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Fırat Üniversitesi Sağlık Bilimleri Tıp Dergisi
2022, Cilt 36, Sayı 3, Sayfa(lar) 248-252
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Ekrin Diferansiyasyonu Olan Adneksiyal Tümörlerin Gelişimi: Literatür Taramasında
Hilal BALTA1, Şenay ERDOĞAN DURMUŞ2, İlknur ÇALIK1, Sevilay ÖZMEN3, Ebru ŞENER3, Ali KURT4, Mehmet Eşref KABALAR4, Nevin KOCAMAN5
1 Firat University, Medical School, Pathology Department, Elazig, TURKEY
2Basaksehir Cam Sakura Hospital, Department of Pathology, Department of Cytopathology Istanbul TURKIYE
3Ataturk University, Medical School, Pathology Department, Erzurum, TURKIYE
4Erzurum Regional Training and Research Hospital, Pathology Department, Erzurum, TURKIYE
5Firat University, Medical School, Department of Histology and Embryology, Elazig, TURKIYE
Anahtar Kelimeler: Deri, Hasta, Adneksiyal tümörler, Ekrin farklılaşması, Literatür
Özet
Amaç: Geniş bir vaka grubu ile kutanöz ekrin tip adneksiyal tümörlerin hasta özelliklerini, sıklığını, lokalizasyonunu ve alt tip sınıflamasını literatüre katkı sağlamak amacıyla incelemeyi amaçladık. Deri eki neoplazmaları, farklı yönlerde farklılaşan nadir tümörlerdir. Ekrin tip deri adneksiyal tümörler hakkında bilgi veren çok az çalışma vardır ve seri sayısı sınırlıdır. Pratikte nadiren görüldükleri için tanı ve ayrımda zorluklara neden olabilirler.

Gereç ve Yöntem: Erzurum bölge eğitim ve araştırma hastanesinde 2009-2017 yılları arasında histopatolojik olarak ekrin diferansiyasyonlu adneksiyal tümör tanısı alan 124 hasta retrospektif olarak incelendi.

Bulgular: Bölümümüzde çalışma süresi boyunca ekrin diferansiyasyonlu toplam 124 deri eki neoplazmı teşhis edildi. Hastaların ortalama yaşı 50.15 idi. Erkeklerde tümörler kadınlara göre daha sık gözlendi. Tümörlerin %92,75'i iyi huyluydu ve çoğu yüz ve gövdeye lokalizeydi.

Sonuç: Adneksiyal deri tümörleri ile ilgili farklı histolojik paternlere sahip hastalar üzerinde incelemeler yapılarak literatüre yeni bilgiler kazandırılmıştır.

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    Skin adnexal neoplasms are rare tumors thought to originate from undifferentiated stem cells rather than mature cells due to their heterogeneous nature 1-4. Because they are rarely seen in practice, they can cause difficulties in diagnosis and distinction 1-6. According to their differentiation, they are classified as hair follicle, sebaceous, eccrine and apocrine tumors 1-11. While benign ones are common, malignant ones are rare 2,6-8,10.

    Recent morphological parameters, ultrastructural and immunohistochemical data, have been put forward, suggesting that some changes have to be made in older classifications 2,4,6,9,11,12. Finally, for this purpose, a new classification considering the embryological development of skin appendages has been proposed and divided into two main groups as skin adnexal tumors, folliculo sebaseous -apocrine units and eccrine tumors 2-10. Lesions usually appear as papules or nodules 1,2. There are very few studies that provide information about eccrine type skin adnexal tumors and their number of series are limited. In this retrospective study, it was aimed to examine the patient characteristics, frequencies, localizations and subtype classifications of cutaneous eccrine type adnexal tumors in the archive of Erzurum regional training and research hospital Pathology Department in order to contribute to the literature with a large case group.

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    We reviewed 124 patients that diagnosed as adnexal tumors with eccrine differentiation histopathologically in Erzurum region training and research hospital between 2009- 2017, retrospectively. The data such as patients’ characteristics such as age, gender, site of lesions, lesions characteristics, histopathological features and subtypes collected from patients’ pathology reports. The results were categorized according to WHO 2010 (Table 1).


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    Table 1: Classification of skin adnexal tumors according to WHO 2010 classification

    Detailed microscopic examination was carried out. Data analysis performed using the SPSS 20.0 program. Descriptive statistics for the evaluation of results have shown in the form of mean, the nominal variables have shown as the number of cases and (%).

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    A total of 124 neoplasms of skin appendages with eccrine differantiation were diagnosed during the study period at our department. Patients’ mean age was 50.15 years (age range: 6–92 years). Tumors were observed more common in males as compared to females with male to female ratio of 71:53 (Table 2 and Table 3). 92.75 % of tumors were benign. Incidence of malignancy was low (7.25 %) (Table 4). The frequency of eccrine tumors and relationship with gender were shown in Table 3. Most of the tumors were localized to face and trunk region (62.9%), followed by extremities and head (37.09 %) (Table 5, Figure 1). Eccrine hydrocystoma were seen in scrotum and conjuctiva, as rare localizations. In only one patient syringoma localized in labia major which is an extremely rare area. The sizes of lesions were ranged of 0.1 cm-12 cm. Eccrine hydrocystomas were usually in milimetric sizes while porocarsinomas were observed in larger measures, such as 2.5cm, 12 cm. The mean age of our malign cases was 57.7 and the female: male rate was 5/4.


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    Table 2: Gender distribution of ecrine type adnexal tumors


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    Table 3: Distribution of tumour type in relation to sex


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    Table 4: Distribution of tumors according to their behavior


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    Table 5: Characterization of tumour with according to anatomical site


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    Figure 1: Histopathology was seen showing lobules of epithelial cells arranged in a puzzle or mosaic pattern. The distinctive red basement membrane-like structure surrounds the tumor lobules. Each lobule shows a peripheral lining by dark basaloid cells and a larger and paler area of cells suggesting the dermal cylinder in the case of cylindiroma (Figure 1A). The case of nodular hydradenoma had histopathology showing a well-encapsulated mass with large cystic spaces inside. Note the granular and slightly eosinophilic cytoplasm surrounding a dark nucleus. Ductal luminal structures can be appreciated (Figure 1B). In the case of eccrine syringofibroadenoma, thin anastomotic reticulated cords extending from the basal layer of the epidermis to the dermis and basaloid monomorphic cuboidal cell lines were observed. Tumor cells are slightly smaller than neighboring keratinocytes (Figure 1C). High-grade tumors (porocarcinoma) may show areas of cystic degeneration with focal necrosis as shown above. Viable mitotic activity is associated with a worse prognosis (Figure 1D).

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    Cutaneous adnexal tumors were first recognised in the later part of nineteenth century. They are relatively uncommon and are thought to have a genetic basis. Diagnosis is essentially based on histopathological examination because clinical features are not very distinctive. They may sometimes display more than one histological differentiation resembling other wide variety of tumors. The frequency of skin eccrine type adnexal tumors in the literature are variable 1-8. For example, in a work done in India, it was found that there was 18/40 (45%) eccrine differentiation skin adnexal tumor and in another study in India, 35/70 (50%) eccrine type skin adnexal tumors were found 1,2. In other studies, skin adnexal tumors with eccrine differantiation frequency was found as 20/39 (51.2%), 161/1016 (15.8%), 19/36 (52.7%), 33/52 (63.4%), 8/21(38.09 %) (4, 6-8, 10). In the study of literature, nodular hidradenomas were the most common eccrine tumors 2,4,6-8. In our study, 124 eccrine type skin adnexal tumors were found in 320,000 pathology samples (Table 3). Amongst the eccrine types, nodular hidradenoma was the most common neoplasm similar to literature. While 34 out of 124 (27.4%) cases were diagnosed as nodular hidradenoma, eccrine syringofibroadenoma was rare. (Fig. 1B,1C). In some studies, sebaceous type skin adnexal tumors, eccrine hydrocystomas and eccrine spiradenomas were frequent 1,11,12.

    The mean age of eccrine type skin adnexal tumors in the literature was 62, 29, 34.5, 33 and 35.5 4-6,8,10. In our study, patients’ mean age was 50.15 years (age range: 6–92 years) and this is this is higher than some studies. In the literature, female: male ratios for these tumors were variable 1,2,4-6,8-10,12. Differently in our study there was a male predominance with the male: female ratio 71:53 (Table 2). The most common localizations in the studies were head, neck, face and body 1-12. In our study extremity, neck, back and head areas were more frequent, respectively (Table 5). In the literature, the sizes of eccrine type skin adnexal tumors are also variable. In a study it has been reported between 1.1 cm and 2 cm. In a different study 5 cm sizes were reported but most of the lesions are smaller than 3 cm 4,10. In our study the size of eccrine type skin adnexal tumors were between 0,1 cm and 12 cm and especially in porocarcinoma the sizes was much larger (2.5 and 12 cm). In literature, the incidence of eccrine type malignant skin adnexal tumors ranged from 4.2% to 25% 1,2,4,6-9,11-13. In our study, it was 7.25% (Fig. 1D) (Table 4). In some studies, sebaceous type skin adnexal tumors, eccrine hydrocystomas and eccrine spiradenomas were frequent 1,11,12.

    The mean age of eccrine type skin adnexal tumors in the literature was 62, 29, 34.5, 33 and 35.5 4-6,8,10. In our study, patients’ mean age was 50.15 years (age range: 6–92 years) and this is this is higher than some studies. In the literature, female: male ratios for these tumors were variable 1,2,4-6,8-10,12. Differently in our study there was a male predominance with the male: female ratio 71:53. The most common localizations in the studies were head, neck, face and body 1-12. In our study extremity, neck, back and head areas were more frequent, respectively. In the literature, the sizes of eccrine type skin adnexal tumors are also variable. In a study it has been reported between 1.1 cm and 2 cm. In a different study 5 cm sizes were reported but most of the lesions are smaller than 3 cm 4,10. In our study the size of eccrine type skin adnexal tumors were between 0,1 cm and 12 cm and especially in porocarcinoma the sizes was much larger (2.5 and 12 cm). In literature, the incidence of eccrine type malignant skin adnexal tumors ranged from 4.2% to 25% 1,2,4,6-9,11-13. In our study, it was 7.25.

    As a result, adnexal skin tumors have distinct histological patterns which differentiates them from other cutaneous tumors. The commonest variants are those of eccrine sweat gland origin. In our study eccrine tumors were most common in males. The frequent localizations were face and trunk. Malignant eccrine tumors were very rare. In our study, malignant tumors’ diameters are larger than the tumor diameters in the literature.

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    1) Marla NJ, Pailoor K, Pai MR, Jayaprakash SK. Clinicopathological study of adnexal tumors of skin. Adv Lab Med Int 2014; 4: 122-127.

    2) Pantola C, Kala S, Agarwal A, Sonal A, Saurabh P. Cutaneous adnexal tumours: A clinicopathological descriptive study of 70 cases. World J Pathol 2013; 2: 77-82.

    3) Ansai SI. Topics in histopathology of sweat gland and sebaceous neoplasms. J Dermatol 2017; 44: 315-326.

    4) Shukla P, Fatima U, Malaviya AK. Histomorphological and immunohistochemical reappraisal of cutaneous adnexal tumours: A hospital based study. Scientifica (Cairo) 2016; 2016: 2173427.

    5) Jayalakshmi P, Looi LM. Cutaneous adnexal neoplasms in biopsy specimens processed in the Department of Pathology, University of Malaya. Ann Acad Med Singapore 1996; 25: 522-525.

    6) Kamyab-Hesari K, Balighi K, Afshar N. et al. Clinicopathological study of 1016 consecutive adnexal skin tumors. Acta Med Iran 2013; 51: 879-85.

    7) Gonzalez L, Martino BD, Rodríguez M. et al. Clinical and epidemiological study of adnexal tumours at the Dermatology Department of the Medical Sciences Faculty Paraguay, 2002-2008. Folia Dermatol Peru 2009; 20 : 135-139.

    8) Samaila MO. Adnexal skin tumors in Zaria, Nigeria. Ann Afr Med 2008; 7: 6-10.

    9) Blake PW, Bradford PT, Devesa SS, Jorge RT. Cutaneous appendageal carcinoma incidence and survival patterns in the United States: A population-based study. Arch Dermatol 2010; 146: 625-632.

    10) Rajalakshmi V. Case Series of Skin Adnexal Tumours. J Clin Diagnostic Res 2014; 8: 8-11.

    11) Hiatt KM, Pillow JL, Smoller BR. Her-2 expression in cutaneous eccrine and apocrine neoplasm. Mod Pathol 2004; 17: 28-32.

    12) Nishida H, Daa T, Kashima K. et al. C-KIT (CD117) Expression in Benign and Malignant Sweat Gland Tumors. Am J Dermatopathol 2015; 7: 898-905.

    13) Tolutope O, Wei T, John K. et al. Malignant adnexal tumors of the skin: A single institution experience. World Journal of Surgical Oncology 2018; 16: 99.

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