Evidence map›Paper›PMID 42703512›Full record

ArticleCureus2026

Early Genotype-Driven Diagnosis of Hermansky-Pudlak Syndrome Type 4 in a Child With Oculocutaneous Albinism: An Ophthalmic Case Report.

Dorottya Szabo, Julie Waldispühl-Geigl, Sandra Kamper, Heidemarie Pilch, Barbara Plecko, Kathrin Vollnhofer, Laura Posch-Pertl

Abstract readCase Reports
In one paragraph

Article in Cureus, 2026. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

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0cells of the map it votes in
0citing papers in PubMed
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1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

0 citing papers in PubMed.

No citing paper in PubMed yet.

4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

7 authors.

Dorottya SzaboDepartment of Ophthalmology, Medical University of Graz, Graz, AUT.
Julie Waldispühl-GeiglDiagnostic and Research Institute of Human Genetics, Medical University of Graz, Graz, AUT.
Sandra KamperDiagnostic and Research Institute of Human Genetics, Medical University of Graz, Graz, AUT.
Heidemarie PilchDepartment of Pediatrics and Adolescent Medicine, Medical University of Graz, Graz, AUT.
Barbara PleckoDepartment of Pediatrics and Adolescent Medicine, Medical University of Graz, Graz, AUT.
Kathrin VollnhoferDepartment of Ophthalmology, Medical University of Graz, Graz, AUT.
Laura Posch-PertlDepartment of Ophthalmology, Medical University of Graz, Graz, AUT.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Hermansky-Pudlak syndrome (HPS) is a rare inherited multisystem condition characterized by oculocutaneous albinism, bleeding diathesis, and subtype-specific systemic complications, including pulmonary fibrosis and granulomatous colitis. Early diagnosis is essential because pulmonary involvement, particularly in HPS type 4, may be life-threatening. We report a boy who was first referred for ophthalmologic evaluation at seven weeks of age due to generalized hypopigmentation and congenital nystagmus. Examination revealed marked iris translucency, diffuse fundus hypopigmentation with prominent choroidal vasculature, pendular horizontal nystagmus, suspected esotropia, and hyperopia. Genetic testing was initiated at five months of age to determine the underlying cause of the oculocutaneous albinism, including possible syndromic causes. It identified biallelic variants in the HPS4 gene, confirming Hermansky-Pudlak syndrome type 4 at eight months of age. Multidisciplinary evaluation, including hematologic and pulmonary baseline assessments, was subsequently initiated. At the latest follow-up, at two years and five months of age, the patient remained clinically stable without evidence of pulmonary or gastrointestinal complications. Visual rehabilitation and early developmental support were implemented. This case underscores the critical role of early ophthalmologic recognition of oculocutaneous albinism in establishing the diagnosis of Hermansky-Pudlak syndrome. Timely genetic confirmation enables structured surveillance for pulmonary fibrosis and other systemic complications associated with HPS type 4.

Indexed as

hermansky-pudlak syndromehermansky-pudlak syndrome type 4hpsocculocutaneous albinismsyndromic albinism

Identifiers

PMID42703512
PMCPMC13546565

What OpenQuestion holds

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Registered trials

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.