We previously reported on two women with breast lesions in whom radiological examination could not exclude malignancy. In both cases, mastectomy was performed, and histological analyses revealed ...papillary lesions lined by fibrovascular stroma and nuclear inverse polarity. Hematoxylin–eosin, p63, and calponin staining indicated an absence of myoepithelial cells. However, it was concluded that the lesions had been non-malignant. These women have now been under long-term surveillance (74 months for one case and 62 months for the other) and have had no disease recurrence. Mucin (MUC)1, MUC2, MUC4, MUC5AC, MUC5B, and MUC6 immunostaining has also been performed in these women to investigate further whether their tumors were malignant or benign. In both cases, the tumors were only positive for MUC1 in apical luminal apical cells, as in normal breast tissue. MUC5B immunostaining, even when weak, can detect early breast cancer but was completely negative in our two cases. Therefore, both tumors were considered benign. Our findings in these cases suggest that nuclear inverse polarity papillary lesions lacking myoepithelial cells are benign. This knowledge should decrease the number of unnecessary operations performed for this tumor and their negative impact on patients’ quality of life.
We report a patient with sigmoid colon cancer who revealed a unique collision of hemorrhagic vascular adrenal cyst and adrenocortical adenoma with myelolipomatous changes. Two months before referral ...to our hospital, anticoagulant therapy was started for acute myocardial infarction. The components of the adrenocortical adenoma demonstrated a typical signal drop in opposed-phase magnetic resonance (MR) images although macroscopic fat was also depicted both on CT and MR images. The components of the vascular adrenal cyst demonstrated peripheral nodular enhancement with progressive enhancement on dynamic contrast-enhanced CT and a hemorrhagic change in the central region, which showed hyper intensity on T1-weighted images (T1WI) and hypo intensity on T2-weighted images (T2WI). Microscopically, the cyst was filled with foci of hemorrhage, fibrin, fibrosis, and hemosiderin. Furthermore, a white thrombus was found that corresponded to the central low signal intensity depicted on T2WI. Dilated vascular channels that were immunohistochemically positive for CD31 and CD34 were identified within the cyst. They were consistent with the pathological findings of hemorrhagic vascular adrenal cyst. Radiologists should be aware that the diagnosis of adrenal vascular cyst could be challenging since image findings may resemble hemangiomas, pheochromocytomas, and malignancy.
We herein report a 61-year-old man with diabetes and primary bilateral macronodular adrenal hyperplasia (PBMAH) associated with an ARMC5 variant who developed thyroid and pancreatic cancer. The ...patient was diagnosed with diabetes and obesity at 41 years old. At 48 years old, PBMAH complicated with subclinical Cushing syndrome was detected during hospitalization for diabetic ketoacidosis. Afterwards, the patient developed bilateral papillary thyroid cancer, so we suspected a genetic mutation. Pathogenic germline mutations in ARMC5, a major tumor suppressor gene detected in PBMAH, may play an important role in extra-adrenal tumorigenesis in PBMAH. In our patient, a heterozygous mutationc.1855C>T (p.R619*) of the gene was confirmed. The patient was treated with liraglutide and insulin, and hemoglobin A1c was maintained at 6 % to 7 %. Unfortunately, however, the patient died of pancreatic cancer at 61 years old. To our knowledge, this is the first report of a case of PBMAH and concomitant pancreatic cancer. Both PBMAH and diabetes are assumed to increase the risk of cancer. Furthermore, approximately half of patients with PBMAH develop glucose intolerance. Thus, strict screening for neoplastic diseases should be recommended in patients with PBMAH complicated with diabetes.
We report on a case of recurrent primary hyperparathyroidism possibly caused by parathyroid adenocarcinoma metastasizing to the lung. A 46-year-old woman with a history of parathyroid adenoma, which ...was extirpated 8 years ago, presented with symptoms of primary hyperparathyroidism, and was found to have a parathyroid and a lung nodule in radiographic assessments. Resections of the tumors in the parathyroid gland as well as the lung were required to improve her condition, and in pathology, both tumors demonstrated benign features consistent with adenoma. However, from the perspective of the clinical course and location of the tumors, we deduced that the tumors were malignant despite being identified as benign by conventional pathological examination. The integration of information based on clinical status and imaging studies is essential to evaluate the malignant potential of tumors if a patient with hyperparathyroidism has tumors located both inside and outside of the parathyroid gland.
Here, cases of a 68‐ (Case 1) and a 44‐year‐old (Case 2) female are presented. They had an abnormality in the breast, and came to our hospital for further examination and treatment. Radiologically, ...malignancy could not completely excluded so breast excision was performed. Histologically, both cases revealed papillary neoplastic lesions lined by fibrovascular core and nuclear inverse polarity without atypia. Loss of myoepithelial cells was observed by HE, p63, and calponin. Previous report indicate CK5/6, ER, p63 and MUC3 are important for distinguishing between papillary lesions according to the differential index (based on Allred score) of (ER total score + MUC3 total score)/(CK5/6 total score + p63 total score + 1). Based on this analysis, our two cases had benign lesions. However, based on immunopositivity for cell‐cycle marker Cyclin‐D1, Case 1 was negative, and Case 2 was about 70% positive. Additionally, the Ki‐67 index was <1% in both cases, and no evidence of disease was observed after a maximum 62 months of follow‐up in both cases, despite lack of additional treatment. Thus, we propose that lack of myoepithelial cells in papillary lesions do not necessarily indicate malignancy and are thought to be, at the most, uncertain malignant potential.
A 44-year-old man demonstrating cold-like symptoms from the end of August 2013 was referred to us with abnormal shadows on his chest radiograph in September 2013. Clinical examination revealed a ...painless red-colored mass on the sole of his left foot that had enlarged over the course of one year. The left leg showed marked non-pitting edema that had gradually progressed over a month. A chest CT revealed mediastinal lymphadenopathy and diffuse interstitial septal thickening, and lymphangitic carcinomatosis was suspected. A lung biopsy and left inguinal lymph node biopsy revealed the diagnosis of amelanotic melanoma with lung metastasis.
Metaplastic carcinoma accounts for only 1% of all breast cancer. Low-grade adenosquamous carcinoma (LGASCa) of the breast belongs to the family of metaplastic carcinomas. Here, we report on a case of ...LGASCa coexisting with sclerosing adenosis (SA). The patient was a 66-year-old woman. Eight years previously, she had an excisional breast biopsy in a hospital and was diagnosed with SA of her left breast. Three years ago, she presented to our hospital with deformity of the left nipple. We performed a core needle biopsy (CNB) and diagnosed SA. One year ago, a rebiopsy by CNB was performed because of the appearance of cysts in her left breast and worsening of the deformity of the left nipple. The diagnosis of the CNB was SA again. A third CNB was performed 4 months ago because the physical findings suggested malignancy; however, the CNB specimen was diagnosed as SA again. We decided to perform total mastectomy for the final diagnosis. Gross examination of the mastectomy material showed multiple cysts, the largest of which measured 3.3 × 2.2 × 2.0 cm. Histologically, the tumor was an irregularly shaped mass with duct proliferation and multiple cysts. The infiltrating tumor cells were scattered at the tumor periphery, and some infiltrating tumor cells resembled squamous cells. The tumor cells consisted of two types: one cell type was immunopositive for cytokeratin(CK)14 and p63, and the other cell type was immunopositive for CK14 and immunonegative for p63. Based on the histological and immunohistochemical findings, the diagnosis was LGASCa coexisting with SA and multiple cysts of the breast.
In this study, we report two cases of appendiceal diverticulitis with perforation. Case 1 was a 50-year-old man who visited our hospital because of epigastralgia and pyrexia. He had rebound ...tenderness in the right lower abdominal quadrant, as well as an increased inflammatory response observed upon biological examination. Computed tomography revealed an enlarged appendix. He was diagnosed with acute appendicitis, and emergency surgery was performed. Based on pathological examination, he was finally diagnosed with appendiceal diverticulitis with perforation. Case 2 was a woman in her twenties with a chief complaint of lower abdominal pain. She had consulted a nearby gynecologist when she first experienced the pain. She presented to our hospital the next day because her symptoms had not improved, even after taking analgesic drugs. There was muscular defense in the lower abdominal quadrant, as well as an increased inflammatory response. Emergency surgery was performed, and she was diagnosed with appendiceal diverticulitis with perforation based on pathological examination. Although appendiceal diverticulitis is rare, perforation has been reported to occur with it frequently. In addition to describing our two cases, we also provide a review of the literature.
症例は80歳の女性で，関節リウマチ（rheumatoid ...arthritis；RA）がありメトトレキサート（methotrexate；以下，MTXと略記）の内服を行っていた．入院4日前から大量帯下を認め，当院を受診した．血液検査では脱水所見，炎症反応の上昇，貧血を認めた．CTでは回腸子宮瘻を認め入院となった．脱水と貧血を補正し，抗菌薬治療を行った後，入院13日目に開腹小腸部分切除術と子宮全摘術を施行した．病理組織学的所見では，回腸と子宮の瘻孔部分に回腸から浸潤する比較的大型の類円形腫瘍細胞を認め，びまん性大細胞性Bリンパ腫（diffuse large B cell lymphoma；DLBCL）と診断した．悪性リンパ腫の原因としてMTXの関与を考え内服を中止し経過観察しているが，現在までに再発を認めていない．悪性リンパ腫による小腸子宮瘻は報告例がなく極めてまれな病態である．自験例のように薬剤誘発性の悪性リンパ腫を背景とすることがあり，小腸子宮瘻の診療の際には悪性リンパ腫を念頭に置き，既往や内服歴を十分に考慮して診断，治療にあたるべきと考えられた．
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