Home Persistent hypoglycemia in congenital syphilis: hyperinsulinemic hypoglycemia with a focal pancreatic lesion
Article
Licensed
Unlicensed Requires Authentication

Persistent hypoglycemia in congenital syphilis: hyperinsulinemic hypoglycemia with a focal pancreatic lesion

  • Mehmet Akyar ORCID logo , İpek Guney Varal ORCID logo , Gaffari Tunç ORCID logo EMAIL logo , Ayşe Ören ORCID logo , Yasemin Denkboy Öngen ORCID logo and Eren Çağan ORCID logo
Published/Copyright: November 28, 2024

Abstract

Objectives

Congenital syphilis can cause severe morbidities such as hydrops fetalis, preterm birth, low birth weight, hepatosplenomegaly, pneumonia, hypoglycemia, etc., and mortality. Despite hypoglycemia being reported in congenital syphilis and hyperinsulinism, only one case was described. In this article, we aimed to present a newborn with congenital syphilis born to a syphilitic mother who was diagnosed with persistent hyperinsulinemic hypoglycemia with a focal lesion during follow-up.

Case presentation

A female patient was born with a weight of 2,450 g in the 32+3rd week of pregnancy from a 30-year-old syphilitic mother who had pleural effusion and ascites, cholestasis, thrombocytopenia, and anemia at birth. Hypoglycemia was detected on the 10th day of the patient’s follow-up (47 mg/dL). The patient was unresponsive to diazoxide, and octreotide, nifedipine, and glucagon treatments were started gradually. No variants were detected in the HH gene panel, but a focal pancreatic head lesion was detected in the pancreatic head in fluorine-18 L-3,4 dihydroxyphenylalanine positron emission tomography-computed tomography. Focal lesionectomy was recommended for the patient.

Conclusions

In this article, we present a neonate with severe early congenital syphilis and focal pancreatic lesions with persistent hyperinsulinemic hypoglycemia unresponsive to diazoxide. Although hypoglycemia can be seen in congenital syphilis and has been reported before, hypoglycemia associated with hyperinsulinism is very rare; only a few patients have been presented, and a focal pancreatic lesion has not been previously reported. The pathology is unknown, but pancreatitis may cause this entity.


Corresponding author: Gaffari Tunç, Associate Professor, Department of Neonatology, University of Health Sciences, Bursa Yuksek İhtisas Training and Research Hospital, Bursa, Türkiye, E-mail:

Acknowledgments

We are grateful to the patient and her family, as reported in this article, for their genuine support.

  1. Research ethics: Not applicable.

  2. Informed consent: The consent form was filled out by the parent.

  3. Author contributions: All the authors have accepted responsibility for the entire content of this submitted manuscript and approved submission.

  4. Use of Large Language Models, AI and Machine Learning Tools: Not applicable.

  5. Conflict of interest: The authors state no conflict of interest.

  6. Research funding: None declared.

  7. Data availability: Not applicable.

References

1. Kingston, M, French, P, Higgins, S, McQuillan, O, Sukthankar, A, Stott, C, et al.. Members of the Syphilis guidelines revision group; UK national guidelines on the management of syphilis 2015. Int J STD AIDS 2016;27:421–46. https://doi.org/10.1177/0956462415624059.Search in Google Scholar PubMed

2. Koliou, M, Chatzicharalampous, E, Charalambous, M, Aristeidou, K. Congenital syphilis as the cause of multiple bone fractures in a young infant case report. BMC Pediatr 2022;22:728. https://doi.org/10.1186/s12887-022-03789-y.Search in Google Scholar PubMed PubMed Central

3. Gϋemes, M, Rahman, SA, Kapoor, RR, Flanagan, S, Houghton, JAL, Misra, S, et al.. Hyperinsulinemic hypoglycemia in children and adolescents: recent advances in understanding of pathophysiology and management. Rev Endocr Metab Disord 2020;21:577–97. https://doi.org/10.1007/s11154-020-09548-7.Search in Google Scholar PubMed PubMed Central

4. Stanley, CA, De Leon, DD. Etiology of the neonatal hypoglycemias. Adv Pediatr 2024;71:119–34. https://doi.org/10.1016/j.yapd.2024.03.001.Search in Google Scholar PubMed

5. Daaboul, JJ, Kartchner, W, Jones, KL. Neonatal hypoglycemia caused by hypopituitarism in infants with congenital syphilis. J Pediatr 1993;123:983–5. https://doi.org/10.1016/s0022-3476(05)80398-1.Search in Google Scholar PubMed

6. Lee, HS, Lee, JI, Jeon, J. Congenital syphilis unusually presenting with prematurity-related severe neonatal morbidities including meconium obstruction: a case report and review of the literature. Medicine (Baltimore) 2020;99:e22321. https://doi.org/10.1097/md.0000000000022321.Search in Google Scholar

7. Newman-Lindsay, S, Lakshminrusimha, S, Sankaran, D. Diazoxide for neonatal hyperinsulinemic hypoglycemia and pulmonary hypertension. Children 2022;10:5. https://doi.org/10.3390/children10010005.Search in Google Scholar PubMed PubMed Central

8. Fiser, RHJ, Yamauchi, T. Hypoglycemia associated with congenital syphilis. J Ark Med Soc 1977;73:494–5.Search in Google Scholar

9. Simmank, KC, Pettifor, JM. Unusual presentation of congenital syphilis. Ann Trop Paediatr 2000;20:105–7. https://doi.org/10.1080/02724936.2000.11748117.Search in Google Scholar PubMed

10. Ismail, D, Smith, VV, de Lonlay, P, Ribeiro, MJ, Rahier, J, Blankenstein, O, et al.. Familial focal congenital hyperinsulinism. J Clin Endocrinol Metab 2011;96:24–8. https://doi.org/10.1210/jc.2010-1524.Search in Google Scholar PubMed PubMed Central

11. Dastamani, A, Yau, D, Gilbert, C, Morgan, K, De Coppi, P, Craigie, RJ, et al.. Variation in glycemic outcomes in focal forms of congenital hyperinsulinism-the UK perspective. J Endocr Soc 2022;6:bvac033. https://doi.org/10.1210/jendso/bvac033.Search in Google Scholar PubMed PubMed Central

12. Shet, TM, Kandalkar, BM, Vora, IM. Neonatal hepatitis--an autopsy study of 14 cases. Indian J Pathol Microbiol 1998;41:77–84.Search in Google Scholar

13. Svane, S. A peculiar case of pancreatic cirrhosis. A result of latent congenital syphilis? Acta Chir Scand 1972;138:95–101.Search in Google Scholar

14. Oppenheimer, EH, Hardy, JB. Congenital syphilis in the newborn infant: clinical and pathological observations in recent cases. Johns Hopkins Med J 1971;129:63–82.Search in Google Scholar

15. Raeburn, C. Gumma of the pancreas in a premature infant. J Pathol Bacteriol 1951;63:158–9. https://doi.org/10.1002/path.1700630117.Search in Google Scholar PubMed

Received: 2024-08-02
Accepted: 2024-11-11
Published Online: 2024-11-28
Published in Print: 2025-01-29

© 2024 Walter de Gruyter GmbH, Berlin/Boston

Articles in the same Issue

  1. Frontmatter
  2. Review
  3. Osteogenesis imperfecta: shifting paradigms in pathophysiology and care in children
  4. Opinion Paper
  5. CRH receptor antagonist crinecerfont – a promising new treatment option for patients with congenital adrenal hyperplasia due to 21-hydroxylase deficiency
  6. Original Articles
  7. Age and sex mark clinical differences in the presentation of pediatric type 1 diabetes mellitus
  8. Geographic information system mapping and predictors of glycemic control in children and youth with type 1 diabetes: a study from Western India
  9. Body composition assessment measured via bioelectrical impedance analysis in euthyroid children with newly diagnosed Hashimoto’s thyroiditis
  10. Outcomes of newborns screened for congenital hypothyroidism in Turkey – a single center experience
  11. High yield of congenital hypothyroidism among infants attending Children Hospital, Nairobi, Kenya. Facility based study in the absence of newborn screening
  12. Immune checkpoint inhibitors and endocrinopathies in pediatric brain tumor patients
  13. Assessment of quality of life in families affected by maple syrup urine disease: a cross sectional study
  14. Case Reports
  15. Reninoma: an unusual cause of growth failure
  16. Persistent hypoglycemia in congenital syphilis: hyperinsulinemic hypoglycemia with a focal pancreatic lesion
  17. Diagnostic challenges in pediatric Cushing’s disease associated with chronic renal failure: a report of three patients
  18. A novel de novo missense OTC mutation in an Iranian girl: a case report
Downloaded on 2.11.2025 from https://www.degruyterbrill.com/document/doi/10.1515/jpem-2024-0365/html
Scroll to top button