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Advances in dermatology and venereology Acta Dermato-Venereologica
Impetigo Herpetiformis Complicated with Intrauterine Growth Restriction Treated Successfully with Granulocyte and Monocyte Apheresis
Natsuko SAITO-SASAKI 1, 2, Kunio IZU 2, Yu SAWADA 1, Ryosuke HINO 1, Ryoji NAKANO 3, Shohei SHIMAJIRI 3, Izumi NISHIMURA 4, Hiromasa NAKAHARA 4, Kazumitsu SUGIURA 5 and Motonobu NAKAMURA 1
1
Department of Dermatology, University of Occupational and Environmental Health, 1-1 Iseigaoka, Yahatanishi-ku, Kitakyushu 807-8555, Divisions of 2 Dermatology, 3 Clinical Pathology and 4 Obstetrics and Gynecology, Japan Community Health Care Organization, Kyushu Hospital, Kyushu, and 5 Department of Dermatology, Nagoya University Graduate School of Medicine, Nagoya, Japan. E-mail: natsuko-saito @ med. uoeh-u. ac. jp Accepted Aug 29, 2016; Epub ahead of print Aug 30, 2016
Impetigo herpetiformis( IH) is a rare type of generalized pustular psoriasis( GPP) occurring in pregnancy. Although patients with IH sometimes experience intrauterine growth restriction( IUGR), possibly due to lower oxygen and nutrition intake from the inflamed placenta( 1), the mechanism underlying the pathogenesis of IHassociated IUGR is unknown. There have been many reports on treatment for IH-associated lower birth weight( 2, 3); however, the number of case reports is insufficient to reach a consensus on IH treatment, especially when focusing on birth weight. We report here a case of IH with placental inflammation treated successfully with granulocyte and monocyte apheresis( GCAP), leading to an improvement in birth weight. We suggest that placental inflammation in IH may lead to restricted intrauterine growth, which is abrogated by GCAP.
CASE REPORT
A 30-year-old woman reported circinate scaly plaques with pustules on the periphery of her trunk and her extremities at 10 weeks into her fourth pregnancy( Fig. 1A). She had experienced similar intractable skin eruptions and lower birth weight during her second and third pregnancies. Laboratory examination revealed a normal leukocyte count of 7,900 / μl and a C-reactive protein level of
0.12 mg / dl( normal < 0.14 mg / dl). Skin biopsy revealed a subcorneal neutrophil-dominant infiltration, psoriasiform epidermis and perivascular infiltration of lymphocytes and a few neutrophils in the dermis( Fig. 1B). Based on the clinical course and histological examination, we diagnosed her skin eruption as an IH. She had no family history of IH or GPP. Genetic examination did not reveal any mutation in IL36RN encoding interleukin-36 receptor antagonist. She was initially treated with oral methylprednisolone 10 mg / day, and cyclosporine, 100 mg / day, between 10 and 16 weeks of gestation; however, her skin eruption aggravated gradually. At that time, IUGR was also observed by ultrasonographic evaluation.
To relieve the intractable systemic inflammation, weekly GCAP treatment( an extracorporeal circulation therapy that removes activated granulocytes and monocytes( 4)) was administered from 16 weeks gestation. The patient’ s skin eruption improved after 5 GCAP treatments, and GCAP was discontinued. Two weeks after discontinuation of GCAP, however, the skin eruption gradually worsened, and then GCAP treatment was resumed. Her skin eruption improved dramatically after 5 GCAP treatments. However, 3 weeks after another cessation of GCAP treatment, her skin eruption again exacerbated. To improve her peri operative condition, we decided to perform GCAP for her IH until 34 weeks gestation.
Fig. 1.( A) Clinical manifestation of impetigo herpetiformis showing annular hyperkeratotic plaques with pustules on the patient’ s trunk.( B) Histopathology of the skin. Haematoxylin and eosin staining of the skin showed a subcorneal neutrophil infiltration, a psoriasiform epidermis and perivascular infiltration of lymphocytes.( C) Immunostaining for tumour necrosis factor alpha( TNF-α) in placenta( arrowheads), during 3 rd( left) and 4 th( right) pregnancies. Scale bar: 100 μm.( D) Number of TNF-α producing cells in placenta for 3 rd and 4 th pregnancies. The mena(+ SEM) number of TNF-α producing cells for 5 different areas was determined( original magnification × 400). * p < 0.05 doi: 10.2340 / 00015555-2527 Acta Derm Venereol 2017; 97: 410 – 411
This is an open access article under the CC BY-NC license. www. medicaljournals. se / acta Journal Compilation © 2017 Acta Dermato-Venereologica.