What is the clinical evidence from this study?

Study design: Case Report. Population: Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) with coronary-pulmonary artery fistula (n=1). Intervention: Medical management (levothyroxine and atorvastatin). Primary outcome: Clinical and laboratory response.

What does this research mean for the field?

Medical management with levothyroxine and atorvastatin resolves facial edema and normalizes thyroid and lipid profiles in adult patients with ALCAPA and concurrent coronary-pulmonary artery fistula who decline surgical intervention. Novelty: ClaimNovelty.INCREMENTAL. Consensus alignment: ConsensusAlignment.NEUTRAL.

August 19, 2026Frontiers in Cardiovascular MedicineOpen Access

Anomalous origin of the left coronary artery from the pulmonary artery with coronary artery fistula in an adult presenting with cardiomegaly, abnormal electrocardiogram, myocardial perfusion defect and pulmonary hypertension: a case report

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Key result

Medical therapy with levothyroxine and atorvastatin resolves symptoms in an ALCAPA patient declining surgery.

Why the study?

Only 10%-15% of untreated patients with ALCAPA develop collateral circulation enabling survival into adulthood with nonspecific or no symptoms, posing diagnostic challenges particularly when co-occurring with other congenital defects.

Design

Case report

Authors

LLLian LuoCardiovascular Institute Hospital XWXuefeng Wang KHKerui HuangAffiliated Hospital of Southwest Medical University

Discussion

Member takes

Implication

May prompt ALCAPA consideration in select adult cases; leaves open optimal management for asymptomatic survivors.

Study Design

Type

Case Report (n=1)

Multicenter

Structured PICO

Population

A 55-year-old female presenting with facial edema and severe hypothyroidism who was incidentally diagnosed with ALCAPA and a coronary-pulmonary artery fistula and followed for 5 months.

Intervention

Medical management with thyroid hormone replacement therapy (levothyroxine titrated to 62.5 µg once daily) and lipid-lowering treatment (atorvastatin 20 mg nightly) after the patient declined surgical intervention.

Outcome

Resolution of facial edema, normalization of thyroid function and lipid profile, and clinical stability over follow-up.

This case underscores the diagnostic challenges of adult ALCAPA coexisting with a coronary-pulmonary artery fistula and emphasizes the need for multimodal imaging in patients with unexplained cardiomegaly and atypical presentations.

Limitations

Single case analysis of ALCAPA with CPAF limits generalizability.
Short follow-up duration of three months is insufficient to definitively ascertain whether cardiac remodeling was solely attributable to ALCAPA and CAF.
Absence of stress perfusion imaging and cardiac magnetic resonance imaging precludes the assessment of myocardial activity.
The patient remains at risk for sudden cardiac death and heart failure after declining surgical intervention, leaving the optimal follow-up strategy indeterminate.
Single case analysis of ALCAPA with CPAF
Follow-up duration is relatively brief and insufficient to definitively ascertain whether cardiac enlargement and pulmonary hypertension were solely attributable to ALCAPA and CAF
Absence of stress perfusion imaging and cardiac magnetic resonance imaging precludes the assessment of myocardial activity
Patient remains at risk for sudden cardiac death and heart failure, yet she declined surgical intervention, making the optimal follow-up strategy indeterminate

Cite This Study

Luo et al. (2026) conducted a case report in Anomalous origin of the left coronary artery from the pulmonary artery (ALCAPA) with coronary-pulmonary artery fistula (n=1). Medical management (levothyroxine and atorvastatin) was evaluated on Clinical and laboratory response. Medical management with levothyroxine and atorvastatin resolved facial edema and normalized thyroid and lipid profiles in a 55-year-old woman with ALCAPA and a coronary-pulmonary artery fistula who declined surgery.

synapsesocial.com/papers/6aa9314904014c9901263d19 https://doi.org/10.3389/fcvm.2026.1844272

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Anomalous origin of the left coronary artery from the pulmonary artery with coronary artery fistula in an adult presenting with cardiomegaly, abnormal electrocardiogram, myocardial perfusion defect and pulmonary hypertension: a case report

Discussion

Member takes

Implication

Study Design

Structured PICO

Limitations

Cite This Study

Anomalous origin of the left coronary artery from the pulmonary artery with coronary artery fistula in an adult presenting with cardiomegaly, abnormal electrocardiogram, myocardial perfusion defect and pulmonary hypertension: a case report

Discussion

Member takes

Implication

Study Design

Structured PICO

Limitations

Cite This Study

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