Clinical and Experimental Dermatology
A reddish, pedunculated, granulomatous nodule on the nasal dorsum A. Korekawa,1 H. Nakano,1 T. Aizu,1 H. Kitamura,2 K. Harada2 and D. Sawamura1 1 Department of Dermatology, Hirosaki University Graduate School of Medicine, Hirosaki, Japan; and 2Department of Dermatology, Aomori Prefectural Central Hospital, Aomori, Japan
doi: 10.1111/ced.12613
Clinical findings A 65-year-old man presented with a 1-year history of a reddish nodule on his nasal dorsum. On physical examination, the lesion was found to be 13 9 10 mm in size, elastic, soft, reddish in colour and partially encrusted (the patient had scratched the lesion a few days previously), and it bled easily. Although it was a pedunculated granulomatous nodule, it was not cystic or translucent (Fig. 1). There was no associated pain or itching. The patient reported he had often scratched and squeezed the lesion because he thought the rash was acne. Based on the clinical appearance, we diagnosed the lesion as a squamous cell carcinoma, and took a skin biopsy.
There were no atypical cells. Immunohistochemically, gross cystic disease fluid protein-15 stained the cytoplasm of the columnar cells (Fig. 2c), and carcinoembryonic antigen stained their surface (Fig. 2d). The cytoplasm of the myoepithelial cells was positive for
Histopathological findings Histopathological examination of the biopsy revealed cystic structures in the dermis, with some papillary projections of the cyst in the cavity, extravasation of red blood cells and dilation of capillaries in the stroma. A prominently lymphocyte infiltration was demonstrated in the upper dermis. The inner surface of the cyst was lined with one to several layers of columnar cells showing decapitation secretion, indicative of apocrine-type secretion, and partial papillary growth (pseudopapillary growth) without fibrovascular stroma (Fig. 2a,b). The outer sides of the columnar cells were lined with a single row of flat cells, indicative of myoepithelial cells. Orthokeratotic hyperkeratosis and acanthosis was visible in the epidermis.
(a)
Correspondence: Dr Daisuke Sawamura, Department of Dermatology, Hirosaki University Graduate School of Medicine, 5 Zaifu-cho, Hirosaki 036-8562, Japan E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 19 September 2014
ª 2015 British Association of Dermatologists
(b) Figure 1 (a,b) A reddish, pedunculated, granulomatous nodule
on the nasal dorsum.
Clinical and Experimental Dermatology (2015) 40, pp703–705
703
D CP
CED
CPD • Clinicopathological case
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CP
Clinicopathological case
(a)
(b)
(e)
(c)
(d)
(f)
Figure 2 (a,b) Cystic structures in the dermis, with some papillary projections of the cyst into the cavity. The inner surface of the cyst was
lined by one or several layers of columnar apocrine-type secretion cells and partial papillary growth (haematoxylin and eosin, original magnification (a) 9 40; (b) 9 400. (c,d) Immunohistochemical analysis of the apocrine-type secretion cells showed that (c) gross cystic disease fluid protein (GCDFP)-15 stained the cytoplasm of the columnar cells, and (d) carcinoembryonic antigen (CEA) stained their surface. Original magnification (c,d) 9 400. (e) The outer side of the columnar cells was lined by myoepithelial cells (arrow) (haematoxylin and eosin, original magnification 9 400). (f) Immunohistochemical analysis of the myoepithelial cells showed that a-smooth muscle actin (SMA) stained the cytoplasm (arrow) (original magnification 9 400).
a-smooth muscle actin (Fig. 2e,f). After a final diagnosis, the lesion was completely excised. Two years after excision, no recurrence of the lesion was observed. What is your diagnosis?
Diagnosis Apocrine hidrocystoma (AH).
Discussion AH (also known as apocrine cystadenoma) is a benign cystic tumour arising from the proliferation of apocrine glands, and commonly occurs as cystic lesions on the head and neck.1 Hidrocystoma was first described as a cystic lesion that represented cystic dilation of the eccrine sweat gland duct.2,3 Mehregan
704
Clinical and Experimental Dermatology (2015) 40, pp703–705
reported 17 cases of a benign neoplasm, and named these tumours ‘apocrine cystadenoma’. He noted that the tumour was an adenomatous cystic proliferation of apocrine glands and not a simple retention cyst.4 According to the histological architecture, apocrine cystic tumours without papillary projections into the cyst should be termed ‘apocrine hidrocystoma’, whereas apocrine adenomatous cystic tumours with papillary projections into the cyst should be termed ‘apocrine cystadenoma’ (AC).2 In our case, histopathological examination showed some papillary projections of the cyst into the cavity without fibrovascular stroma, but the biopsy specimen showed primarily cystic lesions composed of one or several layers of columnar cells and did not show prominent proliferative adenomatous growth. We considered this partial papillary growth as pseudopapillary growth.2
ª 2015 British Association of Dermatologists
Based on the histological characteristics, hidrocystoma have been categorized into two types (apocrine and eccrine). It is difficult to differentiate between AH and eccrine hidrocystoma (EH) because both are cystic lesions with a double lining of epithelial cells. They are distinguished by the presence of decapitation secretion, which is seen in AH, but not in EH. Additionally, EH will grow in size and/or multiply in number during hot or humid weather. Our case did not have this clinical feature, and did have decapitation secretion histopathologically, thus allowing us to confidently diagnose the lesion as AH rather than AC or EH. Both types of apocrine tumours commonly occur as a solid, dome-shaped, translucent, cystic lesion, and vary in colour, ranging from skin-coloured to bluishblack or white. To our knowledge, this is the first case of AH presenting as a reddish, pedunculated, granulomatous nodule. Furthermore, AH is often located on the head and neck, especially around the eyelids, but the nasal dorsum is a very rare location. Our case is the only report of a relatively large, solitary tumour occurring on the nasal dorsum. We suggest that the red colour observed clinically was due to the extravasation of red cells and dilation of capillaries in the stroma. In addition, there is little fat in the nose, as the nasal bone is located just under the skin, preventing the nodule from growing downwards, thereby resulting in a pedunculated nodule. The patient had often scratched and squeezed the lesion because he thought the rash was acne. We propose that the granulomatous skin change observed clinically is due to repeated irritation and inflammation caused by this. In conclusion, we report a case of AH presenting as a large nodule on the nasal dorsum, the first such presentation to our knowledge. This case highlights that if a reddish, pedunculated, granulomatous nodule is
ª 2015 British Association of Dermatologists
observed in a patient, AH should be considered as a differential diagnosis.
Learning points ● AH commonly occurs as a solid, dome-shaped,
cystic, lesion that may vary in colour from skincoloured to bluish-black or white, but may present as a reddish, pedunculated, granulomatous nodule. ● AH is usually located on the head and neck, especially around the eyelids, but may present on the nasal dorsum. ● The nasal dorsum has little fat with the nasal bone located just under the skin, preventing the tumours from growing downwards. ● In the present case, the patient had scratched and squeezed the lesion, causing repeating irritation and inflammation, thereby resulting in a reddish, pedunculated and granulomatous tumour at this site.
References 1 L opez V, Alonso V, Jord a E et al. Apocrine hidrocystoma on the penis of a 40-year-old man. Int J Dermatol 2013; 52: 502–4. 2 Sugiyama A, Sugiura M, Piris A et al. Apocrine cystadenoma and apocrine hidrocystoma: examination of 21 cases with emphasis on nomenclature according to proliferative features. J Cutan Pathol 2007; 34: 912–17. 3 Robinson AR. Hidrocystoma. J Cutan Genitourin Dis 1893; 11: 293. 4 Mehregan AH. Apocrine cystadenoma: a clinicopathologic study with special reference to the pigmented variety. Arch Dermatol 1964; 90: 274–9.
Clinical and Experimental Dermatology (2015) 40, pp703–705
705
D CP
Clinicopathological case
A reddish, pedunculated, granulomatous nodule on the nasal dorsum A. Korekawa,1 H. Nakano,1 T. Aizu,1 H. Kitamura,2 K. Harada2 and D. Sawamura1 1 Department of Dermatology, Hirosaki University Graduate School of Medicine, Hirosaki, Japan; and 2Department of Dermatology, Aomori Prefectural Central Hospital, Aomori, Japan
doi: 10.1111/ced.12613
Clinical findings A 65-year-old man presented with a 1-year history of a reddish nodule on his nasal dorsum. On physical examination, the lesion was found to be 13 9 10 mm in size, elastic, soft, reddish in colour and partially encrusted (the patient had scratched the lesion a few days previously), and it bled easily. Although it was a pedunculated granulomatous nodule, it was not cystic or translucent (Fig. 1). There was no associated pain or itching. The patient reported he had often scratched and squeezed the lesion because he thought the rash was acne. Based on the clinical appearance, we diagnosed the lesion as a squamous cell carcinoma, and took a skin biopsy.
There were no atypical cells. Immunohistochemically, gross cystic disease fluid protein-15 stained the cytoplasm of the columnar cells (Fig. 2c), and carcinoembryonic antigen stained their surface (Fig. 2d). The cytoplasm of the myoepithelial cells was positive for
Histopathological findings Histopathological examination of the biopsy revealed cystic structures in the dermis, with some papillary projections of the cyst in the cavity, extravasation of red blood cells and dilation of capillaries in the stroma. A prominently lymphocyte infiltration was demonstrated in the upper dermis. The inner surface of the cyst was lined with one to several layers of columnar cells showing decapitation secretion, indicative of apocrine-type secretion, and partial papillary growth (pseudopapillary growth) without fibrovascular stroma (Fig. 2a,b). The outer sides of the columnar cells were lined with a single row of flat cells, indicative of myoepithelial cells. Orthokeratotic hyperkeratosis and acanthosis was visible in the epidermis.
(a)
Correspondence: Dr Daisuke Sawamura, Department of Dermatology, Hirosaki University Graduate School of Medicine, 5 Zaifu-cho, Hirosaki 036-8562, Japan E-mail: [email protected] Conflict of interest: the authors declare that they have no conflicts of interest. Accepted for publication 19 September 2014
ª 2015 British Association of Dermatologists
(b) Figure 1 (a,b) A reddish, pedunculated, granulomatous nodule
on the nasal dorsum.
Clinical and Experimental Dermatology (2015) 40, pp703–705
703
D CP
CED
CPD • Clinicopathological case
D
CP
Clinicopathological case
(a)
(b)
(e)
(c)
(d)
(f)
Figure 2 (a,b) Cystic structures in the dermis, with some papillary projections of the cyst into the cavity. The inner surface of the cyst was
lined by one or several layers of columnar apocrine-type secretion cells and partial papillary growth (haematoxylin and eosin, original magnification (a) 9 40; (b) 9 400. (c,d) Immunohistochemical analysis of the apocrine-type secretion cells showed that (c) gross cystic disease fluid protein (GCDFP)-15 stained the cytoplasm of the columnar cells, and (d) carcinoembryonic antigen (CEA) stained their surface. Original magnification (c,d) 9 400. (e) The outer side of the columnar cells was lined by myoepithelial cells (arrow) (haematoxylin and eosin, original magnification 9 400). (f) Immunohistochemical analysis of the myoepithelial cells showed that a-smooth muscle actin (SMA) stained the cytoplasm (arrow) (original magnification 9 400).
a-smooth muscle actin (Fig. 2e,f). After a final diagnosis, the lesion was completely excised. Two years after excision, no recurrence of the lesion was observed. What is your diagnosis?
Diagnosis Apocrine hidrocystoma (AH).
Discussion AH (also known as apocrine cystadenoma) is a benign cystic tumour arising from the proliferation of apocrine glands, and commonly occurs as cystic lesions on the head and neck.1 Hidrocystoma was first described as a cystic lesion that represented cystic dilation of the eccrine sweat gland duct.2,3 Mehregan
704
Clinical and Experimental Dermatology (2015) 40, pp703–705
reported 17 cases of a benign neoplasm, and named these tumours ‘apocrine cystadenoma’. He noted that the tumour was an adenomatous cystic proliferation of apocrine glands and not a simple retention cyst.4 According to the histological architecture, apocrine cystic tumours without papillary projections into the cyst should be termed ‘apocrine hidrocystoma’, whereas apocrine adenomatous cystic tumours with papillary projections into the cyst should be termed ‘apocrine cystadenoma’ (AC).2 In our case, histopathological examination showed some papillary projections of the cyst into the cavity without fibrovascular stroma, but the biopsy specimen showed primarily cystic lesions composed of one or several layers of columnar cells and did not show prominent proliferative adenomatous growth. We considered this partial papillary growth as pseudopapillary growth.2
ª 2015 British Association of Dermatologists
Based on the histological characteristics, hidrocystoma have been categorized into two types (apocrine and eccrine). It is difficult to differentiate between AH and eccrine hidrocystoma (EH) because both are cystic lesions with a double lining of epithelial cells. They are distinguished by the presence of decapitation secretion, which is seen in AH, but not in EH. Additionally, EH will grow in size and/or multiply in number during hot or humid weather. Our case did not have this clinical feature, and did have decapitation secretion histopathologically, thus allowing us to confidently diagnose the lesion as AH rather than AC or EH. Both types of apocrine tumours commonly occur as a solid, dome-shaped, translucent, cystic lesion, and vary in colour, ranging from skin-coloured to bluishblack or white. To our knowledge, this is the first case of AH presenting as a reddish, pedunculated, granulomatous nodule. Furthermore, AH is often located on the head and neck, especially around the eyelids, but the nasal dorsum is a very rare location. Our case is the only report of a relatively large, solitary tumour occurring on the nasal dorsum. We suggest that the red colour observed clinically was due to the extravasation of red cells and dilation of capillaries in the stroma. In addition, there is little fat in the nose, as the nasal bone is located just under the skin, preventing the nodule from growing downwards, thereby resulting in a pedunculated nodule. The patient had often scratched and squeezed the lesion because he thought the rash was acne. We propose that the granulomatous skin change observed clinically is due to repeated irritation and inflammation caused by this. In conclusion, we report a case of AH presenting as a large nodule on the nasal dorsum, the first such presentation to our knowledge. This case highlights that if a reddish, pedunculated, granulomatous nodule is
ª 2015 British Association of Dermatologists
observed in a patient, AH should be considered as a differential diagnosis.
Learning points ● AH commonly occurs as a solid, dome-shaped,
cystic, lesion that may vary in colour from skincoloured to bluish-black or white, but may present as a reddish, pedunculated, granulomatous nodule. ● AH is usually located on the head and neck, especially around the eyelids, but may present on the nasal dorsum. ● The nasal dorsum has little fat with the nasal bone located just under the skin, preventing the tumours from growing downwards. ● In the present case, the patient had scratched and squeezed the lesion, causing repeating irritation and inflammation, thereby resulting in a reddish, pedunculated and granulomatous tumour at this site.
References 1 L opez V, Alonso V, Jord a E et al. Apocrine hidrocystoma on the penis of a 40-year-old man. Int J Dermatol 2013; 52: 502–4. 2 Sugiyama A, Sugiura M, Piris A et al. Apocrine cystadenoma and apocrine hidrocystoma: examination of 21 cases with emphasis on nomenclature according to proliferative features. J Cutan Pathol 2007; 34: 912–17. 3 Robinson AR. Hidrocystoma. J Cutan Genitourin Dis 1893; 11: 293. 4 Mehregan AH. Apocrine cystadenoma: a clinicopathologic study with special reference to the pigmented variety. Arch Dermatol 1964; 90: 274–9.
Clinical and Experimental Dermatology (2015) 40, pp703–705
705
D CP
Clinicopathological case
