Intrapapillary Hemorrhage with Adjacent Peripapillary Subretinal Hemorrhage

Key Points of This Disease

• IHAPSH is a syndrome characterized by simultaneous intrapapillary hemorrhage and adjacent peripapillary subretinal hemorrhage¹⁾.
• It is more commonly reported in young myopic individuals, especially women, but can occur across a wide age range, and bilateral cases have been reported^1,5,6).
• Symptoms mainly include floaters and mild blurred vision, and it may be discovered asymptomatically¹⁾.
• Visual field defects are often limited to an enlarged blind spot or Bjerrum-type scotoma, and corrected visual acuity is generally preserved^1,5).
• A small scleral canal and vitreopapillary traction have been proposed as mechanisms of onset^1,3,13).
• Association with tilted disc and PHOMS has been reported, and differentiation from papilledema is important^14,15,18,19).
• Most cases resolve spontaneously within weeks to months, and observation is the standard management^1,6,10,11).

1. What is Intrapapillary Hemorrhage (IHAPSH)?

Definition

Intrapapillary Hemorrhage with Adjacent Peripapillary Subretinal Hemorrhage (IHAPSH) refers to a clinical picture in which hemorrhage within the optic disc (intrapapillary) and contiguous peripapillary subretinal hemorrhage are observed simultaneously. It may be accompanied by optic disc edema-like findings and is positioned as a benign syndrome that commonly occurs in young, myopic eyes¹⁾.

History

The condition of optic disc hemorrhage and peripapillary subretinal hemorrhage in the same eye dates back to a 1975 report by Cibis et al. on retinal hemorrhage associated with posterior vitreous detachment²⁾. In 1995, Katz and Hoyt reported intrapapillary and peripapillary hemorrhage occurring in young individuals with incomplete posterior vitreous detachment as a finding of vitreopapillary traction³⁾. In 2004, Kokame et al. reported this syndrome in 9 cases (10 eyes; 7 women, 2 men; ages 14–79). Of the 10 eyes, 8 had myopia of −2.50 to −9.50 D, and 8 had tilted optic discs¹⁾.

Epidemiology

The exact incidence is unknown, but case reports in Japan tend to be biased toward young, myopic women. In the initial 9 cases by Kokame et al., the majority were women, and refractive errors ranged from mild to high myopia¹⁾. Bilateral cases are relatively rare, but there are reports of sequential onset in both eyes over a 2-year course in a 25-year-old woman⁵⁾ and similar findings in both eyes at a 2-month interval in a 36-year-old woman⁶⁾. Cases in teenagers are also increasing, with typical cases accumulating in an 11-year-old girl⁷⁾, a 12-year-old^9,10), a 17-year-old¹¹⁾, and a 19-year-old¹⁾.

Q How rare is IHAPSH?

A

The nationwide incidence is not known. Current literature consists mainly of small case series and case reports, and the exact prevalence is unclear¹⁾.

2. Main Symptoms and Clinical Findings

Wang Y, Chen H, Yuan L, et al. Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage of both eyes after COVID-19 infection: a case report. BMC Ophthalmol. 2024;24(1):101. Figure 1. PMCID: PMC10913657. License: CC BY 4.0.

Fundus photographs of the same case: A (right eye) shows a tilted optic disc, intrapapillary hemorrhage, and vitreous hemorrhage. B (left eye) has an indistinct disc margin, but from this image alone, it is not determined that the left eye also has the same hemorrhagic findings.

Subjective Symptoms

Typical subjective symptoms are floaters and mild blurred vision, and it may be discovered asymptomatically. Visual acuity is often well preserved; in 8 of 10 eyes reported by Kokame et al., it was 20/25 or better¹⁾. In Japanese cases, a 12-year-old girl with corrected visual acuity of 0.9/1.2 at initial visit¹⁰⁾, a 17-year-old maintaining 1.2 in both eyes¹¹⁾, and an 11-year-old girl with 1.2/1.2⁷⁾ have been reported.

However, when hemorrhage extends into the vitreous cavity or subretinal hemorrhage occurs near the macula, blurred vision, visual field defects, and central scotoma may occur. A case of a 62-year-old woman with vitreous hemorrhage and peripapillary subretinal hemorrhage whose corrected visual acuity in the right eye decreased to 0.01 has also been reported⁴⁾.

Fundus Findings

Three characteristic hemorrhagic components are observed simultaneously or sequentially^1,4).

• Intrapapillary hemorrhage: Red to dark red hemorrhage spreading radially on the optic disc surface.
• Peripapillary subretinal hemorrhage: Pale red to dark red hemorrhage extending from the disc margin to the nasal to inferonasal side, with less distinct borders than intraretinal hemorrhage.
• Vitreous hemorrhage: In mild cases, it appears as thin hemorrhagic floaters in front of the disc; in severe cases, it may obscure fundus examination.

Peripapillary subretinal hemorrhage is often biased toward the nasal side^4,7,8,10). At onset, mild papilledema-like swelling may be present, requiring differentiation from congestive optic disc⁹⁾. Hwang and Lin reported multilayered hemorrhages on the disc surface, subretinal, and vitreous in adolescent patients with myopia and crowded or tilted discs, and considered vitreopapillary traction as one mechanism²¹⁾.

Visual Field Findings

Visual field changes are mild, such as enlargement of the Mariotte blind spot or Bjerrum-type scotoma, and are often not noticed by the patient. Goldmann or Humphrey perimetry may show arcuate sensitivity loss or nasal step corresponding to the hemorrhage site. After hemorrhage resolution, visual field defects often improve^1,5).

Course and Prognosis

Hemorrhage generally resolves spontaneously over 6 weeks to several months without treatment. In Kokame et al.’s observation, 8 out of 10 eyes achieved 20/25 or better, and no recurrence was noted in the same eye¹⁾. In typical cases in Japan, there is a report of a 12-year-old girl whose vitreous hemorrhage disappeared 6 weeks after the initial visit, with corrected visual acuity improving to 1.0¹⁰⁾, and a report of a 36-year-old woman with bilateral involvement who recovered to 1.2 in the right eye and 1.0 in the left eye within 2 months⁶⁾. Since some cases develop in both eyes at different times, the contralateral eye should also be examined^5,6).

3. Causes and Risk Factors

Related Anatomical Factors

• Small optic disc and small scleral canal: Teng et al. reported that a crowded optic nerve head and small scleral canal are associated with IHAPSH¹³⁾. Dense nerve fibers within the disc and a small scleral canal opening may predispose to hemorrhage under minor mechanical stress.
• Disc tilt: Takahashi et al. used swept-source OCT to show that the disc tilt angle in IHAPSH eyes is larger than in control eyes¹⁴⁾. A case in Japan with combined tilted disc syndrome, PHOMS, and IHAPSH also supports the association between tilt and this condition¹⁵⁾.
• Myopia and axial elongation: Most reported cases have mild to high myopia, and there is a case of a 12-year-old girl who developed IHAPSH during the progressive phase of myopia⁹⁾. Axial elongation and weakening of peripapillary tissue are thought to underlie this condition.
• Coexistence of PHOMS (peripapillary hyperreflective ovoid mass-like structures): PHOMS are ovoid hyperreflective structures observed on Bruch’s membrane around the optic disc and are thought to reflect axoplasmic stasis^16,17). Multiple reports in Japan have described PHOMS in IHAPSH patients^15,18), suggesting common anatomical and mechanical predispositions. In children, PHOMS have been reported as a frequent cause of pseudopapilledema²²⁾, and the prevalence of PHOMS is significantly higher in children with myopia²³⁾.

Related Mechanical Factors

• Posterior vitreous detachment (PVD) and vitreopapillary traction: Cibis et al. reported that PVD can cause optic disc hemorrhage and retinal hemorrhage²⁾, and Katz and Hoyt showed that vitreopapillary traction due to incomplete PVD causes optic disc hemorrhage in young individuals³⁾. In a 62-year-old case in Japan, PVD and rupture of superficial optic disc vessels were presumed to be the bleeding mechanism⁴⁾.
• Valsalva-like venous pressure elevation: This has been proposed as a candidate bleeding mechanism but is not established as a trigger for IHAPSH¹²⁾.

Patient background as risk

• Young (10s–30s) female
• Myopia (mild to high)
• Tilted disc, small scleral canal, PHOMS
• Incomplete PVD, vitreopapillary adhesion
• Valsalva-like venous pressure elevation (hypothetical candidate, not an established trigger)

Among these, a small scleral canal/crowded disc and papilla tilt are associated factors that showed differences from the control group in case-control studies and fellow-eye comparisons^13,14), while a Valsalva-like venous pressure elevation is a candidate at the hypothesis stage¹²⁾. None of these are established risk scores for predicting individual onset^{1,3,12,13,14)}.

4. Diagnosis and examination methods

Basic diagnosis

The starting point for diagnosis is ophthalmoscopic confirmation of “intrapapillary hemorrhage” and “adjacent peripapillary subretinal hemorrhage” in the same eye. When findings such as nasal deviation of subretinal hemorrhage, mild vitreous hemorrhage, and background factors like young age, myopia, and female sex are present, IHAPSH should be strongly suspected^1,4).

Key points for diagnosis

• Confirm intrapapillary hemorrhage and adjacent peripapillary subretinal hemorrhage in the same eye¹⁾.
• Assess visual acuity, visual field, and degree of optic disc edema, and rule out optic neuritis and papilledema.
• Spontaneous absorption of hemorrhage over the course supports the diagnosis^1,12).

Imaging Tests

Wang Y, Chen H, Yuan L, et al. Intrapapillary hemorrhage with adjacent peripapillary subretinal hemorrhage of both eyes after COVID-19 infection: a case report. BMC Ophthalmol. 2024;24(1):101. Figure 2. PMCID: PMC10913657. License: CC BY 4.0.

Vertical OCT scans through the optic nerve head of the same case: A is the right eye, B is the left eye. The original article shows elevation of the nasal optic disc and high-reflectivity material in the peripapillary subretinal space. OCT can noninvasively evaluate the location and layered structure of hemorrhage and is also used to assess coexisting PHOMS and optic disc tilt.

• Optical coherence tomography (OCT): Evaluates the layered structure of intra- and peripapillary hemorrhage. Hemorrhage appears as a hyperreflective lesion extending into the subretinal space, and the presence of PHOMS (peripapillary hyperreflective ovoid mass-like structures) is also assessed^10,15,16). Swept-source OCT is useful for observing optic disc tilt angle, small scleral canal, and PHOMS^14,15).
• OCT angiography (OCTA): An ancillary test to check for abnormal vascular signals around the optic disc. Blood flow signals within PHOMS have also been reported¹⁷⁾.
• Fluorescein angiography (FA) / Indocyanine green angiography (IA): In addition to fluorescence blockage due to hemorrhage, some cases show mild optic disc staining¹⁾. In atypical cases, angiography is used to rule out neovascular lesions¹²⁾.
• Head MRI/MRV: Considered when bilateral optic disc edema, headache, pulsatile tinnitus, etc., suggest papilledema or idiopathic intracranial hypertension (IIH). In a Japanese case of IIH in a patient in their 30s, findings included enlargement of the perioptic subarachnoid space, flattening of the posterior globe, and PHOMS, with a cerebrospinal fluid opening pressure of 300 mmH₂O confirmed¹⁹⁾.

Visual Field and Electrophysiological Tests

Goldmann perimetry and Humphrey perimetry are used to evaluate enlargement of the Mariotte blind spot and the presence of Bjerrum scotoma. In isolated IHAPSH, central visual field is often preserved^1,5). If another optic nerve disease such as optic neuritis is suspected, additional tests are performed based on symptoms and findings.

Differential Diagnosis

Check for decreased visual acuity, RAPD, optic disc edema, and systemic symptoms, and rule out the following diseases based on clinical findings and necessary additional tests^1,19,20,22).

Differential Diagnosis	Key Points for Differentiation
Nonarteritic anterior ischemic optic neuropathy (NAION)	Older age, acute severe vision loss with profound visual field defect, predominantly optic disc edema
Optic neuritis	Pain with eye movement, central scotoma, relative afferent pupillary defect (RAPD)
Glaucomatous optic neuropathy	Chronic course, characteristic optic disc cupping and RNFL defect
Papilledema / IIH	Bilateral optic disc edema, headache, pulsatile tinnitus, elevated CSF pressure
Optic disc drusen	Autofluorescence, OCT, ultrasound for calcification assessment
Tilted disc syndrome alone	Congenital optic disc dysplasia without hemorrhage
Terson syndrome	Preceding CNS event such as subarachnoid hemorrhage
Traumatic optic disc hemorrhage	Clear history of trauma
Optic disc hemorrhage related to hematologic disease	Systemic findings such as severe anemia or thrombocytopenia 20)

5. Standard Treatment

Observation as a Principle

IHAPSH is a benign condition that resolves spontaneously, and no definitive medical or surgical treatment has been established. Many reports indicate that hemorrhage resolves within 6 weeks to a few months with observation alone ^1,6,10,11).

Key Points for Observation

• Initial evaluation: Basic assessments include visual acuity, refractive error, intraocular pressure, visual field, and OCT. Additional tests such as FA/OCTA or neuroimaging may be performed based on findings to rule out other diseases ^1,12,14).
• Observation interval: Set individually according to the extent of hemorrhage, visual acuity, visual field, and certainty of differential diagnosis.
• Lifestyle guidance: No established activity restrictions prevent recurrence of IHAPSH. Discuss lifestyle precautions individually with the attending physician based on symptoms and fundus findings.
• Evaluation of the fellow eye: Since metachronous involvement of the fellow eye can occur, examine the fellow eye at the initial visit and follow up individually based on symptoms and findings ^5,6).

Exceptional Interventions

There is no established medical or surgical treatment for IHAPSH itself ¹⁾. If the hemorrhage extends to the macula, visual impairment persists, or abnormal vascular signals are observed, deviating from the typical course, reassess for CNV or other retinal/optic nerve diseases and treat according to the diagnosis.

Caution

• Even after diagnosing IHAPSH, if visual field deterioration, optic disc pallor, or bilateral optic disc edema appear, reconsider other diseases such as glaucoma, optic neuritis, or IIH ¹⁹⁾.
• Use imaging findings such as OCT to differentiate from optic disc drusen or PHOMS ^16,22).

6. Pathophysiology and Detailed Mechanism

Mechanical Traction Hypothesis

One proposed mechanism is that nerve fibers and blood vessels densely packed in the small scleral canal of the optic disc undergo mechanical stress due to vitreopapillary traction associated with incomplete posterior vitreous detachment (PVD), leading to hemorrhage^1,3,13). Cases with hemorrhage extending to multiple layers, including the disc surface, peripapillary subretinal space, and vitreous cavity, have also been reported²¹⁾.

Vascular and Tissue Fragility

In a case-control study using swept-source OCT, the optic disc tilt angle in eyes with IHAPSH was greater than that in the contralateral eyes¹⁴⁾. Cases of tilted disc syndrome accompanied by PHOMS and IHAPSH have been reported, but whether tilt or PHOMS is the cause of onset has not been established¹⁵⁾.

Association with PHOMS

PHOMS are ovoid hyperreflective structures observed above the Bruch’s membrane opening on OCT. Initially, differentiation from optic disc drusen was a concern, but they are now understood to reflect axoplasmic flow stasis in retinal nerve fibers^16,17). Borrelli et al. demonstrated blood flow signals within PHOMS on OCTA¹⁷⁾. Tokuhisa et al. reported a 12-year-old girl with IHAPSH who had bilateral PHOMS, suggesting that the coexistence of PHOMS and IHAPSH may not be coincidental¹⁸⁾. Yamamoto et al. also presented an 11-year-old case with tilted disc syndrome accompanied by PHOMS and IHAPSH, discussing the possibility that PHOMS may predispose to the development of IHAPSH¹⁵⁾.

Hemodynamic Factors

Valsalva-like venous pressure elevation has been proposed as a candidate mechanism, but it has not been established as a common trigger or actual cause of IHAPSH¹²⁾.

Integrated Model

In IHAPSH, anatomical features of the optic disc, vitreopapillary traction, and transient hemodynamic changes have been considered as candidate mechanisms. However, the combination varies among cases, and a single onset model has not been established^{1,3,12,13,14)}.

7. Recent Research and Future Perspectives

Advances in Imaging Diagnosis

With the widespread use of swept-source OCT and OCTA, it has become possible to noninvasively evaluate PHOMS, optic disc tilt angle, and peripapillary blood flow signals ^14,17). Takahashi et al. quantified a larger optic disc tilt angle in IHAPSH eyes ¹⁴⁾. The coexistence of tilted optic disc and PHOMS has been reported in individual cases, and further case accumulation is needed to determine the frequency ^15,18).

Refinement of Disease Characteristics Through Case Accumulation

Multiple case reports suggest a tendency for onset to cluster in young women (teens) and middle-aged women (30s–40s), but the limited number of cases has not led to a definitive onset pattern ^7,9,10,11). Cases of onset in the contralateral eye at a different time have also been reported ^5,6).

Development of a Differential Diagnosis Algorithm

Since PHOMS is also observed in IIH and demyelinating diseases, it is necessary to carefully rule out coexisting IIH or optic neuritis in IHAPSH with PHOMS. A Japanese report of IIH with PHOMS in a patient in their 30s highlights the importance of systematic evaluation when PHOMS findings are detected ¹⁹⁾. In the future, an algorithm that parameterizes the presence, size, and distribution of PHOMS to quantify the risk of developing IHAPSH is expected.

Prevention and Patient Education

No established method exists to prevent the onset or recurrence of IHAPSH. Myopia is managed according to standard ophthalmic practice, but there is no clinical evidence that myopia progression control or refractive correction prevents IHAPSH.

For Patients: Please Read Carefully

• Optic disc hemorrhage (IHAPSH) is generally a relatively mild condition in which the bleeding is naturally absorbed and vision almost fully recovers.
• However, some diseases with a similar appearance require prompt treatment. Be sure to undergo a thorough eye examination (OCT, visual field testing, and MRI if necessary) at an ophthalmology clinic.
• If you notice increased floaters, a dark area in your vision, or light sensitivity, do not self-diagnose; seek medical attention.
• Regular follow-up visits are very important to confirm absorption of the hemorrhage and to detect any abnormalities in the other eye early. Adhere to the recommended follow-up schedule.
• There are no established activity restrictions to prevent recurrence of IHAPSH. Consult your doctor about any lifestyle precautions based on your examination findings.
• If you or a family member with strong myopia has similar symptoms, do not leave it untreated and see an ophthalmologist as soon as possible.

8. References

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