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doi:10.1111/jog.12530
J. Obstet. Gynaecol. Res. Vol. 41, No. 2: 301–303, February 2015
Case report of a rare dermatosis in pregnancy: Impetigo herpetiformis Mustafa Ulubay1, Ug˘ur Keskin1, Ulas Fidan1, Ali Fuat Çiçek2, Ercan Çalıs¸kan3, Rıza Efendi Karaca1, Fahri Burçin Fıratlıgil1 and Ali Ergün1 Departments of 1Obstetrics and Gynecology, 2Pathology and 3Dermatology, Gulhane Military Medical Academy, Ankara, Turkey
Abstract Impetigo herpetiformis (IH) is a very rare type of dermatosis seen in pregnancy. According to the published work, IH during pregnancy is associated with the risk of stillbirth, and obstetric management in such cases is very important. Early recognition is important to reduce both maternal and fetal morbidity. We present a case of IH resistant to corticosteroid therapy in a 27-year-old pregnant woman where the pregnancy was terminated by the induction of labor. Key words: dermatosis, impetigo herpetiformis, pregnancy.
Introduction
Case Report
Impetigo herpetiformis (IH) was first described by von Hebra in 1872.1 According to the published work, IH is a variant of generalized pustular psoriasis in pregnancy, characterized by an acute pustular eruption.2,3 It is a rare and potentially life-threatening condition, which affects pregnant women, in particular, in the third trimester.2 The exact mechanism underlying IH is unknown.4 Patients with IH often have systemic symptoms, including malaise, fewer, nausea, vomiting, diarrhea, chills and arthralgia. Laboratory findings are leukocytosis, an elevated erythrocyte sedimentation rate and hypocalcemia. Treatment involves i.v. fluid, electrolyte, calcium and vitamin D supplementation, and systemic and topical corticosteroids.
A 27-year-old 37-week pregnant patient was referred to our clinic with painful, itchy and desquamative macular lesions on her body that were accompanied by fatigue (Fig. 1). The lesions were predominately on her abdomen, groin and back. Some lesions were also present on the patient’s upper and lower extremities. Her medical history revealed that the lesions had commenced on her groin and then spread to the rest of her body at 35 weeks of gestation. Our patient or her relatives had no history of IH or pustular psoriasis. She had been treated for rheumatoid arthritis for 2 years with prednisolone before becoming pregnant. She had experienced an uncomplicated pregnancy before the skin disease started. She had normal vital signs. She was not a smoker. A blood examination revealed
Received: March 14 2014. Accepted: June 30 2014. Reprint request to: Assistant Professor Mustafa Ulubay, Gulhane Military Medical Academy, Obstetrics and Gynecology Department, General Tevfik Saglam Caddesi Etlik, Ankara 06100, Turkey. Email: [email protected] Author contribution: M. U., U. K., U. F. and E. Ç. drafted the manuscript and collected data related to the subject. A. F. Ç. and R. E. K. were involved in drafting the manuscript and in the critical revision of the draft. F. B. F. made a substantial contribution to the conception and design of the study. A. E. participated in the design of the manuscript and the coordination of the study. All authors read and approved the final manuscript.
© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology
301
M. Ulubay et al.
a
a
b
Figure 1 Appearance of impetigo herpetiformis on the patient’s body. The characteristic of lesions are macular desquamative pustular lesions. (a) Anterior, (b) posterior.
normal full blood. Renal, liver function and thyroid function tests were normal, as were her calcium levels. An oral glucose test was normal. She tested negative for Herpes simplex virus type 1 and type 2 and hepatitis B and C. A bacteriological examination of the pus was negative. She took oral iron and multivitamin supplements during her pregnancy. When she was admitted to the hospital, she was given 30 mg of oral prednisone, antihistamines and a topical steroid. Data guidelines are lacking on the treatment of IH in pregnant women. Topical steroids are usually the first choice if symptoms are light or mild. In severe cases, oral prednisone is prescribed at 30 mg/day up to 60 or 80 mg/day.5 Highdose prednisone can have adverse effects on the fetus, such as reactivity failure in fetal monitoring.6 Therefore, it was decided to treat the patient with prednisone 30 mg/day. An obstetric examination revealed a normal level of amniotic fluid, and biometric and Doppler measurements were within normal ranges. The patient was followed up for 5 days with non-stress tests and fetal movement counts. Despite the oral prednisolone therapy, the lesions spread. Due to the risk of stillbirth, labor was induced by cervical ripening and stripping and oxytocin infusion. The patient had a healthy 2620-g girl following a normal vaginal birth. The 1- and 5-min Apgar scores were 8 and 10, respectively. There was no dermatosis on the newborn. The patient received systemic corticosteroid treatment (30 mg prednisone) and topical corticosteroid (clobetasol propionate emollient 0.05%) during the post-partum period. Because prednisone can be
302
b
c
Figure 2 (a) Subcorneal pustule formation and vesiculation (hematoxylin–eosin [HE], original magnification ×100). (b) Granular layer is not seen in this section. There is superficial perivascular lymphocytic inflammatory infiltration in the papillary dermis. Inflammation consists of lymphocytes (HE, ×100). (c) There is no keratinocytes within the epidermis showing nuclear or cytoplasmic immunoreactivity for Herpes simplex virus (HSV)-2 rabbit polyclonal antibody by immunohistochemistry (×100).
excreted into breast milk and may have adverse effects on the newborn, the dose of the oral prednisone was selected as 30 mg.7 The lesions did not completely resolve until 60 days post-partum. In the pathological examination, histological sections revealed subcorneal vesiculation and pustules containing neutrophil leukocytes. The granular layer of the epidermis was diminished or absent in many areas. In addition, perivascular dermatitis was present in the superficial dermis (Fig. 2).
Discussion Pustular psoriasis of pregnancy (PPP) is a rare variant of generalized pustular psoriasis. This condition was first described by Von Hebra in 1872, and named IH.1 As a specific dermatosis of the pregnancy, it is commonly occurring during the puerperal period. PPP has maternal and fetal morbidity and it is therefore important to recognize and properly treat this condition. There are no guidelines on the standard treatment of IH, but systemic corticosteroids are the first choice of treatment, followed by cyclosporin in resistant cases.8 In some cases, parenteral calcium and vitamin D may be effective. However, the best treatment for IH is termination of pregnancy, followed by treatment with oral retinoids. Bacterial infection may occur after treatment with immunosuppressives, and antibacterial medication may be necessary. The management strategy depends on the risk to the mother and fetus. Impetigo herpetiformis may be a genetic or a familial disease, with IH reported in several families in the published work.9 However, our patient and her relatives had no history of psoriasis.9,10 The diagnosis of IH is based mainly on a clinical assessment and the
© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology
Rare case of dermatosis in pregnancy
presence of sterile pustules and erythematous macular skin lesions, accompanied by fatigue, fever and nausea. In some cases, gestational hypertension, fetal distress because of placental insufficiency, hypoparathyroidism or diabetes may occur.11–13 The genetic transition pattern of IH is commonly unclear. In 1982, Qumeish et al. reported a girl with IH and her brother with IH again.14 In 1989, Tada et al. reported a non-twin sister with IH having common human leukocyte antigen antigens.15 It is important to diagnose skin manifestations in pregnancy because some skin diseases, such as hepatitis B virus infection, measles or chicken pox, can be contagious. It is also important that pregnant patients are made aware that IH is not infectious. In conclusion, pregnant patients with IH must be observed closely due to the increase in the perinatal risk of mortality. The differential diagnosis should include infectious diseases. The treatment and management of IH differ from those of other skin diseases.
Acknowledgments Written consent was obtained from the patient and her husband for publication of this case report.
Disclosure The authors declare that they have no competing interests.
References 1. von Hebra F. Ueber einzelne während Schwangerschaft, des Wochenbettes und bei Uterinalkrankheiten der Frauen zu beobach-tende Hautkrankheiten. Wien Med Wochenschr 1872; 22: 1197–1202.
2. Kaposi M. Impetigo herpetiformis. Arch Dermatol Syphil 1887; 19: 273–296. 3. Chang SE, Kim HH, Choi JH, Sung KJ, Moon KC, Koh JK. Impetigo herpetiformis followed by generalized pustular psoriasis: More evidence of same disease entity. Int J Dermatol 2003; 42: 754–755. 4. Sauer GC, Geha BJ. Impetigo herpetiformis. Arch Dermatol 1961; 83: 173–180. 5. Umezawa Y, Ozawa A, Kawasima T et al. Therapeutic guidelines for the treatment of generalized pustular psoriasis (GPP) based on a proposed classification of disease severity. Arch Dermatol Res 2003; 295 (Suppl 1): S43–S54. 6. Lockwood CJ, Radunovic N, Nastic D, Petkovic S, Aigner S, Berkowitz GS. Corticotropin-releasing hormone and related pituitary-adrenal axis hormones in fetal and maternal blood during the second half of pregnancy. J Perinat Med 1996; 24: 243–251. 7. Ost L, Wettrell G, Björkhem I, Rane A. Prednisolone excretion in human milk. J Pediatr 1985; 106: 1008–1011. 8. Oumeish OY, Parish JL. Impetigo herpetiformis. Clin Dermatol 2006; 24: 101–104. 9. Vicdan K, Gokay Z, Var T, Danisman N, Gok-men O. Twin sisters with impetigo herpetiformis. Eur J Obstet Gynecol Reprod Biol 1995; 63: 195–196. 10. Erbagci Z, Erkilic S. A case of recurrent impetigo herpetiformis with a positive family history. Int J Clin Pract 2000; 54: 619–620. 11. Fouda UM, Foud RM, Ammar HM, Salem M, Darouti MEI. Impetigo herpetiformis during the puerperium triggered by secondary hypo-parathyroidism: A case report. Cases J 2009; 2: 9338. 12. Wolf R, Tartler U, Stege H, Megahed M, Ru-zicka T. Impetigo herpetiformis with hyper-parathyroidism. J Eur Acad Dermatol Venereol 2005; 19: 743–746. 13. Huang YH, Chen YP, Liang CC, Chang YL, Hsieh CC. Impetigo herpetiformis with gestational hypertension: A case report and literature review. Dermatology 2011; 222: 221– 224. 14. Oumeish OY, Farraj SE, Bataineh AS. Some aspects of impetigo herpetiformis. Arch Dermatol 1982; 118: 103–105. 15. Tada J, Fukushiro S, Fujiwara Y, Akagi Y, Kodama H, Nohara N. Two sisters with impetigo herpetiformis. Clin Exp Dermatol 1989; 14: 82–84.
© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology
303
doi:10.1111/jog.12530
J. Obstet. Gynaecol. Res. Vol. 41, No. 2: 301–303, February 2015
Case report of a rare dermatosis in pregnancy: Impetigo herpetiformis Mustafa Ulubay1, Ug˘ur Keskin1, Ulas Fidan1, Ali Fuat Çiçek2, Ercan Çalıs¸kan3, Rıza Efendi Karaca1, Fahri Burçin Fıratlıgil1 and Ali Ergün1 Departments of 1Obstetrics and Gynecology, 2Pathology and 3Dermatology, Gulhane Military Medical Academy, Ankara, Turkey
Abstract Impetigo herpetiformis (IH) is a very rare type of dermatosis seen in pregnancy. According to the published work, IH during pregnancy is associated with the risk of stillbirth, and obstetric management in such cases is very important. Early recognition is important to reduce both maternal and fetal morbidity. We present a case of IH resistant to corticosteroid therapy in a 27-year-old pregnant woman where the pregnancy was terminated by the induction of labor. Key words: dermatosis, impetigo herpetiformis, pregnancy.
Introduction
Case Report
Impetigo herpetiformis (IH) was first described by von Hebra in 1872.1 According to the published work, IH is a variant of generalized pustular psoriasis in pregnancy, characterized by an acute pustular eruption.2,3 It is a rare and potentially life-threatening condition, which affects pregnant women, in particular, in the third trimester.2 The exact mechanism underlying IH is unknown.4 Patients with IH often have systemic symptoms, including malaise, fewer, nausea, vomiting, diarrhea, chills and arthralgia. Laboratory findings are leukocytosis, an elevated erythrocyte sedimentation rate and hypocalcemia. Treatment involves i.v. fluid, electrolyte, calcium and vitamin D supplementation, and systemic and topical corticosteroids.
A 27-year-old 37-week pregnant patient was referred to our clinic with painful, itchy and desquamative macular lesions on her body that were accompanied by fatigue (Fig. 1). The lesions were predominately on her abdomen, groin and back. Some lesions were also present on the patient’s upper and lower extremities. Her medical history revealed that the lesions had commenced on her groin and then spread to the rest of her body at 35 weeks of gestation. Our patient or her relatives had no history of IH or pustular psoriasis. She had been treated for rheumatoid arthritis for 2 years with prednisolone before becoming pregnant. She had experienced an uncomplicated pregnancy before the skin disease started. She had normal vital signs. She was not a smoker. A blood examination revealed
Received: March 14 2014. Accepted: June 30 2014. Reprint request to: Assistant Professor Mustafa Ulubay, Gulhane Military Medical Academy, Obstetrics and Gynecology Department, General Tevfik Saglam Caddesi Etlik, Ankara 06100, Turkey. Email: [email protected] Author contribution: M. U., U. K., U. F. and E. Ç. drafted the manuscript and collected data related to the subject. A. F. Ç. and R. E. K. were involved in drafting the manuscript and in the critical revision of the draft. F. B. F. made a substantial contribution to the conception and design of the study. A. E. participated in the design of the manuscript and the coordination of the study. All authors read and approved the final manuscript.
© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology
301
M. Ulubay et al.
a
a
b
Figure 1 Appearance of impetigo herpetiformis on the patient’s body. The characteristic of lesions are macular desquamative pustular lesions. (a) Anterior, (b) posterior.
normal full blood. Renal, liver function and thyroid function tests were normal, as were her calcium levels. An oral glucose test was normal. She tested negative for Herpes simplex virus type 1 and type 2 and hepatitis B and C. A bacteriological examination of the pus was negative. She took oral iron and multivitamin supplements during her pregnancy. When she was admitted to the hospital, she was given 30 mg of oral prednisone, antihistamines and a topical steroid. Data guidelines are lacking on the treatment of IH in pregnant women. Topical steroids are usually the first choice if symptoms are light or mild. In severe cases, oral prednisone is prescribed at 30 mg/day up to 60 or 80 mg/day.5 Highdose prednisone can have adverse effects on the fetus, such as reactivity failure in fetal monitoring.6 Therefore, it was decided to treat the patient with prednisone 30 mg/day. An obstetric examination revealed a normal level of amniotic fluid, and biometric and Doppler measurements were within normal ranges. The patient was followed up for 5 days with non-stress tests and fetal movement counts. Despite the oral prednisolone therapy, the lesions spread. Due to the risk of stillbirth, labor was induced by cervical ripening and stripping and oxytocin infusion. The patient had a healthy 2620-g girl following a normal vaginal birth. The 1- and 5-min Apgar scores were 8 and 10, respectively. There was no dermatosis on the newborn. The patient received systemic corticosteroid treatment (30 mg prednisone) and topical corticosteroid (clobetasol propionate emollient 0.05%) during the post-partum period. Because prednisone can be
302
b
c
Figure 2 (a) Subcorneal pustule formation and vesiculation (hematoxylin–eosin [HE], original magnification ×100). (b) Granular layer is not seen in this section. There is superficial perivascular lymphocytic inflammatory infiltration in the papillary dermis. Inflammation consists of lymphocytes (HE, ×100). (c) There is no keratinocytes within the epidermis showing nuclear or cytoplasmic immunoreactivity for Herpes simplex virus (HSV)-2 rabbit polyclonal antibody by immunohistochemistry (×100).
excreted into breast milk and may have adverse effects on the newborn, the dose of the oral prednisone was selected as 30 mg.7 The lesions did not completely resolve until 60 days post-partum. In the pathological examination, histological sections revealed subcorneal vesiculation and pustules containing neutrophil leukocytes. The granular layer of the epidermis was diminished or absent in many areas. In addition, perivascular dermatitis was present in the superficial dermis (Fig. 2).
Discussion Pustular psoriasis of pregnancy (PPP) is a rare variant of generalized pustular psoriasis. This condition was first described by Von Hebra in 1872, and named IH.1 As a specific dermatosis of the pregnancy, it is commonly occurring during the puerperal period. PPP has maternal and fetal morbidity and it is therefore important to recognize and properly treat this condition. There are no guidelines on the standard treatment of IH, but systemic corticosteroids are the first choice of treatment, followed by cyclosporin in resistant cases.8 In some cases, parenteral calcium and vitamin D may be effective. However, the best treatment for IH is termination of pregnancy, followed by treatment with oral retinoids. Bacterial infection may occur after treatment with immunosuppressives, and antibacterial medication may be necessary. The management strategy depends on the risk to the mother and fetus. Impetigo herpetiformis may be a genetic or a familial disease, with IH reported in several families in the published work.9 However, our patient and her relatives had no history of psoriasis.9,10 The diagnosis of IH is based mainly on a clinical assessment and the
© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology
Rare case of dermatosis in pregnancy
presence of sterile pustules and erythematous macular skin lesions, accompanied by fatigue, fever and nausea. In some cases, gestational hypertension, fetal distress because of placental insufficiency, hypoparathyroidism or diabetes may occur.11–13 The genetic transition pattern of IH is commonly unclear. In 1982, Qumeish et al. reported a girl with IH and her brother with IH again.14 In 1989, Tada et al. reported a non-twin sister with IH having common human leukocyte antigen antigens.15 It is important to diagnose skin manifestations in pregnancy because some skin diseases, such as hepatitis B virus infection, measles or chicken pox, can be contagious. It is also important that pregnant patients are made aware that IH is not infectious. In conclusion, pregnant patients with IH must be observed closely due to the increase in the perinatal risk of mortality. The differential diagnosis should include infectious diseases. The treatment and management of IH differ from those of other skin diseases.
Acknowledgments Written consent was obtained from the patient and her husband for publication of this case report.
Disclosure The authors declare that they have no competing interests.
References 1. von Hebra F. Ueber einzelne während Schwangerschaft, des Wochenbettes und bei Uterinalkrankheiten der Frauen zu beobach-tende Hautkrankheiten. Wien Med Wochenschr 1872; 22: 1197–1202.
2. Kaposi M. Impetigo herpetiformis. Arch Dermatol Syphil 1887; 19: 273–296. 3. Chang SE, Kim HH, Choi JH, Sung KJ, Moon KC, Koh JK. Impetigo herpetiformis followed by generalized pustular psoriasis: More evidence of same disease entity. Int J Dermatol 2003; 42: 754–755. 4. Sauer GC, Geha BJ. Impetigo herpetiformis. Arch Dermatol 1961; 83: 173–180. 5. Umezawa Y, Ozawa A, Kawasima T et al. Therapeutic guidelines for the treatment of generalized pustular psoriasis (GPP) based on a proposed classification of disease severity. Arch Dermatol Res 2003; 295 (Suppl 1): S43–S54. 6. Lockwood CJ, Radunovic N, Nastic D, Petkovic S, Aigner S, Berkowitz GS. Corticotropin-releasing hormone and related pituitary-adrenal axis hormones in fetal and maternal blood during the second half of pregnancy. J Perinat Med 1996; 24: 243–251. 7. Ost L, Wettrell G, Björkhem I, Rane A. Prednisolone excretion in human milk. J Pediatr 1985; 106: 1008–1011. 8. Oumeish OY, Parish JL. Impetigo herpetiformis. Clin Dermatol 2006; 24: 101–104. 9. Vicdan K, Gokay Z, Var T, Danisman N, Gok-men O. Twin sisters with impetigo herpetiformis. Eur J Obstet Gynecol Reprod Biol 1995; 63: 195–196. 10. Erbagci Z, Erkilic S. A case of recurrent impetigo herpetiformis with a positive family history. Int J Clin Pract 2000; 54: 619–620. 11. Fouda UM, Foud RM, Ammar HM, Salem M, Darouti MEI. Impetigo herpetiformis during the puerperium triggered by secondary hypo-parathyroidism: A case report. Cases J 2009; 2: 9338. 12. Wolf R, Tartler U, Stege H, Megahed M, Ru-zicka T. Impetigo herpetiformis with hyper-parathyroidism. J Eur Acad Dermatol Venereol 2005; 19: 743–746. 13. Huang YH, Chen YP, Liang CC, Chang YL, Hsieh CC. Impetigo herpetiformis with gestational hypertension: A case report and literature review. Dermatology 2011; 222: 221– 224. 14. Oumeish OY, Farraj SE, Bataineh AS. Some aspects of impetigo herpetiformis. Arch Dermatol 1982; 118: 103–105. 15. Tada J, Fukushiro S, Fujiwara Y, Akagi Y, Kodama H, Nohara N. Two sisters with impetigo herpetiformis. Clin Exp Dermatol 1989; 14: 82–84.
© 2014 The Authors Journal of Obstetrics and Gynaecology Research © 2014 Japan Society of Obstetrics and Gynecology
303
