F. Soumanou, J. Avakoudjo, I. Gandaho, Euodias Djengue
We reported a case of urethral adenocarcinoma in a 67-year-old woman who was admitted for difficulty voiding, irritative voiding symptoms, bloody urethral discharge and urinary incontinence. Physical examination was realised by gyneacologist and conclued to urological pathology. Urological examination was showed urethral ectropion which was bleeding. She had no fewer, no pelvic pain even less loss weight. Her general state was satisfactory. The lymph nodes of right side inguinal were felt. Computed imaging was showed involvement of right side bladder and bilateral iliac inguinal lymph nodes. Urethral ectropion biopsy anatomopathological screening was conclued to clear cell adenocarcinoma but CIS lesions were not found. The tumor staging was T3N2M0. A radiotherapy was perfomed. The patient was improvement of voiding symptoms and decrease lymph nodes size. Primary urethral carcinomas are rare pathology especially in the woman. Several histological types exist. However, chemotherapy and radiotherapy are very effective on very advanced stages. This cancer may be revealed by an urethral ectropion in a year-old woman.
{"title":"Urethral adenocarcinoma with bilateral iliac and inguinal lymph nodes which revelead by urethra ectropion in 67-year-old woman: A case report","authors":"F. Soumanou, J. Avakoudjo, I. Gandaho, Euodias Djengue","doi":"10.5430/CRCP.V5N1P8","DOIUrl":"https://doi.org/10.5430/CRCP.V5N1P8","url":null,"abstract":"We reported a case of urethral adenocarcinoma in a 67-year-old woman who was admitted for difficulty voiding, irritative voiding symptoms, bloody urethral discharge and urinary incontinence. Physical examination was realised by gyneacologist and conclued to urological pathology. Urological examination was showed urethral ectropion which was bleeding. She had no fewer, no pelvic pain even less loss weight. Her general state was satisfactory. The lymph nodes of right side inguinal were felt. Computed imaging was showed involvement of right side bladder and bilateral iliac inguinal lymph nodes. Urethral ectropion biopsy anatomopathological screening was conclued to clear cell adenocarcinoma but CIS lesions were not found. The tumor staging was T3N2M0. A radiotherapy was perfomed. The patient was improvement of voiding symptoms and decrease lymph nodes size. Primary urethral carcinomas are rare pathology especially in the woman. Several histological types exist. However, chemotherapy and radiotherapy are very effective on very advanced stages. This cancer may be revealed by an urethral ectropion in a year-old woman.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"5 1","pages":"8"},"PeriodicalIF":0.0,"publicationDate":"2018-05-10","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V5N1P8","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"45864142","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
B. Vrugt, T. Frauenfelder, H. Dressel, F. Barresi, C. Clarenbach, Barbara K Kuhn
Pleuroparenchymal fibro-elastosis (PPFE) is a rare idiopathic interstitial pneumonia. Secondary forms of PPFE may occur in patients following lung- or bone marrow transplantation. Here we report a middle-aged woman who presented with dyspnea and cough. She had been working as a cleaning woman in an agriculture company and logistic firm. Chest HRCT was highly suggestive for PPFE. Histopathological examination of a surgical biopsy showed a pattern of PPFE with airway-centered fibro-elastosis, constrictive bronchiolitis and birefringent particles in the pathological areas only. Using transmission electron microscopy with X-ray diffraction (TEM/EDX) these particles were identified as quartz and silicates, both components generally found in soil. The present case illustrates that secondary PPFE may result from agricultural dust exposure and stresses the need for a careful workup of the occupational history.
{"title":"Secondary pleuroparenchymal fibro-elastosis with airway-centered fibro-elastosis and con-strictive bronchiolitis associated with agriculture dust exposure","authors":"B. Vrugt, T. Frauenfelder, H. Dressel, F. Barresi, C. Clarenbach, Barbara K Kuhn","doi":"10.5430/CRCP.V5N1P4","DOIUrl":"https://doi.org/10.5430/CRCP.V5N1P4","url":null,"abstract":"Pleuroparenchymal fibro-elastosis (PPFE) is a rare idiopathic interstitial pneumonia. Secondary forms of PPFE may occur in patients following lung- or bone marrow transplantation. Here we report a middle-aged woman who presented with dyspnea and cough. She had been working as a cleaning woman in an agriculture company and logistic firm. Chest HRCT was highly suggestive for PPFE. Histopathological examination of a surgical biopsy showed a pattern of PPFE with airway-centered fibro-elastosis, constrictive bronchiolitis and birefringent particles in the pathological areas only. Using transmission electron microscopy with X-ray diffraction (TEM/EDX) these particles were identified as quartz and silicates, both components generally found in soil. The present case illustrates that secondary PPFE may result from agricultural dust exposure and stresses the need for a careful workup of the occupational history.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"5 1","pages":"4"},"PeriodicalIF":0.0,"publicationDate":"2018-04-24","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V5N1P4","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"42001709","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Ameloblastomas account for 9%-11% of all odontogenic tumors and usually arise from the mandible. Peripherally arising ameloblastomas are exceedingly rare, comprising 1%-2% of all ameloblastomas. We present a case of peripheral ameloblastoma with malignant differentiation in a 72-year-old male patient. The patient underwent a biopsy of the left palatoglossal fold, and histological examination of the specimen demonstrated a well differentiated squamous cell carcinoma in situ with at least superficially invasive growth and features suggestive of peripheral ameloblastic carcinoma. The patient chose radiation therapy as opposed to surgical excision.
{"title":"Malignant peripheral ameloblastoma arising from the gingiva: A case report","authors":"J. Saleh, Laura Malone, J. Papadimitriou","doi":"10.5430/CRCP.V5N1P1","DOIUrl":"https://doi.org/10.5430/CRCP.V5N1P1","url":null,"abstract":"Ameloblastomas account for 9%-11% of all odontogenic tumors and usually arise from the mandible. Peripherally arising ameloblastomas are exceedingly rare, comprising 1%-2% of all ameloblastomas. We present a case of peripheral ameloblastoma with malignant differentiation in a 72-year-old male patient. The patient underwent a biopsy of the left palatoglossal fold, and histological examination of the specimen demonstrated a well differentiated squamous cell carcinoma in situ with at least superficially invasive growth and features suggestive of peripheral ameloblastic carcinoma. The patient chose radiation therapy as opposed to surgical excision.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"5 1","pages":"1"},"PeriodicalIF":0.0,"publicationDate":"2018-01-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V5N1P1","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43023519","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
A 65-year-old female presented to radiation oncology for potential treatment options due to metastatic pancreatic cancer and significant abdominal pain. Imaging demonstrated a large pancreatic mass with lymphadenopathy, vascular encasement, and liver metastases. She initiated palliative radiation treatment and developed persistent nausea and vomiting, as well as significant laboratory derangements. She was subsequently admitted and diagnosed with tumor lysis syndrome, though this diagnosis is usually an oncologic emergency seen with hematologic malignancies following chemotherapy.
{"title":"Tumor lysis syndrome following radiation therapy in metastatic pancreatic cancer: A case report","authors":"E. Song, Julian C. Hong, B. Czito","doi":"10.5430/CRCP.V4N4P22","DOIUrl":"https://doi.org/10.5430/CRCP.V4N4P22","url":null,"abstract":"A 65-year-old female presented to radiation oncology for potential treatment options due to metastatic pancreatic cancer and significant abdominal pain. Imaging demonstrated a large pancreatic mass with lymphadenopathy, vascular encasement, and liver metastases. She initiated palliative radiation treatment and developed persistent nausea and vomiting, as well as significant laboratory derangements. She was subsequently admitted and diagnosed with tumor lysis syndrome, though this diagnosis is usually an oncologic emergency seen with hematologic malignancies following chemotherapy.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"22-22"},"PeriodicalIF":0.0,"publicationDate":"2017-12-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V4N4P22","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43850555","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
A 58-year old male has a past medical history of a rectal low-grade mucinous adenocarcinoma with a KRAS codon 12/13 mutation (GGT>GAT), for which he received neoadjuvant chemotherapy and radiation. Five months after diagnosis, the patient underwent a low anterior resection showing persistent tumor with the pathological staging being (ypT3, ypN0, ypMx). Six months after surgery, follow-up PET scan showed two right upper lung nodules measuring 1.0 and 1.8 cm. Subsequent video-assisted thoracic surgery with a lung wedge resection revealed two tumors: a mucinous adenocarcinoma and an acinar-predominant adenocarcinoma. The mucinous adenocarcinoma showed similar cytologic features as the rectal tumor; however, it showed more of a lepidic pattern. The immunohictochemical profile of the lung mucinous adenocarcinoma was positive CK7 (cytoplasmic), negative CK20, TTF1, napsin-A, and CDX2. KRAS codon 12/13 analysis also showed a mutation in the lung, however it was different compared to the previous rectal tumor and showed a GGT>AGT mutation. The acinar adenocarcinoma had a wild-type KRAS. In this case, we favored a primary lung mucinous carcinoma over a metastasis from the rectum based on the lepidic morphology, CK20 negativity and differing KRAS codon 12 mutations. A year after the resection of the lung adenocarcinomas, the patient presented with dyspnea and abnormal liver functions. Abdominal US and MRI revealed multiple liver lesions (up to 1.9 cm). Core liver biopsies showed a poorly-differentiated mucinous adenocarcinoma with positive CK7 (cytoplasmic) and negative CK20, TTF-1, napsin-A, CDX2 and HSA. KRAS codon 12/13 analysis also showed a mutation GGT>AGT, similar to that of the lung mucinous adenocarcinoma. The immunohistochemical profile and the KRAS mutation sequence of the hepatic tumor suggests a metastasis from the lung primary and corroborates the earlier premise that the lung tumor is distinct from the rectal one. In conclusion, in the unusual circumstance of a lung mucinous adenocarcinoma in a patient with established gastrointestinal mucinous primary, KRAS mutation analysis sequencing could help distinguish whether the lung mucinous is a primary tumor or metastasis from colorectal origin.
{"title":"The utility of KRAS mutation analysis in differentiating a lung primary mucinous carcinoma from a metastatic colorectal mucinous carcinoma","authors":"Mahmoud L. Soliman, Shi Yang, John C. Lee","doi":"10.5430/CRCP.V4N4P17","DOIUrl":"https://doi.org/10.5430/CRCP.V4N4P17","url":null,"abstract":"A 58-year old male has a past medical history of a rectal low-grade mucinous adenocarcinoma with a KRAS codon 12/13 mutation (GGT>GAT), for which he received neoadjuvant chemotherapy and radiation. Five months after diagnosis, the patient underwent a low anterior resection showing persistent tumor with the pathological staging being (ypT3, ypN0, ypMx). Six months after surgery, follow-up PET scan showed two right upper lung nodules measuring 1.0 and 1.8 cm. Subsequent video-assisted thoracic surgery with a lung wedge resection revealed two tumors: a mucinous adenocarcinoma and an acinar-predominant adenocarcinoma. The mucinous adenocarcinoma showed similar cytologic features as the rectal tumor; however, it showed more of a lepidic pattern. The immunohictochemical profile of the lung mucinous adenocarcinoma was positive CK7 (cytoplasmic), negative CK20, TTF1, napsin-A, and CDX2. KRAS codon 12/13 analysis also showed a mutation in the lung, however it was different compared to the previous rectal tumor and showed a GGT>AGT mutation. The acinar adenocarcinoma had a wild-type KRAS. In this case, we favored a primary lung mucinous carcinoma over a metastasis from the rectum based on the lepidic morphology, CK20 negativity and differing KRAS codon 12 mutations. A year after the resection of the lung adenocarcinomas, the patient presented with dyspnea and abnormal liver functions. Abdominal US and MRI revealed multiple liver lesions (up to 1.9 cm). Core liver biopsies showed a poorly-differentiated mucinous adenocarcinoma with positive CK7 (cytoplasmic) and negative CK20, TTF-1, napsin-A, CDX2 and HSA. KRAS codon 12/13 analysis also showed a mutation GGT>AGT, similar to that of the lung mucinous adenocarcinoma. The immunohistochemical profile and the KRAS mutation sequence of the hepatic tumor suggests a metastasis from the lung primary and corroborates the earlier premise that the lung tumor is distinct from the rectal one. In conclusion, in the unusual circumstance of a lung mucinous adenocarcinoma in a patient with established gastrointestinal mucinous primary, KRAS mutation analysis sequencing could help distinguish whether the lung mucinous is a primary tumor or metastasis from colorectal origin.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"17"},"PeriodicalIF":0.0,"publicationDate":"2017-12-12","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V4N4P17","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"47344991","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Dartri Cahyawari, Eva Krishna Sutedja, Unwati Sugiri, H. Gunawan, O. Suwarsa
Pemphigus erythematosus is characterized by fragile vesicles or bullae, erosions, crusts, and scales in seborrheic area. There are several forms of atypical lesions such as erythematous papules and plaques, verrucous plaques, pustules, and lichenification. Here, we report an atypical pemphigus erythematosus with erythematous papules, plaques, and pustules skin lesions. A 52-year-old Indonesian man presented with prominent pruritic erythematous macules, papules, plaques on the scalp, trunk, and extremities, and also a pustule for each on the back and right arm. Clinically, the patient was diagnosed as small-plaque parapsoriasis, but histopathology examination on the pustule revealed a subcorneal acantholysis and direct immunofluorescence staining showed immunoglobulin G and complement C3 on the cell surface of keratinocytes. These result suitable for pemphigus erythematosus. The patient was treated with topical and systemic corticosteroid, and there were significant improvements in the skin lesions. Pemphigus erythematosus may present with prominent erythematous papules, plaques, and a few pustules. A careful assessment of the clinical manifestation and histopathological findings enabled us to make a correct diagnosis and succesfully treat the patient.
{"title":"An unusual case of pemphigus erythematosus","authors":"Dartri Cahyawari, Eva Krishna Sutedja, Unwati Sugiri, H. Gunawan, O. Suwarsa","doi":"10.5430/CRCP.V4N4P14","DOIUrl":"https://doi.org/10.5430/CRCP.V4N4P14","url":null,"abstract":"Pemphigus erythematosus is characterized by fragile vesicles or bullae, erosions, crusts, and scales in seborrheic area. There are several forms of atypical lesions such as erythematous papules and plaques, verrucous plaques, pustules, and lichenification. Here, we report an atypical pemphigus erythematosus with erythematous papules, plaques, and pustules skin lesions. A 52-year-old Indonesian man presented with prominent pruritic erythematous macules, papules, plaques on the scalp, trunk, and extremities, and also a pustule for each on the back and right arm. Clinically, the patient was diagnosed as small-plaque parapsoriasis, but histopathology examination on the pustule revealed a subcorneal acantholysis and direct immunofluorescence staining showed immunoglobulin G and complement C3 on the cell surface of keratinocytes. These result suitable for pemphigus erythematosus. The patient was treated with topical and systemic corticosteroid, and there were significant improvements in the skin lesions. Pemphigus erythematosus may present with prominent erythematous papules, plaques, and a few pustules. A careful assessment of the clinical manifestation and histopathological findings enabled us to make a correct diagnosis and succesfully treat the patient.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"14"},"PeriodicalIF":0.0,"publicationDate":"2017-12-11","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"43117683","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Leptospirosis is the most important global zoonosis and is caused by pathogenic spirochetes of the genus Leptospira . Human leptospirosis ranges in severity from a mild, self-limited febrile illness to a fulminant life-threatening one but their pathogenesis is still unclear. The extracellular release of the nuclear DNA of neutrophils, called NETs, upon activation by microbes is a pathogen-killing mechanism of neutrophils described in 2004 although its presence in human pathology have been observed only very recently. We report a case of fatal fulminant leptospirosis with associated severe pulmonary involvement and shown for the first time, evidence of the presence of NETs in the lung tissue.
{"title":"First observation of neutrophil extracellular traps in human leptospirosis","authors":"E. Scharrig, R. Drut, R. Gómez","doi":"10.5430/CRCP.V4N4P10","DOIUrl":"https://doi.org/10.5430/CRCP.V4N4P10","url":null,"abstract":"Leptospirosis is the most important global zoonosis and is caused by pathogenic spirochetes of the genus Leptospira . Human leptospirosis ranges in severity from a mild, self-limited febrile illness to a fulminant life-threatening one but their pathogenesis is still unclear. The extracellular release of the nuclear DNA of neutrophils, called NETs, upon activation by microbes is a pathogen-killing mechanism of neutrophils described in 2004 although its presence in human pathology have been observed only very recently. We report a case of fatal fulminant leptospirosis with associated severe pulmonary involvement and shown for the first time, evidence of the presence of NETs in the lung tissue.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"10"},"PeriodicalIF":0.0,"publicationDate":"2017-11-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V4N4P10","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"41767396","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Subcorneal pustular dermatosis (SPD) is a rare, chronic, and recurrent pustular eruption characterized histopathologically by subcorneal pustules that contain neutrophils. SPD has been clearly reported conjunction with other diseases. Leprosy reactions are acute inflammatory process that immunologically driven on the chronic course of leprosy. Erythema nodosum leprosum (ENL) is a type II of leprosy reaction putatively can initiate SPD lesions. We report one case of concomitant SPD and ENL in borderline lepromatous leprosy-relapses. A 41-year-old man with the history of using multidrug therapy-multibacillary for leprosy presented with painful erythematous nodules on the trunk and extremities, accompanied by pustules on erythematous base on the face, arms, buttocks, and legs. There were thickening of both ulnar nerves with gloves and stocking hypesthesia. The bacterial index was 3+ and morphological index was 20%. Histopathological examination on the pustule revealed subcorneal pustules with exocytosis of neutrophils which supported the diagnosis of SPD. A possible immunologic mechanism has been suggested in the induction of the occurence both SPD and ENL.
{"title":"A case of concomitant subcorneal pustular dermatosis and erythema nodosum leprosum in borderline lepromatous leprosy-relapses","authors":"H. Gunawan, Nina Roslina, O. Suwarsa","doi":"10.5430/CRCP.V4N4P6","DOIUrl":"https://doi.org/10.5430/CRCP.V4N4P6","url":null,"abstract":"Subcorneal pustular dermatosis (SPD) is a rare, chronic, and recurrent pustular eruption characterized histopathologically by subcorneal pustules that contain neutrophils. SPD has been clearly reported conjunction with other diseases. Leprosy reactions are acute inflammatory process that immunologically driven on the chronic course of leprosy. Erythema nodosum leprosum (ENL) is a type II of leprosy reaction putatively can initiate SPD lesions. We report one case of concomitant SPD and ENL in borderline lepromatous leprosy-relapses. A 41-year-old man with the history of using multidrug therapy-multibacillary for leprosy presented with painful erythematous nodules on the trunk and extremities, accompanied by pustules on erythematous base on the face, arms, buttocks, and legs. There were thickening of both ulnar nerves with gloves and stocking hypesthesia. The bacterial index was 3+ and morphological index was 20%. Histopathological examination on the pustule revealed subcorneal pustules with exocytosis of neutrophils which supported the diagnosis of SPD. A possible immunologic mechanism has been suggested in the induction of the occurence both SPD and ENL.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"6"},"PeriodicalIF":0.0,"publicationDate":"2017-11-14","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V4N4P6","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"46452628","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Cotyledonoid dissecting leiomyoma (CDL) is a rare variant of leiomyoma which shows a dissecting growth pattern of smooth muscle cells within the myometrium, with or without extra-uterine extension, and a macroscopic appearance resembling placenta. We present a unique case of CDL with both intravascular growth and presence of endometrial glands and stroma within the tumour. A 35-year-old female patient presented with menorrhagia. Ultrasound examination revealed a bulky anteverted uterus with a prolapsed submucosal leiomyoma and an intramural leiomyoma. In the interest of preserving fertility, she underwent myomectomy together with laparoscopic resections of the uterine wall deposit and myometrial tissue. The tumour was soft and pink, with a resemblance to placental tissue, and histology demonstrated nodules of neoplastic smooth muscle cells with areas of oedema and myxoid change dissecting into the surrounding myometrial tissue, which is consistent with the features of CDL. Also seen were non-neoplastic endometrial glands cuffed by stroma located within the smooth muscle tumour as well as in the non-neoplastic myometrium. There were foci of intravascular luminal growth confirmed by positive CD31 immunohistochemical staining of the endothelial cells lining the vascular lumina. The present case demonstrates that CDLs may have a unique histological appearance resembling adenomyoma and an intravascular growth pattern. It is important for pathologists, gynaecologists and radiologists to recognize such smooth muscle tumour variants and hence prevent misdiagnosis of malignancy and overtreatment.
{"title":"Cotyledonoid dissecting leiomyoma with intravascular growth pattern and intra-tumoural endometrial glands and stroma: A case report","authors":"Khurshid Merchant, B. Chern, S. Chew","doi":"10.5430/CRCP.V4N4P1","DOIUrl":"https://doi.org/10.5430/CRCP.V4N4P1","url":null,"abstract":"Cotyledonoid dissecting leiomyoma (CDL) is a rare variant of leiomyoma which shows a dissecting growth pattern of smooth muscle cells within the myometrium, with or without extra-uterine extension, and a macroscopic appearance resembling placenta. We present a unique case of CDL with both intravascular growth and presence of endometrial glands and stroma within the tumour. A 35-year-old female patient presented with menorrhagia. Ultrasound examination revealed a bulky anteverted uterus with a prolapsed submucosal leiomyoma and an intramural leiomyoma. In the interest of preserving fertility, she underwent myomectomy together with laparoscopic resections of the uterine wall deposit and myometrial tissue. The tumour was soft and pink, with a resemblance to placental tissue, and histology demonstrated nodules of neoplastic smooth muscle cells with areas of oedema and myxoid change dissecting into the surrounding myometrial tissue, which is consistent with the features of CDL. Also seen were non-neoplastic endometrial glands cuffed by stroma located within the smooth muscle tumour as well as in the non-neoplastic myometrium. There were foci of intravascular luminal growth confirmed by positive CD31 immunohistochemical staining of the endothelial cells lining the vascular lumina. The present case demonstrates that CDLs may have a unique histological appearance resembling adenomyoma and an intravascular growth pattern. It is important for pathologists, gynaecologists and radiologists to recognize such smooth muscle tumour variants and hence prevent misdiagnosis of malignancy and overtreatment.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"1"},"PeriodicalIF":0.0,"publicationDate":"2017-11-09","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V4N4P1","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"45177285","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Robert F. Stanley, M. Licata, A. Sinha, Yanhua Wang
Hemophagocytic lymphohistiocytosis (HLH) is a clinical syndrome that can be inherited or acquired. Herein, we report a case of HLH and pulmonary alveolar proteinosis (PAP) in the setting of lysinuric protein intolerance (LPI) in a male toddler who presented with prolonged fever, respiratory distress, and failure to thrive. On histologic examination, hemophagocytosis was observed in lymph node, bone marrow sections and aspirates. Lung wedge resection was consistent with PAP. LPI was confirmed with genetic sequencing which revealed compound heterozygous mutations in the SLC7A7 gene. LPI is a rare inborn error of metabolism and is not widely known beyond the pediatric group. Though the association of LPI with HLH has been previously described, we believe this is the first reported case of HLH and PAP associated LPI with histopathological correlation. Early recognition of HLH is critical to successful treatment and LPI should be considered in any young infant who presents with HLH- and PAP-related symptoms.
{"title":"Hemophagocytic lymphohistiocytosis and pulmonary alveolar proteinosis in a 13-month-old boy with lysinuric protein intolerance","authors":"Robert F. Stanley, M. Licata, A. Sinha, Yanhua Wang","doi":"10.5430/CRCP.V4N3P19","DOIUrl":"https://doi.org/10.5430/CRCP.V4N3P19","url":null,"abstract":"Hemophagocytic lymphohistiocytosis (HLH) is a clinical syndrome that can be inherited or acquired. Herein, we report a case of HLH and pulmonary alveolar proteinosis (PAP) in the setting of lysinuric protein intolerance (LPI) in a male toddler who presented with prolonged fever, respiratory distress, and failure to thrive. On histologic examination, hemophagocytosis was observed in lymph node, bone marrow sections and aspirates. Lung wedge resection was consistent with PAP. LPI was confirmed with genetic sequencing which revealed compound heterozygous mutations in the SLC7A7 gene. LPI is a rare inborn error of metabolism and is not widely known beyond the pediatric group. Though the association of LPI with HLH has been previously described, we believe this is the first reported case of HLH and PAP associated LPI with histopathological correlation. Early recognition of HLH is critical to successful treatment and LPI should be considered in any young infant who presents with HLH- and PAP-related symptoms.","PeriodicalId":90463,"journal":{"name":"Case reports in clinical pathology","volume":"4 1","pages":"19"},"PeriodicalIF":0.0,"publicationDate":"2017-09-29","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://sci-hub-pdf.com/10.5430/CRCP.V4N3P19","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"48079256","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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