePub Crossref - TDM: Traumatic neuroma (TN), also known as amputation neuroma, is a reactive hyperplasia of nerve fibers and connective tissue arising from Schwann cells after trauma or surgery. TN of the bile duct is usually asymptomatic, but rarely can lead to right upper quadrant pain, biliary obstruction, and acute cholangitis. It is very difficult to discriminate TN from malignancy before surgery, although doing so could avoid an unnecessary radical resection of the lesion. In the course of surgery, TN can be caused by unintentional injury of a nerve fiber near the common bile duct (CBD) and heat damage to an artery, complete ligation of an artery, and excessive manipulation of the CBD. Therefore, to prevent TN after cholecystectomy, surgery should be performed carefully with appropriate consideration of anatomic variations, and a cystic duct should not be resected too close to the CBD. The possibility of TN should be considered if a patient who has undergone CBD resection with hepaticojejunostomy or cholecystectomy long ago experiences symptoms of jaundice, cholangitis, or obliteration of the CBD. In this report, we present a case of TN mimicking cholangiocarcinoma that emerged from a cystic duct stump after cholecystectomy.

A case report of a carotid space abscess due to extraluminal migration of a fishbone into the deep cervical space: Tae-Hun Lee, Ki Ju Cho, Seong Jun Won, Jung Je Park; Kosin Med J. 2023;38(2):151-155. Published online December 19, 2022; DOI: https://doi.org/10.7180/kmj.22.141

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Abstract PDF PubReader ePub Crossref - TDM: Laryngopharyngeal foreign bodies are among the cases most frequently encountered by otolaryngologists. Most foreign bodies can be easily removed without any complications. However, surgical removal is required in some cases. Therefore, a delayed diagnosis or misdiagnosis could cause fatal complications for patients who need a surgical approach. We report a rare case of extraluminal migration of a foreign body to the deep cervical space. The foreign body (a fishbone) was removed by a surgical approach. With a literature review, we also propose an algorithm for the management of suspicious foreign bodies in the neck.

Targeted temperature management in a patient with suspected hypoxic-ischemic brain injury after successful resuscitation from cardiac arrest: a case report: Christine Kang, Won Yong Lim, Young-hoon Jung, Jiseok Baik; Kosin Med J. 2023;38(2):144-150. Published online December 19, 2022; DOI: https://doi.org/10.7180/kmj.22.130

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Abstract PDF PubReader ePub Crossref - TDM: Hypoxic-ischemic brain injury (HIBI) after cardiac arrest (CA) is a leading cause of mortality and long-term neurological disorders in survivors. Targeted temperature management (TTM) has been rigorously studied as a way to improve results compared to a normal body temperature for preventing secondary damage after HIBI. We report a case of successful TTM in a patient who was suspected to have HIBI after resuscitation from cardiovascular collapse due to respiratory failure during elective surgery under brachial plexus block with dexmedetomidine and remifentanil infusion. A 27-year-old male patient developed CA due to apnea during orthopedic surgery. TTM was performed in the surgical intensive care unit for 72 hours after resuscitation, and the patient recovered successfully. TTM application immediately after resuscitation from CA in patients with suspected HIBI may be an appropriate treatment.

Sigmoid colon plexiform neurofibroma as a colonic subepithelial mass: a case report: Hee Won Baek, Eun Jeong Choi, Seung Jung Yu, Myeongpyo Kim, Sang Heon Lee, Sam Ryong Jee, Hyungjoo Baik, Hong Sub Lee; Kosin Med J. 2023;38(2):138-143. Published online November 9, 2022; DOI: https://doi.org/10.7180/kmj.22.123

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Abstract PDF PubReader ePub Crossref - TDM: Plexiform neurofibroma (PN) is an uncommon benign tumor, usually associated with neurofibromatosis type 1. As most PNs involve the craniomaxillofacial region, PN of the colon is very rare. Here we present a case of PN involving the sigmoid colon. A 43-year-old male patient presented to the outpatient clinic for the evaluation of an incidentally discovered sigmoid colon mass. A colonoscopic biopsy was performed for the mass, and the result revealed neuronal proliferation. The patient visited the outpatient clinic a year later with symptoms of abdominal pain and stool caliber change. Biopsy was repeated for the sigmoid colon mass, and the results showed mucosal Schwann cell proliferation and S-100 immunostaining positivity. Computed tomography and magnetic resonance imaging were performed for further evaluation, and neurofibroma or schwannoma was suspected based on the imaging studies. For an accurate diagnosis, the patient underwent surgery to remove the sigmoid colon mass. The final diagnosis of the mass was confirmed as PN. We hereby report a rare case of PN involving the sigmoid colon that could not be diagnosed before surgery.

Disseminated Staphylococcus aureus infection and acute bacterial pericarditis: a case report: Su Hyun Bae, Song-Hyun Lee, Joon-Young Choi, Bong-Joon Kim, Soo-Jin Kim, Sung-Il Im, Hyun-Su Kim, Jung-Ho Heo; Kosin Med J. 2023;38(2):134-137. Published online August 16, 2022; DOI: https://doi.org/10.7180/kmj.22.021

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Abstract PDF PubReader ePub Crossref - TDM: We experienced a case of disseminated Staphylococcus aureus infection with bacterial pericarditis that progressed to septic shock and multiorgan failure despite pericardiocentesis and surgical removal of the original abscess with intensive antibiotic therapy. We report this case because of the patient’s very rare and remarkable echocardiographic findings and highly turbid pericardial effusion.

Secondary hyperparathyroidism due to multiple parathyroid carcinomas in a patient with chronic hemodialysis: a case report: Soree Ryang, Wook Yi, Mijin Kim, Sang Heon Song, Byung Joo Lee, Bo Hyun Kim; Kosin Med J. 2022;37(3):255-259. Published online July 27, 2022; DOI: https://doi.org/10.7180/kmj.22.103

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Abstract PDF PubReader ePub Crossref - TDM: Parathyroid carcinoma (PC) in cases of secondary or tertiary hyperparathyroidism is relatively uncommon, and only a few case reports have described this entity. Although some papers have reported patients with one or two parathyroid malignancies, multiple PC–especially three or more–have been even more rarely reported. Herein, we report a case of secondary hyperparathyroidism due to multiple PCs in a chronic hemodialysis patient. A 54-year-old man with end-stage kidney disease was referred for hyperparathyroidism. He had been diagnosed with chronic kidney disease in 2001 and had begun hemodialysis in 2009. In laboratory tests, intact parathyroid hormone (iPTH) was markedly elevated to 1,144.1 pg/mL (normal range: 15.0–68.3 pg/mL) and serum calcium was mildly elevated to 10.56 mg/dL (normal range: 8.5–10.3 mg/dL). Ultrasonography showed hypoechoic nodules in the posterior part of both thyroid glands. All three nodules showed increased uptake on a 99mTc sestamibi scan. The patient underwent total parathyroidectomy with autotransplantation to the right forearm. Histopathology findings showed three PCs with capsular invasion and one parathyroid hyperplasia. In the immediate postoperative period, the iPTH level dropped from 1,446.8 to 82.4 pg/dL and, after 1 month, to 4.0 pg/dL. This patient needed oral calcium carbonate and active vitamin D to maintain appropriate serum calcium levels. Although multiple PCs are rare, they can cause secondary hyperparathyroidism. Therefore, clinicians should suspect multiple PCs when patients’ serum iPTH levels are exceptionally high. Additionally, since PCs could occur in multiple glands, autotransplantation of the parathyroid gland after parathyroidectomy should be done carefully.

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