TWO DISTINCTIVE TYPES OF LIPID HISTIOCYTES APPEARING IN THE SPLEEN OF IDIOPATHIC THROMBOCYTOPENIC PURPURA — SEA-BLUE HISTIOCYTE AND FOAM CELL —

In 11 of 18 spleens surgically removed from patients with idiopathic thrombocytopenic purpura (ITP), the occurrence of sea-blue histiocytes or foam cells was observed. In a few cases, sea-blue histiocytes were found in the lymph nodes or bone marrow. Histochemically the sea-blue histiocytes were proved to contain ceroid or ceroid-like pigments in their cytoplasm. Ultrastructurally, numerous intracytoplasmic membrane-bound inclusions, mostly containing tightly laminated materials and intermingling electron-dense amorphous materials or osmiophilic lipid droplets, were present in these cells. In the foam cells, accumulation of excess amounts of phospholipids was histochemically demonstrated in frozen sections, often accompanied by deposition of cholesterol, particularly in advanced cases. Electron microscopically, vacuolar membrane-bound inclusions were found to predominate in their cytoplasm, most of which contained irregularly or loosely laminated materials. Transitonal cells between both types of cells were also found light -or electron microscopically and ultrastructural transition of sea-blue histiocytes to foam cells was traced. Enhanced phagocytosis of blood cells, including platelets, by sea-blue histiocytes, and gradual transition of the ingested blood cells to the inclusions were confirmed. ACTA PATH. JAP. 27 : 447–462, 1977.     

COMPOSITE GANGLIONEUROBLASTOMA — A Case Report and Short Term Culture of Tumor Cells —

A 1-year-old male infant showed cerebellar signs and symptoms, and excess urinary excretion of vanillylmandelic acid (VMA). Each of them disappeared spontaneously but a large abdominal tumor occurred at 3 years of age. Surgical specimen of the tumor was diagnosed as neuroblastoma and the patient died at 4 years of age. Autopsy revealed the huge tumor originated from the right adrenal, and occupying the retroperitoneum, pelvic cavity and posterior mediastinum. Histology of the primary lesion was chiefly that of ganglioneuroma, while all of the metastatic ones neuroblastoma. Transitional histology from neuroblastoma to ganglioneuroma was also observed in the primary lesion. The diagnosis was designated as composite ganglioneuroblastoma. The surgical material of the metastatic neuroblastoma was cultured for 2 months in vitro and the tumor cell clumps extended spontaneously abundant long neurites. This phenomenon suggests the high maturation ability of the neuroblastoma cells.     

CARCINOSARCOMA OF THE GALLBLADDER: Report of a Case and Review of the Literature

An extremely rare case of carcinosarcoma of the gallbladder is reported. The tumor presenting as a large, soft polypoid growth was microscopically characterized by an admixture of adenocarcinomatous and sarcomatous ele-ments, the latter containing specific heterologous mesenchymal elements such as osteoid, cartilage, and rhabdomyoblasts. The cytoplasm of neoplastic rhabdomyoblasts offered unequivocal cross striations and was strongly posi-tive for myoglobin, determined by the immunoperoxidase procedure. Metas-tatic tumors in the liver also exhibited a dual composition of adenocarcinoma and rhabdomyosarcoma. This appears to be the second documented case of this type of neoplasm with the rhabdomyosarcomatous component. The clinical settings and morbid prognosis were similar to events in cases of carcinoma of the gallbladder.     

LOBULAR CAPILLARY HEMANGIOMA OF THE ESOPHAGUS A Case Report and Review of the Literature

A case of lobular capillary hemangioma In the lower portion of the eosphagus in a 49-year-old woman was investigated. Lobular capillary hemangioma of the esophagus must be a rare lesion. We have been unable to find s single case in the literature. The excised lesion consisted macroscopically of a single strip of red tissue ulcerated and covered with a white exudate, 7 mm in length, with an attached vascular pedunculated lesion no more than 4 mm in diameter. Microscopically, the pedunculated lesion had the features of a capillary hemangioma with a circumscribed aggregate of capillaries arranged In one or more lobules. Similar cases from the oral and nasal mucous membranes were recorded In 1980 by M ills et al .¹³.     

SQUAMOUS CELL CARCINOMA OF THE URACHUS A Case Report and Review of Literature

A rare case of squamous cell carcinoma (SCC) is reported. A cystic tumor, measuring 4X4 cm was surgically removed from the supravesical portion in a 77-yearold woman. Macroscopically, it had a comunicating canal to the urinary bladder. Microscopically, well differentiated squamous cell carcinoma with pearl formation was noted. An aspiration biopsy was helpful for the histopathological diagnosis at operation. The patient developed purulent peritonitis because of rupture of the tumor which was vulnerable to infection. Six documented cases and the present case were reviewed. The tumors were classified into two types on morphological features: supravesical type and intramural type. Site and local extension of the tumor accounted for clinical features of each type. The authors considered that SCC cells were derived from totipotential epithelial lining of the urachal remnant.     

XANTHOGRANULOMA OF THE ORAL CAVITY IN ADULT A Case Report and Review of the Literature

A case of xanthogranuloma arising in the palatal gingiva of the maxilla in a 38-year-old Japanese male was investigated. Histopathological findings of the excised lesion were indistinguishable from the xanthogranulomas in infants and children (juvenile xanthogranuloma), and like those in the young, the present case was not associated with abnormalities of serum lipids and was not associated with other lesions. A review of the English literature yielded only two histopathologically documented cases of xanthogranuloma in the oral cavity, arising in young patients. A case of xanthogranuloma of oral cavity in an adult was not documented previously. ACTA PATHOL. JPN. 36: 1565-1570, 1986.     

HIGH-GRADE SURFACE OSTEOSARCOMA OF THE LEFT ILIUM A Case Report and Review of the Literature

A rare case of high-grade surface osteosarcoma of the left ilum is reported. Trephine biopsy performed on a 31-year-old woman suffering from a huge tumor of the left buttock revealed high-grade osteosarcoma, and hemipel-vectomy was undertaken. The surgical specimen showed that the tumor was present on the surface of the left ilium. Ten months after the operation, the patient died of brain metastasis. From these results, we considered this case to be one of high-grade surface osteosarcoma. ACTA PATHOL JPN 38: 235 -240, 1988.     

ADAMANTINOMA OF THE TIBIA A Case Report and a Statistical Review of Reported Cases

A case of adamantinoma of the tibia is reported. A 70-year-old man complained of a painful swelling over the lower front of the right tibia. Roentgenograms showed an oval well-circumscribed, multilocular cystic lesion. Curettage and bone grafting were performed, and 17 months later the patient is in good condition. Microscopically the tumor tissue consisted mainly of epithelial Islands with a palisading at the periphery and a loose reticular pattern in the center of the tumor cell nests, showing a close resemblance to the ameloblastoma of the jaw bones. Electron micrographs revealed the epithelial nature of the tumor cells, i.e., a continuous basal lamina, desmosomes and bundles of microfilaments, probably tonofilaments. One hundred fifty six cases of this peculiar primary skeletal bone tumor previously reported in the literature were reviewed and discussed.     

KAPOSI'S SARCOMA–Case Report and Review of Japanese Cases–

A case of Kaposi's sarcoma in a 60 year-old male was examined histologically and electron microscopically. Multiple hemorrhagic skin lesions were confined within the scalp. The major part of initial skin biopsies ekhibited capillary hemangiomatous component, but examination of surgical materials confirmed its nature as hemorrhagic sarcoma. At autopsy, the skin lesions showed various histological pictures ranging from angiomatous or granulomatous to sarcomatous proliferation of spindle-shaped cells. Of intriguing findings were the bone marrow involvement and the pulmonary changes, the former consisting of typical sarcomatous lesion and the latter being characterized by purpuric appearance of the pleura responsible for the terminal pneumohemothorax. Electron microscopically, the tumor cells showed Weibel-Palade granules in the cytoplasm suggesting the endothelial nature. In this article, Japanese autopsy cases of Kaposi's sarcoma so far reported are summarized.     

LEIOMYOSARCOMA OF THE SPERMATIC CORD: A Case Report and a Brief Review of Literature

A case of leiomyosarcoma of the spermatic cord, occurring in a 79-year-old male patient, is reported. The tumor was resected with high inguinal ligation and orchiectomy. It was located at the scrotal part of the spermatic cord on the left side and was shown to have originated from the spermatic duct using the dissecting method. Histologlcally, it was identical to leiomyosarcoma showing a typical morphological appearance of smooth muscle cell tumor in the conventional histochemical preparations as well as a focal positive reaction to antidesmin antibody, a marked nuclear pleomorphism, and abundant mitotic figures. The patient had been untroubled for six months after the resection of the tumor. Leiomyosarcoma arising in spermatic cord is quite rare.     

CLOVERLEAF SKULL SYNDROME An Autopsy Case and Review of Literatures

An autopsy case of cloverleaf skull deformity associated with hydrocephalus, systemic skeletal malformation including facial dysostosis, fused elbow, syndactyly of the toes, odd digits and striking anomaly of tracheal cartilage is presented. Extra-skeletal abnormalities included covered anus, dermal sinus and the absence of corpus callosum and the septum pellucidum. Severe basal skull deformity appeared to be a primary morphologic alteration associated with premature closure of the specific sutures, which terminally resulted in life-threatening hydrocephalus. Histological investigation showed the abnormalities of endochondral ossification in the cartilage at the epi- and meta-physis of the fused elbows. In addition, electron microscopical study revealed unusual fat droplet-containing chondrocytes even in the resting and multiplicative phase. Association of the tracheal anomaly with this syndrome was disclosed in this case. Generalized bone and cartilage abnormalities not only in the skeletal system but also in the internal organs strongly suggest that this disorder involves generalized osteocartilagenous system. The present case makes a total of 14 cases of the cloverleaf skull syndrome reported in the Japanese literatures to date. Major clinical and pathologic findings of these cases were summerized.     

Non-Paraneoplastic Autoimmune Retinopathy: The First Case Report in Korea

Autoimmune retinopathy (AIR) is an immune-mediated retinopathy, resulting from an immunologic process caused by the aberrant recognition of retinal antigens as autoantigens. The diagnosis of AIR involves the detection of antiretinal antibodies with concurrent clinical and electrophysiological evidence of retinopathy. A 40-year-old patient presented with progressive loss of bilateral vision over several months. A fundus examination was unremarkable. Spectral domain optical coherence tomography revealed a blurred photoreceptor ellipsoid zone at the subfoveal region in both eyes with more prominent disruption in the left eye. Fullfield electroretinography (ERG) showed relatively normal rod and cone responses in the right eye, and decreased photopic bwaves with minimal attenuation of a-waves in the left eye. Multifocal ERG demonstrated slightly reduced amplitude of the inner segment ring in the right eye and decreased amplitudes and delayed latencies of all modalities in the left eye. The patient was suspected to have AIR and it was supported by positive Western blots for 23-kDa protein, enolase (46-kDa), aldolase (40-kDa), 62-kDa and 78-kDa proteins and by immunohistochemical staining of human retinal bipolar and ganglion cells. Despite the immunosuppressive treatment, the destruction of the retinal photoreceptors progressed, and immunosuppressive interventions produced very little visual improvement. We report on what is, to the best of our knowledge, the very first case of serologically confirmed nonparaneoplastic AIR in Korea.     

LIPOMA OF THE SUPERIOR MEDULLARY VELUM WITH SCHWANN CELLS:Report of a Case and Review of the Literature

A large intracranial lipoma, located on the superior medullary velum Is reported. Histologically, the tumor consisted of adult adipose tissue. The right trochlear nerve was involved within the tumor, and the scattered nests of Schwann cells and some larger vessels were also present. Lipoma of the dorsal region of the mid-brain is rare and has not been reported from Japan. Schwann cell proliferation in the lipoma is another unusual feature in this case. The histogenesis of this tumor is discussed, and the literature on lipomas of the dorsal region of the mid-brain is reviewed.     

SCANNING ELECTRON MICROSCOPY OF THE ISCHAEMIC KIDNEY — Changes in the Surface Microstructure of Glomerular Epithelial Cells —

Domestic rabbits were used to study the changes that occur during ischaemic conditions in the kidney. With a scanning electron microscope, the microcellular changes at the surface of glomerular epithelial cells were observed regularly from the onset of ischaemia until five hours later. After one hour of ischaemla, the surface of the glomerular epithelial cells showed mild swelling and some change in its smooth appearance. One part had taken on a sponge-like appearance. After 2.5 hours of ischaemia, the epithelial cells had atrophied and the entire cell surface was clearly sponge-like. It was no longer possible to distinguish the small pore-like structures that had been seen on the cell surface in the normal. Moreover, 2.5 hours of ischaemia was the time when, biochemically, cell metabolism had completely ceased and the permeability of the cell membrane had altered. The cell was considered to have undergone irreversible change by this stage. After 5 hours of ischaemia, cells were markedly atrophied and the appearance of the surface had become even more sponge-like.     

Subacute Thyroiditis After Receiving the mRNA COVID-19 Vaccine (Moderna): The First Case Report and Literature Review in Korea

Subacute thyroiditis (SAT) is a painful thyroiditis that often requires steroid therapy. Here, we report the first case of severe SAT in a patient who received the first dose of mRNA coronavirus disease 2019 (COVID-19) vaccination. A 34-year-old man without a viral prodrome felt a lump when swallowing 5 days after his first dose of mRNA-1273 (Moderna) vaccination. Ten days after vaccination, the patient visited the hospital and was advised to rest and take nonsteroidal anti-inflammatory drugs. He revisited the hospital 10 days later as symptoms aggravated with anterior neck pain, headache, fatigue, muscle weakness, and weight loss. Thyroid hormone levels and inflammatory markers were consistent with thyrotoxicosis. A thyroid ultrasound scan revealed typical SAT findings. His symptoms rapidly improved after receiving prednisone. A week later, the patient successfully completed his second dose of the vaccine. The thyroid function test results were nearly normal 1 month after the completion of the vaccination. We report this case to raise awareness of the occurrence of SAT after COVID-19 vaccination. As the risk of COVID-19 outweighs the minor risks of the vaccine, managing the side effects of the first vaccine dose is crucial to complete COVID-19 vaccination.     

A CASE OF OVARIAN GRANULOSA CELL TUMOR WITH INVASIVE ADENOCARCINOMA OF THE ENDOMETRIUM. A Case Report and Review of the Japanese Literature

A 47-year-old woman was admitted complaining of irregular uterine bleeding. On surgery, an ovarian tumor was found together with a broad-based polypoid endometrial mass. These lesions were histologically diagnosed as granulosa cell tumor with invasive adenocarcinoma of the endometrium. To our knowledge, only four other cases of granulosa cell tumor with adenocarcinoma of the endometrium have been reported in Japan, although our case was unique in showing invasion to the uterine myometrium. ACTA PATHOL JPN 38: 947∼951, 1988.     

Crinophagy in Neuroblastoma: A Case Report and Review of the Literature

Abstract

Crinophagy is a well-described ultraphysiological phenomenon encountered in a variety of cells and tissues. This process reflects a form of autophagy in which degradation of excess or nonfunctional cellular constituents occurs, specifically of neuroendocrine granules. The diagnostic ultrastructural features are the identification of neuroendocrine granules within lysosomes, often encased in or accompanied by myelin bodies. An impressive variety of neuroendocrine/secretory cells and tumors have demonstrated crinophagy from the neuroendocrine cells of the pancreas, small bowel, prostate, and urinary tract. To our knowledge, however, crinophagy has not been previously described in neuroblastoma, despite the fact that these tumors characteristically produce neuroendocrine granules in abundance. This case further supports the idea that crinophagy represents a common ultrastructural mechanism for the disposal and degradation of excess neuroendocrine granules.     

CLEAR CELL ADENOCARCINOMA OF THE FEMALE URETHRA A Case Report

A case of urethral clear cell adenocarcinoma (mesonephric carcinoma) in a 62-year-old woman is reported. The patient consulted our hospital because of acute urinary retention. Fine needle aspiration cytology showed a few atypical cells; loose cluster cells with finely vacuolated cytoplasm, large nuclei and prominent nucleoli, and small cluster cells with finely vacuolated, delicate cytoplasm, small pale nuclei and small nucleoli. Cytologic diagnosis was adenocarcinoma and no diagnosis of clear cell adenocarcinoma was obtained. The neoplasia was localized at the posterior wall of the urethra and deep muscular layer of the vaginal wall. Histologically, the tumor revealed mainly small, elongated glands consisting of single-layered cuboidal or columnar cells with scanty cytoplasm and a focally hobnail appearance. In addition, a small cystic lesion resembling nephrogenic adenoma was observed. This finding raises the possibitily that clear cell adenocarcinoma may be a malignant counterpart of nephrogenic adenoma. ACTA PATHOL JPN 38: 217–223, 1988.