Chigusa Nagata

Epiglottic cyst in an infant.

Epiglottic cyst is a rare cause of stridor and respiratory distress in newborns and infants. A 2-year-old girl was referred to our department for the treatment of an epiglottic cyst causing inspiratory stridor. Flexible fiberoptic laryngoscopy and a computed tomography (CT) scan revealed a cystic lesion on the lingual surface of the epiglottis. Frequent episodes of sleep apnea accompanied by desaturation had been observed during her sleep. Endoscopic deroofing was performed under general anesthesia. After the operation, stridor and sleep apnea disappeared.

Increased expression of OCIA domain containing 2 during stepwise progression of ovarian mucinous tumor.

Ovarian cancer immunoreactive antigen domain containing 2 (OCIAD2) has been reported to show cancer‐specific expression in early invasive lung adenocarcinoma. OCIAD2 shows high homology with OCIAD1, which was originally immunoscreened from ascites of a patient with ovarian cancer and found to be a tumor‐specific protein. Therefore, like OCIAD1, OCIAD2 is expected to show high immunoreactivity in ovarian tumors. In this study, we examined the expression pattern of OCIAD2 in 117 ovarian mucinous tumors, and confirmed that it was more highly expressed in borderline tumor and carcinoma (51/74 cases, 69%) than in adenoma (6/43 cases, 14%). The immunoreactivity of OCIAD2 in borderline tumor and carcinoma was more specific than that of OCIAD1 (adenoma, 21/43 cases, 49%), and more sensitive than that of CEA (borderline tumor and carcinoma, 35/74 cases, 47%). Like OCIAD1, OCIAD2 is a cancer‐related protein and its expression level increases during the course of malignant progression and is thought to be a very useful marker for evaluating the malignancy of ovarian mucinous tumors.

Periodic acid Schiff staining‐positive spermatocytic seminoma

To the Editor: A spermatocytic seminoma is a relatively rare tumor that comprises 1–2% of all seminomas. It is completely different from a conventional seminoma in origin and affected patients are known to have a good clinical prognosis. Spermatocytic seminomas generally arise in patients at around the sixth decade of age. Periodic acid-Schiff (PAS) staining is generally negative, which is a key point for judgment. Herein we report a case of spermatocytic seminoma that was difficult to diagnose because of positive PAS staining and occurrence in a relatively young patient. A 32-year-old male underwent a high orchiectomy procedure for a right testis tumor. The excised tumor weight was 95.8 g and it measured 6.5 × 6.0 × 3.5 cm. Macroscopically, the tumor was multi-nodular and yellowish in color, and showed necrotic changes with a focal hemorrhagic area. The tumor was confined to the right testis. Microscopically, the tumor consisted of three types of cells, including small lymphocyte-like cells with dark stained nuclei and scant cytoplasm, intermediate-sized cells with a round nucleus, and giant cells that were monoor partly multinucleated. The latter type showed chromatin distribution with a filamentous or ‘spireme’ appearance, similar to that of meioticphase spermatocytes. The tumor cells were distributed in a solid nest or sheet-like pattern, and the lobulated structure was enclosed by fibrous septa. No lymphocytic infiltration or granulomatous reaction was seen (Fig. 1). Regarding PAS staining, the majority of the tumor cells had PAS positive structures in cytoplasm. We also performed an amylase digestion test and complete digestion was noted. Thus the PAS positive substance was determined to be glycogen, similar to a conventional seminoma (not shown). Immunostaining was done to more precisely investigate this case. Cytokeratin, placental alkaline phosphatase (PLAP), αfetoprotein (AFP), podoplanin, CD30, and octamer-binding transcription factor 3/4 (OCT3/4) were all negative, while c-kit staining was weakly positive. Even though the age of the patient was relatively young and PAS staining was positive, the case was finally diagnosed as spermatocytic seminoma based on morphological and immunostaining results. To confirm our diagnosis, we performed immunostaining for xeroderma pigmentosum type A (XPA), which is negative in a conventional seminoma and positive in a spermatocytic seminoma, and the tumor had positive staining. Ninth chromosomal amplification is specifically observed in spermatocytic seminomas. We performed a fluorescence in situ hybridization (FISH) examination to confirm that appearance in this case. Our results showed that amplification of the 9 chromosome was absent in a control seminoma specimen, whereas it was observed in the present tumor (Fig. 2). We encountered a rare spermatocytic seminoma tumor that was positive for PAS staining and occurred in a relatively young patient. Negative immunostaining for PLAP has been reported to be a good marker of a spermatocytic seminoma. In the present case, seminoma and embryonal carcinomas were denied based on negative findings for cytokeratin, PLAP, AFP, podoplanin, CD30, and OCT3/4. In particular, the sensitivity and specificity for OCT3/4, a Yamanaka factor thought to be an essential protein for proliferation of stem cells, are reported to be greater than 90% in embryonal carcinoma and seminoma cases. The present case showed a weak reaction to c-kit staining, though Kraggerud et al. reported that c-kit positivity was observed in 40% of their spermatocytic seminoma cases. Therefore, we were confident of our diagnosis of spermatocytic seminoma in the present patient based on the immunostaining results. Stoop et al. investigated proteins such as XPA by immunostaining to determine the origin of spermatocytic seminoma. The XPA gene is related to xeroderma pigmentosum type A and individuals in whom the protein is inactivated by mutations are known to have frequent occurrence of cutaneous cancers. That study found that XPA was predominantly expressed in pachytene spermatocytes. Moreover, the protein was shown to be expressed in all spermatocytic seminomas, though not in all seminomas. They concluded that spermatocytic seminomas originate from cells that have undergone greater differentiation as compared to a spermatogonium. We also investigated the XPA antibody in this case and the tumor was positive, as expected, while the control seminoma specimen was negative. It is known that excessive ninth chromosomal amplification is specifically observed in spermatocytic seminomas, thus we investigated that using a FISH method and confirmed amplification. In addition, our diagnosis of spermatocytic seminoma was confirmed by a genetic search using FISH. As for PAS staining, Talerman in 1980 was the first to report a spermatocytic seminoma negative for PAS. In most reviews presented since, this tumor is noted to be PAS negative. None have reported a case that was PAS positive. We considered the origins of spermatocytic seminoma and conventional seminoma as shown by their different reactions to PAS staining. A spermatocytic seminoma is considered to arise from germ cells that have matured to the stage of Pathology International 2014; 64: 192–194 doi:10.1111/pin.12150 bs_bs_banner

Intratubular trophoblasts in the contralateral testis caused elevation of serum human chorionic gonadotropin following complete remission of stage II testicular tumor: a case report.

We report the case of a 22-year-old male who had a history of metastatic right testicular tumor successfully treated with chemotherapy and surgery. Twenty-one months after the initial treatment, the serum human chorionic gonadotropin started to increase gradually, but whole body imaging including the left testis revealed no abnormal finding except testicular microlithiasis. A biopsy of the left testis revealed intratubular germ cell neoplasia, unclassified type. After the human chorionic gonadotropin level reached 6.6 mIU/ml, he underwent left high orchiectomy. Histology demonstrated a small malignant germ cell tumor as well as intratubular germ cell neoplasia, unclassified type, both of which were negative for human chorionic gonadotropin staining. Besides these lesions, there were tiny foci of human chorionic gonadotropin-immunoreactive intratubular trophoblasts. Serum human chorionic gonadotropin normalized immediately after the orchiectomy, and he had no sign of recurrence at 6 months. The present case will provide new insight into the diagnosis of testicular tumor recurrence with isolated elevation of a serum tumor marker.