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Biomedical subjects

Masanobu Kumakiri

Publications and source records attributed to Masanobu Kumakiri.

18 recordsLinked to original sources

Primary cutaneous signet ring cell carcinoma expressing cytokeratin 20 immunoreactivity.

Primary cutaneous signet ring cell carcinoma (PCSRCC) is a very unusual but distinctive clinicopathologic entity that can simulate metastatic adenocarcinomas. It is defined as a diffuse malignant epithelial neoplasia localized in the dermis and subcutis without epidermal involvement, showing variable amounts of signet ring cells, without evidence of visceral adenocarcinoma. We present 2 cases of PCSRCC, which involved eyelids and axilla respectively. Despite thorough systemic workup, primary sources could not be demonstrated in either case. The tumor cells are positive for gross cystic disease fluid protein 15 in addition to a variety of glandular markers. Furthermore, both cases were immunostained with cytokeratin 20 (CK20). In conclusion, we report 2 cases of PCSRCC expressing CK20 immunoreactivity. CK20-positive primary cutaneous tumors should include PCSRCC in addition to Merkel cell carcinoma.

Aged↗

Rippled-pattern sebaceoma: a report of a lesion on the back with a review of the literature.

A 68-year-old Japanese man presented with a nodule that had been present for 5 to 6 years on the right side of the back. Physical examination revealed a dome-shaped, 12 x 13-mm, dark red nodule. It was excised with a 2 to 3-mm margin. The patient remained free of disease during 77 months of follow-up. Microscopic examination revealed a bulb-like tumor in the dermis, contiguous with the overlying epidermis. It was composed of small, monomorphous, cigar-shaped basaloid cells in linear, parallel rows, resembling the palisading of nuclei of Verocay bodies, and presenting a rippled-pattern. There were scattered cells showing sebaceous differentiation with vacuolated cytoplasm and scalloped nuclei. There were tiny, duct-like spaces. The tumor revealed characteristics of rippled-pattern sebaceoma. The present case is the first reported rippled-pattern sebaceous neoplasm on the back. Many spindle cell tumors, such as basal cell carcinoma, pleomorphic adenoma, dermatofibrosarcoma protuberans, myofibroblastoma, and leiomyoblastoma, in addition to trichoblastoma and sebaceoma, can have a rippled-pattern.

Aged↗

Papillary endothelial hyperplasia and dilated lymphatic vessels in bullous pilomatricoma.

This is a report of papillary endothelial hyperplasia in a 9-year-old girl with a pilomatricoma showing bullous appearance. Histologically, papillary proliferation of endothelial cells was found within dilated lymphatic endothelium-lined vascular channels overlying a pilomatricoma. The endothelial cells covering the papillae were of a lymphatic endothelial cell nature proved by immunohistochemistry and electron microscopy. Abundant fibrous long-spacing collagen was observed in the connective tissue and fibroblasts within papillae.

Child↗

Successful treatment of pyoderma gangrenosum that developed in a patient with myelodysplastic syndrome.

We describe the successful treatment of pyoderma gangrenosum (PG) that developed in a patient with myelodysplastic syndrome (MDS). A 63-year-old Japanese man with MDS was admitted to our hospital because of a large skin ulcer on his neck in November 2001. The initial diagnosis was infectious dermatitis, and antimicrobial therapy was performed, using imipenem/cilastatin, isepamicin, and amphotericin B. However, this therapy was not effective, and the lesion worsened. Cultures of blood, throat swab, and ulcer pus yielded no microorganisms. A biopsy of the skin lesion revealed a severe infiltration of neutrophils in the dermis, without any evidence of infection. The lesion was finally diagnosed as PG, and systemic administration of corticosteroid hormone was started in December 2001. The patient was initially pulsed with 1 g methylprednisolone daily for 3 days. The dose was immediately reduced, and the treatment was maintained with 30 mg prednisolone daily. The skin lesion responded markedly to the therapy, and C-reactive protein became negative. The patient was discharged in February 2002 because the lesion was almost cured. Prednisolone administration was tapered after 6-month maintenance therapy. No recurrence of PG was seen, although his MDS transformed into leukemia in April 2003. Only 31 cases of MDS developing PG have been reported in the past 20 years in Japan. This report describes one such rare patient who was successfully treated with the use of high-dose pulse methylprednisolone and long-term maintenance therapy.

Diagnosis, Differential↗

Epidermoid cyst with perforation of the skull.

A rare case of an epidermoid cyst associated with a skull defect is reported. The patient was a 59-year-old healthy woman from Japan who exhibited an elastic, soft, subcutaneous tumor on the left side of the parietal scalp for approximately 10 years without a history of trauma. On radiographic findings, a punched-out lesion was shown. Computed tomography of the cranium revealed a mass on the left parietal side that adhered to the cerebrum through the bone defect. Surgical excision resulted in remission. The tumor was peridurally located and showed no adhesion to the dura mater encephali. Histopathologically, a hybrid epidermoid cyst with no malignant features was seen. We hypothesized that long-term continuous pressure by the cyst resulted in a bone defect called scalloping.

Bone Diseases↗

Apocrine carcinoma of the vulva in a band-like arrangement with inflammatory and telangiectatic metastasis via local lymphatic channels.

BACKGROUND: Primary adenocarcinomas of the vulva have been classified as sweat gland carcinomas, extramammary Paget's disease, and primary breast carcinomas of the vulva. They share some common histopathologic features. METHODS: We describe a 72-year-old Japanese woman with apocrine carcinoma of the vulva and local lymphatic metastasis. RESULTS: The patient presented with a bruise on her inguinal area. Physical examination revealed a 4 cm x 7 cm, dark-red, irregularly elevated tumor on the left labium majora. Dome-shaped, flesh-colored, small papulovesicles were scattered on the abdomen, accompanied by erythema and induration. The lesion showed a band-like arrangement. General examination revealed multiple bone metastases, particularly in the spine. Microscopic examination revealed a moderately differentiated adenocarcinoma with signet ring cells. A few pagetoid clear cells were present in the hypertrophic epidermis. The peripheral papulovesicles demonstrated the same histopathologic view as in inflammatory and telangiectatic, metastatic breast carcinoma. Tumor cells were positive for various ductal and glandular markers. Estrogen and progesterone receptors were not expressed. Ultrastructural findings suggested differentiation towards apocrine or mammary glands because of the presence of an apocrine process and electron-dense mucous granules. The patient died in spite of combination chemotherapy and irradiation therapy. CONCLUSIONS: We report a rare case of apocrine carcinoma of the vulva in a band-like arrangement with local lymphatic metastasis which showed the clinical and histopathologic characteristics of inflammatory and telangiectatic carcinoma.

Adenocarcinoma↗

Linear acral pseudolymphomatous angiokeratoma of children (APACHE): further evidence that APACHE is a cutaneous pseudolymphoma.

Acral pseudolymphomatous angiokeratoma of children (APACHE) is a recently recognized, rare clinical entity. We report a case of APACHE in a 9-year-old Japanese girl with a dark red, linear plaque on the posterior area of the right lower leg. Histopathologic findings revealed further evidence that APACHE was a cutaneous pseudolymphoma rather than a vascular neoplasm because of the presence of distinct primary and secondary lymphoid follicles, and thick-walled, long blood vessels lined with prominent plump endothelial cells similar to high endothelial venules in the paracortical area of the lymph node. In addition to previous descriptions, the patient presented with a distinct linear lesion. Vacuolar alteration of the basal cell layer and scattered eosinophilic bodies in the epidermis were observed, which suggested a lichenoid tissue reaction. We believe that APACHE might show a distinct linear pattern accompanied by a lichenoid tissue reaction.

Angiokeratoma↗

Cutaneous marginal zone B-cell lymphoma: a case accompanied by massive plasmacytoid cells.

A 36-year-old man presented with a red nodule on his left shoulder. Histologically, there were variously sized, irregularly shaped nests throughout the dermis partly extending into the subcutaneous tissue. Masses of centrocyte-like cells were situated in the center of the tumor nests and accompanied by adjacent secondary follicle structures. Partial follicular colonization was seen. Massive plasmacytoid cells were located in the papillary dermis and the periphery of the tumor nests. Immunohistochemically, these centrocyte-like cells were positive for CD19 and alkaline phosphatase, and negative for CD5 and CD10. Cytoplasm of the plasmacytoid cells was positive for IgG and lambda-light chain, and negative for IgM, IgA, and kappa-light chain. Monotypic immunoglobulin staining including light chain restriction was shown. Clonal immunoglobulin heavy chain gene rearrangement by Southern blot analysis was shown in the tumor tissue. Morphologic, immunohistochemical, and molecular studies revealed that this patient had a cutaneous marginal zone B-cell lymphoma of mucosa-associated lymphoid tissue type. Electron beam (total 40 Gy) irradiation was applied. The tumor disappeared completely. Neither local recurrence or metastasis have appeared during 3 years of follow-up.

Adult↗

Dominant expression of CXCR3 is associated with induced expression of IP-10 at hapten-challenged sites of murine contact hypersensitivity: a possible role for interferon-gamma-producing CD8(+) T cells in IP-10 expression.

Murine contact hypersensitivity is elicited as a consequence of immunologic reactions triggered by skin-applied antigen, interactions among Langerhans cells, T cells, keratinocytes and mast cells, and a variety of chemokines generated by cellular interactions. In this study, we sensitized and challenged BALB/c mice with hapten, dinitrofluorobenzene or picryl chloride, and examined the expressions of mRNA for chemokines and their receptors by reverse transcription-polymerase chain reaction in the skin of elicited earlobes. CXC chemokines, IP-10 and Mig, were transcribed 24-48 h after challenge. This was associated with the expression of their agonistic receptor CXCR3, while mRNA for TARC and MDC, and their receptor CCR4 were not detected. Since CXCR3 and CCR4 are expressed preferentially on types 1 (Th1/Tc1) and 2 (Th2) T cells, respectively, the results suggested that the former type of T cells predominantly infiltrate at the elicited sites. Immune lymph node cells of the sensitized mice also expressed mRNA for CXCR3 but not CCR4 with concomitant transcription of interferon-gamma (IFN-gamma) but not interleukin-4 subsequent to challenge. The percentage of lymph node CD8(+) T cells was increased from 16% in naive mice to 30-50% in hapten-challenged mice, and in the immune lymph nodes, CD8(+) cells were the major source of IFN-gamma compared to CD4(+) cells. Since IFN-gamma is known to stimulate keratinocytes to produce IP-10 and Mig, it is suggested that these IFN-gamma-producing CD8(+) T cells enhance the production of these chemokines, thereby functioning as not only the effector cells but also the cytokine source to sustain the challenge reaction.

Animals↗

Spindle cell angiosarcoma following irradiation therapy for cervical carcinoma.

BACKGROUND: Angiosarcomas arise in the scalp and face in the elderly, in association with chronic lymphedema (Stewart-Treves syndrome), and in irradiated areas. Rarely in these settings, angiosarcomas exhibit pure spindle cell phenotype. METHODS: Herein, the clinicopathologic features of a 72-year-old-woman with spindle cell angiosarcoma are described. RESULTS: A 72-year-old woman presented with numerous nodules and diffuse induration from the lower abdomen to the right buttock, corresponding to the area exposed to 60Co-irradiation during treatment for cervical carcinoma 10 years earlier. Histopathological examination revealed inflitrative, atypical, spindle cells that labeled with antibodies to CD31, CD34, and factor VIII-related antigen. Ultrastructurally, these malignant spindle cells contained Weibel-Palade bodies. No features suggesting radiation dermatitis (sclerosis or bizarre, large fibroblasts) were identified, but lymphangiectases and widely spaced collagen bundles(lymphedema) were prominent in the skin surrounding the angiosarcoma. Computed tomographic scan of the abdomen highlighted this histologic finding by demonstrating tumor masses limited to areas of lymphedema. Treatment with intravenous and local injections of recombinant interleukin 2 (rIL 2) followed by electron beam irradiation were initially effective with tumor remission for 2 months. However, the recurrent tumor did not respond to a second course of one-shot injection of rIL 2 through the abdominal aorta and the patient succumbed to her angiosarcoma 19 months after diagnosis. CONCLUSIONS: Radiation-induced lymphedema may be a factor in angiosarcoma associated with radiotherapy.

Aged↗

Multiple eccrine spiradenomas on the hand, forearm and head.

The first case of multifocal eccrine spiradenomas on the hand, forearm and scalp is described. This case is unusual in that the tumors located on the little finger included the nail matrix and occurred in a linear/zosteriform distribution, resulting in a nail deformity. The nodules on the forearm and scalp were in a random distribution. Histologically, each tumor was highly cellular and composed of two cell types: small darkly staining basaloid cells and larger, pale cells. Ultrastructural observations often showed an intracellular lumen with numerous microvilli in larger pale-staining cells.

Adenoma, Sweat Gland↗