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Dermatology Volume 58 Issue 2 March-April 2013 Indian Journal Of Indian Journal of ISSN: 0019-5154 Dermatology Volume 58 Issue 2 March-April 2013 Indian Journal of Highlights of the issue Dermatology • Update on cutaneous calciphylaxis • Macrophage migration inhibitory factor in • V Dermatology olume • Fixed duration therapy in leprosy 58 • Issue • Environmental dermatoses in Ladakh • Demodex folliculorum as a risk factor in 2 • Diagnosing rosacea March-April • Annular lesions in Dermatology 2013 • Pages Clinical and photomicrograph of Mycosis fungoides, PET-CT for staging and response assessment IJD® Symposium: Integrative Dermatology 87-**** Guest Editor: S R Narahari IJD® www.e-ijd.org E‑Case Report Angiolymphoid Hyperplasia with Eosinophilia with Follicular Mucinosis Rameshwar Gutte, Bhavana Doshi, Uday Khopkar From the Department of Dermatology, Seth G. S. Medical College and King Edward Memorial Hospital, Mumbai, India Abstract Follicular mucinosis occurring along with angiolymphoid hyperplasia with eosinophils (ALHE) has been described in a 49-year-old male. The patient presented with pruritic hyperpigmented papules and nodules on the vertex and right parietal scalp. There was no any other complaint. Histopathological examination from one of the papule showed prominent blood vessels in the dermis lined by plump histiocytoid endothelial cells that were surrounded by a dense lymphoid infiltrate with numerous eosinophils; these findings are typical of angiolymphoid hyperplasia with eosinophilia. Features of follicular mucinosis were observed in the same section with 3 hyperplastic follicular infundibula containing pools of mucin in the infundibular epithelium. The concurrent occurrence of these 2 distinct histopathological patterns in the same biopsy specimen has been reported rarely. Key Words: Angiolymphoid hyperplasia, eosinophilia, follicular mucinosis, scalp What was known? perivascular area and other parts of the dermis. These Follicular mucinosis has been described with many other conditions apart features were consistent with angiolymphoid hyperplasia. from alopecia mucinosa. In addition to these, there were several hyperplastic follicles showing plenty of bluish-gray stringy mucin collected in Introduction large pools within the follicular epithelium. [Figures 2 and 3] Alcian blue stain at pH 2.5 confirmed presence of Angiolymphoid hyperplasia with eosinophilia (ALHE) is mucin within dilated follicular infundibula, interstitium, a rare, benign disease with distinctive histopathological and perivascularly. [Figure 4] Based on clinico-pathological features. Follicular mucinosis is not a distinct clinical correlation, a final diagnosis of angiolymphoid hyperplasia entity, but it is a histopathological reaction pattern that has with eosinophilia with follicular mucinosis was made. been described with many conditions apart from alopecia Patient was advised intralesional bleomycin therapy; mucinosa. Concurrent occurrence of follicular mucinosis however, he lost to follow-up. and ALHE is rare.[1,2] In this case, we describe a 49-year-old Indian male with Discussion both these histopathological findings present in the same ALHE was first described in 1969 by Wells and Whimster.[2] biopsy specimen leading to diagnostic dilemma. It is a well-established clinico-pathological entity presumed to arise due to the development of arterio-venous (A-V) Case Report shunts in the skin. Clinically, it presents with erythematous, A 49-year-old married Indian male presented with multiple, pruritic papules and nodules of varying sizes, affecting pruritic hyper-pigmented papular lesions of 0.5-1 cm in most commonly scalp, ears, face, and the neck regions. size [Figure 1] over the vertex and right parietal scalp for a Histopathologically, diagnostic feature is the thick-or duration of 1 year. There was no history of preceding trauma thin-walled vascular structures lined by protruding or surgery at the site. The lesions arose spontaneously with “hobnail” endothelial cells and an accompanying lymphoid gradual increase in size and number. There was no history infiltrate with numerous eosinophils. The density of both of bleeding, but the patient complained of occasional the lymphoid cells and eosinophils is highly variable.[1] pruritus in the lesions. There was no other relevant medical Follicular mucinosis presents with solitary or grouped history. Peripheral blood eosinophilia was not present. erythematous papules, nodules, or plaques that may be A clinical differential diagnosis of prurigo nodularis indurated. Histopathologically, there is varying amounts and angiolymphoid hyperplasia with eosinophilia was of mucin accumulates within the follicular infundibula considered. and the outer root sheath epithelium, accompanied by perifollicular lympho-histiocytic infiltrate that may contain Histopathological examination from a papule revealed normal epidermis with large, thick-walled vessels in the Access this article online upper and mid dermis lined by plump endothelial cells Quick Response Code: protruding into the lumen. There was a dense lymphocytic Website: www.e‑ijd.org infiltrate along with many eosinophils present in the Address for correspondence: Dr. Rameshwar Gutte, Department of DOI: 10.4103/0019‑5154.108081 Dermatology, Seth GS Medical College and KEM Hospital, Parel, Mumbai ‑ 400 012, India. E‑mail: [email protected] Gutte, et al.: Angiolymphoid hyperplasia with eosinophlia with follicular mucinosis Figure 1: Multiple hyper‑pigmented papules over scalp Figure 2: Diffuse infiltrate in dermis with vascular proliferation and dilated follicles with bluish mucin are seen. (H and E, ×40) Figure 3: Close‑up view showing thick walled dilated vessel with plump hobnail endothelial cells and numerous eosinophils in infiltrate, also a dilated follicle with mucin is shown. (H and E, ×100) eosinophils and plasma cells. It is classified into 2 types, primary (idiopathic) and secondary variety. Primary form usually affects children and young adults and has a shorter Figure 4: Alcian blue stain showing bluish mucin deposition within dilated duration with a benign course, while secondary form is follicular infidibulum, interstitium and peri‑vascularly (Alcian blue, ×100) more widespread and almost always a disease of adults. It has been associated with numerous benign and malignant Due to rare occurrence of this association of ALHE and conditions including lymphomas, of which majority are follicular mucinosis, it is not clear whether such cases mycosis fungoides. Other associations include Hodgkin’s should be treated differently. It is important that both disease, cutaneous B-cell lymphoma, syringolymphoid clinicians and pathologist are aware of this rare association hyperplasia with cutaneous T-cell lymphoma, and to avoid misdiagnosis. inflammatory conditions like chronic discoid lupus In conclusion, we report a case of rare association of erythematosus, alopecia areata, angiolymphoid hyperplasia, ALHE with follicular mucinosis, which shows importance eosinophilic pustular folliculitis, spongitic dermatitis, lichen of clinico-pathological correlation. striatus, leprosy, sarcoidosis, and growths such as verrucae [1,3] and melanocytic nevi. What is new? This case highlights a rare occurrence of follicular mucinosis with The concurrent occurrence of follicular mucinosis with angiolymphoid hyperplasia. angiolymphoid hyperplasia is unusual; to date, only few such cases have been described in the literature. Wolff, References et al. reported a case of angiolymphoid hyperplasia with 1. Joshi R. Angiolymphoid hyperplasia with follicular mucinosis. follicular mucinosis in 1978.[4] Indian J Dermatol Venereol Leprol 2007;73:346-7. 2. Kumar JV, Guruprasad KY. Angiolymphoid hyperplasia Bovet and Delacretaz reported similar case in 1979.[5] Joshi with eosinophilia. Indian J Dermatol Venereol Leprol R. reported a similar case in 2007, probably a first case 1998;64:133-4. [1] of this rare association from India. We report a second 3. Loffreda MD. Inflammatory diseases of hair follicles, sweat similar case from India. glands and cartilage. In: Elder DE, editor. Lever’s Histopathology Gutte, et al.: Angiolymphoid hyperplasia with eosinophlia with follicular mucinosis th of the skin.10 ed. New Delhi: Wolter Kluwer (India) Pvt. Ltd; How to cite this article: Gutte R, Doshi B, Khopkar U. Angiolymphoid 2009. p. 459-502. hyperplasia with eosinophilia with follicular mucinosis. Indian J Dermatol 4. Wolff HH, Kinney J, Ackerman AB. Angiolymphoid hyperplasia 2013;58:159. with follicular mucinosis. Arch Dermatol 1978;114:229-32. 5. Bovet R, Delacretaz J. Angiolymphoid hyperplasia with follicular Received: July, 2011. Accepted: August, 2011. mucinosis. Dermatologic 1979;158:343-7 Source of support: Nil, Conflict of Interest: Nil..
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