Browsing by Author "Tellechea, O"
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- Benign follicular tumorsPublication . Tellechea, O; Cardoso, JC; Reis, JP; Ramos, L; Gameiro, AR; Coutinho, I; Baptista, APBenign follicular tumors comprise a large and heterogeneous group of neoplasms that share a common histogenesis and display morphological features resembling one or several portions of the normal hair follicle, or recapitulate part of its embryological development. Most cases present it as clinically nondescript single lesions and essentially of dermatological relevance. Occasionally, however, these lesions be multiple and represent a cutaneous marker of complex syndromes associated with an increased risk of visceral neoplasms. In this article, the authors present the microscopic structure of the normal hair follicle as a basis to understand the type and level of differentiation of the various follicular tumors. The main clinicopathological features and differential diagnosis of benign follicular tumors are then discussed, including dilated pore of Winer, pilar sheath acanthoma, trichoadenoma, trichilemmoma, infundibuloma, proliferating trichilemmal cyst/tumor, trichoblastoma and its variants, pilomatricoma, trichodiscoma/fibrofolliculoma, neurofollicular hamartoma and trichofolliculoma. In addition, the main syndromes presenting with multiple follicular tumors are also discussed, namely Cowden, Birt-Hogg-Dubé, Rombo and Bazex-Dupré-Christol syndromes, as well as multiple tumors of follicular infundibulum (infundibulomatosis) and multiple trichoepitheliomas. Although the diagnosis of follicular tumors relies on histological examination, we highlight the importance of their knowledge for the clinician, especially when in presence of patients with multiple lesions that may be the cutaneous marker of a cancer-prone syndrome. The dermatologist is therefore in a privileged position to recognize these lesions, which is extremely important to provide further propedeutic, appropriate referral and genetic counseling for these patients.
- Bowel-associated dermatosis-arthritis syndrome in an adolescent with short bowel syndromePublication . Pereira, E; Estanqueiro, P; Almeida, S; Ferreira, R; Tellechea, O; Salgado, MBowel-associated dermatosis-arthritis syndrome (BADAS) is a neutrophilic dermatosis, characterized by the occurrence of arthritis and skin lesions related to bowel disease with or without bowel bypass. We report an unusual case of BADAS in a 15-year-old white male with congenital aganglionosis of the colon and hypoganglionosis of the small intestine and multiple bowel surgeries in childhood complicated by short bowel syndrome. He presented with recurrent peripheral polyarthritis, tenosynovitis, and painful erythematous subcutaneous nodules located on the dorsolateral regions of the legs and on the dorsa of the feet. Histological examination disclosed a neutrophilic dermatosis confirming the diagnosis of BADAS.Although an uncommon disease, especially at pediatric age, it is important to evoke the diagnosis of BADAS in children and adolescents with bowel disease, because treatment options and prognosis are distinct from other rheumatologic conditions.
- Childhood hypopigmented mycosis fungoides: a commonly delayed diagnosisPublication . Gameiro, A; Gouveia, M; Tellechea, O; Moreno, APrimary cutaneous lymphomas (PCLs) are exceedingly rare in children and adolescents, with mycosis fungoides (MF) being the most frequent PCL diagnosed in childhood. There are numerous unusual clinical variants of MF, including the hypopigmented type form (HMF). HMF is exceptional overall, but comparatively common among children. We present an 8-year-old boy with a 3-year history of progressive, generalised, scaly, hypopigmented round patches and few erythematous papules. He was first diagnosed with pityriasis alba (PA), and moisturisers were prescribed with no improvement. Skin biopsy showed typical features of MF, and the patient was successfully treated with narrowband ultraviolet B. HMF may simulate atopic dermatitis, PA, pityriasis lichenoides, tinea versicolour, vitiligo, postinflammatory hypopigmentation or leprosy. Therefore, persistent and unusual hypopigmented lesions should be biopsied to rule out this rare variant of MF.
- Cutaneous aspergillosis in a heart-transplant patientPublication . Brinca, A; Serra, D; Brites, MM; Tellechea, O; Figueiredo, A
- Cutaneous pseudallescheria boydii infection in a renal transplant patient: A case reportPublication . Cardoso, JC; Serra, D; Cardoso, R; Reis, JP; Tellechea, O; Figueiredo, AWe describe the case of a 59-year-old male renal transplant recipient who presented with a 1 month history of nodular, erythematous, and crusted lesions on the anterior surface of the left leg. There was no history of trauma. The patient had no systemic signs or symptoms. A skin biopsy revealed evidence of a deep fungal infection, with septate and branching hyphae amongst the dermal inflammatory infiltrate, but the culture was negative. Initial treatment with itraconazole proved ineffective and new lesions appeared. New tissue samples were obtained for culture, allowing the isolation of Pseudallescheria boydii. The patient underwent surgical excision of the lesions combined with voriconazole 400 mg/day for 2 months. After this period no new lesions appeared and the patient has remained without recurrence after 2 years of follow-up. Pseudallescheria boydii is an opportunistic fungus that can cause systemic infection mainly in immunocompromised patients that manifests as pulmonary, osteoarticular, ocular, vascular, cutaneous or central nervous system disease. Resistance to therapy can occur with infection progression and high mortality. Treatment with the combination of an antifungal agent, mainly voriconazole, and surgery, when feasible, probably provide the best results in cutaneous infections.
- Familial Sneddon's syndromePublication . Mascarenhas, R; Santo, G; Ferro, MA; Tellechea, O; Gonçalo, Margarida; Figueiredo, AA syndrome associating Livedo Reticularis (LR) with Cerebrovascular disease (CVD) was described, in 1965, by Sneddon. It occurs sporadically, but a few familial cases of Sneddon's Syndrome (SS) have been reported, like these 3 cases that represent one of the largest number among siblings. We studied three male brothers, aged 28, 37 and 42 years, with CVD (ischaemic stroke in 2 patients and cerebral haemorrhages in the third) and their sister with no CVD. All patients presented with long lasting Livedo Reticularis, extending beyond the lower limbs. Skin biopsy on the centre of the reticular pattern showed, only in the second patient, partial endothelium detachment in dermo-hypodermic blood vessels. The males also had accesses of Livedoid Vasculitis (LV), in which a skin biopsy showed obliteration of several upper dermal vessels with hialin thrombi and a very scarce inflammatory infiltrate. Complementary studies, with an extensive investigation on pro-coagulation/pro-thrombotic features including antiphospholipid antibodies, were repeatedly negative. Their non-consanguineous parents were not affected, but among these kindred of 9 individuals, apart from the 4 patients reported above, LR and LV were present in two other brothers and also in an aunt and uncle, suggesting autosomal dominant pattern of inheritance, with incomplete penetrance. The relationship between Sneddon's Syndrome and Antiphospholipid Antibody Syndrome is controversial. The present cases, having repeatedly negative antiphospholipid antibodies, support the classification of Sneddon's Syndrome as an independent nosological entity.
- Fibrous histiocytomas: histopathologic review of 95 cases.Publication . Canelas, MM; Cardoso, JC; Andrade, PF; Reis, JP; Tellechea, OFibrous histiocytoma (FH) is a heterogeneous tumor composed of fibroblasts, histiocytes, and blood vessels. We conducted a retrospective histopathologic analysis of 95 biopsies, performed over the last 3.5 years, of fibrous histiocytomas to analyze the location, delimitation, epithelial changes, induction of folliculo-sebaceous structures, cellularity, vascularity, collagen pattern, and types of composite cells of the FH. In the majority of the biopsies, we confirmed the classical histopathologic features of fibrous histiocytomas. The presence of lymphoid nodules, mast cells, and sparse infiltrate of inflammatory cells was an interesting finding observed in our study.
- Florid cutaneous papillomatosis and acanthosis nigricans maligna revealing gastric adenocarcinomaPublication . Brinca, A; Cardoso, JC; Brites, MM; Tellechea, O; Figueiredo, AThis paper reports the case of a 57-year-old, previously healthy male with no systemic symptoms who over a short period of time developed multiple wart-like lesions on his trunk, limbs and face, typical lesions of acanthosis nigricans in the major body folds and tripe palms. Diagnostic tests revealed a metastatic gastric adenocarcinoma. Despite the implementation of therapy, which had a transient effect on the tumor and skin lesions, the patient died in 14 months. The association of these three paraneoplastic dermatoses (florid cutaneous papillomatosis, acanthosis nigricans maligna and tripe palms) in the same patient, apparently with a common pathogenic mechanism, is noteworthy.
- Hidradenite plantarPublication . Brites, MM; Tellechea, O; Poiares-Baptista, ATender, plantar nodules occurring in pediatric patients can be associated to different clinical entities, despite similar morphological features, therefore dermatopathology is the best technique for the definitive diagnosis. We describe the case for a 12-year-old patient with tender, red nodules on the left sole 3 days in duration, and with moderate functional disability. The onset of the lesions was preceded by low fever, and occurred 6 days after hepatitis B vaccination (Engerix B). The clinical diagnoses were juvenile plantar erythema nodosum, insect bites, pressure urticaria and plantar hidradenitis; Laboratory and radiographic studies, as well as microscopic examination of a skin biopsy specimen, were performed. The lesions resolved spontaneously in about 10 days, without recurrence after a year. A skin biopsy specimen revealed dense neutrophilic infiltrate surrounding and involving eccrine glans with abscess formation at the dermal-hypodermal junction. The diagnosis of idiopathic plantar hidradenitis was made. We discuss this entity, recently described by Stahr et al, and other erythematous plantar nodules in the pediatric age.
- Infundibulomatosis: A case report with immunohistochemical study and literature review.Publication . Cardoso, JC; Reis, JP; Figueiredo, P; Tellechea, OTumor of the follicular infundibulum was first described in 1961 by Mehregan and Butler in a patient presenting with multiple papules. It is more frequent, however, as an isolated lesion affecting mainly the face, neck, and upper trunk. Clinical presentation is variable, requiring histology for the diagnosis, which reveals typically a plate-like proliferation of keratinocytes in continuity with the epidermis and hair follicles; some morphological features are reminiscent of the outer root sheath of the hair follicle. A well defined network of elastic fibers surrounding the tumor is usually present using the appropriate staining and this finding is specific because it is not found in other benign follicular tumors. Multiple infundibulomas are usually sporadic and there is no apparent association with internal malignancy. The authors report the case of a 30-year-old female patient with a 5-year history of multiple small discrete hypopigmented macules and papules, scattered over the submental and submaxillary regions and anterior neck. Histopathological findings were consistent with the diagnosis of tumor of the follicular infundibulum. Immunohistochemical study was performed to further characterize the proliferation.
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