|Year : 2015 | Volume
| Issue : 1 | Page : 26-28
Acute diffuse and total alopecia of the female scalp associated with borrelia-infection
Ekta K Bhardwaj1, Ralph Michel Trüeb2
1 Renovia Medical Aesthetics and Hair Center, Manchester, England, United Kingdom
2 Center for Dermatology and Hair Diseases, Zurich Wallisellen, Switzerland
|Date of Web Publication||18-Mar-2015|
Ralph Michel Trüeb
Center for Dermatology and Hair Diseases, Bahnhofplatz 1A, CH-8304 Zurich-Wallisellen
Source of Support: None, Conflict of Interest: None
| Abstract|| |
A case of acute diffuse and total alopecia of the female scalp associated with Borrelia-infection (acrodermatitis chronica atrophicans) is presented. Today, acute diffuse and total alopecia of the female scalp is recognized as a distinct variant of alopecia areata (AA) predominantly observed in women. Cases of AA have formerly been reported in association with infections. AA is understood to represent an organ-specific autoimmune disease of the hair follicle. It is conceivable that the antigenic stimulus provided by the infection may act as a trigger for alopecia. Vice versa, alopecia may act as a marker for detection of undiagnosed infection. Treatment of the patient with intravenous ceftriaxone led to the resolution of cutaneous borreliosis, and in addition to topical clobetasol foam to complete recovery of hair.
Keywords: Borrelia -infection, co-morbidity, diffuse alopecia areata
|How to cite this article:|
Bhardwaj EK, Trüeb RM. Acute diffuse and total alopecia of the female scalp associated with borrelia-infection. Int J Trichol 2015;7:26-8
| Introduction|| |
Alopecia areata (AA) is a common dermatologic condition with the typical clinical presentation of nonscarring hair loss in patches. Since AA is considered to represent an organ-specific autoimmune disease, the clinical appearance would seem sufficient to make a diagnosis, and further laboratory investigations would seem unnecessary or even inappropriate. Nevertheless, in unusual cases, or patients presenting with extensive disease, certain laboratory investigations may be indicated to detect associated autoimmune diseases and/or co-morbid conditions that may be relevant to the disease course. An increased incidence of other autoimmune diseases, such as autoimmune thyroid disease,  pernicious anemia,  and celiac disease,  is seen among AA patients, while serum ferritin  or vitamin D levels  may have an influence on the disease course. Finally, there have been some reports of AA in association with various infections. ,,,,,, Here, we report a case of successful treatment of acute diffuse and total alopecia of the female scalp associated with Borrelia-infection.
| Case report|| |
A 65-year-old female patient presented with a 3 months history of scalp hair loss in tufts. She also complained of swelling and bluish red discoloration of the right lower limb and of fatigue.
Clinical examination revealed diffuse alopecia [Figure 1] with a positive pull test and cutaneous swelling of the right lower extremity with a bluish red discoloration.
A hair pluck test (trichogram) revealed 53% (frontal) to 62% (occipital) telogen roots with a proportion of 6% dystrophic anagen roots.
Laboratory evaluation (AA co-morbidity, heavy metal, and vasculitis screening) revealed elevated thyroid peroxidase and thyroglobulin antibodies (>600 resp. 464 IU/mL; normal: <34 resp. 115 IU/mL), elevated thyreotropin (TSH) levels (15.29 mIU/L; normal: 0.27-4.20 mIU/l), and vitamin D deficiency (42.9 nmol/L). CRP, ferritin, Vitamin B 12 , antinuclear antibodies (ANA), anti-SSa-60/52(Ro), anti-SSB (La), anti-Sm, anti-nRNP (ribonuclein), anti-histone, anti-Jo-1, anti-dsDNS (ELIA), anti-parietal cell antibodies, anti-TSH receptor antibodies, cardiolipin antibodies IgG and IgM, antineutrophil cytoplasmic antibodies myeloperoxidase/PR3 antibodies, anti citrullinated protein antibody, anti HBs, Bcore, and C antibodies, complement factors C3 and C4 levels, creatine phosphokinase, copper, cadmium, and mercury levels were all normal. Borrelia serology (Western blot) tested positive for VLsE IgMG (different genospecies), p41 IgG (B. sensu strictu), and p41 IgM (B. sensu strictu).
A diagnosis of acute diffuse and total alopecia of the female scalp associated with Borrelia-infection (acrodermatitis chronica atrophicans), autoimmune thyroid disease, and vitamin D-deficiency was made.
The patient was prescribed intravenous ceftriaxone 2 g daily for 3 weeks, topical clobetasol propionate 0.05% foam twice daily on 5 consecutive days/week for 6 months, thyroid supplementation therapy, and oral vitamin D3 1500 IU/day.
[Figure 2] [Figure 3] [Figure 4] (at 3 months), (at 6 months) and (complete remission at 10 months) show the photographic sequence of the alopecia in the course of treatment. The swelling and discoloration of the lower right limb resolved completely, and the patient reported lesser fatigue.
| Discussion|| |
The patient presented clinically with acute diffuse and total alopecia of the female scalp as originally described in 2002 by Sato-Kawamura et al.  Today it is recognized to be basically identical with a subtype of AA presenting with diffuse hair loss as originally proposed in the German literature by Braun-Falco and Zaun as early as 1962.  AA incognita is yet another synonymous designation for the condition proposed by Rebora in 1987.  The condition predominantly affects women and is characterized by diffuse hair shedding in the absence of typical patches.
Today, AA is understood to represent a T-cell mediated, organ-specific autoimmune disease of the hair follicle,  that nevertheless may occur in association with other autoimmune phenomena, such as circulating autoantibodies  and autoimmune diseases. 
Presence of one or more additional diseases co-occurring with a primary condition or the effect of such additional diseases, is generally termed as co-morbidity. While the concept of co-morbidities has emerged in dermatologic disease, such as psoriasis,  it has largely been ignored in the management of hair loss patients.
Co-morbid conditions reported to be associated with AA include: Autoimmunity (thyroid, celiac disease, parietal cell), ,, deficiencies (iron, vitamin D, zinc), ,, allergies (atopic disease), , and infections. ,,,,,, The latter have included reports on: Epstein-Barr Virus,  human immunodeficiency virus,  swine flu,  viral hepatitis,  cytomegalovirus,  dental infections,  and Helicobacter pylori.  If such associations are confirmed by epidemiological studies designed for this purpose, new therapeutic options could be available for these patients.
Previously, Borrelia burgdorferi has been linked to pseudopelade Brocq,  but so far no association of Borrelia-infection with AA has been reported. Ixodes tick  and other insect bites  have been associated with localized patches of hair loss at the site of the bite. However, this rather seems to be a pharmacologic or toxic than immunologic effect.
The patient herein reported presented diffuse AA, associated with acrodermatitis chronica atrophicans, autoimmune thyroid deficiency, and vitamin D deficiency. She was successfully treated with topical clobetasol propionate 0.05% foam twice daily on 5 consecutive days/week for 6 months, intravenous ceftriaxone 2g daily for 3 weeks, thyroid supplementation therapy, and oral vitamin D3.
Although the various reported associations of AA with infectious agents have remained controversial and need to be confirmed by epidemiological studies, it remains conceivable that the antigenic stimulus provided by co-morbid infections may entertain the autoimmune reaction underlying AA. In the same line is the observation of AA triggered by recombinant hepatitis B vaccination.  Therefore, detection and treatment of possible infection associated with AA should be taken into consideration. Vice versa, AA may act as a marker for detection of hitherto undiagnosed co-morbid conditions, such as thyroid disease, Vitamin D deficiency, and potentially serious infection.
| References|| |
Lyakhovitsky A, Shemer A, Amichai B. Increased prevalence of thyroid disorders in patients with new onset alopecia areata. Australas J Dermatol 2014. [DOI: 10.1111/ajd.12178. [Epub ahead of print]
MacLean KJ, Tidman MJ. Alopecia areata: More than skin deep. Practitioner 2013;257:29-32, 3.
Fessatou S, Kostaki M, Karpathios T. Coeliac disease and alopecia areata in childhood. J Paediatr Child Health 2003;39:152-4.
Kantor J, Kessler LJ, Brooks DG, Cotsarelis G. Decreased serum ferritin is associated with alopecia in women. J Invest Dermatol 2003;121:985-8.
Aksu Cerman A, Sarikaya Solak S, Kivanc Altunay I. Vitamin D deficiency in alopecia areata. Br J Dermatol 2014;170:1299-304.
Ito T, Tokura Y. Alopecia areata triggered or exacerbated by swine flu virus infection. J Dermatol 2012;39:863-4.
Gil Montoya JA, Cutando Soriano A, Jimenez Prat J. Alopecia areata of dental origin. Med Oral 2002;7:303-8.
Rodriguez TA, Duvic M, National Alopecia Areata Registry. Onset of alopecia areata after Epstein-Barr virus infectious mononucleosis. J Am Acad Dermatol 2008;59:137-9.
Stewart MI, Smoller BR. Alopecia universalis in an HIV-positive patient: Possible insight into pathogenesis. J Cutan Pathol 1993;20:180-3.
Campuzano-Maya G. Cure of alopecia areata after eradication of Helicobacter pylori
: A new association? World J Gastroenterol 2011;17:3165-70.
Podányi B, Lengyel G, Hársing J, Becker K, Horváth A. Skin diseases associated with chronic hepatitis C. Orv Hetil 1998;139:2633-7.
Skinner RB Jr, Light WH, Bale GF, Rosenberg EW, Leonardi C. Alopecia areata and presence of cytomegalovirus DNA. JAMA 1995;273:1419-20.
Sato-Kawamura M, Aiba S, Tagami H. Acute diffuse and total alopecia of the female scalp. A new subtype of diffuse alopecia areata that has a favorable prognosis. Dermatology 2002;205:367-73.
Braun-Falco O, Zuan H. On the nature of chronic diffuse alopecia in women. Arch Klin Exp Dermatol 1962;215:165-80.
Rebora A. Alopecia areata incognita: A hypothesis. Dermatologica 1987;174:214-8.
Paus R, Bertolini M. The role of hair follicle immune privilege collapse in alopecia areata: Status and perspectives. J Investig Dermatol Symp Proc 2013;16:S25-7.
Friedmann PS. Alopecia areata and auto-immunity. Br J Dermatol 1981;105:153-7.
Chu SY, Chen YJ, Tseng WC, Lin MW, Chen TJ, Hwang CY, et al.
Comorbidity profiles among patients with alopecia areata: The importance of onset age, a nationwide population-based study. J Am Acad Dermatol 2011;65:949-56.
Yeung H, Takeshita J, Mehta NN, Kimmel SE, Ogdie A, Margolis DJ, et al.
Psoriasis severity and the prevalence of major medical comorbidity: A population-based study. JAMA Dermatol 2013;149:1173-9.
Kumar B, Sharma VK, Sehgal S. Antismooth muscle and antiparietal cell antibodies in Indians with alopecia areata. Int J Dermatol 1995;34:542-5.
Bhat YJ, Manzoor S, Khan AR, Qayoom S. Trace element levels in alopecia areata. Indian J Dermatol Venereol Leprol 2009;75:29-31.
De Weert J, Temmerman L, Kint A. Alopecia areata: A clinical study. Dermatologica 1984;168:224-9.
Kaur S, Sharma VK, Kumar L, Kumar B. Atopy and alopecia areata in North Indians. Indian J Dermatol Venereol Leprol 2002;68:267-9.
Köstler E, Hubl W, Seebacher C. PCR detected Borrelia
burgdorferi DNA in a tissue sample in pseudopelade Brocq. Hautarzt 1999;50:897.
Castelli E, Caputo V, Morello V, Tomasino RM. Local reactions to tick bites. Am J Dermatopathol 2008;30:241-8.
Namazi MR, Jorizzo JL. Ant-induced alopecia: A case report and literature review. Arch Dermatol 2008;144:1526-7.
Wise RP, Kiminyo KP, Salive ME. Hair loss after routine immunizations. JAMA 1997;278:1176-8.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]
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