CASE REPORT
Tinea Cruris and Tinea Corporis Masquerading as Tinea Indecisiva: Case Report and Review of the Literature Sidharth Sonthalia, Archana Singal, and Shukla Das Background: Tinea indecisiva is characterized by concentric scaly rings simulating tinea imbricata but caused by dermatophytes other than Trichophyton concentricum. Objective: Tinea indecisiva has been rarely reported. We report a unique case and review of the previously reported cases, pathogenesis, and management. Methods: An adult Indian man developed extensive tinea cruris and tinea corporis with concentric rings of scaly lesions over the groin, buttocks, and thighs following the use of oral corticosteroids and antifungal-steroid cream for 3 months. Mycologic and immunologic studies were performed for diagnosis. Results: Diagnosis of tinea indecisiva was confirmed on the appearance of ‘‘ring-within-a-ring’’ lesions clinically and isolation of Trichophyton mentagrophytes var. interdigitale as the etiologic agent on mycologic testing. Intradermal testing with Trichophyton extract showed fluctuating hypersensitivity responses. Four-week treatment with daily oral terbinafine resulted in complete resolution. Conclusion: Tinea indecisiva should be considered in a patient with tinea imbricata–like lesions with local immunosuppression caused by a non-concentricum dermatophyte. Contexte: Tinea indecisiva se caracte´rise par des anneaux squameux, concentriques, simulant tinea imbricata, mais l’affection est cause´e par d’autres dermatophytes que Trichophyton concentricum. Objectif: Rare est la description de Tinea indecisiva dans la documentation; nous faisons e´tat ici d’un cas unique en son genre et passons en revue les cas expose´s ante´rieurement ainsi que leur pathogene`se et leur prise en charge. Me´thode: Un Indien adulte a consulte´ pour des zones e´tendues d’ecze´ma margine´ et d’herpe`s circine´, accompagne´s d’anneaux concentriques de le´sions squameuses touchant les aines, les fesses, et les cuisses, qui sont apparues a` la suite de la prise de corticoste´roı¨des oraux et de l’application d’une cre`me associant un antifongique et un ste´roı¨de pendant 3 mois. Des analyses mycologiques et immunologiques ont e´te´ effectue´es pour confirmer le diagnostic. Re´sultats: Le diagnostic de tinea indecisiva a e´te´ confirme´ par l’apparence clinique des le´sions en forme d’« anneau dans un anneau » et par la mise en e´vidence de l’agent causal, Trichophyton mentagrophytes, variante interdigitale, a` l’examen mycologique. Des e´preuves intradermiques d’extraits de Trichophyton ont re´ve´le´ des re´actions d’hypersensibilite´ de type variable. La prise quotidienne de terbinafine orale, durant 4 semaines, a permis la disparition comple`te des le´sions. Conclusions: Le diagnostic de tinea indecisiva devrait eˆtre envisage´ chez les patients pre´sentant des le´sions ressemblant a` celles de tinea imbricata, accompagne´es d’une immunode´pression locale, mais cause´es par un dermatophyte de type non concentricum.
INEA IMBRICATA (TI) caused by Trichophyton concentricum is a chronic dermatophytosis characterized clinically by the presence of multiple annular concentric plaques that later become lamellar with abundant thick scales, giving the appearance of tiles or fish scales.1 TI occurs endemically in certain regions, especially southern Asia. Although TI is caused exclusively by T. concentricum, recently, a few cases have been reported that presented with clinical features simulating TI but caused by species other than T. concentricum. Such peculiar cases have been labeled as ‘‘tinea indecisiva’’ and
T
From SKINNOCENCE: The Skin Clinic & Research Centre, Haryana, India, and Departments of Dermatology and STD and Microbiology, University College of Medical Sciences and Guru Teg Bahadur Hospital, University of Delhi, Delhi, India. Address reprint requests to: Archana Singal, MD, MNAMS, B-14, Law Apartments, Karkardooma, Delhi-110092, India; e-mail: archanasingal@ hotmail.com.
DOI 10.2310/7750.2014.14057 # 2014 Canadian Dermatology Association
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‘‘tinea pseudoimbricata’’ and tend to occur in patients with some degree of immunosuppression.2,3 To the best of our knowledge, only eight such cases have been published to date, each occurring in a different clinical setting with different lesional localization and caused by different dermatophytic species.2–8 In this report, we describe another unique case of tinea indecisiva with the following special characteristics: involvement of the groin, penile shaft, and buttocks (previous cases have primarily involved the trunk, limbs, or scalp) and caused by Trichophyton mentagrophytes var. interdigitale (never described before). We further present a thorough review of previously reported cases of this condition, with an emphasis on the putative pathogenesis. Figure 1. Erythematous, annular, concentric rings with scattered pustules studded over the rings and grayish-white scales overlying the central relatively clear zone, involving the groin and penile shaft.
Case Report An 18-year-old adult Indian male presented with a 3month history of persistent itchy rash over the groin, lower abdomen, and thighs. He gave a history of intermittent application of an over-the-counter antifungal-steroid combination cream (containing miconazole nitrate 2% and clobetasol dipropionate 0.05%) twice daily, oral levocetirizine 5 mg every night, and oral betamethasone (2 mg) once a day for the past 3 months, which resulted in temporary relief of itching and partial resolution of lesions followed by progressive worsening. He had not sought a dermatologic consultation until then and had no history of having taken any oral antifungal drugs. He had no history of diabetes or any other significant medical disorder. A history of high-risk behavior (eg, multiple sex partners and intravenous drug abuse) for acquisition of acquired immunodeficiency was not elicited. A family history of diabetes or recurrent skin infections was absent. His physical and systemic examinations were normal. A cutaneous examination revealed the presence of widespread erythematous, annular, concentric rings with scattered pustules studded over the rings and grayishwhite scales overlying the central relatively clear zone, involving the groin and penile shaft (Figure 1), buttocks (Figure 2), thighs (Figure 3), and lower abdomen, clinically mimicking TI. Examination of the palms and soles, nails, and scalp revealed no abnormality. Hematologic and biochemical investigations, including hemography, fasting and postprandial plasma glucose, and renal and hepatic function tests, were normal. Serology for human immunodeficiency virus (HIV) was negative. Microscopic examination of skin scrapings with 10% potassium hydroxide (KOH) revealed thin hyaline hyphae suggestive of dermatophytic infection. Fungal culture on Sabouraud 172
dextrose agar (SDA) showed flat, white, downy growth (Figure 4). Lactophenol cotton blue preparation revealed fine septate hyaline branched hyphae with sparse macroconidia and clustered microconidia along with welldeveloped spiral hyphae and chlamydospores (Figure 5), confirming T. mentagrophytes var. interdigitale as the etiologic agent. Serologic testing for IgE immunoglobulin against Trichophyton antigen was strongly positive. A final diagnosis of tinea cruris and tinea corporis manifesting as tinea indecisiva (pseudoimbricata) was made. The patient was treated with daily oral terbinafine 250 mg for 4 weeks and oral levocetirizine 5 mg for 2 weeks. Oral steroids were tapered off over the next 6 weeks. The rash completely subsided within a month. The patient came for his last follow-up visit around 6 months after the initial episode, and there was no recurrence until then.
Figure 2. Similar lesions over the buttocks but with fewer pustules and marked scaling.
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Figure 3. Similar lesions over the thighs.
Discussion The characteristic clinical picture seen in our patient simulated features of TI. Tinea imbricata or Tokelau is a chronic dermatophytosis occurring endemically in certain regions, such as southern Asia (China and India), the islands of the South Pacific, and South and Central America.1 It usually affects glabrous skin and is exclusively caused by T. concentricum, a strictly anthropophilic dermatophyte. The morphology is characteristic, with initial lesions presenting as squamous annular, erythematous, concentric plaques that
Figure 4. Fungal culture on Sabouraud dextrose agar (SDA) showing flat, white, downy growth.
Figure 5. Lactophenol cotton blue preparation showing fine septate hyaline branched hyphae with sparse macroconidia and clustered microconidia along with well-developed spiral hyphae and chlamydospores, suggestive of Trichophyton mentagrophytes var. interdigitale (3400 original magnification).
later become lamellar with abundant thick scales adhering to one side, giving the appearance of tiles, fish scales, or lace.1 The term imbricata is derived from the Latin imbrex and refers to the similarity of the rash to overlapping roof tiles. Cases presenting with clinical features simulating TI but caused by species other than T. concentricum have been labeled ‘‘tinea indecisiva’’ by Batta and colleagues and ‘‘tinea pseudoimbricata’’ by Lim and Smith.2,3 Until now, only eight such cases have been reported in the literature (Table 1), of which four were caused by Trichophyton tonsurans and one each by Trichophyton rubrum, Microsporum audouinii, Microsporum gypseum, and T. mentagrophytes var. mentagrophytes.2–8 This case report is the first in which a different variant of T. mentagrophytes, that is, T. mentagrophytes var. interdigitale, was isolated as the etiologic agent. It is an anthropophilic dermatophyte and a frequent cause of tinea pedis, tinea manuum, and onychomycosis. With the exception of the case reported by Narang and colleagues, in which the scalp was involved,8 lesions were present over the trunk and/or limbs in other cases.2–7 The current patient presented with extensive tinea cruris and tinea corporis primarily involving the groin, genitalia, and buttocks, manifesting as tinea indecisiva. Although there were typical
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174 1
1 1 1 1 1
Lim and Smith, 20033
Ouchi et al, 20054 Poonawalla et al, 20065
Hoque and Holden, 20076 Poziomczyk et al, 20127 Narang et al, 20108
Sonthalia et al, 2010 (present case)
2
3 4
5 6 7
8
20/M
44/F 21/F 35/F
28/M 55/M
14/F
1. NM 2. NM
Age/Sex
HIV 5 human immunodeficiency virus; NM 5 not mentioned.
1
2
Number of Patients
Batta et al, 20022
Authors and Year of publication
1
Serial No.
Groin, penile shaft, lower abdomen, and lower limbs
Neck and trunk Extensive involvement of trunk and limbs Trunk and limbs Trunk and lower limbs Scalp
Trunk and limbs
1. Arms, legs, and trunk 2. Legs
Distribution of Lesions
Topical and oral steroids Crusted scabies HIV-positive patient on antiretroviral therapy and co-trimoxazole for the last 15 days Topical steroid-antifungal combination therapy and oral steroids
Renal transplant patient on oral immunosuppressives (tacrolimus and azathioprine) and topical antifungals Topical steroids for 3 mo Mycosis fungoides refractory to therapy
Topical steroids and antifungals
Associated Comorbidity and/or Previous Therapy
Table 1. Summary of Reported Cases of Tinea Pseudoimbricata (Indecisiva), Including the Present Case
T. mentagrophytes var. interdigitale
T. tonsurans Microsporum gypseum Microsporum audouinii
T. tonsurans Trichophyton rubrum
1. Trichophyton mentagrophytes var. mentagrophytes 2. Trichophyton tonsurans T. tonsurans
Species Isolated
Sonthalia et al
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annular, concentrically placed lesions forming a lamellar network, as seen in TI, there was prominent postulation and less scaling in the present case. The immunologic response to dermatophytes depends on two factors: the host’s cell-mediated immunity (CMI) and the species of dermatophyte involved. The host’s CMI mediated by T-cell reactivity forms the primary defense against dermatophytes. Although most patients usually develop specific antifungal antibodies to a particular dermatophyte after infection, these antibodies are largely nonprotective. Many characteristic observations from patients infected with TI indicate that effective CMI does not develop or is suppressed during the course of infection. These include the endemicity of TI in specific geographic locations, indicating inherent susceptibility of the resident population, acquisition of infection in early childhood, a lack of spontaneous remissions, and almost invariable relapses occurring after an initial response to antifungal therapy. This is in striking contrast to the infection of a man with the cattle ringworm T. verrucosum, where lifelong immunity appears to follow initial infection, indicating the additional role of the species of dermatophyte involved.9 The formation of concentric rings with the clinical ‘‘ring-within-a-ring’’ appearance and the development of sequential waves of scaling encountered in TI has been postulated to arise due to immunologic factors. In a study by Hay and colleagues conducted in Papua New Guinea, 78% of patients (with TI) investigated had antibody to T. concentricum and demonstrated either immediate-type hypersensitivity (52%) or negative responses (46%) to intradermal trichophytin.9 The majority of patients failed to develop delayed-type hypersensitivity on both in vivo skin testing and in vitro assessment by leukocyte migration inhibition. Based on these findings, the relevance of negative delayed-type hypersensitivity to the T. concentricum cytoplasmic antigen and T-lymphocyte hyporeactivity of the host, leading to the persistence of the infection, has been emphasized.9 However, in our case, the patient did not want to have the trichophytin skin testing. Moreover, the interpretation of trichophytin is an investigation with very variable responses. The interpretation of the immediate- as well as the delayed-type reactivity and its clinical significance have not been confidently established. The principle behind the occurrence of clinically similar lesions in patients infected with non-concentricum species of Trichophyton (ie, tinea pseudoimbricata) is at present speculative. But there is a strong case favoring the role of local or systemic immunosuppression in its pathogenesis as well, similar to the phenomenon postulated
for TI. All of the previously reported cases of tinea pseudoimbricata had some form of relative immunosuppression, the most common being prolonged use of sequential or cyclical topical steroids with or without oral steroids in four of the eight reported cases.2,4,6 Although the patients reported by Batta and colleagues had used topical antifungals in addition to topical steroids, no patient had received oral antifungals.2 Batta and colleagues attributed the occurrence of tinea pseudoimbricata to the combination of two factors: local immunosuppression due to prolonged topical steroid use and reinfection due to early discontinuation of topical antifungals. In the current case too, prolonged intermittent use of a combination of superpotent topical steroid and antifungal cream along with oral corticosteroids resulted in a state of local as well as systemic immunosuppression. The patients reported by Lim and Smith, Poonawalla and colleagues, and Narang and colleagues were also immmunosuppressed: a renal transplant–recipient child on oral immunosuppressives (tacrolimus plus azathioprine), an adult male with treatment-refractory mycosis fungoides, and an adult HIV-positive female with a CD4 count of 99 cells/mL on antiretroviral therapy, respectively.3,5,8 Although Poziomczyk and colleagues described their case as ‘‘immunocompetent,’’ their patient had apparently not been more thoroughly evaluated for evidence of immunosuppression and suffered from concomitant crusted scabies.7 Crusted scabies characterized by high mite burdens and extensive cutaneous crusting is known to occur in individuals who mount a vehement allergic response to mite infestation, but their strong IgE-driven T-helper 2 response and other humoral responses are totally nonprotective, resulting in uncontrolled growth of the parasite.10 Thus, irrespective of the lesional distribution and species involved in cases of tinea indecisiva, the immunopathogenesis of this peculiar condition is best explained as a simulation of T-cell hyporeactivity and the lack of delayed-type hypersensitivity to the dermatophyte, as encountered in TI. The differentiation between tinea corporis/cruris (unaltered by any local or systemic immunomodulators), tinea indecisiva, and tinea incognito is important. Tinea corporis and tinea cruris in an immunocompetent individual typically present with annular lesions with an erythematous scaly border and central clearing on most skin surfaces, except the scalp, volar areas, and nails. Dermatophytic infections are fairly common, especially in tropical countries, with a higher incidence during the hot, humid season. Treatment with topical imidazoles or allylamines for 2 to 4 weeks is often sufficient. Patients with diabetes are likely to present with extensive lesions.
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For extensive tinea corporis/cruris, oral antifungals such as terbinafine, fluconazole, or itraconazole have to be given for up to 4 weeks. The overall prognosis is excellent in unaltered dermatophytic infections. Cases of tinea incognito (also called tinea atypica), best defined as dermatophytic infections with altered clinical presentation following inappropriate use of systemic or topical corticosteroids or topical calcineurin inhibitors (CNIs), are on the rise. Easy accessibility of over-the-counter high-potency topical steroids in some countries and their inappropriate use by physicians without fungal testing have contributed to this trend. Immunomodulation by corticosteroids or CNIs not only leads to pathomorphosis and misdiagnosis due to simulation of other dermatoses; the course of infection is also further complicated by delayed recovery. Although tinea incognito commonly displays subdued scaling and loss of annular character of lesions, it may simulate a myriad of other clinical conditions, such as impetigo, rosacea, dermatitis, lupus erythematosus, polymorphous light eruption, psoriasis, and widespread folliculitis (Majocchi granuloma–like presentation), among others.11 Tinea indecisiva, on the other hand, presents with sequentially appearing concentric rings and scaling, giving rise to the characteristic ring-within-a-ring appearance. A high index of clinical suspicion and direct microscopic examination of skin scrapings with KOH and fungal culture on SDA are vital for the diagnosis of both tinea incognito and tinea indecisiva. Treatment of tinea incognito consists of combined use of oral and topical antifungals for at least 4 weeks. The prognosis is good if treatment is complied with and further abuse of immunosuppressives is halted. Owing to the paucity of published literature on tinea indecisiva, there is no current consensus on its standard treatment protocol. However, 4 weeks of oral terbinafine seems to be the most effective and sufficient therapy, with an excellent prognosis.
Conclusion Any patient presenting with such a ring-within-a-ring morphology of pustular and/or scaly lesions, that is, TIlike lesions, should always be investigated with fungal
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cultures for speciation. With indiscriminate use of steroid creams by patients, at least in the Indian context, more such cases may surface from time to time. Their early identification and treatment are all that is required to take care of this otherwise simple dermatosis.
Acknowledgments Financial disclosure of authors and reviewers: None reported.
References 1. Bonifaz A, Archer-Dubon C, Sau´l A. Tinea imbricata or Tokelau. Int J Dermatol 2004;43:506–10, doi:10.1111/j.1365-4632.2004.02171.x. 2. Batta K, Ramlogan D, Smith AG, et al. ‘Tinea indecisiva’ may mimic the concentric rings of tinea imbricata. Br J Dermatol 2002; 147:384, doi:10.1046/j.1365-2133.2002.04767.x. 3. Lim SP, Smith AG. ‘‘Tinea pseudoimbricata’’: tinea corporis in a renal transplant recipient mimicking the concentric rings of tinea imbricata. Clin Exp Dermatol 2003;28:332–3, doi:10.1046/j.13652230.2003.01281.x. 4. Ouchi T, Nagao K, Hata Y, et al. Trichophyton tonsurans infection manifesting as multiple concentric annular erythemas. J Dermatol 2005;32:565–8, doi:10.1111/j.1346-8138.2005.tb00799.x. 5. Poonawalla T, Chen W, Duvic M. Mycosis fungoides with tinea pseudoimbricata owing to Trichophyton rubrum infection. J Cutan Med Surg 2006;10:52–6. 6. Hoque SR, Holden CA. Trichophyton tonsurans infection mimicking tinea imbricata. Clin Exp Dermatol 2007;32:345–6, doi:10.1111/ j.1365-2230.2006.02337.x. 7. Poziomczyk CS, Ko¨che B, Becker FL, et al. Tinea pseudoimbricata caused by M. gypseum associated to crusted scabies. An Bras Dermatol 2010;85:558–9, doi:10.1590/S0365-05962010000400022. 8. Narang K, Pahwa M, Ramesh V. Tinea capitis in the form of concentric rings in an HIV positive adult on antiretroviral treatment. Indian J Dermatol 2012;57:288–90, doi:10.4103/0019-5154.97672. 9. Hay RJ, Reid S, Talwat E, Macnamara K. Immune responses of patients with tinea imbricata. Br J Dermatol 1983;108:581–6, doi:10.1111/j.1365-2133.1983.tb01060.x. 10. Walton SF, Pizzutto S, Slender A, et al. Increased allergic immune response to Sarcoptes scabiei antigens in crusted versus ordinary scabies. Clin Vaccine Immunol 2010;17:1428–38, doi:10.1128/CVI. 00195-10. 11. Zisova LG, Dobrev HP, Tchernev G, et al. Tinea atypica: report of nine cases. Wien Med Wochenschr 2013;163:549–55, doi:10.1007/ s10354-013-0230-4.
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Tinea Cruris and Tinea Corporis Masquerading as Tinea Indecisiva: Case Report and Review of the Literature Sidharth Sonthalia, Archana Singal, and Shukla Das Background: Tinea indecisiva is characterized by concentric scaly rings simulating tinea imbricata but caused by dermatophytes other than Trichophyton concentricum. Objective: Tinea indecisiva has been rarely reported. We report a unique case and review of the previously reported cases, pathogenesis, and management. Methods: An adult Indian man developed extensive tinea cruris and tinea corporis with concentric rings of scaly lesions over the groin, buttocks, and thighs following the use of oral corticosteroids and antifungal-steroid cream for 3 months. Mycologic and immunologic studies were performed for diagnosis. Results: Diagnosis of tinea indecisiva was confirmed on the appearance of ‘‘ring-within-a-ring’’ lesions clinically and isolation of Trichophyton mentagrophytes var. interdigitale as the etiologic agent on mycologic testing. Intradermal testing with Trichophyton extract showed fluctuating hypersensitivity responses. Four-week treatment with daily oral terbinafine resulted in complete resolution. Conclusion: Tinea indecisiva should be considered in a patient with tinea imbricata–like lesions with local immunosuppression caused by a non-concentricum dermatophyte. Contexte: Tinea indecisiva se caracte´rise par des anneaux squameux, concentriques, simulant tinea imbricata, mais l’affection est cause´e par d’autres dermatophytes que Trichophyton concentricum. Objectif: Rare est la description de Tinea indecisiva dans la documentation; nous faisons e´tat ici d’un cas unique en son genre et passons en revue les cas expose´s ante´rieurement ainsi que leur pathogene`se et leur prise en charge. Me´thode: Un Indien adulte a consulte´ pour des zones e´tendues d’ecze´ma margine´ et d’herpe`s circine´, accompagne´s d’anneaux concentriques de le´sions squameuses touchant les aines, les fesses, et les cuisses, qui sont apparues a` la suite de la prise de corticoste´roı¨des oraux et de l’application d’une cre`me associant un antifongique et un ste´roı¨de pendant 3 mois. Des analyses mycologiques et immunologiques ont e´te´ effectue´es pour confirmer le diagnostic. Re´sultats: Le diagnostic de tinea indecisiva a e´te´ confirme´ par l’apparence clinique des le´sions en forme d’« anneau dans un anneau » et par la mise en e´vidence de l’agent causal, Trichophyton mentagrophytes, variante interdigitale, a` l’examen mycologique. Des e´preuves intradermiques d’extraits de Trichophyton ont re´ve´le´ des re´actions d’hypersensibilite´ de type variable. La prise quotidienne de terbinafine orale, durant 4 semaines, a permis la disparition comple`te des le´sions. Conclusions: Le diagnostic de tinea indecisiva devrait eˆtre envisage´ chez les patients pre´sentant des le´sions ressemblant a` celles de tinea imbricata, accompagne´es d’une immunode´pression locale, mais cause´es par un dermatophyte de type non concentricum.
INEA IMBRICATA (TI) caused by Trichophyton concentricum is a chronic dermatophytosis characterized clinically by the presence of multiple annular concentric plaques that later become lamellar with abundant thick scales, giving the appearance of tiles or fish scales.1 TI occurs endemically in certain regions, especially southern Asia. Although TI is caused exclusively by T. concentricum, recently, a few cases have been reported that presented with clinical features simulating TI but caused by species other than T. concentricum. Such peculiar cases have been labeled as ‘‘tinea indecisiva’’ and
T
From SKINNOCENCE: The Skin Clinic & Research Centre, Haryana, India, and Departments of Dermatology and STD and Microbiology, University College of Medical Sciences and Guru Teg Bahadur Hospital, University of Delhi, Delhi, India. Address reprint requests to: Archana Singal, MD, MNAMS, B-14, Law Apartments, Karkardooma, Delhi-110092, India; e-mail: archanasingal@ hotmail.com.
DOI 10.2310/7750.2014.14057 # 2014 Canadian Dermatology Association
Canadian Dermatology Association | Journal of Cutaneous Medicine and Surgery, Vol 19, No 2 (March/April), 2015: pp 171–176
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‘‘tinea pseudoimbricata’’ and tend to occur in patients with some degree of immunosuppression.2,3 To the best of our knowledge, only eight such cases have been published to date, each occurring in a different clinical setting with different lesional localization and caused by different dermatophytic species.2–8 In this report, we describe another unique case of tinea indecisiva with the following special characteristics: involvement of the groin, penile shaft, and buttocks (previous cases have primarily involved the trunk, limbs, or scalp) and caused by Trichophyton mentagrophytes var. interdigitale (never described before). We further present a thorough review of previously reported cases of this condition, with an emphasis on the putative pathogenesis. Figure 1. Erythematous, annular, concentric rings with scattered pustules studded over the rings and grayish-white scales overlying the central relatively clear zone, involving the groin and penile shaft.
Case Report An 18-year-old adult Indian male presented with a 3month history of persistent itchy rash over the groin, lower abdomen, and thighs. He gave a history of intermittent application of an over-the-counter antifungal-steroid combination cream (containing miconazole nitrate 2% and clobetasol dipropionate 0.05%) twice daily, oral levocetirizine 5 mg every night, and oral betamethasone (2 mg) once a day for the past 3 months, which resulted in temporary relief of itching and partial resolution of lesions followed by progressive worsening. He had not sought a dermatologic consultation until then and had no history of having taken any oral antifungal drugs. He had no history of diabetes or any other significant medical disorder. A history of high-risk behavior (eg, multiple sex partners and intravenous drug abuse) for acquisition of acquired immunodeficiency was not elicited. A family history of diabetes or recurrent skin infections was absent. His physical and systemic examinations were normal. A cutaneous examination revealed the presence of widespread erythematous, annular, concentric rings with scattered pustules studded over the rings and grayishwhite scales overlying the central relatively clear zone, involving the groin and penile shaft (Figure 1), buttocks (Figure 2), thighs (Figure 3), and lower abdomen, clinically mimicking TI. Examination of the palms and soles, nails, and scalp revealed no abnormality. Hematologic and biochemical investigations, including hemography, fasting and postprandial plasma glucose, and renal and hepatic function tests, were normal. Serology for human immunodeficiency virus (HIV) was negative. Microscopic examination of skin scrapings with 10% potassium hydroxide (KOH) revealed thin hyaline hyphae suggestive of dermatophytic infection. Fungal culture on Sabouraud 172
dextrose agar (SDA) showed flat, white, downy growth (Figure 4). Lactophenol cotton blue preparation revealed fine septate hyaline branched hyphae with sparse macroconidia and clustered microconidia along with welldeveloped spiral hyphae and chlamydospores (Figure 5), confirming T. mentagrophytes var. interdigitale as the etiologic agent. Serologic testing for IgE immunoglobulin against Trichophyton antigen was strongly positive. A final diagnosis of tinea cruris and tinea corporis manifesting as tinea indecisiva (pseudoimbricata) was made. The patient was treated with daily oral terbinafine 250 mg for 4 weeks and oral levocetirizine 5 mg for 2 weeks. Oral steroids were tapered off over the next 6 weeks. The rash completely subsided within a month. The patient came for his last follow-up visit around 6 months after the initial episode, and there was no recurrence until then.
Figure 2. Similar lesions over the buttocks but with fewer pustules and marked scaling.
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Figure 3. Similar lesions over the thighs.
Discussion The characteristic clinical picture seen in our patient simulated features of TI. Tinea imbricata or Tokelau is a chronic dermatophytosis occurring endemically in certain regions, such as southern Asia (China and India), the islands of the South Pacific, and South and Central America.1 It usually affects glabrous skin and is exclusively caused by T. concentricum, a strictly anthropophilic dermatophyte. The morphology is characteristic, with initial lesions presenting as squamous annular, erythematous, concentric plaques that
Figure 4. Fungal culture on Sabouraud dextrose agar (SDA) showing flat, white, downy growth.
Figure 5. Lactophenol cotton blue preparation showing fine septate hyaline branched hyphae with sparse macroconidia and clustered microconidia along with well-developed spiral hyphae and chlamydospores, suggestive of Trichophyton mentagrophytes var. interdigitale (3400 original magnification).
later become lamellar with abundant thick scales adhering to one side, giving the appearance of tiles, fish scales, or lace.1 The term imbricata is derived from the Latin imbrex and refers to the similarity of the rash to overlapping roof tiles. Cases presenting with clinical features simulating TI but caused by species other than T. concentricum have been labeled ‘‘tinea indecisiva’’ by Batta and colleagues and ‘‘tinea pseudoimbricata’’ by Lim and Smith.2,3 Until now, only eight such cases have been reported in the literature (Table 1), of which four were caused by Trichophyton tonsurans and one each by Trichophyton rubrum, Microsporum audouinii, Microsporum gypseum, and T. mentagrophytes var. mentagrophytes.2–8 This case report is the first in which a different variant of T. mentagrophytes, that is, T. mentagrophytes var. interdigitale, was isolated as the etiologic agent. It is an anthropophilic dermatophyte and a frequent cause of tinea pedis, tinea manuum, and onychomycosis. With the exception of the case reported by Narang and colleagues, in which the scalp was involved,8 lesions were present over the trunk and/or limbs in other cases.2–7 The current patient presented with extensive tinea cruris and tinea corporis primarily involving the groin, genitalia, and buttocks, manifesting as tinea indecisiva. Although there were typical
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174 1
1 1 1 1 1
Lim and Smith, 20033
Ouchi et al, 20054 Poonawalla et al, 20065
Hoque and Holden, 20076 Poziomczyk et al, 20127 Narang et al, 20108
Sonthalia et al, 2010 (present case)
2
3 4
5 6 7
8
20/M
44/F 21/F 35/F
28/M 55/M
14/F
1. NM 2. NM
Age/Sex
HIV 5 human immunodeficiency virus; NM 5 not mentioned.
1
2
Number of Patients
Batta et al, 20022
Authors and Year of publication
1
Serial No.
Groin, penile shaft, lower abdomen, and lower limbs
Neck and trunk Extensive involvement of trunk and limbs Trunk and limbs Trunk and lower limbs Scalp
Trunk and limbs
1. Arms, legs, and trunk 2. Legs
Distribution of Lesions
Topical and oral steroids Crusted scabies HIV-positive patient on antiretroviral therapy and co-trimoxazole for the last 15 days Topical steroid-antifungal combination therapy and oral steroids
Renal transplant patient on oral immunosuppressives (tacrolimus and azathioprine) and topical antifungals Topical steroids for 3 mo Mycosis fungoides refractory to therapy
Topical steroids and antifungals
Associated Comorbidity and/or Previous Therapy
Table 1. Summary of Reported Cases of Tinea Pseudoimbricata (Indecisiva), Including the Present Case
T. mentagrophytes var. interdigitale
T. tonsurans Microsporum gypseum Microsporum audouinii
T. tonsurans Trichophyton rubrum
1. Trichophyton mentagrophytes var. mentagrophytes 2. Trichophyton tonsurans T. tonsurans
Species Isolated
Sonthalia et al
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Tinea Indecisiva
annular, concentrically placed lesions forming a lamellar network, as seen in TI, there was prominent postulation and less scaling in the present case. The immunologic response to dermatophytes depends on two factors: the host’s cell-mediated immunity (CMI) and the species of dermatophyte involved. The host’s CMI mediated by T-cell reactivity forms the primary defense against dermatophytes. Although most patients usually develop specific antifungal antibodies to a particular dermatophyte after infection, these antibodies are largely nonprotective. Many characteristic observations from patients infected with TI indicate that effective CMI does not develop or is suppressed during the course of infection. These include the endemicity of TI in specific geographic locations, indicating inherent susceptibility of the resident population, acquisition of infection in early childhood, a lack of spontaneous remissions, and almost invariable relapses occurring after an initial response to antifungal therapy. This is in striking contrast to the infection of a man with the cattle ringworm T. verrucosum, where lifelong immunity appears to follow initial infection, indicating the additional role of the species of dermatophyte involved.9 The formation of concentric rings with the clinical ‘‘ring-within-a-ring’’ appearance and the development of sequential waves of scaling encountered in TI has been postulated to arise due to immunologic factors. In a study by Hay and colleagues conducted in Papua New Guinea, 78% of patients (with TI) investigated had antibody to T. concentricum and demonstrated either immediate-type hypersensitivity (52%) or negative responses (46%) to intradermal trichophytin.9 The majority of patients failed to develop delayed-type hypersensitivity on both in vivo skin testing and in vitro assessment by leukocyte migration inhibition. Based on these findings, the relevance of negative delayed-type hypersensitivity to the T. concentricum cytoplasmic antigen and T-lymphocyte hyporeactivity of the host, leading to the persistence of the infection, has been emphasized.9 However, in our case, the patient did not want to have the trichophytin skin testing. Moreover, the interpretation of trichophytin is an investigation with very variable responses. The interpretation of the immediate- as well as the delayed-type reactivity and its clinical significance have not been confidently established. The principle behind the occurrence of clinically similar lesions in patients infected with non-concentricum species of Trichophyton (ie, tinea pseudoimbricata) is at present speculative. But there is a strong case favoring the role of local or systemic immunosuppression in its pathogenesis as well, similar to the phenomenon postulated
for TI. All of the previously reported cases of tinea pseudoimbricata had some form of relative immunosuppression, the most common being prolonged use of sequential or cyclical topical steroids with or without oral steroids in four of the eight reported cases.2,4,6 Although the patients reported by Batta and colleagues had used topical antifungals in addition to topical steroids, no patient had received oral antifungals.2 Batta and colleagues attributed the occurrence of tinea pseudoimbricata to the combination of two factors: local immunosuppression due to prolonged topical steroid use and reinfection due to early discontinuation of topical antifungals. In the current case too, prolonged intermittent use of a combination of superpotent topical steroid and antifungal cream along with oral corticosteroids resulted in a state of local as well as systemic immunosuppression. The patients reported by Lim and Smith, Poonawalla and colleagues, and Narang and colleagues were also immmunosuppressed: a renal transplant–recipient child on oral immunosuppressives (tacrolimus plus azathioprine), an adult male with treatment-refractory mycosis fungoides, and an adult HIV-positive female with a CD4 count of 99 cells/mL on antiretroviral therapy, respectively.3,5,8 Although Poziomczyk and colleagues described their case as ‘‘immunocompetent,’’ their patient had apparently not been more thoroughly evaluated for evidence of immunosuppression and suffered from concomitant crusted scabies.7 Crusted scabies characterized by high mite burdens and extensive cutaneous crusting is known to occur in individuals who mount a vehement allergic response to mite infestation, but their strong IgE-driven T-helper 2 response and other humoral responses are totally nonprotective, resulting in uncontrolled growth of the parasite.10 Thus, irrespective of the lesional distribution and species involved in cases of tinea indecisiva, the immunopathogenesis of this peculiar condition is best explained as a simulation of T-cell hyporeactivity and the lack of delayed-type hypersensitivity to the dermatophyte, as encountered in TI. The differentiation between tinea corporis/cruris (unaltered by any local or systemic immunomodulators), tinea indecisiva, and tinea incognito is important. Tinea corporis and tinea cruris in an immunocompetent individual typically present with annular lesions with an erythematous scaly border and central clearing on most skin surfaces, except the scalp, volar areas, and nails. Dermatophytic infections are fairly common, especially in tropical countries, with a higher incidence during the hot, humid season. Treatment with topical imidazoles or allylamines for 2 to 4 weeks is often sufficient. Patients with diabetes are likely to present with extensive lesions.
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For extensive tinea corporis/cruris, oral antifungals such as terbinafine, fluconazole, or itraconazole have to be given for up to 4 weeks. The overall prognosis is excellent in unaltered dermatophytic infections. Cases of tinea incognito (also called tinea atypica), best defined as dermatophytic infections with altered clinical presentation following inappropriate use of systemic or topical corticosteroids or topical calcineurin inhibitors (CNIs), are on the rise. Easy accessibility of over-the-counter high-potency topical steroids in some countries and their inappropriate use by physicians without fungal testing have contributed to this trend. Immunomodulation by corticosteroids or CNIs not only leads to pathomorphosis and misdiagnosis due to simulation of other dermatoses; the course of infection is also further complicated by delayed recovery. Although tinea incognito commonly displays subdued scaling and loss of annular character of lesions, it may simulate a myriad of other clinical conditions, such as impetigo, rosacea, dermatitis, lupus erythematosus, polymorphous light eruption, psoriasis, and widespread folliculitis (Majocchi granuloma–like presentation), among others.11 Tinea indecisiva, on the other hand, presents with sequentially appearing concentric rings and scaling, giving rise to the characteristic ring-within-a-ring appearance. A high index of clinical suspicion and direct microscopic examination of skin scrapings with KOH and fungal culture on SDA are vital for the diagnosis of both tinea incognito and tinea indecisiva. Treatment of tinea incognito consists of combined use of oral and topical antifungals for at least 4 weeks. The prognosis is good if treatment is complied with and further abuse of immunosuppressives is halted. Owing to the paucity of published literature on tinea indecisiva, there is no current consensus on its standard treatment protocol. However, 4 weeks of oral terbinafine seems to be the most effective and sufficient therapy, with an excellent prognosis.
Conclusion Any patient presenting with such a ring-within-a-ring morphology of pustular and/or scaly lesions, that is, TIlike lesions, should always be investigated with fungal
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cultures for speciation. With indiscriminate use of steroid creams by patients, at least in the Indian context, more such cases may surface from time to time. Their early identification and treatment are all that is required to take care of this otherwise simple dermatosis.
Acknowledgments Financial disclosure of authors and reviewers: None reported.
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