Buccal carcinoma associated with adult congenital pulmonary hypoplasia: A case report and literature review

Medicine (Baltimore). 2022 Dec 2;101(48):e32142. doi: 10.1097/MD.0000000000032142.

Abstract

Rationale: Congenital pulmonary hypoplasia is a relatively rare lung malformation. The disease is usually identified in the neonatal period or early childhood. Most affected patients die of respiratory insufficiency after birth. Mild congenital dysplasia is asymptomatic, while moderate congenital dysplasia manifests as respiratory distress or does not become evident until adulthood. Cases of congenital pulmonary hypoplasia in adults are rare, especially in combination with malignant tumors.

Patient concerns: We report the case of a 64-year-old man with buccal carcinoma who was accidentally found to have hypoplasia of the left lung during treatment.

Diagnoses: After chest computed tomography and chest radiograph, the diagnosis of adult congenital pulmonary hypoplasia was confirmed.

Interventions: Since the patient had a history of bronchiectasis for more than 30 years and only had 1 healthy lung, the cardiopulmonary compensatory function was poor and the patient could not tolerate surgery, he was given radiotherapy and chemotherapy.

Outcomes: Radiotherapy and chemotherapy were successfully completed, and within the patient's tolerance, there was no serious adverse reaction of respiratory system.

Conclusion: Congenital pulmonary hypoplasia's diagnosis is challenging in adults because the condition can easily be mistaken for a more common disease. However, early diagnosis is very important to enable prompt therapy and ensure proper follow-up to detect and treat complications such as pulmonary infection and pulmonary hypertension in a timely manner.

Publication types

  • Review
  • Case Reports

MeSH terms

  • Adult
  • Carcinoma*
  • Child, Preschool
  • Humans
  • Infant, Newborn
  • Middle Aged