NDLI: Poland syndrome accompanied by internal iliac artery supply disruption sequence: a case report

Content Provider	Springer Nature : BioMed Central
Author	Gonda, Kenji Tachiya, Yosuke Hatakeyama, Yuichi Momma, Tomoyuki Tamaoki, Tomoko Maejima, Yuko Rokkaku, Yuichi Saji, Shigehira Shimomura, Kenju Kono, Koji
Abstract	Background Poland syndrome is a congenital malformation characterized by ipsilateral hand and chest wall depression, including an absence or hypoplasia of the breast and pectoral muscles. These hypoplastic defects are reportedly caused by a subclavian artery supply disruption sequence. Case presentation A 45-year-old Japanese woman, an out-patient, underwent an emergency examination for intense left lower abdominal pain. Computed tomography images revealed a hydronephrotic left kidney and dilatation of the left ureter. No ureteral calculus or neoplasm was found. In addition, no abnormalities connected to her left abdominal pain were found. Nephritis was diagnosed based on the results of urine analysis, and a course of antibiotics was administered. Computed tomography images also revealed a history of breast reconstruction with a custom-made silicone implant in her right breast. The present case showed symptoms of Poland syndrome, which were absence of the sternal head of the right pectoralis major and asymmetrical malformation of the chest wall due to hypoplasia of the right rib cage. In addition to typical Poland syndrome symptoms, she had hypoplasia of her right kidney, hypoplasia of the right gluteus minimus muscle, right-sided pelvic hypoplasia, spinal curvature to the right, and a cystic mass in her right ovary. Conclusions In the present case of Poland syndrome, computed tomography images revealed malformation of the chest wall, absence of the pectoral muscle, and hypoplasia of a left kidney. Unilateral visceral hypoplasia is reported to be caused by a subclavian artery supply disruption sequence that occurs around 7 to 8 weeks of gestation. The present case can be considered a rare atypical phenotype of Poland syndrome with possible subclavian artery supply disruption sequence with internal iliac artery supply disruption.
Related Links	https://jmedicalcasereports.biomedcentral.com/counter/pdf/10.1186/s13256-018-1823-8.pdf
Ending Page	5
Page Count	5
Starting Page	1
File Format	HTM / HTML
ISSN	17521947
DOI	10.1186/s13256-018-1823-8
Journal	Journal of Medical Case Reports
Issue Number	1
Volume Number	12
Language	English
Publisher	BioMed Central
Publisher Date	2018-10-26
Access Restriction	Open
Subject Keyword	Medicine Public Health General Practice Family Medicine Primary Care Medicine Surgical Oncology Internal Medicine Poland syndrome Internal iliac artery supply disruption Pectoral muscle deficit Renal hypoplasia Medicine/Public Health
Content Type	Text
Resource Type	Case study
Subject	Medicine
Journal Impact Factor	0.9/2023

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