J Cutan Pathol 2014: 41: 329–331 doi: 10.1111/cup.12277 John Wiley & Sons. Printed in Singapore
© 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd
Journal of Cutaneous Pathology
Letter to the Editor
Granulomatous alopecia areata Keywords: alopecia areata, dermatopathology, hair loss To the Editor, We read with great interest the recent article of Paniz Mondolfi and coworkers regarding granulomatous alopecia.1 We would like to share our experience of a similar case in which the suspected diagnosis of alopecia areata (AA) was confirmed by clinicopathologic correlation. A 58-year-old male presented with a 4-week history of sudden hair loss at the chin. Physical examination revealed loss of hairs in this area accompanied by induration of the dermis without discharge of pus (Fig. 1A). Because of the location and the patient’s habit of regular shaving, the border of the lesion was difficult to evaluate. Other parts of the skin including scalp and nails did not show any abnormalities. Four months later, the patient developed a typical patch of AA at the right temporal area which measured 3.5 × 2.7 cm in diameter (Fig. 1B). Simultaneously, the induration at the chin decreased in intensity. He subsequently developed another patch (8.0 × 4.5 cm) of alopecia nearby. There was no improvement for 8 months. However, 7 months later, the patient presented with almost complete regrowth of hairs. This unusually long period of resolution is possibly because of the extensive granuloma formation. Routine laboratory work-up including evaluation of blood count, erythrocyte sedimentation rate, liver and renal parameters, hepatitis screen, folic acid, glucose, anti-nuclear antibodies (ANA), human immunodeficiency virus (HIV), syphilis screen, immunoglobulin E (IgE) level, and mitochondrial, gastric and thyroid autoantibodies did not reveal any abnormalities. There was a slight vitamin B12 deficiency at 204 ng/l (range 211–911 ng/l). In addition, the patient was receiving L-thyroxin therapy because of hypothyroidism. A biopsy taken from the chin at onset of the disease showed a granulomatous perifollicular infiltrate (Fig. 2A–D). Deeper parts of the section revealed a circumscribed lymphocytic component to the infiltrate that extended into the subcutaneous fat without a clear-cut connection to
Fig. 1. Areas of hair loss on the left cheek and chin. Individual hairs are preserved (A). The patient subsequently developed a typical alopecia areata patch involving the right temporal area (B).
a hair follicle in serial sections. Special stains for bacteria (Gram) and fungi (periodic acid-Schiff [PAS] and Grocott) were negative. Alopecias are traditionally classified into nonscarring and scarring variants with AA usually belonging to the former group, although some
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Letter to the Editor
Fig. 2. There is an intense inflammatory infiltrate around the bulb and lower part of the follicle composed of lymphocytes, neutrophils and macrophages. Note that there are numerous foreign-body giant cells around the ruptured hair follicle associated with fibrosis and necrotic tissue. Deep in the subcutaneous fat, there is a dense, circumscribed infiltrate of lymphocytes close to a hair follicle that is outside the section plane (panel A; inset). Granuloma formation with foreign body formation is even more extensive around another nearby follicle (panel D).
degree of scarring may be observed in late stages of this disease. Both, the clinical presentation and/or histopathology of alopecia may be misleading, thus making clinicopathologic correlation an important matter. Our observation shows that this may be especially true when the histopathology of the disease process is obscured by a foreign body-type granulomatous reaction because of released hair shaft material. Thus, apart from diseases such as
330
syphilis and sarcoidosis, granulomas in the context of alopecia may be non-specific. They occur in different forms especially scarring variants, in particular folliculitis decalvans, and have also been present in 5/47 of our cases of lichen planopilaris published recently2 . Notably, in lichen planopilaris – this being a lichenoid disease involving the infundibulum of the hair follicle down to the bulge region – these granulomas around free hair shaft material are located very
Letter to the Editor superficially and are usually devoid of suppurative inflammation. We agree with Paniz Mondolfi and coworkers that a granulomatous infiltrate in AA appears to be a rare event, having been briefly mentioned by Ackerman,3 probably because the inflammation around the bulb is mostly not acute and destructive and therefore usually does not expose the hair shaft to the immune system. Histopathologically, given that granulomas may even occur in lichen planopilaris, a vertical section of the biopsy is helpful to determine the level of the disease and is thus able to narrow the differential diagnosis. In addition, in this case, lymphocytes extending to the subcutaneous fat (inset Fig. 2A) served as a clue as well as a criterion to the histopathologic diagnosis of AA. In the final
analysis, clinicopathological correlation established the diagnosis. Michael Wilk, MD1 Bettina G. Zelger, MD2 Bernhard Zelger, MD, MSc3 1 Dermatology and Dermatopathology Dermatohistological Laboratory Nuernberg, Germany 2 Institute of Pathology Medical University Innsbruck, Innsbruck, Austria 3 Clinical Department of Dermatology & Venereology, Medical University Innsbruck, Innsbruck, Austria e-mail: [email protected]
References 1. Paniz Mondolfi AE, Cressey BD, Ahmad A, Tapia-Centola B, Cohen LM, Mahmoodi M. Granulomatous alopecia: a variant of alopecia areata? J Cutan Pathol 2013; 40: 358.
2. Wilk M, Zelger BG, Zelger B. Lichen planopilaris – histologic criteria & clues in vertical sections. Hair Ther Transplant 2013; 3: 111. DOI: 10.4172/2167-0951.1000111.
3. Ackerman AB, Chongchitnant N, Sanchez J, et al. Histologic diagnosis of inflammatory skin diseases, 2nd ed. Baltimore: Williams & Wilkins, 1997; 191.
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© 2014 John Wiley & Sons A/S. Published by John Wiley & Sons Ltd
Journal of Cutaneous Pathology
Letter to the Editor
Granulomatous alopecia areata Keywords: alopecia areata, dermatopathology, hair loss To the Editor, We read with great interest the recent article of Paniz Mondolfi and coworkers regarding granulomatous alopecia.1 We would like to share our experience of a similar case in which the suspected diagnosis of alopecia areata (AA) was confirmed by clinicopathologic correlation. A 58-year-old male presented with a 4-week history of sudden hair loss at the chin. Physical examination revealed loss of hairs in this area accompanied by induration of the dermis without discharge of pus (Fig. 1A). Because of the location and the patient’s habit of regular shaving, the border of the lesion was difficult to evaluate. Other parts of the skin including scalp and nails did not show any abnormalities. Four months later, the patient developed a typical patch of AA at the right temporal area which measured 3.5 × 2.7 cm in diameter (Fig. 1B). Simultaneously, the induration at the chin decreased in intensity. He subsequently developed another patch (8.0 × 4.5 cm) of alopecia nearby. There was no improvement for 8 months. However, 7 months later, the patient presented with almost complete regrowth of hairs. This unusually long period of resolution is possibly because of the extensive granuloma formation. Routine laboratory work-up including evaluation of blood count, erythrocyte sedimentation rate, liver and renal parameters, hepatitis screen, folic acid, glucose, anti-nuclear antibodies (ANA), human immunodeficiency virus (HIV), syphilis screen, immunoglobulin E (IgE) level, and mitochondrial, gastric and thyroid autoantibodies did not reveal any abnormalities. There was a slight vitamin B12 deficiency at 204 ng/l (range 211–911 ng/l). In addition, the patient was receiving L-thyroxin therapy because of hypothyroidism. A biopsy taken from the chin at onset of the disease showed a granulomatous perifollicular infiltrate (Fig. 2A–D). Deeper parts of the section revealed a circumscribed lymphocytic component to the infiltrate that extended into the subcutaneous fat without a clear-cut connection to
Fig. 1. Areas of hair loss on the left cheek and chin. Individual hairs are preserved (A). The patient subsequently developed a typical alopecia areata patch involving the right temporal area (B).
a hair follicle in serial sections. Special stains for bacteria (Gram) and fungi (periodic acid-Schiff [PAS] and Grocott) were negative. Alopecias are traditionally classified into nonscarring and scarring variants with AA usually belonging to the former group, although some
329
Letter to the Editor
Fig. 2. There is an intense inflammatory infiltrate around the bulb and lower part of the follicle composed of lymphocytes, neutrophils and macrophages. Note that there are numerous foreign-body giant cells around the ruptured hair follicle associated with fibrosis and necrotic tissue. Deep in the subcutaneous fat, there is a dense, circumscribed infiltrate of lymphocytes close to a hair follicle that is outside the section plane (panel A; inset). Granuloma formation with foreign body formation is even more extensive around another nearby follicle (panel D).
degree of scarring may be observed in late stages of this disease. Both, the clinical presentation and/or histopathology of alopecia may be misleading, thus making clinicopathologic correlation an important matter. Our observation shows that this may be especially true when the histopathology of the disease process is obscured by a foreign body-type granulomatous reaction because of released hair shaft material. Thus, apart from diseases such as
330
syphilis and sarcoidosis, granulomas in the context of alopecia may be non-specific. They occur in different forms especially scarring variants, in particular folliculitis decalvans, and have also been present in 5/47 of our cases of lichen planopilaris published recently2 . Notably, in lichen planopilaris – this being a lichenoid disease involving the infundibulum of the hair follicle down to the bulge region – these granulomas around free hair shaft material are located very
Letter to the Editor superficially and are usually devoid of suppurative inflammation. We agree with Paniz Mondolfi and coworkers that a granulomatous infiltrate in AA appears to be a rare event, having been briefly mentioned by Ackerman,3 probably because the inflammation around the bulb is mostly not acute and destructive and therefore usually does not expose the hair shaft to the immune system. Histopathologically, given that granulomas may even occur in lichen planopilaris, a vertical section of the biopsy is helpful to determine the level of the disease and is thus able to narrow the differential diagnosis. In addition, in this case, lymphocytes extending to the subcutaneous fat (inset Fig. 2A) served as a clue as well as a criterion to the histopathologic diagnosis of AA. In the final
analysis, clinicopathological correlation established the diagnosis. Michael Wilk, MD1 Bettina G. Zelger, MD2 Bernhard Zelger, MD, MSc3 1 Dermatology and Dermatopathology Dermatohistological Laboratory Nuernberg, Germany 2 Institute of Pathology Medical University Innsbruck, Innsbruck, Austria 3 Clinical Department of Dermatology & Venereology, Medical University Innsbruck, Innsbruck, Austria e-mail: [email protected]
References 1. Paniz Mondolfi AE, Cressey BD, Ahmad A, Tapia-Centola B, Cohen LM, Mahmoodi M. Granulomatous alopecia: a variant of alopecia areata? J Cutan Pathol 2013; 40: 358.
2. Wilk M, Zelger BG, Zelger B. Lichen planopilaris – histologic criteria & clues in vertical sections. Hair Ther Transplant 2013; 3: 111. DOI: 10.4172/2167-0951.1000111.
3. Ackerman AB, Chongchitnant N, Sanchez J, et al. Histologic diagnosis of inflammatory skin diseases, 2nd ed. Baltimore: Williams & Wilkins, 1997; 191.
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![[Alopecia areata].](/assets/img/hold.png)
