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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 .