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Polythelia and associated hydronephrosis: a case report in neonatal age

  • Zora Zakanj EMAIL logo und Lena Kotrulja
Veröffentlicht/Copyright: 31. März 2015

Abstract

Polythelia, the presence of supernumerary nipples, is a common anomaly usually limited to cosmetic concerns. However, the existence of accessory mammary tissue may indicate associated anomalies of the kidneys and the urinary tract or other systemic diseases. The association of these anomalies and systemic disease is often controversial, although some authors report their significance in neonates and infants. We report the association of accessory nipple and right-sided hydronephrosis in a female term newborn. Prenatal ultrasound monitoring did not indicate hydronephrosis and it was diagnosed after birth with an ultrasound examination of the newborn, which was due to a fever and suspected urinary infection. We consider that in symptomatic infants with accessory breast tissue, a thorough physical examination, urine analysis, and renal ultrasound should be done in order to exclude associated urinary tract abnormalities.

Introduction

Accessory mammary tissue (AMT) occurs at a frequency of 0.4%–6%. Polythelia is twice more common than polimasty. These tissues develop along the embryonic milk line which extends on both sides of the chest from armpit to the groin, but the changes are usually located between or below normal localized breast and nipples, but sometimes could be localized outside the mammary line, on the back and gluteal region. These changes belong to the group of minor malformations, and are usually only of cosmetic significance. However, AMT are reported along with other abnormalities or syndromes [1].

Case report

A 29-year-old woman gave birth at term to a female infant from her first regular and controlled pregnancy, with a birth weight of 3480 g and length 50 cm. On the first clinical examination, about 3 cm below the left nipple, a round and slightly darker area of skin with a discrete elevation in the center was observed (Figure 1). On palpation, sparse glandular tissue was perceived, giving a clue to an accessory nipple. In parents and other family members, no skin lesions, AMT, or other congenital anomalies were observed. On the third day of life, the baby became febrile (rectal temperature 38.9°C) with tachycardia (to 168 beats/min), refused meal intake and occasionally vomited. Basic laboratory examination revealed leukocytosis with a share of non-segmented granulocytes of 16%, increased value of C-reactive protein (41.2 mg/L) and procalcitonin (9.2 mg/L). Urinalysis suggested urinary tract infection. The baby underwent ultrasound examination of the kidneys and urinary tract, on which cystic formation in the lower pole of the right kidney, measuring 18×17.7 mm, was detected (Figure 2A), as well as dilatation of pyelon and ureter (Figure 2B). No other associated organ anomalies were detected.

Figure 1: 
					Accessory nipple in the neonate.
Figure 1:

Accessory nipple in the neonate.

Figure 2: 
					(A) Ultrasonography of kidney with hydronephrosis of right kidney. (B) Ultrasonography with hydronephrosis of right kidney.
Figure 2:

(A) Ultrasonography of kidney with hydronephrosis of right kidney. (B) Ultrasonography with hydronephrosis of right kidney.

Discussion

AMT in neonates is often an isolated condition without associated diseases or other organ involvement. On the contrary, a broad spectrum of kidney-ureteral malformations were discovered in association with AMT, such as vesicoureteral reflux, a dual channel system, adult dominant polycystic kidney disease, unilateral renal agenesis, cystic renal dysplasia, and congenital stenosis of the pyeloureteral joint [2]. The results of a few surveys support the assumption that there is no association of AMT and urinary tract malformations, and that no further investigation is required in children. Diagnostic testing performed in our patient was due to urinary tract infection symptoms and was not related to AMT. Ultrasound examination of the kidney in our case proved to be justified. A case-controlled clinical study, published by Ferrara et al., showed a higher incidence of renal anomalies in children with AMT, suggesting the need for additional diagnostic procedure [3]. We believe that the additional diagnostic procedures in terms of seeking associated malformations are necessary in the case of a positive kidney disease family history [4], atypical localization, and presence of clinical signs suspected of associated disease. In view of the potentially malignant transformation of AMT, most authors recommend surgical follow-up and treatment [5]. Further research is needed to establish the clinical significance of AMT in light of its reported associated conditions.


Corresponding author: Zora Zakanj, MD, PhD, Department of Gynaecology and Obstetrics, University Hospital Centre “Sestre milosrdnice”, Vinogradska 29, 10000 Zagreb, Croatia, Tel.: +385 1 378 72 95, E-mail:

References

[1] Köse R, Ozgoönül A, Bingöl I. Intraareolar polythelia: a rare anomaly. J Pak Med Assoc. 2012;62:499–500.Suche in Google Scholar

[2] Grotto I, Browner-Elhanan K, Mimouni D, Varsano I, Cohen HA, Minoumi M. Occurence of supernumerary nipples in children with kidney and urinary tract malformations. Pediatr Dermatol. 2001;18:291–4.10.1046/j.1525-1470.2001.01930.xSuche in Google Scholar PubMed

[3] Ferrara P, Giorgio V, Vitelli O, Gatto A, Romano V, Bufalo FD, et al. Polythelia: still a marker of urinary tract anomalies in children? Scand J Urol Nephrol. 2009;43:47–50.10.1080/00365590802442086Suche in Google Scholar PubMed

[4] Grossl NA. Supernumerary breast tissue: historical perspectives and clinical features. South Med J. 2000;93:29–32.10.1097/00007611-200093010-00005Suche in Google Scholar

[5] Kokavec R, Macúch J, Fedeles J, Ondriás F. Polythelia is not a mere aesthetic issue. Acta Chir Plast. 2002;44:3–6.Suche in Google Scholar

  1. The authors stated that there are no conflicts of interest regarding the publication of this article.

Received: 2014-05-28
Accepted: 2015-02-06
Published Online: 2015-03-31
Published in Print: 2015-09-01

©2015 by De Gruyter

Artikel in diesem Heft

  1. Frontmatter
  2. Case reports – Obstetrics
  3. Minimally invasive procedure for type II canal defect caesarean scar pregnancy with cardiac activity and high hCG titres at 8+2 weeks of gestation
  4. Rare causes of acute abdomen in pregnancy: “ultrasound to the rescue”. A review of two cases
  5. Enlargement of hepatic hemangioma in successive pregnancies
  6. Misdiagnosis of macroamylasemia in pregnancy as pancreatitis
  7. An advanced cervical ectopic pregnancy
  8. Multidisciplinary management of giant genital tract venous malformations during pregnancy: case report and review of the literature
  9. Acute uterine rupture in spontaneous term labour in a healthy primigravida: case report and review of the literature
  10. Massive ascites in a patient with preeclampsia
  11. Loeys-Dietz syndrome in pregnancy
  12. Prenatal diagnosis of periventricular venous infarction in utero: a case with hereditary protein C deficiency
  13. Case reports – Fetus
  14. Placental chorioangioma presenting prenatal hemolytic anemia and consumption coagulopathy: a case report
  15. Management of fetal ovarian cyst using in utero aspiration
  16. A case of fetal cardiac rupture diagnosed by postmortem magnetic resonance image
  17. Unusual presentation of fetus in fetu in triplet pregnancy mimicking abdominal wall defect
  18. Acral necrosis and upper brachial plexus palsy after prenatal fetal thrombosis
  19. Prenatal diagnosis of a giant fetal hepatic hemangioma: a case report
  20. Prenatal diagnosis and outcomes of fetal cardiac rhabdomyomas: evaluation of seven cases
  21. Case reports – Newborn
  22. Polythelia and associated hydronephrosis: a case report in neonatal age
  23. Necrotizing enterocolitis following intensive phototherapy in full-term newborns – is there a possible association?
  24. A case of neonatal toxic shock syndrome-like exanthematous disease concurrent with maternal toxic shock syndrome
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