Majocchi Granuloma in a Pregnant Woman
Although the fungus usually does not invade beyond the epidermis, dermal infection does occur in this disorder. This is thought to be related to either being immunocompromised or trauma. We present Majocchi granuloma beginning in early pregnancy in a patient with no history of trauma or being immunocompromised by medication or chronic illness.
Ruihua Wang, MS, Yong Hu, MS, Hongfeng Tang, PhD, and Tangde Zhang, MS
CASE
BACKGROUND: Majocchi granuloma is a cutaneous dermatophyte infection of the dermal and subcutaneous tissues characterized by chronic erythematous and indurated plaques, which are almost always the result of immunodeficiency and trauma. CASE: Disseminated dermatophyte Majocchi granuloma was diagnosed in a 19-year-old woman at 32 weeks of gestation and had no history of trauma or chronic illness. She was treated with topical antifungal medications until delivery, at which time systemic medications were started. The skin lesions resolved by the time of her 6-week postpartum visit. CONCLUSION: The altered immune state of pregnancy suggests that Majocchi granuloma should occur with some regularity in pregnancy. That this case is the second one described in the literature suggests that it may be underdiagnosed. Punch biopsy should be considered for diagnosis of unusual skin lesions during pregnancy. (Obstet Gynecol 2014;124:423–5) DOI: 10.1097/AOG.0000000000000302
M
ajocchi granuloma is a term applied to a cutaneous dermatophyte (fungal) infection of the dermal and subcutaneous tissues characterized by chronic erythematous and indurated plaques.1 This infection can occur in both immunocompetent and immunocompromised hosts and is most commonly seen in solid organ transplant recipients or in areas of previous injury.2,3 The initiating factor is thought to be an infected follicle in the dermis or subcutaneous tissue,1 complicated by compromise of phagocytic function.4 Trichophyton rubrum is the most commonly isolated fungus in dermatophytes from Majocchi granuloma. From the Departments of Dermatology, the First People’s Hospital of Shunde, Foshan, and Zhujiang Hospital, Guangzhou, China. The authors thank Xiufen Zheng and Ling Huang for their assistance with proofreading. Corresponding author: Hongfeng Tang, No. 1, Peng Lai, Daliang District, Foshan 528300, China; e-mail: [email protected]. Financial Disclosure The authors did not report any potential conflicts of interest. © 2014 by The American College of Obstetricians and Gynecologists. Published by Lippincott Williams & Wilkins. ISSN: 0029-7844/14
VOL. 124, NO. 2, PART 2, AUGUST 2014
A 19-year-old previously healthy woman, gravida 1 para 0, came to the outpatient area of our hospital at 32 weeks of gestation for evaluation of multiple skin lesions. These had begun 6 months previously on her right thigh and back of her right hand, and 3 months later they spread to her abdomen and left facial area. According to the patient, the initial lesions consisted of small itchy annular plaques that evolved to exfoliation and growing papules and pustules after scratching. She had received no treatment previously for these lesions. She denied trauma, use of steroids, history of fungal skin infections, and chronic medical disorders. Family history was negative. The examination of the lesions revealed indurated, welldemarcated erythematous plaques varying in size from 5 cm35 cm to 12 cm315 cm with irregular borders. The plaques were overlaid by multiple pink papules that were 0.5 mm to 2 mm in diameter on the frontofacial area abdomen and thigh and by yellow pustules on the abdomen (Fig. 1). The physical examination revealed that the patient was afebrile and had no lymphadenopathy in the inguinal, cervical, or axillary areas. The symphysis fundus height of 30 cm was consistent with a 32-week pregnancy. She felt pain on the forehead and intermittent itching on the other lesions. Laboratory investigation showed normal values for lymphocyte T and B subpopulations, and serology tests for all known blood-borne infections and sexually transmitted diseases all yielded normal results. The clinical and ultrasonographic examinations revealed no abnormalities of the embryo or genital tract. A punch biopsy was performed on the frontofacial area and the sample was subjected to periodic acid Schiff staining and culture. The skin biopsy sample demonstrated stratum spinosum cell hyperplasia, chronic inflammation around the subcuticular blood vessels, and mixed granulomas with the infiltration of neutrophils and plasmocytes. The periodic acid Schiff staining was positive for fungal elements (Fig. 2). Trichophyton mentagrophytes was isolated from the culture of the biopsy material on potato dextrose agar medium and sabouraud medium. On direct microscopic examination of the thigh, hand, abdomen and frontofacial area, most of the fungal hyphae could be observed by the potassium hydroxide test. Diagnosis of Majocchi granuloma was made and she was treated with topical antifungal bifonazole until delivery. Her itch was relieved and lesions had mild improvement after topical treatment. At 39 weeks of gestation, she underwent cesarean delivery and delivered an appropriate-for-gestational-age neonate. The wound recovered well after the operation.
Wang et al
Majocchi Granuloma in a Pregnant Woman
423
Fig. 1. Well-demarcated erythematous plaques varying in size from 535 cm to 12315 cm with irregular borders on the left frontofacial area (A), abdomen (B), right thigh (C), and back of right hand (D). The plaques were overlaid with multiple pink papules that were 0.5 mm to 2 mm in diameter on the frontofacial area (A), abdomen (B), and thigh (C), with yellow pustules on the abdomen (B). Wang. Majocchi Granuloma in a Pregnant Woman. Obstet Gynecol 2014.
Postoperatively, she was treated with oral itraconazole 200 mg daily for 6 weeks. The skin lesions gradually resolved and were almost healed by 6 weeks without any scarring. She remained well 6 months after the treatment and has had no recurrence.
COMMENT Majocchi granuloma was first described in 1883 by Professor Domenico Majocchi (1849–1929) as an intracutaneous or subcutaneous granulomatous inflammation that arose as a result of invasion by a dermatophytic fungus, such as anthropophilic Trichophyton rubrum. Species from the Aspergillus and Phoma genera occasionally have been detected as etiologic agents of Majocchi granuloma.5 Majocchi granuloma clinically presents with superficial perifollicular granuloma in healthy individuals, which is mainly on the legs, especially in women who shave their legs,6 or with deep plaques or nodular lesions in immunocompromised hosts, which is characterized by the presence of inflammatory papular, pustular, or nodular lesions, usually on the limbs.6,7 Nodules may develop in any hair-bearing part of the body but are most often observed on the forearms, hands, and legs of the infected individuals. Majocchi granuloma has been described in patients with a variety of immunocompromised states, including malnutrition, leukemia, lymphoma, acquired immunodeficiency syndrome, Cushings syndrome, and after solid organ transplantation.1,8
424
Wang et al
A history of physical trauma with introduction of dermatophyte directly into the dermis or indirectly through disrupted hair follicles has also been described.2,7,9 Generally, the source of the infection can be found in the patient’s skin or nails.3,7 However, most cases that have been described in the literature were not associated with tinea pedis.10,11 Involvement of the scalp and face is rarely observed. In a PubMed search with no additional filters that included publication dates, the terms “Majocchi granuloma or Majocchi granuloma” and “pregnancy or pregnant” did not locate any other reports of Majocchi granuloma in pregnancy. After a review of reference materials, we found a single reported case of Majocchi granuloma.1 That patient’s lesion predated pregnancy and was confined to her ankle. The lesion increased in size during pregnancy.12 The small number of reported cases of Majocchi granuloma in pregnancy is somewhat surprising given the well-described altered immune state of pregnancy. For instance, delayed-type skin test reactivity is decreased and impaired rejection of skin grafts, altered lymphocyte blastogenic responses, and the defective chemotaxis of phagocytic cells are known to occur.13,14 Pregnant women have an increased rate of Candida vaginitis. Coccidioidomycosis is believed to disseminate more frequently in pregnant females.15 Our patient lacked a history of immunosuppression. The extensive nature of the lesions suggests that trauma, especially with no history, is not the cause. An
Majocchi Granuloma in a Pregnant Woman
OBSTETRICS & GYNECOLOGY
Majocchi granuloma is an unusual fungal skin infection of the dermis, usually described after skin trauma or in immunocompromised individuals. The immune alterations in pregnancy reasonably suggest that pregnant women would be at increased risk for this disorder. Even so, this is only the second reported case in the literature. This suggests the possibility of underdiagnosis in pregnancy. In pregnant women with complex, inflammatory, and erythematous skin plaques, a punch biopsy should be considered to direct therapy. REFERENCES 1. Ilkit M, Durdu M, Karakas M. Majocchi’s granuloma: a symptom complex caused by fungal pathogens. Med Mycol 2012; 50:449–57. 2. Sequeira M, Burdick AE, Elgart GW, Berma B. New-onset Majocchi’s granuloma in two kidney transplant recipients under tacrolimus treatment. J Am Acad Dermatol 1998;38:486–8. 3. Kurian A, Haber RM. Tinea corporis gladiatorum presenting as a majocchi granuloma. ISRN Dermatol 2011;2011:767589. 4. Jones HE. Immune response and host resistance of humans to dermatophyte infection. J Am Acad Dermatol 1993;28:S12–8. 5. Majocchi D. Sepra una nuova tricofizia (granuloma tricofitico), studi clinici micologici. Bull R Acad Med Roma 1883;9:220. 6. Bressan AL, Silva RS, Fonseca JC, Alves Mde F. Majocchi’s granuloma. An Bras Dermatol 2011;86:797–8.
Fig. 2. Stratum spinosum cell hyperplasia and chronic inflammation around the subcuticular blood vessels showing mixed granulomas with the infiltration of neutrophils and plasmocytes (A) (340). Typical spores in the deep dermis were visualized by the periodic acid Schiff stain (B) (3200). Wang. Majocchi Granuloma in a Pregnant Woman. Obstet Gynecol 2014.
evaluation for occult infectious disease or abnormal Bcell and T-cell function was negative. It is reasonable to consider that the immune alteration of pregnancy contributed to her disease. In general, systemic antifungal medication has been limited to the treatment of life-threatening fungal infections because of the risk of toxicity for both mother and fetus. As such, we treated our patient with topical antifungal medication before delivery and then began systemic therapy postpartum. She chose not to breastfeed.
VOL. 124, NO. 2, PART 2, AUGUST 2014
7. Gill M, Sachdeva B, Gill PS, Arora B, Deep A, Karan J. Majocchi’s granuloma of the face in an immunocompetent patient. J Dermatol 2007;34:702–4. 8. Steiner UC, Trüeb RM, Schad K, Kamarashev J, Koch S, French LE, et al. Trichophyton rubrum-induced Majocchi’s Granuloma in a heart transplant recipient. A therapeutic challenge. J Dermatol 2012;28:70–2. 9. Chen HH, Chiu HC. Facial Majocchi’s granuloma caused by Trichophyton tonsurans in an immunocompetent patient. Acta Derm Venereol 2003;83:65–6. 10. Lepage JC. Source of Majocchi’s granuloma. J Am Acad Dermatol 1983;8:260. 11. Chang SE, Lee DK, Choi JH, Moon KC, Koh JK. Majocchi’s granuloma of the vulva caused by Trichophyton mentagrophytes. Mycoses 2005;48:382–4. 12. Allen GE. Majocchi’s granuloma. Br J Dermatol 1966;78:544. 13. Gall SA. Maternal adjustments in the immune system in normal pregnancy. Clin Obstet Gynecol 1983;26:521–36. 14. Sridama V, Pacini F, Yang SL, Moawad A, Reilly M, Degroot LJ, et al. Decreased levels of helper T cells: a possible cause of immunodeficiency in pregnancy. N Engl J Med 1982; 307:352–6. 15. Jack DS. Use of antifungal drugs in pregnancy. Drug Saf 2000; 23:77–85.
Wang et al
Majocchi Granuloma in a Pregnant Woman
425
Although the fungus usually does not invade beyond the epidermis, dermal infection does occur in this disorder. This is thought to be related to either being immunocompromised or trauma. We present Majocchi granuloma beginning in early pregnancy in a patient with no history of trauma or being immunocompromised by medication or chronic illness.
Ruihua Wang, MS, Yong Hu, MS, Hongfeng Tang, PhD, and Tangde Zhang, MS
CASE
BACKGROUND: Majocchi granuloma is a cutaneous dermatophyte infection of the dermal and subcutaneous tissues characterized by chronic erythematous and indurated plaques, which are almost always the result of immunodeficiency and trauma. CASE: Disseminated dermatophyte Majocchi granuloma was diagnosed in a 19-year-old woman at 32 weeks of gestation and had no history of trauma or chronic illness. She was treated with topical antifungal medications until delivery, at which time systemic medications were started. The skin lesions resolved by the time of her 6-week postpartum visit. CONCLUSION: The altered immune state of pregnancy suggests that Majocchi granuloma should occur with some regularity in pregnancy. That this case is the second one described in the literature suggests that it may be underdiagnosed. Punch biopsy should be considered for diagnosis of unusual skin lesions during pregnancy. (Obstet Gynecol 2014;124:423–5) DOI: 10.1097/AOG.0000000000000302
M
ajocchi granuloma is a term applied to a cutaneous dermatophyte (fungal) infection of the dermal and subcutaneous tissues characterized by chronic erythematous and indurated plaques.1 This infection can occur in both immunocompetent and immunocompromised hosts and is most commonly seen in solid organ transplant recipients or in areas of previous injury.2,3 The initiating factor is thought to be an infected follicle in the dermis or subcutaneous tissue,1 complicated by compromise of phagocytic function.4 Trichophyton rubrum is the most commonly isolated fungus in dermatophytes from Majocchi granuloma. From the Departments of Dermatology, the First People’s Hospital of Shunde, Foshan, and Zhujiang Hospital, Guangzhou, China. The authors thank Xiufen Zheng and Ling Huang for their assistance with proofreading. Corresponding author: Hongfeng Tang, No. 1, Peng Lai, Daliang District, Foshan 528300, China; e-mail: [email protected]. Financial Disclosure The authors did not report any potential conflicts of interest. © 2014 by The American College of Obstetricians and Gynecologists. Published by Lippincott Williams & Wilkins. ISSN: 0029-7844/14
VOL. 124, NO. 2, PART 2, AUGUST 2014
A 19-year-old previously healthy woman, gravida 1 para 0, came to the outpatient area of our hospital at 32 weeks of gestation for evaluation of multiple skin lesions. These had begun 6 months previously on her right thigh and back of her right hand, and 3 months later they spread to her abdomen and left facial area. According to the patient, the initial lesions consisted of small itchy annular plaques that evolved to exfoliation and growing papules and pustules after scratching. She had received no treatment previously for these lesions. She denied trauma, use of steroids, history of fungal skin infections, and chronic medical disorders. Family history was negative. The examination of the lesions revealed indurated, welldemarcated erythematous plaques varying in size from 5 cm35 cm to 12 cm315 cm with irregular borders. The plaques were overlaid by multiple pink papules that were 0.5 mm to 2 mm in diameter on the frontofacial area abdomen and thigh and by yellow pustules on the abdomen (Fig. 1). The physical examination revealed that the patient was afebrile and had no lymphadenopathy in the inguinal, cervical, or axillary areas. The symphysis fundus height of 30 cm was consistent with a 32-week pregnancy. She felt pain on the forehead and intermittent itching on the other lesions. Laboratory investigation showed normal values for lymphocyte T and B subpopulations, and serology tests for all known blood-borne infections and sexually transmitted diseases all yielded normal results. The clinical and ultrasonographic examinations revealed no abnormalities of the embryo or genital tract. A punch biopsy was performed on the frontofacial area and the sample was subjected to periodic acid Schiff staining and culture. The skin biopsy sample demonstrated stratum spinosum cell hyperplasia, chronic inflammation around the subcuticular blood vessels, and mixed granulomas with the infiltration of neutrophils and plasmocytes. The periodic acid Schiff staining was positive for fungal elements (Fig. 2). Trichophyton mentagrophytes was isolated from the culture of the biopsy material on potato dextrose agar medium and sabouraud medium. On direct microscopic examination of the thigh, hand, abdomen and frontofacial area, most of the fungal hyphae could be observed by the potassium hydroxide test. Diagnosis of Majocchi granuloma was made and she was treated with topical antifungal bifonazole until delivery. Her itch was relieved and lesions had mild improvement after topical treatment. At 39 weeks of gestation, she underwent cesarean delivery and delivered an appropriate-for-gestational-age neonate. The wound recovered well after the operation.
Wang et al
Majocchi Granuloma in a Pregnant Woman
423
Fig. 1. Well-demarcated erythematous plaques varying in size from 535 cm to 12315 cm with irregular borders on the left frontofacial area (A), abdomen (B), right thigh (C), and back of right hand (D). The plaques were overlaid with multiple pink papules that were 0.5 mm to 2 mm in diameter on the frontofacial area (A), abdomen (B), and thigh (C), with yellow pustules on the abdomen (B). Wang. Majocchi Granuloma in a Pregnant Woman. Obstet Gynecol 2014.
Postoperatively, she was treated with oral itraconazole 200 mg daily for 6 weeks. The skin lesions gradually resolved and were almost healed by 6 weeks without any scarring. She remained well 6 months after the treatment and has had no recurrence.
COMMENT Majocchi granuloma was first described in 1883 by Professor Domenico Majocchi (1849–1929) as an intracutaneous or subcutaneous granulomatous inflammation that arose as a result of invasion by a dermatophytic fungus, such as anthropophilic Trichophyton rubrum. Species from the Aspergillus and Phoma genera occasionally have been detected as etiologic agents of Majocchi granuloma.5 Majocchi granuloma clinically presents with superficial perifollicular granuloma in healthy individuals, which is mainly on the legs, especially in women who shave their legs,6 or with deep plaques or nodular lesions in immunocompromised hosts, which is characterized by the presence of inflammatory papular, pustular, or nodular lesions, usually on the limbs.6,7 Nodules may develop in any hair-bearing part of the body but are most often observed on the forearms, hands, and legs of the infected individuals. Majocchi granuloma has been described in patients with a variety of immunocompromised states, including malnutrition, leukemia, lymphoma, acquired immunodeficiency syndrome, Cushings syndrome, and after solid organ transplantation.1,8
424
Wang et al
A history of physical trauma with introduction of dermatophyte directly into the dermis or indirectly through disrupted hair follicles has also been described.2,7,9 Generally, the source of the infection can be found in the patient’s skin or nails.3,7 However, most cases that have been described in the literature were not associated with tinea pedis.10,11 Involvement of the scalp and face is rarely observed. In a PubMed search with no additional filters that included publication dates, the terms “Majocchi granuloma or Majocchi granuloma” and “pregnancy or pregnant” did not locate any other reports of Majocchi granuloma in pregnancy. After a review of reference materials, we found a single reported case of Majocchi granuloma.1 That patient’s lesion predated pregnancy and was confined to her ankle. The lesion increased in size during pregnancy.12 The small number of reported cases of Majocchi granuloma in pregnancy is somewhat surprising given the well-described altered immune state of pregnancy. For instance, delayed-type skin test reactivity is decreased and impaired rejection of skin grafts, altered lymphocyte blastogenic responses, and the defective chemotaxis of phagocytic cells are known to occur.13,14 Pregnant women have an increased rate of Candida vaginitis. Coccidioidomycosis is believed to disseminate more frequently in pregnant females.15 Our patient lacked a history of immunosuppression. The extensive nature of the lesions suggests that trauma, especially with no history, is not the cause. An
Majocchi Granuloma in a Pregnant Woman
OBSTETRICS & GYNECOLOGY
Majocchi granuloma is an unusual fungal skin infection of the dermis, usually described after skin trauma or in immunocompromised individuals. The immune alterations in pregnancy reasonably suggest that pregnant women would be at increased risk for this disorder. Even so, this is only the second reported case in the literature. This suggests the possibility of underdiagnosis in pregnancy. In pregnant women with complex, inflammatory, and erythematous skin plaques, a punch biopsy should be considered to direct therapy. REFERENCES 1. Ilkit M, Durdu M, Karakas M. Majocchi’s granuloma: a symptom complex caused by fungal pathogens. Med Mycol 2012; 50:449–57. 2. Sequeira M, Burdick AE, Elgart GW, Berma B. New-onset Majocchi’s granuloma in two kidney transplant recipients under tacrolimus treatment. J Am Acad Dermatol 1998;38:486–8. 3. Kurian A, Haber RM. Tinea corporis gladiatorum presenting as a majocchi granuloma. ISRN Dermatol 2011;2011:767589. 4. Jones HE. Immune response and host resistance of humans to dermatophyte infection. J Am Acad Dermatol 1993;28:S12–8. 5. Majocchi D. Sepra una nuova tricofizia (granuloma tricofitico), studi clinici micologici. Bull R Acad Med Roma 1883;9:220. 6. Bressan AL, Silva RS, Fonseca JC, Alves Mde F. Majocchi’s granuloma. An Bras Dermatol 2011;86:797–8.
Fig. 2. Stratum spinosum cell hyperplasia and chronic inflammation around the subcuticular blood vessels showing mixed granulomas with the infiltration of neutrophils and plasmocytes (A) (340). Typical spores in the deep dermis were visualized by the periodic acid Schiff stain (B) (3200). Wang. Majocchi Granuloma in a Pregnant Woman. Obstet Gynecol 2014.
evaluation for occult infectious disease or abnormal Bcell and T-cell function was negative. It is reasonable to consider that the immune alteration of pregnancy contributed to her disease. In general, systemic antifungal medication has been limited to the treatment of life-threatening fungal infections because of the risk of toxicity for both mother and fetus. As such, we treated our patient with topical antifungal medication before delivery and then began systemic therapy postpartum. She chose not to breastfeed.
VOL. 124, NO. 2, PART 2, AUGUST 2014
7. Gill M, Sachdeva B, Gill PS, Arora B, Deep A, Karan J. Majocchi’s granuloma of the face in an immunocompetent patient. J Dermatol 2007;34:702–4. 8. Steiner UC, Trüeb RM, Schad K, Kamarashev J, Koch S, French LE, et al. Trichophyton rubrum-induced Majocchi’s Granuloma in a heart transplant recipient. A therapeutic challenge. J Dermatol 2012;28:70–2. 9. Chen HH, Chiu HC. Facial Majocchi’s granuloma caused by Trichophyton tonsurans in an immunocompetent patient. Acta Derm Venereol 2003;83:65–6. 10. Lepage JC. Source of Majocchi’s granuloma. J Am Acad Dermatol 1983;8:260. 11. Chang SE, Lee DK, Choi JH, Moon KC, Koh JK. Majocchi’s granuloma of the vulva caused by Trichophyton mentagrophytes. Mycoses 2005;48:382–4. 12. Allen GE. Majocchi’s granuloma. Br J Dermatol 1966;78:544. 13. Gall SA. Maternal adjustments in the immune system in normal pregnancy. Clin Obstet Gynecol 1983;26:521–36. 14. Sridama V, Pacini F, Yang SL, Moawad A, Reilly M, Degroot LJ, et al. Decreased levels of helper T cells: a possible cause of immunodeficiency in pregnancy. N Engl J Med 1982; 307:352–6. 15. Jack DS. Use of antifungal drugs in pregnancy. Drug Saf 2000; 23:77–85.
Wang et al
Majocchi Granuloma in a Pregnant Woman
425
![[Majocchi granuloma. Advantages of optical brightener staining in a case report].](/assets/img/hold.png)
